Chest
Volume 115 • Number 5 • May 1999
Copyright © 1999 The American College of Chest Physicians

Selected Reports

Dr.Sinan DOĞANTÜRK

Ankara

A Case of Endobronchial Endometriosis Treated by Subsegmentectomy

 

Yasuji Terada ¹ MD, FCCP

Fengshi Chen ¹ MD

Tsuyoshi Shoji ¹ MD

Harumi Itoh ² MD

Hiromi Wada ¹ MD

Shigeki Hitomi ¹ MD, FCCP

¹ Departments of Thoracic Surgery (Drs. Terada, Chen, Shoji, Wada, and Hitomi)
² Radiology (Dr. Itoh), Kyoto University Hospital, Kyoto, Japan.

Manuscript received April 17, 1998
revision accepted December 15, 1998.

Correspondence to: Yasuji Terada, MD, Department of Thoracic Surgery, Kyoto University Hospital, Kyoto 606-8507, Japan; e-mail: [email protected]

We present a case of endobronchial endometriosis with catamenial hemoptysis. The lesion was diagnosed as endobronchial endometriosis using helical CT, and the patient underwent a subsegmentectomy of the upper part of the lateral basal segment. A histopathologic examination of the resected specimen revealed findings typical of endobronchial endometriosis with intimal hyperplasia within the bronchus. Since the operation, the patient has been asymptomatic for 11 months with no recurrence of hemoptysis.Key words: catamenial hemoptysis; endobronchial; endometriosis; helical CT; subsegmentectomy Abbreviations: GRH = gonadotropin-releasing hormone; S9a = upper part of the lateral basal segment

 

Periodic hemoptysis occurring in association with the menses (catamenial hemoptysis) is a rare condition. Since the first published case, which attributed the condition to endometriosis of the lung, ^[1] there have been 30 reported cases, ^{[1] [2] [3] [4] [5] [6] [7] [8] [9] [10] [11] [12] [13] [14] [15] [16] [17] [18] [19] [20] [21] [22] [23] [24] [25] [26]} all of which have been attributed to pulmonary endometriosis, although less than one third of these cases have had supportive histologic evidence. In this report, we present a case of pulmonary endometriosis that was characterized by catamenial hemoptysis. The lesion was diagnosed as endobronchial endometriosis by helical CT, and the patient underwent a subsegmentectomy of the upper part of the lateral basal segment (S9a). A histopathologic examination of the resected specimen revealed endobronchial endometriosis.

 

Case Report

A 29-year-old woman presented with a 1-year history of recurrent hemoptysis occurring during every menstrual cycle. At the age of 20, she had given birth. At the age of 27, she had undergone a partial oophorectomy for a right serous cystadenoma and a left dermoid cyst of the ovary. In July 1996 (at the age of 28), she experienced the first episode of catamenial hemoptysis. Occurring just after the start of the menses, the initial hemoptysis episode lasted for a few days and resolved spontaneously. In the following months, she had hemoptysis during every menses. Three months after the onset of symptoms, the patient began danazol therapy at an initial dosage of 400 mg/d, and the treatment was continued for 6 months while she remained amenorrheic. After the patient discontinued the danazol regimen, the hemoptysis recurred at the next menses. She refused further treatment with danazol because of its side effects, and she declined to have an oophorectomy because of her wish to become pregnant. Therefore, she was admitted to our hospital for surgical treatment. She was otherwise asymptomatic, without any associated chest or abdominal discomfort or pain, or dysmenorrhea. A clinical examination, including a gynecologic examination and a pelvic ultrasonography, was performed. The laboratory investigations revealed a normal full blood count, and the erythrocyte sedimentation rate, the urea and electrolyte levels, the liver function and coagulation parameters, and the serum gonadotropin levels were also normal. A bronchoscopic study during and after the menses also showed no abnormality or active bleeding; therefore, no biopsy or bronchial washing was performed. At the time of hemoptysis, there was a slight infiltration shadow in the right lower lung field on the chest radiographs, but this shadow disappeared after the menses. A CT scan of the lung was performed before and during the menses. Before the menses,

1476

there was a nonspecific endobronchial lesion in S9a, and a fairly well demarcated area with blood aspiration was evident during the menses (Fig 1) .

It was assumed that the lesion was endometriosis, and a thoracotomy was performed on November 18, 1997 using an anteroaxillary incision. During the operation, S9a appeared normal and no tumor was detected. Because the lesion was located near the hilum, a wedge resection was not performed. After confirming the pulmonary artery, the bronchus, and the pulmonary veins of the subsegment, a subsegmentectomy of S9a was performed. Macroscopically, the endobronchial space in the upper part of the lateral basal bronchus was filled with endometrium, and a histopathologic examination of the resected specimen revealed findings typical of endobronchial endometriosis with intimal hyperplasia within a bronchus (Fig 2) . In the distal part of the resected specimen, ciliated columnar epithelium was replaced by endometrium, although the smooth muscle layer was retained (Fig 3) . The postoperative course was uneventful. The patient has now been asymptomatic for 11 months without a recurrence of hemoptysis.

