Sasaki Takaya, Okabe Masahiro, Tosaki Takeshi, Honda Yu, Ishikawa Masahiro, Tsuboi Nobuo, Yokoo Takashi
Division of Nephrology and Hypertension, Department of Internal Medicine, The Jikei University School of Medicine, 3-25-8, Nishi-Shimbashi, Minato-ku, Tokyo, Japan.
Department of Nephrology, Kawaguchi Municipal Medical Center, Kawaguchi, Japan.
CEN Case Rep. 2018 Nov;7(2):253-258. doi: 10.1007/s13730-018-0337-y. Epub 2018 May 16.
We report a case of distal partial trisomy 1 from q32.1 to 41 that have exhibited proteinuric glomerulopathy. The patient was a 17-year-old adolescent with clinical features of low birth weight, mild mental retardation and mild deafness, from the birth. He exhibited non-nephrotic range proteinuria with the mild obesity since the age of sixteen. Image studies did not reveal morphological abnormalities of the kidneys. Renal biopsy findings showed no definitive evidence of primary glomerular diseases, and were characterized by a very low glomerular density, glomerulomegaly and focal effacement of podocyte foot processes. Therapies with dietary sodium restriction, body weight reduction and the administration of angiotensin receptor blocker markedly reduced his proteinuria. It was likely that mismatch between congenital reduction in the nephron number and catch-up growth of the whole body size played a major role in the development of glomerular hyperperfusion injury. At present, the direct contribution of genetic factors due to this chromosomal disorder to such a substantial reduction in the nephron number remains uncertain.
我们报告了一例1号染色体从q32.1到41的远端部分三体综合征病例,该病例表现为蛋白尿性肾小球病。患者为一名17岁青少年,自出生起就有低体重、轻度智力障碍和轻度耳聋的临床特征。自16岁起,他出现非肾病范围蛋白尿并伴有轻度肥胖。影像学检查未发现肾脏形态异常。肾活检结果未显示原发性肾小球疾病的确切证据,其特征为肾小球密度极低、肾小球肿大和足细胞足突局灶性消失。饮食限钠、减轻体重和给予血管紧张素受体阻滞剂治疗显著降低了他的蛋白尿。先天性肾单位数量减少与全身尺寸追赶生长之间的不匹配可能在肾小球高灌注损伤的发展中起主要作用。目前,这种染色体疾病导致的遗传因素对肾单位数量大幅减少的直接影响仍不确定。