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1. Crom DB, Smith D, Xiong Z, Onar A, Hudson MM, Merchant TE, Morris EB: Health status in long-term survivors of pediatric craniopharyngiomas. J Neurosci Nurs; 2010 Dec;42(6):323-8; quiz 329-30
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Health status in long-term survivors of pediatric craniopharyngiomas.
  • Craniopharyngiomas are the third most common pediatric brain tumor and most common pediatric suprasellar tumor.
  • Contemporary treatment of craniopharyngiomas uses limited surgery and radiation in an effort to minimize morbidity, but the long-term health status of patients treated in this fashion has not been well described.
  • The purpose of this study was to analyze the health status of long-term survivors of pediatric craniopharyngioma treated primarily with radiation and conservative surgical resection.
  • Medical records of all long-term survivors of craniopharyngioma treated at St. Jude Children's Research Hospital and then transferred to the long-term follow-up clinic were reviewed.
  • Of these, 51 (93%) were alive at the time of this analysis.
  • The median age at diagnosis was 7.1 years (range, 1.2-17.6 years), and 29 (57%) were male.
  • At the time of analysis, the median survival was 7.6 years (range, 5.0-21.3 years).
  • Diagnosis and treatment included surgical biopsy, resection (n = 50), and radiation therapy (n=48).
  • Only 1 patient received chemotherapy.
  • In a small percentage of patients, complications may result in death even during extended remission of craniopharyngioma.
  • Because of the broad spectrum or morbidities experienced, survivors of craniopharyngioma continue to benefit from multidisciplinary care.

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  • (PMID = 21207770.001).
  • [ISSN] 0888-0395
  • [Journal-full-title] The Journal of neuroscience nursing : journal of the American Association of Neuroscience Nurses
  • [ISO-abbreviation] J Neurosci Nurs
  • [Language] ENG
  • [Grant] United States / NCI NIH HHS / CA / P30 CA021765; United States / NCI NIH HHS / CA / CA 21765
  • [Publication-type] Journal Article; Research Support, N.I.H., Extramural; Research Support, Non-U.S. Gov't
  • [Publication-country] United States
  • [Other-IDs] NLM/ NIHMS756555; NLM/ PMC4895693
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2. Nagasaki K, Tsumanuma I, Yoneoka Y, Jinguji S, Ogawa Y, Kikuchi T, Uchiyama M: Metabolic effects of growth hormone replacement in two pediatric patients with growth without growth hormone. Endocr J; 2010;57(9):771-5
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  • Growth without growth hormone (GH) has occasionally been described in patients who have had tumors removed in the hypothalamic-pituitary area.
  • Two children in whom the growth without GH phenomenon occurred after therapy for brain tumors participated in this study.
  • Case 1 is a 15-yr-old Japanese girl, diagnosed as having Langerhans cell histiocytosis with multiple intracranial lesions at the age of two.
  • She showed a slight body fat increase, dyslipidemia and fatty liver.
  • Case 2 is a 10-yr-old Indonesian boy, diagnosed with craniopharyngioma at the age of three.
  • In both cases, GH replacement therapy was started at 0.042 mg/kg/week for 12 months.
  • Body composition, BMD, and visceral abdominal area were measured every 3 months.
  • Serum fasting blood glucose, insulin, ALT, lipid profile, leptin, and adiponectin levels were also measured every 3 months.
  • Case 1 showed improvement of transaminase (ALT from 64 to 16 IU/L) and triglyceride (from 239 to 129 mg/dL) over 12 months, but did not show a decrease in visceral fat area or of body fat percentage.
  • Case 2 showed a decrease in body fat percentage and visceral fat area, accompanied by elevated serum adiponectin and decreased leptin levels.
  • In conclusion, twelve months GH replacement therapy improves metabolic abnormalities in pediatric patients with growth without GH.
  • [MeSH-major] Growth / drug effects. Hormone Replacement Therapy. Human Growth Hormone / therapeutic use
  • [MeSH-minor] Adiponectin / blood. Adolescent. Body Composition / drug effects. Child. Craniopharyngioma / surgery. Dyslipidemias / metabolism. Empty Sella Syndrome / drug therapy. Fatty Liver / metabolism. Female. Humans. Leptin / blood. Male. Pituitary Neoplasms / surgery

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  • (PMID = 20660985.001).
  • [ISSN] 1348-4540
  • [Journal-full-title] Endocrine journal
  • [ISO-abbreviation] Endocr. J.
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article; Research Support, Non-U.S. Gov't
  • [Publication-country] Japan
  • [Chemical-registry-number] 0 / ADIPOQ protein, human; 0 / Adiponectin; 0 / Leptin; 12629-01-5 / Human Growth Hormone
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3. Nejat F, El Khashab M, Rutka JT: Initial management of childhood brain tumors: neurosurgical considerations. J Child Neurol; 2008 Oct;23(10):1136-48
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  • [Title] Initial management of childhood brain tumors: neurosurgical considerations.
  • Intracranial tumors are the most common solid tumors in children.
  • The infratentorial compartment will be the primary site for 60% to 70% of these tumors, including astrocytomas, medulloblastomas, and ependymomas.
  • Several technological advancements have increased our knowledge of the cell biology of pediatric brain tumors, facilitated earlier diagnosis, and improved neurosurgical resections while minimizing neurological deficits.
  • These in turn have not only improved the survival of children with brain tumors but also their quality of life.
  • Current management strategies in most cases rely on surgery coupled with adjuvant therapies, including radiation therapy and chemotherapy.
  • The vulnerability of the immature brain to adjuvant therapies creates many challenges for the treating physician.
  • We review current diagnostic and therapeutic approaches and outcome for children harboring the most common pediatric brain tumors: astrocytomas (low-grade and high-grade glioma), ependymoma, medulloblastoma, and craniopharyngioma.
  • The emphasis will be on the neurosurgical management of children with these tumors.

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  • (PMID = 18952580.001).
  • [ISSN] 1708-8283
  • [Journal-full-title] Journal of child neurology
  • [ISO-abbreviation] J. Child Neurol.
  • [Language] ENG
  • [Grant] United States / NINDS NIH HHS / NS / R13 NS040925; United States / NINDS NIH HHS / NS / 5R13NS040925-09
  • [Publication-type] Journal Article; Research Support, N.I.H., Extramural; Review
  • [Publication-country] United States
  • [Number-of-references] 92
  • [Other-IDs] NLM/ NIHMS487102; NLM/ PMC3714852
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4. Lustig RH, Post SR, Srivannaboon K, Rose SR, Danish RK, Burghen GA, Xiong X, Wu S, Merchant TE: Risk factors for the development of obesity in children surviving brain tumors. J Clin Endocrinol Metab; 2003 Feb;88(2):611-6
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Risk factors for the development of obesity in children surviving brain tumors.
  • Hypothalamic obesity, a syndrome of intractable weight gain due to hypothalamic damage, is an uncommon but devastating complication for children surviving brain tumors.
  • We undertook a retrospective evaluation of the body mass index (BMI) curves for the St. Jude Children's Research Hospital brain tumor population diagnosed between 1965 and 1995 after completion of therapy to determine risk factors for the development of obesity.
  • Inclusion criteria were: diagnosis less than 14 yr of age, no spinal cord involvement, ambulatory, no supraphysiologic hydrocortisone therapy (>12 mg/m(2) x d), treatment and follow-up at St. Jude Children's Research Hospital, and disease-free survival greater than 5 yr (n = 148).
  • Risk factors examined were age at diagnosis, tumor location, histology, extent of surgery, hydrocephalus requiring ventriculoperitoneal shunting, initial high-dose glucocorticoids, cranial radiation therapy, radiation dosimetry to the hypothalamus, intrathecal chemotherapy, and presence of endocrinopathy.
  • Analyses were performed both between groups within a risk factor and against BMI changes for age in normal children older than 5.5 yr (the age of adiposity rebound).
  • Risk factors were: age at diagnosis (P = 0.04), radiation dosimetry to the hypothalamus (51-72 Gy, P = 0.002 even after hypothalamic and thalamic tumor exclusion), and presence of any endocrinopathy (P = 0.03).
  • In addition, risk factors when compared with BMI slope for the general American pediatric population included: tumor location (hypothalamic, P = 0.001), tumor histology (craniopharyngioma, P = 0.009; pilocytic astrocytoma, P = 0.043; medulloblastoma, P = 0.039); and extent of surgery (biopsy, P = 0.03; subtotal resection, P = 0.018).
  • These results verify hypothalamic damage, either due to tumor, surgery, or radiation, as the primary cause of obesity in survivors of childhood brain tumors.
  • In particular, hypothalamic radiation doses of more than 51 Gy are permissive.
  • These results reiterate the importance of the hypothalamus in energy balance, provide risk assessment criteria for preventative measures before the development of obesity in at-risk patients, and suggest therapeutic strategies to reduce the future development of obesity.
  • [MeSH-major] Brain Neoplasms / epidemiology. Craniopharyngioma / epidemiology. Obesity / epidemiology
  • [MeSH-minor] Astrocytoma / drug therapy. Astrocytoma / epidemiology. Astrocytoma / radiotherapy. Cerebellar Neoplasms / drug therapy. Cerebellar Neoplasms / epidemiology. Cerebellar Neoplasms / radiotherapy. Child. Child, Preschool. Disease-Free Survival. Humans. Hypothalamus / physiology. Medulloblastoma / drug therapy. Medulloblastoma / epidemiology. Medulloblastoma / radiotherapy. Retrospective Studies. Risk Factors

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  • (PMID = 12574189.001).
  • [ISSN] 0021-972X
  • [Journal-full-title] The Journal of clinical endocrinology and metabolism
  • [ISO-abbreviation] J. Clin. Endocrinol. Metab.
  • [Language] eng
  • [Grant] United States / NCI NIH HHS / CA / R25 CA023944; United States / NCI NIH HHS / CA / P30CA12765
  • [Publication-type] Journal Article; Research Support, Non-U.S. Gov't; Research Support, U.S. Gov't, P.H.S.
  • [Publication-country] United States
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5. Hubina E, Kovács L, Szabolcs I, Rimanóczy E, Czirják S, Góth M: [Serum tumor marker levels during a 12-months growth hormone replacement therapy in patients with adult growth hormone deficiency]. Orv Hetil; 2002 Mar 24;143(12):601-5
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  • [Title] [Serum tumor marker levels during a 12-months growth hormone replacement therapy in patients with adult growth hormone deficiency].
  • There are also some data suggesting the development of different neoplasms in animals after growth hormone administration and in humans upon growth hormone replacement therapy.
  • AIMS: The general opinion is however, that growth hormone replacement therapy has no oncogenic effect, but the tumor marker levels have not been studied so far.
  • 6 of them had multiple pituitary hormone deficiency and were on replacement therapy (thyroxine: 2, cortisone: 1, sexual steroids: 6 and desmopressin: 2 patients).
  • The cause of growth hormone deficiency was the removal of pituitary tumor (6 patients) or craniopharyngioma (2 patients), and in 1 case the deficiency was idiopathic.
  • The mean dose of growth hormone was 0.53 in female and 0.51 mg/day in male patients.
  • Insulin-like growth factor-1, carcinoembryonal antigen, human choriogonadotropin hormone, alpha-fetoprotein, prostate specific antigen, tissue polypeptide antigen-M, ferritin, gastrointestinal carcinoma antigen, ovarian antigen, breast specific antigen, carcinoma antigen 50 were measured at baseline and after 3, 6 and 12 months of GH replacement.
  • The mean value of all tumor markers remained within the normal range and there was no significant increase within the normal range either.
  • CONCLUSION: The lack of increase of tumor marker levels does not indicate possible oncogenic effect of one-year GH treatment in hypopituitary adults.
  • The authors can not draw any far-reaching conclusions because of the low patient number and the short follow-up, but the measurement of tumor marker levels may provide useful means to follow up long-term therapy and for the early diagnosis of possible occult malignancy.
  • [MeSH-major] Biomarkers, Tumor / blood. Growth Hormone / adverse effects. Growth Hormone / deficiency. Hypopituitarism / drug therapy
  • [MeSH-minor] Adult. Antigens, Tumor-Associated, Carbohydrate / blood. Carcinoembryonic Antigen / blood. Chorionic Gonadotropin / blood. Cortisone / therapeutic use. Deamino Arginine Vasopressin / therapeutic use. Female. Ferritins / blood. Gonadal Steroid Hormones / therapeutic use. Humans. Immunoassay. Insulin-Like Growth Factor I / metabolism. Male. Middle Aged. Prostate-Specific Antigen / blood. Thyroxine / therapeutic use. Time Factors. Tissue Polypeptide Antigen / blood. alpha-Fetoproteins / metabolism

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  • (PMID = 11963397.001).
  • [ISSN] 0030-6002
  • [Journal-full-title] Orvosi hetilap
  • [ISO-abbreviation] Orv Hetil
  • [Language] hun
  • [Publication-type] English Abstract; Journal Article
  • [Publication-country] Hungary
  • [Chemical-registry-number] 0 / Antigens, Tumor-Associated, Carbohydrate; 0 / Biomarkers, Tumor; 0 / Carcinoembryonic Antigen; 0 / Chorionic Gonadotropin; 0 / Gonadal Steroid Hormones; 0 / Tissue Polypeptide Antigen; 0 / alpha-Fetoproteins; 67763-96-6 / Insulin-Like Growth Factor I; 9002-72-6 / Growth Hormone; 9007-73-2 / Ferritins; EC 3.4.21.77 / Prostate-Specific Antigen; ENR1LLB0FP / Deamino Arginine Vasopressin; Q51BO43MG4 / Thyroxine; V27W9254FZ / Cortisone
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6. Müller HL, Heinrich M, Bueb K, Etavard-Gorris N, Gebhardt U, Kolb R, Sörensen N: Perioperative dexamethasone treatment in childhood craniopharyngioma--influence on short-term and long-term weight gain. Exp Clin Endocrinol Diabetes; 2003 Sep;111(6):330-4
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  • [Title] Perioperative dexamethasone treatment in childhood craniopharyngioma--influence on short-term and long-term weight gain.
  • The substitution of dexamethasone during and after surgery of childhood craniopharyngioma is necessary in order to treat and/or prevent brain edema and adrenal insufficiency.
  • In a retrospective analysis of 60 patients with childhood craniopharyngioma we inquired whether dose and duration of perioperative dexamethasone therapy (n = 68) had influence on short-term post-operative weight gain and long-term development of severe obesity.
  • 24 patients (14 f/10 m) developed severe obesity (BMI > 3 SD).
  • 28 patients (10 f/18 m) retained normal weight (BMI < 2 SD).
  • Eight patients presented with a BMI between 2 and 3 SD at the final visit.
  • Differences in terms of age at surgery or follow-up period were non-detectable between the analyzed groups of craniopharyngioma patients.
  • Duration and cumulative dexamethasone doses (mg/m2 BSA) for perioperative dexamethasone therapy were similar for severely obese patients (duration: 8.7 d; 4.5 - 17 d, cumulative dose: 74; 42 - 177 mg/m2 BSA) and normal weight patients (duration: 10.0 d; 1 - 41 d; dose: 76; 9 - 390 mg/m2 BSA).
  • Whereas cumulative dexamethasone doses positively (p < 0.01; rho: 0.424) correlated with weight gain during the first year following surgery, long-term development of severe obesity was not influenced by dose and duration of perioperative dexamethasone treatment.
  • Patients who developed severe obesity during follow-up had a higher (p < 0.001) BMI already at the time of diagnosis.
  • We conclude that dose and duration of perioperative dexamethasone treatment had short-term effects on post-operative weight gain, but not on the development of long-term severe obesity.
  • The results of our retrospective analysis are currently tested in a prospective surveillance study Kraniopharyngeom 2000 (www.kraniopharyngeom.com).
  • [MeSH-major] Craniopharyngioma / drug therapy. Craniopharyngioma / surgery. Dexamethasone / therapeutic use. Pituitary Neoplasms / drug therapy. Pituitary Neoplasms / surgery. Weight Gain / physiology
  • [MeSH-minor] Adolescent. Antineoplastic Agents, Hormonal / therapeutic use. Body Mass Index. Child. Child, Preschool. Combined Modality Therapy. Female. Humans. Infant. Male. Retrospective Studies

