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CASE REPORT
Year : 2020  |  Volume : 18  |  Issue : 3  |  Page : 251-254

Right hemothorax secondary to endometriosis


1 Department of Obstetrics and Gynaecology, Ave Maria Hospital, Victoria Island, Lagos, Nigeria
2 Department of Radiology, Ave Maria Hospital, Victoria Island, Lagos, Nigeria

Date of Submission24-Mar-2020
Date of Decision11-Apr-2020
Date of Acceptance22-Apr-2020
Date of Web Publication10-Jul-2020

Correspondence Address:
Mr. Ernest Ruto Upeh
Department of Radiology, Ave Maria Hospital, Victoria Island, Lagos
Nigeria
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/cmi.cmi_43_20

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  Abstract 


Endometriosis is said to have a low incidence in women with African descent. However, recent studies have shown otherwise. Thoracic involvement is rare; thoracic endometriosis syndrome mostly affects nulliparous women within the ages of 25–35 years. The diagnosis is usually missed or delayed by clinicians. We report the case of a 26-year-old female with catamenial hemothorax secondary to thoracic endometriosis. A high index of suspicion for this condition should be maintained in women of the reproductive age group presenting with dyspnea/chest pain, severe dysmenorrhea, and background history of chronic pelvic pain. A multidisciplinary approach involving the radiographers, gynecologists, pulmonologists, and the thoracic surgeons coupled with hormonal therapy and surgical treatment will go a long way in improving this disease condition and preventing reoccurrences.

Keywords: Endometriosis, hemothorax, pneumothorax, thoracic endometriosis syndrome


How to cite this article:
Ijasan O, Upeh ER. Right hemothorax secondary to endometriosis. Curr Med Issues 2020;18:251-4

How to cite this URL:
Ijasan O, Upeh ER. Right hemothorax secondary to endometriosis. Curr Med Issues [serial online] 2020 [cited 2020 Oct 21];18:251-4. Available from: https://www.cmijournal.org/text.asp?2020/18/3/251/289417




  Introduction Top


Endometriosis is a relatively common condition during which mucosa cells and stroma are implanted in the extrauterine sites. Its prevalence has been calculated to be up to 10% of reproductive-age females, with women within the age limit of 25–35 years principally affected.[1] It is ordinarily encountered within the dependent parts of the pelvis such as the cervix, vagina, labia,  Fallopian tube More Detailss, uterine ligaments, and even the pelvic peritoneum. Therefore, a thoracic involvement is a comparatively rare presentation of this common pathology.[2],[3]

Thoracic endometriosis syndrome usually presents within the first 48–72 h of menstruation,[4] with 73% of this manifestation being catamenial pneumothorax when implantation occurs within the pleura. Approximately, 14%, 7%, and 6% present as catamenial hemothorax, hemoptysis, and lung nodules, respectively.[1] Pericardial endometriosis is uncommon; however, a few of these cases have been reported with pericardial/pleural effusion and ascites as its manifestations. Up to 85% of women diagnosed with catamenial pneumothorax and catamenial hemothorax will have an associated pelvic disease.[2],[5] The diagnosis of thoracic endometriosis is often clinically suspected where patients show classical symptoms of shortness of breath and long-term sequelae of the pleuritic chest or shoulder pain within 24 h before menses or 72 h after menses.[6],[7]

Various imaging modalities can be used in the diagnosis of thoracic endometriosis syndrome. These include, but not limited to digital radiography, computed tomography and magnetic resonance imaging and sonography depending on the sites where these lesions are found.[5] Another modality is a video-assisted thoracoscopy which is a minimally invasive channel through which thoracic surgeons diagnose thoracic endometriosis.[8] However, it is pertinent to point that there is a huge limitation in the diagnosis of thoracic endometriosis syndrome using imaging examinations and histopathological findings as only about one-third of a definitive diagnosis is from histopathological confirmations.[9]

The treatment modalities may include, but not limited to endoscopic resection, video-assisted thoracoscopic pleurodesis accompanied by the administration of gonadotropin-releasing hormone (GnRH) analog. Aromatase inhibitors are also potent in the treatment of thoracic endometriosis.[10],[11]


  Case Report Top


An ambulant 26-year-old nulliparous female presented to the emergency department with 10 weeks history of nonproductive cough, progressive dyspnea, and right-sided pleuritic chest pain. Furthermore, other symptoms elicited from her history include severe dysmenorrhea and bloating. These symptoms usually occurred 3 days before the onset of menses.

