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眼牙指综合征:一例报告

Oculodentodigital syndrome: report of a case.

作者信息

Scheutzel P

机构信息

Department of Restorative Dentistry, Dental Hospital, Westfälische, Wilhelms-Universität, Münster, Germany.

出版信息

Dentomaxillofac Radiol. 1991 Aug;20(3):175-8. doi: 10.1259/dmfr.20.3.1808004.

DOI:10.1259/dmfr.20.3.1808004
PMID:1808004
Abstract

The clinical and radiographic changes in a case of oculodentodigital syndrome are presented. The characteristic features of this rare developmental disorder, microphthalmus with microcornea and iris anomalies, hypertelorism, thin nose with hypoplastic alae and anteverted nostrils, syndactyly with camptodactyly and clinodactyly of the fourth and fifth fingers associated with bony anomalies of the middle phalanges of the fifth fingers and toes, were all present. In addition, histological examination of a lateral incisor showed the enamel dysplasia to be due to enamel hypoplasia; the dentine also showed marked hypocalcification. There were widespread pulp denticles and hypercementosis throughout the dentition. Both the patient and his mother had coronoid hypoplasia.

摘要

本文介绍了一例眼牙指综合征的临床和影像学变化。这种罕见的发育障碍的特征包括小眼合并小角膜和虹膜异常、眼距过宽、鼻瘦伴鼻翼发育不全和鼻孔前倾、并指(趾)合并第四和第五指屈曲指和斜指,以及第五指和趾中节指(趾)骨的骨异常。此外,一颗侧切牙的组织学检查显示釉质发育不全是由于釉质发育不良所致;牙本质也显示出明显的钙化不足。整个牙列中普遍存在牙髓牙本质小体和牙骨质增生。患者及其母亲均有冠状突发育不全。

相似文献

1
Oculodentodigital syndrome: report of a case.眼牙指综合征:一例报告
Dentomaxillofac Radiol. 1991 Aug;20(3):175-8. doi: 10.1259/dmfr.20.3.1808004.
2
Oculodentodigital dysplasia. A case report.
Minerva Stomatol. 2005 Jul-Aug;54(7-8):453-9.
3
Oculodentodigital dysplasia.眼牙指发育不全
Oral Surg Oral Med Oral Pathol. 1986 Apr;61(4):418-21. doi: 10.1016/0030-4220(86)90431-7.
4
Dental management of oculodentodigital dysplasia: report of case.眼牙指发育不全的牙科处理:病例报告
ASDC J Dent Child. 1986 Mar-Apr;53(2):131-4.
5
[A rare dysplasia: the oculo-dento-digital syndrome].
Z Arztl Fortbild (Jena). 1965 May 1;59(9):504-6.
6
Oculodentodigital dysplasia: study of ophthalmological and clinical manifestations in three boys with probably autosomal recessive inheritance.眼牙指发育不全:对三名可能具有常染色体隐性遗传的男孩的眼科和临床表现的研究。
Ophthalmic Genet. 2004 Sep;25(3):227-36. doi: 10.1080/13816810490513424.
7
Oculodentodigital dysplasia syndrome. Report of four cases.眼牙指发育不全综合征。4例报告。
Acta Paediatr Scand. 1977 Sep;66(5):635-8. doi: 10.1111/j.1651-2227.1977.tb07960.x.
8
[Oculodentodigital dysplasia: report of 2 familial cases].
Pathologica. 1994 Feb;86(1):102-5.
9
Three new cases of oculodentodigital (ODD) syndrome: development of the facial phenotype.三例眼牙指(ODD)综合征新病例:面部表型的发展
J Med Genet. 1985 Oct;22(5):386-9. doi: 10.1136/jmg.22.5.386.
10
Oculodentodigital dysplasia.
Mo Med. 1977 Aug;74(8):379-80, 383.

引用本文的文献

1
Oculo-dento-digital dysplasia: a systematic analysis of published dental literature.眼-牙-指发育不全:已发表牙科文献的系统分析
BDJ Open. 2023 Mar 29;9(1):13. doi: 10.1038/s41405-023-00139-7.
2
Generalised hypomineralisation of enamel in oculodentodigital dysplasia: comprehensive dental management of a case.眼-牙-指(趾)发育不全中釉质的普遍矿化不全:1 例病例的全面口腔管理。
BMJ Case Rep. 2021 Jan 11;14(1):e238079. doi: 10.1136/bcr-2020-238079.
3
Oculodentodigital Dysplasia: A Case Report and Major Review of the Eye and Ocular Adnexa Features of 295 Reported Cases.
眼牙指发育不全:一例报告及295例报告病例的眼部和眼附属器特征的主要综述
Case Rep Ophthalmol Med. 2020 Apr 4;2020:6535974. doi: 10.1155/2020/6535974. eCollection 2020.