Successful Endoscopic Nd-YAG Laser Treatment of Endobronchial Endometriosis

Successful Endoscopic Nd-YAG Laser Treatment of Endobronchial Endometriosis

References 1 Haljamae H, Enger E. Human skeletal muscle energy metabolism during and after complete tourniquet ischemia. Ann Surg 1975; 182:9 –14 2 Kr...

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References 1 Haljamae H, Enger E. Human skeletal muscle energy metabolism during and after complete tourniquet ischemia. Ann Surg 1975; 182:9 –14 2 Kralovich KA, Obeid FN. Abdominal compartment syndrome. In: Trunkey, DD, Lewis FR, eds. Current therapy of trauma. 4th ed. St. Louis, MO: Mosby, 1999; 360 –363 3 Kaplan LJ, Trooskin SZ, Santora TA. Thoracic compartment syndrome. J Trauma 1996; 40:291–293 4 Milgater E, Uretzky G, Shimon DV, et al. Delayed sternal closure following cardiac operations. J Cardiovasc Surg 1986; 27:328 –331 5 Mestres CA, Pomar JL, Acosta M, et al. Delayed sternal closure for life-threatening complications in cardiac operations: an update. Ann Thorac Surg 1991; 51:773–776 6 Furnary AP, Magovern JA, Simpons KA, et al. Prolonged open sternotomy and delayed sternal closure after cardiac operations. Ann Thorac Surg 1992; 54:233–239 7 Elami A, Permut LC, Laks H, et al. Cardiac decompression after operation for congenital heart disease in infancy. Ann Thorac Surg 1994; 58:1392–1396 8 Raihi M, Tomatis LA, Schlosser RJ, et al. Cardiac compression due to closure of median sternotomy in open-heart surgery. Chest 1975; 67:113–114 9 Odim JNK, Tchervenkov CI, Dobell ARC. Delayed sternal closure: a lifesaving maneuver after early operation for complex congenital heart disease in the neonate. J Thorac Cardiovasc Surg 1989; 98:413– 416 10 Jones ST, Fullerton DA, Campbell DN, et al. Technique to stent the open sternum after cardiac operations. Ann Thorac Surg 1994; 58:1186 –1187

Successful Endoscopic NdYAG Laser Treatment of Endobronchial Endometriosis* Francesco Puma, MD; Angelo Carloni, MD; Giovanni Casucci, MD; Carla Puligheddu, MD; Moira Urbani, MD; and Giuseppina Porcaro, MD

Catamenial hemoptysis is a rare condition that is associated with the presence of intrapulmonary or endobronchial endometrial tissue. Diagnosis of and therapy for this condition are still a matter of debate. We describe a case of endobronchial endometriosis with catamenial hemoptysis. An endobronchial lesion was diagnosed by spiral CT scan, taken at the onset of the menses, and confirmed with flexible bronchoscopy. The patient was successfully treated with endoscopic Nd-YAG laser therapy with a 1-day in-hospital procedure. We suggest that endoscopic laser treatment should be the first line of therapy for central airway endometriosis, provided that the source of bleeding has been conclusively located and all of the lesions can be reached with the bronchoscope. (CHEST 2003; 124:1168 –1170) Key words: endometriosis; female genital disease; hemoptysis; laser surgery; lung diseases; respiratory tract diseases 1168

hemoptysis is a cyclic pulmonary hemorC atamenial rhage that is synchronous with female menses. It is a rare clinical entity, associated with the presence of intrapulmonary ectopic endometrial tissue, and related both to distal parenchymal and to central airway lesions. Fewer than 40 cases of catamenial hemoptysis have been reported, although endometriosis related to trachea and/or large bronchi involvement is even rarer, with ⬍ 10 proven cases reported in the English literature.1–3 Diagnosis of and therapy for this condition are still a matter of debate. The precise source of bleeding is generally not easy to localize because the chest radiograph, CT scan, and bronchoscopy often show normal findings after menses. Histopathologic confirmation of pulmonary endometriosis is also difficult since both biopsy and resected specimens should be obtained just before the onset of the menses.4 The main criterion for the diagnosis is the finding of periodic hemoptysis that is synchronous with menstruation, and most of the reported cases were diagnosed on the basis of the patient’s clinical history, without a supportive histologic demonstration.5 In this report, we present a case of bronchial endometriosis diagnosed by spiral CT scan with virtual bronchoscopy and cured by endoscopic Nd-YAG laser treatment.

