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一例麦库西克-考夫曼综合征病例。

A case of McKusick-Kaufman syndrome.

作者信息

Son Se-Hyung, Kim Yoon Joo, Kim Eun Sun, Kim Ee-Kyung, Kim Han-Suk, Kim Beyong Il, Choi Jung-Hwan

机构信息

Department of Pediatrics, Seoul National University College of Medicine, Seoul, Korea.

出版信息

Korean J Pediatr. 2011 May;54(5):219-23. doi: 10.3345/kjp.2011.54.5.219. Epub 2011 May 31.

DOI:10.3345/kjp.2011.54.5.219
PMID:21829414
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC3145907/
Abstract

McKusick-Kaufman syndrome (MKS) is an autosomal recessive multiple malformation syndrome characterized by hydrometrocolpos (HMC) and postaxial polydactyly (PAP). We report a case of a female child with MKS who was transferred to the neonatal intensive care unit of Seoul National University Children's Hospital on her 15th day of life for further evaluation and management of an abdominal cystic mass. She underwent abdominal sonography, magnetic resonance imaging, genitography and cystoscopy which confirmed HMC with a transverse vaginal septum. X-rays of the hand and foot showed bony fusion of the left third and fourth metacarpal bones, right fourth dysplastic metacarpal bone and phalanx, right PAP and hypoplastic left foot with left fourth and fifth dysplastic metatarsal bones. In addition, she had soft palate cleft, mild hydronephroses of both kidneys, hypoplastic right kidney with ectopic location and mild rotation, uterine didelphys with transverse vaginal septum and low-type imperforated anus. She was temporarily treated with ultrasound-guided transurethral aspiration of the HMC. Our patient with HMC and PAP was diagnosed with MKS because she has two typical abnormality of MKS and she has no definite complications of retinal disease, learning disability, obesity and renal failure that develop in Bardet-Biedl syndrome, but not in MKS until 33 months of age. Here, we describe a case of a Korean patient with MKS.

摘要

麦库西克-考夫曼综合征(MKS)是一种常染色体隐性遗传的多发性畸形综合征,其特征为阴道积血积脓(HMC)和轴后多指(PAP)。我们报告一例患有MKS的女童,她在出生第15天被转至首尔国立大学儿童医院新生儿重症监护病房,以便对腹部囊性肿块进行进一步评估和处理。她接受了腹部超声、磁共振成像、泌尿生殖系造影和膀胱镜检查,结果证实为伴有横向阴道隔的HMC。手足X线片显示左第三和第四掌骨骨性融合、右第四掌骨发育异常及指骨、右PAP以及左足发育不全伴左第四和第五跖骨发育异常。此外,她还患有软腭裂、双肾轻度肾盂积水、右肾发育不全伴异位及轻度旋转、双子宫伴横向阴道隔以及低位无孔肛门。她接受了超声引导下经尿道抽吸HMC的临时治疗。我们这位患有HMC和PAP的患者被诊断为MKS,因为她有MKS的两种典型异常,且在33个月大时没有出现巴德-比德尔综合征中会出现但MKS中直到此时尚未出现的视网膜疾病、学习障碍、肥胖和肾衰竭等明确并发症。在此,我们描述一例韩国MKS患者的病例。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/ac54/3145907/c5618940f05f/kjped-54-219-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/ac54/3145907/08bf27c5cd73/kjped-54-219-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/ac54/3145907/4b44c5c3ae72/kjped-54-219-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/ac54/3145907/c5618940f05f/kjped-54-219-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/ac54/3145907/08bf27c5cd73/kjped-54-219-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/ac54/3145907/4b44c5c3ae72/kjped-54-219-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/ac54/3145907/c5618940f05f/kjped-54-219-g003.jpg

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本文引用的文献

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A Novel Familial BBS12 Mutation Associated with a Mild Phenotype: Implications for Clinical and Molecular Diagnostic Strategies.一种与轻度表型相关的新型家族性BBS12突变:对临床和分子诊断策略的启示
Mol Syndromol. 2010 Feb;1(1):27-34. doi: 10.1159/000276763. Epub 2010 Jan 15.
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Mutation analysis of the MKKS gene in McKusick-Kaufman syndrome and selected Bardet-Biedl syndrome patients.麦库西克-考夫曼综合征及部分巴德-比埃尔综合征患者中MKKS基因的突变分析
Hum Genet. 2002 Jun;110(6):561-7. doi: 10.1007/s00439-002-0733-3. Epub 2002 May 9.
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Phenotypic overlap of McKusick-Kaufman syndrome with bardet-biedl syndrome: a literature review.
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麦库西克-考夫曼综合征与巴德-比德尔综合征的表型重叠:文献综述
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