Cho Eun-Kyung, Kim Jinsup, Yang Aram, Cho Sung Yoon, Jin Dong-Kyu
Department of Pediatrics, Samsung Medical Center, Sungkyunkwan University School of Medicine, Seoul, Korea.
Ann Pediatr Endocrinol Metab. 2017 Jun;22(2):129-132. doi: 10.6065/apem.2017.22.2.129. Epub 2017 Jun 28.
Chromosome 2q37 deletion syndrome is a rare chromosomal disorder characterized by mild to moderate developmental delay, brachydactyly of the third to fifth digits or toes, short stature, obesity, hypotonia, a characteristic facial appearance, and autism spectrum disorder. Here, we report on a patient with 2q37 deletion presenting with dilated cardiomyopathy (DCMP). Congenital heart malformations have been noted in up to 20% of patients with 2q37 deletions. However, DCMP has not been reported in 2q37 deletion patients previously. The patient exhibited the characteristic facial appearance (a flat nasal bridge, deep-set eyes, arched eyebrows, and a thin upper lip), developmental delay, mild mental retardation, peripheral nerve palsy, and Albright hereditary osteodystrophy (AHO)-like phenotypes (short stature and brachydactyly). Conventional chromosomal analysis results were normal; however, microarray-based comparative genomic hybridization revealed terminal deletion at 2q37.1q37.3. In addition, the patient was confirmed to have partial growth hormone (GH) deficiency and had shown a significant increase in growth rate after substitutive GH therapy. Chromosome 2q37 deletion syndrome should be considered in the differential diagnosis of patients presenting with AHO features, especially in the presence of facial dysmorphism. When patients are suspected of having a 2q37 deletion, high-resolution cytogenetic analysis is recommended.
2q37染色体缺失综合征是一种罕见的染色体疾病,其特征为轻至中度发育迟缓、第三至五指或趾短指畸形、身材矮小、肥胖、肌张力减退、特征性面容以及自闭症谱系障碍。在此,我们报告一例患有2q37缺失且表现为扩张型心肌病(DCMP)的患者。在高达20%的2q37缺失患者中已发现先天性心脏畸形。然而,此前尚未有关于2q37缺失患者出现DCMP的报道。该患者表现出特征性面容(鼻梁扁平、眼窝深陷、眉毛上拱以及上唇较薄)、发育迟缓、轻度智力障碍、周围神经麻痹以及类似奥尔布赖特遗传性骨营养不良(AHO)的表型(身材矮小和短指畸形)。常规染色体分析结果正常;然而,基于微阵列的比较基因组杂交显示2q37.1q37.3存在末端缺失。此外,该患者被证实存在部分生长激素(GH)缺乏,并且在接受替代GH治疗后生长速率显著增加。对于具有AHO特征的患者,尤其是存在面部畸形的患者,在鉴别诊断时应考虑2q37染色体缺失综合征。当怀疑患者存在2q37缺失时,建议进行高分辨率细胞遗传学分析。