Viremia and viruria of trichodysplasia spinulosa‐associated polyomavirus before the development of clinical disease in a kidney transplant recipient

Abstract

Trichodysplasia spinulosa (TS) is a rare disease associated with immunosuppression and induced by a polyomavirus denominated Tricodisplasia Polyomavirus (TSPyV). We report a case of TS 6 months after kidney transplantation in a 65 years-old woman under immunosuppression therapy with prednisone, mycophenolate and tacrolimus. The patient developed follicular papules on the face with a thickening of the skin and alopecia of the eyebrows, leading to distortion of the face and a leonine appearance characteristic of the disease. The skin biopsy confirmed the clinical diagnosis and the presence of TSPyV DNA in the skin was detected. Staining for SV40 was positive. Immunosuppression was changed: mycophenolate was withdrawn, tacrolimus reduced and everolimus added. Intravenous cidofovir and later on leflunomide were added. Although the literature has reported clinical success with topical cidofovir, we were unable to use it because this drug is not available. There was an improvement of skin lesions and on cosmetic appearance. The patient had three rejections (one clinically diagnosed and two other biopsy proven), progressed with renal failure and graft loss. Retrospective analysis of stored urine and blood samples detected TSPyV DNA in some of those samples two months before the TS clinical development. This case highlights the TSPyV detection in blood and urine samples before the development of skin lesions. K E Y W O R D S kidney transplant, polyomavirus, trichodysplasia spinulosa, viremia, viruria

Sustained Trichodysplasia Spinulosa Polyomavirus Viremia Illustrating a Primary Disseminated Infection in a Kidney Transplant Recipient

“…TSPyV has been detected via PCR in skin specimens [9][10][11][12][13] from transplant patients, and replicative virions have been identified with electronic microscopy and immunohistochemistry [9]. The TSPyV genome has also been detected in skin specimens [11] and tonsillar tissues of healthy individuals [14], in skin specimens [3], stool samples and nasal swabs of asymptomatic transplant recipients [15] and in urine samples of a kidney transplant recipient with TS [16], as well as in a kidney allograft biopsy specimen of a kidney transplant recipient [17]. Our observation of viremia preceding the development of skin lesions by a few months confirmed previous reports [13,[16][17][18].…”

Recent developments in trichodysplasia spinulosa disease

“…5 The detection of TSPyV DNA in nasopharyngeal and fecal/urine samples from asymptomatic individuals further supports the theory of a latent viral infection through a respiratory route of transmission. 7 In 2019, a retrospective study by Pierrotti et al 8…”

“…49 Additionally, we report one case of a renal transplant patient who experienced transplant rejection and graft failure after reduction of immunosuppression, highlighting the risk of reducing immunosuppression specifically in organ transplant TS patients (Table 1). 8 Physical extraction of keratin spicules showed significant improvement in a pediatric patient, suggesting a potential cost-effective and safe alternative treatment option, especially for patients who may not be able to undergo reduction of immunosuppressive regimens (Table 1). 50 Interestingly, the previous review highlighted a link between mycophenolate mofetil immunosuppressive exposure and TS disease progression, without a clear explanation.…”

“…5 We provide further evidence of this relationship, as 10 cases reviewed in Table 1 were exposed to mycophenolate mofetil before TS development, and reducing mofetil treatment showed significant improvement in TS disease in 5 of these patients (Table 1). 6,8,11,27,41,49,[51][52][53] Thus, the mechanistic link between mycophenolate mofetil and TS disease development should be further investigated.…”

“…In 2019, a retrospective study by Pierrotti et al 8 indicated that TSPyV DNA was present in blood and urine samples 2 months before TS clinical development in a kidney transplant patient (Table 1). Urbano et al used real‐time PCR (qPCR) to detect and quantify TSPyV in blood samples for the first time.…”