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  • (PMID = 14520598.001).
  • [ISSN] 0947-7349
  • [Journal-full-title] Experimental and clinical endocrinology & diabetes : official journal, German Society of Endocrinology [and] German Diabetes Association
  • [ISO-abbreviation] Exp. Clin. Endocrinol. Diabetes
  • [Language] eng
  • [Publication-type] Journal Article; Research Support, Non-U.S. Gov't
  • [Publication-country] Germany
  • [Chemical-registry-number] 0 / Antineoplastic Agents, Hormonal; 7S5I7G3JQL / Dexamethasone
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7. Lehman NL: The ubiquitin proteasome system in neuropathology. Acta Neuropathol; 2009 Sep;118(3):329-47
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  • In neuropathology, alteration of the UPS, or mutations in UPS target proteins may result in signaling abnormalities leading to the initiation or progression of tumors such as astrocytomas, hemangioblastomas, craniopharyngiomas, pituitary adenomas, and medulloblastomas.
  • Dysregulation of the UPS may also contribute to tumor progression by perturbation of DNA replication and mitotic control mechanisms, leading to genomic instability.
  • In neurodegenerative diseases caused by the expression of mutant proteins, the cellular accumulation of these proteins may overload the UPS, indirectly contributing to the disease process, e.g., sporadic Parkinsonism and prion diseases.
  • Defects or dysfunction of the UPS may also underlie cognitive disorders such as Angelman syndrome, Rett syndrome and autism, and muscle and nerve diseases, e.g., inclusion body myopathy and giant axon neuropathy.
  • This paper describes the basic biochemical mechanisms comprising the UPS and reviews both its theoretical and proven involvement in neuropathological diseases.
  • The potential for the UPS as a target of pharmacological therapy is also discussed.
  • [MeSH-minor] Brain Diseases / metabolism. Brain Neoplasms / metabolism. Humans. Neurodegenerative Diseases / metabolism. Substrate Specificity

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  • (PMID = 19597829.001).
  • [ISSN] 1432-0533
  • [Journal-full-title] Acta neuropathologica
  • [ISO-abbreviation] Acta Neuropathol.
  • [Language] eng
  • [Grant] United States / NINDS NIH HHS / NS / K08 NS045077
  • [Publication-type] Journal Article; Review
  • [Publication-country] Germany
  • [Chemical-registry-number] 0 / Ubiquitin; EC 3.4.25.1 / Proteasome Endopeptidase Complex
  • [Number-of-references] 149
  • [Other-IDs] NLM/ PMC2716447
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8. Maiter D, Abs R, Johannsson G, Scanlon M, Jönsson PJ, Wilton P, Koltowska-Häggström M: Baseline characteristics and response to GH replacement of hypopituitary patients previously irradiated for pituitary adenoma or craniopharyngioma: data from the Pfizer International Metabolic Database. Eur J Endocrinol; 2006 Aug;155(2):253-60
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  • [Title] Baseline characteristics and response to GH replacement of hypopituitary patients previously irradiated for pituitary adenoma or craniopharyngioma: data from the Pfizer International Metabolic Database.
  • OBJECTIVE: To test the hypothesis whether the effects of GH replacement therapy in adults could be affected by prior pituitary irradiation, the baseline characteristics and response to GH were evaluated in adults with severe GH deficiency (GHD), who had received or not irradiation for the treatment of pituitary adenoma or craniopharyngioma.
  • DESIGN: Data from 447 patients, who had received radiotherapy (427 in addition to surgery), and 630 patients, who were operated on but not irradiated for their tumour, were retrieved from Pfizer International Metabolic Database (KIMS) and compared at baseline and 1 and 2 years following the onset of GH replacement.
  • However, irradiated patients had a greater impairment in the quality of life (QoL), a higher fat mass, lower high-density lipoprotein cholesterol levels and a lower bone mineral content (BMC) than non-irradiated patients.
  • Treatment with GH induced similar changes in both groups.
  • These beneficial effects were maintained and the BMC also increased in both groups by the second year of treatment.
  • CONCLUSIONS: This analysis shows that prior irradiation for pituitary adenoma or craniopharyngioma does not compromise the beneficial effects of GH replacement therapy.

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  • (PMID = 16868138.001).
  • [ISSN] 0804-4643
  • [Journal-full-title] European journal of endocrinology
  • [ISO-abbreviation] Eur. J. Endocrinol.
  • [Language] ENG
  • [Publication-type] Journal Article
  • [Publication-country] England
  • [Chemical-registry-number] 12629-01-5 / Human Growth Hormone
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9. Hannon MJ, Behan LA, Agha A: Thyrotoxic periodic paralysis due to excessive L-thyroxine replacement in a Caucasian man. Ann Clin Biochem; 2009 Sep;46(Pt 5):423-5
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  • [Title] Thyrotoxic periodic paralysis due to excessive L-thyroxine replacement in a Caucasian man.
  • Although the paralysis is temporary, it may be lethal if not diagnosed and treated rapidly, as profound hypokalaemia may induce respiratory muscle paralysis or cardiac arrest.
  • Here we describe the case of a patient with panhypopituitarism due to a craniopharyngioma, who developed thyrotoxic periodic paralysis due to excessive L-thyroxine replacement.
  • This disorder has been described in Asian subjects but, to our knowledge, thyrotoxic periodic paralysis secondary to excessive L-thyroxine replacement has never been described in Caucasians.
  • [MeSH-major] Hypokalemic Periodic Paralysis / chemically induced. Hypopituitarism / drug therapy. Thyrotoxicosis / chemically induced. Thyroxine / adverse effects. Thyroxine / therapeutic use
  • [MeSH-minor] Adult. European Continental Ancestry Group. Humans. Male


10. Plotnick L, Rapaport R, Desrosiers P, Fuqua JS: Update from the GHMonitorSM observational registry in children treated with recombinant human growth hormone (Saizen). Pediatr Endocrinol Rev; 2009 Jan;6 Suppl 2:278-82
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  • [Title] Update from the GHMonitorSM observational registry in children treated with recombinant human growth hormone (Saizen).
  • Since 2003, the GH MonitorSM, an observational registry, has collected data on pediatric subjects treated with Saizen (recombinant human growth hormone (r-hGH)) in the United States and Canada.
  • As of August 2007, 1733 subjects were enrolled (68.9% male).
  • The most common primary diagnosis at screening was idiopathic growth hormone deficiency (56.5% of subjects).Of those subjects with available data, mean height standard deviation (SD) score was -2.1+/-1.0, mean weight SD score was -1.4+/-1.5, and mean body mass index SD score was -0.1+/-1.3.
  • Among subjects in whom the presence or absence of other pituitary hormone deficiencies was recorded, 16.1% had multiple pituitary hormone deficiencies.
  • Most patients reported high compliance with therapy (92.6% missed 0-3 doses per month); compliance was similar for all delivery devices (needle/syringe, cool.clickTM or one.clickTM) used.
  • Two serious adverse events related to Saizen (hospitalization for placement of right frontal ventriculostomy and right frontal craniotomy for transcallosal resection of a large recurrent craniopharyngioma and left slipped capitofemoral epiphysis that required pinning of the right hip) were reported in the period from August 2006 to August 2007.
  • [MeSH-major] Growth Disorders / drug therapy. Human Growth Hormone / therapeutic use
  • [MeSH-minor] Adolescent. Child. Child, Preschool. Female. Humans. Infant. Male. Pituitary Hormones / deficiency. Recombinant Proteins / adverse effects. Recombinant Proteins / therapeutic use. Registries. Young Adult

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  • (PMID = 19337182.001).
  • [ISSN] 1565-4753
  • [Journal-full-title] Pediatric endocrinology reviews : PER
  • [ISO-abbreviation] Pediatr Endocrinol Rev
  • [Language] eng
  • [Publication-type] Journal Article; Review
  • [Publication-country] Israel
  • [Chemical-registry-number] 0 / Pituitary Hormones; 0 / Recombinant Proteins; 12629-01-5 / Human Growth Hormone
  • [Number-of-references] 6
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11. Binder G, Weber S, Ehrismann M, Zaiser N, Meisner C, Ranke MB, Maier L, Wudy SA, Hartmann MF, Heinrich U, Bettendorf M, Doerr HG, Pfaeffle RW, Keller E, South German Working Group for Pediatric Endocrinology: Effects of dehydroepiandrosterone therapy on pubic hair growth and psychological well-being in adolescent girls and young women with central adrenal insufficiency: a double-blind, randomized, placebo-controlled phase III trial. J Clin Endocrinol Metab; 2009 Apr;94(4):1182-90
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  • [Title] Effects of dehydroepiandrosterone therapy on pubic hair growth and psychological well-being in adolescent girls and young women with central adrenal insufficiency: a double-blind, randomized, placebo-controlled phase III trial.
  • CONTEXT AND OBJECTIVE: The efficacy of oral dehydroepiandrosterone (DHEA) in the treatment of atrichia pubis and psychological distress in young females with central adrenal insufficiency is unknown.
  • Our study aimed to evaluate this therapy.
  • Inclusion criteria were ACTH deficiency plus two or more additional pituitary deficiencies, serum DHEA less than 400 ng/ml, and pubertal stage more than B2.
  • Exclusion criteria were cerebral radiation with more than 30 Gy, tumor remission less than 1 yr, amaurosis, hypothalamic obesity, psychiatric disorders, and unstable hormone medication.
  • INTERVENTION: Patients were randomized to placebo (n = 12) or 25 mg HPLC-purified DHEA/d (n = 11) orally for 12 months after stratification into a nontumor (n = 7) and a tumor group (n = 16).
  • MAIN OUTCOME MEASURES: Clinical scoring of pubic hair stage was performed at 0, 6, and 12 months (primary endpoint), and psychometrical evaluation (Symptom Check-List-90-R and the Centre for Epidemiological Studies-Depression Scale) at 0 and 12 months (secondary endpoint).
  • RESULTS: In the placebo group, four patients dropped out because of recurrence of craniopharyngioma, manifestation of type 1 diabetes, and change of residence (n = 2); in the DHEA group, one patient dropped out because of recurrent anxiety attacks.
  • DHEA substitution resulted in normalization of DHEA sulfate and androstanediol glucuronide morning serum levels 2 h after drug intake (P < 0.006), and of its 24 h urinary metabolite levels (P < 0.0001), placebo had no effect.
  • Morning serum levels of androstenedione increased in the DHEA group (P < 0.02) but did not normalize.
  • The DHEA group exhibited significant progress in pubic hair growth from Tanner stage I-III to II-V (mean: +1.5 stages), whereas the placebo group did not (relative risk 0.138; 95% confidence interval 0.021-0.914; P = 0.0046).
  • Importantly, eight of the 10 Symptom Check-List-90-R scores, including those for depression, anxiety, and interpersonal sensitivity, and the global severity index improved in the DHEA group in comparison to the placebo group (P < 0.048).
  • CONCLUSIONS: In adolescent girls with central adrenal insufficiency, daily replacement with 25 mg DHEA orally is beneficial: atrichia pubis vanishes, and psychological well-being improves significantly.
  • [MeSH-major] Adrenal Insufficiency / drug therapy. Adrenocorticotropic Hormone / deficiency. Dehydroepiandrosterone / therapeutic use. Hair / growth & development. Hypopituitarism / drug therapy
  • [MeSH-minor] Adolescent. Adult. Blood Pressure / drug effects. Blood Pressure / physiology. Brain Neoplasms / epidemiology. Double-Blind Method. Female. Humans. Hydrocortisone / therapeutic use. Obesity / epidemiology. Young Adult

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  • (PMID = 19126625.001).
  • [ISSN] 1945-7197
  • [Journal-full-title] The Journal of clinical endocrinology and metabolism
  • [ISO-abbreviation] J. Clin. Endocrinol. Metab.
  • [Language] eng
  • [Publication-type] Clinical Trial, Phase III; Journal Article; Randomized Controlled Trial; Research Support, Non-U.S. Gov't
  • [Publication-country] United States
  • [Chemical-registry-number] 459AG36T1B / Dehydroepiandrosterone; 9002-60-2 / Adrenocorticotropic Hormone; WI4X0X7BPJ / Hydrocortisone
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12. Darendeliler F, Karagiannis G, Wilton P, Ranke MB, Albertsson-Wikland K, Anthony Price D, On Behalf Of The Kigs International Board: Recurrence of brain tumours in patients treated with growth hormone: analysis of KIGS (Pfizer International Growth Database). Acta Paediatr; 2006 Oct;95(10):1284-90
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  • [Title] Recurrence of brain tumours in patients treated with growth hormone: analysis of KIGS (Pfizer International Growth Database).
  • BACKGROUND: Growth hormone (GH) has been used successfully in the treatment of short stature secondary to GH deficiency in survivors of childhood brain tumours.
  • AIM: To analyse KIGS (Pfizer International Growth Database) with respect to tumour recurrence in patients with brain tumours.
  • METHODS: Data for tumour recurrence were analysed retrospectively in 1038 patients with craniopharyngiomas, 655 with medulloblastomas, 113 with ependymomas, 297 with germinomas, and 400 with astrocytomas or gliomas.
  • All patients had received recombinant human GH (Genotropin, Pfizer Inc.).
  • RESULTS: Recurrence-free survival rates were 63% at a follow-up of 10.3 y in craniopharyngioma, 69% in 9.1 y in the glial tumours, 71% in 7.4 y in germinomas, 92% in 4.6 y in medulloblastomas and 89% in 2.5 y in ependymomas.
  • Dose of GH and treatment modalities did not differ significantly between patients with and without recurrence.
  • CONCLUSION: Tumour recurrence rates in surviving patients with brain tumours receiving GH treatment do not appear to be increased compared with published reports.
  • [MeSH-major] Brain Neoplasms / drug therapy. Human Growth Hormone / therapeutic use. Neoplasm Recurrence, Local / epidemiology. Recombinant Proteins / therapeutic use
  • [MeSH-minor] Adolescent. Astrocytoma / drug therapy. Astrocytoma / mortality. Astrocytoma / radiotherapy. Astrocytoma / surgery. Child. Child, Preschool. Combined Modality Therapy. Craniopharyngioma / drug therapy. Craniopharyngioma / mortality. Craniopharyngioma / radiotherapy. Craniopharyngioma / surgery. Disease-Free Survival. Ependymoma / drug therapy. Ependymoma / mortality. Ependymoma / radiotherapy. Ependymoma / surgery. Female. Germinoma / drug therapy. Germinoma / mortality. Germinoma / radiotherapy. Germinoma / surgery. Humans. Male. Medulloblastoma / drug therapy. Medulloblastoma / mortality. Medulloblastoma / radiotherapy. Medulloblastoma / surgery

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  • (PMID = 16982503.001).
  • [ISSN] 0803-5253
  • [Journal-full-title] Acta paediatrica (Oslo, Norway : 1992)
  • [ISO-abbreviation] Acta Paediatr.
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] Norway
  • [Chemical-registry-number] 0 / Recombinant Proteins; 12629-01-5 / Human Growth Hormone
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13. Kedzia A, Obara-Moszyńska M, Korman E, Rabska-Pietrzak B, Kopinski P, Trojan J, Goździcka-Józefiak A: Growth hormone treatment in pituitary insufficiency: selected cases of children with craniopharyngioma and medulloblastoma. Rocz Akad Med Bialymst; 2003;48:28-33
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  • [Title] Growth hormone treatment in pituitary insufficiency: selected cases of children with craniopharyngioma and medulloblastoma.
  • PURPOSE: The work concerns the substitution treatment with growth hormone (GH) in hypopituitary children, including cases that occurred in the course of tumor disease, craniopharyngioma (CP) and medulloblastoma (MB).
  • MATERIAL AND METHODS: The studied population concerned 117 children who presented either somatotropic or polyhormonal pituitary insufficiency (the average age was 12.6 years for girls and 13.6 years for boys).
  • The diagnosis of somatotropic pituitary insufficiency (SPI) was based on insulin and clonidin stimulation tests evaluating GH reserve of hypophysis.
  • The computer tomography (CT) and nuclear magnetic resonance (NMR) examinations were carried out before GH substitution in all children.
  • The tumors (four CP cases and one case of MB) were all found in boys and they were treated with surgery and/or radiotherapy.
  • All studied children, including CP and MB operated patients were treated with human GH (hGH)--Genotropin 16 IU, administered in subcutaneous injections.
  • RESULTS: The annual increase of children height before GH therapy was about 3.2 cm.
  • In the first year of GH therapy the difference in children growth between the CP/MB group as compared with the rest of patients was less than 1.0 cm: 9.4 and 10.2 cm/year, resp.
  • Control NMR examination performed in CP/MB patients treated with surgery with subsequent hGH therapy did not demonstrate any recurrence of tumor.
  • CONCLUSIONS: After two years of hGH therapy the final height of hypopituitary children, including CP patients, nearly reached the values observed in healthy children.
  • GH therapy did not induce a recurrence of neoplasm in CP and MB patients.
  • [MeSH-major] Brain Neoplasms / complications. Craniopharyngioma / complications. Human Growth Hormone / therapeutic use. Hypopituitarism / drug therapy. Medulloblastoma / complications
  • [MeSH-minor] Adolescent. Child. Child, Preschool. Combined Modality Therapy. Female. Humans. Male. Treatment Outcome


