Dental anomalies in pediatric patients with familial adenomatous polyposis

被引:11
作者
Septer, Seth [1 ]
Bohaty, Brenda [2 ,3 ]
Onikul, Robin [3 ]
Kumar, Vandana [4 ]
Williams, Karen B. [5 ]
Attard, Thomas M. [1 ]
Friesen, Craig A. [1 ]
Friesen, Lynn Roosa [6 ,7 ]
机构
[1] Childrens Mercy Kansas City, Div Gastroenterol Hepatol & Nutr, 2401 Gillham Rd, Kansas City, MO 64108 USA
[2] Univ Missouri, Sch Dent, Dept Paediat Dent, Kansas City, MO USA
[3] Childrens Mercy Kansas City, Dept Dent, 2401 Gillham Rd, Kansas City, MO 64108 USA
[4] Univ Missouri, Dept Oral Pathol Med & Oral Radiol, Sch Dent, 650 E 25th St, Kansas City, MO 64108 USA
[5] Univ Missouri, Dept Biomed & Hlth Informat, Sch Med, 2411 Holmes St, Kansas City, MO 64108 USA
[6] Univ Missouri, Sch Dent, Dept Corp Clin Res, 650 E 25th St, Kansas City, MO 64108 USA
[7] Univ Missouri, Sch Dent, Dept Res & Grad Programs, 650 E 25th St, Kansas City, MO 64108 USA
关键词
Familial adenomatous polyposis; Adenomatous polyposis coli gene; Colorectal carcinoma; Osteomas; Dental anomalies; FAP; MAXILLOFACIAL MANIFESTATIONS; GARDNERS-SYNDROME; APC GENE; MUTATIONS; PHENOTYPE; TUMORS; JAWS;
D O I
10.1007/s10689-017-0035-5
中图分类号
R73 [肿瘤学];
学科分类号
100214 ;
摘要
Familial adenomatous polyposis patients often present with non-malignant extra-intestinal manifestations which include dental anomalies that may be evident prior to the appearance of the colonic adenomas. The aims of this study were to describe the prevalence and type of dental anomalies and the relationships between gene mutations and dental anomalies in these patients. Twenty-two pediatric familial adenomatous polyposis patients and 46 controls, who were age and gender matched participated. Familial adenomatous polyposis patient's had a dental examination with panoramic radiograph and medical record review for age at diagnosis, the presence of the adenomatous polyposis coli gene mutation, and determination of other extra-intestinal manifestations on the body. The control group was identified from a retrospective chart review and selected if there was a current panoramic radiograph. The only significant difference between familial adenomatous polyposis patients and controls were the presence of jaw osteomas and sclerosis (p = .0001). Patients with a mutation in, or upstream of codon 1309 had a higher frequency of osteomas (77.8%) and jaw-bone sclerosis (44.4%), and 77% of these had at least one dental anomaly. This preliminary study showed an association between a genetic variant at, or upstream of codon 1309, and radiographic dental anomalies.
引用
收藏
页码:229 / 234
页数:6
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