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  1. Shabaruddin FH, Fleeman ND, Payne K
    Pharmgenomics Pers Med, 2015;8:115-26.
    PMID: 26309416 DOI: 10.2147/PGPM.S35063
    Personalized medicine, with the aim of safely, effectively, and cost-effectively targeting treatment to a prespecified patient population, has always been a long-time goal within health care. It is often argued that personalizing treatment will inevitably improve clinical outcomes for patients and help achieve more effective use of health care resources. Demand is increasing for demonstrable evidence of clinical and cost-effectiveness to support the use of personalized medicine in health care. This paper begins with an overview of the existing challenges in conducting economic evaluations of genetics- and genomics-targeted technologies, as an example of personalized medicine. Our paper illustrates the complexity of the challenges faced by these technologies by highlighting the variations in the issues faced by diagnostic tests for somatic variations, generally referring to genetic variation in a tumor, and germline variations, generally referring to inherited genetic variation in enzymes involved in drug metabolic pathways. These tests are typically aimed at stratifying patient populations into subgroups on the basis of clinical effectiveness (response) or safety (avoidance of adverse events). The paper summarizes the data requirements for economic evaluations of genetics and genomics-based technologies while outlining that the main challenges relating to data requirements revolve around the availability and quality of existing data. We conclude by discussing current developments aimed to address the challenges of assessing the cost-effectiveness of genetics and genomics-based technologies, which revolve around two central issues that are interlinked: the need to adapt available evaluation methods and identifying who is responsible for generating evidence for these technologies.
  2. Liew CH, Shabaruddin FH, Dahlui M
    Healthcare (Basel), 2022 Oct 20;10(10).
    PMID: 36292545 DOI: 10.3390/healthcare10102099
    This study aimed to estimate the economic burden on gynaecological cancer patients and their households, in terms of out-of-pocket expenditure, catastrophic health expenditure (CHE) and poverty impact. A cross-sectional study was conducted at an academic tertiary-care health centre in an upper-middle-income country. Data were obtained via structured interviews of 120 gynaecological cancer patients alongside review of medical charts. Mean (SD) and median (IQR) annual household out-of-pocket expenditures were USD 2780 (SD = USD 3926) and USD 1396 (IQR = 3013), respectively. Two thirds (n = 77/120, 64%) of households experienced CHE and 17% (n = 20/120) were impoverished due to out-of-pocket expenditure related to gynaecological cancer. Factors associated with CHE, explored using multivariate logistic regression analysis, estimated that the highest income quintile households, Q5, were 90% less likely to incur CHE compared to the lowest income quintile households, Q1 (adjusted odds ratio = 0.100; p-value < 0.05) and that patients who were not receiving chemotherapy were 88% less likely to incur CHE compared to those receiving chemotherapy (adjusted odds ratio = 0.120; p-value < 0.05). These results indicate the necessity to broaden the coverage of existing financial assistance for patients from low- and middle-income households, such as extending coverage to adult patients of all ages and to those treated in all public hospitals, including academic health centres.
  3. Shabaruddin FH, Chen LC, Elliott RA, Payne K
    Pharmacoeconomics, 2013 Apr;31(4):277-88.
    PMID: 23529208 DOI: 10.1007/s40273-013-0033-x
    BACKGROUND: Chemotherapy offers cancer patients the potential benefits of improved mortality and morbidity but may cause detrimental outcomes due to adverse drug events (ADEs), some of which requiring time-consuming, resource-intensive and costly clinical management. To appropriately assess chemotherapy agents in an economic evaluation, ADE-related parameters such as the incidence, (dis)utility and cost of ADEs should be reflected within the model parameters. To date, there has been no systematic summary of the existing literature that quantifies the utilities of ADEs due to healthcare interventions in general and chemotherapy treatments in particular.

    OBJECTIVE: This review aimed to summarize the current evidence base of reported utility values for chemotherapy-related ADEs.

    METHODS: A structured electronic search combining terms for utility, utility valuation methods and generic terms for cancer treatment was conducted in MEDLINE and EMBASE in June 2011. Inclusion criteria were: (1) elicitation of utility values for chemotherapy-related ADEs and (2) primary data. Two reviewers identified studies and extracted data independently. Any disagreements were resolved by a third reviewer.

