Department of Language and Cognition, University College London, Chandler House,2 Wakefield Street, London, WC1N 1PF, UK.
Great Ormond Street Hospital for Children, Great Ormond Street, London, WC1N 3JH, UK.
Orphanet J Rare Dis. 2024 Jul 4;19(1):253. doi: 10.1186/s13023-024-03259-x.
Eating, drinking and swallowing difficulties are commonly reported morbidities for individuals born with OA/TOF. This study aimed to determine the nature and prevalence of eating, drinking and oro-pharyngeal swallowing difficulties reported in this population.
A systematic review and meta-proportional analysis were conducted (PROSPERO: CRD42020207263). MEDLINE, EMBASE, CINAHL, Pubmed, Scopus, Web of Science databases and grey literature were searched. Quantitative and qualitative data were extracted relating to swallow impairment, use of mealtime adaptations and eating and drinking-related quality of life. Quantitative data were summarised using narrative and meta-proportional analysis methods. Qualitative data were synthesised using a meta-aggregation approach. Where quantitative and qualitative data described the same phenomenon, a convergent segregated approach was used to synthesise data.
Sixty-five studies were included. Six oro-pharyngeal swallow characteristics were identified, and pooled prevalence calculated: aspiration (24%), laryngeal penetration (6%), oral stage dysfunction (11%), pharyngeal residue (13%), nasal regurgitation (7%), delayed swallow initiation (31%). Four patient-reported eating/drinking difficulties were identified, and pooled prevalence calculated: difficulty swallowing solids (45%), difficulty swallowing liquids (6%), odynophagia (30%), coughing when eating (38%). Three patient-reported mealtime adaptations were identified, and pooled prevalence calculated: need for water when eating (49%), eating slowly (37%), modifying textures (28%). Mixed methods synthesis of psychosocial impacts identified 34% of parents experienced mealtime anxiety and 25% report challenging mealtime behaviours reflected in five qualitative themes: fear and trauma associated with eating and drinking, isolation and a lack of support, being aware and grateful, support to cope and loss.
Eating and drinking difficulties are common in adults and children with repaired OA/TOF. Oro-pharyngeal swallowing difficulties may be more prevalent than previously reported. Eating, drinking and swallowing difficulties can impact on psychological well-being and quality of life, for the individual and parents/family members. Long-term, multi-disciplinary follow-up is warranted.
进食、饮水和吞咽困难是患有 OA/TOF 的个体常见的报告发病率。本研究旨在确定该人群报告的进食、饮水和口咽吞咽困难的性质和流行程度。
进行了系统评价和荟萃比例分析(PROSPERO:CRD42020207263)。检索了 MEDLINE、EMBASE、CINAHL、PubMed、Scopus、Web of Science 数据库和灰色文献。提取了与吞咽障碍、用餐适应和与饮食相关的生活质量相关的定量和定性数据。使用叙述性和荟萃比例分析方法总结定量数据。使用元聚合方法综合定性数据。对于描述相同现象的定量和定性数据,使用汇聚分离方法来综合数据。
纳入了 65 项研究。确定了 6 个口咽吞咽特征,并计算了 pooled prevalence:吸入(24%)、喉渗透(6%)、口腔期功能障碍(11%)、咽残留(13%)、鼻反流(7%)、吞咽启动延迟(31%)。确定了 4 个患者报告的进食/饮水困难,并计算了 pooled prevalence:固体吞咽困难(45%)、液体吞咽困难(6%)、吞咽疼痛(30%)、进食时咳嗽(38%)。确定了 3 个患者报告的用餐适应措施,并计算了 pooled prevalence:进食时需要水(49%)、进食缓慢(37%)、改变质地(28%)。心理社会影响的混合方法综合分析确定了 34%的父母经历了用餐焦虑,25%的父母报告了具有挑战性的用餐行为,反映在五个定性主题中:与进食和饮水相关的恐惧和创伤、孤立和缺乏支持、意识到和感激、应对支持和丧失。
修复后的 OA/TOF 成人和儿童中进食和饮水困难很常见。口咽吞咽困难可能比以前报告的更为普遍。进食、饮水和吞咽困难会影响个体和父母/家庭成员的心理健康和生活质量。需要长期的多学科随访。