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Case 1 is a 5-year-old girl, who suffered from frequent episodes of unexplained fever and failure to thrive, Case 2 is an 8-year-old girl. In both cases, polyuria and polydipsia have been noticed since infancy. In both cases, moderate proteinuria was first detected, quite by chance, a few days prior to admission, The intravenous pyelography showed no nephrogram bilaterally in Case 1. In Case 2, the intravenous pyelography showed a delayed excretion of contrast medium bilaterally. By CT scan, the small kidney was bilaterally disclosed in both cases. An open renal biopsy was performed in both cases. The nephrons were found to be greatly reduced in number in the bioptical specimens. The diameter of the glomerulus of oligomeganephronia was approximately twice that of the control. In addition to glomerular hypertrophy with proliferation of mesangial cells, tubular hypertrophy was also detected. Interstitial fibrosis with lymphocytic infiltration was found in both cases, and was quite advanced in Case 2. Also, hyperplasia of the juxtaglomerular apparatus and diffuse or segmental sclerosis of the glomeruls were found in Case 2a In both cases, thickening of the basement membrane, folding of the basement membrane in the mesangial area, as well as fusion of the foot processes were observed. In Case 2, electron dense deposits were seen in the paramesangial area. The water restriction, the administration of hypertonic saline, and the loading of a synthetic vasopressin (DDAVP) resulted in no changes of either the osmotic pressure or the volume of urine. From these results, the distal tubule was considered to be hypofunctional and unresponsive .
- 社団法人 日本腎臓学会の論文
社団法人 日本腎臓学会 | 論文
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