Augarten A, Kerem B S, Yahav Y, Noiman S, Rivlin Y, Tal A, Blau H, Ben-Tur L, Szeinberg A, Kerem E
Department of Paediatrics, Sheba Medical Centre, Ramat Gan, Israel.
Lancet. 1993 Jul 3;342(8862):25-6. doi: 10.1016/0140-6736(93)91885-p.
Different mutations in the cystic fibrosis (CF) gene appear to contribute to heterogeneity of the CF phenotype. We investigated 15 patients with CF who have the 3849 + 10 kb C-->T mutation. All were Ashkenazi Jews. Their clinical features were compared with those of CF patients with the delta F508/delta F508, W1282X/W1282X, W1282X/delta F508 mutations, which are known to be associated with a severe disease. Patients with the 3849 + 10 kb mutation were older, had been diagnosed as having CF at a more advanced age, and were in a better nutritional state. Sweat chloride values were normal (below 60 mmol/L) in 5 3849 + 10 kb patients (33%). 4 of these patients and 6 others (total 66%) had normal pancreatic function. However, age-adjusted pulmonary function did not differ between the two groups. None of the patients with 3849 + 10 kb C-->T had had meconium ileus or had liver disease or diabetes mellitus. We conclude that this mutation is associated with a mild type of CF.
囊性纤维化(CF)基因的不同突变似乎导致了CF表型的异质性。我们研究了15名携带3849 + 10 kb C→T突变的CF患者。他们均为德系犹太人。将他们的临床特征与已知与严重疾病相关的携带ΔF508/ΔF508、W1282X/W1282X、W1282X/ΔF508突变的CF患者的临床特征进行了比较。携带3849 + 10 kb突变的患者年龄较大,在更晚的年龄被诊断为患有CF,且营养状况更好。15名携带3849 + 10 kb突变的患者中有5名(33%)汗液氯化物值正常(低于60 mmol/L)。其中4名患者和另外6名患者(共66%)胰腺功能正常。然而,两组之间经年龄调整的肺功能并无差异。携带3849 + 10 kb C→T突变的患者均未患胎粪性肠梗阻、肝病或糖尿病。我们得出结论,这种突变与一种轻型CF相关。