    RESULTS: Eighteen studies met the inclusion criteria from the 853 abstracts initially identified, collectively reporting 218 utility values for chemotherapy-related ADEs. All 18 studies used short descriptions (vignettes) to obtain the utility values, with nine studies presenting the vignettes used in the valuation exercises. Of the 218 utility values, 178 were elicited using standard gamble (SG) or time trade-off (TTO) approaches, while 40 were elicited using visual analogue scales (VAS). There were 169 utility values of specific chemotherapy-related ADEs (with the top ten being anaemia [34 values], nausea and/or vomiting [32 values], neuropathy [21 values], neutropenia [12 values], diarrhoea [12 values], stomatitis [10 values], fatigue [8 values], alopecia [7 values], hand-foot syndrome [5 values] and skin reaction [5 values]) and 49 of non-specific chemotherapy-related adverse events. In most cases, it was difficult to directly compare the utility values as various definitions and study-specific vignettes were used for the ADEs of interest.

    LIMITATIONS: This review was designed to provide an overall description of existing literature reporting utility values for chemotherapy-related ADEs. The findings were not exhaustive and were limited to publications that could be identified using the search strategy employed and those reported in the English language.

    CONCLUSIONS: This review identified wide ranges in the utility values reported for broad categories of specific chemotherapy-related ADEs. There were difficulties in comparing the values directly as various study-specific definitions were used for these ADEs and most studies did not make the vignettes used in the valuation exercises available. It is recommended that a basic minimum requirement be developed for the transparent reporting of study designs eliciting utility values, incorporating key criteria such as reporting how the vignettes were developed and presenting the vignettes used in the valuation tasks as well as valuing and reporting the utility values of the ADE-free base states. It is also recommended, in the future, for studies valuing the utilities of chemotherapy-related ADEs to define the ADEs according to the National Cancer Institute (NCI) definitions for chemotherapy-related ADEs as the use of the same definition across studies would ease the comparison and selection of utility values and make the overall inclusion of adverse events within economic models of chemotherapy agents much more straightforward.

  4. Mohamed R, Shabaruddin FH, Azzeri A, McDonald SA, Dahlui M
    Journal of virus eradication, 2019 Nov 04;5(4):253.
    PMID: 31754450
  5. McDonald SA, Dahlui M, Mohamed R, Naning H, Shabaruddin FH, Kamarulzaman A
    PLoS One, 2015;10(6):e0128091.
    PMID: 26042425 DOI: 10.1371/journal.pone.0128091
    BACKGROUND: The prevalence of hepatitis C virus (HCV) infection in Malaysia has been estimated at 2.5% of the adult population. Our objective, satisfying one of the directives of the WHO Framework for Global Action on Viral Hepatitis, was to forecast the HCV disease burden in Malaysia using modelling methods.

    METHODS: An age-structured multi-state Markov model was developed to simulate the natural history of HCV infection. We tested three historical incidence scenarios that would give rise to the estimated prevalence in 2009, and calculated the incidence of cirrhosis, end-stage liver disease, and death, and disability-adjusted life-years (DALYs) under each scenario, to the year 2039. In the baseline scenario, current antiviral treatment levels were extended from 2014 to the end of the simulation period. To estimate the disease burden averted under current sustained virological response rates and treatment levels, the baseline scenario was compared to a counterfactual scenario in which no past or future treatment is assumed.

    RESULTS: In the baseline scenario, the projected disease burden for the year 2039 is 94,900 DALYs/year (95% credible interval (CrI): 77,100 to 124,500), with 2,002 (95% CrI: 1340 to 3040) and 540 (95% CrI: 251 to 1,030) individuals predicted to develop decompensated cirrhosis and hepatocellular carcinoma, respectively, in that year. Although current treatment practice is estimated to avert a cumulative total of 2,200 deaths from DC or HCC, a cumulative total of 63,900 HCV-related deaths is projected by 2039.

    CONCLUSIONS: The HCV-related disease burden is already high and is forecast to rise steeply over the coming decades under current levels of antiviral treatment. Increased governmental resources to improve HCV screening and treatment rates and to reduce transmission are essential to address the high projected HCV disease burden in Malaysia.

