Congenital nasal pyriform aperture stenosis: retrospective case series, systematic review, and pooled analysis

IF 1.2 4区医学 Q3 OTORHINOLARYNGOLOGY

International journal of pediatric otorhinolaryngology Pub Date : 2025-04-26 DOI:10.1016/j.ijporl.2025.112359

Amy E. Ensing, Aseeyah Islam, Dorina Kallogjeri, Judith E.C. Lieu

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引用次数: 0

Abstract

Background

Congenital nasal pyriform aperture stenosis (CNPAS) is a rare and potentially life-threatening source of neonatal upper airway obstruction. Treatment can be medical or surgical. Our objective was to describe CNPAS presentation and management.

Methods

Pooled data analysis of records of children identified with CNPAS from relevant publications and between 2014 and 2024 at our institution was performed. Published studies identified by a medical librarian were assessed independently by two reviewers.

Results

From 210 abstracts, 170 were identified as relevant, and 106 studies were included. Data from 10 children with CNPAS from our institution and 310 from the medical literature were analyzed. The majority 74.2 % (n = 230) of our sample received a surgical intervention for CNPAS. Pyriform aperture diameter of children receiving only medical management (mean = 5.3 mm; SD = 1.6) was not different from that of children who received or were recommended surgery (mean = 5.1 mm; SD = 1.5) with a mean difference of 0.2 mm (95 % CI -0.4 to 0.7). Presence of apnea (adjusted odds ratio [aOR] = 9.1; 95 % CI 2.4 to 35) and stridor/noisy breathing (aOR = 3.9; 95 % CI 1.4 to 10.8) were associated with higher odds of having surgery performed or recommended.

Conclusions

Management decisions in CNPAS have been driven by individual patient presentation rather than pyriform aperture diameter. Clinicians should use pyriform aperture diameter as a diagnostic rather than prognostic tool, as a narrow pyriform aperture diameter may not necessitate surgery in the absence of severe symptoms.

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先天性鼻梨状孔狭窄：回顾性病例系列、系统评价和汇总分析

背景：先天性梨状鼻孔狭窄（CNPAS）是一种罕见且可能危及新生儿上气道阻塞的原因。治疗可以是药物或手术。我们的目的是描述CNPAS的表现和管理。方法对我院2014 - 2024年相关文献中确诊为CNPAS患儿的记录进行随机数据分析。由医学图书管理员确定的已发表的研究由两名审稿人独立评估。结果从210篇摘要中筛选出170篇相关文献，纳入106篇研究。我们分析了本院10例CNPAS患儿和310例医学文献中的数据。我们的样本中，大多数74.2% （n = 230）接受了CNPAS的手术干预。仅接受医疗管理的患儿梨状孔直径(平均5.3 mm；SD = 1.6)与接受或推荐手术的儿童无差异(平均= 5.1 mm；SD = 1.5)，平均差为0.2 mm （95% CI -0.4 ~ 0.7）。存在呼吸暂停(校正优势比[aOR] = 9.1；95% CI 2.4 ~ 35)和喘鸣/嘈杂呼吸(aOR = 3.9；95% CI 1.4 - 10.8)与接受手术或推荐手术的几率较高相关。结论CNPAS的管理决策是由患者的个体表现而不是梨状孔直径驱动的。临床医生应使用梨状孔直径作为诊断工具，而不是预后工具，因为在没有严重症状的情况下，狭窄的梨状孔直径可能不需要手术。

本文章由计算机程序翻译，如有差异，请以英文原文为准。

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来源期刊

International journal of pediatric otorhinolaryngology 医学-耳鼻喉科学

CiteScore

3.20

自引率

6.70%

发文量

276

审稿时长

62 days

期刊介绍： The purpose of the International Journal of Pediatric Otorhinolaryngology is to concentrate and disseminate information concerning prevention, cure and care of otorhinolaryngological disorders in infants and children due to developmental, degenerative, infectious, neoplastic, traumatic, social, psychiatric and economic causes. The Journal provides a medium for clinical and basic contributions in all of the areas of pediatric otorhinolaryngology. This includes medical and surgical otology, bronchoesophagology, laryngology, rhinology, diseases of the head and neck, and disorders of communication, including voice, speech and language disorders.