Notes from the 2006 Children’s Arthritis and Rheumatology Research Alliance (CARRA) Annual Scientific Meeting

 

Denver, CO USA

May 5-7, 2006                      

Reporter: Charles Spencer

 

Nature Clinical Rheumatology seeking articles for pediatric rheumatology including clinical rheum, case reports, and series and commentaries.

 

I Keynote speaker: Genomics of juvenile arthritis

David Glass

 

A. Basics

JIA has a female predominance, family history of autoimmunity, predominance of some T cell clones as well as some HLA associations. The paradox is that most families with a JIA child usually do not have any other children with JIA despite the HLA linkage noted; so JIA must be inherited as a complex genetic trait inheritance as seen in other chronic diseases such as asthma and diabetes.

How many genes might be involved? Dr. Glass suggests that 3-10 per trait is reasonable. Modest non-HLA odds ratios are in the range of 1.2-2.0 contrasting to odds ratio obtained in diseases with definite HLA associations which are >3 and often up to 10. Low odds ratio gives us major power problems in JIA studies.
Two approaches are available:

1)  Candidate gene approach by testing for a limited number of polymorphisms. It’s possible to look at pathologic mechanisms of other diseases and then can come up with 2000 possible genes this way, e.g., gene polymorphisms for IgA deficiency. It’s not a hopeless search as there are general autoimmune disease genes that have been discovered, e.g., PTPN22 1p13.2, an intracellular protein tyrosine phosphate which is seen in rheumatoid factor positive arthritis in adults. The general autoimmune gene MIF 22qqq.23 is a mediator of inflammation. CTAL4 also appears to be a general autoimmune gene. Low odd ratios bedevil these genes.

2)  A second approach is a global genome approach genome-wide and utilizes

haplotype or linkage disequilibrium. Five hundred thousand polymorphisms can

cover 90% of the haplotype in the genome and the human genome has 3 x109

haplotype pairs. “SNPs” can be used to characterize small pieces of the genome.

Functional genomics is also being pursued and an integrative approach also has

great potential.

 

B. Linkage: Following inheritance polymorphic patterns

Unfortunately no large families with many children with JIA have been identified and so we cannot use the linkage approach with multiple family members. An alternative is to use allele sharing and affected sib pairs. It is less sensitive than the linkage studies in large families with many affected members.

One thousand sib pairs will be needed and the Cincinnati center now has 300-400 sib pairs. Linkage studies, though, give you relatively low localization. These studies are done with microsatellites. There are chromosome regions with linkage scores of 2 or greater, e.g., 7q 11 (3.47 log score), 2p25 (6 log score), and several others with lower scores. JIA patterns differ by subtype and share areas of genes with other autoimmune diseases. We have a way to go as genes #’s in each chromosome region can be 50-500.    The use of the HLA microsatellites may help lower this number. The microsatellites have been useful in polyarticular JIA patients where peaks have been demonstrated for HLA-DR. Chromosome 7 has a peak for early-onset poly JIA. With transmission disequilibrium testing, the number of genes is down to 3-4!  This effort has not yet found similar peaks in systemic JIA children.

 

 

C. Gene-wide association studies using case/control studies

A considerable number of patients are needed for this approach; You need a SNP map and one is now available for the genome with a SNP for every 5-6 genes; 2 chipsets that cover most of the genome will be soon available. SNPs dinucleotide variants and

SNPs per genes are analyzed.  cSNPs in coding region are a crucial area to look at searching for non-synonymous cSNPs with an amino acid change. It’s also important to look for SNPs in promoter and regulatory regions. Two companies are developing these chips now and are in a stiff competition.

The Cincinnati center has 1397 DNA samples available from JIA patients but needs more for each JIA subtype to obtain sufficient power. Even with big numbers and high speed computing power, there are too many false positives (Type 1 error). To eliminate these false positives, it is important to consider excluding and eliminating problematic cells, set high significant levels (Be aware that this will decrease the power), use cluster of SNPs, follow good genomic logic and use areas previously positive in this research. Overall results and expression levels should make biological sense. Compare 400 SNPs from JIA to 400 SNP’s from another disease and add a case control element. Then you could use family studies to follow up.

 

D. Environmental influences in JIA-e.g., smoking

No clusters have been noted yet.  Altered family structure has been reported in early childhood onset JIA particularly. Mothers with autoimmune diseases or in families with much autoimmunity often have difficulties in pregnancy. Consider the autoimmune environment during pregnancy. One approach was to check reproductive fitness of JIA mothers versus best friends using a reproductive questionnaire on 227 JRA patients with 796 pregnancies and similar of pregnancies for best friends. JIA mothers had no problems conceiving but had more problems with miscarriages, preterm delivery and still births

Thus one should compare two family groups having a similar number of children.

 

Hypothesis:

Stage I: Intrauterine inflammation (Th1 basis) associated with increased numbers of pregnancy complications and increased mediators of inflammation.