  6. Kubas MA, Shabaruddin FH, Mazlan-Kepli W, Jagan N, Mohamed S, Mohamed Nazar NI, et al.
    J Pharm Bioallied Sci, 2020 Nov;12(Suppl 2):S781-S786.
    PMID: 33828378 DOI: 10.4103/jpbs.JPBS_381_19
    Introduction: Non-vitamin K antagonist oral anticoagulants (NOACs), such as dabigatran and rivaroxaban, are now available for stroke prevention in patients with atrial fibrillation (AF) and are often clinically preferred over vitamin K antagonists (VKAs), such as warfarin. Data describing adherence and persistence to NOACs in real-life clinical practice in Malaysia are scarce. This study aimed to assess adherence and persistence to NOACs in patients with AF in two tertiary-care referral centers: Hospital Kuala Lumpur (HKL) and Hospital Serdang (HSDG).

    Materials and Methods: This was a retrospective cohort study that included all patients with AF who were treated with NOACs (dabigatran or rivaroxaban) in HKL and HSDG. Data were obtained from medical records and pharmacy databases. Adherence was assessed using proportion of days covered (PDC) over a 1-year duration. High adherence was defined as PDC ≥80%. A gap of >60 days between two consecutive refills was used to define non-persistence.

    Result: There were 281 patients who met the inclusion criteria, with 54.1% (n = 152) male. There were 75.1% (n = 211) patients on dabigatran and others on rivaroxaban. Only 66.9% (n = 188) of patients achieved high adherence with PDC ≥80% and 69.8% (n = 196) were persistence with >60-day gap over 12 months. Adherence and persistence were both influenced by treatment center, whereas polypharmacy only influenced adherence.

    Conclusion: Overall adherence and persistence to NOACs were suboptimal and varied between treatment centers, potentially due to institution-specific administrative and clinical practice differences. Clinical care and outcomes can potentially be optimized by identifying factors affecting adherence and persistence and by implementing interventions to improving them.

  7. McDonald SA, Azzeri A, Shabaruddin FH, Dahlui M, Tan SS, Kamarulzaman A, et al.
    Appl Health Econ Health Policy, 2018 12;16(6):847-857.
    PMID: 30145775 DOI: 10.1007/s40258-018-0425-3
    INTRODUCTION: The World Health Organisation (WHO) has set ambitious goals to reduce the global disease burden associated with, and eventually eliminate, viral hepatitis.

    OBJECTIVE: To assist with achieving these goals and to inform the development of a national strategic plan for Malaysia, we estimated the long-term burden incurred by the care and management of patients with chronic hepatitis C virus (HCV) infection. We compared cumulative healthcare costs and disease burden under different treatment cascade scenarios.

    METHODS: We attached direct costs for the management/care of chronically HCV-infected patients to a previously developed clinical disease progression model. Under assumptions regarding disease stage-specific proportions of model-predicted HCV patients within care, annual numbers of patients initiated on antiviral treatment and distribution of treatments over stage, we projected the healthcare costs and disease burden [in disability-adjusted life-years (DALY)] in 2018-2040 under four treatment scenarios: (A) no treatment/baseline; (B) pre-2018 standard of care (pegylated interferon/ribavirin); (C) gradual scale-up in direct-acting antiviral (DAA) treatment uptake that does not meet the WHO 2030 treatment uptake target; (D) scale-up in DAA treatment uptake that meets the WHO 2030 target.

    RESULTS: Scenario D, while achieving the WHO 2030 target and averting 253,500 DALYs compared with the pre-2018 standard of care B, incurred the highest direct patient costs over the period 2018-2030: US$890 million (95% uncertainty interval 653-1271). When including screening programme costs, the total cost was estimated at US$952 million, which was 12% higher than the estimated total cost of scenario C.

    CONCLUSIONS: The scale-up to meet the WHO 2030 target may be achievable with appropriately high governmental commitment to the expansion of HCV screening to bring sufficient undiagnosed chronically infected patients into the treatment pathway.

  8. Azzeri A, Dahlui M, Mohamed R, McDonald SA, Jaafar H, Shabaruddin FH
    Front Public Health, 2023;11:1114560.
    PMID: 36935675 DOI: 10.3389/fpubh.2023.1114560
    INTRODUCTION: A scaled-up treatment cascade with direct-acting antiviral (DAA) therapy is necessary to achieve global WHO targets for hepatitis C virus (HCV) elimination in Malaysia. Recently, limited access to sofosbuvir/daclatasvir (SOF/DAC) is available through compulsory licensing, with access to sofosbuvir/velpatasvir (SOF/VEL) expected through voluntary licensing due to recent agreements. SOF/VEL has superior clinical outcomes but has higher drug acquisition costs compared to SOF/DAC. A stratified treatment cascade might be the most cost-efficient approach for Malaysia whereby all HCV patients are treated with SOF/DAC except for patients with cirrhosis who are treated with SOF/VEL.

