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An Egyptian child with erythromelalgia responding to a new line of treatment: a case report and review of the literature

INTRODUCTION: Erythromelalgia is a rare clinical syndrome characterized by episodic erythema, warmth and intense burning pain, which commonly involves the extremities. For those affected, this disorder may lead to significant long-term morbidity. Unfortunately, to date, no definitive therapy is avai...

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Autores principales: Al-Minshawy, Samir M, El-Mazary, Abdel-Azeem M
Formato: Online Artículo Texto
Lenguaje:English
Publicado: BioMed Central 2014
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC3943265/
https://www.ncbi.nlm.nih.gov/pubmed/24568362
http://dx.doi.org/10.1186/1752-1947-8-69
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author Al-Minshawy, Samir M
El-Mazary, Abdel-Azeem M
author_facet Al-Minshawy, Samir M
El-Mazary, Abdel-Azeem M
author_sort Al-Minshawy, Samir M
collection PubMed
description INTRODUCTION: Erythromelalgia is a rare clinical syndrome characterized by episodic erythema, warmth and intense burning pain, which commonly involves the extremities. For those affected, this disorder may lead to significant long-term morbidity. Unfortunately, to date, no definitive therapy is available. This case report describes an Egyptian child with primary erythromelalgia that manifested at an early age and showed partial response to therapy with cetirizine hydrochloride. This anecdotal case report may have a diagnostic value for clinicians who have not seen this disorder. CASE PRESENTATION: A 34-month-old previously healthy right-handed Hamitic boy without any significant past medical history presented at the age of 2 years with episodic bilateral pain in his feet. His mother reported associated warmth and erythema localized to his feet that never extended beyond his ankle joints. This pain is triggered by exertion and/or warm temperature exposure and is relieved by cooling measures. The diagnosis of erythromelalgia was made based on the patient’s medical history and a thorough physical examination during the episodes. No evidence of local or systemic infection was present. Other causes for the symptoms were excluded by a negative extensive diagnostic work-up. Our patient did not respond to ibuprofen (15mg/kg/dose) three times a day but partial improvement with the oral non-sedating antihistaminic cetirizine hydrochloride (2.5mg/kg/once daily) was observed. When the child stopped cetirizine hydrochloride for 1 month as a test, the symptoms became aggravated and were relieved when cetirizine therapy was restarted. Cetirizine hydrochloride had not previously been reported to have this effect in children with erythromelalgia. CONCLUSIONS: Erythromelalgia is a clinical syndrome of which the etiology, diagnosis and management are controversial. We describe a case of a 34-month-old Egyptian child with primary erythromelalgia that manifested at an early age. We believe that this is the first Egyptian case report of this kind in the literature. Partial response of this patient to cetirizine hydrochloride may grant us a new clue to understanding this mysterious condition.
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spelling pubmed-39432652014-03-06 An Egyptian child with erythromelalgia responding to a new line of treatment: a case report and review of the literature Al-Minshawy, Samir M El-Mazary, Abdel-Azeem M J Med Case Rep Case Report INTRODUCTION: Erythromelalgia is a rare clinical syndrome characterized by episodic erythema, warmth and intense burning pain, which commonly involves the extremities. For those affected, this disorder may lead to significant long-term morbidity. Unfortunately, to date, no definitive therapy is available. This case report describes an Egyptian child with primary erythromelalgia that manifested at an early age and showed partial response to therapy with cetirizine hydrochloride. This anecdotal case report may have a diagnostic value for clinicians who have not seen this disorder. CASE PRESENTATION: A 34-month-old previously healthy right-handed Hamitic boy without any significant past medical history presented at the age of 2 years with episodic bilateral pain in his feet. His mother reported associated warmth and erythema localized to his feet that never extended beyond his ankle joints. This pain is triggered by exertion and/or warm temperature exposure and is relieved by cooling measures. The diagnosis of erythromelalgia was made based on the patient’s medical history and a thorough physical examination during the episodes. No evidence of local or systemic infection was present. Other causes for the symptoms were excluded by a negative extensive diagnostic work-up. Our patient did not respond to ibuprofen (15mg/kg/dose) three times a day but partial improvement with the oral non-sedating antihistaminic cetirizine hydrochloride (2.5mg/kg/once daily) was observed. When the child stopped cetirizine hydrochloride for 1 month as a test, the symptoms became aggravated and were relieved when cetirizine therapy was restarted. Cetirizine hydrochloride had not previously been reported to have this effect in children with erythromelalgia. CONCLUSIONS: Erythromelalgia is a clinical syndrome of which the etiology, diagnosis and management are controversial. We describe a case of a 34-month-old Egyptian child with primary erythromelalgia that manifested at an early age. We believe that this is the first Egyptian case report of this kind in the literature. Partial response of this patient to cetirizine hydrochloride may grant us a new clue to understanding this mysterious condition. BioMed Central 2014-02-25 /pmc/articles/PMC3943265/ /pubmed/24568362 http://dx.doi.org/10.1186/1752-1947-8-69 Text en Copyright © 2014 Al-Minshawy and ; licensee BioMed Central Ltd.El-Mazary http://creativecommons.org/licenses/by/2.0 This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/2.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly credited.
spellingShingle Case Report
Al-Minshawy, Samir M
El-Mazary, Abdel-Azeem M
An Egyptian child with erythromelalgia responding to a new line of treatment: a case report and review of the literature
title An Egyptian child with erythromelalgia responding to a new line of treatment: a case report and review of the literature
title_full An Egyptian child with erythromelalgia responding to a new line of treatment: a case report and review of the literature
title_fullStr An Egyptian child with erythromelalgia responding to a new line of treatment: a case report and review of the literature
title_full_unstemmed An Egyptian child with erythromelalgia responding to a new line of treatment: a case report and review of the literature
title_short An Egyptian child with erythromelalgia responding to a new line of treatment: a case report and review of the literature
title_sort egyptian child with erythromelalgia responding to a new line of treatment: a case report and review of the literature
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC3943265/
https://www.ncbi.nlm.nih.gov/pubmed/24568362
http://dx.doi.org/10.1186/1752-1947-8-69
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