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Review of statistical methods for analysing healthcare resources and costs
Open accessAbstract
We review statistical methods for analysing healthcare resource use and costs, their ability to address skewness, excess zeros, multimodality and heavy right tails, and their ease for general use. We aim to provide guidance on analysing resource use and costs focusing on randomised trials, although methods often have wider applicability. Twelve broad categories of methods were identified: (I) methods based on the normal distribution, (II) methods following transformation of data, (III) single-distribution generalized linear models (GLMs), (IV) parametric models based on skewed distributions outside the GLM family, (V) models based on mixtures of parametric distributions, (VI) two (or multi)-part and Tobit models, (VII) survival methods, (VIII) non-parametric methods, (IX) methods based on truncation or trimming of data, (X) data components models, (XI) methods based on averaging across models, and (XII) Markov chain methods. Based on this review, our recommendations are that, first, simple methods are preferred in large samples where the near-normality of sample means is assured. Second, in somewhat smaller samples, relatively simple methods, able to deal with one or two of above data characteristics, may be preferable but checking sensitivity to assumptions is necessary. Finally, some more complex methods hold promise, but are relatively untried; their implementation requires substantial expertise and they are not currently recommended for wider applied work.
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“exponential weighting has previously been used in both medical42–44 and non-medical45 46 error/risk analysis.”
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Cites
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Cameron, A., & Trivedi, P. (1998) · Cambridge University Press eBooks · doi:10.1017/cbo9780511814365 · Cited by 6,406
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“tion of the variance function could lead to inefficiencies, the mean function estimates are usually robust (Manning and Mullahy, 2001).”
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Duan, N.-H., Manning, W., Morris, C., & Newhouse, J. (1983) · Journal of Business and Economic Statistics · doi:10.1080/07350015.1983.10509330 · Cited by 1,410 · cited 13×
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BackgroundMethodInfluentialThe logged dependent variable, heteroscedasticity, and the retransformation problem.
Manning, W. (1998) · Journal of Health Economics · doi:10.1016/s0167-6296(98)00025-3 · Cited by 959
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“…reviewed included an informal comparison of different methods and approaches applied to the same data (Austin et al., 2003; Briggs and Gray, 1998; Buntin and Zaslavsky, 2004; Chen and Zhou, 2006; Cooper et al., 2007; Lipscomb et al., 1998; Manning and Mullahy, 2001; O’Hagan and Stevens, 2003;…”
BackgroundMethodInfluentialMuch ado about two: reconsidering retransformation and the two-part model in health econometrics.
Mullahy, J. (1998) · Journal of Health Economics · doi:10.1016/s0167-6296(98)00030-7 · Cited by 791 · cited 2×
“In this regard, Mullahy (1998) proposes two estimators using exponential conditional mean specifications.”
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“Two common approaches to directly evaluate uncertainty in costs, effects and/or cost-effectiveness in randomised trials are the Central Limit Theorem and the bootstrap approach (Barber and Thompson, 2000; O’Hagan and Stevens, 2003; Thompson and Barber, 2000).”
BackgroundMethodSelf-citation
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Cited by
Sorted by citations
Items 201–300 of 678
Estimating Downstream Budget Impacts in Implementation Research
Wagner, T. H., Dopp, A. R., & Gold, H. T. (2020) · Medical Decision Making · doi:10.1177/0272989x20954387 · Cited by 20
“Costs can also be analyzed using regression models in which the dependent variable is the downstream costs and the treatment group is the key righthand-side covariate.(30) In many studies, the downstream cost data can be summarized over time periods (e.”
MethodTsiplova, K., Ungar, W. J., Flanagan, H. E., den Otter, J., Waddell, C., Murray, P., D’Entremont, B., Léger, N., Garon, N., Bryson, S. E., et al. (2019) · Journal of Autism and Developmental Disorders · doi:10.1007/s10803-019-03993-3 · Cited by 23
Simon, J., Budge, K., Price, J., Goodwin, G. M., & Geddes, J. R. (2017) · European psychiatry · doi:10.1016/j.eurpsy.2017.06.007 · Cited by 19
Ten‐year trends in direct costs of asthma: a population‐based study
Tavakoli, H., Fitzgerald, J., Chen, W., Lynd, L., Kendzerska, T., Aaron, S., Gershon, A., Marra, C., & Sadatsafavi, M. (2017) · Allergy. European Journal of Allergy and Clinical Immunology · doi:10.1111/all.12993 · Cited by 19
“Matching with replacement is recommended when the pool of unexposed/control is smaller than the pool of exposed/case, as it results in the most efficient use of available data (17, 18).”
BackgroundMethodCost of Herpes Zoster in Patients With Selected Immune-Compromised Conditions in the United States
Li, Q., Chen, S.-Y., Burstin, S., Levin, M., & Suaya, J. (2016) · Open Forum Infectious Diseases · doi:10.1093/ofid/ofw067 · Cited by 19
“The statistical significance of the incremental costs was assessed by the unadjusted generalized linear model with a log link and gamma distribution, which allows adjustments for the skewedness in distribution of costs [18].”
