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1. Lacaze E, Kieffer V, Streri A, Lorenzi C, Gentaz E, Habrand JL, Dellatolas G, Kalifa C, Grill J: Neuropsychological outcome in children with optic pathway tumours when first-line treatment is chemotherapy. Br J Cancer; 2003 Dec 1;89(11):2038-44
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  • [Title] Neuropsychological outcome in children with optic pathway tumours when first-line treatment is chemotherapy.
  • Standard treatment of optic pathways gliomas consists of radiotherapy and surgery when feasible.
  • Owing to the toxicity of irradiation, chemotherapy has emerged as an interesting therapeutic option, especially in young children.
  • This study describes the neuropsychological profile of 27 children (aged between 1.5 and 15.7 years) with optic pathways gliomas treated with chemotherapy as first-line treatment.
  • Eight of them also received radiotherapy as salvage treatment.
  • Eight had neurofibromatosis type 1 (NF1).
  • Intellectual outcome was preserved in children treated with chemotherapy only (mean=107+/-17) compared to children also receiving radiotherapy (mean IQ=88+/-24) or children having NF1 and treated with chemotherapy (mean IQ=80+/-13).
  • Scores for abstract reasoning, mental arithmetic, chessboard/coding, perception, judgement of line orientation were lower in children irradiated than in those treated only by chemotherapy.
  • With respect to long-term neuropsychological outcome, our study shows that a chemotherapy-first strategy can preserve the intellectual outcome of these patients either by avoiding the need of radiotherapy or by delaying its use as much as possible.
  • [MeSH-major] Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Brain Neoplasms / drug therapy. Intelligence / drug effects. Optic Nerve Glioma / drug therapy. Optic Nerve Glioma / psychology
  • [MeSH-minor] Adolescent. Child. Child, Preschool. Female. Humans. Infant. Male. Neurofibromatosis 1 / complications. Neuropsychological Tests

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  • (PMID = 14647135.001).
  • [ISSN] 0007-0920
  • [Journal-full-title] British journal of cancer
  • [ISO-abbreviation] Br. J. Cancer
  • [Language] eng
  • [Publication-type] Journal Article; Research Support, Non-U.S. Gov't
  • [Publication-country] England
  • [Other-IDs] NLM/ PMC2376861
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2. Aguilar Moliner I, Costa Orvay JA, Juma K, Costa Clara JM, Cruz Martínez O, Pou Fernández J: [Optic pathway astrocytoma: an unusual cause of failure to thrive in infants]. An Pediatr (Barc); 2007 Jun;66(6):622-4
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  • [Title] [Optic pathway astrocytoma: an unusual cause of failure to thrive in infants].
  • [Transliterated title] Astrocitoma de vías ópticas: una causa infrecuente de retraso ponderal en el lactante.
  • Failure to thrive is a frequent cause of consultation in pediatric services.
  • The main objective in these patients is the early detection of an organic cause, if present.
  • We report a case of low-grade astrocytoma of the optic pathway in a 2-month-old child whose main symptoms at diagnosis were failure to thrive and anorexia.
  • Unfortunately, despite therapeutic efforts, the tumor showed local and metastatic progression refractory to chemotherapy.
  • [MeSH-major] Brain Neoplasms / diagnosis. Failure to Thrive / etiology. Optic Nerve Glioma / diagnosis

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  • (PMID = 17583627.001).
  • [ISSN] 1695-4033
  • [Journal-full-title] Anales de pediatría (Barcelona, Spain : 2003)
  • [ISO-abbreviation] An Pediatr (Barc)
  • [Language] spa
  • [Publication-type] Case Reports; English Abstract; Journal Article
  • [Publication-country] Spain
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3. Dalla Via P, Opocher E, Pinello ML, Calderone M, Viscardi E, Clementi M, Battistella PA, Laverda AM, Da Dalt L, Perilongo G: Visual outcome of a cohort of children with neurofibromatosis type 1 and optic pathway glioma followed by a pediatric neuro-oncology program. Neuro Oncol; 2007 Oct;9(4):430-7
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  • [Title] Visual outcome of a cohort of children with neurofibromatosis type 1 and optic pathway glioma followed by a pediatric neuro-oncology program.
  • We evaluated the visual outcome of a cohort of children with neurofibromatosis type 1 (NF1) and optic pathway glioma (OPG) treated according to standardized therapeutic guidelines.
  • The study population consisted of all consecutive patients with NF1 and OPG referred to a specialized pediatric neuro-oncology program between 1994 and 2004.
  • Treatment was instituted only in cases of progressive disease or clinical deterioration.
  • Treatment modalities were chemotherapy (based on vincristine/carboplatin) for children younger than 5 years and radiotherapy for all others.
  • At a median follow-up time of 78 months, seven patients had been treated with chemotherapy only, four with radiotherapy, and four with chemotherapy plus radiotherapy.
  • Eight patients were treated for progressive visual loss in the face of stable disease, five for tumor volume increase without visual deterioration, and two for symptomatic tumor volume increase.
  • At referral, six children had a visual acuity (VA) of < 30% in both eyes; eight children had 100% VA bilaterally.
  • At referral, the visual field (VF) could be assessed in three children: One had VF loss in both eyes, one had VF loss in one eye, and one had normal VF.
  • Among 11 children who had some visual function, three had VF loss in one eye and three in both eyes, and five had an intact VF.
  • In summary, among the 15 children treated, one had a definitive and two a mild improvement in VA.
  • In conclusion, the visual outcome of this selected cohort of NF1 patients with OPG is unsatisfactory.
  • A critical reappraisal of the therapeutic strategy adopted is needed.
  • [MeSH-major] Antineoplastic Combined Chemotherapy Protocols / adverse effects. Neurofibromatosis 1 / therapy. Optic Nerve Glioma / therapy. Radiotherapy / adverse effects. Vision Disorders / etiology
  • [MeSH-minor] Carboplatin / administration & dosage. Carboplatin / adverse effects. Child. Child, Preschool. Combined Modality Therapy. Contrast Sensitivity / drug effects. Contrast Sensitivity / radiation effects. Evoked Potentials, Visual. Female. Humans. Infant. Male. Vincristine / administration & dosage. Vincristine / adverse effects. Visual Fields / drug effects. Visual Fields / radiation effects


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4. Mahoney DH Jr, Cohen ME, Friedman HS, Kepner JL, Gemer L, Langston JW, James HE, Duffner PK, Kun LE: Carboplatin is effective therapy for young children with progressive optic pathway tumors: a Pediatric Oncology Group phase II study. Neuro Oncol; 2000 10;2(4):213-20
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  • [Title] Carboplatin is effective therapy for young children with progressive optic pathway tumors: a Pediatric Oncology Group phase II study.
  • The Pediatric Oncology Group conducted a phase II study to evaluate the activity of carboplatin in children 5 years or younger with progressive optic pathway tumors (OPTs).
  • Of the 51 patients accrued to this study, 1 was not eligible because the child was older than 6 years.
  • Twenty-one of 50 had evidence of neurofibromatosis type I (NF-1).
  • Therapy consisted of carboplatin 560 mg/m2 at 4-week intervals.
  • Patients with stable disease or better after two courses were continued on therapy for 18 months or until progressive disease.
  • Of the 50 eligible children, 39 had stable disease or better, and 34 completed the 18-month therapy.
  • Fifteen patients progressed during the 18-month therapy, and 6 patients progressed after completing therapy.
  • Carboplatin is active and safe for the treatment of young children with progressive OPTs.
  • The addition of other potentially active drugs may further increase the event-free survival for these children.
  • [MeSH-major] Antineoplastic Agents / administration & dosage. Antineoplastic Agents / adverse effects. Carboplatin / administration & dosage. Glioma / drug therapy. Optic Nerve Neoplasms / drug therapy
  • [MeSH-minor] Child, Preschool. Disease Progression. Female. Humans. Infant. Male. Neoplasm Recurrence, Local / physiopathology. Patient Selection. Survival Analysis. Treatment Outcome

