BACKGROUND: Tuberous sclerosis complex (TSC) is a multi-system genetic disorder characterized by the development of diverse clinical manifestations. The complexity of this disease is likely to result in substantial challenges and costs in disease management throughout the patient's lifetime. This retrospective database study aims to quantify healthcare resources utilized by TSC patients. METHODS: TSC patients in the Clinical Practice Research Datalink linked to the Hospital Episodes Statistics database were identified between January 1987 and June 2013. Analyses were conducted over the most recent 3-year period of data and stratified by pediatric (< 18) and adult patients. Prescriptions, procedures, diagnostic tests, and healthcare encounters were reported in comparison with a matched comparator cohort. Costs and key economic drivers by primary organ system manifestations were also examined. RESULTS: A total of 286 patients with TSC were identified and consistently reported 2-fold greater resource use than the matched presumably healthy controls. Despite this comparatively greater resource use, half of TSC patients did not record any procedures, and 20% of patients did not record any diagnostic tests; however, inpatient hospitalizations were greater for the TSC cohort (3.1 vs 1.3), but length of stay was comparable. TSC patients had costs totaling £12,681 per patient over the 3-year period, a figure 2.7-fold greater than the total costs in the comparator cohort (£4,777). Costs for patients with specific primary manifestations were even greater, with brain manifestations incurring £22,139 per affected patient. Kidney and nervous system manifestations were the main cost drivers. CONCLUSIONS: The economic burden of TSC and its impact on NHS healthcare resources is mostly attributable to the broad spectrum of manifestations that develop within multiple organ systems. TSC patients may benefit from co-ordinated care based on their requirement for high numbers of healthcare visits across specialties.
BACKGROUND:Tuberous sclerosis complex (TSC) is a multi-system genetic disorder characterized by the development of diverse clinical manifestations. The complexity of this disease is likely to result in substantial challenges and costs in disease management throughout the patient's lifetime. This retrospective database study aims to quantify healthcare resources utilized by TSC patients. METHODS: TSC patients in the Clinical Practice Research Datalink linked to the Hospital Episodes Statistics database were identified between January 1987 and June 2013. Analyses were conducted over the most recent 3-year period of data and stratified by pediatric (< 18) and adult patients. Prescriptions, procedures, diagnostic tests, and healthcare encounters were reported in comparison with a matched comparator cohort. Costs and key economic drivers by primary organ system manifestations were also examined. RESULTS: A total of 286 patients with TSC were identified and consistently reported 2-fold greater resource use than the matched presumably healthy controls. Despite this comparatively greater resource use, half of TSC patients did not record any procedures, and 20% of patients did not record any diagnostic tests; however, inpatient hospitalizations were greater for the TSC cohort (3.1 vs 1.3), but length of stay was comparable. TSC patients had costs totaling £12,681 per patient over the 3-year period, a figure 2.7-fold greater than the total costs in the comparator cohort (£4,777). Costs for patients with specific primary manifestations were even greater, with brain manifestations incurring £22,139 per affected patient. Kidney and nervous system manifestations were the main cost drivers. CONCLUSIONS: The economic burden of TSC and its impact on NHS healthcare resources is mostly attributable to the broad spectrum of manifestations that develop within multiple organ systems. TSC patients may benefit from co-ordinated care based on their requirement for high numbers of healthcare visits across specialties.
Authors: Raouf M Seyam; Waleed K Alkhudair; Said A Kattan; Mohamed F Alotaibi; Hassan M Alzahrani; Waleed M Altaweel Journal: J Kidney Cancer VHL Date: 2017-10-16
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Authors: Johann Philipp Zöllner; Janina Grau; Felix Rosenow; Matthias Sauter; Markus Knuf; Gerhard Kurlemann; Thomas Mayer; Christoph Hertzberg; Astrid Bertsche; Ilka Immisch; Karl Martin Klein; Susanne Knake; Klaus Marquard; Sascha Meyer; Anna H Noda; Felix von Podewils; Hannah Schäfer; Charlotte Thiels; Laurent M Willems; Bianca Zukunft; Susanne Schubert-Bast; Adam Strzelczyk Journal: Orphanet J Rare Dis Date: 2021-06-02 Impact factor: 4.123
Authors: Johann Philipp Zöllner; Nadine Conradi; Matthias Sauter; Markus Knuf; Susanne Knake; Gerhard Kurlemann; Thomas Mayer; Christoph Hertzberg; Astrid Bertsche; Ilka Immisch; Karl Martin Klein; Klaus Marquard; Sascha Meyer; Anna H Noda; Felix von Podewils; Hannah Schäfer; Charlotte Thiels; Bianca Zukunft; Susanne Schubert-Bast; Janina Grau; Laurent M Willems; Felix Rosenow; Jens-Peter Reese; Adam Strzelczyk Journal: Neurol Res Pract Date: 2021-06-28
Authors: Nicholas M P Annear; Richard E Appleton; Zahabiyah Bassi; Rupesh Bhatt; Patrick F Bolton; Pamela Crawford; Alex Crowe; Maureen Tossi; Frances Elmslie; Eric Finlay; Daniel P Gale; Alex Henderson; Elizabeth A Jones; Simon R Johnson; Shelagh Joss; Larissa Kerecuk; Graham Lipkin; Patrick J Morrison; Finbar J O'Callaghan; Jill Cadwgan; Albert C M Ong; Julian R Sampson; Charles Shepherd; J Chris Kingswood Journal: Front Neurol Date: 2019-11-06 Impact factor: 4.003