Evidence map›Paper›PMID 29183089›Full record

ArticleHormone and metabolic research = Hormon- und Stoffwechselforschung = Hormones et metabolisme2018

Successful Treatment of Estrogen Excess in Primary Bilateral Macronodular Adrenocortical Hyperplasia with Leuprolide Acetate.

Fady Hannah-Shmouni, Andreas G Moraitis, Vladimir Valera Romero, Fabio R Faucz, Spyridon A Mastroyannis, Annabel Berthon, Richard A Failor, Maria Merino, Andrew P Demidowich, Constantine A Stratakis

Abstract readCase Reports
In one paragraph

Article in Hormone and metabolic research = Hormon- und Stoffwechselforschung = Hormones et metabolisme, 2018. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 6 papers.

0numbers the graph read from it
0cells of the map it votes in
6citing papers in PubMed
1.1field-weighted citation impact, top 21% 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

6 citing papers in PubMed, 11 citations in OpenAlex.

  1. Review
  2. Article
  3. Article
  4. Inhibin A as a tumor marker for primary bilateral macronodular adrenal hyperplasia.Endocrinology, diabetes & metabolism case reports · 2020
    Article
  5. Article
  6. 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

10 authors at 4 institutions in 1 country.

Fady Hannah-Shmouni *Section on Endocrinology & Genetics (SEGEN), Eunice Kennedy Shriver National Institute of Child Health and Human Development (NICHD), National Institutes of Health (NIH), Bethesda, MD, USA.
Andreas G Moraitis *Section on Endocrinology & Genetics (SEGEN), Eunice Kennedy Shriver National Institute of Child Health and Human Development (NICHD), National Institutes of Health (NIH), Bethesda, MD, USA.
Vladimir Valera RomeroLaboratory of Pathology, National Cancer Institute (NCI), NIH, Bethesda, MD, USA.
Fabio R FauczSection on Endocrinology & Genetics (SEGEN), Eunice Kennedy Shriver National Institute of Child Health and Human Development (NICHD), National Institutes of Health (NIH), Bethesda, MD, USA.
Spyridon A MastroyannisSection on Endocrinology & Genetics (SEGEN), Eunice Kennedy Shriver National Institute of Child Health and Human Development (NICHD), National Institutes of Health (NIH), Bethesda, MD, USA.
Annabel BerthonSection on Endocrinology & Genetics (SEGEN), Eunice Kennedy Shriver National Institute of Child Health and Human Development (NICHD), National Institutes of Health (NIH), Bethesda, MD, USA.
Richard A FailorDivision of Endocrinology, Metabolism, & Nutrition University of Washington, Seattle, WA, USA.
Maria MerinoLaboratory of Pathology, National Cancer Institute (NCI), NIH, Bethesda, MD, USA.
Andrew P DemidowichSection on Endocrinology & Genetics (SEGEN), Eunice Kennedy Shriver National Institute of Child Health and Human Development (NICHD), National Institutes of Health (NIH), Bethesda, MD, USA.
Constantine A StratakisSection on Endocrinology & Genetics (SEGEN), Eunice Kennedy Shriver National Institute of Child Health and Human Development (NICHD), National Institutes of Health (NIH), Bethesda, MD, USA.
Eunice Kennedy Shriver National Institute of Child Health and Human Development · USNational Institutes of Health · USNational Cancer Institute · USUniversity of Washington · US

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
Pediatric Endocrinology Training ProgramZIEHD008944 · NICHD · EUNICE KENNEDY SHRIVER NATIONAL INSTITUTE OF CHILD HEALTH & HUMAN DEVELOPMENT · PI YANOVSKI, JACK A. · 2014 to 2025
$12.1M
Translational Surgical PathologyZIESC000853 · NCI · DIVISION OF CLINICAL SCIENCES - NCI · PI MERINO, MARIA · 2009 to 2025
$4.3M
Intramural NIH HHS ZIA HD008920
6 · The paper itself

Abstract

Primary bilateral macronodular adrenocortical hyperplasia (PBMAH) is an uncommon cause of adrenal Cushing syndrome (CS) in which cortisol and occasionally other steroid hormones can be secreted under the influence of aberrantly expressed G-protein coupled receptors (GPCRs) in the adrenal cortex. We describe the unique case of a 64-year-old postmenopausal female with PBMAH whose adrenal lesions expressed luteinizing hormone receptors (LHr). She presented initially with CS and underwent right adrenalectomy; a few years later she presented with macromastia and mastodynia, possibly due to estrogen excess from her remaining left adrenocortical masses. Testing before and after treatment with quarterly leuprolide acetate therapy and immunohistochemistry on tissue and targeted sequencing of the genes of interest were performed. Tissue from the patient's right adrenal was tested for P450 aromatase (CYP19A1) and LHr expression; both were expressed throughout the hyperplastic cortex, although expression was more intense in the adenomatous areas. Targeted sequencing revealed a pathogenic

Indexed as

3',5'-Cyclic-GMP PhosphodiesterasesAdrenal Hyperplasia, CongenitalAgedBase SequenceEstrogensFollow-Up StudiesHumansImmunohistochemistryLeuprolideMiddle AgedPhosphoric Diester HydrolasesReceptors, LHSequence Analysis, DNAStaining and LabelingTomography, X-Ray ComputedTreatment Outcome3',5'-Cyclic-GMP PhosphodiesterasesEstrogensLeuprolidePDE11A protein, humanPhosphoric Diester HydrolasesReceptors, LH

Identifiers

PMID29183089
PMCPMC6343127
OpenAlexW2768344580

What OpenQuestion holds

Textmetadata
LicenceTDM
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.