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Case Reports
. 2009 Jan;60(1):169-72.
doi: 10.1016/j.jaad.2008.07.051.

Trichodysplasia of immunosuppression treated with oral valganciclovir

Aton M Holzer, Lauren C Hughey
Case Reports

Trichodysplasia of immunosuppression treated with oral valganciclovir

Aton M Holzer et al. J Am Acad Dermatol. 2009 Jan.

Abstract

Trichodysplasia of immunosuppression (TOI) is a newly described rare clinicopathologic entity usually found in transplant patients on chronic immunosuppressive medications or with hematologic malignancies. TOI consists of a cutaneous eruption of spiny papules predominantly affecting the face and arms. The distinct histopathologic picture includes abnormally maturing anagen follicles with hyperkeratotic infundibula and excessive inner root sheath differentiation. Given that intranuclear viral particles have been noted on electron microscopy of skin biopsy in several patients, a viral etiology is suspected. These viral particles are similar in shape and size to a polyoma virus, however, the virus has not been identified to date. There are reports of improvement of TOI with topical cidofovir or tazarotene 0.5% gel, but to our knowledge there has not been a report of near-complete resolution with oral valganciclovir. We report a case of TOI in a heart transplant patient on a chronic immunosuppressive regimen of sirolimus, mycophenolic acid and prednisone that demonstrated almost complete response to treatment with systemic valganciclovir.

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Figures

Figure 1
Figure 1
Clinical image of patient immediately prior to initiation of valganciclovir therapy.
Figure 2
Figure 2
A. Low power view (20X) demonstrating abnormally maturing anagen hair follicles with excessive inner root sheath differentiation, no normal hair papillae and hyperkeratotic, slightly dilated infundibula. B. Intermediate power view (100X) demonstrating the hyperkeratotic infundibula and the absence of mature hair sheaths.
Figure 2
Figure 2
A. Low power view (20X) demonstrating abnormally maturing anagen hair follicles with excessive inner root sheath differentiation, no normal hair papillae and hyperkeratotic, slightly dilated infundibula. B. Intermediate power view (100X) demonstrating the hyperkeratotic infundibula and the absence of mature hair sheaths.
Figure 3
Figure 3
Clinical image of patient one year subsequent to initiation of valganciclovir therapy.
See this image and copyright information in PMC

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