Propylthiouracil-induced vasculitis with alveolar hemorrhage after 31 years of treatment: a case report.

Ro'a Khalaf, Deema Mhesin, Hasan Abu Salim, Sari Taha, Waddah Abed
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Abstract

Background: Propylthiouracil is an antithyroid medication that is associated with ANCA positivity, often in the absence of clinically overt vasculitis. Propylthiouracil-induced ANCA-associated vasculitis (AAV) is characterized by multiple antigenicity and presents similarly to other forms of drug-induced vasculitis.

Case presentation: We report a case of a 49-year-old woman who developed propylthiouracil-induced AAV after 31 years of intermittent therapy. The patient presented with progressive respiratory and generalized symptoms. She was diagnosed with propylthiouracil-induced AAV with alveolar hemorrhage based on detection of ANCA and anti-MPO antibodies, exclusion of primary vasculitis, and symptom resolution following propylthiouracil withdrawal. The patient was treated with intravenous methylprednisolone, followed by oral prednisolone. Her symptoms resolved, and her laboratory and radiological tests improved within one week.

Conclusions: Research linking the duration of propylthiouracil use to the development of vasculitis is restricted by multiple methodological limitations. Maintaining a high index of clinical suspicion is a more reliable approach to detecting propylthiouracil-induced AAV than regular laboratory monitoring, given that patients with Graves' disease may show ANCA positivity in the absence of medication use. Therefore, clinicians should remain vigilant for the varied presentations of propylthiouracil-induced AAV. Moreover, further research is recommended to investigate the possible link between the duration of PTU use and the onset of clinically overt vasculitis.

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治疗 31 年后丙基硫氧嘧啶诱发血管炎伴肺泡出血:病例报告。
背景:丙基硫氧嘧啶是一种抗甲状腺药物,与ANCA阳性相关,但临床上往往没有明显的血管炎。丙基硫氧嘧啶诱导的 ANCA 相关性血管炎(AAV)具有多重抗原性的特点,其表现与其他形式的药物诱导性血管炎相似:我们报告了一例 49 岁女性的病例,她在接受了 31 年的间歇性治疗后,患上了丙基硫氧嘧啶诱导的 AAV。患者出现进行性呼吸道症状和全身症状。根据 ANCA 和抗 MPO 抗体的检测结果、原发性血管炎的排除情况以及停用丙基硫氧嘧啶后症状的缓解情况,她被诊断为丙基硫氧嘧啶诱发的 AAV 并伴有肺泡出血。患者接受了静脉甲基强的松龙治疗,随后又口服了强的松龙。她的症状在一周内得到缓解,实验室和放射学检查也有所改善:将丙基硫氧嘧啶的使用时间与脉管炎的发生联系起来的研究受到多种方法学限制。鉴于巴塞杜氏病患者在未用药的情况下也可能出现ANCA阳性,因此与定期进行实验室监测相比,保持高度的临床怀疑指数是检测丙基硫氧嘧啶诱发的血管炎的更可靠方法。因此,临床医生应对丙基硫脲嘧啶诱发的AAV的各种表现保持警惕。此外,还建议进一步研究使用 PTU 的时间长短与临床上明显的血管炎发病之间可能存在的联系。
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来源期刊
自引率
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发文量
7
审稿时长
8 weeks
期刊介绍: Clinical Diabetes and Endocrinology is an open access journal publishing within the field of diabetes and endocrine disease. The journal aims to provide a widely available resource for people working within the field of diabetes and endocrinology, in order to improve the care of people affected by these conditions. The audience includes, but is not limited to, physicians, researchers, nurses, nutritionists, pharmacists, podiatrists, psychologists, epidemiologists, exercise physiologists and health care researchers. Research articles include patient-based research (clinical trials, clinical studies, and others), translational research (translation of basic science to clinical practice, translation of clinical practice to policy and others), as well as epidemiology and health care research. Clinical articles include case reports, case seminars, consensus statements, clinical practice guidelines and evidence-based medicine. Only articles considered to contribute new knowledge to the field will be considered for publication.
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