Evidence map›Paper›PMID 35133616›Full record

SynthesisJournal of endocrinological investigation2022

Global Cushing's disease epidemiology: a systematic review and meta-analysis of observational studies.

G Giuffrida, S Crisafulli, F Ferraù, A Fontana, Y Alessi, F Calapai, M Ragonese, N Luxi, S Cannavò, G Trifirò

Abstract readMeta-AnalysisSystematic Review
PubMed Publisher
In one paragraph

Synthesis in Journal of endocrinological investigation, 2022. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 29 papers, 1 of them a synthesis that pooled it.

0numbers the graph read from it
0cells of the map it votes in
29citing papers in PubMed, 1 pooled it
4.7field-weighted citation impact, top 4% 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

29 citing papers in PubMed, 1 synthesis or guideline pooled it, 41 citations in OpenAlex.

  1. Pooled it
  2. Prospective Multicenter Evaluation of [Journal of nuclear medicine : official publication, Society of Nuclear Medicine · 2025
    Trial
  3. Distinct plasma protein profiles after long-term remission of Cushing's disease.The Journal of clinical endocrinology and metabolism · 2026
    Article
  4. Cushing's disease across the lifespan: a case-based clinical review.Archives of endocrinology and metabolism · 2026
    Review
  5. Review
  6. The value of targeting CXCR4 withEuropean journal of nuclear medicine and molecular imaging · 2026
    Article
  7. Article
  8. The clinical value of [Pituitary · 2026
    Observational
  9. Article
  10. Article
  11. Article
  12. Article
  13. Article
  14. Article
  15. Article
  16. Review
  17. Article
  18. Review
  19. Article
  20. Review
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 3 institutions in 2 countries.

G Giuffrida *Department of Human Pathology of Adulthood and Childhood "G. Barresi", University of Messina, Messina, Italy.
S Crisafulli *Department of Medicine, University of Verona, Verona, Italy.
F FerraùDepartment of Human Pathology of Adulthood and Childhood "G. Barresi", University of Messina, Messina, Italy. fferrau@unime.it.ORCID http://orcid.org/0000-0002-7472-6598
A FontanaUnit of Biostatistics, Fondazione IRCCS Casa Sollievo della Sofferenza, San Giovanni Rotondo, Italy.
Y AlessiEndocrine Unit, University Hospital "G. Martino", Messina, Italy.
F CalapaiDepartment of Chemical, Biological, Pharmaceutical, Environmental Sciences, University of Messina, Messina, Italy.
M RagoneseDepartment of Human Pathology of Adulthood and Childhood "G. Barresi", University of Messina, Messina, Italy.
N LuxiDepartment of Diagnostics and Public Health, University of Verona, Verona, Italy.
S Cannavò *Department of Human Pathology of Adulthood and Childhood "G. Barresi", University of Messina, Messina, Italy.
G Trifirò *Department of Diagnostics and Public Health, University of Verona, Verona, Italy.
University of Messina · ITUniversity of Verona · ITCasa Sollievo della Sofferenza · IT

Funding

Ministero dell'Istruzione, dell'Università e della Ricerca PRIN 2017N8CK4KMinistero dell'Istruzione, dell'Università e della Ricerca Prin 2017S55RXB
6 · The paper itself

Abstract

purposeCushing's disease (CD), 70% of endogenous hypercortisolism cases, is a rare disease caused by adrenocorticotropic hormone (ACTH)-secreting pituitary adenomas. To date, no systematic reviews and meta-analyses on its global epidemiology have been published. We provide a systematic review and meta-analysis of CD global epidemiology, also evaluating the quality of study reporting for the identified studies.

methodsMEDLINE and EMBASE databases were searched for studies on CD epidemiology from inception until November 30th, 2020, including original observational studies in English about CD prevalence and/or incidence for well-defined geographic areas. Two reviewers independently extracted data and assessed reporting quality. CD prevalence/incidence pooled estimates were derived from a random-effects meta-analysis. Reporting quality was assessed using a STrengthening the Reporting of OBservational studies in Epidemiology (STROBE) checklist adapted for observational studies on rare diseases, heterogeneity using the Cochran's Q-test and its derived measure of inconsistency (I

resultsThirteen studies were included. The pooled CD prevalence was 2.2 [95% CI 1.1-4.8] per 100,000, while the incidence rate was 0.24 [95% CI 0.15-0.33] per 100,000 person-years. For both parameters, considerable between-studies heterogeneity was found (I

conclusionOverall, our systematic meta-analysis demonstrated CD epidemiology to be similarly reported across different areas of the world, with some exceptions regarding regional differences or observation period intervals. Keeping into account the methodological differences between each paper, large-scale studies on CD epidemiology are warranted. Setting up national specific registries, based on standardized diagnostic and clinical parameters, with clearly defined selection and analysis criteria, and a strong cooperation between the scientific national societies for endocrinology is crucial to exclude other causes of variability (i.e. geographical differences due to other factors like (epi)genetic changes), and to support public health decision making.

Indexed as

ACTH-Secreting Pituitary AdenomaAdenomaPituitary ACTH HypersecretionHumansIncidenceObservational Studies as TopicPrevalenceACTH-dependent hypercortisolismACTH-secreting PitNETsCushing’s disease

Identifiers

PMID35133616
OpenAlexW4210947023

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.