ArticleThe Journal of clinical endocrinology and metabolism2015
Duration of Diabetes Predicts Aortic Pulse Wave Velocity and Vascular Events in Alström Syndrome.
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Article in The Journal of clinical endocrinology and metabolism, 2015. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 9 papers, 1 of them a synthesis that pooled it.
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9citing papers in PubMed, 1 pooled it
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Who cites it
9 citing papers in PubMed, 1 synthesis or guideline pooled it, 15 citations in OpenAlex.
- Consensus clinical management guidelines for Alström syndrome.Orphanet journal of rare diseases · 2020Pooled it
- Female Alms1-deficient mice develop echocardiographic features of adult but not infantile Alström syndrome cardiomyopathy.Disease models & mechanisms · 2024Article
- Alström syndrome: an ultra-rare monogenic disorder as a model for insulin resistance, type 2 diabetes mellitus and obesity.Endocrine · 2021Review
- Relative Adipose Tissue Failure in Alström Syndrome Drives Obesity-Induced Insulin Resistance.Diabetes · 2021Article
- Cardiovascular Implications in Idiopathic and Syndromic Obesity in Childhood: An Update.Frontiers in endocrinology · 2020Review
- Comprehensive Endocrine-Metabolic Evaluation of Patients With Alström Syndrome Compared With BMI-Matched Controls.The Journal of clinical endocrinology and metabolism · 2018Article
- Duration of Diabetes and Prediabetes During Adulthood and Subclinical Atherosclerosis and Cardiac Dysfunction in Middle Age: The CARDIA Study.Diabetes care · 2018Article
- Prediction of protective sensory loss, neuropathy and foot ulceration in type 2 diabetes.BMJ open diabetes research & care · 2016Article
- High quality, patient centred and coordinated care for Alstrom syndrome: a model of care for an ultra-rare disease.Orphanet journal of rare diseases · 2015Article
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Authors and funding
12 authors at 7 institutions in 3 countries.
Richard B PaiseyDiabetes Research Unit (R.B.P., J.S., C.C., S.B.), Horizon Centre, Torbay Hospital NHS Foundation Trust, Lawes Bridge, Torquay, Devon TQ2 7AA, United Kingdom; School of Clinical and Experimental Medicine (T.B.), College of Medicine and Dentistry, University of Birmingham, Edgbaston, Birmingham B15 2TT, United Kingdom; Leeds Children's Hospital (F.C.), Leeds, West Yorkshire LS1 3EX, United Kingdom; Internal Medicine 3 (P.M.), Department of Medicine, University Hospital of Padua, 35122 Padua, Italy; The Jackson Laboratory (J.D.M.), Bar Harbor, Maine 04609; University of Nottingham Medical School (C.P.), Nottingham NG7 2UH, United Kingdom; and Department of Cardiology (R.P.S., N.C.E.), and Department of Endocrinology (T.G.), University Hospitals Birmingham NHS Foundation Trust, Queen Elizabeth Hospital, Queen Elizabeth Medical Centre, Birmingham B15 2TH, United Kingdom.
Jamie SmithDiabetes Research Unit (R.B.P., J.S., C.C., S.B.), Horizon Centre, Torbay Hospital NHS Foundation Trust, Lawes Bridge, Torquay, Devon TQ2 7AA, United Kingdom; School of Clinical and Experimental Medicine (T.B.), College of Medicine and Dentistry, University of Birmingham, Edgbaston, Birmingham B15 2TT, United Kingdom; Leeds Children's Hospital (F.C.), Leeds, West Yorkshire LS1 3EX, United Kingdom; Internal Medicine 3 (P.M.), Department of Medicine, University Hospital of Padua, 35122 Padua, Italy; The Jackson Laboratory (J.D.M.), Bar Harbor, Maine 04609; University of Nottingham Medical School (C.P.), Nottingham NG7 2UH, United Kingdom; and Department of Cardiology (R.P.S., N.C.E.), and Department of Endocrinology (T.G.), University Hospitals Birmingham NHS Foundation Trust, Queen Elizabeth Hospital, Queen Elizabeth Medical Centre, Birmingham B15 2TH, United Kingdom.