14. Mottolese C, Stan H, Hermier M, Berlier P, Convert J, Frappaz D, Lapras C: Intracystic chemotherapy with bleomycin in the treatment of craniopharyngiomas. Childs Nerv Syst; 2001 Dec;17(12):724-30
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  • [Title] Intracystic chemotherapy with bleomycin in the treatment of craniopharyngiomas.
  • OBJECT: Since bleomycin has not yet been used very frequently in the treatment of patients with craniopharyngioma, it seemed important to document the course of a series of such patients treated with this preparation.
  • METHODS AND RESULTS: Local chemotherapy with bleomycin was performed in 24 patients (20 children and 4 adults), 16 of whom presented with cystic or mixed (solid/cystic) craniopharyngioma and 8, with recurrent cystic craniopharyngioma.
  • The drug was administered through an Ommaya reservoir, which was placed either by using a direct surgical approach (6 patients) or a stereotactic approach (16 patients), or with endoscopic assistance in patients with hydrocephaly (2 patients).
  • Each patient received a 3-mg dose of bleomycin every other day.
  • Most patients (17, or 70%) were treated only with intracystic chemotherapy.
  • Chemotherapy was followed by surgery in 7 patients.
  • Five were operated on at the beginning of our study, and 2 required surgery because chemotherapy yielded poor results.
  • The follow-up period ranged from 2 years to 10 years.
  • CONCLUSION: Our results show that bleomycin can be an alternative in the treatment of cystic craniopharyngiomas or cystic recurrences, as it reduces surgical morbidity and improves clinical results.
  • [MeSH-major] Antibiotics, Antineoplastic / therapeutic use. Bleomycin / therapeutic use. Craniopharyngioma / drug therapy. Pituitary Neoplasms / drug therapy
  • [MeSH-minor] Adolescent. Child. Child, Preschool. Drug Administration Routes. Drug Administration Schedule. Female. Follow-Up Studies. Humans. Infant. Male. Middle Aged. Neoplasm Recurrence, Local. Retrospective Studies. Tomography, X-Ray Computed

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  • (PMID = 11862438.001).
  • [ISSN] 0256-7040
  • [Journal-full-title] Child's nervous system : ChNS : official journal of the International Society for Pediatric Neurosurgery
  • [ISO-abbreviation] Childs Nerv Syst
  • [Language] eng
  • [Publication-type] Clinical Trial; Journal Article
  • [Publication-country] Germany
  • [Chemical-registry-number] 0 / Antibiotics, Antineoplastic; 11056-06-7 / Bleomycin
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15. Moschovi M, Alexiou GA, Dastamani A, Stefanaki K, Prodromou N, Hatzigiorgi H, Karamolegou K, Tzortzatou-Stathopoulou F: Alpha-fetoprotein secretion in a craniopharyngioma. Are craniopharyngiomas part of the germ cell tumor family? Acta Neurol Belg; 2010 Sep;110(3):272-5
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  • [Title] Alpha-fetoprotein secretion in a craniopharyngioma. Are craniopharyngiomas part of the germ cell tumor family?
  • Brain CT and MRI revealed a suprasellar cystic and partially solid mass with calcifications.
  • These findings were suggestive for a brain germ cell tumor.
  • Therefore, systemic chemotherapy was started.
  • After two courses there was a reduction in the levels of AFP but the tumor size remained unchanged.
  • Subtotal tumor excision was performed that revealed the presence of a craniopharyngioma.
  • One month later there was enlargement of the cystic part of the tumor, while serum AFP was elevated.
  • The child received again systemic chemotherapy and placement of a reservoir into the cystic part of the tumor.
  • Analysis of the intracystic flouid revealed the presence of beta-HCG and AFP.
  • One year later the patient was stable but with complete loss of vision.
  • These observations support the theory of a germ cell tumor family, in which craniopharyngioma and germ cell tumor present the two sides of the same entity, while between them a wide variety of tumors, with variable type of secretion of AFP and/or beta-HCG, may exist.
  • [MeSH-major] Craniopharyngioma. Neoplasms, Germ Cell and Embryonal / classification. Pituitary Neoplasms. alpha-Fetoproteins / secretion
  • [MeSH-minor] Female. Humans. Infant. Tomography, X-Ray Computed

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  • (PMID = 21114138.001).
  • [ISSN] 0300-9009
  • [Journal-full-title] Acta neurologica Belgica
  • [ISO-abbreviation] Acta Neurol Belg
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] Belgium
  • [Chemical-registry-number] 0 / AFP protein, human; 0 / alpha-Fetoproteins
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16. Requejo F, Schumacher M, van Velthoven V: Coating the wall of an injured intracranial carotid artery during tumor removal with n-butyl-2-cyanoacrylate: technical case report. Neurosurgery; 2006 Oct;59(4 Suppl 2):ONSE484-5; discussion ONSE485
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  • [Title] Coating the wall of an injured intracranial carotid artery during tumor removal with n-butyl-2-cyanoacrylate: technical case report.
  • OBJECTIVE: Carotid artery injury close to the clinoid process is difficult to repair, and is even more so when the vessel is firmly attached to a calcified tumor.
  • We treated a patient with an intraoperative carotid lesion by coating the vessel wall with n-butyl-2-cyanoacrylate (NBCA).
  • CLINICAL PRESENTATION: A 7-year-old boy was referred to our clinic with a 3-month history of somnolence, apathy, and headache.
  • The cranial magnetic resonance imaging and computed tomographic scans showed a sellar and suprasellar calcified mass with heterogeneous contrast enhancement, a cyst component in the upper part of the tumor displaced upward and back from the mesencephalic and diencephalic structures.
  • Using a microsurgical technique, the suprasellar part of the craniopharyngioma was removed.
  • For this, 0.5 ml of NBCA was distributed on the surface of the injured segment and surrounding subarachnoid space by injection through a needle.
  • A digital cerebral angiogram obtained a few days after the procedure did not show vasospasm, stenosis, or pseudoaneurysm in the supraclinoidal segment of the carotid artery.
  • A magnetic resonance angiogram obtained 3 years later showed a normal shape of the internal carotid artery and a stable residual tumor without inflammatory signs.
  • The child is now attending school and is under hormonal therapy.
  • CONCLUSION: For hemostatic purposes, the technique of coating an injured arterial wall with NBCA may be useful in cases in which a microsuture is impossible and a permanent artery occlusion is unwanted because of a risk of an ischemic stroke.
  • [MeSH-major] Brain Neoplasms / surgery. Carotid Artery Injuries / drug therapy. Carotid Artery Injuries / etiology. Enbucrilate / analogs & derivatives. Neurosurgical Procedures / adverse effects. Tissue Adhesives / administration & dosage
  • [MeSH-minor] Child. Humans. Male. Treatment Outcome

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  • (PMID = 17041522.001).
  • [ISSN] 1524-4040
  • [Journal-full-title] Neurosurgery
  • [ISO-abbreviation] Neurosurgery
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article; Technical Report
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / Tissue Adhesives; F8CEP82QNP / Enbucrilate
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17. Birkholz D, Korpal-Szczyrska M, Kamińska H, Bień E, Połczyńska K, Stachowicz-Stencel T, Szołkiewicz A: [Influence of surgery and radiotherapy on growth and pubertal development in children treated for brain tumour]. Med Wieku Rozwoj; 2005 Jul-Sep;9(3 Pt 2):463-9
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  • [Title] [Influence of surgery and radiotherapy on growth and pubertal development in children treated for brain tumour].
  • INTRODUCTION: The increasing number of childhood cancer survivors has resulted in a growing interest in the late effects, which depend on type of treatment.
  • Frequently, a brain tumour and its therapy in children are endocrinologically devastating.
  • AIM OF STUDY: The aim of study was to compare growth and pubertal development in children after brain tumour therapy treated or not treated with recombinant growth hormone (rGH).
  • Group I - (12/18) not treated with rGH, after total resection of brain tumour: craniopharyngeoma (8/12), astrocytoma (2/12) ependymoma (1/12), germinoma (1/12).
  • Mean time of remission was 5,0yrs (+/- 0,9).
  • Group II - (6/12) treated with rGH, after subtotal resection of craniopharyngeoma (4/6), ependymoma (1/6), medulloblastoma (1/6) and cranial irradiation with mean total doses 46,5 Gy (+/- 5,65).
  • Children were qualified for rGH replacement according to deceleration of growth and lower growth hormone secretion (< 10 ng/ml) in stimulating tests.
  • Mean time of remission was 6,5 yrs (+/- 2,41).
  • Growth, height in centimeters converted to standard deviation score--SDS, body mass index (BMI), pubertal status and hormonal tests, were also evaluated.
  • 100% children of group I needed substitution because of secondary hypothyreosis, 83% due to secondary adrenal insufficiency and 53% of diabetes insipidus.
  • Mean height after brain tumour surgical treatment in group I was - 1,24 SDS (+/- 0,85) and did not significantly change in the time of observation.
  • Mean BMI after total resection of brain tumour was 18,09 (+/- 4,20) and significantly increased to 23,73 (+/- 2,82).
  • In group II - all children presented multihormonal pituitary insufficiency.
  • Mean deviation score of height before rGH treatment was - 3,84 SDS (+/- 2,87) and after mean time of rGH therapy of 1,5 yrs (+/- 1,2) decreased to 2,6 (+/- 1,06).
  • Mean BMI before treatment with rGH 18, 06 (+/- 4,4) increased to 22,41 (+ 0,74) in the time of observation and decreased to 18,5 (+/- 2,87) after 1,5 years (+/- 1,2) of rGH treatment.
  • Children treated with surgery for brain tumour need substitution for secondary hypothyroidism, part of then need treatment for secondary adrenal and gonadal insufficiency and diabetes incipidus.
  • 2. Children who were treated with surgery and/or cranial irradiation developed multihormonal pituitary insufficiency, growth failure and replacement rGh therapy was needed.
  • 3. Total resection of brain tumour without chemo- and radiotherapy did not impair growth in first years after surgery.
  • [MeSH-major] Brain Neoplasms / therapy. Growth Disorders / drug therapy. Growth Disorders / etiology. Human Growth Hormone / therapeutic use
  • [MeSH-minor] Adolescent. Astrocytoma / complications. Astrocytoma / therapy. Body Height / radiation effects. Child. Cranial Irradiation / adverse effects. Craniopharyngioma / complications. Craniopharyngioma / therapy. Ependymoma / complications. Ependymoma / therapy. Female. Germinoma / complications. Germinoma / therapy. Humans. Male. Puberty. Radiation Injuries / etiology. Treatment Outcome

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  • (PMID = 16719158.001).
  • [Journal-full-title] Medycyna wieku rozwojowego
  • [ISO-abbreviation] Med Wieku Rozwoj
  • [Language] pol
  • [Publication-type] Controlled Clinical Trial; English Abstract; Journal Article
  • [Publication-country] Poland
  • [Chemical-registry-number] 12629-01-5 / Human Growth Hormone
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18. Verhelst J, Kendall-Taylor P, Erfurth EM, Price DA, Geffner M, Koltowska-Häggström M, Jönsson PJ, Wilton P, Abs R: Baseline characteristics and response to 2 years of growth hormone (GH) replacement of hypopituitary patients with GH deficiency due to adult-onset craniopharyngioma in comparison with patients with nonfunctioning pituitary adenoma: data from KIMS (Pfizer International Metabolic Database). J Clin Endocrinol Metab; 2005 Aug;90(8):4636-43
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  • [Title] Baseline characteristics and response to 2 years of growth hormone (GH) replacement of hypopituitary patients with GH deficiency due to adult-onset craniopharyngioma in comparison with patients with nonfunctioning pituitary adenoma: data from KIMS (Pfizer International Metabolic Database).
  • Patients with hypopituitarism caused by a craniopharyngioma (CP) and/or its treatment have a higher mortality than patients with other etiologies, such as a nonfunctioning pituitary adenoma (NFPA).
  • PATIENTS: Baseline characteristics were studied in 351 CP patients (189 men and 162 women; mean age, 42.5 yr) and compared with 370 NFPA patients, matched for age and sex (185 men and 185 women; mean age, 42.5 yr).
  • RESULTS: At baseline, both CP and NFPA patients had characteristic features of GH deficiency, with low serum IGF-I, increased body fat, dyslipidemia, and reduced quality of life.
  • Male CP patients were significantly more obese (30.0 vs. 28.2 kg/m2; P = 0.0003) compared with NFPA patients, had a higher waist/hip ratio (P = 0.004), higher triglycerides (P = 0.003), and lower high-density lipoprotein cholesterol (P = 0.03).
  • The incidence of previous fractures, hypertension, coronary heart disease, claudication, and diabetes mellitus was high, but not different, between CP and NFPA patients.
  • After 2 yr of GH replacement therapy, similar significant improvements were evident in both groups in fat-free mass, total and low-density lipoprotein cholesterol, and Quality-of-Life-Assessment in GH Deficient Adults score compared with baseline.
  • In contrast to NFPA patients, CP patients had no significant decrease in body fat with GH therapy.
  • CONCLUSIONS: In the KIMS database, patients with CP have more often undergone surgery by the transcranial route than patients with NFPA, have a higher prevalence of pituitary deficiencies, are more obese (predominantly males), and have more dyslipidemia.
  • CP patients respond equally well to GH therapy in fat-free mass, lipids, and quality of life, but are less likely to lose body fat.
  • We assume that this difference in response merely reflects the stronger tendency of CP patients to accumulate fat over time.
  • [MeSH-major] Adenoma / complications. Craniopharyngioma / complications. Human Growth Hormone / therapeutic use. Hypopituitarism / drug therapy. Hypopituitarism / etiology. Pituitary Neoplasms / complications
  • [MeSH-minor] Adult. Age of Onset. Blood Glucose. Body Composition. Comorbidity. Databases, Factual. Fasting. Female. Hemoglobin A, Glycosylated / metabolism. Humans. Insulin-Like Growth Factor I / metabolism. Lipids / blood. Male. Middle Aged. Prevalence. Quality of Life. Treatment Outcome


19. Kobayashi K, Higashima M, Mutou K, Kidani T, Tachibana O, Yamashita J, Koshino Y: Severe delirium due to basal forebrain vascular lesion and efficacy of donepezil. Prog Neuropsychopharmacol Biol Psychiatry; 2004 Nov;28(7):1189-94
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  • A 68-year-old man had suffered for a month from delirium of mixed type caused by the right basal forebrain vascular lesion after surgery for craniopharyngioma.
  • Magnetic resonance imaging (MRI) showed hemorrhagic infarcts in the head of the right caudate nucleus and the right basal forebrain of the medial septal nucleus, diagonal band of Broca and nucleus basalis of Meynert.
  • Donepezil administration dramatically improved his intractable delirium at the 19th post-donepezil administration day, but this was followed by amnestic symptoms.
  • [MeSH-major] Basal Ganglia Cerebrovascular Disease / drug therapy. Delirium / drug therapy. Delirium / etiology. Indans / therapeutic use. Nootropic Agents / therapeutic use. Piperidines / therapeutic use. Prosencephalon / pathology
  • [MeSH-minor] Aged. Basal Nucleus of Meynert / pathology. Craniopharyngioma / complications. Craniopharyngioma / surgery. Humans. Magnetic Resonance Imaging. Male. Pituitary Neoplasms / complications. Pituitary Neoplasms / surgery. Postoperative Complications / drug therapy. Postoperative Complications / psychology. Sleep Wake Disorders / drug therapy. Sleep Wake Disorders / etiology. Vision Disorders / complications

