---
title: "Molgramostim Inhalation Solution in Autoimmune pulmonary alveolar proteinosis: cost-effectiveness analyses and utility weights"
description: "Published cost-effectiveness analyses and health-state utility weights for Molgramostim Inhalation Solution in Autoimmune pulmonary alveolar proteinosis, each value quoted from the article: 8 cost-effectiveness analyses and 7 studies reporting utility weights. Analyses: Nintedanib and pirfenidone versus symptom management in idiopathic pulmonary fibrosis, United States: Dempsey et al. (2022); Nintedanib versus placebo, both with usual care, in progressive fibrosing interstitial lung disease, Netherlands: Westerink et al. (2022); Augmentation therapy until FEV1 is below 35% predicted or for life versus no augmentation therapy in severe alpha1-antitrypsin deficiency, United States: Gildea et al. (2003); Tobramycin inhalation powder versus tobramycin inhalation solution in cystic fibrosis with chronic Pseudomonas aeruginosa infection, United States: Panguluri et al. (2017); 4 further articles. Utility weights: Utility weights by percent-predicted forced vital capacity and after acute exacerbation, idiopathic pulmonary fibrosis: Dempsey et al. (2022); Utility weights by percent predicted forced vital capacity and for acute exacerbations, idiopathic pulmonary fibrosis: Porte et al. (2018); Utility weights and disability weights for progression-free, progressed, and acute exacerbation states, idiopathic pulmonary fibrosis: Zhao et al. (2023); Utility weights for the lung transplant waiting list, after lung transplantation, and end-stage pulmonary disease: Groen et al. (2004); 3 further articles. Every value is a verbatim quote from the primary article it is shown with."
canonical: "https://www.evicenter.com/drugs/molgramostim-inhalation-solution--autoimmune-pulmonary-alveolar-proteinosis/cost-effectiveness-analyses"
published: "2026-10-22T07:49:06-04:00"
modified: "2026-10-22T07:49:06-04:00"
---
# Molgramostim Inhalation Solution in Autoimmune pulmonary alveolar proteinosis: cost-effectiveness analyses and utility weights
Published cost-effectiveness analyses and health-state utility weights that a model of this product could cite, adapt, or compare against, grouped by how each article relates to the product and its indication. Every value is quoted from the article it cites, and section 3 lists the searches that found the articles.
## Overview
| Item | Detail |
| --- | --- |
| Literature searched | Tufts Medical Center Cost-Effectiveness Analysis Registry, searched October 21, 2026; the queries are listed in section 3. |
| Cost-effectiveness analyses | 8 |
| Studies reporting utility weights | 7 |
## 1 Cost-effectiveness analyses
### 1.1 Analyses of Molgramostim Inhalation Solution
No evidence found.
### 1.2 Treatments for the same indication
No evidence found.
### 1.3 The same disease in other populations or settings
No evidence found.
### 1.4 Related diseases and analogous treatments
#### Nintedanib and pirfenidone versus symptom management in idiopathic pulmonary fibrosis, United States: Dempsey et al. (2022)
Relevance: Evaluates nintedanib and pirfenidone against symptom management in idiopathic pulmonary fibrosis from a United States health care payer perspective, with utilities assigned by percent-predicted forced vital capacity.
> “We used a previously published Markov model (from the UK) that incorporates U.S.-specific data and project the lifetime costs and health benefits of treating IPF with: (1) symptom management; (2) pirfenidone; or (3) nintedanib”
> “We assumed a health care payer’s perspective.”
> “All future costs and quality-adjusted life years (QALYs) were discounted at 3% per year”
> “Symptom management was associated with lifetime costs and benefits of $79,815 and 3.78 QALYs, respectively.”
> “Nintedanib was associated with lifetime costs and benefits of $675,544 and 4.15 QALYs, respectively.”
> “This amounts to an incremental cost-effectiveness ratio of $1.6 million per QALY”
> “Pirfenidone had lifetime costs of $688,778 and benefits of 4.10 QALYs.”