Catherine CareyDiabetes Research Unit (R.B.P., J.S., C.C., S.B.), Horizon Centre, Torbay Hospital NHS Foundation Trust, Lawes Bridge, Torquay, Devon TQ2 7AA, United Kingdom; School of Clinical and Experimental Medicine (T.B.), College of Medicine and Dentistry, University of Birmingham, Edgbaston, Birmingham B15 2TT, United Kingdom; Leeds Children's Hospital (F.C.), Leeds, West Yorkshire LS1 3EX, United Kingdom; Internal Medicine 3 (P.M.), Department of Medicine, University Hospital of Padua, 35122 Padua, Italy; The Jackson Laboratory (J.D.M.), Bar Harbor, Maine 04609; University of Nottingham Medical School (C.P.), Nottingham NG7 2UH, United Kingdom; and Department of Cardiology (R.P.S., N.C.E.), and Department of Endocrinology (T.G.), University Hospitals Birmingham NHS Foundation Trust, Queen Elizabeth Hospital, Queen Elizabeth Medical Centre, Birmingham B15 2TH, United Kingdom.
Timothy BarrettDiabetes Research Unit (R.B.P., J.S., C.C., S.B.), Horizon Centre, Torbay Hospital NHS Foundation Trust, Lawes Bridge, Torquay, Devon TQ2 7AA, United Kingdom; School of Clinical and Experimental Medicine (T.B.), College of Medicine and Dentistry, University of Birmingham, Edgbaston, Birmingham B15 2TT, United Kingdom; Leeds Children's Hospital (F.C.), Leeds, West Yorkshire LS1 3EX, United Kingdom; Internal Medicine 3 (P.M.), Department of Medicine, University Hospital of Padua, 35122 Padua, Italy; The Jackson Laboratory (J.D.M.), Bar Harbor, Maine 04609; University of Nottingham Medical School (C.P.), Nottingham NG7 2UH, United Kingdom; and Department of Cardiology (R.P.S., N.C.E.), and Department of Endocrinology (T.G.), University Hospitals Birmingham NHS Foundation Trust, Queen Elizabeth Hospital, Queen Elizabeth Medical Centre, Birmingham B15 2TH, United Kingdom.
Fiona CampbellDiabetes Research Unit (R.B.P., J.S., C.C., S.B.), Horizon Centre, Torbay Hospital NHS Foundation Trust, Lawes Bridge, Torquay, Devon TQ2 7AA, United Kingdom; School of Clinical and Experimental Medicine (T.B.), College of Medicine and Dentistry, University of Birmingham, Edgbaston, Birmingham B15 2TT, United Kingdom; Leeds Children's Hospital (F.C.), Leeds, West Yorkshire LS1 3EX, United Kingdom; Internal Medicine 3 (P.M.), Department of Medicine, University Hospital of Padua, 35122 Padua, Italy; The Jackson Laboratory (J.D.M.), Bar Harbor, Maine 04609; University of Nottingham Medical School (C.P.), Nottingham NG7 2UH, United Kingdom; and Department of Cardiology (R.P.S., N.C.E.), and Department of Endocrinology (T.G.), University Hospitals Birmingham NHS Foundation Trust, Queen Elizabeth Hospital, Queen Elizabeth Medical Centre, Birmingham B15 2TH, United Kingdom.
Pietro MaffeiDiabetes Research Unit (R.B.P., J.S., C.C., S.B.), Horizon Centre, Torbay Hospital NHS Foundation Trust, Lawes Bridge, Torquay, Devon TQ2 7AA, United Kingdom; School of Clinical and Experimental Medicine (T.B.), College of Medicine and Dentistry, University of Birmingham, Edgbaston, Birmingham B15 2TT, United Kingdom; Leeds Children's Hospital (F.C.), Leeds, West Yorkshire LS1 3EX, United Kingdom; Internal Medicine 3 (P.M.), Department of Medicine, University Hospital of Padua, 35122 Padua, Italy; The Jackson Laboratory (J.D.M.), Bar Harbor, Maine 04609; University of Nottingham Medical School (C.P.), Nottingham NG7 2UH, United Kingdom; and Department of Cardiology (R.P.S., N.C.E.), and Department of Endocrinology (T.G.), University Hospitals Birmingham NHS Foundation Trust, Queen Elizabeth Hospital, Queen Elizabeth Medical Centre, Birmingham B15 2TH, United Kingdom.
