Pharmacologic Treatment of Congenital Nephrogenic Diabetes Insipidus

NCT ID: NCT00478335

Last Updated: 2018-02-12

Study Results

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Basic Information

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Recruitment Status

COMPLETED

Clinical Phase

NA

Total Enrollment

4 participants

Study Classification

INTERVENTIONAL

Study Start Date

2007-05-31

Study Completion Date

2012-10-31

Brief Summary

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The purpose of this research study is to determine if two investigational medications will be more effective in decreasing urine output than the currently available and routinely used medications in patients with congenital nephrogenic diabetes insipidus (NDI).

Detailed Description

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The study involves the use of the investigational medications sildenafil and calcitonin. These medications have shown promise as treatment for NDI in laboratory (non-human) studies but have not been used for treatment of NDI in humans. At this time, there is no guarantee that these investigational medications will provide additional benefit to people with NDI.

The study is open to males, between the ages of 5 and 25 years who have been diagnosed with Nephrogenic diabetes insipidus (NDI) and who have normal kidney and bladder function. A total of 40 patients with NDI will be enrolled in the study. The study will involve two outpatient clinic visits, followed by a 9-night hospital stay, followed by a final follow-up outpatient clinic visit. All visits will take place within a 20-day time period.

At the first clinic visit, blood and urine testing for kidney and liver function and blood count will be performed. If the genetic alteration which causes your NDI has not been previously identified, blood for DNA testing will also be obtained. If a kidney and bladder ultrasound has not been performed in the past 6 months, it will be obtained. The ultrasound is to make sure that there is no problem with drainage of urine from the kidneys and bladder. Subjects will be asked to fill out food preference questionnaires to use for planning of meals for the hospital stay. Subjects will be given containers to collect two consecutive 24-hour urine samples at home. These urine collections will help determine how well the subjects routine medicines are working to control their NDI.

At the second clinic visit, subjects will bring in the two 24-hour urine samples. Blood will again be collected for further testing of kidney function. Subjects will be given containers to collect another 24-hour urine just prior to the hospital visit.

The third visit requires hospital admission and will be scheduled at the study site closest to the subjects home (The Children's Hospital, Denver, Colorado; University of Aarhus, Denmark). For the hospital visit, subjects will need to stop their usual NDI medications for 48 hours prior to the visit. Subjects will perform another 24-hour urine collection on the day prior to your hospital admission. This urine sample will be turned in to the laboratory when you are admitted to the hospital for the research study. The length of the hospital stay is 10 days/9 nights. During the stay, subjects can expect to have their weight, heart rate, and blood pressure checked three times a day. All urine will be collected. Blood testing will be performed every other day. Subjects will need to eat the meals provided at the hospital; all meals will be provided according to a low-salt diet restriction. Subjects may drink fluids as desired but they will need to avoid caffeine-containing beverages and alcohol.

On the first day of the hospital stay, testing will be performed to confirm the diagnosis of NDI. This test involves administration of the medicine dDAVP (Desmopressin) through an IV catheter (into a vein) with collection of urine every 30 minutes for 4 hours. subjects will be randomized (like the toss of a coin) to receive either the investigational medication treatment for 4 days followed by the routine medication treatment for 4 days or vice versa. When subjects receive the routine medication treatment, they will receive placebos (inactive substances like a sugar pill) in place of the investigational medicines. In this way, neither the subject nor the investigator will know whether the subjects are receiving the investigational or the routine medication treatment first. Medicines will be given twice a day during the hospital stay. On the last day of the hospital stay, subjects will be instructed to resume their normal diet and medications.

At a final outpatient clinic visit, blood testing and urinalysis will be performed.

Potential benefits of participation include a no-cost health examination, laboratory studies, and an evaluation of current management of NDI. There is no cost for participation in this research study. No pay will be given to participants in this research study.

This research study has been approved by the ethical review boards of the following institutions: Colorado Multiple Institutional Review Board (#06-0588), Emory University Institutional Review Board (#729-2005), and the University of Aarhus (#20050183). Individuals who decide to take part in this research study will need to sign a specific consent form at a participating institution as well as a release for use of personal health information (HIPAA form).

Conditions

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Nephrogenic Diabetes Insipidus

Study Design

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Allocation Method

RANDOMIZED

Intervention Model

CROSSOVER

Primary Study Purpose

TREATMENT

Blinding Strategy

QUADRUPLE

Participants Caregivers Investigators Outcome Assessors

Study Groups

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Active Therapy

4-day treatment with hydrochlorothiazide/amiloride, indomethacin, calcitonin, sildenafil

Group Type EXPERIMENTAL

sildenafil

Intervention Type DRUG

25 mg quaque die (QD) or 50 mg QD x 4 days based on subject weight

calcitonin

Intervention Type DRUG

one nasal spray daily for 4 days

hydrochlorothiazide/amiloride

Intervention Type DRUG

25 mg/2.5 mg BID or 50 mg/5 mg BID x 8 days depending on subject weight

indomethacin

Intervention Type DRUG

50 mg QD or 50 mg BID x 8 days depending on subject weight

Placebo Control

4-day treatment with hydrochlorothiazide/amiloride, indomethacin, placebo for calcitonin, placebo for sildenafil

