Literature DB >> 26800857

An estimation of the incidence of tuberous sclerosis complex in a nationwide retrospective cohort study (1997-2010).

C-H Hong1,2, H-P Tu3, J-R Lin4, C-H Lee5,6.   

Abstract

BACKGROUND: Tuberous sclerosis complex (TSC) is caused by mutations in TSC1 and TSC2, leading to mammalian target of rapamycin hyperactivation. Patients with TSC develop hamartomas in brain, lungs, liver and skin. Two epidemiological studies, performed in Minnesota, U.S.A., have estimated the incidence of TSC to be 0·28-0·56 per 100 000 person-years (PY), based on < 12 patients. Furthermore, whether common comorbidities are associated with this rare disease is not known.
OBJECTIVES: To estimate the incidence of TSC and investigate the associations of TSC with other comorbidities, including diabetes, peptic ulcers, stroke and myocardial infarction.
METHODS: We estimated the incidence and prevalence of TSC and its comorbidities from 1997 to 2010, based on the Catastrophic Illness Certificate disease database and a beneficiary cohort of 1 million people.
RESULTS: The incidence of TSC in Taiwan is 0·153 per 100 000 PY. The number of patients identified with TSC in Taiwan doubled from 206 in 2006 to 471 in 2010. In 2010, the prevalence of TSC in Taiwan was estimated to be 1·58 in 100 000. We confirmed that female patients with TSC are more likely to develop renal tumours than male patients. Surprisingly, patients with TSC have a significantly decreased risk of developing peptic ulcers compared with controls.
CONCLUSIONS: This is the first large-scale and longitudinal incidence study of TSC. This study provides compelling evidence that TSC mutations in humans are associated with a decreased risk of peptic ulcers.
© 2016 British Association of Dermatologists.

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Year:  2016        PMID: 26800857     DOI: 10.1111/bjd.14415

Source DB:  PubMed          Journal:  Br J Dermatol        ISSN: 0007-0963            Impact factor:   9.302


  14 in total

1.  Rapamycin and dexamethasone during pregnancy prevent tuberous sclerosis complex-associated cystic kidney disease.

Authors:  Morris Nechama; Yaniv Makayes; Elad Resnick; Karen Meir; Oded Volovelsky
Journal:  JCI Insight       Date:  2020-07-09

2.  Deceased Donor Renal Transplantation Combined with Bilateral Nephrectomy in a Patient with Tuberous Sclerosis and Renal Failure.

Authors:  R Novotny; J Chlupac; T Marada; S Bloudickova-Rajnochova; H Vavrinova; L Janousek; J Fronek
Journal:  Case Rep Transplant       Date:  2019-03-06

3.  First comprehensive TSC1/TSC2 mutational analysis in Mexican patients with Tuberous Sclerosis Complex reveals numerous novel pathogenic variants.

Authors:  Miriam E Reyna-Fabián; Nancy L Hernández-Martínez; Miguel A Alcántara-Ortigoza; Jorge T Ayala-Sumuano; Sergio Enríquez-Flores; José A Velázquez-Aragón; Alfredo Varela-Echavarría; Carlos G Todd-Quiñones; Ariadna González-Del Angel
Journal:  Sci Rep       Date:  2020-04-20       Impact factor: 4.379

Review 4.  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

Review 5.  Renal tumors in tuberous sclerosis complex.

Authors:  Peter Trnka; Sean E Kennedy
Journal:  Pediatr Nephrol       Date:  2020-10-01       Impact factor: 3.714

Review 6.  Direct and indirect costs and cost-driving factors in adults with tuberous sclerosis complex: a multicenter cohort study and a review of the literature.

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

7.  Quality of life and its predictors in adults with tuberous sclerosis complex (TSC): a multicentre cohort study from Germany.

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

8.  Incidence of tuberous sclerosis and age at first diagnosis: new data and emerging trends from a national, prospective surveillance study.

Authors:  Daniel Ebrahimi-Fakhari; Lilian Lisa Mann; Martin Poryo; Norbert Graf; Rüdiger von Kries; Beate Heinrich; Darius Ebrahimi-Fakhari; Marina Flotats-Bastardas; Ludwig Gortner; Michael Zemlin; Sascha Meyer
Journal:  Orphanet J Rare Dis       Date:  2018-07-17       Impact factor: 4.123

9.  Sex-Selective Effects on Behavior in a Mouse Model of Tuberous Sclerosis Complex.

Authors:  Rachel Michelle Saré; Abigail Lemons; Christopher Figueroa; Alex Song; Merlin Levine; Carolyn Beebe Smith
Journal:  eNeuro       Date:  2020-04-29

10.  Rare inherited kidney diseases: an evolving field in Nephrology.

Authors:  Mariana Faucz Munhoz da Cunha; Gabriela Sevignani; Giovana Memari Pavanelli; Mauricio de Carvalho; Fellype Carvalho Barreto
Journal:  J Bras Nefrol       Date:  2020-03-20
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