Assessing disease experience across the life span for individuals with osteogenesis imperfecta: challenges and opportunities for patient-reported outcomes (PROs) measurement: ... View Full Text


Ontology type: schema:ScholarlyArticle      Open Access: True


Article Info

DATE

2019-12

AUTHORS

Laura L. Tosi, Marianne K. Floor, Christina M. Dollar, Austin P. Gillies, Members of the Brittle Bone Disease Consortium, Tracy S. Hart, David D. Cuthbertson, V. Reid Sutton, Jeffrey P. Krischer

ABSTRACT

BACKGROUND: Patient reported outcome (PRO) information is crucial for establishing better patient-provider communication, improving shared decision-making between clinicians and patients, assessing patient responses to therapeutic interventions, and increasing satisfaction with care. We used the Brittle Bones Disease Consortium (BBDC) Contact Registry for People with OI, managed by the Rare Disease Clinical Research Network (RDCRN) to (1) to evaluate the construct validity of the Patient-Reported Outcome Measurement Information System® (PROMIS®) to record important components of the disease experience among individuals with OI; and (2) explore the feasibility of using a registry to recruit individuals with OI to report on health status. Our long-term goal is to enhance communication of health and disease management findings back to the OI community, especially those who do not have access to major OI clinical centers. RESULTS: We demonstrated the construct validity of PROMIS instruments in OI. Our results confirm that the scores from most domains differ significantly from the general US population: individuals with OI have worse symptom burden and functioning. We found no excessive floor or ceiling effects. Our study demonstrates that the BBDC Contact Registry can be used to recruit participants for online health status surveys. However, there are numerous challenges that must be addressed: lack of self-knowledge of OI type, under-representation of men, limited ethnic diversity, and imperfect questionnaire completion rates. CONCLUSION: Our pilot study demonstrated the feasibility of using a contact registry to recruit respondents from the OI community and to obtain analyzable PROMIS data regarding disease experience. Because the results differ from the general population and avoid excessive floor and ceiling effects, PROMIS instruments can be used to assess response to therapeutic interventions in individuals with OI. Future directions will include (1) development and validation of an OI-specific patient-based classification system that aggregates persons with similar clinical characteristics and risks for complications to identify treatment needs; and (2) integrating these PRO tools into routine patient care and research studies. More... »

PAGES

23

Identifiers

URI

http://scigraph.springernature.com/pub.10.1186/s13023-019-1004-x

DOI

http://dx.doi.org/10.1186/s13023-019-1004-x

DIMENSIONS

https://app.dimensions.ai/details/publication/pub.1111767155

PUBMED

https://www.ncbi.nlm.nih.gov/pubmed/30696467


Indexing Status Check whether this publication has been indexed by Scopus and Web Of Science using the SN Indexing Status Tool
Incoming Citations Browse incoming citations for this publication using opencitations.net

JSON-LD is the canonical representation for SciGraph data.

TIP: You can open this SciGraph record using an external JSON-LD service: JSON-LD Playground Google SDTT