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  • (PMID = 11265230.001).
  • [ISSN] 1522-8517
  • [Journal-full-title] Neuro-oncology
  • [ISO-abbreviation] Neuro-oncology
  • [Language] eng
  • [Grant] United States / NCI NIH HHS / CA / CA-33587; United States / NCI NIH HHS / CA / CA-28439; United States / NCI NIH HHS / CA / CA-20549; United States / NCI NIH HHS / CA / CA-33625; United States / NCI NIH HHS / CA / CA-29691; United States / NCI NIH HHS / CA / CA-31566; United States / NCI NIH HHS / CA / CA-69428; United States / NCI NIH HHS / CA / CA-28383; United States / NCI NIH HHS / CA / CA-28476; United States / NCI NIH HHS / CA / CA-25408; United States / NCI NIH HHS / CA / CA-15525; United States / NCI NIH HHS / CA / CA-32053; United States / NCI NIH HHS / CA / CA-05587; United States / NCI NIH HHS / CA / CA-30969; United States / NCI NIH HHS / CA / CA-29293; United States / NCI NIH HHS / CA / CA-15989; United States / NCI NIH HHS / CA / CA-03161; United States / NCI NIH HHS / CA / CA-41573; United States / NCI NIH HHS / CA / CA-29139; United States / NCI NIH HHS / CA / CA-15898; United States / NCI NIH HHS / CA / CA-69177; United States / NCI NIH HHS / CA / CA-07431
  • [Publication-type] Clinical Trial; Clinical Trial, Phase II; Journal Article; Multicenter Study; Research Support, U.S. Gov't, P.H.S.
  • [Publication-country] England
  • [Chemical-registry-number] 0 / Antineoplastic Agents; BG3F62OND5 / Carboplatin
  • [Other-IDs] NLM/ PMC1920597
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5. Vanderveen DK, Nihalani BR, Barron P, Anderson RL: Optic nerve sheath fenestration for an isolated optic nerve glioma. J AAPOS; 2009 Feb;13(1):88-90
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  • [Title] Optic nerve sheath fenestration for an isolated optic nerve glioma.
  • The treatment modalities for neurofibromatosis type 1-associated optic gliomas include chemotherapy, radiation therapy, and surgical excision.
  • The current recommendation is to consider treatment for an optic nerve glioma only if there is clear evidence of either ophthalmologic or radiographic progression with significant visual dysfunction.
  • We report a case of a child with neurofibromatosis type 1 and an isolated optic nerve glioma with documented progression and visual loss in which clinical signs improved and visual deterioration stabilized after optic nerve sheath fenestration.

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  • (PMID = 18930671.001).
  • [ISSN] 1528-3933
  • [Journal-full-title] Journal of AAPOS : the official publication of the American Association for Pediatric Ophthalmology and Strabismus
  • [ISO-abbreviation] J AAPOS
  • [Language] ENG
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] United States
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6. Mantadakis E, Raissaki M, Danilatou V, Kambourakis A, Stiakaki E, Kalmanti M: Remission of a chiasmatic glioma in a non-NF1 patient after brief chemotherapy with vincristine and carboplatin: case report and literature review. J Neurooncol; 2004 Mar-Apr;67(1-2):95-100
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  • [Title] Remission of a chiasmatic glioma in a non-NF1 patient after brief chemotherapy with vincristine and carboplatin: case report and literature review.
  • We describe the case of an 8-year-old girl without neurofibromatosis, who presented with total loss of vision on the left eye, due to a chiasmatic mass with imaging characteristics of glioma, accompanied by a second asymptomatic mass in the middle cranial fossa, along the intracranial route of the right trigeminal nerve.
  • The patient received a total of 10 weekly injections of vincristine and four injections of carboplatin every 3 weeks and achieved a very good partial response (97% volume reduction) after the nineth week of therapy with acceptable toxicity.
  • However, we believe the quick response accompanied by visual improvement was most likely due to chemotherapy.
  • [MeSH-major] Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Glioma / drug therapy. Optic Chiasm / pathology. Optic Nerve Neoplasms / drug therapy
  • [MeSH-minor] Antineoplastic Agents / administration & dosage. Carboplatin / administration & dosage. Child. Female. Humans. Magnetic Resonance Imaging. Neurofibromatoses / pathology. Vincristine / administration & dosage

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  • (PMID = 15072453.001).
  • [ISSN] 0167-594X
  • [Journal-full-title] Journal of neuro-oncology
  • [ISO-abbreviation] J. Neurooncol.
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / Antineoplastic Agents; 5J49Q6B70F / Vincristine; BG3F62OND5 / Carboplatin
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7. Gnekow AK, Kortmann RD, Pietsch T, Emser A: Low grade chiasmatic-hypothalamic glioma-carboplatin and vincristin chemotherapy effectively defers radiotherapy within a comprehensive treatment strategy -- report from the multicenter treatment study for children and adolescents with a low grade glioma -- HIT-LGG 1996 -- of the Society of Pediatric Oncology and Hematology (GPOH). Klin Padiatr; 2004 Nov-Dec;216(6):331-42
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  • [Title] Low grade chiasmatic-hypothalamic glioma-carboplatin and vincristin chemotherapy effectively defers radiotherapy within a comprehensive treatment strategy -- report from the multicenter treatment study for children and adolescents with a low grade glioma -- HIT-LGG 1996 -- of the Society of Pediatric Oncology and Hematology (GPOH).
  • BACKGROUND: Low grade gliomas arise in all CNS-locations and age groups, chiasmatic-hypothalamic tumors occur especially in young children.
  • Early radiotherapy (RT) shall be deferred by chemotherapy (CT) within the concept of the HIT-LGG 1996 study, offering a comprehensive treatment strategy for all age groups.
  • PATIENTS: 198 of 905 protocol patients (21.9 %) had a chiasmatic (34), chiasmatic-hypothalamic (144) or hypothalamic (20) primary tumor, median age at diagnosis 3.6 years (0.2-16.3 y.), 54 had neurofibromatosis (27.3 %), 108 female (54.5 %).
  • 98 children had severe visual impairment as their first symptom.
  • Histology showed 132 pilocytic astrocytoma I degrees , 6 astrocytoma II degrees /nos and 2 DIGG/DIA I degrees (3 not known).
  • RESULTS: 82 children were treated at diagnosis, 68 upon clinical or radiological progression following observation times of 3.0 to 115.0 months.
  • At a median observation time of 50.1 months 21 tumors are stable, 3 regressive (2 not evaluable, 1 death).
  • 44/123 tumors were progressive after median 22.5 months, 37 with a chiasmatic-hypothalamic primary, 16/44 were irradiated.
  • At a median observation time of 44.7 months 2 children are in complete remission, 92 tumors are stable, 8 regressive, 9 progressive.
  • CONCLUSION: Within the comprehensive treatment strategy for low grade glioma HIT-LGG 1996 chemotherapy is effective to delay the need for early radiotherapy in chiasmatic-hypothalamic glioma.
  • [MeSH-major] Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Brain Neoplasms / therapy. Glioma / therapy. Hypothalamus. Optic Chiasm. Optic Nerve Neoplasms / therapy
  • [MeSH-minor] Adolescent. Age Factors. Antineoplastic Agents / administration & dosage. Antineoplastic Agents, Phytogenic / administration & dosage. Carboplatin / administration & dosage. Child. Child, Preschool. Combined Modality Therapy. Data Interpretation, Statistical. Female. Follow-Up Studies. Humans. Infant. Male. Radiotherapy Dosage. Time Factors. Vincristine / administration & dosage

