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Pulmonary Endometriosis - a Rare Differential Diagnosis of Lung Cancer

Pulmonary Endometriosis - a Rare Differential Diagnosis of Lung Cancer 10.2478/chilat-2014-0108 ACTA CHIRURGICA LATVIENSIS • 2014 (14/1) CASE REPORT Pulmonary Endometriosis – a Rare Differential Diagnosis of Lung Cancer Agita Jukna, Ilze Strumfa, Inese Drike, Andrejs Vanags, Janis Gardovskis Riga Stradins University, Latvia SUMMARY Endometriosis is characterised by the presence of ectopic functional endometrial tissue outside the uterus. The disease most frequently affects pelvic tissues and organs but any site can be involved including gastrointestinal tract, lungs and other organs. Extra-pelvic endometriosis mostly occurs in females aged 35-40 years. Here we report a well-documented, morphologically proved and surgically treated case of pulmonary endometriosis in a 50-year-old woman in order to heighten the awareness of endometriosis as the differential diagnosis for nodular lung lesions. Key words: endometriosis, lung, lung tumour, differential diagnosis th AIM OF THE DEMONSTRATION cm in diameter, was detected in the 8 segment of the The aim of this demonstration is to report a rare case left lung. Thoracotomy and marginal resection of the of extra-pelvic endometriosis in order to increase the affected segment was performed. Cytological smear of awareness about endometriosis as the differential the operation material showed cylindrical epithelial diagnosis of lung cancer. cells and groups of proliferating cells with traits of atypia on a RBC-rich background. Cytologist thus CASE REPORT could not exclude malignant tumour. The resected lung A fifty-year-old female underwent routine chest fragment with a collapsed subpleural cavity measuring roentgenography. As a peripheral nodule was disclosed 1 cm in diameter was submitted to the pathological in her left lung, the family doctor referred the patient to examination. By histology, the resection margins were thoracic surgeon. To decide about the further assessment composed of lung tissue lacking any disease process. and treatment strategy, chest computed tomography The wall of the cystic nodule had dual structure with was performed repeatedly after a month. However, the hypercellular stroma which consisted of small spindled diagnosis was not clear and tumour could not be ruled cells and dilated cystic glands lined by low cubic out completely, therefore the lady was admitted to the epithelium (Fig. 1). None of these components showed hospital for surgical treatment. cell atypia. By immunohistochemistry, the pathological The patient had no complaints. However, on detailed tissues expressed oestrogen and progesterone receptors questioning she recalled occasional, dry cough. The lady but lacked pulmonary markers including TTF-1 (Fig. 2). had had no treatment or specific examination due to The proliferation fraction (by Ki-67) and oncoprotein non-disturbing intensity of these symptoms, and also spectrum (p53, Bcl-2) did not suggest malignant tumour. was unable to describe the duration of the cough. Her Thus, endometriosis was diagnosed in accordance with gynaecological anamnesis was characterised by irregular the morphological structure and immunophenotype. menstruations during the last year. Previously, her There were no postoperative complications. The patient th menstrual cycle was regular, with the average duration was released from hospital on 9 postoperative day for 28 days and bleeding of normal intensity lasting 3-4 further treatment under supervision of gynaecologist days. The menstrual bleeding was not preceded by or with the final diagnosis of nodular endometriosis in the th associated with pain, and the patient did not reveal 8 lung segment. use of any hormonal medications. She had had three pregnancies of which one resulted in term birth but two DISCUSSION were terminated by medical abortion. Endometriosis is an oestrogen-dependent disease By laboratory investigation, the levels of red blood characterized by functional endometrial tissue cells (RBC), white blood cells and platelets as well as outside the uterus. Most commonly it affects pelvic biochemical parameters (ASAT, ALAT, creatinine, tissues and organs but any system can be involved, sodium, potassium, C-reactive protein) were within including gastrointestinal tract, kidneys, spleen, lungs laboratory reference ranges. The gynaecologic and other sites. The cause of endometriosis is largely ultrasound evaluation data were consistent with unknown. Its incidence in the developed Western uterine adenomyosis and endometrioid cyst of the right countries is increasing during the previous 4-5 decades ovary, measuring 2.0x1.7 cm. The pulmonary function [Porth, 2011]. The disorder mostly develops during tests yielded no abnormality. By thoracic radiography, reproductive age. Its prevalence has been estimated sharply demarcated smooth nodule, measuring 2.2 as 1-10% of women reaching even 15-25% among 38 ACTA CHIRURGICA LATVIENSIS • 2014 (14/1) infertile ladies [Ellenson et al., 2010; Machairiotis