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2. 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.
  • 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.

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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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3. 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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  • [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.
  • 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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4. 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.
  • 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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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).
  • 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.
  • [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. 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].
  • 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.
  • CONCLUSION: The current experience confirms the still remarkable challenges in the treatment of craniopharyngioma in childhood.
  • 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.
  • [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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8. 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.
  • 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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9. Habrand JL, De Crevoisier R: Radiation therapy in the management of childhood brain tumors. Childs Nerv Syst; 2001 Feb;17(3):121-33
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  • [Title] Radiation therapy in the management of childhood brain tumors.
  • Radiation therapy (RT) still plays a major role in the management of intracranial malignancies, together with surgical resection and, more recently, chemotherapy.
  • Technical innovations have also greatly benefited gliomas: modern techniques dealing with 3D CT and MRI-based treatment combined with stereotactic positioning of the patients, achieve a high degree of conformity that might improve both local control and longterm neurocognitive and growth sequelae.
  • [MeSH-major] Brain Neoplasms / radiotherapy
  • [MeSH-minor] Brain / radiation effects. Child. Child Development / radiation effects. Cognition / radiation effects. Craniopharyngioma / radiotherapy. Dose Fractionation. Dose-Response Relationship, Radiation. Glioma / radiotherapy. Humans. Medulloblastoma / radiotherapy. Neuroectodermal Tumors, Primitive / radiotherapy. Pinealoma / radiotherapy. Radiotherapy / adverse effects. Radiotherapy / methods. Radiotherapy / psychology

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  • (PMID = 11305764.001).
  • [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] Journal Article; Review
  • [Publication-country] Germany
  • [Number-of-references] 156
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10. Karavitaki N, Cudlip S, Adams CB, Wass JA: Craniopharyngiomas. Endocr Rev; 2006 Jun;27(4):371-97
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  • Craniopharyngiomas are rare, mainly sellar/parasellar, epithelial tumors diagnosed during childhood or adult life.
  • Despite their benign histological appearance, they often show an unpredictable growth pattern, which, combined with the lack of randomized studies, poses significant difficulties in the establishment of an optimal therapeutic protocol.
  • This should focus on the prevention of recurrence(s), improvement of survival, reduction of the significant disease and treatment-related morbidity (endocrine, visual, hypothalamic, neurobehavioral, and cognitive), and preservation of the quality of life.
  • Currently, surgical excision followed by external beam irradiation, in cases of residual tumor, is the main treatment option.
  • Intracystic irradiation or bleomycin, stereotactic radiosurgery, or radiotherapy and systemic chemotherapy are alternative approaches; their place in the management plan remains to be assessed in adequately powered long-term trials.
  • Apart from the type of treatment, the identification of clinical and imaging parameters that will predict patients with a better prognosis is difficult.
  • The central registration of patients with these challenging tumors may provide correlates between treatments and outcomes and establish prognostic factors at the pathological or molecular level that may further guide us in the future.
  • [MeSH-major] Craniopharyngioma / therapy. Pituitary Neoplasms / therapy
  • [MeSH-minor] Antibiotics, Antineoplastic / therapeutic use. Bleomycin / adverse effects. Bleomycin / therapeutic use. Cognition Disorders / etiology. Humans. Hypothalamic Diseases / etiology. Neoplasm Recurrence, Local / etiology. Radiotherapy / adverse effects. Radiotherapy / methods

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  • (PMID = 16543382.001).
  • [ISSN] 0163-769X
  • [Journal-full-title] Endocrine reviews
  • [ISO-abbreviation] Endocr. Rev.
  • [Language] eng
  • [Publication-type] Journal Article; Review
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / Antibiotics, Antineoplastic; 11056-06-7 / Bleomycin
  • [Number-of-references] 256
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11. Ohmori K, Collins J, Fukushima T: Craniopharyngiomas in children. Pediatr Neurosurg; 2007;43(4):265-78
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  • BACKGROUND: The modern era of pediatric craniopharyngioma treatment includes multiple modalities including microsurgical resection, irradiation, brachytherapy or chemotherapy.
  • No clear consensus as to the best therapeutic approach has yet been established.
  • The aim of this study was to describe the techniques and strategies for the treatment of pediatric craniopharyngiomas in light of a literature review with particular attention to the incidence of adverse postoperative effects.
  • METHODS: Twenty-seven pediatric patients (median age 9.0 years) who were surgically treated for craniopharyngiomas were evaluated.
  • According to the literature, recurrence rates range from zero to 52.8% (average 16.1%) in the gross total resection group, 51.1% in the subtotal resection without radiation therapy group, and 33.5% in the subtotal resection with radiation therapy group.
  • CONCLUSIONS: For patients in whom early postoperative MRI reveals complete craniopharyngioma removal, a very low rate of recurrence is anticipated.
  • [MeSH-major] Craniopharyngioma / surgery. Neurosurgical Procedures / methods. Pituitary Neoplasms / surgery

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  • [Copyright] Copyright (c) 2007 S. Karger AG, Basel.
  • (PMID = 17627142.001).
  • [ISSN] 1016-2291
  • [Journal-full-title] Pediatric neurosurgery
  • [ISO-abbreviation] Pediatr Neurosurg
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article; Review
  • [Publication-country] Switzerland
  • [Number-of-references] 61
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12. 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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  • [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.
  • 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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13. 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.
  • 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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14. Cannavò S, Marini F, Trimarchi F: Patients with craniopharyngiomas: therapeutical difficulties with growth hormone. J Endocrinol Invest; 2008 Sep;31(9 Suppl):56-60
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  • Craniopharyngioma (CP) is a rare and benign tumor of the pituitary region.
  • In patients with GH deficiency (GHD), recombinant GH (rGH) replacement is recommended, after a near complete surgical excision of CP and exclusion of tumor progression.
  • The risk of CP recurrence or progression due to rGH therapy is unproven.
  • Treatment with rGH improves significantly the quality of life (QoL), although body composition and lipid abnormalities are not modified.
  • They had worse QoL than their childhood onset (CO)-counterpart.
  • During 3-yr rGH treatment mean body mass index, fat mass percentage, and both hip and waist circumferences decreased significantly only in CO patients.
  • [MeSH-major] Craniopharyngioma / drug therapy. Human Growth Hormone / adverse effects. Human Growth Hormone / therapeutic use. Pituitary Neoplasms / drug therapy
  • [MeSH-minor] Aged. Body Mass Index. Child. Databases, Factual. Hormone Replacement Therapy / adverse effects. Humans. Hypopituitarism / drug therapy. Hypopituitarism / epidemiology. Hypopituitarism / etiology. Hypopituitarism / psychology. Middle Aged. Prevalence. Quality of Life

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  • (PMID = 19020388.001).
  • [ISSN] 0391-4097
  • [Journal-full-title] Journal of endocrinological investigation
  • [ISO-abbreviation] J. Endocrinol. Invest.
  • [Language] eng
  • [Publication-type] Comparative Study; Evaluation Studies; Journal Article
  • [Publication-country] Italy
  • [Chemical-registry-number] 12629-01-5 / Human Growth Hormone
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15. 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).
  • Mean time of remission was 6,5 yrs (+/- 2,41).
  • 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).
  • 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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16. 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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  • 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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17. 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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  • 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.
  • 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.
  • 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

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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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18. 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).
  • [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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19. 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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  • 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.
  • 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.
  • Their natural history is more benign than in other age groups, except for CNS tumors and very large or obstructing tumors.
  • Complete surgical excision is the treatment of choice, most cases not need adjuvant chemotherapy.
  • [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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