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一名患有漂浮港综合征的14岁患者出现少牙畸形和舌系带过短的新发现。

A novel finding of oligodontia and ankyloglossia in a 14-year-old with Floating-Harbor syndrome.

作者信息

Singh Akshara, Bhatia Hind Pal, Sood Shveta, Sharma Naresh, Mohan Amit

机构信息

Assistant Professor, Department of Pedodontics and Preventive Dentistry, Manav Rachna Dental College, Faridabad, Haryana, India.

Professor & Head, Department of Pedodontics and Preventive Dentistry, Manav Rachna Dental College, Faridabad, Haryana, India.

出版信息

Spec Care Dentist. 2017 Nov;37(6):318-321. doi: 10.1111/scd.12257. Epub 2017 Dec 6.

DOI:10.1111/scd.12257
PMID:29210485
Abstract

Floating-Harbor syndrome (FHS) is a rare genetic condition characterized by distinct facial features, short stature and delayed skeletal development. Here we present case of a 14-year-old boy with short stature, typical facial features, impaired voice quality, clinodactyly, cryptorchidism and unilateral agenesis of kidney. In addition he had oligodontia and ankyloglossia with features suggestive of FHS. Treatment rendered was restoration of caries, application of pit and fissure sealants followed by frenectomy. The purpose of this report is to highlight the oral developmental anomalies and the management of a patient with FHS and to add to the current knowledge of the literature on this syndrome.

摘要

弗洛廷-哈伯综合征(FHS)是一种罕见的遗传性疾病,其特征为独特的面部特征、身材矮小和骨骼发育延迟。在此,我们报告一例14岁男孩,他身材矮小、具有典型的面部特征、音质受损、手指弯曲、隐睾症和单侧肾发育不全。此外,他还有少牙症和舌系带过短,这些特征提示为FHS。所采取的治疗措施为龋齿修复、窝沟封闭,随后进行舌系带切除术。本报告的目的是强调FHS患者的口腔发育异常及管理,并补充该综合征目前的文献知识。

相似文献

1
A novel finding of oligodontia and ankyloglossia in a 14-year-old with Floating-Harbor syndrome.一名患有漂浮港综合征的14岁患者出现少牙畸形和舌系带过短的新发现。
Spec Care Dentist. 2017 Nov;37(6):318-321. doi: 10.1111/scd.12257. Epub 2017 Dec 6.
2
Chiari I malformation as part of the Floating-Harbor syndrome?Chiari I型畸形是漂浮港综合征的一部分?
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Growth hormone deficiency: an unusual presentation of floating harbor syndrome.生长激素缺乏症:漂浮港综合征的一种不常见表现。
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引用本文的文献

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Floating-Harbor Syndrome: A Systematic Literature Review and Case Report.浮港综合征:一项系统文献综述与病例报告
J Clin Med. 2024 Jun 12;13(12):3435. doi: 10.3390/jcm13123435.
2
Oligodontia in the Clinical Spectrum of Syndromes: A Systematic Review.综合征临床谱中的少牙症:一项系统评价
Dent J (Basel). 2023 Dec 4;11(12):279. doi: 10.3390/dj11120279.
3
[Floating-Harbor syndrome: a case report and literature review].[浮动港综合征:一例报告及文献综述]
Zhongguo Dang Dai Er Ke Za Zhi. 2019 Dec;21(12):1208-1211. doi: 10.7499/j.issn.1008-8830.2019.12.011.
4
Novel genotypes and phenotypes among Chinese patients with Floating-Harbor syndrome.中国 Floating-Harbor 综合征患者的新型基因型和表型。
Orphanet J Rare Dis. 2019 Jun 14;14(1):144. doi: 10.1186/s13023-019-1111-8.