Case Report A 25-year-old nonsmoking woman presented for evaluation of recurrent hemoptysis in July 2001. She underwent her first uneventful pregnancy, and the infant was delivered by a cesarean section in July 2000. In February 2001, just after the onset of menses, she suddenly expectorated roughly 10 mL red blood. Hemoptysis lasted for 1 day and stopped spontaneously. There were no other symptoms. Similar episodes occurred on the first day of every following menses until the time of evaluation. The volume of blood varied from 5 to 15 mL. The findings of a chest radiograph and flexible fiberoptic bronchoscopy, which were performed elsewhere in June 2001, were normal. The findings of a physical examination and laboratory study were normal. The patient underwent, after and during menses, high-resolution CT scans of the chest, and a conventional enhanced, single-breathhold, spiral CT scan supplemented with multiplanar reconstructions and virtual bronchoscopy. The CT scan performed after menses did not reveal any bronchopulmonary abnormality. The second examination, obtained at the onset of menses, demonstrated some ground-glass opacifications with a patchy distribution at the level of the left lower lobe that was related to the presence of blood in the alveoli (Fig 1). Furthermore, a small area of bronchial mucosal thickening was observed at the origin of the left upper bronchus by virtual bronchoscopy (Fig 2). These findings were not visible with the first CT scan (Fig 3). A pelvic CT scan and gynecologic examination did not *From the Division of Thoracic Surgery (Drs. Puma, Casucci, Urbani, and Porcaro), University of Perugia, Ospedale Civile S. Maria, Terni, Perugia, Italy; and the Department of Radiology (Drs. Carloni and Puligheddu), Ospedale di Citta` di Castello, Castello, Italy. Manuscript received August 7, 2002; revision accepted March 14, 2003. Reproduction of this article is prohibited without written permission from the American College of Chest Physicians (e-mail: [email protected]). Correspondence to: Francesco Puma, MD, Chirurgia Toracica, Ospedale Civile S. Maria, 05100 Terni, Italy; e-mail: [email protected] Selected Reports

Figure 1. CT scan obtained on the first day of menses. Ground-glass opacification can be seen at the level of the apical segment of the left lower lobe and is related to the presence of blood in the alveoli.

reveal pelvic endometriosis. At the onset of the next menses, the patient underwent flexible fiberoptic bronchoscopy that showed a tiny submucosal red spot at the origin of the left upper bronchus with signs of recent bleeding. No further abnormalities were noticeable. Cytologic examination of the brushing specimens showed clusters of nonmucinous cylindrical cells without a glandular pattern. Options for therapy, including an attempt at endoscopic laser treatment, were discussed with the patient. In September 2001, she was intubated with an 8.5-mm ventilating rigid bronchoscope. The endoscopic findings were totally normal. The mucosa at the origin of the left upper bronchus, on the side of the interlobar carina, was vaporized by Nd-YAG laser, with a total amount of 648 J delivered. The patient was discharged from the hospital 8 h after undergoing the operation. She has remained asymptomatic since having the endoscopic operation, and no recurrence of hemoptysis has been observed with a follow-up of 17 months. The findings of fiberoptic bronchoscopy performed 3 months after rigid bronchoscopy was normal, and the mucosal scar of the previous laser treatment was no longer visible.

