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

Abstract

Kazem S, van der Meijden E, Feltkamp MCW. The trichodysplasia spinulosa-associated polyomavirus: virological background and clinical implications. APMIS 2013; 121: 770-82.Trichodysplasia spinulosa-associated polyomavirus (TSPyV) is a new species of the family Polyomaviridae that was discovered in 2010. TSPyV infects humans and is associated with the development of a rare disease called trichodysplasia spinulosa. Trichodysplasia spinulosa is a skin disease of severely immunocompromised hosts characterized by follicular distention and keratotic spine formation especially on the face. Electron microscopy, immunohistochemistry, and viral load measurements indicate an etiological role of active TSPyV infection in the development of this disease. This review will address virological and pathogenic properties of TSPyV, as well as epidemiologic, diagnostic, and therapeutic aspects of TSPyV infection.

Seroreactivity against Merkel cell polyomavirus and other polyomaviruses in chronic lymphocytic leukaemia, the MCC-Spain study

“…However, JCPyV and TSPyV, both reported as opportunistic diseases in subjects with haematologic disorders (Amend et al, 2010;Kazem et al, 2013), showed higher seroprevalences at lower seroreactivity levels among treated subjects when compared to untreated ones. This effect might be explained by a sustained poor immune response independent of treatment status.…”

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

“…2,4,11,14 TS is an emerging entity in the setting of immunosuppression. Given the number of immunocompromised patients, and the likely high prevalence of the causative virus, as seroepidemiological studies indicate that TS-associated polyomavirus is ubiquitous and latently infects 70% of the healthy individuals in adulthood, 13,16,17 we believe that TS is probably underdiagnosed, possibly leading to delays in diagnosis. As organ transplantation and immunosuppression are becoming more common, the frequency of TS could increase.…”

“…4 Infection is acquired at an early age without apparent disease and probably persists subclinically. 13 Immunosuppression, alone or in combination with other no known factors, is the predisposing factor that triggers folliculotropic polyomavirus reactivation, and alters follicular maturation. 4,5,8,9,11 , Routine testing with polymerase chain reaction assay, gene-sequencing studies, immunohistochemical stains, or electron microscopy, are not necessary in all cases, as routine histologic staining can be sufficient to make the diagnosis in cases of classic clinical presentations.…”

“…4 Medical treatment options of clinical TS lesions remain limited. 5 The reduction or change of immunosuppressants has demonstrated clinical improvement, 13 whereas topical and oral retinoids are minimally effective. Topical steroids, topical tacrolimus, topical antibiotics, imiquimod cream, keratolytics, benzyl peroxide, and minocycline are ineffective.…”

“…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…”