Ricardo Barbosa Lima, Taíssa Cássia de Souza Furtado, Paulo Nelson-Filho, Raquel Assed Bezerra da Silva, Francisco Wanderley Garcia Paula-Silva, Fabrício Kitazono de Carvalho, Alexandra Mussolino de Queiroz
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引用次数: 0
摘要
目的:本病例报告的目的是描述一个RUNX2无义突变与锁骨颅发育不良(CCD)与不寻常的牙齿特征相关。患者是一名12岁的巴西女孩,因乳牙过度固位和未长出恒牙而寻求牙科治疗。临床和x线检查显示多颗阻生恒牙,上颌门牙的永久阻生带突出,除骨骼异常外,牙釉质缺陷(发育不全和低矿化)。没有多余的牙齿。提出了CCD的诊断假设,并将患者转介到遗传医学服务,在那里诊断得到证实。经过RUNX2基因筛选,包括聚合酶链反应和两条DNA链的测序,在外显子2 (c.193)鉴定出一个杂合无义突变C > T [Q65X])。本文报道一例CCD患者的牙齿特征异常。
RUNX2 Nonsense Mutation Associated with Cleidocranial Dysplasia with Unusual Dental Features.
Purpose: The purpose of this case report is to describe a RUNX2 nonsense mutation associated with cleidocranial dysplasia (CCD) with unusual dental features. The patient was a 12-year-old Brazilian girl who sought dental care due to over-retention of primary teeth and absence of erupted permanent teeth. Clinical and radiographic examinations revealed multiple impacted permanent teeth, a prominent cingulum of the permanent impacted maxillary incisors and enamel defects (hypoplasia and hypomineralization) in addition to skeletal abnormalities. No supernumerary teeth were present. The diagnostic hypothesis of CCD was raised and the patient was refer- red to the genetic medical service, where the diagnosis was cofirmed. After RUNX2 genetic screening, including polymerase chain reaction and sequencing of both DNA strands, a heterozygous nonsense mutation was identified in exon 2 (c.193 C>T [Q65X]). This article reports unusual dental features in a patient with CCD.
期刊介绍:
Acquired after the merger between the American Society of Dentistry for Children and the American Academy of Pediatric Dentistry in 2002, the Journal of Dentistry for Children (JDC) is an internationally renowned journal whose publishing dates back to 1934. Published three times a year, JDC promotes the practice, education and research specifically related to the specialty of pediatric dentistry. It covers a wide range of topics related to the clinical care of children, from clinical techniques of daily importance to the practitioner, to studies on child behavior and growth and development. JDC also provides information on the physical, psychological and emotional conditions of children as they relate to and affect their dental health.