Cost-utility analysis of competing treatment strategies for drug-resistant epilepsy in children with Tuberous Sclerosis Complex

Fallah, Aria; Weil, Alexander G.; Wang, Shelly; Lewis, Evan; Baca, Christine B.; Mathern, Gary W.

doi:10.1016/j.yebeh.2016.07.034

Cited by 22 publications

(41 citation statements)

References 40 publications

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“…Direct costs are reported by 10 studies [23, 35, 104, 106, 107, 110-112, 114, 119] and four studies assessed drivers of direct cost [23,35,110,111]. The measures of DALY and QALY were only used by one study that calculated projected costs of different treatment strategies for epilepsy in TSC [109]. Fourteen studies reported on quality of life [53,56,101,103,105,106,108,115,118,119,[121][122][123]125].…”

Section: Burden Of Illness and Resource Usementioning

confidence: 99%

“…Fallah et al [109] estimated the theoretical costeffectiveness of four different therapy strategies in pediatric TSC patients with drug-refractory epilepsy, specifically epilepsy surgery, VNS, ketogenic diet, and carbamazepine as an additional third ASD. The costeffectiveness was modeled based on adjusted historic costs and data from an open cost-effectiveness registry.…”

Section: Projected Costsmentioning

confidence: 99%

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A systematic review on the burden of illness in individuals with tuberous sclerosis complex (TSC)

Zöllner

Franz

Hertzberg

et al. 2020

Orphanet J Rare Dis

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Objective: This review will summarize current knowledge on the burden of illness (BOI) in tuberous sclerosis complex (TSC), a multisystem genetic disorder manifesting with hamartomas throughout the body, including mainly the kidneys, brain, skin, eyes, heart, and lungs. Methods: We performed a systematic analysis of the available literature on BOI in TSC according to the PRISMA guidelines. All studies irrespective of participant age that reported on individual and societal measures of disease burden (e.g. health care resource use, costs, quality of life) were included. Results: We identified 33 studies reporting BOI in TSC patients. Most studies (21) reported health care resource use, while 14 studies reported quality of life and 10 studies mentioned costs associated with TSC. Only eight research papers reported caregiver BOI. Substantial BOI occurs from most manifestations of the disorder, particularly from pharmacoresistant epilepsy, neuropsychiatric, renal and skin manifestations. While less frequent, pulmonary complications also lead to a high individual BOI. The range for the mean annual direct costs varied widely between 424 and 98,008 International Dollar purchasing power parities (PPP-$). Brain surgery, end-stage renal disease with dialysis, and pulmonary complications all incur particularly high costs. There is a dearth of information regarding indirect costs in TSC. Mortality overall is increased compared to general population; and most TSC related deaths occur as a result of complications from seizures as well as renal complications. Long term studies report mortality between 4.8 and 8.3% for a follow-up of 8 to 17.4 years. Conclusions: TSC patients and their caregivers have a high burden of illness, and TSC patients incur high costs in health care systems. At the same time, the provision of inadequate treatment that does not adhere to published guidelines is common and centralized TSC care is received by no more than half of individuals who need it, especially adults. Further studies focusing on the cost effectiveness and BOI outcomes of coordinated TSC care as well as of new treatment options such as mTOR inhibitors are necessary.

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Section: Burden Of Illness and Resource Usementioning

confidence: 99%

Section: Projected Costsmentioning

confidence: 99%

A systematic review on the burden of illness in individuals with tuberous sclerosis complex (TSC)

Zöllner

Franz

Hertzberg

et al. 2020

Orphanet J Rare Dis

View full text Add to dashboard Cite

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“…For instance, Fallah, Wang, Lewis, Baca, and Mathern (2016) showed that resective epilepsy surgery resulted as the most costeffective treatment option in TSC children with DRE after the introduction of a third AED but before vagus nerve stimulation, and the authors concluded that the most cost-effective treatment should be chosen based on available resources. For instance, Fallah, Wang, Lewis, Baca, and Mathern (2016) showed that resective epilepsy surgery resulted as the most costeffective treatment option in TSC children with DRE after the introduction of a third AED but before vagus nerve stimulation, and the authors concluded that the most cost-effective treatment should be chosen based on available resources.…”

Section: Surgical Treatmentmentioning

confidence: 99%

“…Finally, cost effectiveness is another parameter that should be kept in mind when evaluating possible surgery or other treatment options. For instance, Fallah, Wang, Lewis, Baca, and Mathern (2016) showed that resective epilepsy surgery resulted as the most costeffective treatment option in TSC children with DRE after the introduction of a third AED but before vagus nerve stimulation, and the authors concluded that the most cost-effective treatment should be chosen based on available resources.…”

Section: Surgical Treatmentmentioning

confidence: 99%

“…In a series of 11 individuals with drug resistant epilepsy and TSC, Elliott et al (2009) reported that nine of them (82%) experienced at least a 67% reduction in seizure burden. Finally, a cost-utility analysis found VNS implantation to be more expensive and less effective than alternative strategies, and suggested that it should not be prioritized (Fallah et al, 2016).…”

Section: Vagus Nerve Stimulationmentioning

confidence: 99%

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Current concepts on epilepsy management in tuberous sclerosis complex

Canevini

Curatolo

Briola

et al. 2018

American J of Med Genetics Pt C

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Tuberous sclerosis complex (TSC) is an autosomal dominant neurocutaneous disease affecting approximately 1 in 6,000 people, and represents one of the most common genetic causes of epilepsy. Epilepsy affects 90% of the patients and appears in the first 2 years of life in the majority of them. Early onset of epilepsy in the first 12 months of life is associated with high risk of cognitive decline and neuropsychiatric problems including autism. Prenatal or early infantile diagnosis of TSC, before the onset of epilepsy, provides a unique opportunity to monitor EEG before the onset of clinical seizures, thus enabling early intervention in the process of epileptogenesis. In this review, we discuss the current status of knowledge on epileptogenesis in TSC, and present recommendations of American and European experts in the field of epilepsy.

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Pharmacoresistance in Epilepsy

San-Juan,

Peralta

2023

Pharmacoresistance in Epilepsy

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Cost-utility analysis of competing treatment strategies for drug-resistant epilepsy in children with Tuberous Sclerosis Complex

Cited by 22 publications

References 40 publications

A systematic review on the burden of illness in individuals with tuberous sclerosis complex (TSC)

A systematic review on the burden of illness in individuals with tuberous sclerosis complex (TSC)

Current concepts on epilepsy management in tuberous sclerosis complex

Pharmacoresistance in Epilepsy

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