Stage II Maternal inflammatory mediators pass via cord blood into the fetus resulting in increased fetal Th1 level as well as the presence of susceptibility JIA genes.

 

E. Methotrexate pharmocogenomics

Pilot SNP study involves separating JIA DNA into pools of methotrexate responders and nonresponders and then evaluates the SNP’s. Then it should be possible to separate out top 10 SNP’s findings Then it might eventually be feasible to have a SNP test that will evaluate whether your patient will respond to methotrexate or Enbrel or other drugs.

In conclusion, presently there are extraordinary opportunities to explore the JIA genome and determine why a child might get JIA, whether a child will do well, and whether a child will do well on a particular drug.

 

 

II Reports from the Subcommittees of CARRA

 

A. JIA and spondyloarthropathy subcommittee

1.   RFA for uveitis-several proposals sent in; One was chosen from Duke that is a Multicenter Prospective Registry of Infliximab Use in Uveitis. It will use a Delphi questionnaire and look at an important question, the treatment of uveitis-Deborah Levy and Egla Rabinovich will head up the study.  The hypothesis is that infliximab is effective for the treatment of uveitis. The gold standard would be a placebo trial but not feasible ethically with this serious eye involvement. This study has reviewed retrospective data on 35 patients. This approach has its strengths but also some weaknesses. There was a lack of a standardized regimen with a standardized dose, schedule, and outcome measures.


          The study will assess the ability of infliximab to treat uveitis using different parameters. It will compare

efficacy between two doses of rituximab: 5 mg/kg/dose and 10 mg mg/kg/dose, both every 4 weeks. The study

will utilize a web-based, prospective collection of data. A child must to have been determined to need infliximab before treatment. The study will attempt to standardize treatment using standard ophthalmologic techniques.

These techniques include use of a recent classification of uveitis (SUN) based upon anatomy. There is a SUN grading system of flare as well as a SUN uveitis activity scale of worsening, stable, and improving.

     Ten to 12 CARRA centers will be sought. Each center must be motivated and reliable. Each center must have an excellent collaborative relationship with their pediatric ophthalmologists. The goal is for each center to recruit 4 patients over 6 month’s entry period.

     Inclusion criteria include: persistent non-infectious JIA uveitis, onset < 16 years of age, present for 3 months and already treated appropriately. The anterior chamber should have at least 1+uveitis by SUN criteria; The child needs to have failed conventional ophthalmologic drops and at least 1 DMARD. Any methotrexate dose must be stable. No intraocular steroid injection or surgery is allowed within 6 weeks of entry.

Exclusion criteria include: No biological drug utilized in current treatment program and must have been off Enbrel or Humira for >60 days.

 

          SUN criteria will be used. Methotrexate dose may be 0.5-1.0 mg/kg/day with a stable dose for > 6 weeks. Centers will be randomized for 5 or 10 mg/kg of infliximab with an escalating dose scheme. Infliximab will not be tapered until a low dose methotrexate is obtained and only 1 ophthalmologic eye drop medication is being utilized. Outcome measures will include: Time to improvement, time to relapse, rate of disease flare, and rate of taper. The schedule is: 0-4 month’s selection of sites and IRB, 4-10 months for entry start, then 12-18 months to finish.  The study will pay $1000 for IRB submission and $100 per patient visit sheet returned.

 

B. Long-term JIA outcome-Sue Bowyer

Presently Cohort 1 has 347 patients. They are mostly from the middle to the late period of the use of methotrexate (1980’s-1990’s), some even in the time of early use of methotrexate. Cohort 2 has a goal of 500 children that will cover the era of early use of methotrexate and biologics singly or in combination. Parents are filling out CHAQ, global assessment, etc., medications used, side effects of medications, PT/OT, and hospital admissions. Barriers to care are now being collected including referral source, specialists seen before, insurance problems, etc.

Yet new Changes being considered in JRA outcome study Some of these changes include quality of life measure, a modified CHAQ (compare to previous CHAQ), repeat RF level, collect remission criteria and test criteria, and possible collaboration with DNA collection of David Glass (but no $$ yet)

 

C. Dermatomyositis-Brian Feldman

 The JDM priorities voted on last year include:

1) treatment for new patients

Our plan-Survey of current practice-medications, other therapies, evaluation, and how long to treat. A consensus practice conference will be held. The survey will be reviewed and 2-3 treatment protocols will be developed. The funding will be provided by Friends of CARRA. Cases have been written and reviewed-survey to be written; survey will be send out soon.

            2) REM trial for JDM and JDM/PM with refractory disease

It will involve validation of disease activity measures that have been developed as well as a basic science study of responders and non-responders. The primary aim is the efficacy of treatment while the secondary aim remains the science, both clinical and basic.

In the REM trial, the patients will be randomized into 2 groups based on different doses of infliximab. Basic science studies include B cell studies, differences in immunophenotypic characteristics, Fc receptor polymorphism, and HLA associations and polymorphisms.