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  • (PMID = 15565548.001).
  • [ISSN] 0300-8630
  • [Journal-full-title] Klinische Pädiatrie
  • [ISO-abbreviation] Klin Padiatr
  • [Language] eng
  • [Publication-type] Clinical Trial; Comparative Study; Journal Article; Multicenter Study; Research Support, Non-U.S. Gov't
  • [Publication-country] Germany
  • [Chemical-registry-number] 0 / Antineoplastic Agents; 0 / Antineoplastic Agents, Phytogenic; 5J49Q6B70F / Vincristine; BG3F62OND5 / Carboplatin
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8. Silva MM, Goldman S, Keating G, Marymont MA, Kalapurakal J, Tomita T: Optic pathway hypothalamic gliomas in children under three years of age: the role of chemotherapy. Pediatr Neurosurg; 2000 Sep;33(3):151-8
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  • [Title] Optic pathway hypothalamic gliomas in children under three years of age: the role of chemotherapy.
  • OBJECTIVES: Optic pathway/hypothalamic gliomas (OPHGs) tend to occur in young children.
  • Treatment options consist of surgical resection, radiation therapy (RT) and chemotherapy.
  • Due to complications induced by surgery and RT, chemotherapy has gained significant recognition for the treatment of OPHG in young children.
  • We analyzed 14 patients who were treated with chemotherapy with or without surgery.
  • Five patients had partial tumor resection and 4 had endoscopic biopsy at the time of ventriculoperitoneal shunt placement.
  • All patients received chemotherapy: carboplatin in 8, a combination of carboplatin and vincristine in 4 and a combination of other agents in 2.
  • RESULTS: Eight (57%) of 14 patients had a sustained reduction of tumor during the follow-up time between 15 months and 8 years.
  • These tumor reductions were often accompanied by clinical improvements.
  • Diencephalic syndrome responded to chemotherapy alone in 4 of 6 patients.
  • However, 5 others had progressive disease; 3 during the treatment and 2 following the treatment (9 months and 2 years, respectively).
  • All these 5 patients had a partial tumor resection prior to chemotherapy.
  • CONCLUSION: A majority of OPHGs responds to chemotherapy.
  • Due to slow progression of these tumors and adverse effects of other therapeutic modalities, we recommend chemotherapy as a primary treatment for OPHGs.
  • Our present data indicates that partial surgical resection does not enhance chemotherapy effectiveness for OPHGs in infants or children younger than 3 years.
  • [MeSH-major] Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Hypothalamic Neoplasms / diagnosis. Hypothalamic Neoplasms / drug therapy. Optic Nerve Glioma / diagnosis. Optic Nerve Glioma / drug therapy
  • [MeSH-minor] Astrocytoma / drug therapy. Child, Preschool. Disease-Free Survival. Female. Humans. Infant. Magnetic Resonance Imaging. Male. Retrospective Studies. Survival Analysis. Treatment Outcome

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  • [Copyright] Copyright 2000 S. Karger AG, Basel
  • (PMID = 11096362.001).
  • [ISSN] 1016-2291
  • [Journal-full-title] Pediatric neurosurgery
  • [ISO-abbreviation] Pediatr Neurosurg
  • [Language] eng
  • [Publication-type] Comparative Study; Journal Article
  • [Publication-country] Switzerland
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9. Diaz RJ, Laughlin S, Nicolin G, Buncic JR, Bouffet E, Bartels U: Assessment of chemotherapeutic response in children with proptosis due to optic nerve glioma. Childs Nerv Syst; 2008 Jun;24(6):707-12
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  • [Title] Assessment of chemotherapeutic response in children with proptosis due to optic nerve glioma.
  • INTRODUCTION: Some children with optic pathway gliomas present with proptosis related to intraorbital tumor extension.
  • The radiological assessment of chemotherapeutic response in these patients can be complicated by irregular tumor shape and lack of relation between tumor volume and cosmetic effect.
  • METHOD: We propose that proptosis measurements and derivation of a proptosis index can be a useful adjunct to the measurement of tumor volume in the radiological assessment of chemotherapeutic response.
  • A series of six patients with proptosis and the diagnosis of an optic nerve tumor from an optic pathway glioma registry demonstrate by case example the correlation between the proptosis index and the clinical and radiographic response to chemotherapy.
  • [MeSH-major] Drug Therapy / methods. Exophthalmos / drug therapy. Exophthalmos / etiology. Optic Nerve Glioma / complications. Optic Nerve Neoplasms / complications. Outcome Assessment (Health Care) / methods
  • [MeSH-minor] Child, Preschool. Female. Humans. Infant. Magnetic Resonance Imaging / methods. Male. Retrospective Studies. Tomography Scanners, X-Ray Computed. Vision, Ocular / physiology

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  • [Cites] J Neurosurg. 1997 May;86(5):747-54 [9126887.001]
  • [Cites] Klin Padiatr. 2004 Nov-Dec;216(6):331-42 [15565548.001]
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  • (PMID = 18157537.001).
  • [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; Research Support, Non-U.S. Gov't
  • [Publication-country] Germany
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10. Kaufman LM, Doroftei O: Optic glioma warranting treatment in children. Eye (Lond); 2006 Oct;20(10):1149-64
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Optic glioma warranting treatment in children.
  • PURPOSE: To describe cases of optic pathway glioma (OPG) warranting treatment in children.
  • METHODS: This is a retrospective review of pediatric patients treated for OPG.
  • The clinical data and imaging studies were obtained from the medical records and radiology files of patients seen at the Pediatric Neuro-Ophthalmology Clinic at the University of Illinois, Chicago and the private office of the author (LMK).
  • Three of the patients were also ultimately diagnosed with neurofibromatosis type 1.
  • Six of the patients were treated with intravenous chemotherapy, with three patients requiring a second chemotherapy cycle.
  • One patient was successfully treated with an en-bloc optic nerve excision.
  • Both chemotherapy and en-bloc excision can be employed for treatment.
  • [MeSH-major] Optic Nerve Glioma / therapy
  • [MeSH-minor] Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Child, Preschool. Disease Progression. Exophthalmos / etiology. Eye Enucleation. Female. Humans. Infant. Magnetic Resonance Imaging. Male. Neoplasm Invasiveness. Neurofibromatosis 1 / complications. Neurofibromatosis 1 / diagnosis. Neurofibromatosis 1 / pathology. Neurofibromatosis 1 / therapy. Optic Nerve / pathology. Optic Nerve / surgery. Retrospective Studies. Treatment Outcome. Vision Disorders / etiology

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  • (PMID = 17019413.001).
  • [ISSN] 0950-222X
  • [Journal-full-title] Eye (London, England)
  • [ISO-abbreviation] Eye (Lond)
  • [Language] eng
  • [Grant] United States / NEI NIH HHS / EY / EY 1792
  • [Publication-type] Journal Article; Research Support, N.I.H., Extramural; Research Support, Non-U.S. Gov't
  • [Publication-country] England
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11. Jahraus CD, Tarbell NJ: Optic pathway gliomas. Pediatr Blood Cancer; 2006 May 1;46(5):586-96
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Optic pathway gliomas.
  • Optic pathway gliomas represent approximately 5% of all pediatric intracranial tumors.
  • Their propensity to occur in very young children and infants further complicates selection of therapy.
  • Historically, surgery and radiotherapy have played a primary role in management, however, in the last 15 years, chemotherapy has evolved into the first-line treatment of choice.
  • Nonetheless, chemotherapy frequently fails, but serves to delay implementation of radiotherapy or surgery until the child has progressed neuropsychologically.
  • An overall favorable prognosis for this tumor emphasizes the need for careful selection of therapy.
  • Herein, we review the major features of optic pathway glioma, including epidemiology, pathology, therapeutic interventions, outcome, and treatment sequelae.
  • [MeSH-major] Cranial Nerve Neoplasms. Optic Nerve Glioma
  • [MeSH-minor] Combined Modality Therapy. Humans. Treatment Outcome