> “In our model, pirfenidone was slightly more expensive than nintedanib and provided the same amount of benefit”
Source: [Dempsey et al. (2022)](https://pmc.ncbi.nlm.nih.gov/articles/PMC8744245/)
#### Nintedanib versus placebo, both with usual care, in progressive fibrosing interstitial lung disease, Netherlands: Westerink et al. (2022)
Relevance: Evaluates nintedanib against placebo in progressive fibrosing interstitial lung disease in the Netherlands, with lung transplantation among its modeled states; section 2 lists lung transplantation as an option for refractory autoimmune pulmonary alveolar proteinosis.
> “An individual patient simulation model was built in Microsoft Excel.”
> “The long-term clinical effects, costs and cost-effectiveness were forecasted for treatment with nintedanib versus placebo, both on top of UC in patients with PF-ILD, using a 10-year time horizon as the follow-up period.”
> “To account for future devaluations, a societal perspective with discounting rates of 4.0% for costs and 1.5% for effects and a time horizon of 10 years were applied, in line with Dutch guideline recommendations”
> “Overall, treatment with nintedanib was predicted to result in 2.87 discounted QALYs versus 2.00 discounted QALYs for placebo, both on top of UC.”
> “The total costs per patient over 10 years of treatment were €91,301 for nintedanib versus €38,358 for placebo, both on top of UC.”
> “this produced an incremental cost-effectiveness ratio (ICER) of €40,418 per life year gained (LYG) and an ICER of €60,690 per QALY.”
> “When lung transplantation was excluded from the model, the ICER increased to €84,926 per QALY.”
Source: [Westerink et al. (2022)](https://pmc.ncbi.nlm.nih.gov/articles/PMC9440187/)
#### Augmentation therapy until FEV1 is below 35% predicted or for life versus no augmentation therapy in severe alpha1-antitrypsin deficiency, United States: Gildea et al. (2003)
Relevance: Evaluates augmentation therapy for life and until a lung function threshold against no augmentation in severe alpha1-antitrypsin deficiency in the United States, with health states defined by percent-predicted FEV1 and by lung transplantation.
> “A Markov-based decision model was created to assess the cost-effectiveness of augmentation therapy (Aug) for severe alpha1-antitrypsin deficiency, comparing strategies of: (1) no Aug, (2) Aug for life, and (3) Aug until FEV1 is below 35% predicted.”
> “A hypothetical cohort of 46-year-old patients with FEV1 49% predicted was followed over time using Monte Carlo simulation across five possible health states: (1) FEV1 50 to 79% predicted, (2) FEV1 35 to 49% predicted, (3) FEV1 below 35% predicted, (4) status-post-lung transplantation, and (5) dead.”
> “Treatment for life yielded 7.19 quality-adjusted life-years (QALYs) and cost 895,243 dollars.”
> “Treating until FEV1 is below 35% predicted cost 511,930 dollars and produced 6.64 QALYs.”
> “"No Aug" cost 92,091 dollars with 4.62 QALYs.”
> “The incremental cost-effectiveness ratio was 207,841 dollars/QALY for Aug until FEV1 is below 35% predicted and 312,511 dollars/QALY for the "Aug for life" strategy.”
> “In all sensitivity analyses, the incremental cost-effectiveness ratio for Aug for life exceeded 100,000 dollars.”
Source: [Gildea et al. (2003)](https://pubmed.ncbi.nlm.nih.gov/12574076/)
#### Tobramycin inhalation powder versus tobramycin inhalation solution in cystic fibrosis with chronic Pseudomonas aeruginosa infection, United States: Panguluri et al. (2017)
Relevance: Evaluates tobramycin inhalation powder against tobramycin inhalation solution for chronic Pseudomonas aeruginosa infection in cystic fibrosis from a United States payer perspective.
> “A patient-level simulation model was developed to compare TIP with TIS.”
> “Both costs and benefits were predicted over a 10-year time horizon from a payer’s perspective, and were discounted annually at 3%.”
> “All costs were presented in 2016 US dollars.”
> “The model was developed from the US healthcare perspective over a time horizon of 10 years, with a cycle length of 6 months.”
