Annangi Srinadh, Pasha Sara
University of Kentucky College of Medicine, Division of Pulmonary Critical Care and Sleep Medicine, Lexington, KY, USA.
Department of Pulmonary and Critical Care Medicine, Harrison Memorial Hospital, Cynthiana, KY, USA.
Ann Transl Med. 2021 Apr;9(7):584. doi: 10.21037/atm-20-4212.
Oral isotretinoin is a synthetic analog of vitamin - A, reserved for cases with severe resistant acne. We hereby report a case of drug-induced vasculitis (DV) from isotretinoin exposure leading to life-threatening pulmonary-renal syndrome requiring immunosuppression and plasmapheresis. A previously healthy 21-year-old female receiving oral isotretinoin presented with a 10-day history of worsening myalgias, arthralgias, and abdominal pain. Soon after admission she progressed to severe pulmonary-renal syndrome requiring intubation and renal replacement therapy. Urinalysis revealed >50 dysmorphic RBC with casts and renal ultrasound was unremarkable. Serological testing was only positive for antineutrophil cytoplasmic antibodies (ANCA) at 1:80 with Anti- proteinase 3 (PR3) at 830 AU/mL and Anti-histone Ab at 2.9. As clinical presentation and serology are highly suggestive of ANCA associated DV, plasmapheresis, and rituximab were also initiated along with the continuation of steroids. She clinically improved but remained dialysis dependent and received a live donor renal transplant. The temporal relationship of symptom onset and drug initiation with no other possible identifiable etiologies-DV in our case was attributed to isotretinoin exposure. Though considered safe, oral Isotretinoin in rare instances can cause the life-threatening pulmonary-renal syndrome. Given its widespread use, it is prudent that prescribers should educate patients regarding the possible symptoms of vasculitis and to seek immediate medical attention when warranted. Physicians should also be vigilant of this complication and should act swiftly to avoid uneventful outcomes.
口服异维甲酸是维生素A的合成类似物，仅用于治疗重度难治性痤疮。我们在此报告一例因接触异维甲酸导致药物性血管炎（DV）的病例，该血管炎引发了危及生命的肺肾综合征，需要进行免疫抑制和血浆置换治疗。一名此前健康的21岁女性在接受口服异维甲酸治疗后，出现了为期10天的肌痛、关节痛和腹痛加重症状。入院后不久，她进展为严重的肺肾综合征，需要插管和肾脏替代治疗。尿液分析显示每高倍视野有超过50个异形红细胞且有管型，肾脏超声检查无异常。血清学检测仅抗中性粒细胞胞浆抗体（ANCA）呈阳性，滴度为1:80，抗蛋白酶3（PR3）为830 AU/mL，抗组蛋白抗体为2.9。由于临床表现和血清学高度提示为ANCA相关的DV，在继续使用类固醇的同时，也开始了血浆置换和利妥昔单抗治疗。她的临床症状有所改善，但仍依赖透析，并接受了活体供肾移植。在我们的病例中，症状出现与药物使用的时间关系以及没有其他可识别的病因，DV被归因于异维甲酸暴露。尽管口服异维甲酸被认为是安全的，但在罕见情况下可导致危及生命的肺肾综合征。鉴于其广泛使用，开处方者应告知患者血管炎的可能症状，并在必要时寻求立即医疗关注，这是谨慎的做法。医生也应警惕这种并发症，并应迅速采取行动以避免不良后果。