Discussion Catamenial hemoptysis is a rare condition that is generally related to pulmonary endometriosis,6 even though

Figure 2. Virtual bronchoscopic image obtained on the first day of menses. The arrow indicates a small (3-mm) thickening of the lateral wall of the left upper bronchus. www.chestjournal.org

Figure 3. Virtual bronchoscopic image obtained after menses showing a normal left upper bronchus.

the possibility of tracheobronchial lesions must be kept in mind. The proper diagnosis and treatment of this condition are still controversial. The role of bronchoscopy and CT scanning in the diagnosis of catamenial hemoptysis remains undefined. For some authors, bronchoscopy is not indicated in the diagnostic workup when clinical and CT scan findings are present.7,8 On the other hand, a low diagnostic yield from the CT scan has been reported in tracheobronchial endometriosis without parenchymal involvement.2 CT scans obtained during menses can demonstrate only small ground-glass opacifications representing the areas of bleeding or nodular lesions, thin-walled cavities, and bullous formations.9 In the interval between menses, the CT scan findings may be normal, or a change in the size of the described lesions may be observed. In patients with central airway endometriosis, a CT scan may show only mild thickening of the bronchial wall.2 In our case, such findings were clearly evident with the virtual bronchoscopy. Flexible fiberoptic bronchoscopy can be normal even if performed within 24 h of the hemoptysis. Multiple normal bronchoscopy findings do not prevent the achievement of a correct diagnosis after serial examinations.1 In other patients, fiberoptic bronchoscopy can clearly localize bleeding without producing a tissue diagnosis because the lesion lay too distal in the bronchial tree.10 In central airway endometriosis, endoscopic findings at the onset of menses vary from single or scattered purplish-red submucosal patches to white cystic lesions.2 The lesions disappear after menses, and a crypt can be observed, as a sign of the cyst’s healing.1 The use of both CT scanning and flexible fiberoptic bronchoscopy are mandatory in the diagnosis of catamenial hemoptysis. The proper timing of such examinations plays the most important role for the diagnosis, and serial diagnostic procedures may be required. Even though central airway endometriosis is a much rarer phenomenon than parenchymal lesions, flexible fiberoptic bronchoscopy within the first day of menses should be performed in every patient with catamenial hemoptysis. In our patient, both the findings of the first CT scan and flexible bronchoscopy performed after menses were normal, but both were important for the diagnosis. The second CT scan CHEST / 124 / 3 / SEPTEMBER, 2003

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conducted during menses showed a small area of bronchial mucosa thickening, particularly evident with the virtual bronchoscopy and not visible during the first examination. Comparisons between the two CT scans led to the correct diagnosis. On the basis of such findings, flexible fiberoptic bronchoscopy, performed on the first day of menses, disclosed a tiny submucosal red spot in the left upper bronchus with signs of recent bleeding. Medical therapy has been recommended as the first choice in pulmonary endometriosis. It consists of the suppression of endometrial tissue with progesterone (ie, pseudopregnancy) or danazol (ie, pseudomenopause). Danazol is a synthetic steroid with antiestrogenic and light androgenic effects that affects ovarian hormone synthesis.11 It has proved to be effective in curing or controlling symptoms, even in patients who are nonresponsive to ovulation suppression,12 but a variable recurrence rate after the cessation of therapy has been reported.1,13 Furthermore, heavy side effects of the hormonal therapy often are observed, including climacteric symptoms, virilization, weight gain, and sterility.4 Surgery should be the preferred method if the patient wishes to become pregnant, if the side effects of hormonal therapy are intolerable, or in case of recurrence when the drug therapy is discontinued. Pulmonary resection is indicated when a single point of bleeding has been located definitively. For peripheral lesions, thoracoscopic wedge resections have been successfully performed.4,14 In patients with centrally located bronchial endometriosis, subsegmentectomy, segmentectomy, or lobectomy are required.3,15–17 Treatment with oophorectomy has been reported in the literature,18 but it seems to be an extreme solution and should be avoided. The precise endoscopic identification of the tracheobronchial lesions brings new therapeutic options. With the combination of CT scanning and flexible fiberoptic bronchoscopy, the source of bleeding should be located in every tracheobronchial endometriosis. In our patient, we decided to proceed to laser treatment because the lesion had been precisely located in a bronchus, which is easily reachable by the bronchoscope. We preferred the ventilating rigid bronchoscopy mainly for our extensive experience in the use of this procedure. Nevertheless, a successful laser ablation of the lesion also could have been performed through the flexible fiberoptic bronchoscope. No previous endoscopic treatments of this condition have been reported in the literature. Endoscopic Nd-YAG laser can eliminate mucosal and submucosal lesions with a minimally invasive procedure, without significant operative risk. Such treatment could be the first line of therapy for central airway endometriosis, provided that the source of bleeding has been conclusively located and the lesions can be reached with the bronchoscope. Endoscopic ablation potentially can achieve good and probably long-term outcomes without the adverse effects of pharmacologic therapy and surgical therapy.

