Bilateral neuralgic amyotrophy in a patient with livestock-associated hepatitis E virus infection.

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Date: Apr. 4, 2022
From: CMAJ: Canadian Medical Association Journal(Vol. 194, Issue 13)
Publisher: CMA Impact Inc.
Document Type: Clinical report
Length: 2,650 words
Lexile Measure: 1820L

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A 52-year-old, previously healthy, right-handed male livestock trader woke in the middle of the night with acute, 9/10 right shoulder and forearm pain. He was unable to go back to sleep because of the pain, despite taking analgesics. The following day, he noticed weakness in his right hand and presented to the emergency department. He was found to have severe right shoulder pain and decreased active range of motion. Hand weakness was not documented. A presumptive diagnosis of inflammatory arthritis was made. Blood work showed normal complete blood count, electrolytes, creatinine kinase and erythrocyte sedimentation rate and elevated hepatic enzymes: aspartate aminotransferase 205 (normal range 16-51) U/L, alanine aminotransferase 472 (normal < 52) U/L, lactate dehydrogenase 861 (normal range 230-490) U/L, Y-glutamyl transferase 442 (normal range 5-38) U/L, and alkaline phosphatase 186 (normal range 36-144) U/L. The elevated hepatic enzymes were felt to be unrelated to the working diagnosis. The patient was prescribed prednisone 50 mg orally daily and analgesics and instructed to follow up with his family physician.

The following day, he presented to the emergency department again. Pain had spread to his left arm and right-hand weakness had progressed, leading to difficulty performing activities of daily living. An examination showed that he was unable to flex the first or second digits of his right hand; no other neurologic abnormalities were noted. Radiographs of the upper extremity were unremarkable. The case was discussed over the phone with the on-call neurologist, who felt the symptoms were likely related to the peripheral nervous system. Referral to neuromuscular medicine was recommended.

We assessed the patient in the neuromuscular medicine clinic 6 days after symptom onset. He had discontinued prednisone after 1 dose owing to perceived ineffectiveness. He described a constant, deep, aching 8/10 pain in both shoulders and forearms. He said he had no neck pain or sensory symptoms. Examination showed weakness in multiple nerve territories (Table 1), most notably in the anterior interosseous nerve (AIN) distribution, a branch of the median nerve. The patient could not flex the distal interphalangeal joints of digits 1 or 2 on the right (Figure 1) and had bilateral scapular winging (Figure 2). He had decreased sensation in the right axillary nerve territory. Reflexes were present (2+) and symmetric.

Acute pain followed by weakness involving the AIN, long thoracic and suprascapular nerves suggested neuralgic amyotrophy (NA). We considered cervical radiculopathy to be less likely, given the patient's weakness in multiple myotomes and absence of neck pain and paresthesia. Testing for vasculitic mononeuritis multiplex showed negative antineutrophil cytoplasm antibodies and normal C-reactive protein. Neuralgic amyotrophy may be provoked by infections. Given the bilateral involvement, elevated liver enzymes and occupational risk factor, we tested only for hepatitis E virus (HEV); HEV immunoglobulin M (IgM) and G (IgG) antibodies were positive. We started the patient on prednisone 60 mg orally daily for treatment of NA, with a 5-week taper. Within 1 week, his pain had resolved.

At 5-month follow-up, he reported no improvement in hand strength. Electromyography was performed...

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Gale Document Number: GALE|A698824074