title: Bone marrow engraftment and associated dermatologic sequelae in a three-yr-old after liver transplantation.
abstract: We present a case of a three-yr-old child with a history of multisystem Langerhans cell histiocytosis treated with systemic chemotherapy, who developed progressive liver failure and received an orthotopic split liver transplant while continuing on chemotherapy. One month following transplant, he developed acute graft-vs.-host disease of the skin and gastrointestinal tract. Peripheral blood chimerism studies post-transplant demonstrated an increasing predominance of donor lymphocytes and granulocytes. Shortly after, the patient developed vitiligo, and two yr after transplantation, the patient developed skin manifestations of psoriasis. We discuss and review the current literature, which demonstrates that chimerism following liver transplantation is rare and in our patient may be related to his profound immunosuppression around the time of liver transplant as well the development of acute graft-versus-host disease. While autoimmune disease can occur after solid organ and stem cell transplant, our patient developed skin manifestations of autoimmunity after liver transplantation, which is also rarely described.
authors.affiliation: Harvard Medical School, Boston, MA, USA.
authors.forename: Johanna
authors.initials: J
authors.lastname: Sheu
authors.forename: Arturo P
authors.initials: AP
authors.lastname: Saavedra
authors.forename: Barbara A
authors.initials: BA
authors.lastname: Degar
authors.forename: Christine N
authors.initials: CN
authors.lastname: Duncan
authors.forename: Rima
authors.initials: R
authors.lastname: Fawaz
authors.forename: Jennifer K
authors.initials: JK
authors.lastname: Tan
authors.forename: Birgitta A
authors.initials: BA
authors.lastname: Schmidt
authors.forename: Heung B
authors.initials: HB
authors.lastname: Kim
authors.forename: Jennifer T
authors.initials: JT
authors.lastname: Huang
mesh_terms: D015551:Autoimmunity; D001706:Biopsy; D001853:Bone Marrow / Q000473:pathology*; D002675:Child, Preschool; D041981:Gastrointestinal Tract / Q000503:physiopathology; D006086:Graft vs Host Disease; D006098:Granulocytes / Q000166:cytology; D015614:Histiocytosis / Q000503:physiopathology; D006801:Humans; D016031:Liver Transplantation / Q000009:adverse effects* / Q000379:methods; D008214:Lymphocytes / Q000166:cytology; D008297:Male; D011183:Postoperative Complications; D011565:Psoriasis / Q000150:complications / Q000503:physiopathology; D012871:Skin Diseases / Q000503:physiopathology*; D018183:Transplantation Chimera; D014820:Vitiligo / Q000150:complications / Q000503:physiopathology
keywords: Langerhans cell histiocytosis; autoimmune; bone marrow engraftment; chimerism; liver transplantation; post-transplant