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  • (PMID = 16411210.001).
  • [ISSN] 1545-5009
  • [Journal-full-title] Pediatric blood & cancer
  • [ISO-abbreviation] Pediatr Blood Cancer
  • [Language] eng
  • [Publication-type] Journal Article; Review
  • [Publication-country] United States
  • [Number-of-references] 110
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12. Shamji MF, Benoit BG: Syndromic and sporadic pediatric optic pathway gliomas: review of clinical and histopathological differences and treatment implications. Neurosurg Focus; 2007;23(5):E3
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Syndromic and sporadic pediatric optic pathway gliomas: review of clinical and histopathological differences and treatment implications.
  • Optic pathway gliomas (OPGs) are the most common primary neoplasm of the optic pathway.
  • These lesions usually present in childhood and can arise anywhere along the optic pathway; they occur more frequently in women; and they rarely undergo late progression.
  • Neurofibromatosis Type 1 (NF1), with aberrant oncogenic signaling and consequent predisposition to intracranial tumors, is the most common associated syndrome, with nearly 20% of NF1 patients developing OPGs.
  • A comorbid NF1 diagnosis has implications for tumor location with greater predilection for optic nerve involvement, whereas chiasmal and postchiasmal lesions are more frequently seen in sporadic cases.
  • When treatment is indicated, however, the molecular abnormalities that constitute this syndrome can limit the available chemotherapy and radiotherapy options because clinicians fear secondary malignancy and cerebrovascular complications.
  • Furthermore, radiotherapy early in life can impair an individual's intellectual development, endocrine function, and physical growth, thereby limiting the role of this modality in the treatment of this childhood lesion.
  • Differential gene expression and histogenesis among sporadic and syndromic OPGs may account for the different tumor behaviors, but studies correlating specific genetic and proteomic changes with patient outcome are pending.
  • Loss of heterozygosity at 10 and 17q are more common among patients with NF1, and Ki67 labeling intensity of 2-3% and low p53 labeling intensity seem prognostic of aggressive tumor behavior.
  • Recent advances in the development of a preclinical mouse model of NF1-associated OPG will permit investigation into improved detection strategies and chemotherapeutic and radiotherapy treatment protocols.
  • [MeSH-major] Glioma / diagnosis. Glioma / therapy. Optic Nerve Neoplasms / diagnosis. Optic Nerve Neoplasms / therapy
  • [MeSH-minor] Animals. Antineoplastic Agents / therapeutic use. Child. Diagnostic Imaging / methods. Disease-Free Survival. Female. Follow-Up Studies. Humans. Male. Mass Screening / instrumentation. Mass Screening / methods. Neoplasm Regression, Spontaneous. Neurofibromatosis 1 / complications. Neurofibromatosis 1 / diagnosis. Prognosis. Radiotherapy, Adjuvant. Survival Rate

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  • (PMID = 18004965.001).
  • [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 / Antineoplastic Agents
  • [Number-of-references] 92
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13. Campagna M, Opocher E, Viscardi E, Calderone M, Severino SM, Cermakova I, Perilongo G: Optic pathway glioma: long-term visual outcome in children without neurofibromatosis type-1. Pediatr Blood Cancer; 2010 Dec 1;55(6):1083-8
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  • [Title] Optic pathway glioma: long-term visual outcome in children without neurofibromatosis type-1.
  • BACKGROUND: Little is known about the visual outcome of children affected by an optic pathway glioma (OPG).
  • PROCEDURES: We evaluated the long-term visual outcome of 32 consecutive children affected by OPG without neurofibromatosis type-1 referred to the Pediatric Department of Padua University and managed according to standardized strategies.
  • RESULTS: Eight children received chemotherapy, 10 radiotherapy, 7 both chemotherapy and radiotherapy, whereas 7 were untreated.
  • At presentation, visual acuity (VA) was normal in 22 children (13 unilaterally and 9 bilaterally), and reduced in 10.
  • Visual field, assessed in 29 children, was normal in 9 and reduced in 20.
  • The number of children with some grade of visual impairment increased from 7 to 10 during follow-up.
  • Of the 17 children in whom the tumor became significantly smaller, VA improved in 6, was stable in 3, and worse in 8.
  • CONCLUSIONS: The visual prognosis of children with OPG is unsatisfactory.
  • Older children treated with radiotherapy seem to have a better visual outcome than younger children.
  • Severe optic pallor at diagnosis or during follow-up may be indicative of a negative prognosis.
  • [MeSH-major] Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Optic Nerve Glioma / physiopathology. Optic Nerve Neoplasms / physiopathology. Visual Acuity / physiology. Visual Fields / physiology. Visual Pathways / physiopathology
  • [MeSH-minor] Adolescent. Child. Child, Preschool. Combined Modality Therapy. Female. Humans. Infant. Male. Neurofibromatosis 1 / physiopathology. Radiotherapy Dosage. Treatment Outcome

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  • (PMID = 20979170.001).
  • [ISSN] 1545-5017
  • [Journal-full-title] Pediatric blood & cancer
  • [ISO-abbreviation] Pediatr Blood Cancer
  • [Language] eng
  • [Publication-type] Clinical Trial; Journal Article; Research Support, Non-U.S. Gov't
  • [Publication-country] United States
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14. Nicolin G, Parkin P, Mabbott D, Hargrave D, Bartels U, Tabori U, Rutka J, Buncic JR, Bouffet E: Natural history and outcome of optic pathway gliomas in children. Pediatr Blood Cancer; 2009 Dec 15;53(7):1231-7
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Natural history and outcome of optic pathway gliomas in children.
  • BACKGROUND: The optimal management of optic pathway gliomas (OPGs) is complicated by their variable natural history, the association with neurofibromatosis type 1 (NF1) and difficulties in defining progression and response to treatment.
  • RESULTS: Of the 133 children included, 78 (59%) had NF1; 87 (71 NF1) were observed initially, of whom 23 (11 NF1) subsequently required treatment.
  • Forty-six patients received immediate treatment.
  • Initial treatment, without or with an observation period, comprised chemotherapy alone (32, 11 NF1); debulking + chemotherapy (15, 4 NF1); gross total resection (6); radiotherapy (2); debulking + radiotherapy (3); and debulking only (12, 3 NF1).
  • Four children died (overall survival at 5 and 10 years was 97.6% and 94.6% for those who required treatment).
  • Progression-free survival (PFS) for the 69 patients who needed treatment was 48%.
  • There was no difference in PFS between chemotherapy versus chemotherapy + debulking or debulking alone.
  • PFS for the NF1 patients who required treatment was similar to that of non-NF1 patients.
  • Mean follow-up time was 9.0 (range 0.6-18.0, median 8.6) years.
  • CONCLUSIONS: The study confirms the complexity of OPGs and that NF1 is a major determinant of the resultant behavior of the tumor.
  • [MeSH-major] Optic Nerve Glioma / epidemiology
  • [MeSH-minor] Adolescent. Antineoplastic Combined Chemotherapy Protocols / adverse effects. Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Brain Damage, Chronic / etiology. Carboplatin / administration & dosage. Child. Child, Preschool. Combined Modality Therapy. Cranial Irradiation / adverse effects. Disease Progression. Disease-Free Survival. Female. Follow-Up Studies. Humans. Infant. Intellectual Disability / etiology. Lomustine / administration & dosage. Male. Neurofibromatosis 1 / epidemiology. Procarbazine / administration & dosage. Retrospective Studies. Survival Analysis. Thioguanine / administration & dosage. Treatment Outcome. Vinblastine / administration & dosage. Vincristine / administration & dosage. Vision Disorders / etiology

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  • [Copyright] (c) 2009 Wiley-Liss, Inc.
  • (PMID = 19621457.001).
  • [ISSN] 1545-5017
  • [Journal-full-title] Pediatric blood & cancer
  • [ISO-abbreviation] Pediatr Blood Cancer
  • [Language] eng
  • [Publication-type] Journal Article; Research Support, Non-U.S. Gov't
  • [Publication-country] United States
  • [Chemical-registry-number] 35S93Y190K / Procarbazine; 5J49Q6B70F / Vincristine; 5V9KLZ54CY / Vinblastine; 7BRF0Z81KG / Lomustine; BG3F62OND5 / Carboplatin; FTK8U1GZNX / Thioguanine
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15. Massimi L, Tufo T, Di Rocco C: Management of optic-hypothalamic gliomas in children: still a challenging problem. Expert Rev Anticancer Ther; 2007 Nov;7(11):1591-610

  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Management of optic-hypothalamic gliomas in children: still a challenging problem.
  • Optic pathway-hypothalamic gliomas (OPHGs) are rare, often unresectable tumors that mostly occur in childhood.
  • Their biological behavior is unpredictable, although they tend to follow an aggressive clinical course in infants and a benign course in children with neurofibromatosis type 1.
  • Surgery is advocated for progressing tumors to decompress the optic pathways, obtain a quick relief from intracranial hypertension and allow histologic examination (when needed).
  • Chemotherapy is increasingly used in the management of OPHGs, especially in infants, to delay radiotherapy.
  • Carboplatin and vincristine are the most frequently used drugs, although several chemotherapeutic agents in different combinations are currently employed with good results.
  • [MeSH-major] Hypothalamic Neoplasms / therapy. Optic Nerve Glioma / therapy
  • [MeSH-minor] Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Child. Combined Modality Therapy. Humans

