We report a rare case of a 21-year-old man with a bilateral absence of abductor pollicis brevis and opponens pollicis muscles, which has not been previously reported. He presented with weakness in both of his hands. On physical examination there was atrophy on the thenar eminences of both of his hands and there was an absence of gross functional impairment in his hand functions. On magnetic resonance imaging, abductor pollicis brevis and opponens pollicis muscles were bilaterally absent.
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