Yalçın Selma, Ensarioglu Kerem, Kurt Bahar, Özişler Cem
Department of Pulmonology, Ordu Fatsa State Hospital, Ordu, TUR.
Department of Pulmonology, University of Health Sciences, Ankara Atatürk Sanatorium Training and Research Hospital, Ankara, TUR.
Cureus. 2023 Apr 4;15(4):e37121. doi: 10.7759/cureus.37121. eCollection 2023 Apr.
Hughes-Stovin Syndrome (HSS) is a rare clinical condition characterized by thrombophlebitis as well as multiple pulmonary and bronchial aneurysms. It commonly presents with coughing, dyspnea, fever, chest pain, and hemoptysis, and its management usually consists of surgical and medical approaches. In this report, we discuss a case of a patient with HSS. A 30-year-old male patient was admitted to the pulmonary medicine ward for hemoptysis. After evaluation with chest CT, bilateral pulmonary embolism and pulmonary aneurysms were observed. Due to a history of aphthous lesions, Behçet's disease (BD) was considered the initial diagnosis; however, the patient did not fit the criteria and was later diagnosed with HSS. Intravenous methylprednisolone was initiated, along with a maintenance treatment with cyclophosphamide. Treatment response was observed in the fourth month; however, due to the persistence of hemoptysis, additional cycles of cyclophosphamide were later required, under which the patient's condition has been stable. HSS currently lacks clear diagnostic criteria, and further studies are needed to investigate genetic backgrounds, familial transmissions, and treatment alternatives.
休斯-斯托文综合征（HSS）是一种罕见的临床病症，其特征为血栓性静脉炎以及多发性肺和支气管动脉瘤。它通常表现为咳嗽、呼吸困难、发热、胸痛和咯血，其治疗通常包括手术和药物治疗方法。在本报告中，我们讨论一例HSS患者。一名30岁男性患者因咯血入住呼吸内科病房。经胸部CT评估后，发现双侧肺栓塞和肺动脉瘤。由于有复发性口腔溃疡病史，最初考虑诊断为白塞病（BD）；然而，该患者不符合诊断标准，后来被诊断为HSS。开始静脉注射甲泼尼龙，并联合环磷酰胺进行维持治疗。在第四个月观察到治疗反应；然而，由于咯血持续存在，后来需要额外的环磷酰胺疗程，在此期间患者病情一直稳定。HSS目前缺乏明确的诊断标准，需要进一步研究以探讨其遗传背景、家族遗传情况和治疗方案。
