Hepatitis E and diaphragmatic dysfunction: Case series and review of the literature.

INTRODUCTION: The causes of diaphragmatic paresis are manifold. An association between neuralgic amyotrophy (NA) and hepatitis E virus (HEV) infection has been reported. We wondered about the prevalence of diaphragmatic disfunction and hepatitis E infection in our clinic. METHODS: From July 1st, 2020 to August 31st, 2023, patients presenting with diaphragmatic dysfunction and simultaneous clinical symptoms of an acute NA, or a history of NA, as well as patients with previously unexplained diaphragmatic dysfunction were examined for HEV infection. RESULTS: By August 31st, 2023, 13 patients with diaphragmatic dysfunction and HEV infection were diagnosed (4 women, 9 men). Mean age was 59 ± 10 years. Liver values were normal in all patients. The median latency to diagnosis was five months (range: 1-48 months); nine patients, 4 of them with typical symptoms of NA, presented with acute onset three patients showed bilateral diaphragmatic dysfunction. All patients had a positive IgG immunoblot. Seven patients, three with NA, had an elevated hepatitis E IgM titer and six of them also a positive IgM immunoblot. In all cases, O2C hepatitis genotype 3 was identified. In eight cases, all those with a high IgG titer >125, the O2 genotype 1 was also detected. CONCLUSION: NA that shows involvement of the phrenic nerve resulting in diaphragmatic dysfunction and dyspnoea, may be associated with HEV infection. The observation of 13 patients with diaphragmatic dysfunctions and HEV infection within a period of three years indicates a high number of undetected HEV-associated diaphragmatic dysfunction in the population, especially in the absence of NA symptoms. Therefore, even in diaphragmatic dysfunction without NA symptoms and causative damaging event, HEV infection should be considered, as it may represent a subform of NA with only phrenic nerve involvement. Therapy of HEV-associated diaphragmatic dysfunction in the acute phase is an open question. In view of the poor prognosis for recovery, antiviral therapy should be discussed. However, no relevant data are currently available.

Phrenic Nerve Involvement in Neuralgic Amyotrophy (Parsonage-Turner Syndrome).

Neuralgic amyotrophy is a poorly understood neuromuscular disorder affecting peripheral nerves mostly within the brachial plexus distribution but can also involve other sites including the phrenic nerve. In the classic form of the syndrome it causes proximal upper limb and neck pain on the affected side with subsequent muscle weakness that can be highly heterogeneous. Nocturnal noninvasive ventilation support is a first-line treatment after phrenic mononeuropathy. The regular monitoring of diaphragm function with spirometry and diaphragm ultrasound can help determine prognosis and inform decision-making.

Parsonage-Turner syndrome associated with hepatitis E infection in immunocompetent patients.

Introduction The hepatitis E virus (HEV) is the leading cause of acute hepatitis around the world. In recent years, knowledge has increased concerning extrahepatic manifestations caused by HEV, including neurological manifestations such as Parsonage-Turner syndrome (PTS). PTS is characterized by severe shoulder or arm pain and patchy paresis with muscle weakness. The aim of the present study was to assess the association between HEV and PTS. Materials and Methods We reported two cases of PTS associated with HEV, which were diagnosed in a short period of time in the same village. PTS was diagnosed by physical examination and electrophysiological studies, and serology testing for IgM, low-avidity IgG, and RNA of HEV established the diagnosis of acute HEV infection. Results A 44-year-old man who presented cervicobrachial pain accompanied by paresthesia, dyspnea, and isolated derangement of liver enzymes and 57-year-old women with cervical pain radiated to upper limbs, paresthesia, and liver cytolysis, although, this patient was initially diagnosed as having drug-induced hepatitis. Finally, the diagnosis was Parsonage- Turner syndrome associated with hepatitis e virus. In both patients, symptoms were bilateral and they required hospital admission. Both consumed vegetables are grown in a local patch and the phylogenetic analysis showed genotype 3f. Then, we reviewed the literature on PTS and HEV and we found 62 previously described cases that were more likely to be men (86.20 %) with more frequent bilateral symptoms (85.71 %). Genotype 3 is the most commonly associated. Three of those cases were diagnosed in Spain. Conclusions According to our findings, HEV should be considered in patients with neuralgic amyotrophy, including those with the absence of liver cytolysis.

