Evidence map›Paper›PMID 37214872›Full record

ArticlemedRxiv : the preprint server for health sciences2023

Systematic Review of Treatment of Beta-Cell Monogenic Diabetes.

Rochelle N Naylor, Kashyap A Patel, Jarno L T Kettunen, Jonna M E Männistö, Julie Støy, Jacques Beltrand, Michel Polak, ADA/EASD PMDI, Tina Vilsbøll, Siri A W Greeley and 2 more

Open access · greenAbstract readPreprint
In one paragraph

Article in medRxiv : the preprint server for health sciences, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.

0numbers the graph read from it
0cells of the map it votes in
0citing papers in PubMed
–field-weighted citation impact
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

0 citing papers in PubMed, 9 citations in OpenAlex.

No citing paper in PubMed yet.

4 · The record

Corrections and comments

5 · Who and what money

Authors and funding

12 authors at 7 institutions in 6 countries.

Rochelle N NaylorDepartments of Pediatrics and Medicine, University of Chicago, Chicago, Illinois, USA.
Kashyap A PatelUniversity of Exeter Medical School, Department of Clinical and Biomedical Sciences, Exeter, Devon, UK.
Jarno L T KettunenHelsinki University Hospital, Abdominal Centre/Endocrinology, Helsinki, Finland; Folkhalsan Research Center, Helsinki, Finland; Institute for Molecular Medicine Finland FIMM, University of Helsinki, Helsinki, Finland.
Jonna M E MännistöDepartments of Pediatrics and Clinical Genetics, Kuopio University Hospital, Kuopio, Finland; Department of Medicine, University of Eastern Finland, Kuopio, Finland.
Julie StøySteno diabetes center Aarhus, Aarhus university hospital, Aarhus, Denmark.
Jacques BeltrandAPHP Centre Hôpital Necker Enfants Malades Université Paris Cité, Paris France; Inserm U1016 Institut Cochin Paris France.
Michel PolakDepartment of pediatric endocrinology gynecology and diabetology, Hôpital Universitaire Necker Enfants Malades, IMAGINE institute, INSERM U1016, Paris, France; Université Paris Cité, Paris, France.
ADA/EASD PMDI
Tina VilsbøllDepartment of Clinical Medicine, University of Copenhagen.
Siri A W GreeleyDepartments of Pediatrics and Medicine, University of Chicago, Chicago, Illinois, USA.
Andrew T HattersleyUniversity of Exeter Medical School, Department of Clinical and Biomedical Sciences, Exeter, Devon, UK.
Tiinamaija TuomiLund University Diabetes Center, Malmo, Sweden.
Hôpital Necker-Enfants Malades · FRLund University · SEUniversity of Copenhagen · DKUniversity of Exeter · GBUniversity of Chicago · USUniversity of Eastern Finland · FIUniversity of Helsinki · FI

Funding

RADIANT Clinic and Data Coordinating CenterU54DK118612 · NIDDK · UNIVERSITY OF CHICAGO · PI Louis H. Philipson, Miriam Sargon Udler · 2018 to 2026
$21.9M
Monogenic Diabetes: Next Generation Diagnosis, Treatment and ComplicationsR01DK104942 · NIDDK · UNIVERSITY OF CHICAGO · PI GREELEY, SIRI ATMA W., PHILIPSON, LOUIS H. · 2016 to 2024
$4.7M
NIDDK NIH HHS R01 DK104942NIDDK NIH HHS U54 DK118612Wellcome Trust
6 · The paper itself

Abstract

Background: Beta-cell monogenic forms of diabetes are the area of diabetes care with the strongest support for precision medicine. We reviewed treatment of hyperglycemia in GCK-related hyperglycemia, HNF1A-HNF4A- and HNF1B-diabetes, Mitochondrial diabetes (MD) due to m.3243A>G variant, 6q24-transient neonatal diabetes (TND) and SLC19A2-diabetes. Methods: Systematic reviews with data from PubMed, MEDLINE and Embase were performed for the different subtypes. Individual and group level data was extracted for glycemic outcomes in individuals with genetically confirmed monogenic diabetes. Results: 147 studies met inclusion criteria with only six experimental studies and the rest being single case reports or cohort studies. Most studies had moderate or serious risk of bias.For GCK-related hyperglycemia, six studies (N=35) showed no deterioration in HbA1c on discontinuing glucose lowering therapy. A randomized trial (n=18 per group) showed that sulfonylureas (SU) were more effective in HNF1A-diabetes than in type 2 diabetes, and cohort and case studies supported SU effectiveness in lowering HbA1c. Two crossover trials (n=15 and n=16) suggested glinides and GLP-1 receptor agonists might be used in place of SU. Evidence for HNF4A-diabetes was limited. While some patients with HNF1B-diabetes (n=301) and MD (n=250) were treated with oral agents, most were on insulin. There was some support for the use of oral agents after relapse in 6q24-TND, and for thiamine improving glycemic control and reducing insulin requirement in SLC19A2-diabetes (less than half achieved insulin-independency). Conclusion: There is limited evidence to guide the treatment in monogenic diabetes with most studies being non-randomized and small. The data supports: no treatment in GCK-related hyperglycemia; SU for HNF1A-diabetes. Further evidence is needed to examine the optimum treatment in monogenic subtypes.

Indexed as

6q24GCKGeneticsHNF1AHNF1BHNF4Am.3243A>GMitochondrial diabetesMODYMonogenic DiabetesNeonatal DiabetesPrecision MedicineSLC19A2Syndromic Diabetes

Identifiers

PMID37214872
PMCPMC10197799
OpenAlexW4376270912

What Socratic holds

Textmetadata
LicenceCC BY-NC-ND
Read underepoch 390

Registered trials

None linked

Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the Socratic graph.