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| Name | Class |
|---|---|
| Bayer | INDUSTRY |
| Rush University Medical Center | OTHER |
| Phoenix Children's Hospital | OTHER |
| The University of Texas Health Science Center, Houston |
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The original Joint Outcome Study (JOS) enrolled 65 boys with hemophilia from 16 sites nationally. The subjects were randomized to one of two arms (prophylaxis or an enhanced episode-based treatment)and were followed prospectively until the age of six. At the age of six, the proportion of children on each treatment arm who developed bone or cartilage damage as determined by X-Ray or MRI was assessed. In addition, the function and structure of the index joints (defined as knees, ankles, and elbows)were evaluated using a physical assessment scale specially designed for preschool children.
The specific aim of the Joint Outcome Study Continuation (JOSC) is to extend observations of the children participating in the original JOS until the subjects reach the age of 18 years in order to determine the natural history of joint development in hemophilia and the impact of primary or secondary prophylaxis on the prevention, limitation, or reversal of hemophilic arthropathy. In addition, plasma and DNA will be collected and banked yearly for current and future studies of biomarkers and predictors of hemophilia outcomes.
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| Measure | Description | Time Frame |
|---|---|---|
| Determine the natuaral history of joint development in hemophilia and the impact of primary or secondary prophylaxis on the prevention, limitation, or reversal of hemophilic arthropathy. | Enrollment, age 14, and study exit at age 18 |
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Inclusion Criteria:
Exclusion Criteria:
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Study population will be invited based on their enrollment in the original Joint Outcome Study (JOS). This population
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| Name | Affiliation | Role |
|---|---|---|
| Marilyn Manco-Johnson, MD | University of Colorado at Denver Health and Sciences Center | Principal Investigator |
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| PubMed Identifier | Type | Citation | Retractions |
|---|---|---|---|
| 32492157 | Derived | Warren BB, Thornhill D, Stein J, Fadell M, Ingram JD, Funk S, Norton KL, Lane HD, Bennett CM, Dunn A, Recht M, Shapiro A, Manco-Johnson MJ. Young adult outcomes of childhood prophylaxis for severe hemophilia A: results of the Joint Outcome Continuation Study. Blood Adv. 2020 Jun 9;4(11):2451-2459. doi: 10.1182/bloodadvances.2019001311. |
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| ID | Term |
|---|---|
| D006467 | Hemophilia A |
| D004194 | Disease |
| D007592 | Joint Diseases |
| ID | Term |
|---|---|
| D025861 | Blood Coagulation Disorders, Inherited |
| D001778 | Blood Coagulation Disorders |
| D006402 | Hematologic Diseases |
| D006425 | Hemic and Lymphatic Diseases |
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| OTHER |
| Oregon Health and Science University | OTHER |
| Emory University | OTHER |
| University of Texas Southwestern Medical Center | OTHER |
| Prisma Health-Midlands | OTHER |
| Indiana University | OTHER |
| Intermountain Health Care, Inc. | OTHER |
| Ann & Robert H Lurie Children's Hospital of Chicago | OTHER |
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Whole Blood - EDTA, Citrate Plasma, and DNA
| D020147 | Coagulation Protein Disorders |
| D006474 | Hemorrhagic Disorders |
| D030342 | Genetic Diseases, Inborn |
| D009358 | Congenital, Hereditary, and Neonatal Diseases and Abnormalities |
| D010335 | Pathologic Processes |
| D013568 | Pathological Conditions, Signs and Symptoms |
| D009140 | Musculoskeletal Diseases |