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Hourglass-like constrictions are fascicular conditions confirmed definitively by interfascicular neurolysis. Certain peripheral nerves have vulnerable areas such as around the elbow in the posterior interosseous nerve. We report the first hourglass-like constriction in the brachial plexus supplying the radial innervated forearm musculature. Preoperative magnetic resonance imaging (MRI) findings of the brachial plexus were consistent with neuralgic amyotrophy (NA).A 9-yr-old boy experienced worsening left arm pain and difficulty in elevating the shoulder. Sequentially, severe palsy emerged when extending the wrist, thumb, and fingers. Based on the clinical picture, we diagnosed him with NA. The oblique coronal T2-weighted short-tau inversion recovery images showed mildly diffuse enlargement and hyperintensity of the brachial plexus. He showed few signs of improvement and interfascicular neurolysis was performed 11 mo after the onset. One of the fascicles in the posterior cord had developed an hourglass-like constriction. Electrical stimulation confirmed that the fascicle supplied forearm muscles. His wrist and finger extension had almost recovered at the 12-mo postoperative visit.Hourglass-like constrictions can occur in the brachial plexus. Although surgical approaches for the constrictions are still controversial, several reports demonstrated their effectiveness. Meanwhile, concerning NA treatment, evidence on the surgical intervention is lacking. Brachial plexus MRI might help in discerning the lesion and planning treatment options including surgical interventions. Hourglass-like constrictions are a possible etiology for certain NA patients with residual symptoms or paresis.