Evidence map›Paper›PMID 37399459›Full record

ArticleThe Journal of rheumatology2023

Feasibility of Conducting Comparative Effectiveness Research and Validation of a Clinical Disease Activity Score for Chronic Nonbacterial Osteomyelitis.

Eveline Y Wu, Melissa Oliver, Joshua Scheck, Sivia Lapidus, Ummusen Kaya Akca, Shima Yasin, Sara M Stern, Antonella Insalaco, Manuela Pardeo, Gabriele Simonini and 21 more

Registry-linked trialOpen access · bronzeAbstract read
PubMed Full text at PMC Publisher
In one paragraph

Article in The Journal of rheumatology, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. It is linked to trial NCT04725422 (CHronic Nonbacterial Osteomyelitis International Registry), which is not on this map. Cited by 13 papers.

0numbers the graph read from it
0cells of the map it votes in
13citing papers in PubMed
4.2field-weighted citation impact, top 5% 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.

NCT04725422 recruitingnot on this map

CHronic Nonbacterial Osteomyelitis International Registry (CHOIR)

TypeobservationalSponsorSeattle Children's HospitalRan2018 to 2050Enrolled2,000ConditionsChronic Nonbacterial Osteomyelitis, Chronic Recurrent Multifocal OsteomyelitisArmsMethotrexate, Sulfasalazine, Leflunomide, Pamidronate, Zoledronic acid
3 · Its place in the literature

Who cites it

13 citing papers in PubMed, 17 citations in OpenAlex.

  1. Review
  2. Review
  3. Article
  4. Review
  5. Review
  6. Article
  7. Observational
  8. Article
  9. Article
  10. Autoinflammatory Bone Diseases.Balkan medical journal · 2025
    Review
  11. Pediatric Musculoskeletal Imaging: Beginning of a New Journey.The Indian journal of radiology & imaging · 2025
    Article
  12. Observational
  13. 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

31 authors at 20 institutions in 8 countries.

