Evidence map›Paper›PMID 26671997›Full record

ArticleEndocrine-related cancer2016

GHRH excess and blockade in X-LAG syndrome.

Adrian F Daly, Philippe A Lysy, Céline Desfilles, Liliya Rostomyan, Amira Mohamed, Jean-Hubert Caberg, Veronique Raverot, Emilie Castermans, Etienne Marbaix, Dominique Maiter and 8 more

Open access · bronzeAbstract readCase Reports
In one paragraph

Article in Endocrine-related cancer, 2016. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 23 papers, 1 of them a synthesis that pooled it.

0numbers the graph read from it
0cells of the map it votes in
23citing papers in PubMed, 1 pooled it
3.5field-weighted citation impact, top 7% 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

23 citing papers in PubMed, 1 synthesis or guideline pooled it, 70 citations in OpenAlex.

  1. Guideline
  2. Genetics of familial acromegaly and pituitary gigantism.The Journal of clinical endocrinology and metabolism · 2026
    Review
  3. Article
  4. Review
  5. Review
  6. Article
  7. Pituitary Tumorigenesis-Implications for Management.Medicina (Kaunas, Lithuania) · 2023
    Review
  8. Review
  9. Pasireotide-a novel somatostatin receptor ligand after 20 years of use.Reviews in endocrine & metabolic disorders · 2022
    Review
  10. Article
  11. Genetics of Acromegaly and Gigantism.Journal of clinical medicine · 2021
    Review
  12. Review
  13. Review
  14. Potential markers of disease behavior in acromegaly and gigantism.Expert review of endocrinology & metabolism · 2020
    Review
  15. Review
  16. The causes and consequences of pituitary gigantism.Nature reviews. Endocrinology · 2018
    Review
  17. Article
  18. Article
  19. Article
  20. Article
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

18 authors at 7 institutions in 5 countries.