            3) IMACS-Ingrid Lundberg

This project involves several aspects including a classification study of JDM, a Twin Sib study, and a Canadian IMACs registry. See more information on the IMACS website.

            4) Ann Reeds lab-

Dendritic cells and pathogenesis, gene profiling in JDM, and T cell repertoire work.

            5) Lauren Pachman: Biology of calcification-Dr. Pachman needs Ca++  specimens.

            6) European PRINTO is doing a three limb treatment study for newly diagnosed JDM

    comparing prednisone alone, pred + cyclosporine, and prednisone + methotrexate.

    CARRA has not given approval for participation of US centers in this study and

    PRINTO will not enroll US centers without a coordinated approval by CARRA.

            7) PReS European JDM network in participating in a classification of overlap

                  syndrome project.

 

D. Lupus studies-Lupus Committee Project Update Laura Schaunberg

  1. Clinical: Hermine Brunner-4 studies approved

a)     Meaning Outcomes-objective is to find minimally clinical important differences of disease activity measures-also the validity of SLE criteria for children and the validity of outcome measures.

b)     Renal biomarkers-NGAL Neutrophil gelatonaise

c)      Development of evidence-based use of steroids in cSLE

as well the use of steroids effect on outcome of cSLE.

d)     Effect of hormonal changes in adolescents with cSLE on disease and antibodies levels

      2.  Clinical Science-not approved

         a) Helping educate lupus patients-Larry Vogler

                             b) Lupus Registry in Georgia

                             c) Tripoelin in protecting ovaries in patients with SLE

      3.  Basic Research-approved

         a) Nitric oxide in cSLE-another researcher

                             b) Maternal microchimerism in cSLE-Ann Reed

      4.  Basic Science-not improved-multiple studies-see CARRA website

 

       5. Ongoing studies-APPLE

This study is testing the efficacy of atorvastatin (Lipitor) in preventing atherosclerosis in cSLE over 3 years. It is a placebo controlled study. The secondary goals are to assess Limiter’s anti-inflammatory effect on SLE and to assess the safety of Lipitor. There have been 24 study centers involved in the study but 2 centers have terminated participation. Many study problems have occurred involving institutional approval. This has occurred at different levels including at Duke, FDA, NIH, and other centers. Prevention trials are difficult to sell. Having said that, a number of study centers have had unanticipated problems. It has even taken 36 months to get final activation of one site!

So far 22 sites have been activated with 2 sites terminated. Enrollment began in Sept 2003 and since then 143 patients have been randomized, 163 consented (169 with verbal consents). The goal of enrollment is 280 patients. The current timeline is to consent 180 by 6/2006 and finish all entry by the end of 2006.

About ½ of approached cSLE patients have been successfully enrolled. There has been only a 4% dropout rate. Compliance (defined as missing less than 25% of doses) has thus far been excellent at 90%.

APPLE adverse events include 24 SAE's (serious adverse events).  The SAE’s involve a lupus flares (5 patients), infections (7), related complications (3), e.g., MAS or enteritis, and liver enzymes or muscle enzymes elevation (9).

What have we learned?

a) Enthusiasm is necessary but not sufficient

b) Sites need enough money

c) Sites need outside support both doing things for sites and being available for questions.

d) Sites need education of staff.

e) Sites need personal attention.

f)  RCT’s (randomized control trials) need longer start up as there are multiple regulatory groups         

    (NIAMS, FDA, OMB, DSMB) and multiple IRB’s to deal with. A site visit with the sponsor can  

    help.

g) Data collected;

 

1) The mean for IRB approval was 25 weeks! (0-Duke-to 85 weeks)

            2) The mean time to IMT certification of scan technicians for a site was 37 weeks (11-157)

            3) Site activation mean was 63 weeks (25-152 weeks)

            4) Even after site approved, the time to enrollment of the first patient was a mean of 12 weeks.

 

How can we optimize recruitment? The role of the patient’s physician (rheumatologist) appears to be critical. Make sure it’s clear whose patient it is and have the primary pediatric rheumatologist initiate the approach to the patient and family.

The importance of PI cannot be over-emphasized. The PI must be very active in the study from the beginning and remain so and not leave it all to the clinical coordinator.

The overall belief system that research is good is important and that this particular trial is valuable and worth doing is essential. Once these beliefs are in place, effectiveness in recruitment can be taught.

Additional strategies might include:

  • Repeat approaches to a patient can be successful.
  • Review consent face to face and let the patient and family review it at home.
  • Monthly site calls may help.
  • Individual site action plans may be useful.
  • Study PI and NIAMS visiting sites may also help.
  • Increase site payments may increase the likelihood of a study’s success with coverage of the local study coordinator cost and patient transportation.
  • Work on retention strategies

 

From the study PI’s perspective, it might be best to focus on the best clinical study sites with the best track record. Who can provide your most potential patients and who will follow through well?