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  • (PMID = 18020927.001).
  • [ISSN] 1744-8328
  • [Journal-full-title] Expert review of anticancer therapy
  • [ISO-abbreviation] Expert Rev Anticancer Ther
  • [Language] eng
  • [Publication-type] Journal Article; Review
  • [Publication-country] England
  • [Number-of-references] 148
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16. Zeid JL, Charrow J, Sandu M, Goldman S, Listernick R: Orbital optic nerve gliomas in children with neurofibromatosis type 1. J AAPOS; 2006 Dec;10(6):534-9
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Orbital optic nerve gliomas in children with neurofibromatosis type 1.
  • PURPOSE: To describe the clinical course and treatment of symptomatic orbital optic nerve gliomas in children with neurofibromatosis type-1 (NF-1).
  • METHODS: A retrospective review of the records of patients with NF-1 and symptomatic orbital optic nerve gliomas seen in a large multidisciplinary NF-1 clinic of a tertiary care children's hospital.
  • The main outcome measures included presenting symptoms and signs, ophthalmologic examination at diagnosis, the presence of progressive disease following diagnosis, type of therapy, and the reasons therapy was instituted.
  • RESULTS: Twelve patients with symptomatic orbital optic nerve gliomas, all of which led to proptosis (eight girls, four boys), were identified.
  • The mean age of diagnosis of NF-1 was 20 months; the mean age of diagnosis of the orbital optic nerve glioma was 26 months.
  • At the time of diagnosis of the tumor, 10 of 12 patients (83%) had decreased visual acuity in the affected eye.
  • Three patients underwent optic nerve resection; eight received chemotherapy, and one was observed without therapy.
  • Of the eight children who received chemotherapy, progressive disease prior to treatment could be documented in only three; none of these eight children had a reproducible improvement in vision following chemotherapy.
  • There was no demonstrable improvement in vision in any treated patient with NF-1-associated orbital optic nerve gliomas.
  • CONCLUSIONS: Although not definitively proven, our data and previous studies suggest that NF-1-associated orbital optic nerve gliomas should not be treated unless there is clear evidence of either ophthalmologic or radiographic progression.

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  • (PMID = 17189147.001).
  • [ISSN] 1091-8531
  • [Journal-full-title] Journal of AAPOS : the official publication of the American Association for Pediatric Ophthalmology and Strabismus
  • [ISO-abbreviation] J AAPOS
  • [Language] ENG
  • [Publication-type] Journal Article; Research Support, Non-U.S. Gov't
  • [Publication-country] United States
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17. Sims-McCallum RP: Outpatient carboplatin desensitization in a pediatric patient with bilateral optic glioma. Ann Pharmacother; 2000 Apr;34(4):477-8
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Outpatient carboplatin desensitization in a pediatric patient with bilateral optic glioma.
  • OBJECTIVE: To report a case of successful outpatient carboplatin desensitization in a pediatric patient with bilateral optic glioma.
  • CASE SUMMARY: A 10-year-old white girl with bilateral optic glioma developed a hypersensitivity reaction to carboplatin after nine courses.
  • A desensitization regimen was administered, and she has tolerated all subsequent courses of carboplatin therapy.
  • DISCUSSION: Carboplatin is an important chemotherapeutic agent in the treatment of a variety of pediatric brain tumors.
  • Hypersensitivity to this agent appears to develop after frequent exposure such as with the once-weekly regimens often used to treat brain tumors in pediatric patients.
  • This regimen could prove useful for other patients who develop hypersensitivity reactions to carboplatin and allow therapy to continue.
  • [MeSH-major] Antineoplastic Agents / therapeutic use. Carboplatin / therapeutic use. Optic Nerve Glioma / drug therapy
  • [MeSH-minor] Child. Drug Administration Schedule. Drug Hypersensitivity / etiology. Female. Humans

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  • (PMID = 10772434.001).
  • [ISSN] 1060-0280
  • [Journal-full-title] The Annals of pharmacotherapy
  • [ISO-abbreviation] Ann Pharmacother
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / Antineoplastic Agents; BG3F62OND5 / Carboplatin
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18. Piccirilli M, Lenzi J, Delfinis C, Trasimeni G, Salvati M, Raco A: Spontaneous regression of optic pathways gliomas in three patients with neurofibromatosis type I and critical review of the literature. Childs Nerv Syst; 2006 Oct;22(10):1332-7
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Spontaneous regression of optic pathways gliomas in three patients with neurofibromatosis type I and critical review of the literature.
  • CASE REPORTS: The authors report their experience about three children (two girls, one boy; average age 1.6 years) with a spontaneous regression of optic gliomas.
  • All of them had a previous diagnosis of neurofibromatosis type 1 (NF 1).
  • None of them underwent surgery or biopsy nor received chemotherapy or radiotherapy.
  • LITERATURE REVIEW: Moreover, the authors analyze the features of the 16 cases previously reported in English literature of spontaneously regressed optic gliomas with an overview of the different therapeutic strategies.
  • The knowledge that this kind of tumor, particularly in young patients, may regress is important in the decision of the best therapeutic approach.
  • [MeSH-major] Neurofibromatosis 1 / complications. Optic Nerve Glioma / complications. Optic Nerve Neoplasms / complications
  • [MeSH-minor] Child, Preschool. Female. Humans. Magnetic Resonance Imaging / methods. Male. Review Literature as Topic

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  • (PMID = 16639629.001).
  • [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] Case Reports; Journal Article; Review
  • [Publication-country] Germany
  • [Number-of-references] 32
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19. Steinbok P: Optic pathway tumors in children. J Chin Med Assoc; 2003 Jan;66(1):4-12
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Optic pathway tumors in children.
  • Tumors of the optic pathways are sub-divided in this review into those that arise in one or both optic nerves anterior to the chiasm (optic nerve tumors); those that arise within the chiasm and do not extend significantly into the hypothalamus (optic chiasmatic tumors) and the large exophytic tumors that involve both the optic chiasm and the hypothalamus to a lesser or greater degree (optic chiasmatic/hypothalamic tumors).
  • The management of optic chiasmatic gliomas is controversial, partly related to failure to separate out chiasmatic tumors from the chiasmatic/hypothalamic tumors.
  • The optic nerve tumors are reviewed briefly, since they rarely extend intracranially.
  • On the other hand, chiasmatic/hypothalamic tumors grow like typical neoplasms.
  • Modern management has trended away from radical surgical resection, which has significant morbidity, to chemotherapy as the first line of treatment.
  • In this review, the clinical presentation and management of different types of optic pathway tumors are discussed.
  • [MeSH-major] Hypothalamic Neoplasms / therapy. Optic Chiasm. Optic Nerve Neoplasms / therapy
  • [MeSH-minor] Child. Glioma / diagnosis. Glioma / therapy. Humans

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  • (PMID = 12728968.001).
  • [ISSN] 1726-4901
  • [Journal-full-title] Journal of the Chinese Medical Association : JCMA
  • [ISO-abbreviation] J Chin Med Assoc
  • [Language] eng
  • [Publication-type] Journal Article; Review
  • [Publication-country] China (Republic : 1949- )
  • [Number-of-references] 34
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20. Akiyama H, Nakamizo S, Kawamura A, Nagashima T, Takeda H, Hasegawa D, Kosaka Y, Yoshida M: [Management of chiasmatic-hypothalamic gliomas in children: report of nine pediatric cases]. No Shinkei Geka; 2007 Nov;35(11):1079-85
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] [Management of chiasmatic-hypothalamic gliomas in children: report of nine pediatric cases].
  • Radical resection of chiasmatic-hypothalamic glioma (CHG) carries a significant risk of morbidity and the optimum treatment remains undecided.
  • The authors reported 9 children with CHG, who were treated with surgical resection with or without postoperative chemotherapy.
  • Age at the time of diagnosis ranged from 4 months to 7.7 years (mean 3.1 years), and no patient had evidence of neurofibromatosis type 1.
  • Surgical resections of the tumors were performed in all patients because of severe visual impairment or intracranial hypertension caused by large tumors.
  • Seven patients with residual tumors received postoperative chemotherapy consisting of cisplatin, cyclophosphamide, etoposide and vincristine.
  • Reduction in tumor size was noticed in 5 patients, although 2 patients had no response and switched to local radiotherapy.
  • Although minor complications of chemotherapy were noticed in 5 patients, severe sequelae such as neuropsychological deficits or endocrinopathies did not occur, and all patients completed chemotherapy programs.
  • Additional treatments are recommended in case of incomplete tumor resections, because our experience demonstrates that the majority of the residual tumors have potential to progress.
  • Our present data suggests that the chemotherapy of the aforementioned regimen is effective in controlling CHGs after partial resections and is relatively well tolerated even in young children who are vulnerable to radiotherapy.
  • [MeSH-major] Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Glioma / surgery. Hypothalamic Neoplasms / surgery. Optic Chiasm. Optic Nerve Glioma / surgery
  • [MeSH-minor] Child. Child, Preschool. Cisplatin / administration & dosage. Combined Modality Therapy. Cyclophosphamide / administration & dosage. Drug Administration Schedule. Etoposide / administration & dosage. Female. Humans. Infant. Male. Vincristine / administration & dosage

