MedPath

Transthyretin Amyloidosis Outcome Survey (THAOS)

Completed
Conditions
Transthyretin Gene Mutations
Transthyretin Amyloidosis
Interventions
Other: None. Observational Study.
Registration Number
NCT00628745
Lead Sponsor
Pfizer
Brief Summary

THAOS is a global, multi-center, longitudinal observational survey open to all patients with transthyretin amyloidosis (ATTR), including ATTR-PN (polyneuropathy), ATTR-CM (cardiomyopathy) and wild-type ATTR-CM. It is open-ended with a minimum duration of 10 years. Patients will be followed as long as they are able to participate. The principal aims of this outcome survey are to better understand and characterize the natural history of the disease by studying a large and heterogenous patient population. Survey data may be used to develop new treatment guidelines and recommendations, and to inform and educate clinicians about the management of this disease.

Detailed Description

n/a NA

Recruitment & Eligibility

Status
COMPLETED
Sex
All
Target Recruitment
6718
Inclusion Criteria

Patients must meet all of the following inclusion criteria to be eligible for inclusion into THAOS:

  1. Evidence of a personally signed and dated informed consent document indicating that the participant (or a legally acceptable representative) has been informed of all pertinent aspects of the study.

  2. Males and females greater than or equal to 18 years of age.

  3. Confirmed genotyped TTR mutation with or without a diagnosis of hereditary or wild-type ATTR amyloidosis. Confirmation of ATTRwt amyloidosis will be determined by genotyped confirmation that patient does not possess a known mutation in TTR gene (ie, is a carrier of wild-type allele only) via genetic testing and one of the following set of criteria (a, b, or c):

    1. Presence of amyloid in cardiac biopsy tissue confirmed as TTR amyloid by mass spectrometry or immunohistochemistry; or
    2. Evidence of cardiac involvement by echocardiogram as defined by left ventricle wall thickness of >12 mm, and presence of amyloid in non-cardiac tissue confirmed as TTR amyloid by mass spectrometry or immunohistochemistry; or
    3. Evidence of cardiac involvement by echocardiogram as defined by left ventricle wall thickness of >12 mm, and presence of amyloid in cardiac tissue indirectly confirmed by scintigraphy with a "bone seeking tracer" eg, 99mTC-DPD [99mTC-3,3-diphosphono-1,2-propano-dicarboxylic acid], 99mTC- PYP [Pyrophosphate], and 99mTC-HMDP [hydroxymethylene diphosphonate] with Perugini grade greater than or equal to 2.

Exclusion Criteria

Patients meeting any of the following will not be included in the study:

  1. Patient has evidence of primary (light chain) or secondary amyloidosis.
Read More
Exclusion Criteria

Not provided

Read More

Study & Design

Study Type
OBSERVATIONAL
Study Design
Not specified
Arm && Interventions
GroupInterventionDescription
ObservationalNone. Observational Study.-
Primary Outcome Measures
NameTimeMethod
Number of Participants With Treatment-Emergent Adverse Events (AEs) and Serious Adverse Events (SAEs)From the start of data collection at Baseline (Day 1) to the end of data collection (Up to 14.4 years)

An AE was any untoward medical occurrence in a participant who administered a medicinal product without regard to possibility of causal relationship with the study treatment. SAE was defined as one of the following: resulted in death; was life-threatening (immediate risk of death); requires inpatient hospitalization or prolongation of hospitalization; resulted in persistent or significant disability/incapacity (substantial disruption of the ability to conduct normal life functions); constituted a congenital anomaly/birth defect. Treatment-emergent AE was defined as an AE with onset date occurring during the on-treatment period. AEs included all SAEs and non-SAEs.

Number of Participants With Treatment Emergent Treatment Related AEs and SAEsFrom the start of data collection at Baseline (Day 1) to the end of data collection (Up to 14.4 years)

A treatment-related AE was any untoward medical occurrence attributed to the administered medicinal product in a participant who received study drug. Treatment emergent AEs included both SAEs and all non-SAEs. A treatment-related SAE was a treatment-related AE and was defined as any untoward medical occurrence at any dose that: resulted in death; was life-threatening (immediate risk of death); requires inpatient hospitalization or prolongation of hospitalization; resulted in persistent or significant disability/incapacity (substantial disruption of the ability to conduct normal life functions); constituted a congenital anomaly/birth defect. Causality was assessed by the investigator.

