Taylor Benoit1, Rocky Bacelieri, Dean S Morrell, John Metcalf. 1. Department of Dermatology and Dermatologic Surgery, Medical University of South Carolina, 135 Rutledge Ave., Charleston, SC 29425, USA. benoitm@musc.edu
Abstract
BACKGROUND: Viral-associated trichodysplasia of immunosuppression is an increasingly recognized entity characterized by follicular-based papules, primarily in the central part of the face, that produce variable degrees of alopecia and dysmorphic features. It has been primarily described in transplant recipients but has recently been recognized in patients receiving chemotherapy for leukemia and lymphoma. It is associated with distinctive histologic features such as dilated anagen hair follicles, absent hair papillae, and abrupt cornification of the inner root sheath. OBSERVATIONS: A 5-year-old boy presented with spiny follicular papules that caused thickening of the skin of the face 1 year after cardiac transplantation. He had been exposed to several immunosuppressive agents, including mycophenolate mofetil, tacrolimus, intravenous immunoglobulin, rituximab, cylcophosphamide, and prednisone. Despite the failure of multiple topical treatments, our patient's eruption improved with systemic valganciclovir therapy. CONCLUSIONS: We describe the youngest patient (to our knowledge) with viral-associated trichodysplasia of immunosuppression and discuss the characteristic clinicopathologic features. Our report supports the theory that immunosuppression is the predisposing factor to a folliculotropic papovavirus that alters follicular maturation.
BACKGROUND: Viral-associated trichodysplasia of immunosuppression is an increasingly recognized entity characterized by follicular-based papules, primarily in the central part of the face, that produce variable degrees of alopecia and dysmorphic features. It has been primarily described in transplant recipients but has recently been recognized in patients receiving chemotherapy for leukemia and lymphoma. It is associated with distinctive histologic features such as dilated anagen hair follicles, absent hair papillae, and abrupt cornification of the inner root sheath. OBSERVATIONS: A 5-year-old boy presented with spiny follicular papules that caused thickening of the skin of the face 1 year after cardiac transplantation. He had been exposed to several immunosuppressive agents, including mycophenolate mofetil, tacrolimus, intravenous immunoglobulin, rituximab, cylcophosphamide, and prednisone. Despite the failure of multiple topical treatments, our patient's eruption improved with systemic valganciclovir therapy. CONCLUSIONS: We describe the youngest patient (to our knowledge) with viral-associated trichodysplasia of immunosuppression and discuss the characteristic clinicopathologic features. Our report supports the theory that immunosuppression is the predisposing factor to a folliculotropic papovavirus that alters follicular maturation.
Authors: Mark R Matthews; Richard C Wang; Robert L Reddick; Victor A Saldivar; John C Browning Journal: J Cutan Pathol Date: 2011-01-19 Impact factor: 1.587
Authors: Els van der Meijden; Siamaque Kazem; Manda M Burgers; Rene Janssens; Jan Nico Bouwes Bavinck; Hester de Melker; Mariet C W Feltkamp Journal: Emerg Infect Dis Date: 2011-08 Impact factor: 6.883
Authors: Siamaque Kazem; Els van der Meijden; Richard C Wang; Arlene S Rosenberg; Elena Pope; Taylor Benoit; Philip Fleckman; Mariet C W Feltkamp Journal: PLoS One Date: 2014-10-07 Impact factor: 3.240