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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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Detalles Bibliográficos
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
Descripción
Sumario: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.