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  • (PMID = 18044225.001).
  • [ISSN] 0301-2603
  • [Journal-full-title] No shinkei geka. Neurological surgery
  • [ISO-abbreviation] No Shinkei Geka
  • [Language] jpn
  • [Publication-type] Case Reports; English Abstract; Journal Article
  • [Publication-country] Japan
  • [Chemical-registry-number] 5J49Q6B70F / Vincristine; 6PLQ3CP4P3 / Etoposide; 8N3DW7272P / Cyclophosphamide; Q20Q21Q62J / Cisplatin
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21. Mitchell AE, Elder JE, Mackey DA, Waters KD, Ashley DM: Visual improvement despite radiologically stable disease after treatment with carboplatin in children with progressive low-grade optic/thalamic gliomas. J Pediatr Hematol Oncol; 2001 Dec;23(9):572-7
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Visual improvement despite radiologically stable disease after treatment with carboplatin in children with progressive low-grade optic/thalamic gliomas.
  • BACKGROUND: The purpose of this study was to examine the clinical and radiologic response to carboplatin by children with progressive optic/thalamic gliomas.
  • PATIENTS AND METHODS: Between July 1997 and July 1999, 12 consecutive children were treated with monthly carboplatin for progressive optic/thalamic gliomas.
  • RESULTS: Five children have completed 12 cycles of carboplatin and five children are currently receiving treatment.
  • Nine children have stable radiologic disease and one child has had a partial radiologic response to chemotherapy.
  • Eight children have had regular visual assessments.
  • One child has had deterioration in vision despite radiologically stable disease.
  • CONCLUSIONS: The results suggest that the clinical response of optic/thalamic gliomas to carboplatin, as measured by visual acuity and visual fields, may be better than predicted by radiologic assessment.
  • These data suggest that a prospective clinical study is warranted of the role of carboplatin in children with progressive optic/thalamic gliomas and visual impairment.
  • [MeSH-major] Antineoplastic Agents, Alkylating / therapeutic use. Brain Neoplasms / drug therapy. Carboplatin / therapeutic use. Glioma / drug therapy. Optic Chiasm. Optic Nerve Neoplasms / drug therapy. Thalamus. Vision Disorders / etiology
  • [MeSH-minor] Adolescent. Anemia / chemically induced. Child. Child, Preschool. Combined Modality Therapy. Disease Progression. Female. Humans. Infusions, Intravenous. Male. Neurofibromatosis 1 / complications. Neutropenia / chemically induced. Remission Induction. Retrospective Studies. Thrombocytopenia / chemically induced. Treatment Outcome. Visual Acuity. Visual Fields

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  • (PMID = 11902299.001).
  • [ISSN] 1077-4114
  • [Journal-full-title] Journal of pediatric hematology/oncology
  • [ISO-abbreviation] J. Pediatr. Hematol. Oncol.
  • [Language] eng
  • [Publication-type] Journal Article; Review
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / Antineoplastic Agents, Alkylating; BG3F62OND5 / Carboplatin
  • [Number-of-references] 25
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22. Liang CL, Lu K, Liliang PC, Chen HJ: Gamma Knife surgery for optic glioma. Report of 2 cases. J Neurosurg; 2010 Dec;113 Suppl:44-7
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Gamma Knife surgery for optic glioma. Report of 2 cases.
  • Optic pathway/hypothalamic gliomas represent approximately 2%-5% of brain tumors in children.
  • Total excision, subtotal excision, subtotal excision followed by irradiation, radiation therapy alone, chemotherapy, and no treatment at all have been reported.
  • In this article the authors discuss the results of Gamma Knife surgery (GKS) for optic gliomas in 2 children.
  • Two pediatric patients, a boy and a girl, underwent GKS for optic gliomas at our hospital between March 2005 and August 2005.
  • The tumor involved the optic chiasm in 1 patient and the right optic nerve in the other patient.
  • Treatments were planned with the prescription of 11 Gy to the 50% isodose line for the optic chiasm glioma and 15 Gy to the 50% isodose line for the optic nerve glioma.
  • During the follow-up period, neither of the patients developed any endocrine dysfunction.
  • Gamma Knife surgery permits treatment of optic glioma with good tumor control and no clinically relevant morbidity.
  • With the ability to deliver a high dose to the tumor while sparing normal brain tissue, especially the optic nerve, optic chiasm, and pituitary gland, GKS should be the choice of treatment for optic gliomas.
  • [MeSH-major] Astrocytoma / surgery. Optic Nerve / surgery. Optic Nerve Glioma / surgery. Radiosurgery / instrumentation
  • [MeSH-minor] Adolescent. Child. Female. Humans. Magnetic Resonance Imaging. Male. Treatment Outcome

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  • (PMID = 21121786.001).
  • [ISSN] 1933-0693
  • [Journal-full-title] Journal of neurosurgery
  • [ISO-abbreviation] J. Neurosurg.
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] United States
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23. Suárez JC, Viano JC, Zunino S, Herrera EJ, Gomez J, Tramunt B, Marengo I, Hiramatzu E, Miras M, Pena M, Sonzini Astudillo B: Management of child optic pathway gliomas: new therapeutical option. Childs Nerv Syst; 2006 Jul;22(7):679-84

  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Management of child optic pathway gliomas: new therapeutical option.
  • OBJECTIVE: To present our experience in the treatment of child optic pathway gliomas in the last 25 years.
  • The most frequent symptoms have been ophthalmologic and visual alterations in all 17 patients, endocrine alterations in 10, and neurological signs in 6.
  • One of the patients presented neurofibromatosis type 1 (NF1), another patient had Down syndrome.
  • Diagnosed using computed tomography or/and magnetic resonance imaging, histological studies showed pilocytic astrocytomas in 13 cases and a fibrillary astrocytoma grade II in 1 case.
  • The treatment consisted of surgery, external beam radiotherapy, chemotherapy, and brachytherapy with iodine 125, separately or combined.
  • Five patients died; the causes were secondary tumors in two children, tumor recurrence in one, sepsis secondary to respiratory and urinary tract infections in the child with Down syndrome, and finally, hydrocephaly due to hyperproteinorachia of tumor origin in one.
  • CONCLUSION: Chemotherapy and brachytherapy are therapeutic methods to be considered, especially in children under 5.
  • Marsupialization of the residual cyst into the ventricular system postradio or oncolytic treatment through endoscopic or stereotactic techniques is useful in the treatment of endocranial hypertension and/or hypothalamic compression in these patients.
  • [MeSH-major] Glioma / therapy. Optic Nerve Glioma / therapy. Optic Nerve Neoplasms / therapy
  • [MeSH-minor] Child. Child, Preschool. Female. Humans. Infant. Magnetic Resonance Imaging / methods. Male. Neurosurgery. Radiotherapy. Tomography, X-Ray Computed / methods

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  • (PMID = 16389565.001).
  • [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; Comparative Study; Journal Article
  • [Publication-country] Germany
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24. Jaing TH, Lin KL, Tsay PK, Hsueh C, Hung PC, Wu CT, Tseng CK: Treatment of optic pathway hypothalamic gliomas in childhood: experience with 18 consecutive cases. J Pediatr Hematol Oncol; 2008 Mar;30(3):222-4