Eveline Y WuE.Y. Wu, MD, MSCR, L.K. Kovalick, PNP, Division of Rheumatology, Department of Pediatrics, The University of North Carolina at Chapel Hill, Chapel Hill, North Carolina, USA.ORCID 0000-0002-6394-6843
Melissa OliverM. Oliver, MD, MS, Division of Pediatric Rheumatology, Indiana University School of Medicine, Indianapolis, Indiana, USA.ORCID 0000-0002-7689-2573
Joshua ScheckJ. Scheck, BS, N. Rosenwasser, MD, G. Casselman, BS, A. Liau, BS, Y. Shao, BS, C. Yang, BS, Y. Zhao, MD, PhD, Division of Pediatric Rheumatology, Seattle Children's Hospital, University of Washington, Seattle, Washington, USA.
Sivia LapidusS. Lapidus, MD, Division of Rheumatology, Department of Pediatrics, Joseph M. Sanzari Children's Hospital, Hackensack University Medical Center and Hackensack Meridian School of Medicine, Hackensack, New Jersey, USA.
Ummusen Kaya AkcaU. Kaya Akca, MD, S. Ozen, MD, Division of Rheumatology, Department of Pediatrics, Hacettepe University, Ankara, Turkey.ORCID 0000-0002-0426-9432
Shima YasinS. Yasin, MD, MSc, P.J. Ferguson, MD, Division of Rheumatology, Allergy and Immunology, Department of Pediatrics, University of Iowa Carver College of Medicine, Iowa City, Iowa, USA.
Sara M SternS.M. Stern, MD, Division of Rheumatology, Department of Pediatrics, The University of Utah, Salt Lake City, Utah, USA.
Antonella InsalacoA. Insalaco, MD, M. Pardeo, MD, Division of Rheumatology, ERN RITA Center, IRCCS Ospedale Pediatrico Bambino Gesù, Roma, Italy.
Manuela PardeoA. Insalaco, MD, M. Pardeo, MD, Division of Rheumatology, ERN RITA Center, IRCCS Ospedale Pediatrico Bambino Gesù, Roma, Italy.
Gabriele SimoniniG. Simonini, MD, E. Marrani, MD, Rheumatology Unit, ERN ReCONNET Center, Meyer Children's Hospital IRCCS, Florence, Italy.
Edoardo MarraniG. Simonini, MD, E. Marrani, MD, Rheumatology Unit, ERN ReCONNET Center, Meyer Children's Hospital IRCCS, Florence, Italy.ORCID 0000-0002-0346-3268
Xing WangX. Wang, PhD, Biostatistics Epidemiology and Analytics in Research, Seattle Children's Research Institute, Seattle, Washington, USA.
Bin HuangB. Huang, PhD, Division of Biostatistics and Epidemiology, Cincinnati Children's Hospital Medical Center, University of Cincinnati College of Medicine, Cincinnati, Ohio, USA.
Leonard K KovalickE.Y. Wu, MD, MSCR, L.K. Kovalick, PNP, Division of Rheumatology, Department of Pediatrics, The University of North Carolina at Chapel Hill, Chapel Hill, North Carolina, USA.
Natalie RosenwasserJ. Scheck, BS, N. Rosenwasser, MD, G. Casselman, BS, A. Liau, BS, Y. Shao, BS, C. Yang, BS, Y. Zhao, MD, PhD, Division of Pediatric Rheumatology, Seattle Children's Hospital, University of Washington, Seattle, Washington, USA.
Gabriel CasselmanJ. Scheck, BS, N. Rosenwasser, MD, G. Casselman, BS, A. Liau, BS, Y. Shao, BS, C. Yang, BS, Y. Zhao, MD, PhD, Division of Pediatric Rheumatology, Seattle Children's Hospital, University of Washington, Seattle, Washington, USA.ORCID 0000-0001-6950-7502
Adriel LiauJ. Scheck, BS, N. Rosenwasser, MD, G. Casselman, BS, A. Liau, BS, Y. Shao, BS, C. Yang, BS, Y. Zhao, MD, PhD, Division of Pediatric Rheumatology, Seattle Children's Hospital, University of Washington, Seattle, Washington, USA.
Yurong ShaoJ. Scheck, BS, N. Rosenwasser, MD, G. Casselman, BS, A. Liau, BS, Y. Shao, BS, C. Yang, BS, Y. Zhao, MD, PhD, Division of Pediatric Rheumatology, Seattle Children's Hospital, University of Washington, Seattle, Washington, USA.
Claire YangJ. Scheck, BS, N. Rosenwasser, MD, G. Casselman, BS, A. Liau, BS, Y. Shao, BS, C. Yang, BS, Y. Zhao, MD, PhD, Division of Pediatric Rheumatology, Seattle Children's Hospital, University of Washington, Seattle, Washington, USA.
Doaa Mosad MosaD.M. Mosa, MD, Rheumatology and Rehabilitation Department, Mansoura University Hospitals, Mansoura University, Mansoura City, Egypt.
Lori TuckerL. Tucker, MD, Division of Rheumatology, BC Children's Hospital, University of British Columbia, Vancouver, British Columbia, Canada.
Hermann GirschickH. Girschick, Department of Pediatrics, Vivantes Clinic Friedrichshain, Berlin, Germany.
Ronald M LaxerR.M. Laxer, The Hospital for Sick Children and the University of Toronto, Toronto, Ontario, Canada.
Jonathan D AkikusaJ.D. Akikusa, Royal Children's Hospital, Melbourne, Australia.
Christian M HedrichC.M. Hedrich, MD, PhD, Alder Hey Children's NHS Foundation Trust, Eaton Road, Liverpool, UK.
Karen OnelK. Onel, MD, Division of Pediatric Rheumatology, Hospital for Special Surgery, New York, New York, USA.ORCID 0000-0002-1238-5361
Fatma DedeogluF. Dedeoglu, MD, Division of Immunology, Boston Children's Hospital, Harvard Medical School, Boston, Massachusetts, USA.
Marinka TwiltM. Twilt, MD, Division of Rheumatology, Department of Paediatrics, Alberta Children's Hospital, Calgary, Alberta, Canada.ORCID 0000-0003-1954-2822
Polly J FergusonS. Yasin, MD, MSc, P.J. Ferguson, MD, Division of Rheumatology, Allergy and Immunology, Department of Pediatrics, University of Iowa Carver College of Medicine, Iowa City, Iowa, USA.
Seza OzenU. Kaya Akca, MD, S. Ozen, MD, Division of Rheumatology, Department of Pediatrics, Hacettepe University, Ankara, Turkey.ORCID 0000-0003-2883-7868
Yongdong ZhaoJ. Scheck, BS, N. Rosenwasser, MD, G. Casselman, BS, A. Liau, BS, Y. Shao, BS, C. Yang, BS, Y. Zhao, MD, PhD, Division of Pediatric Rheumatology, Seattle Children's Hospital, University of Washington, Seattle, Washington, USA; ydzhao@uw.edu.ORCID 0000-0003-3618-1379
Seattle Children's Hospital · USBambino Gesù Children's Hospital · ITHacettepe University · TRMeyer Children's Hospital · ITUniversity of Iowa · USUniversity of North Carolina at Chapel Hill · USAlberta Children's Hospital · CAAlder Hey Children's NHS Foundation Trust · GBBC Children's Hospital · CABoston Children's Hospital · USCancer Research And Biostatistics · USCincinnati Children's Hospital Medical Center · USHackensack University Medical Center · USHospital for Sick Children · CAHospital for Special Surgery · USIndiana University – Purdue University Indianapolis · USKlinikum im Friedrichshain · DEMansoura University · EGRoyal Children's Hospital · AUUniversity of Utah · US

