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Alport SyndromeAlport-XXLNCT02378805

Alport Therapy Registry - European Initiative Towards Delaying Renal Failure in Alport Syndrome

An observational study of Alport Syndrome and Hereditary Kidney Disease, run by University Hospital Goettingen at 1 site in Germany.

Recruiting

Looking for participants now. Contact the study team or a site below to ask about taking part.

Sites and contacts
Official record
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Trial summary

The hereditary type IV collagen disease Alport syndrome leads to kidney failure early in life. Currently there are no specific medications approved for treatment, however, several therapies have been evaluated preclinically and could improve outcome. Read more about this study

Who is it for?
Any ageHealthy volunteers can take part
What is being tested?
ACE-inhibitor, Angiotensin-receptor blocker (ARB) and HMG-Coenzyme inhibitor (statin)
Type of treatment
Injection or infusion
How many people
800 participants
Where
1 siteGermany
Study dates
Jul 1995to 1 Mar 2036
How long will I take part?
30 Years
Phase
Observational
Are there results?
Not yet: the study is under way

Key requirements

Who can take part, as listed by the sponsor. Only the study team can confirm whether you are eligible, so take this list to your doctor.

  • Ages Any age
  • All sexes
  • Healthy volunteers accepted
  • Child, Adult, Older adult

Study population: For Alport XXL, eligible patients were children or young adults with a definitive diagnosis of AS by genetic analysis or kidney biopsy and at early stages of AS (stages 0, I or II) at baseline. AS stages were defined as: Stage 0 Microhematuria without microalbuminuria Stage I Microalbuminuria: UACR 30-300 mg/gCreatinine Stage II Proteinuria: UACR >300 with eGFR>60 ml/min/1.73m2 Stage III eGFR<60 ml/min/1.73m2 Stage IV end stage kidney failure Stage V death (from all causes)

Who may be able to join

  • Diagnosis of Alport syndrome (AS) by kidney biopsy or mutation analysis (or both).
  • Any type of genetic variant is accepted for X-linked, autosomal or digenic Alport syndrome (COL4A3, 4 or 5 genes).

Who cannot join

  • Patients not willing to give informed consent. Patient with suspected diagnosis, whcih cannot be confirmed.

Trial locations and contacts

1 site in 1 country. Contact the study team or a site near you to ask about taking part.

Germany1 site, 1 recruiting
  • University Medical Center Göttingen

    Göttingen, Lower Saxony, 37075

    Oliver Gross, MD, +49-551-39- ext. 60488, gross.oliver@med.uni-goettingen.de

    Recruiting

Other trials in Germany

About this study

Official title: European Alport Therapy Registry - European Initiative Towards Delaying Renal Failure in Alport Syndrome: Current and Novel Therapies

The hereditary type IV collagen disease Alport syndrome leads to kidney failure early in life. Currently there are no specific medications approved for treatment, however, several therapies have been evaluated preclinically and could improve outcome. For that reason, this non-interventional, observational study investigates, if medications (1) delay disease progression; (2) delay time to kidney failure; (3) improve life-expectancy compared to untreated patients (relatives). This observational study started in 2006 as an European registry. Since 2019, this registry has been expanded to "Alport XXL" via the International Alport Alliance as a global effort across all continents. From 2020 on to present, "Alport XXL" has a special focus on the outcomes of early therapy in young patients on ACE-inhibitors vs. Angiotensin-receptor blockers vs. their combination.

Read the detailed description

Early diagnosis in children with Alport syndrome (AS) with isolated hematuria opens a "window of opportunity" for early intervention. In the Alport mouse-model, this early intervention with the ACE-inhibitor Ramipril let to a delay of kidney failure by 111%. In order to observe treatment approaches for AS in humans, this registry has been established in 2006 to collect data over several generations of Alport families across Europe. In the meantime, this registry has been expanded to "Alport XXL" via the International Alport Alliance as a global effort across all continents.