MethodWillis, T., Hartley, S., Glidewell, L., Farrin, A., Lawton, R., Mceachan, R., Ingleson, E., Heudtlass, P., Collinson, M., Clamp, S., et al. (2015) · Implementation Science · doi:10.1186/s13012-016-0387-5 · Cited by 19
Hong, S. N., Lai, F. T. T., Wang, B., Choi, E. P. H., Wong, I. C. K., Lam, C. L. K., & Wan, E. Y. F. (2024) · Journal of Epidemiology and Global Health · doi:10.1007/s44197-024-00256-y · Cited by 25
“To account for the highly positively skewed distribu - tion of cost data, gamma generalised linear model with log link function adjusting for age and sex was used to exam - ine the association between the number of morbidities and annual public direct medical costs [24, 25 ].”
MethodBetter performance for right-skewed data using an alternative gamma model
Veazie, P., Intrator, O., Kinosian, B. P., & Phibbs, C. (2023) · BMC Medical Research Methodology · doi:10.1186/s12874-023-02113-1 · Cited by 18
“Nonetheless, parametric methods remain a common and useful approach [13–15].”
MethodChen, L.-Y., Hu, Z.-Y., Hu, X.-T., & Xu, G.-N. (2023) · Journal of Cleaner Production · doi:10.1016/j.jclepro.2023.136797 · Cited by 18
DEEP Study: Modeling Outcomes and Costs of Persistent Orofacial Pain
Durham, J., Breckons, M., Vale, L. D., & Shen, J. (2021) · JDR Clinical & Translational Research · doi:10.1177/23800844211063870 · Cited by 15
The out-of-pocket burden of chronic diseases: the cases of Belgian, Czech and German older adults
Krůtilová, V. K., Bahnsen, L., & Graeve, D. D. (2021) · BMC Health Services Research · doi:10.1186/s12913-021-06259-w · Cited by 18
“In order to deal with these issues, the literature provides a variety of different approaches [37, 38].”
BackgroundMallucci, C., Jenkinson, M., Conroy, E., Hartley, J., Brown, M., Moitt, T., Dalton, J., Kearns, T., Griffiths, M., Culeddu, G., et al. (2020) · Health Technology Assessment · doi:10.3310/hta24170 · Cited by 18
Maisog, J., Li, W., Xu, Y., Hurley, B., Shah, H. N., Lemberg, R., Borden, T., Bandeian, S., Schline, M., Cross, R., et al. (2019) · arXiv.org · doi:10.48550/arxiv.1912.13032 · Cited by 18
“Historically, the problem of high-cost claimants was studied by actuarial scientists at the population level, with emphasis on parameter estimation and statistical significance tests [15–19].”
BackgroundPerformance of Safety-Net Hospitals in Year 1 of the Comprehensive Care for Joint Replacement Model
Thirukumaran, C. P., Glance, L. G., Cai, X.-Y., Balkissoon, R., Mesfin, A., & Li, Y. (2019) · Health Affairs · doi:10.1377/hlthaff.2018.05264 · Cited by 18
Estimating Informal Caregiving Time from Patient EQ-5D Data: The Informal CARE Effect (iCARE) Tool
Gheorghe, M., Hoefman, R., Versteegh, M., & van Exel, J. (2018) · PharmacoEconomics (Auckland) · doi:10.1007/s40273-018-0706-6 · Cited by 18
Racial and Ethnic Differences in Out-of-Pocket Expenses among Adults with Diabetes
Simmons, M., Bishu, K. G., Williams, J. S., Walker, R. J., Dawson, A. Z., & Egede, L. E. (2018) · Journal of the National Medical Association · doi:10.1016/j.jnma.2018.04.004 · Cited by 18
Rhon, D., Snodgrass, S., Cleland, J., Greenlee, T. A., Sissel, C. D., & Cook, C. (2018) · Physical Therapy · doi:10.1093/ptj/pzy019 · Cited by 18
Oregon Medicaid Expenditures After the 2014 Affordable Care Act Medicaid Expansion
Springer, R., Marino, M., O'Malley, J., Lindner, S., Huguet, N., & DeVoe, J. (2018) · Medical Care · doi:10.1097/mlr.0000000000000907 · Cited by 18
Health shocks and child time allocation decisions by households: evidence from Ethiopia
Dinku, Y., Fielding, D., & Genç, M. (2018) · doi:10.1186/s40172-018-0064-9 · Cited by 18
“6 Poisson models can be appropriate for continuous variables that include zero observations, as long as the conditional mean equation is correctly specified (Manning & Mullahy, 2001; Mihaylova et al., 2011; Wooldridge, 2010).”
