Neuralgic amyotrophy and hepatitis E infection: 6 prospective case reports

Abstract

IntroductionHepatitis E virus (HEV) represents the main cause of enterically transmitted hepatitis worldwide. It is known that neuralgic amyotrophy (NA) is one of the most frequent neurological manifestations of HEV. However, clinical, electrodiagnostic (EDX) and MRI characteristics, as well as long-term follow-up of HEV-related NA have not been fully described yet.Case reportsWe describe longitudinally clinical, EDX, biological and MRI results of six cases of HEV-associated NA, diagnosed from 2012 to 2017. Patients were between the ages of 33 and 57 years old and had a positive HEV serology. Clinical patterns showed the whole spectrum of NA, varying from extensive multiple mononeuropathy damage to single mononeuropathy. EDX results showed that the patients totalised 26 inflammatory mononeuropathies (1 to 8 per patient). These involved classical nerves such as suprascapular (6/6 cases), long thoracic (5/6 cases) and accessory spinal nerves (2/6 cases) and, some less frequent more distal nerves like anterior interosseous nerve (3/6 cases), as well as some unusual ones such as the lateral antebrachial cutaneous nerve (1/6 case), sensory fibres of median nerve (1/6 case) and phrenic nerves (1/6 case). After 2 to 8 years, all nerves had clinically recovered (muscle examination above 3/5 on MRC scale for all muscles except in one patient).DiscussionHEV should be systematically screened when NA is suspected, whatever the severity, if the onset is less than 4 months (before IgM HEV-antibodies disappear) and appears to be frequently associated with severe clinical and EDX pattern, without increasing the usual recovery time.

Neuralgic amyotrophy: an underrecognized entity

“…In addition, the lumbosacral plexus and the anterior and posterior interosseous, cranial, and phrenic nerves are peripheral nerves that are frequently involved. [6][7][8][9][10] Approximately 70% to 80% of patients manifest sensory deficits during episodes of NA; however, these deficits are usually mild compared with the degree of weakness. 14 Hyperesthesia and/or paresthesia are the most common sensory symptoms of NA, and hypoesthesia can also occur.…”

“…However, gadolinium-enhanced MRI can be helpful for the diagnosis of NA because inflammatory sites in the involved nerves show high signal intensity. 7,33 Before confirming a diagnosis of NA, MRI of the spine and ultrasound or MRI of the shoulder should be performed to rule out radiculopathy caused by a herniated disc or spinal stenosis and rotator cuff tear, respectively. 13 In addition, high-resolution magnetic resonance neurography is helpful for the diagnosis of hourglass-like constriction neuropathy (a subtype of NA).…”

“…In addition, the lumbosacral plexus and the anterior and posterior interosseous, cranial, and phrenic nerves are peripheral nerves that are frequently involved. 6–10…”

“…[2][3][4][5] Although NA affects the brachial plexus, this disorder also affects various other peripheral nerves, such as the suprascapular, long thoracic, phrenic, anterior interosseous, and radial nerves. [6][7][8][9][10] In many studies, NA has been described as a rare disorder. However, recent reports suggest that NA is significantly underdiagnosed in clinical practice and has an actual incidence rate of about 1 in 1000 per year.…”

“…2–5 Although NA affects the brachial plexus, this disorder also affects various other peripheral nerves, such as the suprascapular, long thoracic, phrenic, anterior interosseous, and radial nerves. 6–10…”