Liu Jingwei, Yan Haibo, Yang Chunfeng, Li Yumei
Department of Pediatric Intensive Care Unit, First Hospital of Jilin University, Changchun, China.
Front Pediatr. 2023 Mar 2;11:1116166. doi: 10.3389/fped.2023.1116166. eCollection 2023.
Stevens-Johnson syndrome/toxic epidermal necrolysis has a severe impact on patients' eyes, genital mucosa, and many other organs. Bronchiolitis obliterans is a rare complication of Stevens-Johnson syndrome/toxic epidermal necrolysis.
We report a case of bronchiolitis obliterans associated with toxic epidermal necrolysis in our department. Furthermore, we examined the patients with bronchiolitis obliterans induced by Stevens-Johnson syndrome/toxic epidermal necrolysis and summarized the clinical characteristics, treatment, and prognosis. Databases available online in English including PubMed, Medline, and Web of Science were consulted.
We report one case and review 23 published case reports. Of the 24 patients, 13 were female, the oldest patient was 59 years old and the youngest was 5 years old. The time of bronchiolitis obliterans onset after Stevens-Johnson syndrome/toxic epidermal necrolysis varied from 5 days to 5 months. Bronchoscopy examination showed ulceration, exudative lesions, occlusion, and inflammation. The CT of lung manifestation included mosaic perfusion, bronchiectasis, consolidation, air trapping, pneumatocele, pleural thickening, lung collapse, larger central airway dilatation, lung overinflation, oligemia, and pneumomediastinum. Most cases indicated pulmonary function tests with obstructive ventilation dysfunction. The prognosis was poor; six of the patients died.
Patients with Stevens-Johnson syndrome/toxic epidermal necrolysis may develop bronchitis obliterans at different stages, so all patients with Stevens-Johnson syndrome/toxic epidermal necrolysis should be followed up for possible respiratory complications.
史蒂文斯-约翰逊综合征/中毒性表皮坏死松解症对患者的眼睛、生殖器黏膜及许多其他器官有严重影响。闭塞性细支气管炎是史蒂文斯-约翰逊综合征/中毒性表皮坏死松解症的一种罕见并发症。
我们报告了我科1例与中毒性表皮坏死松解症相关的闭塞性细支气管炎病例。此外,我们对史蒂文斯-约翰逊综合征/中毒性表皮坏死松解症所致闭塞性细支气管炎患者进行了检查,并总结了其临床特征、治疗及预后情况。查阅了包括PubMed、Medline和Web of Science在内的英文在线可用数据库。
我们报告了1例病例并回顾了23篇已发表的病例报告。这24例患者中,女性13例,年龄最大59岁,最小5岁。史蒂文斯-约翰逊综合征/中毒性表皮坏死松解症后闭塞性细支气管炎发病时间从5天至5个月不等。支气管镜检查显示有溃疡、渗出性病变、阻塞及炎症。肺部CT表现包括马赛克灌注、支气管扩张、实变、气体潴留、肺气囊、胸膜增厚、肺不张、中央大气道扩张、肺过度充气、肺血减少及纵隔气肿。多数病例肺功能检查提示存在阻塞性通气功能障碍。预后较差;6例患者死亡。
史蒂文斯-约翰逊综合征/中毒性表皮坏死松解症患者可能在不同阶段发生闭塞性细支气管炎,因此所有史蒂文斯-约翰逊综合征/中毒性表皮坏死松解症患者均应进行随访,以排查可能出现的呼吸道并发症。