School of Medicine, University of North Carolina at Chapel Hill.
Department of Dermatology, University of North Carolina at Chapel Hill.
JAMA Dermatol. 2022 Jul 1;158(7):787-790. doi: 10.1001/jamadermatol.2022.1090.
Cases of photodistributed Stevens-Johnson syndrome (SJS) and toxic epidermal necrolysis (TEN) have been infrequently reported since the first documented case in 1989. This emerging clinical entity and its underlying mechanism have yet to be fully characterized.
To report a case of photodistributed SJS/TEN and highlight similarities to other cases reported in the literature.
DESIGN, SETTING, AND PARTICIPANTS: Case report and literature review of published cases of photodistributed SJS/TEN. The case report describes a 29-year-old woman with recent lamotrigine and trimethoprim-sulfamethoxazole exposure who developed TEN in a photodistributed pattern 1 day after prolonged sun exposure. A search of PubMed using the keywords toxic epidermal necrolysis, Stevens-Johnson syndrome, photo-distributed, photo-induced, and sun-exposed was performed to identify other cases reported in the literature.
Literature review revealed 8 previously reported cases of healthy individuals with known drug and UV radiation (UVR) exposures who subsequently developed SJS or TEN with photodistribution. Cases reviewed were skewed demographically to young women aged 19 to 48 years (8 of 9 patients) with all cases reporting UVR exposure 24 to 72 hours prior to the onset of symptoms.
Photodistributed TEN has been increasingly described in the literature and may represent a distinct variant of SJS/TEN. While the pathogenesis remains unclear, the role of UVR as a "second hit" is suggested by the data presented in the cases documented thus far.
自 1989 年首例确诊病例以来,光分布型史蒂文斯-约翰逊综合征(SJS)和中毒性表皮坏死松解症(TEN)的病例报道罕见。这种新兴的临床实体及其潜在机制尚未得到充分描述。
报告 1 例光分布型 SJS/TEN 病例,并强调与文献中报道的其他病例的相似之处。
设计、设置和参与者:光分布型 SJS/TEN 的病例报告和文献回顾,以及对已发表病例的回顾。该病例报告描述了 1 例 29 岁女性,近期有拉莫三嗪和甲氧苄啶-磺胺甲噁唑暴露史,在长时间暴露于阳光后第 1 天出现 TEN,表现为光分布型。通过在 PubMed 上使用关键词毒性表皮坏死松解症、史蒂文斯-约翰逊综合征、光分布、光诱导和暴露于阳光下进行检索,以确定文献中报道的其他病例。
文献回顾显示,此前有 8 例健康个体在已知药物和紫外线辐射(UVR)暴露后出现 SJS 或 TEN,伴有光分布。回顾的病例在人口统计学上偏向于 19 至 48 岁的年轻女性(9 例患者中有 8 例),所有病例均报告在症状出现前 24 至 72 小时有 UVR 暴露。
光分布型 TEN 在文献中越来越多地被描述,可能代表 SJS/TEN 的一种独特变异。虽然发病机制尚不清楚,但迄今为止记录的病例中的数据提示 UVR 作为“第二次打击”的作用。