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)

Bilateral Parsonage-Turner syndrome after COVID-19 vaccination. A case report and review of the literature / Kétoldali Parsonage-Turner-szindróma COVID-19-vakcinációt követoen. Esetismertetés és az irodalom áttekintése

Parsonage---Turner syndrome (PTS; neuralgic amyotrophy) is a generally unilateral neuritis with sudden onset, severe shoulder or upper arm pain. Although the intense pain is usually self-limiting, two-thirds of patients experience pro-gressive motor weakness, narrowed range of motion, reflex changes, dysesthesias and chronic neuropathic pain in the shoulder girdle musculature and proximal upper limb muscles. The aetiology is unclear, in addition to some idiopath-ic cases the most common triggers of PTS are surgery, trauma, infection or vaccination. It is reported after SARS-CoV-2 infection, and unilateral PTS has been described in some cases following different types of COVID-19 vaccines. We are currently presenting the case of a middle-aged woman who developed partial neuralgic amyotrophy on the right shoulder one month after receiving the second dose of the BNT162b2 COVID-19 mRNA vaccine (Pfizer-BioNTech), and seven months later the symptoms appeared in the contralateral upper limb. The diagnosis of PTS was also confirmed by magnetic resonance and electrodiagnostic examination. The PTS is not an uncommon condi-tion, but in the absence of knowledge it is rarely thought of. The purpose of this report is to draw attention to the possibility of PTS in shoulder or upper arm pain following both SARS-CoV-2 infection and COVID-19 vaccination, as early diagnosis and adequate therapy may help to shorten the course of the disease.

Pearls & Oy-sters: Bilateral Mononeuropathic Neuralgic Amyotrophy Triggered by Bartonella henselae Infection Responsive to Immunoglobulin.

We present a case of a cat owner with a scar on his right thenar eminence, followed by lymphadenopathy in the right axilla, general malaise and fever, and subsequent onset of bilateral neuralgic amyotrophy within one week. After a comprehensive workup, cat scratch disease caused by Bartonella henselae was confirmed serologically and adequately treated. Despite antibiotic treatment, the patient presented clinically with persistent bilateral, asymmetric neuropathy of the median nerve, predominantly the interosseous anterior nerve, which was confirmed by multifocal swelling and hyperintense signal of the nerves on T2-weighted MR neurography. Electrophysiological examination confirmed axonal median neuropathies bilaterally. After an unsuccessful steroid treatment trial, the patient showed an excellent and sustained response to intravenous immunoglobulin despite a delay from symptom onset to treatment of 10 months.

Parsonage-Turner Syndrome Following COVID-19 Vaccination: MR Neurography.

Vaccination is one of the several known triggers of Parsonage-Turner syndrome (PTS). This case series describes two individuals with clinical presentations of PTS whose symptoms began 13 hours and 18 days following receipt of the Pfizer-BioNTech BNT162b2 and Moderna mRNA-1273 COVID-19 vaccine, respectively. The diagnosis of PTS was confirmed by using both electrodiagnostic testing and 3.0-T MR neurography. Although research is needed to understand the association between PTS and COVID-19 vaccination, MR neurography may be used to help confirm suspected cases of PTS as COVID-19 vaccines continue to be distributed worldwide.

Parsonage-Turner posterior a vacunación antirrábica por mordedura de murciélago: comunicación de un caso / Parsonage-Turner after rabies vaccination due to bat bite: case report