 

In summary, the Apple study has accomplished some things and learned much. Look at the study data available on CARRA website. It includes a well developed and refined CRF. The study has established site activation benchmarks. We have much data on cSLE and the most experience on Lipitor in children for any disease. In the next year Apple enrollment will be ended. A shared data base will be available. Several other lupus studies are nearing publication; and several other accomplishments are noteworthy. Please see the CARRA website.

 

E. Vasculitis/Scleroderma/autoinflammatory disease subcommittee-Kathleen O’Neil

1. Scleroderma-Gloria Higgins

a) CARRA is currently indigitating with International Inception Juvenile Systemic Sclerosis Cohort Study.

b) Investigation of topical alprostadil for severe Raynaud’s phenomenon.

c) COMPAQ-1H severe immunosuppressive study-Andy Reiff

d) Vasculitis registries have been started including-Wegner's Registry, CNS Vasculitis

e) Autoinflammatory diseases-FMF meeting occurred in Bethesda in 2005

f)  Registry for autoinflammatory diseases has begun.

 

2. Juvenile systemic sclerosis cohort study:

            a) Disease for <18 months.

            b) Collect data q 6 months for 36 months: skin and organ involvement, lab results, quality of life

              measured; Note: Patient # will be given for confidentiality

c) Chronic vasculitis registry-David Cabral

d) Case report forms are being developed.

e) Diseases are rare!-Only 64 Wegener’s and 327 unspecified vasculitis patients have been entered in

    the registry so far.

Why important?

High disease burden occurs and there is limited pediatric specific data available. Why do it? Because of our belief in the need for and value of hypothesis driven research.

The vasculitis registry needs sufficient resources!!! Is it feasible even if this registry remains unfunded by CARRA and non-CARRA organizations? Yes, it is feasible! But start simple with Wegener’s granulomatosis, Microscopic angiitis, Churg-Stauss syndrome, and CNS vasculitis syndromes. Data Registry on internet; once at diagnosis only-many benefits, e.g., be able to test formation and function of database and web system; It will provide a framework for expansion of the registry and other benefits.

 

Things to be done: Obtain more centers and help with IRB applications with suggested consents. Time frame for launching is 3-4 months. There is a plan for followup studies.

 

2. CNS vasculitis-Sue Benseler-

As mentioned above and previously at last year meeting, CARRA has envisioned developing a framework for prospective network studies of pediatric rheumatologic diseases. Some networks that may serve as a model for ours might include two existing research framework of pediatric neurology: International Stroke registry (1000 children) and a Canadian study of pediatric demyelinating diseases; This study allows for multiple visits.

Features of this CNS vasculitis network will include:

1)     A secure web based interactive repository which is Oracle-based and easy to use.

2)     A database developed in collaboration with Computer Biology at Sick Kids Hospital in Toronto.

3)     Project manager: Stacy Ou Wai who will have 2 programmers to help.

4)     The browser will be hosted at Sick Kids.

     5)  Two CNS vasculitis studies are planned

-CNS vasculitis prospective outcome study-test monitoring and outcome tools,   

            predictors of adverse outcome-see website

 

Work to be done: organization at this meeting for the startup-discuss data collection forms and overall functionality of study databases; discuss and tackle all challenges around the organization; finalize the projects, so that the programmers can start. Fortunately, there are some startup funds available.

 

F. Pain and outcome and quality of life-Gary Walco

Introduction: Questions to be discussed: Should this group continue at all? The group is small and many of the participants have roles on other committees. You could also argue that pain not a disease. Some good things to mention: Members of the pain group have a major interest in pain and QOL. It would appear likely focus on pain within CARRA would be diminished if the group was disbanded. The idiopathic pain focus would be lost.

It has been decided to continue.

 

1. Studies

  • Methadone study in JIA for chronic pain.
  • It is organized out of Hackensack University Medical Center and Duke University.
  • Four patients have been entered so far.

2. Idiopathic pain syndromes

a. Musculoskeletal pain registry

Last CARRA meeting there was much discussion on beginning a longitudinal, prospective observation study of all children presenting with chronic or recurrent musculoskeletal pain in order to generate regression models. This would be a registry. This project is still under discussion.

b. Taxonomy

This is a joint endeavor of CARRA and the American Pain Society Special Interest Group on Pain in Infants, Children, and Adolescents. A summit is planned for 2007.

The planning meeting for the summit in Vancouver June 2006. This planning meeting is in coordination with the Committee on Taxonomy of the IASP.

Funding sources being pursued.

c. Other studies in progress: Jennifer Weiss-Analgesia for joint injections; P Malleson, Yuki Kimura, and J Weiss are doing a pain survey; Fibromyalgia studies begun in Cincinnati including sleep patterns in pain syndrome children and the relationship of celiac disease and pain syndromes.

 

III Committee Concurrent Sessions-JDM-Meeting #1 Brian Feldman

  1. Goals of these two meeting:
    1. Discuss scientific priorities of subcommittee and CARRA in JDM.

     2.  Pick top priorities of this subcommittee

This effort will include addressing the most commonly seen JDM as well as the most severe forms (e.g., the JDM ICU patient). It will leverage current technology and scientific progress. It will require multicenter collaboration. It will be pediatric focused.

