A Case of Treatment-Resistant Catamenial Disease: Insufficiency in Mainstay Treatment Options

A. Faisal, S. Fatima, F. Masud, I. Ratnani
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Abstract

Introduction: Thoracic endometriosis syndrome (TES) is a rare phenomenon that affects 6-10% of women of reproductive age. It is characterized by presence of ectopic endometrial tissue within the thoracic cavity, mainly the lung parenchyma and pleura. TES presents as catamenial pneumothorax (CP), catamenial hemothorax (CH), pulmonary nodules or hemoptysis. Hormonal therapy is considered first line treatment followed by operative thoracoscopy for removal of ectopic endometrial implants. Our case highlights the limited number of treatment options in a young patient with recurrent catamenial disease.Description: A 37-year-old G4P0040 female presented to the emergency department (ED) with worsening shortness of breath (SOB) for 5 days. Her SOB was sudden in onset, progressively worsening, and associated with cough and right-sided chest pain. Patient was admitted 5 months ago for similar symptoms. She has a past medical history significant for hypertension, obesity, iron deficiency anemia, severe endometriosis and TES manifesting as recurrent, right sided pleural effusions, CPs and CHs requiring surgical treatment. Previous biopsies of the ectopic endometrial tissue revealed dense fibroconnective tissue with focal endometriosis and no indication for malignancy. In the ED, she appeared in distress and had a BP of 166/102. Physical examination revealed tachypnea, accessory muscle usage, respiratory distress and retractions. Breath sounds were decreased in the right lung. EKG revealed normal sinus rhythm, and a COVID-19 test yielded negative results. Chest x-ray and CT were consistent with findings for right tension hemothorax. She was started on BiPAP initially and thoracic surgery was consulted. A tube thoracostomy was performed in the ED and a chest tube was placed. Moreover, a CBC revealed Hb of 5.7g/dl. She was transfused 4 units pRBC and admitted to the ICU for further monitoring. Ob/gyn was consulted and leuprolide was administered to prevent recurrence of a hemothorax. She remained in the hospital for 3 days and was subsequently discharged after resolution of symptoms confirmed by imaging.Conclusion: Catamenial hemothoraces are a late finding of TES. There is mixed data regarding the effectiveness of GnRH agonists on recurrence rates of CH, however subsequent surgery has shown to be preventative in resistant cases. This unique case illustrates the insufficiency in both mainstay treatments. Despite the fact the patient had previous lobectomies using VATS, lysis of adhesions and hormonal therapy, she continued to have recurrent manifestations of TES. Novel treatments in conjunction with a multidisciplinary team approach should be used to effectively treat TES and prevent disease relapse.
难治性羊膜病1例:主要治疗方案不足
简介:胸段子宫内膜异位症(TES)是一种罕见的现象,影响6-10%的育龄妇女。其特点是胸腔内存在异位子宫内膜组织,主要是肺实质和胸膜。TES表现为连膜性气胸(CP)、连膜性血胸(CH)、肺结节或咯血。激素治疗被认为是一线治疗,其次是手术胸腔镜切除异位子宫内膜植入物。我们的病例强调了有限的治疗方案,在一个年轻的病人复发性肛管疾病。描述:一名37岁的G4P0040女性因呼吸急促加重5天来到急诊科(ED)。她的呜咽发作突然,逐渐加重,并伴有咳嗽和右侧胸痛。病人5个月前因类似症状入院既往有高血压、肥胖、缺铁性贫血、严重子宫内膜异位症和TES,表现为复发性右侧胸腔积液、CPs和CHs需要手术治疗。先前异位子宫内膜组织活检显示致密纤维结缔组织伴局灶性子宫内膜异位症,无恶性迹象。在急诊科,她看起来很痛苦,血压为166/102。体格检查显示呼吸急促,副肌使用,呼吸窘迫和收缩。右肺呼吸音减少。心电图显示窦性心律正常,COVID-19检测结果为阴性。胸部x线及CT表现与右侧紧张性血胸一致。她最初开始使用BiPAP,并咨询了胸外科手术。在急诊科进行了管式开胸术,并放置了胸管。此外,CBC显示Hb为5.7g/dl。患者输注4单位pRBC并入院ICU进一步监测。我们咨询了妇产科医生,并给药leuprolide以防止血胸复发。她在医院住了3天,随后在影像学证实症状消退后出院。结论:肠系血胸是TES的晚期发现。关于GnRH激动剂对CH复发率的有效性,有不同的数据,但随后的手术已显示对耐药病例具有预防作用。这个独特的病例说明了两种主要治疗方法的不足。尽管患者之前曾使用VATS进行过肺叶切除术、松解粘连和激素治疗,但她仍然有TES的复发表现。新的治疗方法应与多学科团队的方法相结合,以有效地治疗TES和预防疾病复发。
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