Patients Treated for Wiskott-Aldrich Syndrome (WAS) Since 1990

NCT ID: NCT02064933

Last Updated: 2020-08-10

Study Results

Results pending

The study team has not published outcome measurements, participant flow, or safety data for this trial yet. Check back later for updates.

Basic Information

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Recruitment Status

COMPLETED

Total Enrollment

305 participants

Study Classification

OBSERVATIONAL

Study Start Date

2014-02-02

Study Completion Date

2019-05-01

Brief Summary

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Wiskott - Aldrich syndrome (WAS) is a rare serious medical condition that causes problems both with the immune system and with easy bruising and bleeding. The immune abnormalities cause patients with WAS to be very susceptible to infections. Depending on the specific type of primary immune deficiency diseases, there are effective treatments, including antibiotics, cellular therapy and gene therapy, but studies of large numbers of patients are needed to determine the full range of causes, natural history, or the best methods of treatment for long term success.

This multicenter study combines retrospective, prospective and cross-sectional analyses of the transplant experiences for patients with WAS who have already received HCT since 1990, or who will undergo Hematopoietic cell transplant (HCT) during the study period. The retrospective and prospective portions of the study will address the impact of a number of pre and post-transplant factors on post-transplant disease correction and ultimate benefit from HCT and the cross-sectional portion of the study will assess the benefit of HCT 2 years post-HCT in consenting surviving patients.

Detailed Description

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Conditions

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Wiskott-Aldrich Syndrome

Study Design

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Observational Model Type

COHORT

Study Time Perspective

OTHER

Study Groups

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Retrospective Cohort (Longitudinal Analysis)

Participants with WAS treated at consortium centers since 1990 who have received transplant (Stratum A) or gene therapy (Stratum B)

No interventions assigned to this group

Prospective Cohort (Longitudinal Analysis)

Participants treated at consortium centers since 1990 who will receive transplant (Stratum A) or gene therapy (Stratum B)

No interventions assigned to this group

Cross-Sectional Cohort (Cross-sectional Analysis)

Living participants with WAS who have received transplant (Stratum A) or gene therapy (Stratum B) at Consortium Centers 1990-Present And \>= 2 Years Post-Transplant

No interventions assigned to this group

Eligibility Criteria

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Inclusion Criteria

* WAS participants will be defined as males who have:

1. thrombocytopenia (\< 100K) AND EITHER molecular diagnosis of WAS OR reduced WASP expression; OR
2. thrombocytopenia (\< 100K) AND positive family history consistent with WAS diagnosis; OR
3. chronic thrombocytopenia (\< 100K for minimum of 3 months) AND low mean platelet volume (MPV below normal range for age) AND EITHER recurrent and/or severe infections requiring treatment and/or eczema OR lack of antibody response to polysaccharide antigens or low IgM.
* Longitudinal Analysis (Retrospective and Prospective)

1. Stratum A. Participants with WAS who have or will Receive HCT

* Participants with WAS who have received an HCT since January 1, 1990
2. Stratum B. Participants with WAS who have or will Receive Gene Transfer

* Participants in which the intention is to treat with gene transfer with autologous modified cells
* Cross-Sectional Analysis (Strata A and B) 1. Participants with WAS who are surviving and at least 2 years after the most recent HCT or gene therapy.
Eligible Sex

MALE

Accepts Healthy Volunteers

No

Sponsors

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Primary Immune Deficiency Treatment Consortium (PIDTC)

OTHER

Sponsor Role collaborator

Rare Diseases Clinical Research Network

NETWORK

Sponsor Role collaborator

National Institute of Allergy and Infectious Diseases (NIAID)

NIH

Sponsor Role lead

Responsible Party

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Responsibility Role SPONSOR

Principal Investigators

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Lauri M. Burroughs, MD

Role: STUDY_CHAIR

Fred Hutchinson Cancer Center

David J. Rawlings, MD

Role: STUDY_CHAIR

Department of Pediatrics, University of Washington-Seattle Children's Hospital

Luigi D. Notarangelo, MD

Role: STUDY_CHAIR

National Institute of Allergy and Infectious Diseases, NIH

Alexandra H. Filipovich, MD

Role: STUDY_CHAIR

Children's Hospital Medical Center, Cincinnati

Locations

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Department of Pediatrics, University of Alabama at Birmingham