    METHODS: This study aimed to conduct a 5-year budget impact analysis of the proposed stratified treatment cascade for HCV treatment in Malaysia. A disease progression model that was developed based on model-predicted HCV epidemiology data was used for the analysis, where all HCV patients in scenario A were treated with SOF/DAC for all disease stages while in scenario B, SOF/DAC was used only for non-cirrhotic patients and SOF/VEL was used for the cirrhotic patients. Healthcare costs associated with DAA therapy and disease stage monitoring were included to estimate the downstream cost implications.

    RESULTS: The stratified treatment cascade with 109 in Scenario B was found to be cost-saving compared to Scenario A. The cumulative savings for the stratified treatment cascade was USD 1.4 million over 5 years.

    DISCUSSION: A stratified treatment cascade with SOF/VEL was expected to be cost-saving and can result in a budget impact reduction in overall healthcare expenditure in Malaysia.

  9. Lim KK, Yoon SY, Mohd Taib NA, Shabaruddin FH, Dahlui M, Woo YL, et al.
    Appl Health Econ Health Policy, 2018 06;16(3):395-406.
    PMID: 29572724 DOI: 10.1007/s40258-018-0384-8
    OBJECTIVE: Previous studies showed that offering BRCA mutation testing to population subgroups at high risk of harbouring the mutation may be cost effective, yet no evidence is available for low- or middle-income countries (LMIC) and in Asia. We estimated the cost effectiveness of BRCA mutation testing in early-stage breast cancer patients with high pre-test probability of harbouring the mutation in Malaysia, an LMIC in Asia.

    METHODS: We developed a decision analytic model to estimate the lifetime costs and quality-adjusted life-years (QALYs) accrued through BRCA mutation testing or routine clinical surveillance (RCS) for a hypothetical cohort of 1000 early-stage breast cancer patients aged 40 years. In the model, patients would decide whether to accept testing and to undertake risk-reducing mastectomy, oophorectomy, tamoxifen, combinations or neither. We calculated the incremental cost-effectiveness ratio (ICER) from the health system perspective. A series of sensitivity analyses were performed.

    RESULTS: In the base case, testing generated 11.2 QALYs over the lifetime and cost US$4815 per patient whereas RCS generated 11.1 QALYs and cost US$4574 per patient. The ICER of US$2725/QALY was below the cost-effective thresholds. The ICER was sensitive to the discounting of cost, cost of BRCA mutation testing and utility of being risk-free, but the ICERs remained below the thresholds. Probabilistic sensitivity analysis showed that at a threshold of US$9500/QALY, 99.9% of simulations favoured BRCA mutation testing over RCS.

    CONCLUSIONS: Offering BRCA mutation testing to early-stage breast cancer patients identified using a locally-validated risk-assessment tool may be cost effective compared to RCS in Malaysia.

  10. Hiebert L, Hecht R, Soe-Lin S, Mohamed R, Shabaruddin FH, Syed Mansor SM, et al.
    Value Health Reg Issues, 2019 May;18:112-120.
    PMID: 30921591 DOI: 10.1016/j.vhri.2018.12.005
    BACKGROUND: In Malaysia, more than 330 000 individuals are estimated to be chronically infected with hepatitis C virus (HCV), but less than 2% have been treated to date.

    OBJECTIVES: To estimate the required coverage and costs of a national screening strategy to inform the launch of an HCV elimination program.

    METHODS: We designed an HCV screening strategy based on a "stepwise" approach. This approach relied on targeting of people who inject drugs in the early years, with delayed onset of widespread general population screening. Annual coverage requirements and associated costs were estimated to ensure that the World Health Organization elimination treatment targets were met.

    RESULTS: In total, 6 million individuals would have to be screened between 2018 and 2030. Targeting of people who inject drugs in the early years would limit annual screening coverage to less than 1 million individuals from 2018 to 2026. General population screening would have to be launched by 2026. Total costs were estimated at MYR 222 million ($58 million). Proportional to coverage targets, 60% of program costs would fall from 2026 to 2030.

