Specific Detection of Trichodysplasia Spinulosa–Associated Polyomavirus DNA in Skin and Renal Allograft Tissues in a Patient With Trichodysplasia Spinulosa

Abstract

Background: Trichodysplasia spinulosa (TS) is a rare, disfiguring skin condition that affects immunosuppressed patients, universally involving the central face. New data point to the recently discovered TS-associated polyomavirus (TSPyV) as the causative agent. Observations:We report a case of TS in a 48-year-old African American man after renal transplant; via polymerase chain reaction and sequencing, confirm the detection of TSPyV in lesional skin; and report the novel detection of TSPyV DNA in renal allograft tissue. Results of polymerase chain reaction analysis were negative for Merkel cell polyomavirus in lesional skin. Fifteen months later, urine cytologic findings showed morphologic evidence of a urinary tract polyomavirus infection. Results of SV40 immunohistochemical analysis were negative in lesional skin, renal allograft, and urine specimens. Conclusions:To our knowledge, this is the first reported case in which TSPyV DNA has been detected in extracutaneous tissues and the third with combined ultrastructural and molecular confirmation of the presence of TSPyV in lesional skin. Lack of detection of other pathogenic human polyomaviruses in this patient's skin supports the specific role of this polyomavirus in the genesis of TS. Further basic science studies are needed to determine the exact pathomechanisms of this polyomavirus and to explore possible tumorigenic roles in other skin diseases.

The trichodysplasia spinulosa‐associated polyomavirus: virological background and clinical implications

“…Interestingly, in a recent study, TSPyV DNA was detected in stool and nasopharyngeal swabs of a 13-year-old immunocompromised heart transplant patient 1 month after immunosuppression showing no TS symptoms, but repeated analysis in the following months remained negative (92). Occasionally also urine and kidney samples were found positive for TSPyV DNA (87). The meaning of these findings, in particular their role in TS diagnosis, remains unclear at the moment.…”

Tricodisplasia espinulosa asociada a síndrome mielodisplásico e inmunosupresión

“…Lamentablemente, en nuestro país no No existe un tratamiento establecido para TS, aunque se ha visto mejoría clínica con aplicación de cidofovir tópico al 1% o 3% (no disponible en nuestro país) y valganciclovir oral, siendo una terapia de alto costo. También podría ser efectivo el ajuste de esquema inmunosupresor en el caso de pacientes trasplantados 9 .…”

Trichodysplasia Spinulosa in a 20-Month-Old Girl With a Good Response to Topical Cidofovir 1%

“…10 There are other less typical histologic findings that include multiple small hair shafts or hair shaft-like material within affected follicles, vacuolated keratinocytes with pyknotic nuclei and coarse keratohyalin granules in the upper layers of the perifollicular epithelium, gray-blue cytoplasmic material in the proliferative inner root-sheath cells, and focal perifolliculitis and a sparse lymphocytic interface dermatitis. 10 Correlation between skin biopsy and clinical features is required for definitive diagnosis. However, many times the diagnosis may be missed if the sample does not contain anagenphase follicles that demonstrate characteristic features.…”

“…Topical steroids, topical tacrolimus, topical antibiotics, imiquimod cream, keratolytics, benzyl peroxide, and minocycline are ineffective. Oral valganciclovir hydrochloride and 1% to 3% topical cidofovir have been reported to be efficacious 3,5,10,11,[13][14][15] Differential diagnoses for TS would include other digitate keratotic conditions, as multiple minute digitate hyperkeratoses, palmoplantar filiform hyperkeratosis, paraneoplastic facial hyperkeratotic spicules associated plasma cell dyscrasias, lichen spinulosus, spiny…”

“…6,7 The overall disease course of TS is somewhat unpredictable 10 : some cases of TS fully resolve after months to years, whereas others are reported with only slight improvement even with different treatments.…”