Birmingham, Alabama, United States

Site Status

Phoenix Children's Hospital

Phoenix, Arizona, United States

Site Status

Cancer and Blood Disease Institute, Children's Hospital Los Angeles, Keck School of Medicine, University of Southern California

Los Angeles, California, United States

Site Status

Department of Pediatrics, David Geffen School of Medicine at University of California, Los Angeles,

Los Angeles, California, United States

Site Status

Lucile Salter Packard Children's Hospital at Stanford

Palo Alto, California, United States

Site Status

University of California, San Francisco Benioff Children's Hospital

San Francisco, California, United States

Site Status

Children's Hospital Denver, University of Colorado

Denver, Colorado, United States

Site Status

Nemours Alfred I. duPont Hospital for Children

Wilmington, Delaware, United States

Site Status

Children's National Hospital-George Washington University School of Medicine and Health Sciences

Washington D.C., District of Columbia, United States

Site Status

Blood and Marrow Transplant Program, Johns Hopkins All Children's Hospital

St. Petersburg, Florida, United States

Site Status

Aflac Cancer and Blood Disorders Center, Emory/Children's Healthcare of Atlanta

Atlanta, Georgia, United States

Site Status

Ann & Robert H. Lurie Children's Hospital of Chicago

Chicago, Illinois, United States

Site Status

Center for Cancer and Blood Disorders, Children's Hospital/Louisiana State University

New Orleans, Louisiana, United States

Site Status

Boston Children's Hospital

Boston, Massachusetts, United States

Site Status

Department of Pediatrics, C. S. Mott Children's Hospital, University of Michigan

Ann Arbor, Michigan, United States

Site Status

Division of Pediatric Blood and Marrow Transplantation, University of Minnesota

Minneapolis, Minnesota, United States

Site Status

Mayo Clinic Children's Center

Rochester, Minnesota, United States

Site Status

Cardinal Glennon Children's Hospital, Saint Louis University

St Louis, Missouri, United States

Site Status

Saint Louis Children's Hospital, Washington University

St Louis, Missouri, United States

Site Status

Institute for Pediatric Cancer and Blood Disorders, Hackensack University Medical Center

Hackensack, New Jersey, United States

Site Status

Department of Pediatrics, Memorial Sloan Kettering Cancer Center

New York, New York, United States

Site Status

Department of Pediatrics, Golisano Children's Hospital, University of Rochester

Rochester, New York, United States

Site Status

Maria Fareri Children's Hospital, New York Medical College

Valhalla, New York, United States

Site Status

Duke University Medical Center

Durham, North Carolina, United States

Site Status

Cincinnati Children's Hospital Medical Center, University of Cincinnati

Cincinnati, Ohio, United States

Site Status

Rainbow Babies and Children's Hospital

Cleveland, Ohio, United States

Site Status

Nationwide Children's Hospital

Columbus, Ohio, United States

Site Status

Division of Pediatric Hematology/Oncology, Oregon Health and Science University

Portland, Oregon, United States

Site Status

Children's Hospital of Philadelphia, University of Pennsylvania

Philadelphia, Pennsylvania, United States

Site Status

University of Pittsburgh Children's Hospital of Pittsburgh

Pittsburgh, Pennsylvania, United States

Site Status

St. Jude Children's Research Hospital

Memphis, Tennessee, United States

Site Status

Pediatrics, University of Texas Southwestern Medical Center

Dallas, Texas, United States

Site Status

Baylor College of Medicine Section of Immunology, Allergy, and Retrovirology, Texas Children's Hospital

Houston, Texas, United States

Site Status

Texas Transplant Institute, Methodist Children's Hospital

San Antonio, Texas, United States

Site Status

Primary Children's Hospital, University of Utah

Salt Lake City, Utah, United States

Site Status

Fred Hutchinson Cancer Research Center and University of Washington-Seattle Children's Hospital

