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Endobronchial Hamartoma Associated with Bronchioloalveolar Cell Carcinoma

1988; Elsevier BV; Volume: 94; Issue: 5 Linguagem: Inglês

10.1378/chest.94.5.1094

1931-3543

D. Mompoint, O Groussard, Philippe Greniér,

Head and Neck Anomalies

Computed tomographic findings in a 60-year-old man with lobar bronchioloalveolar cell carcinoma distal to endobronchial hamartoma are described. Computed tomographic findings in a 60-year-old man with lobar bronchioloalveolar cell carcinoma distal to endobronchial hamartoma are described. Computed tomography (CT) findings in a case of central, endobronchial hamartoma associated with lobar bronchioloalveolar carcinoma in the subtended lung parenchyma are described. This pathologic association has not, to our knowledge, been previously reported. A 60-year-old male smoker, with a history of chronic obstructive pulmonary disease, presented with loss of weight and hemoptysis of three months' duration following an episode of bronchial infection. Chest roentgenograms showed a persistent ill-defined alveolar consolidation in apical-posterior and inferior lingular segments of the left upper lobe (Fig 1). Bronchoscopic examination revealed a polypoid, elastic, mobile mass arising from the posterior wall of the left upper lobe bronchus, extending to its superior division and occluding the lingular bronchus. Bronchoscopic biopsy was negative for cancer. Computed tomographic scans showed a smooth-edged endobronchial intraluminal left upper lobe mass, 2 cm wide, extended to the lingular lumen; thin-section scans revealed small foci of fat inside the mass and no detectable calcium deposits (Fig 2). The diagnosis of endobronchial hamartoma was established by CT on the evidence of fat within the lesion. The alveolar consolidation visible in the upper and lower segments of the left upper lobe was considered obstructive pneumonitis.FIGURE 2CT scan at the level of left upper lobe bronchus shows endoluminal, rounded mass with areas of low density (–30 UH) (arrow). A calcification on the anterior bronchial wall is visible.View Large Image Figure ViewerDownload (PPT) Left upper lobectomy was performed. The examination of he specimen disclosed a pedunculated endobronchial tumor, 2 cm long, obliterating the apicodorsal bronchus. The tumor base was implanted on the bifurcation of this bronchus; the head emerged at the surgical section. This white, smooth, firm tumor had a chondroid aspect. There was no wall infiltration; the pulmonary parenchyma of the dorsal and apical segments was infiltrated by an ill-defined, whitish, tumorous tissue (Fig 3). The endobronchial tumor was a hamartochondroma composed of a central, cartilaginous, dystrophic area associated with fibrous and adipose tissue. The surface was lined by a normal bronchial epithelium. The infiltrated dorsal and apical regions corresponded to a bronchioloalveolar cell carcinoma, with mucin-producing cells lining alveolar walls of normal thickness. A lymph node draining the apicodorsal bronchus was involved with carcinoma. Thoracic hamartoma, including central, intrabronchial hamartoma, rarely encountered in comparison to the more common intrapulmonary variety, can be confidently diagnosed with CT. Siegelman et al,1Siegelman S.S. Khouri N.F. Scott Jr., W.W. Leo F.P. Hamper U.M. Fishman E.K. Zerhouni E.A. Pulmonary hamartoma: CT findings.Radiology. 1986; 160: 313-317Crossref PubMed Scopus (244) Google Scholar using thin-section CT, described the criteria for pulmonary hamartoma (diameter of 2.5 cm or less, smooth edge, focal collections of fat or fat alternating with areas of calcification). The same criteria can be confidently extended to endobronchial hamartoma because of its similar histologic composition.2Bateson E.M. Relationship between intrapulmonary and endobronchial cartilage containing tumors (so-called hamartomata).Thorax. 1965; 20: 447-461Crossref Google Scholar Chronic lobar consolidation is a rare pattern of bronchioloalveolar cell carcinoma.3Hill C.A. Bronchioloalveolar carcinoma: a review.Radiology. 1984; 150: 15-20Crossref PubMed Scopus (108) Google Scholar,4Epstein D.M. Gefter W.B. Miller W.T. Lobar bronchioalveolar cell carcinoma.AJR. 1982; 139: 463-468Crossref PubMed Scopus (32) Google Scholar This neoplasm represents 5 percent of primary lung carcinomas.4Epstein D.M. Gefter W.B. Miller W.T. Lobar bronchioalveolar cell carcinoma.AJR. 1982; 139: 463-468Crossref PubMed Scopus (32) Google Scholar Bronchioloalveolar carinoma usually appears radiographically as a unique localized nodule or mass, diffuse nodules, or diffuse areas of consolidation.3Hill C.A. Bronchioloalveolar carcinoma: a review.Radiology. 1984; 150: 15-20Crossref PubMed Scopus (108) Google Scholar, 4Epstein D.M. Gefter W.B. Miller W.T. Lobar bronchioalveolar cell carcinoma.AJR. 1982; 139: 463-468Crossref PubMed Scopus (32) Google Scholar, 5Im J.G. Choi B.Y. Chang K.H. Yeon K.M. Han M.C. Kim C.W. CT findings of lobar bronchioloalveolar carinoma.J Comput Assist Tomogr. 1986; 10: 320-322Crossref PubMed Scopus (12) Google Scholar, 6Fraser R.G. Paré J.A.P. Diagnosis of disease in the chest. WB Saunders, Philadelphia1978: 1055Google Scholar Radiographically, chronic lobar consolidation is nonspecific and may be encountered in a variety of situations, including pneumonia, obstructive pneumonia, lymphoma, and tuberculosis. According to Im et al,5Im J.G. Choi B.Y. Chang K.H. Yeon K.M. Han M.C. Kim C.W. CT findings of lobar bronchioloalveolar carinoma.J Comput Assist Tomogr. 1986; 10: 320-322Crossref PubMed Scopus (12) Google Scholar an air bronchogram made of narrow, elongated, rigid bronchi is a characteristic CT finding in infiltrative lobar bronchioloalveolar carcinoma. The prognosis of lobar bronchioloalveolar carcinoma is poor, comparable to that of diffuse disease.4Epstein D.M. Gefter W.B. Miller W.T. Lobar bronchioalveolar cell carcinoma.AJR. 1982; 139: 463-468Crossref PubMed Scopus (32) Google Scholar Parenchymal consolidation distal to an endobronchial lesion is usually assumed to be secondary to the bronchial obstruction caused by the lesion. However, in rare circumstances, the possibility of coexistent abnormalities should be kept in mind. Alveolar cell carcinoma has been reported in association with scleroderma, diffuse interstitial fibrosis, and mineral oil aspiration pneumonia.6Fraser R.G. Paré J.A.P. Diagnosis of disease in the chest. WB Saunders, Philadelphia1978: 1055Google Scholar,7Felson B. Ralaisomay G. Carcinoma of the lung complicating lipoid pneumonia.AJR. 1983; 141: 901-907Crossref PubMed Scopus (37) Google Scholar Extensive cicatrization often accompanies bronchioloalveolar cell carcinoma,8Naidich D.P. Zerhouni E.A. Hutchins G.M. Genieser N.B. McCauley D.I. Siegelman S.S. Computed tomography of the pulmonary parenchyma. Distal air-space disease.J Thorac Imag. 1985; 1: 39-53Crossref PubMed Scopus (51) Google Scholar in up to 50 percent of cases, according to some researchers. Thus, one is inclined to evoke the possibility in this case that postobstructive pneumonia led to alveolar cell carcinoma. However, since there are no other reports of alveolar cell carcinoma distal to an obstructing neoplasm, their coexistence should be viewed as coincidental.