Familial congenital laryngotracheal stenosis: A systematic review

被引：0

作者：

Saniasiaya, Jeyasakthy ^{[1
,2
]}

van der Meer, Graeme ^{[1
]}

Toll, Ed C. ^{[1
]}

McCaffer, Craig ^{[1
]}

Barber, Colin ^{[1
]}

Neeff, Michel ^{[1
]}

机构：

[1] Starship Childrens Hosp, Dept Otorhinolaryngol, Te Whatu Ora Te Toka Tumai, Auckland, New Zealand

[2] Univ Malaya, Fac Med, Dept Otorhinolaryngol, Kuala Lumpur, Malaysia

来源：

INTERNATIONAL JOURNAL OF PEDIATRIC OTORHINOLARYNGOLOGY | 2024年 / 177卷

关键词：

Congenital upper airway obstruction; Familial; Laryngotracheal stenosis; Genetic predisposition; Outcome; SUBGLOTTIC STENOSIS;

D O I：

10.1016/j.ijporl.2023.111841

中图分类号：

R76 [耳鼻咽喉科学];

学科分类号：

100213 ;

摘要：

Background: Congenital laryngotracheal stenosis (CLS) is a rare cause of stridor among newborns. Evidence has shown that several family members can be affected by CLS. Knowledge of the pathophysiology of familial congenital laryngotracheal stenosis (FCLS) will enable more effective therapeutic strategies. Objective: To determine the clinical course and outcome of familial congenital laryngotracheal stenosis (FCLS). Methods: A literature search was conducted over a period of one month (September 2023) by searching several databases to identify studies published from inception to 31st August 2023. Results: Of 256 papers identified, five articles met the inclusion criteria. A total of 17 patients with slight female predominance (59 %) were identified. Familial congenital tracheal stenosis was reported in female twins (100 %). A variety of clinical presentations were listed. An endoscopic airway study was performed on all patients. 64.8 % of the included children were managed surgically. Genetic studies performed on 41 % of children could not locate genetic abnormalities. Conclusion: Consanguinity, twin births, and female gender could be predisposing factors for FCLS, although the quality of evidence is low due to the rarity of the condition.

引用

页数：5

共 22 条

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