[
  {
    "@context": "https://springernature.github.io/scigraph/jsonld/sgcontext.json", 
    "about": [
      {
        "id": "http://purl.org/au-research/vocabulary/anzsrc-for/2008/1117", 
        "inDefinedTermSet": "http://purl.org/au-research/vocabulary/anzsrc-for/2008/", 
        "name": "Public Health and Health Services", 
        "type": "DefinedTerm"
      }, 
      {
        "id": "http://purl.org/au-research/vocabulary/anzsrc-for/2008/11", 
        "inDefinedTermSet": "http://purl.org/au-research/vocabulary/anzsrc-for/2008/", 
        "name": "Medical and Health Sciences", 
        "type": "DefinedTerm"
      }
    ], 
    "author": [
      {
        "affiliation": {
          "alternateName": "Children\u2019s National Health System", 
          "id": "https://www.grid.ac/institutes/grid.239560.b", 
          "name": [
            "Bone Health Program, Children\u2019s National Health System, 111 Michigan Avenue, NW, 20010, Washington, DC, USA"
          ], 
          "type": "Organization"
        }, 
        "familyName": "Tosi", 
        "givenName": "Laura L.", 
        "type": "Person"
      }, 
      {
        "affiliation": {
          "alternateName": "Children\u2019s National Health System", 
          "id": "https://www.grid.ac/institutes/grid.239560.b", 
          "name": [
            "Bone Health Program, Children\u2019s National Health System, 111 Michigan Avenue, NW, 20010, Washington, DC, USA"
          ], 
          "type": "Organization"
        }, 
        "familyName": "Floor", 
        "givenName": "Marianne K.", 
        "type": "Person"
      }, 
      {
        "affiliation": {
          "alternateName": "Children\u2019s National Health System", 
          "id": "https://www.grid.ac/institutes/grid.239560.b", 
          "name": [
            "Bone Health Program, Children\u2019s National Health System, 111 Michigan Avenue, NW, 20010, Washington, DC, USA"
          ], 
          "type": "Organization"
        }, 
        "familyName": "Dollar", 
        "givenName": "Christina M.", 
        "type": "Person"
      }, 
      {
        "affiliation": {
          "alternateName": "Children\u2019s National Health System", 
          "id": "https://www.grid.ac/institutes/grid.239560.b", 
          "name": [
            "Bone Health Program, Children\u2019s National Health System, 111 Michigan Avenue, NW, 20010, Washington, DC, USA"
          ], 
          "type": "Organization"
        }, 
        "familyName": "Gillies", 
        "givenName": "Austin P.", 
        "type": "Person"
      }, 
      {
        "familyName": "Members of the Brittle Bone Disease Consortium", 
        "type": "Person"
      }, 
      {
        "affiliation": {
          "alternateName": "Osteogenesis Imperfecta Foundation", 
          "id": "https://www.grid.ac/institutes/grid.423291.f", 
          "name": [
            "Osteogenesis Imperfecta Foundation, Gaithersburg, MD, USA"
          ], 
          "type": "Organization"
        }, 
        "familyName": "Hart", 
        "givenName": "Tracy S.", 
        "type": "Person"
      }, 
      {
        "affiliation": {
          "alternateName": "University of South Florida", 
          "id": "https://www.grid.ac/institutes/grid.170693.a", 
          "name": [
            "College of Medicine, University of South Florida, Tampa, Florida, USA"
          ], 
          "type": "Organization"
        }, 
        "familyName": "Cuthbertson", 
        "givenName": "David D.", 
        "type": "Person"
      }, 
      {
        "affiliation": {
          "alternateName": "Texas Children's Hospital", 
          "id": "https://www.grid.ac/institutes/grid.416975.8", 
          "name": [
            "Department of Molecular and Human Genetics, Baylor College of Medicine, Houston, TX, USA", 
            "Texas Children\u2019s Hospital, Houston, TX, USA"
          ], 
          "type": "Organization"
        }, 
        "familyName": "Sutton", 
        "givenName": "V. Reid", 
        "type": "Person"
      }, 
      {
        "affiliation": {
          "alternateName": "University of South Florida", 
          "id": "https://www.grid.ac/institutes/grid.170693.a", 
          "name": [
            "College of Medicine, University of South Florida, Tampa, Florida, USA"
          ], 
          "type": "Organization"
        }, 
        "familyName": "Krischer", 
        "givenName": "Jeffrey P.", 
        "type": "Person"
      }
    ], 
    "citation": [
      {
        "id": "https://doi.org/10.1016/j.cct.2012.02.012", 
        "sameAs": [
          "https://app.dimensions.ai/details/publication/pub.1003111252"
        ], 
        "type": "CreativeWork"
      }, 
      {
        "id": "sg:pub.10.1097/gim.0b013e3181a1ff7b", 
        "sameAs": [
          "https://app.dimensions.ai/details/publication/pub.1003253020", 
          "https://doi.org/10.1097/gim.0b013e3181a1ff7b"
        ], 
        "type": "CreativeWork"
      }, 
      {
        "id": "sg:pub.10.1097/gim.0b013e3181a1ff7b", 
        "sameAs": [
          "https://app.dimensions.ai/details/publication/pub.1003253020", 
          "https://doi.org/10.1097/gim.0b013e3181a1ff7b"
        ], 
        "type": "CreativeWork"
      }, 
      {
        "id": "https://doi.org/10.1002/ajmg.a.37365", 
        "sameAs": [
          "https://app.dimensions.ai/details/publication/pub.1005010730"
        ], 
        "type": "CreativeWork"
      }, 
      {
        "id": "https://doi.org/10.1002/ajmg.a.36545", 
        "sameAs": [
          "https://app.dimensions.ai/details/publication/pub.1005173401"
        ], 
        "type": "CreativeWork"
      }, 
      {
        "id": "https://doi.org/10.1136/jmg.16.2.101", 
        "sameAs": [
          "https://app.dimensions.ai/details/publication/pub.1005607842"
        ], 
        "type": "CreativeWork"
      }, 
      {
        "id": "https://doi.org/10.1016/s0140-6736(13)61091-0", 
        "sameAs": [
