Parsonage-Turner syndrome, affecting suprascapular nerve and especially to infraspinatus muscles after COVID-19 vaccination in a professional wrestler, a case report and literature review of causes and treatments.

BACKGROUND: Acute peripheral neuropathy, also known as Parsonage-Turner syndrome or neuralgic amyotrophy, mostly affects the upper brachial plexus trunks, which include the shoulder girdle. It is typically accompanied by abrupt, intense pain, weakness, and sensory disruption. The etiology and causes of this disease are still unknown because of its low prevalence, however viral reactions-induced inflammation is one of its frequent causes. CASE PRESENTATION: Here, we introduce a professional wrestler patient who was diagnosed with PTS after vaccination and was treated, and we review some articles in this field. CONCLUSION: When it comes to shoulder-girdle complaints and pain, Parsonage-Turner syndrome can be a differential diagnosis. Corticosteroids during the acute period, followed by physical therapy, appear to be an efficient way to manage pain, inflammation, muscular atrophy, and the process of recovering to full nerve regeneration.

Asociación de trasplante de progenitores hematopoyéticos y síndrome de Parsonage-Turner: ¿coincidencia o relación causa-efecto? / Association between hematopoietic stem cell transplantation and Parsonage Turner syndrome: coincidence or cause-effect relationship?

El síndrome de Parsonage-Turner o plexopatía braquial idiopática es una inflamación total o parcial del plexo braquial cuya presentación típica es una omalgia intensa y súbita, seguida de debilidad braquial y amiotrofia precoz. La etiología es desconocida, aunque se propone un mecanismo inmunomediado.El trasplante de progenitores hematopoyéticos es un tratamiento bien establecido de las neoplasias hematológicas y tiene un papel creciente en el tratamiento de enfermedades autoinmunes. Los efectos adversos neurológicos son probablemente infradiagnosticados.La asociación del síndrome de Parsonage-Turner y el trasplante de progenitores hematopoyéticos es muy poco conocida. Describimos dos casos clínicos de plexopatía braquial idiopática tras trasplante de células stem (progenitores) hematopoyéticas (TPH).La reconstitución del sistema inmune tras un trasplante de progenitores hematopoyéticos puede ser un desencadenante de plexopatía braquial, aunque se necesitan más estudios para entender la fisiopatología de esta entidad y establecer su relación causal con el trasplante. (AU)

Parsonage-Turner syndrome or idiopathic brachial neuritis is a total or partial inflammation of the brachial plexus, with a typical presentation as a sudden and very intense pain in the shoulder, followed by weakness and early amyotrophy. The etiology is still unknown, although an immune mediated mechanism is thought to be involved.Hematopoietic stem cell transplantation is a well-established treatment for hematological malignancies, but with a growing implication in the treatment of autoimmune diseases. The neurological side effects are probably underdiagnosed.The association of the Parsonage-Turner syndrome and the hematopoietic stem cell transplantation is scarce. We describe two clinical cases of idiopathic brachial plexopathy after hematopoietic stem cell transplantation.The reconstruction of the immune system after a transplant may be the trigger of a brachial plexopathy, but more studies are necessary for the etiology of this disease to be understood and to establish a cause-effect relation with the transplant. (AU)

Parsonage Turner syndrome after needle electromyography: a needle in a haystack.

.

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.

COVID-19 Vaccine Related Cervical Radiculitis and Parsonage-Turner Syndrome: Case Report and Review of the Literature.

Parsonage-Turner Syndrome, or neuralgic amyotrophy, is an acute-onset upper limb and shoulder girdle palsy that can occur in a post-viral, post-surgical or idiopathic setting. There have also been some reported cases of the syndrome occurring following vaccinations. The pathophysiology of neuralgic amyotrophy is not completely understood and many of the commonly used diagnostic imaging modalities we use to try and diagnose this syndrome are inaccurate and misleading. We present the case of a 40-year-old gentleman who presented with acute onset burning pain and fasciculations in his right upper extremity following vaccination with the second dose of the Pfizer-BioNTech COVID-19 vaccine. His symptoms progressed to weakness in isolated muscle groups with electromyographic evidence of decreased nerve conduction. MRI of the cervical spine demonstrated multilevel central and foraminal stenosis, suggesting a diagnosis of cervical radiculopathy. The patient underwent a C4-5/C5-6 and C6-7 laminoforaminotomy and tolerated the procedure well. Post-operatively, the patient has experienced gradual symptom improvement with residual right triceps and pectoralis muscle weakness as well as paresthesias of the right elbow and forearm. Parsonage-Turner Syndrome is a brachial plexus palsy that can affect one or multiple branches of the brachial plexus. It causes acute-onset pain and weakness, and the diagnosis can be difficult to make with the commonly used diagnostic imaging methods. We reviewed other case reports about neuralgic amyotrophy following vaccinations as well as the current literature on more accurate diagnostic imaging modalities that may help our diagnosis and understanding of the pathophysiology of this condition.

