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Spiky Skin in a Renal Transplant Recipient

Educational Objective
Based on this clinical scenario and the accompanying image, understand how to arrive at a correct diagnosis.
1 Credit CME

A woman in her 70s presented with a 1-year history of a facial cutaneous eruption initially affecting malar cheeks and eyebrows. Subsequently it spread to involve nose, chin, upper trunk, and extremities. The lesions were mildly pruritic. Her medical history included a cadaveric renal transplant 2 years earlier for end-stage renal failure. Immunosuppressant medications included mycophenolate mofetil, 200 mg, twice daily and tacrolimus, 8 mg, twice daily. Tacrolimus levels were therapeutic. Physical examination revealed multiple 1-mm flesh-colored follicular papules and keratin spines against a diffuse erythematous background affecting the face (Figure, A and B) and upper trunk. Her scalp and eyebrow hairs were unremarkable. Serology results were unremarkable. Results of skin biopsy from the right ear demonstrated dilatation and keratotic plugging of the hair infundibula with marked dystrophy and expansion of the inner root sheath. The inner root sheath cells were enlarged with irregular trichohyaline granules and apoptotic cells with abrupt cornification without formation of a granular layer (Figure, C). Immunohistochemical analysis for SV40 polyomavirus was positive (Figure, D).

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A. Trichodysplasia spinulosa

Taking the clinical and histopathological findings together, the diagnosis is consistent with trichodysplasia spinulosa. Treatment options included reduction in tacrolimus dose; however, owing to the risk of transplant rejection this was not deemed to be a safe option. To date, the rash persists and has further spread to involve most of the trunk and extremities.

Trichodysplasia spinulosa is a rare viral infection reported in immunosuppressed patients with a medical history of organ transplantation or patients who are immunosuppressed from hematologic malignant abnormalities treated with chemotherapy. Trichodysplasia spinulosa-associated polyomavirus (TSPγV) was first identified in 2010.1 The causal mechanism by which this TSPγV human polyomavirus causes trichodysplasia spinulosa remains unknown. Despite this, on a molecular level, TSPγV has been shown to activate factors implicated in the mitogen-activated protein kinase (MAPK) pathway leading to cellular proliferation characteristics of trichodysplasia spinulosus.2

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Article Information

Corresponding Author: Flora Poon, MBBS, MRT, MHSc, Department of Dermatology, Monash Health, 246 Clayton Rd, Clayton, Victoria, Australia 3168 (flora.poon@hotmail.com).

Published Online: July 18, 2018. doi:10.1001/jamadermatol.2018.1267

Conflict of Interest Disclosures: None reported.

Additional Contributions: We thank Richard Turner, MBBCh, Oxford University Hospitals, for provision of photos. We also thank the patient for granting permission to publish this information.

References
1.
van der Meijden  E, Janssens  RWA, Lauber  C,  et al.  Discovery of a new human polyomavirus associated with trichodysplasia spinulosa in an immunocompromized patient.  PLoS Pathog. 2010;6(7):e1001024.PubMedGoogle ScholarCrossref
2.
Wu  JH, Simonette  RA, Nguyen  HP, Rady  PL, Tyring  SK.  Small T-antigen of the TS-associated polyomavirus activates factors implicated in the MAPK pathway.  J Eur Acad Dermatol Venereol. 2016;30(6):1061-1062.PubMedGoogle ScholarCrossref
3.
Bialasiewicz  S, Byrom  L, Fraser  C, Clark  J.  Potential route of transmission for trichodysplasia spinulosa polyomavirus.  J Infect Dis. 2017;215(7):1175-1176.PubMedGoogle ScholarCrossref
4.
Celeiro-Muñoz  C, González-Vilas  D, Sánchez-Aguilar  D, Suárez-Peñaranda  JM.  Viral-associated trichodysplasia secondary to antineoplastic treatment in a patient with lymphoblastic leukemia.  Am J Dermatopathol. 2014;36(6):e105-e107.PubMedGoogle ScholarCrossref
5.
Matthews  MR, Wang  RC, Reddick  RL, Saldivar  VA, Browning  JC.  Viral-associated trichodysplasia spinulosa: a case with electron microscopic and molecular detection of the trichodysplasia spinulosa-associated human polyomavirus.  J Cutan Pathol. 2011;38(5):420-431.PubMedGoogle ScholarCrossref
6.
Cook  L.  Polyomaviruses.  Microbiol Spectr. 2016;4(4).PubMedGoogle Scholar
7.
Wanat  KA, Holler  PD, Dentchev  T,  et al.  Viral-associated trichodysplasia: characterization of a novel polyomavirus infection with therapeutic insights.  Arch Dermatol. 2012;148(2):219-223.PubMedGoogle ScholarCrossref
8.
Schwieger-Briel  A, Balma-Mena  A, Ngan  B, Dipchand  A, Pope  E.  Trichodysplasia spinulosa—a rare complication in immunosuppressed patients.  Pediatr Dermatol. 2010;27(5):509-513.PubMedGoogle ScholarCrossref
9.
Benoit  T, Bacelieri  R, Morrell  DS, Metcalf  J.  Viral-associated trichodysplasia of immunosuppression: report of a pediatric patient with response to oral valganciclovir.  Arch Dermatol. 2010;146(8):871-874.PubMedGoogle ScholarCrossref
10.
Kassar  R, Chang  J, Chan  AW, Lilly  LB, Al Habeeb  A, Rotstein  C.  Leflunomide for the treatment of trichodysplasia spinulosa in a liver transplant recipient.  Transpl Infect Dis. 2017;19(4):e12702.PubMedGoogle ScholarCrossref
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