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2017年美国小儿骨科学会尼古拉斯·安德里奖:通过转化研究推动马蹄内翻足的个性化医疗。

The 2017 ABJS Nicolas Andry Award: Advancing Personalized Medicine for Clubfoot Through Translational Research.

作者信息

Dobbs Matthew B, Gurnett Christina A

机构信息

Department of Orthopaedics, St. Louis Children's Hospital, 1 Children's Place, Suite 4S-60, St. Louis, MO, 63110, USA.

Department of Neurology, Washington University School of Medicine in St Louis, St. Louis, MO, USA.

出版信息

Clin Orthop Relat Res. 2017 Jun;475(6):1716-1725. doi: 10.1007/s11999-017-5290-0. Epub 2017 Feb 24.

Abstract

BACKGROUND

Clubfoot is one of the most common pediatric orthopaedic disorders. While the Ponseti method has revolutionized clubfoot treatment, it is not effective for all patients. When the Ponseti method does not correct the foot, patients are at risk for lifelong disability and may require more-extensive surgery.

QUESTIONS/PURPOSES: (1) What genetic and morphologic abnormalities contribute to the development of clubfoot? (2) How can this information be used to devise personalized treatment paradigms for patients with clubfoot?

METHODS

Human gene sequencing, molecular genetic engineering of mouse models of clubfoot, MRI of clubfoot, and development of new treatment methods all have been used by our group to understand the biological basis and improve therapy for this group of disorders.

RESULTS

We gained new insight into clubfoot pathogenesis from our discovery that mutations in the PITX1-TBX4-HOXC transcriptional pathway cause familial clubfoot and vertical talus in a small number of families, with the unique lower limb expression of these genes providing an explanation for the lack of upper extremity involvement in these disorders. MRI studies revealed corresponding morphologic abnormalities, including hypomorphic muscle, bone, and vasculature, that are not only associated with these gene mutations, but also are biomarkers for treatment-resistant clubfoot.

CONCLUSIONS

Based on an understanding of the underlying biology, we improved treatment methods for neglected and syndromic clubfoot, developed new treatment for congenital vertical talus based on the principles of the Ponseti method, and designed a new dynamic clubfoot brace to improve strength and compliance.

摘要

背景

马蹄内翻足是最常见的儿科骨科疾病之一。虽然庞塞蒂方法彻底改变了马蹄内翻足的治疗方式,但并非对所有患者都有效。当庞塞蒂方法无法矫正足部时,患者有终身残疾的风险,可能需要更广泛的手术。

问题/目的:(1)哪些基因和形态学异常导致马蹄内翻足的发生?(2)如何利用这些信息为马蹄内翻足患者设计个性化的治疗方案?

方法

我们团队利用人类基因测序、马蹄内翻足小鼠模型的分子基因工程、马蹄内翻足的磁共振成像以及新治疗方法的开发,来了解这组疾病的生物学基础并改善治疗效果。

结果

我们发现PITX1-TBX4-HOXC转录途径中的突变在少数家族中导致家族性马蹄内翻足和垂直距骨,这使我们对马蹄内翻足的发病机制有了新的认识,这些基因在下肢的独特表达解释了这些疾病为何不累及上肢。磁共振成像研究揭示了相应的形态学异常,包括发育不全的肌肉、骨骼和脉管系统,这些不仅与这些基因突变有关,也是治疗抵抗性马蹄内翻足的生物标志物。

结论

基于对潜在生物学的理解我们改进了 neglected 和综合征性马蹄内翻足的治疗方法,根据庞塞蒂方法的原则开发了先天性垂直距骨的新治疗方法,并设计了一种新的动态马蹄内翻足支具以提高强度和顺应性。

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本文引用的文献

1
Design and descriptive data of the randomized Clubfoot Foot Abduction Brace Length of Treatment Study (FAB24).
J Pediatr Orthop B. 2017 Mar;26(2):101-107. doi: 10.1097/BPB.0000000000000387.
2
A comparison of the Dobbs method for correction of idiopathic and teratological congenital vertical talus.
J Child Orthop. 2016 Apr;10(2):93-9. doi: 10.1007/s11832-016-0727-7. Epub 2016 Apr 2.
3
Deletions of 5' HOXC genes are associated with lower extremity malformations, including clubfoot and vertical talus.
J Med Genet. 2016 Apr;53(4):250-5. doi: 10.1136/jmedgenet-2015-103505. Epub 2016 Jan 4.
6
Serial casting for neuromuscular flatfoot and vertical talus in an adolescent with hereditary spastic paraplegia.
Pediatr Phys Ther. 2014 Summer;26(2):253-64. doi: 10.1097/PEP.0000000000000023.
7
Genome-wide association study identifies new disease loci for isolated clubfoot.
J Med Genet. 2014 May;51(5):334-9. doi: 10.1136/jmedgenet-2014-102303. Epub 2014 Mar 25.

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