The clinical features of classic carpal tunnel syndrome are well known. However, some patients who may respond equally well to carpal tunnel release (CTR) display atypical signs and symptoms. The chief differential features are allodynia (painful dysesthesias), lack of finger flexion, and, on examination, pain on passive finger flexion. The goal of the study was to present the clinical features, increase awareness, facilitate accurate diagnosis, and report the outcomes after surgery. Thirty-five hands, from 22 patients with the main features of allodynia and lack of full finger flexion, were gathered in the period 2014-2021. The other common complaints included sleeping disturbances (20 patients), hand swelling (31 hands), and shoulder pain on the same side as the hand problem with limited range of motion (30 sides). The Tinel or Phalen signs were obscured by the pain. However, pain with passive flexion of the fingers was universally present. All the patients were treated with carpal tunnel release through a mini-incision approach: four patients had a trigger finger, which was treated concomitantly in six hands, and one patient underwent contralateral CTR for carpal tunnel syndrome with a more standard presentation. At a minimum of 6 months of follow-up (mean, 22 months; range, 6-60 months), the pain decreased by 7.5 ± 1.9 points on the Numerical Rating Scale, which ranges from 0 to 10. The pulp-to-palm distance improved from 3.7 to 0.3 cm. The mean Disabilities of the Arm, Shoulder, and Hand score decreased from 67 to 20. The mean Single-Assessment Numeric Evaluation score for the whole group was 9.7 ± 0.6. Hand allodynia and lack of finger flexion may be indications of median neuropathy in the carpal canal, which responds to CTR. Awareness of this condition is important because the uncharacteristic clinical presentation may not be considered an indication for surgery that can be beneficial. Therapeutic IV.