Trichodysplasia spinulosa is characterized by active polyomavirus infection Journal Article


Authors: Kazem, S.; van der Meijden, E.; Kooijman, S.; Rosenberg, A. S.; Hughey, L. C.; Browning, J. C.; Sadler, G.; Busam, K.; Pope, E.; Benoit, T.; Fleckman, P.; de Vries, E.; Eekhof, J. A.; Feltkamp, M. C. W.
Article Title: Trichodysplasia spinulosa is characterized by active polyomavirus infection
Abstract: Background: Recently a new polyomavirus was identified in a patient with trichodysplasia spinulosa (TS), a rare follicular skin disease of immunocompromised patients characterized by facial spines and overgrowth of inner root sheath cells. Seroepidemiological studies indicate that TSPyV is ubiquitous and latently infects 70% of the healthy individuals. Objective: To corroborate the relationship between active TSPyV infection and TS disease by analyzing the presence, load, and precise localization of TSPyV infection in TS patients and in controls. Study design: TS lesional and non-lesional skin samples were retrieved from TS patients through a PubMed search. Samples were analyzed for the presence and load of TSPyV DNA with quantitative PCR, and for expression and localization of viral protein with immunofluorescence. Findings obtained in TS patients (n= 11) were compared to those obtained in healthy controls (n= 249). Results: TSPyV DNA detection was significantly associated with disease (P<0.001), with 100% positivity of the lesional and 2% of the control samples. Quantification revealed high TSPyV DNA loads in the lesional samples (∼10 6copies/cell), and low viral loads in the occasionally TSPyV-positive non-lesional and control samples (<10 2copies/cell). TSPyV VP1 protein expression was detected only in lesional TS samples, restricted to the nuclei of inner root sheath cells over-expressing trichohyalin. Conclusions: The high prevalence and load of TSPyV DNA only in TS lesions, and the abundant expression of TSPyV protein in the affected hair follicle cells demonstrate a tight relation between TSPyV infection and TS disease, and indicate involvement of active TSPyV infection in TS pathogenesis. © 2011 Elsevier B.V.
Keywords: adolescent; adult; child; clinical article; controlled study; human tissue; preschool child; protein expression; school child; child, preschool; middle aged; unclassified drug; case-control studies; pathogenesis; polymerase chain reaction; protein localization; disease association; skin biopsy; immunofluorescence; skin; chromatin; polyoma virus; dna, viral; polyomavirus; polyomavirus infections; tumor virus infections; skin disease; virus load; histocytochemistry; intermediate filament proteins; virus protein; etiology; virus dna; viral load; polyomavirus infection; capsid proteins; trichodysplasia spinulosa; trichodysplasia spinulosa-associated polyomavirus; trichohyalin; skin diseases, viral
Journal Title: Journal of Clinical Virology
Volume: 53
Issue: 3
ISSN: 1386-6532
Publisher: Elsevier B.V.  
Date Published: 2012-03-01
Start Page: 225
End Page: 230
Language: English
DOI: 10.1016/j.jcv.2011.11.007
PROVIDER: scopus
PUBMED: 22196870
DOI/URL:
Notes: --- - "Export Date: 2 April 2012" - "CODEN: JCVIF" - "Source: Scopus"
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  1. Klaus J Busam
    688 Busam