Adrian F DalyDepartment of EndocrinologyCentre Hospitalier Universitaire de Liège, University of Liège, Domaine Universitaire du Sart-Tilman, 4000 Liège, BelgiumDepartment of Human GeneticsCentre Hospitalier Universitaire de Liège, University of Liège, Domaine Universitaire du Sart-Tilman, 4000 Liège, BelgiumPediatric Endocrinology UnitUniversité Catholique de Louvain, Avenue Hippocrate 10, 1200 Brussels, BelgiumCliniques Universitaires Saint Luc and Department of PathologyUniversité Catholique de Louvain, Avenue Hippocrate 10, 1200 Brussels, BelgiumLaboratory of Molecular BiologyAPHM, Hôpital la Conception, 13385, Marseille, FranceAix Marseille UniversitéCRNS, CRN2M-UMR 7286, 13344, Marseille, FranceSection on Endocrinology and GeneticsProgram on Developmental Endocrinology and Genetics (PDEGEN) and Pediatric Endocrinology Inter-institute Training Program, Eunice Kennedy Shriver National Institute of Child Health and Human Development (NICHD), National Institutes of Health (NIH), Bethesda, Maryland 20892, USALaboratoire d'Hormonologie - CBPEGroupement Hospitalier Est, Hospices Civils de Lyon, 59 bd Pinel - 69677 Bron Cedex, FranceUniversité Catholique de LouvainPole d'endocrinologie, diabete et nutrition (EDIN), Brussels, BelgiumDepartment of NeurosurgeryUniversité Catholique de Louvain, Brussels, Belgium albert.beckers@chu.ulg.ac.be.
Philippe A LysyDepartment of EndocrinologyCentre Hospitalier Universitaire de Liège, University of Liège, Domaine Universitaire du Sart-Tilman, 4000 Liège, BelgiumDepartment of Human GeneticsCentre Hospitalier Universitaire de Liège, University of Liège, Domaine Universitaire du Sart-Tilman, 4000 Liège, BelgiumPediatric Endocrinology UnitUniversité Catholique de Louvain, Avenue Hippocrate 10, 1200 Brussels, BelgiumCliniques Universitaires Saint Luc and Department of PathologyUniversité Catholique de Louvain, Avenue Hippocrate 10, 1200 Brussels, BelgiumLaboratory of Molecular BiologyAPHM, Hôpital la Conception, 13385, Marseille, FranceAix Marseille UniversitéCRNS, CRN2M-UMR 7286, 13344, Marseille, FranceSection on Endocrinology and GeneticsProgram on Developmental Endocrinology and Genetics (PDEGEN) and Pediatric Endocrinology Inter-institute Training Program, Eunice Kennedy Shriver National Institute of Child Health and Human Development (NICHD), National Institutes of Health (NIH), Bethesda, Maryland 20892, USALaboratoire d'Hormonologie - CBPEGroupement Hospitalier Est, Hospices Civils de Lyon, 59 bd Pinel - 69677 Bron Cedex, FranceUniversité Catholique de LouvainPole d'endocrinologie, diabete et nutrition (EDIN), Brussels, BelgiumDepartment of NeurosurgeryUniversité Catholique de Louvain, Brussels, Belgium.
Céline DesfillesDepartment of EndocrinologyCentre Hospitalier Universitaire de Liège, University of Liège, Domaine Universitaire du Sart-Tilman, 4000 Liège, BelgiumDepartment of Human GeneticsCentre Hospitalier Universitaire de Liège, University of Liège, Domaine Universitaire du Sart-Tilman, 4000 Liège, BelgiumPediatric Endocrinology UnitUniversité Catholique de Louvain, Avenue Hippocrate 10, 1200 Brussels, BelgiumCliniques Universitaires Saint Luc and Department of PathologyUniversité Catholique de Louvain, Avenue Hippocrate 10, 1200 Brussels, BelgiumLaboratory of Molecular BiologyAPHM, Hôpital la Conception, 13385, Marseille, FranceAix Marseille UniversitéCRNS, CRN2M-UMR 7286, 13344, Marseille, FranceSection on Endocrinology and GeneticsProgram on Developmental Endocrinology and Genetics (PDEGEN) and Pediatric Endocrinology Inter-institute Training Program, Eunice Kennedy Shriver National Institute of Child Health and Human Development (NICHD), National Institutes of Health (NIH), Bethesda, Maryland 20892, USALaboratoire d'Hormonologie - CBPEGroupement Hospitalier Est, Hospices Civils de Lyon, 59 bd Pinel - 69677 Bron Cedex, FranceUniversité Catholique de LouvainPole d'endocrinologie, diabete et nutrition (EDIN), Brussels, BelgiumDepartment of NeurosurgeryUniversité Catholique de Louvain, Brussels, Belgium Department of EndocrinologyCentre Hospitalier Universitaire de Liège, University of Liège, Domaine Universitaire du Sart-Tilman, 4000 Liège, BelgiumDepartment of Human GeneticsCentre Hospitalier Universitaire de Liège, University of Liège, Domaine Universitaire du Sart-Tilman, 4000 Liège, BelgiumPediatric Endocrinology UnitUniversité Catholique de Louvain, Avenue Hippocrate 10, 1200 Brussels, BelgiumCliniques Universitaires Saint Luc and Department of PathologyUniversité Catholique de Louvain, Avenue Hippocrate 10, 1200 Brussels, BelgiumLaboratory of Molecular BiologyAPHM, Hôpital la Conception, 13385, Marseille, FranceAix Marseille UniversitéCRNS, CRN2M-