          "https://app.dimensions.ai/details/publication/pub.1008584059"
        ], 
        "type": "CreativeWork"
      }, 
      {
        "id": "https://doi.org/10.1200/jco.2013.53.5948", 
        "sameAs": [
          "https://app.dimensions.ai/details/publication/pub.1009488783"
        ], 
        "type": "CreativeWork"
      }, 
      {
        "id": "https://doi.org/10.1111/j.1399-0004.1989.tb03198.x", 
        "sameAs": [
          "https://app.dimensions.ai/details/publication/pub.1012028028"
        ], 
        "type": "CreativeWork"
      }, 
      {
        "id": "https://doi.org/10.1016/j.acra.2014.05.008", 
        "sameAs": [
          "https://app.dimensions.ai/details/publication/pub.1012470650"
        ], 
        "type": "CreativeWork"
      }, 
      {
        "id": "https://doi.org/10.1159/000351374", 
        "sameAs": [
          "https://app.dimensions.ai/details/publication/pub.1013811272"
        ], 
        "type": "CreativeWork"
      }, 
      {
        "id": "https://doi.org/10.4137/hsi.s11093", 
        "sameAs": [
          "https://app.dimensions.ai/details/publication/pub.1014229919"
        ], 
        "type": "CreativeWork"
      }, 
      {
        "id": "https://doi.org/10.1097/aud.0000000000000242", 
        "sameAs": [
          "https://app.dimensions.ai/details/publication/pub.1015057003"
        ], 
        "type": "CreativeWork"
      }, 
      {
        "id": "https://doi.org/10.1097/aud.0000000000000242", 
        "sameAs": [
          "https://app.dimensions.ai/details/publication/pub.1015057003"
        ], 
        "type": "CreativeWork"
      }, 
      {
        "id": "https://doi.org/10.1016/j.ejogrb.2006.09.003", 
        "sameAs": [
          "https://app.dimensions.ai/details/publication/pub.1017677486"
        ], 
        "type": "CreativeWork"
      }, 
      {
        "id": "https://doi.org/10.1111/j.1754-4505.2008.00070.x", 
        "sameAs": [
          "https://app.dimensions.ai/details/publication/pub.1023581999"
        ], 
        "type": "CreativeWork"
      }, 
      {
        "id": "https://doi.org/10.1097/mcd.0b013e32836032f0", 
        "sameAs": [
          "https://app.dimensions.ai/details/publication/pub.1023820074"
        ], 
        "type": "CreativeWork"
      }, 
      {
        "id": "https://doi.org/10.1097/mcd.0b013e32836032f0", 
        "sameAs": [
          "https://app.dimensions.ai/details/publication/pub.1023820074"
        ], 
        "type": "CreativeWork"
      }, 
      {
        "id": "https://doi.org/10.1097/mcd.0b013e32836032f0", 
        "sameAs": [
          "https://app.dimensions.ai/details/publication/pub.1023820074"
        ], 
        "type": "CreativeWork"
      }, 
      {
        "id": "https://doi.org/10.1097/mcd.0b013e32836032f0", 
        "sameAs": [
          "https://app.dimensions.ai/details/publication/pub.1023820074"
        ], 
        "type": "CreativeWork"
      }, 
      {
        "id": "https://doi.org/10.1097/mcd.0b013e32836032f0", 
        "sameAs": [
          "https://app.dimensions.ai/details/publication/pub.1023820074"
        ], 
        "type": "CreativeWork"
      }, 
      {
        "id": "sg:pub.10.1007/s11606-014-2894-x", 
        "sameAs": [
          "https://app.dimensions.ai/details/publication/pub.1024673878", 
          "https://doi.org/10.1007/s11606-014-2894-x"
        ], 
        "type": "CreativeWork"
      }, 
      {
        "id": "https://doi.org/10.1016/j.atherosclerosis.2016.08.023", 
        "sameAs": [
          "https://app.dimensions.ai/details/publication/pub.1026091770"
        ], 
        "type": "CreativeWork"
      }, 
      {
        "id": "https://doi.org/10.1097/mlr.0b013e31829b1d84", 
        "sameAs": [
          "https://app.dimensions.ai/details/publication/pub.1026300673"
        ], 
        "type": "CreativeWork"
      }, 
      {
        "id": "https://doi.org/10.1097/mlr.0b013e31829b1d84", 
        "sameAs": [
          "https://app.dimensions.ai/details/publication/pub.1026300673"
        ], 
        "type": "CreativeWork"
      }, 
      {
        "id": "https://doi.org/10.3109/14767058.2016.1151870", 
        "sameAs": [
          "https://app.dimensions.ai/details/publication/pub.1027833043"
        ], 
        "type": "CreativeWork"
      }, 
      {
        "id": "sg:pub.10.1007/s00428-004-0985-5", 
        "sameAs": [
          "https://app.dimensions.ai/details/publication/pub.1028748750", 
          "https://doi.org/10.1007/s00428-004-0985-5"
        ], 
        "type": "CreativeWork"
      }, 
      {
        "id": "sg:pub.10.1007/s00198-005-1905-5", 
        "sameAs": [
          "https://app.dimensions.ai/details/publication/pub.1028789286", 
          "https://doi.org/10.1007/s00198-005-1905-5"
        ], 
        "type": "CreativeWork"
      }, 
      {
        "id": "sg:pub.10.1007/s00198-005-1905-5", 
        "sameAs": [
          "https://app.dimensions.ai/details/publication/pub.1028789286", 
          "https://doi.org/10.1007/s00198-005-1905-5"
        ], 
        "type": "CreativeWork"
      }, 
      {
        "id": "sg:pub.10.1007/s00198-005-1905-5", 
        "sameAs": [
          "https://app.dimensions.ai/details/publication/pub.1028789286", 
          "https://doi.org/10.1007/s00198-005-1905-5"
        ], 
        "type": "CreativeWork"
      }, 
      {
        "id": "https://doi.org/10.1002/ajmg.a.33380", 
        "sameAs": [
          "https://app.dimensions.ai/details/publication/pub.1031491402"
        ], 
        "type": "CreativeWork"
      }, 
      {
        "id": "https://doi.org/10.1002/ajmg.a.33380", 
        "sameAs": [
          "https://app.dimensions.ai/details/publication/pub.1031491402"
        ], 
        "type": "CreativeWork"
      }, 
      {
        "id": "https://doi.org/10.3766/jaaa.19.8.3", 
        "sameAs": [
          "https://app.dimensions.ai/details/publication/pub.1032598647"
        ], 
        "type": "CreativeWork"
      }, 
      {