Funding

North Carolina Translational and Clinical Science Institute (NC TraCS) KL2KL2TR002490 · NCATS · UNIV OF NORTH CAROLINA CHAPEL HILL · PI WEINBERGER, MORRIS · 2018 to 2022
$11.0M
Pediatric musculOskeletal & RheumaTology Innovation COre center (PORTICO)P30AR076316 · NIAMS · CINCINNATI CHILDRENS HOSP MED CTR · PI DUMOULIN, CHARLES L. · 2019 to 2023
$3.4M
NCATS NIH HHS KL2 TR002490NIAMS NIH HHS P30 AR076316
6 · The paper itself

Abstract

objectiveProspective comparative effectiveness research (CER) in chronic nonbacterial osteomyelitis (CNO) is lacking. Our objectives were to (1) determine the use and safety of each consensus treatment plan (CTP) regimen for CNO, (2) assess the feasibility of using the Chronic Nonbacterial Osteomyelitis International Registry (CHOIR) data for CER, and (3) develop and validate a CNO clinical disease activity score (CDAS) using CHOIR.

methodsConsenting children or young adults with CNO were enrolled into CHOIR. Demographic, clinical, and imaging data were prospectively collected. The CNO CDAS was developed through a Delphi survey and nominal group technique. External validation surveys were administered to CHOIR participants.

resultsOne hundred forty (78.2%) CHOIR participants enrolled between August 2018 and September 2020 received at least 1 CTP regimen. Baseline characteristics from different CTP groups were well matched. Patient pain, patient global assessment, and clinical CNO lesion count were key variables included in the CNO CDAS. The CDAS showed a strong correlation with patient/parent report of difficulty using a limb, back, or jaw and patient/parent report of disease severity, but a weak correlation with patient/parent report of fatigue, sadness, and worry. The change in CDAS was significant in patients reporting disease worsening or improvement (

conclusionThe CNO CDAS was developed and validated for disease monitoring and assessment of treatment effectiveness. CHOIR provided a comprehensive framework for future CER.

Indexed as

Comparative Effectiveness ResearchOsteomyelitisChildChronic DiseaseFeasibility StudiesHumansProspective StudiesYoung Adultchronic nonbacterial osteomyelitischronic recurrent multifocal osteomyelitisclinical disease activity scorecomparative effectiveness researchconsensus treatment plans

Identifiers

PMID37399459
PMCPMC10543471
OpenAlexW4382795108

What OpenQuestion holds

Textmetadata
LicenceTDM
Read underepoch 390

Registered trials

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.