Table 3 Modeling approach and the sources of parameter values in the included studies

Margaret C. Garin [13]

NA

1-year

3-month

FC I (0)

FC II (0)

FC III (90%)

FC IV (10%)

Death (0)

Drug costs; hospitalization costs;

outpatient costs;

cost of follow-up examination;

cost of ADR treatment

(All the above costs are from medical insurance reimbursement data)

The transition probability between each FC state (RCT);

the transition probability from each FC state to the death state (RCT)

The process of discounting was not carried out

The survey conducted by Keogh et al.

The sensitivity analysis was conducted on specific parameters

Y-F Chen [12]

Healthcare system

30-year

3-month

FC I (0)

FC II (0 or 100%)

FC III (100% or 0)

FC IV (0)

Death (0)

Drug costs (national drug formulary);

hospitalization costs (published literature);

cost of liver-function tests (hospital data);

cost of ADR treatment related to epoprostenol (National Health Service reference cost)

The transition probability between each FC state (RCT);

the transition probability from each FC state to the death state (RCT)

The costs and health outcomes were both discounted annually at a rate of 3.5%

The survey conducted by Keogh et al. (base-case analysis); the study by Kirsch et al. (sensitivity analysis); the study by Olschewski et al. (sensitivity analysis)

The sensitivity analyses were conducted by employing diverse utility values

Antonio Roman [14]

Healthcare system

3-year

3-month

FC I (0)

FC II (0)

FC III (100%)

FC IV (10%)

Death (0)

Drug costs (ex-factory price of drugs);

costs of disease management (Spanish costs database)

The transition probability between each FC state (RCT);

the transition probability from each FC state to the death state (RCT)

The costs and health outcomes were both discounted annually at a rate of 3%

The survey conducted by Keogh et al. (base-case analysis);

Spanish population-based utility provided by an expert panel (sensitivity analysis)

Deterministic sensitivity analyses;

probabilistic sensitivity analysis

CADTH

2015 [15]

Healthcare system

30-year

3-month

FC I

FC II

FC III

FC IV

Death

(The distribution of different FC states at the initial state was not provided.)

Drug costs (pharmaceutical companies);

cost of follow-up examination (published literature);

hospitalization costs (published literature);

cost of ADR treatment (published literature)

The transition probability between each FC state (network meta-analysis);

the transition probability from each FC state to the death state (published literature and Canadian life tables)

The costs and health outcomes were both discounted annually at a rate of 5%

The survey conducted by Keogh et al.

Deterministic sensitivity analyses;

probabilistic sensitivity analysis

Watsamon

Thongsri [18]

Social

Lifetime

3-month

FC I (0)

FC II (0 or 100%)

FC III (100% or 0)

FC IV (0)

Death (0)

Drug costs (pharmaceutical companies);

cost of outpatient visits (hospital database);

cost of hospital admission (hospital database);

direct nonmedical cost and indirect cost (interviewing patients using questionnaire)

The transition probability between each FC state (published literature);

the transition probability from each FC state to the death state (published literature)

The costs and health outcomes were both discounted annually at a rate of 3%

The utility scores were collected from patients using the EQ-5D questionnaire

Deterministic sensitivity analyses;

threshold sensitivity analysis;

probabilistic sensitivity analysis

Fan [17]

NA

Lifetime

1-year

FC I (3.14%)

FC II (45.59%)

FC III (41.92%)

FC IV (5.36%)

Death (0)

Drug costs;

cost of follow-up examination

(all the above costs are from the data of the hospital)

The transition probability between each FC state (Bosentan Charity Project);

the transition probability from each FC state to the death state (national registry)

The costs were discounted annually at a rate of 3.5%;

the health outcomes were not discounted.

The survey conducted by Kirsch et al. (base-case analysis);

The survey conducted by Highland et al. (sensitivity analysis)

The sensitivity analysis was conducted by employing diverse utilities

Kathryn Coyle [16]

Healthcare system

30-year

3-month

FC I (0)

FC II (0 or 100%)

FC III (100% or 0)

FC IV (0)

Death (0)

Drug costs (provincial drug formulary);

cost of follow-up examination

(schedule of benefits for physician services under the Health Insurance Act)

The transition probability between each FC state (network meta-analysis);

the transition probability from each FC state to the death state (published literature and Canadian life tables)

The costs and health outcomes were both discounted annually at a rate of 5%

The survey conducted by Keogh et al.