BackgroundEconomic burden of relapsed or refractory multiple myeloma: Results from an international trial
Robinson, D., Orlowski, R., Stokes, M., He, J.-M., Huse, S., Chitnis, A., Kranenburg, B., & Lam, A. (2017) · European Journal of Haematology · doi:10.1111/ejh.12876 · Cited by 18
Illou, M. M. A., Haddad, S., Agier, I., & Ridde, V. (2015) · BMC Health Services Research · doi:10.1186/s12913-015-0957-2 · Cited by 18
“The literature suggests a variety of methods for taking into account these distinctive distributions [36–38].”
BackgroundCost of Care for Patients With Cirrhosis
Kanwal, F., Nelson, R., Liu, Y., Kramer, J., Hernaez, R., Cholankeril, G., Rana, A., Flores, A., Smith, D. L., Cao, Y., et al. (2023) · American Journal of Gastroenterology · doi:10.14309/ajg.0000000000002472 · Cited by 17
“…of patients did not incur any outpatient (3.5% of cases and 20% of controls) or inpatient costs (67.3% of cases, 95.2% controls), we used 2-part regression to model cost outcomes: (i) probability of incurring any costs vs none and (ii) continuous cost (conditional on nonzero costs) (21,22).”
MethodInfluentialLarkin, J., Walsh, B., Moriarty, F., Clyne, B., Harrington, P., & Smith, S. M. (2022) · BMJ Open · doi:10.1136/bmjopen-2021-060502 · Cited by 17
Ismail, K., Stewart, K., Ridge, K., Britneff, E., Freudenthal, R., Stahl, D. R., McCrone, P., Gayle, C., & Doherty, A. M. (2019) · Diabetic Medicine · doi:10.1111/dme.13918 · Cited by 19
“We used a bootstrapped model with 1,000 replicates, which makes no assumptions about the underlying data distribution, to estimate the 95% confidence intervals of coefficients, as regression analyses of cost data often result in non-normally distributed residuals.(29) We did not transform the data as this would distor…”
MethodA doubly robust approach for cost–effectiveness estimation from observational data
Li, J., Vachani, A., Epstein, A. E., & Mitra, N. (2018) · Statistical Methods in Medical Research · doi:10.1177/0962280217693262 · Cited by 17
Belger, M., Haro, J., Reed, C., Happich, M., Kahle-Wrobleski, K., Argimón, J., Bruno, G., Dodel, R., Jones, R. W., Vellas, B., et al. (2016) · BMC Medical Research Methodology · doi:10.1186/s12874-016-0188-1 · Cited by 17
“[30].”
Jacobs, M. A., Kim, J., Tetley, J. C., Schmidt, S., Brimhall, B., Mika, V., Wang, C.-P., Manuel, L. S., Damien, P., & Shireman, P. (2022) · Journal of the American College of Surgeons · doi:10.1097/xcs.0000000000000468 · Cited by 16
Tran, A. D., Chen, R., Nielsen, S., Zahra, E., Degenhardt, L., Santo, T., Farrell, M., & Larance, B. (2021) · The International journal on drug policy · doi:10.1016/j.drugpo.2021.103472 · Cited by 16
Rodriguez-Acevedo, A., Chan, R., Olsen, C., Pandeya, N., Whiteman, D., & Gordon, L. (2021) · BMC Cancer · doi:10.1186/s12885-021-08756-x · Cited by 16
“To measure differences in out-of-pocket expenses each year, we performed a two-part model analysis [26] to account for the excess zeros from individuals with no out-of-pocket expenses during the first year of cancer treatment (i.”
MethodDeterminants of Medical Direct Costs of Care among Patients of a Memory Center
Dauphinot, V., Garnier‐Crussard, A., Moutet, C., Delphin‐Combe, F., Späth, H.-M., & Krolak‐Salmon, P. (2021) · The Journal of Prevention of Alzheimer s Disease · doi:10.14283/jpad.2021.16 · Cited by 16
“As the distribution of the global cost was skewed, the relationship between each patient’s characteristics and the global cost was studied using generalized linear model (GLM) with log link and gamma distribution (38, 39).”
Utilisation of Personal Care Services in Scotland: The Influence of Unpaid Carers
Lemmon, E. (2020) · Journal of Long-Term Care · doi:10.31389/jltc.23 · Cited by 33
“…models have widely been used and discussed in the health economics literature (Buntin and Zaslavsky, 2004; Duan et al., 1984; Mihaylova et al., 2011; Mullahy, 1998) and have often been shown to outperform other models when a large proportion of zeroes exist in the data (Mihaylova et al., 2011).”
BackgroundMethodInfluentialHow do low-income enrollees in the Affordable Care Act marketplaces respond to cost-sharing?