> “The use of TIP over TIS was associated with cost savings per patient over a time horizon of 10 years for nebuliser and exacerbation costs when high adherence to TIP and low adherence to TIS was assumed”
> “The incremental total costs were lower (by US$36,168) and QALYs gained were higher (by 0.27) with TIP as compared to TIS.”
> “This equates to lower costs with TIP per QALY gained (−US$133,195/QALY) for the base case (ICER).”
> “TIP was dominant over TIS in 100% of the simulations”
Source: [Panguluri et al. (2017)](https://pmc.ncbi.nlm.nih.gov/articles/PMC5509782/)
#### Aztreonam lysine for inhalation versus tobramycin solution for inhalation in cystic fibrosis with chronic Pseudomonas aeruginosa infection, United States: Schechter et al. (2015)
Relevance: Evaluates aztreonam lysine for inhalation against tobramycin solution for inhalation in cystic fibrosis from a United States third-party payer perspective, with health states defined by percent-predicted FEV1.
> “A cost-effectiveness analysis with a 3-year time horizon was performed to simulate the economic consequences of either treatment from the perspective of a third party payer in the United States.”
> “A Markov structure was used to consider transitions between health states, defined principally by levels of percent predicted of FEV1.”
> “Use of aztreonam lysine for inhalation was associated with an average cost saving of $41,947 per patient over 3 years, as well as greater quality-adjusted life-years and total life-years.”
> “Scenario analyses demonstrated that these findings were robust to changes in key assumptions.”
Source: [Schechter et al. (2015)](https://pubmed.ncbi.nlm.nih.gov/26053185/)
#### Inhaled mannitol added to best supportive care versus best supportive care in cystic fibrosis, Australia: Warren et al. (2019)
Relevance: Evaluates inhaled mannitol added to best supportive care against best supportive care alone in cystic fibrosis from the perspective of the Australian government-funded national healthcare system.
Access: Not open access
Not quoted: The results by concomitant dornase alfa use are reported in the full text, which is closed access.
> “The objective of this study was to assess the cost effectiveness of inhaled mannitol (in addition to best supportive care) in the Australian setting from the perspective of a government-funded national healthcare system.”
> “A probabilistic patient-level simulation Markov model estimated life-time costs and outcomes of mannitol when added to best supportive care, compared with best supportive care alone in patients aged 6 years and older.”
> “The utility value for the cystic fibrosis health state was as measured in the trials using the Health Utility Index, whereas the impact of pulmonary exacerbations and lung transplantation on utility was ascertained from the published literature.”
> “The addition of inhaled mannitol to best supportive care resulted in a discounted cost per quality-adjusted life-year of AU$39,165.”
> “The result was robust with 77% of probabilistic sensitivity analysis samples below a willingness-to-pay threshold of AU$45,000/quality-adjusted life-year.”
> “our model suggests inhaled mannitol provides a cost-effective addition to best supportive care in patients with cystic fibrosis, irrespective of concomitant dornase alfa use.”
Source: [Warren et al. (2019)](https://pubmed.ncbi.nlm.nih.gov/30666534/)
#### Lung transplantation versus no transplantation by type of end-stage pulmonary disease, Netherlands: Groen et al. (2004)
Relevance: Evaluates lung transplantation by type of end-stage pulmonary disease in the Netherlands; section 2 lists lung transplantation as an option for refractory autoimmune pulmonary alveolar proteinosis.
Access: Free full text
> “A microsimulation model was used, based on empirical data from the Dutch lung transplantation program, collected between 1991 and 1999.”
> “We assessed life-years, quality-adjusted life-years, and costs with and without transplantation for the diagnostic categories alfa-1 antitrypsin deficiency, COPD/emphysema, bronchiectasis, primary and secondary pulmonary hypertension, cystic fibrosis, and pulmonary fibrosis.”
> “All cost categories considered relevant from a societal perspective were included in the model”
> “The inflow period is followed by a 25-year follow-up period.”
> “For the purpose of the current evaluation, all costs were indexed to price levels of the year 2000.”
> “Because of the length of the simulated time period, future costs and effects were discounted at 5% per year.”
> “Secondary pulmonary hypertension and pulmonary fibrosis had the lowest survival gain and the lowest gain of quality-adjusted life-years.”
