Viral-Associated Trichodysplasia of Immunosuppression

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.

Complete resolution of trichodysplasia spinulosa in a pediatric renal transplant patient: Case report and literature review

“…Since the first case of TS was published in 1999 by Halcox et al., over 25 cases have been described in both adult and pediatric immunosuppressed patients. There are eight reported cases in pediatric transplant recipients, including this case report, who were diagnosed with TS (Table ). Using electron microscopy, high viral loads were found within the hair follicles in many patients with clinical TS; however, as was the scenario with our patient, other cases did not demonstrate viral particle inclusions despite classical clinical presentation of TS …”

“…TS is a disfiguring condition and difficult to treat . A variety of approaches have been tried with varying success.…”

Trichodysplasia Spinulosa: Rare Presentation of Polyomavirus Infection in Immunocompromised Patients

“…Systemic valganciclovir and valacyclovir and topical acyclovir and cidofovir have been reported to be effective in treating TS.1--14,11,27,28 Of these antiviral medications, topical cidofovir 1% or 3% cream, applied twice daily, and valganciclovir 900 mg, applied twice daily, have been most com m only used. 6,[12][13][14]19,22,26 Oral valganciclovir is reported to be the best treatment, with patients showing nearly complete resolution. 6,[12][13][14]19,22,26 Other treatments that have been used include topical steroids, topical and systemic antibacterials, topical and systemic retinoids, and topical imiquimod, all of which have either provided minimal benefit or been ineffective.1,5,6,16,20 A small proportion of patients (three of nine reported), experi enced some improvement with the use of topical retinoids (tretinoin or tazarotene).14,16,1"'22 It is unclear if these topical vitamin A derivatives alter the state of viral infection or simply diminish the cutaneous manifestations of TS.…”

Viral-Associated Trichodysplasia

“…Our case demonstrates an impressive, persistent response to topical cidofovir therapy without any adverse effects (Figure 1A and C), a finding that is consistent with prior reports that suggested improvement in 3 patients who used topical cidofovir cream or ointment (1%–3%) 2,4,8 and 1 report in which there was no response to topical cidofovir therapy but there was improvement with systemic valganciclovir therapy. 13 Also, other patients experienced improvement with oral valacyclovir or valganciclovir therapy when being treated for herpes zoster 17 or cytomegalovirus prophylaxis. 12 A mixture of topical acyclovir, 2-deoxy-D-glucose, and epigallocatechin as a cost-effective alternative to topical cidofovir also has been demonstrated to be a moderately effective topical therapy in a single report.…”