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颅面表型与罗宾诺综合征相关。

Craniofacial phenotypes associated with Robinow syndrome.

机构信息

Division of Plastic Surgery, Michael E. DeBakey Department of Surgery, Baylor College of Medicine, Houston, Texas, USA.

Division of Plastic Surgery, Texas Children's Hospital, Houston, Texas, USA.

出版信息

Am J Med Genet A. 2021 Dec;185(12):3606-3612. doi: 10.1002/ajmg.a.61986. Epub 2020 Nov 25.

DOI:10.1002/ajmg.a.61986
PMID:33237614
Abstract

Robinow syndrome is characterized by mesomelic limb shortening, hemivertebrae, and genital hypoplasia. Due to low prevalence and considerable phenotypic variability, it has been challenging to definitively characterize features of Robinow syndrome. While craniofacial abnormalities associated with Robinow syndrome have been broadly described, there is a lack of detailed descriptions of genotype-specific phenotypic craniofacial features. Patients with Robinow syndrome were invited for a multidisciplinary evaluation conducted by specialist physicians at our institution. A focused assessment of the craniofacial manifestations was performed by a single expert examiner using clinical examination and standard photographic images. A total of 13 patients with clinical and molecular diagnoses consistent with either dominant Robinow syndrome (DRS) or recessive Robinow syndrome (RRS) were evaluated. On craniofacial examination, gingival hyperplasia was nearly ubiquitous in all patients. Orbital hypertelorism, a short nose with anteverted and flared nares, a triangular mouth with a long philtrum, cleft palate, macrocephaly, and frontal bossing were not observed in all individuals but affected individuals with both DRS and RRS. Other anomalies were more selective in their distribution in this patient cohort. We present a comprehensive analysis of the craniofacial findings in patients with Robinow Syndrome, describing associated morphological features and correlating phenotypic manifestations to underlying genotype in a manner relevant for early recognition and focused evaluation of these patients.

摘要

罗宾诺综合征的特征为中胚层肢体缩短、半椎体和生殖器发育不全。由于发病率低且表型变异较大,因此明确罗宾诺综合征的特征一直具有挑战性。虽然与罗宾诺综合征相关的颅面异常已被广泛描述,但缺乏对特定基因型表型颅面特征的详细描述。罗宾诺综合征患者应邀在我们机构接受由专科医生进行的多学科评估。由一位专家检查者使用临床检查和标准摄影图像对颅面表现进行了集中评估。共评估了 13 名具有明确临床和分子诊断的患者,这些患者符合显性罗宾诺综合征(DRS)或隐性罗宾诺综合征(RRS)。在颅面检查中,所有患者几乎都存在牙龈增生。眶距增宽、短鼻伴前鼻孔倾斜和外展、三角口伴长人中、腭裂、大头畸形和额骨突出并非所有患者都存在,但 DRS 和 RRS 患者都存在这些特征。其他异常在该患者队列中的分布更具选择性。我们对罗宾诺综合征患者的颅面发现进行了全面分析,描述了相关的形态特征,并将表型表现与潜在的基因型相关联,这对于早期识别和重点评估这些患者具有重要意义。