> “As compared with COPD/emphysema, alfa-1 antitrypsin deficiency, bronchiectasis, and CF had 25%, 40% and 19% more favorable cost-effectiveness ratios, respectively.”
> “Cost-utility ratios varied less, with values of –7%, –14% and –11% for alfa-1 antitrypsin deficiency, bronchiectasis, and primary pulmonary hypertension, respectively, compared with COPD.”
> “Table 4 Cost-effectiveness ratios and cost-utility ratios of model outcomes per diagnosis at various discount rates (×1000 US dollars)”
> “COPD = chronic obstructive pulmonary disease, ATD = alfa-1 antitrypsin deficiency, BE = bronchiectasis, PPH = primary pulmonary hypertension, SPH = secondary pulmonary hypertension, CF = cystic fibrosis, PF = pulmonary fibrosis, CER = cost-effectiveness ratio, CUR = cost-utility ratio.”
Source: [Groen et al. (2004)](https://doi.org/10.1111/j.1600-6143.2004.00479.x)
| Diagnosis | Mean cost-utility ratio (SE), 5% discount rate (×1000 US dollars) |
| --- | --- |
| “COPD” | “89.9 (0.77)” |
| “ATD” | “83.4 (0.55)” |
| “BE” | “77.0 (0.37)” |
| “PPH” | “79.9 (0.62)” |
| “SPH” | “88.7 (0.94)” |
| “CF” | “83.2 (0.59)” |
| “PF” | “86.9 (0.70)” |
Source: [Groen et al. (2004)](https://doi.org/10.1111/j.1600-6143.2004.00479.x)
#### Continuous and nocturnal long-term oxygen therapy versus no oxygen therapy in chronic obstructive pulmonary disease, United States: Oba (2009)
Relevance: Evaluates continuous and nocturnal long-term oxygen therapy in chronic obstructive pulmonary disease in the United States; section 2 lists oxygen supplementation as supportive care, and the IMPALA-2 protocol permitted it.
> “A Markov model was developed to estimate the incremental cost-effectiveness ratios (ICERs) for continuous and nocturnal oxygen therapies.”
> “The maximum time horizon was set to 5 years.”
> “Cost variables were based on the current Medicare reimbursement rate and on appropriate sources.”
> “The ICER for continuous oxygen therapy ($16,124 per quality-adjusted life-year [QALY]) was within bounds considered to be cost-effective, while that of nocturnal oxygen therapy was not ($306,356/QALY).”
> “The estimated ICER for continuous oxygen therapy was robust (95% confidence interval, $13,153-$24,658/QALY)”
> “The ICER for nocturnal oxygen therapy was sensitive to variation in the mortality rate; it could be as low as $18,267/QALY gained.”
> “At the other end, nocturnal oxygen therapy could be less effective than no oxygen therapy, despite additional costs.”
Source: [Oba (2009)](https://pubmed.ncbi.nlm.nih.gov/19284806/)
## 2 Utility weights
### 2.1 Utility weights from analyses of Molgramostim Inhalation Solution
No evidence found.
### 2.2 Utility weights for the same indication
No evidence found.
### 2.3 Utility weights for the same disease in other populations or settings
No evidence found.
### 2.4 Utility weights for related diseases and analogous treatments
#### Utility weights by percent-predicted forced vital capacity and after acute exacerbation, idiopathic pulmonary fibrosis: Dempsey et al. (2022)
Relevance: EQ-5D utilities by percent-predicted lung function band and post-exacerbation disutilities in idiopathic pulmonary fibrosis, from a United States model.
> “used the EQ-5D to measure QOL by % forced vital capacity (FVC)”
> “assumed that exacerbations decreased QOL by 0.140 in the initial cycle that it is experienced and decreased QOL by 0.078 in subsequent months”
Source: [Dempsey et al. (2022)](https://pmc.ncbi.nlm.nih.gov/articles/PMC8744245/)
| Health state | Utility |
| --- | --- |
| “90–110%” | “0.8380” |
| “80–89.9%” | “0.8105” |
| “70–79.9%” | “0.7800” |
| “60–69.9%” | “0.7657” |
| “50–59.9%” | “0.7387” |
| “40–49.9%” | “0.6634” |
Source: [Dempsey et al. (2022)](https://pmc.ncbi.nlm.nih.gov/articles/PMC8744245/)
#### Utility weights by percent predicted forced vital capacity and for acute exacerbations, idiopathic pulmonary fibrosis: Porte et al. (2018)
Relevance: Utilities by percent-predicted forced vital capacity and for acute exacerbations in idiopathic pulmonary fibrosis, from a lifetime model adapted to the French setting.
