• 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

帕利斯特-基利安综合征:产前绒毛膜绒毛、产后淋巴细胞及生长缓慢的表皮细胞的核型正常,但皮肤成纤维细胞存在12号染色体短臂的嵌合性四体。

Pallister-Killian syndrome: normal karyotype in prenatal chorionic villi, in postnatal lymphocytes, and in slowly growing epidermal cells, but mosaic tetrasomy 12p in skin fibroblasts.

作者信息

Horn D, Majewski F, Hildebrandt B, Körner H

机构信息

Institute of Medical Genetics, School of Medicine (Charité), Humboldt University, Berlin, Germany.

出版信息

J Med Genet. 1995 Jan;32(1):68-71. doi: 10.1136/jmg.32.1.68.

DOI:10.1136/jmg.32.1.68
PMID:7897632
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC1050184/
Abstract

We report on two patients with Pallister-Killian syndrome: an 18 month old male infant followed since the neonatal period and a 4 year old boy. Prenatal diagnosis by chorionic villi sampling (CVS) in the first case showed a normal karyotype without mosaicism. Chromosome analysis on peripheral lymphocytes of the newborn also showed a normal karyotype. The clinical diagnosis of Pallister-Killian syndrome was made after the first year of life because of the typical facial dysmorphism and other characteristic clinical features, such as frontotemporal alopecia, depigmented area of the skin, sensorineural hearing loss, and severe psychomotor retardation. Chromosome analysis from skin fibroblasts now showed an isochromosome 12p mosaicism. The origin of the extra chromosome was confirmed by in situ hybridisation using a chromosome 12 specific library. In the second case chromosomal analysis from peripheral lymphocytes at the age of 19 months showed a normal karyotype 46,XY. Following the clinical diagnosis of Pallister-Killian syndrome a superficial skin biopsy was performed which showed very poor and slow growth of cells and a normal karyotype. Because of the typical symptoms a larger and deeper skin biopsy was performed from which there was rapid growth of fibroblasts. Now the diagnosis was established on the basis of the presence of an i(12p) extra chromosome in 69% of the metaphases.

摘要

我们报告了两名患有帕利斯特-基利安综合征的患者:一名自新生儿期起就接受随访的18个月大男婴和一名4岁男孩。第一例通过绒毛取样(CVS)进行的产前诊断显示核型正常,无嵌合体现象。新生儿外周淋巴细胞的染色体分析也显示核型正常。由于典型的面部畸形及其他特征性临床症状,如额颞部脱发、皮肤色素脱失区、感音神经性听力损失和严重精神运动发育迟缓,在患儿1岁后做出了帕利斯特-基利安综合征的临床诊断。此时皮肤成纤维细胞的染色体分析显示存在12号染色体短臂等臂染色体嵌合体。使用12号染色体特异性文库进行原位杂交证实了额外染色体的来源。在第二例中,19个月大时外周淋巴细胞的染色体分析显示核型正常,为46,XY。在帕利斯特-基利安综合征临床诊断后,进行了一次浅表皮肤活检,结果显示细胞生长非常缓慢且不良,核型正常。由于出现了典型症状,于是进行了一次更大且更深部的皮肤活检,从中发现成纤维细胞生长迅速。现在根据69%的中期相中存在一条额外的12号染色体短臂等臂染色体确诊。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/a2f6/1050184/9e2cf735812b/jmedgene00268-0072-c.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/a2f6/1050184/2504aa38a77a/jmedgene00268-0071-a.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/a2f6/1050184/d21b0d9d214e/jmedgene00268-0071-b.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/a2f6/1050184/bac374ad5db0/jmedgene00268-0072-a.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/a2f6/1050184/471a6469ee01/jmedgene00268-0072-b.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/a2f6/1050184/9e2cf735812b/jmedgene00268-0072-c.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/a2f6/1050184/2504aa38a77a/jmedgene00268-0071-a.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/a2f6/1050184/d21b0d9d214e/jmedgene00268-0071-b.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/a2f6/1050184/bac374ad5db0/jmedgene00268-0072-a.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/a2f6/1050184/471a6469ee01/jmedgene00268-0072-b.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/a2f6/1050184/9e2cf735812b/jmedgene00268-0072-c.jpg

