Yang S S, Chen H, Williams P, Cacciarelli A, Misra R P, Bernstein J
Arch Pathol Lab Med. 1980 Apr;104(4):208-11.
Spondyloepiphyseal dysplasia congenita is a short-trunk chondrodysplasia trait. The abnormalities are present at birth and involve primarily the spine, the epiphyses of long bones, and the pelvis. Postmortem examinations of two patients with this disease who died shortly after birth showed a mild disorganization of chondrocytic columnization in the physeal growth zone. The chondrocytes contained PAS-positive cytoplasmic inclusions after diastase digestion to eliminate glycogen. Ultrastructural examination of the inclusions in one patient showed them to be accumulations of finely granular material in dilated cisterns of rough endoplasmic reticulum. To our knowledge, similar findings have been seen only in three other types of chondrodysplasia. The inclusions, which therefore are of diagnostic importance, may be of importance also in reflecting the primary metabolic abnormality.
先天性脊柱骨骺发育不良是一种短躯干软骨发育异常特征。这些异常在出生时就存在,主要累及脊柱、长骨骨骺和骨盆。对两名出生后不久死亡的该疾病患者进行的尸检显示,生长板生长区的软骨细胞柱状排列轻度紊乱。在经过淀粉酶消化以去除糖原后,软骨细胞含有PAS阳性的细胞质内含物。对一名患者内含物的超微结构检查显示,它们是粗面内质网扩张池内细颗粒物质的积聚。据我们所知,类似的发现仅在其他三种软骨发育异常类型中见过。因此,这些内含物具有诊断重要性,在反映原发性代谢异常方面可能也很重要。