Trial of Oral Valproic Acid for Retinitis Pigmentosa

NCT ID: NCT01233609

Last Updated: 2017-12-02

Study Results

Results available

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Basic Information

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Recruitment Status

COMPLETED

Clinical Phase

PHASE2

Total Enrollment

90 participants

Study Classification

INTERVENTIONAL

Study Start Date

2010-11-30

Study Completion Date

2015-12-31

Brief Summary

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The objectives of this study are to evaluate the efficacy of Valproic Acid (VPA) to both slow the progression of visual function loss and/or to restore visual function in patients with Autosomal Dominant Retinitis Pigmentosa (RP) and to collect safety and tolerability information.

Detailed Description

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Retinitis Pigmentosa (RP) is an incurable and untreatable group of heterogeneous retinal degenerative diseases that cause severe visual loss. There is currently no therapeutic that substantially slows the progression of this disease, and certainly none that can restore vision in RP patients. The Valproic Acid (VPA) study is designed as a six-site, interventional, prospective, randomized, placebo controlled, double-blinded study of 90 participants to evaluate the efficacy of oral Valproic Acid to both slow the progression of visual function loss and/or to restore visual function in patients with an Autosomal Dominant RP genetic mutation and to collect safety and tolerability information. Patients that participate in the study will be randomized to either placebo or VPA in a 1:1 ratio.

Conditions

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Retinitis Pigmentosa

Keywords

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retinitis pigmentosa valproic acid visual field

Study Design

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Allocation Method

RANDOMIZED

Intervention Model

PARALLEL

Primary Study Purpose

TREATMENT

Blinding Strategy

QUADRUPLE

Participants Caregivers Investigators Outcome Assessors

Study Groups

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Valproic Acid

Subjects who receive valproic acid

Group Type ACTIVE_COMPARATOR

Valproic Acid

Intervention Type DRUG

One to four 250mg softgels by mouth daily (dose determined by body weight)

Placebo

Subjects who receive placebo

Group Type PLACEBO_COMPARATOR

Placebo

Intervention Type DRUG

Dosage per subject weight- same schedule as the active comparator

Interventions

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Valproic Acid

One to four 250mg softgels by mouth daily (dose determined by body weight)

Intervention Type DRUG

Placebo

Dosage per subject weight- same schedule as the active comparator

Intervention Type DRUG

Other Intervention Names

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Valproate

Eligibility Criteria

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Inclusion Criteria

1. Understand/sign the IRB-approved study informed consent document.
2. Age greater than or equal to 18 years, no upper age limit
3. Males and non-child bearing females must weigh ≥40 Kg and ≤158.9 Kg; Females of child bearing potential must weigh ≥40 Kg and ≤74.9 Kg.
4. Diagnosis of Retinitis Pigmentosa (RP).
5. Visual acuity of greater than or equal to 35 letters in at least one eye as measured by the EVA-ETDRS (equivalent to 20/200 on a Snellen chart).
6. Genotyped as autosomal dominant form of RP.
7. Female subjects of childbearing potential and male subjects able to father children must have (or have a partner who has) had a hysterectomy or vasectomy, be completely abstinent from intercourse or must commit to practice at least two acceptable methods of contraception to minimize the chance of pregnancy during the study and for the 13 week period after stopping the study drug.
8. Female subjects of childbearing potential must have a negative urine pregnancy test at study entry and throughout the duration of the study.
9. Willingness to comply with the protocol.

Exclusion Criteria

1. Medical problems that make consistent follow-up over the treatment period unlikely (e.g. stroke, severe MI, end stage malignancy), or in general a poor medical risk because of other systemic diseases or active uncontrolled infections.
2. Other retinal diseases: Glaucoma, retinal inflammatory disease (CME is allowable), cataract worse than +2 NS, or herpes simplex virus of the eye.
3. Intact visual field of 5⁰ or less.
4. Subject unable to provide reliable perimetry measurements in both eyes for both static and kinetic visual field, as determined by the Reading Center.
5. Diabetes.
6. History of cancer (other than non-melanoma skin cancer) diagnosed, or requiring treatment within the past 2 years.
7. A hemoglobin concentration, a platelet count or an absolute neutrophil count below the lower limit of normal at study entry.
8. Suspected liver dysfunction determined by having liver function values elevated above the upper limit of normal.
9. History of pancreatitis by clinical features and/or laboratory abnormalities in the last 12 months.
10. Renal dysfunction based on serum creatinine,(MDRD) equation.
11. Urea cycle disorders.
12. History of neurological conditions including epilepsy, history of brain injury, encephalitis, or any organic brain syndrome.
13. History of schizophrenia, schizoaffective disorder, bipolar disorder, suicidality or organic mental disorders.
14. Currently receiving valproic acid or other anti-convulsants.
15. Sensitive to or have ever had an allergic reaction to valproic Acid.
16. Sensitive to or have ever had an allergic reaction to peanuts as peanut oil is an inactive ingredient in valproic acid capsules and the placebo.
17. Has taken one of the disallowed drugs at least 2 weeks prior to randomization.
18. Pregnant women.
19. Lactating mothers who are breast feeding their babies.
20. RP patients involved in other clinical trials within the last 3 months.
21. Require enrollment by consent of a legally authorized representative.
22. Persons who are unable to read are not allowed to consent for themselves or others to participate in this study.
23. The potential participant lives in the same household as a current participant in this protocol.
Minimum Eligible Age

