title: Dramatic radiographic response resulting in cerebrospinal fluid rhinorrhea associated with sunitinib therapy in recurrent atypical meningioma: case report.
abstract: Sunitinib is a multiple tyrosine kinase inhibitor with antiangiogenic, cytostatic, and antimigratory activity for meningiomas. A recent clinical trial of sunitinib for treatment of recurrent Grade II and III meningiomas suggested potential efficacy in this population, but only 2 patients exhibited significant radiographic response with tumor volume reduction. The authors illustrate another such case and discuss a complication related to this dramatic tumor volume reduction in aggressive skull base meningiomas. The authors describe the case of a 39-year-old woman who had undergone repeat surgical interventions and courses of radiotherapy over the previous 11 years for recurrent cranial and spinal meningiomas. Despite 4 operations over the course of 4 years on her right petroclival meningioma with cavernous sinus and jugular fossa extensions, she had progressive neurological deficits and tumor recurrences. The specimen histology progressed from WHO Grade I initially to Grade II at the time of the third recurrence. The lesion was then irradiated 3 times using stereotactic radiosurgery for further recurrences. More recently, the tumor size increased rapidly on imaging, in association with progressive neurological symptoms arising from brainstem compression and vasogenic edema. Institution of sunitinib therapy yielded a dramatic radiographic response, with marked reduction in the tumor volume and reduction of brainstem vasogenic edema within a few weeks of initiation of treatment. The significant radiographic response of tumor in the clival region was also associated with CSF rhinorrhea from a dural breach created by resolution of the invasive skull base meningioma, which necessitated withholding the sunitinib medication. To address the leak, the authors undertook surgical exploration and transsphenoidal packing using an autologous fat graft and a vascularized pedicled nasoseptal flap. The patient has done well during follow-up of 3 months after packing, with no evidence of recurrent CSF leak, and the medication was subsequently restarted. Prior clinical data and the dramatic radiographic response in this patient suggest that sunitinib holds promising therapeutic potential in carefully selected patients with recurrent atypical meningiomas where conventional strategies have been exhausted. There is a potential risk of associated CSF rhinorrhea, especially in more invasive skull base lesions showing dramatic radiographic response.
authors.affiliation: Departments of 1 Neurosurgery and.
authors.forename: Amol
authors.initials: A
authors.lastname: Raheja
authors.affiliation: Departments of 1 Neurosurgery and.
authors.forename: Howard
authors.initials: H
authors.lastname: Colman
authors.affiliation: Pathology, University of Utah, Salt Lake City, Utah.
authors.forename: Cheryl A
authors.initials: CA
authors.lastname: Palmer
authors.affiliation: Departments of 1 Neurosurgery and.
authors.forename: William T
authors.initials: WT
authors.lastname: Couldwell
mesh_terms: D000328:Adult; D002559:Cerebrospinal Fluid Rhinorrhea; D005260:Female; D006801:Humans; D008577:Meningeal Neoplasms / Q000601:surgery*; D008579:Meningioma / Q000601:surgery*; D009364:Neoplasm Recurrence, Local; D016634:Radiosurgery; D012189:Retrospective Studies; D000077210:Sunitinib
chemical_list: D000077210:Sunitinib
keywords: OS = overall survival; PDGFR = platelet-derived growth factor receptor; PFS = progression-free survival; PI-3 K/AKT = phosphatidyl inositol-3 kinase; Raf-MAPK = mitogen-associated protein kinase; SRS = stereotactic radiosurgery; VEGF = vascular endothelial growth factor; VEGFR = VEGF receptor; cerebrospinal fluid rhinorrhea; oncology; radiographic response; recurrent atypical meningioma; sunitinib