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Arnolfo P, Péréon Y, Berthelot JM. Neuralgic amyotrophy of Parsonage and Turner in daily practice: More often mononeuropathic than previously assumed. Joint Bone Spine 2025; 92:105846. [PMID: 39892588 DOI: 10.1016/j.jbspin.2025.105846] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Academic Contribution Register] [Received: 11/12/2024] [Revised: 12/15/2024] [Accepted: 01/14/2025] [Indexed: 02/04/2025]
Affiliation(s)
- Paul Arnolfo
- Rheumatology Unit, Nantes University Hospital, Place Alexis Ricordeau, 44093 Nantes, France
| | - Yann Péréon
- Centre de Référence Maladies Neuromusculaires Rares AOC, Filnemus, Euro-NMD, Hôtel-Dieu, CHU de Nantes, Place Alexis Ricordeau, 44093 Nantes, France
| | - Jean-Marie Berthelot
- Rheumatology Unit, Nantes University Hospital, Place Alexis Ricordeau, 44093 Nantes, France.
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Seror P. Neuralgic amyotrophy of Parsonage and Turner. Which nerves are most frequently involved in daily practice? Data from 355 patients. Joint Bone Spine 2025; 92:105777. [PMID: 39303939 DOI: 10.1016/j.jbspin.2024.105777] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Academic Contribution Register] [Received: 03/11/2024] [Revised: 08/26/2024] [Accepted: 09/04/2024] [Indexed: 09/22/2024]
Abstract
OBJECTIVES The aim of this study was to conduct a retrospective analysis of clinical patterns associated with neuralgic amyotrophy of Parsonage and Turner (NAPT) in a series of 355 patients seen in "ambulatory care". METHODS Clinical, epidemiological and electrodiagnostic (EDX) data were collected by means of an electrodiagnosis consultation. Data were obtained on age, sex, body mass index, side involved, nerves involved, number of nerves involved per attack, number of attacks per patient, number of bilateral, recurrent, and hereditary cases. RESULTS Mean age was 42.7 years, 218 patients (61%) were male, mean body mass index was 23.8; the right side was involved in 232 cases (65%). Inflammatory mononeuropathy (MN) was the most frequent elementary nerve lesion. It was unique (UMN) in 253 patients (71%), and multiple (MMN) in 102 (29%), with a total of 495 neuropathies, and a mean of 2.38 nerves involved in one patient with multiple mononeuropathy (MMN). Five nerves are more frequently involved: long thoracic (n=138), suprascapular (n=129), anterior interosseous (n=53), spinal accessory (SAN) (n=51), lateral antebrachial cutaneous (n=41) nerves. Other nerves (axillary, posterior interosseous, sensory median, phrenic, ulnar and medial antebrachial cutaneous nerves, and lumbo-sacral plexus) are less frequently involved, and less suggestive for NAPT diagnosis. Bilateral, recurrent and hereditary cases were observed in 22, 10 and 0 cases, respectively. CONCLUSIONS The clinical patterns of NAPT in ambulatory care thorough an EDX consultation have showed that unique mononeuropathy (UMN) are more frequent than MMN, what results in milder cases; and that five nerves lesions are more frequent and more suggestive of NAPT.
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Affiliation(s)
- Paul Seror
- Laboratoire d'électroneuromyographie, 146, avenue Ledru-Rollin, 75011 Paris, France; Laboratoire d'électroneuromyographie, hôpital de l'Est Parisien, Ramsay GS, 93600 Aulnay-sous-Bois, France.
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Drakou A, Altsitzioglou P, Roustemis AG, Vourda E, Papakonstantinou ME, Sioutis S, Koulalis D. Parsonage-Turner Syndrome and SARS-CoV-2 Infection: A Literature Review With Case Presentation. Cureus 2024; 16:e63305. [PMID: 39070412 PMCID: PMC11283305 DOI: 10.7759/cureus.63305] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Academic Contribution Register] [Accepted: 06/27/2024] [Indexed: 07/30/2024] Open
Abstract
Neuralgic amyotrophy, also known as Parsonage-Turner syndrome (PTS), is characterized by severe pain and muscle wasting affecting the anterior body, including the head, shoulder, upper limb, and chest wall. Often triggered by an antecedent event, such as infection, PTS encompasses various conditions historically identified as separate entities. In 1948, Parsonage and Turner unified these conditions under the term neuralgic amyotrophy based on shared features of intense pain and muscular atrophy. Recent studies have highlighted PTS as a spectrum disorder with diverse manifestations, including pure sensory neuropathy, extensive neuropathy, spinal accessory nerve involvement, and diaphragmatic palsy. We reviewed 26 documented cases of PTS following SARS-CoV-2 infection, emphasizing the importance of considering PTS in individuals with a history of COVID-19 due to varied clinical presentations. Standardized diagnostic methods and comprehensive evaluations are crucial for accurate diagnosis and management. Future research should focus on consistent evaluation methods and employing a comprehensive differential diagnosis approach.
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Affiliation(s)
| | - Pavlos Altsitzioglou
- 1st Department of Orthopaedic Surgery, National and Kapodistrian University of Athens, Attikon Hospital, Athens, GRC
| | - Anastasios G Roustemis
- 1st Department of Orthopaedic Surgery, National and Kapodistrian University of Athens, Attikon Hospital, Athens, GRC
| | - Eleni Vourda
- Department of Allergy and Immunology, National and Kapodistrian University of Athens, Attikon Hospital, Athens, GRC
| | | | - Spyridon Sioutis
- 1st Department of Orthopaedic Surgery, National and Kapodistrian University of Athens, Attikon Hospital, Athens, GRC
| | - Dimitrios Koulalis
- 1st Department of Orthopaedic Surgery, National and Kapodistrian University of Athens, Attikon Hospital, Athens, GRC
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Granata G, Tomasello F, Sciarrone MA, Stifano V, Lauretti L, Luigetti M. Neuralgic Amyotrophy and Hourglass Nerve Constriction/Nerve Torsion: Two Sides of the Same Coin? A Clinical Review. Brain Sci 2024; 14:67. [PMID: 38248282 PMCID: PMC10813384 DOI: 10.3390/brainsci14010067] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Academic Contribution Register] [Received: 11/29/2023] [Revised: 12/29/2023] [Accepted: 01/09/2024] [Indexed: 01/23/2024] Open
Abstract
Neuralgic amyotrophy, also called Parsonage-Turner syndrome, in its classic presentation is a brachial plexopathy or a multifocal neuropathy, involving mainly motor nerves of the upper limb with a monophasic course. Recently, a new radiological entity was described, the hourglass constriction, which is characterized by a very focal constriction of a nerve, or part of it, usually associated with nerve thickening proximally and distally to the constriction. Another condition, which is similar from a radiological point of view to hourglass constriction, is nerve torsion. The pathophysiology of neuralgic amyotrophy, hourglass constriction and nerve torsion is still poorly understood, and a generic role of inflammation is proposed for all these conditions. It is now widely accepted that nerve imaging is necessary in identifying hourglass constrictions/nerve torsion pre-surgically in patients with an acute mononeuropathy/plexopathy. Ultrasound and MRI are useful tools for diagnosis, and they are consistent with intraoperative findings. The prognosis is generally favorable after surgery, with a high rate of good motor recovery.
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Affiliation(s)
- Giuseppe Granata
- Dipartimento di Neuroscienze, Organi di Senso e Torace, Fondazione Policlinico Universitario Agostino Gemelli IRCCS, 00168 Rome, Italy; (G.G.); (V.S.); (L.L.)
- Dipartimento di Neuroscienze, Università Cattolica del Sacro Cuore, 00168 Rome, Italy; (F.T.); (M.A.S.)
| | - Fabiola Tomasello
- Dipartimento di Neuroscienze, Università Cattolica del Sacro Cuore, 00168 Rome, Italy; (F.T.); (M.A.S.)
| | - Maria Ausilia Sciarrone
- Dipartimento di Neuroscienze, Università Cattolica del Sacro Cuore, 00168 Rome, Italy; (F.T.); (M.A.S.)
| | - Vito Stifano
- Dipartimento di Neuroscienze, Organi di Senso e Torace, Fondazione Policlinico Universitario Agostino Gemelli IRCCS, 00168 Rome, Italy; (G.G.); (V.S.); (L.L.)
| | - Liverana Lauretti
- Dipartimento di Neuroscienze, Organi di Senso e Torace, Fondazione Policlinico Universitario Agostino Gemelli IRCCS, 00168 Rome, Italy; (G.G.); (V.S.); (L.L.)
- Dipartimento di Neuroscienze, Università Cattolica del Sacro Cuore, 00168 Rome, Italy; (F.T.); (M.A.S.)
| | - Marco Luigetti
- Dipartimento di Neuroscienze, Organi di Senso e Torace, Fondazione Policlinico Universitario Agostino Gemelli IRCCS, 00168 Rome, Italy; (G.G.); (V.S.); (L.L.)
- Dipartimento di Neuroscienze, Università Cattolica del Sacro Cuore, 00168 Rome, Italy; (F.T.); (M.A.S.)
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5
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Dalati Y, Ashraf U. Parsonage-Turner Syndrome With an Uncommon Trigger: A Case Study and Literature Review. Cureus 2023; 15:e49589. [PMID: 38156184 PMCID: PMC10754625 DOI: 10.7759/cureus.49589] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Academic Contribution Register] [Accepted: 11/21/2023] [Indexed: 12/30/2023] Open
Abstract
Patients with Parsonage-Turner syndrome (PTS) are often misdiagnosed due to overlapping symptoms with other conditions and coinciding procedures. Because it is most commonly seen following viral infection, it is often not considered in other cases. We present a rare case in which a 79-year-old female, with no significant past medical history, was diagnosed with PTS two months after a biopsy of the right levator scapulae muscle. Forty-eight hours after the procedure, she developed sudden-onset pain and weakness in the right scapulae and neck, followed by worsened weakness. This case report highlights the importance of considering PTS before proceeding with treatment. Patients with suspected PTS should undergo electromyography (EMG) to confirm diagnosis and monitor disease progression and resolution.
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Affiliation(s)
- Yaman Dalati
- Department of Medicine, College of Osteopathic Medicine, Michigan State University, East Lansing, USA
| | - Usman Ashraf
- Department of Physical Medicine and Rehabilitation, Trinity Health, Ypsilanti, USA
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Rusin G, Morga R, Spaczyńska-Boczar M, Rudnicki W, Kwinta BM, Luczynska E, Słowik A, Antczak J. Neuralgic amyotrophy with bilateral radial nerve torsion: A unique case and review of the literature. Neurologia 2023; 38:707-710. [PMID: 37996215 DOI: 10.1016/j.nrleng.2022.11.002] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Academic Contribution Register] [Received: 04/22/2022] [Accepted: 11/29/2022] [Indexed: 11/25/2023] Open
Affiliation(s)
- G Rusin
- Department of Neurology, University Hospital in Krakow, Jakubowskiego 2, Krakow, Poland.
| | - R Morga
- Department of Neurosurgery, University Hospital in Krakow, Jakubowskiego 2, Krakow, Poland
| | - M Spaczyńska-Boczar
- Department of Neurology, University Hospital in Krakow, Jakubowskiego 2, Krakow, Poland
| | - W Rudnicki
- Electroradiology Department, Jagiellonian University Medical College, Jakubowskiego 2, Krakow, Poland
| | - B M Kwinta
- Department of Neurosurgery and Neurotraumatology, Jagiellonian University Medical College, Jakubowskiego 2, Krakow, Poland
| | - E Luczynska
- Electroradiology Department, Jagiellonian University Medical College, Jakubowskiego 2, Krakow, Poland
| | - A Słowik
- Department of Neurology, University Hospital in Krakow, Jakubowskiego 2, Krakow, Poland; Department of Neurology, Jagiellonian University Medical College, Jakubowskiego 2, Krakow, Poland
| | - J Antczak
- Department of Neurology, University Hospital in Krakow, Jakubowskiego 2, Krakow, Poland; Department of Neurology, Jagiellonian University Medical College, Jakubowskiego 2, Krakow, Poland
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Maślanka K, Zielinska N, Karauda P, Balcerzak A, Georgiev G, Borowski A, Drobniewski M, Olewnik Ł. Congenital, Acquired, and Trauma-Related Risk Factors for Thoracic Outlet Syndrome-Review of the Literature. J Clin Med 2023; 12:6811. [PMID: 37959276 PMCID: PMC10648912 DOI: 10.3390/jcm12216811] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Academic Contribution Register] [Received: 08/21/2023] [Revised: 10/17/2023] [Accepted: 10/19/2023] [Indexed: 11/15/2023] Open
Abstract
Thoracic outlet syndrome is a group of disorders that affect the upper extremity and neck, resulting in compression of the neurovascular bundle that exits the thoracic outlet. Depending on the type of compressed structure, the arterial, venous, and neurogenic forms of TOS are distinguished. In some populations, e.g., in certain groups of athletes, some sources report incidence rates as high as about 80 cases per 1000 people, while in the general population, it is equal to 2-4 per 1000. Although the pathogenesis of this condition appears relatively simple, there are a very large number of overlapping risk factors that drive such a high incidence in certain risk groups. Undoubtedly, a thorough knowledge of them and their etiology is essential to estimate the risk of TOS or make a quick and accurate diagnosis.
