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Congenital self-healing reticulohistiocytosis in a newborn: unusual oral and cutaneous manifestations

BACKGROUND: Congenital self-healing reticulohistiocytosis (CSHRH), also called Hashimoto-Pritzker disease, is a rare and benign variant of Langerhans cell histiocytosis, characterized by cutaneous lesions without extracutaneous involvement. CASE PRESENTATION: We present a case of CSHRH with diffuse...

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Autores principales: Rizzoli, Alessandra, Giancristoforo, Simona, Haass, Cristina, De Vito, Rita, Gaspari, Stefania, Scapillati, Eleonora, Diociaiuti, Andrea, El Hachem, May
Formato: Online Artículo Texto
Lenguaje:English
Publicado: BioMed Central 2021
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8194160/
https://www.ncbi.nlm.nih.gov/pubmed/34112212
http://dx.doi.org/10.1186/s13052-021-01082-9
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author Rizzoli, Alessandra
Giancristoforo, Simona
Haass, Cristina
De Vito, Rita
Gaspari, Stefania
Scapillati, Eleonora
Diociaiuti, Andrea
El Hachem, May
author_facet Rizzoli, Alessandra
Giancristoforo, Simona
Haass, Cristina
De Vito, Rita
Gaspari, Stefania
Scapillati, Eleonora
Diociaiuti, Andrea
El Hachem, May
author_sort Rizzoli, Alessandra
collection PubMed
description BACKGROUND: Congenital self-healing reticulohistiocytosis (CSHRH), also called Hashimoto-Pritzker disease, is a rare and benign variant of Langerhans cell histiocytosis, characterized by cutaneous lesions without extracutaneous involvement. CASE PRESENTATION: We present a case of CSHRH with diffuse skin lesions and erosions in the oral mucosa, present since birth and lasting for 2 months, and we perform a review of the literature on Pubmed in the last 10 years. CONCLUSIONS: Our case confirm that lesions on oral mucosa, actually underestimated, may be present in patients with CSHRH. Patients affected by CSHRH require a close follow-up until the first years of life, due to the unpredictable course of Langerhans cell histiocytosis, in order to avoid missing diagnosis of more aggressive types of this disorder.
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spelling pubmed-81941602021-06-15 Congenital self-healing reticulohistiocytosis in a newborn: unusual oral and cutaneous manifestations Rizzoli, Alessandra Giancristoforo, Simona Haass, Cristina De Vito, Rita Gaspari, Stefania Scapillati, Eleonora Diociaiuti, Andrea El Hachem, May Ital J Pediatr Case Report BACKGROUND: Congenital self-healing reticulohistiocytosis (CSHRH), also called Hashimoto-Pritzker disease, is a rare and benign variant of Langerhans cell histiocytosis, characterized by cutaneous lesions without extracutaneous involvement. CASE PRESENTATION: We present a case of CSHRH with diffuse skin lesions and erosions in the oral mucosa, present since birth and lasting for 2 months, and we perform a review of the literature on Pubmed in the last 10 years. CONCLUSIONS: Our case confirm that lesions on oral mucosa, actually underestimated, may be present in patients with CSHRH. Patients affected by CSHRH require a close follow-up until the first years of life, due to the unpredictable course of Langerhans cell histiocytosis, in order to avoid missing diagnosis of more aggressive types of this disorder. BioMed Central 2021-06-10 /pmc/articles/PMC8194160/ /pubmed/34112212 http://dx.doi.org/10.1186/s13052-021-01082-9 Text en © The Author(s) 2021 https://creativecommons.org/licenses/by/4.0/Open AccessThis article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article's Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article's Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/ (https://creativecommons.org/licenses/by/4.0/) . The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/ (https://creativecommons.org/publicdomain/zero/1.0/) ) applies to the data made available in this article, unless otherwise stated in a credit line to the data.
spellingShingle Case Report
Rizzoli, Alessandra
Giancristoforo, Simona
Haass, Cristina
De Vito, Rita
Gaspari, Stefania
Scapillati, Eleonora
Diociaiuti, Andrea
El Hachem, May
Congenital self-healing reticulohistiocytosis in a newborn: unusual oral and cutaneous manifestations
title Congenital self-healing reticulohistiocytosis in a newborn: unusual oral and cutaneous manifestations
title_full Congenital self-healing reticulohistiocytosis in a newborn: unusual oral and cutaneous manifestations
title_fullStr Congenital self-healing reticulohistiocytosis in a newborn: unusual oral and cutaneous manifestations
title_full_unstemmed Congenital self-healing reticulohistiocytosis in a newborn: unusual oral and cutaneous manifestations
title_short Congenital self-healing reticulohistiocytosis in a newborn: unusual oral and cutaneous manifestations
title_sort congenital self-healing reticulohistiocytosis in a newborn: unusual oral and cutaneous manifestations
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8194160/
https://www.ncbi.nlm.nih.gov/pubmed/34112212
http://dx.doi.org/10.1186/s13052-021-01082-9
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