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

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

ABBREVIATIONS

BMIz = BMI z-score; CD = cyst decompression; DI = diabetes insipidus; GTR = gross-total resection; HyOb = hypothalamic obesity; IMRT = intensity-modulated radiation therapy; OS = overall survival; PBT = proton beam therapy; PFS = progression-free survival; RT = radiation therapy; STR = subtotal resection.

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Contributor Notes

Correspondence Guillermo Aldave: Texas Children’s Hospital, Houston, TX. gxaldave@texaschildrens.org

INCLUDE WHEN CITING Published online May 28, 2021; DOI: 10.3171/2020.11.PEDS20803.

Disclosures The authors report no conflict of interest concerning the materials or methods used in this study or the findings specified in this paper.

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