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

Abstract

TS of immunosuppression is a rare, disfiguring dermatologic condition caused by TS-associated polyomavirus in immunosuppressed patients. It is difficult to treat, with no clearly described approach to resolve the condition completely and safely. We report a child with a renal transplant who developed TS and was treated with significant reduction in immunosuppression and transient use of cidofovir cream. The combined approach, primarily with significant long-term reduction in immunosuppression guided by monitoring BK viremia in our patient, led to complete resolution of TS without recurrence or graft rejection by 5 years after transplant. This outcome was superior to all other reports of TS in children after transplantation. Closely monitoring for BK viremia, as a surrogate marker of over-immunosuppression, can guide adjustment in immunosuppressant medication to treat polyomavirus disease without developing the complication of graft rejection in a patient at significant risk.

Primary trichodysplasia spinulosa polyomavirus infection in a kidney transplant child displaying virus‐infected decoy cells in the urine

“…Intriguingly, only those patients presenting with classic TS display high TSPyV skin prevalence and viral loads in both skin swabs and spines. In this regard, the evidence so far gathered from a very small‐sized international cohort (<40 TS patients) seems to indicate that TS could be a manifestation of primary TSPyV infection during deep immunosuppression …”

Trichodysplasia spinulosa mimicking lichen nitidus in a renal transplant patient

“…TS typically presents on the central face and ears with multiple skin‐colored, folliculocentric papules and central keratotic spicules. The additional presence of the rash on the trunk, arms, and legs has also been reported . Skin lesions are usually asymptomatic, but are occasionally associated with mild pruritus.…”

“…Characteristic histopathologic changes, electron microscopy, or PCR of either a biopsy sample or skin scraping can confirm the diagnosis of TS . Histologically, TS is characterized by dilated hair follicles with proliferation of the inner root sheath cells and the presence of cytoplasmic eosinophilic inclusions consisting of trichohyalin . Electron microscopy can be used to confirm the diagnosis by identifying intranuclear viral inclusions within the hair follicle keratinocytes .…”

“…Human polyomaviruses are ubiquitous and include BK and JC viruses, which are known to cause severe disease in immunocompromised individuals . In fact, BK viremia is a serious infectious cause of kidney transplant dysfunction and can lead to graft loss .…”

“…The presentation of TS is therefore considered a condition of immunosuppression. Given the current difficulty of measuring the TSPyV viral load, it has been proposed that BK viral loads may be used as a surrogate marker for TSPyV viral load …”