A 27-year-old woman with bruxism suffered a spider bite (Loxosceles rufescens) on the left cheek that caused severe local cellulitis, facial palsy, and painful hyperesthesia over the two lower trigeminal nerve divisions. Facial but not trigeminal neuropathy improved, and she developed a labial dystonia that only corrected while pressing the right medial incisor. A specially designed maxillary splint that continuously pressed it suppressed both dystonia and related spontaneous firing of motor unit potentials in electromyography. Overstimulation of the contralateral trigeminal territory possibly compensated for the altered left trigeminal nerve input, balanced proprioceptive influences at the central inhibitory-excitatory circuitry, and controlled dystonia.
Background/Aims: Cramp-fasciculation syndrome (CFS) is an acquired, chronic, usually benign and rather heterogeneous condition with isolated fasciculations and muscle cramps generally induced by physical exercise. They commonly involve calf and quadriceps muscles. The pathophysiology of CFS is related to peripheral nerve hyperexcitability, most often located at the motor nerve terminal or intramuscular arborization. Methods: A 21-year-old man presented with a progressive syndrome of bronchospasm, cramps and muscle twitches related to physical exercise. Spirography showed bronchial hyperresponsiveness, so he received inhaled corticosteroids and β2-agonists that improved respiratory symptoms. Electrodiagnostic studies were consistent with CFS. Gabapentin was then introduced. Results: Both respiratory and muscle symptoms improved. A new spirogram after all inhaled medication had been discontinued was normal. Conclusion: This picture suggests a concomitant involvement of the peripheral motor nerves of both skeletal and airway autonomic smooth muscle, a presentation not previously reported in CFS.
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