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

Tebbe, W.; Wittkowski, Helmut; Tebbe, Johannes J.; Hülskamp, Georg

doi:10.3389/fped.2022.888282

Cited by 2 publications

(1 citation statement)

References 97 publications

Supporting

Mentioning

Contrasting

Order By: Relevance

“…A recent analysis published by Hillel et al identified a specific microbiota of Moraxella (Prevotella and Streptococcus) that occurs significantly more often in patients with ITS compared to control group ( 23 ). A possible effect of a locally altered microbiome and a successful therapy of a idiopathic stenosis with macrolides in an 8-year-old child was reported by Tebbe et al ( 24 ). Those might be additional factors favorizing the development of “idiopathic” tracheal stenosis.…”

Section: Discussionmentioning

confidence: 80%

The role of local expression of hormone receptors in the genesis of idiopathic tracheal stenosis

Schweipert¹,

Riediger²,

Balandat³

et al. 2023

J Thorac Dis

View full text Add to dashboard Cite

Background Tracheal stenosis in adults is usually the result of mechanical injuries either from direct trauma, tracheotomy or intubation. Idiopathic stenosis in the cricotracheal section is a rare condition and occurs almost exclusively in females. Therefore, an influence of the female sexual hormones estrogen and progesterone has been assumed previously. Methods Tracheal specimens of 27 patients who received tracheal resection for either idiopathic tracheal stenosis (ITS) (n=11) or posttraumatic tracheal stenosis (PTTS) (n=16) between 2008 and 2019 in our surgical department were included and retrospectively analyzed. Immunohistochemical staining of tracheal specimens concerning the hormone receptor status of progesterone and estrogen was performed. Results While post-tracheotomy stenosis occurred in males (n=6) as well as in females (n=10), none of the patients with idiopathic stenosis were males. All of the idiopathic stenosis (n=11; 100%) showed a strong expression of the estrogen receptors (ERs) in the fibroblasts and expression of progesterone receptors (PRs) in fibroblasts in 8 of 11 (72.7%). In the post-tracheotomy patients, only 3/16 (18.8%) showed slight staining of PRs and 6/16 (37.5%) of ERs. Of those, only one male patient presented with expression of ERs and PRs and another male patient presented with isolated PRs. Oral intake of hormone compounds was seen in 11/27 (40.7%) patients: 7/11 (63.6%) in the ITS group and 4/16 (25%) in the PTTS (noteworthy that the PTTS group included 6 male patients). Conclusions Although the number of patients is small, our results show that the expression of female sexual hormone receptors in the fibroblasts of the trachea is a persistent finding in ITS. Surgery provided good results with a favorable long-term outcome without recurrence of stenosis for ITS and PTTS. Further investigation with a special focus on hormones is needed to assist in the prevention of this rare disease.

show abstract

Section: Discussionmentioning

confidence: 80%

The role of local expression of hormone receptors in the genesis of idiopathic tracheal stenosis

Schweipert¹,

Riediger²,

Balandat³

et al. 2023

J Thorac Dis

View full text Add to dashboard Cite

show abstract

Symptom‐Free Intervals Following Laser Wedge Excision for Recurrent Idiopathic Subglottic Stenosis

Xie,

Bowen,

O'Byrne

et al. 2024

Otolaryngol.--head neck surg.

View full text Add to dashboard Cite

ObjectiveAnalyze the duration of symptom‐free intervals following laser wedge excision (LWE) for recurrent idiopathic subglottic stenosis (iSGS). Secondary aim includes evaluating the influence of patient‐related or disease factors.Study DesignRetrospective review.SettingTertiary center.MethodsReview of iSGS patients who underwent LWE between 2002 and 2021. LWE patients without prior airway surgery were labeled LWE primary (LWEP) and those with prior history of dilation were labeled LWE secondary (LWES). A conditional frailty repeated events model was used to analyze the median time to recurrence (MTR) for each nth recurrence. Secondary analysis included stratification by use of medical therapy and initial preoperative characteristics of scar (Myer‐Cotton grade, distance between the glottis and superior‐most aspect of scar, DGS; length of scar, DL).ResultsTwo hundred and ten iSGS patients underwent LWE (131 LWEP, 79 LWES). The proportion of patients experiencing at least 1, 3, 6, and 12 recurrences, respectively, was 68.0% (n = 143), 40.7% (n = 85), 20.0% (n = 42), and 5.2% (n = 11). There was exponential time‐shortening from the 1st to 12th recurrence (P < .0001). While MTR was 4.1 years after the first LWE, this fell to 2.8, 1.7, 1.0, and 0.7 years for the 2nd, 3rd, 6th, and 12th recurrences. Furthermore, LWEP patients experienced longer MTR than LWES counterparts within the first 6 recurrences (P < .01). There was no significant relationship between intersurgical interval and medication adherence, DL, DGS, or grade for recurrences beyond the first (P = .207, P = .20, P = .43, P = .16).ConclusionSymptom‐free intervals in iSGS shorten with each subsequent recurrence and LWE. The difference in MTR between LWEP and LWES groups was significant within the first 6 recurrences with LWEP having longer MTR.Level of Evidence3.

show abstract

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

Cited by 2 publications

References 97 publications

The role of local expression of hormone receptors in the genesis of idiopathic tracheal stenosis

The role of local expression of hormone receptors in the genesis of idiopathic tracheal stenosis

Symptom‐Free Intervals Following Laser Wedge Excision for Recurrent Idiopathic Subglottic Stenosis

Contact Info

Product

Resources

About