Harris Andrew M, Hall Bryan, Kriss Vesna M, Fowlkes John L, Kiessling Stefan G
University of Kentucky, Department of Pediatrics, Lexington, KY 40536, USA.
Pediatr Nephrol. 2007 Dec;22(12):2141-4. doi: 10.1007/s00467-007-0600-5. Epub 2007 Sep 12.
Rabson-Mendenhall syndrome is a rare genetic disorder characterized by severe insulin resistance, extreme hyperinsulinemia, postprandial hyperglycemia, growth retardation, and dysmorphisms. Enlargement of the kidneys and nephrocalcinosis have been described previously. We report a 10-year-old boy who presented with gross hematuria, unilateral hydronephrosis, and the initial diagnosis of bilateral extensive medullary nephrocalcinosis. Medullary sponge kidney (MSK) was included in the differential diagnosis given the ultrasound findings. Further evaluation by intravenous pyelogram confirmed the suspected bilateral MSK. Given the patient's history of hydronephrosis due to an obstructing renal stone and MSK, urine calcium excretion was assessed and found to be markedly increased at 9.5 mg/kg per day. To our knowledge, this is the first report of Rabson-Mendenhall syndrome and an association with MSK. We recommend evaluation for nephrocalcinosis, MSK, and hypercalciuria in all children diagnosed with Rabson-Mendenhall syndrome.
拉布森-门登霍尔综合征是一种罕见的遗传性疾病,其特征为严重胰岛素抵抗、极度高胰岛素血症、餐后高血糖、生长发育迟缓及畸形。此前已有关于肾脏增大和肾钙质沉着症的描述。我们报告一名10岁男孩,他出现肉眼血尿、单侧肾积水,最初诊断为双侧广泛性髓质肾钙质沉着症。鉴于超声检查结果,鉴别诊断中考虑了髓质海绵肾(MSK)。静脉肾盂造影进一步评估证实了疑似双侧MSK。鉴于该患者因梗阻性肾结石和MSK导致肾积水的病史,对尿钙排泄进行了评估,发现其明显增加,为每天9.5毫克/千克。据我们所知,这是拉布森-门登霍尔综合征与MSK关联的首例报告。我们建议对所有诊断为拉布森-门登霍尔综合征的儿童进行肾钙质沉着症、MSK和高钙尿症的评估。