Stiff limb syndrome: end of spectrum or a separate entity?

BACKGROUND: Stiff-person syndrome is a rare disorder characterized by rigidity of axial or limb muscles with episodes of co-contraction of agonist and antagonist muscles during the spasms. In some patients axial or limb involvement may predominate and may have unusual manifestations.

DESIGN: Case report.

SETTING: Tertiary care teaching hospital.

PATIENT: A 42-year-old farmer presented with seasonal occurrence of hiccup and vomiting during summer months for the last 3 years. He had painful lower limb spasms lasting for 2-3 minutes every 10-15 minutes for the past 20 days. His neurological examination was normal, erythrocyte sedimentation rate (ESR) was 50 mm at 1st hour, and cerebrospinal fluid protein 78 mg/dL without pleocytosis. Radiograph of chest, abdominal ultrasound, and craniospinal magnetic resonance imaging were normal. The patient improved on diazepam.

CONCLUSION: Our patient is a forme fruste of stiff person syndrome with hiccups and vomiting due to diaphragmatic spasm.