Number of All-Cause DeathsFrom the start of data collection at Baseline (Day 1) to the end of data collection (Up to 14.4 years)

Number of deaths due to any cause was analyzed as time from enrollment or first treatment of tafamidis.

Secondary Outcome Measures
NameTimeMethod
Number of Participants With Modified Polyneuropathy Disability (mPND) Scores at BaselineAt the start of data collection at Baseline (Day 1)

Modified Polyneuropathy Disability (mPND) is a score that categorizes participants into six stages (0, I,II, IIIa, IIIb, IV) based on mobility status. 0 = No sensory disturbances in the feet and able to walk without difficulty; I=Sensory disturbances in the feet but able to walk without difficulty; II=Some difficulties with walking but can walk without aid; IIIa=Able to walk with 1 stick or crutch; IIIb=Able to walk with 2 sticks or crutches; IV=Not ambulatory, confined to a wheelchair or bedridden. Higher stage indicates lower mobility status.

Number of Participants With Coutinho Disease Stages at BaselineAt the start of data collection at Baseline (Day 1)

Coutinho disease stages is the most common classification used to capture ATTR (Transthyretin Amyloidosis) disease progression. Participants with stage 0 disease are asymptomatic, Participants with stage 1 (mild) disease are ambulatory, Participants with stage 2 (moderate) disease are ambulatory but require assistance and/or have involvement of the upper limbs, and Participants with stage 3 (severe) disease are bedridden or wheelchair-bound.

Number of Participants With Karnofsky Performance Index at BaselineAt the start of data collection at Baseline (Day 1)

Karnofsky performance score is used to quantify participant's general well-being and activities of daily life and participants are classified based on their functional impairment. Karnofsky performance score is 10 level score which ranges between 10 (moribund) to 100 (normal, no evidence of disease). Higher score means higher ability to perform daily tasks.

Number of Participants With Heart Failure at BaselineAt the start of data collection at Baseline (Day 1)

Heart failure, also known as congestive heart failure is a cardiovascular event.

Number of Participants With New York Heart Association (NYHA) Classifications at BaselineAt the start of data collection at Baseline (Day 1)

New York Health Association (NYHA) functional classification included: Class I (no limitation in physical activity, no dyspnea with normal activity), Class II (slight limitation of physical activity; fatigue, palpitation, or dyspnea with ordinary physical activity), Class III (marked limitation of physical activity; fatigue, palpitation, or dyspnea with less than ordinary physical activity) and Class IV (cannot perform a physical activity without any symptoms, dyspnea at rest).

Number of Participants Diagnosed With ATTR at BaselineAt the start of data collection at Baseline (Day 1)

Participants diagnosed with Transthyretin Amyloidosis (ATTR) at baseline with assessed category of yes, no, and unknown.

Number of Participants With Prior Misdiagnosis at BaselineAt the start of data collection at Baseline (Day 1)

Number of participants with ATTR and participants who had prior misdiagnosis at the baseline were reported.

Number of Participants With ATTR Genotypes at BaselineAt the start of data collection at Baseline (Day 1)

Genetic mutation leads to misfolding of protein transthyretin (TTR) which results in ATTR. In this outcome, number of participants with ATTRv mutation type and wild type TTR were reported.

Number of Participants With Past or Current Clinical Trial Participation at BaselineAt the start of data collection at Baseline (Day 1)

Number of participants had previous or current participant in any clinical trials at the baseline.

Number of Participants With Past or Current Tafamidis Compassionate Use/Early Access or Other Non-commercial Program at BaselineAt the start of data collection at Baseline (Day 1)

Number of participants being allowed for the use of Tafamidis when under strict conditions, Tafamidis was in development and made available to groups of participants who have a disease with no satisfactory authorised therapies and who cannot enter clinical trials.

