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Título

Case report: Belly-dancer syndrome in a patient with Neuromyelitis Optica Spectrum Disorder

RESUMO

Case report: Female, 46 years old, diagnosed with Neuromyelitis Optica Spectrum Disorder (AQP4 positive) in 2010, with episodes of inflammation characterized by optic neuritis, left hemiparesis and hemiparesthesia. She was treated with methotrexate, azathioprine and, after a new attack, rituximab was initiated until now. The last episode was in July 2020, characterized by parestesia below T8 vertebral level. In January 2021, she reported abdominal involuntary movements, radiating to the dorsal region at the thoracolumbar transition. It was interpreted as bilateral internal and external oblique muscle contraction. On electroneuromyography, spontaneous discharges of action potentials of arrhythmic activity and variable frequency was found, suggestive of myokymia at rest in the rectus abdominis muscles and more markedly in the left oblique. Considering the diagnostic hypothesis of Belly-dancer Syndrome, levetiracetam 250mg 12/12h was started, with no satisfactory response either when associated with carbamazepine 200mg 12/12h and gabapentin 300mg 8/8h. Opted for the application of botulinum toxin in the left rectus abdominis and left external oblique with partial improvement of movements. Discussion: Belly-Dancer Syndrome or diaphragmatic flutter is a rare condition that involves repetitive and involuntary movements of the abdominal wall muscles. The probable etiologies are drug (levodopa, biperiden, quetiapine), postoperative abdominal surgery, encephalitis, stroke, osmotic demyelination, metabolic changes and trauma. Such changes lead to abnormal excitation of the phrenic nerve, via the central nervous system or along the nerve. Another proposed mechanism is irritation of the diaphragm muscle itself. Diagnosis can be made with ultrasound, thoracic videofluoroscopy, or electromyography. Supposed treatment includes phenytoin, carbamazepine, clonazepam, fluoxetine, gabapentin and valproic acid. The use of botulinum toxin is one of the most recent therapies. In general, treatment attempts have been largely ineffective. Final comments: The present case of Belly-Dancer Syndrome in a patient with Neuromyelitis Optica Spectrum Disorder, may be associated with the last episode inflammation of the disease. The syndrome is little known and that more descriptions of cases and treatments are needed to provide a better evaluation of treatment.

Palavras Chave

Belly-Dancer syndrome; diaphragmatic flutter; Neuromyelitis Optica Spectrum Disorder