Brachial Plexitis After Stem Cell Transplant.

BACKGROUND: Brachial plexitis is characterized by acute onset pain followed by weakness due to dysfunction of the nerves constituting the brachial plexus. The exact pathophysiology of this condition remains unknown but it is known to follow acute infections, such as a viral illness. CASE DESCRIPTION: We describe a case of idiopathic brachial plexitis in a patient after a peripheral stem cell transplant. The acute denervation of nerves in the brachial plexus may be due to reconstitution of his immune system. This is a rare entity and can be a diagnostic dilemma. CONCLUSIONS: Brachial plexitis may follow peripheral stem cell transplantation. Awareness of this phenomenon is helpful to clinicians involved in the care of these patients.

Backpack palsy and other brachial plexus neuropathies in the military population.

Brachial plexus neuropathy is often seen in the military population, especially due to pressure (backpack palsy, BPP) or idiopathic (neuralgic amyotrophy, NA). We aimed to gain insight in the disease characteristics of soldiers with brachial plexus neuropathies in the Dutch military population and to compare disease characteristics between patients with BPP and NA. In this retrospective chart review study we aimed to include all patients with brachial plexus neuropathy, who presented in the Joint Military Hospital between 1 January, 2011 and 31 December, 2016. We calculated the incidence of NA and BPP and Chi-square tests or Student t tests were performed for differences in patient characteristics between NA and BPP. We included 127 patients, 63 with BPP, 45 with NA, 10 with traumatic brachial plexus neuropathy, and 9 with other plexopathy. The incidence of brachial plexus neuropathy was 50/100 000 person years overall, 25/100 000 person years for BPP, and 18/100 000 person years for NA. Patients in the BPP group differed from the NA with regard to pain (BPP 41% vs NA 93%, P = .000), atrophy (13% BPP vs 29% NA, P = .049), and sensory symptoms (83% BPP vs 44% NA, P = .000). In the BPP group 90% had incomplete recovery and in the NA group 78%. Our study showed a high incidence of BPP and NA in the military population and suggests recovery is not so benevolent as previously thought. Future research is necessary to improve insight and outcome of military patients with brachial plexus neuropathies.

Síndrome de Parsonage-Turner en paciente sometido a cirugía de revascularización miocárdica. caso clínico / Parsonage-Turner syndrome: report of one case

We report a 59-year-old male who, three weeks after a coronary revascularization surgery, reported a sudden intense burning pain in his left upper limb. Two weeks later, he reports a paresis with difficulty to extend his left wrist and fingers. The electromyography showed a severe axonal damage of the radial nerve with distal denervation signs. This clinical picture probably corresponds to a neuralgic amyotrophy, an inflammatory disorder of the brachial plexus known by a number of terms, including Parsonage-Turner syndrome.

NA-CONTROL: a study protocol for a randomised controlled trial to compare specific outpatient rehabilitation that targets cerebral mechanisms through relearning motor control and uses self-management strategies to improve functional capability of the upper extremity, to usual care in patients with neuralgic amyotrophy.