18 Years

Eligible Sex

ALL

Accepts Healthy Volunteers

No

Sponsors

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United States Department of Defense

FED

Sponsor Role collaborator

Foundation Fighting Blindness

OTHER

Sponsor Role lead

Responsible Party

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Responsibility Role SPONSOR

Principal Investigators

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Patricia Zilliox, PhD

Role: STUDY_DIRECTOR

Foundation Fighting Blindness

Locations

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University of Miami, Bascom Palmer Eye Institute

Miami, Florida, United States

Site Status

University of Michigan, Ann Arbor

Ann Arbor, Michigan, United States

Site Status

Oregon Health & Science University

Portland, Oregon, United States

Site Status

University of Tennessee, Hamilton Eye Institute

Memphis, Tennessee, United States

Site Status

Retina Foundation of the Southwest

Dallas, Texas, United States

Site Status

University of Utah School of Medicine, Moran Eye Center

Salt Lake City, Utah, United States

Site Status

Countries

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United States

References

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Berson EL, Rosner B, Weigel-DiFranco C, Dryja TP, Sandberg MA. Disease progression in patients with dominant retinitis pigmentosa and rhodopsin mutations. Invest Ophthalmol Vis Sci. 2002 Sep;43(9):3027-36.

Reference Type BACKGROUND
PMID: 12202526 (View on PubMed)

Bryant AE 3rd, Dreifuss FE. Valproic acid hepatic fatalities. III. U.S. experience since 1986. Neurology. 1996 Feb;46(2):465-9. doi: 10.1212/wnl.46.2.465.

Reference Type BACKGROUND
PMID: 8614514 (View on PubMed)

Berson EL, Sandberg MA, Rosner B, Birch DG, Hanson AH. Natural course of retinitis pigmentosa over a three-year interval. Am J Ophthalmol. 1985 Mar 15;99(3):240-51. doi: 10.1016/0002-9394(85)90351-4.

Reference Type BACKGROUND
PMID: 3976802 (View on PubMed)

Chen PS, Wang CC, Bortner CD, Peng GS, Wu X, Pang H, Lu RB, Gean PW, Chuang DM, Hong JS. Valproic acid and other histone deacetylase inhibitors induce microglial apoptosis and attenuate lipopolysaccharide-induced dopaminergic neurotoxicity. Neuroscience. 2007 Oct 12;149(1):203-12. doi: 10.1016/j.neuroscience.2007.06.053. Epub 2007 Jul 28.

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PMID: 17850978 (View on PubMed)

Delyfer MN, Leveillard T, Mohand-Said S, Hicks D, Picaud S, Sahel JA. Inherited retinal degenerations: therapeutic prospects. Biol Cell. 2004 May;96(4):261-9. doi: 10.1016/j.biolcel.2004.01.006.

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Dragunow M, Greenwood JM, Cameron RE, Narayan PJ, O'Carroll SJ, Pearson AG, Gibbons HM. Valproic acid induces caspase 3-mediated apoptosis in microglial cells. Neuroscience. 2006 Jul 21;140(4):1149-56. doi: 10.1016/j.neuroscience.2006.02.065.

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PMID: 16600518 (View on PubMed)

Gaby AR. Nutritional therapies for ocular disorders: Part Three. Altern Med Rev. 2008 Sep;13(3):191-204.

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Gottlicher M, Minucci S, Zhu P, Kramer OH, Schimpf A, Giavara S, Sleeman JP, Lo Coco F, Nervi C, Pelicci PG, Heinzel T. Valproic acid defines a novel class of HDAC inhibitors inducing differentiation of transformed cells. EMBO J. 2001 Dec 17;20(24):6969-78. doi: 10.1093/emboj/20.24.6969.

Reference Type BACKGROUND
PMID: 11742974 (View on PubMed)

Hartong DT, Berson EL, Dryja TP. Retinitis pigmentosa. Lancet. 2006 Nov 18;368(9549):1795-809. doi: 10.1016/S0140-6736(06)69740-7.

