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卡塔尔囊性纤维化患儿的鼻窦疾病与囊性纤维化跨膜传导调节因子(CFTR)I1234V突变

Sinonasal Disease in Pediatric Patients With Cystic Fibrosis and the Cystic Fibrosis Transmembrane Conductance Regulator (CFTR) I1234V Mutation in Qatar.

作者信息

Odeh Mohammad, Al-Naimi Amal R, Younes Abbas

机构信息

Pediatric Pulmonology Medicine, Sidra Medicine, Doha, QAT.

Pediatric Pulmonary Medicine, Sidra Medicine, Doha, QAT.

出版信息

Cureus. 2025 Jul 21;17(7):e88481. doi: 10.7759/cureus.88481. eCollection 2025 Jul.

Abstract

Background and objective Sinonasal disease is a common but underrecognized manifestation in patients with cystic fibrosis (CF). The CF transmembrane conductance regulator (CFTR) I1234V mutation is the most prevalent CF mutation in Qatar, yet its specific impact on upper airway pathology remains poorly characterized. This study aimed to evaluate the prevalence and clinical features of sinonasal disease in pediatric patients with CF who are homozygous for the CFTR I1234V mutation in Qatar. Methodology A retrospective cross-sectional study was conducted at Sidra Medicine, including pediatric patients diagnosed with homozygous CFTR I1234V mutation between September 1, 2017, and June 30, 2024. Clinical data, sinonasal symptoms, ENT examination findings, computed tomography (CT) imaging results, and surgical interventions were collected and analyzed. Results A total of 29 patients were included (16, 55.1%, female, mean age 10.4 years). More than half (16, 55.1%) were asymptomatic for sinonasal complaints. However, 13 (44.8%) reported nasal congestion, and 8 (27.5%) had nasal polyps on endoscopy. CT scans performed in nine patients revealed sinusitis in all cases (100%) and polyposis in seven (77.7%). Surgical intervention was required in 8 (27.5%) patients, primarily functional endoscopic sinus surgery. Co-colonization with methicillin-resistant Staphylococcus aureus (MRSA) and Pseudomonas was noted in three (10.3%) patients. Conclusions Pediatric patients with CF who are homozygous for the I1234V mutation in Qatar exhibit variable expression of sinonasal disease, with many demonstrating radiologic or endoscopic abnormalities despite being asymptomatic. These findings highlight the importance of routine ENT assessment in this population, even in the absence of complaints, to guide early intervention and potentially reduce respiratory complications.

摘要

背景与目的

鼻窦疾病是囊性纤维化(CF)患者中常见但未得到充分认识的表现。CF跨膜传导调节因子(CFTR)I1234V突变是卡塔尔最常见的CF突变,但其对上呼吸道病理的具体影响仍未得到充分描述。本研究旨在评估卡塔尔CFTR I1234V突变纯合子的儿科CF患者鼻窦疾病的患病率和临床特征。方法:在西德拉医学中心进行了一项回顾性横断面研究,纳入2017年9月1日至2024年6月30日期间诊断为CFTR I1234V突变纯合子的儿科患者。收集并分析临床数据、鼻窦症状、耳鼻喉科检查结果、计算机断层扫描(CT)成像结果和手术干预情况。结果:共纳入29例患者(16例,55.1%为女性,平均年龄10.4岁)。超过一半(16例,55.1%)的患者鼻窦无主诉症状。然而,13例(44.8%)报告有鼻塞,8例(27.5%)在内镜检查时有鼻息肉。9例患者进行的CT扫描显示,所有病例(100%)均有鼻窦炎,7例(77.7%)有息肉病。8例(27.5%)患者需要手术干预,主要是功能性内镜鼻窦手术。3例(10.3%)患者同时感染耐甲氧西林金黄色葡萄球菌(MRSA)和铜绿假单胞菌。结论:卡塔尔CFTR I1234V突变纯合子的儿科CF患者鼻窦疾病表现各异,许多患者尽管无症状,但仍有放射学或内镜异常。这些发现凸显了对该人群进行常规耳鼻喉科评估的重要性,即使在无主诉症状的情况下,以指导早期干预并可能减少呼吸道并发症。

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