Neuromuscular hamartoma is a possible primary pathology of oculomotor ophthalmoplegic migraine
Department of Neurosurgery, Tokyo Medical University, Japan. Cephalalgia
(Impact Factor: 4.89).
12/2011; 32(2):171-4. DOI: 10.1177/0333102411431331
BACKGROUND: Oculomotor ophthalmoplegic migraine (O-OPM) occurs in many children, and in some cases MRI shows a small mass in the root exit zone (REZ) of the oculomotor nerve. This mass is considered to result from nerve hypertrophy caused by repeated demyelination. CASE RESULTS: A 51-year-old man has been on oral medication for O-OPM, which he had from 6 years of age. However, the frequency and intensity of his migraine attacks have gradually increased. Brain magnetic resonance imaging (MRI) revealed a small nodular mass in the REZ of the oculomotor nerve. The mass was initially diagnosed as oculomotor schwannoma and tumor resection was attempted. However, as the mass was tightly adhered to the oculomotor nerve and hemorrhagic, biopsy was performed. The pathological diagnosis was neuromuscular hamartoma. CONCLUSION: The small nodular mass in the REZ of the oculomotor nerve may be a hamartoma associated with congenital factors and may possibly be the primary pathology of O-OPM in this case.
Available from: Vijay Shankar
- "Clinically, patients are usually asymptomatic, except for the presence of a palpable mass, which may be solitary or multinodular. However, depending on the site of involvement, pain, neurologic dysfunction in the form of muscle weakness, parasthesia and headache including oculomotor ophthalmoplegic migraine, has been reported.6,8,9 Computed tomography scans provide a good guide to suggest the diagnosis and serve to define the entire scope of the lesion, especially in the central type of tumors.6 "
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ABSTRACT: Benign triton tumors (BTT) are very rare lesions composed of mature skeletal muscle and neural tissue. We report a case of a 14-year-old boy who presented with asymptomatic swelling of the chin over an 18-month duration which increased gradually to involve the left side of the lower lip. Clinically, a diagnosis of neurofibroma was made. Excisional biopsy confirmed the diagnosis of a BTT. Having an affinity for large nerve trunks like the brachial and sciatic, these tumors rarely occur in the head and neck region. When they do, they may involve the large central cranial nerve trunk and present as intracranial masses or involve the smaller peripheral nerve twigs and present as asymptomatic skin nodules, of which only four cases involving peripheral nerves are reported in the English literature. Here, we report the fifth documented case of a BTT involving the mental branch of the trigeminal nerve. A brief review of the literature is also provided.
Available from: Monica Margoni
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