    CONCLUSIONS: This exercise was one of the first attempts to conduct a detailed analysis of the required screening coverage and costs of a national HCV elimination strategy. These findings suggest that the stepwise approach could delay the onset of general population screening by more than 5 years after the program's launch. This delay would allow additional time to mobilize investments required for a successful general population screening program and also minimize program costs. This strategy prototype could inform the design of effective screening strategies in other countries.

  11. Chong HY, Lim KS, Fong SL, Shabaruddin FH, Dahlui M, Mei Lai PS, et al.
    Br J Clin Pharmacol, 2023 Nov;89(11):3340-3351.
    PMID: 37294011 DOI: 10.1111/bcp.15818
    AIMS: Despite the availability of newer antiseizure medications, carbamazepine (CBZ) remains the gold standard. However, patients of Asian ancestry are susceptible to CBZ-related severe cutaneous adverse reactions. Universal HLA-B*15:02 screening is a promising intervention to address this. With the increasing recognition of integrating real-world evidence in economic evaluations, the cost-effectiveness of universal HLA-B*15:02 screening was assessed using available real-world data in Malaysia.

    METHODS: A hybrid model of a decision tree and Markov model was developed to evaluate 3 strategies for treating newly diagnosed epilepsy among adults: (i) CBZ initiation without HLA-B*15:02 screening (current practice); (ii) universal HLA-B*15:02 screening prior to CBZ initiation; and (iii) alternative prescribing without HLA-B*15:02 screening. The model was populated with real-world inputs derived from the Malaysian population. From a societal perspective, base-case analysis and sensitivity analyses estimated the costs and outcomes over a lifetime. Incremental cost-effectiveness ratios were calculated.

    RESULTS: In the base-cases analysis, universal HLA-B*15:02 screening yielded the lowest total costs and the highest total quality-adjusted life years (QALYs) gained. Compared with current practice, universal screening was less costly by USD100 and more effective by QALYs increase of 0.1306, while alternative prescribing resulted in 0.1383 QALYs loss at additional costs of USD332. The highest seizure remission rate (56%) was estimated for universal HLA-B*15:02 screening vs. current practice (54%) and alternative prescribing (48%).

    CONCLUSION: Our study suggests that universal HLA-B*15:02 screening is a cost-effective intervention in Malaysia. With the demonstrated value of real-world evidence in economic evaluations, more relevant standardization efforts should be emphasized to better inform decision-making.

  12. Snyder SR, Hao J, Cavallari LH, Geng Z, Elsey A, Johnson JA, et al.
    Public Health Genomics, 2018;21(5-6):217-227.
    PMID: 31189173 DOI: 10.1159/000500725
    BACKGROUND/AIMS: Economic evaluation is integral to informed public health decision-making in the rapidly growing field of precision and personalized medicine (PM); however, this research requires specialized expertise and significant resources. Generic models are a novel innovation to efficiently address a critical PM evidence shortage and implementation barrier by enabling use of population-specific input values. This is a generic PM economic evaluation model proof-of-concept study for a pharmacogenomic use case.

    METHODS: An 8-step generic economic model development process was applied to the use case of human leukocyte antigen (HLA)-B*15:02genotyping for prediction of carbamazepine-induced cutaneous reactions, with a user-friendly decision-making tool relying on user-provided input values. This generic model was transparently documented and validated, including cross-validation comparing cost-effectiveness results with 3 country-specific models.

    RESULTS: A generic pharmacogenomic use case cost-effectiveness model with decision-making tool was successfully developed and cross-validated using input values for 6 populations which produced consistent results for HLA-B*15:02 screening at country-specific cost-effectiveness threshold values. Differences between the generic and country-specific model results were largely due to differences in model structure and assumptions.

    CONCLUSION: This proof on concept demonstrates the feasibility of generic models to provide useful PM economic evidence, supporting their use as a pragmatic and timely approach to address a growing need.

