Background and Purpose: Frozen shoulder (FS) is a common musculoskeletal condition. Researchers identified abnormalities in shoulder muscle performance in people with FS, such as muscle guarding, muscle stiffness, and alterations in muscle activation. Evidence has indicated that the central nervous system is also reorganized in patients with chronic musculoskeletal pain, which may contribute to altered neuromuscular control. Nevertheless, no study has investigated whether the central excitability changes with FS. The purposes of the study were to investigate the changes in shoulder kinematics, central excitability, muscle activation, and muscle tone in patients with FS. Methods: Thirteen participates with FS and 13 healthy controls were recruited. The outcomes included measurement of corticospinal excitability (active motor threshold, motor evoked potential, cortical silent period, short-interval intracortical inhibition, and short-interval intracortical facilitation), shoulder kinematics, muscle tone, and muscle activation of the upper trapezius (UT), infraspinatus (Isp), and pectoralis major (PM). Results: The FS group had a higher active motor threshold (77.09 ± 10.53 vs. 68.00 ± 15.56, p = 0.041), longer cortical silent period (124.42 ± 17.06 vs. 77.02 ± 29.72, p = 0.008), less short-interval intracortical facilitation (106.08 ± 24.25 vs. 88.26 ± 11.46, p = 0.021) of the UT, a tendency of lower activation of the UT (22.64 ± 8.08 vs. 31.70 ± 10.95, p = 0.072) and higher activation of the PM (8.00 ± 3.76 vs. 4.70 ± 1.75, p = 0.014), less range of humeral elevation (p = 0.001-0.029), scapular upward rotation (37.36 ± 15.74 vs. 55.60 ± 11.78, p = 0.005), and internal rotation (11.85 ± 6.90 vs. 18.70 ± 8.92, p = 0.043) during the functional tasks, and lower elasticity of the Isp (1.29 ± 0.18 vs. 1.17 ± 0.26, p = 0.046) than the healthy group. There were no differences in the findings of the other tests between the two groups. Conclusions: There were differences in the corticospinal excitability of the UT between patients with and without FS, along with abnormal muscle activation and shoulder kinematics during functional activities. These findings indicated that the neuromuscular control of the shoulder might be impaired and influence the motor performance in the patients with FS. Clinical Relevance: Clinicians could pay more attention to both central and peripheral neuromuscular control of the shoulder complex while evaluating and treating patients with FS.