Demarquez J L, Vital C, Babin J P, Allain D, Bondonny J M, Sautarel M, Rancon F, Martin C
Arch Fr Pediatr. 1977 Feb;34(2):179-91.
A case of hyperinsulinism occuring in a newborn, with a birthweight of 4,050 g, is reported. The hypoglycaemia was refractory to the usual therapy (increase of glucose administration per os, and I.V., corticosteroids, glucagon, diazoxide). At surgery, undertaken at 9 days of age, an adenomatous nodule was removed along with the left part of the pancreas. Death occurred at 18 days, after the child had developed a transitory acidoketosic diabetes and an encephalopathy. Measurement of insulin by radio-immunoassay revealed a strong increase in the ratio insulin/glycaemia, characteristic of nesidioblastoma, as well as a high concentration of insulin in the tumor as compared to normal tissue. On the ultrastructural level, the observed features differed from those seen in children and adults and showed an abnormal overload of dense deposits in the cytoplasm of some histiocytes.
报告了一例出生体重4050克的新生儿发生高胰岛素血症的病例。低血糖对常规治疗(口服和静脉输注葡萄糖增加、皮质类固醇、胰高血糖素、二氮嗪)无效。在9日龄时进行手术,切除了腺瘤样结节以及胰腺左部。患儿在出现短暂性酸酮症糖尿病和脑病后于18日龄死亡。通过放射免疫测定法测量胰岛素发现,胰岛素/血糖比值显著升高,这是胰岛细胞瘤的特征,并且与正常组织相比,肿瘤中的胰岛素浓度很高。在超微结构水平上,观察到的特征与儿童和成人所见不同,一些组织细胞的细胞质中出现了致密沉积物异常过载的情况。