et al., REFERENCES 2013]. Extra-pelvic endometriosis is more frequent in 1. Augoulea A, Lambrinoudaki I, Christodoulakos G. females aged 35-40 years. The clinical picture can be Thoracic endometriosis syndrome // Respiration, variable depending on the localization of endometriosis 2008; 75:113 – 119 but particularly it is characterized by abnormal bleeding 2. Bergqvist A. Different types of extragenital or pain [Machairiotis et al., 2013]. In recent years, endometriosis: a review // Gynecol Endocrinol, treatment of extra-pelvic endometriosis has shifted from 1993; 7:207 – 221 medical management toward a surgical approach as 3. Ellenson LH, Pirog EC. The female genital tract // surgery clearly improves disease outcome [Veeraswamy In: Robbins I, Stanley L, Kumar V. Pathologic basis et al., 2010]. of disease. 8th ed. Philadelphia: Saunders Elsevier; Endometriosis of the lung is rare. About 150 cases 2010; 721 have been reported in the lungs and pleura; pleural 4. Huang H, Li C, Zarogoulidis P, Darwiche K, endometriosis is by far the commonest type among Machairiotis N, Yang L, Simoff M, Celis E, Zhao these [Bergqvist, 1993; Sutton et al., 2006]. The patients T, Zarogoulidis K, Katsikogiannis N, Hohenforst- may present with symptoms such as shortness of breath, Schmidt W, Li Q. Endometriosis of the lung: report chest pain or they may be asymptomatic [Nezhat et al., of a case and literature review // Eur J Med Res, 2012]. An important diagnostic issue includes the cyclic 2013; 18:13 nature of the symptoms which frequently occur along 5. Machairiotis N, Stylianaki A, Dryllis G, Zarogoulidis with the menstrual cycle. Regarding the association with P, Kouroutou P, Tsiamis N, Katsikogiannis N, Sarika menstrual cycle, the thoracic endometriosis can manifest E, Courcoutsakis N, Tsiouda T, Gschwendtner A, as catamenial pneumothorax, haemothorax, chest Zarogoulidis K, Sakkas L, Baliaka A, Machairiotis C. pain or haemoptysis, non-catamenial endometriosis- Extrapelvic endometriosis: a rare entity or an under related pneumothorax and lung nodules [Rousset et diagnosed condition? // Diagn Pathol, 2013; 8:194. al., 2014]. Distant spread of endometriosis is explained 6. Nezhat C, King LP, Paka C, Odegaard J, Beygui R. by hypothesis of venous or lymphatic circulation Bilateral thoracic endometriosis affecting the lung [Augoulea et al., 2008; Huang et al., 2013] despite the and diaphragm // JSLS, 2012; 16:140 – 142 fact that endometriosis is not a malignant tumour. 7. Porth CM. Essentials of pathophysiology. 3rd ed. Pulmonary endometriosis requires careful differential Philadelphia: Lippincott Williams & Wilkins; 2011; diagnosis, including lung cancer [Yu et al., 2013]. In 1047 – 1048 our case, the characteristic catamenial complaints were 8. Rousset P, Rousset-Jablonski C, Alifano M, absent and the menopausal age was not typical for Mansuet-Lupo A, Buy JN, Revel MP. Thoracic endometriosis causing further confusion. Pulmonary endometriosis syndrome: CT and MRI features // endometriosis is a diagnosis of exclusion. Neither Clin Radiol, 2014; 69:323 – 330 computed tomography nor endoscopy is specific for it 9. Sutton C, Adamson DG, Jones JD. Modern [Augoulea et al., 2008]. Magnetic resonance imaging management of endometriosis. United Kingdom: can be superior by showing haemorrhage [Rousset et al., Taylor & Francis; 2005; 410 2014]. For the establishing of diagnosis it is important 10. Verasswamy A, Lewis M, Mann A, Kotikela S, to interpret clinical and radiological data together Hajhosseini B, Nezhat C. Extragenital endometri- with morphological investigation from the biopsies or osis // Clin Obstet Gynecol, 2010; 53:449 – 466 operation material. 11. Yu JH, Lin XY, Wang L, Liu Y, Fan CF, Zhang Y, In conclusion, the reported case represents an unusual Wang EH. Endobronchial endometriosis presenting distant extra-pelvic manifestation of nodular pulmonary as central-type lung cancer: a case report // Diagn endometriosis in an atypical age. Increased awareness Pathol, 2013; 8:53 of this diagnosis is necessary to avoid over-diagnosis of lung cancer. Morphologic evaluation represents the gold standard of evidence-based diagnosis, and can be Address: further supported by immunohistochemistry. Agita Jukna Department of Pathology, Riga Stradins University Conflict of interest: None Dzirciema Street 16, Riga, LV-1007, Latvia E-mail: agita.jukna@gmail.com 39 ACTA CHIRURGICA LATVIENSIS • 2014 (14/1) Fig. 1. Tissue structure of the pulmonary endometriosis. 1A. Overview of the cystic and nodular architecture. Haematoxylin-eosin (HE), original magnification (OM) 50x. 1B. Fragment of endometrial cyst wall lined by low cubical epithelium. Note the lack of cell anaplasia. HE, OM 400x. Fig. 2. Immunophenotype of pulmonary endometriosis. 2A, Progesterone receptor (PR) expression in pulmonary endometriosis. Note the lack of reactivity in lung tissues. Immunoperoxidase (IP), anti-PR, OM 100x. 2B, Absence of TTF-1 in the endometriosis cells. IP, anti-TTF-1, OM 100x. http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png Acta Chirurgica Latviensis de Gruyter