Group Type PLACEBO_COMPARATOR

hydrochlorothiazide/amiloride

Intervention Type DRUG

25 mg/2.5 mg BID or 50 mg/5 mg BID x 8 days depending on subject weight

indomethacin

Intervention Type DRUG

50 mg QD or 50 mg BID x 8 days depending on subject weight

Placebo for sildenafil

Intervention Type DRUG

one tablet daily for 4 days

placebo for calcitonin

Intervention Type DRUG

one nasal spray daily

Interventions

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sildenafil

25 mg quaque die (QD) or 50 mg QD x 4 days based on subject weight

Intervention Type DRUG

calcitonin

one nasal spray daily for 4 days

Intervention Type DRUG

hydrochlorothiazide/amiloride

25 mg/2.5 mg BID or 50 mg/5 mg BID x 8 days depending on subject weight

Intervention Type DRUG

indomethacin

50 mg QD or 50 mg BID x 8 days depending on subject weight

Intervention Type DRUG

Placebo for sildenafil

one tablet daily for 4 days

Intervention Type DRUG

placebo for calcitonin

one nasal spray daily

Intervention Type DRUG

Other Intervention Names

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Viagra Miacalcic Moduret, Moduretic Indocin

Eligibility Criteria

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Inclusion Criteria

* Known diagnosis of Congenital Nephrogenic Diabetes Insipidus (CNDI)
* Age 5 to 25 years
* Normal kidney function
* Post-void residual urine \< 200 ml (determined by bladder ultrasound)

Exclusion Criteria

* Impaired kidney function
* Known urinary retention or bladder dysfunction
* High blood pressure
* Other significant chronic medical disease (e.g., heart failure, liver disease, etc.)
* Allergy to study drugs
Minimum Eligible Age

5 Years

Maximum Eligible Age

25 Years

Eligible Sex

MALE

Accepts Healthy Volunteers

No

Sponsors

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University of Aarhus

OTHER

Sponsor Role collaborator

University of Colorado, Denver

OTHER

Sponsor Role lead

Responsible Party

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Responsibility Role SPONSOR

Principal Investigators

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Melissa A Cadnapaphornchai, MD

Role: PRINCIPAL_INVESTIGATOR

University of Colorado, Denver

Locations

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University of Colorado at Denver and Health Sciences Center

Aurora, Colorado, United States

Site Status

University of Aarhus

Aarhus, , Denmark

Site Status

Countries

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United States Denmark

References

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Bichet DG. Nephrogenic diabetes insipidus. Adv Chronic Kidney Dis. 2006 Apr;13(2):96-104. doi: 10.1053/j.ackd.2006.01.006.

Reference Type BACKGROUND
PMID: 16580609 (View on PubMed)

Fujiwara TM, Bichet DG. Molecular biology of hereditary diabetes insipidus. J Am Soc Nephrol. 2005 Oct;16(10):2836-46. doi: 10.1681/ASN.2005040371. Epub 2005 Aug 10.

Reference Type BACKGROUND
PMID: 16093448 (View on PubMed)

Schrier RW, Cadnapaphornchai MA. Renal aquaporin water channels: from molecules to human disease. Prog Biophys Mol Biol. 2003 Feb;81(2):117-31. doi: 10.1016/s0079-6107(02)00049-4.

Reference Type BACKGROUND
PMID: 12565698 (View on PubMed)

Schrier RW, Cadnapaphornchai MA, Umenishi F. Water-losing and water-retaining states: role of water channels and vasopressin receptor antagonists. Heart Dis. 2001 May-Jun;3(3):210-4. doi: 10.1097/00132580-200105000-00014.

Reference Type BACKGROUND
PMID: 11975794 (View on PubMed)

Nielsen S, Kwon TH, Frokiaer J, Agre P. Regulation and dysregulation of aquaporins in water balance disorders. J Intern Med. 2007 Jan;261(1):53-64. doi: 10.1111/j.1365-2796.2006.01760.x.

Reference Type BACKGROUND
PMID: 17222168 (View on PubMed)

Kotnik P, Nielsen J, Kwon TH, Krzisnik C, Frokiaer J, Nielsen S. Altered expression of COX-1, COX-2, and mPGES in rats with nephrogenic and central diabetes insipidus. Am J Physiol Renal Physiol. 2005 May;288(5):F1053-68. doi: 10.1152/ajprenal.00114.2004. Epub 2005 Jan 11.

Reference Type BACKGROUND
PMID: 15644490 (View on PubMed)

Nielsen S, Frokiaer J, Marples D, Kwon TH, Agre P, Knepper MA. Aquaporins in the kidney: from molecules to medicine. Physiol Rev. 2002 Jan;82(1):205-44. doi: 10.1152/physrev.00024.2001.

Reference Type BACKGROUND
PMID: 11773613 (View on PubMed)

Bouley R, Pastor-Soler N, Cohen O, McLaughlin M, Breton S, Brown D. Stimulation of AQP2 membrane insertion in renal epithelial cells in vitro and in vivo by the cGMP phosphodiesterase inhibitor sildenafil citrate (Viagra). Am J Physiol Renal Physiol. 2005 Jun;288(6):F1103-12. doi: 10.1152/ajprenal.00337.2004. Epub 2005 Jan 11.

Reference Type BACKGROUND
PMID: 15644488 (View on PubMed)

Related Links

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http://www.ndif.org

Nephrogenic Diabetes Insipidus Foundation

Other Identifiers

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06-0588

Identifier Type: -

Identifier Source: org_study_id

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