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克莱夫斯特拉综合征的口腔表现与需求:一例病例报告及文献综述

Kleefstra Syndrome-Dental Manifestations and Needs: A Case Report with a Literature Review.

作者信息

Karlak Victoria, Jankowski Jakub, Kolasińska Julia, Nijakowski Kacper

机构信息

University Centre of Dentistry and Specialised Medicine, 60-812 Poznan, Poland.

Student's Scientific Group in Department of Conservative Dentistry and Endodontics, Poznan University of Medical Sciences, 60-812 Poznan, Poland.

出版信息

Case Rep Dent. 2023 Sep 22;2023:2478465. doi: 10.1155/2023/2478465. eCollection 2023.

DOI:10.1155/2023/2478465
PMID:37780946
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC10541297/
Abstract

Kleefstra syndrome (KS) is a rare genetic disorder (prevalence < 1/1 000 000) characterised by autistic spectrum disorder (ASD), childhood hypotonia, and seizures. A typical facial appearance includes microcephaly, arched eyebrows, synophrism, hypertelorism, a short nose, midface hypoplasia, prognathism, and tongue protrusion. This case report presents a 19-year-old female with KS from a dentist's perspective. The patient demonstrates limited mouth opening with a slight deviation of the mandible on the left side. The mandibular prognathism and anterior open bite are visible. A generalised inflammatory gingival enlargement is most likely a response to local irritants like plaque and calculus but is enhanced by the patient's mouth breathing habit. Also, the tongue is unusually large-macroglossia. Dental anomalies were diagnosed by digital panoramic radiograph, including dilaceration of the maxillary left second premolar and taurodontism. The patient was qualified to be treated under general anaesthesia with multiple extractions, restorations, hygienisation procedures, and tooth remineralisation. This individual should also be considered for orthodontic treatment and an eventual tongue reduction procedure. In the case of KS, multidisciplinary cooperation as well as frequent dental check-ups with fluoride prophylaxis are recommended. Unfortunately, dental treatment is still not an integral part of taking care of disabled children and youth with rare diseases.

摘要

克莱夫斯特拉综合征(KS)是一种罕见的遗传性疾病(患病率<1/1,000,000),其特征为自闭症谱系障碍(ASD)、儿童期肌张力减退和癫痫发作。典型的面部特征包括小头畸形、拱形眉毛、皱眉、眼距过宽、短鼻、面中部发育不全、下颌前突和伸舌。本病例报告从牙医的角度介绍了一名19岁的KS女性患者。患者表现为张口受限,下颌向左侧轻度偏斜。下颌前突和前牙开合可见。广泛性炎症性牙龈增生很可能是对牙菌斑和牙结石等局部刺激物的反应,但患者的口呼吸习惯加剧了这种情况。此外,舌头异常大——巨舌症。通过数字化全景X线片诊断出牙列异常,包括上颌左侧第二前磨牙牙根弯曲和牛牙症。该患者符合在全身麻醉下进行多次拔牙、修复、洁治程序和牙齿再矿化治疗的条件。该患者还应考虑进行正畸治疗以及最终的缩舌手术。对于KS患者,建议多学科合作以及频繁进行牙科检查并进行氟化物预防。不幸的是,牙科治疗仍然不是照顾患有罕见疾病的残疾儿童和青少年的一个组成部分。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/1caa/10541297/5d101edf8aec/CRID2023-2478465.005.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/1caa/10541297/35722ebc24a7/CRID2023-2478465.001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/1caa/10541297/7ebed79aa026/CRID2023-2478465.002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/1caa/10541297/c53e0b737230/CRID2023-2478465.003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/1caa/10541297/dc94e4d07811/CRID2023-2478465.004.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/1caa/10541297/5d101edf8aec/CRID2023-2478465.005.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/1caa/10541297/35722ebc24a7/CRID2023-2478465.001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/1caa/10541297/7ebed79aa026/CRID2023-2478465.002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/1caa/10541297/c53e0b737230/CRID2023-2478465.003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/1caa/10541297/dc94e4d07811/CRID2023-2478465.004.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/1caa/10541297/5d101edf8aec/CRID2023-2478465.005.jpg