        "id": "https://doi.org/10.1111/j.1540-8175.2009.00973.x", 
        "sameAs": [
          "https://app.dimensions.ai/details/publication/pub.1035917253"
        ], 
        "type": "CreativeWork"
      }, 
      {
        "id": "https://doi.org/10.1111/j.1540-8175.2009.00973.x", 
        "sameAs": [
          "https://app.dimensions.ai/details/publication/pub.1035917253"
        ], 
        "type": "CreativeWork"
      }, 
      {
        "id": "https://doi.org/10.1016/j.jbi.2008.08.010", 
        "sameAs": [
          "https://app.dimensions.ai/details/publication/pub.1039152312"
        ], 
        "type": "CreativeWork"
      }, 
      {
        "id": "https://doi.org/10.1002/jbmr.1555", 
        "sameAs": [
          "https://app.dimensions.ai/details/publication/pub.1040635552"
        ], 
        "type": "CreativeWork"
      }, 
      {
        "id": "sg:pub.10.1007/s10792-006-9012-y", 
        "sameAs": [
          "https://app.dimensions.ai/details/publication/pub.1041094728", 
          "https://doi.org/10.1007/s10792-006-9012-y"
        ], 
        "type": "CreativeWork"
      }, 
      {
        "id": "sg:pub.10.1186/s13023-015-0362-2", 
        "sameAs": [
          "https://app.dimensions.ai/details/publication/pub.1041535695", 
          "https://doi.org/10.1186/s13023-015-0362-2"
        ], 
        "type": "CreativeWork"
      }, 
      {
        "id": "https://doi.org/10.1001/archderm.138.7.909", 
        "sameAs": [
          "https://app.dimensions.ai/details/publication/pub.1041713671"
        ], 
        "type": "CreativeWork"
      }, 
      {
        "id": "https://doi.org/10.1097/maj.0b013e318158e981", 
        "sameAs": [
          "https://app.dimensions.ai/details/publication/pub.1041846128"
        ], 
        "type": "CreativeWork"
      }, 
      {
        "id": "https://doi.org/10.1097/maj.0b013e318158e981", 
        "sameAs": [
          "https://app.dimensions.ai/details/publication/pub.1041846128"
        ], 
        "type": "CreativeWork"
      }, 
      {
        "id": "https://doi.org/10.1111/cge.12409", 
        "sameAs": [
          "https://app.dimensions.ai/details/publication/pub.1042226937"
        ], 
        "type": "CreativeWork"
      }, 
      {
        "id": "sg:pub.10.1007/s10545-006-0438-1", 
        "sameAs": [
          "https://app.dimensions.ai/details/publication/pub.1044396590", 
          "https://doi.org/10.1007/s10545-006-0438-1"
        ], 
        "type": "CreativeWork"
      }, 
      {
        "id": "https://doi.org/10.1016/j.cct.2008.09.002", 
        "sameAs": [
          "https://app.dimensions.ai/details/publication/pub.1045002035"
        ], 
        "type": "CreativeWork"
      }, 
      {
        "id": "https://doi.org/10.1053/ajot.2002.128037", 
        "sameAs": [
          "https://app.dimensions.ai/details/publication/pub.1046369974"
        ], 
        "type": "CreativeWork"
      }, 
      {
        "id": "https://doi.org/10.1053/ajot.2002.128037", 
        "sameAs": [
          "https://app.dimensions.ai/details/publication/pub.1046369974"
        ], 
        "type": "CreativeWork"
      }, 
      {
        "id": "https://doi.org/10.1097/scs.0b013e3181ecc55a", 
        "sameAs": [
          "https://app.dimensions.ai/details/publication/pub.1046714429"
        ], 
        "type": "CreativeWork"
      }, 
      {
        "id": "https://doi.org/10.1097/scs.0b013e3181ecc55a", 
        "sameAs": [
          "https://app.dimensions.ai/details/publication/pub.1046714429"
        ], 
        "type": "CreativeWork"
      }, 
      {
        "id": "https://doi.org/10.1097/scs.0b013e3181ecc55a", 
        "sameAs": [
          "https://app.dimensions.ai/details/publication/pub.1046714429"
        ], 
        "type": "CreativeWork"
      }, 
      {
        "id": "https://doi.org/10.1016/j.ahj.2010.11.006", 
        "sameAs": [
          "https://app.dimensions.ai/details/publication/pub.1048412135"
        ], 
        "type": "CreativeWork"
      }, 
      {
        "id": "https://doi.org/10.3174/ajnr.a0731", 
        "sameAs": [
          "https://app.dimensions.ai/details/publication/pub.1050503510"
        ], 
        "type": "CreativeWork"
      }, 
      {
        "id": "https://doi.org/10.1002/ajmg.a.33909", 
        "sameAs": [
          "https://app.dimensions.ai/details/publication/pub.1051316900"
        ], 
        "type": "CreativeWork"
      }, 
      {
        "id": "https://doi.org/10.1002/ajmg.a.33909", 
        "sameAs": [
          "https://app.dimensions.ai/details/publication/pub.1051316900"
        ], 
        "type": "CreativeWork"
      }, 
      {
        "id": "https://doi.org/10.1002/ajmg.a.33909", 
        "sameAs": [
          "https://app.dimensions.ai/details/publication/pub.1051316900"
        ], 
        "type": "CreativeWork"
      }, 
      {
        "id": "https://doi.org/10.1097/00004694-199809000-00001", 
        "sameAs": [
          "https://app.dimensions.ai/details/publication/pub.1060186948"
        ], 
        "type": "CreativeWork"
      }, 
      {
        "id": "https://doi.org/10.1097/00004694-199809000-00001", 
        "sameAs": [
          "https://app.dimensions.ai/details/publication/pub.1060186948"
        ], 
        "type": "CreativeWork"
      }, 
      {
        "id": "https://doi.org/10.1097/00004694-199809000-00001", 
        "sameAs": [
          "https://app.dimensions.ai/details/publication/pub.1060186948"
        ], 
        "type": "CreativeWork"
      }, 
      {
        "id": "https://doi.org/10.2106/jbjs.o.00494", 
        "sameAs": [
          "https://app.dimensions.ai/details/publication/pub.1068904073"
        ], 
        "type": "CreativeWork"
      }, 
      {
        "id": "https://doi.org/10.2106/jbjs.o.00494", 
        "sameAs": [
          "https://app.dimensions.ai/details/publication/pub.1068904073"
        ], 
        "type": "CreativeWork"
      }, 
      {
        "id": "https://doi.org/10.2106/jbjs.o.00494", 
        "sameAs": [
