Sabry M A, Farag T I, Shaltout A A, Zaki M, Al-Mazidi Z, Abulhassan S J, Al-Torki N, Quishawi A, Al Awadi S A
Kuwait Medical Genetics Centre.
Clin Genet. 1999 Jan;55(1):44-9. doi: 10.1034/j.1399-0004.1999.550108.x.
We describe 2 unrelated Bedouin girls who met the criteria for the diagnosis of Kenny-Caffey syndrome. The girls had some unusual features--microcephaly and psychomotor retardation--that distinguish the Kenny-Caffey syndrome profile in Arab children from the classical Kenny-Caffey syndrome phenotype characterized by macrocephaly and normal intelligence. The 2 girls did not harbor the 22q11 microdeletion (the hallmark of the DiGeorge cluster of diseases) that we previously reported in another Bedouin family with the Kenny-Caffey syndrome (Sabry et al. J Med Genet 1998: 35(1): 31-36). This indicates considerable genetic heterogeneity for this syndrome. We also review previously reported 44 Arab/Bedouin patients with the same profile of hypoparathyroidism, short stature, seizures, mental retardation and microcephaly. Our results suggest that these patients represent an Arab variant of Kenny-Caffey syndrome with characteristic microcephaly and psychomotor retardation. We suggest that all patients with Kenny-Caffey syndrome should be investigated for the 22q11 microdeletion. Other possible genetic causes for the Kenny-Caffey syndrome or its Arab variant include chromosome 10p abnormalities.
我们描述了两名符合肯尼-卡菲综合征诊断标准的非亲属贝都因女孩。这两名女孩有一些不寻常的特征——小头畸形和精神运动发育迟缓——这些特征将阿拉伯儿童中的肯尼-卡菲综合征表现型与以大头畸形和智力正常为特征的经典肯尼-卡菲综合征表型区分开来。这两名女孩没有携带我们先前在另一个患有肯尼-卡菲综合征的贝都因家族中报道的22q11微缺失(迪乔治综合征群疾病的标志)(Sabry等人,《医学遗传学杂志》1998年:35(1): 31 - 36)。这表明该综合征存在相当大的遗传异质性。我们还回顾了先前报道的44名具有甲状旁腺功能减退、身材矮小、癫痫、智力发育迟缓及小头畸形相同表现型的阿拉伯/贝都因患者。我们的结果表明,这些患者代表了具有特征性小头畸形和精神运动发育迟缓的肯尼-卡菲综合征的阿拉伯变异型。我们建议对所有肯尼-卡菲综合征患者进行22q11微缺失检测。肯尼-卡菲综合征或其阿拉伯变异型的其他可能遗传原因包括10号染色体短臂异常。