Allogeneic bone marrow transplantation in a 7-year-old girl with congenital erythropoietic porphyria: a treatment dilemma

Br J Dermatol. 2007 Mar;156(3):567-71. doi: 10.1111/j.1365-2133.2006.07699.x.

Abstract

Congenital erythropoietic porphyria (CEP, Günther's disease) has a very variable phenotype. In the more severely affected, bone marrow transplantation (BMT) is potentially curative, but is not without risks. We describe a 7-year-old girl with CEP characterized by severe photosensitivity but only mild anaemia, in whom the difficult decision to proceed with allogeneic BMT was made after discussion in a multidisciplinary team. She has shown successful engraftment, accompanied by biochemical and clinical resolution of her metabolic disease. She remains well 3 years later, the oldest patient with CEP receiving BMT to survive beyond 12 months. However, she has experienced significant morbidity including florid cutaneous graft-versus-host disease with postinflammatory hypopigmentation. Her case is important in highlighting the delay in diagnosis not uncommon in this condition and the complex decision-making process involved in proceeding with BMT.

Publication types

  • Case Reports

MeSH terms

  • Bone Marrow Transplantation* / adverse effects
  • Child
  • Female
  • Graft vs Host Disease / etiology
  • Graft vs Host Disease / pathology
  • Humans
  • Hypopigmentation / etiology
  • Hypopigmentation / pathology
  • Porphyria, Erythropoietic / diagnosis
  • Porphyria, Erythropoietic / pathology
  • Porphyria, Erythropoietic / therapy*