[Neuralgic amyotrophy: an inflammatory neuropathy and its surgical treatment]. / Neuralgische Amyotrophie ­ eine entzündliche Neuropathie und ihre operative Behandlung.

BACKGROUND: Neuralgic amyotrophy (NA) is a monofocal or oligofocal inflammatory neuropathy whose incidence has been significantly underestimated. A connection between constrictions and torsions of peripheral nerves with this disease has been increasingly established in recent years. Modern imaging techniques such as high-resolution nerve ultrasound and MR neurography have contributed to a better understanding of the pathophysiology and a better assessment of the prognosis of the disease. This has led to the concept of treating patients with such focal changes surgically in order to improve the prognosis. This review presents current ideas on the pathophysiology, clinical presentation, diagnosis and treatment of the disease. PATIENTS AND METHODS: In a retrospective study, pre-, intra- and postoperative findings of 22 patients with 23 constrictions/torsions of peripheral nerves of the upper extremity were analysed. The patients underwent surgery at a nerve surgery centre over a period of 3.5 years (Dec. 2019-May 2023). The median nerve was most frequently affected (N=9), followed by the suprascapular nerve (N=6) and radial nerve (N=4). The axillary nerve (N=3) and the accessory nerve (N=1) were also involved. Surgical exploration revealed nerve torsions (N=9), nerve constrictions (N=5), fascicular torsions (N=12) and fascicular constrictions (N=9). Depending on the intraoperative findings, epineuriotomies (N=1), epi- and perineuriotomies (N=33), end-to-end sutures (N=2), and one epi- and one perineural suture were performed. RESULTS: After an average follow-up of 10 months (3-28 months), 17 patients were re-examined. All of them reported a clear subjective improvement in motor deficits. Clinically and electromyographically, a reinnervation and significant increase in strength from a pre-existing strength grade of M0 to at least M3 in the vast majority of affected muscles was demonstrated in these patients. SUMMARY: The incidence of NA continues to be underestimated and, in a significant proportion of patients, leads to permanent motor deficits, most likely due to constrictions and torsions of affected nerves. Surgical treatment is recommended as early as possible. Very good results can usually be achieved with epi- and perineuriotomy. In rare cases, end-to-end neurorrhaphy or nerve grafting is required.

Parsonage-Turner Syndrome and Hereditary Brachial Plexus Neuropathy.

Parsonage-Turner syndrome and hereditary brachial plexus neuropathy (HBPN) present with indistinguishable attacks of rapid-onset severe shoulder and arm pain, disabling weakness, and early muscle atrophy. Their combined incidence ranges from 3 to 100 in 100,000 persons per year. Dominant mutations of SEPT9 are the only known mutations responsible for HBPN. Parsonage and Turner termed the disorder "brachial neuralgic amyotrophy," highlighting neuropathic pain and muscle atrophy. Modern electrodiagnostic and imaging testing assists the diagnosis in distinction from mimicking disorders. Shoulder and upper limb nerves outside the brachial plexus are commonly affected including the phrenic nerve where diaphragm ultrasound improves diagnosis. Magnetic resonance imaging can show multifocal T2 nerve and muscle hyperintensities with nerve hourglass swellings and constrictions identifiable also by ultrasound. An inflammatory immune component is suggested by nerve biopsies and associated infectious, immunization, trauma, surgery, and childbirth triggers. High-dose pulsed steroids assist initial pain control; however, weakness and subsequent pain are not clearly responsive to steroids and instead benefit from time, physical therapy, and non-narcotic pain medications. Recurrent attacks in HBPN are common and prophylactic steroids or intravenous immunoglobulin may reduce surgical- or childbirth-induced attacks. Rehabilitation focusing on restoring functional scapular mechanics, energy conservation, contracture prevention, and pain management are critical. Lifetime residual pain and weakness are rare with most making dramatic functional recovery. Tendon transfers can be used when recovery does not occur after 18 months. Early neurolysis and nerve grafts are controversial. This review provides an update including new diagnostic tools, new associations, and new interventions crossing multiple medical disciplines.