相似文献

1
Pallister-Killian syndrome: normal karyotype in prenatal chorionic villi, in postnatal lymphocytes, and in slowly growing epidermal cells, but mosaic tetrasomy 12p in skin fibroblasts.帕利斯特-基利安综合征:产前绒毛膜绒毛、产后淋巴细胞及生长缓慢的表皮细胞的核型正常,但皮肤成纤维细胞存在12号染色体短臂的嵌合性四体。
J Med Genet. 1995 Jan;32(1):68-71. doi: 10.1136/jmg.32.1.68.
2
Pallister-Killian syndrome: Cytogenetics and molecular investigations of mosaic tetrasomy 12p in prenatal chorionic villus and in amniocytes. Strategy of prenatal diagnosis.帕利斯特-基利安综合征:产前绒毛膜绒毛和羊水中12号染色体短臂镶嵌四体的细胞遗传学和分子研究。产前诊断策略。
Taiwan J Obstet Gynecol. 2016 Dec;55(6):863-866. doi: 10.1016/j.tjog.2016.07.010.
3
A case of mosaic tetrasomy 12p (Pallister-Killian Syndrome) diagnosed prenatally: comparison of chromosome analyses of various cells obtained from the patient.1例产前诊断的12p染色体镶嵌体三体(帕利斯特-基利安综合征):对该患者不同细胞的染色体分析比较
Am J Perinatol. 1997 Nov;14(10):641-3. doi: 10.1055/s-2008-1040769.
4
Clinical, cytogenetic, and molecular observations in a patient with Pallister-Killian-syndrome with an unusual karyotype.一名患有具有异常核型的帕利斯特-基利安综合征患者的临床、细胞遗传学和分子观察结果。
Am J Med Genet A. 2003 Dec 15;123A(3):296-300. doi: 10.1002/ajmg.a.20339.
5
Pallister-Killian syndrome in older children and adolescents.大龄儿童及青少年的帕利斯特-基利安综合征
Pediatr Neurol. 1993 Jul-Aug;9(4):312-5. doi: 10.1016/0887-8994(93)90071-j.
6
Prenatal diagnosis of the Pallister-Killian mosaic aneuploidy syndrome by CVS.经绒毛取样术对帕利斯特-基利安嵌合非整倍体综合征进行产前诊断。
Am J Med Genet. 1992 Mar 1;42(5):747-50. doi: 10.1002/ajmg.1320420525.
7
The use of interphase FISH for prenatal diagnosis of Pallister-Killian syndrome.
Prenat Diagn. 1997 Mar;17(3):255-65. doi: 10.1002/(sici)1097-0223(199703)17:3<255::aid-pd49>3.0.co;2-t.
8
Tissue-limited mosaicism in Pallister-Killian syndrome -- a case in point.帕利斯特-基利安综合征中的组织局限性嵌合体——一个典型案例。
J Perinatol. 2002 Jul-Aug;22(5):420-3. doi: 10.1038/sj.jp.7210712.
9
Report of two new cases of Pallister-Killian syndrome confirmed by FISH: tissue-specific mosaicism and loss of i(12p) by in vitro selection.通过荧光原位杂交(FISH)确诊的两例帕利斯特-基利安综合征新病例报告:组织特异性嵌合体以及体外选择导致i(12p)丢失
Am J Med Genet. 1997 Oct 3;72(1):106-10. doi: 10.1002/(sici)1096-8628(19971003)72:1<106::aid-ajmg21>3.0.co;2-u.
10
Isochromosome 12p mosaicism (Pallister-Killian syndrome): newborn diagnosis by direct bone marrow analysis.
Am J Med Genet. 1988 Dec;31(4):835-9. doi: 10.1002/ajmg.1320310416.