Seattle, Washington, United States

Site Status

American Family Children's Hospital, University of Wisconsin

Madison, Wisconsin, United States

Site Status

Children's Hospital of Wisconsin-Milwaukee

Milwaukee, Wisconsin, United States

Site Status

Alberta Children's Hospital

Calgary, Alberta, Canada

Site Status

Children's & Women's Health Centre of British Columbia

Vancouver, British Columbia, Canada

Site Status

Cancer Care Manitoba, University of Manitoba

Winnipeg, Manitoba, Canada

Site Status

The Hospital for Sick Children

Toronto, Ontario, Canada

Site Status

CHU Sainte-Justine, Department of Pediatrics, University of Montreal

Montreal, Quebec, Canada

Site Status

Countries

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United States Canada

References

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Griffith LM, Cowan MJ, Kohn DB, Notarangelo LD, Puck JM, Schultz KR, Buckley RH, Eapen M, Kamani NR, O'Reilly RJ, Parkman R, Roifman CM, Sullivan KE, Filipovich AH, Fleisher TA, Shearer WT. Allogeneic hematopoietic cell transplantation for primary immune deficiency diseases: current status and critical needs. J Allergy Clin Immunol. 2008 Dec;122(6):1087-96. doi: 10.1016/j.jaci.2008.09.045. Epub 2008 Nov 6.

Reference Type BACKGROUND
PMID: 18992926 (View on PubMed)

Griffith LM, Cowan MJ, Notarangelo LD, Kohn DB, Puck JM, Pai SY, Ballard B, Bauer SC, Bleesing JJ, Boyle M, Brower A, Buckley RH, van der Burg M, Burroughs LM, Candotti F, Cant AJ, Chatila T, Cunningham-Rundles C, Dinauer MC, Dvorak CC, Filipovich AH, Fleisher TA, Bobby Gaspar H, Gungor T, Haddad E, Hovermale E, Huang F, Hurley A, Hurley M, Iyengar S, Kang EM, Logan BR, Long-Boyle JR, Malech HL, McGhee SA, Modell F, Modell V, Ochs HD, O'Reilly RJ, Parkman R, Rawlings DJ, Routes JM, Shearer WT, Small TN, Smith H, Sullivan KE, Szabolcs P, Thrasher A, Torgerson TR, Veys P, Weinberg K, Zuniga-Pflucker JC; workshop participants. Primary Immune Deficiency Treatment Consortium (PIDTC) report. J Allergy Clin Immunol. 2014 Feb;133(2):335-47. doi: 10.1016/j.jaci.2013.07.052. Epub 2013 Oct 15.

Reference Type RESULT
PMID: 24139498 (View on PubMed)

Griffith LM, Cowan MJ, Notarangelo LD, Kohn DB, Puck JM, Shearer WT, Burroughs LM, Torgerson TR, Decaluwe H, Haddad E; workshop participants. Primary Immune Deficiency Treatment Consortium (PIDTC) update. J Allergy Clin Immunol. 2016 Aug;138(2):375-85. doi: 10.1016/j.jaci.2016.01.051. Epub 2016 Apr 22.

Reference Type RESULT
PMID: 27262745 (View on PubMed)

Pai SY, Logan BR, Griffith LM, Buckley RH, Parrott RE, Dvorak CC, Kapoor N, Hanson IC, Filipovich AH, Jyonouchi S, Sullivan KE, Small TN, Burroughs L, Skoda-Smith S, Haight AE, Grizzle A, Pulsipher MA, Chan KW, Fuleihan RL, Haddad E, Loechelt B, Aquino VM, Gillio A, Davis J, Knutsen A, Smith AR, Moore TB, Schroeder ML, Goldman FD, Connelly JA, Porteus MH, Xiang Q, Shearer WT, Fleisher TA, Kohn DB, Puck JM, Notarangelo LD, Cowan MJ, O'Reilly RJ. Transplantation outcomes for severe combined immunodeficiency, 2000-2009. N Engl J Med. 2014 Jul 31;371(5):434-46. doi: 10.1056/NEJMoa1401177.

Reference Type RESULT
PMID: 25075835 (View on PubMed)

Haddad E, Allakhverdi Z, Griffith LM, Cowan MJ, Notarangelo LD. Survey on retransplantation criteria for patients with severe combined immunodeficiency. J Allergy Clin Immunol. 2014 Feb;133(2):597-9. doi: 10.1016/j.jaci.2013.10.022. Epub 2013 Dec 10. No abstract available.

