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Peptide Receptor Radionuclide Therapy in Patients With Neurofibromatosis Type 2: Initial Experience
Olivia Kertels1, Maria Breun2, Heribert Hänscheid3
1From the Institute of Diagnostic Radiology.
Purpose:
Neurofibromatosis type 2 (NF2) is a genetic disorder that is associated with multiple tumors of the nervous system, and approximately one half of patients present with meningiomas. For patients with multifocal disease, somatostatin receptor-targeted peptide receptor radionuclide therapy (PRRT) might be a suitable systemic treatment option.
Patients And Methods:
Between March 2015 and August 2017, 11 NF2 patients (7 females and 4 males; mean age, 39 ± 12 years) with multifocal, progressive meningiomas underwent a median of 4 cycles of PRRT (range, 2-6 cycles). Acute and chronic adverse events were recorded according to National Institutes of Health's Common Toxicity Criteria (CTC) version 5.0. Follow-up MRIs (every 3 to 6 months), using the Response Assessment in Neuro-Oncology response criteria for meningiomas, were used to assess treatment responses.
Results:
Peptide receptor radionuclide therapy was well tolerated in all patients without any relevant acute adverse effects. Transient hematologic toxicity (CTC grade 3) was observed in 2 subjects. Somatostatin receptor-directed radiopeptide therapy resulted in radiological disease stabilization in 6 of 11 patients. Median progression-free survival was 12 months (range, 1-55 months), and overall survival was 37 months (range, 5-61 months).
Conclusions:
Based on our retrospective pilot data, PRRT is feasible and well-tolerated in NF2 patients. It might offer a suitable treatment option in subjects with multiple, recurrent, or treatment-refractory meningiomas.
Insights
Peptide receptor radionuclide therapy (PRRT) is a feasible and well-tolerated treatment for Neurofibromatosis type 2 (NF2) patients with meningiomas. This therapy showed disease stabilization in most patients, offering a new option for difficult cases.
Area of Science:
- Neuro-oncology
- Medical physics
- Pharmacology
Background:
- Neurofibromatosis type 2 (NF2) is a genetic disorder frequently associated with nervous system tumors.
- Meningiomas occur in approximately 50% of NF2 patients, often presenting as multifocal disease.
- Systemic treatment options for multifocal meningiomas in NF2 are limited.
Purpose of the Study:
- To evaluate the feasibility and safety of somatostatin receptor-targeted peptide receptor radionuclide therapy (PRRT) in NF2 patients with multifocal meningiomas.
- To assess the treatment response and survival outcomes of PRRT in this patient population.
Main Methods:
- 11 NF2 patients with progressive meningiomas received a median of 4 cycles of PRRT.
- Adverse events were graded using the National Institutes of Health's Common Toxicity Criteria (CTC) v5.0.
- Treatment response was assessed using Response Assessment in Neuro-Oncology (RANO) criteria via MRI every 3-6 months.
Main Results:
- PRRT was well-tolerated with no significant acute adverse effects.
- Transient grade 3 hematologic toxicity occurred in 2 patients.
- Radiological disease stabilization was achieved in 6 of 11 patients.
- Median progression-free survival was 12 months, and median overall survival was 37 months.
Conclusions:
- PRRT is a feasible and well-tolerated treatment for NF2 patients.
- This therapy may be a suitable option for patients with multiple, recurrent, or treatment-refractory meningiomas.
- Further investigation in larger cohorts is warranted.
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