Introducción:Paciente adulto masculino que consulta por mordedura de murciélago. Se realiza aplicación de vacuna antirrábica y toxoide tetánico, tras lo cual presenta alteraciones motoras, sensitivas y dolor de difícil manejo en el miembro superior derecho. Siendo un reto diagnóstico de manera inicial, posterior a estudios electrofisiológicos se consideró un síndrome de Parsonage-Turner. Este caso es relevante por la asociación de vacuna antirrábica no descrita en antecedentes de inmunización en revisiones previas.Caso clínico:Se le realizaron varias pruebas diagnósticas e imágenes como ecografías, resonancia magnética y estudios electrofisiológicos los cuales confirmaron el diagnóstico de neuritis braquial (síndrome de Parsonage-Turner). El paciente recibió intervenciones quirúrgicas con fascitomías y neurólisis, además de múltiples manejos farmacológicos para dolor, incluyendo opioides fuertes, neuromoduladores, antidepresivos y medicamentos por bomba intratecal, con disminución solo del 50 % de los síntomas de dolor y afectación a su calidad de vida.Conclusión:El caso nos muestra cómo el síndrome de Parsonage-Turner, una enfermedad neurológica con etiología desconocida, se puede cronificar en un síndrome doloroso al no tener un diagnóstico temprano o someter al paciente procedimientos no indicados. Este síndrome debe considerarse en la atención de urgencias y consulta externa por sus consecuencias a largo plazo y el difícil manejo de los síntomas crónicos. Hay una relación causal reportada en la literatura con el toxoide tetánico, pero este se aplicó previo a los síntomas; no hay referencias de inicio de síntomas posterior a la vacuna antirrábica, lo cual puede generar a futuro, una relación causal si se encontraran nuevos casos.(AU)

Introduction:Adult male patient who suffers a bat bite, after which rabies vaccine and tetanus toxoid are administered; later, he presents on the right upper limb severe motor, sensory and pain disorders. Initially a diagnostic challenge, after electrophysiological studies, Parsonage-Turner syndrome was considered. This case is relevant since the association between rabies vaccine and this syndrome has not been described.Clinical case:Several diagnostic tests and images were performed, including ultrasound, magnetic resonance and electrophysiological studies, which confirmed the diagnosis of brachial neuritis (Parsonage-Turner syndrome). Even though the patient received surgical interventions with fasciotomies and neurolysis as well as multiple pharmacological pain management with strong opioids, neuromodulators, antidepressants and intrathecal pump medications, there was a 50 % decrease in pain symptoms and an impairment of their quality of life.Conclusion:The case shows a Parsonage-Turner syndrome, a neurological disease with unknown etiology, with difficult diagnosis which can lead to chronic pain syndrome or unnecessary surgical procedures. This syndrome should be considered in emergency care and outpatient care due to its long-term consequences and the difficult management of chronic symptoms. There is a causal relationship reported in the literature with tetanus toxoid, but it was applied prior to symptoms; there are no reports of onset of symptoms after the rabies vaccine, which may generate a causal relationship in the future if new cases are found.(AU)

Forma de presentación "prono-doloroso-like" del síndrome de Parsonage-Turner / Parsonage-Turne síndrome "pulled elbow-like" presentation form

Introducción: El síndrome de Parsonage-Turner es una neuritis del plexo braquial de etiología desconocida. Se ha des-crito su aparición tras procesos infecciosos, por lo que se sospecha un mecanismo autoinmune. Su descripción en la edad pediátrica se limita a casos aislados. En los adultos, la sintomatología típica es un dolor súbito e intenso, localizado en el hombro y la región proximal de la extremidad superior, seguido de parálisis flácida y amiotrofia. Caso clínico: Lactante de 14 meses de edad que consulta por presentar una impotencia funcional de los miembros superiores de 48 horas de evolución. Los días previos mostró una infección respiratoria de las vías altas. Presentaba el brazo derecho en posición de prono, debilidad de la musculatura proximal de ambas extremidades superiores, sobre todo la derecha, e incapacidad para la movilización. Se detectó rhinovirus en la reacción en cadena de la polimerasa de moco nasal. El electromiogra-ma (EMG) del deltoides demostró una denervación aguda intensa de los músculos proximales de ambas extremidades superiores, sobre todo la derecha, compatible con una plexitis braquial bilateral. Conclusiones: El espectro clínico de presentación de la neuralgia amiotrófica puede ser distinto en la edad pediátrica. El dolor, característico de la forma adulta, puede no estar presente. Su forma de presentación puede asemejarse a un prono doloroso, por lo que deberemos incluirlo en su diagnóstico diferencial. El diagnóstico se basa en hallazgos clínicos; son de utilidad la resonancia magnética y el EMG, que debe realizarse 2-3 semanas tras el inicio del cuadro. El conocimiento de esta entidad permite establecer un diagnóstico precoz, lo que evita tratamientos potencialmente yatrogénicos, y anticipar el pronóstico

