Literature DB >> 17502954

Holt-Oram syndrome.

Corina Lichiardopol1, C Militaru, B Popescu, G Hila, F Mixich.   

Abstract

The Holt-Oram syndrome or atriodigital dysplasia is an autosomal dominant disorder with near complete penetrance and variable expression, caused by mutations of the TBX5 gene (12q24.1), affecting one in 100 000 live births. 60% of cases are familial and 40% sporadic. We present the case of a 24 years old male patient with a personal history of bilateral coxa vara surgically corrected on the right at the age of 8 years, complicated by osteochondritis, short stature (160 cm), underweight (37 kg, BMI 14.45 kg/cm(2)), triangular face, micrognathia, down slanting palpebral fissures, hypertelorism, low set ears, scoliosis, narrow shoulders, shortened left arm, left thumb agenesia, limited supination, abnormal toes, hypoplastic muscles, atrial septal defect ostium secundum type, incomplete right bundle branch block, hypoacusia and normal intelligence.

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Year:  2007        PMID: 17502954

Source DB:  PubMed          Journal:  Rom J Morphol Embryol        ISSN: 1220-0522            Impact factor:   1.033


  8 in total

Review 1.  MicroRNAs: pleiotropic players in congenital heart disease and regeneration.

Authors:  Sarah C Hoelscher; Stefanie A Doppler; Martina Dreßen; Harald Lahm; Rüdiger Lange; Markus Krane
Journal:  J Thorac Dis       Date:  2017-03       Impact factor: 2.895

2.  T-box family of transcription factor-TBX5, insights in development and disease.

Authors:  Ting Zhu; Longwei Qiao; Qian Wang; Rui Mi; Jinnan Chen; Yaojuan Lu; Junxia Gu; Qiping Zheng
Journal:  Am J Transl Res       Date:  2017-02-15       Impact factor: 4.060

3.  Anaesthetic management of emergency cesarean section in a patient with holt oram syndrome.

Authors:  Babu N Girish; S Rajesh; P Somasekharam; Pradeep Kumar
Journal:  J Anaesthesiol Clin Pharmacol       Date:  2010-10

4.  Atypical carpal tunnel syndrome in a holt oram patient: a case report and literature review.

Authors:  James Mace; Srikanth Reddy; Randeep Mohil
Journal:  Open Orthop J       Date:  2014-12-29

Review 5.  Holt-Oram Syndrome in a Patient with Crohn's Disease: a Rare Case Report and Literature Review.

Authors:  Panagiotis-Theofanis Arkoumanis; Antonios Gklavas; Margarita Karageorgou; Polyxeni Gourzi; Gerassimos Mantzaris; Malena Pantou; Ioannis Papaconstantinou
Journal:  Med Arch       Date:  2018-10

6.  TBX5-encoded T-box transcription factor 5 variant T223M is associated with long QT syndrome and pediatric sudden cardiac death.

Authors:  Alexandra M Markunas; Perathu K R Manivannan; Jordan E Ezekian; Agnim Agarwal; William Eisner; Katherina Alsina; Hugh D Allen; Gregory A Wray; Jeffrey J Kim; Xander H T Wehrens; Andrew P Landstrom
Journal:  Am J Med Genet A       Date:  2020-12-23       Impact factor: 2.802

7.  Coexisting urogenital anomaly and duodenal atresia in two atypical Holt-Oram syndrome.

Authors:  Tuncer Ahmet Ali; Karavelioğlu Afra; Baskin Embleton Didem; Elmas Muhsin
Journal:  J Indian Assoc Pediatr Surg       Date:  2016 Oct-Dec

8.  A novel de novo TBX5 mutation in a patient with Holt-Oram syndrome leading to a dramatically reduced biological function.

Authors:  Martina Dreßen; Harald Lahm; Armin Lahm; Klaudia Wolf; Stefanie Doppler; Marcus-André Deutsch; Julie Cleuziou; Jelena Pabst von Ohain; Patric Schön; Peter Ewert; Ivan Malcic; Rüdiger Lange; Markus Krane
Journal:  Mol Genet Genomic Med       Date:  2016-07-14       Impact factor: 2.183

  8 in total

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