Autosomal dominant cutis laxa and critical stenosis of the left main coronary artery in a 21-year-old female with an intronic mutation in the elastin gene.
Nikolaj Thure KrarupMarie HvidbjergTomás ZarembaMette SommerlundMartin Kirk ChristensenPublished in: American journal of medical genetics. Part A (2022)
Cutis laxa (CL) is a rare, inherited or acquired connective tissue disorder characterized by abnormal elastic fibers causing loose and redundant skin and a prematurely aged appearance. The syndrome has been associated with hypertension, but cases with early-onset ischemic heart disease have never been described. Here, we report a 21-year-old Danish female with activity-related shortness of breath and oedema of the lower extremities. The patient had a clinical diagnosis of autosomal dominant CL, but no genotyping had been performed prior to the index admission. The patient was diagnosed with ischemic heart disease, based on results of non-invasive cardiovascular imaging (including MRI and PET-CT) followed by invasive treatment of a critical left main coronary artery stenosis. Subsequent referral to genetic testing revealed a likely pathogenic intronic variant in ELN. This case report includes the clinical findings and relates these to known molecular mechanisms of CL.
Keyphrases
- case report
- coronary artery
- early onset
- pet ct
- pulmonary artery
- late onset
- magnetic resonance imaging
- genome wide
- blood pressure
- emergency department
- primary care
- positron emission tomography
- soft tissue
- contrast enhanced
- computed tomography
- copy number
- high throughput
- magnetic resonance
- photodynamic therapy
- replacement therapy
- mass spectrometry