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Affiliation(s)
- Krystian Maślanka
- Department of Anatomical Dissection and Donation, Medical University of Lodz, 90-419 Lodz, Poland; (K.M.); (N.Z.); (P.K.); (A.B.)
| | - Nicol Zielinska
- Department of Anatomical Dissection and Donation, Medical University of Lodz, 90-419 Lodz, Poland; (K.M.); (N.Z.); (P.K.); (A.B.)
| | - Piotr Karauda
- Department of Anatomical Dissection and Donation, Medical University of Lodz, 90-419 Lodz, Poland; (K.M.); (N.Z.); (P.K.); (A.B.)
| | - Adrian Balcerzak
- Department of Anatomical Dissection and Donation, Medical University of Lodz, 90-419 Lodz, Poland; (K.M.); (N.Z.); (P.K.); (A.B.)
| | - Georgi Georgiev
- Department of Orthopaedics and Traumatology, University Hospital Queen Giovanna—ISUL, Medical University of Sofia, 1527 Sofia, Bulgaria;
| | - Andrzej Borowski
- Orthopaedics and Paediatric Orthopaedics Department, Medical University of Lodz, 90-419 Lodz, Poland; (A.B.); (M.D.)
| | - Marek Drobniewski
- Orthopaedics and Paediatric Orthopaedics Department, Medical University of Lodz, 90-419 Lodz, Poland; (A.B.); (M.D.)
| | - Łukasz Olewnik
- Department of Anatomical Dissection and Donation, Medical University of Lodz, 90-419 Lodz, Poland; (K.M.); (N.Z.); (P.K.); (A.B.)
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Yu M, Ding W, Shao G, Li M, Zhou X, Liu L, Li X. Application of a nerve stereoscopic reconstruction technique based on ultrasonic images in the diagnosis of neuralgic amyotrophy. Front Physiol 2023; 14:1201275. [PMID: 37791346 PMCID: PMC10543234 DOI: 10.3389/fphys.2023.1201275] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Academic Contribution Register] [Received: 04/06/2023] [Accepted: 08/30/2023] [Indexed: 10/05/2023] Open
Abstract
Objective: To propose a nerve stereoscopic reconstruction technique based on ultrasound imaging for site diagnosis, intuitive reflection of disease severity, and classification of neuralgic amyotrophy (NA). Methods: We enrolled 44 patients with NA who underwent high-frequency ultrasonography examination. Multiple sites on the normal side and the affected side were scanned to calculate the ratio of the cross-section area (CSA) of the affected side to the normal side at each location measured, i.e., the cross-section area swelling ratio (CSASR). The CSASR of 44 patients and 30 normal controls was analyzed to determine their threshold value for the diagnosis of NA. Then, ultrasound images of the cross-section were used to reconstruct the stereoscopic model of the nerve on the affected side and the normal side. Using the CSASR values in each measurement location, a CSASR stereoscopic model was developed. Results: The threshold value of CSASR for ultrasound diagnosis of NA was 1.55. The average diseased segments per patient was 2.49 ± 1.97, with an average overall length of 10.03 ± 7.95 cm. Nerve stereoscopic reconstruction could be conducted for swelling, torsion, incomplete constriction, and complete constriction. Conclusion: The ultrasound image reconstruction method proposed in this study can accurately determine the site, range, and type of neuropathies in patients with NA, and simultaneously provide complete and accurate data information and intuitive morphological information.
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Affiliation(s)
- Miao Yu
- Department of Hand Surgery, Ningbo No. 6 Hospital, Ningbo, China
- Department of Plastic Reconstructive Surgery, Ningbo No. 6 Hospital, Ningbo, China
| | - Wenquan Ding
- Department of Hand Surgery, Ningbo No. 6 Hospital, Ningbo, China
- Department of Plastic Reconstructive Surgery, Ningbo No. 6 Hospital, Ningbo, China
| | - Guoqing Shao
- Department of Ultrasonic Medicine, Ningbo No. 6 Hospital, Ningbo, China
| | - Miaozhong Li
- Department of Hand Surgery, Ningbo No. 6 Hospital, Ningbo, China
- Department of Plastic Reconstructive Surgery, Ningbo No. 6 Hospital, Ningbo, China
| | - Xiaoling Zhou
- Department of Hand Surgery, Ningbo No. 6 Hospital, Ningbo, China
- Department of Plastic Reconstructive Surgery, Ningbo No. 6 Hospital, Ningbo, China
| | - Linhai Liu
- Department of Hand Surgery, Ningbo No. 6 Hospital, Ningbo, China
- Department of Plastic Reconstructive Surgery, Ningbo No. 6 Hospital, Ningbo, China
| | - Xueyuan Li
- Department of Hand Surgery, Ningbo No. 6 Hospital, Ningbo, China
- Department of Plastic Reconstructive Surgery, Ningbo No. 6 Hospital, Ningbo, China
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He R, Yu JL, Jin HL, Ng L, Wang JC, Li X, Gai TT, Zhou Y, Li DP. Hourglass-like constriction of the anterior interosseous nerve in the left forearm: A case report. World J Clin Cases 2023; 11:4194-4201. [PMID: 37388800 PMCID: PMC10303608 DOI: 10.12998/wjcc.v11.i17.4194] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Academic Contribution Register] [Received: 04/18/2023] [Revised: 05/01/2023] [Accepted: 05/19/2023] [Indexed: 06/12/2023] Open
Abstract
BACKGROUND Hourglass-like constriction neuropathy is a rare neurological disorder. The main clinical manifestation is peripheral nerve injury with no apparent cause, and the pathomorphological change is an unexplained narrowing of the diseased nerve. The diagnosis and treatment of the disease are challenging and there is no accepted diagnostic or therapeutic approach.
CASE SUMMARY This report describes a rare hourglass constriction of the anterior interosseous nerve in the left forearm in a 47-year-old healthy male who was treated surgically and gradually recovered function over a 6-mo follow-up period.
CONCLUSION Hourglass-like constriction neuropathy is a rare disorder. With the development of medical technology, more examinations are now available for diagnosis. This case aims to highlight the rare manifestations of Hourglass-like constriction neuropathy and provides a reference for enriching the clinical diagnosis and treatment experience.
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Affiliation(s)
- Rong He
- Bone and Hand Microsurgery, Yantai Hospital of Shandong Wendeng Osteopathic & Traumatology, Yantai 264009, Shandong Province, China
| | - Jian-Long Yu
- Bone and Hand Microsurgery, Yantai Hospital of Shandong Wendeng Osteopathic & Traumatology, Yantai 264009, Shandong Province, China
| | - Hai-Long Jin
- Bone and Hand Microsurgery, Shandong Wendeng Osteopathic Hospital, Weihai 264400, Shandong Province, China
| | - Liqi Ng
- Institute of Orthopaedic and Musculoskeletal Science, University College London, London HA7 4LP, United Kingdom
| | - Jin-Chao Wang
- Bone and Hand Microsurgery, Yantai Hospital of Shandong Wendeng Osteopathic & Traumatology, Yantai 264009, Shandong Province, China
| | - Xin Li
- Foot and Ankle Surgery, Chongqing Orthopedic Hospital of Traditional Chinese Medicine, Chongqing 400012, China
| | - Ting-Ting Gai
- Bone and Hand Microsurgery, Yantai Hospital of Shandong Wendeng Osteopathic & Traumatology, Yantai 264009, Shandong Province, China
| | - Yu Zhou
- Foot and Ankle Surgery, Chongqing Orthopedic Hospital of Traditional Chinese Medicine, Chongqing 400012, China
| | - Da-Peng Li
- Bone and Hand Microsurgery, Yantai Hospital of Shandong Wendeng Osteopathic & Traumatology, Yantai 264009, Shandong Province, China
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Parsonage-Turner Syndrome Following SARS-CoV-2 Infection: A Systematic Review. Biomedicines 2023; 11:biomedicines11030837. [PMID: 36979815 PMCID: PMC10045449 DOI: 10.3390/biomedicines11030837] [Citation(s) in RCA: 3] [Impact Index Per Article: 1.5] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Academic Contribution Register] [Received: 02/21/2023] [Revised: 03/07/2023] [Accepted: 03/08/2023] [Indexed: 03/12/2023] Open
Abstract
Parsonage-Turner syndrome (PTS) is an inflammatory disorder of the brachial plexus. Hypothesized underlying causes focus on immune-mediated processes, as more than half of patients present some antecedent event or possible predisposing condition, such as infection, vaccination, exercise, or surgery. Recently, PTS was reported following the severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2) infection. We aimed to investigate data on PTS triggered by SARS-CoV-2 infection to provide an extensive perspective on this pathology and to reveal what other, more specific, research questions can be further addressed. In addition, we aimed to highlight research gaps requiring further attention. We systematically reviewed two databases (LitCOVID and the World Health Organization database on COVID-19) to January 2023. We found 26 cases of PTS in patients with previous SARS-CoV-2 infection. The clinical and paraclinical spectrum was heterogeneous, ranging from classical PTS to pure sensory neuropathy, extended neuropathy, spinal accessory nerve involvement, and diaphragmatic palsy. Also, two familial cases were reported. Among them, 93.8% of patients had severe pain, 80.8% were reported to present a motor deficit, and 53.8% of patients presented muscle wasting. Paresthesia was noted in 46.2% of PTS individuals and a sensory loss was reported in 34.6% of patients. The present systematic review highlights the necessity of having a high index of suspicion of PTS in patients with previous SARS-CoV-2 infection, as the clinical manifestations can be variable. Also, there is a need for a standardized approach to investigation and reporting on PTS. Future studies should aim for a comprehensive assessment of patients. Factors including the baseline characteristics of the patients, evolution, and treatments should be consistently assessed across studies. In addition, a thorough differential diagnosis should be employed.
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11
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Zhang G, Yan F, He F, Liu D, Wang L. A Rare Case of Neuralgic Amyotrophy Associated with Brucella Infection. Infect Drug Resist 2023; 16:1145-1151. [PMID: 36861015 PMCID: PMC9969863 DOI: 10.2147/idr.s400228] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Academic Contribution Register] [Received: 12/04/2022] [Accepted: 01/27/2023] [Indexed: 02/25/2023] Open
Abstract
Multiple micro-organisms associated with Neuralgic Amyotrophy (NA) have been reported and Brucella species should be an important and overlooked infectious cause or trigger.We report a rare case of neuralgic amyotrophy associated with Brucella infection and is believed to be the first such case report in China. A 42-year-old male with brucellosis was confirmed serologically, who presented recurrent fever and fatigue and suddenly developed severe pain in the right shoulder within one week, followed by the inability to lift and abduct the proximal end of the right upper limb. Based on typical clinical manifestions, MRI neuroimaging of the brachial plexus and neuro-electrophysiological studies to confirm a diagnosis of NA and presented spontaneous recovery during this period, immunomodulatory treatment with corticosteroid or intravenous immunoglobulin had not been attempted, leaving a serious movement disorder in the right upper limb. Even rare, NA and other neurobrucellosis forms should be considered as complications of Brucella infection.
