Ji-Xu Antonio, Artounian Kimberly, Fung Maxwell A, Burrall Barbara A
Department of Dermatology, University of California, Davis, Sacramento, California, USA.
Dermatol Online J. 2022 Dec 15;28(6). doi: 10.5070/D328659724.
Trichodysplasia spinulosa (TS) is a rare skin condition that occurs mainly in immunosuppressed patients. Although initially postulated to be an adverse effect of immunosuppressants, TS-associated polyomavirus (TSPyV) has since been isolated from TS lesions and is now considered to be the causative agent. Trichodysplasia spinulosa presents with folliculocentric papules with protruding keratin spines, most commonly on the central face. Trichodysplasia spinulosa can be diagnosed clinically, but the diagnosis can be confirmed with histopathological examination. Histological findings include the presence of hyperproliferating inner root sheath cells containing large eosinophilic trichohyaline granules. Polymerase chain reaction (PCR) can also be used to detect and quantify TSPyV viral load. Owing to the paucity of reports in the literature, TS is frequently misdiagnosed and there is no high-quality evidence to guide management. Herein, we present a renal transplant recipient with TS that did not respond to topical imiquimod but improved upon treatment with valganciclovir and reduction of the mycophenolate mofetil dose. Our case highlights the inverse relationship between immune status and disease progression in this condition.
棘状毛囊发育不良（TS）是一种罕见的皮肤病，主要发生于免疫抑制患者。尽管最初推测是免疫抑制剂的不良反应，但此后已从TS皮损中分离出TS相关多瘤病毒（TSPyV），现在认为它是病原体。棘状毛囊发育不良表现为以毛囊为中心的丘疹，伴有突出的角质棘，最常见于面部中央。棘状毛囊发育不良可通过临床诊断，但组织病理学检查可确诊。组织学表现包括存在含有大的嗜酸性透明角质颗粒的增殖过度的内根鞘细胞。聚合酶链反应（PCR）也可用于检测和定量TSPyV病毒载量。由于文献报道较少，TS常被误诊，且缺乏高质量证据指导治疗。在此，我们报告1例肾移植受者患有TS，局部应用咪喹莫特无效，但接受缬更昔洛韦治疗并减少霉酚酸酯剂量后病情改善。我们的病例突出了这种情况下免疫状态与疾病进展之间的负相关关系。
