• 文献检索
  • 文档翻译
  • 深度研究
  • 学术资讯
  • Suppr Zotero 插件Zotero 插件
  • 邀请有礼
  • 套餐&价格
  • 历史记录
应用&插件
Suppr Zotero 插件Zotero 插件浏览器插件Mac 客户端Windows 客户端微信小程序
定价
高级版会员购买积分包购买API积分包
服务
文献检索文档翻译深度研究API 文档MCP 服务
关于我们
关于 Suppr公司介绍联系我们用户协议隐私条款
关注我们

Suppr 超能文献

核心技术专利:CN118964589B侵权必究
粤ICP备2023148730 号-1Suppr @ 2026

文献检索

告别复杂PubMed语法,用中文像聊天一样搜索,搜遍4000万医学文献。AI智能推荐,让科研检索更轻松。

立即免费搜索

文件翻译

保留排版,准确专业,支持PDF/Word/PPT等文件格式,支持 12+语言互译。

免费翻译文档

深度研究

AI帮你快速写综述,25分钟生成高质量综述,智能提取关键信息,辅助科研写作。

立即免费体验

相似文献

1
Macrocerebellum, epilepsy, intellectual disability, and gut malrotation in a child with a 16q24.1-q24.2 contiguous gene deletion.一名患有16q24.1 - q24.2连续基因缺失的儿童出现巨小脑、癫痫、智力残疾和肠道旋转不良。
Am J Med Genet A. 2014 Aug;164A(8):2062-8. doi: 10.1002/ajmg.a.36569. Epub 2014 Apr 9.
2
Partial deletion of ANKRD11 results in the KBG phenotype distinct from the 16q24.3 microdeletion syndrome.ANKRD11 部分缺失导致 KBG 表型与 16q24.3 微缺失综合征不同。
Am J Med Genet A. 2013 Apr;161A(4):835-40. doi: 10.1002/ajmg.a.35739. Epub 2013 Mar 12.
3
Deletions in 16q24.2 are associated with autism spectrum disorder, intellectual disability and congenital renal malformation.16q24.2 缺失与自闭症谱系障碍、智力障碍和先天性肾脏畸形有关。
J Med Genet. 2013 Mar;50(3):163-73. doi: 10.1136/jmedgenet-2012-101288. Epub 2013 Jan 18.
4
KBG syndrome: 16q24.3 microdeletion in an Indian patient.KBG综合征:一名印度患者的16号染色体长臂24.3区微缺失
Clin Dysmorphol. 2017 Jul;26(3):161-166. doi: 10.1097/MCD.0000000000000168.
5
Refinement of the deletion in 8q22.2-q22.3: the minimum deletion size at 8q22.3 related to intellectual disability and epilepsy.8q22.2-q22.3区域缺失的细化:与智力残疾和癫痫相关的8q22.3最小缺失大小。
Am J Med Genet A. 2014 Aug;164A(8):2104-8. doi: 10.1002/ajmg.a.36604. Epub 2014 May 6.
6
Haploinsufficiency for ANKRD11-flanking genes makes the difference between KBG and 16q24.3 microdeletion syndromes: 12 new cases.ANKRD11侧翼基因的单倍剂量不足导致KBG综合征和16q24.3微缺失综合征的差异:12例新病例
Eur J Hum Genet. 2017 Jun;25(6):694-701. doi: 10.1038/ejhg.2017.49. Epub 2017 Apr 19.
7
Clinical and molecular findings in 39 patients with KBG syndrome caused by deletion or mutation of ANKRD11.39例由ANKRD11缺失或突变引起的KBG综合征患者的临床和分子学发现
Am J Med Genet A. 2016 Nov;170(11):2847-2859. doi: 10.1002/ajmg.a.37878. Epub 2016 Sep 8.
8
High resolution mapping of interstitial long arm deletions of chromosome 16: relationship to phenotype.16号染色体间质性长臂缺失的高分辨率图谱:与表型的关系
J Med Genet. 1993 Oct;30(10):828-32. doi: 10.1136/jmg.30.10.828.
9
The epileptology of Koolen-de Vries syndrome: Electro-clinico-radiologic findings in 31 patients.Koolen-de Vries 综合征的癫痫学:31 例患者的电临床 - 影像学表现。
Epilepsia. 2017 Jun;58(6):1085-1094. doi: 10.1111/epi.13746. Epub 2017 Apr 25.
10
Microcephaly, intellectual impairment, bilateral vesicoureteral reflux, distichiasis, and glomuvenous malformations associated with a 16q24.3 contiguous gene deletion and a Glomulin mutation.小头畸形、智力障碍、双侧输尿管反流、双睫和丛状静脉畸形与 16q24.3 连续基因缺失和 Glomulin 突变相关。
Am J Med Genet A. 2012 Apr;158A(4):839-49. doi: 10.1002/ajmg.a.35229. Epub 2012 Mar 9.