Reference Type BACKGROUND
PMID: 17113430 (View on PubMed)

Henry TR. The history of valproate in clinical neuroscience. Psychopharmacol Bull. 2003;37 Suppl 2:5-16.

Reference Type BACKGROUND
PMID: 14624229 (View on PubMed)

Hoffman DR, Locke KG, Wheaton DH, Fish GE, Spencer R, Birch DG. A randomized, placebo-controlled clinical trial of docosahexaenoic acid supplementation for X-linked retinitis pigmentosa. Am J Ophthalmol. 2004 Apr;137(4):704-18. doi: 10.1016/j.ajo.2003.10.045.

Reference Type BACKGROUND
PMID: 15059710 (View on PubMed)

Jacobson SG, Cideciyan AV, Huang Y, Hanna DB, Freund CL, Affatigato LM, Carr RE, Zack DJ, Stone EM, McInnes RR. Retinal degenerations with truncation mutations in the cone-rod homeobox (CRX) gene. Invest Ophthalmol Vis Sci. 1998 Nov;39(12):2417-26.

Reference Type BACKGROUND
PMID: 9804150 (View on PubMed)

Kim HJ, Rowe M, Ren M, Hong JS, Chen PS, Chuang DM. Histone deacetylase inhibitors exhibit anti-inflammatory and neuroprotective effects in a rat permanent ischemic model of stroke: multiple mechanisms of action. J Pharmacol Exp Ther. 2007 Jun;321(3):892-901. doi: 10.1124/jpet.107.120188. Epub 2007 Mar 19.

Reference Type BACKGROUND
PMID: 17371805 (View on PubMed)

Mangione CM, Lee PP, Gutierrez PR, Spritzer K, Berry S, Hays RD; National Eye Institute Visual Function Questionnaire Field Test Investigators. Development of the 25-item National Eye Institute Visual Function Questionnaire. Arch Ophthalmol. 2001 Jul;119(7):1050-8. doi: 10.1001/archopht.119.7.1050.

Reference Type BACKGROUND
PMID: 11448327 (View on PubMed)

Noorwez SM, Ostrov DA, McDowell JH, Krebs MP, Kaushal S. A high-throughput screening method for small-molecule pharmacologic chaperones of misfolded rhodopsin. Invest Ophthalmol Vis Sci. 2008 Jul;49(7):3224-30. doi: 10.1167/iovs.07-1539. Epub 2008 Mar 31.

Reference Type BACKGROUND
PMID: 18378578 (View on PubMed)

Nowomiejska K, Vonthein R, Paetzold J, Zagorski Z, Kardon R, Schiefer U. Comparison between semiautomated kinetic perimetry and conventional Goldmann manual kinetic perimetry in advanced visual field loss. Ophthalmology. 2005 Aug;112(8):1343-54. doi: 10.1016/j.ophtha.2004.12.047.

Reference Type BACKGROUND
PMID: 15996734 (View on PubMed)

Nowomiejska K, Vonthein R, Paetzold J, Zagorski Z, Kardon R, Schiefer U. Reaction time during semi-automated kinetic perimetry (SKP) in patients with advanced visual field loss. Acta Ophthalmol. 2010 Feb;88(1):65-9. doi: 10.1111/j.1755-3768.2008.01407.x. Epub 2009 Dec 16.

Reference Type BACKGROUND
PMID: 19094165 (View on PubMed)

Peterson GM, Naunton M. Valproate: a simple chemical with so much to offer. J Clin Pharm Ther. 2005 Oct;30(5):417-21. doi: 10.1111/j.1365-2710.2005.00671.x. No abstract available.

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Reference Type BACKGROUND
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Birch DG, Bernstein PS, Iannacone A, Pennesi ME, Lam BL, Heckenlively J, Csaky K, Hartnett ME, Winthrop KL, Jayasundera T, Hughbanks-Wheaton DK, Warner J, Yang P, Fish GE, Teske MP, Sklaver NL, Erker L, Chegarnov E, Smith T, Wahle A, VanVeldhuisen PC, McCormack J, Lindblad R, Bramer S, Rose S, Zilliox P, Francis PJ, Weleber RG. Effect of Oral Valproic Acid vs Placebo for Vision Loss in Patients With Autosomal Dominant Retinitis Pigmentosa: A Randomized Phase 2 Multicenter Placebo-Controlled Clinical Trial. JAMA Ophthalmol. 2018 Aug 1;136(8):849-856. doi: 10.1001/jamaophthalmol.2018.1171.

Reference Type DERIVED
PMID: 29879277 (View on PubMed)

Related Links

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http://www.blindness.org/index.php?option=com_content&view=section&id=7&itemid=101

Click here for more information about this study: Clinical Trial page of the Foundation Fighting Blindness Website.

Other Identifiers

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H-13371

Identifier Type: -

Identifier Source: org_study_id