Pregnancy-associated idiopathic laryngotracheal stenosis: presentation, management and results of surgical treatment

Eur J Cardiothorac Surg. 2021 Jan 4;59(1):122-129. doi: 10.1093/ejcts/ezaa296.

Abstract

Objectives: Idiopathic laryngotracheal stenosis (ILTS) is an uncommon problem arising mostly in women. In some, it arises during or is exacerbated by pregnancy. Experience with management of patients with this rare association is limited. This study seeks to evaluate the management of patients with pregnancy-associated ILTS and compare outcomes to cases not associated with pregnancy.

Methods: Retrospective review of 15 patients undergoing surgical treatment of pregnancy-associated ILTS from 1971 to 2013. Variables of interest and airway outcomes were compared to patients with non-pregnancy-associated ILTS.

Results: Pregnancy-associated ILTS was observed in 15/263 (5.7%) patients. Symptoms developed during their first pregnancy. When compared to non-pregnancy patients, these patients were younger (37 vs 47 years; P = 0.0003), had more prior tracheostomies (26.7% vs 10.9%; P = 0.085) and had more preoperative airway dilatations (86.7% vs 57.7%; P = 0.030). All patients completed pregnancy without complications and ultimately underwent laryngotracheal resection. The expression of hormonal receptors in the surgical specimens was similar in both groups (oestrogen receptors: 100% vs 75% and progesterone receptors: 71.4% vs 72.1%, in pregnancy and non-pregnancy patients, respectively). Airway outcomes were good/excellent in 13 (86.7%) patients with pregnancy-associated ILTS and 225 (90.7%) patients without pregnancy association (P = 0.642), and did not change when adjusting for other risk factors.

Conclusions: Pregnancy-associated ILTS is rare. The pathophysiology is unclear, but appearance of symptoms during pregnancy may suggest hormonal factors. To minimize foetal risk, dilatation during pregnancy followed by laryngotracheal resection after delivery is the preferred treatment. Pregnancy association does not seem to affect outcomes with expected satisfactory results in most patients.

Keywords: Idiopathic subglottic tracheal stenosis; Laryngotracheal stenosis; Pregnancy; Trachea; Tracheal disease.

MeSH terms

  • Constriction, Pathologic
  • Female
  • Humans
  • Laryngostenosis* / diagnosis
  • Laryngostenosis* / surgery
  • Pregnancy
  • Retrospective Studies
  • Trachea
  • Tracheal Stenosis* / diagnosis
  • Tracheal Stenosis* / surgery
  • Tracheostomy
  • Treatment Outcome