A cachectic 55-year-old man presented with ataxia and metabolic instability. On physical examination, he had prominent myoedema in all muscle groups. In addition, a CT scan and lumbar puncture showed extensive nonobstructive hydrocephalus. Further clinical evaluation revealed elevated creatine phosphokinase and liver enzyme levels, although the patient was euthyroid. The patient improved neurologically and metabolically with supportive therapy but the myoedema persisted. Previous cases have emphasized that myoedema is a localized, electrically silent, benign myopathic disorder of unknown cause. As with a previous case with ventricular enlargement, myoedema may be part of systemic pathology. Finally, as in most other reports, myoedema is a rare condition; only 3 cases (of 44) with palpable (but not visible) myoedema were uncovered in this study.