Lavetti, K., DeLeire, T., & Ziebarth, N. R. (2019) · Journal of Risk and Insurance · doi:10.3386/w26430 · Cited by 16
Elder, C., DeBar, L., Ritenbaugh, C., Dickerson, J. F., Vollmer, W., Deyo, R., Johnson, E. S., & Haas, M. (2018) · Journal of general internal medicine · doi:10.1007/s11606-018-4539-y · Cited by 16
Forma, L., Jylhä, M., Pulkki, J., Aaltonen, M. S., Raitanen, J., & Rissanen, P. (2017) · BMC Health Services Research · doi:10.1186/s12913-017-2615-3 · Cited by 16
“Survival methods such as Cox proportion hazard models may be employed when the data are skewed or multimodal, have heavy tails, or consist of excess zeroes [23].”
Method[Checklist for the Development and Assessment of Cost-of-Illness Studies].
Müller, D., Stock, S., Dintsios, C., Chernyak, N., Gerber-Grote, A., Gloede, T. D., Hermann, B., Huppertz, E., Jülich, F., Mostardt, S., et al. (2017) · Das Gesundheitswesen · doi:10.1055/s-0042-124664 · Cited by 16
The impact of comorbidities on productivity loss in asthma patients
Ehteshami-Afshar, S., Fitzgerald, J., Carlsten, C., Tavakoli, H., Rousseau, R., Tan, W., Rolf, J., & Sadatsafavi, M. (2016) · Respiratory Research · doi:10.1186/s12931-016-0421-9 · Cited by 16
“As the productivity loss data were zero-inflated, we used two-part models for statistical inference [24].”
MethodAnalysis of contemporary HIV/AIDS health care costs in Germany
Treskova, M., Kuhlmann, A., Bogner, J., Hower, M., Heiken, H., Stellbrink, H., Mahlich, J., von der Schulenburg, J. G., & Stoll, M. (2016) · Medicine · doi:10.1097/md.0000000000003961 · Cited by 16
“[16] In this analysis, we examine composition of the annual costs, determine major cost drivers, [17] and estimate relative cost ratios [18] between patients with varying characteristics.”
MethodChay, J., Tang, R. W. C., Tan, T.-E., Chan, C. M., Mathur, R., Lee, B. J. H., Chan, H. H., Sim, S. S. K. P., Farooqui, S., Teo, K. Y. C., et al. (2023) · Eye · doi:10.1038/s41433-023-02624-7 · Cited by 15
“A two-part regression model was used to estimate the expected median income, controlling for a quadratic function of age and gender [24].”
MethodDirect Health Care Costs Associated With Multiple Sclerosis
Khakban, A., Llorian, E. R., Michaux, K. D., Patten, S. B., Traboulsee, A. L., Oh, J., Lynd, L. D., & on behalf of CanProCo Study Group (2022) · Neurology · doi:10.1212/wnl.0000000000201645 · Cited by 17
“Given the large sample size of our analysis, the Central Limit Theorem will guarantee near normality of sample means.(32) The regression coefficient for cohort membership (MS vs control) represents the adjusted excess cost of MS relative to the non-MS population.”
BackgroundAdvance directives completion and hospital out‐of‐pocket expenditures
Zhu, Y., & Enguidanos, S. M. (2022) · Journal of Hospital Medicine · doi:10.1002/jhm.12839 · Cited by 15
Brusco, N. K., Ekegren, C. L., Taylor, N. F., Hill, K., Lee, A. L., Somerville, L., Lannin, N. A., Wade, D. T., Abdelmotaleb, R., Callaway, L., et al. (2021) · BMC Health Services Research · doi:10.1186/s12913-021-06462-9 · Cited by 14
“The mean cost difference will be determined between the two groups using a linear regression model [48].”
MethodShah, K., Elder, D., Nguyen, M., Turner, L., Doyle, M., Woldendorp, K., Seco, M., Law, C., Wilson, M., Keech, A., et al. (2021) · Heart, Lung and Circulation · doi:10.1016/j.hlc.2021.05.088 · Cited by 15
Klein, S., Jiang, S., Morey, J., Pai, A., Mancini, D., Lala, A., & Ferket, B. (2021) · Circulation: Heart Failure · doi:10.1161/circheartfailure.120.007763 · Cited by 15
Okunrintemi, V., Valero-Elizondo, J., Michos, E., Salami, J. A., Ogunmoroti, O., Osondu, C. U., Tibuakuu, M., Benson, E.-M. A., Pawlik, T., Blaha, M., et al. (2019) · Journal of general internal medicine · doi:10.1007/s11606-019-05325-8 · Cited by 15
“, most expenditures are seen in only a small proportion of the population), two-part models were utilized to study expenditures.(22) Two-part models are often used to model healthcare expenditures and are the product of (1) the probability that any given individual had any expenditures and (2) their mean expenditures.”
Economic evaluation of a behavior-modifying intervention to enhance antiepileptic drug adherence.
Plumpton, C. O., Brown, I., Reuber, M., Marson, A., & Hughes, D. (2015) · Epilepsy & Behavior · doi:10.1016/j.yebeh.2015.01.035 · Cited by 15
“Cost and utility decrement models were generated as generalized linear models, accounting for relevant covariates and the positively skewed nature of the data [32].”
