Thoracic endometriosis-related non-catamenial pneumothorax with peculiar histological findings
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CASE REPORT
Thoracic endometriosis‑related non‑catamenial pneumothorax with peculiar histological findings Elisa Scarnecchia1,3 · Francesco Inzirillo1 · Paolo Declich2 · Claudio Della Pona1 Received: 17 June 2019 / Accepted: 28 July 2019 © The Japanese Association for Thoracic Surgery 2019
Abstract Thoracic endometriosis-related non-catamenial pneumothorax is a rare entity whose pathogenesis is still less unclear than catamenial pneumothorax one. Hormonal therapy and/or talc pleurodesis are not sufficient for successful management. Surgical videothoracoscopic resection has a central role in the treatment. We displace a case of thoracic endometriosis-related non-catamenial pneumothorax presenting with recurrent right pneumothorax, surgically treated three times and misdiagnosed at first two interventions. At third operation, unusual histological findings on diaphragmatic and pulmonary specimens were disclosed. These results could partially clarify the presentation of some complicated misdiagnosed cases. More has to be investigated about pathogenesis of the disease and influence of the hormonal balance on it. Keywords Thoracic endometriosis · Catamenial pneumothorax · Videothoracoscopic surgery
Introduction Catamenial pneumothorax (CP) is an entity occurring in females of childbearing age in the perimenstrual period. By definition, the “perimenstrual period” usually includes the day before to 72 h after the onset of menstruation [1]. CP is mostly related to thoracic endometriosis (TE) and accounts for 20–35% of pneumothorax occurring in young women. Nevertheless, the mean age for diagnosis of CP is slightly higher if compared to spontaneous pneumothorax [1, 2]. Based on the finding of TE in women of childbearing age presenting with recurrent pneumothorax in the intermenstrual period, Alifano and co-workers identified two different nosological entities: thoracic endometriosis related catamenial pneumothorax (TER-CP) with CP occurring in the perimenstrual period and TER-nonCP arising in the intermenstrual period, with TER-nonCP accounting for one-third of * Elisa Scarnecchia [email protected] 1
Department of Thoracic Surgery and Thoracic Endoscopy, Eugenio Morelli Hospital, via Zubiani 33, Sondalo, SO, Italy
2
Department of Anatomic Pathology and Histology, Eugenio Morelli Hospital, Sondalo, Italy
3
Department of Medical and Surgical Sciences (DIMEC), Alma Mater Studiorum, University of Bologna, Bologna, Italy
all cases [1, 3]. Three different theories have been proposed to explain TE: retrograde migration with endometrial seeding through diaphragmatic pores; lymphatic/vascular embolism of endometrial foci on the visceral pleura; congenital metaplasia [1, 4]. TER non-CP pathogenesis is even less clear and still debated. Accordingly, more has to be investigate to better understand CP and TE pathogenesis, mainly for TER-nonCP. Here we report a case of TER-nonCP with unusual histological findings.
Case presentation A 45-year-old non-smoker woman was referred to our service after a 5 years p
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