 

Discussion

Thoracic endometriosis can involve the pleura and can manifest itself as a catamenial hemothorax or pneumothorax, or it can involve the pulmonary parenchyma, resulting in catamenial hemoptysis. In a review ^[27] of 65 cases of thoracic endometriosis collected up to 1981, 54 cases (83%) were pleural and only 11 cases (17%) were parenchymal. The latter 11 cases were designated as parenchymal disease by a chest radiograph, which showed coin lesions, and by catamenial hemoptysis or localization of the pulmonary bleeding site . Catamenial hemoptysis is rare, with only 30 reported cases ^{[1] [2] [3] [4] [5] [6] [7] [8] [9] [10] [11] [12] [13] [14] [15] [16] [17] [18] [19] [20] [21] [22] [23] [24] [25] [26]} in the English literature since Latters et al ^[1] first described cyclical hemoptysis associated with the menses due to pulmonary endometriosis (Table 1) . All of the cases reported were attributed to pulmonary endometriosis, although histopathologic confirmation of the diagnosis was obtained in only one third of the cases. ^[28]

There are several hypotheses for the cause of extraperitoneal endometriosis. ^{[29] [30] [31]} Pleural endometriosis may result either from local metaplasia of the celomic epithelium

Figure 1. Left: Before the menses, there was a region of nonspecific bronchial thickening in S9a. Right: During the menses, a fairly well-demarcated area with blood oozing from the nodule was evident.

Figure 2. Left: Macroscopically, the endobronchial space of the lateral basal bronchus was filled with endometrium. Right: The histopathologic findings of the resected specimen revealed findings typical of endobronchial endometriosis with intimal hyperplasia within a bronchus (hematoxylin-eosin, original ×10).

(the metaplasia theory) or from retrograde menses with a transdiaphragmatic passage and the subsequent implantation of endometrium inside the thoracic cavity (the transplantation theory). In contrast, parenchymal endometriosis is thought to result from the filtering function of the pulmonary vascular network with the trapping of endometrial particles that, through a hematogenous or lymphogenous process, have spread from the pelvic organs, often following surgery or childbirth. ^[32] In the present case, the patient's history of ovarian tumor surgery might have been the cause.

It is often difficult to diagnose parenchymal pulmonary endometriosis. The most important criterion is the patient's history of catamenial symptoms. The diagnostic use of bronchoscopy is limited because most cases of pulmonary endometriosis involve the distal pulmonary parenchyma rather than the mucosa of the large bronchi, and because the bleeding site may only be evident during the

Figure 3. The distal part of the histopathologically examined resected specimen. To the right of the arrow, ciliated columnar epithelium has been replaced by endometrium, although the smooth muscle layer has been retained. Hemosiderosis can be seen in the lung parenchyma (hematoxylin-eosin, original ×40).

1477

 

*VATS = video-assisted thoracoscopic surgery.

menses. A tissue diagnosis has been obtained in only four reported cases, two following a transbronchial biopsy ^{[7] [24]} and two by cytologic diagnosis following a bronchial lavage. ^{[10] [16]} Although chest radiographs often show normal findings, they can reveal solitary or multiple pulmonary nodules displaying cyclical changes in size. CT findings in pulmonary endometriosis may include ill-defined or well-defined opacities, nodular lesions, thin-walled cavities, or bullous formations, ^{[17] [23]} but most of the cases examined during hemoptysis have revealed transient radiologic densities in the affected part of the lung. In the present case, helical CT confirmed the presence of an endobronchial lesion preceding the menses, as well as parenchymal blood oozing around the lesion during the menses.

The treatment of pulmonary endometriosis usually consists of hormonal therapy with danazol or gonadotropin-releasing hormone (GRH) analogs. Danazol is a synthetic steroid with an anti-estrogenic and weakly androgenic effect; however, side effects, such as weight gain, climacteric symptoms, and virilization, are common. The drug is also expensive, and symptoms often recur after therapy is discontinued. GRH analogs inhibit the release of GRH from the pituitary gland, resulting in levels of sex hormones equal to levels seen after surgical castration. The indications for pulmonary surgery are hormonal therapy failure, intolerable drug side effects, or symptom recurrence after the cessation of medical treatment. The longest reported follow-up periods have been 10 months after surgical treatment ^[25] and 12 months after treatment with danazol ^[6] and buserelin. ^[20] Pleural manifestations, however, are often difficult to treat surgically because the lesions tend to be multifocal. ^[33] Therefore, a single focus of bleeding must be conclusively located before surgery. When the lesion is multiple or when its location cannot be detected, an oophorectomy should be considered. In this case, we confirmed the endobronchial lesion by helical CT before surgery and removed it successfully by subsegmentectomy.

