Evidence map›Paper›PMID 33203450›Full record

Observational studyOrphanet journal of rare diseases2020

Pain drawings as a diagnostic tool for the differentiation between two pain-associated rare diseases (Ehlers-Danlos-Syndrome, Guillain-Barré-Syndrome).

Larissa Wester, Martin Mücke, Tim Theodor Albert Bender, Julia Sellin, Frank Klawonn, Rupert Conrad, Natasza Szczypien

Abstract readObservational Study
In one paragraph

Observational study in Orphanet journal of rare diseases, 2020. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 2 papers, 1 of them a synthesis that pooled it.

0numbers the graph read from it
0cells of the map it votes in
2citing papers in PubMed, 1 pooled it
–field-weighted citation impact
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

2 citing papers in PubMed, 1 synthesis or guideline pooled it.

  1. Pooled it
  2. 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

7 authors.

Larissa WesterCenter for Rare Diseases Bonn (ZSEB), University Hospital Bonn, Bonn, Germany.
Martin MückeCenter for Rare Diseases Bonn (ZSEB), University Hospital Bonn, Bonn, Germany. muecke@uni-bonn.de.ORCID 0000-0002-9275-9035
Tim Theodor Albert BenderCenter for Rare Diseases Bonn (ZSEB), University Hospital Bonn, Bonn, Germany.
Julia SellinCenter for Rare Diseases Bonn (ZSEB), University Hospital Bonn, Bonn, Germany.
Frank KlawonnInstitute for Information Engineering, Ostfalia University of Applied Sciences, Wolfenbüttel, Germany.
Rupert ConradDepartment of Psychosomatic Medicine and Psychotherapy, University Hospital Bonn, Bonn, Germany.
Natasza SzczypienInstitute for Information Engineering, Ostfalia University of Applied Sciences, Wolfenbüttel, Germany.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

backgroundThe diagnosis of rare diseases poses a particular challenge to clinicians. This study analyzes whether patients' pain drawings (PDs) help in the differentiation of two pain-associated rare diseases, Ehlers-Danlos Syndrome (EDS) and Guillain-Barré Syndrome (GBS).

methodThe study was designed as a prospective, observational, single-center study. The sample comprised 60 patients with EDS (3 male, 52 female, 5 without gender information; 39.2 ± 11.4 years) and 32 patients with GBS (10 male, 20 female, 2 without gender information; 50.5 ± 13.7 years). Patients marked areas afflicted by pain on a sketch of a human body with anterior, posterior, and lateral views. PDs were electronically scanned and processed. Each PD was classified based on the Ružička similarity to the EDS and the GBS averaged image (pain profile) in a leave-one-out cross validation approach. A receiver operating characteristic (ROC) curve was plotted.

results60-80% of EDS patients marked the vertebral column with the neck and the tailbone and the knee joints as pain areas, 40-50% the shoulder-region, the elbows and the thumb saddle joint. 60-70% of GBS patients marked the dorsal and plantar side of the feet as pain areas, 40-50% the palmar side of the fingertips, the dorsal side of the left palm and the tailbone. 86% of the EDS patients and 96% of the GBS patients were correctly identified by computing the Ružička similarity. The ROC curve yielded an excellent area under the curve value of 0.95.

conclusionPDs are a useful and economic tool to differentiate between GBS and EDS. Further studies should investigate its usefulness in the diagnosis of other pain-associated rare diseases. This study was registered in the German Clinical Trials Register, No. DRKS00014777 (Deutsches Register klinischer Studien, DRKS), on 01.06.2018.

Indexed as

Ehlers-Danlos SyndromeGuillain-Barre SyndromeFemaleHumansMalePainProspective StudiesRare DiseasesDifferential diagnosisORPHA: 2103ORPHA: 98249Orphan diseasesRužička similaritySelf-report instrument

Identifiers

PMID33203450
PMCPMC7672863

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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.