Liliya RostomyanDepartment of EndocrinologyCentre Hospitalier Universitaire de Liège, University of Liège, Domaine Universitaire du Sart-Tilman, 4000 Liège, BelgiumDepartment of Human GeneticsCentre Hospitalier Universitaire de Liège, University of Liège, Domaine Universitaire du Sart-Tilman, 4000 Liège, BelgiumPediatric Endocrinology UnitUniversité Catholique de Louvain, Avenue Hippocrate 10, 1200 Brussels, BelgiumCliniques Universitaires Saint Luc and Department of PathologyUniversité Catholique de Louvain, Avenue Hippocrate 10, 1200 Brussels, BelgiumLaboratory of Molecular BiologyAPHM, Hôpital la Conception, 13385, Marseille, FranceAix Marseille UniversitéCRNS, CRN2M-UMR 7286, 13344, Marseille, FranceSection on Endocrinology and GeneticsProgram on Developmental Endocrinology and Genetics (PDEGEN) and Pediatric Endocrinology Inter-institute Training Program, Eunice Kennedy Shriver National Institute of Child Health and Human Development (NICHD), National Institutes of Health (NIH), Bethesda, Maryland 20892, USALaboratoire d'Hormonologie - CBPEGroupement Hospitalier Est, Hospices Civils de Lyon, 59 bd Pinel - 69677 Bron Cedex, FranceUniversité Catholique de LouvainPole d'endocrinologie, diabete et nutrition (EDIN), Brussels, BelgiumDepartment of NeurosurgeryUniversité Catholique de Louvain, Brussels, Belgium.
Amira MohamedDepartment of EndocrinologyCentre Hospitalier Universitaire de Liège, University of Liège, Domaine Universitaire du Sart-Tilman, 4000 Liège, BelgiumDepartment of Human GeneticsCentre Hospitalier Universitaire de Liège, University of Liège, Domaine Universitaire du Sart-Tilman, 4000 Liège, BelgiumPediatric Endocrinology UnitUniversité Catholique de Louvain, Avenue Hippocrate 10, 1200 Brussels, BelgiumCliniques Universitaires Saint Luc and Department of PathologyUniversité Catholique de Louvain, Avenue Hippocrate 10, 1200 Brussels, BelgiumLaboratory of Molecular BiologyAPHM, Hôpital la Conception, 13385, Marseille, FranceAix Marseille UniversitéCRNS, CRN2M-UMR 7286, 13344, Marseille, FranceSection on Endocrinology and GeneticsProgram on Developmental Endocrinology and Genetics (PDEGEN) and Pediatric Endocrinology Inter-institute Training Program, Eunice Kennedy Shriver National Institute of Child Health and Human Development (NICHD), National Institutes of Health (NIH), Bethesda, Maryland 20892, USALaboratoire d'Hormonologie - CBPEGroupement Hospitalier Est, Hospices Civils de Lyon, 59 bd Pinel - 69677 Bron Cedex, FranceUniversité Catholique de LouvainPole d'endocrinologie, diabete et nutrition (EDIN), Brussels, BelgiumDepartment of NeurosurgeryUniversité Catholique de Louvain, Brussels, Belgium Department of EndocrinologyCentre Hospitalier Universitaire de Liège, University of Liège, Domaine Universitaire du Sart-Tilman, 4000 Liège, BelgiumDepartment of Human GeneticsCentre Hospitalier Universitaire de Liège, University of Liège, Domaine Universitaire du Sart-Tilman, 4000 Liège, BelgiumPediatric Endocrinology UnitUniversité Catholique de Louvain, Avenue Hippocrate 10, 1200 Brussels, BelgiumCliniques Universitaires Saint Luc and Department of PathologyUniversité Catholique de Louvain, Avenue Hippocrate 10, 1200 Brussels, BelgiumLaboratory of Molecular BiologyAPHM, Hôpital la Conception, 13385, Marseille, FranceAix Marseille UniversitéCRNS, CRN2M-
Jean-Hubert CabergDepartment of EndocrinologyCentre Hospitalier Universitaire de Liège, University of Liège, Domaine Universitaire du Sart-Tilman, 4000 Liège, BelgiumDepartment of Human GeneticsCentre Hospitalier Universitaire de Liège, University of Liège, Domaine Universitaire du Sart-Tilman, 4000 Liège, BelgiumPediatric Endocrinology UnitUniversité Catholique de Louvain, Avenue Hippocrate 10, 1200 Brussels, BelgiumCliniques Universitaires Saint Luc and Department of PathologyUniversité Catholique de Louvain, Avenue Hippocrate 10, 1200 Brussels, BelgiumLaboratory of Molecular BiologyAPHM, Hôpital la Conception, 13385, Marseille, FranceAix Marseille UniversitéCRNS, CRN2M-UMR 7286, 13344, Marseille, FranceSection on Endocrinology and GeneticsProgram on Developmental Endocrinology and Genetics (PDEGEN) and Pediatric Endocrinology Inter-institute Training Program, Eunice Kennedy Shriver National Institute of Child Health and Human Development (NICHD), National Institutes of Health (NIH), Bethesda, Maryland 20892, USALaboratoire d'Hormonologie - CBPEGroupement Hospitalier Est, Hospices Civils de Lyon, 59 bd Pinel - 69677 Bron Cedex, FranceUniversité Catholique de LouvainPole d'endocrinologie, diabete et nutrition (EDIN), Brussels, BelgiumDepartment of NeurosurgeryUniversité Catholique de Louvain, Brussels, Belgium.