Number of Participants With Known Family History of Symptomatic ATTR at BaselineAt the start of data collection at Baseline (Day 1)

Number of participants whether with family history of symptomatic ATTR amyloidosis at Baseline were reported.

Number of Affected Generations at BaselineAt the start of data collection at Baseline (Day 1)

The mean of affected generations in participants with a known family history was reported.

Derived Neuropathy Impairment Score-Lower Limb (NIS-LL) at BaselineAt the start of data collection at Baseline (Day 1)

Neuropathy Impairment Score - Lower Limb (NIS-LL) assessed motor, sensory and reflex activity specifically in the lower limbs and combined total scores for the lower limbs were collected and reported. Derived NIS LL score extends from 0 (normal functions) to a maximum possible value of 88 points, the scale is additive for all deficits and is applied bilaterally for each modality tested: 1) muscle strength: 0 (normal)-4 (paralysis), higher score = more weakness; 2) sensory and 3) reflex testings: 0=normal, 1=decreased, or 2=absent. Reflex score: 0 (normal)-10 (present), higher score =present in more anatomic sites; Motor score: 0-160 (full range of motion with maximum resistance across all anatomical sites), higher score=more impairment. Sensory Score has a range of 0 to the normal value of 124 where ratings are coded as 0=absent; 1=decreased; 2=normal.

Body Mass Index (BMI) at BaselineAt the start of data collection at Baseline (Day 1)

BMI was calculated by weight divided by height squared and measured as kilogram per square meter (kg/m\^2).

Modified Body Mass Index (mBMI) at BaselineAt the start of data collection at Baseline (Day 1)

mBMI was calculated by multiplying BMI by serum albumin levels \[gram/liter (g/L)\]. mBMI was measured as kg/m\^2\*g/L. A progressive decline in mBMI indicated worsening of disease severity.

Sitting Systolic and Diastolic Blood Pressures (SBP and DBP) at BaselineAt the start of data collection at Baseline (Day 1)

BP (Blood Pressure) is the pressure of the blood within the arteries. It is produced primarily by the contraction of the heart muscle. BP measurement is recorded by 2 numbers: systolic BP (SBP, BP when heart is contracting; it is the maximum arterial pressure during contraction of left ventricle) and diastolic BP (DBP, BP when heart is relaxing; it is the minimum arterial pressure during relaxation and dilation of ventricles). Available sitting SBP and DBP at Baseline were reported.

Left Ventricular (LV) Septum Thickness at BaselineAt the start of data collection at Baseline (Day 1)

Cardiac amyloidosis is attributable to intramyocardial amyloid infiltration, which leads to a progressive increase of ventricular wall thickness and stiffness. A left ventricular (LV) wall thickness ≥12 mm plus at least one red flag should raise the suspicion of cardiac amyloidosis.

Left Ventricular (LV) Ejection Fraction at BaselineAt the start of data collection at Baseline (Day 1)

Left ventricular dysfunction, a condition in which the left ventricle of the heart exhibits a decreased functionality, was assessed based on systolic ejection fraction. Systolic ejection fraction was the fraction of the end-diastolic volume (EDV) that was ejected out of left ventricle with each contraction.

Euro Quality of Life-5 Dimensions-3 Level (EQ-5D-3L): Visual Analog Scale (VAS) Overall Health Score at BaselineAt the start of data collection at Baseline (Day 1)

EQ-5D-3L VAS: participant rated questionnaire to assess generic health status in two parts: single utility score and visual analog scale. The VAS component rated the current health state on a scale ranging from 0 (worst imaginable health state) to 100 (best imaginable health state); higher scores indicating a better health state.

EQ-5D-3L: VAS Derived Index at BaselineAt the start of data collection at Baseline (Day 1)

The EQ-5D-3L VAS derived index is calculated by subtracting the values of the descriptive EQ-5D system from the numerical value. This corresponds to the best possible health status, the scale of the Derived Index is 0 \[death\] to 1 \[perfect health\]. EQ-5D-3L VAS overall health score and derived score data were sourced from different part of the EQ-5D questionnaire and are conceptually different from each other as EQ VAS is a 0-100 scale and EQ-5D index is a value attached to an EQ-5D profile according to a set of weights that reflect, on average, participant's preferences about how good or bad the state is. More data were collected for EQ-5D index score compared to EQ VAS overall health score at the baseline.