Small children with AS first develop microscopic hematuria (stage 0), proceeding to microalbuminuria (stage I), overt proteinuria (stage II), impaired kidney function (stage III) and finally can end up with kidney failure (stage IV), leading to impaired quality of life and premature death (stage V). This registry uses these stages to assess if earlier initiation of medications such as ACE-inhibition at earlier stages of disease is more effective than later therapy in delaying the time to disease progression (doubeling or tripeling of albuminuria), delaying loss of estimated glomerular filtration rate (eGFR), and if therapy improves life-expectancy.

Untreated children with autosomal-recessive AS, digenic AS, and boys with X-linked AS typically all develop kidney failure early in life. Untreated girls with X-linked AS have a 30-40% risk of kidney failure, typically later in life (40 years or older). Untreated heterozygous patients with COL4A3/COL4A4 variants typically have a less severe phenotype (in former times also called "familial benign hematuria" or "thin basement membrane nephropathy" (TBMN)) and a 1-2% risk of kidney failure.

Several interim results of this registry have been published since 2012.

Alport XXL is designed and conducted as strictly observational, non-interventional data acquisition with prospective (and in parts retrospective) data analysis. Young patients with AS in disease stages 0,I,II from all over the world are included. The renewed version from 2021 has been re-approved by the Ethics Committee of the University Medical Center Göttingen as "Alport XXL", a further development of the former European Alport Therapy Registry (AZ 10/11/06). "Alport XXL" registry and data storage are in conformity with Good Clinical Practice guidelines.

ICH-GCP-conform patient information and data exchange is secured by data transfer and cooperation agreements between all international trial centers and the coordinating principal investigator at University Medical Center Goettingen. At baseline, data collection including retrospective data is performed using a standardized, ICH-GCP-conform and pseudonymized questionnaire assessing age, sex, weight, height, mode of inheritance (X-linked, autosomal, compound heterozygous/homozygous, number of missense variants), family history, albumin in 24-hour or spontaneous urine, serum-creatinine, RAS-blockade with preparation and dose. Follow-up visits include same data than baseline plus blood-pressure, smoking-status, serum-potassium, eGFR, hearing loss and eye involvement, other symptoms such as leiomyomatosis, comorbidities and adverse events (adverse events of special interest defined as hyperkalemia, cough, hypotension, acute renal failure, malignancy, death).

Being tested
ACE-inhibitor, Angiotensin-receptor blocker (ARB), HMG-Coenzyme inhibitor (statin), Spironolactone or Finerenone, Paricalcitol
Keywords
Alport syndrome, thin basement membrane disease, familial benign hematuria
Medical subject headings
Nephritis, Hereditary, Hematuria, Benign Familial

What participants receive

What participants receive. In a randomised study each person is assigned to one group by chance.

  • ACE-inhibitorDrug

    observational study

  • Angiotensin-receptor blocker (ARB)Drug

    observational study

  • HMG-Coenzyme inhibitor (statin)Drug

    observational study

  • Spironolactone or FinerenoneDrug

    observational study

  • ParicalcitolDrug

    observational study!