Veronique RaverotDepartment of EndocrinologyCentre Hospitalier Universitaire de Liège, University of Liège, Domaine Universitaire du Sart-Tilman, 4000 Liège, BelgiumDepartment of Human GeneticsCentre Hospitalier Universitaire de Liège, University of Liège, Domaine Universitaire du Sart-Tilman, 4000 Liège, BelgiumPediatric Endocrinology UnitUniversité Catholique de Louvain, Avenue Hippocrate 10, 1200 Brussels, BelgiumCliniques Universitaires Saint Luc and Department of PathologyUniversité Catholique de Louvain, Avenue Hippocrate 10, 1200 Brussels, BelgiumLaboratory of Molecular BiologyAPHM, Hôpital la Conception, 13385, Marseille, FranceAix Marseille UniversitéCRNS, CRN2M-UMR 7286, 13344, Marseille, FranceSection on Endocrinology and GeneticsProgram on Developmental Endocrinology and Genetics (PDEGEN) and Pediatric Endocrinology Inter-institute Training Program, Eunice Kennedy Shriver National Institute of Child Health and Human Development (NICHD), National Institutes of Health (NIH), Bethesda, Maryland 20892, USALaboratoire d'Hormonologie - CBPEGroupement Hospitalier Est, Hospices Civils de Lyon, 59 bd Pinel - 69677 Bron Cedex, FranceUniversité Catholique de LouvainPole d'endocrinologie, diabete et nutrition (EDIN), Brussels, BelgiumDepartment of NeurosurgeryUniversité Catholique de Louvain, Brussels, Belgium.
Emilie CastermansDepartment of EndocrinologyCentre Hospitalier Universitaire de Liège, University of Liège, Domaine Universitaire du Sart-Tilman, 4000 Liège, BelgiumDepartment of Human GeneticsCentre Hospitalier Universitaire de Liège, University of Liège, Domaine Universitaire du Sart-Tilman, 4000 Liège, BelgiumPediatric Endocrinology UnitUniversité Catholique de Louvain, Avenue Hippocrate 10, 1200 Brussels, BelgiumCliniques Universitaires Saint Luc and Department of PathologyUniversité Catholique de Louvain, Avenue Hippocrate 10, 1200 Brussels, BelgiumLaboratory of Molecular BiologyAPHM, Hôpital la Conception, 13385, Marseille, FranceAix Marseille UniversitéCRNS, CRN2M-UMR 7286, 13344, Marseille, FranceSection on Endocrinology and GeneticsProgram on Developmental Endocrinology and Genetics (PDEGEN) and Pediatric Endocrinology Inter-institute Training Program, Eunice Kennedy Shriver National Institute of Child Health and Human Development (NICHD), National Institutes of Health (NIH), Bethesda, Maryland 20892, USALaboratoire d'Hormonologie - CBPEGroupement Hospitalier Est, Hospices Civils de Lyon, 59 bd Pinel - 69677 Bron Cedex, FranceUniversité Catholique de LouvainPole d'endocrinologie, diabete et nutrition (EDIN), Brussels, BelgiumDepartment of NeurosurgeryUniversité Catholique de Louvain, Brussels, Belgium.
Etienne MarbaixDepartment of EndocrinologyCentre Hospitalier Universitaire de Liège, University of Liège, Domaine Universitaire du Sart-Tilman, 4000 Liège, BelgiumDepartment of Human GeneticsCentre Hospitalier Universitaire de Liège, University of Liège, Domaine Universitaire du Sart-Tilman, 4000 Liège, BelgiumPediatric Endocrinology UnitUniversité Catholique de Louvain, Avenue Hippocrate 10, 1200 Brussels, BelgiumCliniques Universitaires Saint Luc and Department of PathologyUniversité Catholique de Louvain, Avenue Hippocrate 10, 1200 Brussels, BelgiumLaboratory of Molecular BiologyAPHM, Hôpital la Conception, 13385, Marseille, FranceAix Marseille UniversitéCRNS, CRN2M-UMR 7286, 13344, Marseille, FranceSection on Endocrinology and GeneticsProgram on Developmental Endocrinology and Genetics (PDEGEN) and Pediatric Endocrinology Inter-institute Training Program, Eunice Kennedy Shriver National Institute of Child Health and Human Development (NICHD), National Institutes of Health (NIH), Bethesda, Maryland 20892, USALaboratoire d'Hormonologie - CBPEGroupement Hospitalier Est, Hospices Civils de Lyon, 59 bd Pinel - 69677 Bron Cedex, FranceUniversité Catholique de LouvainPole d'endocrinologie, diabete et nutrition (EDIN), Brussels, BelgiumDepartment of NeurosurgeryUniversité Catholique de Louvain, Brussels, Belgium.
