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先天性心脏病作为诊断22q11.2缺失的警示信号。

Congenital Heart Disease as a Warning Sign for the Diagnosis of the 22q11.2 Deletion.

作者信息

Grassi Marcília S, Jacob Cristina M A, Kulikowski Leslie D, Pastorino Antonio C, Dutra Roberta L, Miura Nana, Jatene Marcelo B, Pegler Stephanie P, Kim Chong A, Carneiro-Sampaio Magda

机构信息

Instituto da Criança, HC, FMUSP, São Paulo, SP, Brazil.

Departamento de Patologia, Faculdade de Medicina, Universidade de São Paulo, São Paulo, SP, Brazil.

出版信息

Arq Bras Cardiol. 2014 Nov;103(5):382-390. doi: 10.5935/abc.20140145. Epub 2014 Oct 10.

Abstract

Background: To alert for the diagnosis of the 22q11.2 deletion syndrome (22q11.2DS) in patients with congenital heart disease (CHD). Objective: To describe the main CHDs, as well as phenotypic, metabolic and immunological findings in a series of 60 patients diagnosed with 22q11.2DS. Methods: The study included 60 patients with 22q11.2DS evaluated between 2007 and 2013 (M:F=1.3, age range 14 days to 20 years and 3 months) at a pediatric reference center for primary immunodeficiencies. The diagnosis was established by detection of the 22q11.2 microdeletion using FISH (n = 18) and/or MLPA (n = 42), in association with clinical and laboratory information. Associated CHDs, progression of phenotypic facial features, hypocalcemia and immunological changes were analyzed. Results: CHDs were detected in 77% of the patients and the most frequent type was tetralogy of Fallot (38.3%). Surgical correction of CHD was performed in 34 patients. Craniofacial dysmorphisms were detected in 41 patients: elongated face (60%) and/or elongated nose (53.3%), narrow palpebral fissure (50%), dysplastic, overfolded ears (48.3%), thin lips (41.6%), elongated fingers (38.3%) and short stature (36.6%). Hypocalcemia was detected in 64.2% and decreased parathyroid hormone (PTH) level in 25.9%. Decrease in total lymphocytes, CD4 and CD8 counts were present in 40%, 53.3% and 33.3%, respectively. Hypogammaglobulinemia was detected in one patient and decreased concentrations of immunoglobulin M (IgM) in two other patients. Conclusion: Suspicion for 22q11.2DS should be raised in all patients with CHD associated with hypocalcemia and/or facial dysmorphisms, considering that many of these changes may evolve with age. The 22q11.2 microdeletion should be confirmed by molecular testing in all patients.

摘要

背景

提高对先天性心脏病(CHD)患者22q11.2缺失综合征(22q11.2DS)的诊断警惕性。目的:描述一系列60例诊断为22q11.2DS患者的主要先天性心脏病,以及其表型、代谢和免疫学表现。方法:该研究纳入了60例2007年至2013年间在一家原发性免疫缺陷病儿科参考中心接受评估的22q11.2DS患者(男∶女 = 1.3,年龄范围为14天至20岁零3个月)。通过荧光原位杂交(FISH,n = 18)和/或多重连接探针扩增技术(MLPA,n = 42)检测22q11.2微缺失,并结合临床和实验室信息来确诊。分析相关的先天性心脏病、面部特征表型进展、低钙血症和免疫学变化。结果:77%的患者检测到先天性心脏病,最常见的类型是法洛四联症(38.3%)。34例患者接受了先天性心脏病的手术矫正。41例患者检测到颅面部畸形:长脸(60%)和/或长鼻(53.3%)、睑裂狭窄(50%)、发育异常且折叠过度的耳朵(48.3%)、薄唇(41.6%)、手指细长(38.3%)和身材矮小(36.6%)。64.2%的患者检测到低钙血症,25.9%的患者甲状旁腺激素(PTH)水平降低。总淋巴细胞、CD4和CD8计数分别在40%、53.3%和33.3%的患者中出现下降。1例患者检测到低丙种球蛋白血症,另外2例患者免疫球蛋白M(IgM)浓度降低。结论:对于所有伴有低钙血症和/或面部畸形的先天性心脏病患者,应怀疑22q11.2DS,因为这些变化中的许多可能会随年龄发展。所有患者均应通过分子检测来确诊22q11.2微缺失。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/96d3/4262098/3e37d99d6bb7/abc-103-05-0382-g01.jpg

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