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Affiliation(s)
- Gaozan Zhang
- Department of Neurology, China-Japan Union Hospital of Jilin University, Changchun, 130031, People’s Republic of China
| | - Fenfen Yan
- Department of Neurology, China-Japan Union Hospital of Jilin University, Changchun, 130031, People’s Republic of China
| | - Fei He
- Department of Neurology, China-Japan Union Hospital of Jilin University, Changchun, 130031, People’s Republic of China
| | - Dingxi Liu
- Department of Clinical Medicine, Zunyi Medical University, Zhuhai, 519041, People’s Republic of China,Correspondence: Dingxi Liu; Libo Wang, Email ;
| | - Libo Wang
- Department of Neurology, China-Japan Union Hospital of Jilin University, Changchun, 130031, People’s Republic of China
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12
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Salomon M, Marruganti S, Cucinotta A, Lorusso M, Bortolotti P, Brindisino F. Parsonage-Turner Syndrome mimicking musculoskeletal shoulder pain: A case report during the SARS-CoV-2 pandemic era. J Telemed Telecare 2023; 29:133-146. [PMID: 35678699 PMCID: PMC9184833 DOI: 10.1177/1357633x221100059] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Academic Contribution Register] [Indexed: 02/02/2023]
Abstract
Parsonage-Turner Syndrome or neuralgic amyotrophy is a peripheral neuropathy typically characterized by an abrupt onset of pain, followed by progressive neurological deficits (e.g. weakness, atrophy, occasionally sensory abnormalities) that involve the upper limb, mainly the shoulder, encompassing an extensive spectrum of clinical manifestations, somehow difficult to recognize. This case report describes the proper management of a 35-year-old, bank employee and sports amateur who reported subtle and progressive upper limb disorder with previous history of neck pain. SARS-CoV-2 pandemic era made patient's access to the healthcare system more complicated. Nevertheless, proper management of knowledge, relevant aspects of telerehabilitation-based consultation for musculoskeletal pain, advanced skills, tools and technologies led the physiotherapist to suspect an atypical presentation of Parsonage-Turner Syndrome. Further, neurologist consultation and electromyography suggested signs of denervation in the serratus anterior and supraspinatus muscle. Therefore, an appropriate physiotherapist's screening for referral is conducted to correct diagnosis and thorough treatment.
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Affiliation(s)
- Mattia Salomon
- Department of Clinical Sciences and
Translational Medicine, University of Roma “Tor Vergata” c/o
Medicine and Surgery School, Rome, Italy,Mattia Salomon, Department of Clinical
Sciences and Translational Medicine, University of Roma “Tor Vergata” c/o
Medicine and Surgery School, Rome, 00133, Italy.
| | - Sharon Marruganti
- Department of Clinical Sciences and
Translational Medicine, University of Roma “Tor Vergata” c/o
Medicine and Surgery School, Rome, Italy
| | - Andrea Cucinotta
- Department of Clinical Sciences and
Translational Medicine, University of Roma “Tor Vergata” c/o
Medicine and Surgery School, Rome, Italy
| | - Mariangela Lorusso
- Department of Clinical Sciences and
Translational Medicine, University of Roma “Tor Vergata” c/o
Medicine and Surgery School, Rome, Italy
| | - Paolo Bortolotti
- Department of Medicine and Health
Science “Vincenzo Tiberio”, University of Molise C/da Tappino c/o
Cardarelli Hospital, Campobasso, Italy
| | - Fabrizio Brindisino
- Department of Medicine and Health
Science “Vincenzo Tiberio”, University of Molise C/da Tappino c/o
Cardarelli Hospital, Campobasso, Italy
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Saade F, Bouteille C, Quemener-Tanguy A, Obert L, Rochet S. Parsonage-Turner syndrome and SARS-CoV-2 infection: A case report. HAND SURGERY & REHABILITATION 2023; 42:90-92. [PMID: 36273762 PMCID: PMC9583683 DOI: 10.1016/j.hansur.2022.10.002] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Subscribe] [Academic Contribution Register] [Received: 06/28/2022] [Revised: 10/13/2022] [Accepted: 10/15/2022] [Indexed: 02/07/2023]
Affiliation(s)
- F Saade
- Orthopedics, Traumatology, Plastic & Reconstructive Surgery Unit, Hand Surgery Unit, University, Hospital J. Minjoz, Besançon, France; NanoMedecine Research Unit University of Franche-Comté, 19 Rue Ambroise Paré, 25030 Besançon Cedex, France.
| | - C Bouteille
- Orthopedics, Traumatology, Plastic & Reconstructive Surgery Unit, Hand Surgery Unit, University, Hospital J. Minjoz, Besançon, France; NanoMedecine Research Unit University of Franche-Comté, 19 Rue Ambroise Paré, 25030 Besançon Cedex, France
| | - A Quemener-Tanguy
- Orthopedics, Traumatology, Plastic & Reconstructive Surgery Unit, Hand Surgery Unit, University, Hospital J. Minjoz, Besançon, France; NanoMedecine Research Unit University of Franche-Comté, 19 Rue Ambroise Paré, 25030 Besançon Cedex, France
| | - L Obert
- Orthopedics, Traumatology, Plastic & Reconstructive Surgery Unit, Hand Surgery Unit, University, Hospital J. Minjoz, Besançon, France; NanoMedecine Research Unit University of Franche-Comté, 19 Rue Ambroise Paré, 25030 Besançon Cedex, France
| | - S Rochet
- Orthopedics, Traumatology, Plastic & Reconstructive Surgery Unit, Hand Surgery Unit, University, Hospital J. Minjoz, Besançon, France; NanoMedecine Research Unit University of Franche-Comté, 19 Rue Ambroise Paré, 25030 Besançon Cedex, France
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Macionis V. Chronic pain and local pain in usually painless conditions including neuroma may be due to compressive proximal neural lesion. FRONTIERS IN PAIN RESEARCH 2023; 4:1037376. [PMID: 36890855 PMCID: PMC9986610 DOI: 10.3389/fpain.2023.1037376] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Academic Contribution Register] [Received: 09/05/2022] [Accepted: 01/12/2023] [Indexed: 02/22/2023] Open
Abstract
It has been unexplained why chronic pain does not invariably accompany chronic pain-prone disorders. This question-driven, hypothesis-based article suggests that the reason may be varying occurrence of concomitant peripheral compressive proximal neural lesion (cPNL), e.g., radiculopathy and entrapment plexopathies. Transition of acute to chronic pain may involve development or aggravation of cPNL. Nociceptive hypersensitivity induced and/or maintained by cPNL may be responsible for all types of general chronic pain as well as for pain in isolated tissue conditions that are usually painless, e.g., neuroma, scar, and Dupuytren's fibromatosis. Compressive PNL induces focal neuroinflammation, which can maintain dorsal root ganglion neuron (DRGn) hyperexcitability (i.e., peripheral sensitization) and thus fuel central sensitization (i.e., hyperexcitability of central nociceptive pathways) and a vicious cycle of chronic pain. DRGn hyperexcitability and cPNL may reciprocally maintain each other, because cPNL can result from reflexive myospasm-induced myofascial tension, muscle weakness, and consequent muscle imbalance- and/or pain-provoked compensatory overuse. Because of pain and motor fiber damage, cPNL can worsen the causative musculoskeletal dysfunction, which further accounts for the reciprocity between the latter two factors. Sensitization increases nerve vulnerability and thus catalyzes this cycle. Because of these mechanisms and relatively greater number of neurons involved, cPNL is more likely to maintain DRGn hyperexcitability in comparison to distal neural and non-neural lesions. Compressive PNL is associated with restricted neural mobility. Intermittent (dynamic) nature of cPNL may be essential in chronic pain, because healed (i.e., fibrotic) lesions are physiologically silent and, consequently, cannot provide nociceptive input. Not all patients may be equally susceptible to develop cPNL, because occurrence of cPNL may vary as vary patients' predisposition to musculoskeletal impairment. Sensitization is accompanied by pressure pain threshold decrease and consequent mechanical allodynia and hyperalgesia, which can cause unusual local pain via natural pressure exerted by space occupying lesions or by their examination. Worsening of local pain is similarly explainable. Neuroma pain may be due to cPNL-induced axonal mechanical sensitivity and hypersensitivity of the nociceptive nervi nervorum of the nerve trunk and its stump. Intermittence and symptomatic complexity of cPNL may be the cause of frequent misdiagnosis of chronic pain.
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Surgical Treatment of Parsonage Turner Syndrome With Primary Nerve Transfers: A Case Series and Cadaver Dissection. Ann Plast Surg 2022; 89:301-305. [PMID: 35993685 DOI: 10.1097/sap.0000000000003265] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Academic Contribution Register] [Indexed: 11/25/2022]
Abstract
ABSTRACT Parsonage Turner syndrome (PTS) is the development of severe, spontaneous pain with subsequent nerve palsy. Unfortunately, many patients never achieve full functional recovery, and many have chronic pain. The use of nerve transfers in PTS has not been reported in the literature. We present 4 cases of PTS treated surgically with primary nerve transfer and neurolysis of the affected nerve following the absence of clinical and electrodiagnostic recovery at 5 months from onset. In addition, we present a cadaver dissection demonstrating an interfascicular dissection of the anterior interosseous nerve (AIN) into its components to enable a fascicular transfer in partial AIN neuropathy. Two patients with complete axillary neuropathy underwent a neurorrhaphy between the nerve branch to the lateral head of the triceps and the anterior/middle deltoid nerve branch of the axillary nerve. Two patients with partial AIN neuropathy involving the FDP to the index finger (FDP2) underwent a neurorrhaphy between an extensor carpi radialis brevis nerve branch and the FDP2 nerve branch. All patients had neurolysis of the affected nerves. All subjects recovered at least M4 motor strength. The cadaver dissection demonstrates 3 separate nerve fascicles of the AIN into FPL, FDP2, and pronator quadratus that can be individually selected for reinnervation with a fascicular nerve transfer. Functional recovery for patients with PTS with neurolysis alone is variable. Surgical treatment with neurolysis and a nerve transfer to improve functional recovery when no recovery is seen by 5 months is an option.
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Mechanisms of bone pain: Progress in research from bench to bedside. Bone Res 2022; 10:44. [PMID: 35668080 PMCID: PMC9170780 DOI: 10.1038/s41413-022-00217-w] [Citation(s) in RCA: 16] [Impact Index Per Article: 5.3] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Academic Contribution Register] [Received: 04/12/2022] [Revised: 04/29/2022] [Accepted: 05/09/2022] [Indexed: 12/27/2022] Open
Abstract
AbstractThe field of research on pain originating from various bone diseases is expanding rapidly, with new mechanisms and targets asserting both peripheral and central sites of action. The scope of research is broadening from bone biology to neuroscience, neuroendocrinology, and immunology. In particular, the roles of primary sensory neurons and non-neuronal cells in the peripheral tissues as important targets for bone pain treatment are under extensive investigation in both pre-clinical and clinical settings. An understanding of the peripheral mechanisms underlying pain conditions associated with various bone diseases will aid in the appropriate application and development of optimal strategies for not only managing bone pain symptoms but also improving bone repairing and remodeling, which potentially cures the underlying etiology for long-term functional recovery. In this review, we focus on advances in important preclinical studies of significant bone pain conditions in the past 5 years that indicated new peripheral neuronal and non-neuronal mechanisms, novel targets for potential clinical interventions, and future directions of research.
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Damiris K, Aghaie Meybodi M, Niazi M, Pyrsopoulos N. Hepatitis E in immunocompromised individuals. World J Hepatol 2022; 14:482-494. [PMID: 35582299 PMCID: PMC9055194 DOI: 10.4254/wjh.v14.i3.482] [Citation(s) in RCA: 12] [Impact Index Per Article: 4.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Academic Contribution Register] [Received: 10/12/2021] [Revised: 12/15/2021] [Accepted: 02/13/2022] [Indexed: 02/06/2023] Open
Abstract
Hepatitis E virus (HEV) originally identified as a cause of acute icteric hepatitis in developing countries has grown to be a cause of zoonotic viral hepatitis in developed countries such as the United States. While there are eight identified genotypes to date, genotype 1 (HEV1), HEV2, HEV3, HEV4 are the most common to infect humans. HEV1 and HEV2 are most common in developing countries including Latina America, Africa and Asia, and are commonly transmitted through contaminated water supplies leading to regional outbreaks. In contrast HEV3 and HEV4 circulate freely in many mammalian animals and can lead to occasional transmission to humans through fecal contamination or consumption of undercooked meat. The incidence and prevalence of HEV in the United States is undetermined given the absence of FDA approved serological assays and the lack of commercially available testing. In majority of cases, HEV infection is a self-limiting hepatitis requiring only symptomatic treatment. However, this is not the case in immunocompromised individuals, including those that have undergone solid organ or stem cell transplantation. In this subset of patients, chronic infection can be life threatening as hepatic insult can lead to inflammation and fibrosis with subsequent cirrhosis and death. The need for re-transplantation as a result of post-transplant hepatitis is of great concern. In addition, there have been many reported incidents of extrahepatic manifestations, for which the exact mechanisms remain to be elucidated. The cornerstone of treatment in immunocompromised solid organ transplant recipients is reduction of immunosuppressive therapies, while attempting to minimize the risk of organ rejection. Subsequent treatment options include ribavirin, and pegylated interferon alpha in those who have demonstrated ribavirin resistance. Further investigation assessing safety and efficacy of anti-viral therapy is imperative given the rising global health burden. Given this concern, vaccination has been approved in China with other investigations underway throughout the world. In this review we introduce the epidemiology, diagnosis, clinical manifestations, and treatment of HEV, with emphasis on immunocompromised individuals in the United States.