引用本文的文献

1
Prenatal diagnosis of a case with complete and uniform tetrasomy 12p by the utility of noninvasive prenatal testing.应用无创性产前检测对一例完全且均一性 12p 三体综合征进行产前诊断。
J Assist Reprod Genet. 2023 Sep;40(9):2233-2240. doi: 10.1007/s10815-023-02896-8. Epub 2023 Jul 28.
2
Pallister-Killian syndrome in a two-year-old boy.一名两岁男孩患帕利斯特-基利安综合征。
Clin Case Rep. 2017 Apr 8;5(6):774-777. doi: 10.1002/ccr3.892. eCollection 2017 Jun.
3
Rare case of Killian-Pallister syndrome associated with idiopathic short stature detected with fluorescent in situ hybridization on buccal smear.

本文引用的文献

1
Late fetal pontine destruction.晚期胎儿脑桥破坏。
Pediatr Neurol. 1993 May-Jun;9(3):213-5. doi: 10.1016/0887-8994(93)90087-s.
2
Prenatal diagnosis of tetrasomy 47,XY,+i(12p) confirmed by in situ hybridization.
Prenat Diagn. 1988 Feb;8(2):85-91. doi: 10.1002/pd.1970080202.
3
Risk effect of maternal age in Pallister i(12p) syndrome.
Clin Genet. 1988 Sep;34(3):181-4. doi: 10.1111/j.1399-0004.1988.tb02860.x.
4
通过颊黏膜涂片荧光原位杂交检测出的与特发性身材矮小相关的Killian-Pallister综合征罕见病例。
Mol Cytogenet. 2016 May 3;9:38. doi: 10.1186/s13039-016-0239-7. eCollection 2016.
4
Genome-wide expression analysis in fibroblast cell lines from probands with Pallister Killian syndrome.帕利斯特-基利安综合征先证者成纤维细胞系的全基因组表达分析。
PLoS One. 2014 Oct 16;9(10):e108853. doi: 10.1371/journal.pone.0108853. eCollection 2014.
5
Pallister-Killian syndrome.帕利斯特-基利安综合征
Am J Case Rep. 2014 May 7;15:194-8. doi: 10.12659/AJCR.890614. eCollection 2014.
6
45,X/46,XY mosaicism and Oculo-Auriculo-Vertebral Spectrum following an IVF pregnancy: a report and a discussion of their interrelationships.体外受精妊娠后出现的45,X/46,XY嵌合体与眼-耳-脊椎综合征:一例报告及其相互关系探讨
J Maxillofac Oral Surg. 2009 Sep;8(3):279-82. doi: 10.1007/s12663-009-0068-x. Epub 2009 Nov 21.
Mosaic tetrasomy 12p.
Clin Genet. 1985 Dec;28(6):495-502. doi: 10.1111/j.1399-0004.1985.tb00416.x.
5
Pallister-Killian syndrome: cytogenetic and molecular studies.帕利斯特-基利安综合征:细胞遗传学和分子研究
Clin Genet. 1987 Jun;31(6):399-405. doi: 10.1111/j.1399-0004.1987.tb02832.x.
6
Tetrasomy 12p (Pallister-Killian syndrome).12号染色体短臂四体(帕利斯特-基利安综合征)。
J Med Genet. 1991 Feb;28(2):122-5. doi: 10.1136/jmg.28.2.122.
7
Pallister Killian--mosaic tetrasomy 12 p syndrome. Another prenatally diagnosed case.
Prenat Diagn. 1991 Apr;11(4):271-5. doi: 10.1002/pd.1970110409.
8
Pallister-Killian syndrome diagnosed by chorionic villus sampling.经绒毛取样诊断的帕利斯特-基利安综合征
Prenat Diagn. 1991 Jul;11(7):477-9. doi: 10.1002/pd.1970110712.
9
Prenatal diagnosis of Pallister-Killian syndrome.帕利斯特-基利安综合征的产前诊断。
Am J Med Genet. 1990 Apr;35(4):526-8. doi: 10.1002/ajmg.1320350417.
10
Failure of PHA-stimulated i(12p) lymphocytes to divide in Pallister-Killian syndrome.
Am J Med Genet. 1992 Apr 1;42(6):815-9. doi: 10.1002/ajmg.1320420614.