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Delayed effort-induced swelling with myofasciitis and systemic manifestations: A so far unrecognized type of pressure-induced urticaria

Diseases characterized by recurrent symptoms with prolonged intervals without any clinical manifestations can pose diagnostic difficulties. Some diagnoses will be obvious but other situations can be very challenging. To nosologically delineate a new entity characterized by recurrent flares of indura...

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Autores principales: Bursztejn, Anne-Claire, Lipsker, Dan
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Wolters Kluwer Health 2017
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5319520/
https://www.ncbi.nlm.nih.gov/pubmed/28207531
http://dx.doi.org/10.1097/MD.0000000000006112
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author Bursztejn, Anne-Claire
Lipsker, Dan
author_facet Bursztejn, Anne-Claire
Lipsker, Dan
author_sort Bursztejn, Anne-Claire
collection PubMed
description Diseases characterized by recurrent symptoms with prolonged intervals without any clinical manifestations can pose diagnostic difficulties. Some diagnoses will be obvious but other situations can be very challenging. To nosologically delineate a new entity characterized by recurrent flares of induration of the forearms and legs with swelling of the extremities accompanied by intense fatigue and variable other symptoms. Retrospective observational study of patients recorded from 2000 to 2015. All patients included were seen during a consultation at the Dermatology Department of the University Hospital of Strasbourg, France. We retrieved the medical records from patients seen and recorded over the last 16 years having induration of the extremities, the forearm and the legs occurring between 4 and 12 hours after a physical effort accompanied by systemic signs that lasted for a few days. We analyzed in detail the clinical and biological features, evolution, and treatments of these patients. We included 6 males, with a mean age of 47 years; mean age at disease onset was 42. All patients were initially misdiagnosed as having rheumatic disorders. The mean delay before diagnosis was 5 years. The main complaint was painful induration or muscle soreness of the forearms and the legs associated with transient functional impairment and prolonged asthenia for a mean duration of 3.5 days. Induration of the deep soft tissues was very suggestive of myofasciitis. The delay between the triggering physical effort and the swelling was between 6 and 12 hours. Physical effort as triggering factor was never spontaneously mentioned. Two patients had partial response to high dose antihistamines and 2 other patients to the interleukin-1 inhibitor anakinra. One patient responded to hydroxychloroquine. The very stereotypical presentation in those 6 patients suggests that this is a recognizable entity characterized by effort-induced induration of forearms and/or legs, due to deep edematous myofascial involvement, occurring a few hours after a physical effort. We suggest to name this entity delayed effort-induced swelling with myofasciitis and systemic manifestations.
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spelling pubmed-53195202017-03-02 Delayed effort-induced swelling with myofasciitis and systemic manifestations: A so far unrecognized type of pressure-induced urticaria Bursztejn, Anne-Claire Lipsker, Dan Medicine (Baltimore) 4000 Diseases characterized by recurrent symptoms with prolonged intervals without any clinical manifestations can pose diagnostic difficulties. Some diagnoses will be obvious but other situations can be very challenging. To nosologically delineate a new entity characterized by recurrent flares of induration of the forearms and legs with swelling of the extremities accompanied by intense fatigue and variable other symptoms. Retrospective observational study of patients recorded from 2000 to 2015. All patients included were seen during a consultation at the Dermatology Department of the University Hospital of Strasbourg, France. We retrieved the medical records from patients seen and recorded over the last 16 years having induration of the extremities, the forearm and the legs occurring between 4 and 12 hours after a physical effort accompanied by systemic signs that lasted for a few days. We analyzed in detail the clinical and biological features, evolution, and treatments of these patients. We included 6 males, with a mean age of 47 years; mean age at disease onset was 42. All patients were initially misdiagnosed as having rheumatic disorders. The mean delay before diagnosis was 5 years. The main complaint was painful induration or muscle soreness of the forearms and the legs associated with transient functional impairment and prolonged asthenia for a mean duration of 3.5 days. Induration of the deep soft tissues was very suggestive of myofasciitis. The delay between the triggering physical effort and the swelling was between 6 and 12 hours. Physical effort as triggering factor was never spontaneously mentioned. Two patients had partial response to high dose antihistamines and 2 other patients to the interleukin-1 inhibitor anakinra. One patient responded to hydroxychloroquine. The very stereotypical presentation in those 6 patients suggests that this is a recognizable entity characterized by effort-induced induration of forearms and/or legs, due to deep edematous myofascial involvement, occurring a few hours after a physical effort. We suggest to name this entity delayed effort-induced swelling with myofasciitis and systemic manifestations. Wolters Kluwer Health 2017-02-17 /pmc/articles/PMC5319520/ /pubmed/28207531 http://dx.doi.org/10.1097/MD.0000000000006112 Text en Copyright © 2017 the Author(s). Published by Wolters Kluwer Health, Inc. http://creativecommons.org/licenses/by-nc-nd/4.0 This is an open access article distributed under the terms of the Creative Commons Attribution-Non Commercial-No Derivatives License 4.0 (CCBY-NC-ND), where it is permissible to download and share the work provided it is properly cited. The work cannot be changed in any way or used commercially without permission from the journal. http://creativecommons.org/licenses/by-nc-nd/4.0
spellingShingle 4000
Bursztejn, Anne-Claire
Lipsker, Dan
Delayed effort-induced swelling with myofasciitis and systemic manifestations: A so far unrecognized type of pressure-induced urticaria
title Delayed effort-induced swelling with myofasciitis and systemic manifestations: A so far unrecognized type of pressure-induced urticaria
title_full Delayed effort-induced swelling with myofasciitis and systemic manifestations: A so far unrecognized type of pressure-induced urticaria
title_fullStr Delayed effort-induced swelling with myofasciitis and systemic manifestations: A so far unrecognized type of pressure-induced urticaria
title_full_unstemmed Delayed effort-induced swelling with myofasciitis and systemic manifestations: A so far unrecognized type of pressure-induced urticaria
title_short Delayed effort-induced swelling with myofasciitis and systemic manifestations: A so far unrecognized type of pressure-induced urticaria
title_sort delayed effort-induced swelling with myofasciitis and systemic manifestations: a so far unrecognized type of pressure-induced urticaria
topic 4000
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5319520/
https://www.ncbi.nlm.nih.gov/pubmed/28207531
http://dx.doi.org/10.1097/MD.0000000000006112
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