Literature DB >> 25771998

Tuberous sclerosis complex: a survey of health care resource use and health burden.

Anne M Rentz1, Anne M Skalicky2, Zhimei Liu3, James W Wheless4, David W Dunn5, Michael D Frost6, JoAnne Nakagawa7, Matthew Magestro3, Judith Prestifilippo3.   

Abstract

PURPOSE: Tuberous sclerosis complex (TSC) is a multiorgan, autosomal-dominant genetic disorder with incomplete penetrance.
METHODS: This analysis of a web-based survey focuses on the clinical presentation, management, and associated burden of patients with TSC in the United States.
RESULTS: A total of 676 TSC patients or caregivers responded. Both pediatric and adult patient groups experienced skin lesions (77% and 44%), seizures (77% and 24%), and kidney complications (33% and 25%) as well as other manifestations. Patient groups averaged 22 visits to a physician, nine procedures/tests, two emergency room visits, and two hospital admissions in the past year. Standardized tests were administered for health-related quality of life and TSC patients reported significantly worse mental health scores and better physical health scores compared to a normative sample of cancer patients.
CONCLUSION: Results demonstrate that TSC is associated with significant clinical burden, resource utilization, and decreased mental health well-being.
Copyright © 2015 Elsevier Inc. All rights reserved.

Entities:  

Keywords:  children and adults; health care utilization; health-related quality of life; tuberous sclerosis complex (TSC)

Mesh:

Year:  2014        PMID: 25771998     DOI: 10.1016/j.pediatrneurol.2014.11.013

Source DB:  PubMed          Journal:  Pediatr Neurol        ISSN: 0887-8994            Impact factor:   3.372


  10 in total

1.  The Adult Phenotype of Tuberous Sclerosis Complex.

Authors:  Neera Nathan; Kristina Burke; Cory Trickett; Joel Moss; Thomas N Darling
Journal:  Acta Derm Venereol       Date:  2016-02       Impact factor: 4.437

2.  Clinical, genetic and quality-of-life study of a cohort of adult patients with tuberous sclerosis.

Authors:  Elena Cristina De Sautu De Borbón; Juan Manuel Guerra Vales; Carlos Lumbreras Bermejo; Felix Guerrero Ramos; María José Buj Padilla; Jesús González de la Aleja; Montserrat Morales Conejo
Journal:  Orphanet J Rare Dis       Date:  2021-05-31       Impact factor: 4.123

Review 3.  A systematic review on the burden of illness in individuals with tuberous sclerosis complex (TSC).

Authors:  Johann Philipp Zöllner; David Neal Franz; Christoph Hertzberg; Rima Nabbout; Felix Rosenow; Matthias Sauter; Susanne Schubert-Bast; Adelheid Wiemer-Kruel; Adam Strzelczyk
Journal:  Orphanet J Rare Dis       Date:  2020-01-21       Impact factor: 4.123

4.  Treatment Patterns and Use of Resources in Patients With Tuberous Sclerosis Complex: Insights From the TOSCA Registry.

Authors:  Ruben Marques; Elena Belousova; Mirjana P Benedik; Tom Carter; Vincent Cottin; Paolo Curatolo; Maria Dahlin; Lisa D'Amato; Guillaume Beaure d'Augères; Petrus J de Vries; José C Ferreira; Martha Feucht; Carla Fladrowski; Christoph Hertzberg; Sergiusz Jozwiak; John A Lawson; Alfons Macaya; Rima Nabbout; Finbar O'Callaghan; Jiong Qin; Valentin Sander; Matthias Sauter; Seema Shah; Yukitoshi Takahashi; Renaud Touraine; Sotiris Youroukos; Bernard Zonnenberg; John C Kingswood; Anna C Jansen
Journal:  Front Neurol       Date:  2019-10-25       Impact factor: 4.003

Review 5.  The research landscape of tuberous sclerosis complex-associated neuropsychiatric disorders (TAND)-a comprehensive scoping review.

Authors:  Stephanie Vanclooster; Stacey Bissell; Agnies M van Eeghen; Nola Chambers; Liesbeth De Waele; Anna W Byars; Jamie K Capal; Sebastián Cukier; Peter Davis; Jennifer Flinn; Sugnet Gardner-Lubbe; Tanjala Gipson; Tosca-Marie Heunis; Dena Hook; J Christopher Kingswood; Darcy A Krueger; Aubrey J Kumm; Mustafa Sahin; Eva Schoeters; Catherine Smith; Shoba Srivastava; Megumi Takei; Robert Waltereit; Anna C Jansen; Petrus J de Vries
Journal:  J Neurodev Disord       Date:  2022-02-13       Impact factor: 4.025

6.  Understanding the health economic burden of patients with tuberous sclerosis complex (TSC) with epilepsy: a retrospective cohort study in the UK Clinical Practice Research Datalink (CPRD).

Authors:  Charles Shepherd; Matthias Koepp; Melissa Myland; Keyur Patel; Cristiana Miglio; Vathani Siva; Elizabeth Gray; Maureen Neary
Journal:  BMJ Open       Date:  2017-10-05       Impact factor: 2.692

7.  Clinical and genetic analysis of tuberous sclerosis complex-associated renal angiomyolipoma in Chinese pedigrees.

Authors:  Shuqiang Li; Yushi Zhang; Jinxing Wei; Xuepei Zhang
Journal:  Oncol Lett       Date:  2017-09-27       Impact factor: 2.967

8.  Natural clusters of tuberous sclerosis complex (TSC)-associated neuropsychiatric disorders (TAND): new findings from the TOSCA TAND research project.

Authors:  Petrus J de Vries; Elena Belousova; Mirjana P Benedik; Tom Carter; Vincent Cottin; Paolo Curatolo; Lisa D'Amato; Guillaume Beure d'Augères; José C Ferreira; Martha Feucht; Carla Fladrowski; Christoph Hertzberg; Sergiusz Jozwiak; John A Lawson; Alfons Macaya; Ruben Marques; Rima Nabbout; Finbar O'Callaghan; Jiong Qin; Valentin Sander; Matthias Sauter; Seema Shah; Yukitoshi Takahashi; Renaud Touraine; Sotiris Youroukos; Bernard Zonnenberg; J Chris Kingswood; Anna C Jansen
Journal:  J Neurodev Disord       Date:  2020-09-01       Impact factor: 4.025

9.  Prevalence, mortality and healthcare economic burden of tuberous sclerosis in Hong Kong: a population-based retrospective cohort study (1995-2018).

Authors:  William Ching-Yuen Chu; Lorraine Lok-Wing Chiang; Dorothy Chi-Ching Chan; Wilfred Hing-Sang Wong; Godfrey Chi-Fung Chan
Journal:  Orphanet J Rare Dis       Date:  2020-09-25       Impact factor: 4.123

10.  Patient and Caregiver Health State Utilities in Tuberous Sclerosis Complex.

Authors:  Siu Hing Lo; Jade Marshall; Hanna Skrobanski; Andrew Lloyd
Journal:  Pharmacoecon Open       Date:  2021-09-15
  10 in total

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