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TRACHEAL STENOSIS CAUSED BY TRACHEOPATHIA OSTEOPLASTICA: TREATMENT BY CERVICAL TRACHEAL RESECTION WITH RECONSTRUCTION

Beth Israel Deaconess Medical Center, Boston, MA

INTRODUCTION: Tracheopathia Osteoplastica (TPO) is a disorder characterized by submucosal nodules developing on the cartilaginous tracheal rings sparing the membranous trachea. Patients are predominantly asymptomatic and the disorder is diagnosed as an incidental finding at bronchoscopy, CT or autopsy. Airway obstruction is extremely rare and we report a case of segmental tracheal stenosis from TPO that was effectively treated with cervical tracheal resection and reanastamosis.

CASE PRESENTATION: A 76 year-old male presented with dyspnea of 3 years duration. At presentation, he could only climb one to two flights of stairs. He had cough and had trouble expectorating secretions. He denied any recent or recurrent pulmonary infections, chest pain, weight-loss, changes in voice, orthopnea or lower extremity swelling. His past medical history was unremarkable. He was a lifetime non-smoker. On examination, an audible wheeze was evident, especially with panting. Remainder of his examination was unremarkable. Flexible bronchoscopy revealed a saber sheath shaped trachea, approximation of lateral walls with abnormal cartilaginous growth causing 70–80% stenosis of the proximal trachea, and several submucosal nodules involving the cartilaginous rings of distal trachea, diagnostic for TPO. Computed tomography (CT) scan of the airway confirmed a predominant 4 cm segment of stenosis in proximal trachea. He underwent a cervical tracheal resection and reconstruction with the removal of the stenotic segment and reanastamosis with excellent improvement in symptoms. The resected tracheal segment showed extensive ossification of cartilage, consistent with the diagnosis of TPO.

DISCUSSIONS: The incidence of TPO is unknown as the patients are usually asymptomatic and the diagnosis is made incidentally. When symptomatic, patients present with cough, hemoptysis, dyspnea on exertion, wheeze or recurrent infections. Airway compromise, as seen in our patient, is extremely rare and has been reported in five patients, all requiring surgical interventions^1,2. Non-surgical interventions such as balloon dilation or laser resection or stent placements are unsuccessful due to the rigid nature of the bony lesions. Linear tracheoplasty has been described as a surgical option. Our patient underwent resection of the stenotic cervical tracheal stent with end-to-end anastamosis without post-operative complications and with excellent results.

CONCLUSION: For symptomatic airway obstruction from TPO, tracheal resection with reanastomosis offers an excellent surgical option with prompt improvement in symptoms.

DISCLOSURE: Chakravarthy Reddy, No Financial Disclosure Information; No Product/Research Disclosure Information

REFERENCES:

1. Prakash UB, McCullough AE, Edell ES, et al. Tracheopathia Osteoplastica: Familial occurrence. Mayo Clin Proc1989; 64 (9):1091 –1096
2. Grillo HC, Wright CD. Airway obstruction owing to tracheopathia osteoplastica: Treatment by linear tracheoplasty. Ann Thorac Surg2005; 79 :1676 –1681