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Affiliation(s)
- Konstantinos Damiris
- Department of Medicine, Rutgers - New Jersey Medical School, Newark, NJ 07103, United States
| | - Mohamad Aghaie Meybodi
- Department of Medicine, Rutgers - New Jersey Medical School, Newark, NJ 07103, United States
| | - Mumtaz Niazi
- Department of Medicine - Gastroenterology and Hepatology, Rutgers - New Jersey Medical School, Newark, NJ 07103, United States
| | - Nikolaos Pyrsopoulos
- Department of Medicine - Gastroenterology and Hepatology, Rutgers - New Jersey Medical School, Newark, NJ 07103, United States
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Dedaev S, Druzhinina E, Druzhinin D, Tumurov D, Ilyasov S. Neuralgic amyotrophy. Zh Nevrol Psikhiatr Im S S Korsakova 2022; 122:8-14. [DOI: 10.17116/jnevro20221220118] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Academic Contribution Register] [Indexed: 11/17/2022]
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Seliverstova EG, Sinkin MV, Kordonsky AY, Aleinikova IB, Tikhomirov IV, Grin AA. [Electrodiagnostic evaluation in differential diagnosis and neurosurgical treatment of radiculopathies caused by spine disorders. Diagnostic value and methodology]. ZHURNAL VOPROSY NEIROKHIRURGII IMENI N. N. BURDENKO 2022; 86:109-118. [PMID: 35412720 DOI: 10.17116/neiro202286021109] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Subscribe] [Academic Contribution Register] [Indexed: 06/14/2023]
Abstract
UNLABELLED One of the most common causes of radiculopathy requiring neurosurgical treatment is a herniated disc. Magnetic resonance imaging (MRI) is still the main diagnostic approach for this lesion. However, MRI does not allow assessing the functional state of the root. Neurophysiological examination assesses the function of peripheral nervous system. These methods are used for differential diagnosis of causes of neurological symptoms and determine the level of the nerve root injury. OBJECTIVE To assess the role of electromyography including paraspinal muscle mapping in diagnosis of radiculopathies following spine diseases. MATERIAL AND METHODS We have analyzed literature data in the Scopus, Pubmed, and RSCI databases and selected 93 references for primary reviewing. Final analysis enrolled the manuscripts with a detailed description of neurophysiological examinations and data on sensitivity/specificity of these methods. RESULTS Needle electromyography (EMG) is the most informative neurophysiological method for diagnosis of radicular damage. Sensitivity of EMG is up to 90% for lumbosacral radiculopathy. Electromyography of the paraspinal muscles can be used in case of of cervical, thoracic and lumbar radiculopathy in addition to EMG of limb muscles. Therefore sensitivity increases to 100%. Diagnostic value of nerve conduction study (NCS) is low, and performing NCS without EMG is not useful. CONCLUSION In neurosurgical practice electrodiagnostic (EDX) should be performed for differential diagnosis of radiculopathy and peripheral nervous system lesions, to determine the level of radicular compression, and when physical examination does not correspond with neuroimaging or MRI is not possible to perform.
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Affiliation(s)
- E G Seliverstova
- Sklifosovsky Research Institute for Emergency Care, Moscow, Russia
| | - M V Sinkin
- Sklifosovsky Research Institute for Emergency Care, Moscow, Russia
- Evdokimov Moscow State University of Medicine and Dentistry, Moscow, Russia
| | - A Yu Kordonsky
- Sklifosovsky Research Institute for Emergency Care, Moscow, Russia
| | - I B Aleinikova
- Sklifosovsky Research Institute for Emergency Care, Moscow, Russia
| | - I V Tikhomirov
- Sklifosovsky Research Institute for Emergency Care, Moscow, Russia
| | - A A Grin
- Sklifosovsky Research Institute for Emergency Care, Moscow, Russia
- Evdokimov Moscow State University of Medicine and Dentistry, Moscow, Russia
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Kim SI, Seok HY, Yi J, Cho JH. Leg paralysis after AstraZeneca COVID-19 vaccination diagnosed as neuralgic amyotrophy of the lumbosacral plexus: a case report. J Int Med Res 2021; 49:3000605211056783. [PMID: 34816739 PMCID: PMC8649466 DOI: 10.1177/03000605211056783] [Citation(s) in RCA: 16] [Impact Index Per Article: 4.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Academic Contribution Register] [Indexed: 12/24/2022] Open
Abstract
The ongoing global administration of vaccines for coronavirus disease 2019 (COVID-19)
means that increasing numbers of patients are likely to present with post-vaccination
complications. We describe the first reported case of neuralgic amyotrophy (NA) involving
the lumbosacral plexus occurring after AstraZeneca COVID-19 vaccination. The patient
presented with acute-onset leg paralysis following administration of the vaccine. Based on
the clinical, electrodiagnostic, and radiologic findings, the patient was diagnosed with
post-vaccination NA. We speculate that the COVID-19 vaccine elicited an immune-mediated
inflammatory response to the injected antigen due to inflammatory immunity in a patient
with predisposed susceptibility to NA.
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Affiliation(s)
- Se Il Kim
- Department of Rehabilitation Medicine, 37976Keimyung University School of Medicine, Keimyung University Dongsan Hospital, Keimyung University School of Medicine, Daegu, Republic of Korea
| | - Hung Youl Seok
- Department of Neurology, 37976Keimyung University School of Medicine, Keimyung University Dongsan Hospital, Keimyung University School of Medicine, Daegu, Republic of Korea
| | - Jaehyuck Yi
- Department of Radiology, 37976Keimyung University School of Medicine, Keimyung University Dongsan Hospital, Keimyung University School of Medicine, Daegu, Republic of Korea
| | - Jang Hyuk Cho
- Department of Rehabilitation Medicine, 37976Keimyung University School of Medicine, Keimyung University Dongsan Hospital, Keimyung University School of Medicine, Daegu, Republic of Korea
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Crespo Burillo JA, Loriente Martínez C, García Arguedas C, Mora Pueyo FJ. [Amyotrophic neuralgia secondary to Vaxzevria (AstraZeneca) COVID-19 vaccine]. Neurologia 2021; 36:571-572. [PMID: 38620927 PMCID: PMC8173493 DOI: 10.1016/j.nrl.2021.05.007] [Citation(s) in RCA: 7] [Impact Index Per Article: 1.8] [Reference Citation Analysis] [Track Full Text] [Download PDF] [Journal Information] [Subscribe] [Academic Contribution Register] [Indexed: 12/03/2022] Open
Affiliation(s)
| | | | | | - F J Mora Pueyo
- Servicio de Neurología, Hospital de Barbastro, Barbastro, España
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Garofoli R, Zauderer J, Seror P, Roren A, Guerini H, Rannou F, Drapé JL, Nguyen C, Lefèvre-Colau MM. Neuralgic amyotrophy and hepatitis E infection: 6 prospective case reports. RMD Open 2021; 6:rmdopen-2020-001401. [PMID: 33219125 PMCID: PMC8011528 DOI: 10.1136/rmdopen-2020-001401] [Citation(s) in RCA: 5] [Impact Index Per Article: 1.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Academic Contribution Register] [Received: 07/17/2020] [Revised: 09/30/2020] [Accepted: 11/01/2020] [Indexed: 12/23/2022] Open
Abstract
Introduction Hepatitis E virus (HEV) represents the main cause of enterically transmitted hepatitis
worldwide. It is known that neuralgic amyotrophy (NA) is one of the most frequent
neurological manifestations of HEV. However, clinical, electrodiagnostic (EDX) and MRI
characteristics, as well as long-term follow-up of HEV-related NA have not been fully
described yet. Case reports We describe longitudinally clinical, EDX, biological and MRI results of six cases of
HEV-associated NA, diagnosed from 2012 to 2017. Patients were between the ages of 33 and
57 years old and had a positive HEV serology. Clinical patterns showed the whole
spectrum of NA, varying from extensive multiple mononeuropathy damage to single
mononeuropathy. EDX results showed that the patients totalised 26 inflammatory
mononeuropathies (1 to 8 per patient). These involved classical nerves such as
suprascapular (6/6 cases), long thoracic (5/6 cases) and accessory spinal nerves (2/6
cases) and, some less frequent more distal nerves like anterior interosseous nerve (3/6
cases), as well as some unusual ones such as the lateral antebrachial cutaneous nerve
(1/6 case), sensory fibres of median nerve (1/6 case) and phrenic nerves (1/6 case).
After 2 to 8 years, all nerves had clinically recovered (muscle examination above
3/5 on MRC scale for all muscles except in one patient). Discussion HEV should be systematically screened when NA is suspected, whatever the severity, if
the onset is less than 4 months (before IgM HEV-antibodies disappear) and appears
to be frequently associated with severe clinical and EDX pattern, without increasing the
usual recovery time.
