Trichodysplasia Spinulosa – A Newly Described Folliculocentric Viral Infection in an Immunocompromised Host

Abstract

This is a case report of an immunocompromised individual who presented with progressive alopecia, friable follicular spinous processes, and erythematous, indurated papules. Examination of skin biopsies using light microscopy and immunohistochemistry revealed pathologic changes of the follicular inner root sheath epithelium with dystrophic trichohyaline granules. Electron microscopy of thin sections of tissue revealed intracellular viral particles with a size and appearance consistent with those in the Papovaviridae family. Electron microscopy of negatively stained extract from a homogenized lesion also demonstrated icosahedral viruses with papovavirus morphology. We believe this is a previously unreported folliculocentric viral infection in an immunosuppressed human host and have termed this entity "trichodysplasia spinulosa".

The trichodysplasia spinulosa‐associated polyomavirus: virological background and clinical implications

“…Furthermore, enlargement of hair follicles with excessive proliferative cells is observed (72). Compared with normal skin, TS follicles are absent of hair shafts, and papilla with abnormal corneocytes that fill the infundibula of the follicles (Fig.…”

“…In 1999, Haycox and colleagues fully described the disease and introduced the term "trichodysplasia spinulosa" (72). They showed for the first time the presence of virus particles and suggested a viral etiology.…”

Entry, infection, replication, and egress of human polyomaviruses: an update

“…The intracellular presence of virus particles in a patient with trichodysplasia spinulosa (TS) was first demonstrated in 1999 (Haycox et al 1999). Identification of the virus was not achieved until much later in plucked facial spines of a heart transplant patient with TS (van der Meijden et al 2010).…”

“…Identification of the virus was not achieved until much later in plucked facial spines of a heart transplant patient with TS (van der Meijden et al 2010). TS is a rare skin disease in immunocompromised hosts and is characterised by the development of facial follicular papules and keratotic protrusions (spicules or spines) with alopecia of the eyelashes and brows (Haycox et al 1999;Heaphy et al 2004;Osswald et al 2007;Sadler et al 2007;Lee et al 2008;Schwieger-Briel et al 2010). The presence and high seroprevalence of TSPyV in healthy individuals suggest a subclinical latent infection.…”

Trichodysplasia Spinulosa in a 20-Month-Old Girl With a Good Response to Topical Cidofovir 1%

“…Trichodysplasia spinulosa (TS) is a follicular eruption that affects immunocompromised patients, and was first reported by Haycox et al in 1999, 1 who described a skin eruption with friable spinous processes, indurated erythematous papules, and eyebrow alopecia in a patient who was receiving cyclosporine after renal and pancreas transplantation. After that, in 2004, Sperling et al 2 used the term viral-associated trichodysplasia to report a kidney transplant recipient on a multidrug immunosuppression regimen.…”