Jan D MarshallDiabetes Research Unit (R.B.P., J.S., C.C., S.B.), Horizon Centre, Torbay Hospital NHS Foundation Trust, Lawes Bridge, Torquay, Devon TQ2 7AA, United Kingdom; School of Clinical and Experimental Medicine (T.B.), College of Medicine and Dentistry, University of Birmingham, Edgbaston, Birmingham B15 2TT, United Kingdom; Leeds Children's Hospital (F.C.), Leeds, West Yorkshire LS1 3EX, United Kingdom; Internal Medicine 3 (P.M.), Department of Medicine, University Hospital of Padua, 35122 Padua, Italy; The Jackson Laboratory (J.D.M.), Bar Harbor, Maine 04609; University of Nottingham Medical School (C.P.), Nottingham NG7 2UH, United Kingdom; and Department of Cardiology (R.P.S., N.C.E.), and Department of Endocrinology (T.G.), University Hospitals Birmingham NHS Foundation Trust, Queen Elizabeth Hospital, Queen Elizabeth Medical Centre, Birmingham B15 2TH, United Kingdom.
Christopher PaiseyDiabetes Research Unit (R.B.P., J.S., C.C., S.B.), Horizon Centre, Torbay Hospital NHS Foundation Trust, Lawes Bridge, Torquay, Devon TQ2 7AA, United Kingdom; School of Clinical and Experimental Medicine (T.B.), College of Medicine and Dentistry, University of Birmingham, Edgbaston, Birmingham B15 2TT, United Kingdom; Leeds Children's Hospital (F.C.), Leeds, West Yorkshire LS1 3EX, United Kingdom; Internal Medicine 3 (P.M.), Department of Medicine, University Hospital of Padua, 35122 Padua, Italy; The Jackson Laboratory (J.D.M.), Bar Harbor, Maine 04609; University of Nottingham Medical School (C.P.), Nottingham NG7 2UH, United Kingdom; and Department of Cardiology (R.P.S., N.C.E.), and Department of Endocrinology (T.G.), University Hospitals Birmingham NHS Foundation Trust, Queen Elizabeth Hospital, Queen Elizabeth Medical Centre, Birmingham B15 2TH, United Kingdom.
Richard P SteedsDiabetes Research Unit (R.B.P., J.S., C.C., S.B.), Horizon Centre, Torbay Hospital NHS Foundation Trust, Lawes Bridge, Torquay, Devon TQ2 7AA, United Kingdom; School of Clinical and Experimental Medicine (T.B.), College of Medicine and Dentistry, University of Birmingham, Edgbaston, Birmingham B15 2TT, United Kingdom; Leeds Children's Hospital (F.C.), Leeds, West Yorkshire LS1 3EX, United Kingdom; Internal Medicine 3 (P.M.), Department of Medicine, University Hospital of Padua, 35122 Padua, Italy; The Jackson Laboratory (J.D.M.), Bar Harbor, Maine 04609; University of Nottingham Medical School (C.P.), Nottingham NG7 2UH, United Kingdom; and Department of Cardiology (R.P.S., N.C.E.), and Department of Endocrinology (T.G.), University Hospitals Birmingham NHS Foundation Trust, Queen Elizabeth Hospital, Queen Elizabeth Medical Centre, Birmingham B15 2TH, United Kingdom.
Nicola C EdwardsDiabetes Research Unit (R.B.P., J.S., C.C., S.B.), Horizon Centre, Torbay Hospital NHS Foundation Trust, Lawes Bridge, Torquay, Devon TQ2 7AA, United Kingdom; School of Clinical and Experimental Medicine (T.B.), College of Medicine and Dentistry, University of Birmingham, Edgbaston, Birmingham B15 2TT, United Kingdom; Leeds Children's Hospital (F.C.), Leeds, West Yorkshire LS1 3EX, United Kingdom; Internal Medicine 3 (P.M.), Department of Medicine, University Hospital of Padua, 35122 Padua, Italy; The Jackson Laboratory (J.D.M.), Bar Harbor, Maine 04609; University of Nottingham Medical School (C.P.), Nottingham NG7 2UH, United Kingdom; and Department of Cardiology (R.P.S., N.C.E.), and Department of Endocrinology (T.G.), University Hospitals Birmingham NHS Foundation Trust, Queen Elizabeth Hospital, Queen Elizabeth Medical Centre, Birmingham B15 2TH, United Kingdom.
