Neurogenic thoracic outlet syndrome (NTOS) is an impairing painful condition. Patients usually report upper-limb pain, weakness and paresthesia. Shoulder weakness is frequently reported but has never been described with objective strength evaluation. We aimed to compare isokinetic shoulder strength between patients with NTOS and healthy controls. Patients and controls were prospectively evaluated with an isokinetic strength test at 60 and 180°/s, and an endurance test (30 repetitions at 180°/s) of the shoulder rotators. Patients were functionally assessed with QuickDASH questionnaires. One hundred patients and one hundred healthy subjects were included. Seventy-one percent of patients with NTOS were females with a mean age of 39.4 ± 9.6. They were compared to controls, 73% females and the mean age of 38.8 ± 9.8. Patients’ mean QuickDASH was 58.3 ± 13.9. Concerning the peak of strength at 60°/s, the symptomatic limbs of patients with NTOS had significantly 21% and 29% less strength than the control limbs for medial and lateral rotators, respectively (p ≤ 0.001). At 180°/s, the symptomatic limbs had significantly 23% and 20% less strength than the controls for medial and lateral rotators, respectively (p ≤ 0.001). The symptomatic limbs had significantly 45% and 30% less endurance than the controls for medial and lateral rotators, respectively (p ≤ 0.001). These deficits were correlated to the QuickDASH. Patients with NTOS presented a significant deficit of strength and endurance of the shoulder rotators correlated to disability. This highlights the interest in upper-limb strength evaluation in the diagnostic process and the follow-up of NTOS.