  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Treatment of optic pathway hypothalamic gliomas in childhood: experience with 18 consecutive cases.
  • The aim of this study was to present our 17-year experience (1989 to 2006) in the treatment of optic pathway/hypothalamic gliomas (OPHG) in 18 children younger than 17 years (median age, 66 mo).
  • OPHG was diagnosed using computed tomography and/or magnetic resonance imaging.
  • Treatment included partial tumor resection in 12 patients, chemotherapy in 5, and radiotherapy in 3.
  • Ophthalmologic and visual alterations occurred in 12 patients, endocrine alterations in 6, and neurologic signs in 5.
  • All treatment modalities led to tumor shrinkage and stabilization for a variable period, but none of them totally eradicated the tumor.
  • Fourteen (78%) of 18 patients had a sustained reduction of tumor size between 6 months and 17 years.
  • Two patients died, none with neurofibromatosis-1, with a hypothalamic/chiasmatic tumor with suprasellar extension and accompanying electrolyte abnormalities.
  • Because progression of these tumors is slow and associated with endocrinopathy, we recommend chemotherapy as a primary treatment of OPHG if the disease progresses.
  • [MeSH-major] Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Hypothalamic Neoplasms / therapy. Optic Nerve Glioma / therapy. Visual Pathways / pathology
  • [MeSH-minor] Adolescent. Child. Child, Preschool. Disease Progression. Female. Follow-Up Studies. Humans. Infant. Magnetic Resonance Imaging. Male. Predictive Value of Tests. Retrospective Studies. Survival Rate. Treatment Outcome

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  • (PMID = 18376285.001).
  • [ISSN] 1077-4114
  • [Journal-full-title] Journal of pediatric hematology/oncology
  • [ISO-abbreviation] J. Pediatr. Hematol. Oncol.
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] United States
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25. Shofty B, Constantini S, Freedman S, Ben-Sira L, Kesler A: [Optic pathway gliomas--current position and future directions]. Harefuah; 2010 Nov;149(11):721-5, 748

  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] [Optic pathway gliomas--current position and future directions].
  • Optic pathway gliomas (OPG) are the most common primary tumors of the visual pathways, and constitute 1% of all brain tumors and 5% of all brain tumors in children.
  • Among Neurofibromatosis type 1 patients (a hereditary genetic disorder which is characterized by higher incidence of tumors from a neurocutaneous origin) it is the most frequent tumor and it constitutes between 15 to 20 percent of all nervous system tumors.
  • These tumors are stable most of the time and remain indolent for many years after diagnosis, especially in patients suffering from Neurofibromatosis type 1.
  • However, amongst some of the patients suffering from OPG, these tumors develop progressive characteristics and can cause visual disturbances, endocrine dysfunction, blindness and even death.
  • Patients with aggressive tumors will need treatment, which can be either surgery, chemotherapy or radiation therapy.
  • Today, the treating physicians face substantial difficulty in estimating the course the tumor will take, choosing the right candidates for oncological treatment and the type of therapy most suited to the case, due to lack of reliable information in the relevant literature.
  • This article characterizes the tumors, presents updates from recent literature, as well as recommendations for treatment and follow-up.
  • [MeSH-major] Neurofibromatosis 1 / surgery. Optic Nerve Glioma / surgery
  • [MeSH-minor] Adolescent. Child. Glioma / drug therapy. Glioma / radiography. Glioma / radiotherapy. Glioma / surgery. Humans. Magnetic Resonance Imaging

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  • (PMID = 21250414.001).
  • [ISSN] 0017-7768
  • [Journal-full-title] Harefuah
  • [ISO-abbreviation] Harefuah
  • [Language] heb
  • [Publication-type] English Abstract; Journal Article; Review
  • [Publication-country] Israel
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26. Marec-Berard P, Szathmari A, Conter C, Mottolese C, Berlier P, Frappaz D: Improvement of diencephalic syndrome after partial surgery of optic chiasm glioma. Pediatr Blood Cancer; 2009 Sep;53(3):502-4
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Improvement of diencephalic syndrome after partial surgery of optic chiasm glioma.
  • MRI showed a chiasma of the hypothalamic mass.
  • Three cycles of chemotherapy were given, resulting in stable disease on MRI, but growth failure despite attempts at enteral feeding.
  • A 30% tumor reduction was observed on post-operative imaging.
  • After surgery, the child gained weight and his growth curve returned to normal.
  • [MeSH-major] Hypothalamic Diseases / etiology. Optic Chiasm. Optic Nerve Glioma / complications. Optic Nerve Glioma / surgery

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  • [Copyright] (c) 2009 Wiley-Liss,
  • (PMID = 19489055.001).
  • [ISSN] 1545-5017
  • [Journal-full-title] Pediatric blood & cancer
  • [ISO-abbreviation] Pediatr Blood Cancer
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] United States
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27. Peyrl A, Azizi A, Czech T, Gruber-Olipitz M, Jones N, Haberler C, Prayer D, Autzinger E, Slavc I: Tumor stabilization under treatment with imatinib in progressive hypothalamic-chiasmatic glioma. Pediatr Blood Cancer; 2009 Apr;52(4):476-80
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Tumor stabilization under treatment with imatinib in progressive hypothalamic-chiasmatic glioma.
  • BACKGROUND: Hypothalamic-chiasmatic gliomas (HCG) account for up to 20% of tumors in patients under the age of 3 years.
  • While most children respond to chemotherapy, alternative treatment approaches are needed for those with progressive disease refractory to chemotherapy.
  • PROCEDURE: Six patients (median age: 5.5 years) with progressive HCG were treated with imatinib for 3-29 months at a median daily oral dose of 270 mg/m(2).
  • Disease control lasted from 5 to 46 months and was sustained longer in comparison to their last prior chemotherapy.
  • Immunohistochemistry in our patients' tumor cells revealed focal expression of arg and PDGFR-alpha in one patient, in the remaining five patients no expression of any of the five known targets of imatinib could be detected.
  • Expression of PDGFR-alpha and PDGFR-beta was detected in endothelial cells of tumor capillaries of all six patients.
  • CONCLUSIONS: We conclude that imatinib has possible activity in progressive HCG and may present an additional therapeutic option for patients who are too young or whose tumor is too extensive to receive radiotherapy.
  • [MeSH-major] Antineoplastic Agents / therapeutic use. Glioma / drug therapy. Hypothalamic Neoplasms / drug therapy. Optic Nerve Glioma / drug therapy. Piperazines / therapeutic use. Pyrimidines / therapeutic use
  • [MeSH-minor] Benzamides. Child, Preschool. Female. Humans. Imatinib Mesylate. Immunohistochemistry. Infant. Male. Neoplasm Recurrence, Local / drug therapy. Optic Chiasm / drug effects. Optic Chiasm / pathology. Treatment Outcome

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  • [Copyright] Copyright 2008 Wiley-Liss, Inc.
  • (PMID = 19061223.001).
  • [ISSN] 1545-5017
  • [Journal-full-title] Pediatric blood & cancer
  • [ISO-abbreviation] Pediatr Blood Cancer
  • [Language] eng
  • [Publication-type] Clinical Trial; Journal Article
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / Antineoplastic Agents; 0 / Benzamides; 0 / Piperazines; 0 / Pyrimidines; 8A1O1M485B / Imatinib Mesylate
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28. Ogura T, Adachi J, Nishikawa R, Hirose T, Matsutani M: Synchronous optic and pineal pilocytic astrocytomas in a paediatric patient with neurofibromatosis type 1. Pediatr Neurosurg; 2004 Nov-Dec;40(6):301-5
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Synchronous optic and pineal pilocytic astrocytomas in a paediatric patient with neurofibromatosis type 1.
  • A 12-year-old girl with neurofibromatosis type 1 presented with headache, visual acuity and visual field disturbance.
  • Computed tomography and magnetic resonance imaging revealed an enhanced solid mass involving her right optic nerve and optic chiasm, and a cystic lesion in the pineal region that had resulted in obstructive hydrocephalus.
  • An open biopsy of the right optic nerve tumour was performed, and it was histologically identified as a pilocytic astrocytoma.
  • Local irradiation of 50 Gy to the optic pathway tumour was performed, and the tumour has remained stable for more than 29 months.
  • Chemotherapy with cisplatin and vincristine was performed after a second surgery, and the pineal tumour has not re-grown in 18 months.
  • To our knowledge, this is the first case report to describe synchronous optic and pineal pilocytic astrocytomas associated with neurofibromatosis type 1.
  • [MeSH-major] Astrocytoma / etiology. Brain Neoplasms / etiology. Neurofibromatosis 1 / complications. Optic Nerve Glioma / etiology. Pineal Gland
  • [MeSH-minor] Child. Female. Humans


