Mathlouthi A, Ben Rehouma C, Ben M'Rad S, Dumon J F, Labbene N, Driss B, Sellami M, Djenayah F
Centre Laser, Service du Dr J. F. Dumon, Hôpital Sainte-Marguerite, Marseille, France.
Rev Pneumol Clin. 1993;49(3):156-62.
Tracheobronchobathia osteoplastica (TBO) is a benign disease first described in 1855 and rather rare since only 371 cases have been reported so far. The disease is characterized by the presence of subepithelial osteocartilaginous focal lesions without any relation to tracheal rings. The mechanism of its occurrence remains controverted. We report a case of TBO which is particular in that is was associated with atrophic rhinitis, polydactylia and disorder of oesophageal motricity. From a compilation of 113 cases published by French and Anglo-Saxon authors since 1970, it appears that the clinico-radiological and laboratory picture is of little help in the positive diagnosis which in fact rests on three key-points: bronchial endoscopy, pathological examination of bronchial biopsy fragments and thoracic imaging techniques including computerized topography and magnetic resonance imaging. The usually benign course of the disease can be marked by severe complications, such as infections, haemorrhages, acute dyspnoea by extension to the larynx or significant narrowing of the tracheal lumen which may need tracheotomy or surgical resection of the lesions.