Moyamoya following cranial irradiation for primary brain tumors in children

Ullrich, Nicole J.; Robertson, Richard L.; Kinnamon, Daniel D.; Scott, R. Michael; Kieran, Mark W.; Turner, Christopher D.; N., S.; Goumnerova, Liliana; Proctor, Mary C.; Tarbell, Nancy J.; Marcus, Karen J.; Pomeroy, Scott L.

doi:10.1212/01.wnl.0000257095.33125.48

Cited by 298 publications

(203 citation statements)

References 48 publications

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“…In a study of pediatric patients with primary brain tumors treated at the Dana‐Farber Cancer Institute between 1990 and 2000, the crude incidence of post‐RT moyamoya syndrome was found to be 9/31 (29%) for optic glioma, 1/23 (4.3%) for craniopharyngioma, and 1/65 (1.5%) for medulloblastoma 11. Those data are not significantly different from those of this study.…”

Section: Discussioncontrasting

confidence: 76%

“…Moreover, optic glioma is associated with NF1. Patients with NF1 who received higher doses of radiation to the circle of Willis have increased risk of moyamoya syndrome development 11.…”

Section: Discussionmentioning

confidence: 99%

“…Post‐RT moyamoya syndrome, although known to be radiation related, is infrequently described in the literature. Between 1971 and 2015, there have been a total of 84 reported cases of moyamoya syndrome following cranial RT, of which 76 occurred in children 4, 5, 6, 7, 8, 9, 10, 11. The long‐term clinical outcome of these patients with post‐RT moyamoya syndrome also remains unclear.…”

Section: Introductionmentioning

confidence: 99%

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Incidence and long‐term outcome of postradiotherapy moyamoya syndrome in pediatric patients with primary brain tumors: a single institute experience in Taiwan

Chang

Liang

et al. 2016

Cancer Medicine

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We aimed to investigate the incidence and long‐term outcome of moyamoya syndrome in pediatric patients with primary brain tumors after receiving cranial radiotherapy (RT) in a single institute in Taiwan. The complete medical records, medical images, and RT notes of 391 pediatric patients with primary brain tumors treated with cranial RT between January 1975 and December 2005 in Taipei Veterans General Hospital (TVGH), Taiwan, were entered into an electronic registry and reviewed. Eight (2%) cases of post‐RT moyamoya syndrome were identified in the sample of 391 patients. The median latency was 3 years post‐RT. Among the eight patients, three had craniopharyngioma, two had optic glioma, two had medulloblastoma, and one had a suprasellar astrocytoma. The prescribed physical doses of RT were in the range of 40–54 Gy. The incidence was highest in those with optic glioma (0.039/person‐year), followed by craniopharyngioma (0.013/person‐year), astrocytoma (0.003/person‐year), and medulloblastoma (0.002/person‐year). No patients died of vasculopathy. No difference in crude incidence was found between our results and those of other series. The incidence of moyamoya syndrome was diagnosis dependent, with the highest incidence among patients with optic glioma. No regional difference in incidence was found. Long‐term, stable neurological function may be achieved following timely surgical intervention.

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Section: Discussioncontrasting

confidence: 76%

“…Moreover, optic glioma is associated with NF1. Patients with NF1 who received higher doses of radiation to the circle of Willis have increased risk of moyamoya syndrome development 11.…”

Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

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Incidence and long‐term outcome of postradiotherapy moyamoya syndrome in pediatric patients with primary brain tumors: a single institute experience in Taiwan

Chang

Liang

et al. 2016

Cancer Medicine

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“…While clearly effective, radiotherapy is, however, associated with a number of late effects, including neurocognitive impairment, second-tumor formation, and increasing reports of the occurrence of moyamoya syndrome. 2,5,10,11,36,45 One of the patients in this series required revascularization surgery for this unusual but potentially devastating complication. Radiotherapy has been also associated with increased rates of endocrine deficit (especially growth hormone deficiency) compared with other treatment modalities.…”