MethodFrey, S., Linder, R., Juckel, G., & Stargardt, T. (2013) · The European Journal of Health Economics · doi:10.1007/s10198-013-0460-9 · Cited by 16
“The probability to be prescribed any comedication that is commonly used to treat disease and medication-related side effects was estimated using logistic regression [59, 60].”
Burke, J. F., Kovacs, B., Borton, L., & Sander, S. D. (2012) · Postgraduate Medicine · doi:10.3810/pgm.2012.03.2539 · Cited by 16
Estimating and examining the costs of inpatient diabetes care in an Irish Public Hospital
Friel, K. M., Gillespie, P., Coates, V., McCauley, C., McCann, M., O'Kane, M., McGuigan, K., Khamis, A., & Manktelow, M. (2021) · Diabetic Medicine · doi:10.1111/dme.14753 · Cited by 14
Does the amount of formal care affect informal care? Evidence among over-60s in France
Perdrix, E., & Roquebert, Q. (2021) · European Journal of Health Economics · doi:10.1007/s10198-021-01370-5 · Cited by 14
“See Appendix A for details and [25] for further details on the resolution of this maximization program.”
BackgroundInfluentialMartin, B., Brodke, D., Wilson, F. A., Chaiyakunapruk, N., & Nelson, R. (2021) · Medical Care · doi:10.1097/mlr.0000000000001496 · Cited by 14
Prediction of out-of-pocket health expenditures in Rwanda using machine learning techniques
Muremyi, R., Haughton, D., Kabano, I., & Niragire, F. (2020) · The Pan African Medical Journal · doi:10.11604/pamj.2020.37.357.27287 · Cited by 14
“Furthermore, one of the high priorities in sustainable development goals (SDGs) was to provide universal health care services specifically for the populations of low- and middle-income countries [4,5].”
BackgroundEby, E. L., Bajpai, S., Faries, D. E., Haynes, V. S., & Lage, M. J. (2020) · Journal of Managed Care & Specialty Pharmacy · doi:10.18553/jmcp.2020.26.9.1081 · Cited by 16
Rhon, D., O'Hagan, E. T., Mysliwiec, V., & Lentz, T. A. (2019) · Spine · doi:10.1097/brs.0000000000003114 · Cited by 14
“Health care visits and cost data are often positively skewed and overdispersed leading to underestimation of standard errors and overstatement of regression parameter significance using ordinary least squares (OLS) or Poisson regression.(32) For health care visit data, we used a generalized linear model (GENMOD proced…”
MethodMorton, K., Sanderson, E., Dixon, P., King, A. J. L., Jenkins, S., MacNeill, S., Shaw, A. R. G., Metcalfe, C. D., Chalder, M., Hollingworth, W., et al. (2019) · Health Services and Delivery Research · doi:10.3310/hsdr07210 · Cited by 10
Rhon, D. I., Greenlee, T. A., & Fritz, J. M. (2018) · Pain Medicine · doi:10.1093/pm/pny212 · Cited by 20
Burnhope, E., Rodríguez-Guadarrama, Y., Waring, M., Guilder, A., Malhotra, B., Razavi, R., Rinaldi, C. A., Pennington, M., & Carr-White, G. (2019) · Journal of Medical Economics · doi:10.1080/13696998.2019.1581621 · Cited by 14
“We applied Generalized Linear Modelling following recommendations by Mihaylova et al.29 and used the Park test to select the distribution.”
MethodScuffham, P., Byrnes, J., Pollicino, C. A., Cross, D. B., Goldstein, S., & Ng, S. (2018) · Journal of General Internal Medicine · doi:10.1007/s11606-018-4682-5 · Cited by 16
Smoking Cessation Is Associated With Lower Indirect Costs
Baker, C. L., Bruno, M., Emir, B., Li, V. W., & Goren, A. (2018) · Journal of Occupational and Environmental Medicine · doi:10.1097/jom.0000000000001302 · Cited by 14
“have no loss in work productivity and absenteeism, single-distribution generalized linear models often cannot adequately model this type of data.(42,43) Two-part models have been commonly used to better model indirect costs and to more accurately predict mean costs.”
BackgroundPilot evaluation of the management of chronic oedema in community settings project.
Humphreys, I., Thomas, M. J., & Morgan, K. (2017) · British Journal of Community Nursing · doi:10.12968/bjcn.2017.22.12.578 · Cited by 14
Ensemble Risk Model of Emergency Admissions (ERMER)
Mesgarpour, M., Chaussalet, T., & Chahed, S. (2017) · International Journal of Medical Informatics · doi:10.1016/j.ijmedinf.2017.04.010 · Cited by 16
“There are three main layers of difficulties in the preparation of features: correlations, recategorisations and selections (Mihaylova et al., 2011; Walpole et al., 2014; Yang et al., 2005).”
