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.

REFERENCES

1. Kwong JS, Muller NL, Miller RR. Diseases 01 the trachea and

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

chea. Semin Roenlgeno /1983 ; 18: 38-50

3. Onitsuka H, Hirose N, Watanabe K, et al. Computed tomography

。1 tracheopathia osteoplastica. AJR 1983 ; 140 : 268-270

4. Young RH , Sandstorm RE , Mark GJ. Tracheopathia osteopla-

stica. Clinical , radiol ogic , and pathological correlations. J

Thorac CardiovascSurg 1980 ; 79 : 537-541

5. Scully RE , Mark EJ , McNeely WF , McNeely BU. Case records 01

the Massachusetts General Hospital. Weekly clinicopahtological

exercises. Case 46-1992. N Eng J Med 1992 ; 19: 1512-1518

6. Prakash P, Tang E. Tracheopath ia osteoplastica in the larynx. J

Laryngol 010/1985 ; 99 : 305-31 0

7. Way SPB. Tracheopathia osteoplastica. J Clin Palho/1967 ; 20

814-820

8. Pilis 1. Trach eopathia chondro-osteoplastica. Les Bronches

1983; 23 : 6-21

9. Stain JP. Morere P, Woll LM , Nouvet G, Tayot J, Andrieu­

Guitrancourt J. Etude de la Secretion de I’Hormone de crois­

sance dans le cadre des Facteu rs Etiopathogeniques de la

Tracheo-Bronchopathie Chondro-Osteoplastique. Revue fran­

caise des Maladies Respiraloires 1976 ; 4 : 917-924

10. Hirsch M. Goldstein J , Tovi F, Gerzol SG. Diagnosis 01 trach­

eopathi a osteoplastica by computed tomography. Ann 0101

Rh inol Laryngo/ 1985; 94: 217-219

대 한 방 사 선 의 학회 지 1995 ; 33( 6) : 899-901

후두를침범한기관골신생증의 CT 소견 :1예 보고1

1 인제대학교의과대학진단방사선과학교실

2인제대학교 의과대학 해부병리학교실

김 종 덕·윤 혜 경2

기관 글신생즘은 기관과 주기관지의 점막하를 따라 글 · 연굴조직이 형성되어 비후됨으로써 기도의 변형과 협착을 초래하

는 앙성질환이며 드물게 볼 수 있다 기관과 주기관지의 후벽에는 연골이 없기 때문에 침범되지 않고 전벽과 후벽을 침범하

는 것이 다른 질환과 구별되는 특징적인 소견이므로 이 질환의 진단에는 CT가 가장 좋다. 또한 후두가 침범되는 경우는 매우

드물어서 문헌상에 단지 몇 예의 보고만 되어있을 뿐이다.62세된 남자환자에서 후두와 기관 및 주기관지를 연속적으로 침

범한 1예의 기관 글신생증을 문헌고찰과 함께 보고한다.

- 901 -

<: ICR ’96(19th International ç:ongress oJ~ Radiology) 안내

일 시 : 1996년 6월 9일(일 )-13일(목)

장 소 : Beijing, China

Registration

Delegate

Student

Accompaning person

초록마감

1996년 3월 15일까지

제출처 및 연락처

Dai Jianping, MD

ICR ’96 Beijing

1996. 1. 15. 이 전

$50

$50

$50

Dept. of Neuroradiology

Beijing Tiantan Hi spital

# 6 Tiantan Xili, Beijing 10050, China

Tel 86-10- 7021886

Fax 86-10-5112164

1996. 1. 15일 이 후

$75

$75

$75

듬록 및 초록 Form은 본학회로 언락하시면 송부하여 드리겠습니다.

쉽터원고모집안내

현장등록

$100

$100

$100

쉽터의 원고는 방사선의학 또는 영상과 관계가 있는 사진 또는 그림으로서 독자들에게 흥미를 줄 수 있는 내

용이어야합니다.

한페이지 단위가되어야하며 두 페이지 이상인 경우는독립적으로가치가 있어야하며，게재여부는대한방사

선의학회 편집위원회에서 결정하며 게재료는 학회에서 부담함니다.

- 902