Article, Gynecology

A rare massive presentation of catamenial hemothorax

a b s t r a c t

Catamenial hemothorax is a rare manifestation of thoracic endometriosis syndrome. It is commonly seen associ- ated with pelvic endometriosis in nulliparous reproductive-age women. Most cases are minor and self-limiting. We present a case of a 32-year-old woman who presented with prolonged worsening dyspnea and was found to have a massive hemothorax on evaluation.

(C) 2019

Introduction

Thoracic endometriosis syndrome (TES) is the term given when en- dometrial tissue occurs within the thoracic cavity. The most common clinical presentations of TES include chest pain, cough, and dyspnea. TES can manifest as one of the following entities: catamenial pneumo- thorax (73% of cases), catamenial hemothorax (14% of cases), hemopty- sis (7% of cases) or lung nodules (6% of cases) [1].

Clinical manifestations are defined as catamenial when they occur between 72 h before and 72 h after the onset of menses [2]. Catamenial hemothorax (CH) is an extremely rare manifestation of TES that is usu- ally mild and self-limiting. We report a case of a young woman who pre- sented with massive CH.

case presentation“>Case presentation

A 32-year-old woman presented to the emergency department (ED) with a one-week history of worsening shortness of breath (SOB) exacerbated by physical activity. She also complained of right-sided pleuritic chest pain and dry cough during deep inspira- tion. No history of fever, trauma, travel, sick contacts or similar epi- sodes in the past. The patient had an extensive past history of

* Corresponding author.

E-mail address: [email protected] (S. Pillai).

dysmenorrhea diagnosed as endometriosis and had been on treat- ment for the last 12 years.

At the time of this presentation, she was on the fourth day of her menstrual cycle. In the ED, she appeared dyspneic on examination. She had a blood pressure of 96/60 mm Hg with a heart rate of 106 beats/min. Physical examination showed absent breath sounds on the right lower lobe of the lung. Chest x-ray was done imme- diately, which showed a moderate to massive sized right pleural ef- fusion without a mediastinal shift (Fig. 1). All investigations for a malignant or infectious etiology were unremarkable. Ultrasound- guided diagnostic pleural aspiration was done and 250 ml of hemor- rhagic chocolate-colored fluid was aspirated. A computed tomogra- phy (CT) scan of the chest (Fig. 2) showed gross Right-sided pleural effusion causing a near-total collapse of the right lung. Enhancing soft tissue densities were seen in the right cardiophrenic angle sug- gestive of endometriotic deposits. Cardiothoracic surgery was consulted immediately.

Video-assisted thoracoscopic surgical (VATS) decortication was done within 24 h, with excision of the endometrial lesion and talc pleurodesis. A mesh plug was applied after removal of an endometrial lesion in the right cardiophrenic angle. Approximately 1.8 L of reddish fluid were drained and an intercostal drain (ICD) placed. Histopatholog- ical examination of decorticated tissue (Fig. 3) was consistent with endometriosis.

She underwent aggressive deep breathing exercises and in- centive spirometry post-operatively. Sequential imaging showed significant improvement with a complete resolution of

https://doi.org/10.1016/j.ajem.2019.158474

0735-6757/(C) 2019

1695.e2 A. Somani et al. / American Journal of Emergency Medicine 38 (2020) 1695.e11695.e3

Discussion

Fig. 1. Chest Xray P-A view showing opacity of the right hemithorax (arrow).

the hemothorax, following which the ICD was taken out. She was eventually discharged on dienogest 2 mg once daily and analge- sics. A follow-up visit 2 months later showed no recurrence.

Endometriosis is prevalent in 50% of women with infertility, and up to 70% of women with pelvic pain [3,4]. The thoracic cavity is the most common extra pelvic location involving the pleura, lung parenchyma, diaphragm, or bronchus.

CH is a late manifestation of TES (14% of cases) and indicates the presence of a significant burden of proliferating pleural endometrial im- plants [5]. It is considered to be the final end of the spectrum following hemoptysis and Catamenial pneumothorax, each being a sequential en- dometrial involvement of the lung tissue and pleura [5]. A review of cases of CH in literature showed chest pain and dyspnea as the most common presenting symptoms each occurring in 53% of cases [5]. The mean age of patients was 35 years [5]. There was documented pelvic en- dometriosis in more than 80% cases [5]. A definitive diagnosis of CH couldn’t be made until at least four clinical episodes of hemothorax in 20% of cases [5]. Most cases of CH are minor and self-limiting. To the best of our knowledge, there is only one other reported instance, in the literature of massive (Blood loss of >= 1000 ml) CH [7].

A catamenial etiology is very rarely suspected in hemothorax with trauma, infection, malignancy, or coagulopathy being the com- mon causes [7]. A catamenial etiology is seldom suspected in a mas- sive hemothorax, a condition in which cardioVascular injury is usually the sole cause. Hence such a massive presentation of CH is re- markable and can be a cause of diagnostic confusion. The mean dura- tion of symptoms prior to a definitive diagnosis of thoracic endometriosis is about 8-19 months [1]. Since the patient described in our case had a history of pelvic endometriosis, the suspicion for CH was higher. However, there were at least 18 cases described in the literature with CH as the primary manifestation of endometriosis [6]. This suggests a diagnostic challenge for the emergency physician

Fig. 2. a, b, c from left to right: CT scan thorax: a-b (Coronal view), c (Axial view). a-Extensive pleural effusion in the right hemithorax (red arrow). b-Nodular hyper dense lesion in right anterior cardio phrenic location (red arrow). c-Right massive plural effusion (blue arrow) with nodular enhancing hyper dense lesion in right cardio phrenic location (red arrow).

Fig. 3. a, b from left to right: Histopathology. a: Decorticated pleural tissue showing fibrocollagenous tissue (black arrow) with decidualised endometrial stromal tissue and areas of hemorrhage (red arrow) and foreign body giant cell reaction (green arrow). b: Endometrial nodule with siderophages or hemosiderin laden macrophages (blue arrow) seen.

A. Somani et al. / American Journal of Emergency Medicine 38 (2020) 1695.e11695.e3 1695.e3

where management of hemothorax without removing ectopic endo- metrial tissue, would lead to Recurrent episodes. The emergency physician should have a high index of suspicion especially when signs of respiratory distress correlate temporally with the menstrual cycle in young women with a history of pelvic endometriosis or infertility.

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