CClinicalTrials.gg
RecruitingNCT04115774ROIUpdated Nov 20, 2025

Registry of Osteogenesis Imperfecta

An observational study in Osteogenesis Imperfecta, sponsored by Luca Sangiorgi. Recruiting at 1 site in Italy. Per ClinicalTrials.gov, last updated 2025-11-20.

Sponsored by Luca Sangiorgi · Observational

Study type
Observational
Model
Cohort
Time perspective
Other
Enrollment
5,000
Sex
All
01

Study summary

ROI is a retrospective and prospective registry, finalized for care and research purposes. It is articulated in main sections - strongly related and mutually dependent on each other - corresponding to different data domains: personal information, clinical data, genetic data, genealogical data, surgeries, etc. This approach has been developed to corroborate and integrate data from different sources evaluating several aspects of diseases and to correlate genetic background and phenotypic outcomes, in order to better investigate diseases pathophysiology. Due to legal requirements, institutional directives and organizational issues, we are unable to include individuals residing outside Italy in the registry at this time. We are currently engaged in the preparation of a recruitment process for individuals residing outside Italy.

Read the detailed description

The traditional method of collecting patient information is often chaotic, inconvenient and sometimes even unsafe, particularly when dealing with rare diseases. In 2013, the need to simplify the diagnostic process and to overcome the difficulties of data storage and analysis, led to the suggestion of implementing the Registry of Osteogenesis Imperfecta (ROI).

The ROI relies on an IT platform named Genotype-phenotype Data Integration platform - GeDI. This solution was developed through a collaboration between Rare Skeletal Disease Department and a local software company (Dilaxia) and is General Data Protection Regulation (GDPR)-compliant, multi-client and web-accessible. It has been designed according to current medical informatics standards, including the Orphanet code, the International Classification of Diseases (ICD), the Human Genome Variants Society, aiming to follow FAIR (Findability Accessibility Interoperability Reusability) principles. GeDI is continuously being implemented to improve the management of people with Osteogenesis Imperfecta and to assist researchers in analyzing the information collected. ROI is divided into the following main sections:

  • Personal data: it comprises general information, birth details and residence data;
  • Patient data: including the patients internal code, the hospital code and other patient details;
  • Diagnostic Process: the diagnosis, the status (affected, suspected, etc.), age at diagnosis, comorbidities, allergies, etc.;
  • Genogram: a tool for designing the family transmission of the disease, alongside information on the disease status of all relatives included;
  • Clinical events: it records a long list of signs and symptoms of Multiple Osteochondromas as well as several additional items to describe the disease
  • Genetic Analysis and Alteration: including analytical technique, sample information, analysis duration, etc. This section also comprises detailed information on any detected pathological variants (e.g. gene, international reference, DNA change, protein change, genomic position, etc.);
  • Visits: this section includes visit type (genetic, orthopedic, rehabilitation, pediatric, etc.), the date of the visit, prescriptions, imaging, etc.;
  • Treatments: this section comprises information of a wide range of treatments including pharmacological, devices, supplements, and other treatments such as psychological, nutritional, etc.;
  • Surgeries: this section contains information on the type of surgeries, the age of the patients, the site/localization of the procedures, etc.
  • Documents: this repository allow us to store all types of documents (radiological reports, imaging, consents, clinical reports, etc.);
  • Consents: this section provides a comprehensive overview of all consents collected, including the collection date;
  • Samples: this section includes information on the samples, like the type, date of collection, etc.;
  • PROs: this section collects information on patients reported outcomes such as the quality of life or ABC scale.
02

Conditions studied

  • Osteogenesis Imperfecta

Keywords

  • Disease Registry
  • Natural History Study
  • Disease Evolution
  • Genotype-Phenotype Correlation
03

Who can participate

Ages eligible
Child (0–17), Adult (18–64), Older adult (65+)
Sexes eligible
All
Accepts healthy volunteers
Yes
Sampling method
Probability sample

Study population

Patients affected by Osteogenesis Imperfecta. The Registry will include also data on foetuses (prenatal and abortion).

