Evidence map›Paper›PMID 27585961›Full record

ArticleNature reviews. Endocrinology2017

Diagnosis and management of Silver-Russell syndrome: first international consensus statement.

Emma L Wakeling, Frédéric Brioude, Oluwakemi Lokulo-Sodipe, Susan M O'Connell, Jennifer Salem, Jet Bliek, Ana P M Canton, Krystyna H Chrzanowska, Justin H Davies, Renuka P Dias and 26 more

Open access · hybridAbstract readConsensus Statement
PubMed Publisher
In one paragraph

Article in Nature reviews. Endocrinology, 2017. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 251 papers, 3 of them syntheses that pooled it.

0numbers the graph read from it
0cells of the map it votes in
251citing papers in PubMed, 3 pooled it
35.7field-weighted citation impact, top 1% of its field
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

251 citing papers in PubMed, 3 syntheses or guidelines pooled it, 496 citations in OpenAlex.

  1. Pooled it
  2. Pooled it
  3. Guideline
  4. Article
  5. Article
  6. Review
  7. Review
  8. Article
  9. Article
  10. Review
  11. Article
  12. Article
  13. Article
  14. Review
  15. Review
  16. Article
  17. Article
  18. Article
  19. Article
  20. Article

191 more citing papers are in PubMed but not listed here.

4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

36 authors at 20 institutions in 14 countries.

Emma L WakelingNorth West Thames Regional Genetics Service, London North West Healthcare NHS Trust, Watford Road, Harrow HA1 3UJ, UK.
Frédéric BrioudeAP-HP, Hôpitaux Universitaires Paris Est (AP-HP) Hôpital des Enfants Armand Trousseau, Service d'Explorations Fonctionnelles Endocriniennes, 26 avenue du Dr Arnold Netter, 75012 Paris, France.
Oluwakemi Lokulo-SodipeHuman Development and Health, Faculty of Medicine, University of Southampton, Southampton SO17 1BJ, UK.
Susan M O'ConnellDepartment of Paediatrics and Child Health, Cork University Hospital, Wilton, Cork T12 DC4A, Ireland.
Jennifer SalemMAGIC Foundation, 6645 W. North Avenue, Oak Park, Illinois 60302, USA.
Jet BliekAcademic Medical Centre, Department of Clinical Genetics, Laboratory for Genome Diagnostics, Meibergdreef 15, 1105AZ Amsterdam, Netherlands.
Ana P M CantonUnidade de Endocrinologia Genetica, Laboratorio de Endocrinologia Celular e Molecular LIM/25, Disciplina de Endocrinologia da Faculdade de Medicina da Universidade de Sao Paulo, Av. Dr. Arnaldo, 455 5° andar sala 5340 (LIM25), 01246-000 São Paulo, SP, Brazil.
Krystyna H ChrzanowskaDepartment of Medical Genetics, The Children's Memorial Health Institute, Al. Dzieci Polskich 20, 04-730 Warsaw, Poland.
Justin H DaviesDepartment of Paediatric Endocrinology, University Hospital Southampton, Tremona Road, Southampton SO16 6YD, UK.
Renuka P DiasInstitutes of Metabolism and Systems Research, Vincent Drive, University of Birmingham, Birmingham B15 2TT, UK.
Béatrice DubernAP-HP, Hôpitaux Universitaires Paris Est (AP-HP) Hôpital des Enfants Armand Trousseau, Nutrition and Gastroenterology Department, 26 avenue du Dr Arnold Netter, 75012 Paris, France.
Miriam ElbrachtInsitute of Human Genetics, Technical University of Aachen, Pauwelsstr. 30, D-52074 Aachen, Germany.
Eloise GiabicaniAP-HP, Hôpitaux Universitaires Paris Est (AP-HP) Hôpital des Enfants Armand Trousseau, Service d'Explorations Fonctionnelles Endocriniennes, 26 avenue du Dr Arnold Netter, 75012 Paris, France.
Adda GrimbergPerelman School of Medicine, University of Pennsylvania, The Children's Hospital of Philadelphia, 3401 Civic Center Boulevard, Suite 11NW30, Philadelphia, Pennsylvania 19104, USA.
Karen GrønskovApplied Human Molecular Genetics, Kennedy Center, Department of Clinical Genetics, Copenhagen University Hospital, Rigshospitalet, Gl. Landevej 7, 2600 Glostrup, Copenhagen, Denmark.
Anita C S Hokken-KoelegaErasmus University Medical Center, Pediatrics, Subdivision of Endocrinology, Wytemaweg 80, 3015 CN, Rotterdam, Netherlands.
Alexander A JorgeUnidade de Endocrinologia Genetica, Laboratorio de Endocrinologia Celular e Molecular LIM/25, Disciplina de Endocrinologia da Faculdade de Medicina da Universidade de Sao Paulo, Av. Dr. Arnaldo, 455 5° andar sala 5340 (LIM25), 01246-000 São Paulo, SP, Brazil.
Masayo KagamiDepartment of Molecular Endocrinology, National Research Institute for Child Health and Development, 2-10-1 Ohkura, Setagayaku, Tokyo 157-8535, Japan.
Agnes LinglartAPHP, Department of Pediatric Endocrinology, Reference Center for Rare Disorders of the Mineral Metabolism and Plateforme d'Expertise Paris Sud Maladies Rares, Hospital Bicêtre Paris Sud, 78 Rue du Général Leclerc, 94270 Le Kremlin-Bicêtre, France.
Mohamad MaghnieIRCCS Istituto Giannina Gaslini, University of Genova, Via Gerolamo Gaslini 5, 16147 Genova, Italy.
Klaus MohnikeOtto-von-Guericke University, Department of Pediatrics, Leipziger Street 44, 39120 Magdeburg, Germany.
David MonkImprinting and Cancer Group, Cancer Epigenetic and Biology Program, Bellvitge Biomedical Research Institute, Gran via 199-203, Hospital Duran i Reynals, 08908, Barcelona, Spain.
Gudrun E MooreFetal Growth and Development Group, Institute of Child Health, University College London, 30 Guilford Street, London WC1N 1EH, UK.
Philip G MurrayCentre for Paediatrics and Child Health, Institute of Human Development, Royal Manchester Children's Hospital, Oxford Road, Manchester M13 9WL, UK.
Tsutomu OgataDepartment of Pediatrics, Hamamatsu University School of Medicine, 1-20-1 Handayama, Higashi-ku, Hamamatsu 431-3192, Japan.
Isabelle Oliver PetitPediatric Endocrinology, Genetic, Bone Disease &Gynecology Unit, Children's Hospital, TSA 70034, 31059 Toulouse, France.
Silvia RussoInstituto Auxologico Italiano, Cytogenetic and Molecular Genetic Laboratory, via Ariosto 13 20145 Milano, Italy.
Edith SaidDepartment of Anatomy &Cell Biology, Centre for Molecular Medicine &Biobanking, Faculty of Medicine &Surgery, University of Malta, Msida MSD2090, Malta.
Meropi ToumbaIASIS Hospital, 8 Voriou Ipirou, 8036, Paphos, Cyprus.
Zeynep TümerApplied Human Molecular Genetics, Kennedy Center, Department of Clinical Genetics, Copenhagen University Hospital, Rigshospitalet, Gl. Landevej 7, 2600 Glostrup, Copenhagen, Denmark.
Gerhard BinderUniversity Children's Hospital, Pediatric Endocrinology, Hoppe-Seyler-Strasse 1, 72070 Tuebingen, Germany.
Thomas EggermannInsitute of Human Genetics, Technical University of Aachen, Pauwelsstr. 30, D-52074 Aachen, Germany.
Madeleine D HarbisonMount Sinai School of Medicine, 5 E 98th Street #1192, New York, New York 10029, USA.
I Karen TempleHuman Development and Health, Faculty of Medicine, University of Southampton, Southampton SO17 1BJ, UK.
Deborah J G MackayHuman Development and Health, Faculty of Medicine, University of Southampton, Southampton SO17 1BJ, UK.
Irène NetchineAP-HP, Hôpitaux Universitaires Paris Est (AP-HP) Hôpital des Enfants Armand Trousseau, Service d'Explorations Fonctionnelles Endocriniennes, 26 avenue du Dr Arnold Netter, 75012 Paris, France.
Inserm · FRCopenhagen University Hospital · DKHospital das Clínicas da Faculdade de Medicina da Universidade de São Paulo · BRPrincess Anne Hospital · GBRWTH Aachen University · DEAmsterdam UMC Location University of Amsterdam · NLAssistance Publique – Hôpitaux de Paris · FRBirmingham Children's Hospital · GBChildren's Hospital of Philadelphia · USChildren's Memorial Health Institute · PLCork University Hospital · IEErasmus MC · NLHamamatsu University School of Medicine · JPIcahn School of Medicine at Mount Sinai · USInstitut d'Investigació Biomédica de Bellvitge · ESIRCCS Istituto Auxologico Italiano · ITIstituto Giannina Gaslini · ITLondon North West Healthcare NHS Trust · GBNational Center For Child Health and Development · JPOtto-von-Guericke University Magdeburg · DE