Pulmonary Endometriosis - a Rare Differential Diagnosis of Lung Cancer

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Abstract

10.2478/chilat-2014-0108 ACTA CHIRURGICA LATVIENSIS • 2014 (14/1) CASE REPORT Pulmonary Endometriosis – a Rare Differential Diagnosis of Lung Cancer Agita Jukna, Ilze Strumfa, Inese Drike, Andrejs Vanags, Janis Gardovskis Riga Stradins University, Latvia SUMMARY Endometriosis is characterised by the presence of ectopic functional endometrial tissue outside the uterus. The disease most frequently affects pelvic tissues and organs but any site can be involved including gastrointestinal tract, lungs and other organs. Extra-pelvic endometriosis mostly occurs in females aged 35-40 years. Here we report a well-documented, morphologically proved and surgically treated case of pulmonary endometriosis in a 50-year-old woman in order to heighten the awareness of endometriosis as the differential diagnosis for nodular lung lesions. Key words: endometriosis, lung, lung tumour, differential diagnosis th AIM OF THE DEMONSTRATION cm in diameter, was detected in the 8 segment of the The aim of this demonstration is to report a rare case left lung. Thoracotomy and marginal resection of the of extra-pelvic endometriosis in order to increase the affected segment was performed. Cytological smear of awareness about endometriosis as the differential the operation material showed cylindrical epithelial diagnosis of lung cancer. cells and groups of proliferating cells with traits of atypia on a RBC-rich background. Cytologist thus CASE REPORT could not exclude malignant tumour. The resected lung A fifty-year-old female underwent routine chest fragment with a collapsed subpleural cavity measuring roentgenography. As a peripheral nodule was disclosed 1 cm in diameter was submitted to the pathological in her left lung, the family doctor referred the patient to examination. By histology, the resection margins were thoracic surgeon. To decide about the further assessment composed of lung tissue lacking any disease process. and treatment strategy, chest computed tomography The wall of the cystic nodule had dual structure with was performed repeatedly after a month. However, the hypercellular stroma which consisted of small spindled diagnosis was not clear and tumour could not be ruled cells and dilated cystic glands lined by low cubic out completely, therefore the lady was admitted to the epithelium (Fig. 1). None of these components showed hospital for surgical treatment. cell atypia. By immunohistochemistry, the pathological The patient had no complaints. However, on detailed tissues expressed oestrogen and progesterone receptors questioning she recalled occasional, dry cough. The lady but lacked pulmonary markers including TTF-1 (Fig. 2). had had no treatment or specific examination due to The proliferation fraction (by Ki-67) and oncoprotein non-disturbing intensity of these symptoms, and also spectrum (p53, Bcl-2) did not suggest malignant tumour. was unable to describe the duration of the cough. Her Thus, endometriosis was diagnosed in accordance with gynaecological anamnesis was characterised by irregular the morphological structure and immunophenotype. menstruations during the last year. Previously, her There were no postoperative complications. The patient th menstrual cycle was regular, with the average duration was released from