Complete endobronchial excision of leiomyoma of the bronchus

Complete endobronchial excision of leiomyoma of the bronchus

Complete endobronchial excision of leiomyoma of the bronchus A leiomyoma of the left main-stem bronchus was diagnosed and resected bronchoscopically. ...

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Complete endobronchial excision of leiomyoma of the bronchus A leiomyoma of the left main-stem bronchus was diagnosed and resected bronchoscopically. At repeat bronchoscopic examination and biopsy 14 months later, there was no evidence of any residual or recurrent leiomyoma. The previous world-wide experience is reviewed. and the salient features of the lesion are discussed. The importance of early diagnosis and conservative therapy are emphasized.

David M. Shahian, M.D., and M. Terry McEnany, M.D., Boston and Cambridge, Mass.

Benign tumors of the lung occur infrequently; they represent five to twenty percent of alI solitary pulmonary lesions. 1-7 Most present as asymptomatic peripheral nodules, although benign endobronchial tumors do occur. Primary pulmonary leiomyomas can present either as parenchymal or endobronchial lesions, although only nineteen such endobronchial tumors have been identified. I, 6, 8-23 We present here the second such tumor to have been resected bronchoscopicalIy and emphasize the importance of early diagnosis and a conservative, parenchyma-sparing operation in the management of benign endobronchial lesions.

Case report A 30-year-old woman (MAH No. 163325) first presented to the Mount Auburn Hospital in January, 1976, complaining of progressive shortness of breath and dyspnea on exertion. She had recently been hospitalized at another institution because of a "spontaneous left pneumothorax," and a tube thoracostomy had been performed. Her course at that time had been complicated by prolonged collapse of the left lower lobe, but this ultimately resolved. Because of continuing respiratory symptoms, the patient was referred for further evaluation. The patient denied any history of aspiration or pneumonia, occupational exposure, or thoracic trauma, and she was a nonsmoker. There was no family history of respiratory disease. She had always been healthy and active, but over the previous year she had noted increasing exertional dyspnea, From the Department of Surgery, Harvard Medical School, Boston, Mass., the Thoracic and Cardiovascular Surgical Service, Mount Auburn Hospital, Cambridge, Mass., and the Department of Surgery, Massachusetts General Hospital, Boston, Mass. Received for publication June 7, 1978. Accepted for publication July 7, 1978. Address for reprints: M. Terry McEnany, M.D., Department of Surgery, The Miriam Hospital, 164 Summit Ave., Providence, R. I. 02906.

Fig. 1. Admission chest x-ray film showing hyperinflation of the left lung. particularly upon climbing stairs. She had also been aware of an intermittent "musical sound" over the left hemithorax. At the time of admission to the hospital, the patient was a healthy appearing young woman in no acute distress, with normal vital signs. The trachea was in the midline. Chest examination revealed normal breath sounds on the right side. On the left, the breath sounds were diminished, but there were no rhonchi or wheezes. The left hemithorax was hyperresonant. Results of the cardiac, abdominal, and pelvic examinations were normal.

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Fig. 3. Tomogram reveals lesion arising from inferior surface of the left main-stem bronchus .

Fig. 2. Inspiratory (A) and expiratory (B) roentgenograms demonstrate air-trapping and mediastinal shift.

The admission hemogram and laboratory chemistry profile were normal. Examination of the arterial blood gases with the woman breathing room air revealed a P0 2 of 78 mm. Hg with a PC02 of 33 mm . Hg and a pH of 7.44 . The admission chest roentgenogram showed overinflation of the left lung with a depressed left hemidiaphragm (Fig . I) . Inspiratory and expiratory films confirmed air trapping on the left (Fig. 2). Decreased expansion on the left and a mediastinal shift to that side with rapid inspiration was defined by fluoroscopy. Laminagrams demonstrated a smooth, noncalcified, nearly occlusive nodule arising from the inferior medial surface of the left main-stem bronchus near the carina (Fig. 3). On Jan . 28, 1976, bronchoscopic examination was performed with a rigid ventilating bronchoscope. On the left, I em . distal to the carina, was a glistening, nonvascular, slightly lobulated , irregular, yellow tumor, 1.5 by 2 em. , arising from the medial wall of the main-stem bronchus . Staging biopsies were taken around the base of the lesion and the lobar bronchial orifices; these were reported as chronic bronchitis. The lesion itself was then completely resected in multiple bites through the bronchoscope. Histopathological examination (Fig . 4) revealed a smooth muscle tumor composed of interlacing spindle cells covered by intact but hyperplastic bronchial epithelium . There were no abnormal nuclei. Special stains were confirmatory, and the final pathological diagnosis was benign intrabronchial leiomyoma. Normal bronchial mucosa was present at the resection margins .

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Fig. 5. Distribution of the previously reported endobronchial leiomyomas .

Fig . 4. Microscopic examination demonstrates a benign smooth muscle tumor covered by hyperplastic bronchial epithelium . The intraoperative and postoperative course was uncomplicated. and the patient was discharged several days later. She was completely asymptomatic. The patient was readmitted March 10. 1977, for a followup bronchoscopic examination. result s of which were totally normal. Biopsies of the previous resection site at that time showed normal bronchial mucosa.