Introduction: Parsonage-Turner syndrome is a brachial plexus neuritis of unknow etiology. An autoinmune mecha-nism is suspected because of its appearance after infections. It predominantly affects proximal muscles of upper ex-tremities, with a first painful phase, which evolves to paresis and atrophy of the affected muscles. His description in pediatric patients is limited to isolated cases. Case report: 14 months infant consulting for weakness of upper limbs. He had an upper respiratory tract infection the previous days. Examination revealed weakness of the proximal muscles of the both arms, more marked on the right side and inability to active mobility simulating a pulled elbow. Magnetic resonance imaging of neck and shoulder showed no structural pathology. The electromyogram performed at 3 weeks of the onset of symptoms demonstrated acute denervation of the proximal arm muscles compatible with bilateral brachial plexitis. Rhinovirus was detected in polimerasa chain reaction in nasopharyngeal aspirate. Conclusions: Parsonage-Turner syndrome is an entity to consider in painful shoulder or upper limb functional impo-tence, as pulled elbow. Diagnosis is based on clinical findings. It's useful to rule one MRI and EMG (which must be per-formed 2-3 weeks after onset). It has spontaneous evolution partially favorable to rehabilitation and anti-inflammatory treatment. Knowledge of this entity allows proper handling, avoiding iatrogenic treatments, and allows anticipate the outcome

Extremely Painful Multifocal Acquired Predominant Axonal Sensorimotor Neuropathy of the Upper Limb.

The differential diagnosis of upper extremity mononeuritis multiplex includes neuralgic amyotrophy, vasculitic neuropathy, and Lewis-Sumner syndrome. We describe 3 patients initially suspected of neuralgic amyotrophy, who had an extremely painful, protracted, progressive disease course, not fitting one of these established diagnoses. Nerve ultrasonography showed focal caliber changes of the roots, plexus, and limb nerves. Electromyography showed predominant multifocal axonopathy. Ongoing autoimmune neuropathy was suspected. Steroid treatment provided temporary relief, and intravenous immunoglobulin A sustained pain decrease and functional improvement. These patients appear to have extremely painful axonal inflammatory neuropathy, with a good response to immune-modulating treatment.

Clinical phenotype and outcome of hepatitis E virus-associated neuralgic amyotrophy.

OBJECTIVE: To determine the clinical phenotype and outcome in hepatitis E virus-associated neuralgic amyotrophy (HEV-NA). METHODS: Cases of NA were identified in 11 centers from 7 European countries, with retrospective analysis of demographics, clinical/laboratory findings, and treatment and outcome. Cases of HEV-NA were compared with NA cases without evidence of HEV infection. RESULTS: Fifty-seven cases of HEV-NA and 61 NA cases without HEV were studied. Fifty-six of 57 HEV-NA cases were anti-HEV IgM positive; 53/57 were IgG positive. In 38 cases, HEV RNA was recovered from the serum and in 1 from the CSF (all genotype 3). Fifty-one of 57 HEV-NA cases were anicteric; median alanine aminotransferase 259 IU/L (range 12-2,961 IU/L); in 6 cases, liver function tests were normal. HEV-NA cases were more likely to have bilateral involvement (80.0% vs 8.6%, p < 0.001), damage outside the brachial plexus (58.5% vs 10.5%, p < 0.01), including phrenic nerve and lumbosacral plexus injury (25.0% vs 3.5%, p = 0.01, and 26.4% vs 7.0%, p = 0.001), reduced reflexes (p = 0.03), sensory symptoms (p = 0.04) with more extensive damage to the brachial plexus. There was no difference in outcome between the 2 groups at 12 months. CONCLUSIONS: Patients with HEV-NA are usually anicteric and have a distinct clinical phenotype, with predominately bilateral asymmetrical involvement of, and more extensive damage to, the brachial plexus. Involvement outside the brachial plexus is more common in HEV-NA. The relationship between HEV and NA is likely to be causal, but is easily overlooked. Patients presenting with NA should be tested for HEV, irrespective of liver function test results. Prospective treatment/outcome studies of HEV-NA are warranted.

Neuralgic Amyotrophy Attributed Incorrectly to Block-Related Injury: Understanding Errors in Clinical Reasoning.