Inclusion criteria

  • All Osteogenesis Imperfecta patients, including prenatal and fetal diagnosis of Osteogenesis Imperfecta

Exclusion criteria

Exclusion Criteria:

  • Any condition unrelated to Osteogenesis Imperfecta
04

Study design

Observational model
Cohort
Time perspective
Other
Enrollment
5,000 participants (estimated)
Target follow-up
25 Years
Patient registry
Yes
Biospecimen retention
Samples with dna

Groups and cohorts

  • Osteogenesis Imperfecta patients

    The group comprises all patients affected by Osteogenesis Imperfecta, including prenatal and fetal diagnosis of Osteogenesis Imperfecta

    Drug: bisphosphonates

Interventions

  • Drugbisphosphonates

    Since this is an observational study, the investigators collect general information on bisphosphonates treatment/impact

05

What researchers measure

Primary outcomes

  1. Natural History and Epidemiology

    To maintain an established registry in order to assess epidemiology and natural history. Collection of physical examinations (severity of the disease), orthopaedics and functionals data (number of fractures, fracture sites, deafness, etc.), genetics background (target gene, type of mutation, etc.), family history (inheritance in maternal or paternal line, etc.) and treatment information (pharmacological, devices, supplements, and other treatments). Clinical, orthopedic, surgical, treatment and functional features are updated at each follow up. Clinical reports, medical charts and imaging are the primary source of data.

    Time frame: 25 years

Secondary outcomes

  1. Genotype-Phenotype Correlation

    The secondary outcome comprises the correlation between genotype and phenotype. This includes, but is not limited to clinical features and genetic background. This will be pursued using the information collected during visits and follow-ups and the genetic information resulting from molecular investigations.

    Time frame: 25 years

Other outcomes

  1. Disease evolution

    This outcome aims to investigate the evolution of Osteogenesis Imperfecta during time. This will be evaluated within the families and among the families. Main clinical features, such as height, number of fractures, bone evaluations, will be collected both retrospectively and prospectively. An evaluation of these parameters will be performed at each visit to keep track on the progression of clinical manifestations.

