Abstract

A 65-year-old female with multiple myeloma, status-post allogeneic CAR-T cell therapy, presented for an asymptomatic eruption of small papules with keratotic spiculations predominantly affecting her nose. Salicylic acid cleanser was ineffective. Folliculitis was suspected clinically, and a punch biopsy of the left nasal ala was performed. Histologic sections demonstrated enlarged and dilated hair follicles lacking hair shafts, plugging of the follicular infundibula, expansion of the inner root sheath, and enlarged, eosinophilic trichohyaline granules, consistent with a diagnosis of trichodysplasia spinulosa (TS). TS is a rare polyomavirus-associated follicular dermatosis first described in 1995 as an adverse effect of cyclosporine in a kidney transplant recipient. Four years later, a case of TS was reported in a patient with multiple myeloma and thought to represent a cutaneous manifestation of that disease based on the observation that the eosinophilic “deposits” were cryoprecipitates composed of IgG-kappa. The characteristic lesion of TS is a skin-colored papule with a protruding keratin spine, or “spicule”, emanating from the follicular ostium. Lesions are most often found on the central face of immunocompromised patients. Leonine-changes of the face are a rare complication. The causative agent, TS-associated polyomavirus (TSPyV), was first characterized in 2010 and is now known to be ubiquitous. Recent data suggests that TS is caused by a primary TSPyV infection, rather than re-activation of latent virus. Diagnosis can be confirmed with PCR. Reported successful treatments include reduced immunosuppression, cidofovir, tazarotene gel, oral leflunomide, and valganciclovir.

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