BACKGROUND: Neuralgic amyotrophy (NA) is a distinct peripheral neurological disorder of the brachial plexus with a yearly incidence of 1/1000, which is characterised by acute severe upper extremity pain. Weakness of the stabilising shoulder muscles in the acute phase leads to compensatory strategies and abnormal motor control of the shoulder - scapular dyskinesia. Despite peripheral nerve recovery, scapular dyskinesia often persists, leading to debilitating residual complaints including pain and fatigue. Evidence suggests that persistent scapular dyskinesia in NA may result from maladaptive cerebral neuroplasticity, altering motor planning. Currently there is no proven effective causative treatment for the residual symptoms in NA. Moreover, the role of cerebral mechanisms in persistent scapular dyskinesia remains unclear. METHODS: NA-CONTROL is a single-centre, randomised controlled trial comparing specific rehabilitation to usual care in NA. The rehabilitation programme combines relearning of motor control, targeting cerebral mechanisms, with self-management strategies. Fifty patients will be included. Patients are recruited through the Radboud university medical center Nijmegen, the Netherlands. Patients with a (suspected) diagnosis of NA, with lateralized symptoms and scapular dyskinesia in the right upper extremity, who are 18 years or older and not in the acute phase can be included. The primary outcome is the Shoulder Rating Questionnaire score, which measures functional capability of the upper extremity. Secondary clinical outcomes include measures of pain, fatigue, participation, reachable workspace, muscle strength and quality of life. In addition, motor planning is assessed with first-person motor imagery and functional magnetic resonance imaging. In a sub-study the patients are compared to 25 healthy participants, to determine the involvement of cerebral mechanisms. This will enable interpretation of cerebral changes associated with the rehabilitation programme and functional impairments in NA. DISCUSSION: NA-CONTROL is the first randomised trial to investigate the effect of specific rehabilitation on residual complaints in NA. It also is the first study into the cerebral mechanisms that might underlie persistent scapular dyskinesia in NA. It thus may aid the further development of mechanism-based interventions for disturbed motor control in NA and in other peripheral neurological disorders. TRIAL REGISTRATION: ClinicalTrials.gov, NCT03441347 . Registered on 20 February 2018.

Suprascapular neuropathy: A review of 87 cases.

INTRODUCTION: Suprascapular neuropathy (SSN) is rare, with an estimated prevalence of 4.3% in patients with shoulder pain. METHODS: This retrospective chart review included patients with SSN seen during a 16-year period. Demographics and clinical information were recorded. Descriptive statistics, including percentages, means, and standard deviations, were computed for the variables of interest for all patients. RESULTS: Of 87 patients included in this study, trauma (n = 27) was the most common cause of SSN, followed by neuralgic amyotrophy (n = 21). Fifty-seven patients had isolated SSN. Others had SSN associated with axillary neuropathy (23 patients), brachial plexopathy (3 patients), and long thoracic, radial, or spinal accessory neuropathy (1 patient each). DISCUSSION: SSN is commonly associated with axillary neuropathy. Trauma remains the most common cause of SSN. Electrodiagnostic findings aid in the initial diagnosis and may indicate the need for close clinical follow-up based on the severity of the axonal injury.

Compound heterozygous CACNA1H mutations associated with severe congenital amyotrophy.

Neuromuscular disorders encompass a wide range of conditions often associated with a genetic component. In the present study, we report a patient with severe infantile-onset amyotrophy in whom two compound heterozygous variants in the gene CACNA1H encoding for Cav3.2 T-type calcium channels were identified. Functional analysis of Cav3.2 variants revealed several alterations of the gating properties of the channel that were in general consistent with a loss-of-channel function. Taken together, these findings suggest that severe congenital amyoplasia may be related to CACNA1H and would represent a new phenotype associated with mutations in this gene.

A CARE-compliant article: a case report of idiopathic brachial neuritis treated with ultrasound-guided electroacupuncture.