Orthopnea secondary to brachial plexitis with bilateral diaphragmatic paralysis.

BACKGROUND: Diaphragmatic paralysis can present with orthopnea. We report a unique presentation of bilateral diaphragmatic paralysis, an uncommon diagnosis secondary to an unusual cause, brachial plexitis. This report thoroughly describes the patient's presentation, workup, management, and outcome. It also reviews the literature on diaphragmatic paralysis and Parsonage-Turner syndrome. CASE PRESENTATION: A 50-year-old male patient developed insidious orthopnea associated with left shoulder and neck pain over three months with no associated symptoms. On examination, marked dyspnea was observed when the patient was asked to lie down; breath sounds were present and symmetrical, and the neurological examination was normal. The chest radiograph showed an elevated right hemidiaphragm. Echocardiogram was normal. There was a 63% positional reduction in Forced Vital Capacity and maximal inspiratory and expiratory pressures on pulmonary function testing. The electromyogram was consistent with neuromuscular weakness involving both brachial plexus and diaphragmatic muscle (Parsonage and Turner syndrome). CONCLUSIONS: Compared to unilateral, bilateral diaphragmatic paralysis may be more challenging to diagnose. On PFT, reduced maximal respiratory pressures, especially the maximal inspiratory pressure, are suggestive. Parsonage-Turner syndrome is rare, usually with unilateral diaphragmatic paralysis, but bilateral cases have been reported.

[Association between hematopoietic stem cell transplantation and Parsonage Turner syndrome: coincidence or cause-effect relationship?] / Asociación de trasplante de progenitores hematopoyéticos y síndrome de Parsonage-Turner: ¿coincidencia o relación causa-efecto?

Parsonage-Turner syndrome or idiopathic brachial neuritis is a total or partial inflammation of the brachial plexus, with a typical presentation as a sudden and very intense pain in the shoulder, followed by weakness and early amyotrophy. The etiology is still unknown, although an immune mediated mechanism is thought to be involved. Hematopoietic stem cell transplantation is a well-established treatment for hematological malignancies, but with a growing implication in the treatment of autoimmune diseases. The neurological side effects are probably underdiagnosed. The association of the Parsonage-Turner syndrome and the hematopoietic stem cell transplantation is scarce. We describe two clinical cases of idiopathic brachial plexopathy after hematopoietic stem cell transplantation. The reconstruction of the immune system after a transplant may be the trigger of a brachial plexopathy, but more studies are necessary for the etiology of this disease to be understood and to establish a cause-effect relation with the transplant.

Effects of indirect foraminal decompression during anterior cervical disc fusion procedure: preliminary results of a prospective study with clinical and radiological outcomes.

INTRODUCTION: The respective effects of direct and indirect decompression in the clinical outcome after anterior cervical disc fusion (ACDF) is still debated. The main purpose of this study was to analyze the effects of indirect decompression on foraminal volumes during ACDF performed in patients suffering from cervico-brachial neuralgias due to degenerative foraminal stenosis, i.e. to determine whether implant height was associated with increased postoperative foraminal height and volume. METHODS: A prospective follow-up of patients who underwent ACDF for cervicobrachial neuralgias due to degenerative foraminal stenosis was conducted. Patient had performed a CT-scan pre and post-operatively. Disc height, foraminal heights and foraminal volumes were measured pre and post operatively. RESULTS: 37 cervical disc fusions were successfully performed in 20 patients, with a total of 148 foramina studied. Foraminal height and volume were measured bilaterally on the pre- and post-operative CT scans (148 foramina studied). After univariate analysis, it was found a significant improvement for every radiological parameter, with a significant increase in disc height, foraminal height and foraminal volume being respectively +3,22 mm (p < 0,001), +2,12 mm (p < 0,001) and +54 mm3 (p < 0,001). Increase in disc height was significantly associated with increase in foraminal height (p < 0,001) and foraminal volume (p < 0,001). At the same time, increase in foraminal height was significantly correlated with foraminal volume (p < 0,001), and seems to be the major component affecting increasing in foraminal volume. CONCLUSION: Indirect decompression plays an important part in the postoperative foraminal volume increase after ACDF performed for cervicobrachial neuralgias.

