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Trial registered on ANZCTR


Registration number
ACTRN12619000445178
Ethics application status
Not required
Date submitted
8/03/2019
Date registered
18/03/2019
Date last updated
18/03/2019
Date data sharing statement initially provided
18/03/2019
Date results information initially provided
18/03/2019
Type of registration
Retrospectively registered

Titles & IDs
Public title
Clinical outcomes of an inflammatory bone disorder that may be associated with skin disorders.
Scientific title
Long-term clinical outcomes in Synovitis Acne Pustulosis Hyperostosis and Osteitis syndrome
Secondary ID [1] 297442 0
None
Universal Trial Number (UTN)
U1111-1228-8056
Trial acronym
SAPHO
Linked study record

Health condition
Health condition(s) or problem(s) studied:
Synovitis Acne Pustulosis Hyperostosis and Osteitis syndrome 311623 0
Condition category
Condition code
Inflammatory and Immune System 310247 310247 0 0
Autoimmune diseases
Musculoskeletal 310574 310574 0 0
Other muscular and skeletal disorders

Intervention/exposure
Study type
Observational
Patient registry
False
Target follow-up duration
Target follow-up type
Description of intervention(s) / exposure
The clinical features and treatment outcomes in a cohort of 21 patients diagnosed with Synovitis, Acne, Pustulosis, Hyperostosis and Osteitis or SAPHO syndrome, in Western Australia were reviewed retrospectively over a 32-year period by chart review and electronic medical record interrogation.
No involvement or consent was required. This was a retrospective data audit .
Intervention code [1] 313698 0
Not applicable
Comparator / control treatment
No control group
Control group
Uncontrolled

Outcomes
Primary outcome [1] 319136 0

Primary outcome 1 (composite outcome): describe the clinical and treatment outcomes as well as the natural history of SAPHO in a cohort of patients as assessed by data linkage to medical records
Timepoint [1] 319136 0
Retrospectively followed for up to 32 years
Secondary outcome [1] 367070 0
The clinical effect of tumour necrosis factor inhibitor (TNFi) on SAPHO as assessed by exposure to TNFi using data linkage to medical records.
Timepoint [1] 367070 0
Retrospectively followed for up to 32 years

Eligibility
Key inclusion criteria
Diagnosis of SAPHO as defined as multifocal osteitis with/without skin symptoms, sterile acute or chronic joint inflammation with either palmoplantar pustulosis or psoriasis, or acne or hidradenitis, with sterile osteitis with any one of these sets of criteria deemed sufficient for diagnosis.
Minimum age
18 Years
Maximum age
No limit
Gender
Both males and females
Can healthy volunteers participate?
No
Key exclusion criteria
Those people that did not meet the criteria required to be diagnosed with SAPHO (multifocal osteitis with/without skin symptoms, sterile acute or chronic joint inflammation with either palmoplantar pustulosis or psoriasis, or acne or hidradenitis, with sterile osteitis with any one of these sets of criteria).

Study design
Purpose
Screening
Duration
Longitudinal
Selection
Defined population
Timing
Retrospective
Statistical methods / analysis

Recruitment
Recruitment status
Completed
Date of first participant enrolment
Anticipated
Actual
Date of last participant enrolment
Anticipated
Actual
Date of last data collection
Anticipated
Actual
Sample size
Target
Accrual to date
Final
Recruitment in Australia
Recruitment state(s)
WA
Recruitment hospital [1] 13180 0
Fiona Stanley Hospital - Murdoch
Recruitment hospital [2] 13181 0
Fremantle Hospital and Health Service - Fremantle
Recruitment hospital [3] 13182 0
Sir Charles Gairdner Hospital - Nedlands
Recruitment hospital [4] 13183 0
Royal Perth Hospital - Perth
Recruitment postcode(s) [1] 25735 0
6150 - Murdoch
Recruitment postcode(s) [2] 25736 0
6160 - Fremantle
Recruitment postcode(s) [3] 25737 0
6009 - Nedlands
Recruitment postcode(s) [4] 25738 0
6000 - Perth