Maruotti, A., Raponi, V., & Lagona, F. (2016) · Biometrical journal. Biometrische Zeitschrift · doi:10.1002/bimj.201400121 · Cited by 14
Kranker, K. (2016) · Journal of Health Economics · doi:10.1016/j.jhealeco.2016.01.008 · Cited by 14
Comparative Analysis of Treatment Costs in EUROHOPE.
Iversen, T., Aas, E., Rosenqvist, G., & Häkkinen, U. (2015) · Health Economics · doi:10.1002/hec.3262 · Cited by 14
Lage, M. J., Carroll, C. A., & Fairman, K. A. (2013) · Journal of Medical Economics · doi:10.3111/13696998.2013.823868 · Cited by 15
In-hospital falls and fall-related injuries: a protocol for a cost of fall study
Morello, R., Barker, A., Haines, T., Zavarsek, S., Watts, J., Hill, K., Brand, C., Sherrington, C., Wolfe, R., Bohensky, M., et al. (2013) · Injury Prevention · doi:10.1136/injuryprev-2012-040706 · Cited by 14
The cost of outpatient pneumonia in children <5 years of age in Fiji
Temple, B., Griffiths, U., Mulholland, E., Ratu, F. T., Tikoduadua, L., & Russell, F. (2012) · Tropical medicine & international health · doi:10.1111/j.1365-3156.2011.02897.x · Cited by 14
“Patient-specific costs were summarised as arithmetic mean costs per site, the statistic most relevant to policy makers because of its relationship with total costs (Mihaylova et al. 2011).”
BackgroundGalante, J., Montero-Marín, J., Vainre, M., Dufour, G., García-Campayo, J., & Jones, P. B. (2024) · PLoS ONE · doi:10.1371/journal.pone.0305928 · Cited by 13
“The two-part model, widely used for mixed discrete-continuous outcomes with a mass of zero values [51], comprised two steps.”
Lin, K., Singer, D., Ko, D., Glynn, R. J., Najafzadeh, M., Lee, S. B., Bessette, L. G., Cervone, A., DiCesare, E. C., & Kim, D. H. (2023) · JAMA Network Open · doi:10.1001/jamanetworkopen.2023.42264 · Cited by 13
Healthcare expenditure trends among adult stroke patients in the United States, 2011-2020.
Ramamoorthy, V., Chan, K., Roy, M., Saxena, A., Ahmed, M., Zhang, Z.-W., Appunni, S., Thomas, R., McGranaghan, P., McDermott, M., et al. (2023) · Journal of Stroke & Cerebrovascular Diseases · doi:10.1016/j.jstrokecerebrovasdis.2023.107333 · Cited by 13
d’Elbée, M., Terris-Prestholt, F., Briggs, A., Griffiths, U., Larmarange, J., Medley, G., & Gomez, G. B. (2023) · Health Economics · doi:10.1002/hec.4722 · Cited by 13
Self-citationUtilisation and costs of mental health-related service use among adolescents
Ziebold, C., Ribeiro, W. S., King, D., McDaid, D., Hoffmann, M. S., Romeo, R., Pan, P. M., Miguel, E. C., Bressan, R. A., Rohde, L. A., et al. (2022) · PLoS ONE · doi:10.1371/journal.pone.0273628 · Cited by 15
“Annual costs for each participant were included in the models as a scalar dependent variable, with a Gamma distribution [44], using the log-link function.”
Chang, H.-Y., Ferris, L. M., Eisenberg, M. D., Krawczyk, N., Schneider, K., Lemke, K. W., Richards, T., Jackson, K., Murthy, V., Weiner, J., et al. (2020) · Medical Care · doi:10.1097/mlr.0000000000001389 · Cited by 13
Muratov, S., Lee, J., Holbrook, A., Guertin, J., Mbuagbaw, L., Paterson, J., Gomes, T., Pequeno, P., & Tarride, J. (2019) · BMJ Open · doi:10.1136/bmjopen-2018-028637 · Cited by 13
Evensen, S., Wisløff, T., Lystad, J. U., Bull, H., Martinsen, E., Ueland, T., & Falkum, E. (2019) · BMC Psychiatry · doi:10.1186/s12888-019-2130-7 · Cited by 13
Singh, J., Valero-Elizondo, J., Salami, J. A., Warraich, H. J., Ogunmoroti, O., Spatz, E., Desai, N., Rana, J., Virani, S., Blankstein, R., et al. (2018) · Journal of the American Heart Association : Cardiovascular and Cerebrovascular Disease · doi:10.1161/jaha.117.007874 · Cited by 13
“most expenditures are seen in only a small proportion of the population), 2-part models were utilized to study expenditures.(18) Two-part models are often used to model healthcare expenditures, and are the product of (1) the probability that any given individual had any expenditures; and (2) their mean expenditures.”
