Literature DB >> 26078697

Tuberous sclerosis complex; single center experience.

İlknur Erol1, Tülin Savaş1, Sevda Şekerci2, Nalan Yazıcı3, Ayşe Erbay3, Şenay Demir4, Semra Saygı1, Özlem Alkan4.   

Abstract

AIM: This study was planned with the aim of retrospectively reviewing the clinical and laboratory findings and therapies of our patients diagnosed with tuberous sclerosis and redefining the patients according to the diagnostic criteria revised by the 2012 International Tuberous Sclerosis Complex Consensus Group and comparing them with the literature. MATERIALS AND
METHOD: Twenty patients diagnosed with tuberous sclerosis complex in the Pediatric Neurology Clinic were examined retrospectively in terms of clinical findings and therapies. The diagnoses were compared again according to 1998 and 2012 criteria.
RESULTS: It was observed that the complaint at presentation was seizure in 17 of 20 patients and hypopigmented spots on the skin in 3 of 20 patients. On the initial physical examination, findings related with the disease were found in the skin in 17 of the patients, in the eye in 5, in the kidneys in 7 and in the brain with imaging in 17. No cardiac involvement was observed in the patients. Infantile spasm was observed in 7 of the patients who presented because of seizure (n=17), partial seizure was observed in 7 and multiple seizure types were observed in 3. It was found that sirolimus treatment was given to 9 of 20 patients because of different reasons, 7 of these 9 patients had epileptic seizures and sirolimus treatment had no effect on epileptic seizures. According to 2012 diagnostic criteria, no marked change occured in the diagnoses of our patients.
CONCLUSIONS: It was observed that the signs and symptoms of our patients were compatible with the literature. Molecular genetic examination was planned for the patients who were being followed up because of probable tuberous sclerosis complex. It was observed that sirolimus treatment had no marked effect on the seizure frequency of our patients.

Entities:  

Keywords:  Diagnostic criteria; mammalian target of rapamycin (mTOR); tuberous sclerosis complex

Year:  2015        PMID: 26078697      PMCID: PMC4462331          DOI: 10.5152/tpa.2015.2138

Source DB:  PubMed          Journal:  Turk Pediatri Ars


  38 in total

1.  Multiple cardiac rhabdomyomas as a sole symptom of tuberous sclerosis complex: case report with molecular confirmation.

Authors:  Sergiusz Jóźwiak; Dorota Domańska-Pakieła; David J Kwiatkowski; Katarzyna Kotulska
Journal:  J Child Neurol       Date:  2005-12       Impact factor: 1.987

2.  The cutaneous features of tuberous sclerosis: a population study.

Authors:  D W Webb; A Clarke; A Fryer; J P Osborne
Journal:  Br J Dermatol       Date:  1996-07       Impact factor: 9.302

Review 3.  Current development of mTOR inhibitors as anticancer agents.

Authors:  Sandrine Faivre; Guido Kroemer; Eric Raymond
Journal:  Nat Rev Drug Discov       Date:  2006-08       Impact factor: 84.694

4.  Mutational analysis in a cohort of 224 tuberous sclerosis patients indicates increased severity of TSC2, compared with TSC1, disease in multiple organs.

Authors:  S L Dabora; S Jozwiak; D N Franz; P S Roberts; A Nieto; J Chung; Y S Choy; M P Reeve; E Thiele; J C Egelhoff; J Kasprzyk-Obara; D Domanska-Pakiela; D J Kwiatkowski
Journal:  Am J Hum Genet       Date:  2000-12-08       Impact factor: 11.025

5.  Tuberous sclerosis and infantile spasms.

Authors:  R Riikonen; O Simell
Journal:  Dev Med Child Neurol       Date:  1990-03       Impact factor: 5.449

Review 6.  Burden of disease and unmet needs in tuberous sclerosis complex with neurological manifestations: systematic review.

Authors:  Lindsay Hallett; Talia Foster; Zhimei Liu; Marissa Blieden; Joice Valentim
Journal:  Curr Med Res Opin       Date:  2011-06-21       Impact factor: 2.580

7.  Neuro-epileptic determinants of autism spectrum disorders in tuberous sclerosis complex.

Authors:  Patrick F Bolton; Rebecca J Park; J Nicholas P Higgins; Paul D Griffiths; Andrew Pickles
Journal:  Brain       Date:  2002-06       Impact factor: 13.501

8.  Cardiac rhabdomyomas and their association with tuberous sclerosis.

Authors:  D W Webb; R D Thomas; J P Osborne
Journal:  Arch Dis Child       Date:  1993-03       Impact factor: 3.791

9.  Cognitive impairment in tuberous sclerosis complex is a multifactorial condition.

Authors:  F E Jansen; K L Vincken; A Algra; P Anbeek; O Braams; M Nellist; B A Zonnenberg; A Jennekens-Schinkel; A van den Ouweland; D Halley; A C van Huffelen; O van Nieuwenhuizen
Journal:  Neurology       Date:  2007-11-21       Impact factor: 9.910

10.  Behavior problems in children with tuberous sclerosis complex and parental stress.

Authors:  Claudine M C Kopp; David A Muzykewicz; Brigid A Staley; Elizabeth A Thiele; Margaret B Pulsifer
Journal:  Epilepsy Behav       Date:  2008-07-07       Impact factor: 2.937

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  4 in total

1.  Renal involvement in tuberous sclerosis complex.

Authors:  Alper Alp
Journal:  Turk Pediatri Ars       Date:  2015-12-01

2.  Assessment of tumors in children with tuberous sclerosis: a single centre's experience.

Authors:  Suna Emir; Şadan Hacısalihoğlu; Derya Özyörük; Filiz Ekici; Aydan Değerliyurt; Alev Güven; İlker Çetin
Journal:  Turk Pediatri Ars       Date:  2017-03-01

3.  CD99: A potential Diagnostic Marker for Differentiating Sub-ependymal Giant Cell Astrocytoma From Other Mimickers: A Report of Five Cases.

Authors:  Alireza Sadeghipour; Navid Abdi; Pegah Babaheidarian
Journal:  Iran J Pathol       Date:  2017-07-01

Review 4.  Efficacy and safety of mTOR inhibitors (rapamycin and its analogues) for tuberous sclerosis complex: a meta-analysis.

Authors:  Min Li; Ying Zhou; Chaoyang Chen; Ting Yang; Shuang Zhou; Shuqing Chen; Ye Wu; Yimin Cui
Journal:  Orphanet J Rare Dis       Date:  2019-02-13       Impact factor: 4.123

  4 in total

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