Hamazaki M, Saito A
Acta Pathol Jpn. 1979 Jan;29(1):99-107. doi: 10.1111/j.1440-1827.1979.tb01295.x.
The case presented here is a three-month-old male infant with the Beckwith-Wiedemann's syndrome. Clinically, the patient was characterized by macroglossia, visceromegaly, umbilical hernia, microcephalus and other multiple malformations such as facial flame nevus or ear lobe grooves. The patient died of bronchopneumonia at the age of three months, and an autopsy was performed. Morphological examination revealed adrenal cytomegaly, hyperplasia and hypertrophy of the pancreatic islets, adrenal rest tissue in the right testis or hypertrophy of muscle fibers of the tongue associated with fibrous degenerative change, in addition to bronchopneumonia of the lung, causing his death. On electron microscopical examination, the cytomegalic cell of the adrenal was characteristic of large pleomorphic nucleus and granular substances with high density in the cytoplasm. In this case, thorough histologic search revealed no evidence of tumorous growth in the organs, though the exomphalos-macroglossia-gigantism syndrome has been of interest in its relationship to the occasional occurrence of Wilms tumor, adrenal carcinoma or other tumors.
此处所呈现的病例是一名患有贝克威思-维德曼综合征的三个月大男婴。临床上,该患者的特征为巨舌、内脏肿大、脐疝、小头畸形以及其他多种畸形,如面部火焰状痣或耳垂沟。该患者于三个月大时死于支气管肺炎,并进行了尸检。形态学检查显示肾上腺细胞肿大、胰岛增生肥大、右侧睾丸存在肾上腺残余组织或舌肌纤维肥大并伴有纤维性退行性变,此外还有肺部支气管肺炎,这导致了他的死亡。电子显微镜检查显示,肾上腺的巨细胞具有多形性大核以及细胞质中高密度的颗粒物质的特征。在该病例中,尽管脐膨出-巨舌-巨人症综合征因其与偶尔发生的肾母细胞瘤、肾上腺癌或其他肿瘤的关系而备受关注,但全面的组织学检查未发现器官中有肿瘤生长的证据。