          "https://app.dimensions.ai/details/publication/pub.1068904073"
        ], 
        "type": "CreativeWork"
      }, 
      {
        "id": "https://doi.org/10.2196/jmir.4736", 
        "sameAs": [
          "https://app.dimensions.ai/details/publication/pub.1069286336"
        ], 
        "type": "CreativeWork"
      }, 
      {
        "id": "https://app.dimensions.ai/details/publication/pub.1077688423", 
        "type": "CreativeWork"
      }, 
      {
        "id": "https://doi.org/10.1016/j.metabol.2017.06.001", 
        "sameAs": [
          "https://app.dimensions.ai/details/publication/pub.1085932779"
        ], 
        "type": "CreativeWork"
      }, 
      {
        "id": "sg:pub.10.1007/s11136-017-1751-x", 
        "sameAs": [
          "https://app.dimensions.ai/details/publication/pub.1095857389", 
          "https://doi.org/10.1007/s11136-017-1751-x"
        ], 
        "type": "CreativeWork"
      }, 
      {
        "id": "https://doi.org/10.1001/jama.2017.21903", 
        "sameAs": [
          "https://app.dimensions.ai/details/publication/pub.1100829981"
        ], 
        "type": "CreativeWork"
      }
    ], 
    "datePublished": "2019-12", 
    "datePublishedReg": "2019-12-01", 
    "description": "BACKGROUND: Patient reported outcome (PRO) information is crucial for establishing better patient-provider communication, improving shared decision-making between clinicians and patients, assessing patient responses to therapeutic interventions, and increasing satisfaction with care. We used the Brittle Bones Disease Consortium (BBDC) Contact Registry for People with OI, managed by the Rare Disease Clinical Research Network (RDCRN) to (1) to evaluate the construct validity of the Patient-Reported Outcome Measurement Information System\u00ae (PROMIS\u00ae) to record important components of the disease experience among individuals with OI; and (2) explore the feasibility of using a registry to recruit individuals with OI to report on health status. Our long-term goal is to enhance communication of health and disease management findings back to the OI community, especially those who do not have access to major OI clinical centers.\nRESULTS: We demonstrated the construct validity of PROMIS instruments in OI. Our results confirm that the scores from most domains differ significantly from the general US population: individuals with OI have worse symptom burden and functioning. We found no excessive floor or ceiling effects. Our study demonstrates that the BBDC Contact Registry can be used to recruit participants for online health status surveys. However, there are numerous challenges that must be addressed: lack of self-knowledge of OI type, under-representation of men, limited ethnic diversity, and imperfect questionnaire completion rates.\nCONCLUSION: Our pilot study demonstrated the feasibility of using a contact registry to recruit respondents from the OI community and to obtain analyzable PROMIS data regarding disease experience. Because the results differ from the general population and avoid excessive floor and ceiling effects, PROMIS instruments can be used to assess response to therapeutic interventions in individuals with OI. Future directions will include (1) development and validation of an OI-specific patient-based classification system that aggregates persons with similar clinical characteristics and risks for complications to identify treatment needs; and (2) integrating these PRO tools into routine patient care and research studies.", 
    "genre": "research_article", 
    "id": "sg:pub.10.1186/s13023-019-1004-x", 
    "inLanguage": [
      "en"
    ], 
    "isAccessibleForFree": true, 
    "isFundedItemOf": [
      {
        "id": "sg:grant.3805114", 
        "type": "MonetaryGrant"
      }
    ], 
    "isPartOf": [
      {
        "id": "sg:journal.1036535", 
        "issn": [
          "1750-1172"
        ], 
        "name": "Orphanet Journal of Rare Diseases", 
        "type": "Periodical"
      }, 
      {
        "issueNumber": "1", 
        "type": "PublicationIssue"
      }, 
      {
        "type": "PublicationVolume", 
        "volumeNumber": "14"
      }
    ], 
    "name": "Assessing disease experience across the life span for individuals with osteogenesis imperfecta: challenges and opportunities for patient-reported outcomes (PROs) measurement: a pilot study", 
    "pagination": "23", 
    "productId": [
      {
        "name": "readcube_id", 
        "type": "PropertyValue", 
        "value": [
          "c804f548a987bde0f70164b67e86c2593875fe5b47a7c8fdae69c3a1285b9fb4"
        ]
      }, 
      {
        "name": "pubmed_id", 
        "type": "PropertyValue", 
        "value": [
          "30696467"
        ]
      }, 
      {
        "name": "nlm_unique_id", 
        "type": "PropertyValue", 
        "value": [
          "101266602"
        ]
      }, 
      {
        "name": "doi", 
        "type": "PropertyValue", 
        "value": [
          "10.1186/s13023-019-1004-x"
        ]
      }, 
      {
        "name": "dimensions_id", 
        "type": "PropertyValue", 
        "value": [
          "pub.1111767155"
        ]
      }
    ], 
    "sameAs": [
      "https://doi.org/10.1186/s13023-019-1004-x", 
      "https://app.dimensions.ai/details/publication/pub.1111767155"
    ], 
    "sdDataset": "articles", 
    "sdDatePublished": "2019-04-11T09:55", 
    "sdLicense": "https://scigraph.springernature.com/explorer/license/", 
    "sdPublisher": {
      "name": "Springer Nature - SN SciGraph project", 
      "type": "Organization"
    }, 
    "sdSource": "s3://com-uberresearch-data-dimensions-target-20181106-alternative/cleanup/v134/2549eaecd7973599484d7c17b260dba0a4ecb94b/merge/v9/a6c9fde33151104705d4d7ff012ea9563521a3ce/jats-lookup/v90/0000000347_0000000347/records_89801_00000004.jsonl", 
    "type": "ScholarlyArticle", 
    "url": "https://link.springer.com/10.1186%2Fs13023-019-1004-x"
  }
]
 