RATIONALE: Electroacupuncture is commonly used for treating nerve injury. However, studies published in recent years have not described an appropriate method for accurately identifying the location and depth of injured nerves beneath the acupoints. PATIENT CONCERNS: A 26-year-old male patient had left shoulder pain and weakness after tetanus, diphtheria, and pertussis vaccination and was diagnosed with idiopathic brachial neuritis 24 months before this study. The patient had undergone prednisone and ibuprofen treatment in another hospital, but the therapeutic effect was poor and limited. DIAGNOSES: The nerve conduction studies showed decreased amplitude over the left supraspinatus and deltoid muscles. Electromyography showed increased giant waves and polyphasic waves with reduced recruitments in the left deltoid muscle and increased giant waves with reduced recruitment in the left supraspinatus muscle. The condition was diagnosed with idiopathic brachial neuritis. INTERVENTIONS: Ultrasound was used to identify the location and depth of axillary and suprascapular nerves, and direct electroacupuncture was conducted at the quadrangular space and suprascapular notch to stimulate the nerves. Other needles were placed according to deltoid and supraspinatus muscles origins and insertions. The procedure was conducted once a week, and rehabilitation activities were conducted daily. OUTCOMES: The patient experienced significant improvements of left shoulder pain and muscle weakness after ultrasound-guided electroacupuncture treatment. The total shoulder pain and disability index score declined from 49.23% to 11.54%. The scores of both pain and disability domains improved and maintained stable declining after the intervention. The disability of the arm, shoulder; and hand scores declined from 60 to 23.3. According to amplitude data from nerve conduction studies, the injured axillary nerve showed remarkable improvement in the third month. Muscle strength improved to the normal state. The patient was generally satisfied with the ultrasound-guided electroacupuncture treatment. LESSON: Ultrasound-guided electroacupuncture was based on anatomical correlations between nerves and muscles and on electrical stimulation theories. The results suggest that this intervention might be an alternative therapy for idiopathic brachial neuritis. Furthermore, in this study, it had minimal adverse effects. This therapy is demonstrated to be effective in future controlled studies.

Bilateral idiopathic neuralgic amyotrophy involving selective branches of peripheral nerves with a stepwise progression: A case report.

RATIONALE: This is a report about a rare case of idiopathic neuralgic amyotrophy (INA) involving selective peripheral nerve branches of bilateral upper extremities, which exhibited a stepwise progression. PATIENT CONCERN: A 66-year-old woman presented with paresis of selective branches of bilateral median nerves, followed by paresis of bilateral posterior interosseous nerve (PIN) 8 weeks later. DIAGNOSES: We diagnosed it as INA involving the selective motor branches of bilateral median nerves and bilateral PINs. Forearm magnetic resonance imaging combined with electrodiagnostic testing helped accurately identify the affected regions, and ultrasonography demonstrated a severe constriction of the left PIN. INTERVENTIONS: Intravenous methylprednisolone partially relieved the pain and paralysis. Surgical neurolysis of the constricted left PIN was done for persistent paralysis. OUTCOMES: The muscle power of the bilateral median nerve territories was recovered to nearly normal, but the muscle power of the left PIN territories remained at grade 1. LESSONS: This case indicates that INA can manifest as a multiple mononeuropathy involving individual fascicular levels of peripheral nerve branches with focal constriction, and electrodiagnostic study combined with forearm MRI and ultrasonography can help in identifying affected lesion and predicting the prognosis.

[Parsonage-Turner syndrome: Unknown but not infrequent. Apropos of 6 cases]. / Síndrome de Parsonage-Turner: desconocido pero no infrecuente. A propósito de 6 casos.

Shoulder pain is a common reason for seeking emergency, primary and specialist care. Parsonage-Turner syndrome should form part of the differential diagnosis of shoulder pain. Familiarity with the clinical and electrophysiological characteristics of this syndrome could help prevent iatrogenic disease. We present 6 cases of Parsonage-Turner syndrome, a syndrome of unknown etiology with a typical clinical picture. All patients had acute onset of the syndrome with intense shoulder pain of approximately 3 weeks' duration. After this phase ended, there was noticeable weakness of the affected arm, causing muscular atrophy depending on the muscles affected by the neuropathy. Winged scapula was present in all patients. One patient showed alteration of sensitivity and another showed hemidiaphragmatic paralysis. After a complete physical examination, the patients underwent electromyographic study that confirmed the diagnosis. Analgesic treatment and rehabilitation were prescribed.

[Parsonage-Turner syndrome. Report of one case]. / Síndrome de Parsonage-Turner en paciente sometido a cirugía de revascularización miocárdica. Caso clínico.