References 1 Bateman ED, Morrison SC. Catamenial hemoptysis from endobronchial endometriosis: a case report and review of 1170

previously reported cases. Respir Med 1990; 84:157–161 2 Wang HC, Kuo PH, Kuo SH, et al. Catamenial hemoptysis from tracheobronchial endometriosis: reappraisal of diagnostic value of bronchoscopy and bronchial brush cytology. Chest 2000; 118:1205–1208 3 Terada Y, Chen F, Shoji T, et al. A case of endobronchial endometriosis treated by subsegmentectomy. Chest 1999; 115:1475–1478 4 Inoue T, Kurokawa Y, Kaiwa Y, et al. Video-assisted thoracoscopic surgery for catamenial hemoptysis. Chest 2001; 120: 655– 658 5 Wood DJ, Krishna K, Stocks P, et al. Catamenial hemoptysis: a rare cause. Thorax 1993; 48:1048 –1049 6 Joseph J, Sahn SA. Thoracic endometriosis syndrome: new observations from an analysis of 110 cases. Am J Med 1996; 100:164 –170 7 Guidry GG, George RB. Diagnostic studies in catamenial hemoptysis. Chest 1990; 98:260 –261 8 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 9 Volkart JR. CT findings in pulmonary endometriosis. J Comput Assisted Tomogr 1995; 19:156 –157 10 Karpel JP, Appel D, Merav A. Pulmonary endometriosis. Lung 1985; 163:151–159 11 Madanes AE, Farber M. Danazol. Ann Intern Med 1982; 96:625– 630 12 Elliot DL, Barker AF, Dixon LM. Catamenial hemoptysis: new methods of diagnosis and therapy. Chest 1985; 87:687– 688 13 Lawrence HC. Pulmonary endometriosis in pregnancy. Am J Obstet Gynecol 1988; 159:733–734 14 Cassina PC, Hauser M, Kacl G, et al. Catamenial hemoptysis: diagnosis with MRI. Chest 1997; 111:1447–1450 15 Assor D. Endometriosis of the lung: report of a case. Am J Clin Pathol 1972; 57:311–315 16 Kristianen K, Fjeld NB. Pulmonary endometriosis causing hemoptysis: report of a case treated with lobectomy. Scand J Thorac Cardiovasc Surg 1993; 27:113–115 17 Weber F. Catamenial hemoptysis. Ann Thorac Surg 2001; 72:1750 –1751 18 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

Successful Management of Pregnancy in a Patient With Eisenmenger Syndrome With Epoprostenol* Chris Geohas, MD; and Vallerie V. McLaughlin, MD, FCCP

Pregnancy in the setting of pulmonary hypertension and Eisenmenger physiology is associated with a substantial maternal and fetal risk. Such patients are advised against pregnancy. We report a case of a woman with an Eisenmenger atrial septal defect diagnosed during the last trimester of pregnancy. On presentation, she was critically ill and there was evidence of fetal distress. She was emergently Selected Reports