Norfolk Total Quality of Life (QoL) Score at BaselineAt the start of data collection at Baseline (Day 1)

Norfolk QOL: 35-item participant-rated questionnaire used to assess impact of neuropathy on the quality of life of participants diagnosed with ATTR. Scoring was based on 35 questions that yield a TQOL as well as 5 subscale scores: activities of daily living, large fiber neuropathy/physical functioning, small fiber neuropathy, autonomic neuropathy, and symptoms. TQOL= sum of all the items, total possible score range= -2 to 138, where higher score=worse quality of life.

Number of Participants With Abnormal Electrocardiogram (ECG) at BaselineAt the start of data collection at Baseline (Day 1)

Following parameters were analyzed: heart rate, PR interval, QT interval, QRS interval and QT interval corrected using Fridericia's formula (QTcF). Abnormal findings in ECG were based on investigator's discretion.

Number of Participants With Atrial Fibrillation/Flutter, Pacemaker Implanted, and Implantable Cardioverter/Defibrillator (ICD) at BaselineAt the start of data collection at Baseline (Day 1)

Number of participants with atrial fibrillation/flutter (rapid, irregular heart rhythm), implanted artificial cardiac pacemaker, and implantable cardioverter-defibrillator (ICD) (detects and stops irregular heartbeats, also called arrhythmias) were reported.

Trial Locations

Locations (112)

The University of Utah Health Sciences Center

🇺🇸

Salt Lake City, Utah, United States

Medical College of Wisconsin

🇺🇸

Milwaukee, Wisconsin, United States

Hospital Italiano de Buenos Aires (HIBA)

🇦🇷

Buenos Aires, Argentina

FLENI

🇦🇷

Ciudad Autonoma de Buenos aires, Argentina

Instituto De Investigaciones Medicas Dr Alfredo Lanari

🇦🇷

Ciudad Autonoma De Buenos Aires, Argentina

Afdeling Klinische Cardiologie, O&N I

🇧🇪

Leuven, Belgium

Hospital Universitário Clementino Fraga Filho -HUCFF Universidade Federal do Rio de Janeiro

