Evidence map›Paper›PMID 36520683›Full record

ArticleEndocrine connections2023

Multiple endocrine neoplasia type 4: a new member of the MEN family.

Hélène Singeisen, Mariko Melanie Renzulli, Vojtech Pavlicek, Pascal Probst, Fabian Hauswirth, Markus K Muller, Magdalene Adamczyk, Achim Weber, Reto Martin Kaderli, Pietro Renzulli

Open access · goldAbstract read
In one paragraph

Article in Endocrine connections, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 24 papers.

0numbers the graph read from it
0cells of the map it votes in
24citing papers in PubMed
5.3field-weighted citation impact, top 3% 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

24 citing papers in PubMed, 37 citations in OpenAlex.

  1. Observational
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  4. Article
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  6. Article
  7. Article
  8. Heritable hyperparathyroidism: Genetic insights and clinical implications.Best practice & research. Clinical endocrinology & metabolism · 2025
    Review
  9. Pituitary Neuroendocrine Tumors in Multiple Endocrine Neoplasia.Endocrinology and metabolism (Seoul, Korea) · 2025
    Review
  10. p27Cells · 2025
    Article
  11. Neuroendocrine Tumors: Germline Genetics and Hereditary Syndromes.Current treatment options in oncology · 2025
    Review
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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 1 country.

Hélène SingeisenDepartment of Internal Medicine, Endocrinology, Cantonal Hospital Thurgau, Münsterlingen, Switzerland.
Mariko Melanie RenzulliInstitute of Radiology, Cantonal Hospital Thurgau, Frauenfeld, Switzerland.
Vojtech PavlicekDepartment of Internal Medicine, Endocrinology, Cantonal Hospital Thurgau, Münsterlingen, Switzerland.
Pascal ProbstDepartment of Surgery, Cantonal Hospital Thurgau, Frauenfeld, Switzerland.
Fabian HauswirthDepartment of Surgery, Cantonal Hospital Thurgau, Münsterlingen, Switzerland.
Markus K MullerDepartment of Surgery, Cantonal Hospital Thurgau, Frauenfeld, Switzerland.
Magdalene AdamczykDepartment of Pathology and Molecular Pathology, University Hospital Zurich and University of Zurich, Zurich, Switzerland.
Achim WeberDepartment of Pathology and Molecular Pathology, University Hospital Zurich and University of Zurich, Zurich, Switzerland.
Reto Martin KaderliDepartment of Visceral Surgery and Medicine, Bern University Hospital, University of Bern, Bern, Switzerland.
Pietro RenzulliDepartment of Surgery, Cantonal Hospital Thurgau, Münsterlingen, Switzerland.ORCID 0000-0001-5557-5512
University of Zurich · CHSpital Thurgau (Switzerland) · CHUniversity of Bern · CH

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Objective: Multiple endocrine neoplasia type 4 (MEN4) is caused by a CDKN1B germline mutation first described in 2006. Its estimated prevalence is less than one per million. The aim of this study was to define the disease characteristics. Methods: A systematic review was performed according to the PRISMA 2020 criteria. A literature search from January 2006 to August 2022 was done using MEDLINE® and Web of ScienceTM. Results: Forty-eight symptomatic patients fulfilled the pre-defined eligibility criteria. Twenty-eight different CDKN1B variants, mostly missense (21/48, 44%) and frameshift mutations (17/48, 35%), were reported. The majority of patients were women (36/48, 75%). Men became symptomatic at a median age of 32.5 years (range 10-68, mean 33.7 ± 23), whereas the same event was recorded for women at a median age of 49.5 years (range 5-76, mean 44.8 ± 19.9) (P = 0.25). The most frequently affected endocrine organ was the parathyroid gland (36/48, 75%; uniglandular disease 31/36, 86%), followed by the pituitary gland (21/48, 44%; hormone-secreting 16/21, 76%), the endocrine pancreas (7/48, 15%), and the thyroid gland (4/48, 8%). Tumors of the adrenal glands and thymus were found in three and two patients, respectively. The presenting first endocrine pathology concerned the parathyroid (27/48, 56%) and the pituitary gland (11/48, 23%). There were one (27/48, 56%), two (13/48, 27%), three (3/48, 6%), or four (5/48, 10%) syn- or metachronously affected endocrine organs in a single patient, respectively. Conclusion: MEN4 is an extremely rare disease, which most frequently affects women around 50 years of age. Primary hyperparathyroidism as a uniglandular disease is the leading pathology.

Indexed as

CDKN1Bmultiple endocrine neoplasia type 4parathyroid glandpituitary gland

Identifiers

PMID36520683
PMCPMC9874964
OpenAlexW4311588642

What OpenQuestion 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 OpenQuestion graph.