Activación muscular del hombro y dolor en nadadores infantiles / Ativação muscular do ombro e dor em crianças nadadoras / Shoulder muscle activation and pain in child swimmers

Objetivo: Comparar la activación electromiográfica durante la simulación de la fase de propulsión de la brazada de nado crol con banda elástica de ungrupo de nadadores con dolor y un grupo control, antes y después de entrenamiento preventivo (EP) de hombro y en la fase de desentrenamiento, enrelación al dolor y a una contracción voluntaria máxima (CVM) de 11 músculos de la cintura escapular.Método: Se utilizó una muestra de 12 nadadores de la categoría infantil (11 a 12 años). Fueron evaluados con cinco pruebas clínicas, cada una específicaa músculo o tendón en relación al gesto deportivo. Fue utilizada una escala de dolor. Para los datos mioeléctricos fueron colocados electrodos desuperficie en los 11 músculo y evaluado la CVM y el movimiento de la brazada. Los atletas realizaron en un período de diez semanas ejercicios defortalecimiento muscular y estabilizadores de escápula, con frecuencia semanal de dos y duración de 45 min cada sesión, luego de ello se volvió aejecutar el protocolo de evaluación nuevamente.Resultados: Se constataron diferencias entre los grupos en las pruebas clínicas y en comparación a la EMG en la fase pre EP, post EP y desentrenamiento.Conclusión: Los EP tuvieron efectos positivos, alteraciones en los patrones musculares y en la disminución del dolor de hombros de los atletas.(AU)

Objective: To compare the electromyographic activation during the simulation of the propulsion phase of the crawl stroke with elastic band of a group ofswimmers with pain and a control group, before and after preventive shoulder training (PT) and in the detraining phase, in relation to pain and amaximum voluntary contraction (MVC) of 11 muscles of the shoulder girdle.Method: A sample of 12 swimmers from the children's category (11-12 years old). They were evaluated with five clinical tests, each one specific to muscleor tendon in relation to the sporting gesture. A pain scale was used. For myoelectric data, surface electrodes were placed over 11 muscle evaluated, wasperformed a MVC for each muscle and a propulsive swimming movement. The athletes performed muscle strengthening and scapula stabilizing exercisesfor a period of ten weeks, with a weekly frequency of two and a duration of 45 min each session, after which the evaluation protocol was performedagain.Results: Differences were found betweengroups in the clinical tests and in comparison to electromyographic activation in the pre-PT, post-PT anddetraining phases.Conclusion: Preventive exercises had positive effects, on muscle patterns and decreasing shoulder pain in athletes.(AU)

Objetivo: Comparar a ativação eletromiográfica durante a simulação da fase de propulsão do nado crawl, fora da água, com uma faixa elástica de umgrupo de nadadores com dor e um grupo controle (atletas sem dor). Antes e depois do treinamento preventivo (EP) de ombros e na fase dedestreinamento, em relação a dor ea contração voluntária máxima (CVM) de 1l músculos da cintura escapular.Método: Participaram deste estudo 12 nadadores da categoria infantil (11 a 12 anos). Foram avaliados com cinco testes clínicos, cada um específico para músculo ou tendão em relação ao gesto esportivo, através da escala de dor. Foram colocados sensores para a eletromiografia de superfície, foi realizadaavaliação da CVM e movimento da braçada. Os atletas realizaram em um período de dez semanas os exercícios de fortalecimento muscular eestabilizadores da escápula (supervisionados por profissional), com frequência semanal de duas e duração de 45 min cada sessão, após os quais oprotocolo de avaliação foi executado novamente.Resultados: Foram encontradas diferenças na comparação com a eletromiografia nas fases pré-EP, pós-EP e destreinamento.Conclusão: EP teve efeitos positivos, como possíveis alterações nos padrões musculares e na redução da dor no ombro em atletas infantis.(AU)

Un caso de neuralgia amiotrófica tras vacunación frente a COVID-19 / A case of neuralgic amyotrophy after vaccination against COVID-19

