Carpal tunnel syndrome (CTS) is often found in adults and may be associated with rare anatomical abnormalities in the hand, such as the presence of a palmaris profundus tendon. Although the muscle was originally thought to be a variant of the palmaris longus muscle, some case studies have described both structures during operative treatment with open carpal tunnel release. We present a case of a 51-year-old woman who underwent open carpal tunnel release to treat CTS of the right hand. Preoperatively, results of electrodiagnostic tests and clinical examinations, respectively, were normal and equivocal. Intraoperatively, a palmaris profundus tendon was found compressing the median nerve and the patient had a palmaris longus tendon in the wrist. At 6-week follow-up, no recurrence of symptoms was reported. Abnormal nerve structures, although rarely encountered, should be considered in diagnosing CTS of the hand. Resection of the palmaris profundus tendon may help avoid possible recurrence of symptoms.

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