Buserelin

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.

(CHEST 1999; 115:1475-1478)

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,[1-26] 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, there was a nonspecific endobronchial lesion in S9a, and a fairly well demarcated area with blood aspiration was evident during the menses (Fig 1).

[Figure 1 ILLUSTRATION OMITTED]

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.

[Figures 2-3 ILLUSTRATION OMITTED]

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 parenchymai 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[1-26] 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]

Table 1--Summary of the Literature on Catamenial Hemoptysis(*)

(*) VATS = video-assisted thoracoscopic surgery.

There are several hypotheses for the cause of extraperitoneal endometriosis.[29-31] Pleural endometriosis may result either from local metaplasia of the celomic epithelium (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 menses. A tissue diagnosis has been obtained in only four reported cases, two following a transbronehial 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 in-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 gonadotropinreleasing 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.

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(*) From the Departments of Thoracic Surgery (Drs. Terada, Chen, Shoji, Wada, and Hitomi) and 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: yaterada@kuhp.kyoto-u.ac.jp

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