hospital on 9 postoperative day for 28 days and bleeding of normal intensity lasting 3-4 further treatment under supervision of gynaecologist days. The menstrual bleeding was not preceded by or with the final diagnosis of nodular endometriosis in the th associated with pain, and the patient did not reveal 8 lung segment. use of any hormonal medications. She had had three pregnancies of which one resulted in term birth but two DISCUSSION were terminated by medical abortion. Endometriosis is an oestrogen-dependent disease By laboratory investigation, the levels of red blood characterized by functional endometrial tissue cells (RBC), white blood cells and platelets as well as outside the uterus. Most commonly it affects pelvic biochemical parameters (ASAT, ALAT, creatinine, tissues and organs but any system can be involved, sodium, potassium, C-reactive protein) were within including gastrointestinal tract, kidneys, spleen, lungs laboratory reference ranges. The gynaecologic and other sites. The cause of endometriosis is largely ultrasound evaluation data were consistent with unknown. Its incidence in the developed Western uterine adenomyosis and endometrioid cyst of the right countries is increasing during the previous 4-5 decades ovary, measuring 2.0x1.7 cm. The pulmonary function [Porth, 2011]. The disorder mostly develops during tests yielded no abnormality. By thoracic radiography, reproductive age. Its prevalence has been estimated sharply demarcated smooth nodule, measuring 2.2 as 1-10% of women reaching even 15-25% among 38 ACTA CHIRURGICA LATVIENSIS • 2014 (14/1) infertile ladies [Ellenson et al., 2010; Machairiotis et al., REFERENCES 2013]. Extra-pelvic endometriosis is more frequent in 1. Augoulea A, Lambrinoudaki I, Christodoulakos G. females aged 35-40 years. The clinical picture can be Thoracic endometriosis syndrome // Respiration, variable depending on the localization of endometriosis 2008; 75:113 – 119 but particularly it is characterized by abnormal bleeding 2. Bergqvist A. Different types of extragenital or pain [Machairiotis et al., 2013]. In recent years, endometriosis: a review // Gynecol Endocrinol, treatment of extra-pelvic endometriosis has shifted from 1993; 7:207 – 221 medical management toward a surgical approach as 3. Ellenson LH, Pirog EC. The female genital tract // surgery clearly improves disease outcome [Veeraswamy In: Robbins I, Stanley L, Kumar V. Pathologic basis et al., 2010]. of disease. 8th ed. Philadelphia: Saunders Elsevier; Endometriosis of the lung is rare. About 150 cases 2010; 721 have been reported in the lungs and pleura; pleural 4. Huang H, Li C, Zarogoulidis P, Darwiche K, endometriosis is by far the commonest type among Machairiotis N, Yang L, Simoff M, Celis E, Zhao these [Bergqvist, 1993; Sutton et al., 2006]. The patients T, Zarogoulidis K, Katsikogiannis N, Hohenforst- may present with symptoms such as shortness of breath, Schmidt W, Li Q. Endometriosis of the lung: report chest pain or they may be asymptomatic [Nezhat et al., of a case and literature review // Eur J Med Res, 2012]. An important diagnostic issue includes the cyclic 2013; 18:13 nature of the symptoms which frequently occur along 5. Machairiotis N, Stylianaki A, Dryllis G, Zarogoulidis with the menstrual cycle. Regarding the association with P, Kouroutou P, Tsiamis N, Katsikogiannis N, Sarika menstrual cycle, the thoracic