引用本文的文献

1
Characteristics of the Kelch domain containing (KLHDC) subfamily and relationships with diseases.含kelch结构域(KLHDC)亚家族的特征及其与疾病的关系。
FEBS Lett. 2025 Apr;599(8):1094-1112. doi: 10.1002/1873-3468.15108. Epub 2025 Jan 30.
2
MiR-3571 modulates traumatic brain injury by regulating the PI3K/AKT signaling pathway via Fbxo31.miR-3571 通过调节 Fbxo31 调控 PI3K/AKT 信号通路来调节创伤性脑损伤。
Cell Biochem Biophys. 2024 Dec;82(4):3629-3643. doi: 10.1007/s12013-024-01452-0. Epub 2024 Jul 31.
3
Huntington disease-like 2: insight into neurodegeneration from an African disease.亨廷顿病样2型:从一种非洲疾病中洞察神经退行性变
Nat Rev Neurol. 2024 Jan;20(1):36-49. doi: 10.1038/s41582-023-00906-y. Epub 2023 Dec 19.
4
Microdeletion of 16q24.1-q24.2-A unique etiology of Lymphedema-Distichiasis syndrome and neurodevelopmental disorder.16q24.1-q24.2 微缺失——淋巴水肿-并指(趾)畸形综合征和神经发育障碍的独特病因。
Am J Med Genet A. 2022 Jul;188(7):1990-1996. doi: 10.1002/ajmg.a.62730. Epub 2022 Mar 21.
5
Compound genetic etiology in a patient with a syndrome including diabetes, intellectual deficiency and distichiasis.患者同时患有糖尿病、智力缺陷和睫毛乱生综合征,存在复合遗传病因。
Orphanet J Rare Dis. 2022 Feb 28;17(1):86. doi: 10.1186/s13023-022-02248-2.
6
The amino acid transporter Slc7a5 regulates the mTOR pathway and is required for granule cell development.氨基酸转运蛋白 Slc7a5 调节 mTOR 通路,是颗粒细胞发育所必需的。
Hum Mol Genet. 2020 Nov 4;29(18):3003-3013. doi: 10.1093/hmg/ddaa186.
7
Isolated macrocerebellum: description of six cases and literature review.孤立性小脑增大:6例病例描述及文献综述
Quant Imaging Med Surg. 2016 Oct;6(5):496-503. doi: 10.21037/qims.2016.06.10.
8
SCF ubiquitin ligase-targeted therapies.SCF泛素连接酶靶向疗法。
Nat Rev Drug Discov. 2014 Dec;13(12):889-903. doi: 10.1038/nrd4432. Epub 2014 Nov 14.

