Literature DB >> 22520346

Emerging treatments in the management of tuberous sclerosis complex.

Michael H Kohrman1.   

Abstract

Tuberous sclerosis complex is a genetic disorder characterized by the formation of nonmalignant hamartomas in the brain, heart, skin, kidney, lung, and other organs. It is associated with autism, epilepsy, and other neurocognitive and behavioral disabilities. Wide phenotypic variation occurs in disease severity and natural course: some patients demonstrate minimal effects, e.g., skin changes; others manifest profound seizures and mental retardation. Tuberous sclerosis complex is caused by mutations in either the tuberous sclerosis complex 1 or 2 gene (coding for hamartin and tuberin, respectively). The tuberous sclerosis complex 1/tuberous sclerosis complex 2 protein dimer complex is a crucial inhibitory element in the mammalian target of rapamycin pathway, regulating cell growth and proliferation. Until recently, few options existed, other than surgery, for treating symptoms of tuberous sclerosis complex related to the growth of hamartomas. Increased understanding of the genetic cause of the disease and underlying dysregulation of the mammalian target of rapamycin pathway has led to clinical trials of mammalian target of rapamycin inhibitors, including sirolimus and everolimus. This review gives an overview of tuberous sclerosis complex and its molecular causes, and summarizes results from recent clinical trials of mammalian target of rapamycin inhibitors in patients with the disease.
Copyright © 2012 Elsevier Inc. All rights reserved.

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Year:  2012        PMID: 22520346     DOI: 10.1016/j.pediatrneurol.2012.02.015

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


  20 in total

Review 1.  Tuberous sclerosis complex: new insights into clinical and therapeutic approach.

Authors:  Angela Volpi; Gabriele Sala; Elena Lesma; Francesca Labriola; Marco Righetti; Rosa Maria Alfano; Mario Cozzolino
Journal:  J Nephrol       Date:  2018-11-07       Impact factor: 3.902

2.  Tuberous sclerosis complex; single center experience.

Authors:  İlknur Erol; Tülin Savaş; Sevda Şekerci; Nalan Yazıcı; Ayşe Erbay; Şenay Demir; Semra Saygı; Özlem Alkan
Journal:  Turk Pediatri Ars       Date:  2015-03-01

Review 3.  A case of subependymal giant cell astrocytoma without tuberous sclerosis complex and review of the literature.

Authors:  Michael O'Rawe; Arjun S Chandran; Stuti Joshi; Alexandre Simonin; Jason M Dyke; Sharon Lee
Journal:  Childs Nerv Syst       Date:  2020-08-17       Impact factor: 1.475

Review 4.  Hypomelanoses in children.

Authors:  Nanja van Geel; Marijn Speeckaert; Ines Chevolet; Sofie De Schepper; Hilde Lapeere; Barbara Boone; Reinhart Speeckaert
Journal:  J Cutan Aesthet Surg       Date:  2013-04

5.  TOSCA - first international registry to address knowledge gaps in the natural history and management of tuberous sclerosis complex.

Authors:  John C Kingswood; Paolo Bruzzi; Paolo Curatolo; Petrus J de Vries; Carla Fladrowski; Christoph Hertzberg; Anna C Jansen; Sergiusz Jozwiak; Rima Nabbout; Matthias Sauter; Renaud Touraine; Finbar O'Callaghan; Bernard Zonnenberg; Stefania Crippa; Silvia Comis; Guillaume Beaure d'Augères; Elena Belousova; Tom Carter; Vincent Cottin; Maria Dahlin; José Carlos Ferreira; Alfons Macaya; Mirjana Perkovic Benedik; Valentin Sander; Sotirios Youroukos; Ramon Castellana; Bulent Ulker; Martha Feucht
Journal:  Orphanet J Rare Dis       Date:  2014-11-26       Impact factor: 4.123

Review 6.  Genetics of tuberous sclerosis complex: implications for clinical practice.

Authors:  Carolina Caban; Nubaira Khan; Daphne M Hasbani; Peter B Crino
Journal:  Appl Clin Genet       Date:  2016-12-21

7.  TuberOus SClerosis registry to increase disease Awareness (TOSCA) - baseline data on 2093 patients.

Authors:  John C Kingswood; Guillaume B d'Augères; Elena Belousova; José C Ferreira; Tom Carter; Ramon Castellana; Vincent Cottin; Paolo Curatolo; Maria Dahlin; Petrus J de Vries; Martha Feucht; Carla Fladrowski; Gabriella Gislimberti; Christoph Hertzberg; Sergiusz Jozwiak; John A Lawson; Alfons Macaya; Rima Nabbout; Finbar O'Callaghan; Mirjana P Benedik; Jiong Qin; Ruben Marques; Valentin Sander; Matthias Sauter; Yukitoshi Takahashi; Renaud Touraine; Sotiris Youroukos; Bernard Zonnenberg; Anna C Jansen
Journal:  Orphanet J Rare Dis       Date:  2017-01-05       Impact factor: 4.123

8.  Endoplasmic reticulum stress and cell death in mTORC1-overactive cells is induced by nelfinavir and enhanced by chloroquine.

Authors:  Charlotte E Johnson; David K Hunt; Marie Wiltshire; Terry P Herbert; Julian R Sampson; Rachel J Errington; D Mark Davies; Andrew R Tee
Journal:  Mol Oncol       Date:  2014-11-22       Impact factor: 6.603

Review 9.  Everolimus in the treatment of subependymal giant cell astrocytomas, angiomyolipomas, and pulmonary and skin lesions associated with tuberous sclerosis complex.

Authors:  David Neal Franz
Journal:  Biologics       Date:  2013-10-10

10.  EFFECTS: an expanded access program of everolimus for patients with subependymal giant cell astrocytoma associated with tuberous sclerosis complex.

Authors:  Andras Fogarasi; Liesbeth De Waele; Gabriella Bartalini; Sergiusz Jozwiak; Nicola Laforgia; Helene Verhelst; Borivoj Petrak; Jean-Michel Pedespan; Olaf Witt; Ramon Castellana; Stefania Crippa; Gabriella Gislimberti; Zsuzsanna Gyorsok
Journal:  BMC Neurol       Date:  2016-08-08       Impact factor: 2.474

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