Objectives.
Tracheal compression by postero-lateral displaced origin of the innominate artery is extremely rare.
The management can be complicated by late presentation.

Methods.
An ex-premature triplet presented with nine month history of persistent wheeze with episodes of choking and cyanosis, recurrent chest infections, failure to thrive due to poor feeding.
Physical examination demonstrated wheezing, but no desaturation or stridor. Echocardiogram was normal.
Broncoscopy performed at 10 months of age demonstrated collapse of the anterior tracheal wall with anterior pulsation. Contrast-enhanced CT scan revealed severe reduction of the antero-posterior tracheal diameter by extrinsic compression due to the presence of postero-lateral displaced origin of the innominate artery.
Surgical relief of the extrinsic tracheal compression was therefore indicated.

Results.
Aortopexy was performed via median sternotomy, with mobilization of ascending aorta and innominate artery and anterior suspension through the sternum. Complete relief of the tracheal compression was confirmed by intra-operative bronchoscopy.
Despite uneventful post-operative recovery and discharge on 4th post-operative day, 5 months after surgery the child remains well but with mild stridor and wheezing, suggesting the persistence of some tracheomalacia.

Conclusions.
Because the symptoms of tracheal compression can be deceivingly mild, the diagnosis of anomalous origin and course of the innominate artery can easily be either missed or delayed without a high index of suspicion and a plan of appropriate investigations.
Unfortunately, failure to diagnose and treat the tracheal compression early, results in chronic tracheo-malacia persisting long-term despite adequate surgical relief.
Early referral with appropriate decision-making and management may avoid long-term respiratory problems.