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.