Multiple micro-organisms associated with Neuralgic Amyotrophy (NA) have been reported and Brucella species should be an important and overlooked infectious cause or trigger.We report a rare case of neuralgic amyotrophy associated with Brucella infection and is believed to be the first such case report in China. A 42-year-old male with brucellosis was confirmed serologically, who presented recurrent fever and fatigue and suddenly developed severe pain in the right shoulder within one week, followed by the inability to lift and abduct the proximal end of the right upper limb. Based on typical clinical manifestions, MRI neuroimaging of the brachial plexus and neuro-electrophysiological studies to confirm a diagnosis of NA and presented spontaneous recovery during this period, immunomodulatory treatment with corticosteroid or intravenous immunoglobulin had not been attempted, leaving a serious movement disorder in the right upper limb. Even rare, NA and other neurobrucellosis forms should be considered as complications of Brucella infection.