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  • (PMID = 15610933.001).
  • [ISSN] 0278-5846
  • [Journal-full-title] Progress in neuro-psychopharmacology & biological psychiatry
  • [ISO-abbreviation] Prog. Neuropsychopharmacol. Biol. Psychiatry
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] England
  • [Chemical-registry-number] 0 / Indans; 0 / Nootropic Agents; 0 / Piperidines; 8SSC91326P / donepezil
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20. Jakacki RI, Cohen BH, Jamison C, Mathews VP, Arenson E, Longee DC, Hilden J, Cornelius A, Needle M, Heilman D, Boaz JC, Luerssen TG: Phase II evaluation of interferon-alpha-2a for progressive or recurrent craniopharyngiomas. J Neurosurg; 2000 Feb;92(2):255-60
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  • [Title] Phase II evaluation of interferon-alpha-2a for progressive or recurrent craniopharyngiomas.
  • OBJECT: Craniopharyngiomas originate from the same cells as squamous cell skin carcinoma, which can be treated successfully with interferon-alpha (IFNalpha)-2a.
  • The authors evaluated the activity and toxicity of systemic IFN in young patients with craniopharyngiomas.
  • METHODS: Fifteen patients between the ages of 4.2 and 19.8 years who had progressive or recurrent craniopharyngiomas were enrolled in this study.
  • Nine of these patients had never received external-beam radiation therapy.
  • Therapy consisted of 8,000,000 U/m2 IFNalpha-2a administered daily for 16 weeks (induction phase) followed by the same dose three times per week for an additional 32 weeks (maintenance phase).
  • Of the 12 patients who could be evaluated, radiological studies demonstrated a response to treatment in three with predominantly cystic tumors (one minor response, one partial response, and one complete response); one of these patients also showed improvement in visual fields.
  • The size of the cystic component of the tumors often increased temporarily during the first several months of therapy.
  • Three patients met the criteria for progressive disease during therapy.
  • The median time to progression was 25 months.
  • The need for radiation therapy in patients treated with IFN was delayed for 18 to 35 months (median 25 months) in six patients.
  • All patients developed transient flulike symptoms shortly after receiving the first dose of IFN.
  • Other toxicities (predominantly hepatic, neurological, and cutaneous) were seen in nine (60%) of the 15 patients during the first 8 weeks of treatment but resolved after temporary discontinuation and/or dose reduction.
  • CONCLUSIONS: Interferon-alpha-2a is active against some childhood craniopharyngiomas; its toxicity precludes administration of high daily doses, and the optimum dose level and schedule remain to be defined.
  • [MeSH-major] Craniopharyngioma / drug therapy. Interferon-alpha / administration & dosage. Neoplasm Recurrence, Local / drug therapy. Pituitary Neoplasms / drug therapy
  • [MeSH-minor] Adolescent. Adult. Child. Child, Preschool. Combined Modality Therapy. Cranial Irradiation. Disease Progression. Dose-Response Relationship, Drug. Drug Administration Schedule. Female. Humans. Injections, Subcutaneous. Magnetic Resonance Imaging. Male. Pituitary Gland / pathology. Radiotherapy, Adjuvant. Recombinant Proteins. Treatment Outcome

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  • (PMID = 10659012.001).
  • [ISSN] 0022-3085
  • [Journal-full-title] Journal of neurosurgery
  • [ISO-abbreviation] J. Neurosurg.
  • [Language] eng
  • [Publication-type] Case Reports; Clinical Trial; Clinical Trial, Phase II; Journal Article
  • [Publication-country] UNITED STATES
  • [Chemical-registry-number] 0 / Interferon-alpha; 0 / Recombinant Proteins; 76543-88-9 / interferon alfa-2a
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21. Pettorini BL, Inzitari R, Massimi L, Tamburrini G, Caldarelli M, Fanali C, Cabras T, Messana I, Castagnola M, Di Rocco C: The role of inflammation in the genesis of the cystic component of craniopharyngiomas. Childs Nerv Syst; 2010 Dec;26(12):1779-84
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  • [Title] The role of inflammation in the genesis of the cystic component of craniopharyngiomas.
  • BACKGROUND: Craniopharyngioma accounts for 5-10% of childhood tumors and, despite of the benign histological features, its clinical course can be malignant because of critical anatomical relationships with neural and vascular structures and the possible morbidity associated to resection.
  • Only a few studies have addressed the molecular characterization of the cyst fluid so far and the mechanisms of action of intracystic agents are not clearly understood yet.
  • METHODS: The acidic soluble proteins contained in the cystic fluid of six patients with cystic craniopharyngioma, three of them treated with intratumoral interferon-α, were analyzed.
  • A high performance liquid chromatography electrospray ionization mass spectrometry analysis was performed.
  • FINDINGS: The antimicrobial peptides α-defensins 1-3 relevant for innate immunity were detected in the cystic fluid before the intratumoral treatment.
  • Amount of peptides significantly decreased in cystic fluid during pharmacological treatment.
  • INTERPRETATION: Detection of α-defensins 1-3 excludes that cyst fluid formation can derive from disruption of blood-brain barrier and suggests the involvement of innate immune response in pathology of craniopharyngioma cyst formation.
  • The reduction of α-defensins could derive both from direct antitumoral effect of interferon-α on squamous epithelial cells of craniopharyngioma cyst and from its immuno-modulatory effects on the recruitment of cells of innate immune systems.
  • Additional studies will be necessary to establish the role of these molecules in the pathogenesis of craniopharyngioma, and further investigations will be necessary to confirm the efficacy of the antitumoral activity of interferon-α.
  • [MeSH-major] Craniopharyngioma / immunology. Cysts / immunology. Inflammation / immunology. Pituitary Neoplasms / immunology
  • [MeSH-minor] Child. Child, Preschool. Chromatography, High Pressure Liquid. Cyst Fluid / chemistry. Cyst Fluid / immunology. Female. Humans. Immunity, Innate / immunology. Immunologic Factors / administration & dosage. Injections, Intraventricular. Interferon-alpha / administration & dosage. Male. Spectrometry, Mass, Electrospray Ionization. alpha-Defensins / analysis. alpha-Defensins / immunology. alpha-Defensins / metabolism

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  • (PMID = 20668862.001).
  • [ISSN] 1433-0350
  • [Journal-full-title] Child's nervous system : ChNS : official journal of the International Society for Pediatric Neurosurgery
  • [ISO-abbreviation] Childs Nerv Syst
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] Germany
  • [Chemical-registry-number] 0 / Immunologic Factors; 0 / Interferon-alpha; 0 / alpha-Defensins
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22. Linnert M, Gehl J: Bleomycin treatment of brain tumors: an evaluation. Anticancer Drugs; 2009 Mar;20(3):157-64
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  • [Title] Bleomycin treatment of brain tumors: an evaluation.
  • Bleomycin has been used in the treatment of brain tumors for over 30 years.
  • Currently, we are evaluating electrochemotherapy (the use of electric pulses to enhance uptake of bleomycin) for patients with secondary brain tumors.
  • Using the keywords 'brain' and 'bleomycin', a database search without date restriction was performed and over 500 articles were found.
  • Twenty-five articles were used for this study based on relevance determined by: (i) clinical studies, (ii) use of bleomycin, and (iii) direct injection into brain tissue or cysts.
  • There were two main indications for the use of bleomycin directly into the brain: (i) cystic tumors in the form of craniopharyngiomas and (ii) solid brain tumors such as glioblastomas and astrocytomas.
  • One death was directly related to this treatment, where very high doses were used.
  • Two patients developed loss of vision and two patients had hearing loss because of the treatment.
  • All cases with severe and moderate adverse effects except one were patients with craniopharyngiomas and probably because of tumor localization in the deep brain.
  • In conclusion, bleomycin injection into the brain has been fairly well tolerated at doses much higher than that used in electrochemotherapy.
  • [MeSH-major] Antimetabolites, Antineoplastic / therapeutic use. Bleomycin / therapeutic use. Brain Neoplasms / drug therapy
  • [MeSH-minor] Adolescent. Adult. Aged. Blindness / chemically induced. Brain Edema / chemically induced. Child. Child, Preschool. Craniopharyngioma / drug therapy. DNA Damage. Deafness / chemically induced. Electroporation. Female. Glioma / drug therapy. Humans. Infant. Injections, Spinal. Male. Middle Aged. Pituitary Neoplasms / drug therapy. Young Adult

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  • (PMID = 19396014.001).
  • [ISSN] 1473-5741
  • [Journal-full-title] Anti-cancer drugs
  • [ISO-abbreviation] Anticancer Drugs
  • [Language] eng
  • [Publication-type] Journal Article; Review
  • [Publication-country] England
  • [Chemical-registry-number] 0 / Antimetabolites, Antineoplastic; 11056-06-7 / Bleomycin
  • [Number-of-references] 43
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23. Cavalheiro S, Di Rocco C, Valenzuela S, Dastoli PA, Tamburrini G, Massimi L, Nicacio JM, Faquini IV, Ierardi DF, Silva NS, Pettorini BL, Toledo SR: Craniopharyngiomas: intratumoral chemotherapy with interferon-alpha: a multicenter preliminary study with 60 cases. Neurosurg Focus; 2010 Apr;28(4):E12
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  • [Title] Craniopharyngiomas: intratumoral chemotherapy with interferon-alpha: a multicenter preliminary study with 60 cases.
  • OBJECT: The authors assessed the efficacy of intratumoral interferon-alpha (IFNalpha)-based chemotherapy in pediatric patients with cystic craniopharyngiomas.
  • METHODS: In a prospective multicenter study of 60 pediatric patients, the authors assessed the efficacy of intratumoral INFalpha2A-based chemotherapy.
  • RESULTS: Sixty cases of cystic craniopharyngioma were analyzed.
  • The cohort consisted of 35 male and 25 female children (mean age 11 years).
  • CONCLUSIONS: This has been the largest documented series of intratumoral chemotherapy using INFalpha for the control of cystic craniopharyngiomas.
  • The treatment has proved efficacious; there was no mortality, and morbidity rates were low.
  • [MeSH-major] Craniopharyngioma / drug therapy. Interferon-alpha / administration & dosage. Pituitary Neoplasms / drug therapy
  • [MeSH-minor] Child. Child, Preschool. Cohort Studies. Drug Administration Schedule. Female. Humans. Infant. Injections, Intralesional. Magnetic Resonance Imaging. Male. Neuronavigation. Prospective Studies. Treatment Outcome. Tumor Burden

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  • (PMID = 20367356.001).
  • [ISSN] 1092-0684
  • [Journal-full-title] Neurosurgical focus
  • [ISO-abbreviation] Neurosurg Focus
  • [Language] eng
  • [Publication-type] Clinical Trial; Comparative Study; Journal Article; Multicenter Study
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / Interferon-alpha
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24. Kitahara M, Ishikawa S, Hara K, Kanno S, Ogata F, Naruo K, Yaguchi A: [Changes in peripheral lymphocytes count in unconscious patients treated at home with Japanese herbal medicines (Hozai)]. Gan To Kagaku Ryoho; 2002 Dec;29 Suppl 3:526-9
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  • We investigated changes in peripheral white blood cells count, lymphocytes count, and serum albumin level in five unconscious patients administered Japanese herbal medicines (Hozai).
  • The causes of unconsciousness were intracerebral hemorrhage in 3, cerebral infarction in 1, and craniopharyngioma in 1.
  • To assess the effect of Hozai on the immune system of unconscious patients, we investigated these parameters before and after treatment.
  • We observed normalization of WBC counts, increase of lymphocytes count, and unchanged levels of serum albumin after Hozai treatment.
  • One of the problems affecting unconscious patients treated at home is infectious diseases, especially respiratory infections.
  • These results suggest that Hozai treatment might be useful adjuvants to support the general condition of the patients treated at home.
  • [MeSH-major] Consciousness Disorders / immunology. Drugs, Chinese Herbal / therapeutic use. Medicine, Kampo. Phytotherapy. Serum Albumin / metabolism
  • [MeSH-minor] Aged. Cerebral Hemorrhage / complications. Cerebral Hemorrhage / drug therapy. Female. Humans. Leukocyte Count. Lymphocyte Count. Male. Middle Aged

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  • (PMID = 12536844.001).
  • [ISSN] 0385-0684
  • [Journal-full-title] Gan to kagaku ryoho. Cancer & chemotherapy
  • [ISO-abbreviation] Gan To Kagaku Ryoho
  • [Language] jpn
  • [Publication-type] English Abstract; Journal Article
  • [Publication-country] Japan
  • [Chemical-registry-number] 0 / Drugs, Chinese Herbal; 0 / Serum Albumin
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25. Trivin C, Busiah K, Mahlaoui N, Recasens C, Souberbielle JC, Zerah M, Sainte-Rose C, Brauner R: Childhood craniopharyngioma: greater hypothalamic involvement before surgery is associated with higher homeostasis model insulin resistance index. BMC Pediatr; 2009;9:24
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  • [Title] Childhood craniopharyngioma: greater hypothalamic involvement before surgery is associated with higher homeostasis model insulin resistance index.
  • BACKGROUND: Obesity seems to be linked to the hypothalamic involvement in craniopharyngioma.
  • We evaluated the pre-surgery relationship between the degree of this involvement on magnetic resonance imaging and insulin resistance, as evaluated by the homeostasis model insulin resistance index (HOMA).
  • METHODS: 27 children with craniopharyngioma were classified as either grade 0 (n = 7, no hypothalamic involvement), grade 1 (n = 8, compression without involvement), or grade 2 (n = 12, severe involvement).
  • RESULTS: Despite having similar body mass indexes (BMI), the grade 2 patients had higher glucose, insulin and HOMA before surgery than the grade 0 (P = 0.02, <0.05 and 0.02 respectively) and 1 patients (P < 0.02 and <0.03 for both insulin and HOMA).
  • The grade 0 (5.8 +/- 4.9) and 1 (7.2 +/- 5.3) patients gained significantly less weight (kg) during the year after surgery than did the grade 2 (16.3 +/- 7.4) patients.
  • The pre-surgery HOMA was positively correlated with these weight changes (P < 0.03).
  • The data for the whole population before and 6-18 months after surgery showed increases in BMI (P < 0.0001), insulin (P < 0.005), and leptin (P = 0.0005), and decreases in sOB-R (P < 0.04) and ghrelin (P < 0.03).
  • CONCLUSION: The hypothalamic involvement by the craniopharyngioma before surgery seems to determine the degree of insulin resistance, regardless of the BMI.
  • This suggests that obesity should be prevented by reducing inn secretion in those cases with hypothalamic involvement.
  • [MeSH-major] Craniopharyngioma / pathology. Hypothalamus / pathology. Insulin Resistance. Obesity / etiology. Pituitary Neoplasms / pathology
  • [MeSH-minor] Adolescent. Blood Glucose / analysis. Child. Child, Preschool. Female. Ghrelin / blood. Homeostasis. Hormone Replacement Therapy. Humans. Hydrocortisone / blood. Hypophysectomy. Hypopituitarism / drug therapy. Hypopituitarism / etiology. Insulin-Like Growth Factor I / analysis. Leptin / blood. Male. Models, Biological. Receptors, Leptin / blood. Retrospective Studies. Single-Blind Method. Thyroxine / blood. Weight Gain

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  • (PMID = 19341477.001).
  • [ISSN] 1471-2431
  • [Journal-full-title] BMC pediatrics
  • [ISO-abbreviation] BMC Pediatr
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] England
  • [Chemical-registry-number] 0 / Blood Glucose; 0 / Ghrelin; 0 / Leptin; 0 / Receptors, Leptin; 67763-96-6 / Insulin-Like Growth Factor I; Q51BO43MG4 / Thyroxine; WI4X0X7BPJ / Hydrocortisone
  • [Other-IDs] NLM/ PMC2675525
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26. Fraioli MF, Moschettoni L, Catena E, Fraioli C: Cystic craniopharyngioma: trans-sphenoidal surgery and intra-cystic apposition of "bleomycin wax". Acta Neurochir (Wien); 2010 Feb;152(2):293-6
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  • [Title] Cystic craniopharyngioma: trans-sphenoidal surgery and intra-cystic apposition of "bleomycin wax".
  • BACKGROUND: The current therapeutic approach to craniopharyngioma is multidisciplinary.
  • Sub-total removal, followed by adjuvant treatments, especially in large cystic tumours, is an accepted regime reported by many authors.
  • CASE REPORT: A young patient with an intra- and suprasellar cystic craniopharyngioma was operated on via a microsurgical trans-sphenoidal approach, achieving sub-total removal and bleomycin mixed with bone wax ("bleomycin wax") applied to the capsular remnant.
  • There was no evidence of tumour recurrence after a follow-up period of 5.4 years.
  • CONCLUSIONS: The intra-operative use of "bleomycin-wax" should be limited to those patients in whom intra-operative CSF fistula does not occur.
  • [MeSH-major] Bleomycin / administration & dosage. Craniopharyngioma / drug therapy. Craniopharyngioma / surgery. Pituitary Neoplasms / drug therapy. Pituitary Neoplasms / surgery. Sphenoid Bone / surgery
  • [MeSH-minor] Antibiotics, Antineoplastic / administration & dosage. Child. Cysts / drug therapy. Cysts / pathology. Diabetes Insipidus / drug therapy. Diabetes Insipidus / etiology. Drug Combinations. Epithelial Cells / pathology. Humans. Magnetic Resonance Imaging. Male. Nasal Cavity / anatomy & histology. Nasal Cavity / surgery. Neurosurgical Procedures. Optic Chiasm / pathology. Optic Chiasm / surgery. Palmitates / administration & dosage. Pituitary Gland / pathology. Pituitary Gland / surgery. Postoperative Complications / drug therapy. Postoperative Complications / etiology. Sella Turcica / pathology. Sella Turcica / surgery. Treatment Outcome. Waxes

