Pegylated interferon-α-2b for children with recurrent craniopharyngioma

Clinical article

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  • 1 Department of Neurosurgery, University of Pittsburgh School of Medicine; and
  • 2 Departments of Neurosurgery,
  • 3 Radiology, and
  • 4 Pediatrics, Children's Hospital of Pittsburgh, University of Pittsburgh School of Medicine, Pittsburgh, Pennsylvania
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Object

Previous studies of systemic and intralesional administration of nonpegylated interferon have shown efficacy against craniopharyngioma. Pegylaion of interferon-α-2b (PI) prolongs the half-life, allowing sustained exposure of the drug over time, and enhances efficacy. The authors report the results of the use of PI in 5 children with recurrent craniopharyngiomas.

Methods

Five children, ranging in age from 9 to 15 years, with recurrent craniopharyngiomas were treated for up to 2 years with subcutaneous injections of PI at a dose of 1–3 μg/kg/week. Tumor response was assessed using MRI.

Results

All patients had stable disease or better in response to PI. One patient experienced a recurrence after gross-total resection (GTR). She initially showed an increase in the predominantly cystic tumor after 3 months of treatment, followed by a complete response. She required no further intervention and remains without evidence of disease 10 years after starting treatment. Another patient experienced recurrence 3.3 years after subtotal resection (STR) and radiation therapy. He had complete disappearance of the predominantly cystic component after 4 months of treatment, and a small residual calcified mass remains 5 years later. The third patient experienced recurrence after 3 GTRs. He had a complete response after 7 months of treatment and remains without evidence of disease 19 months after starting treatment. The fourth patient experienced recurrence after 2 STRs. He had a 30% decrease in tumor size after 4 months of treatment, which was maintained for 12 months at which point the cyst began to increase in size. The final patient experienced recurrence after GTR and has stable disease 6 months after starting treatment with PI.

Conclusions

The use of PI in children with recurrent craniopharyngiomas can result in significant and durable responses and potentially delay or avoid the need for radiation therapy.

Abbreviations used in this paper:GTR = gross-total resection; PI = pegylated interferon-α-2b; STR = subtotal resection.

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Contributor Notes

Address correspondence to: Regina Jakacki, M.D., Department of Pediatrics, Children's Hospital of Pittsburgh, 4401 Penn Avenue, Pittsburgh, Pennsylvania 15224. email: regina.jakacki@chp.edu.

Please include this information when citing this paper: published online October 12, 2012; DOI: 10.3171/2012.9.PEDS12225.

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