  • SGLT2 inhibitorDrug

    observational study

Study groups in NCT02378805
Study groupRoleWhat they receive
no-T: untreated patientsuntreated patients, typically uncles or grandfathers of present patients. No Intervention (means no therapy until CKD stage V, on renal replacement therapy)
T-III: late therapy in patientspatients treated with medications with low eGFR (below 60 ml/min) (starts at patients with CKD stages III and IV).
Receives: Drug: ACE-inhibitor; Drug: Angiotensin-receptor blocker (ARB); Drug: HMG-Coenzyme inhibitor (statin); Drug: Spironolactone or Finerenone; Drug: Paricalcitol; Drug: SGLT2 inhibitor
T-II: early therapy in patientstherapy starts in patients with albuminuria >300mg/gCreatinine and eGFR higher than 60 ml/min.
Receives: Drug: ACE-inhibitor; Drug: Angiotensin-receptor blocker (ARB); Drug: HMG-Coenzyme inhibitor (statin); Drug: Spironolactone or Finerenone; Drug: Paricalcitol; Drug: SGLT2 inhibitor
T-I: very early therapy in patientstherapy starts in patients with microhematuria only (usually at birth) or microalbuminuria (30-300 mg albumin per gCreatinine).
Receives: Drug: ACE-inhibitor; Drug: Angiotensin-receptor blocker (ARB); Drug: HMG-Coenzyme inhibitor (statin); Drug: Spironolactone or Finerenone; Drug: Paricalcitol; Drug: SGLT2 inhibitor
no therapy in heterozygous patientsheterozygous patients without therapy
therapy in heterozygous patientsheterozygous patients with therapy (which also can be divided into subgroups stage T-0, I, II, III)
Receives: Drug: ACE-inhibitor; Drug: Angiotensin-receptor blocker (ARB); Drug: HMG-Coenzyme inhibitor (statin); Drug: Spironolactone or Finerenone; Drug: Paricalcitol; Drug: SGLT2 inhibitor

What is being measured

The primary outcome is the main question the study is designed to answer.

  1. age at onset of end stage kidney failure (need for renal replacement therapy)

    kidney disease in Alport syndrome starts at birth, age at onset of end stage kidney failure in years

    Measured: until end of observation in 2037

  2. life-expectancy in years (age at death)

    Alport syndrome starts at birth, age at death of patients in years

    Measured: until end of observation in 2037

  3. yearly loss of estimated glomerular filtration rate (eGFR)

    kidney disease in Alport syndrome starts at birth, eGFR-loss per year in ml/min/1.73m2

    Measured: until end of observation in 2037

  4. time in years until doubling or tripling of urinary albumin to creatinine ratio (UACR)

    kidney disease in Alport syndrome starts at birth; time since birth to doubling (AS stages I or II) or tripling (AS stage 0) of UACR in years.

    Measured: until end of observation in 2037

Secondary outcomes (8)
  1. amount of albuminuria over time

    kidney disease in Alport syndrome starts at birth; change in albuminuria after birth

    Measured: until end of observation in 2037

  2. amount of proteinuria over time

    kidney disease in Alport syndrome starts at birth; change in proteinuria after birth

    Measured: until end of observation in 2037

  3. number of patients with X-chromosomal or autosomal inheritance

    specific genetic variant: X-linked, digenic, autosomal heterozygous, homozygous, compound heterozygous

    Measured: until end of observation in 2037

  4. number of patients experiencing side effects (AEs)

    AEs of special interest: acute kidney renal failure, angioedema, hyperkalemia, dry cough, symptomatic hypotension (orthostatic collapse) and others, and death from all causes.

    Measured: until end of observation in 2037

  5. number of patients with hearing loss

    Alport syndrome is a genetic disease, hearing loss delevops over time since birth

    Measured: until end of observation in 2037

  6. number of patients with eye involvement

    Alport syndrome is a genetic disease, eye symptoms delevop over time since birth

    Measured: until end of observation in 2037

  7. number of patients with positive family history of end stage kidney failure

    number of relatives with kidney failure

    Measured: until end of observation in 2037

  8. age of relatives at end stage kidney failure

    age of relatives at start of renal replacement therapy in years

    Measured: until end of observation in 2037

Timeline

From the start of the study to its completion; the mark is today. Estimated dates are the sponsor’s plan.

  1. First posted4 Mar 2015 (estimated)
  2. Study startJul 1995
  3. Primary completion1 Mar 2036 (estimated)
  4. Study completion1 Mar 2036 (estimated)

Resources

More details

For doctors and researchers: how the study is designed, who runs it, and its registry IDs.