We report a 59-year-old male who, three weeks after a coronary revascularization surgery, reported a sudden intense burning pain in his left upper limb. Two weeks later, he reports a paresis with difficulty to extend his left wrist and fingers. The electromyography showed a severe axonal damage of the radial nerve with distal denervation signs. This clinical picture probably corresponds to a neuralgic amyotrophy, an inflammatory disorder of the brachial plexus known by a number of terms, including Parsonage-Turner syndrome.

Herpes zoster increased risk of neuralgic amyotrophy: a retrospective, population-based matched cohort study.

Although neuralgic amyotrophy (NA) has occasionally been reported to be associated with reactivated herpes zoster, their associated risk remains unknown. The aim of this study was to assess the risk of developing NA following preceding herpes zoster. The authors used the National Health Insurance Research Database of Taiwan to select 41,548 patients with newly diagnosed herpes zoster during the period 2000 to 2010 and randomly extracted 166,192 matched control subjects. All participants in the study and control groups were followed for 3 months after the diagnosis to identify those who developed NA. Cox proportional hazards regression analyses were performed to evaluate the subsequent risk of NA. Twenty-one subjects from the group with herpes zoster (0.05%) developed NA over the 3-month period and 46 from the group without herpes zoster (0.03%). The patients with herpes zoster had a higher risk of developing NA (adjusted hazard ratio = 1.408, 95% confidence interval = 1.013-2.319, P = 0.030). In the patients with herpes zoster, female sex, age ≥ 65, hepatitis E virus (HEV), and having had a recent infectious event including pneumonia and influenza were risk factors for developing NA (adjusted HR 2.746, 1.998, 2.735, 2.016, and 1.718, respectively, all P < 0.05). Patients with herpes zoster attack have a higher risk of developing NA over a 3-month period after diagnosis, especially those who are female, age ≥ 65, HEV, or have experienced a recent infectious event or pneumonia and influenza.

Is pharmacologic treatment better than neural mobilization for cervicobrachial pain? A randomized clinical trial.

Purpose: This study aim was to compare the effectiveness of the median nerve neural mobilization (MNNM) and cervical lateral glide (CLG) intervention versus oral ibuprofen (OI) in subjects who suffer cervicobrachial pain (CP). Methods: This investigation was a, multicenter, blinded, randomized controlled clinical trial (NCT02595294; NCT02593721). A number of 105 individuals diagnosed with CP were enrolled in the study and treated in 2 different medical facilities from July to November 2015. Participants were recruited and randomly assigned into 3 groups of 35 subjects. Intervention groups received MNNM or CLG neurodynamic treatments, and the (active treatment) control group received an OI treatment for 6 weeks. Primary outcome was pain intensity reported through the Numeric Rating Scale for Pain (NRSP). Secondary outcomes were physical function involving the affected upper limb using the Quick DASH scale, and ipsilateral cervical rotation (ICR) using a cervical range of motion (CROM) device. Assessments were performed before and 1 hour after treatment for NRSP (baseline, 3 and 6 weeks) and CROM (baseline and 6 weeks), as well as only 1 assessment for Quick DASH (baseline and 6 weeks). Results: Repeated-measures ANOVA intergroup statistically significant differences were shown for CP intensity (F(2,72) = 22.343; P < .001; Eta2 = 0.383) and Quick DASH (F(2,72) = 15.338; P < .001; Eta2 = 0.299), although not for CROM (F(2,72) = 1.434; P = .245; Eta2 = 0.038). Indeed, Bonferroni´s correction showed statistically significant differences for CP intensity (P < .01; 95% CI = 0.22 - 3.26) and Quick DASH reduction (P < .01; 95% CI = 8.48 - 24.67) in favor of the OI treatment at all measurement moments after baseline. Conclusions: OI pharmacologic treatment may reduce pain intensity and disability with respect to neural mobilization (MNNM and CLG) in patients with CP during six weeks. Nevertheless, the non-existence of between-groups ROM differences and possible OI adverse effects should be considered.