MethodMcCormick, N., Marra, C. A., Sadatsafavi, M., & Aviña-Zubieta, J. (2018) · Lupus · doi:10.1177/0961203318768882 · Cited by 13
“10 year, two-part models(34) were used for cost calculation.”
MethodMitchell, D., Guertin, J., Dubois, A., Dubé, M.-P., Tardif, J., Iliza, A., Fanton-Aita, F., Matteau, A., & LeLorier, J. (2018) · Molecular Diagnosis & Therapy · doi:10.1007/s40291-018-0323-2 · Cited by 13
Chinkhumba, J., de Allegri, M., Mazalale, J., Brenner, S., Mathanga, D., Muula, A., & Robberstad, B. (2017) · PLoS ONE · doi:10.1371/journal.pone.0182326 · Cited by 13
Li, F., Chen, S., Wei, C., & Jia, J. (2017) · BMC Neurology · doi:10.1186/s12883-017-0802-9 · Cited by 11
“For base case analyses, GLMs will be developed using the gamma distribution with a log-link function [35].”
MethodLong-Term Health Spending Persistence among the Privately Insured in the US
Hirth, R., Calonico, S., Gibson, T., Levy, H., Smith, J., & Das, A. (2016) · doi:10.1111/j.1475-5890.2016.12120 · Cited by 13
“Mihaylova et al. (2011) review several statistical methods for analysing health care resource use and costs and assess their ability to address skewness, excess zeros, multimodality and heavy right tails.”
MethodLorgelly, P., Doble, B., Knott, R., & The Cancer 2015 Investigators (2016) · PharmacoEconomics (Auckland) · doi:10.1007/s40273-015-0343-2 · Cited by 13
“expenditure data [21]; however, of more relevance is the high proportion of zeros.”
BackgroundHealth Care Needs and Costs for Children Exposed to Prenatal Substance Use to Adulthood
Lee, E., Schofield, D., Dronavalli, M., Lawler, K., Uebel, H., Burns, L., Bajuk, B., Page, A., Gu, Y.-Y., Eastwood, J., et al. (2024) · JAMA pediatrics · doi:10.1001/jamapediatrics.2024.2281 · Cited by 12
The economic impact of comorbidity in multiple sclerosis
Ponzio, M., Monti, M., Mallucci, G., Borrelli, P., Fusco, S., Tacchino, A., Brichetto, G., Tronconi, L., Montomoli, C., & Bergamaschi, R. (2022) · Neurological Sciences · doi:10.1007/s10072-022-06517-7 · Cited by 12
“These latter models are appropriate for analyzing zero-inflated cost data with skewness, [21] which is typical in medical expenditure data [22].”
BackgroundAssociation of Insurance Type With Inpatient Surgery 30-Day Complications and Costs
Simon, R. C., Kim, J.-S., Schmidt, S., Brimhall, B. B., Salazar, C. I., Wang, C., Wang, Z., Sarwar, Z. U., Manuel, L., Damien, P., et al. (2022) · Journal of Surgical Research · doi:10.1016/j.jss.2022.09.006 · Cited by 15
Santini, Z., Nielsen, L., Hinrichsen, C., Nelausen, M., Meilstrup, C., Koyanagi, A., McDaid, D., Lyubomirsky, S., VanderWeele, T., & Koushede, V. (2021) · Mental health & prevention · doi:10.1016/j.mhp.2021.200222 · Cited by 12
Applying random forest in a health administrative data context: a conceptual guide
King, C., & Strumpf, E. (2021) · Health Services and Outcomes Research Methodology · doi:10.1007/s10742-021-00255-7 · Cited by 11
Wu, J., Wu, Y. Y., Wang, Z., Wu, Y., Wu, T., Wang, M., Wang, X., Wang, S., Wang, J., Yu, H., et al. (2021) · International Journal of Environmental Research and Public Health · doi:10.3390/ijerph18073693 · Cited by 12
Choi, I., Lee, D., Son, K., & Bae, S. (2020) · BMC Public Health · doi:10.1186/s12889-020-09161-y · Cited by 12
McCormick, N., Marra, C. A., Sadatsafavi, M., & Aviña-Zubieta, J. (2020) · Seminars in Arthritis & Rheumatism · doi:10.1016/j.semarthrit.2019.06.010 · Cited by 12
“The second part (GLM with log link and negative binomial distribution) assessed costs only among those with non-zero costs.”
InfluentialVariation in Bariatric Surgery Costs and Complication Rates in the Military Health System.
Luan, W. P., Leroux, T., Olsen, C., Robb, D., Skinner, J. S., & Richard, P. (2019) · Military Medicine · doi:10.1093/milmed/usz454 · Cited by 12
Kähm, K., Laxy, M., Schneider, U., & Holle, R. (2018) · PharmacoEconomics (Auckland) · doi:10.1007/s40273-018-0699-1 · Cited by 12
Kaul, S., Avila, J., Jupiter, D., Rodriguez, A. M., Kirchhoff, A. C., & Kuo, Y. (2017) · Journal of Cancer Research and Clinical Oncology · doi:10.1007/s00432-017-2494-3 · Cited by 12
Laxy, M., Wilson, E., Boothby, C. E., & Griffin, S. (2017) · Value in Health · doi:10.1016/j.jval.2017.05.018 · Cited by 12
“[30] Mihaylova B, Briggs A, O’Hagan A, Thompson SG.”
