Hyperpigmentation Associated with the Use of Topical Cidofovir for Treatment of Trichodysplasia Spinulosa in an Immunosuppressed Adult: Case Report and Review of the Literature
Trichodysplasia spinulosa (TS) is a rare, opportunistic infectious skin disease caused by the polyoma virus. Clinically, TS is characterized by follicular papules, keratin spicules, and alopecia most classically in a midfacial distribution. Since its discovery in 2010, no standard of treatment has been established, though use of oral acitretin, valganciclovir, lefludomide, topical cidofovir, physical extraction, and modification of immunosuppressive medications have been reported in the literature. We describe the case of a 52-year old female with a painful midfacial eruption and alopecia of the bilateral eyebrows ultimately diagnosed with TS and treated with topical cidofovir 3%. Though the TS eruption resolved, treatment resulted in hyperpigmentation of the affected area. Hyperpigmentation associated with cidofovir use has been reported in cases of molluscum contagiosum, however, no such association has been described in the treatment of TS to our knowledge. Therefore, we report this case to highlight an underreported adverse effect of topical cidofovir in the setting of this rare disease.
van der Meijden E, Janssens RW, Lauber C, Bouwes Bavinck JN, Gorbalenya AE, Feltkamp MC. Discovery of a new human polyomavirus associated with trichodysplasia spinulosa in an immunocompromized patient. PLoS pathogens. 2010;6(7):e1001024.
An P, Saenz Robles MT, Duray AM, Cantalupo PG, Pipas JM. Human polyomavirus BKV infection of endothelial cells results in interferon pathway induction and persistence. PLoS pathogens. 2019;15(1):e1007505.
Wieland U, Silling S, Hellmich M, Potthoff A, Pfister H, Kreuter A. Human polyomaviruses 6, 7, 9, 10 and Trichodysplasia spinulosa-associated polyomavirus in HIV-infected men. The Journal of general virology. 2014;95(Pt 4):928-932.
Haycox CL, Kim S, Fleckman P, et al. Trichodysplasia spinulosa--a newly described folliculocentric viral infection in an immunocompromised host. The journal of investigative dermatology Symposium proceedings. 1999;4(3):268-271.
DeCrescenzo AJ, Philips RC, Wilkerson MG. Trichodysplasia spinulosa: A rare complication of immunosuppression. JAAD case reports. 2016;2(4):307-309.
Calista D. Topical cidofovir for severe cutaneous human papillomavirus and molluscum contagiosum infections in patients with HIV/AIDS. A pilot study. Journal of the European Academy of Dermatology and Venereology : JEADV. 2000;14(6):484-488.
Scherrer MA, Rocha VB, Andrade AR. Contact dermatitis to methylisothiazolinone. Anais brasileiros de dermatologia. 2015;90(6):912-914.
Nguyen NV, Burgos A, Prok L. Spiny Papules in an Immunosuppressed Child. Pediatric dermatology. 2015;32(6):e296-297.
Coogle LP, Holland KE, Pan C, Van Why SK. Complete resolution of trichodysplasia spinulosa in a pediatric renal transplant patient: Case report and literature review. Pediatric transplantation. 2017;21(2).
Leitenberger JJ, Abdelmalek M, Wang RC, Strasfeld L, Hopkins RS. Two cases of trichodysplasia spinulosa responsive to compounded topical cidofovir 3% cream. JAAD case reports. 2015;1(6):S33-35.
Santesteban R, Feito M, Mayor A, Beato M, Ramos E, de Lucas R. Trichodysplasia Spinulosa in a 20-Month-Old Girl With a Good Response to Topical Cidofovir 1%. Pediatrics. 2015;136(6):e1646-1649.
Digiovanna JJ, Mauro T, Milstone LM, Schmuth M, Toro JR. Systemic retinoids in the management of ichthyoses and related skin types. Dermatologic therapy. 2013;26(1):26-38.
Kassar R, Chang J, Chan AW, Lilly LB, Al Habeeb A, Rotstein C. Leflunomide for the treatment of trichodysplasia spinulosa in a liver transplant recipient. Transplant infectious disease : an official journal of the Transplantation Society. 2017;19(4).
Holzer AM, Hughey LC. Trichodysplasia of immunosuppression treated with oral valganciclovir. Journal of the American Academy of Dermatology. 2009;60(1):169-172.
Barton M, Lockhart S, Sidbury R, Wang R, Brandling-Bennett H. Trichodysplasia Spinulosa in a 7-Year-Old Boy Managed Using Physical Extraction of Keratin Spicules. Pediatric dermatology. 2017;34(2):e74-e76.
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