Literature DB >> 24913039

Prenatal diagnosis of giant cardiac rhabdomyoma in tuberous sclerosis complex: a new therapeutic option with everolimus.

E Mlczoch1, A Hanslik, D Luckner, E Kitzmüller, D Prayer, I Michel-Behnke.   

Abstract

Tuberous sclerosis complex (TSC) is a genetic disorder characterized by abnormal cell proliferation and tumor growth in a number of organ systems, primarily the brain, kidneys, eyes and heart. Clinical symptoms vary according to the location of the tumor. The most common disorders are seizures, neurodevelopmental disorders, renal failure and arrhythmias. TSC was found to be influenced by inhibitors of the protein kinase mammalian target of rapamycin (mTOR), which regulates abnormal cellular proliferation. mTOR inhibitors have been studied effectively in patients with subependymal giant-cell astrocytomas and renal angiolipomas in the context of TSC. We describe a prenatally diagnosed case of giant rhabdomyoma, due to right ventricular outflow tract obstruction, which presented as a duct-dependent lesion. Postnatal treatment with the mTOR inhibitor everolimus initiated significant regression of the cardiac tumor. This finding suggests that mTOR inhibitor therapy is an option for giant rhabdomyomas that develop in the neonatal period.
Copyright © 2014 ISUOG. Published by John Wiley & Sons Ltd.

Entities:  

Keywords:  brain cortical tumor; mTOR inhibitor; magnetic resonance imaging; right ventricular outflow obstruction

Mesh:

Substances:

Year:  2015        PMID: 24913039     DOI: 10.1002/uog.13434

Source DB:  PubMed          Journal:  Ultrasound Obstet Gynecol        ISSN: 0960-7692            Impact factor:   7.299


  8 in total

1.  Accelerated Cardiac Rhabdomyoma Regression with Everolimus in Infants with Tuberous Sclerosis Complex.

Authors:  Fatou Aw; Isabelle Goyer; Marie-Josée Raboisson; Christine Boutin; Philippe Major; Nagib Dahdah
Journal:  Pediatr Cardiol       Date:  2016-11-23       Impact factor: 1.655

Review 2.  Cardiovascular manifestations of tuberous sclerosis complex and summary of the revised diagnostic criteria and surveillance and management recommendations from the International Tuberous Sclerosis Consensus Group.

Authors:  Robert B Hinton; Ashwin Prakash; Robb L Romp; Darcy A Krueger; Timothy K Knilans
Journal:  J Am Heart Assoc       Date:  2014-11-25       Impact factor: 5.501

3.  Regression of Neonatal Cardiac Rhabdomyoma in Two Months Through Low-Dose Everolimus Therapy: A Report of Three Cases.

Authors:  Jeng-Sheng Chang; Ping-Yun Chiou; Shu-Hui Yao; I-Ching Chou; Ching-Yuang Lin
Journal:  Pediatr Cardiol       Date:  2017-08-05       Impact factor: 1.655

Review 4.  Perfect match: mTOR inhibitors and tuberous sclerosis complex.

Authors:  Cong Luo; Wen-Rui Ye; Wei Shi; Ping Yin; Chen Chen; Yun-Bo He; Min-Feng Chen; Xiong-Bin Zu; Yi Cai
Journal:  Orphanet J Rare Dis       Date:  2022-03-04       Impact factor: 4.123

Review 5.  Treatment of Cardiac Rhabdomyomas with mTOR Inhibitors in Children with Tuberous Sclerosis Complex-A Systematic Review.

Authors:  Monika Sugalska; Anna Tomik; Sergiusz Jóźwiak; Bożena Werner
Journal:  Int J Environ Res Public Health       Date:  2021-05-05       Impact factor: 3.390

6.  Congenital subependymal giant cell astrocytomas in patients with tuberous sclerosis complex.

Authors:  Katarzyna Kotulska; Julita Borkowska; Marek Mandera; Marcin Roszkowski; Elzbieta Jurkiewicz; Wiesława Grajkowska; Małgorzata Bilska; Sergiusz Jóźwiak
Journal:  Childs Nerv Syst       Date:  2014-09-17       Impact factor: 1.475

7.  Safety and efficacy of mTOR inhibitor treatment in patients with tuberous sclerosis complex under 2 years of age - a multicenter retrospective study.

Authors:  Afshin Saffari; Ines Brösse; Adelheid Wiemer-Kruel; Bernd Wilken; Paula Kreuzaler; Andreas Hahn; Matthias K Bernhard; Cornelis M van Tilburg; Georg F Hoffmann; Matthias Gorenflo; Sven Hethey; Olaf Kaiser; Stefan Kölker; Robert Wagner; Olaf Witt; Andreas Merkenschlager; Andreas Möckel; Timo Roser; Jan-Ulrich Schlump; Antje Serfling; Juliane Spiegler; Till Milde; Andreas Ziegler; Steffen Syrbe
Journal:  Orphanet J Rare Dis       Date:  2019-05-03       Impact factor: 4.123

8.  A case report of severe tuberous sclerosis complex detected in utero and linked to a novel duplication in the TSC2 gene.

Authors:  Valérie Mongrain; Nicolaas H van Doesburg; Françoise Rypens; Catherine Fallet-Bianco; Justine Maassen; Julien Dufort-Gervais; Lucie Côté; Philippe Major
Journal:  BMC Neurol       Date:  2020-09-01       Impact factor: 2.474

  8 in total

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