  13. Pok LSL, Shabaruddin FH, Dahlui M, Sockalingam S, Mohamed Said MS, Rosman A, et al.
    Int J Rheum Dis, 2018 May;21(5):943-951.
    PMID: 29314744 DOI: 10.1111/1756-185X.13256
    AIM: To determine the incidence and direct costs of NSAID-induced upper GI adverse events in Malaysian rheumatology patients.
    METHODS: A retrospective, multi-centre, cohort study of rheumatology patients on long-term NSAIDs was conducted. Clinical data of patients treated between 2010 and 2013 were collected for a 24-month follow-up period. The costs of managing upper GI adverse events were based on patient level resource use data.
    RESULTS: Six hundred and thirty-four patients met the inclusion criteria: mean age 53.4 years, 89.9% female, diagnosis of rheumatoid arthritis (RA; 59.3%), osteoarthritis (OA; 10.3%) and both RA and OA (30.3%). Three hundred and seventy-one (58.5%) patients were prescribed non-selective NSAIDs and 263 (41.5%) had cyclo-oxygenase-2 inhibitors. Eighty-four upper GI adverse events occurred, translating into a risk of 13.2% and an incidence rate of 66.2 per 1000 person-years. GI adverse events comprised: dyspepsia n = 78 (12.3%), peptic ulcer disease (PUD) n = 5 (0.79%) and upper GI bleeding (UGIB) n = 1 (0.16%). The total direct healthcare cost of managing adverse events was Malaysian Ringgit (MR) 37 352 (US dollars [USD] 11 419) with a mean cost of MR 446.81 ± 534.56 (USD 136.60 ± 163.42) per patient, consisting mainly of GI pharmacotherapy (33.8%), oesophagoduodenoscopies (23.1%) and outpatient clinic visits (18.2%). Mean cost per patient by GI events were: dyspepsia, MR 408.98 ± 513.29 (USD125.03 ± 156.92); PUD, MR 805.93 ± 578.80 (USD 246.39 ± 176.95); UGIB, MR 1601.94 (USD 489.74, n = 1).
    CONCLUSION: The economic burden of GI adverse events due to long-term NSAIDs use in Malaysian patients with chronic rheumatic diseases is modest.
    Study site: Rheumatology clinics, Hospital Putrajaya, Hospital Selayang, Pusat Perubatan Universiti Kebangsaan Malaysia (PPUKM), University Malaya Medical Centre (UMMC), Kuala Lumpur, Malaysia
  14. Chong HY, Mohamed Z, Tan LL, Wu DBC, Shabaruddin FH, Dahlui M, et al.
    Br J Dermatol, 2017 Oct;177(4):1102-1112.
    PMID: 28346659 DOI: 10.1111/bjd.15498
    BACKGROUND: A strong association has been documented between HLA-B*15:02 and carbamazepine-induced severe cutaneous adverse reactions (SCARs) in Asians. Human leucocyte antigen testing is potentially valuable in many countries to facilitate early recognition of patient susceptibility to SCARs.

    OBJECTIVES: To determine the cost-effectiveness of universal HLA-B*15:02 screening in preventing carbamazepine-induced Stevens-Johnson syndrome/toxic epidermal necrolysis in an ethnically diverse Malaysian population.

    METHODS: A hybrid model of a decision tree and Markov model was developed to evaluate three strategies for treating newly diagnosed epilepsy among adults: (i) carbamazepine initiation without HLA-B*15:02 screening (current practice); (ii) universal HLA-B*15:02 screening prior to carbamazepine initiation; and (iii) alternative treatment [sodium valproate (VPA)] prescribing without HLA-B*15:02 screening. Base-case analysis and sensitivity analyses were performed over a lifetime time horizon. Incremental cost-effectiveness ratios were calculated.

    RESULTS: Both universal HLA-B*15:02 screening and VPA prescribing were dominated by current practice. Compared with current practice, universal HLA-B*15:02 screening resulted in a loss of 0·0255 quality-adjusted life years (QALYs) at an additional cost of 707 U.S. dollars (USD); VPA prescribing resulted in a loss of 0·2622 QALYs at an additional cost of USD 4127, owing to estimated differences in antiepileptic treatment efficacy.

    CONCLUSIONS: Universal HLA-B*15:02 screening is unlikely to be a cost-effective intervention in Malaysia. However, with the emergence of an ethnically diverse population in many other countries, this may render HLA-B*15:02 screening a viable intervention when an increasing proportion of the population is at risk and an equally effective yet safer antiepileptic drug is available.

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