OBJECTIVE: We report a case of misdiagnosed neuralgic amyotrophy (brachial plexus neuritis, Parsonage-Turner syndrome). Our primary objective is to review the scientific basis for errors in clinical reasoning. CASE REPORT: We herein report a patient in whom signs and symptoms compatible with neuralgic amyotrophy presented after shoulder surgery. The patient's brachial plexopathy was attributed incorrectly as a complication of interscalene brachial plexus block. The true diagnosis was made only after the patient developed neuralgic amyotrophy in the contralateral upper extremity after a subsequent shoulder surgery on that side, this time without a brachial plexus block. CONCLUSIONS: Cognitive bias may lead to errors in clinical reasoning and consequent misdiagnosis. Temporal proximity may falsely implicate regional anesthesia as the causative agent.

Síndrome de Parsonage-Turner: a propósito de un caso / Parsonage-turner syndrome: about a case

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Neuritis braquial o síndrome de Parsonage-Turner: un problema de responsabilidad. A propósito de 3 casos / Brachial neuritis or Parsonage-Turner syndrome: a problem of liability. A presentation of 3 cases

La amiotrofia neurálgica, neuritis braquial o síndrome de Parsonage-Turner es una afectación neuromuscular rara de etiología desconocida. Cuando se inicia en relación con un acto asistencial, como puede ser un parto o una cirugía, es frecuente que se arguya mala praxis como causa del adverso resultado, normalmente una incorrecta posición del paciente en la mesa de quirófano, circunstancia en la que se implica directamente el área de anestesia. Presentamos 3 casos de síndrome de Parsonage-Turner a raíz de intervenciones quirúrgicas bien distintas, con resultados dispares en lo referente al pronóstico. Se revisa y comenta la bibliografía ante la posibilidad de que este tipo de circunstancias sean objeto de reclamación de responsabilidad sanitaria por mala praxis, con especial hincapié en las teorías etiopatogénicas más aceptadas actualmente y en la relación de este síndrome con la cirugía como aspecto médico legal determinante. Se proponen parámetros de valoración (AU)

Neuralgic amyotrophy, brachial neuritis or Parsonage-Turner syndrome is a rare neuromuscular involvement of unknown aetiology. When it onsets in connection with a health care act, such as childbirth or surgery, a malpractice argument is often used as a cause of adverse outcome, usually due to an incorrect position of the patient on the operating table, a circumstance which directly involves the anesthesia area. Three cases are presented of Parsonage-Turner syndrome following very different surgery, with different results as regards prognosis. A review and discussion of bibliography is presented on the possibility that such circumstances are the subject of malpractice claims. Special emphasis is placed on the most currently accepted aetiopathogenic theories, and the relationship of this syndrome with the surgical act as a determining medico-legal aspect. Valuation parameters are proposed (AU)

Infección por virus de la hepatitis E como factor desencadenante del síndrome de Parsonage-Turner / Infection by the hepatitis E virus as a precipitating factor of Parsonage-Turner syndrome

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Treatment of chronic cervicobrachial pain with periradicular injection of meloxicam.

BACKGROUND: Cervicobrachial pain (CBP) is often resistant to conventional oral analgesics. We hypothesized that the periradicular injection of meloxicam would produce a significant reduction in their intractable CBP. The secondary objective was to assess the impact of the treatment on functional recovery. METHODS: 48 patients with persistent CBP (>3 months of duration) despite multimodal analgesic therapy received 1-3 periradicular injections of meloxicam, 5-20 mg, at the dermatomal level(s) corresponding to their pain symptoms. Pain level (0=none to 10=severe), rescue analgesics, and functional activity were recorded at baseline and for 90d after the last injection. The injection was repeated if the pain score remained >3 or paresthesia persisted. RESULTS: The mean pain score was reduced from a baseline of 8.9 (±1) to 1.7 (±2.2) at 90 days after the last meloxicam injection. Following meloxicam treatment(s), only 13% of the patients required oral analgesic rescue medication. All patients increased their functional activity level. CONCLUSIONS: Cervical periradicular injection of meloxicam reduced CBP by 81% at 90-day follow-up and also improved functional recovery.