🇧🇷

Rio de Janeiro, Brazil

Instituto Dante Pazzanese De Cardiologia

🇧🇷

Sao Paulo, Brazil

Alexandrovska University Hospital Clinic of Neurology

🇧🇬

Sofia, Bulgaria

University of Alberta Foothills Medical Centre

🇨🇦

Calgary, Alberta, Canada

Toronto General Hospital-University Health Network

🇨🇦

Toronto, Ontario, Canada

Toronto General Hospital

🇨🇦

Toronto, Ontario, Canada

Cyprus Institute of Neurology and Genetics

🇨🇾

Nicosia, Cyprus

Aarhus University Hospital, Skejby

🇩🇰

Aarhus, Denmark

CHU de Bicetre

🇫🇷

Cedex, France

Hopital Louis Pasteur

🇫🇷

Colmar, France

CHU Henri Mondor

🇫🇷

Créteil, France

Chu De Fort De France

🇫🇷

Fort De France, France

CHRU de Lille, Hopital Claude Huriez

🇫🇷

Lille, France

Hopital Salengro - CHRU de Lille

🇫🇷

Lille, France

CHU de Toulouse - Hopital Rangueil

🇫🇷

Toulouse cedex 09, France

University Hospital of RWTH Aachen

🇩🇪

Aachen, Germany

Charite CAmpus Rudolf-Virchow-Klinikum

🇩🇪

Berlin, Germany

Medical University of Heidelberg

🇩🇪

Heidelberg, Germany

Johann-Gutenberg-Universität

🇩🇪

Mainz, Germany

University Medical Center, Johannes Gutenberg-University Mainz

🇩🇪

Mainz, Germany

Universitätsmedizin der Johannes Gutenberg Universität Mainz KöR

🇩🇪

Mainz, Germany

Universitatsklinikum Muenster - Transplant Hepatology

🇩🇪

Muenster, Germany

Wolfson Medical Center

🇮🇱

Holon, Israel

Fondazione Policlinico Gemelli - Universita Cattolica del Sacro Cuore

🇮🇹

Roma, Italy

Sheba Medical Center

🇮🇱

Ramat Gan, Israel

Comitato Etico Indipendente dell Azienda

🇮🇹

Bologna, Italy

Azienda Ospedaliero-Universitaria di Careggi

🇮🇹

Firenze, Italy

AOU Policlinico G. Martino - Messina - Comitato Etico Scientifico

🇮🇹

Messina, Italy

AOU Policlinico G. Martino - Messina - Dr. Vita

🇮🇹

Messina, Italy

Centro per lo Studio e la Cura delle Amiloidosi Sistemiche - Pavia - Prof. Merlini

🇮🇹

Pavia, Italy

Comitato di Bioetica della Fondazione IRCCS Policlinico S. Matteo

🇮🇹

Pavia, Italy

Fondazione Toscana Gabriele Monasterio Per La Ricerca Medica E Di Sanita Pubblica (Ftgm)

🇮🇹

Pisa, Italy

Shinshu University School of Medicine

🇯🇵

Matsumoto, JP, Japan

Chiba University Hospital

🇯🇵

Chiba, Japan

Kumamoto University

🇯🇵

Kumamoto, Japan

Samsung Medical Center, Sungkyunkwan University School of Medicine

🇰🇷

Seoul, Korea, Republic of

Konkuk University Medical Center

🇰🇷

Seoul, Korea, Republic of

University Malaya Medical Centre (UMMC)

🇲🇾

Kuala Lumpur, Wilayah Persekutuan, Malaysia

Instituto Nacional de Ciencia Medicas y Nutricion Salvador Zubiran

🇲🇽

Distrito Federal, Mexico

University Medical Center Groningen

🇳🇱

Groningen, Netherlands

Centro Hospitalar Do Alto Ave, Epe

🇵🇹

Guimaraes, Portugal

Centro Hospitalar Lisboa Norte (CHLN) EPE - Hospital de Santa Maria

🇵🇹

Lisboa, Portugal

Hospital Santa Maria

🇵🇹

Lisboa, Portugal

Unidade Clinica de Paramiloidose-Centro Hospitalar Porto,EPE-Hospital Geral Santo Antonio

🇵🇹

Porto, Portugal

Institutul De Cardiologie Prof. Dr. C. C. Iliescu Spitalului Fundeni

🇷🇴

Bucuresti, Romania

King Faisal Specialist Hospital and Research Center

🇸🇦

Riyadh, Saudi Arabia

Hospital Universitario Donostia

🇪🇸

Gipuzkoa - SanSebastian, Donostia, Spain

Hospital Universitario Puerta de Hierro

🇪🇸

Majadahonda, Madrid, Spain

Institut Clinic de Nefrologia i Urologia - ICNU, Hospital Clinic i Provincial de Barcelona