Section: Radiotherapymentioning

confidence: 96%

The role of surgery in optic pathway/hypothalamic gliomas in children

Goodden¹,

Pizer

Pettorini³

et al. 2014

PED

105

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Object Optic pathway/hypothalamic gliomas (OPHGs) are generally benign tumors situated in an exquisitely sensitive brain region. The location and natural history of OPHGs has led to much debate about optimal treatment. This paper revisits the role of and optimal timing of debulking surgery in OPHG. Methods This paper presents a series of cases managed by the neuro-oncology team at Alder Hey Children's Hospital and a single surgeon. Data were collected retrospectively for periods prior to 2009 and prospectively thereafter. Tailored treatment strategies were used, including observation and combinations of surgery, chemotherapy, and radiotherapy. Tumor control rates and outcomes are reviewed. Results Forty-two patients were treated between 1998 and 2011. Their median age at diagnosis was 5 years 7 months. Nineteen patients were positive for neurofibromatosis Type 1 (NF1) and 23 patients were negative for NF1. The median duration of follow-up was 77 months (range 21.8–142.3 months). Presenting symptoms included visual impairment (in 50% of cases), headache (in 24%), and hypothalamic/pituitary dysfunction (in 29%). Twenty-two debulking procedures were performed in 21 patients. Four biopsies (3 open, 1 endoscopic) were also performed. The histological diagnosis was pilocytic astrocytoma in 21 patients and pilomyxoid astrocytoma in 2 patients. Ten patients (Group 1) had primary surgical debulking alone and were then observed. Four patients (Group 2) had surgical debulking, plus planned chemotherapy within 3 months. Seven patients (Group 3) required surgical debulking for progressive disease following a variety of treatments. Patient age had the greatest impact on subsequent tumor progression. In total, 13 patients received chemotherapy, 4 on initial presentation, 4 in combination with surgery, and 5 for further tumor progression. Five patients were treated with radiotherapy, 3 prior to referral to Alder Hey. Eleven patients required shunt insertion for hydrocephalus. Vision was stabilized for 74% of patients. The number of patients with hypothalamic/pituitary dysfunction increased from 12 at presentation to 16 by the end of treatment. The overall survival rate was 93%. Three patients died—1 from tumor progression, 1 from infective complications from tumor biopsy, and 1 from a spontaneous posterior fossa hemorrhage. NF1 was associated with improved outcome—fewer patients required active intervention and rates of visual impairment and/or or hypothalamic/pituitary dysfunction were lower. Conclusions Good long-term survival and functional outcomes can be achieved in children with OPHG. Tumor control was achieved through an individualized approach using surgery, chemotherapy, or radiotherapy in varied combinations. The authors aim to limit radiotherapy to cases involving older children in whom other therapies have failed, due to the well-described and often devastating late effects associated with midline cranial irradiation. Surgery has a clear role for diagnosis, tumor control, and relief of mass effect. In particular, primary surgical debulking of tumor (without adjuvant therapy) is safe and effective. Recent advances in intraoperative MRI may add value and need further assessment.

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“…2). This is potentially a serious complication of whole brain radiotherapy in children with brain tumours (19). However, this risk is probably insignificant when compared with the morbidity and mortality associated with the tumour itself, radical resection or repeat surgery.…”

Section: Discussionmentioning

confidence: 99%

‘Do no harm’: management of craniopharyngioma

Gleeson¹,

Amin²,

Maghnie

2008

European Journal of Endocrinology

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Craniopharyngioma management is challenging. Although histology is benign, the tumour can be clinically aggressive with local invasion and frequent recurrences. Extensive morbidity may be present at diagnosis and furthermore, occurs as a consequence of neurosurgery and radiotherapy. Hypothalamic symptoms can have a devastating effect on quality of life and may reduce life expectancy. This case highlights both the challenge of managing hyperphagia and morbid obesity and the importance of initial treatment preserving existing hypothalamic function and the need to avoid tumour recurrence and further surgery.A 11-year old boy presented with hydrocephalus secondary to a craniopharyngioma (he had visual failure and hypopituitarism but few hypothalamic symptoms). He underwent radical resection followed by radiotherapy. Following this treatment, he developed psychological and behavioural problems and hyperphagia. Weight gain in the first year (an increase from C1.4 to C3.7 S.D.) resulted in poor mobility and a fall which caused a slipped femoral epiphysis. In the next year, there was a 6-month period of unexpected weight loss (C4.2 to C3.8 S.D.) that culminated in emergency treatment for diabetic ketoacidosis secondary to severe insulin resistance. He developed a left hemiplegia, and a subsequent cerebral angiogram identified multiple stenoses of the Circle of Willis with a Moyamoya appearance secondary to radiotherapy. Weight gain has continued (C3.8 to C5.5 S.D.) so that bariatric surgery is a management option.European Journal of Endocrinology 159 S95-S99

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Moyamoya following cranial irradiation for primary brain tumors in children

Cited by 298 publications

References 48 publications

Incidence and long‐term outcome of postradiotherapy moyamoya syndrome in pediatric patients with primary brain tumors: a single institute experience in Taiwan

Incidence and long‐term outcome of postradiotherapy moyamoya syndrome in pediatric patients with primary brain tumors: a single institute experience in Taiwan

The role of surgery in optic pathway/hypothalamic gliomas in children

‘Do no harm’: management of craniopharyngioma

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