Neuralgia amiotrófica asociada a parálisis frénica bilateral tratada con ventilación mecánica no invasiva / Amyotrophic neuralgia associated with bilateral phrenic paralysis treated with non-invasive mechanical ventilation

La neuralgia amiotrófica es una neuropatía infrecuente caracterizada por la aparición de un dolor intenso unilateral en el hombro. Es rara la afectación aislada o concomitante de otros nervios motores periféricos derivados del plexo cervicobraquial, como el nervio frénico o el laríngeo. Su etiología es desconocida, aunque se han planteado varios factores que pueden condicionar su aparición. La afectación del nervio frénico, unilateral o bilateral, es aún más infrecuente. El diagnóstico se basa en la historia clínica, la exploración funcional, técnicas de imagen y en los hallazgos del electromiograma. Presentamos el caso de una mujer de 48 años con antecedentes de trasplante renal por glomerulonefritis proliferativa y rechazo posterior del mismo, que fue definitivamente diagnosticada de neuralgia amiotrófica con afectación frénica bilateral, que precisó ventilación mecánica no invasiva de forma prolongada (AU)

Amyotrophic neuralgia is an uncommon neuropathy characterized by severe unilateral shoulder pain. Isolated or concomitant involvement of other peripheral motor nerves depending on the brachial plexus such as phrenic or laryngeal nerves is unusual. Its etiology is unknown, yet several explanatory factors have been proposed. Phrenic nerve involvement, either unilateral or bilateral, is exceedingly rare. Diagnosis relies on anamnesis, functional and imaging investigations and electromyogram. We report the case of a 48-year-old woman with a past history of renal transplantation due to proliferative glomerulonephritis with subsequent transplant rejection, who was eventually diagnosed with amyotrophic neuralgia with bilateral phrenic involvement, and who required sustained non-invasive mechanical ventilation (AU)

Neuralgic amyotrophy complicating acute hepatitis E infection: a rare association.

Hepatitis E virus infection (HEV) is an emerging pathogen that is under-recognised in developed countries. Preceding infection manifested by acute transaminitis has been associated with neurological manifestations, predominately involving the peripheral nervous system, even in immunocompetent patients. We present a case of a 65-year-old previously fit and well Caucasian man with bilateral neuralgic amyotrophy (NA) and acute transaminitis. Serology testing for immunoglobulin (Ig) M and G established the diagnosis of acute HEV infection. The patient received immunomodulatory treatment with an excellent long-term outcome. The temporal association of the clinical presentation of bilateral NA and acute transaminitis from HEV infection suggested the causal association of HEV to NA. We propose screening for HEV in patients presenting with NA and acute hepatitis.

Cervical foraminal steroid injections under CT guidance: retrospective study of in situ contrast aspects in a serial of 248 cases.

OBJECTIVES: To describe all the CT findings after in situ contrast injection just before steroid injection and to recognize the abnormal aspects associated with intravascular contamination. MATERIAL AND METHODS: We retrospectively evaluated 248 cervical transforaminal steroid injections done at the university hospital in Dijon, France, in 2008-2012, to treat cervicobrachial neuralgia inadequately improved by optimal medical treatment for at least 3 weeks. Features describing the opacification patterns were recorded. RESULTS: Five main nonvascular opacification patterns were identified: clumps of contrast agent outside the foramen (16 %), a crab claw pattern surrounding the ganglion (13 %), a "French" circumflex accent pattern (15 %), reflux along the needle (7 %), and facet joint capsule opacification (22 %). Concerning the situations requiring a change in needle position, intravenous injection occurred in 26 % of the patients, with a crab claw pattern in half the cases and a clump pattern in half the cases. Intraarteriolar injection was noted in two patients. CONCLUSION: CT after in situ contrast injection ensures proper needle positioning outside the blood vessels before steroid injection. Penetration of the needle tip into a vein is very common, whereas arteriolar puncture is extremely rare.

[Neuralgic amyotrophy--a case report]. / Neuralgia amiotroficzna--opis przypadku.

We describe a case of a 54-year-old woman reporting weakness of the right upper limb, preceded by a period of severe pain in the right shoulder. Despite several orthopedic consultations and two cycles of physical therapy, the symptoms deteriorated over a period of 6 months, and progressive paresis of the extensor muscles of the upper limb and elbow flexion contracture were observed. On the basis of the clinical presentation and results of medical tests including an electrophysiological study that showed axonal damage to the long thoracic nerve, axillary nerve, and posterior interosseous nerve, neuralgic amyotrophy was diagnosed. A treatment with prednisone was introduced, and the patient's condition significantly improved.