Journal of th e Korean Radiological Society 1995: 33(6) : 899 - 901 CT Findings of Laryngotracheobronchiallnvolvement in Tracheopathia Osteoplastica : A Case Report 1 Jong Deok Kim , M.D. , Hye KyoungYoon , M.D.2 We report a case of tracheopathia osteoplastica (TO) that .involved both larynx and tracheobronchus. On CTscans , the laryngeal andtracheobronchial cartilages werethickened with irregularcalcification . Multiple nodules with orwithout cal - cification were seen protruding into the lumen from the anterior and lateral walls. Index Words : Trachea , CT Larynx , CT INTRODUCTION Tracheopathia osteoplastica(TO) is a rare , benign condition involving the trachea and major bronchi , in which islands of osseous tissue form within the submucosa of the anterior and lateral walls. The pos- ter ior walls are spared , as they contain no cartilage. Lesions involving the larynx have only occasionally been reported(1 - 6) . CASE REPORT A 62 - year - old man was admitted because of severe dyspnea and hoarseness for 45 days , which was aggra- vated progressively with a recent upper respiratory tract infection. He had exper ienced less degree of dyspnea and hoarseness 2 years previously , which had not been worsened until adm ission. Difficulties were present dur ing a tracheostomy with a la rge amount of emphysema along the neck and upper mediastinum. Endoscopy revealed almpst fused vocal cords except a 3 mm opening at the posterior commissural region. CT scans showed irregular enlargement of both arytenoid and cricoid cartilages bilaterally. Glottic and infrag- lottic larynx was nar r owed and elongated , with nodula r calcification in its walls(Fig . 1a). The trachea and right ' Oepartment of O iagnostic R adiology . College of Medicine, Inje U niversity P usan Paik H ospital ' Oepartment of Anatomical Pathology , College of Medicine , Inje Universi ty P usan Paik H ospital Rece i vedJu ly 11 , 1995 ; Accepted October 13, 1995 Address reprint requests to : Jong Oeok Kim , M.D. , Department of Diag n ostic Rad iology , Co li ege of Med i cine , Inje Un ivers i ty P usa n Pa ik H ospi tal, 633-1 65 Kegum.dong , Pu sanjin .ku , Pu san , 614.735 Ko rea. Te l. 82-51-890-6549 F ax 82.51.896. 1085 -8 99 main bronchus were deformed and triangular in shape. Thick , horeseshoe -shaped , irregular calcific deposits were spread cont i nuously along the anterior and lat- eral walls(Fig . 1 b , c). With protrusion into the tracheal and bronchial lumen , tracheal and bronchial lumina were distorted with crescentic configuration . No cal- cific depositis were found in the posterior wall of the trachea and right main bronchus . There was no evi- dence of an extratracheal or extralaryngeal mass explain the cause of these deformity. Biopsy under a ventilating bronchoscope was performed and histo- pathology of specimen revealed resp i ratory mucosa with focal squamous metaplasia and compact bone trabeculae with fatty or fibrotic mar r ow ti ssues in the underlying stroma. Chronic inflammato ry cell infil- tration was associated(Fig. 1 DISCUSSION Tracheopathia Osteoplastica was first described macroscop i cally by Rokitansky in 1855 and mic ro- by Wilks in 1857. Pathologically , the lesions are composed of submucosal islands of hyaline ca rti- lage with areas of lameliar bone and occasional mar- row elements. The mucosal surface is i ntact. A connec- tion to the the perichondrium is often evident , sugge- sting t hat the les ions arise from native cartilage(2 , 5). When the larynx is affected , sympotoms arise at an earlier stage as in this and other cases ; the symptoms may include hoarseness and dyspnea and , because of the close relation to the uppermost part of the eso- phagus , a sensation of foreign body , salivation , pain and dysphagia with weight loss may also occu r( 6) . There has been only a few reports of TO involving the larynx since the first description of the disease more
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Journal of the Korean Radiological Society 1995: 33(6) : 899- 901
CT Findings of Laryngotracheobronchiallnvolvement in Tracheopathia Osteoplastica : A Case Report1
Jong Deok Kim, M.D. , Hye KyoungYoon, M.D.2
We report a case of tracheopathia osteoplastica (TO) that.involved both larynx and tracheobronchus. On CTscans, the laryngeal andtracheobronchial cartilages werethickened with irregularcalcification . Multiple nodules with orwithout cal cification were seen protruding into the lumen from the anterior and lateral walls.
Index Words : Trachea, CT Larynx,CT
INTRODUCTION
Tracheopathia osteoplastica(TO) is a rare , benign condition involving the trachea and major bronchi , in which islands of osseous tissue form within the submucosa of the anterior and lateral walls. The poster ior walls are spared , as they contain no cartilage. Lesions involving the larynx have only occasionally been reported(1 -6).
