Primary trichodysplasia spinulosa polyomavirus infection in a kidney transplant child displaying virus‐infected decoy cells in the urine

Borgogna, Cinzia; Albertini, Silvia; Zavattaro, Elisa; Veronese, Federica; Peruzzi, Licia; Meijden, E. van der; Feltkamp, Mariet C.W.; Tosoni, Antonella; Volpe, Alessandro; Boldorini, Renzo; Gariglio, Marisa

doi:10.1002/jmv.25519

Cited by 11 publications

(20 citation statements)

References 16 publications

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“…All patients presented with predominately centrofacial folliculocentric papules with keratinaceous spines. Twelve of the 19 cases demonstrated generalized involvement including the trunk and extremities 2,4,7,9‐15,18 . Lateral eyebrow alopecia and less commonly eyelash alopecia were reported as well 2,7,8,11,12,17,20 .…”

Section: Resultsmentioning

confidence: 98%

“…Diagnosis was most commonly made using clinical presentation and histology. In three cases, PCR for TSPyV was run on plucked keratinaceous spines and found to be positive 4,8,18 . SV40T antibody immunostaining from tissue samples was also completed in two cases.…”

Section: Resultsmentioning

confidence: 99%

“…8 In one case, IgG titers showed seroconversion after the patient's second transplant associated with the appearance of cutaneous disease. 4 van der Meijden et al reported two cases of trichodysplasia spinulosa, where high viral loads of TSPyV were detected in the blood and CSF supporting primary disseminated TSPyV infection as the cause of the cutaneous manifestations. 26 This may explain the rarity of cases relative to the high seropositivity in immunosuppressed patients.…”

Section: Re Sultsmentioning

confidence: 99%

“…In 2010, van der Meijden et al 2 discovered a novel human polyomavirus that they named trichodysplasia spinulosa‐associated polyomavirus (TSPyV). In healthy populations, the seroprevalence of TSPyV is estimated at 70%, but clinical manifestations present only in a very small percentage of infected individuals, principally transplant recipients and leukemia and lymphoma patients 3,4 . Reported successful treatment modalities include reduction in immunosuppression as well as topical and systemic antiviral medications 5,6 …”

Section: Introductionmentioning

confidence: 99%

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Pediatric trichodysplasia spinulosa: A report of 2 cases and review of the literature

Rusk

Crute

Goebel

et al. 2020

Pediatric Dermatology

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Trichodysplasia spinulosa is a rare disorder caused by the ubiquitous trichodysplasia spinulosa-associated polyomavirus (TSPyV) and characterized clinically by predominately centrofacial, but often generalized, folliculocentric papules with protuberant keratinaceous spines. Although seroprevalence reaches up to 70% in adult populations, TSPyV causes clinical manifestations in a small percentage of patients who are immunosuppressed. Diagnosis can be made using typical clinical and histologic features, SV40T antibody immunostaining, and PCR of various tissues including the keratinaceous spine, skin, serum, urine, and CSF. Various topical and systemic medications have demonstrated variable success. Decreasing or discontinuing immunosuppression has also been shown to improve or alleviate clinical manifestations.

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Section: Resultsmentioning

confidence: 98%

Section: Resultsmentioning

confidence: 99%

Section: Re Sultsmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

See 2 more Smart Citations

Pediatric trichodysplasia spinulosa: A report of 2 cases and review of the literature

Rusk

Crute

Goebel

et al. 2020

Pediatric Dermatology

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“…They described for the first time the presence of degenerated TSPyV‐infected decoy epithelial cells in the patient's urine that are morphologically different from BK or JC polyomavirus‐infected (BKPyV and JCPyV) decoy cells 14 . TSPyV decoy cells harbored vacuoles in the cytoplasm along with large eosinophilic granules resembling those observed in the inner root sheath cells and strongly reacted with anti‐TSPyV‐specific antibodies 14 . These results are supportive of a model where TSPyV, similar to JCPyV and BKPyV, can be shed in the urine of kidney transplant recipients.…”

Section: Viral Associationmentioning

confidence: 99%

Recent developments in trichodysplasia spinulosa disease

Narayanan

Rády

Tyring

2020

Transplant Infectious Dis

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Trichodysplasia Spinulosa (TS) is a rare proliferative skin disease that occurs primarily in immunocompromised patients, specifically organ transplant recipients. TS is characterized by uncontrolled inner root sheath cell proliferation and folliculocentric papular eruption that can progress to disfiguring leonine facies when left untreated. TS presents with distinct histological features including the presence of large eosinophilic, trichohyaline granules within hyperproliferating inner root sheath cells of the hair bulb. The discovery of the Trichodysplasia Spinulosa Polyomavirus (TSPyV) and recent studies highlighting the role of TSPyV tumor antigens in cell proliferation pathways have provided new insight into the mechanisms of TS development. In this review, we discuss the expansion of our understanding of TS, specifically over the past 5 years. We summarize novel cases of TS and recent developments in the mechanisms underlying TSPyV-mediated disease progression. We also evaluate advancements in diagnostic methods and treatment options. As the incidence of TS continues to rise, it is becoming critical for clinicians to understand the clinical features of TS and emerging research regarding pathogenesis and therapeutics for early treatment of this potentially disfiguring disease.

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