Tracheobronchial stents in mucopolysaccharidosis

Int J Pediatr Otorhinolaryngol. 2016 Apr:83:187-92. doi: 10.1016/j.ijporl.2016.02.015. Epub 2016 Feb 22.

Abstract

Introduction: The mucopolysaccharidoses are a group of hereditary disorders pathologically characterized by tissue accumulation of glycosaminoglycans due to deficient lysosomal metabolism which often leads to progressive airway stenosis and respiratory insufficiency. Relentless and treatment-refractory narrowing of the lower airways with ensuing severe limitation of quality of life is a challenging problem in mucopolysaccharidoses.

Case reports: We report 2 cases of MPS (Hunter's and Maroteaux-Lamy's syndrome resp.) in whom tracheal stents were placed to relieve severe tracheal obstruction. The first patient could be weaned from mechanical ventilation after stent placement but showed significant long-term stent-related morbidity. The second patient suffered a severe procedure-related complication due to positioning problems typical for MPS.

Conclusions: Very good short-term success can be achieved with airway stent placement in patients with MPS and severe lower airway stenosis but a high risk of severe complications and important long-term morbidity have to be weighed against potential individual benefit.

Keywords: Airway stenosis; Bronchoscopy; Mucopolysaccharidosis; Tracheal stent.

Publication types

  • Case Reports

MeSH terms

  • Adolescent
  • Airway Obstruction / etiology
  • Airway Obstruction / surgery*
  • Bronchography
  • Child
  • Humans
  • Male
  • Mucopolysaccharidosis II / complications
  • Mucopolysaccharidosis II / surgery*
  • Mucopolysaccharidosis VI / complications
  • Mucopolysaccharidosis VI / surgery*
  • Quality of Life
  • Stents*
  • Tomography, X-Ray Computed
  • Trachea / pathology
  • Trachea / surgery
  • Tracheal Stenosis / etiology
  • Tracheal Stenosis / surgery*
  • Tracheostomy / adverse effects
  • Tracheostomy / methods*