Study design
Study type
Observational
Observational model
Cohort
Time perspective
Cross-sectional
Enrolment
800 participants (estimated)
Target duration
30 Years
IDs, dates and publications
ClinicalTrials.gov ID
NCT02378805
Sponsor's protocol ID
Alport-UMG2010
Acronym
Alport-XXL
Status last verified
Mar 2025
First submitted
26 Feb 2015
Last updated
6 Mar 2025
Results posted
No
Data monitoring committee
No
FDA-regulated device
No
Individual data sharing
Undecided: Investigators plan to share the full data set with other researchers upon reasonable request via written cooperation agreements.

Publications

  • Kashtan CE, Gross O. Clinical practice recommendations for the diagnosis and management of Alport syndrome in children, adolescents, and young adults-an update for 2020. Pediatr Nephrol. 2021 Mar;36(3):711-719. doi: 10.1007/s00467-020-04819-6. Epub 2020 Nov 6.

    PubMed 33159213
  • Weinstock BA, Feldman DL, Fornoni A, Gross O, Kashtan CE, Lagas S, Lennon R, Miner JH, Rheault MN, Simon JF; Workshop Participants. Clinical trial recommendations for potential Alport syndrome therapies. Kidney Int. 2020 Jun;97(6):1109-1116. doi: 10.1016/j.kint.2020.02.029. Epub 2020 Apr 6.

    PubMed 32386680
  • Temme J, Kramer A, Jager KJ, Lange K, Peters F, Muller GA, Kramar R, Heaf JG, Finne P, Palsson R, Reisaeter AV, Hoitsma AJ, Metcalfe W, Postorino M, Zurriaga O, Santos JP, Ravani P, Jarraya F, Verrina E, Dekker FW, Gross O. Outcomes of male patients with Alport syndrome undergoing renal replacement therapy. Clin J Am Soc Nephrol. 2012 Dec;7(12):1969-76. doi: 10.2215/CJN.02190312. Epub 2012 Sep 20.

    PubMed 22997344
  • Temme J, Peters F, Lange K, Pirson Y, Heidet L, Torra R, Grunfeld JP, Weber M, Licht C, Muller GA, Gross O. Incidence of renal failure and nephroprotection by RAAS inhibition in heterozygous carriers of X-chromosomal and autosomal recessive Alport mutations. Kidney Int. 2012 Apr;81(8):779-83. doi: 10.1038/ki.2011.452. Epub 2012 Jan 11.

    PubMed 22237748
  • Gross O, Licht C, Anders HJ, Hoppe B, Beck B, Tonshoff B, Hocker B, Wygoda S, Ehrich JH, Pape L, Konrad M, Rascher W, Dotsch J, Muller-Wiefel DE, Hoyer P; Study Group Members of the Gesellschaft fur Padiatrische Nephrologie; Knebelmann B, Pirson Y, Grunfeld JP, Niaudet P, Cochat P, Heidet L, Lebbah S, Torra R, Friede T, Lange K, Muller GA, Weber M. Early angiotensin-converting enzyme inhibition in Alport syndrome delays renal failure and improves life expectancy. Kidney Int. 2012 Mar;81(5):494-501. doi: 10.1038/ki.2011.407. Epub 2011 Dec 14.

    PubMed 22166847
  • Stock J, Kuenanz J, Glonke N, Sonntag J, Frese J, Tonshoff B, Hocker B, Hoppe B, Feldkotter M, Pape L, Lerch C, Wygoda S, Weber M, Muller GA, Gross O. Prospective study on the potential of RAAS blockade to halt renal disease in Alport syndrome patients with heterozygous mutations. Pediatr Nephrol. 2017 Jan;32(1):131-137. doi: 10.1007/s00467-016-3452-z. Epub 2016 Jul 11.

    PubMed 27402170
  • Frese J, Kettwig M, Zappel H, Hofer J, Grone HJ, Nagel M, Sunder-Plassmann G, Kain R, Neuweiler J, Gross O. Kidney Injury by Variants in the COL4A5 Gene Aggravated by Polymorphisms in Slit Diaphragm Genes Causes Focal Segmental Glomerulosclerosis. Int J Mol Sci. 2019 Jan 26;20(3):519. doi: 10.3390/ijms20030519.