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Affiliation(s)
- Romain Garofoli
- Service de Rééducation et de Réadaptation de l'Appareil Locomoteur et des Pathologies du Rachis, Hôpital Cochin, AP-HP.Centre-Université de Paris, Paris, France
| | - Jennifer Zauderer
- Service de Rééducation et de Réadaptation de l'Appareil Locomoteur et des Pathologies du Rachis, Hôpital Cochin, AP-HP.Centre-Université de Paris, Paris, France
| | - Paul Seror
- Département De Neurophysiologie, Univ. Paris Pierre Et Marie Curie, Hôpital Pitié-Salpêtrière, Assistance Publique-Hôpitaux de Paris, Paris, France
| | - Alexandra Roren
- Service de Rééducation et de Réadaptation de l'Appareil Locomoteur et des Pathologies du Rachis, Hôpital Cochin, AP-HP.Centre-Université de Paris, Paris, France
| | - Henri Guerini
- Service de Radiologie ostéo-articulaire, Hôpital Cochin, Paris, France
| | - François Rannou
- Service de Rééducation et de Réadaptation de l'Appareil Locomoteur et des Pathologies du Rachis, Hôpital Cochin, AP-HP.Centre-Université de Paris, Paris, France.,Université Paris Descartes, PRES Sorbonne Paris Cité, Paris, France
| | - Jean-Luc Drapé
- Service de Radiologie ostéo-articulaire, Hôpital Cochin, Paris, France
| | - Christelle Nguyen
- Service de Rééducation et de Réadaptation de l'Appareil Locomoteur et des Pathologies du Rachis, Hôpital Cochin, AP-HP.Centre-Université de Paris, Paris, France.,Université de Paris, Faculté de Santé, UFR de Médecine, Paris, France.,INSERM UMR-S 1124, Toxicité Environnementale, Cibles Thérapeutiques, Signalisation Cellulaire et Biomarqueurs (T3S), Campus Saint-Germain-des-Prés, Paris, France
| | - Marie-Martine Lefèvre-Colau
- Service de Rééducation et de Réadaptation de l'Appareil Locomoteur et des Pathologies du Rachis, Hôpital Cochin, AP-HP.Centre-Université de Paris, Paris, France
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Díaz C, Contreras JJ, Muñoz M, Osorio M, Quiroz M, Pizarro R. Parsonage-Turner syndrome association with SARS-CoV-2 infection. JSES REVIEWS, REPORTS, AND TECHNIQUES 2021; 1:252-256. [PMID: 34913044 PMCID: PMC8059331 DOI: 10.1016/j.xrrt.2021.04.002] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Download PDF] [Figures] [Subscribe] [Academic Contribution Register] [Indexed: 11/18/2022]
Affiliation(s)
- Cristóbal Díaz
- Shoulder and Elbow Unit, Instituto Traumatológico, Santiago, Chile
- Department of Orthopedics and Trauma, Universidad de Chile, Santiago, Chile
| | - Julio J Contreras
- Shoulder and Elbow Unit, Instituto Traumatológico, Santiago, Chile
- Department of Orthopedics and Trauma, Universidad de Chile, Santiago, Chile
- Shoulder and Elbow Unit, Pontificia Universidad Católica de Chile, Santiago, Chile
- Department of Orthopedics and Trauma, Pontificia Universidad Católica de Chile, Santiago, Chile
| | - Martín Muñoz
- Department of Orthopedics and Trauma, Universidad de Chile, Santiago, Chile
| | - Matías Osorio
- Shoulder and Elbow Unit, Instituto Traumatológico, Santiago, Chile
- Department of Orthopedics and Trauma, Universidad de Chile, Santiago, Chile
| | - Milton Quiroz
- Department of Radiology, Clínica Cordillera, Santiago, Chile
| | - Renato Pizarro
- Department of Radiology, Clínica Cordillera, Santiago, Chile
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Crespo Burillo JA, Loriente Martínez C, García Arguedas C, Mora Pueyo FJ. Amyotrophic neuralgia secondary to Vaxzevri (AstraZeneca) COVID-19 vaccine. NEUROLOGÍA (ENGLISH EDITION) 2021; 36:571-572. [PMID: 34330677 PMCID: PMC8316086 DOI: 10.1016/j.nrleng.2021.05.002] [Citation(s) in RCA: 7] [Impact Index Per Article: 1.8] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Download PDF] [Journal Information] [Subscribe] [Academic Contribution Register] [Received: 05/13/2021] [Accepted: 05/18/2021] [Indexed: 11/18/2022] Open
Affiliation(s)
| | | | | | - F J Mora Pueyo
- Servicio de Neurología, Hospital de Barbastro, Barbastro, Spain
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Viatgé T, Noel-Savina E, Prévot G, Faviez G, Plat G, De Boissezon X, Cintas P, Didier A. [Parsonage-Turner syndrome following severe SARS-CoV-2 infection]. Rev Mal Respir 2021; 38:853-858. [PMID: 34325956 PMCID: PMC8233958 DOI: 10.1016/j.rmr.2021.06.004] [Citation(s) in RCA: 5] [Impact Index Per Article: 1.3] [Reference Citation Analysis] [Abstract] [Key Words] [Download PDF] [Figures] [Journal Information] [Subscribe] [Academic Contribution Register] [Received: 01/13/2021] [Accepted: 06/01/2021] [Indexed: 01/08/2023]
Abstract
Introduction Les complications de l’infection par le SARS-CoV-2 peuvent affecter la sphère neurologique ; il s’agit habituellement de cas de polyradiculonévrite aiguë inflammatoire ou de plexite. Observation Le cas clinique rapporté concerne un patient de 20 ans sans comorbidités, atteint d’une infection sévère à la COVID-19 compliquée d’un syndrome de détresse respiratoire aiguë, avec manifestations thromboemboliques et surinfection bactérienne. Ce patient souffrait également d’un trouble neurologique non spécifique lié au SARS-Cov-2 avec une paralysie pseudobulbaire (IRM, EMG et ponction lombaire étaient normaux), associé à des troubles neurologiques persistants 4 mois plus tard, caractérisés par un déficit à prédominance motrice de l’épaule gauche et une insuffisance respiratoire. Le bilan respiratoire et neurologique conclut à un syndrome de Parsonage-Turner, ou amyotrophie névralgique, affectant les racines nerveuses en C5-C6, le nerf pectoral latéral et le nerf phrénique à l’origine de l’amyotrophie de la ceinture scapulaire et de la paralysie du diaphragme gauche. Conclusion Ce cas montre que la dyspnée persistante après une infection à la COVID-19 doit faire rechercher une cause diaphragmatique et que celle-ci n’est pas toujours secondaire à la neuropathie de réanimation, mais peut aussi être le témoin d’une amyotrophie névralgique.
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Affiliation(s)
- T Viatgé
- Service de pneumologie, hôpital Larrey, CHU de Toulouse, 31059 Toulouse cedex 9, France.
| | - E Noel-Savina
- Service de pneumologie, hôpital Larrey, CHU de Toulouse, 31059 Toulouse cedex 9, France
| | - G Prévot
- Service de pneumologie, hôpital Larrey, CHU de Toulouse, 31059 Toulouse cedex 9, France
| | - G Faviez
- Service de pneumologie, hôpital Larrey, CHU de Toulouse, 31059 Toulouse cedex 9, France
| | - G Plat
- Service de pneumologie, hôpital Larrey, CHU de Toulouse, 31059 Toulouse cedex 9, France
| | - X De Boissezon
- Service de médecine physique et de réadaptation, hôpital Rangueil, CHU de Toulouse, 31059 Toulouse cedex 9, France
| | - P Cintas
- Service de neurologie, hôpital Purpan, CHU de Toulouse, 31059 Toulouse cedex 9, France
| | - A Didier
- Service de pneumologie, hôpital Larrey, CHU de Toulouse, 31059 Toulouse cedex 9, France
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27
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Answer to Cabona et al « Isolated musculocutaneous nerve involvement in COVID-19 related Neuralgic amyotrophy» Joint Bone Spine 2021;88:105238 and to Finsterer and Scorza « SARS-CoV-2 or SARS-CoV-2 vaccination associated Parsonage-Turner syndrome». Joint Bone Spine 2021;88:105239. Joint Bone Spine 2021; 88:105240. [PMID: 34146696 PMCID: PMC8270742 DOI: 10.1016/j.jbspin.2021.105240] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Subscribe] [Academic Contribution Register] [Accepted: 06/09/2021] [Indexed: 11/29/2022]
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Abstract
Neuralgic amyotrophy (NA) is markedly underdiagnosed in clinical practice, and
its actual incidence rate is about 1 per 1000 per year. In the current article,
we provide an overview of essential information about NA, including the
etiology, clinical manifestations, diagnostic investigations, differential
diagnosis, treatment, and prognosis. The causes of NA are multifactorial and
include immunological, mechanical, or genetic factors. Typical clinical findings
are a sudden onset of pain in the shoulder region, followed by patchy flaccid
paralysis of muscles in the shoulder and/or arm. A diagnosis of NA is based on a
patient’s clinical history and physical examination. Gadolinium-enhanced
magnetic resonance imaging and high-resolution magnetic resonance neurography
are useful for confirming the diagnosis and choosing the appropriate treatment.
However, before a diagnosis of NA is confirmed, other disorders with similar
symptoms, such as cervical radiculopathy or rotator cuff tear, need to be ruled
out. The prognosis of NA depends on the degree of axonal damage. In conclusion,
many patients with motor weakness and pain are encountered in clinical practice,
and some of these patients will exhibit NA. It is important that clinicians
understand the key features of this disorder to avoid misdiagnosis.
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Affiliation(s)
- Tae Uk Kim
- Department of Physical Medicine and Rehabilitation, College of Medicine, Dankook University, Cheonan, Republic of Korea
| | - Min Cheol Chang
- Department of Physical Medicine and Rehabilitation, College of Medicine, Yeungnam University, Daegu, Republic of Korea
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Firmino GF, Schulze ML, Schlindwein MAM, Rampeloti B, Gonçalves MVM, Maçaneiro CH, Dos Santos RA. Neuralgic Amyotrophy: Its Importance in Orthopedics Practice. Spine Surg Relat Res 2021; 5:232-237. [PMID: 34435146 PMCID: PMC8356235 DOI: 10.22603/ssrr.2021-0014] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Academic Contribution Register] [Received: 01/29/2021] [Accepted: 03/17/2021] [Indexed: 11/23/2022] Open
Abstract
The present academic work aims to contribute to an early diagnosis of neuralgic amyotrophy (NA) because of its high prevalence in the population. This disease is a neuromuscular syndrome with unclear etiology; it affects mostly the brachial plexus, causing acute pain in the affected shoulder, paralysis, and disabilities. Considering the importance of an early treatment that can modify the prognosis of the patient, knowing the last updates about the syndrome as its clinical presentation is important. Data analysis was conducted through an online non-systematic review that indicated the epidemiology, pathophysiology, and differential diagnosis and prognosis of NA. Knowledge of the clinical features of NA is not common; however, it is important in orthopedic practice because it requires differentiation from spine pathologies.
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Affiliation(s)
- George Fagundes Firmino
- Medical Student at Department of Medicine, University of the Region of Joinville, Joinville, Brazil
| | - Milena Luisa Schulze
- Medical Student at Department of Medicine, University of the Region of Joinville, Joinville, Brazil
| | | | - Breno Rampeloti
- Medical Student at Department of Medicine, University of the Region of Joinville, Joinville, Brazil
| | | | - Carlos Henrique Maçaneiro
- Professor of Orthopedics and Traumatology, Department of Medicine, University of the Region of Joinville, Joinville, Brazil
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Off-Label Use of Intravenous Immunoglobulin with Methylprednisolone to Treat Parsonage-Turner Syndrome in a United States Marine. Case Rep Med 2021; 2021:6663755. [PMID: 33868407 PMCID: PMC8035001 DOI: 10.1155/2021/6663755] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Academic Contribution Register] [Received: 12/09/2020] [Revised: 03/05/2021] [Accepted: 03/14/2021] [Indexed: 11/17/2022] Open
Abstract
Neuralgic amyotrophy (NA) also known as Parsonage-Turner syndrome is an inflammatory disorder of the brachial plexus characterized by sudden, acute onset of severe pain of the arm and/or shoulder followed by muscle weakness and sensory abnormalities. Although management may involve physical therapy, immunomodulatory drugs, and analgesics, there is nothing specific for the treatment of NA. Full functional recovery can take months to years, but recurrence and/or persistence of symptoms and disability are frequent. This case reports a 22-year-old male who recovered from NA within 3 months following treatment with 1000 mg of methylprednisolone and off-label use of 0.5 g/kg of intravenous immunoglobulins (IVIG) for four consecutive days. Three years later, the patient experienced soreness and paresthesia of the shoulder following a military shooting exercise, and 0.75 g/kg of IVIG and 1000 mg of MP were prescribed for 2 consecutive days resulting in complete recovery and no recurrences to date. EMG findings, 3.5-year postinitial treatment, revealed improvement in the brachial plexopathy. This provides support for the combined use of IVIG and glucocorticoids in the treatment of NA and highlights the need for further studies investigating whether this combined treatment regimen may accelerate recovery and improve long-term outcomes for patients diagnosed with NA.
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31
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Poetschke J, Schwarz D, Kremer T, Rein S. [Lesions of the anterior interosseous nerve: differentiating between compression neuropathy and neuritis]. HANDCHIR MIKROCHIR P 2021; 53:31-39. [PMID: 33588494 DOI: 10.1055/a-1349-4989] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Academic Contribution Register] [Indexed: 10/22/2022] Open
Abstract
BACKGROUND In cases of anterior interosseous nerve (AIN) syndrome, it is often difficult to differentiate between compression neuropathy and neuritis. MATERIAL AND METHODS This review analyses the clinical aspects of the neuritic AIN syndrome and the different diagnostic tools for securing the diagnosis and differentiating the condition from compression neuropathy. Based on these data, the current therapeutic options are proposed. RESULTS The AIN syndrome often results from neuritis of the AIN fascicles within the trunk of the median nerve. The differentiation between neuritis and compression neuropathy of the AIN is based on dedicated neurophysiological examinations as well as nerve sonography and MRI neurography. Although conservative treatment is the gold standard, microsurgical interventions have become more important in recent years. CONCLUSION A dedicated diagnostic workup of the AIN syndrome is paramount for optimal treatment. Conservative treatment remains the standard to date. However, if torsions and constrictions of nerve fascicles are detected, intrafascicular neurolysis should be considered, as current research shows the potential for an improved outcome in such cases.
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Affiliation(s)
- Julian Poetschke
- Klinikum Sankt Georg gGmbH Klinik für Plastische und Handchirurgie mit Schwerbrandverletztenzentrum
| | - Daniel Schwarz
- Abteilung für Neuroradiologie, AG MR-Neurographie, Neurologische Klinik, Universitätsklinikum Heidelberg
| | - Thomas Kremer
- Klinikum Sankt Georg gGmbH Klinik für Plastische und Handchirurgie mit Schwerbrandverletztenzentrum
| | - Susanne Rein
- Klinikum Sankt Georg gGmbH Klinik für Plastische und Handchirurgie mit Schwerbrandverletztenzentrum.,Martin-Luther-Universität Halle-Wittenberg, Halle (Saale)
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32
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Hu X, Jing M, Feng J, Tang J. Four cases of pediatric neuralgic amyotrophy treated with immunotherapy: one-year follow-up and literature review. J Int Med Res 2021; 48:300060520912082. [PMID: 32228355 PMCID: PMC7132571 DOI: 10.1177/0300060520912082] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Academic Contribution Register] [Indexed: 11/17/2022] Open
Abstract
Neuralgic amyotrophy (NA) is a neurological disease that occurs across all age
groups, but its prognosis in children is controversial. The present report adds
to the knowledge about its prognosis by describing four cases of pediatric NA in
which the patients were treated with immunotherapy and followed up for 1 year.