Download the RDF metadata as:  json-ld nt turtle xml License info

HOW TO GET THIS DATA PROGRAMMATICALLY:

JSON-LD is a popular format for linked data which is fully compatible with JSON.

curl -H 'Accept: application/ld+json' 'https://scigraph.springernature.com/pub.10.1186/s13023-019-1004-x'

N-Triples is a line-based linked data format ideal for batch operations.

curl -H 'Accept: application/n-triples' 'https://scigraph.springernature.com/pub.10.1186/s13023-019-1004-x'

Turtle is a human-readable linked data format.

curl -H 'Accept: text/turtle' 'https://scigraph.springernature.com/pub.10.1186/s13023-019-1004-x'

RDF/XML is a standard XML format for linked data.

curl -H 'Accept: application/rdf+xml' 'https://scigraph.springernature.com/pub.10.1186/s13023-019-1004-x'


 

This table displays all metadata directly associated to this object as RDF triples.

267 TRIPLES      21 PREDICATES      74 URIs      21 LITERALS      9 BLANK NODES

Subject Predicate Object
1 sg:pub.10.1186/s13023-019-1004-x schema:about anzsrc-for:11
2 anzsrc-for:1117
3 schema:author Ne098418287af4fa6a907691b9583d2ab
4 schema:citation sg:pub.10.1007/s00198-005-1905-5
5 sg:pub.10.1007/s00428-004-0985-5
6 sg:pub.10.1007/s10545-006-0438-1
7 sg:pub.10.1007/s10792-006-9012-y
8 sg:pub.10.1007/s11136-017-1751-x
9 sg:pub.10.1007/s11606-014-2894-x
10 sg:pub.10.1097/gim.0b013e3181a1ff7b
11 sg:pub.10.1186/s13023-015-0362-2
12 https://app.dimensions.ai/details/publication/pub.1077688423
13 https://doi.org/10.1001/archderm.138.7.909
14 https://doi.org/10.1001/jama.2017.21903
15 https://doi.org/10.1002/ajmg.a.33380
16 https://doi.org/10.1002/ajmg.a.33909
17 https://doi.org/10.1002/ajmg.a.36545
18 https://doi.org/10.1002/ajmg.a.37365
19 https://doi.org/10.1002/jbmr.1555
20 https://doi.org/10.1016/j.acra.2014.05.008
21 https://doi.org/10.1016/j.ahj.2010.11.006
22 https://doi.org/10.1016/j.atherosclerosis.2016.08.023
23 https://doi.org/10.1016/j.cct.2008.09.002
24 https://doi.org/10.1016/j.cct.2012.02.012
25 https://doi.org/10.1016/j.ejogrb.2006.09.003
26 https://doi.org/10.1016/j.jbi.2008.08.010
27 https://doi.org/10.1016/j.metabol.2017.06.001
28 https://doi.org/10.1016/s0140-6736(13)61091-0
29 https://doi.org/10.1053/ajot.2002.128037
30 https://doi.org/10.1097/00004694-199809000-00001
31 https://doi.org/10.1097/aud.0000000000000242
32 https://doi.org/10.1097/maj.0b013e318158e981
33 https://doi.org/10.1097/mcd.0b013e32836032f0
34 https://doi.org/10.1097/mlr.0b013e31829b1d84
35 https://doi.org/10.1097/scs.0b013e3181ecc55a
36 https://doi.org/10.1111/cge.12409
37 https://doi.org/10.1111/j.1399-0004.1989.tb03198.x
38 https://doi.org/10.1111/j.1540-8175.2009.00973.x
39 https://doi.org/10.1111/j.1754-4505.2008.00070.x
40 https://doi.org/10.1136/jmg.16.2.101
41 https://doi.org/10.1159/000351374
42 https://doi.org/10.1200/jco.2013.53.5948
43 https://doi.org/10.2106/jbjs.o.00494
44 https://doi.org/10.2196/jmir.4736
45 https://doi.org/10.3109/14767058.2016.1151870
46 https://doi.org/10.3174/ajnr.a0731
47 https://doi.org/10.3766/jaaa.19.8.3
48 https://doi.org/10.4137/hsi.s11093
49 schema:datePublished 2019-12
50 schema:datePublishedReg 2019-12-01
51 schema:description BACKGROUND: Patient reported outcome (PRO) information is crucial for establishing better patient-provider communication, improving shared decision-making between clinicians and patients, assessing patient responses to therapeutic interventions, and increasing satisfaction with care. We used the Brittle Bones Disease Consortium (BBDC) Contact Registry for People with OI, managed by the Rare Disease Clinical Research Network (RDCRN) to (1) to evaluate the construct validity of the Patient-Reported Outcome Measurement Information System® (PROMIS®) to record important components of the disease experience among individuals with OI; and (2) explore the feasibility of using a registry to recruit individuals with OI to report on health status. Our long-term goal is to enhance communication of health and disease management findings back to the OI community, especially those who do not have access to major OI clinical centers. RESULTS: We demonstrated the construct validity of PROMIS instruments in OI. Our results confirm that the scores from most domains differ significantly from the general US population: individuals with OI have worse symptom burden and functioning. We found no excessive floor or ceiling effects. Our study demonstrates that the BBDC Contact Registry can be used to recruit participants for online health status surveys. However, there are numerous challenges that must be addressed: lack of self-knowledge of OI type, under-representation of men, limited ethnic diversity, and imperfect questionnaire completion rates. CONCLUSION: Our pilot study demonstrated the feasibility of using a contact registry to recruit respondents from the OI community and to obtain analyzable PROMIS data regarding disease experience. Because the results differ from the general population and avoid excessive floor and ceiling effects, PROMIS instruments can be used to assess response to therapeutic interventions in individuals with OI. Future directions will include (1) development and validation of an OI-specific patient-based classification system that aggregates persons with similar clinical characteristics and risks for complications to identify treatment needs; and (2) integrating these PRO tools into routine patient care and research studies.
52 schema:genre research_article
53 schema:inLanguage en
54 schema:isAccessibleForFree true
55 schema:isPartOf N63b908a2ab5941ada256bb1a8256ed73
56 Ne74dacba06fe431da4d1f8bb3283645e
57 sg:journal.1036535
58 schema:name Assessing disease experience across the life span for individuals with osteogenesis imperfecta: challenges and opportunities for patient-reported outcomes (PROs) measurement: a pilot study
59 schema:pagination 23
60 schema:productId N44592bc78b09407792e557c0c0ea769e
61 N91793b5c36eb4c23a261da1f954fc32e
62 Nb01c06174ffa4449bf5caed76d86530b
63 Nc5dc7d85ca16456d93de9581fa93a888
64 Nc83d5ac9839546978f15c75038145959
65 schema:sameAs https://app.dimensions.ai/details/publication/pub.1111767155
66 https://doi.org/10.1186/s13023-019-1004-x
67 schema:sdDatePublished 2019-04-11T09:55
68 schema:sdLicense https://scigraph.springernature.com/explorer/license/
