Painless idiopathic neuralgic amyotrophy after COVID-19 vaccination: A case report.
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ABSTRACT:
SUBMITTER: Diaz-Segarra N
PROVIDER: S-EPMC8250535 | biostudies-literature |
REPOSITORIES: biostudies-literature
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Project description:Neuralgic amyotrophy is an idiopathic neuropathy characterized by acute-onset pain, typically in the upper extremity or shoulder, followed by weakness of the associated muscles. Phrenic nerve involvement is rare. We report a 63-year-old man who presented with dyspnea and right shoulder pain after severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2) vaccination. His chest radiograph showed an elevated right hemidiaphragm that was absent before vaccination. A pulmonary function test showed a restrictive pattern with a significant reduction (40%) in forced vital capacity in the supine position. Diaphragm ultrasonography revealed a reduction in both diaphragmatic excursion and a thickening fraction of the right hemidiaphragm. Electrophysiological studies suggested a right upper brachial plexopathy. Considering the temporal relationship between the vaccination and absence of other causes, SARS-CoV-2 vaccination was thought to be the reason for neuralgic amyotrophy with diaphragmatic dysfunction. As there was no evidence of hypoventilation or sleep disturbance that may require noninvasive ventilation, the patient was followed with conservative treatment with analgesics. During 8 months of follow-up, his shoulder pain was relieved significantly but dyspnea improved only slightly. Neuralgic amyotrophy is an under-diagnosed etiology of diaphragmatic dysfunction and should be considered in patients with dyspnea and shoulder pain.
| S-EPMC9530308 | biostudies-literature
Project description:RationaleThis 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 concernA 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.DiagnosesWe 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.InterventionsIntravenous methylprednisolone partially relieved the pain and paralysis. Surgical neurolysis of the constricted left PIN was done for persistent paralysis.OutcomesThe 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.LessonsThis 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.
| S-EPMC6531285 | biostudies-literature
Project description:Neuralgic amyotrophy is a distinct clinical syndrome with acute severe pain and patchy paresis in the shoulder and arm region. The clinical phenotype was recently found to be more comprehensive and the long-term prognosis less optimistic than usually assumed for many patients. The disorder can be idiopathic or hereditary in an autosomal dominant fashion, with only few phenotypical variations between the two. This article provides a practical overview of current knowledge on the clinical presentation, diagnosis, pathogenesis and the treatment of pain and complications.
| S-EPMC2780663 | biostudies-literature
Project description:We report the findings in five muscle and three sural nerve biopsies, and in one postmortem plexus specimen, from six patients with hereditary neuralgic amyotrophy (HNA). We found that the sensory nerves are definitely involved in HNA despite the mainly motor symptoms, and that lesions in nerves and muscles are more widespread throughout the peripheral nervous system than clinically presumed, but, simultaneously, very focally affect isolated fascicles within individual nerves.
| S-EPMC1739556 | biostudies-other
Project description:ObjectiveTo determine whether there is an association between an acute preceding hepatitis E virus (HEV) infection and neuralgic amyotrophy (NA), and if so, whether patients with HEV-related NA differ from patients without an associated HEV infection.MethodsHEV testing was conducted in a retrospective cohort of 28 Cornish patients with NA (2011-2013) and a prospective cohort of 38 consecutive Dutch patients with NA (2004-2007). Acute-phase serum samples were analyzed for the presence of anti-HEV immunoglobulin (Ig) M and IgG and HEV RNA (quantitative real-time PCR).ResultsFive cases (10.6%) of acute hepatitis E infection were identified in a total group of 47 patients with NA of whom serum samples were available. In 4 patients, HEV RNA was detected in serum samples taken at presentation. All patients with HEV-associated NA had clinical and electrophysiologic evidence of bilateral brachial plexus involvement. Anti-HEV IgM positivity was not related to age, sex, disease severity, disease course, or outcome.ConclusionsAcute hepatitis E is found in 10% of patients with NA from the United Kingdom and the Netherlands. Further research is required to investigate the role of HEV in NA in other geographical locations and to determine pathophysiologic mechanisms.
