Spiny Papules in an Immunosuppressed Child

Abstract

Trichodysplasia spinulosa is a rare folliculocentric polyomavirus infection observed in the setting of immunosuppression. We report a 7-year-old boy with pre-B-cell lymphoblastic leukemia who presented with folliculocentric spiny papules on the face. Histologic evaluation revealed hypertrophic bulbs, an expanded inner root sheath, and numerous brightly eosinophilic trichohyalin granules. We present this case to raise awareness of this rare but recognizable entity and to highlight the availability of appropriate diagnostic and therapeutic modalities.

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

“…Topical cidofovir 1% cream daily was used in five cases and cidofovir 3% cream in one case with response ranging from moderate to significant subjective improvement 2,7,14,15,17,18 . Topical valacyclovir once a day was also reported with complete resolution over 6 months 19 .…”

“…Patient ages ranged from 20 months to 15 years. Lymphoblastic leukemia was the most common underlying diagnosis, reported in seven patients 7‐13 . Renal transplant was the second most common, reported in five patients 4,14‐16 .…”

“…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 . Diagnosis was most commonly made using clinical presentation and histology.…”

“…Lymphoblastic leukemia was the most common underlying diagnosis, reported in seven patients. [7][8][9][10][11][12][13] Renal transplant was the second most common, reported in five patients. 4,[14][15][16] Other underlying diagnoses included heart transplant, colon transplant, lung transplant, and multiorgan transplant.…”

“…19 Five patients significantly improved or cleared following reduction in immunosuppression or discontinuation of chemotherapy. 5,7,14,15,20 In one reported case, IV cidofovir 0.5 mg/kg/wk for 16 doses in combination with reduction of immunotherapy, intravenous immunoglobulin (IVIg), and cidofovir 1% cream daily led to significant improvement. 15 Manual extraction of spines was also performed in one case with significant improvement.…”