Evidence map›Paper›PMID 33247381›Full record

ArticleChild's nervous system : ChNS : official journal of the International Society for Pediatric Neurosurgery2021

Constitutional mismatch repair deficiency (CMMRD) presenting with high-grade glioma, multiple developmental venous anomalies and malformations of cortical development-a multidisciplinary/multicentre approach and neuroimaging clues to clinching the diagnosis.

Sahil Chhabda, Sniya Sudhakar, Kshitij Mankad, Mette Jorgensen, Fernando Carceller, Thomas S Jacques, Ashirwad Merve, Miren Aizpurua, Jane Chalker, Elisa Garimberti and 1 more

Abstract readCase ReportsMulticenter Study
PubMed Publisher
In one paragraph

Article in Child's nervous system : ChNS : official journal of the International Society for Pediatric Neurosurgery, 2021. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 4 papers, 1 of them a synthesis that pooled it.

0numbers the graph read from it
0cells of the map it votes in
4citing papers in PubMed, 1 pooled it
0.4field-weighted citation impact, top 36% 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, 1 synthesis or guideline pooled it, 7 citations in OpenAlex.

  1. Genotype-phenotype correlations inOncology reviews · 2025
    Pooled it
  2. Review
  3. Review
  4. 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

11 authors at 2 institutions in 1 country.

Sahil ChhabdaGreat Ormond Street Hospital for Children NHS Foundation Trust, Great Ormond Street, London, WC1N 3JH, UK. schhabda@nhs.net.ORCID 0000-0003-1191-1187
Sniya SudhakarGreat Ormond Street Hospital for Children NHS Foundation Trust, Great Ormond Street, London, WC1N 3JH, UK.
Kshitij MankadGreat Ormond Street Hospital for Children NHS Foundation Trust, Great Ormond Street, London, WC1N 3JH, UK.
Mette JorgensenGreat Ormond Street Hospital for Children NHS Foundation Trust, Great Ormond Street, London, WC1N 3JH, UK.
Fernando CarcellerThe Royal Marsden Hospital, Downs Road, Sutton, SM2 5PT, UK.
Thomas S JacquesPaediatric Neuropathology, UCL GOS Institute of Child Health, 30 Guilford Street, London, WC1N 1EH, UK.
Ashirwad MerveGreat Ormond Street Hospital for Children NHS Foundation Trust, Great Ormond Street, London, WC1N 3JH, UK.
Miren AizpuruaGreat Ormond Street Hospital for Children NHS Foundation Trust, Great Ormond Street, London, WC1N 3JH, UK.
Jane ChalkerSIHMDS-AG Laboratory, London North Genomic Laboratory Hub, Level 2, Camelia Botnar Laboratories, Great Ormond Street Hospital for Children NHS Foundation Trust, Great Ormond Street, London, WC1N 3JH, UK.
Elisa GarimbertiSIHMDS-AG Laboratory, London North Genomic Laboratory Hub, Level 2, Camelia Botnar Laboratories, Great Ormond Street Hospital for Children NHS Foundation Trust, Great Ormond Street, London, WC1N 3JH, UK.
Felice D'ArcoGreat Ormond Street Hospital for Children NHS Foundation Trust, Great Ormond Street, London, WC1N 3JH, UK.
Great Ormond Street Hospital for Children NHS Foundation Trust · GBInstitute of Cancer Research · GB

Funding

Medical Research Council G0902001
6 · The paper itself

Abstract

Constitutional mismatch repair deficiency syndrome (CMMRD) is a rare cancer-predisposition syndrome associated with a high risk of developing a spectrum of malignancies in childhood and adolescence, including brain tumours. In this report, we present the case of an 8-year-old boy with acute headache, vomiting and an episode of unconsciousness in whom brain imaging revealed a high-grade glioma (HGG). The possibility of an underlying diagnosis of CMMRD was suspected radiologically on the basis of additional neuroimaging findings, specifically the presence of multiple supratentorial and infratentorial developmental venous anomalies (DVAs) and malformations of cortical development (MCD), namely, heterotopic grey matter. The tumour was debulked and confirmed to be a HGG on histopathology. The suspected diagnosis of CMMRD was confirmed on immunohistochemistry and genetic testing which revealed mutations in PMS2 and MSH6. The combination of a HGG, multiple DVAs and MCD in a paediatric or young adult patient should prompt the neuroradiologist to suggest an underlying diagnosis of CMMRD. A diagnosis of CMMRD has an important treatment and surveillance implications not only for the child but also the family in terms of genetic counselling.

Indexed as

Brain NeoplasmsColorectal NeoplasmsGliomaMalformations of Cortical DevelopmentNeoplastic Syndromes, HereditaryAdolescentChildDNA Mismatch RepairHumansMaleMismatch Repair Endonuclease PMS2MutationNeuroimagingMismatch Repair Endonuclease PMS2CMMRDConstitutional mismatch repair deficiency syndromeDevelopmental venous anomalyGrey matter heterotopiaHigh-grade gliomaMalformation of cortical development

Identifiers

PMID33247381
OpenAlexW3110294661

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.