Her vitals were a respiratory rate of 19 breaths/min, blood pressure of 120/80 mmHg, and a pulse rate of 100 beats/min. She was afebrile, maintaining an oxygen saturation of 99% on room air. On physical examination, reduced breath sounds in the right lung field were noted.

Chest X-ray (CXR) revealed a severe right-sided pleural effusion, atelectasis, a suspected mediastinal cavity, and mild left-sided mediastinal shift [Figure 1]a. Abdominal scans were suggestive of minimal ascites and a right-sided pleural effusion. Within the lung base, multiple tortuous echogenic strands were noted with no vascularity on color Doppler sonography raising suspicion of the presence of endometrial stroma and gland tissue [Figure 2]a and [Figure 2]b.
Figure 1: (a) Chest X-ray on the 1st day of presentation showing massive right-sided pleural effusion (green arrow) with a suspected mediastinal cavity (blue arrow) and a tracheal shift to the left (white arrow). (b) Chest X-ray with tube thoracostomy in situ(yellow arrow), The image shows a reduction in the right-sided pleural effusion (green arrow) and pneumothorax (red arrow). (c) Chest X-ray on the 5th day showing expanded right lung with subtle pneumothorax (red arrow) and pleural effusion (green arrow).

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Figure 2: (a) Thoracic ultrasound on 1st day of the presentation showing massive pleural fluid (green arrow) with low-level echoes and echogenic strands (white arrow). (b) Thoracic ultrasound showing a right massive pleural fluid (green arrow) with echogenic strands. CDI = Color Doppler imaging. (c) Thoracic ultrasound showing expanded right lung with subtle pleural effusion (green arrow), gallbladder (yellow arrow).

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Needle aspiration with the placement of a right-sided pigtail catheter was performed immediately. The full blood counts and electrolyte and urea results were essentially within the normal limits. Pleural fluid hematocrit to serum hematocrit was 0.6 suggestive of hemothorax. Pleural fluid cytology showed clusters of epithelial cells without atypia with background histiocytes. After the patient's consent was obtained, a right tube thoracostomy was done on the 2nd day of admission, with a total of 1300 ml of free-flowing sanguineous fluid drained. Approximately 1288 ml, 2042 ml, 2600 ml, and 1610 ml were drained on the 3rd, 4th, 5th, and 6th days of admission, respectively. Throughout admission, 8840 ml of sanguineous fluid was drained in total. Furthermore, she had intrapleural streptokinase, prophylactic antibiotics, analgesia, and chest physiotherapy. It was decided to use streptokinase to help achieve intrapleural fibrinolysis, break down loculated blood, and facilitate drainage of the hemothorax. Marked symptomatic improvement was noted. A repeat CXR and thoracic sonography done on the 5th day showed remarkable improvement and minimal pleural effusion [Figure 1]b and [Figure 2]c. On the 6th day, the thoracostomy tube was removed [Figure 1]c. She was counseled about her condition, administered 10.8 mg of GnRH analog (zoladex), to prevent reoccurrence and told to report to the clinic in 3 months. At her 3-month follow-up visit, she was clinically stable. CXR showed minimal pleural effusion. She was administered another dose of 10.8 mg zoladex and told to report to the clinic after another 3 months for possible placement on danazol.


