Borgo G, Gasparini P, Bonizzato A, Cabrini G, Mastella G, Pignatti P F
Centro Regionale Veneto Fibrosi Cistica, Ospedale Civile Maggiore, Verona, Italy.
Eur J Pediatr. 1993 Dec;152(12):1006-11. doi: 10.1007/BF01957227.
In an attempt to ascertain a relationship between genotype and phenotype, we studied the pulmonary and nutritional status of 123 cystic fibrosis patients with known genotype at an age of 8.5-10 years. Patients represent a cohort as they are almost all those born and diagnosed in a given area and period. They were followed at a single centre using uniform diagnostic and treatment protocols. Pulmonary and nutritional status of homozygous delta 508 patients did not differ from that of compound heterozygotes or of patients with other unspecified genotypes. Pulmonary manifestations varied widely in all genotype groups. With the given number of patients, a slightly higher mortality of delta F508 homozygotes could have been coincidental. We conclude that up to the age of 8.5-10 years the severity of pulmonary lesions and nutritional deficiencies is not related to the delta F508 mutation.
为了确定基因型与表型之间的关系,我们研究了123名已知基因型的囊性纤维化患者在8.5至10岁时的肺部和营养状况。这些患者几乎都是在特定地区和时期出生并被诊断出患病的,因此构成了一个队列。他们在单一中心接受随访,采用统一的诊断和治疗方案。纯合子Δ508患者的肺部和营养状况与复合杂合子或其他未明确基因型的患者并无差异。所有基因型组的肺部表现差异很大。鉴于患者数量,ΔF508纯合子的死亡率略高可能只是巧合。我们得出结论,在8.5至10岁之前,肺部病变的严重程度和营养缺乏与ΔF508突变无关。