Funding

Department of Health PB-PG-1111-26003Medical Research Council G0801438Medical Research Council MR/J000329/1
6 · The paper itself

Abstract

This Consensus Statement summarizes recommendations for clinical diagnosis, investigation and management of patients with Silver-Russell syndrome (SRS), an imprinting disorder that causes prenatal and postnatal growth retardation. Considerable overlap exists between the care of individuals born small for gestational age and those with SRS. However, many specific management issues exist and evidence from controlled trials remains limited. SRS is primarily a clinical diagnosis; however, molecular testing enables confirmation of the clinical diagnosis and defines the subtype. A 'normal' result from a molecular test does not exclude the diagnosis of SRS. The management of children with SRS requires an experienced, multidisciplinary approach. Specific issues include growth failure, severe feeding difficulties, gastrointestinal problems, hypoglycaemia, body asymmetry, scoliosis, motor and speech delay and psychosocial challenges. An early emphasis on adequate nutritional status is important, with awareness that rapid postnatal weight gain might lead to subsequent increased risk of metabolic disorders. The benefits of treating patients with SRS with growth hormone include improved body composition, motor development and appetite, reduced risk of hypoglycaemia and increased height. Clinicians should be aware of possible premature adrenarche, fairly early and rapid central puberty and insulin resistance. Treatment with gonadotropin-releasing hormone analogues can delay progression of central puberty and preserve adult height potential. Long-term follow up is essential to determine the natural history and optimal management in adulthood.

Indexed as

Disease ManagementInternationalityGonadotropin-Releasing HormoneHuman Growth HormoneHumansSilver-Russell SyndromeGonadotropin-Releasing HormoneHuman Growth Hormone

Identifiers

PMID27585961
OpenAlexW2507689999

What OpenQuestion holds

Textmetadata
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 OpenQuestion graph.