key: cord-0690660-n8yttzat
authors: Coll, Clemence; Tessier, Muriel; Vandendries, Christophe; Seror, Paul
title: Neuralgic amyotrophy and Covid 19 infection: 2 cases of spinal accessory nerve palsy
date: 2021-04-24
journal: Joint Bone Spine
DOI: 10.1016/j.jbspin.2021.105196
sha: 948c6b3bbfee4b65536b11a6050fc9dbd5f22962
doc_id: 690660
cord_uid: n8yttzat

Objective Neuralgic amyotrophy (NA), also known as Parsonage Turner Syndrome is often triggered by mechanical stress or upper respiratory tract viral infections. We reported 2 cases of shoulder weakness and amyotrophy related to spinal accessory nerve (SAN) palsy due to neuralgic amyotrophy occurring after COVID-19 infection. Methods For both patients, clinical history, clinical examination, electrodiagnostic (EDX), and imaging examinations invalidated other diagnoses but confirmed NA diagnosis. Results The NA involved only the SAN in both cases. EDX revealed a characteristic axonal lesion found in NA. SAN conduction study revealed normal latencies and low compound motor action potential amplitude for trapezius muscle when needle examination demonstrated a neurogenic pattern and denervation signs in the trapezius muscle. Both patient's MRI revealed denervation T2 hyper signal in impaired muscles, and hyper signal of the involved roots, trunks, or nerves without any mass, cyst, injury, fibrous band, or tearing signs along SAN course. Conclusions The Covid-19 infection could be the trigger for NA as many other viruses, and as it is a possible trigger for Guillain Barré Syndrome.

The most frequent and serious symptoms due to COVID-19 infection are related to severe acute respiratory syndrome (SARS) [1] . Neurological disorders have also been described, which may involve the central and the peripheral nervous systems, from the most frequent and benign such as anosmia to the rare and severe Guillain Barre Syndrome (GBS) [1, 2] . Neuralgic amyotrophy (NA) is defined as an acute and painful monophasic peripheral axonal neuropathy, with single or multiple nerve lesions that cause weakness, amyotrophy, and sensory loss in an asymmetric and patchy distribution, involving especially the upper limbs [3] . As GBS, it is presumed to have auto-immune and inflammatory pathophysiology. It is usually triggered by mechanical stress or upper respiratory tract viral infections [3, 4] . Three cases of neuralgic amyotrophy (NA) related to Covid-19 respiratory infections have been reported [5] [6] [7] . One was purely sensitive [5] , the second one involved supraspinatus, infraspinatus, teres minor, teres major, and trapezius muscles [6] ; and the third one involved the median nerve [7] . Hereafter, we have reported two cases of NA involving spinal accessory nerve (SAN) following documented SARS related to Covid-19 infection.

A 63-year-old man presented with a SARS related to Covid-19 infection, documented by positive nasopharyngeal swab PCR and suggestive chest CT-scan. Coronavirus infection was treated with Hydroxychloroquine, Azithromycin, Baracitinib, Ceftriaxone, and Dexamethasone. Due to worsening of respiratory distress, he required Mechanical Ventilation Resuscitation for 6 weeks; first by orotracheal intubation, then by percutaneous tracheotomy. When discharged from the intensive care unit (ICU), the patient experienced a period of mental confusion and agitation [8, 9] . Lumbar punction, electroencephalogram, and neuro-imagings were normal. So it is only one month after ICU discharge that the patient first complained of pain and disability in his right shoulder. The first clinical evaluation found J o u r n a l P r e -p r o o f 5 an active and passive limitation of shoulder range of motion, and muscular atrophy of upper trapezius muscle and supra-infra-spinous fossae [10, 11] . Other muscles' strength was normal especially the sternocleidomastoid (SCM) muscle. Adhesive capsulitis was diagnosed but this did not explain the importance of shoulder amyotrophy. Tendon reflexes were preserved. The patient also complained of paresthesia in the ulnar area of both forearms. Dynamic examination of the shoulder showed altered scapulothoracic rhythm: the scapula was mildly winged in the lateral position, and abruptly slid during the lateral elevation of the upper limb ( Figure 1 ) [11, 12] . An Electrodiagnostic examination (EDX) was One could see an abrupt sliding of the scapula laterally and downward during upper limb lateral elevation [11, 12] . Tendon reflexes were preserved. EDX performed 4 months after the Covid-19 infection onset, 

The SAN is the external terminal division of the eleventh cranial nerve; it crosses the cervical area to innervate the sternocleidomastoid muscle (SCM) and the three bundles of the trapezius muscle [11, 12] .

When it is damaged, consequences are atrophy and weakness of the trapezius muscle. It causes limitation of the elevation of shoulder with lateral position of the scapula at rest, mild lateral winging, and abrupt sliding of the scapula during lateral elevation. This clinical pattern is typical of what was found in both reported cases. As usual, the muscular strength of SCM was preserved [10] [11] [12] .

Three cases of neuralgic amyotrophy (NA) related to Covid-19 infections have already been reported [5] [6] [7] . One case was purely sensitive [5] and involves only the antebrachial cutaneous nerve. Another case involved supraspinatus, infraspinatus, teres minor, teres major, and trapezius muscles. MRI abnormalities were typical of NA but there was no EDX data available [6] . The third one [7] was described as an astonishing mild lesion (for NA diagnosis) of the median nerve with a partial conduction block in the forearm, and no denervation signs with needle examination.

NA is frequently an elimination diagnosis [3] . Before evoking NA in our two patients, we have ruled out damage of the SAN, with clinical examination, EDX, and MRIs. We first eliminated cervical spine or brachial plexus trauma or stretching, due to patient mobilizations in prone position, while they were under curare. History found neither surgery of the cervical region, nor venous jugular access, which could have been the cause of a SAN injury. The patients carried homolateral subclavian catheters, distant from the path of the SAN. One of them also had a controlateral catheter. Both patients had other associated upper limb conditions that could not explain the pain and that have been confounding for a time. In case 1 the patient had adhesive capsulitis that explained the limitation of passive shoulder mobility. This association Figures 1 (a, b) . These images of Case 1 shows the atrophy of the right upper trapezius muscle with a "pseudo atrophy of supraspinatus fossa" and a mild winging of the scapula: scapula is lateral, away from the spine, contrary to winging due to serratus anterior palsy that is important, and medial, near the spine [11, 12] .

Traces of SAN nerve conduction study of case 1 that demonstrates an important axonal lesion of the right SAN. One can see that latencies of the right side are similar to those of the normal left side, when compound motor action potential amplitudes are very low 2.4mV vs 7.5mV for upper trapezius muscle (TM), and 0.5mV vs 5.1mV for lower TM, which correspond respectively to a 66% and 90% axonal loss [11] .

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