Idiopathic cervical tracheal stenosis in an 11-year-old male

Hoffman, Matthew R.; Thompson, Katrina; Beek-King, Jessica Van

doi:10.1016/j.ijporl.2018.05.013

Cited by 2 publications

(11 citation statements)

References 20 publications

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“…It is suspected when other causes of tracheal stenosis are excluded such as congenital narrowing of the trachea, sequelae after intubation, granulomatosis with polyangiitis (Wegener), bilateral vocal cord affections, vocal cord dysfunction, gastro-laryngeal reflux disease and especially asthma ( 2 , 4 , 7 – 11 ). Since spreading into adjacent bronchi can occur ( 8 , 11 – 13 ) some authors call this condition idiopathic tracheal stenosis. The differentiation between the two entities is not well-defined.…”

Section: Reviewmentioning

confidence: 99%

Section: Reviewmentioning

confidence: 99%

“…The mean time interval between onset of symptoms and diagnosis is 2–3 years ( 2 , 4 , 8 , 17 , 18 , 23 ). Most common symptoms are dyspnea ( 2 , 10 , 18 ) both in rest and during exertion ( 8 , 23 , 40 , 41 ), cough, stridor ( 18 , 42 ), hoarseness, and wheezing ( 8 , 10 , 18 , 43 , 44 ). Before establishing diagnosis, patients commonly receive an erroneous diagnosis of chronic obstructive pulmonary disease or asthma ( 10 , 24 , 45 ).…”

Section: Reviewmentioning

confidence: 99%

“…Histologic evaluation of tracheal tissue biopsies usually shows unspecific acute and chronic active inflammation with epithelial ulceration ( 5 , 13 ) and subsequent squamous metaplasia ( 5 , 8 – 10 , 13 , 44 , 57 , 58 ), scattered eosinophilic infiltration ( 13 , 57 ), normal cartilage ( 23 , 57 ), and dilatation of mucus glands and ducts ( 5 , 23 ). Taking biopsies serves mainly to exclude other inflammatory causes of tracheal stenosis ( 4 ) as histopathological findings are not pathognomonic in iSGS.…”

Section: Reviewmentioning

confidence: 99%

“…One report shows that anti-reflux therapy greatly lengthened the interval between recurrent interventions ( 7 ). Among the few reports in children, one affected boy had an episode of gastritis, probably GERD, prior to presentation ( 8 ). A direct causal relationship between iSGS and GERD is questioned by some investigators, who found no difference between history or treatment of reflux in iSGS patients compared to those with granulomatosis-associated tracheal stenosis ( 12 ).…”

Section: Reviewmentioning

confidence: 99%

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Case report: Idiopathic subglottic stenosis in a girl; successful treatment with macrolides

et al. 2022

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An 8-year-old girl presented with treatment-refractory cough and inspiratory stridor. Bronchoscopies showed progressive scarring leading to narrowing of the proximal trachea (Myer-Cotton Grade 2) and epithelial metaplasia of the tracheal and bronchial mucosa. After excluding other causes of congenital and acquired tracheal stenosis, an idiopathic subglottic tracheal stenosis (iSGS) was diagnosed. Because of the patient's young age, a judicious therapeutic approach seemed appropriate. Therapy with azithromycin, followed by roxithromycin, was started. Symptoms almost completely subsided, spirometry normalized, and endoscopic and histologic findings improved considerably. Therapy has been continued for more than 3 years with normal lung function values, and no compromise on physical activities and development. In instances of iSGS, therapy with macrolides is worth considering before more invasive procedures such as dilatation, laser, intralesional injections, or surgical resection are performed.

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