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  • (PMID = 19390776.001).
  • [ISSN] 0942-0940
  • [Journal-full-title] Acta neurochirurgica
  • [ISO-abbreviation] Acta Neurochir (Wien)
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] Austria
  • [Chemical-registry-number] 0 / Antibiotics, Antineoplastic; 0 / Drug Combinations; 0 / Palmitates; 0 / Waxes; 11056-06-7 / Bleomycin; 8021-48-5 / bone wax
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27. Chanson JB, Anheim M, Lagha-Boukbiza O, Fleury M, Sellal F, Tranchant C: [Severe generalized dystonia due to postradiotherapy cerebral calcifications]. Rev Neurol (Paris); 2008 May;164(5):477-80
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  • [Transliterated title] Dystonie généralisée sévère liée à des calcifications cérébrales postradiques.
  • We report a very severe case of generalized dystonia due to postradiotherapy basal ganglia calcifications.
  • CASE REPORT: An 8-year-old girl received 53 grays radiotherapy after surgery for craniopharyngioma.
  • One year later she developed generalized dystonia.
  • Computed tomography showed bilateral basal ganglia calcifications, especially of the lenticular nuclei.
  • Pharmacological treatment with tetrabenazine, clonazepam and trihexiphenydile allowed a very limited improvement of dystonia; the course was complicated by dystonic storms and decompensations resulting from the iatrogenous panhypopituitarism.
  • CONCLUSION: This case illustrates a severe complication of cranial irradiation which should be considered in the indications of this treatment, especially for children.
  • [MeSH-major] Anti-Dyskinesia Agents / therapeutic use. Calcinosis / complications. Calcinosis / etiology. Dystonia / drug therapy. Dystonia / etiology. Radiotherapy / adverse effects
  • [MeSH-minor] Basal Ganglia / pathology. Brain Neoplasms / complications. Brain Neoplasms / radiotherapy. Child. Clonazepam / therapeutic use. Craniopharyngioma / complications. Craniopharyngioma / radiotherapy. Female. GABA Modulators / therapeutic use. Humans. Hypopituitarism / drug therapy. Hypopituitarism / etiology. Tetrabenazine / therapeutic use. Tomography, X-Ray Computed. Trihexyphenidyl / therapeutic use

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  • (PMID = 18555882.001).
  • [ISSN] 0035-3787
  • [Journal-full-title] Revue neurologique
  • [ISO-abbreviation] Rev. Neurol. (Paris)
  • [Language] fre
  • [Publication-type] Case Reports; English Abstract; Journal Article
  • [Publication-country] France
  • [Chemical-registry-number] 0 / Anti-Dyskinesia Agents; 0 / GABA Modulators; 5PE9FDE8GB / Clonazepam; 6RC5V8B7PO / Trihexyphenidyl; Z9O08YRN8O / Tetrabenazine
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28. Hilczer M, Smyczynska J, Stawerska R, Lewinski A: Final height and growth hormone secretion after completion of growth hormone therapy in patients with idiopathic growth hormone deficiency and with abnormalities of the hypothalamic-pituitary region. Neuro Endocrinol Lett; 2005 Feb;26(1):19-24
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  • [Title] Final height and growth hormone secretion after completion of growth hormone therapy in patients with idiopathic growth hormone deficiency and with abnormalities of the hypothalamic-pituitary region.
  • AIMS: The aim of the study was an evaluation of final height and growth hormone (GH) secretion after completion of GH therapy (retesting) in patients with GH deficiency (GHD).
  • PATIENTS AND METHODS: The analysis comprised 53 patients (43 boys, 10 girls) with childhood-onset GHD, who completed GH therapy and reached final height.
  • Magnetic resonance imaging (MRI), performed in all the patients, led to the following groups: pituitary stalk interruption syndrome (PSIS), pituitary hypoplasia (HP), craniopharyngioma (CP) -- patients after tumour excision, patients with normal hypothalamic-pituitary region (NP).
  • In retesting, GH secretion was significantly (p<0.005) lower in PSIS and CP than in HP and in NP and also (p<0.05) in HP than in NP.
  • Permanent severe GHD was confirmed in all the patients with PSIS and CP and in some patients with HP (37.5%), while it was excluded in all the patients with normal pituitary in MRI.
  • CONCLUSIONS: It seems that in patients with PSIS and CP, the confirmation of persistent character of GHD needs no retesting, while in patients with normal MRI results, GHD diagnosis should be established with special attention.
  • [MeSH-major] Body Height / drug effects. Growth Hormone / therapeutic use. Human Growth Hormone / blood. Human Growth Hormone / deficiency. Hypothalamo-Hypophyseal System / abnormalities
  • [MeSH-minor] Adolescent. Age of Onset. Child. Craniopharyngioma / surgery. Female. Humans. Magnetic Resonance Imaging. Male. Pituitary Diseases / blood. Pituitary Diseases / drug therapy. Pituitary Diseases / surgery. Pituitary Gland / abnormalities. Pituitary Neoplasms / surgery

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  • (PMID = 15726014.001).
  • [ISSN] 0172-780X
  • [Journal-full-title] Neuro endocrinology letters
  • [ISO-abbreviation] Neuro Endocrinol. Lett.
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] Sweden
  • [Chemical-registry-number] 12629-01-5 / Human Growth Hormone; 9002-72-6 / Growth Hormone
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29. Marcus KJ, Goumnerova L, Billett AL, Lavally B, Scott RM, Bishop K, Xu R, Young Poussaint T, Kieran M, Kooy H, Pomeroy SL, Tarbell NJ: Stereotactic radiotherapy for localized low-grade gliomas in children: final results of a prospective trial. Int J Radiat Oncol Biol Phys; 2005 Feb 1;61(2):374-9
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  • [Title] Stereotactic radiotherapy for localized low-grade gliomas in children: final results of a prospective trial.
  • PURPOSE: To evaluate the efficacy of stereotactic radiotherapy (SRT) for small, localized, pediatric brain tumors and to determine the patterns of failure.
  • Of the 81 patients, 50 had low-grade astrocytoma, 23 had residual or recurrent craniopharyngioma, 4 had posterior fossa ependymoma, and 4 had other histologic types.
  • All patients underwent biopsy for diagnosis, with the exception of patients with neurofibromatosis and radiographic evidence of an optic system tumor.
  • This report focused on the patients with low-grade gliomas only.
  • Of the 50 patients, 26 were males and 24 females; the median age was 9 years (range, 2-26 years).
  • The indications for treatment of patients with low-grade gliomas were progression during or after chemotherapy or progression after surgery alone.
  • Immobilization was accomplished with a removable head-frame.
  • CT and MRI fusion was used for treatment planning.
  • The target volume generally included the preoperative tumor plus a 2-mm margin for the planning target volume.
  • Three to nine arcs were used to deliver a mean total dose of 52.2 Gy in 1.8-Gy daily fractions.
  • RESULTS: With a median follow-up of 6.9 years (range, 0.9-10.2 years), the progression-free survival rate was 82.5% at 5 years and 65% at 8 years.
  • Five patients, all with optic system/hypothalamic primary tumors, developed central nervous system dissemination 1.0-7.4 years after SRT.
  • One patient developed a presumed radiation-induced primitive neuroectodermal tumor 6 years after initial treatment.
  • Six patients died, three of dissemination, two of progression to higher grade tumors, and one of a secondary radiation-induced tumor.
  • All 6 cases of local progression were within the primary tumor bed at the time of progression and had received the full prescription dose.
  • CONCLUSION: Stereotactic radiotherapy provides excellent local control for children with small, localized low-grade glial tumors.
  • Marginal failures have not been observed, supporting the use of limited margins to minimize late sequelae using stereotactic immobilization and planning techniques.
  • [MeSH-major] Astrocytoma / surgery. Brain Neoplasms / surgery. Radiosurgery
  • [MeSH-minor] Adolescent. Adult. Child. Child, Preschool. Confidence Intervals. Disease Progression. Disease-Free Survival. Female. Humans. Male. Neoplasm Recurrence, Local / drug therapy. Prospective Studies. Radiotherapy Dosage

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  • (PMID = 15667955.001).
  • [ISSN] 0360-3016
  • [Journal-full-title] International journal of radiation oncology, biology, physics
  • [ISO-abbreviation] Int. J. Radiat. Oncol. Biol. Phys.
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] United States
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30. Bommakanti K, Panigrahi M, Yarlagadda R, Sundaram C, Uppin MS, Purohit AK: Optic chiasmatic-hypothalamic gliomas: is tissue diagnosis essential? Neurol India; 2010 Nov-Dec;58(6):833-40

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  • [Title] Optic chiasmatic-hypothalamic gliomas: is tissue diagnosis essential?
  • The advocated treatment is mainly primary radiotherapy without a histological diagnosis.
  • Hence primary radiotherapy without histological confirmation may have deleterious consequences.
  • AIM: The aim of the paper was to analyze the sensitivity and specificity of magnetic resonance imaging (MRI) in these lesions and to analyze the feasibility of primary radiotherapy.
  • They were grouped into three groups on the basis of radiological features and treated with a suspected diagnosis.
  • The final diagnosis was correlated with preoperative diagnosis, and the feasibility of managing these lesions without a histopathological confirmation is discussed.
  • RESULTS: The three radiological groups were: Group-1 solid tumors with or without microcysts in 9 patients (histology: 8 pilocystic astrocytomas and 1 tuberculoma); Group-2 mixed tumors with solid and cystic components in 9 patients (histology: 7 pilocytic astrocytomas and 2 craniopharyngiomas); Group-3 ring enhancing lesions in 6 patients (all the 6 patients initially received antituberculous treatment, in 3 patients the lesion resolved and in the remaining 3 patients the lesion was subjected to biopsy as it did not resolve, the biopsy was suggestive of pilocytic astrocytoma).
  • Thus, MRI was shown to have a sensitivity of 83.33% and a specificity of 50% for diagnosing optic chiasmatic-hypothalamic gliomas.
  • CONCLUSIONS: Various lesions like craniopharyngiomas, tuberculomas can mimic optic chiasmatic-hypothalamic gliomas radiologically, and it is not possible to diagnose them with certainty on the basis of radiological findings alone.
  • Biopsy and tissue diagnosis should always be sought before instituting radiotherapy or chemotherapy for optic chiasmatic-hypothalamic gliomas.
  • [MeSH-major] Glioma / diagnosis. Hypothalamic Neoplasms / diagnosis. Optic Chiasm / pathology. Optic Nerve Neoplasms / diagnosis
  • [MeSH-minor] Adolescent. Biopsy / methods. Child. Child, Preschool. Contrast Media. Female. Humans. Magnetic Resonance Imaging / methods. Male. Young Adult

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  • [CommentIn] Neurol India. 2011 Jan-Feb;59(1):144 [21339694.001]
  • (PMID = 21150045.001).
  • [ISSN] 0028-3886
  • [Journal-full-title] Neurology India
  • [ISO-abbreviation] Neurol India
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] India
  • [Chemical-registry-number] 0 / Contrast Media
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31. Serowka K, Chiu Y, Gonzalez I, Gilles F, McComb G, Krieger M, Dhall G, Britt B, Ji L, Sposto R, Finlay JL: Central nervous system (CNS) tumors in the first six months of life: the Children's Hospital Los Angeles experience, 1979-2005. Pediatr Hematol Oncol; 2010 Mar;27(2):90-102
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  • [Title] Central nervous system (CNS) tumors in the first six months of life: the Children's Hospital Los Angeles experience, 1979-2005.
  • BACKGROUND: The authors report the experience at the Children's Hospital Los Angeles with brain tumors diagnosed before 6 months of age, describing the characteristics of the patients, their tumors, treatment strategies, and prognostic factors.
  • METHODS: Thirty-three children who were identified between 1979 and 2005 were included.
  • There were 11 gliomas, 9 choroid plexus tumors, 8 medulloblastomas and supratentorial primitive neuroectodermal tumors (PNET), 2 atypical teratoid/rhabdoid tumors (ATRT), and 1 each of ependymoma, craniopharyngioma, and immature teratoma.
  • Locations of primary tumors included 21 supratentorial (64%) and 7 posterior fossa, and 5 tumors involved both compartments.
  • The treatment strategies included 5 patients with biopsy only, 18 less than gross total resections (<GTRx), and 9 GTRx.
  • Fourteen children (42%) received chemotherapy.
  • Three patients (9%) received irradiation, 1 at initial diagnosis and 2 at relapse.
  • Nine patients (27%) demonstrated metastases, 6 at diagnosis and 3 at relapse.
  • RESULTS: The Kaplan Meier analysis of event-free survival (EFS) and overall survival (OS) for all patients is 21 +/- 9% and 35 +/- 9% at 5 years.
  • The 5-year OS for children achieving a GTRx is 64 +/- 21% and for those with <GTRx is 27 +/- 10% (p = .08).
  • [MeSH-major] Brain Neoplasms
  • [MeSH-minor] Female. Hospitals, Pediatric. Humans. Infant. Infant, Newborn. Los Angeles / epidemiology. Male. Prognosis. Survival Rate. Treatment Outcome

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  • (PMID = 20201690.001).
  • [ISSN] 1521-0669
  • [Journal-full-title] Pediatric hematology and oncology
  • [ISO-abbreviation] Pediatr Hematol Oncol
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] England
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32. Pancucci G, Massimi L, Caldarelli M, D'Angelo L, Sturiale C, Tamburrini G, Tufo T, Di Rocco C: [Pediatric craniopharyngioma: long-term results in 61 cases]. Minerva Pediatr; 2007 Jun;59(3):219-31
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  • [Title] [Pediatric craniopharyngioma: long-term results in 61 cases].
  • [Transliterated title] Craniofaringioma pediatrico: risultati a lungo termine in 61 casi.
  • AIM: The aim of this study was to analyze the long-term results of the surgical management of craniopharyngioma in children by reviewing a series of patients consecutively treated in a single institution, and to assess the efficacy of intratumoral chemotherapy with interferon-alpha.
  • METHODS: Sixty-one paediatric patients (38 males and 23 females; mean age: 8 years) have been surgically treated in the last 20 years.
  • The goal of surgery was to remove the tumour as much as possible.
  • RESULTS: All the 55 surviving patients enjoy a normal social life, except for 3 of them (visual and/or neurological deficits); endocrine function, compromised in 3/5 of cases, is managed by chronic hormone replacement; neuropsychological assessment is satisfactory in almost all the cases.
  • Although obesity does not seem to be an important social limit, it represents a real management problem.
  • Interferon-a was useful in transitorily arresting the growing cystic craniopharyngiomas.
  • CONCLUSION: The current experience confirms the still remarkable challenges in the treatment of craniopharyngioma in childhood.
  • Surgery should aim not only at removing the tumour, but also at preserving the late neuro-endocrinological functions.
  • Intracystic chemotherapy with interferon-alpha might represent an effective option to postpone the surgical operation until the maturation of the hypothalamic-hypophyseal pathway is completed.
  • However, it can not replace the traditional surgical management.
  • [MeSH-major] Brain / pathology. Craniopharyngioma / therapy. Hypophysectomy. Pituitary Neoplasms / therapy
  • [MeSH-minor] Adolescent. Antineoplastic Agents / therapeutic use. Chemotherapy, Adjuvant. Child. Child, Preschool. Female. Follow-Up Studies. Humans. Infant. Injections, Intralesional. Interferon-alpha / therapeutic use. Magnetic Resonance Imaging. Male. Radiotherapy, Adjuvant. Recombinant Proteins. Retrospective Studies. Treatment Outcome

