Leflunomide for the treatment of trichodysplasia spinulosa in a liver transplant recipient

Kassar, Rawan; Chang, Janis; Chan, An‐Wen; Lilly, Leslie; Habeeb, Ayman Al; Rotstein, Coleman

doi:10.1111/tid.12702

Cited by 23 publications

(27 citation statements)

References 26 publications

(30 reference statements)

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“…TS typically presents on the central face and ears with multiple skin‐colored, folliculocentric papules and central keratotic spicules. The additional presence of the rash on the trunk, arms, and legs has also been reported . Skin lesions are usually asymptomatic, but are occasionally associated with mild pruritus.…”

Section: Discussionmentioning

confidence: 96%

“…A reduction in immunosuppressive medications allows immune recovery and facilitates a reduction in viral load. Alternatives that have been reported to be successful include IV cidofovir, topical cidofovir (1%‐3%) cream, oral valganciclovir, and leflunomide . Additionally, physical extraction of keratin spicules has been utilized as a cost‐effective and safe option for temporary improvement in appearance .…”

Section: Discussionmentioning

confidence: 99%

“…Alternatives that have been reported to be successful include IV cidofovir, topical cidofovir (1%-3%) cream, oral valganciclovir, and leflunomide. 2,6,[10][11][12] Additionally, physical extraction of keratin spicules has been utilized as a cost-effective and safe option for temporary improvement in appearance. 5 This technique may not be feasible when substantial body surface areas are involved, and the lesions may return after extraction in the case of ongoing immunosuppression.…”

Section: Discussionmentioning

confidence: 99%

“…2,9,10 The additional presence of the rash on the trunk, arms, and legs has also been reported. 1,7,8,11,12 Skin lesions are usually asymptomatic, but are occasionally associated with mild pruritus. Despite the lack of symptoms, the rash can be conspicuous and disfiguring.…”

mentioning

confidence: 99%

“…TSPyV has been consistently identified in cases of TS, suggesting that TSPyV infection is causative [2][3][4][5][6][7]. TS has been reported in both adult and pediatric patients who have undergone chemotherapy for a lymphoreticular malignancy or have undergone organ transplantation 1,2,[4][5][6][7][8][9][10][11][12]. Attempts to estimate the seroprevalence of TSPyV have found the virus to be quite ubiquitous in the general population: near 10% in healthy children, 80% in healthy adults, and 89% in a subset of 80 renal transplant patients in the Netherlands.…”

mentioning

confidence: 99%

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Trichodysplasia spinulosa mimicking lichen nitidus in a renal transplant patient

Barone

Brockman

Johnson

et al. 2019

Pediatric Transplantation

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Trichodysplasia spinulosa (TS) is a rare cutaneous condition associated with the TSPyV and characterized by skin‐colored, folliculocentric papules with keratin spicule formation. TS is seen almost exclusively in immunosuppressed individuals, often presenting in patients with a history of solid organ transplantation or chemotherapy for a lymphoreticular malignancy. We report a case of widespread TS in a 9‐year‐old girl with a history of renal transplantation complicated by BK viremia, which is also caused by a polyomavirus, BKPyV. The clinical presentation of TS in this case morphologically resembled the more common, harmless skin condition known as “lichen nitidus,” and was more extensive than expected for TS, creating a diagnostic challenge. This case illustrates an important presentation of severe TS of which transplant teams, oncologists, primary care providers, and dermatologists should be aware.

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Section: Discussionmentioning

confidence: 96%

Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

mentioning

confidence: 99%

mentioning

confidence: 99%

See 3 more Smart Citations

Trichodysplasia spinulosa mimicking lichen nitidus in a renal transplant patient

Barone

Brockman

Johnson

et al. 2019

Pediatric Transplantation

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Spiky Skin in a Renal Transplant Recipient

2018

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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 granulesandapoptoticcellswithabruptcornificationwithoutformationofagranularlayer(Figure , C). Immunohistochemical analysis for SV40 polyomavirus was positive (Figure , D). Diagnosis A. Trichodysplasia spinulosa DiscussionTaking the clinical and histopathological findings together, the diagnosis is consistent with trichodysplasia spinulosa.

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Trichodysplasia Spinulosa in a Kidney Transplant Recipient

2021

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A kidney transplant recipient in her 60s (who was taking mycophenolate mofetil and belatacept) presented with a 1-year history of pruritic pink follicular papules with central white spicules on her face, ears, and extremities associated with eyebrow and eyelash alopecia (Figure). Skin biopsy results demonstrated distended hair follicles occluded by hyperkeratotic and parakeratotic debris. At the suprabulbar level, the outer root sheath had central plugging with enlarged intracytoplasmic trichohyaline granules diagnostic of trichodysplasia spinulosa (TS). Polymerase chain reaction confirmed the presence of TS polyoma virus.Treatment with compounded topical 1% cidofovir cream, applied twice daily, produced significant clinical improvement within 2 months of treatment that was maintained at 6-month and 1-year follow-up visits. Trichodysplasia spinulosa is a rare folliculocentric infection seen almost exclusively in patients with immunosuppression, including transplant recipients and patients with a hematologic malignancy. 1 In 2010, the TS human polyoma virus was identified by electron microscopy, sequenced, and named. 2 To our knowledge, only 30 cases of TS have been reported in association with iatrogenic immunosuppression.Trichodysplasia spinulosa is characterized by numerous erythematous to skin-colored follicular papules with central white spicules distributed predominantly over the central face, often resulting in loss of the eyebrows and eyelashes. While the TS human polyoma virus has been identified in saliva, blood, and nasal swabs, the virus appears to primarily affect the skin, with unknown longterm consequences. 2 Trichodysplasia spinulosa can be diagnosed clinically. However, because the differential diagnosis is broad, a skin biopsy provides diagnostic confirmation. The differential diagnosis may include lichen spinulosus, multiple minute digitate hyperkeratosis, spiculate demodicosis, lichen planopilaris, keratosis pilaris, trichostasis spinulosa and the follicular spicules of multiple myeloma.Treatments for TS include 1% to 3% topical cidofovir, oral valganciclovir, and a reduction of immunosuppression when clinically appropriate. 3 Given the risk of transplant rejection when reducing immunosuppression, as well as potential adverse effects of valganciclovir, including creatinine elevations and bone marrow suppression, topical cidofovir was chosen as the initial option. More recently, leflunomide has also been described as an oral treatment. 4 This patient highlights a rare condition that was effectively and safely treated with topical therapy.

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Leflunomide for the treatment of trichodysplasia spinulosa in a liver transplant recipient

Cited by 23 publications

References 26 publications

Trichodysplasia spinulosa mimicking lichen nitidus in a renal transplant patient

Trichodysplasia spinulosa mimicking lichen nitidus in a renal transplant patient

Spiky Skin in a Renal Transplant Recipient

Trichodysplasia Spinulosa in a Kidney Transplant Recipient

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