Evidence map›Paper›PMID 31527860›Full record

ArticleEuropean journal of human genetics : EJHG2020

Declining detection rates for APC and biallelic MUTYH variants in polyposis patients, implications for DNA testing policy.

Diantha Terlouw, Manon Suerink, Sunny S Singh, Hans J J P Gille, Frederik J Hes, Alexandra M J Langers, Hans Morreau, Hans F A Vasen, Yvonne J Vos, Tom van Wezel and 3 more

Open access · bronzeAbstract read
In one paragraph

Article in European journal of human genetics : EJHG, 2020. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 9 papers, 1 of them a synthesis that pooled it.

0numbers the graph read from it
0cells of the map it votes in
9citing papers in PubMed, 1 pooled it
1.1field-weighted citation impact, top 23% 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

9 citing papers in PubMed, 1 synthesis or guideline pooled it, 22 citations in OpenAlex.

  1. Guideline
  2. Article
  3. Optimizing genetic testing strategy for suspected attenuated adenomatous polyposis: effective solutions in public health systems.Clinical & translational oncology : official publication of the Federation of Spanish Oncology Societies and of the National Cancer Institute of Mexico · 2025
    Article
  4. Article
  5. Article
  6. Review
  7. Article
  8. Article
  9. 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

13 authors at 4 institutions in 1 country.

Diantha TerlouwDepartment of Clinical Genetics, Leiden University Medical Center, Leiden, the Netherlands.
Manon SuerinkDepartment of Clinical Genetics, Leiden University Medical Center, Leiden, the Netherlands.
Sunny S SinghDepartment of Internal Medicine, Erasmus University Medical Center, Rotterdam, the Netherlands.
Hans J J P GilleDepartment of Clinical Genetics, VUMC, Amsterdam, the Netherlands.
Frederik J HesDepartment of Clinical Genetics, Leiden University Medical Center, Leiden, the Netherlands.ORCID http://orcid.org/0000-0002-3025-0882
Alexandra M J LangersDepartment of Gastroenterology and Hepatology, Leiden University Medical Center, Leiden, the Netherlands.
Hans MorreauDepartment of Pathology, Leiden University Medical Center, Leiden, the Netherlands.
Hans F A VasenDepartment of Gastroenterology and Hepatology, Leiden University Medical Center, Leiden, the Netherlands.
Yvonne J VosDepartment of Clinical Genetics, University of Groningen, University Medical Center Groningen, Groningen, the Netherlands.
Tom van WezelDepartment of Pathology, Leiden University Medical Center, Leiden, the Netherlands.ORCID http://orcid.org/0000-0001-5773-7730
Carli M TopsDepartment of Clinical Genetics, Leiden University Medical Center, Leiden, the Netherlands.
Sanne W Ten BroekeDepartment of Clinical Genetics, Leiden University Medical Center, Leiden, the Netherlands.
Maartje NielsenDepartment of Clinical Genetics, Leiden University Medical Center, Leiden, the Netherlands. m.nielsen@lumc.nl.ORCID http://orcid.org/0000-0002-5351-1870
Leiden University Medical Center · NLUniversity Medical Center Groningen · NLAmsterdam UMC Location Vrije Universiteit Amsterdam · NLErasmus MC · NL

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

This study aimed to determine the prevalence of APC-associated familial adenomatous polyposis (FAP) and MUTYH-associated polyposis (MAP) in a large cohort, taking into account factors as adenoma count and year of diagnosis. All application forms used to send patients in for APC and MUTYH variant analysis between 1992 and 2017 were collected (n = 2082). Using the data provided on the application form, the APC and biallelic MUTYH prevalence was determined and possible predictive factors were examined using multivariate multinomial logistic regression analysis in SPSS. The prevalence of disease causing variants in the APC gene significantly increases with adenoma count while MAP shows a peak prevalence in individuals with 50-99 adenomas. Logistic regression analysis shows significant odds ratios for adenoma count, age at diagnosis, and, interestingly, a decline in the chance of finding a variant in either gene over time. Moreover, in 22% (43/200) of patients with FAP-related extracolonic manifestations a variant was identified. The overall detection rates are above 10% for patients with >10 adenomas aged <60 and >20 adenomas aged <70. Patients with variants outside these criteria had FAP-related extracolonic manifestations, colorectal cancer aged <40, somatic KRAS c.34G > T variant in the tumor or a first-degree relative with >10 adenomas. Therefore, APC and MUTYH testing in patients with >10 adenomas aged <60 and with >20 adenomas aged <70 is advised. Almost all FAP and MAP patients not meeting these criteria showed other characteristics that can be used as an indication to prompt genetic testing.

Indexed as

Adenomatous Polyposis ColiAdolescentAdultAgedAllelesChildDNA GlycosylasesFemaleGenetic TestingHumansMaleMiddle AgedMutationPrevalenceDNA GlycosylasesmutY adenine glycosylase

Identifiers

PMID31527860
PMCPMC6974599
OpenAlexW2973381268

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.