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Pigmented villo-nodular synovitis/tenosynovitis (PVNS) is a rare cause of combined ulnar and median nerve compression neuropathy at the wrist. In our case, a 53-year-old house-wife had sensorial and motor complaints at her left hand. In clinical examination, painless soft tissue mass was palpated at her wrist and both the tenar and hypotenar muscles were atrophic. Electromyography showed prolonged distal latencies for median and ulnar nerve. A space-occupying soft tissue lesion was revealed in magnetic resonance imaging. Carpal tunnel and Guyon canal were released and lesion was excised. PVNS was confirmed by histopathological examination. If compression neuropathy of medial and ulnar nerves together is caused by a space-occupying lesion, PVNS should be considered in etiology.