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We report a case of unilateral congenital aplasia of the carpal scaphoid bone discovered in adulthood, which probably caused a severe painful intercarpal degenerative arthritis. This case was not associated with hypoplasia or aplasia of thenar and forearm muscles, abnormality of the skeleton of the thumb ray, absence of the sesamoid bones, hypoplasia of the forearm bones, as in cases reported in the literature; the only additional anatomic abnormality was a mild hypoplasia of the radial styloid. A surgical treatment (4-bone arthrodesis) has been performed to treat the advanced carpal collapse and the arthritis.