Access: Not open access
Not quoted: The utility weights are reported in the full text, which is closed access.
> “A previously developed lifetime Markov model was adapted to the French setting by simulating the progression of IPF patients in terms of lung function decline, incidence of acute exacerbations, and death.”
> “Considering the effect of IPF on patients' quality-of-life, a CUA integrating quality adjusted life years (QALY) was chosen as the primary outcome measure in the main analysis.”
Source: [Porte et al. (2018)](https://pubmed.ncbi.nlm.nih.gov/29368948/)
#### Utility weights and disability weights for progression-free, progressed, and acute exacerbation states, idiopathic pulmonary fibrosis: Zhao et al. (2023)
Relevance: Utilities and disability weights for progression-free, progressed, and acute exacerbation states in idiopathic pulmonary fibrosis, with utilities abstracted preferentially as EQ-5D from the trials of a network meta-analysis.
> “For estimating QALYs, data were collected from the enrolled RCTs in our NMA, which reported utility scores of quality of life in different health states related to the economic model, comprising EuroQol Five Dimensions Questionnaire (EQ-5D) and St. George's Respiratory Questionnaire (SGRQ) index.”
> “EQ-5D was preferentially abstracted, when reported.”
> “Global DALYs were sourced from Global Burden of Disease (GBD) Study 2017.”
Source: [Zhao et al. (2023)](https://pmc.ncbi.nlm.nih.gov/articles/PMC10331814/)
| Health state | Utility |
| --- | --- |
| “Progression-free survival (QALYs)” | “0.88” |
| “Progressed disease (QALYs)” | “0.82” |
| “Acute exacerbation (QALYs)” | “0.60” |
| “Progression-free survival (DALYs)” | “0.02” |
| “Progressed disease (DALYs)” | “0.22” |
| “Acute exacerbation (DALYs)” | “0.41” |
Source: [Zhao et al. (2023)](https://pmc.ncbi.nlm.nih.gov/articles/PMC10331814/)
#### Utility weights for the lung transplant waiting list, after lung transplantation, and end-stage pulmonary disease: Groen et al. (2004)
Relevance: Utilities for the lung transplant waiting list, after lung transplantation, and for end-stage pulmonary disease, for the lung transplantation that section 2 lists for refractory disease.
Access: Free full text
> “Utility scores were derived from EuroQol questionnaires (12) taken every 3 months on the waiting list, 1, 4 and 7 months after transplantation, and every 6 months thereafter.”
> “On the waiting list, average utility values were 0.55 during the first 6 months, 0.50 between 6 and 9 months, 0.45 between 9 and 12 months, and 0.40 after 1 year.”
> “The utility score during the screening phases was assumed to be 0.50, the average of the waiting list data.”
> “For the purpose of the model, utility was set at 0.85 during the first 2 years after transplantation and to 0.90 after that period.”
> “In any phase, the utility during the final 3 months before a patient died was 0.31.”
> “The small number of transplantations in the Netherlands did not allow distinction by diagnosis.”
Source: [Groen et al. (2004)](https://doi.org/10.1111/j.1600-6143.2004.00479.x)
#### Utility weights after lung transplantation and for gastrointestinal adverse events, progressive fibrosing interstitial lung disease: Westerink et al. (2022)
Relevance: Utilities derived from EQ-5D-3L data, with different baseline utilities after lung transplantation and decrements for treatment-related gastrointestinal adverse events, in progressive fibrosing interstitial lung disease.
Access: Free full text
Not quoted: The utility and disutility values are reported in Appendix C of the Electronic Supplementary Material, which the article text does not include.
