Evidence map›Paper›PMID 33757522›Full record

ArticlePediatric rheumatology online journal2021

Magnetic resonance imaging findings in children with Parry-Romberg syndrome and en coup de sabre.

Harry Knights, Elizabeth Minas, Faraan Khan, Lindsay Shaw, Muthana Al Obaidi, Kshitij Mankad, Despina Eleftheriou

Open access · goldAbstract read
PubMed Full text at PMC Publisher
In one paragraph

Article in Pediatric rheumatology online journal, 2021. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 5 papers, 1 of them a synthesis that pooled it.

0numbers the graph read from it
0cells of the map it votes in
5citing papers in PubMed, 1 pooled it
1.2field-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

5 citing papers in PubMed, 1 synthesis or guideline pooled it, 11 citations in OpenAlex.

  1. Pooled it
  2. Article
  3. Article
  4. Morphea: The 2023 update.Frontiers in medicine · 2023
    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

7 authors at 3 institutions in 1 country.

Harry KnightsDepartment of Paediatric Rheumatology, University College London Institute of Child Health and Great Ormond Street Hospital for Children NHS Foundation Trust, London, UK. hdk116@ic.ac.uk.ORCID https://orcid.org/0000-0002-8184-7208
Elizabeth MinasDepartment of Radiology, Charing Cross Hospital, London, UK.
Faraan KhanDepartment of Radiology, St George's University Hospitals NHS Foundation Trust, London, UK.
Lindsay ShawDepartment of Paediatric Dermatology, University College London Institute of Child Health and Great Ormond Street Hospital for Children NHS Foundation Trust, London, UK.
Muthana Al ObaidiDepartment of Paediatric Rheumatology, University College London Institute of Child Health and Great Ormond Street Hospital for Children NHS Foundation Trust, London, UK.
Kshitij Mankad *Department of Radiology, Great Ormond Street Hospital for Children NHS Foundation Trust, London, UK.
Despina Eleftheriou *Department of Paediatric Rheumatology, University College London Institute of Child Health and Great Ormond Street Hospital for Children NHS Foundation Trust, London, UK.
Great Ormond Street Hospital for Children NHS Foundation Trust · GBCharing Cross Hospital · GBSt George’s University Hospitals NHS Foundation Trust · GB

Funding

Versus Arthritis 20164 and 12593
6 · The paper itself

Abstract

backgroundThe aim of this study was to: (i) describe the abnormalities seen on brain imaging in a group of children with en coup de sabre (EDCS) with/without Parry-Romberg syndrome (PRS); and (ii) identify clinical predictors of brain imaging abnormalities.

methodsThis was a single centre (Great Ormond Street Hospital, London) retrospective case series of patients with ECDS/PRS seen from 2000 to 2018. We identified patients with cutaneous manifestations consistent with the clinical descriptions of ECDS/PRS. Presenting clinical, laboratory, and radiological brain findings are described. Results are expressed as medians and ranges or frequencies and percentages. Fisher's exact test was used to identify clinical associations with magnetic resonance imaging (MRI) abnormalities.

resultsFourteen patients were studied: 6 males and 8 females; median age 14 years (range 3-20). We observed neuroimaging abnormalities in 2/6 ECDS and 5/8 ECDS/PRS patients. White matter signal abnormality, dystrophic calcification, leptomeningeal enhancement, and sulcal crowding were the typical findings on brain imaging. A total of 50% of patients had no MRI abnormality despite some of these patients having neurological symptoms. The presence of seizures was significantly associated with ipsilateral enhanced white matter signalling on MRI (p < 0.05).

conclusionsIn summary, we observed several distinct radiographic patterns associated with ECDS/PRS. Seizure disorder was strongly associated with the presence of ipsilateral enhanced white matter signalling. Improved neuroimaging techniques that combine morphological with functional imaging may improve the detection rate of brain involvement in children with ECDS/PRS in the future.

Indexed as

Magnetic Resonance ImagingAdolescentChildChild, PreschoolFacial HemiatrophyFemaleHumansMaleRetrospective StudiesScleroderma, LocalizedYoung AdultEn coup de sabreEpilepsyLinear sclerodermaMagnetic resonance imagingParry-Romberg syndrome

Identifiers

PMID33757522
PMCPMC7986399
OpenAlexW3138702828

What OpenQuestion holds

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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.