Recall some of last CARRA meetings JDM subcommittee priorities (not complete list):

  • Original Treatment protocols for new onset and resistant disease.
  • Validate new and evolving measures of disease activity and damage.
  • Identify genetic and environmental disease susceptibility factors
  • Define diagnostic criteria and criteria for flare, remission and course
  • Develop predictors of disease course

 

Recall the ranking list-top 8-Revisit every 2 years

  • Treatment protocol for new patients
  • Treatment for resistant disease-REM addressing this question, at least with Remicade.
  • Criteria flare activity measures, remission;
  • Determination of best markers of disease activity.
  • Correlation with JDM outcome muscle biopsy and MRI.
  • Prospective registry
  • Prevention and treatment of complications-osteoporosis, lipodystrophy, calcinosis
  • Genomics, pharmacogenetics

 

A survey will be sent out soon of 11 cases of JDM. It has been reviewed by multiple committee members. It includes an ICU case. Hopefully it will prove to be a very user- friendly survey. Then a conference for development of consensus treatment protocols will be organized. Four protocols will be created and then ranked in priority. The results of the protocol development conference will be reviewed by the individual JDM results subcommittee. The time line will be: Cases to Brian in next two weeks; Develop web survey with cases by July 1; Send out surveys and analyze over 3-6 months; By January 1, 2007, Brian will choose a working group to review the survey; A conference will then be organized (but not have occurred) by the next CARRA.

 

Juvenile dermatomyositis subcommmittee-2nd meeting. Brian Feldman

 

What additional studies should be done now that we have one study (survey and development of 4 treatment protocols) ongoing?

Let’s review the priorities:

The NIH study for adults and children (Ingrid Lundberg’s study) may help us with classification and criteria; Everyone should read her protocol and decide if want to participate.

PRINTO is working with JDM activity and damage indices.

 

Brainstorming sessions next occurred on ideas for the next priorities: Three groups of 8-12 subcommittee members.

 

1. Group A Priorities:

  • How to treat retractable skin disease?
  • Survey on use of Biologics
  • Survey on initial assessment of JDM
  • Survey on evaluation of bone density and bone health
  • Survey on regimens of tapering methotrexate and prednisone
  • Prognostic subsets within JDM patients
  • What are remission criteria?

 

2. Group B Priorities:

  • Mild disease-mild treatment or none at all.
  • How to treat resistant disease and how to diminish steroid side effects?
  • Does Methotrexate work? When does it work?
  • What to add next to steroids if steroids insufficient or too toxic-What’s the best sequence of second line agents?
  • Define good and bad responses/outcome-Does Biopsy and MRI help with prediction?
  • What are best diagnostic criteria now? We’ve been using Bohan and Peter for a long time.

 

Group C

  • Brief retrospective study of 5-10 year patients of JDM/center with data collection of course data.
  • Analyze data of retrospective study.
  • Begin prospective outcome study using results of retrospective study.
  • Physical therapy study on efficacy and how effects outcome.

 

Group Consensus in 2nd meeting

  • Do first survey/consensus working group/3-4 treatment proposals/devise the study-major work; Not rigid randomized control study. NIH and CARRA funding to be sought
  • Outcomes-predictors-What to study?-A retrospective study (chart review), then start a prospective study will be considered-deferred.
  • Survey of tapering medication using case histories.
  • Make data between clinical protocols and retro/prospective course study interactive.
  • Global goal, perhaps too ambitious, would be to combine results of treatment protocols and course/prognostic indicators to arrive at a beginning treatment protocols for each of several types of presentations of JDM in a hematology/oncology protocol paradigm.

 

Pain syndromes Committee

 

What study to do? Much to discuss. Perhaps a prospective study on pain syndrome patients should be performed. Consider also a naturalistic study using a registry that collects all the data such as temperament, early pain experience, quality of life, taxonomy. How to fund?

 

We could develop a denominator by doing a brief survey of generalists’ exposure to chronic msk pain or could you use Finnish data instead as the denominator.

Hypotheses to be studied might include:

-Frequency of idiopathic msk pain in primary care setting.

-Socioeconomic factors in development of pain syndromes.

 

Should we collect data by naturalistic study or intervention program or both?

It could be make easier for the physician team with the patient and family filling out the registry or study forms. It may be best to have it computer-based. Should the target population for a study be all children with different types of musculoskeletal pain or just those with idiopathic pain?

 

Who can do this work and how can it be paid for? The NIH is interested in observational studies, outcome measures, and statistical measures as they apply to pain. Thus it is a surprisingly good atmosphere for funding for pain in children. A small workshop may further this along, perhaps in 2007 with big meeting funded by NIH.

 

Pain Subcommittee-2nd meeting

Again, what are we interested in studying?