  Discussion Top


Endometriosis is distinguished by the presence of endometrial tissue, including the stroma and glands being implanted outside the uterine cavity. This occurs mostly within the pelvis, resulting in symptoms such as dysmenorrhea, dysuria, and dyspareunia. Endometriosis is not limited to the pelvis alone, as it could affect other fewer common sites such as the labia, abdomen, and thorax.[2] Thoracic endometriosis is relatively rare and is singularized by the implantation of endometrial glands within the pleura, pericardium, respiratory tracts, or diaphragm.[1],[2],[3]

Over the years, there has been a right-sided preponderance of catamenial pneumothorax and hemothorax than the left.[2],[12] This was also the case in our patient. In an attempt to explain this phenomenon, several theories have generated. The leading postulates are those of endometrial tissues emanating from the paracolic gutters, being trapped by the falciform ligament in the liver, preventing it from moving across to the left and are thereby propelled by intraperitoneal pressure through diaphragmatic fenestrated defects into the right lung zone.[2],[12],[13],[14],[15] Others believed that catamenial hemothorax and catamenial pneumothorax may be as a result of the rupture of bullae orchestrated by prostaglandin F2-alpha because of the endometriosis deposition.[8],[16] Another explanation for these are the effects of venous or lymphatic transport and coelomic metaplasia.[14],[15] However, none of the above postulates alone accounts for all the clinical manifestations of thoracic endometriosis syndrome.[17] Therefore, the etiology of thoracic endometriosis syndrome is presumably multifaceted.

The clinical manifestation of thoracic endometriosis syndrome in the patient discussed was a cough, progressive dyspnea with a background history of severe dysmenorrhea, chronic pelvic pain, right-sided pleuritic chest pain, and abdominal bloating in synchrony with her menstrual period. These clinical symptoms are consistent with other studies.[2],[3],[5],[16]

In our patient, several echogenic strands were found in the right basal pleura suspected to be of endometrial origin on the sonogram [Figure 2]a and [Figure 2]b. Pneumothorax and hemothorax were right-sided, which is in agreement with almost 85%–95% of all the cases reported.[13],[16] Although the incidence of thoracic endometriosis syndrome is reportedly low among women of African descent,[18] we are of the view that this is probably due to underreporting of cases, low index of suspicion, and generally poor awareness of the condition among physicians. On the other hand, with more access to tertiary institutions and specialist care, the prevalence of thoracic endometriosis is on the increase.[6] A huge referral of gynecological surgeries (thoracic endometriosis syndrome-related) and its associated cost with long-term sequelae of this disease is the sweeping cause for reduced quality of life for women within the median age of 30 years.[13]

Established reports suggest that imaging modalities are nonspecific for the diagnosis of thoracic endometriosis syndrome.[15] However, the suspected diagnosis in our patient was supported with the findings on CXR and thoracic/abdominal ultrasound.[19] Furthermore, pleural fluid cytology results were those of clustered epithelial cells without atypia with background histiocytes. Another nonspecific marker that can be employed generally in the diagnoses of endometriosis is elevated cancer antigen 125 levels.[1],[7],[16]

Our patient was planned for the administration of GnRH analog for at least 6 months. This will inhibit the activity of the ectopic endometrium until effective pleurodesis occurs with the formation of pleural adhesions.[15],[16] Other hormonal agents employed in the management of endometriosis and its sequelae include combined oral contraceptives, progestogens, aromatase inhibitors, and androgens such as danazol. Consequently, when hormone therapy fails or catamenial pneumothorax/catamenial hemothorax recurs, surgical treatment is advised for better results.


  Conclusion Top


The thoracic endometrial syndrome should be suspected in females of the reproductive age group who show symptoms of right pleuritic chest pain and recurrent right-sided hemothorax or pneumothorax.

The first line of treatment should be hormonal therapy which will stop a reoccurrence in more than 50% of cases. Subsequently, where this fails, surgical ablation and pleurodesis should be considered with hormonal therapy as an adjunct to treatment.

A multidisciplinary approach involving radiologists, gynecologists, pulmonologists, and cardiothoracic surgeons is important in the management of complicated patients with this syndrome.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Lopes EN, Damasio LC, Baracat EC, Sra AK, Sra JL, Albuquerque DM. Pleural endometriosis associated with catamenial hemothorax: A case report. MOJ Womens Health 2017;5:313-31.  Back to cited text no. 1
    
2.
Rawala MS, Khaliq MF, Iqbal MA, Naqvi ST, Farhan K, Myers A, et al. A rare case of cyclical hemothorax: Thoracic endometriosis syndrome. Case Rep Pulmonol 2018;(2018):9830797.  Back to cited text no. 2
    