References

1. Latters R, Shepard F, Tovell H, et al. A clinical and pathologic study of endometriosis of the lung. Surg Gynecol Obstet 1956; 103:552-558  

2. Rodman MH, Jones CW. Catamenial hemoptysis due to bronchial endometriosis. N Engl J Med 1962; 266:805-808  

3. Assor D. Endometriosis of the lung: report of a case. Am J Clin Pathol 1972; 57:311-315  citation

4. Lindenberg K, Schmid J, Ruttner J, et al. Endometriosis of the lung: case report. Arch Gynakol 1975; 218:219-226  abstract

5. Rosenberg SM, Riddick DH. Successful treatment of catamenial hemoptysis with danazol. Obstet Gynecol 1981; 57:130-132  abstract

6. Ronnberg L, Ylostalo P. Treatment of pulmonary endometriosis with danazol. Acta Obstet Gynecol Scand 1981; 60:77-78  abstract

7.

1478

Suginami H, Hamada K, Yano K. A case of endometriosis of the lung treated with danazol. Obstet Gynecol 1985; 66(3 Suppl):68S-71S  abstract

8. Karpel JP, Appel D, Merav A. Pulmonary endometriosis. Lung 1985; 163:151-159  citation

9. Elliot DL, Barker AF, Dixon LM. Catamenial hemoptysis: new methods of diagnosis and therapy. Chest 1985; 87:687-688  abstract

10. Johnson WM III, Tyndal CM. Pulmonary endometriosis: treatment with danazol. Obstet Gynecol 1987; 69:506-507  abstract

11. Hertzanu Y, Heimer D, Hirsch M. Computed tomography of pulmonary endometriosis. Comput Radiol 1987; 11:81-84  abstract

12. Virutamasen P, Boonjunwetwat D. Menstrual bleeding associated with hemoptysis: a case report. J Med Assoc Thai 1988; 71:115-117  citation

13. Grimm MH, Grady KJ, Golish JA. Bronchopulmonary endometriosis: a rare cause of hemoptysis. South Med J 1988; 81:1198-1199  abstract

14. Lawrence HC III. Pulmonary endometriosis in pregnancy. Am J Obstet Gynecol 1988; 159:733-734  abstract

15. Maguire PJ. Catamenial hemoptysis. J Okla State Med Assoc 1988; 81:729-730  citation

16. Bateman ED, Morrison SC. Catamenial hemoptysis from endobronchial endometriosis: a case report and review of previously reported cases [review]. Respir Med 1990; 84:157-161  citation

17. Katoh O, Yamada H, Aoki Y, et al. Utility of angiograms in patients with catamenial hemoptysis. Chest 1990; 98:1296-1297  abstract

18. Guidry GG, George RB, Payne DK. Catamenial hemoptysis: a case report and review of the literature. J La State Med Soc 1990; 142:27-30  abstract

19. Svendstrup F, Husby H. Parenchymal pulmonary endometriosis. J Laryngol Otol 1991; 105:235-236  abstract

20. Espaulella J, Armengol J, Bella F, et al. Pulmonary endometriosis: conservative treatment with GnRH agonists. Obstet Gynecol 1991; 78:535-537  abstract

21. Kristianen K, Fjeld NB. Pulmonary endometriosis causing hemoptysis: report of a case treated with lobectomy. Scand J Thorac Cardiovasc Surg 1993; 27:113-115  abstract

22. Joseph J, Reed CE, Sahn SA. Thoracic endometriosis: recurrence following hysterectomy with bilateral salpingo-oophorectomy and successful treatment with talc pleurodesis. Chest 1994; 106:1894-1896  abstract

23. Volkart JR. CT findings in pulmonary endometriosis. J Comput Assisted Tomogr 1995; 19:156-157  

24. Kuo PH, Wang HC, Liaw YS, et al. Bronchoscopic and angiographic findings in tracheobronchial endometriosis. Thorax 1996; 51:1060-1061  abstract

25. Cassina PC, Hauser M, Kacl G, et al. Catamenial hemoptysis: diagnosis with MRI. Chest. 1997; 111:1447-1450  full text

26. Morita Y, Tsutsumi O, Taketani Y. Successful hormonal treatment of pulmonary parenchymal endometriosis. Int J Gynaecol Obstet 1997; 59:61-63  citation

27. Foster DC, Stern JL, Buscema J, et al. Pleural and parenchymal pulmonary endometriosis. Obstet Gynecol 1981; 58:552-556  citation

28. Wood DJ, Krishnan K, Stocks P, et al. Catamenial hemoptysis: a rare cause. Thorax 1993; 48:1048-1049  abstract

29. Hobbs JE, Bortnick R. Endometriosis of the lung: an experimental and clinical study. Am J Obstet Gynecol 1940; 40:832-843  

30. Ramey JK, Archer DF. Peritoneal fluid: its relevance to the development of endometriosis. Fertil Steril 1993; 60:1-14  abstract

31. Gaetije R, Kotzian S, Herrmann G, et al. Invasiveness of endometriotic cells in vitro. Lancet 1995; 346:1463-1464  abstract

32. Park W. The occurrence of decidual tissue within the lung: report of a case. J Pathol Bacteriol 1954; 67:563-570  

33. Seltzer VL, Benjamin F. Treatment of pulmonary endometriosis with a long-acting GnRH agonist. Obstet Gynecol 1990; 76:929-931  abstract

 

Ana sayfaya donmek icin tiklayin http://www.geocities.com/sinandoganturk/kadindogum1.html