We also present a summary of relevant cases of pediatric NA treated with
immunotherapy. The clinical features of the four present cases were similar to
those of previously reported cases, and their symptoms improved after
immunotherapy. At the 1-year follow-up, three of the children gained near
complete recovery, and their improvement was significantly better than that
observed at the 2-month follow-up. A review of the literature showed that most
previously reported children with NA showed improvement after immunotherapy, but
no more than half of the patients recovered fully. These findings indicate that
in children with NA, immunotherapy is fairly effective and its benefits improve
with time. Thus, long-term follow-up is needed in these patients to determine
their prognosis.
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Affiliation(s)
- Xiaoyue Hu
- Department of Neurology, Children's Hospital of Soochow University, Suzhou, Jiangsu Province, China.,Department of Neurology, Wuxi Children's Hospital, Wuxi, Jiangsu Province, China
| | - Miao Jing
- Department of Neurology, Wuxi Children's Hospital, Wuxi, Jiangsu Province, China
| | - Jun Feng
- Department of Neurology, Children's Hospital of Soochow University, Suzhou, Jiangsu Province, China
| | - Jihong Tang
- Department of Neurology, Children's Hospital of Soochow University, Suzhou, Jiangsu Province, China
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33
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Speicher TE, Cui JW, Scharmann SD. The Role of Positional Release Therapy in Treating Recalcitrant Brachial Plexus Neuritis: A Case Report. J Athl Train 2020; 56:1124-1131. [PMID: 33351937 DOI: 10.4085/jat003-19] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Academic Contribution Register] [Indexed: 11/09/2022]
Abstract
A 17-year-old female Caucasian soccer player presented with severe right shoulder pain and scapular winging due to brachial plexus neuritis. Over the course of 6 weeks, the patient received Positional Release Therapy once a week coupled with electrical modalities, massage and a daily home exercise program. The form of brachial plexus neuritis the patient was diagnosed with was Parsonage-Turner Syndrome, a rare condition often resistant to traditional physical therapy, typically persists for six months to years, at times requiring surgical intervention. This case report is unique because it is the first to utilize Positional Release Therapy for its treatment and one which resolved more quickly than typically reported.
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Affiliation(s)
- Timothy E Speicher
- President, Positional Release Therapy Institute, 1702E. 5600S. Ogden, UT 84403, (801) 689-2546-Phone, (385) 206-8657-Fax, , https://twitter.com/speichertim
| | - Jia-Wen Cui
- Institute of Rehabilitation Medicine, Shanghai University of Traditional Chinese Medicine.,School of Rehabilitation Science, Shanghai University of Traditional Chinese Medicine
| | - Stephen D Scharmann
- Assistant Residency Director, McKay Dee Family Medicine Residency, Medical Director, Weber State University, Department of Athletic Training,
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Farr E, D'Andrea D, Franz CK. Phrenic Nerve Involvement in Neuralgic Amyotrophy (Parsonage-Turner Syndrome). Sleep Med Clin 2020; 15:539-543. [PMID: 33131663 DOI: 10.1016/j.jsmc.2020.08.002] [Citation(s) in RCA: 10] [Impact Index Per Article: 2.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Academic Contribution Register] [Indexed: 12/17/2022]
Abstract
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.
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Affiliation(s)
- Ellen Farr
- Shirley Ryan AbilityLab, McGaw Medical Center, Northwestern University, 355 East Erie Street, 26N (Biologics), Chicago, IL 60611, USA
| | - Dom D'Andrea
- Shirley Ryan AbilityLab, 355 East Erie Street, 26N (Biologics), Chicago, IL 60611, USA
| | - Colin K Franz
- Shirley Ryan AbilityLab, Feinberg School of Medicine, Northwestern University, 355 East Erie Street, 26N (Biologics), Chicago, IL 60611, USA.
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35
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Vetrano IG, Nazzi V. Letter to the Editor. Treatment of spontaneous anterior interosseous nerve palsy. J Neurosurg 2020; 133:1274-1275. [PMID: 32679556 DOI: 10.3171/2020.4.jns201156] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Academic Contribution Register] [Indexed: 11/06/2022]
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36
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Rubin DI. Brachial and lumbosacral plexopathies: A review. Clin Neurophysiol Pract 2020; 5:173-193. [PMID: 32954064 PMCID: PMC7484503 DOI: 10.1016/j.cnp.2020.07.005] [Citation(s) in RCA: 25] [Impact Index Per Article: 5.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Academic Contribution Register] [Received: 06/18/2020] [Revised: 07/09/2020] [Accepted: 07/31/2020] [Indexed: 12/11/2022] Open
Abstract
Diseases of the brachial and lumbosacral plexus are uncommon and complex. The diagnosis of plexopathies is often challenging for the clinician, both in terms of localizing a patient's symptoms to the plexus as well as determining the etiology. The non-specific clinical features and similar presentations to other root, nerve, and non-neurologic disorders emphasize the importance of a high clinical index of suspicion for a plexopathy and comprehensive clinical evaluation. Various diagnostic tests, including electrodiagnostic (EDX) studies, neuroimaging (including ultrasound, MRI, or PET), serologic studies, and genetic testing, may be used to confirm a plexopathy and assist in identifying the underlying etiology. EDX testing plays an important role in confirming a plexopathy defining the localization, pathophysiology, chronicity, severity, and prognosis. Given the complexity of the plexus anatomy, multiple common and uncommon NCS and an extensive needle examination is often required, and a comprehensive, individualized approach to each patient is necessary. Treatment of plexopathies often focuses on symptomatic management although, depending on the etiology, specific targeted treatments may improve outcome. This article reviews the clinical features, EDX approaches, and evaluation and treatment of brachial and lumbosacral plexopathies.
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Affiliation(s)
- Devon I. Rubin
- Electromyography Laboratory, Mayo Clinic, Jacksonville, FL, USA
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37
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Gstoettner C, Mayer JA, Rassam S, Hruby LA, Salminger S, Sturma A, Aman M, Harhaus L, Platzgummer H, Aszmann OC. Neuralgic amyotrophy: a paradigm shift in diagnosis and treatment. J Neurol Neurosurg Psychiatry 2020; 91:879-888. [PMID: 32487526 DOI: 10.1136/jnnp-2020-323164] [Citation(s) in RCA: 64] [Impact Index Per Article: 12.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Academic Contribution Register] [Received: 03/07/2020] [Revised: 05/07/2020] [Accepted: 05/10/2020] [Indexed: 01/22/2023]
Abstract
Neuralgic amyotrophy (NA), also known as Parsonage-Turner syndrome, is characterised by sudden pain attacks, followed by patchy muscle paresis in the upper extremity. Recent reports have shown that incidence is much higher than previously assumed and that the majority of patients never achieve full recovery. Traditionally, the diagnosis was mainly based on clinical observations and treatment options were confined to application of corticosteroids and symptomatic management, without proven positive effects on long-term outcomes. These views, however, have been challenged in the last years. Improved imaging methods in MRI and high-resolution ultrasound have led to the identification of structural peripheral nerve pathologies in NA, most notably hourglass-like constrictions. These pathognomonic findings have paved the way for more accurate diagnosis through high-resolution imaging. Furthermore, surgery has shown to improve clinical outcomes in such cases, indicating the viability of peripheral nerve surgery as a valuable treatment option in NA. In this review, we present an update on the current knowledge on this disease, including pathophysiology and clinical presentation, moving on to diagnostic and treatment paradigms with a focus on recent radiological findings and surgical reports. Finally, we present a surgical treatment algorithm to support clinical decision making, with the aim to encourage translation into day-to-day practice.
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Affiliation(s)
- Clemens Gstoettner
- Clinical Laboratory for Bionic Extremity Reconstruction, Department of Surgery, Medical University of Vienna, Vienna, Austria
| | - Johannes A Mayer
- Clinical Laboratory for Bionic Extremity Reconstruction, Department of Surgery, Medical University of Vienna, Vienna, Austria.,Department for Hand, Plastic, Reconstructive and Burn Surgery, BG Trauma Center Tuebingen at the Eberhard Karls University Tuebingen, Tuebingen, Germany
| | - Stephanie Rassam
- Clinical Laboratory for Bionic Extremity Reconstruction, Department of Surgery, Medical University of Vienna, Vienna, Austria.,Department of General, Visceral, Endocrine and Transplantation Surgery, Cantonal Hospital St. Gallen, St. Gallen, Switzerland
| | - Laura A Hruby
- Clinical Laboratory for Bionic Extremity Reconstruction, Department of Surgery, Medical University of Vienna, Vienna, Austria.,Department of Orthopaedics and Trauma Surgery, Medical University of Vienna, Vienna, Austria
| | - Stefan Salminger
- Clinical Laboratory for Bionic Extremity Reconstruction, Department of Surgery, Medical University of Vienna, Vienna, Austria.,Division of Plastic and Reconstructive Surgery, Department of Surgery, Medical University of Vienna, Vienna, Austria
| | - Agnes Sturma
- Clinical Laboratory for Bionic Extremity Reconstruction, Department of Surgery, Medical University of Vienna, Vienna, Austria.,Department of Bioengineering, Imperial College London, London, UK
| | - Martin Aman
- Clinical Laboratory for Bionic Extremity Reconstruction, Department of Surgery, Medical University of Vienna, Vienna, Austria.,Department of Hand, Plastic and Reconstructive Surgery, Burn Center-Hand and Plastic Surgery, University of Heidelberg, BG Trauma Center Ludwigshafen, Ludwigshafen, Germany
| | - Leila Harhaus
- Department of Hand, Plastic and Reconstructive Surgery, Burn Center-Hand and Plastic Surgery, University of Heidelberg, BG Trauma Center Ludwigshafen, Ludwigshafen, Germany
| | - Hannes Platzgummer
- Department of Biomedical Imaging and Image-guided Therapy, Medical University of Vienna, Vienna, Austria
| | - Oskar C Aszmann
- Clinical Laboratory for Bionic Extremity Reconstruction, Department of Surgery, Medical University of Vienna, Vienna, Austria .,Division of Plastic and Reconstructive Surgery, Department of Surgery, Medical University of Vienna, Vienna, Austria
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38
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Fleet JL, Harish S, Bain J, Baker SK. Arm Numbness at 45 Degrees Abduction: A Case Report of Thoracic Outlet Syndrome After Brachial Neuritis. JOURNAL OF REHABILITATION MEDICINE - CLINICAL COMMUNICATIONS 2020; 3:1000034. [PMID: 33884136 PMCID: PMC8008736 DOI: 10.2340/20030711-1000034] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Subscribe] [Academic Contribution Register] [Accepted: 04/16/2020] [Indexed: 11/21/2022]
Abstract
Objective To describe a case of nerve kinking correlating with surgical findings in neurogenic thoracic outlet syndrome in a patient with history of brachial neuritis. Thoracic outlet syndrome and brachial neuritis are briefly reviewed. Case report A 32-year-old woman with a history of bilateral brachial neuritis presented with paraesthesias in her hand when abducting her shoulder to 45° or higher. A kink in the superior trunk of the brachial plexus, as well as asymmetrically narrowed costoclavicular space, was found on magnetic resonance imaging with the shoulder abducted. Conservative measures failed, leading to partial anterior scalenectomy and neurolysis, which led to improvement in her symptoms. Conclusion Anatomical variations in combination with biomechanical changes after brachial neuritis can be associated with neurogenic thoracic outlet syndrome.