Dominique MaiterDepartment of EndocrinologyCentre Hospitalier Universitaire de Liège, University of Liège, Domaine Universitaire du Sart-Tilman, 4000 Liège, BelgiumDepartment of Human GeneticsCentre Hospitalier Universitaire de Liège, University of Liège, Domaine Universitaire du Sart-Tilman, 4000 Liège, BelgiumPediatric Endocrinology UnitUniversité Catholique de Louvain, Avenue Hippocrate 10, 1200 Brussels, BelgiumCliniques Universitaires Saint Luc and Department of PathologyUniversité Catholique de Louvain, Avenue Hippocrate 10, 1200 Brussels, BelgiumLaboratory of Molecular BiologyAPHM, Hôpital la Conception, 13385, Marseille, FranceAix Marseille UniversitéCRNS, CRN2M-UMR 7286, 13344, Marseille, FranceSection on Endocrinology and GeneticsProgram on Developmental Endocrinology and Genetics (PDEGEN) and Pediatric Endocrinology Inter-institute Training Program, Eunice Kennedy Shriver National Institute of Child Health and Human Development (NICHD), National Institutes of Health (NIH), Bethesda, Maryland 20892, USALaboratoire d'Hormonologie - CBPEGroupement Hospitalier Est, Hospices Civils de Lyon, 59 bd Pinel - 69677 Bron Cedex, FranceUniversité Catholique de LouvainPole d'endocrinologie, diabete et nutrition (EDIN), Brussels, BelgiumDepartment of NeurosurgeryUniversité Catholique de Louvain, Brussels, Belgium.
Chloe BrunelleDepartment of EndocrinologyCentre Hospitalier Universitaire de Liège, University of Liège, Domaine Universitaire du Sart-Tilman, 4000 Liège, BelgiumDepartment of Human GeneticsCentre Hospitalier Universitaire de Liège, University of Liège, Domaine Universitaire du Sart-Tilman, 4000 Liège, BelgiumPediatric Endocrinology UnitUniversité Catholique de Louvain, Avenue Hippocrate 10, 1200 Brussels, BelgiumCliniques Universitaires Saint Luc and Department of PathologyUniversité Catholique de Louvain, Avenue Hippocrate 10, 1200 Brussels, BelgiumLaboratory of Molecular BiologyAPHM, Hôpital la Conception, 13385, Marseille, FranceAix Marseille UniversitéCRNS, CRN2M-UMR 7286, 13344, Marseille, FranceSection on Endocrinology and GeneticsProgram on Developmental Endocrinology and Genetics (PDEGEN) and Pediatric Endocrinology Inter-institute Training Program, Eunice Kennedy Shriver National Institute of Child Health and Human Development (NICHD), National Institutes of Health (NIH), Bethesda, Maryland 20892, USALaboratoire d'Hormonologie - CBPEGroupement Hospitalier Est, Hospices Civils de Lyon, 59 bd Pinel - 69677 Bron Cedex, FranceUniversité Catholique de LouvainPole d'endocrinologie, diabete et nutrition (EDIN), Brussels, BelgiumDepartment of NeurosurgeryUniversité Catholique de Louvain, Brussels, Belgium.
Giampaolo TrivellinDepartment of EndocrinologyCentre Hospitalier Universitaire de Liège, University of Liège, Domaine Universitaire du Sart-Tilman, 4000 Liège, BelgiumDepartment of Human GeneticsCentre Hospitalier Universitaire de Liège, University of Liège, Domaine Universitaire du Sart-Tilman, 4000 Liège, BelgiumPediatric Endocrinology UnitUniversité Catholique de Louvain, Avenue Hippocrate 10, 1200 Brussels, BelgiumCliniques Universitaires Saint Luc and Department of PathologyUniversité Catholique de Louvain, Avenue Hippocrate 10, 1200 Brussels, BelgiumLaboratory of Molecular BiologyAPHM, Hôpital la Conception, 13385, Marseille, FranceAix Marseille UniversitéCRNS, CRN2M-UMR 7286, 13344, Marseille, FranceSection on Endocrinology and GeneticsProgram on Developmental Endocrinology and Genetics (PDEGEN) and Pediatric Endocrinology Inter-institute Training Program, Eunice Kennedy Shriver National Institute of Child Health and Human Development (NICHD), National Institutes of Health (NIH), Bethesda, Maryland 20892, USALaboratoire d'Hormonologie - CBPEGroupement Hospitalier Est, Hospices Civils de Lyon, 59 bd Pinel - 69677 Bron Cedex, FranceUniversité Catholique de LouvainPole d'endocrinologie, diabete et nutrition (EDIN), Brussels, BelgiumDepartment of NeurosurgeryUniversité Catholique de Louvain, Brussels, Belgium.
