Dipartimento di Scienze Mediche Traslazionali, Sezione di Pediatria, Università di Napoli Federico II, CRR Fibrosi Cistica del Bambino, Naples, Italy.
Dipartimento di Sanità Pubblica, Università di Napoli Federico II, Naples, Italy.
Sci Rep. 2020 Apr 2;10(1):5806. doi: 10.1038/s41598-020-62747-9.
The clinical manifestation of cystic fibrosis (CF) is heterogeneous also in patients with the same cystic fibrosis transmembrane regulator (CFTR) genotype and in affected sibling pairs. Other genes, inherited independently of CFTR, may modulate the clinical manifestation and complications of patients with CF, including the severity of chronic sinonasal disease and the occurrence of chronic Pseudomonas aeruginosa colonization. The T2R38 gene encodes a taste receptor and recently its functionality was related to the occurrence of sinonasal diseases and upper respiratory infections. We assessed the T2R38 genotype in 210 patients with CF and in 95 controls, relating the genotype to the severity of sinonasal disease and to the occurrence of P. aeruginosa pulmonary colonization. The frequency of the PAV allele i.e., the allele associated with the high functionality of the T2R38 protein, was significantly lower in i) CF patients with nasal polyposis requiring surgery, especially in patients who developed the complication before 14 years of age; and ii) in CF patients with chronic pulmonary colonization by P. aeruginosa, especially in patients who were colonized before 14 years of age, than in control subjects. These data suggest a role for T2R38 as a novel modifier gene of sinonasal disease severity and of pulmonary P. aeruginosa colonization in patients with CF.
囊性纤维化(CF)的临床表现在具有相同囊性纤维化跨膜转导调节因子(CFTR)基因型的患者中和受影响的同胞对中也是异质的。其他独立于 CFTR 遗传的基因可能调节 CF 患者的临床表现和并发症,包括慢性鼻-鼻窦炎疾病的严重程度和慢性铜绿假单胞菌定植的发生。T2R38 基因编码味觉受体,最近其功能与鼻-鼻窦疾病和上呼吸道感染的发生有关。我们评估了 210 例 CF 患者和 95 例对照者的 T2R38 基因型,将基因型与鼻-鼻窦疾病的严重程度和铜绿假单胞菌肺部定植的发生相关联。PAV 等位基因的频率(即与 T2R38 蛋白高功能相关的等位基因)在以下情况下显著降低:i)需要手术治疗的 CF 患者伴鼻息肉,尤其是在 14 岁之前发生该并发症的患者;ii)在慢性铜绿假单胞菌肺部定植的 CF 患者中,尤其是在 14 岁之前定植的患者中,显著低于对照组。这些数据表明 T2R38 作为 CF 患者鼻-鼻窦疾病严重程度和肺部铜绿假单胞菌定植的新型修饰基因发挥作用。