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1.
Clin Genet ; 104(6): 713-715, 2023 12.
Article in English | MEDLINE | ID: mdl-37612261

ABSTRACT

This study reports variants in BBS1 and BBS7 in patients with Bardet-Biedl syndrome from the Canadian Maritime provinces. The BBS1 variant NM_024649.5:c.1169T>G was identified as a recurrent variant in Prince Edward Island.


Subject(s)
Bardet-Biedl Syndrome , Microtubule-Associated Proteins , Humans , Canada , Microtubule-Associated Proteins/genetics , Mutation , Prince Edward Island
2.
Am J Med Genet A ; 191(2): 554-558, 2023 02.
Article in English | MEDLINE | ID: mdl-36308391

ABSTRACT

Congenital heart defect (CHD) is a birth defect that affects the structure of the heart. Although CHD is often multifactorial, it can also be inherited as part of a Mendelian disorder such as in congenital heart defect and ectodermal dysplasia (CHDED). This disorder is caused by de novo variants in PRKD1. Here, we describe a patient with a novel de novo variant of PRKD1 with phenotypic features consistent with CHDED. Previously unreported features were noted including high intracranial pressure (ICP), partial anomalous pulmonary venous return (PAPVR), and bifid uvula. We suggest that these features may be associated with CHDED.


Subject(s)
Cleft Palate , Ectodermal Dysplasia , Heart Defects, Congenital , Humans , Intracranial Pressure , Heart Defects, Congenital/complications , Heart Defects, Congenital/diagnosis , Heart Defects, Congenital/genetics , Ectodermal Dysplasia/complications , Ectodermal Dysplasia/diagnosis , Ectodermal Dysplasia/genetics , Phenotype
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