3.
Bataduwaarachchi VR, Jayawardhana J, Weerasinghe C, Sadikeen A, Constantine R, Gunasekara K. An asymptomatic haemorrhagic pleural effusion mimicking extra-pulmonary tuberculosis in a young woman with thoracic endometriosis syndrome: A case report. Int J RespirPulm Med 2018;5:82.  Back to cited text no. 3
    
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Bačić I, Petani B, Morović D, Kovačić I. Rare case of thoracic endometriosis syndrome: Catamenial hemothorax. Signavitae 2017;13:2.  Back to cited text no. 4
    
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Chamié LP, Ribeiro DM, Tiferes DA, Macedo Neto AC, Serafini PC. Atypical sites of deeply infiltrative endometriosis: Clinical characteristics and imaging findings. Radiographics 2018;38:309-28.  Back to cited text no. 5
    
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Okyere I, Glover PS, Forson PK, Okyere P, Blood-Dzraku D. Catamenial pneumothorax in Ghana: Case report and literature review. Pan Afr Med J 2019;33:287.  Back to cited text no. 6
    
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Celik S, Erşen E. Catamenial Pneumothorax, Pneumothorax, Khalid Amer, Intech Open, (ISBN: 978-1-83968-066-3) 2019; DOI: 10.5772/intechopen.82564.  Back to cited text no. 7
    
8.
Alifano M. “Catamenial Pneumothorax.” National Organization for Rare Disorder. 2012. Available from: https://rarediseases.org/rare-diseases/catamenial-pneumothorax/. [Last retrieved on 2019 Sep 22].  Back to cited text no. 8
    
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Pankratjevaite L, Samiatina-Morkuniene D. A case report of thoracic endometriosis - A rare cause of haemothorax. Int J Surg Case Rep 2017;33:139-42.  Back to cited text no. 9
    
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Junejo SZ, Singh Lubana S, Shina SS, Tuli SS. A case of thoracic endometriosis syndrome presenting with recurrent catamenial pneumothorax. Am J Case Rep 2018;19:573-6.  Back to cited text no. 10
    
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Chadwick TH, Audlin K. Letrozole for the treatment of thoracic endometriosis: A case report. Obstetrics Gynecol 2019;133:100-11.  Back to cited text no. 11
    
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Narula N, Ngu S, Avula A, Mansour W, Chalhoub M. Left-sided catamenial pneumothorax: A rare clinical entity. Cureus 2018;10:e2567.  Back to cited text no. 12
    
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Bobbio A, Canny E, Mansuet Lupo A, Lococo F, Legras A, Magdeleinat P, et al. Thoracic endometriosis syndrome other than pneumothorax: Clinical and pathological findings. Ann Thorac Surg 2017;104:1865-71.  Back to cited text no. 13
    
14.
McCloud A, Onugha O. Recurrent catamenial pneumothorax after failed doxycycline pleurodesis. Clin Surg 2019;4:2531.  Back to cited text no. 14
    
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Ajayi A, Ajayi V, Oyetunji I, Biobaku O, Aikhuele H, Afolabi BM. Abdominal scar endometriosis and hemothorax in a Nigerian woman: A case report. Gynecol Obstet (Sunnyvale) 2017;7:430.  Back to cited text no. 15
    
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AlAqeel S, AlJehani Y, AlMuhaish M. Bilateral catamenial hemopneumothorax: Diagnostic management challenges. Int J Surg Case Rep 2019;61:271-4.  Back to cited text no. 16
    
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Nezhat C, Lindheim SR, Backhus L, Vu M, Vang N, Nezhat A, et al. Thoracic Endometriosis Syndrome: A Review of Diagnosis and Management. JSLS 2019;23:e2019.00029.  Back to cited text no. 17
    
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Ajani MA, Salami A, Nwanji ID, Olusanya AA, Fatunla OE. Distribution and characteristics of endometriotic lesions in South-Western Nigeria: A single institutional experience. Med J Zambia 2019;46:28-32.  Back to cited text no. 18
    
19.
Brogi E, Gargani L, Bignami E, Barbariol F, Marra A, Forfori F, et al. Thoracic ultrasound for pleural effusion in the intensive care unit: A narrative review from diagnosis to treatment. Crit Care 2017;21:325.  Back to cited text no. 19
    


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