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  • (PMID = 17519867.001).
  • [ISSN] 0026-4946
  • [Journal-full-title] Minerva pediatrica
  • [ISO-abbreviation] Minerva Pediatr.
  • [Language] ita
  • [Publication-type] English Abstract; Journal Article
  • [Publication-country] Italy
  • [Chemical-registry-number] 0 / Antineoplastic Agents; 0 / Interferon-alpha; 0 / Recombinant Proteins; 76543-88-9 / interferon alfa-2a
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33. Giavoli C, Ferrante E, Bergamaschi S, Ronchi CL, Lania AG, Spada A, Beck-Peccoz P: An unusual case of recurrent autoimmune hypophysitis. Exp Clin Endocrinol Diabetes; 2010 May;118(5):287-90
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  • [Title] An unusual case of recurrent autoimmune hypophysitis.
  • Autoimmune hypophysitis (AH) is an inflammatory disease that can present either as empty sella or as pituitary mass.
  • A 16-years-old girl was admitted at our Unit for primary amenorrhea.
  • A pituitary MRI performed 2 years before for severe headache demonstrated a large sellar and suprasellar lesion.
  • As a craniopharyngioma was suspected, the consultant neurosurgeon suggested the removal of the lesion.
  • Hormonal replacement therapy was started, obtaining a good clinical and biochemical control.
  • Four years later, severe headache and a MRI suggestive of pituitary adenoma recurred.
  • A MRI performed 3 months later did not show any pituitary lesion and empty sella was again described.
  • This patient represents one of the few reported cases of recurrent hypophysitis and demonstrates that both pituitary enlargement and empty-sella can be seen in the same patient at different times of his history.
  • [MeSH-major] Pituitary Diseases / radiography
  • [MeSH-minor] Adolescent. Adrenal Cortex Hormones / therapeutic use. Amenorrhea / etiology. Autoimmune Diseases / pathology. Empty Sella Syndrome / pathology. Female. Human Growth Hormone / therapeutic use. Humans. Hypogonadism / drug therapy. Hypopituitarism / pathology. Pituitary Neoplasms / pathology. Tomography, X-Ray Computed

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  • (PMID = 19691013.001).
  • [ISSN] 1439-3646
  • [Journal-full-title] Experimental and clinical endocrinology & diabetes : official journal, German Society of Endocrinology [and] German Diabetes Association
  • [ISO-abbreviation] Exp. Clin. Endocrinol. Diabetes
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article; Research Support, Non-U.S. Gov't
  • [Publication-country] Germany
  • [Chemical-registry-number] 0 / Adrenal Cortex Hormones; 12629-01-5 / Human Growth Hormone
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34. Faienza MF, Delvecchio M, Indrio F, Francavilla R, Acquafredda A, Cavallo L: Acute pancreatitis in a girl with panhypopituitarism due to craniopharyngioma on growth hormone treatment. A combination of risk factors. Horm Res; 2009;71(6):372-5
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  • [Title] Acute pancreatitis in a girl with panhypopituitarism due to craniopharyngioma on growth hormone treatment. A combination of risk factors.
  • Craniopharyngioma is a rare, benign, suprasellar brain tumor associated with a significant number of endocrine and metabolic impairments.
  • Growth hormone deficiency, caused by the tumor itself or by its subsequent surgical treatment, is the most common hormone deficiency in these patients and replacement is frequently necessary.
  • Hypothalamic obesity observed after surgery treatment, whether combined with radiotherapy or not, presents with increased abdominal fat and altered lipid profiles and is likely caused by both disruption of the mechanisms controlling satiety, hunger and energy balance and impairment of sensitivity to leptin, insulin and ghrelin axis.
  • It is well known that hyperlipemia is associated with acute pancreatitis, both as a precipitant and as an epiphenomenon.
  • Moreover, the increased incidence of acute pancreatitis during growth hormone therapy is possibly due to increased enzyme production (e.g., amylase, lipase and elastase).
  • We report the case of a 13-year-old girl affected by craniopharyngioma on growth hormone replacement treatment who developed acute pancreatitis.
  • We suggest including routine evaluation of lipid profile during follow-up of all children on growth hormone treatment, especially those affected by hypopituitarism secondary to craniopharyngioma, given pancreatic adverse effects of growth hormone replacement therapy and associated metabolic impairment due to hypothalamic obesity.
  • [MeSH-major] Craniopharyngioma / drug therapy. Hormone Replacement Therapy. Human Growth Hormone / adverse effects. Hypopituitarism / drug therapy. Pancreatitis / chemically induced. Recombinant Proteins / adverse effects
  • [MeSH-minor] Adolescent. Amylases / blood. Fasting / blood. Female. Humans. Lipase / blood. Lipids / blood


35. Moll GW, Bock HG: Two tumors detected by thyroid assessment in two children. Endocr Pract; 2001 Nov-Dec;7(6):467-73
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  • [Title] Two tumors detected by thyroid assessment in two children.
  • OBJECTIVE: To describe the early detection of two tumors in two children by recognition of unusual features in initial thyroid assessments.
  • In addition, we describe RET proto-oncogene studies in one of them.
  • RESULTS: A 14.5-year-old boy was referred for assessment because of short stature in conjunction with lack of physical growth and development.
  • His physical examination was remarkable for height at the 50th percentile (height age, 11.5 years), weight at the 50th percentile (weight age, 13 years), and prepubertal male status.
  • A parasellar tumor was detected and removed; histopathologic examination revealed that it was a craniopharyngioma.
  • The patient requires lifelong multihormonal therapy for his panhypopituitarism and has responded with physical growth.
  • Our second patient, a 7.5-year-old girl, was referred because of a painless left thyroid nodule of 4 months' duration.
  • Her physical examination was remarkable for (1) upper lip swelling, (2) intermittent rash, and (3) a goiter with painless mobile left and right nodules.
  • Genetic studies showed that she was positive for the RET multiple endocrine neoplasia IIB mutation.
  • CONCLUSION: Attention to thyroid physical findings and laboratory studies can promptly lead to correct diagnoses and management of some rare and life-threatening tumors in children.
  • [MeSH-major] Carcinoma, Medullary / diagnosis. Craniopharyngioma / diagnosis. Thyroid Neoplasms / diagnosis
  • [MeSH-minor] Adolescent. Child. Female. Humans. Hypopituitarism / drug therapy. Male. Multiple Endocrine Neoplasia / genetics. Puberty, Delayed / etiology. Thyroidectomy. Thyrotropin / blood. Thyroxine / blood

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  • (PMID = 11747285.001).
  • [ISSN] 1530-891X
  • [Journal-full-title] Endocrine practice : official journal of the American College of Endocrinology and the American Association of Clinical Endocrinologists
  • [ISO-abbreviation] Endocr Pract
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] United States
  • [Chemical-registry-number] 9002-71-5 / Thyrotropin; Q51BO43MG4 / Thyroxine
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36. Saito N, Aoki K, Sakurai T, Ito K, Hayashi M, Hirata Y, Sato K, Harashina J, Akahata M, Iwabuchi S: Linezolid treatment for intracranial abscesses caused by methicillin-resistant Staphylococcus aureus--two case reports. Neurol Med Chir (Tokyo); 2010;50(6):515-7
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  • [Title] Linezolid treatment for intracranial abscesses caused by methicillin-resistant Staphylococcus aureus--two case reports.
  • Two patients were treated for intracranial infections involving methicillin-resistant Staphylococcus aureus (MRSA).
  • After decompressive craniectomy, the patient developed an epidural abscess.
  • The infection did not improve after intravenous vancomycin (VCM) administration for 15 days.
  • However, after administration of linezolid (LZD) for 14 days, the infection had improved, and the white blood cell count and C-reactive protein values had normalized.
  • A 53-year-old woman had previously undergone 3 operations for craniopharyngioma before the age of 35 years.
  • Magnetic resonance imaging with contrast medium revealed a brain abscess caused by MRSA.
  • After 14 days of intravenous administration of VCM, the infection had not improved and intravenous administration of LZD was initiated.
  • After administration of LZD for 14 days intravenously and 14 days orally, the infection had improved, and the white blood cell count and C-reactive protein values had normalized.
  • LZD has good CNS penetration, so should be considered for secondline antibiotic therapy for VCM-resistant intracranial MRSA infection.
  • [MeSH-major] Acetamides / administration & dosage. Brain Abscess / drug therapy. Brain Abscess / microbiology. Epidural Abscess / drug therapy. Epidural Abscess / microbiology. Methicillin-Resistant Staphylococcus aureus / drug effects. Oxazolidinones / administration & dosage. Staphylococcal Infections / drug therapy

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  • (PMID = 20587984.001).
  • [ISSN] 1349-8029
  • [Journal-full-title] Neurologia medico-chirurgica
  • [ISO-abbreviation] Neurol. Med. Chir. (Tokyo)
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] Japan
  • [Chemical-registry-number] 0 / Acetamides; 0 / Oxazolidinones; 0 / Protein Synthesis Inhibitors; ISQ9I6J12J / Linezolid
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37. Okońska M, Birkholz D, Korpal-Szczyrska M, Adamkiewicz-Drozyńska E, Alska A, Magnuszewska H: [The evaluation of the influence of growth hormone therapy on growing process and metabolic functions in patients after treatment of craniopharyngioma]. Pediatr Endocrinol Diabetes Metab; 2010;16(1):19-24
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  • [Title] [The evaluation of the influence of growth hormone therapy on growing process and metabolic functions in patients after treatment of craniopharyngioma].
  • [Transliterated title] Ocena wpływu terapii hormonem wzrostu na przebieg wzrastania i funkcje metaboliczne u pacjentów po leczeniu craniopharyngioma.
  • INTRODUCTION: Craniopharyngioma (CP) is a tumor, which damages pituitary function because of its localization.
  • The growth hormone therapy (rGH) profits in the increase of growth rate and also may have metabolic effects like body weight reduction.
  • AIM OF THE STUDY: The evaluation of benefits from rGH therapy in patients cured from CP.
  • MATERIAL AND METHODS: 12 patients (7 boys and 5 girls) treated for CP with surgery; 3 of them also underwent radiotherapy.
  • The mean age at examination time was 11.7 yrs; remission time 2.96 yrs; rGH therapy started on average 3.69 yrs after the surgery.
  • Height (hSDS), weight, BMI were measured after the surgery, before and after 1 yr of rGH treatment.
  • Height velocity (HV) was evaluated before and after 1 yr of rGH therapy.
  • Pituitary GH-function was assessed.
  • Cholesterol, LDL, HDL, triglycerides and HbA1c were estimated before and after one year of rGH therapy.
  • HSDS after oncological treatment (OT) average -1.66 SD and decreased significantly until rGH therapy; weight after OT average 28.45 kg and until rGH therapy increased significantly; BMI after OT average 19.26 and increased significantly until rGH therapy as well.
  • After one year of rGH therapy hSDS and HV increased significantly; they average -1.65SD and 10.21 cm/yr respectively.
  • During rGH therapy neither tumor recurrence nor severe side effects were observed.
  • CONCLUSIONS: rGH therapy of patients cured from CP influences profitably not only growth rate, but also BMI reduction and the decrease in cholesterol LDL and HbA1c.
  • [MeSH-major] Craniopharyngioma / complications. Growth Disorders / drug therapy. Human Growth Hormone / therapeutic use. Pituitary Neoplasms / complications
  • [MeSH-minor] Child. Female. Humans. Male. Overweight / drug therapy. Overweight / etiology. Recombinant Proteins

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  • (PMID = 20529601.001).
  • [ISSN] 2081-237X
  • [Journal-full-title] Pediatric endocrinology, diabetes, and metabolism
  • [ISO-abbreviation] Pediatr Endocrinol Diabetes Metab
  • [Language] pol
  • [Publication-type] Clinical Trial; English Abstract; Journal Article
  • [Publication-country] Poland
  • [Chemical-registry-number] 0 / Recombinant Proteins; 12629-01-5 / Human Growth Hormone
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38. Bahuleyan B, Menon G, Nair S: Immediate postoperative death due to hypothalamic injury following surgery for craniopharyngioma. J Clin Neurosci; 2009 Jun;16(6):850-1
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  • [Title] Immediate postoperative death due to hypothalamic injury following surgery for craniopharyngioma.
  • Autonomic disturbances due to hypothalamic injury that result in postoperative death are rare complications following surgery for craniopharyngioma.
  • We discuss the case of a child who died due to hypothalamic injury following radical excision of a multi-compartmental craniopharyngioma.
  • [MeSH-major] Autonomic Nervous System Diseases / etiology. Craniopharyngioma / surgery. Hypothalamus / injuries. Neurosurgical Procedures / adverse effects. Pituitary Neoplasms / surgery. Postoperative Complications / etiology
  • [MeSH-minor] Child. Craniotomy. Decompression, Surgical. Diabetes Insipidus / drug therapy. Diabetes Insipidus / etiology. Fatal Outcome. Heart Failure / etiology. Heart Failure / physiopathology. Humans. Hypotension / etiology. Hypotension / physiopathology. Magnetic Resonance Imaging. Male. Malignant Hyperthermia / etiology. Third Ventricle / pathology. Third Ventricle / surgery


39. Hofman R, Rosingh HJ: Unilateral hearing loss as primary symptom of craniopharyngioma in a six-year-old girl. J Laryngol Otol; 2008 Mar;122(3):e10
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  • [Title] Unilateral hearing loss as primary symptom of craniopharyngioma in a six-year-old girl.
  • OBJECTIVE: We report a rare case of otological presentation of craniopharyngioma.
  • METHOD: Case report and review of world literature concerning presentations of craniopharyngioma.
  • Magnetic resonance scanning revealed a massive, cystic craniopharyngioma exerting pressure on the patient's ventricular system and brainstem and also invading the internal acoustic canal.
  • The patient's hearing loss completely recovered, and she experienced no neurological or endocrinological side effects of the treatment.
  • Craniopharyngioma have a prevalence of 0.13-2:100,000.
  • CONCLUSION: Craniopharyngioma is a rare disease.
  • [MeSH-major] Craniopharyngioma / complications. Hearing Loss, Unilateral / etiology. Pituitary Neoplasms / complications
  • [MeSH-minor] Child. Craniotomy / methods. Female. Humans. Reflex, Babinski / drug therapy. Treatment Outcome

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  • (PMID = 18252012.001).
  • [ISSN] 1748-5460
  • [Journal-full-title] The Journal of laryngology and otology
  • [ISO-abbreviation] J Laryngol Otol
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article; Review
  • [Publication-country] England
  • [Number-of-references] 6
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40. Chung TT, Drake WM, Evanson J, Walker D, Plowman PN, Chew SL, Grossman AB, Besser GM, Monson JP: Tumour surveillance imaging in patients with extrapituitary tumours receiving growth hormone replacement. Clin Endocrinol (Oxf); 2005 Sep;63(3):274-9
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  • [Title] Tumour surveillance imaging in patients with extrapituitary tumours receiving growth hormone replacement.
  • In most cases, AO-GHD arises as a result of pituitary/peripituitary tumours and/or their treatment, but the effect of GH replacement on recurrence/regrowth of these tumours is unknown.
  • The aim of this study was to examine the effect of GH replacement in a group of patients with primary tumours of the parasellar region, many of which (e.g. craniopharyngioma, glioma or germ cell tumours) might be anticipated to have a higher recurrence rate than secretory and nonsecretory anterior pituitary tumours.
  • PATIENTS AND DESIGN: We report here our experience of prospective imaging in 50 consecutive patients (21 males; mean age 45.9 years) with nonanterior pituitary parasellar tumours treated with GH.
  • All had severe GHD (peak serum GH 9 mU/l or less on dynamic testing) and were treated with an identical dose-titration regimen to maintain serum IGF-I concentrations between the median and upper end of the age-adjusted normal range.
  • The primary diagnoses were: craniopharyngioma (28), germ cell tumour (8), arachnoid cyst (4), meningioma (4), glioma (4) and mensenchymal tumour (2).
  • External pituitary irradiation had been given to 37 (74%) of patients.
  • Measurements Surveillance imaging (magnetic resonance imaging (MRI) 70%, computed tomography (CT) 16%, both 14%) was performed at baseline (prior to GH), at 6--12 months, and then again yearly or as clinically indicated.
  • RESULTS: Four patients had an apparent increase in tumour volume but in only one patient was it considered necessary to abandon GH replacement.
  • In two of the four cases marginal increases in cystic parasellar tumours were not progressive; and in the fourth case apparent recurrence of a suprasellar germ cell tumour was shown to be acellular fibrous tissue only on biopsy.
  • In all other cases either the appearances were unchanged or the amount of tissue was reduced during long-term follow-up on GH.
  • CONCLUSIONS: Overall, GH appears safe with respect to tumour recurrence over this time period in this patient group.
  • Comparison with similar prospective series in patients not receiving GH replacement is desirable.
  • [MeSH-major] Brain / pathology. Growth Hormone / therapeutic use. Hormone Replacement Therapy. Hypopituitarism / drug therapy. Magnetic Resonance Imaging. Neoplasm Recurrence, Local / diagnosis
  • [MeSH-minor] Adult. Brain Neoplasms / complications. Brain Neoplasms / therapy. Combined Modality Therapy. Craniopharyngioma / complications. Craniopharyngioma / therapy. Female. Follow-Up Studies. Germinoma / complications. Germinoma / therapy. Glioma / complications. Glioma / therapy. Humans. Male. Middle Aged. Pituitary Irradiation