Use a model of the useful lupus study group-answer one question and get it going.

e.g., taxonomy and classification-classification schema, and data, then biomarkers with treatment intervention; Have the NIH or a drug company support a conference to set up the study initially.

 

First, establish a Web-based data registry or study. Plan a conference to set up a questionnaire for web-based registry and then establish the registry in the next 18 months.

Next pilot the registry.  Second, tie in a study limb on biomarkers and cytokines for pain sensitivity or pain response as well as genetics.  Again, initial funding could be through the NIH or a drug company. In two years ready to go for a RO-1 with biology and intervention portions.

 

The plan is to set up a website on the CARRA website and link it to SharePoint or similar commercial site; The entire subcommittee should then together put together a form by e-mail. The versions of the form can be debated over the internet with everyone posting comments or questions on CARRA website. Each subcommittee member would fill out the form on 20 patients each. Deadline: ACR and CARRA meeting there in November.  In the meantime subcommittee members research questions and literature of our field as well as a large pain literature that we may not all be aware of.

 

Peter Malleson will send out e-mail to ask for input on the website; Please look for the disease group (Pain) and give your input and monitor other’s input. Peter will spearhead it.  The Steering group meeting in June at Pain Meeting in Vancouver-Saturday and Sunday.

 

Please remember the ongoing study of the joint injection questionnaire. It has started at Hackensack and coordinated with Vancouver. The study will look at local anesthesia arms, with and without lidocaine with iodopheresis (note: tingling side effect).

The study coordinators will send out an IRB protocol and grant application.

 

Transition study-Patience White

Their group will survey the pediatric rheumatology community and how are they following the current guidelines from the consensus conference of 2002. The group is using the Survey Monkey. Peter Malleson will fill out the form CARRA requires-It will be sent out thru CARRA and then sent for publication.

 

IV-CARRA funding sources discussions

What is the current atmosphere for clinical research in rheumatic disease? We’ll review the current status and describe some funding and career opportunities and new initiatives

  1. NIAMS/NIH (USA)
  2. Arthritis Foundation (USA)
  3. IMHA (Institute of Musculoskeletal Health and Arthritis)/CIHR (Canadian Institute of Health Research)-Canada

 

A. NIH/NIAMS

Let’s review current status of funding-Is there money?

1. Budget

Two hundred and forty-four new and competing continuation applications accepted by NIAMS in 2005 (20.2% acceptance rate); overall rate for NIH was 22%; NIAMS is usually less than the NIH rate due to its 10% rate on high risk research applications. The information on grants available on NIAMS website at www.NIAMS.gov.

 

What is the picture for 2006 and 2007 for funding? Congress has cut Fiscal Year (FY) NIAMS 2006 funding by 3.2 million dollars over FY 2005-In response,  NIAMS is decreasing non-competing continuing grants  by 3.2%. The pay line for FY 2006 is 14.5% and overall success rate of grants will be 18-19%. All program projects (e.g., P01) that are 10 years old or over will be eliminated.

 

FY 2007 is going to be tough; NIH total budget will be cut to $28.6 billion-NIAMS will go down to a budget of 504.5 million; NIAMS success rate will be likely be in the neighborhood of 16% compared to 19% NIH overall.

 

2. New career opportunities

a. New career funding opportunities (K99)

This is a NIH pathway to independence award program. The program features an opportunity for promising postdoc scientists to receive both mentored and independent research support from the same award This was started  partly in recognition that the mean age currently of  new RO1 awardees is now 42years. NIH will award 150-200 awards in its initial year, beginning in fall 2006. NIH hopes to continue this level of awards for each of the following five years. This is a major component of the NIH effort to support new scientists as they transition to research independence. All NIH institutes and Centers are participating in this award. Award applications will be reviewed by K study sections. This is a career transition grant. The grant application can be viewed at the website http://grants.nih.gov/grants/guide/pa-files.  The mentored phase direct costs are $50,000 per year, with $20,000 for research costs. Independent phase requires review and if continued, $250,000 per year is budgeted. Many submission dates are planned including a 4/6, 10/6;

 

b. Exploratory/Development Research Grants

High risk research will major risk of failure, but if works, then it is a significant result-2 year grant-$275,000 total. Success rate is low at 10%.

 

3. NIAMS Small Grant Award for New Investigators (R03)

This grant is for funding for a pilot study to obtain preliminary data. If you’ve had a large grant with the NIH in the past you are ineligible. It provides 3 years of support with up to $50,000 per year.

 

4. NIH Loan Repayment Program for Clinical Research (L30) for subspecialty trainees. If the applicant’s research application is chosen (it is competitive), the trainee must do a minimum of 2 years of research. It is expected that the applicant for the L30 has received outside funding for the research, though it does not have to be from the NIH. The applicant will receive repayment of $35,000/year towards his or her loans.

 

5. CTSA-This program may replace the GCRP. It is for the development of a center for translational clinical and basic research projects so that new treatments can be developed and be available much more quickly to patients. CTSA’s purpose is to create a definable academic home for the discipline of clinical and transitional science that encourage novel methods and approaches, enhance informatics and technology, and improve training and mentoring to ensure that new investigators can navigate the increasingly complex research system.

