Schauer G M, Dunn L K, Godmilow L, Eagle R C, Knisely A S
Department of Pathology, Pennsylvania Hospital, Philadelphia.
Am J Med Genet. 1990 Dec;37(4):583-91. doi: 10.1002/ajmg.1320370433.
Sonography permitted the diagnosis of Fraser syndrome (cryptophthalmos-syndactyly syndrome) at 18.5 weeks of gestation in a fetus whose parents had had a previous affected child. The karyotype of that child was 46,XX,inv(9)(p11q21); the karyotype of the phenotypically normal father and of the fetus was 46,XY,inv(9)(p11q21). Findings on sonography included oligohydramnios with nonvisualization of kidneys, hypertelorism and microphthalmia, and markedly enlarged lungs. On autopsy at 19 weeks, findings included renal agenesis, cryptophthalmos with multiple abnormalities of the eyes and ocular adnexa, laryngeal atresia, pulmonary hyperplasia with accelerated maturation, absence of the Eustachian tube with connective tissue occupying the tympanic cavity and bone occluding the external acoustic meatus, and soft-tissue webbing between the digits. This is the second reported instance of prenatal diagnosis of Fraser syndrome in the second trimester. The histopathologic findings in Fraser syndrome at this gestational age, in particular the eye and ear, have not been described previously.
超声检查在一名父母曾有过患病孩子的胎儿妊娠18.5周时确诊了弗雷泽综合征(隐眼-并指综合征)。该患病孩子的核型为46,XX,inv(9)(p11q21);表型正常的父亲和胎儿的核型为46,XY,inv(9)(p11q21)。超声检查结果包括羊水过少伴肾脏不可见、眼距过宽和小眼畸形,以及肺部明显增大。妊娠19周时尸检发现包括肾缺如、隐眼伴眼和眼附属器的多种异常、喉闭锁、肺增生伴成熟加速、咽鼓管缺如伴结缔组织占据鼓室且骨阻塞外耳道,以及手指间软组织蹼。这是第二例关于孕中期产前诊断弗雷泽综合征的报道病例。此前尚未描述过该孕周弗雷泽综合征的组织病理学发现,尤其是眼和耳的发现。