MethodBrennan, A., Jackson, A., Horgan, M. M., Bergin, C. J., & Browne, J. P. (2015) · BMC Health Services Research · doi:10.1186/s12913-015-0816-1 · Cited by 13
“Generalized linear models were used to model costs ppm on HAART as they are suitable for modelling cost data, which tend to be skewed, without the need for transformation [25].”
Chen, S.-Y., Lee, Y.-C., Alas, V., Greene, M., & Brixner, D. (2014) · Endocrine Practice · doi:10.4158/ep13269.or · Cited by 12
The Association Between Health Assessment-Derived Summary Health Scores and Health Care Costs
Pronk, N. P., Lowry, M., Maciosek, M. V., & Gallagher, J. M. (2011) · Journal of Occupational and Environmental Medicine · doi:10.1097/jom.0b013e318223d464 · Cited by 12
Wei, Y.-J., Palumbo, F., Simoni-Wastila, L., Shulman, L., Stuart, B., Beardsley, R., & Brown, C. H. (2015) · Parkinsonism & Related Disorders · doi:10.1016/j.parkreldis.2014.10.021 · Cited by 12
van Veen, S. V., van Kleef, R. V., van de Ven, W., & van Vliet, R. (2014) · European Journal of Health Economics · doi:10.1007/s10198-014-0567-7 · Cited by 12
“Several studies have shown that when sample sizes are large (enough), OLS may provide the same model fit as more complicated models, such as 2PMs or GLMs [11, 18, 29, 34, 54].”
MethodFeldman, D. I., Valero-Elizondo, J., Salami, J. A., Rana, J. S., Ogunmoroti, O., Osondu, C. U., Spatz, E. S., Virani, S. S., Blankstein, R., Blaha, M. J., et al. (2017) · Atherosclerosis · doi:10.1016/j.atherosclerosis.2017.02.004 · Cited by 11
Pereira, C. F. R., Dijxhoorn, A.-F. Q., Koekoek, B., van den Broek, M. V. D., Vandersteen, K., Engel, M., van Rijn, M., Meijers, J., Hasselaar, J., van der Heide, A., et al. (2024) · International Journal of Integrated Care · doi:10.5334/ijic.7504 · Cited by 11
Metadata
- Authors
- Mihaylova, B., Briggs, A., O'Hagan, A., & Thompson, S. G.
- Affiliations
- University of Oxford (GB)
- University of Glasgow (GB)
- Department of Probability and Statistics, University of Sheffield, Sheffield, UK
- MRC Biostatistics Unit (GB)
- Corresponding author
- Mihaylova, Borislava
- Venue
- Health Economics
- Publisher
- Wiley
- Published
- 2010-08-27
- Volume
- 20
- Issue
- 8
- Pages
- 897-916
- ISSN
- 1057-9230 (ISSN-L)
- 1099-1050
- PMC
- PMC3470917
- OpenAlex
- W2041616439
- Cited by
- 767
- References
- 99
- Citations by year
- 2026: 35 · 2025: 47 · 2024: 51 · 2023: 48 · 2022: 79 · 2021: 85 · 2020: 77 · 2019: 67 · 2018: 58 · 2017: 47 · 2016: 41 · 2015: 39 · 2014: 30 · 2013: 26 · 2012: 23
- FWCI
- 5.09
- Type
- journal article
- Language
- en
- Open access
- bronze
- doi.org
- Licence
- Locations
- Health Economics (publishedVersion)
- Europe PMC (PubMed Central) (publishedVersion, cc-by)
- PubMed Central (publishedVersion, cc-by)
- PubMed (publishedVersion)
- ENLIGHTEN (Jurnal Bimbingan dan Konseling Islam) (submittedVersion)
- Funders
- British Heart Foundation (RG/08/014/24067)
- Topics
- Social Sciences › Economics, Econometrics and Finance › Economics and Econometrics › Health Systems, Economic Evaluations, Quality of Life
- Physical Sciences › Mathematics › Statistics and Probability › Statistical Methods and Bayesian Inference
- Physical Sciences › Mathematics › Statistics and Probability › Statistical Methods in Clinical Trials
- Keywords
- health care utilization, health care costs, skewed data, excess zeros, multimodality, generalized linear models, mixture distributions, two-part model, Tobit model, survival analysis, non-parametric methods, data truncation, model averaging, Markov chain analysis, randomised trials, statistical method selection
- MeSH terms
- Data Interpretation, Statistical, Health Resources, Markov Chains, Linear Models, Randomized Controlled Trials as Topic, Health Care Costs
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