Comparison of multimodal surgical and radiation treatment methods for pediatric craniopharyngioma: long-term analysis of progression-free survival and morbidity

Vijay M Ravindra; M Fatih Okcu; Lucia Ruggieri; Thomas S Frank; Arnold C Paulino; Susan L McGovern; Vincent E Horne; Robert C Dauser; William E Whitehead; Guillermo Aldave

doi:10.3171/2020.11.PEDS20803

Comparison of multimodal surgical and radiation treatment methods for pediatric craniopharyngioma: long-term analysis of progression-free survival and morbidity

J Neurosurg Pediatr. 2021 May 28:1-8. doi: 10.3171/2020.11.PEDS20803. Online ahead of print.

Authors

Affiliations

¹ 1Division of Pediatric Neurosurgery, Texas Children's Hospital, Department of Neurosurgery, Baylor College of Medicine, Houston.
² 6Department of Neurosurgery, Naval Medical Center San Diego; and.
³ 7Division of Pediatric Neurosurgery, University of California, San Diego, California.
⁴ 2Texas Children's Cancer Center, Department of Pediatrics, Texas Children's Hospital, Baylor College of Medicine, Houston.
⁵ 3Department of Neurosurgery, University of Texas Medical Branch, Galveston.
⁶ 4Department of Radiation Oncology, Texas Children's Hospital, Houston.
⁷ 5Section of Pediatric Diabetes and Endocrinology, Department of Pediatrics, Texas Children's Hospital, Baylor College of Medicine, Houston, Texas.

PMID: 34049281
DOI: 10.3171/2020.11.PEDS20803

Abstract

Objective: The authors compared survival and multiple comorbidities in children diagnosed with craniopharyngioma who underwent gross-total resection (GTR) versus subtotal resection (STR) with radiation therapy (RT), either intensity-modulated radiation therapy (IMRT) or proton beam therapy (PBT). The authors hypothesized that there are differences between multimodal treatment methods with respect to morbidity and progression-free survival (PFS).

Methods: The medical records of children diagnosed with craniopharyngioma and treated surgically between February 1997 and December 2018 at Texas Children's Hospital were reviewed. Surgical treatment was stratified as GTR or STR + RT. RT was further stratified as PBT or IMRT; PBT was stratified as STR + PBT versus cyst decompression (CD) + PBT. The authors used Kaplan-Meier analysis to compare PFS and overall survival, and chi-square analysis to compare rates for hypopituitarism, vision loss, and hypothalamic obesity (HyOb).

Results: Sixty-three children were included in the analysis; 49% were female. The mean age was 8.16 years (95% CI 7.08-9.27). Twelve of 14 children in the IMRT cohort underwent CD. The 5-year PFS rates were as follows: 73% for GTR (n = 31), 54% for IMRT (n = 14), 100% for STR + PBT (n = 7), and 77% for CD + PBT (n = 11; p = 0.202). The overall survival rates were similar in all groups. Rates of hypopituitarism (96% GTR vs 75% IMRT vs 100% STR + PBT, 50% CD + PBT; p = 0.023) and diabetes insipidus (DI) (90% GTR vs 61% IMRT vs 85% STR + PBT, 20% CD + PBT; p = 0.004) were significantly higher in the GTR group. There was no significant difference in the HyOb or vision loss at the end of study follow-up among the different groups. Within the PBT group, 2 patients presented a progressive vasculopathy with subsequent strokes. One patient experienced a PBT-induced tumor.

Conclusions: GTR and CD + PBT presented similar rates of 5-year PFS. Hypopituitarism and DI rates were higher with GTR, but the rate of HyOb was similar among different treatment modalities. PBT may reduce the burden of hypopituitarism and DI, although radiation carries a risk of potential serious complications, including progressive vasculopathy and secondary malignancy. Further prospective study comparing neurocognitive outcomes is necessary.

Keywords: craniopharyngioma; oncology; radiation; surgery; treatment.