Funding & Sponsors
Funding source category [1] 302014 0
Self funded/Unfunded
Name [1] 302014 0
Address [1] 302014 0
Country [1] 302014 0
Primary sponsor type
Individual
Name
Graeme Carroll
Address
ArthroCare
19A Guildford Road,
Mt Lawley
WA 6050
Country
Australia
Secondary sponsor category [1] 301799 0
None
Name [1] 301799 0
Address [1] 301799 0
Country [1] 301799 0

Ethics approval
Ethics application status
Not required

Summary
Brief summary
Synovitis, Acne, Pustulosis, Hyperostosis and Osteitis or SAPHO syndrome, is a rare immuno-inflammatory bone disease that may be associated with skin changes. There are no clear guidelines for treatment. To assess the outcome of previously observed therapeutic treatments in SAPHO, we examined outcomes in 21 patients collected over more than 30 years of clinical experience. Mostly good or very good long-term treatment outcomes were observed in older bisphosphonate medications and newer biologic treatment called tumour necrosis factor inhibitor (TNFi) treatment.
Trial website
Trial related presentations / publications
Public notes
This project was registered and approved as a quality assurance project (QA26150) by Fiona Stanley Hospital Human Research Ethics Committee on 19 of March 2018 and, as such, did not require formal Human Research Ethics Committee Review.

Contacts
Principal investigator
Name 91070 0
A/Prof Graeme Carroll
Address 91070 0
ArthroCare
19A Guildford Rd
Mount Lawley
WA 6050
Country 91070 0
Australia
Phone 91070 0
+61 08 92716306
Fax 91070 0
+61 08 9370 3957
Email 91070 0
gjcarrollmd@gmail.com
Contact person for public queries
Name 91071 0
A/Prof Graeme Carroll
Address 91071 0
ArthroCare
19A Guildford Rd
Mount Lawley
WA 6050
Country 91071 0
Australia
Phone 91071 0
+61 08 92716306
Fax 91071 0
+61 08 9370 3957
Email 91071 0
gjcarrollmd@gmail.com
Contact person for scientific queries
Name 91072 0
A/Prof Graeme Carroll
Address 91072 0
ArthroCare
19A Guildford Rd
Mount Lawley
WA 6050
Country 91072 0
Australia
Phone 91072 0
+61 08 92716306
Fax 91072 0
+61 08 9370 3957
Email 91072 0
gjcarrollmd@gmail.com

Data sharing statement
Will individual participant data (IPD) for this trial be available (including data dictionaries)?
Yes
What data in particular will be shared?
All of the individual participant data collected during the trial, after de-identification; individual participant data underlying published results only.
When will data be available (start and end dates)?
Immediately following publication, no end date.
Available to whom?
Researchers who provide a methodologically sound proposal, case-by-case basis at the discretion of Primary Sponsor
Available for what types of analyses?
For further research into SAPHO; only to achieve the aims in the approved proposal; for IPD meta-analyses.
How or where can data be obtained?
Access subject to approvals by Principal Investigator, requirement to sign data access agreement
What supporting documents are/will be available?
No other documents available
Summary results
Have study results been published in a peer-reviewed journal?
No
Other publications
Have study results been made publicly available in another format?
Yes
Other publication details
Citation type [1] 1464 0
Conference poster
Citation/DOI/link/details [1] 1464 0
Presented at the Australasian College of Dermatologists Scientific Meeting, Gold Coast, May 2018
Attachments [1] 1464 0
Results – plain English summary
This report illustrates the broad spectrum of disease presentations and severity in SAPHO. It also illustrates the disease progression over time and the responses to a variety of treatments or, in three cases, with no treatment. Mostly good or very good long-term treatment outcomes were observed in disease modifying agents (DMARDs) and biologic treatments. Suboptimal treatment may be associated with poorer clinical outcomes and greater skeletal damage.
Just over half the participants studied were treated with biologic agents. Nine of 12 (75%) had a good primary response, extending for up to 14 years. It was concluded that TNFi therapy is mostly effective and moderately sustainable.
The study has several limitations. The numbers of participants are small; most patients studies were Caucasian and therefore the results may not be generalizable to the wider population.