CASE REPORT
A 62 -year -old man was admitted because of severe dyspnea and hoarseness for 45 days, which was aggravated progressively with a recent upper respiratory tract infection. He had exper ienced less degree of dyspnea and hoarseness 2 years previously , which had not been worsened until admission. Difficulties were present during a tracheostomy with a large amount of emphysema along the neck and upper mediastinum. Endoscopy revealed almpst fused vocal cords except a 3 mm opening at the posterior commissural region. CT scans showed irregular enlargement of both arytenoid and cr icoid cartilages bilaterally. Glottic and infraglottic larynx was narrowed and elongated , with nodular calcification in its walls(Fig. 1 a). The trachea and right
'Oepartment of Oiagnostic Radiology. College of Medicine, Inje University Pusan Paik Hospital 'Oepartment of Anatomical Pathology, College of Medicine , Inje University Pusan Paik Hospital ReceivedJuly 11 , 1995 ; Accepted October 13, 1995 Address reprint requests to : Jong Oeok Kim , M.D. , Department of Diagnostic Radiology, Co li ege of Medicine, Inje Un iversity Pusan Paik Hospi tal, 633-1 65 Kegum.dong, Pusanjin.ku, Pusan, 614.735 Korea. Tel. 82-51-890-6549 Fax 82.51.896.1085
-899
main bronchus were deformed and triangular in shape. Thick , horeseshoe -shaped , irregular calcific deposits were spread continuously along the anterior and lateral walls(Fig. 1 b, c). With protrusion into the tracheal and bronchial lumen , tracheal and bronchial lumina were distorted with crescentic configuration. No cal cific depositis were found in the posterior wall of the trachea and r ight main bronchus. There was no evi dence of an extratracheal or extralaryngeal mass t。
explain the cause of these deformity. Biopsy under a ventilating bronchoscope was performed and histopathology of specimen revealed resp iratory mucosa with focal squamous metaplasia and compact bone trabeculae with fatty or fibrotic marrow tissues in the underlying stroma. Chronic inflammatory cell infiltration was associated(Fig. 1 이
DISCUSSION
Tracheopathia Osteoplastica was first described macroscopically by Rokitansky in 1855 and microSCOplC허 Iy by Wilks in 1857. Pathologically , the lesions are composed of submucosal islands of hyal ine cart ilage with areas of lameliar bone and occasional marrow elements. The mucosal surface is intact. A connection to the the perichondrium is often evident , suggesting that the lesions arise from native cartilage(2 , 5).
When the laryn x is affected , sympotoms arise at an earlier stage as in this and other cases ; the symptoms may include hoarseness and dyspnea and , because of the close relation to the uppermost part of the esophagus , a sensation of foreign body , salivation , pain and dysphagia with weight loss may also occu r(6) . There has been only a few reports of TO involving the larynx since the first description of the disease more
Journal of the Korean Radiological Society 1995; 33(6 ) ; 899- 901
a b
c d Fig. 1. CT scan obtained at the level of infraglottic larynx(a) demonstrates moderate narrowing of laryngeallumen , thickening of cricoid cartilage, and nodular calcifications in the laryngeal wal l. At the tachea(b) and main bronchus(c) levels , trachea and right main bronchu s are deformed with thickened cartilage and calcific deposits protruding into the lumen. Specimen hist이 ogy(d) reveals th ick lam ellated bony trabeculae with fatty marrow tissues under the mucosa with incomplete squamous metaplasia(H & E, X 1 00)
than 100 years ago(6 -9). The histopathologic diagnosis has been made after
endotracheal biopsy , segmental resection of the trachea , or lobectomy. Conventional tomography and computed tomography have been reportedly useful in making the diagnosis(1 -6, 9). On CT scans(1 -3, 5, 9) , the tracheal carti lages are thickened with irregular calcification. Multiple nodules with or without calcification may be seen protruding into the lumen from the anterior and lateral walls. This is considered to be pathognomonic for TO. Typically , a long segment of the trachea is involved with possible extension to the main stem bronchi . Demonstration of calcification is essential for the radiologic diagnosis ofcartilagenous tumors in general and for TO in particular. Conventional tomography and CT are the only modalities for demonstration of the cal c ification. However the CT has been superior to conventional tomography because CT can show that not all of the calcifications are located within the endotracheal nodules , but are also in submucosal
900
plaques , and it reveals nodules devoid of calcification (10) . There are several diffuse diseases of the trachea and main - stem bronchi that decrease the airway diameter ; relapsing polychondritis, amyloidosis , sarcoidosis , Wegener’s granulomatosis , TO, tracheobronchitis associated with ulcerative colitis , saber - sheath trachea , tracheomalacia and bronchomalacia, and infectious disorders. Of these calcification may be seen in relapsing polychondritis , amyloidα3is , TO, and saber - sheath trachea. Relapsing p이ychondritis and amyloidosis are systemic disorders in which the tracheobronchial tree is usually involved as a part of characteristic systemic manifestations. Episodic inflammation of the ears , nose, upper airways , and joints with cauliflower ear, softtissue changes with calcification in the cartilage , and saddle nose deformity are specific findings of relapsing polychondritis. The nodules in amyloidosis may be circumferential , but those of TO typically spare the posterior membranous wal l. Saber sheath trachea affects only the intrathoracic trachea
Jong Deok Kim, et 81: CT Findings of Laryngotracheobronchial Involvement in Tracheopathia Osteoplastica
and the coronal diameter is markedly reduced , resul
ting in a saber - sheath configuration(1 , 5 , 10).
In summary , if, on CT scans , small patchy calcific
deposits were spread along the inner aspect of the
thickened laryngeal and tracheobronchial cartilages , protruding into the lumen and sparing the posterior
wall , TO should be considered.
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main-stem bronchi: Correlation 01 CT with pathologic linding
RadioGraphics 1992 ; 12 : 645-647
2. Chop lin RH , Wehnut WD , Theros EG. Diffuse les ion s 01 the tra
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3. Onitsuka H, Hirose N, Watanabe K, et al. Computed tomography