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Affiliation(s)
- Jamie L Fleet
- Division of Physical Medicine & Rehabilitation, Department of Medicine, McMaster University, Hamilton, Canada
| | | | - James Bain
- Division of Plastic Surgery, Department of Surgery, McMaster University, Hamilton, Canada
| | - Steven K Baker
- Division of Physical Medicine & Rehabilitation, Department of Medicine, McMaster University, Hamilton, Canada
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39
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Hamada H, Sugaya H, Takahashi N, Matsuki K, Tokai M, Ueda Y, Hoshika S, Kuniyoshi K. Incidence of Axillary Nerve Injury After Arthroscopic Shoulder Stabilization. Arthroscopy 2020; 36:1555-1564. [PMID: 32109573 DOI: 10.1016/j.arthro.2020.02.016] [Citation(s) in RCA: 11] [Impact Index Per Article: 2.2] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Academic Contribution Register] [Received: 08/11/2019] [Revised: 02/07/2020] [Accepted: 02/08/2020] [Indexed: 02/02/2023]
Abstract
PURPOSE To investigate the incidence of axillary nerve palsy after arthroscopic shoulder stabilization and to measure the distance between the nerve and capsule in shoulders with a capsular lesion. METHODS This retrospective study included 2,027 shoulders (1,909 patients; 1,433 male and 476 female patients; mean age, 32 years [age range, 13-81 years]) subjected to arthroscopic soft-tissue stabilization for recurrent shoulder instability from 2005 to 2017. The exclusion criteria were bone grafting or transfer and preoperative axillary nerve symptoms. We retrospectively reviewed patient records and investigated the incidence and clinical features of axillary nerve palsy. We measured the closest distance between the axillary nerve and capsule on preoperative magnetic resonance images. RESULTS Postoperative axillary nerve palsy occurred in 4 shoulders (0.2% of all arthroscopic stabilizations). Capsular repair was performed in 2 shoulders (1.2% of 160 capsular repairs); humeral avulsion of the glenohumeral ligament (HAGL) repair, 1 shoulder (2% of 47 HAGL repairs); and isolated Bankart repair, 1 shoulder (0.05% of 1,941 Bankart repairs). The closest distance between the nerve and capsule was 3.4 ± 3.2 mm in shoulders with capsular or HAGL lesions and less than 1 mm in the 3 shoulders with palsy. The common symptoms in axillary nerve palsy cases were shoulder discomfort, delayed recovery of range of motion, and deltoid weakness and atrophy. A definitive diagnosis was made with electromyography in all cases. Nerve injury by a suture was confirmed during revision surgery in 3 shoulders subjected to capsular or HAGL repair during the initial operation. The palsy was transient and fully recovered in 1 shoulder with isolated Bankart repair. CONCLUSIONS The incidence of axillary nerve palsy after arthroscopic soft-tissue shoulder stabilization was low but higher in shoulders subjected to capsular or HAGL repair. We should always consider the possibility of axillary nerve palsy in shoulders that require capsular or HAGL repair. LEVEL OF EVIDENCE Level IV, therapeutic case series.
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Affiliation(s)
- Hiroshige Hamada
- Sports Medicine & Joint Center, Funabashi Orthopaedic Hospital, Funabashi, Japan
| | - Hiroyuki Sugaya
- Sports Medicine & Joint Center, Funabashi Orthopaedic Hospital, Funabashi, Japan.
| | - Norimasa Takahashi
- Sports Medicine & Joint Center, Funabashi Orthopaedic Hospital, Funabashi, Japan
| | - Keisuke Matsuki
- Sports Medicine & Joint Center, Funabashi Orthopaedic Hospital, Funabashi, Japan
| | - Morihito Tokai
- Sports Medicine & Joint Center, Funabashi Orthopaedic Hospital, Funabashi, Japan
| | - Yusuke Ueda
- Sports Medicine & Joint Center, Funabashi Orthopaedic Hospital, Funabashi, Japan
| | - Shota Hoshika
- Sports Medicine & Joint Center, Funabashi Orthopaedic Hospital, Funabashi, Japan
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40
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Goetsch MR, Shen J, Jones JA, Memon A, Chatham W. Neuralgic Amyotrophy Presenting with Multifocal Myonecrosis and Rhabdomyolysis. Cureus 2020; 12:e7382. [PMID: 32337110 PMCID: PMC7179979 DOI: 10.7759/cureus.7382] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Academic Contribution Register] [Indexed: 11/29/2022] Open
Abstract
Neuralgic amyotrophy (NA), also known as Parsonage-Turner syndrome, is an idiopathic disorder characterized by rapid-onset unilateral upper extremity pain, paralysis, and sensory disturbance in the distribution of the brachial plexus. The etiology is unknown, and there is a multitude of alternative clinical presentations as well as secondary triggers, which make the diagnosis challenging. To date, there has been no report of NA presenting with frank myonecrosis. In this report, we document the first case of NA presenting with multifocal myonecrosis of the shoulder girdle muscles and rhabdomyolysis. This case posed a unique challenge in the diagnostic workup and management as many causes of myonecrosis present similarly to NA, and NA is a diagnosis of exclusion. Our patient underwent exhaustive testing and several trials of therapy before diagnosis could be made. Such evaluations are expensive and carry risks for patients. As such, it is important that physicians recognize this unique presentation of NA.
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Affiliation(s)
| | - Jeffrey Shen
- Internal Medicine, University of Alabama at Birmingham, Birmingham, USA
| | - Jeffrey A Jones
- Pathology, University of Alabama at Birmingham, Birmingham, USA
| | - Adeel Memon
- Neurology, University of Alabama at Birmingham, Birmingham, USA
| | - Walter Chatham
- Medicine, University of Alabama at Birmingham, Birmingham, USA
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Seror P, Roren A, Lefevre-Colau MM. Infraspinatus muscle palsy involving suprascapular nerve, brachial plexus or cervical roots related to inflammatory or mechanical causes: Experience of 114 cases. Neurophysiol Clin 2020; 50:103-111. [PMID: 32147283 DOI: 10.1016/j.neucli.2020.02.002] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Academic Contribution Register] [Received: 07/13/2019] [Revised: 02/11/2020] [Accepted: 02/11/2020] [Indexed: 11/17/2022] Open
Abstract
OBJECTIVES To report a large series of neurogenic infraspinatus muscle (ISM) palsy, in order to improve knowledge of diagnosis, diverse etiologies and care management. METHODS Clinical and electrodiagnostic (EDX) data for 114 cases of ISM palsy were collected over a 21-year period. Cases were attributed to 4 clinical conditions: (1) isolated suprascapular nerve mononeuropathy (n=48), (2) multiple mononeuropathies (n=33), (3) plexus lesions (n=17), and (4) cervical radiculopathy (n=16). These were related to 2 mechanisms: inflammatory (dysimmune) and mechanical. RESULTS Group 1 cases were younger, had the most severe ISM palsies, were mostly related to inflammatory lesions (81%) such as neuralgic amyotrophy (NA), and frequently had delayed diagnosis because disability was mild. Group 2 cases were all related to inflammatory lesions and had slightly less severe ISM palsies that were frequently hidden by winged scapula. In groups 3 and 4, ISM palsies were milder and all cases were related to mechanical lesions such as brachial plexus trauma or C4-C5-C6 radiculopathy. In these cases, deltoid and ISM palsies were equal in frequency and severity whereas biceps brachii impairment was less frequent and much milder. Deltoid palsy frequently appeared predominant as compared with ISM palsy, because upper limb elevation palsy was more disabling than external rotation palsy. CONCLUSIONS ISM palsy is a rare condition, often under-diagnosed and misidentified. The 4 main conditions of ISM palsy may be recognized by careful clinical, EDX and other examinations when necessary. Analysis of the present series highlights some clinical and EDX points that should help non-specialist and even specialist clinicians who are faced with this rare condition, to distinguish mechanical and inflammatory causes, and thus adapt patient management accordingly.
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Affiliation(s)
- Paul Seror
- Laboratoire d'électroneuromyographie, 146, avenue Ledru Rollin, 75011 Paris, France; Ramsay générale de santé, hôpital privé de l'Est Parisien, Aulnay sous-Bois, France.
| | - Alexandra Roren
- INSERM UMR-S 1153, Service de Rééducation et de Réadaptation de l'Appareil Locomoteur et des Pathologies du Rachis, PRES Sorbonne Paris Cité, ECaMO team, Institut Fédératif de Recherche sur le Handicap, Hôpital Cochin, Université Paris Descartes, AP-HP, Paris, France
| | - Marie Martine Lefevre-Colau
- INSERM UMR-S 1153, Service de Rééducation et de Réadaptation de l'Appareil Locomoteur et des Pathologies du Rachis, PRES Sorbonne Paris Cité, ECaMO team, Institut Fédératif de Recherche sur le Handicap, Hôpital Cochin, Université Paris Descartes, AP-HP, Paris, France
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42
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Clinico-diagnostic features of neuralgic amyotrophy in childhood. Neurol Sci 2020; 41:1735-1740. [PMID: 32140911 DOI: 10.1007/s10072-020-04314-8] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Academic Contribution Register] [Received: 11/04/2019] [Accepted: 02/21/2020] [Indexed: 10/24/2022]
Abstract
Neuralgic amyotrophy (NA), even known as Personage-Turner's syndrome (PTS), is a neurologic condition, affecting the lower motor neurons of brachial plexus and/or individual nerves or nerve branches, characterized by pain, muscle weakness/atrophy, and sensory symptoms. NA has an acute/subacute onset, after an infection or vaccination; it is more common in male and is rare in the pediatric population. The etiology remains uncertain, being considered heterogeneous and multifactorial. A severe acute neurologic pain around the shoulder girdle is the classic presenting symptom at onset. As the pain subsides, weakness and paresis develop. NA is usually unilateral, but sometimes, a subclinical contralateral limb involvement could be present and bilateral affection has been described. The diagnosis is clinical, through a comprehensive history and neurological examination. However, electrophysiological testing and imaging are critical, because there is no diagnostic test for PTS and it remains a diagnosis of exclusion. Upper brachial plexus peripheral involvement with weakness of periscapular and perihumeral muscles is the classic presentation, associated with electrophysiological evidence of denervation in the affected muscles. Imaging, laboratory, and genetic testing can be useful for the differential diagnosis. NA is in most cases a self-limiting condition, and it is characterized by good recovery. Treatment of NA usually involves a combination of corticosteroids, analgesics, immobilization, and physical therapy, even if limited data are available in children. Physiotherapy is required to maintain muscle strength.
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Haber P, Moro PL, Ng C, Dores GM, Perez-Vilar S, Marquez PL, Cano M. Safety review of tetanus toxoid, reduced diphtheria toxoid, acellular pertussis vaccines (Tdap) in adults aged ≥65 years, Vaccine Adverse Event reporting System (VAERS), United States, September 2010-December 2018. Vaccine 2019; 38:1476-1480. [PMID: 31883809 DOI: 10.1016/j.vaccine.2019.11.074] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Academic Contribution Register] [Received: 08/13/2019] [Revised: 11/26/2019] [Accepted: 11/27/2019] [Indexed: 01/10/2023]
Abstract
INTRODUCTION The Advisory Committee on Immunization Practices (ACIP) recommends vaccination with tetanus toxoid, reduced diphtheria toxoid, and acellular pertussis vaccine (Tdap) in persons ≥65 years of age. To date, few studies have assessed the safety of Tdap in this population. We aimed to summarize reports submitted to the Vaccine Adverse Event Reporting System (VAERS) following receipt of Tdap in this age group. METHODS We searched for and analyzed U.S. VAERS reports of Tdap among individuals ≥65 years of age submitted from September 1, 2010 through December 31, 2018. We classified reports according to concurrent vaccination, seriousness, and outcome (death, non-death) and determined the frequency of reported adverse events (AEs). For serious reports, we reviewed available medical records. Data mining analyses were undertaken to detect disproportionality in reporting. RESULTS VAERS received a total of 1,798 reports following Tdap, of which 104 (6%) were serious. The most common AEs were injection site erythema (26%; n = 468), injection site pain (19%; n = 335), injection site swelling (18%; n = 329), and erythema (18%; n = 321). We identified seven deaths; none were attributed to Tdap. Among serious non-death reports, nervous system disorders (35.1%; n = 34) and infections and infestations (n = 18.6%; n = 18) were most commonly reported. Data mining did not identify any vaccine-AE combination reported more frequently than expected. CONCLUSIONS We did not identify any new safety concern over nearly a decade of recommended Tdap use among adults ≥65 years of age. Findings from this post-marketing review are consistent with prior post-marketing observations and pre-licensure studies.