> “A regression equation was used to determine patient-specific health state utilities based on EQ-5D-3L data”
> “UK preference weights were used to derive the utilities because no Dutch patients were included in INBUILD”
> “The economic model assumed utility decrements associated with treatment-related adverse events, and in the case of lung transplantation, different baseline utilities were applied.”
Source: [Westerink et al. (2022)](https://pmc.ncbi.nlm.nih.gov/articles/PMC9440187/)
#### Utility weights before and after liquid oxygen and concentrator treatment, chronic hypoxaemia: Andersson et al. (1998)
Relevance: Utilities in chronic hypoxaemia before and after domiciliary oxygen treatment.
Access: Free full text
Not quoted: The utility weights are reported in the full text, a PDF at the publisher’s site that could not be retrieved for checking, so this page does not quote them.
> “A prospective, randomized multicentre trial comparing C/C with L for LTOT was conducted during a six-month period.”
> “Fifty-one patients (29 on L and 22 on C/C) with chronic hypoxaemia, regularly active outside the home, participated in the study initially.”
> “Health-related quality of life was measured by the Sickness Impact Profile (SIP) and the EuroQol, instruments at the start and after 6 months.”
Source: [Andersson et al. (1998)](https://pubmed.ncbi.nlm.nih.gov/9877478/)
#### Utility weights for long-term oxygen therapy without and with moderate or severe exacerbation, chronic obstructive pulmonary disease: Groessl et al. (2023)
Relevance: Utilities for chronic obstructive pulmonary disease requiring long-term oxygen therapy without exacerbation and with moderate or severe exacerbation, from a United States healthcare system model.
> “With our analysis based on a US healthcare system perspective, we estimated the utility of our sample to be 0.630 when no exacerbations were present using estimates from the National Health and Wellness Survey (NHWS)”
> “utility decrements of 15% for moderate exacerbations and 50% for severe exacerbations resulted in monthly utility values of 0.045 and 0.026”
> “This translates to a monthly decrement of 0.008 and 0.027 for moderate and severe exacerbations, respectively.”
Source: [Groessl et al. (2023)](https://pmc.ncbi.nlm.nih.gov/articles/PMC10083032/)
| Health state | Utility |
| --- | --- |
| “COPD needing LTOT – no exacerbation” | “0.630” |
| “COPD needing LTOT – moderate exacerbation” | “0.536” |
| “COPD needing LTOT – severe exacerbation” | “0.315” |
Source: [Groessl et al. (2023)](https://pmc.ncbi.nlm.nih.gov/articles/PMC10083032/)
## 3 Search strategy
| Dataset | Query | Results |
| --- | --- | --- |
| Methods | "alveolar proteinosis" OR molgramostim OR sargramostim OR "whole lung lavage" | 0 |
| Ratios | "alveolar proteinosis" OR molgramostim OR sargramostim OR "lung lavage" OR "GM-CSF" | 0 |
| Weights | "alveolar proteinosis" OR "lung lavage" OR "GM-CSF" | 0 |
| Ratios | "granulocyte-macrophage" OR "granulocyte macrophage" OR "bronchoalveolar lavage" OR "pulmonary alveolar" | 6 |
| Ratios | "idiopathic pulmonary fibrosis" OR "interstitial lung disease" | 43 |
| Ratios | "alpha-1 antitrypsin" OR "alpha1-antitrypsin" OR "lung transplantation" OR "lung transplant" | 67 |
| Ratios | "nebulized" OR "nebulised" OR "nebulizer" | 11 |
| Weights | "diffusing capacity" OR "DLCO" OR "gas transfer" OR "St George" | 16 |
| Weights | "pulmonary fibrosis" OR "interstitial lung disease" | 98 |
| Weights | "long-term oxygen" OR "supplemental oxygen" OR "home oxygen" OR "oxygen therapy" | 78 |
| Ratios | "long-term oxygen" OR "home oxygen" OR "rare lung" OR "orphan lung" | 11 |
| Ratios | "antitrypsin" OR "augmentation therapy" | 12 |
| Ratios | "cystic fibrosis" AND (inhaled OR inhalation) | 9 |
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