Literature DB >> 25180276

Rapid regression of left ventricular outflow tract rhabdomyoma after sirolimus therapy.

Colm Breathnach1, Jane Pears2, Orla Franklin1, David Webb3, Colin J McMahon4.   

Abstract

The neonatal presentation of cardiac rhabdomyomas varies in severity from severe outflow tract obstruction to minimal cardiac dysfunction. The natural history for these lesions is spontaneous regression in the majority of cases. We describe a newborn boy with severe left ventricular outflow tract obstruction secondary to a large rhabdomyoma. The tumor infiltrated the paraaortic area and extended around the origin of the right coronary artery, making surgical resection challenging. Oral sirolimus therapy resulted in a rapid regression of the tumor and alleviation of outflow tract obstruction within 1 month of treatment. This is the first report of sirolimus therapy in alleviating critical left ventricular outflow tract obstruction in this condition.
Copyright © 2014 by the American Academy of Pediatrics.

Entities:  

Keywords:  obstruction; rhabdomyoma; sirolimus; tuberous sclerosis; tumor

Mesh:

Substances:

Year:  2014        PMID: 25180276     DOI: 10.1542/peds.2013-3293

Source DB:  PubMed          Journal:  Pediatrics        ISSN: 0031-4005            Impact factor:   7.124


  10 in total

1.  Successful use of sirolimus for refractory atrial ectopic tachycardia in a child with cardiac rhabdomyoma.

Authors:  Sanja Ninic; Marija Kalaba; Bosiljka Jovicic; Vladislav Vukomanovic; Sergej Prijic; Biljana Vucetic; Ruzica Kravljanac; Ana Vujic; Jovan Kosutic
Journal:  Ann Noninvasive Electrocardiol       Date:  2017-02-19       Impact factor: 1.468

2.  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

3.  Innovative Strategies for the Management of a Massive Neonatal Rhabdomyoma.

Authors:  N Prabhu; N Osifodunrin; D Murphy; S Butler; L E Hunter
Journal:  J Pediatr Intensive Care       Date:  2017-09-12

4.  Maternal Sirolimus Therapy for Fetal Cardiac Rhabdomyomas.

Authors:  Benjamin T Barnes; David Procaccini; Jude Crino; Karin Blakemore; Priya Sekar; Katelynn G Sagaser; Angie C Jelin; Lasya Gaur
Journal:  N Engl J Med       Date:  2018-05-10       Impact factor: 91.245

5.  Multiple cardiac rhabdomyomas in tuberous sclerosis complex: case report and review of the literature.

Authors:  Paula Frudit; Bruno Kusznir Vitturi; Flavia Cristina Navarro; Ivan Rondelli; Geanete Pozzan
Journal:  Autops Case Rep       Date:  2019-09-30

6.  Genotype and Phenotype Landscape of 283 Japanese Patients with Tuberous Sclerosis Complex.

Authors:  Sumihito Togi; Hiroki Ura; Hisayo Hatanaka; Yo Niida
Journal:  Int J Mol Sci       Date:  2022-09-22       Impact factor: 6.208

Review 7.  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

Review 8.  Is mTOR Inhibitor Good Enough for Treatment All Tumors in TSC Patients?

Authors:  Samy L Habib; Noor Y Al-Obaidi; Maciej Nowacki; Katarzyna Pietkun; Barbara Zegarska; Tomasz Kloskowski; Wojciech Zegarski; Tomasz Drewa; Edward A Medina; Zhenze Zhao; Sitai Liang
Journal:  J Cancer       Date:  2016-07-21       Impact factor: 4.207

9.  Sirolimus in Infants with Multiple Cardiac Rhabdomyomas Associated with Tuberous Sclerosis Complex.

Authors:  Maurizio Lucchesi; Enrico Chiappa; Flavio Giordano; Francesco Mari; Lorenzo Genitori; Iacopo Sardi
Journal:  Case Rep Oncol       Date:  2018-06-28

10.  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

  10 in total

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