Introducción: La neuralgia amiotrófica es un trastorno inflamatorio del plexo braquial con una fisiopatología poco conocida, posiblemente relacionada con fenómenos inmunológicos. Se caracteriza por dolor agudo e intenso, acompañado de debilidad muscular y alteración de los resultados en los estudios electromiográficos y de conducción nerviosa. Al no tener un tratamiento específico, se aborda a través de fisioterapia y el control sintomático. Caso clínico: Presentamos el caso de una mujer de 37 años, sin antecedentes relevantes, que desarrolló el cuadro tras ser vacunada contra el SARS-CoV-2. La paciente respondió de forma discreta a la fisioterapia, analgesia convencional y corticosteroides. Después de 10 meses de tratamiento, se tomó la decisión de utilizar radiofrecuencia pulsada y bloqueo eco-guiado del plexo, lo que logró mejores resultados. Un año después de la aparición del dolor, los síntomas de la paciente habían mejorado, aunque no había podido reincorporarse a su actividad profesional previa como policía. Discusión: Se han publicado muy pocos casos de neuralgia amiotrófica después de la vacunación contra el SARS-CoV-2. El diagnóstico diferencial incluye diversos trastornos musculoesqueléticos y neurológicos comunes, que pueden despistar al profesional y retrasar la identificación de esta entidad, especialmente en el contexto de campañas de vacunación masivas con gran volumen de reacciones adversas. La analgesia convencional a menudo es insuficiente para abordar los problemas de estos pacientes, por lo que se debe prever la necesidad de programar técnicas invasivas. Dada la rareza de la afección y su impacto en la vida personal y profesional del paciente, se destaca la importancia de un diagnóstico precoz y una comunicación fluida. Finalmente, se subraya el valor de la declaración de reacciones adversas como un signo de profesionalidad y un activo para establecer una relación médico-paciente constructiva.(AU)

Introduction: neuralgic amyotrophy is an inflammatory disorder of the brachial plexus with a poorly understood pathophysiology, possibly related to immunological phenomena. It is characterized by acute and intense pain, accompanied by muscle weakness and altered results in electromyographic and nerve conduction studies. Having no specific treatment, it is approached through physiotherapy and symptomatic control. Case history: we present the case of a 37-year-old woman, with no relevant history, who developed the condition after being vaccinated against SARS-CoV-2. The patient responded discreetly to physical therapy, conventional analgesia and corticosteroids. After 10 months of treatment, the decision was made to use pulsed radiofrequency and ultrasound-guided plexus block of the plexus, which achieved better results. A year after the onset of pain, the patient’s symptoms had improved, although she had not been able to return to her professional activity as a policewoman. Discussion: very few cases of neuralgic amyotrophy have been reported after SARS-CoV-2 vaccination. The differential diagnosis includes many common muskuloskeletal and neurologic disorders, which can mislead professionals and delay identification of the disease, especially in the context of massive vaccination campaigns. Conventional analgesia is often insufficient to address these patients’ complaints; thus, scheduled invasive techniques need to be considered. Given the rarity of the condition and its impact on the personal and professional life of the patient, we highlight the importance of an early diagnosis and smooth communication with the patient. Finally, we stress the value of declaring adverse reactions as a sign of professionalism and an asset in establishing a constructive doctor-patient relationship.(AU)

Dolor y rehabilitación en síndrome de Parsonage-Tuner por SARS-CoV-2: a propósito de un caso / Pain and rehabilitation in Parsonage-Tuner syndrome due to SARS-CoV-2: a case report

El síndrome de Parsonage-Tuner es una entidad neurológica poco frecuente de etiología desconocida, asociada con infecciones virales, trauma y antecedentes vacunales. En el marco de la pandemia por COVID-19 se han reportado casos de esta entidad neurológica como complicación infecciosa, con un desarrollo clínico del dolor, la debilidad y estudios electrofisiológicos típicos. Se genera la necesidad de considerar al síndrome de Parsonage-Tuner en los diagnósticos diferencias de las monoplejías en los pacientes con infección por COVID-19, además de fomentar estrategias de abordaje clínico. En la literatura se han descrito menos de 7 casos, siendo este el primero en Latinoamérica.(AU)

Parsonage-Tuner syndrome is a rare neurological entity of unknown etiology, associated with viral infections, trauma, and vaccinal history. In the context of the COVID-19 pandemic, cases of this neurological entity have been reported as an infectious complication, with the clinical development of pain, weakness, and typical electrophysiological studies. There is a need to consider Parsonage-Turner syndrome in the differential diagnosis of monoplegia in patients with COVID-19 infection and to promote clinical management strategies. Less than 7 cases have been described in the literature, this being the first in Latin America.(AU)