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  • (PMID = 16117814.001).
  • [ISSN] 0300-0664
  • [Journal-full-title] Clinical endocrinology
  • [ISO-abbreviation] Clin. Endocrinol. (Oxf)
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] England
  • [Chemical-registry-number] 9002-72-6 / Growth Hormone
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41. Mottolese C, Szathmari A, Berlier P, Hermier M: Craniopharyngiomas: our experience in Lyon. Childs Nerv Syst; 2005 Aug;21(8-9):790-8
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  • [Title] Craniopharyngiomas: our experience in Lyon.
  • OBJECTIVE: We reviewed our experience in surgical treatment of craniopharyngiomas.
  • Surgical treatment of craniopharyngiomas in children represents a challenge for neurosurgeons because it presents a different set of surgical problems.
  • Two groups of patients were distinguished: a group of 36 patients treated with surgical direct surgery; a second group of 24 patients treated only with intracystic chemotherapy with bleomycin (18 patients) or associated with surgery (six patients).
  • In the first group, the removal of lesion was total in 74% of cases.
  • All patients presented ante-pituitary insufficiency and diabetes insipidus, which required substitutive treatment.
  • In the group treated with bleomycin, 18 patients presented a primary cystic or a mixed form of craniopharyngioma and six patients showed a cystic recurrence.
  • The dose used varied from 30 mg to a maximal dose of 150 mg, with a middle dose of 60 mg in the large majority of cases.
  • In this group, the cyst disappeared in 12 patients and reduced to 30% of its initial volume, and stabilization of the lesion was achieved in the other six patients.
  • CONCLUSION: Our experience showed that in the group treated with intracystic chemotherapy alone, results were better with a low rate of morbidity and mortality.
  • In cases of cystic craniopharyngiomas, we considered bleomycin as the treatment of choice.
  • For solid forms or for cases resistant to intracystic chemotherapy with bleomycin, direct surgery has to be proposed.
  • [MeSH-major] Craniopharyngioma / surgery. Cysts / surgery. Pituitary Neoplasms / surgery
  • [MeSH-minor] Adolescent. Child. Child, Preschool. Female. France. Humans. Infant. Magnetic Resonance Imaging. Male. Neurosurgical Procedures. Radiosurgery. Tomography, X-Ray Computed

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  • (PMID = 15971075.001).
  • [ISSN] 0256-7040
  • [Journal-full-title] Child's nervous system : ChNS : official journal of the International Society for Pediatric Neurosurgery
  • [ISO-abbreviation] Childs Nerv Syst
  • [Language] eng
  • [Publication-type] Comparative Study; Journal Article; Review
  • [Publication-country] Germany
  • [Number-of-references] 46
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42. Stieber VW: Radiation therapy for visual pathway tumors. J Neuroophthalmol; 2008 Sep;28(3):222-30
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  • [Title] Radiation therapy for visual pathway tumors.
  • The multimodality management of visual pathway tumors frequently involves radiation.
  • Most commonly, photons are delivered via multiple focused beams aimed at the tumor while sparing adjacent tissues.
  • The dose can be delivered in multiple treatments (radiation therapy) or in a single treatment (radiosurgery).
  • Children with visual pathway gliomas should be treated with chemotherapy alone, delaying the use of radiation therapy until progression.
  • Definitive radiation therapy of optic nerve sheath meningiomas results in stable vision in most patients.
  • Radiation therapy or radiosurgery for pituitary tumors can result in control of both tumor growth and hormone hypersecretion.
  • Postoperative radiation therapy or radiosurgery of craniopharyngiomas significantly improves local control rates compared with surgery alone.
  • Radiation therapy is highly effective for eradicating orbital pseudolymphoma and lymphoma.
  • The risk of complications from radiation treatment is dependent on the organ at risk, the cumulative dose it receives, and the dose delivered per fraction.
  • [MeSH-major] Optic Nerve / radiation effects. Optic Nerve Diseases / radiotherapy. Optic Nerve Glioma / radiotherapy. Radiotherapy / methods
  • [MeSH-minor] Craniopharyngioma / complications. Craniopharyngioma / pathology. Craniopharyngioma / radiotherapy. Humans. Lymphoma / complications. Lymphoma / pathology. Lymphoma / radiotherapy. Meningioma / complications. Meningioma / pathology. Meningioma / radiotherapy. Pituitary Neoplasms / complications. Pituitary Neoplasms / pathology. Pituitary Neoplasms / radiotherapy. Pseudolymphoma / complications. Pseudolymphoma / pathology. Pseudolymphoma / radiotherapy. Radiation Dosage

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  • (PMID = 18769290.001).
  • [ISSN] 1536-5166
  • [Journal-full-title] Journal of neuro-ophthalmology : the official journal of the North American Neuro-Ophthalmology Society
  • [ISO-abbreviation] J Neuroophthalmol
  • [Language] eng
  • [Publication-type] Journal Article; Review
  • [Publication-country] United States
  • [Number-of-references] 106
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43. Steinbok P, Hukin J: Intracystic treatments for craniopharyngioma. Neurosurg Focus; 2010 Apr;28(4):E13
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  • [Title] Intracystic treatments for craniopharyngioma.
  • Craniopharyngioma is a benign tumor histopathologically and in theory should be curable by radical resection.
  • In practice, this tumor behaves like a chronic disease, with many issues related to the effect of the tumor itself and the various treatments on the adjacent structures, such as the pituitary stalk and gland, hypothalamus, visual apparatus, and suprasellar arteries.
  • A multimodality approach to the management of these tumors may produce the optimal outcome, balancing disease control and quality of life.
  • In this paper, the role of intracystic therapies is reviewed, with the major focus on intracystic bleomycin and interferon-alpha.
  • [MeSH-major] Antibiotics, Antineoplastic / administration & dosage. Antibiotics, Antineoplastic / therapeutic use. Bleomycin / therapeutic use. Craniopharyngioma / drug therapy. Immunologic Factors / therapeutic use. Interferon-alpha / therapeutic use. Pituitary Neoplasms / drug therapy
  • [MeSH-minor] Combined Modality Therapy. Humans. Injections, Intralesional. Radiotherapy, Conformal. Treatment Outcome

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  • (PMID = 20367357.001).
  • [ISSN] 1092-0684
  • [Journal-full-title] Neurosurgical focus
  • [ISO-abbreviation] Neurosurg Focus
  • [Language] eng
  • [Publication-type] Journal Article; Review
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / Antibiotics, Antineoplastic; 0 / Immunologic Factors; 0 / Interferon-alpha; 11056-06-7 / Bleomycin
  • [Number-of-references] 51
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44. Olchovsky D, Ezra D, Vered I, Hadani M, Shimon I: Symptomatic hyponatremia as a presenting sign of hypothalamic-pituitary disease: a syndrome of inappropriate secretion of antidiuretic hormone (SIADH)-like glucocorticosteroid responsive condition. J Endocrinol Invest; 2005 Feb;28(2):151-6
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  • [Title] Symptomatic hyponatremia as a presenting sign of hypothalamic-pituitary disease: a syndrome of inappropriate secretion of antidiuretic hormone (SIADH)-like glucocorticosteroid responsive condition.
  • Hyponatremia associated with high urine osmolality is usually caused by inappropriate secretion of antidiuretic hormone.
  • As secondary hypoadrenalism requires a specific treatment, a high index of suspicion and appropriate hormonal testing are required to differentiate between these two entities.
  • We retrospectively studied 10 patients with a previously undiagnosed hypothalamic-pituitary disease who presented with an acute symptomatic hyponatremia.
  • Two of these 3 patients did not respond adequately to the low-dose ACTH test.
  • Endocrine evaluation disclosed partial or complete hypopituitarism in all 10 patients.
  • Six patients had pituitary macroadenomas, one had a craniopharyngioma, one patient had a large aneurysm of the internal carotid with sellar destruction and two others had empty sella.
  • Treatment by fluid restriction did not affect serum sodium levels significantly.
  • [MeSH-major] Glucocorticoids / therapeutic use. Hyponatremia / drug therapy. Hyponatremia / etiology. Hypothalamic Diseases / complications. Inappropriate ADH Syndrome / complications. Pituitary Diseases / complications
  • [MeSH-minor] Adrenocorticotropic Hormone / administration & dosage. Aged. Dose-Response Relationship, Drug. Female. Humans. Hydrocortisone / blood. Male. Middle Aged. Retrospective Studies

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  • (PMID = 15887861.001).
  • [ISSN] 0391-4097
  • [Journal-full-title] Journal of endocrinological investigation
  • [ISO-abbreviation] J. Endocrinol. Invest.
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] Italy
  • [Chemical-registry-number] 0 / Glucocorticoids; 9002-60-2 / Adrenocorticotropic Hormone; WI4X0X7BPJ / Hydrocortisone
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45. Albert A, Cruz O, Montaner A, Vela A, Badosa J, Castañón M, Morales L: [Congenital solid tumors. A thirteen-year review]. Cir Pediatr; 2004 Jul;17(3):133-6
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  • [Title] [Congenital solid tumors. A thirteen-year review].
  • [Transliterated title] Tumores sólidos congénitos. Revisión de 13 años.
  • Tumors diagnosed during the first month of life are infrequent: 0.5 to 2% of all childhood neoplasms.
  • This is an interesting group of tumors because their type, relative incidence, natural history and response to treatment differ from those seen in older children.
  • AIM: To contribute the experience of our institution in congenital tumors the last 13 years.
  • MATERIAL AND METHODS: The records of all neonates (< 31 days old) diagnosed with solid tumors since January 1990 to December 2002 have been retrospectively reviewed.
  • RESULTS: Twenty-seven neonates have been diagnosed with tumors in the last 13 years.
  • Neuroblastoma was the commonest tumor (10 cases, 37%), of which 4 were stage I, 4 stage IV-S and 2 stage III.
  • There were 8 teratomas (3 sacrocoxigeal, 1 retroperitoneal, 1 in the CNS, 1 orbitary and two oronasal), two hepatic tumors (1 hepatoblastoma, 1 hemangioendothelioma, two CNS tumors, two giant nevus (one on a hamartoma), and one each Wilms tumor, infantile fibrosarcoma and myofibroblastic tumor.
  • Treatment was surgical resection alone in 17 cases (68%) and surgery + chemotherapy in 8 (32%) (5 neuroblastomas, one CNS tumor, one Wilms tumor and one presacral teratoma who developed a yolk sac tumor); 3 patients died (11%): one at surgery, one of tumoural airway obstruction at birth and one with craniopharyngioma.
  • Among the 14 tumors that were initially not malignant, two can be locally agressive, one was an immature teratoma, the giant nevus with hamartoma developed in situ melanoma, the other nevus had meningeal melanosis with hydrocephalus, and one mature presacral teratoma developed a yolk sac tumor.
  • CONCLUSIONS: Diagnosis of congenital tumors is performed earlier in recent years due to the wide use of prenatal ultrasound.
  • Their natural history is more benign than in other age groups, except for CNS tumors and very large or obstructing tumors.
  • The histological patern is not determinant of the outcome.
  • Complete surgical excision is the treatment of choice, most cases not need adjuvant chemotherapy.
  • We ought to pass this message on to our colleagues in prenatal diagnosis, so parents get reliable information.
  • [MeSH-major] Central Nervous System Neoplasms / congenital. Kidney Neoplasms / congenital. Liver Neoplasms / congenital. Neuroblastoma / congenital. Skin Neoplasms / congenital. Soft Tissue Neoplasms / congenital. Teratoma / congenital. Wilms Tumor / congenital
  • [MeSH-minor] Female. Follow-Up Studies. Humans. Infant, Newborn. Male. Neoplasm Recurrence, Local. Postoperative Complications. Pregnancy. Prenatal Diagnosis. Time Factors

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  • (PMID = 15503950.001).
  • [ISSN] 0214-1221
  • [Journal-full-title] Cirugía pediátrica : organo oficial de la Sociedad Española de Cirugía Pediátrica
  • [ISO-abbreviation] Cir Pediatr
  • [Language] spa
  • [Publication-type] Comparative Study; English Abstract; Journal Article
  • [Publication-country] Spain
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46. Rittinger O, Kranzinger M, Jones R, Jones N: Malignant astrocytoma arising 10 years after combined treatment of craniopharyngioma. J Pediatr Endocrinol Metab; 2003 Jan;16(1):97-101
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  • [Title] Malignant astrocytoma arising 10 years after combined treatment of craniopharyngioma.
  • Craniopharyngioma is the third most common intracranial tumor in childhood.
  • Following surgery, virtually all patients present with hypopituitarism and are at considerable risk of tumor recurrence.
  • Secondary tumors, however, are rare, occurring usually 10 years after diagnosis and associated with poor prognosis.
  • We report on a 5 year-old boy in whom craniopharyngioma was diagnosed due to unilateral visual loss.
  • After surgery he underwent conventional radiation therapy with a total tumor dose of 55 Gy, and had hormonal support with DDAVP, thyroxine, and a variable dose of hydrocortisone.
  • Growth velocity declined slowly in the first 4 years, but improved later on again without GH therapy despite abnormal provocative tests.
  • At the age of 15 years he developed peripheral facial nerve palsy due to a malignant astrocytoma (WHO grade III/IV).
  • Repeated conventional radiation therapy with an additional stereotactic boost and chemotherapy could not prevent the fatal outcome.
  • This observation may temper the use of radiosurgery in benign intracranial tumors.
  • [MeSH-major] Astrocytoma / diagnosis. Brain Stem Neoplasms / diagnosis. Craniopharyngioma / radiotherapy. Craniopharyngioma / surgery. Neoplasms, Second Primary / diagnosis. Pituitary Neoplasms / radiotherapy. Pituitary Neoplasms / surgery
  • [MeSH-minor] Child, Preschool. Combined Modality Therapy. Fatal Outcome. Hormones / blood. Humans. Magnetic Resonance Imaging. Male. Neoplasm Invasiveness. Time Factors

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  • (PMID = 12585346.001).
  • [ISSN] 0334-018X
  • [Journal-full-title] Journal of pediatric endocrinology & metabolism : JPEM
  • [ISO-abbreviation] J. Pediatr. Endocrinol. Metab.
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] England
  • [Chemical-registry-number] 0 / Hormones
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47. Park DH, Park JY, Kim JH, Chung YG, Lee HK, Lee KC, Suh JK: Outcome of postoperative intratumoral bleomycin injection for cystic craniopharyngioma. J Korean Med Sci; 2002 Apr;17(2):254-9
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  • [Title] Outcome of postoperative intratumoral bleomycin injection for cystic craniopharyngioma.
  • Total excision is a treatment of choice in preventing the relapse of craniopharyngioma, but for tumors involving an extensive area, it is often associated with an increased risk of complications.
  • We have performed a partial or subtotal tumor removal followed by repeated injection of bleomycin into the remaining tumor through a subcutaneous reservoir as postoperative adjuvant therapy.
  • A retrospective review of clinical, radiological, and surgical data was performed for 10 patients (5 males and 5 females; age, 3-65 yr; follow-up duration, 12-79 months) with cystic craniopharyngiomas.
  • The shrinkage and/or stabilization of tumor was initially noted in all cases.
  • The recurrence of tumor was seen in 4 cases (40%).
  • The decreased or increased level of LDH was interpreted as tumor shrinkage or recurrence, respectively.
  • Our study demonstrates that postoperative bleo-mycin injection for cystic craniopharyngioma, although does not appear to eradicate the tumor, decreases and stabilizes the tumor size, when used as an adjuvant therapy in young patients.
  • [MeSH-major] Antibiotics, Antineoplastic / therapeutic use. Bleomycin / therapeutic use. Craniopharyngioma / drug therapy. Pituitary Neoplasms / drug therapy. Postoperative Care
  • [MeSH-minor] Adolescent. Adult. Aged. Brain / radiation effects. Child. Child, Preschool. Female. Follow-Up Studies. Humans. Injections. L-Lactate Dehydrogenase / metabolism. Male. Middle Aged. Retrospective Studies. Tomography, X-Ray Computed / methods