    PubMed 30691124
  • Boeckhaus J, Hoefele J, Riedhammer KM, Nagel M, Beck BB, Choi M, Gollasch M, Bergmann C, Sonntag JE, Troesch V, Stock J, Gross O. Lifelong effect of therapy in young patients with the COL4A5 Alport missense variant p.(Gly624Asp): a prospective cohort study. Nephrol Dial Transplant. 2022 Nov 23;37(12):2496-2504. doi: 10.1093/ndt/gfac006.

    PubMed 35022790
  • Zhang Y, Bockhaus J, Wang F, Wang S, Rubel D, Gross O, Ding J. Genotype-phenotype correlations and nephroprotective effects of RAAS inhibition in patients with autosomal recessive Alport syndrome. Pediatr Nephrol. 2021 Sep;36(9):2719-2730. doi: 10.1007/s00467-021-05040-9. Epub 2021 Mar 27.

    PubMed 33772369
  • Boeckhaus J, Gross O. Sodium-Glucose Cotransporter-2 Inhibitors in Patients with Hereditary Podocytopathies, Alport Syndrome, and FSGS: A Case Series to Better Plan a Large-Scale Study. Cells. 2021 Jul 18;10(7):1815. doi: 10.3390/cells10071815.

    PubMed 34359984
  • Boeckhaus J, Gale DP, Simon J, Ding J, Zhang Y, Bergmann C, Turner AN, Hall M, Sayer JA, Srivastava S, Kang HG, Cerkauskaite-Kerpauskiene A, Gillion V, Claes KJ, Krueger B, de Fallois J, Walden U, Choi M, Schueler M, Mueller RU, Todorova P, Hohenstein B, Zeisberg M, Friede T, Knebelmann B, Halbritter J, Gross O. SGLT2-Inhibition in Patients With Alport Syndrome. Kidney Int Rep. 2024 Sep 24;9(12):3490-3500. doi: 10.1016/j.ekir.2024.09.014. eCollection 2024 Dec.

    PubMed 39698346

Questions about this trial

What is this clinical trial studying?

An observational study of Alport Syndrome and Hereditary Kidney Disease, run by University Hospital Goettingen at 1 site in Germany.

The full registered description is in the About this study section and the official record.

Who may be eligible to take part?

The sponsor lists 2 inclusion and 1 exclusion criteria. Eligibility is decided by the study team after screening, so read the full criteria and contact a listed site.

Is this clinical trial currently recruiting?

The record says: looking for participants now, as of the sponsor's last update on 6 Mar 2025. Status can change between updates, so confirm with the study contact before making plans.

Where is the study taking place?

The record lists 1 site in Germany. Whether a site is taking part right now should be confirmed with that site. See every site.

What are the possible benefits and risks?

They depend on the study and the treatment, and no study can promise a benefit. The study team explains the known risks during informed consent, before anyone joins; the record itself does not list them.

Are results available for this study?

This study is still under way. Results are not available in the source record yet.

How can a doctor refer a patient or contact the research team?

Contact Oliver Gross, MD (+49-551-39- ext. 60488, gross.oliver@med.uni-goettingen.de), or a site near the patient. AzTrials does not enrol participants or take referrals.

What does the primary outcome measure?

age at onset of end stage kidney failure (need for renal replacement therapy) (time frame: until end of observation in 2037). The study lists 4 primary outcomes in all. It is what the study was designed to measure, not a finding.

Where can I read the original study information?

On the ClinicalTrials.gov record for NCT02378805, which also lists 11 linked publications.

Does a listing mean the treatment is approved or proven to work?

No. A listing means the study is registered. It does not show that the treatment is safe, works, or is approved by a regulator.

How often is this information updated?

AzTrials copies changed records from ClinicalTrials.gov every night and reloads the whole registry every week. The sponsor last updated this record on 6 Mar 2025.

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