Jezela-Stanek Aleksandra, Dobrzańska Anna, Maksym-Gąsiorek Dorota, Trzeciakowski Wojciech, Gutkowska Anna, Olczak-Kowalczyk Dorota, Gajdulewicz Maria, Spodar Krystyna, Czech-Kowalska Justyna, Krajewska-Walasek Małgorzata
Departments of Medical Genetics Neonatal Pathology and Intensive Care Oral Pathology, Children's Memorial Health Institute, Warsaw, Poland.
Clin Dysmorphol. 2009 Jan;18(1):13-17. doi: 10.1097/MCD.0b013e328317c884.
We describe the clinical characteristics of two patients with cat-eye syndrome (CES, MIM #115470) resulting from a supernumerary marker chromosome that includes 22pter-q12.3. They both presented a constellation of features typical of CES, including coloboma, auricular malformations, heart and renal anomalies, as well as hepatic dysfunction, which led to severe effects. In one case Pierre Robin sequence was diagnosed which has not been described earlier in this trisomy. Although CES is a well known, but infrequently diagnosed disorder, we draw attention both to its clinical overlaps with other disorders and, in view of the clinical variability being identified within the 22q11 region, to the importance of careful molecular examination of proximal 22q in patients with suggestive clinical signs.
我们描述了两名患有猫眼综合征(CES,MIM #115470)患者的临床特征,该综合征由一条包含22pter-q12.3的额外标记染色体导致。他们均表现出一系列典型的CES特征,包括虹膜缺损、耳部畸形、心脏和肾脏异常以及肝功能障碍,这些导致了严重后果。其中一例诊断出Pierre Robin序列,此前在该三体综合征中尚未有过描述。尽管CES是一种广为人知但诊断不常见的疾病,但我们既关注其与其他疾病的临床重叠情况,又鉴于在22q11区域发现的临床变异性,强调对具有提示性临床体征的患者进行22q近端仔细分子检查的重要性。