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  • (PMID = 11961313.001).
  • [ISSN] 1011-8934
  • [Journal-full-title] Journal of Korean medical science
  • [ISO-abbreviation] J. Korean Med. Sci.
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] Korea (South)
  • [Chemical-registry-number] 0 / Antibiotics, Antineoplastic; 11056-06-7 / Bleomycin; EC 1.1.1.27 / L-Lactate Dehydrogenase
  • [Other-IDs] NLM/ PMC3054855
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48. Liu AK, Bagrosky B, Fenton LZ, Gaspar LE, Handler MH, McNatt SA, Foreman NK: Vascular abnormalities in pediatric craniopharyngioma patients treated with radiation therapy. Pediatr Blood Cancer; 2009 Feb;52(2):227-30
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  • [Title] Vascular abnormalities in pediatric craniopharyngioma patients treated with radiation therapy.
  • BACKGROUND: Craniopharyngioma is a benign brain tumor that can be treated with some combination of surgery, intracystic chemotherapy and radiation therapy.
  • Treatment for craniopharyngioma, especially radiation therapy, is associated with a variety of long-term toxicities including vascular abnormalities.
  • We report on the incidence of vascular abnormalities seen in the children with craniopharyngioma who received radiation therapy at our institution.
  • PROCEDURE: We reviewed our experience with craniopharyngioma patients who received radiation therapy from 1995 to 2008.
  • We reviewed clinical data including surgery, chemotherapy, radiation therapy and imaging for vasculopathy.
  • RESULTS: Twenty of the 22 children with craniopharyngioma who received radiation therapy had imaging available.
  • Six of the 20 were found to have some type of vasculopathy.
  • One had bilateral temporal cavernomas, one had moyamoya syndrome, one had an aneurysm of the internal carotid artery and three children had decreases in the caliber of the carotid or cerebral arteries, but were asymptomatic.
  • Two of the six children with abnormalities also received intracystic bleomycin prior to radiation therapy.
  • CONCLUSIONS: We report a high incidence of vascular abnormalities in children with craniopharyngioma.
  • [MeSH-major] Craniopharyngioma / complications. Craniopharyngioma / radiotherapy. Vascular Diseases / etiology
  • [MeSH-minor] Adolescent. Bleomycin / adverse effects. Bleomycin / therapeutic use. Child. Child, Preschool. Combined Modality Therapy. Female. Humans. Male. Radiotherapy / adverse effects. Retrospective Studies

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  • [Copyright] (c) 2008 Wiley-Liss, Inc.
  • (PMID = 18937328.001).
  • [ISSN] 1545-5017
  • [Journal-full-title] Pediatric blood & cancer
  • [ISO-abbreviation] Pediatr Blood Cancer
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] United States
  • [Chemical-registry-number] 11056-06-7 / Bleomycin
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49. Darendeliler F, Karagiannis G, Wilton P: Headache, idiopathic intracranial hypertension and slipped capital femoral epiphysis during growth hormone treatment: a safety update from the KIGS database. Horm Res; 2007;68 Suppl 5:41-7
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  • [Title] Headache, idiopathic intracranial hypertension and slipped capital femoral epiphysis during growth hormone treatment: a safety update from the KIGS database.
  • BACKGROUND: Several uncommon adverse effects may be related to growth hormone (GH) treatment.
  • Three potential side effects, headache, idiopathic intracranial hypertension (IIH) and slipped capital femoral epiphysis (SCFE), will be discussed.
  • Data from 57,968 children in the KIGS (Pfizer International Growth Study database) were analyzed to determine the effects of recombinant human GH (Genotropin) on these side effects.
  • The diagnostic groups were idiopathic GH deficiency (IGHD) (n = 27,690), congenital GHD (CGHD) (n = 2,547), craniopharyngioma (n = 1,155), cranial tumours (n = 2,203), Turner syndrome (TS) (n = 6,092), idiopathic short stature (ISS) (n = 5,286), small for gestational age (SGA) (n = 2,973), chronic renal insufficiency (CRI) (n = 1,753) and Prader-Willi syndrome (PWS) (n = 1,368).
  • RESULTS: Total incidence (per 100,000 treatment years) of headache was 793.5 (n = 569).
  • The incidence was significantly higher in the groups of patients with craniopharyngiomas, CGHD and cranial tumours than in the other diagnostic groups (p < 0.05 for all).
  • IIH occurred in 41 children resulting in a total incidence (per 100,000 treatment years) of 27.7.
  • The incidence (per 100,000 treatment years) was significantly lower in patients with IGHD (12.2) than in those with TS (56.4) (p = 0.0004), CGHD (54.5) (p = 0.0064), PWS (68.3) (p = 0.0263) and CRI (147.8) (p < 0.001).
  • No cases of IIH were reported in the ISS group of patients.
  • The median duration from onset of GH therapy to IIH ranged from 0.01 to 1.3 years in various diagnostic groups.
  • SCFE was observed in a total of 52 children resulting in a total incidence (per 100,000 treatment years) of 73.4.
  • The incidence (per 100,000 treatment years) was significantly lower in patients with IGHD (18.3) and in those children with ISS (14.5) than in the TS (84.5), cranial tumours (86.1) and craniopharyngioma groups (120.5) (p < 0.05 for all).
  • The median duration from onset of GH therapy to SCFE ranged from 0.4 to 2.5 years.
  • CONCLUSIONS: The incidences of IIH and SCFE in this analysis are lower than the values reported in previous KIGS analyses and comparable to other databases.
  • [MeSH-major] Epiphyses, Slipped / chemically induced. Femur Head / drug effects. Headache / chemically induced. Human Growth Hormone / adverse effects. Pseudotumor Cerebri / chemically induced

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  • (PMID = 18174706.001).
  • [ISSN] 1423-0046
  • [Journal-full-title] Hormone research
  • [ISO-abbreviation] Horm. Res.
  • [Language] eng
  • [Publication-type] Journal Article; Review
  • [Publication-country] Switzerland
  • [Chemical-registry-number] 0 / Recombinant Proteins; 12629-01-5 / Human Growth Hormone
  • [Number-of-references] 48
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50. Rohrer TR, Langer T, Grabenbauer GG, Buchfelder M, Glowatzki M, Dörr HG: Growth hormone therapy and the risk of tumor recurrence after brain tumor treatment in children. J Pediatr Endocrinol Metab; 2010 Sep;23(9):935-42
MedlinePlus Health Information. consumer health - Childhood Brain Tumors.

  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Growth hormone therapy and the risk of tumor recurrence after brain tumor treatment in children.
  • To assess the effect of human growth hormone (hGH) therapy and other factors on tumor recurrence after treatment of pediatric brain tumors (BTs), we retrospectively analyzed data from 108 craniopharyngioma, medulloblastoma, and ependymoma patients.
  • Risk factors were identified using multifactorial univariate regression analysis.
  • There were significant correlations for completeness of tumor removal and recurrence-free survival (RFS).
  • This difference was found only for medulloblastomas and accounted for by higher rates of incomplete tumor removal in non-hGH patients.
  • Craniopharyngioma recurrence correlated only with RFS.
  • Malignant BT recurrence correlated with completeness of tumor removal, chemotherapy, and RFS.
  • 4 children developed SMNs, 3/4 after hGH therapy.
  • We conclude that hGH therapy after treatment of pediatric BTs does not increase tumor recurrence risk.
  • [MeSH-major] Brain Neoplasms / therapy. Human Growth Hormone / adverse effects. Neoplasm Recurrence, Local / etiology
  • [MeSH-minor] Adolescent. Child. Child, Preschool. Female. Humans. Infant. Infant, Newborn. Male. Neoplasms, Second Primary / etiology. Retrospective Studies. Risk

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  • (PMID = 21175094.001).
  • [ISSN] 0334-018X
  • [Journal-full-title] Journal of pediatric endocrinology & metabolism : JPEM
  • [ISO-abbreviation] J. Pediatr. Endocrinol. Metab.
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] England
  • [Chemical-registry-number] 12629-01-5 / Human Growth Hormone
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51. Geffner M, Lundberg M, Koltowska-Häggström M, Abs R, Verhelst J, Erfurth EM, Kendall-Taylor P, Price DA, Jonsson P, Bakker B: Changes in height, weight, and body mass index in children with craniopharyngioma after three years of growth hormone therapy: analysis of KIGS (Pfizer International Growth Database). J Clin Endocrinol Metab; 2004 Nov;89(11):5435-40
MedlinePlus Health Information. consumer health - Body Weight.

  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Changes in height, weight, and body mass index in children with craniopharyngioma after three years of growth hormone therapy: analysis of KIGS (Pfizer International Growth Database).
  • Extreme degrees of obesity may occur in association with hypothalamic tumors, usually after surgical intervention.
  • This phenomenon has been reported to occur in as many as 25-75% of children undergoing extensive surgical extirpation of craniopharyngiomas (Cranio).
  • Because less is known about the auxology of children with Cranio with milder alterations in growth, we undertook a 3-yr longitudinal analysis, using the KIGS database (Pfizer International Growth Database), to study their growth patterns and evolution of weight.
  • We compared the effect of GH therapy on height, weight, and body mass index (BMI) in 199 prepubertal children with diagnosed Cranio treated by surgery and/or radiotherapy to two other groups of children with other causes of organic GH deficiency (OGHD): one with postsurgical and/or postirradiated OGHD (OGHD + S/I; n = 92) and the other with OGHD not due to Cranio and not having undergone either surgery or irradiation (OGHD - S/I; n = 85).
  • At the start of GH therapy, 1) mean chronological (P < 0.0001) and bone (P = 0.0002) ages were youngest in OGHD - S/I and oldest in OGHD + S/I;.
  • 3) mean weight and BMI SDS were greatest in Cranio and least in OGHD - S/I (both P < 0.0001); and 4) the mean initial GH dose prescribed was highest in OGHD - S/I and comparable in the other two groups (P < 0.0001).
  • After 3 yr of GH therapy, 1) mean bone age remained youngest in OGHD - S/I and oldest in OGHD + S/I (P < 0.0001);.
  • 2) mean height SDS was highest in Cranio and comparably lower in the other two groups (P = 0.0159);.
  • 3) mean weight and BMI SDS remained greatest in Cranio and least in OGHD - S/I (P < 0.0001 and P = 0.0003, respectively); and 4) the mean GH dose remained highest in the OGHD - S/I group and least in the Cranio group (P = 0.0082).
  • There were statistically significant increases within each group between the start of treatment and after 3 yr of GH therapy in height and weight, but not in BMI SDS.
  • Lastly, after 3 yr of GH treatment, children in the Cranio group continued to have disproportionately heavier weight and higher BMI (with the greatest values in those with lower stimulated peak GH concentrations) compared with members of the other two groups, with no salutary effect of GH treatment on weight SDS and a mild improvement in BMI SDS.
  • After S/I treatment, children with Cranio are disproportionately prone to varying degrees of weight gain compared with children with other forms of OGHD.
  • In the present cohort of prepubertal children with Cranio, GH therapy induced excellent linear growth, but failed to have an ameliorative effect on weight gain and had only a slight beneficial effect on BMI gain.
  • Because affected children may have resultant significant long-term medical morbidity and diminished quality of life, it is critical that the mechanism of this phenomenon be determined to devise helpful preventive or therapeutic interventions.
  • [MeSH-major] Body Height / drug effects. Body Mass Index. Body Weight / drug effects. Craniopharyngioma / drug therapy. Growth Hormone / therapeutic use
  • [MeSH-minor] Human Growth Hormone / deficiency. Humans

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  • (PMID = 15531494.001).
  • [ISSN] 0021-972X
  • [Journal-full-title] The Journal of clinical endocrinology and metabolism
  • [ISO-abbreviation] J. Clin. Endocrinol. Metab.
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] United States
  • [Chemical-registry-number] 12629-01-5 / Human Growth Hormone; 9002-72-6 / Growth Hormone
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52. Feldt-Rasmussen U, Wilton P, Jonsson P, KIMS Study Group, KIMS International Board: Aspects of growth hormone deficiency and replacement in elderly hypopituitary adults. Growth Horm IGF Res; 2004 Jun;14 Suppl A:S51-8
Hazardous Substances Data Bank. CHOLESTEROL .

  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • Normal ageing is associated with a decline in spontaneous growth hormone (GH) secretion, and although elderly hypopituitary adults demonstrate an increase in total and central fat compared with age-matched controls and are distinguishable from control subjects in terms of GH responsiveness on dynamic testing, there are few data available on the response to GH replacement in older subjects.
  • We have studied the baseline characteristics of 295 patients (173 males and 122 females) aged >65 years of age who began GH replacement therapy at the time of entry into the KIMS program (Pfizer International Metabolic Database) and the effects of GH replacement in 125 patients who completed at least 12 months of GH replacement therapy.
  • Data were compared with those of 2469 (1249 males and 1220 females) patients aged <65 years with adult-onset GH deficiency (GHD).
  • There was a higher proportion of pituitary adenoma relative to craniopharyngioma in the older age group (P<0.001), but there was no difference between groups in the degree of hypopituitarism (number of additional hormone deficiencies).
  • Blood pressure, cholesterol and low-density lipoprotein (LDL) cholesterol levels were positively correlated with age, and older patients had a predictably higher prevalence of diabetes mellitus, coronary heart disease, stroke and history of hypertension.
  • Quality of life (Assessment of Growth Hormone Deficiency in Adults (AGHDA) score) was impaired in both groups before the start of GH therapy.
  • GH replacement doses were lower in older patients with GHD as compared with patients <65 years old.
  • After 12 months of GH replacement, significant improvements were evident in waist circumference, waist/hip ratio, lean body mass, diastolic blood pressure, total and LDL cholesterol levels and AGHDA scores in patients aged <65 years.
  • These data indicate a positive benefit from GH replacement in older patients with hypopituitarism - particularly in relation to quality of life - using a lower dose of GH for replacement and with appropriate age-related safety controls.
  • [MeSH-major] Hormone Replacement Therapy. Human Growth Hormone / deficiency. Human Growth Hormone / therapeutic use. Hypopituitarism / drug therapy
  • [MeSH-minor] Adenoma / complications. Age Factors. Aged. Cholesterol / blood. Craniopharyngioma / complications. Female. Humans. Hypertension / complications. Male. Middle Aged. Pituitary Neoplasms / complications

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  • (PMID = 15135778.001).
  • [ISSN] 1096-6374
  • [Journal-full-title] Growth hormone & IGF research : official journal of the Growth Hormone Research Society and the International IGF Research Society
  • [ISO-abbreviation] Growth Horm. IGF Res.
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] Scotland
  • [Chemical-registry-number] 12629-01-5 / Human Growth Hormone; 97C5T2UQ7J / Cholesterol
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53. Sadatomo T, Sakoda K, Yamanaka M, Kutsuna M, Kurisu K: Mazindol administration improved hyperphagia after surgery for craniopharyngioma--case report. Neurol Med Chir (Tokyo); 2001 Apr;41(4):210-2
Hazardous Substances Data Bank. MAZINDOL .

  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Mazindol administration improved hyperphagia after surgery for craniopharyngioma--case report.
  • A 54-year-old man presented with visual disturbance and polydipsia.
  • Bifrontal craniotomy was performed and the tumor was totally removed.
  • Histological findings confirmed the diagnosis of craniopharyngioma.
  • About one month after the operation the patient manifested hyperphagia and he gained 15 kg in one month.
  • [MeSH-major] Appetite Depressants / therapeutic use. Craniopharyngioma / surgery. Hyperphagia / drug therapy. Mazindol / therapeutic use. Postoperative Complications / drug therapy. Supratentorial Neoplasms / surgery
  • [MeSH-minor] Humans. Male. Middle Aged. Treatment Outcome

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  • (PMID = 11381681.001).
  • [ISSN] 0470-8105
  • [Journal-full-title] Neurologia medico-chirurgica
  • [ISO-abbreviation] Neurol. Med. Chir. (Tokyo)
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] Japan
  • [Chemical-registry-number] 0 / Appetite Depressants; C56709M5NH / Mazindol
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