69 schema:sdPublisher N8c6a4d448ded4860b053d32850f98112
70 schema:url https://link.springer.com/10.1186%2Fs13023-019-1004-x
71 sgo:license sg:explorer/license/
72 sgo:sdDataset articles
73 rdf:type schema:ScholarlyArticle
74 N149d2fd6fa6348b1919398a6e89bee83 schema:affiliation https://www.grid.ac/institutes/grid.170693.a
75 schema:familyName Cuthbertson
76 schema:givenName David D.
77 rdf:type schema:Person
78 N44592bc78b09407792e557c0c0ea769e schema:name pubmed_id
79 schema:value 30696467
80 rdf:type schema:PropertyValue
81 N46e365da3b364ce7bc8b4143f51d41f1 rdf:first Nce9e820d07a842d08cd7534bcbb63d4b
82 rdf:rest N702bee2f3a3c42599c277fc9f80ba24d
83 N5416d085e3374681ab40563e2939ebe5 schema:affiliation https://www.grid.ac/institutes/grid.239560.b
84 schema:familyName Dollar
85 schema:givenName Christina M.
86 rdf:type schema:Person
87 N55f6df644d05491b8e9919bf52c2b753 rdf:first N149d2fd6fa6348b1919398a6e89bee83
88 rdf:rest N46e365da3b364ce7bc8b4143f51d41f1
89 N5e4695afca014be7bf803464e90122af schema:affiliation https://www.grid.ac/institutes/grid.423291.f
90 schema:familyName Hart
91 schema:givenName Tracy S.
92 rdf:type schema:Person
93 N63b908a2ab5941ada256bb1a8256ed73 schema:issueNumber 1
94 rdf:type schema:PublicationIssue
95 N702bee2f3a3c42599c277fc9f80ba24d rdf:first N7079701ceb064dac9307252e1ac47da1
96 rdf:rest rdf:nil
97 N7079701ceb064dac9307252e1ac47da1 schema:affiliation https://www.grid.ac/institutes/grid.170693.a
98 schema:familyName Krischer
99 schema:givenName Jeffrey P.
100 rdf:type schema:Person
101 N70fd411f17f749cbb9dcb3eb5fc4483c rdf:first Ndb1365ceae594bb491bed1cfbc613723
102 rdf:rest Nc168d58913f644c8bba4807789e4105a
103 N7abf80fc344248869a3bf94884c80ae4 rdf:first N5416d085e3374681ab40563e2939ebe5
104 rdf:rest N70fd411f17f749cbb9dcb3eb5fc4483c
105 N8c6a4d448ded4860b053d32850f98112 schema:name Springer Nature - SN SciGraph project
106 rdf:type schema:Organization
107 N91793b5c36eb4c23a261da1f954fc32e schema:name dimensions_id
108 schema:value pub.1111767155
109 rdf:type schema:PropertyValue
110 N943a2035e78b4575a142ef6ab733f0ca rdf:first N5e4695afca014be7bf803464e90122af
111 rdf:rest N55f6df644d05491b8e9919bf52c2b753
112 Nb01c06174ffa4449bf5caed76d86530b schema:name doi
113 schema:value 10.1186/s13023-019-1004-x
114 rdf:type schema:PropertyValue
115 Nb8b2d40206f14598a1e22f63d7283b95 schema:affiliation https://www.grid.ac/institutes/grid.239560.b
116 schema:familyName Tosi
117 schema:givenName Laura L.
118 rdf:type schema:Person
119 Nbbb02ce260e64937a2b8e42fd0ab2806 schema:affiliation https://www.grid.ac/institutes/grid.239560.b
120 schema:familyName Floor
121 schema:givenName Marianne K.
122 rdf:type schema:Person
123 Nc07b28deb42047d280f553f533b8efe8 schema:familyName Members of the Brittle Bone Disease Consortium
124 rdf:type schema:Person
125 Nc168d58913f644c8bba4807789e4105a rdf:first Nc07b28deb42047d280f553f533b8efe8
126 rdf:rest N943a2035e78b4575a142ef6ab733f0ca
127 Nc5dc7d85ca16456d93de9581fa93a888 schema:name readcube_id
128 schema:value c804f548a987bde0f70164b67e86c2593875fe5b47a7c8fdae69c3a1285b9fb4
129 rdf:type schema:PropertyValue
130 Nc83d5ac9839546978f15c75038145959 schema:name nlm_unique_id
131 schema:value 101266602
132 rdf:type schema:PropertyValue
133 Nce9e820d07a842d08cd7534bcbb63d4b schema:affiliation https://www.grid.ac/institutes/grid.416975.8
134 schema:familyName Sutton
135 schema:givenName V. Reid
136 rdf:type schema:Person
137 Ndb1365ceae594bb491bed1cfbc613723 schema:affiliation https://www.grid.ac/institutes/grid.239560.b
138 schema:familyName Gillies
139 schema:givenName Austin P.
140 rdf:type schema:Person
141 Ne098418287af4fa6a907691b9583d2ab rdf:first Nb8b2d40206f14598a1e22f63d7283b95
142 rdf:rest Nef1989831c5c4d58bba20d6d8220d712
143 Ne74dacba06fe431da4d1f8bb3283645e schema:volumeNumber 14
144 rdf:type schema:PublicationVolume
145 Nef1989831c5c4d58bba20d6d8220d712 rdf:first Nbbb02ce260e64937a2b8e42fd0ab2806
146 rdf:rest N7abf80fc344248869a3bf94884c80ae4
147 anzsrc-for:11 schema:inDefinedTermSet anzsrc-for:
148 schema:name Medical and Health Sciences
149 rdf:type schema:DefinedTerm
150 anzsrc-for:1117 schema:inDefinedTermSet anzsrc-for:
151 schema:name Public Health and Health Services
152 rdf:type schema:DefinedTerm
153 sg:grant.3805114 http://pending.schema.org/fundedItem sg:pub.10.1186/s13023-019-1004-x
154 rdf:type schema:MonetaryGrant
155 sg:journal.1036535 schema:issn 1750-1172
156 schema:name Orphanet Journal of Rare Diseases
157 rdf:type schema:Periodical
158 sg:pub.10.1007/s00198-005-1905-5 schema:sameAs https://app.dimensions.ai/details/publication/pub.1028789286
159 https://doi.org/10.1007/s00198-005-1905-5
160 rdf:type schema:CreativeWork
161 sg:pub.10.1007/s00428-004-0985-5 schema:sameAs https://app.dimensions.ai/details/publication/pub.1028748750
162 https://doi.org/10.1007/s00428-004-0985-5
163 rdf:type schema:CreativeWork
164 sg:pub.10.1007/s10545-006-0438-1 schema:sameAs https://app.dimensions.ai/details/publication/pub.1044396590
165 https://doi.org/10.1007/s10545-006-0438-1
166 rdf:type schema:CreativeWork
167 sg:pub.10.1007/s10792-006-9012-y schema:sameAs https://app.dimensions.ai/details/publication/pub.1041094728
168 https://doi.org/10.1007/s10792-006-9012-y
169 rdf:type schema:CreativeWork
170 sg:pub.10.1007/s11136-017-1751-x schema:sameAs https://app.dimensions.ai/details/publication/pub.1095857389
171 https://doi.org/10.1007/s11136-017-1751-x
172 rdf:type schema:CreativeWork
173 sg:pub.10.1007/s11606-014-2894-x schema:sameAs https://app.dimensions.ai/details/publication/pub.1024673878
174 https://doi.org/10.1007/s11606-014-2894-x
175 rdf:type schema:CreativeWork
176 sg:pub.10.1097/gim.0b013e3181a1ff7b schema:sameAs https://app.dimensions.ai/details/publication/pub.1003253020
177 https://doi.org/10.1097/gim.0b013e3181a1ff7b
178 rdf:type schema:CreativeWork
179 sg:pub.10.1186/s13023-015-0362-2 schema:sameAs https://app.dimensions.ai/details/publication/pub.1041535695
180 https://doi.org/10.1186/s13023-015-0362-2
181 rdf:type schema:CreativeWork
182 https://app.dimensions.ai/details/publication/pub.1077688423 schema:CreativeWork
183 https://doi.org/10.1001/archderm.138.7.909 schema:sameAs https://app.dimensions.ai/details/publication/pub.1041713671
184 rdf:type schema:CreativeWork
185 https://doi.org/10.1001/jama.2017.21903 schema:sameAs https://app.dimensions.ai/details/publication/pub.1100829981
