Tuberc Respir Dis.  2008 Sep;65(3):235-238.

Tracheopathia Osteochondroplastica with Recurrent Massive Hemoptysis and Atelectasis: A Case Report

• Oh IJ
• Ju JY
• Choi YD

Abstract

We treated a rare case of TO that presented with recurrent massive hemoptysis that resulted in total obstruction of the bronchus intermedius by very large blood clots. Bronchoscopic intervention resulted in a full recovery from the atelectasis. However, there are no guidelines for preventing recurrence of the hemoptysis or disease progression. Conservative and expectant management are used to treat these patients and most do well.

Keyword

Tracheopathia osteochondroplastica; Massive hemoptysis; Atelectasis

MeSH Terms

Bronchi
Disease Progression
Hemoptysis
Humans
Pulmonary Atelectasis
Recurrence

Figure

• 

  Figure 1 (A) The initial chest radiograph shows atelectasis of the right middle and lower lobes. The coronal (B) and sagittal (C) reformatted images of the precontrast CT shows diffuse multiple fine nodularity and calcifications along the anterolateral tracheal and both main bronchial walls (arrows) with sparing of the posterior tracheal wall of (arrowheads) and hemorrhage in the right bronchus intermedius (star) with distal obstructive atelectasis.

• 

  Figure 2 The bronchoscopic findings show (A, B) multiple fine nodules in the artilaginous tracheal wall from just below vocal cord to 2 cm above carina with (C) total obstruction of the right bronchus intermedius by (D) a very large blood clot; bronchoscopic removal of the clot was performed.

• 

  Figure 3 The pathologic findings showed a submuosal nodule with metaplastic bone and cartilage (H&E stain, x40).

• 

  Figure 4 Virtual CT bronchoscope image also shows multiple small irregular nodules along the anterolateral wall of the trachea (arrows) with preservation of the posterior wall (P).

Reference

1. Harma RA, Suurkari S. Tracheopathia chondro-osteoplastica. A clinical study of thirty cases. Acta Otolaryngol. 1977. 84:118–123.

2. Leske V, Lazor R, Coetmeur D, Crestani B, Chatte G, Cordier JF. Groupe d'Etudes et de Recherche sur les Maladies "Orphelines" Pulmonaires (GERM"O"P). Tracheobronchopathia osteochondroplastica: a study of 41 patients. Medicine (Baltimore). 2001. 80:378–390.

3. Park SS, Shin DH, Lee DH, Jeon SC, Lee JH, Lee JD. Tracheopathia osteoplastica simulating asthmatic symptoms. Diagnosis by bronchoscopy and computerized tomography. Respiration. 1995. 62:43–45.

4. Nienhuis DM, Prakash UB, Edell ES. Tracheobronchopathia osteochondroplastica. Ann Otol Rhinol Laryngol. 1990. 99:689–694.

5. Meyer CN, Dossing M, Broholm H. Tracheobronchopathia osteochondroplastica. Respir Med. 1997. 91:499–502.

6. Coetmeur D, Bovyn G, Leroux P, Niel-Duriez M. Tracheobronchopathia osteochondroplastica presenting at the time of a difficult intubation. Respir Med. 1997. 91:496–498.

7. Lee EJ, Ku KM, Lee CK, Lee HK, Kim S, Oh YH, et al. Tracheopathia osteoplastica associated with anthracofibrosis: case report. J Korean Radiol Soc. 2004. 50:251–253.

8. Lee Y, Cho H, Choi S, Choi H, Jung Y, Shin H, et al. Two cases of tracheopathia osteoplastica. Tuberc Respir Dis. 2004. 56:198–202.

9. Moon JY, Yoo JH, Cho Y, Kang HM, Sung DW. One case of tracheopathia osteoplastica. Tuberc Respir Dis. 2001. 51:76–79.

10. Kim HB, Jeon HG, Jang IW, Cho HI. A case of tracheopathia osteoplastica managed with electrical drill. Korean J Otolaryngol - Head Neck Surg. 2000. 43:895–898.

11. Yoon HK, Kim JD. CT findings of laryngotracheobronchial involvement in tracheopathia osteoplastica: a case report. J Korean Radiol Soc. 1995. 33:899–901.

12. Yum HK, Jeon WK, Kim DS. A case of tracheobronchopathia osteoplastica. Tuberc Respir Dis. 1993. 40:714–718.

13. Del Grande F, Russi EW, Hailemariam S, Hauser M. Extensive tracheobronchial wall thickening and luminal narrowing in a patient with longstanding hoarseness. Respiration. 2002. 69:186–190.

14. Yokoyama T, Fujiyoshi Y, Ninomiya H, Tashiro K, Kajiki A, Takamoto M, et al. Tracheobronchopathia osteoplastica with "rock garden" Findings on bronchoscopy. Nihon Kyobu Shikkan Gakkai Zasshi. 1997. 35:477–480.

15. Akyol MU, Martin AA, Dhurandhar N, Miller RH. Tracheobronchopathia osteochondroplastica: a case report and a review of the literature. Ear Nose Throat J. 1993. 72:347–350.