相似文献

1
Craniofacial phenotypes associated with Robinow syndrome.颅面表型与罗宾诺综合征相关。
Am J Med Genet A. 2021 Dec;185(12):3606-3612. doi: 10.1002/ajmg.a.61986. Epub 2020 Nov 25.
2
Extremity anomalies associated with Robinow syndrome.与罗宾诺综合征相关的肢体异常。
Am J Med Genet A. 2021 Dec;185(12):3584-3592. doi: 10.1002/ajmg.a.61884. Epub 2020 Sep 25.
3
Autosomal dominant Robinow syndrome associated with a novel DVL3 splice mutation.与新型DVL3剪接突变相关的常染色体显性罗宾诺综合征
Am J Med Genet A. 2018 Apr;176(4):992-996. doi: 10.1002/ajmg.a.38635.
4
Phenotypic and mutational spectrum of ROR2-related Robinow syndrome.ROR2 相关性罗宾诺综合征的表型和突变谱。
Hum Mutat. 2022 Jul;43(7):900-918. doi: 10.1002/humu.24375. Epub 2022 May 10.
5
Clinical characterization of autosomal dominant and recessive variants of Robinow syndrome.罗伯诺综合征常染色体显性和隐性变异的临床特征
Am J Med Genet A. 2007 Feb 15;143(4):320-5. doi: 10.1002/ajmg.a.31592.
6
A novel de-novo WNT5A mutation in a Chinese patient with Robinow syndrome.一名患有Robinow综合征的中国患者中发现一种新的WNT5A新生突变。
Clin Dysmorphol. 2016 Oct;25(4):186-9. doi: 10.1097/MCD.0000000000000130.
7
An osteosclerotic form of Robinow syndrome.罗伯诺综合征的骨硬化型。
Am J Med Genet A. 2014 Oct;164A(10):2638-42. doi: 10.1002/ajmg.a.36677. Epub 2014 Jul 14.
8
Robinow syndrome: Genital analysis, genetic heterogeneity, and associated psychological impact.罗宾诺综合征:生殖器分析、遗传异质性及相关心理影响。
Am J Med Genet A. 2021 Dec;185(12):3601-3605. doi: 10.1002/ajmg.a.61981. Epub 2020 Dec 5.
9
DVL3 Alleles Resulting in a -1 Frameshift of the Last Exon Mediate Autosomal-Dominant Robinow Syndrome.导致最后一个外显子发生 -1 移码的 DVL3 等位基因介导常染色体显性遗传性罗宾诺综合征。
Am J Hum Genet. 2016 Mar 3;98(3):553-561. doi: 10.1016/j.ajhg.2016.01.005. Epub 2016 Feb 25.
10
One gene, two phenotypes: ROR2 mutations in autosomal recessive Robinow syndrome and autosomal dominant brachydactyly type B.一个基因,两种表型:常染色体隐性遗传的Robinow综合征和常染色体显性遗传的B型短指症中的ROR2突变
Hum Mutat. 2003 Jul;22(1):1-11. doi: 10.1002/humu.10233.

引用本文的文献

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Wnt5a gain- and loss-of-function present distinctly in craniofacial bone.Wnt5a功能获得和功能缺失在颅面骨中表现明显不同。
bioRxiv. 2025 Jul 22:2025.07.21.665966. doi: 10.1101/2025.07.21.665966.
2
Craniofacial studies in chicken embryos confirm the pathogenicity of human FZD2 variants associated with Robinow syndrome.鸡胚颅面研究证实了与 Robinow 综合征相关的人类 FZD2 变异体的致病性。
Dis Model Mech. 2024 Jun 1;17(6). doi: 10.1242/dmm.050584. Epub 2024 Jul 5.
3
ROR2-Related Skeletal Dysplasia Reveals Disrupted Chondrocyte Polarity through Modulation of BMP/TGF-β Signaling.
ROR2 相关骨骼发育不良通过调节 BMP/TGF-β 信号通路破坏软骨细胞极性。
Aging Dis. 2024 Feb 1;15(1):282-294. doi: 10.14336/AD.2023.0531.
4
Mechanistic studies in Drosophila and chicken give new insights into functions of DVL1 in dominant Robinow syndrome.在果蝇和鸡中的机制研究为 DVL1 在显性 Robinow 综合征中的功能提供了新的见解。
Dis Model Mech. 2023 Apr 1;16(4). doi: 10.1242/dmm.049844. Epub 2023 Apr 13.
5
Phenotypic and mutational spectrum of ROR2-related Robinow syndrome.ROR2 相关性罗宾诺综合征的表型和突变谱。
Hum Mutat. 2022 Jul;43(7):900-918. doi: 10.1002/humu.24375. Epub 2022 May 10.
6
Novel pathogenic variants and quantitative phenotypic analyses of Robinow syndrome: WNT signaling perturbation and phenotypic variability.Robinow综合征的新型致病变异与定量表型分析:WNT信号通路扰动与表型变异性
HGG Adv. 2021 Dec 3;3(1):100074. doi: 10.1016/j.xhgg.2021.100074. eCollection 2022 Jan 13.