Reference Type RESULT
PMID: 24331379 (View on PubMed)

Shearer WT, Dunn E, Notarangelo LD, Dvorak CC, Puck JM, Logan BR, Griffith LM, Kohn DB, O'Reilly RJ, Fleisher TA, Pai SY, Martinez CA, Buckley RH, Cowan MJ. Establishing diagnostic criteria for severe combined immunodeficiency disease (SCID), leaky SCID, and Omenn syndrome: the Primary Immune Deficiency Treatment Consortium experience. J Allergy Clin Immunol. 2014 Apr;133(4):1092-8. doi: 10.1016/j.jaci.2013.09.044. Epub 2013 Nov 28.

Reference Type RESULT
PMID: 24290292 (View on PubMed)

Dvorak CC, Cowan MJ, Logan BR, Notarangelo LD, Griffith LM, Puck JM, Kohn DB, Shearer WT, O'Reilly RJ, Fleisher TA, Pai SY, Hanson IC, Pulsipher MA, Fuleihan R, Filipovich A, Goldman F, Kapoor N, Small T, Smith A, Chan KW, Cuvelier G, Heimall J, Knutsen A, Loechelt B, Moore T, Buckley RH. The natural history of children with severe combined immunodeficiency: baseline features of the first fifty patients of the primary immune deficiency treatment consortium prospective study 6901. J Clin Immunol. 2013 Oct;33(7):1156-64. doi: 10.1007/s10875-013-9917-y. Epub 2013 Jul 2.

Reference Type RESULT
PMID: 23818196 (View on PubMed)

Griffith LM, Cowan MJ, Notarangelo LD, Puck JM, Buckley RH, Candotti F, Conley ME, Fleisher TA, Gaspar HB, Kohn DB, Ochs HD, O'Reilly RJ, Rizzo JD, Roifman CM, Small TN, Shearer WT; Workshop Participants. Improving cellular therapy for primary immune deficiency diseases: recognition, diagnosis, and management. J Allergy Clin Immunol. 2009 Dec;124(6):1152-60.e12. doi: 10.1016/j.jaci.2009.10.022.

Reference Type RESULT
PMID: 20004776 (View on PubMed)

Burroughs LM, Petrovic A, Brazauskas R, Liu X, Griffith LM, Ochs HD, Bleesing JJ, Edwards S, Dvorak CC, Chaudhury S, Prockop SE, Quinones R, Goldman FD, Quigg TC, Chandrakasan S, Smith AR, Parikh S, Davila Saldana BJ, Thakar MS, Phelan R, Shenoy S, Forbes LR, Martinez C, Chellapandian D, Shereck E, Miller HK, Kapoor N, Barnum JL, Chong H, Shyr DC, Chen K, Abu-Arja R, Shah AJ, Weinacht KG, Moore TB, Joshi A, DeSantes KB, Gillio AP, Cuvelier GDE, Keller MD, Rozmus J, Torgerson T, Pulsipher MA, Haddad E, Sullivan KE, Logan BR, Kohn DB, Puck JM, Notarangelo LD, Pai SY, Rawlings DJ, Cowan MJ. Excellent outcomes following hematopoietic cell transplantation for Wiskott-Aldrich syndrome: a PIDTC report. Blood. 2020 Jun 4;135(23):2094-2105. doi: 10.1182/blood.2019002939.

Reference Type RESULT
PMID: 32268350 (View on PubMed)

Related Links

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https://www.rarediseasesnetwork.org/cms/pidtc/

Primary Immune Deficiency Treatment Consortium (PIDTC), Rare Clinical Diseases Research Network, National Institutes of Health

https://www.niaid.nih.gov/about/dait

Division of Allergy, Immunology, and Transplantation (DAIT)

https://www.niaid.nih.gov/

National Institute of Allergy and Infectious Diseases (NIAID)

Other Identifiers

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DAIT RDCRN PIDTC-6904

Identifier Type: -

Identifier Source: org_study_id

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