Entry - %610926 - TOOTH AGENESIS, SELECTIVE, 5; STHAG5 - OMIM - (MIRROR)
% 610926

TOOTH AGENESIS, SELECTIVE, 5; STHAG5


Alternative titles; symbols

HYPODONTIA/OLIGODONTIA 5
HE-ZHAO DEFICIENCY


HGNC Approved Gene Symbol: STHAG5

Cytogenetic location: 10q11.2-q21   Genomic coordinates (GRCh38) : 10:41,600,001-68,800,000


Gene-Phenotype Relationships
Location Phenotype Phenotype
MIM number
Inheritance Phenotype
mapping key
10q11.2-q21 Tooth agenesis, selective, 5 610926 2
Phenotypic Series
 


TEXT

For a general phenotypic description and a discussion of genetic heterogeneity of selective tooth agenesis, see STHAG1 (106600).


Clinical Features

Wang et al. (2000) described a form of agenesis of permanent teeth in a large Chinese kindred. Penetrance was estimated to be 0.88, and no significant clinical manifestations other than the oligodontia were found. Agenesis ranged from a few teeth to the entire set of teeth, involved only the permanent teeth, and usually appeared at age 7 or 8 years, when primary teeth are normally replaced by permanent teeth. Some of the affected individuals had development of first and second molars. In some cases, it appeared that the remaining teeth were not permanent teeth; instead, they were primary and sometimes remained until the forties. Because of the impression that the tooth abnormality in this kindred had been hitherto undescribed, Wang et al. (2000) referred to it as 'He-Zhao deficiency.' The kindred came from a small village in Xunyi County of Shaanxi Province in northwest China.


Mapping

By linkage analysis in the large Chinese kindred with selective tooth agenesis studied by Wang et al. (2000), Liu et al. (2001) found maximum 2-point and multipoint lod scores of 13.29 at theta = zero (on marker D10S196) and 18.09 (between markers D10S1772 and D10S1766), respectively. Haplotype analysis narrowed the assignment to a 5.5-cM interval flanked by markers D10S604 and D10S568 on chromosome 10q11.2.


REFERENCES

  1. Liu, W., Wang, H., Zhao, S., Zhao, W., Bai, S., Zhao, Y., Xu, S., Wu, C., Huang, W., Chen, Z., Feng, G., He, L. The novel gene locus for agenesis of permanent teeth (He-Zhao deficiency) maps to chromosome 10q11.2. J. Dent. Res. 80: 1716-1720, 2001. [PubMed: 11669481, related citations] [Full Text]

  2. Wang, H., Zhao, S., Zhao, W., Feng, G., Jiang, S., Liu, W., Li, S., Xue, H., He, L. Congenital absence of permanent teeth in a six-generation Chinese kindred. Am. J. Med. Genet. 90: 193-198, 2000. [PubMed: 10678655, related citations]


Creation Date:
Carol A. Bocchini : 4/11/2007
carol : 07/22/2009
carol : 4/11/2007
carol : 4/11/2007

% 610926

TOOTH AGENESIS, SELECTIVE, 5; STHAG5


Alternative titles; symbols

HYPODONTIA/OLIGODONTIA 5
HE-ZHAO DEFICIENCY


HGNC Approved Gene Symbol: STHAG5

ORPHA: 99798;   DO: 0050591;  


Cytogenetic location: 10q11.2-q21   Genomic coordinates (GRCh38) : 10:41,600,001-68,800,000


Gene-Phenotype Relationships

Location Phenotype Phenotype
MIM number
Inheritance Phenotype
mapping key
10q11.2-q21 Tooth agenesis, selective, 5 610926 2

TEXT

For a general phenotypic description and a discussion of genetic heterogeneity of selective tooth agenesis, see STHAG1 (106600).


Clinical Features

Wang et al. (2000) described a form of agenesis of permanent teeth in a large Chinese kindred. Penetrance was estimated to be 0.88, and no significant clinical manifestations other than the oligodontia were found. Agenesis ranged from a few teeth to the entire set of teeth, involved only the permanent teeth, and usually appeared at age 7 or 8 years, when primary teeth are normally replaced by permanent teeth. Some of the affected individuals had development of first and second molars. In some cases, it appeared that the remaining teeth were not permanent teeth; instead, they were primary and sometimes remained until the forties. Because of the impression that the tooth abnormality in this kindred had been hitherto undescribed, Wang et al. (2000) referred to it as 'He-Zhao deficiency.' The kindred came from a small village in Xunyi County of Shaanxi Province in northwest China.


Mapping

By linkage analysis in the large Chinese kindred with selective tooth agenesis studied by Wang et al. (2000), Liu et al. (2001) found maximum 2-point and multipoint lod scores of 13.29 at theta = zero (on marker D10S196) and 18.09 (between markers D10S1772 and D10S1766), respectively. Haplotype analysis narrowed the assignment to a 5.5-cM interval flanked by markers D10S604 and D10S568 on chromosome 10q11.2.


REFERENCES

  1. Liu, W., Wang, H., Zhao, S., Zhao, W., Bai, S., Zhao, Y., Xu, S., Wu, C., Huang, W., Chen, Z., Feng, G., He, L. The novel gene locus for agenesis of permanent teeth (He-Zhao deficiency) maps to chromosome 10q11.2. J. Dent. Res. 80: 1716-1720, 2001. [PubMed: 11669481] [Full Text: https://doi.org/10.1177/00220345010800080701]

  2. Wang, H., Zhao, S., Zhao, W., Feng, G., Jiang, S., Liu, W., Li, S., Xue, H., He, L. Congenital absence of permanent teeth in a six-generation Chinese kindred. Am. J. Med. Genet. 90: 193-198, 2000. [PubMed: 10678655]


Creation Date:
Carol A. Bocchini : 4/11/2007

Edit History:
carol : 07/22/2009
carol : 4/11/2007
carol : 4/11/2007