    Time frame: 25 years

06

Study locations

1 of 1 sites recruiting
07

References and documents

Publications

  • Ablin DS, Sane SM. Non-accidental injury: confusion with temporary brittle bone disease and mild osteogenesis imperfecta. Pediatr Radiol. 1997 Feb;27(2):111-3. doi: 10.1007/s002470050079. PubMed 9028840 ↗
  • Davie MW, Haddaway MJ. Bone mineral content and density in healthy subjects and in osteogenesis imperfecta. Arch Dis Child. 1994 Apr;70(4):331-4. doi: 10.1136/adc.70.4.331. PubMed 8185368 ↗
  • Chapman S, Hall CM. Non-accidental injury or brittle bones. Pediatr Radiol. 1997 Feb;27(2):106-10. doi: 10.1007/s002470050078. PubMed 9028839 ↗
  • Lindert U, Cabral WA, Ausavarat S, Tongkobpetch S, Ludin K, Barnes AM, Yeetong P, Weis M, Krabichler B, Srichomthong C, Makareeva EN, Janecke AR, Leikin S, Rothlisberger B, Rohrbach M, Kennerknecht I, Eyre DR, Suphapeetiporn K, Giunta C, Marini JC, Shotelersuk V. MBTPS2 mutations cause defective regulated intramembrane proteolysis in X-linked osteogenesis imperfecta. Nat Commun. 2016 Jul 6;7:11920. doi: 10.1038/ncomms11920. PubMed 27380894 ↗
  • Martin E, Shapiro JR. Osteogenesis imperfecta:epidemiology and pathophysiology. Curr Osteoporos Rep. 2007 Sep;5(3):91-7. doi: 10.1007/s11914-007-0023-z. PubMed 17925189 ↗
  • Miller ME, Hangartner TN. Temporary brittle bone disease: association with decreased fetal movement and osteopenia. Calcif Tissue Int. 1999 Feb;64(2):137-43. doi: 10.1007/s002239900592. PubMed 9914321 ↗
  • Moore MS, Minch CM, Kruse RW, Harcke HT, Jacobson L, Taylor A. The role of dual energy x-ray absorptiometry in aiding the diagnosis of pediatric osteogenesis imperfecta. Am J Orthop (Belle Mead NJ). 1998 Dec;27(12):797-801. PubMed 9880097 ↗
  • Roughley PJ, Rauch F, Glorieux FH. Osteogenesis imperfecta--clinical and molecular diversity. Eur Cell Mater. 2003 Jun 30;5:41-7; discussion 47. doi: 10.22203/ecm.v005a04. PubMed 14562271 ↗
  • Sillence DO, Senn A, Danks DM. Genetic heterogeneity in osteogenesis imperfecta. J Med Genet. 1979 Apr;16(2):101-16. doi: 10.1136/jmg.16.2.101. PubMed 458828 ↗
  • Maioli M, Gnoli M, Boarini M, Tremosini M, Zambrano A, Pedrini E, Mordenti M, Corsini S, D'Eufemia P, Versacci P, Celli M, Sangiorgi L. Genotype-phenotype correlation study in 364 osteogenesis imperfecta Italian patients. Eur J Hum Genet. 2019 Jul;27(7):1090-1100. doi: 10.1038/s41431-019-0373-x. Epub 2019 Mar 18. PubMed 30886339 ↗
  • Zionts LE, Nash JP, Rude R, Ross T, Stott NS. Bone mineral density in children with mild osteogenesis imperfecta. J Bone Joint Surg Br. 1995 Jan;77(1):143-7. PubMed 7822373 ↗
  • Hill CL, Baird WO, Walters SJ. Quality of life in children and adolescents with Osteogenesis Imperfecta: a qualitative interview based study. Health Qual Life Outcomes. 2014 Apr 16;12:54. doi: 10.1186/1477-7525-12-54. PubMed 24742068 ↗
  • Hald JD, Folkestad L, Harslof T, Brixen K, Langdahl B. Health-Related Quality of Life in Adults with Osteogenesis Imperfecta. Calcif Tissue Int. 2017 Nov;101(5):473-478. doi: 10.1007/s00223-017-0301-4. Epub 2017 Jul 4. PubMed 28676897 ↗
  • Vanz AP, van de Sande Lee J, Pinheiro B, Zambrano M, Brizola E, da Rocha NS, Schwartz IVD, de Souza Pires MM, Felix TM. Health-related quality of life of children and adolescents with osteogenesis imperfecta: a cross-sectional study using PedsQL. BMC Pediatr. 2018 Mar 2;18(1):95. doi: 10.1186/s12887-018-1077-z. PubMed 29499676 ↗
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Registry details

Key details

Study ID
NCT04115774
Lead sponsor
Luca Sangiorgi
Responsible party
Luca Sangiorgi (Head of Departement of Rare Skeletal Disorders, Istituto Ortopedico Rizzoli) — Sponsor-investigator
First posted
Oct 4, 2019
Start date
Jun 28, 2013
Primary completion
Jul 2018
Completion
Feb 29, 2032 (estimated)
Last update
Nov 20, 2025

Study contacts

Marina Mordenti, PhD
Contact
registri.malattierare@ior.it
+39 05 6366062
Marcella Lanza, PhD
Contact
registri.malattierare@ior.it
+39 05 6366169
Luca Sangiorgi, PhD
principal investigator · Istituto Ortopedico Rizzoli

Oversight

Data monitoring committee
No
FDA-regulated drug
No
FDA-regulated device
No
View the source record on ClinicalTrials.gov ↗

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