Tracheostomy in children with mucopolysaccharidosis: A systematic review

IF 1.2 4区医学 Q3 OTORHINOLARYNGOLOGY

International journal of pediatric otorhinolaryngology Pub Date : 2024-11-19 DOI:10.1016/j.ijporl.2024.112167

Francesca Galluzzi , Werner Garavello

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引用次数: 0

Abstract

Objectives

Children with mucopolysaccharidosis (MPS) with difficult airways may require tracheostomy, and surgery can be challenging. This review aims to study the indications, surgical aspects, postoperative complications and outcomes of tracheostomy in MPS children.

Methods

A systematic review following Preferred Reporting Items for Systematic Reviews and Meta-Analyses guidelines was conducted. Pubmed and Embase were searched for studies on tracheostomy in children with MPS. The Joanna Briggs Institute was used for quality appraisal.

Results

A total of 9 studies were included. Three were retrospectives, one was prospective, and five were case series. Overall, 34 subjects who underwent a tracheostomy were included. The mean age was 11 years and the male/female ratio was 1.7. MPS II was the most common type included (16/34). The percentage of tracheotomized MPS children was 10 % (ranging from 3.5 % to 15.5 %). Indications for tracheostomy were: progressive or urgent airway obstruction unresponsive to conservative treatments, before a planned surgical procedure, and in case of failed intubation/extubation. Characteristics clinical features make surgery difficult and predispose complications such as tracheal granulations, wound infection, stomal narrowing, persistent of abundant secretions and tracheitis. Considering tracheostomy outcome, two patients underwent decannulation, but one of them required reinsertion tracheostomy after one year. The duration of follow-up was 8.4 and 9.98 years.

Conclusion

Tracheostomy is an effective means of ensuring airway patency in children with MPS and is considered essentially permanent. Management can be challenging due to typical clinical features and progression of MPS. A planned multidisciplinary approach may prevent complications.

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黏多醣症儿童的气管造口术：系统综述

目的患有粘多糖病（MPS）、呼吸道困难的儿童可能需要进行气管切开术，而手术可能具有挑战性。本综述旨在研究 MPS 儿童气管切开术的适应症、手术方面、术后并发症和疗效。方法按照《系统综述和元分析首选报告项目》指南进行系统综述。在 Pubmed 和 Embase 中检索了有关 MPS 儿童气管切开术的研究。结果共纳入 9 项研究。其中三项为回顾性研究，一项为前瞻性研究，五项为病例系列研究。总共纳入了 34 名接受气管造口术的受试者。平均年龄为 11 岁，男女比例为 1.7。MPS II 是最常见的类型（16/34）。接受气管切开术的 MPS 儿童占 10%（从 3.5% 到 15.5% 不等）。气管切开术的适应症包括：对保守治疗无效的进行性或紧急气道阻塞、计划中的外科手术前以及插管/拔管失败的情况。气管造口术的临床特征会给手术带来困难，并容易引起气管肉芽、伤口感染、气管狭窄、持续大量分泌物和气管炎等并发症。考虑到气管造口术的结果，两名患者接受了气管造口术，但其中一人需要在一年后重新插入气管造口术。结论气管造口术是确保 MPS 患儿气道通畅的有效方法，而且基本上是永久性的。由于 MPS 的典型临床特征和病情发展，管理可能具有挑战性。有计划的多学科方法可预防并发症。

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.