CATAMENIAL PNEUMOTHORAX: A MANAGEMENT CHALLENGE

Abstract

SESSION TITLE: Medical Student/Resident Pulmonary Manifestations of Systemic Disease 3 SESSION TYPE: Med Student/Res Case Rep Postr PRESENTED ON: 10/09/2018 01:15 PM - 02:15 PM INTRODUCTION: Catamenial pneumothorax (CP) is a rare clinical entity presenting with recurrent pneumothorax in women of reproductive age (1). Presence of ectopic endometrial tissue in thorax leads to their activation with each menstrual cycle. We report a unique case of a young female who presented with recurrent spontaneous pneumothorax despite video assisted thoracoscopic surgery (VATS). CASE PRESENTATION: A 36-year-old female with history of endometriosis, CP status post VATS about 4 months prior to this presentation, DVT, was referred to our ER for admission after she was found to have large right pneumothorax on a CT chest done as outpatient for chest discomfort. She was hemodynamically stable with SpO2 97% on room air. CT chest revealed a very large loculated right pneumothorax with left mediastinal shift and extensive pleural thickening. Initially she underwent a pigtail catheter placement in the largest pocket. Despite the pigtail drainage, patient continued to have air leak and mediastinal shift. Thus, a decision was made to perform VATS again. Pleural biopsy and VATS pleurodesis were performed with placement of 12F, 19F, 28F chest tubes. Mediastinal shift resolved but she continued to have small pneumothorax. On day 16, she was discharged with a chest tube and outpatient follow-up. Chest tube was removed on one week follow up in the clinic. She continued to have small loculated right sided pneumothorax. Pleural biopsy obtained during VATS, showed positive paired-box gene8 (PAX8) in epithelial lining compatible with presence of endometrial tissue in pleura. She is currently on progesterone only mini pill and is closely followed up in clinic. DISCUSSION: Thoracic endometriosis is the most common site for extra pelvic endometriosis with CP being a common presentation. It mostly affects right side (85-90%) like in our case. Characteristic findings associated with CP include defects of diaphragm like perforation, stomata, pores and fenestrations, which can vary in size, shape and numbers (2). With this case, we emphasize that the physicians should be aware of this relatively uncommon clinical entity when encountering young female with recurrent pneumothorax. Even after confirmed diagnosis, CP poses a treatment challenge as it can continue to cause recurrent large pneumothoraces despite VATS and hormonal therapy. Patients can develop pleural thickening and loculations which makes management of future pneumothoarcses even more difficult. No clear guidelines exist on management of CP. With the disease affecting young population and having a significant effect on lifestyle, it is prudent that more research should be done for CP management. CONCLUSIONS: CP is an uncommon cause of recurrent pneumothorax. Histologic finding of endometrial tissue on pleural biopsy is often required to confirm the diagnosis. CP is usually managed with hormonal therapy and VATS but needs further investigation. Reference #1: Bagan P, et al. Catamenial pneumothorax: retrospective study of surgical treatment. Ann Thorac Surg 2003;75:378-81. Reference #2: Visouli AN, et al. Catamenial pneumothorax: a rare entity? Report of 5 cases and review of the literature. J Thorac Dis. 2012 Nov;4(Suppl 1):17–31. DISCLOSURES: No relevant relationships by Nidhi Aggarwal, source=Web Response No relevant relationships by Vivek Kumar, source=Web Response No relevant relationships by Yizhak Kupfer, source=Web Response No relevant relationships by Jignesh Patel, source=Web Response No relevant relationships by Anand Rai, source=Web Response No relevant relationships by Arjun Saradna, source=Web Response No relevant relationships by Ankur Sinha, source=Web Response No relevant relationships by Parita Soni, source=Web Response