Constantine A StratakisDepartment of EndocrinologyCentre Hospitalier Universitaire de Liège, University of Liège, Domaine Universitaire du Sart-Tilman, 4000 Liège, BelgiumDepartment of Human GeneticsCentre Hospitalier Universitaire de Liège, University of Liège, Domaine Universitaire du Sart-Tilman, 4000 Liège, BelgiumPediatric Endocrinology UnitUniversité Catholique de Louvain, Avenue Hippocrate 10, 1200 Brussels, BelgiumCliniques Universitaires Saint Luc and Department of PathologyUniversité Catholique de Louvain, Avenue Hippocrate 10, 1200 Brussels, BelgiumLaboratory of Molecular BiologyAPHM, Hôpital la Conception, 13385, Marseille, FranceAix Marseille UniversitéCRNS, CRN2M-UMR 7286, 13344, Marseille, FranceSection on Endocrinology and GeneticsProgram on Developmental Endocrinology and Genetics (PDEGEN) and Pediatric Endocrinology Inter-institute Training Program, Eunice Kennedy Shriver National Institute of Child Health and Human Development (NICHD), National Institutes of Health (NIH), Bethesda, Maryland 20892, USALaboratoire d'Hormonologie - CBPEGroupement Hospitalier Est, Hospices Civils de Lyon, 59 bd Pinel - 69677 Bron Cedex, FranceUniversité Catholique de LouvainPole d'endocrinologie, diabete et nutrition (EDIN), Brussels, BelgiumDepartment of NeurosurgeryUniversité Catholique de Louvain, Brussels, Belgium.
Vincent BoursDepartment of EndocrinologyCentre Hospitalier Universitaire de Liège, University of Liège, Domaine Universitaire du Sart-Tilman, 4000 Liège, BelgiumDepartment of Human GeneticsCentre Hospitalier Universitaire de Liège, University of Liège, Domaine Universitaire du Sart-Tilman, 4000 Liège, BelgiumPediatric Endocrinology UnitUniversité Catholique de Louvain, Avenue Hippocrate 10, 1200 Brussels, BelgiumCliniques Universitaires Saint Luc and Department of PathologyUniversité Catholique de Louvain, Avenue Hippocrate 10, 1200 Brussels, BelgiumLaboratory of Molecular BiologyAPHM, Hôpital la Conception, 13385, Marseille, FranceAix Marseille UniversitéCRNS, CRN2M-UMR 7286, 13344, Marseille, FranceSection on Endocrinology and GeneticsProgram on Developmental Endocrinology and Genetics (PDEGEN) and Pediatric Endocrinology Inter-institute Training Program, Eunice Kennedy Shriver National Institute of Child Health and Human Development (NICHD), National Institutes of Health (NIH), Bethesda, Maryland 20892, USALaboratoire d'Hormonologie - CBPEGroupement Hospitalier Est, Hospices Civils de Lyon, 59 bd Pinel - 69677 Bron Cedex, FranceUniversité Catholique de LouvainPole d'endocrinologie, diabete et nutrition (EDIN), Brussels, BelgiumDepartment of NeurosurgeryUniversité Catholique de Louvain, Brussels, Belgium.
Christian RaftopoulosDepartment of EndocrinologyCentre Hospitalier Universitaire de Liège, University of Liège, Domaine Universitaire du Sart-Tilman, 4000 Liège, BelgiumDepartment of Human GeneticsCentre Hospitalier Universitaire de Liège, University of Liège, Domaine Universitaire du Sart-Tilman, 4000 Liège, BelgiumPediatric Endocrinology UnitUniversité Catholique de Louvain, Avenue Hippocrate 10, 1200 Brussels, BelgiumCliniques Universitaires Saint Luc and Department of PathologyUniversité Catholique de Louvain, Avenue Hippocrate 10, 1200 Brussels, BelgiumLaboratory of Molecular BiologyAPHM, Hôpital la Conception, 13385, Marseille, FranceAix Marseille UniversitéCRNS, CRN2M-UMR 7286, 13344, Marseille, FranceSection on Endocrinology and GeneticsProgram on Developmental Endocrinology and Genetics (PDEGEN) and Pediatric Endocrinology Inter-institute Training Program, Eunice Kennedy Shriver National Institute of Child Health and Human Development (NICHD), National Institutes of Health (NIH), Bethesda, Maryland 20892, USALaboratoire d'Hormonologie - CBPEGroupement Hospitalier Est, Hospices Civils de Lyon, 59 bd Pinel - 69677 Bron Cedex, FranceUniversité Catholique de LouvainPole d'endocrinologie, diabete et nutrition (EDIN), Brussels, BelgiumDepartment of NeurosurgeryUniversité Catholique de Louvain, Brussels, Belgium.
