Evidence map›Paper›PMID 28685649›Full record

ArticleThe neuroradiology journal2018

Misdiagnosis: CNS Erdheim-Chester disease mimicking CLIPPERS.

Jillian Berkman, Caleb Ford, Emily Johnson, Beth A Malow, Joseph M Aulino

Abstract readCase Reports
In one paragraph

Article in The neuroradiology journal, 2018. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 9 papers.

0numbers the graph read from it
0cells of the map it votes in
9citing papers in PubMed
1.4field-weighted citation impact, top 18% 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, 19 citations in OpenAlex.

  1. Article
  2. "Crab sign" in an ataxia patient with central nervous system Erdheim Chester disease: a novel neuroimaging finding with review of literature.Neurological sciences : official journal of the Italian Neurological Society and of the Italian Society of Clinical Neurophysiology · 2025
    Review
  3. Neuroimaging Spectrum of Erdheim-Chester Disease: An Image-Based Review.AJNR. American journal of neuroradiology · 2025
    Review
  4. Review
  5. Erdheim-Chester Disease.AJNR. American journal of neuroradiology · 2023
    Article
  6. Article
  7. Brain biopsy in patients with CLIPPERS syndrome: why and when.Therapeutic advances in neurological disorders · 2022
    Article
  8. Article
  9. 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

5 authors at 2 institutions in 1 country.

Jillian Berkman1 Vanderbilt University School of Medicine, Nashville, TN, USA.
Caleb Ford1 Vanderbilt University School of Medicine, Nashville, TN, USA.
Emily Johnson2 Vanderbilt University Medical Center, Department of Neurology, Nashville, TN, USA.
Beth A Malow2 Vanderbilt University Medical Center, Department of Neurology, Nashville, TN, USA.
Joseph M Aulino3 Vanderbilt University Medical Center, Department of Radiology and Radiological Sciences, Nashville, TN, USA.
Vanderbilt University Medical Center · USVanderbilt University · US

Funding

MEDICAL SCIENTIST TRAINING PROGRAMT32GM007347 · NIGMS · VANDERBILT UNIVERSITY · PI WILLIAMS, CHRISTOPHER S. · 1985 to 2023
$26.3M
Research Project: Sensory and Multisensory Contributions to AutismU54HD083211 · NICHD · VANDERBILT UNIVERSITY MEDICAL CENTER · PI DYKENS, ELISABETH MAY · 2015 to 2019
$7.1M
NICHD NIH HHS U54 HD083211NIGMS NIH HHS T32 GM007347
6 · The paper itself

Abstract

Introduction Chronic lymphocytic inflammation with pontine perivascular enhancement responsive to steroids (CLIPPERS) is an inflammatory central nervous system (CNS) disorder with particular involvement of the pons. Diagnostic criteria include a range of clinical symptoms related to the underlying brainstem pathology, visible with magnetic resonance imaging (MRI). MRI findings include the appearance of punctuate and curvilinear gadolinium enhancement 'peppering' the pons. We discuss a patient presenting with clinical and radiographic characteristics of CLIPPERS who was diagnosed with Erdheim-Chester disease (ECD). Case report A 52-year-old male presented with 2 years of progressive spasticity, dysarthria, and gait instability. Initially, he was diagnosed with Parkinson's disease at an outside hospital, based on tremor, rigidity, and gait instability; however, he failed to improve with a trial of levodopa. Brain MRI showed small enhancing parenchymal nodules coalescing in the central pons, but also affecting the cerebellum and cerebellar peduncles, with more punctate enhancing lesions in the cerebral lobar subcortical white matter. When the patient's response to steroids was inadequate, further imaging was done, revealing perinephric processes. Subsequent biopsy revealed ECD. Conclusions A review of the literature for cases of CLIPPERS demonstrated a subset of patients later found to have various malignancies involving the CNS. This case report uses the patient's unique radiographic and clinical presentation to demonstrate the importance of the exclusion criteria within the CLIPPERS diagnostic requirements and stresses red flags suggestive of alternative diagnoses. This distinction is of high importance when differentiating a relatively benign process such as CLIPPERS from more malignant diseases.

Indexed as

BrainCentral Nervous System DiseasesDiagnostic ErrorsErdheim-Chester DiseaseHumansMaleMiddle Agedcentral nervous system imagingCLIPPERSErdheim–Chester disease

Identifiers

PMID28685649
PMCPMC6111432
OpenAlexW2776465796

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.