Discussion

Although one of the most common benign lesions of the gastrointestinal tract, a leiomyoma is rarely found in the lung . Fewer than 40 have been previously reported . l • 2 . 4 . 6 . 8-38 At the Mayo Clinic, of 130 benign lung tumors found between 1958 and 1968, only two were leiomyomas .! The three most common lesionshamartoma (76.9 percent), benign fibrous mesothelioma (12 .3 percent), and pseudotumors (5 .4 percent)made up 95 percent of that series . Parenchymal pulmonary leiomyomas occur more frequently in women, whereas endobronchial lesions show no such sex predilection . Patients of all ages, from children to the elderly , have been afflicted . The

anatomic location of 19 previously described endobronchial leiomyomas is shown in Fig. 5. Only three were found in main-stem bronchi , the remainder being distributed evenly among the lobes on the right and left sides . Pulmonary leiomyomas are thought to arise from the smooth muscle of bronchi or bronchioles," although some inve stigators have suggested an origin from pulmonary arterioles or embryonic rests. There are no diagnostic gross pathological features. Histologic examination shows densely packed eosinophilic spindle cells in interlacing bundles, with pale elongated nuclei. Trichrome or phosphotungstic acid-hematoxylin stains confirm their smooth muscle origin and differentiate leiomyoma from fibroma, neurofibroma, and neurolemmoma . The tumor is usually covered by bronchial epithelium but can extend beyond it into the lumen. Hyperplasia or squamous metaplasia of the overlying bronchial epithelium can occur, as with other benign bronchial lesions. The tumor does not transgress the connective tissue or cartilagenous components of the bronchial wall. When histologically benign, these lesions behave so biologically as well. Leiom yosarcoma , the malignant counterpart , is more common and should always be considered in the differential diagnosis. Synchronous benign leiomyomas have been reported'" as have metachronous leiomyomas in separate sites .P! "Benign metastasizing Ieiomyomas" have been described in patients with uterine fibroid

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tumors, although the nature of their malignant potential remains controversial. 30. 36. 37, 39 Pulmonary leiomyomas can also occur in association with identical cutaneous lesions." These benign tumors may present as asymptomatic parenchymal nodules, discovered on routine chest roentgenograms. An endobronchial lesion, on the other hand, may not itself be apparent on a plain x-ray film, but its obstructive sequelae suggest its presence. Wheezing, hemoptysis, cough, chest pain, atelectasis, recurrent or atypical pneumonitis, and unexplained dyspnea are the most common premonitory signs. The corresponding chest x-ray findings range from atelectasis and collapse to unilateral emphysema, hyperlucency, and expiratory air-trapping on the affected side.": 38 Our patient presented with a pneumothorax secondary to air trapping. In this group of patients, the danger of postobstructive pneumonitis, bronchiectasis, and ultimate lung destruction places a high premium on correct early diagnosis. Of the 19 previously described leiomyomas within major bronchi, only 10 were diagnosed correctly by bronchoscopic study. Eleven of the 19 were removed by lobectomy, six by pneumonectomy, and only one was removed via the bronchoSCOpe.6 The remaining case was diagnosed at autopsy. There are several significant problems with bronchoscopic diagnosis. The tumor presents no distinguishing gross characteristics, and even microscopic examination may require special stains. In several cases, the lesion has been mistaken for a bronchial adenoma by the surgeon and biopsy has been deferred in favor of thoracotomy and resection. 15. 18 Others have described an inaccurate suspicion of carcinoma, leading to pneumonectomy. 18. 20 Even if a correct diagnosis is made, lung removal may well be necessary because of chronic infection or advanced parenchymal destruction or both.v 9. 12. 21 The importance of early, accurate diagnosis of these lesions cannot be overestimated. Sears and associates" have recently emphasized this point in connection with benign endobronchial polyps. The lesion in this patient was hard, yellow, and glistening, and it did not have the surface characteristics of a bronchial adenoma. Careful biopsy was considered appropriate when there was no significant bleeding on touching the surface, and the quick frozen diagnosis showed it to be benign. Complete endobronchial excision was therefore performed. The site of excision was totally normal more than I year later. A high level of suspicion should be maintained in all patients with obstructive pulmonary symptoms. Plain chest roentgenography is supplemented with tomography, fluoroscopy, and bronchography as indicated. Pulmonary function tests with flow-volume loops may

be helpful. Bronchoscopy is the main-stay of diagnosis. A wary approach to lesions which might be bronchial adenomas is mandatory, but even these may be cautiously touched with the bronchoscope and a biopsy then taken if appropriate. Adequate suction and the capacity to perform an immediate thoracotomy are, of course, required. 41 If the diagnosis of leiomyoma or similar benign lesion is made, and if complete bronchoscopic removal seems impossible, a variety of conservative procedures short of major pulmonary resection are still available. These include bronchotomy and local resection under direct vision or bronchoplastic resection. Jensik and associates" have used the latter technique with good success in bronchial adenomas. Parenchymal resection should be done only for end-stage infection or solitary parenchymal nodules. REFERENCES

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47:179, 1965 7 Steele JD: The solitary pulmonary nodule. J THORAC CARDIOVASC SURG 46:21, 1963 8 Aakus T, Mylius EA: Leiomyoma of the lung. Acta Chir Scand 124:372, 1962 9 Berkheiser SW, Szypulski JT: Leiomyoma of intermediate bronchial origin. J THORAC CARDIOVASC SURG

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