本文引用的文献

1
The centrosomal E3 ubiquitin ligase FBXO31-SCF regulates neuronal morphogenesis and migration.中心体 E3 泛素连接酶 FBXO31-SCF 调节神经元形态发生和迁移。
PLoS One. 2013;8(2):e57530. doi: 10.1371/journal.pone.0057530. Epub 2013 Feb 28.
2
Deletions in 16q24.2 are associated with autism spectrum disorder, intellectual disability and congenital renal malformation.16q24.2 缺失与自闭症谱系障碍、智力障碍和先天性肾脏畸形有关。
J Med Genet. 2013 Mar;50(3):163-73. doi: 10.1136/jmedgenet-2012-101288. Epub 2013 Jan 18.
3
Macrocerebellum: significance and pathogenic considerations.大脑小脑:意义和发病学考虑。
Cerebellum. 2012 Dec;11(4):1026-36. doi: 10.1007/s12311-012-0379-1.
4
Microcephaly, intellectual impairment, bilateral vesicoureteral reflux, distichiasis, and glomuvenous malformations associated with a 16q24.3 contiguous gene deletion and a Glomulin mutation.小头畸形、智力障碍、双侧输尿管反流、双睫和丛状静脉畸形与 16q24.3 连续基因缺失和 Glomulin 突变相关。
Am J Med Genet A. 2012 Apr;158A(4):839-49. doi: 10.1002/ajmg.a.35229. Epub 2012 Mar 9.
5
Loss of junctophilin-3 contributes to Huntington disease-like 2 pathogenesis.衔接蛋白-3 的缺失导致亨廷顿病样 2 型发病机制。
Ann Neurol. 2012 Feb;71(2):245-57. doi: 10.1002/ana.22598.
6
Total and regional brain volumes in a population-based normative sample from 4 to 18 years: the NIH MRI Study of Normal Brain Development.基于人群的从 4 岁至 18 岁正常样本的全脑和区域性脑容量:NIH 正常脑发育 MRI 研究。
Cereb Cortex. 2012 Jan;22(1):1-12. doi: 10.1093/cercor/bhr018. Epub 2011 May 25.
7
Cerebellum enlargement and corpus callosum agenesis: a longitudinal case report.
J Child Neurol. 2011 Jun;26(6):756-60. doi: 10.1177/0883073810387140.
8
An antisense CAG repeat transcript at JPH3 locus mediates expanded polyglutamine protein toxicity in Huntington's disease-like 2 mice.JPH3 基因座的反义 CAG 重复转录本介导亨廷顿病样 2 型小鼠中扩增的多聚谷氨酰胺蛋白毒性。
Neuron. 2011 May 12;70(3):427-40. doi: 10.1016/j.neuron.2011.03.021.
9
Phenotypic analysis of individuals with Costello syndrome due to HRAS p.G13C.HRAS p.G13C 所致 Costello 综合征患者的表型分析。
Am J Med Genet A. 2011 Apr;155A(4):706-16. doi: 10.1002/ajmg.a.33884. Epub 2011 Mar 15.
10
Increased cerebellar volume in the early stage of fucosidosis: a case control study.黏多糖贮积症患者小脑体积在早期增加:病例对照研究。
Neuroradiology. 2011 Jul;53(7):509-16. doi: 10.1007/s00234-011-0855-1. Epub 2011 Mar 8.

一名患有16q24.1 - q24.2连续基因缺失的儿童出现巨小脑、癫痫、智力残疾和肠道旋转不良。

Macrocerebellum, epilepsy, intellectual disability, and gut malrotation in a child with a 16q24.1-q24.2 contiguous gene deletion.

作者信息

Seeley Andrea H, Durham Mark A, Micale Mark A, Wesolowski Jeffrey, Foerster Bradley R, Martin Donna M

机构信息

Department of Pediatrics and Communicable Diseases, University of Michigan Medical School, Ann Arbor, Michigan.

出版信息

Am J Med Genet A. 2014 Aug;164A(8):2062-8. doi: 10.1002/ajmg.a.36569. Epub 2014 Apr 9.

DOI:10.1002/ajmg.a.36569
PMID:24719385
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC4266592/
Abstract

Macrocerebellum is a rare condition characterized by enlargement of the cerebellum with conservation of the overall shape and cytoarchitecture. Here, we report on a child with a distinctive constellation of clinical features including macrocerebellum, epilepsy, apparent intellectual disability, dysautonomia, gut malrotation, and poor gut motility. Oligonucleotide chromosome microarray analysis identified a 16q24.1-q24.2 deletion that included four OMIM genes (FBXO31, MAP1LC3B, JPH3, and SLC7A5). Review of prior studies describing individuals with similar or overlapping16q24.1-q24.2 deletions identified no other reports of macrocerebellum. These observations highlight a potential genetic cause of this rare disorder and raise the possibility that one or more gene(s) in the 16q24.1-q24.2 interval regulate cerebellar development.

摘要

巨小脑是一种罕见病症,其特征为小脑增大而整体形状和细胞结构保持不变。在此,我们报告一名儿童,其具有一系列独特的临床特征,包括巨小脑、癫痫、明显的智力残疾、自主神经功能障碍、肠道旋转不良和肠道蠕动不佳。寡核苷酸染色体微阵列分析确定了一个16q24.1-q24.2缺失,其中包含四个OMIM基因(FBXO31、MAP1LC3B、JPH3和SLC7A5)。回顾先前描述具有相似或重叠16q24.1-q24.2缺失个体的研究,未发现其他关于巨小脑的报告。这些观察结果突出了这种罕见疾病的潜在遗传原因,并增加了16q24.1-q24.2区间内一个或多个基因调节小脑发育的可能性。