🇪🇸

Barcelona, Spain

Hospital Universitari de Bellvitge

🇪🇸

Barcelona, Spain

Hospital de Rehabilitacion y Traumatologia Virgen de las Nieves

🇪🇸

Granada, Spain

Hospital Juan Ramon Jimenez

🇪🇸

Huelva, Spain

Hospital Gregorio Marañón

🇪🇸

Madrid, Spain

Hospital Clinico San Carlos

🇪🇸

Madrid, Spain

Piteå Älvdals Hospital

🇸🇪

Piteå, Sweden

Karolinska University Hospital, Huddinge

🇸🇪

Stockholm, Sweden

Umeå University Hospital

🇸🇪

Umeå, Sweden

National Taiwan University Hospital

🇨🇳

Taipei, Taiwan

Istanbul University,Istanbul Faculty of Medicine,Department of Neurology

🇹🇷

Istanbul, Turkey

Cleveland Clinic Abu Dhabi LLC

🇦🇪

Abu Dhabi, United Arab Emirates

Johns Hopkins Hospital

🇺🇸

Baltimore, Maryland, United States

Harvard Vanguard Medical Associates

🇺🇸

Boston, Massachusetts, United States

University of Michigan

🇺🇸

Ann Arbor, Michigan, United States

Mayo Clinic

🇺🇸

Rochester, Minnesota, United States

Washington University School of Medicine

🇺🇸

Saint Louis, Missouri, United States

Cardiology Clinic at Montefiore Hutchinson Campus

🇺🇸

Bronx, New York, United States

Montefiore Medical Center-Jack D. Weiler Hospital

🇺🇸

Bronx, New York, United States

Congestive Heart Failure Clinic

🇺🇸

Bronx, New York, United States

Montefiore Moses Division

🇺🇸

Bronx, New York, United States

NYU Medical Center

🇺🇸

New York, New York, United States

Columbia University Medical Center

🇺🇸

New York, New York, United States

Cleveland Clinic Foundation

🇺🇸

Cleveland, Ohio, United States

The Ohio University College of Medicine

🇺🇸

Columbus, Ohio, United States

OSU Wexner Medical Center

🇺🇸

Columbus, Ohio, United States

Oregon Health and Science University

🇺🇸

Portland, Oregon, United States

Peters Township Health and Wellness Pavillion

🇺🇸

McMurray, Pennsylvania, United States

Penn Presbyterian Medical Center-University of Pennsylvania

🇺🇸

Philadelphia, Pennsylvania, United States

University of Pennsylvania - Perelman Center for Advanced Medicine

🇺🇸

Philadelphia, Pennsylvania, United States

Temple University School of Medicine

🇺🇸

Philadelphia, Pennsylvania, United States

Allegheny General Hospital

🇺🇸

Pittsburgh, Pennsylvania, United States

Admin

🇺🇸

Pittsburgh, Pennsylvania, United States

University of Pittsburgh Medical Center (UPMC)

🇺🇸

Pittsburgh, Pennsylvania, United States

Wexford Health and Wellness Pavillion

🇺🇸

Wexford, Pennsylvania, United States

IRB

🇺🇸

Nashville, Tennessee, United States

Vanderbilt University School of Medicine

🇺🇸

Nashville, Tennessee, United States

University of Maryland

🇺🇸

Baltimore, Maryland, United States

Hospital Son Llàtzer

🇪🇸

Palma de Mallorca, Spain

University of Alabama at Birmingham

🇺🇸

Birmingham, Alabama, United States

Mayo Clinic Arizona

🇺🇸

Phoenix, Arizona, United States

VA Greater Los Angeles Healthcare System

🇺🇸

Los Angeles, California, United States

University of California, Irvine

🇺🇸

Orange, California, United States

University of California - San Francisco, UCSF Department of Neurology

🇺🇸

San Francisco, California, United States

Office of Sponsored Research

🇺🇸

San Francisco, California, United States

Stanford University School of Medicine

🇺🇸

Stanford, California, United States

UC Denver

🇺🇸

Aurora, Colorado, United States

Smilow Cancer Hospital at Yale-New Haven

🇺🇸

New Haven, Connecticut, United States

UHealth Deerfield Beach

🇺🇸

Deerfield Beach, Florida, United States

University of Miami Hospital & Clinics

🇺🇸

Miami, Florida, United States

UHealth Plantation

🇺🇸

Plantation, Florida, United States

Clinical Trials Unit

🇺🇸

Chicago, Illinois, United States

Northwestern University

🇺🇸

Chicago, Illinois, United States

University of Chicago Medical Center IRB

🇺🇸

Chicago, Illinois, United States

University of Chicago Medical Center

🇺🇸

Chicago, Illinois, United States

Advocate Christ Medical Centre

🇺🇸

Oak Lawn, Illinois, United States

John Ochsner Heart & Vascular Institute

🇺🇸

New Orleans, Louisiana, United States

University of Maryland School of Medicine

🇺🇸

Baltimore, Maryland, United States

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