Other programs are continuing:

6. Outcomes RFA; To promote the development of instruments to measure outcomes and clinical response. ($11 million)

7. Clinical Trial Planning Grants (PA)

8. RDCRN (RFA about to be reissued)-rare disease center and research network-look to this RFA

9. Rheumatic Disease Core Centers (RFA)

10. NIH wide: Gain (Genetic association information network) and GEI (genes and environment)

 

B. Arthritis Foundation

Debbie McCoy

AF Vice President for Research

1. Introduction to Arthritis Foundation

The mission activities for the Arthritis Foundation (AF) include research, public health, and public policy. The top priority diseases currently are OA, RA, and JIA. Peer review includes ten study sections. On each study section, members serve 3 year terms. Selection of the members of the study sections is based upon scientific background. Most clinical studies are in clinical immunology, clinical outcomes and therapeutics, and technology and biomechanics.

Grant decisions are made by the Medical and Scientific Advisory Board of the AF. It determines funding priorities fro any given year and allocation for award programs. The award programs are defined by their funding mechanism. Funds are actually a bit better in 2006! Sept 1 is the deadline for these research grants.

2. Types of awards:

a. Post-doc fellowships for MD or PhD scientists. This grant provides salary support of $50,000 for up to 3 years.

b. Doctor dissertation. This grant awards sufficient funds to finish a dissertation.

c. Career Development Grant-This grant requires > 3 years research experience and allows a young I            investigator to set up own lab. The grant provides $330,000 over 4 years.

d. Established Investigator Awards-e.g., Innovation Research Grant. This grant is for innovative, novel, and special research opportunities to address unique and relevant research that has substantial risk of having negative results. Little or no preliminary data is necessary. The grant provides $100,000 per year for 2 years;

 

3. How does the AF spend its money for research every year? On developing young researchers! Eight-two percent of AF funds are in training and career development grants. Seventeen percent fund established investigators; Innovative research applications have a success rate of 10% and career development/training grants are at 40%.

 

4. Changes in the AF: A working group has been established

There has been consideration of a merger of AJAO strategic goals and AF goals; e.g., AJAO goal by 2010 for 10 million dollars, AF has a 3 million dollar target for Juvenile idiopathic arthritis funding.

Charge of the working group is to discuss if the AJAO and the AF Strategic Initiatives can be combined-$10 million fund-raising is feasible and how these may be best utilized and how collaborate with other organizations and funding sources. The report from this working group is due by June- The M&S committee will make a recommendation to Executive Board of the Arthritis Foundation.

 

5. Contact information

www.arthritis.org

dmccoy@arthritis.org (404-965-9625)

Don’t forget the summer fellowship meeting of pediatric rheumatology fellows at the AJAO in July which is organized by the American Academy of Pediatrics and funded by the AF.

 

C. Institute of Musculoskeletal Health and Arthritis (IHMA)-Louise Desjardins, BSc, MSc, PhD

IHMA is one of 13 institutes of Canadian Institute of Health Research (CIHR)

There are 3 dimensions:

1. A focus on access (arthritis, bone, skin, muscle, oral, musculoskeletal rehab), strategic priorities (tissue injury and repair, physical activity, mobility, and health/pain disability), and knowledge translation. All three areas built on ethics base.

 

2. All research projects are done in concert with commercial partners.

Partnership contributions can be diverse and include research agendas.

 

3. Types of grants (Go to website for more detail on grants)

a. One year pilot grants

b. Three to five year individual and team grants up to $500,000/year-e.g., New and Emerging Teams.

c. Visiting Professorships

d. Canadian wide science fair

e. Pfizer summer science scholarship

f. Strategic training in health research: e.g., pain and Inflammation (Pfizer), pain and disability; also operating grants in arthritis, injury initiatives, regenerative medicine, and osteoarthritis (Partners put in 5-6 parts to one part government)

g. Knowledge Translation: Choosing new research directions, informing relevant policies, encouraging product development-all done with partners-Doing two such transitional knowledge projects at present.

 

2. Stakeholders in the IHMA: Researchers, health care providers, policy makers, the Canadian public, private sector, and patients/public groups.

 

3. Recent conferences:

a. Current research- topics and RFA’s

b. Canadian arthritis standards of care-access to meds, education of providers, testing innovative    

    models of care

c. Pain

 

Anyone interested in more details may e-mail Dr. Desjardin for the 2005 IHMA annual report and visit the IHMA website.

 

 

 

V Clinical Research-Advanced session Ed Giannini Cincinnati

A. Introduction

Topics today will include: Budget, Summary of required documents, Training, Certification, and Resources, and

Audits-types of audits, what to do when told an audit is going to occur, what to do before an audit to prepare, what to do during audit, possible outcomes.

