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Jacky T. Yeung, Ian F. Pollack, Ashok Panigrahy and Regina I. Jakacki

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.

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Mark D. Krieger, Ashok Panigrahy, J. Gordon McComb, Marvin D. Nelson, Xiaodong Liu, Ignacio Gonzalez-Gomez, Floyd Gilles and Stefan Bluml

Object

The management of pediatric intraventricular tumors is highly dependent on identification of the tumor type. Choroid plexus papillomas, a common intraventricular tumor in children, can be difficult to distinguish radiographically from choroid plexus carcinomas and other common pediatric central nervous system (CNS) tumors. In this study to overcome the limitations of current noninvasive imaging modalities, the authors use novel magnetic resonance (MR) spectroscopy techniques in vivo to elucidate the identifying biochemical features of choroid plexus tumors that may facilitate diagnosis and treatment.

Methods

Based on an Internal Review Board–approved protocol, six children with newly diagnosed, untreated intraventricular brain tumors were identified. On retrospective review, this series included three choroid plexus papillomas and three choroid plexus carcinomas. Single-voxel proton MR spectroscopy with a short echo time was performed, and absolute metabolite concentrations (in mmol/kg) were determined using fully automated quantitation. These results were compared with MR spectroscopy profiles obtained in 54 other untreated CNS neoplasms in children.

The myo-inositol (mI) level was significantly higher in choroid plexus papillomas (> 10 mmol/kg), uniquely distinguishing these tumors from choroid plexus carcinomas and all other tumors. Choroid plexus carcinomas, on the other hand, had significantly elevated levels of choline when compared with choroid plexus papillomas.

Conclusions

In this study the authors find that mI is a biochemical constituent that uniquely identifies choroid plexus papillomas and can be used as a noninvasive means of diagnosis and for follow-up evaluations in patients with this disease.

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Gurpreet S. Gandhoke, Jason S. Hauptman, David J. Salvetti, Gregory M. Weiner, Ashok Panigrahy, Sabri Yilmaz and Ian F. Pollack

The authors report a unique case of a transosseous CSF fistula that was detected more than 10 years after treatment of a symptomatic Chiari I malformation. This lesion initially presented as an intraosseous cystic lesion involving the C-2 vertebra, which was found to communicate freely with the subarachnoid space through a tiny dural opening. Surgical management involved hemilaminectomy and repair of the dural defect followed by reinforcement of the bony defect with demineralized bone matrix. Following closure of the fistula, symptoms of elevated intracranial pressure developed, necessitating a ventriculoperitoneal shunt for CSF diversion.