Italian Journal of Pediatrics (Jun 2021)

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

  • Alessandra Rizzoli,
  • Simona Giancristoforo,
  • Cristina Haass,
  • Rita De Vito,
  • Stefania Gaspari,
  • Eleonora Scapillati,
  • Andrea Diociaiuti,
  • May El Hachem

DOI
https://doi.org/10.1186/s13052-021-01082-9
Journal volume & issue
Vol. 47, no. 1
pp. 1 – 4

Abstract

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Abstract 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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