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Background: Monomelic Amyotrophy is a rare self-limiting motor neuron disease affecting anterior horn cells of spinal cord. It usually has a juvenile onset, male predominance, single upper limb involvement (although bilateral asymmetric or even symmetric involvement has been reported) and is usually described in India and Far-East Asia with few reports from North America.
Case Report: A North-American Hispanic female with monomelic amyotrophy simultaneously involving the right upper and the left lower extremities with unusual sequence of muscle involvement in the lower limb, namely involving only the anterior and medial compartments of the thigh.
Conclusions: Although monomelic amyotrophy has several well known phenotypes or patterns of muscular involvement in certain races or geographical distribution, unusual or unreported presentations and topography of muscular involvement may still be possible and the disease should be considered regardless the race and geographical area.

Keywords: Neuromuscular Manifestations, Neuromuscular Monitoring