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Affiliation(s)
- Penina Haber
- Immunization Safety Office, Division of Healthcare Quality Promotion, National Center for Emerging and Zoonotic Infectious Diseases, Centers for Disease Control and Prevention, United States.
| | - Pedro L Moro
- Immunization Safety Office, Division of Healthcare Quality Promotion, National Center for Emerging and Zoonotic Infectious Diseases, Centers for Disease Control and Prevention, United States
| | - Carmen Ng
- Immunization Safety Office, Division of Healthcare Quality Promotion, National Center for Emerging and Zoonotic Infectious Diseases, Centers for Disease Control and Prevention, United States
| | - Graça M Dores
- Division of Epidemiology, Office of Biostatistics and Epidemiology, Center for Biologics Evaluation and Research, U.S. Food and Drug Administration, United States
| | - Silvia Perez-Vilar
- Division of Epidemiology, Office of Biostatistics and Epidemiology, Center for Biologics Evaluation and Research, U.S. Food and Drug Administration, United States
| | - Paige L Marquez
- Immunization Safety Office, Division of Healthcare Quality Promotion, National Center for Emerging and Zoonotic Infectious Diseases, Centers for Disease Control and Prevention, United States
| | - Maria Cano
- Immunization Safety Office, Division of Healthcare Quality Promotion, National Center for Emerging and Zoonotic Infectious Diseases, Centers for Disease Control and Prevention, United States
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Sneag DB, Arányi Z, Zusstone EM, Feinberg JH, Queler SC, Nwawka OK, Lee SK, Wolfe SW. Fascicular constrictions above elbow typify anterior interosseous nerve syndrome. Muscle Nerve 2019; 61:301-310. [DOI: 10.1002/mus.26768] [Citation(s) in RCA: 21] [Impact Index Per Article: 3.5] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Academic Contribution Register] [Received: 07/01/2019] [Revised: 11/20/2019] [Accepted: 11/24/2019] [Indexed: 01/28/2023]
Affiliation(s)
- Darryl B. Sneag
- Department of Radiology and ImagingHospital for Special Surgery New York New York
| | | | | | - Joseph H. Feinberg
- Department of Physiatry, Sports MedicineHospital for Special Surgery New York New York
| | - Sophie C. Queler
- Department of Radiology and ImagingHospital for Special Surgery New York New York
| | - O. Kenechi Nwawka
- Department of Radiology and ImagingHospital for Special Surgery New York New York
| | - Steve K. Lee
- Department of Orthopedic Surgery, Hand, and Upper ExtremityHospital for Special Surgery New York New York
| | - Scott W. Wolfe
- Department of Orthopedic Surgery, Hand, and Upper ExtremityHospital for Special Surgery New York New York
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Jahic D, Marjanovic B, Merkac J, Mirnik N, Babic N. Neuralgic Amyotrophy in a 66-year-old Hiker: a Case Report. Mater Sociomed 2019; 31:224-226. [PMID: 31762709 PMCID: PMC6853743 DOI: 10.5455/msm.2019.31.224-226] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Academic Contribution Register] [Indexed: 11/03/2022] Open
Abstract
Introduction Neuralgic amyotrophy (NA) or Parsonage Turner syndrome is a clinical syndrome characterized by sudden attack of neuropathic pain, motor weakness and sensory loss that could be more or less clinically present. Different interpretations regarding the differential diagnosis, symptoms, cause and treatment were given till now. Aim We report our experience with a 66-year-old male who had a sudden pain attack and palsy in shoulder region, without sensory loss, and associated calcific tendinitis of rotator cuff and degenerative changes in cervical spine. Case report Patient came to our hospital with strong pain in shoulder area and signs of frozen shoulder. Active abduction and anteflexion was only to 30 degrees. He experienced the intense pain with visual analogue scale (VAS) 10/10 a night before, during his sleep. No trauma. Neurontin (gabapentin) was given to the patient. After 4 days, he felt better with abduction and anteflexion to 90 degrees. After 2 weeks VAS was 3/10, abduction and anteflection to 100 degrees. Conclusion Neuralgic amyotrophy (NA) is a self-limiting inflammatory disorder usually with idiopathic etiology. The condition can be challenging to treat since many associated symptoms and diagnostic tests and procedures may mimic NA. In that case, accurate differential diagnosis is essential.
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Affiliation(s)
- Dzenan Jahic
- Orthopaedics and Traumatology Clinic, University Clinical Center Sarajevo, Sarajevo, Bosnia and Herzegovina.,Faculty of Sport and Physical Education, University of Sarajevo, Sarajevo, Bosnia and Herzegovina
| | | | - Jakob Merkac
- Orthopedic hospital Valdoltra, Ankaran, Slovenia
| | - Nino Mirnik
- Orthopedic hospital Valdoltra, Ankaran, Slovenia
| | - Nermina Babic
- Department of Physiology, Faculty of Medicine, University of Sarajevo, Sarajevo, Bosnia and Herzegovina
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Maharaj N, Cockcroft DW. Reversible bilateral phrenic nerve paralysis. Respir Med Case Rep 2019; 28:100953. [PMID: 31681533 PMCID: PMC6818340 DOI: 10.1016/j.rmcr.2019.100953] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Academic Contribution Register] [Received: 06/10/2019] [Revised: 10/15/2019] [Accepted: 10/15/2019] [Indexed: 12/02/2022] Open
Abstract
Bilateral phrenic nerve paralysis is a rare potentially life-threatening condition which is usually due to trauma (including surgery) or neurologic disease. We present a patient with apparent rapid onset bilateral phrenic nerve paralysis whose primary symptom was severe positional (supine) dyspnea with profound supine oxygen desaturation. Nerve conduction study abnormalities of the phrenic nerves and some left brachial plexus nerves suggested a diagnosis of ALS. He was treated with supportive night time ventilatory assistance (BiPAP) and over 4 years his condition recovered essentially completely. In retrospect the most likely diagnosis was a rare brachial plexopathy referred to as neuralgic amyotrophy.
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Affiliation(s)
- Neil Maharaj
- Division of Respirology, Critical Care & Sleep Medicine, Department of Medicine, University of Saskatchewan, Royal University Hospital, 103 Hospital Drive, Saskatoon, Saskatchewan, S7N 0W8, Canada
| | - Donald W Cockcroft
- Division of Respirology, Critical Care & Sleep Medicine, Department of Medicine, University of Saskatchewan, Royal University Hospital, 103 Hospital Drive, Saskatoon, Saskatchewan, S7N 0W8, Canada
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Fousekis FS, Mitselos IV, Christodoulou DK. Extrahepatic manifestations of hepatitis E virus: An overview. Clin Mol Hepatol 2019; 26:16-23. [PMID: 31601068 PMCID: PMC6940480 DOI: 10.3350/cmh.2019.0082] [Citation(s) in RCA: 31] [Impact Index Per Article: 5.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Academic Contribution Register] [Received: 09/03/2019] [Accepted: 09/16/2019] [Indexed: 12/14/2022] Open
Abstract
Hepatitis E virus (HEV) is a significant health problem with approximately 20 million individuals infected annually. HEV infection has been associated with a wide spectrum of extrahepatic manifestations, including neurological, hematological and renal disorders. Guillain-Barré syndrome and neuralgic amyotrophy are the most frequent neurological manifestations. In addition, HEV infection has been observed with other neurological diseases, such as encephalitis, myelitis and Bell’s palsy. Hematologic manifestations include anemia due to glucose-6-phospate dehydrogonase deficiency, autoimmune hemolytic anemia and severe thrombocytopenia. Membranoproliferative glomerulonephritis and relapse IgA nephropathy with or without coexisting cryoglobulinemia appear to be the most common renal injuries related with HEV infection. Also, HEV infection has been associated with acute pancreatitis and other immune-mediated manifestations, such as arthritis and myocarditis. However, the pathophysiologic mechanisms of HEV-related extrahepatic manifestations are still largely unclear.
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Affiliation(s)
- Fotios S Fousekis
- Department of Gastroenterology and Hepatology, University Hospital of Ioannina, School of Health Sciences, University of Ioannina, Ioannina, Greece
| | - Ioannis V Mitselos
- Department of Gastroenterology and Hepatology, University Hospital of Ioannina, School of Health Sciences, University of Ioannina, Ioannina, Greece
| | - Dimitrios K Christodoulou
- Department of Gastroenterology and Hepatology, University Hospital of Ioannina, School of Health Sciences, University of Ioannina, Ioannina, Greece
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Serin HM, Yılmaz S, Kanmaz S, Şimşek E, Aktan G, Tekgül H, Gökben S. A rare cause of brachial plexopathy: hereditary neuralgic amyotrophy. Turk Arch Pediatr 2019; 54:189-191. [PMID: 31619932 PMCID: PMC6776451 DOI: 10.5152/turkpediatriars.2018.5837] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Subscribe] [Academic Contribution Register] [Received: 06/05/2017] [Accepted: 01/24/2018] [Indexed: 11/22/2022]
Abstract
Neuralgic amyotrophy is characterized by recurrent, painful, unilateral neuropathy involving mainly the upper brachial plexus followed by muscle weakness and muscle wasting. There are two forms: idiopathic and hereditary. Hereditary neuralgic amyotrophy is an autosomal dominant disease that is often linked to a mutation of SEPT9, a gene of the Septin family. The phenotypic spectrum of the disease may include hypotelorism, cleft palate, and other minor dysmorphisms. The age of onset is from infancy to adulthood. Hereditary neuralgic amyotrophy can be triggered by external stimuli such as infections, vaccinations, cold, stress, surgery, and strenuous exercise. Here, we report a six-year-old girl who was found to have mutation in the SEPT9 gene when she presented with recurrent attacks of painful brachial plexopathy following vaccinations, and was diagnosed as having hereditary neuralgic amyotrophy.
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Affiliation(s)
- Hepsen Mine Serin
- Division of Pediatric Neurology, Department of Pediatrics, Ege University Faculty of Medicine, İzmir, Turkey
| | - Sanem Yılmaz
- Division of Pediatric Neurology, Department of Pediatrics, Ege University Faculty of Medicine, İzmir, Turkey
| | - Seda Kanmaz
- Division of Pediatric Neurology, Department of Pediatrics, Ege University Faculty of Medicine, İzmir, Turkey
| | - Erdem Şimşek
- Division of Pediatric Neurology, Department of Pediatrics, Ege University Faculty of Medicine, İzmir, Turkey
| | - Gül Aktan
- Division of Pediatric Neurology, Department of Pediatrics, Ege University Faculty of Medicine, İzmir, Turkey
| | - Hasan Tekgül
- Division of Pediatric Neurology, Department of Pediatrics, Ege University Faculty of Medicine, İzmir, Turkey
| | - Sarenur Gökben
- Division of Pediatric Neurology, Department of Pediatrics, Ege University Faculty of Medicine, İzmir, Turkey
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Njabom CN, Gilbert A, Brasseur E, Zandona R, Ghuysen A, D'Orio V. Parsonage-Turner Syndrome as a Rare Extrahepatic Complication of Hepatitis E Infection. Eur J Case Rep Intern Med 2019; 6:001208. [PMID: 31508389 PMCID: PMC6726346 DOI: 10.12890/2019_001208] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Subscribe] [Academic Contribution Register] [Received: 07/17/2019] [Accepted: 07/26/2019] [Indexed: 01/12/2023] Open
Abstract
Parsonage-Turner syndrome, also known as neuralgic amyotrophy, is a rare disorder characterized by painful clinical manifestations mainly involving the upper limbs. This syndrome seems to be triggered, among other factors, by some viral infections, although its pathophysiology remains unclear. Moreover, it has rarely been related to hepatitis E virus infection. We report the case of a 33-year-old man who was diagnosed with Parsonage-Turner syndrome following acute hepatitis E infection. LEARNING POINTS Parsonage-Turner syndrome is a painful and disabling condition.Hepatitis E infection can lead to extra-hepatic manifestations such as neurological complications.The association of Parsonage-Turner syndrome with hepatitis E infection is rare but some cases have been reported previously in the literature.
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Affiliation(s)
- Claude Ngah Njabom
- C.H.U. Liège, Emergency Department, site Notre Dame des Bruyères, Chênée, Belgium
| | - Allison Gilbert
- C.H.U. Liège, Emergency Department, site Notre Dame des Bruyères, Chênée, Belgium
| | - Edmond Brasseur
- C.H.U. Liège, Emergency Department, site Notre Dame des Bruyères, Chênée, Belgium
| | - Régine Zandona
- C.H.U. Liège, Emergency Department, site Sart Tilman, Angleur, Belgium
| | - Alexandre Ghuysen
- C.H.U. Liège, Emergency Department, site Sart Tilman, Angleur, Belgium
| | - Vincent D'Orio
- C.H.U. Liège, Emergency Department, site Sart Tilman, Angleur, Belgium
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