Evidence map›Paper›PMID 36071555›Full record

ReviewThe Journal of clinical endocrinology and metabolism2022

Familial Male-limited Precocious Puberty (FMPP) and Testicular Germ Cell Tumors.

Cezanne D Kooij, Annelies M C Mavinkurve-Groothuis, Idske C L Kremer Hovinga, Leendert H J Looijenga, Tuula Rinne, Jacques C Giltay, Laetitia M O de Kort, Aart J Klijn, Ronald R de Krijger, Annemarie A Verrijn Stuart

Open access · bronzeAbstract readReview
In one paragraph

Review in The Journal of clinical endocrinology and metabolism, 2022. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 4 papers.

0numbers the graph read from it
0cells of the map it votes in
4citing papers in PubMed
2.0field-weighted citation impact, top 14% 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

4 citing papers in PubMed, 9 citations in OpenAlex.

  1. Review
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  3. Familial Male-limited Precocious Puberty (FMPP) and Testicular Germ Cell Tumors.The Journal of clinical endocrinology and metabolism · 2022
    Review
  4. 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 4 institutions in 2 countries.

Cezanne D KooijPrincess Máxima Center for Pediatric Oncology, 3584 CS Utrecht, The Netherlands.ORCID 0000-0001-5985-0075
Annelies M C Mavinkurve-GroothuisPrincess Máxima Center for Pediatric Oncology, 3584 CS Utrecht, The Netherlands.ORCID 0000-0002-6984-3626
Idske C L Kremer HovingaDepartment of Pediatric Endocrinology, Wilhelmina Children's Hospital, 3584 EA Utrecht, The Netherlands.ORCID 0000-0002-6244-0429
Leendert H J LooijengaPrincess Máxima Center for Pediatric Oncology, 3584 CS Utrecht, The Netherlands.ORCID 0000-0002-8146-1911
Tuula RinneDepartment of Human Genetics, Radboud University Medical Center, 6525 GA Nijmegen, The Netherlands.ORCID 0000-0003-4350-8106
Jacques C GiltayDepartment of Medical Genetics, University Medical Center Utrecht, 3508 AB Utrecht, The Netherlands.ORCID 0000-0002-7946-1613
Laetitia M O de KortDepartment of Urology, University Medical Center Utrecht, 3584 CX Utrecht, The Netherlands.ORCID 0000-0002-9239-8356
Aart J KlijnDepartment of Pediatric Urology, Wilhelmina Children's Hospital, 3584 EA Utrecht, The Netherlands.ORCID 0000-0002-3460-2961
Ronald R de KrijgerPrincess Máxima Center for Pediatric Oncology, 3584 CS Utrecht, The Netherlands.ORCID 0000-0001-6871-1296
Annemarie A Verrijn StuartDepartment of Pediatric Endocrinology, Wilhelmina Children's Hospital, 3584 EA Utrecht, The Netherlands.
Princess Máxima Center · NLUniversity Medical Center Utrecht · NLWilhelmina Children's Hospital · NLRadboud University Nijmegen · NL

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

objectiveThe purpose of this study is to report development of a malignant testicular germ cell tumor (GCT) in 2 young adult males with familial male-limited precocious puberty (FMPP) because of LHCGR pathogenic variants in 2 families. Secondarily, to study the possible relation between FMPP and testicular tumors and to investigate whether FMPP might predispose to development of malignant testicular tumors in adulthood a literature review is conducted.

methodsData on 6 cases in 2 families are obtained from the available medical records. In addition, a database search is performed in Cochrane, PubMed, and Embase for studies that report on a possible link between FMPP and testicular tumors.

resultsThe characteristics of 6 males with FMPP based on activating LH receptor (LHCGR) germline pathogenic variants are described, as are details of the testicular GCTs. Furthermore, a literature review identified 4 more patients with signs of FMPP and a (precursor of) testicular GCT in adolescence or adulthood (age 15-35 years). Additionally, 12 patients with signs of precocious puberty and, simultaneously, occurrence of a Leydig cell adenoma or Leydig cell hyperplasia are reported.

conclusionThere is a strong suggestion that FMPP might increase the risk of development of testicular GCTs in early adulthood compared with the risk in the general population. Therefore, prolonged patient monitoring from mid-pubertal age onward including instruction for self-examination and periodic testicular ultrasound investigation in patients with a germline LHCGR pathogenic variant might contribute to early detection and thus early treatment of testicular GCT.

Indexed as

Puberty, PrecociousTesticular NeoplasmsAdolescentAdultHumansMaleNeoplasms, Germ Cell and EmbryonalYoung Adultactivating luteinizing hormone receptorfamilial male limited precocious pubertygerm cell tumorsperipheral precocious pubertytesticular tumorstestotoxicosis

Identifiers

PMID36071555
PMCPMC9681611
OpenAlexW4294992844

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.