Trichodysplasia Spinulosa in a 7‐Year‐Old Boy Managed Using Physical Extraction of Keratin Spicules

Abstract

Trichodysplasia spinulosa is an uncommon skin disease characterized by a folliculocentric papular eruption and keratin spine formation, classically appearing on the central face and ears. It almost always occurs in immunosuppressed patients and is linked to a viral etiology. Diagnostic tests including PCR are available for detection of the TS-associated polyomavirus (TSPyV). Effective treatment options include topical cidofovir and oral valganciclovir. We present a case diagnosed by PCR from skin scrapings and treated with physical extraction of the keratin spicules. Significant improvement was noted suggesting a safe, cost-effective treatment alternative.

Trichodysplasia spinulosa in a child: Identification of trichodysplasia spinulosa‐associated polyomavirus in skin, serum, and urine

“…Clinical diagnosis may be confirmed by the characteristic histopathology, electron microscopy, or molecular demonstration of virus in affected hairs . Similar to a previously reported case, we were able to confirm the diagnosis by PCR from plucked spicules and suggest that this noninvasive method could be used as a diagnostic test in children or adults when a biopsy is not preferred.…”

“…Trichodysplasia spinulosa–associated polyomavirus has been detected in the skin, urine, blood, respiratory secretions, and cerebrospinal fluid of TS patients . Although our patient had clear evidence of a systemic TSPyV infection with virus detectable in the blood and urine, the skin was the only site of end‐organ dysfunction.…”

Trichodysplasia spinulosa mimicking lichen nitidus in a renal transplant patient

“…TS, first described in 1999 by Haycox et al, is a rare cutaneous complication of systemic immunosuppression and is associated with the TSPyV . TSPyV has been consistently identified in cases of TS, suggesting that TSPyV infection is causative . TS has been reported in both adult and pediatric patients who have undergone chemotherapy for a lymphoreticular malignancy or have undergone organ transplantation .…”

“…The development of keratin spicules on the face and ears in the context of immunosuppression, particularly organ transplantation or lymphoreticular malignancy, should raise suspicion for TS. Characteristic histopathologic changes, electron microscopy, or PCR of either a biopsy sample or skin scraping can confirm the diagnosis of TS . Histologically, TS is characterized by dilated hair follicles with proliferation of the inner root sheath cells and the presence of cytoplasmic eosinophilic inclusions consisting of trichohyalin .…”

“…Electron microscopy can be used to confirm the diagnosis by identifying intranuclear viral inclusions within the hair follicle keratinocytes . TSPyV PCR may also be helpful for confirming viremia, but this test is not yet widely available …”