Susan BunceDiabetes Research Unit (R.B.P., J.S., C.C., S.B.), Horizon Centre, Torbay Hospital NHS Foundation Trust, Lawes Bridge, Torquay, Devon TQ2 7AA, United Kingdom; School of Clinical and Experimental Medicine (T.B.), College of Medicine and Dentistry, University of Birmingham, Edgbaston, Birmingham B15 2TT, United Kingdom; Leeds Children's Hospital (F.C.), Leeds, West Yorkshire LS1 3EX, United Kingdom; Internal Medicine 3 (P.M.), Department of Medicine, University Hospital of Padua, 35122 Padua, Italy; The Jackson Laboratory (J.D.M.), Bar Harbor, Maine 04609; University of Nottingham Medical School (C.P.), Nottingham NG7 2UH, United Kingdom; and Department of Cardiology (R.P.S., N.C.E.), and Department of Endocrinology (T.G.), University Hospitals Birmingham NHS Foundation Trust, Queen Elizabeth Hospital, Queen Elizabeth Medical Centre, Birmingham B15 2TH, United Kingdom.
Tarekegn GeberhiwotDiabetes Research Unit (R.B.P., J.S., C.C., S.B.), Horizon Centre, Torbay Hospital NHS Foundation Trust, Lawes Bridge, Torquay, Devon TQ2 7AA, United Kingdom; School of Clinical and Experimental Medicine (T.B.), College of Medicine and Dentistry, University of Birmingham, Edgbaston, Birmingham B15 2TT, United Kingdom; Leeds Children's Hospital (F.C.), Leeds, West Yorkshire LS1 3EX, United Kingdom; Internal Medicine 3 (P.M.), Department of Medicine, University Hospital of Padua, 35122 Padua, Italy; The Jackson Laboratory (J.D.M.), Bar Harbor, Maine 04609; University of Nottingham Medical School (C.P.), Nottingham NG7 2UH, United Kingdom; and Department of Cardiology (R.P.S., N.C.E.), and Department of Endocrinology (T.G.), University Hospitals Birmingham NHS Foundation Trust, Queen Elizabeth Hospital, Queen Elizabeth Medical Centre, Birmingham B15 2TH, United Kingdom.
Torbay Hospital · GBJackson Laboratory · USLeeds Teaching Hospitals NHS Trust · GBQueen Elizabeth Hospital · GBUniversity of Birmingham · GBUniversity of Nottingham · GBUniversity of Padua · IT
Funding
MOLECULAR GENETIC CHARACTERIZATION OF ALSTROM SYNDROMER01HD036878 · NICHD · JACKSON LABORATORY · PI NAGGERT, JUERGEN K. · 1998 to 2011
$4.2MNICHD NIH HHS HD036878NICHD NIH HHS R01 HD036878Wellcome Trust
Abstract
contextAlström syndrome is characterized by increased risk of cardiovascular disease from childhood.
objectiveTo explore the association between risk factors for cardiovascular disease, aortic pulse wave velocity, and vascular events in Alström syndrome.
designCross-sectional analyses with 5-year follow-up.
settingThe UK NHS nationally commissioned specialist clinics for Alström syndrome. PATIENTS: Thirty-one Alström patients undertook vascular risk assessment, cardiac studies, and aortic pulse wave velocity measurement. Subsequent clinical outcomes were recorded.
interventionsInsulin resistance was treated with lifestyle intervention and metformin, and diabetes with the addition of glitazones, glucagon-like peptide 1 agonists, and/or insulin. Thyroid and T deficiencies were corrected. Dyslipidemia was treated with statins and nicotinic acid derivatives. Cardiomyopathy was treated with standard therapy as required.
main outcome measuresThe associations of age, gender, and risk factors for cardiovascular disease with aortic pulse wave velocity were assessed and correlated with the effects of reduction in left ventricular function. Vascular events were monitored for 5 years.
resultsAortic pulse wave velocity was positively associated with the duration of diabetes (P = .001) and inversely with left ventricular ejection fraction (P = .036). Five of the cohort with cardiovascular events had higher aortic pulse wave velocity (P = .0247), and all had long duration of diabetes.
conclusionsDuration of diabetes predicted aortic pulse wave velocity in Alström syndrome, which in turn predicted cardiovascular events. This offers hope of secondary prevention because type 2 diabetes can be delayed or reversed by lifestyle interventions.
Indexed as
Pulse Wave AnalysisAdolescentAdultAlstrom SyndromeCardiovascular DiseasesChildCross-Sectional StudiesDiabetes Mellitus, Type 2FemaleFollow-Up StudiesHumansMalePrognosisTime FactorsYoung Adult
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