Veronique BeauloyeDepartment of EndocrinologyCentre Hospitalier Universitaire de Liège, University of Liège, Domaine Universitaire du Sart-Tilman, 4000 Liège, BelgiumDepartment of Human GeneticsCentre Hospitalier Universitaire de Liège, University of Liège, Domaine Universitaire du Sart-Tilman, 4000 Liège, BelgiumPediatric Endocrinology UnitUniversité Catholique de Louvain, Avenue Hippocrate 10, 1200 Brussels, BelgiumCliniques Universitaires Saint Luc and Department of PathologyUniversité Catholique de Louvain, Avenue Hippocrate 10, 1200 Brussels, BelgiumLaboratory of Molecular BiologyAPHM, Hôpital la Conception, 13385, Marseille, FranceAix Marseille UniversitéCRNS, CRN2M-UMR 7286, 13344, Marseille, FranceSection on Endocrinology and GeneticsProgram on Developmental Endocrinology and Genetics (PDEGEN) and Pediatric Endocrinology Inter-institute Training Program, Eunice Kennedy Shriver National Institute of Child Health and Human Development (NICHD), National Institutes of Health (NIH), Bethesda, Maryland 20892, USALaboratoire d'Hormonologie - CBPEGroupement Hospitalier Est, Hospices Civils de Lyon, 59 bd Pinel - 69677 Bron Cedex, FranceUniversité Catholique de LouvainPole d'endocrinologie, diabete et nutrition (EDIN), Brussels, BelgiumDepartment of NeurosurgeryUniversité Catholique de Louvain, Brussels, Belgium.
Anne BarlierDepartment of EndocrinologyCentre Hospitalier Universitaire de Liège, University of Liège, Domaine Universitaire du Sart-Tilman, 4000 Liège, BelgiumDepartment of Human GeneticsCentre Hospitalier Universitaire de Liège, University of Liège, Domaine Universitaire du Sart-Tilman, 4000 Liège, BelgiumPediatric Endocrinology UnitUniversité Catholique de Louvain, Avenue Hippocrate 10, 1200 Brussels, BelgiumCliniques Universitaires Saint Luc and Department of PathologyUniversité Catholique de Louvain, Avenue Hippocrate 10, 1200 Brussels, BelgiumLaboratory of Molecular BiologyAPHM, Hôpital la Conception, 13385, Marseille, FranceAix Marseille UniversitéCRNS, CRN2M-UMR 7286, 13344, Marseille, FranceSection on Endocrinology and GeneticsProgram on Developmental Endocrinology and Genetics (PDEGEN) and Pediatric Endocrinology Inter-institute Training Program, Eunice Kennedy Shriver National Institute of Child Health and Human Development (NICHD), National Institutes of Health (NIH), Bethesda, Maryland 20892, USALaboratoire d'Hormonologie - CBPEGroupement Hospitalier Est, Hospices Civils de Lyon, 59 bd Pinel - 69677 Bron Cedex, FranceUniversité Catholique de LouvainPole d'endocrinologie, diabete et nutrition (EDIN), Brussels, BelgiumDepartment of NeurosurgeryUniversité Catholique de Louvain, Brussels, Belgium Department of EndocrinologyCentre Hospitalier Universitaire de Liège, University of Liège, Domaine Universitaire du Sart-Tilman, 4000 Liège, BelgiumDepartment of Human GeneticsCentre Hospitalier Universitaire de Liège, University of Liège, Domaine Universitaire du Sart-Tilman, 4000 Liège, BelgiumPediatric Endocrinology UnitUniversité Catholique de Louvain, Avenue Hippocrate 10, 1200 Brussels, BelgiumCliniques Universitaires Saint Luc and Department of PathologyUniversité Catholique de Louvain, Avenue Hippocrate 10, 1200 Brussels, BelgiumLaboratory of Molecular BiologyAPHM, Hôpital la Conception, 13385, Marseille, FranceAix Marseille UniversitéCRNS, CRN2M-
Albert BeckersDepartment of EndocrinologyCentre Hospitalier Universitaire de Liège, University of Liège, Domaine Universitaire du Sart-Tilman, 4000 Liège, BelgiumDepartment of Human GeneticsCentre Hospitalier Universitaire de Liège, University of Liège, Domaine Universitaire du Sart-Tilman, 4000 Liège, BelgiumPediatric Endocrinology UnitUniversité Catholique de Louvain, Avenue Hippocrate 10, 1200 Brussels, BelgiumCliniques Universitaires Saint Luc and Department of PathologyUniversité Catholique de Louvain, Avenue Hippocrate 10, 1200 Brussels, BelgiumLaboratory of Molecular BiologyAPHM, Hôpital la Conception, 13385, Marseille, FranceAix Marseille UniversitéCRNS, CRN2M-UMR 7286, 13344, Marseille, FranceSection on Endocrinology and GeneticsProgram on Developmental Endocrinology and Genetics (PDEGEN) and Pediatric Endocrinology Inter-institute Training Program, Eunice Kennedy Shriver National Institute of Child Health and Human Development (NICHD), National Institutes of Health (NIH), Bethesda, Maryland 20892, USALaboratoire d'Hormonologie - CBPEGroupement Hospitalier Est, Hospices Civils de Lyon, 59 bd Pinel - 69677 Bron Cedex, FranceUniversité Catholique de LouvainPole d'endocrinologie, diabete et nutrition (EDIN), Brussels, BelgiumDepartment of NeurosurgeryUniversité Catholique de Louvain, Brussels, Belgium.
Centre Hospitalier Universitaire de Liège · BEUCLouvain · BECentre de Recherche en Neurobiologie - Neurophysiologie de Marseille · FRUniversity of Liège · BEEunice Kennedy Shriver National Institute of Child Health and Human Development · USHôpital de la Conception · FRHospices Civils de Lyon · FR

