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Titolo:
Cutaneous manifestations of maternal engraftment in patients with severe combined immunodeficiency: a clinicopathologic study
Autore:
Denianke, KS; Frieden, IJ; Cowan, MJ; Williams, ML; McCalmont, TH;
Indirizzi:
Univ Calif San Francisco, Sch Med, San Francisco, CA 94143 USA Univ Calif San Francisco San Francisco CA USA 94143 ancisco, CA 94143 USA Univ Calif San Francisco, Dept Dermatol, San Francisco, CA 94143 USA Univ Calif San Francisco San Francisco CA USA 94143 ancisco, CA 94143 USA Univ Calif San Francisco, Dept Pediat, San Francisco, CA 94143 USA Univ Calif San Francisco San Francisco CA USA 94143 ancisco, CA 94143 USA Univ Calif San Francisco, Dept Pathol, Dermatopathol Sect, San Francisco, CA 94143 USA Univ Calif San Francisco San Francisco CA USA 94143 ancisco, CA 94143 USA
Titolo Testata:
BONE MARROW TRANSPLANTATION
fascicolo: 3, volume: 28, anno: 2001,
pagine: 227 - 233
SICI:
0268-3369(200108)28:3<227:CMOMEI>2.0.ZU;2-Q
Fonte:
ISI
Lingua:
ENG
Soggetto:
BONE-MARROW TRANSPLANTATION; VERSUS-HOST DISEASE; T-CELLS; OMENNS SYNDROME; GRAFT; DIVERSITY; CHIMERISM; DIAGNOSIS;
Keywords:
severe combined immunodeficiency disease (SCID); bone marrow transplant (BMT); graft-versus-host disease (GVHD); maternal engraftment;
Tipo documento:
Article
Natura:
Periodico
Settore Disciplinare:
Clinical Medicine
Life Sciences
Citazioni:
17
Recensione:
Indirizzi per estratti:
Indirizzo: Frieden, IJ Univ Calif San Francisco, Sch Med, San Francisco, CA 94143 USAUniv Calif San Francisco San Francisco CA USA 94143 94143 USA
Citazione:
K.S. Denianke et al., "Cutaneous manifestations of maternal engraftment in patients with severe combined immunodeficiency: a clinicopathologic study", BONE MAR TR, 28(3), 2001, pp. 227-233

Abstract

SCID is a heterogeneous group of disorders characterized by defective T cell and B cell function. Eczematous and morbilliform eruptions are common, and graft-versus-host disease (GVHD) due to maternal engraftment has been documented. We sought to better characterize SCID-related cutaneous disease observed prior to BMT and to compare the eruption to conventional GVHD. Medical records of 51 patients with SCID treated between 1982 and 1999 were reviewed. Ten of 51 (20%) had rash and evidence of maternal engraftment prior to BMT (study group). Eleven of 51 (22%) had no rash or evidence of engraftment pre-BMT but developed GVHD following transplant (control group). Skin biopsies were available for review for 8/10 of the study group and for 8/11of the control group. Cutaneous findings consisted of a scaling, erythematous maculopapular eruption spread widely over the trunk and extremities, with near-erythroderma in some patients. Microscopically, biopsies from the study group differed significantly from controls. Key differences included parakeratosis (P less than or equal to 0.01), psoriasiform hyperplasia (P less than or equal to 0.04) and spongiosis (P less than or equal to 0.04). The dermatopathologic findings of transplacental GVHD differ from the patternof post-transplant GVHD. A 'psoriasi-form-lichenoid-sponglotic' pattern with necrotic keratinocytes should trigger consideration of SCID and maternalengraftment in the dermatopathologic evaluation of eruptions of infancy.

ASDD Area Sistemi Dipartimentali e Documentali, Università di Bologna, Catalogo delle riviste ed altri periodici
Documento generato il 05/07/20 alle ore 12:28:10