29. Allen JC: Initial management of children with hypothalamic and thalamic tumors and the modifying role of neurofibromatosis-1. Pediatr Neurosurg; 2000 Mar;32(3):154-62
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Initial management of children with hypothalamic and thalamic tumors and the modifying role of neurofibromatosis-1.
  • Optic pathway/hypothalamus gliomas (OPG) arise primarily from a slower-growing juvenile pilocytic astrocytoma, and thalamic gliomas arise primarily from a fibrillary astrocytoma which can become clinically and histologically more aggressive.
  • The major therapeutic challenge for these patients is to maximize their quality of life by preserving visual and endocrine function while minimizing treatment-related morbidity.
  • Treatment is often initiated at diagnosis in infants and toddlers who have a major visual impairment or the diencephalic syndrome.
  • The judicious application of chemotherapy may serve to forestall the need for radiotherapy or surgery.
  • However, over 90% of children with OPG without NF-1 will require some form of therapy.
  • Surgical intervention is often required to relieve intracranial pressure and establish the histologic identity of the tumor.
  • Current multimodality therapy is relatively ineffective.
  • The bithalamic variant behaves similarly to a pontine glioma.
  • [MeSH-major] Astrocytoma / surgery. Brain Neoplasms / surgery. Hypothalamic Neoplasms / surgery. Neurofibromatosis 1 / surgery. Thalamic Diseases / surgery
  • [MeSH-minor] Child. Child, Preschool. Humans. Hypothalamus / pathology. Infant. Magnetic Resonance Imaging. Neoadjuvant Therapy. Optic Nerve Glioma / diagnosis. Optic Nerve Glioma / surgery. Thalamus / pathology

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  • [Copyright] Copyright 2000 S. Karger AG, Basel
  • (PMID = 10867564.001).
  • [ISSN] 1016-2291
  • [Journal-full-title] Pediatric neurosurgery
  • [ISO-abbreviation] Pediatr Neurosurg
  • [Language] eng
  • [Publication-type] Journal Article; Research Support, Non-U.S. Gov't; Review
  • [Publication-country] Switzerland
  • [Number-of-references] 21
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30. Montiel Herrera JM, Góngora-Melendez MA, Pineda-Maldonado ML, Medina-Sanson A, Del Rio Chivardi JM, Del Río-Navarro BE, Rosas-Vargas MA: Carboplatin hypersensitivity and desensitization in an infant. Ther Drug Monit; 2010 Aug;32(4):525-8
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  • A 1-year-old Mexican girl weighing 10 kg who had optic nerve glioma was initially scheduled to receive 12 cycles of 600 mg/m2 carboplatin (CBP) as two 300-mg/m2 intravenous infusions administered over 1 hour on 2 different days and a 1-hour intravenous infusion of 1.5 mg/m2 vincristine every 4 weeks.
  • The patient had no history of drug allergies or any type of adverse drug reaction, but she developed itchiness, maculopapular rash, sweating, respiratory distress, and anxiety during the seventh cycle of CBP.
  • According to the algorithm developed by Naranjo et al, the adverse drug reaction was classified as definite secondary to CBP and confirmed by positive skin tests indicating hypersensitivity to the drug.
  • After evaluating the clinical course of the adverse drug reaction and considering the need to continue cancer treatment, a decision was made to desensitize the patient to CBP.
  • The desensitization procedure took 8 hours and was performed during each new chemotherapy cycle until the 12 cycles of chemotherapy were successfully completed.
  • [MeSH-major] Antineoplastic Agents / adverse effects. Carboplatin / adverse effects. Desensitization, Immunologic / methods. Drug Hypersensitivity / therapy
  • [MeSH-minor] Anaphylaxis / blood. Antineoplastic Agents, Phytogenic / therapeutic use. Drug Monitoring. Drug Therapy, Combination. Exanthema / chemically induced. Female. Humans. Immunoglobulin E / immunology. Infant. Optic Nerve Glioma / complications. Optic Nerve Glioma / drug therapy. Optic Nerve Neoplasms / complications. Optic Nerve Neoplasms / drug therapy. Referral and Consultation. Skin Tests. Vincristine / therapeutic use

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  • (PMID = 20463633.001).
  • [ISSN] 1536-3694
  • [Journal-full-title] Therapeutic drug monitoring
  • [ISO-abbreviation] Ther Drug Monit
  • [Language] eng
  • [Publication-type] Case Reports; Clinical Conference; Journal Article
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / Antineoplastic Agents; 0 / Antineoplastic Agents, Phytogenic; 37341-29-0 / Immunoglobulin E; 5J49Q6B70F / Vincristine; BG3F62OND5 / Carboplatin
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31. Kortmann RD, Timmermann B, Taylor RE, Scarzello G, Plasswilm L, Paulsen F, Jeremic B, Gnekow AK, Dieckmann K, Kay S, Bamberg M: Current and future strategies in radiotherapy of childhood low-grade glioma of the brain. Part II: Treatment-related late toxicity. Strahlenther Onkol; 2003 Sep;179(9):585-97
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Current and future strategies in radiotherapy of childhood low-grade glioma of the brain. Part II: Treatment-related late toxicity.
  • BACKGROUND: For more than 60 years, radiation therapy has been an integral part in the management of childhood low-grade glioma.
  • As this tumor carries an excellent long-term prognosis, the risk of late effects is of particular clinical importance and impinges upon radiotherapeutic treatment strategies.
  • MATERIAL AND METHODS: Studies on the use of radiation therapy in children with low-grade glioma were systematically reviewed for data on radiotherapy-induced side effects on brain parenchyma, endocrine dysfunction, growth retardation, neurocognitive dysfunction, vasculopathy, and secondary neoplasms.
  • Past reports included only retrospective series from the 1930s to present days, a time during which treatment policies and radiation techniques widely varied and considerably changed in recent years.
  • Often, considerable uncertainty existed regarding pretreatment health status and radiotherapy-related factors (e.g., total dose, dose per fraction, treatment fields).
  • The risk of radiation-induced disturbances in visual function is low (no case reported).
  • Modern treatment techniques appear to reduce the risk of radiation-induced late effects.
  • CONCLUSIONS: More studies and clear definitions of clinical endpoints such as neurocognitive and endocrinological outcome are needed in order to clarify the impact of radiation therapy on the risk of late sequelae.
  • These information and the contribution of tumor, surgery and chemotherapy will help to define the role of radiation therapy in the future management of childhood low-grade glioma and whether the use of highly sophisticated and expensive treatment techniques is justifiable.
  • The recently initiated prospective study of the APRO (Pediatric Radiooncology Working Party) on documentation of dose prescription to organs at risk and the network of the GPOH to explore late effects as well as the forthcoming prospective SIOP/GPOH (International Society of Pediatric Oncology/German Society of Pediatric Oncology and Hematology) LGG 2003 trial are addressing these issues.
  • [MeSH-major] Brain Neoplasms / radiotherapy. Glioma / radiotherapy. Radiotherapy / adverse effects
  • [MeSH-minor] Adolescent. Adult. Age Factors. Child. Child, Preschool. Developmental Disabilities / etiology. Growth Disorders / etiology. Humans. Infant. Moyamoya Disease / etiology. Neurofibromatoses / complications. Optic Nerve Glioma / radiotherapy. Prognosis. Radiotherapy Dosage. Retrospective Studies. Risk Factors. Stroke / etiology. Time Factors. Treatment Outcome

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  • (PMID = 14628124.001).
  • [ISSN] 0179-7158
  • [Journal-full-title] Strahlentherapie und Onkologie : Organ der Deutschen Röntgengesellschaft ... [et al]
  • [ISO-abbreviation] Strahlenther Onkol
  • [Language] eng
  • [Publication-type] Comparative Study; Journal Article; Review
  • [Publication-country] Germany
  • [Number-of-references] 74
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