Abstract
Introduction
The treatment of pediatric intracranial low-grade gliomas (LGG) generally begins with maximal safe resection. Radiation therapy (RT) and chemotherapy are typically reserved for patients with incomplete resection and/or disease progression. We report long-term treatment outcomes and toxicities in a cohort of pediatric patients with LGG after RT.
Methods
Thirty-four patients <21 years old with intracranial LGG who were treated with RT at the Johns Hopkins Hospital were included in this retrospective analysis. Patients were evaluated for overall survival (OS), progression-free survival (PFS), recurrence patterns, and treatment toxicities using descriptive statistics, Kaplan-Meier curves, and Cox proportional hazard regressions.
Results
The mean age at diagnosis was 7.9 years (range 1.2–18.3 years) and mean age at RT was 9.8 years (range 3.0–28.9 years). The median follow-up time was 9.8 years after radiation (range 0.8–33.3 years). The estimated 10-year OS and PFS after RT were 92 and 74 %, respectively. Twelve patients had disease progression after RT, and all recurrences were local. Two patients died due to disease progression 2.3 and 9.1 years after RT. One patient had malignant transformation of LGG to high-grade glioma. No significant predictors of PFS were identified on uni- or multivariate analysis. Late effects of LGG and treatment seen were endocrine deficiencies in 16 patients, visual problems in 10 patients, hearing loss in 4 patients, special education requirements for 5 patients, and a vascular injury/demyelination secondary to RT in 1 patient.
Conclusion
Our study suggests that the use of radiation in patients with intracranial LGG results in excellent OS and PFS with acceptable toxicity at long-term follow-up.
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References
Pollack IF, Claassen D, Al-Shboul Q, et al. (1995) Low-grade gliomas of the cerebral hemispheres in children: an analysis of 71 cases. J Neurosurg 82:536–547
Kortmann RD, Timmermann B, Taylor RE, et al. (2003) Current and future strategies in radiotherapy of childhood low-grade glioma of the brain. Part I: treatment modalities of radiation therapy. Strahlenther Onkol 179:509–520
Kortmann RD, Timmermann B, Taylor RE, et al. (2003) Current and future strategies in radiotherapy of childhood low-grade glioma of the brain. Part II: treatment-related late toxicity. Strahlenther Onkol 179:585–597
Gnekow AK, Falkenstein F, von Horstein S, et al. (2012) Long-term follow-up of the multicenter, multidisciplinary treatment study HIT-LGG-1996 for low-grade glioma in children and adolescents of the German Speaking Society of Pediatric Oncology and Hematology. Neuro-Oncology 14:1265–1284
Muller K, Gnekow A, Falkenstein F, et al. (2013) Radiotherapy in pediatric pilocytic astrocytomas: a subgroup analysis within the prospective multicenter study HIT-LGG 1996 by the German Society of Pediatric Oncology and Hematology (GPOH). Strahlenther Onkol 189:647–655
Marcus KJ, Goumnerova L, Billett AL, et al. (2005) Stereotactic radiotherapy for localized low-grade gliomas in children: final results of a prospective trial. Int J Rad Onc Biol Phys 61:374–379
Chadderton RD, West CG, Schuller S, et al. (1995) Radiotherapy in the treatment of low-grade astrocytomas: II—the physical and cognitive sequelae. Childs Nerv Syst 11:443–448
Brauner R, Malandry F, Rappaport R, et al. (1990) Growth and endocrine disorders in optic glioma. Eur J Pediatr 149:825–828
Grill J, Couanet D, Cappelli C, et al. (1999) Radiation induced cerebral vasculopathy in children with neurofibromatosis and optic pathway glioma. Ann Neurol 45:393–396
Lacaze E, Kieffer V, Streri A, et al. (2003) Neuropsychological outcome in children with optic pathway tumours when first-line treatment is chemotherapy. Brit J Canc 289:2038–2044
Paulino AC, Mazloom A, Terashima K, et al. (2013) Intensity-modulated radiotherapy (IMRT) in pediatric low-grade glioma. Cancer 119:2654–2659
Aarsen FK, Paquier PF, Reddingius RE, et al. (2005) Functional outcome after low-grade astrocytoma treatment in childhood. Cancer 106:396–402
Danoff BF, Cowchock FS, Marquette C, et al. (1982) Assessment of the long-term effects of primary radiation therapy for brain tumors in children. Cancer 49:1580–1586
Benesch M, Lackner H, Sovinz P, et al. (2006) Late sequela after treatment of childhood low-grade gliomas: a retrospective analysis of 69 long-term survivors treated between 1983 and 2003. J Neuro-Oncol 78:199–205
Merchant TE, Kun LE, Wu S, et al. (2009) Phase II trial of conformal radiation therapy for pediatric low-grade glioma. J Clin Onc 27:3598–3604
Merchant TE, Conklin HM, Wu S, et al. (2009) Late effects of conformal radiation therapy for pediatric patients with low-grade glioma: prospective evaluation of cognitive, endocrine, and hearing deficits. J Clin Onc 27:3691–3697
Mishra KK, Puri DR, Missett BT, et al. (2006) The role of up-front radiation therapy for incompletely resected pediatric WHO grade II low-grade gliomas. Neuro-Oncol 8:166–174
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Huynh-Le, MP., Walker, A.J., Burger, P.C. et al. Management of pediatric intracranial low-grade gliomas: long-term follow-up after radiation therapy. Childs Nerv Syst 32, 1425–1430 (2016). https://doi.org/10.1007/s00381-016-3100-8
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DOI: https://doi.org/10.1007/s00381-016-3100-8