Funding

Molecular Genetics of Endocrine Tumors and Related DisordersZIAHD008920 · NICHD · EUNICE KENNEDY SHRIVER NATIONAL INSTITUTE OF CHILD HEALTH & HUMAN DEVELOPMENT · PI STRATAKIS, CONSTANTINE A. · 2012 to 2021
$25.8M
Intramural NIH HHS ZIA HD008920
6 · The paper itself

Abstract

X-linked acrogigantism (X-LAG) syndrome is a newly described form of inheritable pituitary gigantism that begins in early childhood and is usually associated with markedly elevated GH and prolactin secretion by mixed pituitary adenomas/hyperplasia. Microduplications on chromosome Xq26.3 including the GPR101 gene cause X-LAG syndrome. In individual cases random GHRH levels have been elevated. We performed a series of hormonal profiles in a young female sporadic X-LAG syndrome patient and subsequently undertook in vitro studies of primary pituitary tumor culture following neurosurgical resection. The patient demonstrated consistently elevated circulating GHRH levels throughout preoperative testing, which was accompanied by marked GH and prolactin hypersecretion; GH demonstrated a paradoxical increase following TRH administration. In vitro, the pituitary cells showed baseline GH and prolactin release that was further stimulated by GHRH administration. Co-incubation with GHRH and the GHRH receptor antagonist, acetyl-(d-Arg(2))-GHRH (1-29) amide, blocked the GHRH-induced GH stimulation; the GHRH receptor antagonist alone significantly reduced GH release. Pasireotide, but not octreotide, inhibited GH secretion. A ghrelin receptor agonist and an inverse agonist led to modest, statistically significant increases and decreases in GH secretion, respectively. GHRH hypersecretion can accompany the pituitary abnormalities seen in X-LAG syndrome. These data suggest that the pathology of X-LAG syndrome may include hypothalamic dysregulation of GHRH secretion, which is in keeping with localization of GPR101 in the hypothalamus. Therapeutic blockade of GHRH secretion could represent a way to target the marked hormonal hypersecretion and overgrowth that characterizes X-LAG syndrome.

Indexed as

Antineoplastic Agents, HormonalChild, PreschoolFemaleGenetic Diseases, X-LinkedGigantismGrowth HormoneGrowth Hormone-Releasing HormoneHumansOctreotidePituitary NeoplasmsProlactinReceptors, GhrelinSomatostatinSyndromeTumor Cells, CulturedAntineoplastic Agents, HormonalGrowth HormoneGrowth Hormone-Releasing HormoneOctreotidepasireotideProlactinReceptors, GhrelinSomatostatinGHRHgigantismGPR101growth hormonepituitary

Identifiers

PMID26671997
PMCPMC6300999
OpenAlexW2259967879

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