| S-EPMC3937863 | biostudies-literature
Project description:Abstract Background The Coronavirus disease 2019 (COVID-19) pandemic has led to the rapid development of COVID-19 vaccine. The Centers for Disease Control and Prevention (CDC) has recently reported increase in myopericarditis incidence post-COVID-19 vaccination. Post-vaccination myopericarditis as side effect has been reported, however, is infrequent. We described a case of pericarditis post-first dose of Pfizer-BioNTech vaccine. Case summary A patient presented with typical symptoms of pericarditis and related electrocardiogram and echocardiogram changes, 7 days post receiving the first dose of COVID-19 vaccine. No other causes were identified from series of investigations. Patient had good symptomatic relief with non-steroidal anti-inflammatory medication. Discussion The incidence of pericarditis post-vaccination is rare, with limited reporting in previous literatures. No causal relationship has yet to be established due to small number of cases. The benefits of COVID-19 vaccination currently outweigh the side effect profile and are recommended as the first-line approach to control the current pandemic.
| S-EPMC8522432 | biostudies-literature
Project description:BACKGROUND:Hereditary neuralgic amyotrophy (HNA) is an autosomal dominant disorder that manifests as recurrent, episodic, painful brachial neuropathies. A gene for HNA maps to chromosome 17q25.3 where mutations in SEPT9, encoding the septin-9 protein, have been identified. OBJECTIVE:To determine the frequency and type of mutations in the SEPT9 gene in a new cohort of 42 unrelated HNA pedigrees. METHODS:DNA sequencing of all exons and intron-exon boundaries for SEPT9 was carried out in an affected individual in each pedigree from our HNA cohort. Genotyping using microsatellite markers spanning the SEPT9 gene was also used to identify pedigrees with a previously reported founder haplotype. RESULTS:Two missense mutations were found: c.262C>T (p.Arg88Trp) in seven HNA pedigrees and c.278C>T (p.Ser93Phe) in one HNA pedigree. Sequencing of other known exons in SEPT9 detected no additional disease-associated mutations. A founder haplotype, without defined mutations in SEPT9, was present in seven pedigrees. CONCLUSIONS:We provide further evidence that mutation of the SEPT9 gene is the molecular basis of some cases of hereditary neuralgic amyotrophy (HNA). DNA sequencing of SEPT9 demonstrates a restricted set of mutations in this cohort of HNA pedigrees. Nonetheless, sequence analysis will have an important role in mutation detection in HNA. Additional techniques will be required to find SEPT9 mutations in an HNA founder haplotype and other pedigrees.
| S-EPMC2683739 | biostudies-literature
Project description:Since severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2) was noted to cause coronavirus disease 2019 (COVID-19) in 2019, there have been many trials to develop vaccines against the virus. Messenger ribonucleic acid (mRNA) vaccine as a type of the vaccine has been developed and commercialized rapidly, but there was not enough time to verify the long-term safety. An 82-year-old female patient was admitted to the emergency room with dyspnea accompanied by stridor three days after the 3rd COVID-19 mRNA vaccination (Comirnaty, Pfizer-BioNTech, USA). The patient was diagnosed with bilateral vocal fold paralysis (VFP) by laryngoscope. Respiratory distress was improved after the intubation and tracheostomy in sequence. The brain, chest, and neck imaging tests, serological tests, cardiological analysis, and immunological tests were performed to evaluate the cause of bilateral VFP. However, no definite cause was found except for the precedent vaccination. Because bilateral VFP can lead to a fatal condition, a quick evaluation is necessary in consideration of VFP when dyspnea with stridor occurs after vaccination.
| S-EPMC9239840 | biostudies-literature
Project description:Septin 9 (SEPT9) interacts with microtubules (MTs) and is mutated in hereditary neuralgic amyotrophy (HNA), an autosomal-dominant neuropathy. The mechanism of SEPT9 interaction with MTs and the molecular basis of HNA are unknown. Here, we show that the N-terminal domain of SEPT9 contains the novel repeat motifs K/R-x-x-E/D and R/K-R-x-E, which bind and bundle MTs by interacting with the acidic C-terminal tails of ?-tubulin. Alanine scanning mutagenesis revealed that the K/R-R/x-x-E/D motifs pair electrostatically with one another and the tails of ?-tubulin, enabling septin–septin interactions that link MTs together. SEPT9 isoforms lacking repeat motifs or containing the HNA-linked mutation R88W, which maps to the R/K-R-x-E motif, diminished intracellular MT bundling and impaired asymmetric neurite growth in PC-12 cells. Thus, the SEPT9 repeat motifs bind and bundle MTs, and thereby promote asymmetric neurite growth. These results provide the first insight into the mechanism of septin interaction with MTs and the molecular and cellular basis of HNA.
| S-EPMC3871440 | biostudies-literature
Project description: Not available
| S-EPMC8343468 | biostudies-literature