Deterministic sensitivity analyses;

probabilistic sensitivity analysis

CADTH

2017 [19]

Payer

30-year

3-month

FC I (0)

FC II (40%)

FC III (60%)

FC IV (0)

Death (0)

Drug costs (pharmaceutical companies, local drug formularies, expert consultation);

cost of ADR treatment (expert consultation, local government departments);

hospitalization costs (the data of the hospital)

The transition probability between each FC state (RCT);

the transition probability from each FC state to the death state (RCT)

The discount value was not specified

The survey conducted by Keogh et al.

Deterministic sensitivity analyses;

probabilistic sensitivity analysis; scenario analysis

Oktavia Lilyasari [20]

Social

Lifetime

3-month

FC I

FC II

FC III

FC IV

Death

(The patients were included in the model while being in a functional class II or III state)

Drug costs (hospital database);

cost of outpatient visits (hospital database);

cost of hospital admission (hospital database);

direct nonmedical cost (interviewing patients using questionnaire)

The transition probability between each FC state (published literature);

the transition probability from each FC state to the death state (published literature)

The costs and health outcomes were both discounted annually at a rate of 3%

Interviewing patients using questionnaire

Deterministic sensitivity analyses;

probabilistic sensitivity analysis

Marzieh Nosrati [21]

Healthcare system

Lifetime

3-month

FC I (0)

FC II (100%)

FC III (0)

FC IV (0)

Death (0)

Drug costs (local generic market price);

cost of outpatient visits (published literature);

cost of follow-up examination (published literature)

The transition probability between each FC state (published literature);

the transition probability from each FC state to the death state (published literature and life tables)

The costs were discounted annually at a rate of 5%;

the health outcomes were discounted annually at a rate of 3%

Published literature

Deterministic sensitivity analyses;

model validation

Zhao [22]

Healthcare system

5-year

10-year

Lifetime

3-month

FC I (3.14%)

FC II (49.59%)

FC III (41.92%)

FC IV (5.36%)

Death

(The initial distribution data of the cohort were obtained from the Patient Registry of the Bosentan Charity Program)

Drug costs (hospital database);

cost of follow-up examination (hospital database)

The transition probability between each FC state (network meta-analysis);

the transition probability from each FC state to the death state (published literature)

The costs and health outcomes were both discounted annually at a rate of 4.1%

The survey conducted by Keogh et al.

Deterministic sensitivity analyses;

probabilistic sensitivity analysis

Mahna Ekhlasi [23]

Healthcare system

Lifetime

3-month

FC I (0)

FC II (50.7%)

FC III (49.3%)

FC IV (0)

Death (0)

(The initial distribution data of the cohort were obtained from the published)

Drug costs;

cost of outpatient visits;

cost of hospital admission;

cost of follow-up examination

cost of ADR treatment

(All costs were extracted from the official healthcare services price list of Iran)

The transition probability between each FC state (RCT);

the transition probability from each FC state to the death state (published literature and life tables)

The costs were discounted annually at a rate of 7.2%;

the health outcomes were discounted annually at a rate of 5%

The survey conducted by Keogh et al.

Deterministic sensitivity analyses;

probabilistic sensitivity analysis

Dong [24]

Healthcare system

30-year

3-month

FC I (0)

FC II (50%)

FC III (50%)

FC IV (0)

Death

Drug costs (local generic market price);

cost of outpatient visits (local generic market price);

cost of hospital admission (published literature);

cost of follow-up examination (local generic market price)

The transition probability between each FC state (RCT);

the transition probability from each FC state to the death state (published literature and life tables)

The costs and health outcomes were both discounted annually at a rate of 5%

The survey conducted by Keogh et al.

Deterministic sensitivity analyses;

probabilistic sensitivity analysis; scenario analysis