Phenotypic variations of cartilage hair hypoplasia: granulomatous skin inflammation and severe T cell immunodeficiency as initial clinical presentation in otherwise well child with short stature

J Clin Immunol. 2014 Jan;34(1):42-8. doi: 10.1007/s10875-013-9962-6. Epub 2013 Nov 12.

Abstract

We report a child with short stature since birth who was otherwise well, presenting at 2.8 years with progressive granulomatous skin lesions when diagnosed with severe T cell immunodeficiency. When previously investigated for short stature, and at the time of current investigations, she had no radiological skeletal features characteristics for cartilage hair hypoplasia, but we found a disease causing RMRP (RNase mitochondrial RNA processing endoribonuclease) gene mutation. Whilst search for HLA matched unrelated donor for haematopoietic stem cell transplantation (HSCT) was underway, she developed rapidly progressive EBV-related lymphoproliferative disorder requiring laparotomy and small bowel resection, and was treated with anti-B cell monoclonal antibody and eventually curative allogeneic HSCT. Screening for RMRP gene mutations should be part of immunological evaluation of patients with 'severe and/or combined' T cell immunodeficiency of unknown origin, especially when associated with short stature and regardless of presence or absence of radiological skeletal features.

Publication types

  • Case Reports

MeSH terms

  • Bone and Bones / diagnostic imaging
  • Child, Preschool
  • Dermatitis / pathology
  • Dwarfism
  • Female
  • Granuloma / pathology
  • Hair / abnormalities*
  • Hematopoietic Stem Cell Transplantation / adverse effects
  • Hirschsprung Disease / diagnosis*
  • Hirschsprung Disease / genetics
  • Hirschsprung Disease / therapy
  • Humans
  • Immunologic Deficiency Syndromes / diagnosis*
  • Immunologic Deficiency Syndromes / genetics
  • Immunologic Deficiency Syndromes / therapy
  • Immunophenotyping
  • Osteochondrodysplasias / congenital*
  • Osteochondrodysplasias / diagnosis
  • Osteochondrodysplasias / genetics
  • Osteochondrodysplasias / therapy
  • Phenotype*
  • Primary Immunodeficiency Diseases
  • Radiography
  • Receptors, Antigen, T-Cell, alpha-beta / metabolism
  • T-Lymphocytes / metabolism
  • Transplantation, Homologous
  • Treatment Outcome

Substances

  • Receptors, Antigen, T-Cell, alpha-beta

Supplementary concepts

  • Cartilage-hair hypoplasia
  • T cell immunodeficiency primary