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本文引用的文献

1
De Novo Mutation in KMT2C Manifesting as Kleefstra Syndrome 2: Case Report and Literature Review.表现为克莱夫斯特拉综合征2型的KMT2C基因新发突变:病例报告及文献综述
Pediatr Rep. 2022 Mar 11;14(1):131-139. doi: 10.3390/pediatric14010019.
2
New Insights into Kleefstra Syndrome: Report of Two Novel Cases with Previously Unreported Features and Literature Review.克莱夫斯特拉综合征的新见解:两例具有先前未报道特征的新病例报告及文献综述
Cytogenet Genome Res. 2018;156(3):127-133. doi: 10.1159/000494532. Epub 2018 Nov 17.
3
Hypogonadotropic Hypogonadism and Kleefstra Syndrome due to a Pathogenic Variant in the Gene: An Underrecognized Association.
由于该基因的致病性变异导致的低促性腺激素性性腺功能减退和克莱夫斯特拉综合征:一种未被充分认识的关联。
Case Rep Endocrinol. 2018 Oct 2;2018:4283267. doi: 10.1155/2018/4283267. eCollection 2018.
4
Pulmonary hypertension in patients with 9q34.3 microdeletion-associated Kleefstra syndrome.9q34.3 微缺失相关 Kleefstra 综合征患者的肺动脉高压。
Am J Med Genet A. 2018 Aug;176(8):1773-1777. doi: 10.1002/ajmg.a.38852. Epub 2018 Jul 31.
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Kleefstra Syndrome: The First Case Report From Iran.克莱夫斯特拉综合征:来自伊朗的首例病例报告。
Acta Med Iran. 2017 Oct;55(10):650-654.
6
A novel frameshift deletion in in a patient with Kleefstra Syndrome results in decreased H3K9 dimethylation.一名患有克莱夫斯特拉综合征患者的一种新型移码缺失导致H3K9二甲基化减少。
Mol Genet Genomic Med. 2017 Jan 26;5(2):141-146. doi: 10.1002/mgg3.268. eCollection 2017 Mar.
7
A Novel Kleefstra Syndrome-associated Variant That Affects the Conserved TPL Motif within the Ankyrin Repeat of EHMT1 Leads to Abnormal Protein Folding.一种与克莱夫斯特拉综合征相关的新型变异体,它影响EHMT1锚蛋白重复序列内保守的TPL基序,导致蛋白质折叠异常。
J Biol Chem. 2017 Mar 3;292(9):3866-3876. doi: 10.1074/jbc.M116.770545. Epub 2017 Jan 5.
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Kleefstra syndrome in Hungarian patients: additional symptoms besides the classic phenotype.匈牙利患者的克莱夫斯特拉综合征:除经典表型外的其他症状
Mol Cytogenet. 2016 Feb 25;9:22. doi: 10.1186/s13039-016-0231-2. eCollection 2016.
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Severe neonatal presentation of Kleefstra syndrome in a patient with hypoplastic left heart syndrome and 9q34.3 microdeletion.一名患有左心发育不全综合征和9q34.3微缺失的患者出现严重的新生儿期克莱夫斯特拉综合征表现。
Birth Defects Res A Clin Mol Teratol. 2014 Dec;100(12):985-90. doi: 10.1002/bdra.23324. Epub 2014 Nov 7.
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Intragenic duplication of EHMT1 gene results in Kleefstra syndrome.EHMT1基因的基因内重复导致克莱夫斯特拉综合征。
Mol Cytogenet. 2014 Oct 23;7(1):74. doi: 10.1186/s13039-014-0074-7. eCollection 2014.