186 rdf:type schema:CreativeWork
187 https://doi.org/10.1002/ajmg.a.33380 schema:sameAs https://app.dimensions.ai/details/publication/pub.1031491402
188 rdf:type schema:CreativeWork
189 https://doi.org/10.1002/ajmg.a.33909 schema:sameAs https://app.dimensions.ai/details/publication/pub.1051316900
190 rdf:type schema:CreativeWork
191 https://doi.org/10.1002/ajmg.a.36545 schema:sameAs https://app.dimensions.ai/details/publication/pub.1005173401
192 rdf:type schema:CreativeWork
193 https://doi.org/10.1002/ajmg.a.37365 schema:sameAs https://app.dimensions.ai/details/publication/pub.1005010730
194 rdf:type schema:CreativeWork
195 https://doi.org/10.1002/jbmr.1555 schema:sameAs https://app.dimensions.ai/details/publication/pub.1040635552
196 rdf:type schema:CreativeWork
197 https://doi.org/10.1016/j.acra.2014.05.008 schema:sameAs https://app.dimensions.ai/details/publication/pub.1012470650
198 rdf:type schema:CreativeWork
199 https://doi.org/10.1016/j.ahj.2010.11.006 schema:sameAs https://app.dimensions.ai/details/publication/pub.1048412135
200 rdf:type schema:CreativeWork
201 https://doi.org/10.1016/j.atherosclerosis.2016.08.023 schema:sameAs https://app.dimensions.ai/details/publication/pub.1026091770
202 rdf:type schema:CreativeWork
203 https://doi.org/10.1016/j.cct.2008.09.002 schema:sameAs https://app.dimensions.ai/details/publication/pub.1045002035
204 rdf:type schema:CreativeWork
205 https://doi.org/10.1016/j.cct.2012.02.012 schema:sameAs https://app.dimensions.ai/details/publication/pub.1003111252
206 rdf:type schema:CreativeWork
207 https://doi.org/10.1016/j.ejogrb.2006.09.003 schema:sameAs https://app.dimensions.ai/details/publication/pub.1017677486
208 rdf:type schema:CreativeWork
209 https://doi.org/10.1016/j.jbi.2008.08.010 schema:sameAs https://app.dimensions.ai/details/publication/pub.1039152312
210 rdf:type schema:CreativeWork
211 https://doi.org/10.1016/j.metabol.2017.06.001 schema:sameAs https://app.dimensions.ai/details/publication/pub.1085932779
212 rdf:type schema:CreativeWork
213 https://doi.org/10.1016/s0140-6736(13)61091-0 schema:sameAs https://app.dimensions.ai/details/publication/pub.1008584059
214 rdf:type schema:CreativeWork
215 https://doi.org/10.1053/ajot.2002.128037 schema:sameAs https://app.dimensions.ai/details/publication/pub.1046369974
216 rdf:type schema:CreativeWork
217 https://doi.org/10.1097/00004694-199809000-00001 schema:sameAs https://app.dimensions.ai/details/publication/pub.1060186948
218 rdf:type schema:CreativeWork
219 https://doi.org/10.1097/aud.0000000000000242 schema:sameAs https://app.dimensions.ai/details/publication/pub.1015057003
220 rdf:type schema:CreativeWork
221 https://doi.org/10.1097/maj.0b013e318158e981 schema:sameAs https://app.dimensions.ai/details/publication/pub.1041846128
222 rdf:type schema:CreativeWork
223 https://doi.org/10.1097/mcd.0b013e32836032f0 schema:sameAs https://app.dimensions.ai/details/publication/pub.1023820074
224 rdf:type schema:CreativeWork
225 https://doi.org/10.1097/mlr.0b013e31829b1d84 schema:sameAs https://app.dimensions.ai/details/publication/pub.1026300673
226 rdf:type schema:CreativeWork
227 https://doi.org/10.1097/scs.0b013e3181ecc55a schema:sameAs https://app.dimensions.ai/details/publication/pub.1046714429
228 rdf:type schema:CreativeWork
229 https://doi.org/10.1111/cge.12409 schema:sameAs https://app.dimensions.ai/details/publication/pub.1042226937
230 rdf:type schema:CreativeWork
231 https://doi.org/10.1111/j.1399-0004.1989.tb03198.x schema:sameAs https://app.dimensions.ai/details/publication/pub.1012028028
232 rdf:type schema:CreativeWork
233 https://doi.org/10.1111/j.1540-8175.2009.00973.x schema:sameAs https://app.dimensions.ai/details/publication/pub.1035917253
234 rdf:type schema:CreativeWork
235 https://doi.org/10.1111/j.1754-4505.2008.00070.x schema:sameAs https://app.dimensions.ai/details/publication/pub.1023581999
236 rdf:type schema:CreativeWork
237 https://doi.org/10.1136/jmg.16.2.101 schema:sameAs https://app.dimensions.ai/details/publication/pub.1005607842
238 rdf:type schema:CreativeWork
239 https://doi.org/10.1159/000351374 schema:sameAs https://app.dimensions.ai/details/publication/pub.1013811272
240 rdf:type schema:CreativeWork
241 https://doi.org/10.1200/jco.2013.53.5948 schema:sameAs https://app.dimensions.ai/details/publication/pub.1009488783
242 rdf:type schema:CreativeWork
243 https://doi.org/10.2106/jbjs.o.00494 schema:sameAs https://app.dimensions.ai/details/publication/pub.1068904073
244 rdf:type schema:CreativeWork
245 https://doi.org/10.2196/jmir.4736 schema:sameAs https://app.dimensions.ai/details/publication/pub.1069286336
246 rdf:type schema:CreativeWork
247 https://doi.org/10.3109/14767058.2016.1151870 schema:sameAs https://app.dimensions.ai/details/publication/pub.1027833043
248 rdf:type schema:CreativeWork
249 https://doi.org/10.3174/ajnr.a0731 schema:sameAs https://app.dimensions.ai/details/publication/pub.1050503510
250 rdf:type schema:CreativeWork
251 https://doi.org/10.3766/jaaa.19.8.3 schema:sameAs https://app.dimensions.ai/details/publication/pub.1032598647
252 rdf:type schema:CreativeWork
253 https://doi.org/10.4137/hsi.s11093 schema:sameAs https://app.dimensions.ai/details/publication/pub.1014229919
254 rdf:type schema:CreativeWork
255 https://www.grid.ac/institutes/grid.170693.a schema:alternateName University of South Florida
256 schema:name College of Medicine, University of South Florida, Tampa, Florida, USA
257 rdf:type schema:Organization
258 https://www.grid.ac/institutes/grid.239560.b schema:alternateName Children’s National Health System
259 schema:name Bone Health Program, Children’s National Health System, 111 Michigan Avenue, NW, 20010, Washington, DC, USA
260 rdf:type schema:Organization
261 https://www.grid.ac/institutes/grid.416975.8 schema:alternateName Texas Children's Hospital
262 schema:name Department of Molecular and Human Genetics, Baylor College of Medicine, Houston, TX, USA
263 Texas Children’s Hospital, Houston, TX, USA
264 rdf:type schema:Organization
265 https://www.grid.ac/institutes/grid.423291.f schema:alternateName Osteogenesis Imperfecta Foundation
266 schema:name Osteogenesis Imperfecta Foundation, Gaithersburg, MD, USA
267 rdf:type schema:Organization
 




Preview window. Press ESC to close (or click here)


...