Stewart D H, Streeten B W, Brockhurst R J, Anderson D R, Hirose T, Gass D M
Department of Ophthalmology, State University of New York Health Science Center, Syracuse 13210.
Arch Ophthalmol. 1991 Jul;109(7):1017-25. doi: 10.1001/archopht.1991.01080070129050.
Ten patients with bilateral nanophthalmos underwent sclerectomies for uveal effusion. Ultrastructural examination of the sclera revealed abnormal collagen in seven patients. Four showed dramatic fraying of the collagen fibrils into fine filaments 2 to 3 nm in diameter. In three of these cases and three other cases without fraying, there were foci of 10- to 35-nm small collagen fibrils, some appearing to arise by splitting of otherwise normal collagen fibrils. In areas of fraying, elastic fibers were absent. All patients had a wider range of collagen diameters than did control subjects. The youngest patient with fraying also had Hallermann-Streiff syndrome. In three patients, no collagen abnormality was found. The clinical feature correlating best with the presence of abnormal collagen was an extremely small eye, since the three patients without collagen abnormality had the largest eyes (range of anteroposterior diameters, 19.2 to 20.3 mm). Nanophthalmos appears to result from several distinct defects.
10例双侧小眼球患者因葡萄膜渗漏接受了巩膜切除术。对巩膜进行超微结构检查发现,7例患者存在胶原异常。4例患者表现出胶原纤维显著磨损成直径为2至3纳米的细丝。在其中3例以及另外3例未出现磨损的病例中,存在10至35纳米的小胶原纤维灶,一些似乎是由原本正常的胶原纤维分裂形成的。在磨损区域,弹性纤维缺失。所有患者的胶原直径范围均比对照组受试者更宽。最年轻的出现磨损的患者还患有哈勒曼-施特雷夫综合征。3例患者未发现胶原异常。与胶原异常存在最密切相关的临床特征是眼球极小,因为3例无胶原异常的患者眼球最大(前后径范围为19.2至20.3毫米)。小眼球似乎是由几种不同的缺陷导致的。