 

B. Budget:

1)     Look at your study protocol, flowsheet, and budget and make sure all 3 match up well.

2)     Some sponsors believe that study coordinators and investigators should not be compensated. If that is the case, it may be possible to merge costs into other areas.

3)     Prepare an internal budget exactly like the one provided by the pharmaceutical company and compare the two budgets.

4)     Underestimating study coordinator time needed is a major budget problem. Remember to include monitoring visits by the sponsor or CRC-When the monitor is there, his or her time is totally utilized.

5)     Allow for time to prepare and ship specimens in coordinator’s time.

6)     Don’t be afraid to approach the sponsor company for budget renegotiation if some costs are not being covered. They expect you to come back.

7)     Ask for non-refundable startup funds and fees upfront and these funds should be paid even if no patients are ever enrolled.

C. Required study documents:

1)     Important points in preparing study documents:

a)     Need to document delegation of authority for any study.

b)     Document everything when communicating with sponsor or coordinating center-Have a log of contacts, especially phone calls. The pharmaceutical companies do.

c)      Act as if it’s a medical record and you have to go to court-have all the documents you would need.

D. Training, certification, and resources

1) Required now or soon to be required everywhere:

                        a) CITI training is now the standard-Course in the Protection of Human Research Subjects

b) GCP-Good Clinical Practice training-provided not at Drug study meetings but at separate   

    training programs.

c) HIPAA-training by institution you’re at.

2) Recommended

a) You should take CITI training-check with your institution-you may be able to take it there-You

    can also use on-line training at www.citiprogram.org  and follow explicit instructions as an

    outside applicant, e.g., at Cincinnati Children’s

b) Institutions are seeking AAHRAP certification and requiring you to take CITI and other training.

c) You will need GCP training soon-Good clinical practice guidelines-these guidelines give you  

    help in designing and performing a study properly.

d)     Training may soon be required to take GCP training e.g., at RAN Institute, it might cost $175 for an online course by yourself and $87.50 for a discount. Soon training at drug company investigators will not be sufficient to the FDA as they have been tired of mistakes.

e)     It is not required yet to get Certified Physician Investigator (CPA) online through Academy of Pharmaceutical Physicians and Investigators-This is advanced certification.

3) Resources  

There are a number of texts and references e.g., The CRC Guide to Coordinating Clinical

Research. Other good references to use:

a) Ellenberg SS, Fleming TR, DeMets DL: Data Monitoring Committees in Clinical

                            Trials: A Practical Perspective.

b) Nylen: The ultimate step by step guide to conducting pharmaceutical studies in the

                            USA

4) HIPAA requirements-Check with your institutional requirements.

 

E. Audits

This discussion will focus on FDA, Internal Audits, and Drug Company Audits.

1. What trigger audits, internal or external?

  1. Previous poor performance: For cause audits, if “official action indicated” letter is sent, one of three possible results of this type of audit. These “official action indicated” letters are quite serious and get you on a black list, the ultimate punishment by the FDA for clinical researchers.
  2. The FDA can be notified by the drug company and will audit and will find errors if it looks hard enough and then could possibly look at some or all of the clinical studies of the institution (You would then wear on scarlet R on your lab coat).
  3. Other reasons: safety findings or efficacy findings that are inconsistent with other centers, if the study is a pivotal study for FDA application.

 

2. If you receive notice that you are going to be audited:

a. Notify everyone-Sponsor, IRB (your mother, the neighbors)

b. Get ready using an audit checklist

c. Some "do’s and don’ts" might include:

1)  No clinic for the PI or subPI’s that day-Don’t have patients in clinic or inpatient duties. It insults the

auditors and gets everything off on a bad foot.

2) Answer as briefly and concisely as possible and don’t sign anything. Pretend that you are a defendant

in front of a judge.

3) Offer only water, coffee, soft drinks-no special entertainment, dinner, free lunch.

4) Have all patient notebooks, ICDs, training and source documents, computerized records at hand for

the audit. The auditor will try to see if all the records jibe. Have a room setup before hand.

 

3. Outcomes of an audit:

a. FDA-deviations/violations: may get form 483-shows their findings and requires a response.

b. Correction needed right away-You will get a warning letter if the inspector finds something wrong that needs correction right away. Respond immediately.

c. The official Establishment Inspection Report (EIR) comes several months later: 3 outcomes:

a) No action indicated (NAI):

b) Voluntary action indicated (VAI)-not signing CRF’s in a timely manner,

     unknown investigator signs CRF

c) Official Action Indicated (OAI)-the worse-you’re put on the List of

    Disqualified and Restricted Investigator list. You will never get another study

    And should resign from research groups (CARRA, PRCSG).

    Example: Patient in study not observed overnight as indicated after study

    medicine given. Patient became sick overnight and was brought into the

    institution’s ER. No one at the ER knew anything about the study. The PI could

    not be contacted.

   

 

 

V SUMMARY OF MEETING

A. JIA Committee

1. Studies to mention

a. Uveitis Remicad