endometriosis can manifest E, Courcoutsakis N, Tsiouda T, Gschwendtner A, as catamenial pneumothorax, haemothorax, chest Zarogoulidis K, Sakkas L, Baliaka A, Machairiotis C. pain or haemoptysis, non-catamenial endometriosis- Extrapelvic endometriosis: a rare entity or an under related pneumothorax and lung nodules [Rousset et diagnosed condition? // Diagn Pathol, 2013; 8:194. al., 2014]. Distant spread of endometriosis is explained 6. Nezhat C, King LP, Paka C, Odegaard J, Beygui R. by hypothesis of venous or lymphatic circulation Bilateral thoracic endometriosis affecting the lung [Augoulea et al., 2008; Huang et al., 2013] despite the and diaphragm // JSLS, 2012; 16:140 – 142 fact that endometriosis is not a malignant tumour. 7. Porth CM. Essentials of pathophysiology. 3rd ed. Pulmonary endometriosis requires careful differential Philadelphia: Lippincott Williams & Wilkins; 2011; diagnosis, including lung cancer [Yu et al., 2013]. In 1047 – 1048 our case, the characteristic catamenial complaints were 8. Rousset P, Rousset-Jablonski C, Alifano M, absent and the menopausal age was not typical for Mansuet-Lupo A, Buy JN, Revel MP. Thoracic endometriosis causing further confusion. Pulmonary endometriosis syndrome: CT and MRI features // endometriosis is a diagnosis of exclusion. Neither Clin Radiol, 2014; 69:323 – 330 computed tomography nor endoscopy is specific for it 9. Sutton C, Adamson DG, Jones JD. Modern [Augoulea et al., 2008]. Magnetic resonance imaging management of endometriosis. United Kingdom: can be superior by showing haemorrhage [Rousset et al., Taylor & Francis; 2005; 410 2014]. For the establishing of diagnosis it is important 10. Verasswamy A, Lewis M, Mann A, Kotikela S, to interpret clinical and radiological data together Hajhosseini B, Nezhat C. Extragenital endometri- with morphological investigation from the biopsies or osis // Clin Obstet Gynecol, 2010; 53:449 – 466 operation material. 11. Yu JH, Lin XY, Wang L, Liu Y, Fan CF, Zhang Y, In conclusion, the reported case represents an unusual Wang EH. Endobronchial endometriosis presenting distant extra-pelvic manifestation of nodular pulmonary as central-type lung cancer: a case report // Diagn endometriosis in an atypical age. Increased awareness Pathol, 2013; 8:53 of this diagnosis is necessary to avoid over-diagnosis of lung cancer. Morphologic evaluation represents the gold standard of evidence-based diagnosis, and can be Address: further supported by immunohistochemistry. Agita Jukna Department of Pathology, Riga Stradins University Conflict of interest: None Dzirciema Street 16, Riga, LV-1007, Latvia E-mail: agita.jukna@gmail.com 39 ACTA CHIRURGICA LATVIENSIS • 2014 (14/1) Fig. 1. Tissue structure of the pulmonary endometriosis. 1A. Overview of the cystic and nodular architecture. Haematoxylin-eosin (HE), original magnification (OM) 50x. 1B. Fragment of endometrial cyst wall lined by low cubical epithelium. Note the lack of cell anaplasia. HE, OM 400x. Fig. 2. Immunophenotype of pulmonary endometriosis. 2A, Progesterone receptor (PR) expression in pulmonary endometriosis. Note the lack of reactivity in lung tissues. Immunoperoxidase (IP), anti-PR, OM 100x. 2B, Absence of TTF-1 in the endometriosis cells. IP, anti-TTF-1, OM 100x.

Journal

Acta Chirurgica Latviensisde Gruyter

Published: Nov 24, 2014

Keywords: Medicine; Clinical Medicine; Surgery; Surgery, other

References