Literature DB >> 35449882

A Case Report of Topiramate for Severe Breath Holding Spells in a Teenage Boy with Pitt-Hopkins Syndrome.

Megan Bone1, Kimberly Goodspeed1.   

Abstract

Pitt-Hopkins syndrome is a rare genetic neurodevelopmental disorder characterized by intellectual disability, delayed motor development, and absent speech. Patients often show symptoms of respiratory dysrhythmia, including episodes of hyperpnea followed by apnea with cyanosis. These spells occur while awake and do not have ictal correlate on electroencephalogram (EEG). The episodes can become quite frequent and can be challenging to treat. We present a case of a teenage patient with Pitt-Hopkins syndrome who had very frequent apneic spells that responded well to treatment with topiramate after limited response to acetazolamide.
© The Author(s) 2022.

Entities:  

Keywords:  autism; developmental disability; genetics; intellectual disability; neurodevelopment

Year:  2022        PMID: 35449882      PMCID: PMC9016566          DOI: 10.1177/2329048X221093172

Source DB:  PubMed          Journal:  Child Neurol Open        ISSN: 2329-048X


  12 in total

1.  Acetazolamide for severe apnea in Pitt-Hopkins syndrome.

Authors:  Stijn L Verhulst; J De Dooy; J Ramet; N Bockaert; R Van Coster; B Ceulemans; W De Backer
Journal:  Am J Med Genet A       Date:  2012-03-09       Impact factor: 2.802

Review 2.  Pitt-Hopkins Syndrome: A Review of Current Literature, Clinical Approach, and 23-Patient Case Series.

Authors:  Kimberly Goodspeed; Cassandra Newsom; Mary Ann Morris; Craig Powell; Patricia Evans; Sailaja Golla
Journal:  J Child Neurol       Date:  2018-01-10       Impact factor: 1.987

3.  Successful use of acetazolamide for central apnea in a child with Pitt-Hopkins syndrome.

Authors:  Claire Gaffney; Paul McNally
Journal:  Am J Med Genet A       Date:  2015-04-21       Impact factor: 2.802

4.  Clinical and polygraphic improvement of breathing abnormalities after valproate in a case of Pitt-Hopkins syndrome.

Authors:  Ilenia Maini; Gaetano Cantalupo; Emanuela Claudia Turco; Fernando De Paolis; Cinzia Magnani; Liborio Parrino; Mario Giovanni Terzano; Francesco Pisani
Journal:  J Child Neurol       Date:  2012-02-28       Impact factor: 1.987

5.  Effect of topiramate on seizures and respiratory dysrhythmia in Rett syndrome.

Authors:  Monisha Goyal; Mary Ann O'Riordan; Max Wiznitzer
Journal:  J Child Neurol       Date:  2004-08       Impact factor: 1.987

6.  Severe respiratory dysrhythmia in Rett syndrome treated with topiramate.

Authors:  Natalija Krajnc
Journal:  J Child Neurol       Date:  2013-12-05       Impact factor: 1.987

7.  Two types of early epileptic encephalopathy in a Pitt-Hopkins syndrome patient with a novel TCF4 mutation.

Authors:  Hinako Kirikae; Mitsugu Uematsu; Yurika Numata-Uematsu; Naoya Saijo; Yu Katata; Yoshitsugu Oikawa; Atsuo Kikuchi; Kumiko Yanagi; Tadashi Kaname; Kazuhiro Haginoya; Shigeo Kure
Journal:  Brain Dev       Date:  2021-09-24       Impact factor: 1.961

Review 8.  Central sleep apnea: misunderstood and mistreated!

Authors:  Jerome A Dempsey
Journal:  F1000Res       Date:  2019-06-28

9.  Psychopharmacology of topiramate: from epilepsy to bipolar disorder.

Authors:  Marco Mula; Andrea E Cavanna; Francesco Monaco
Journal:  Neuropsychiatr Dis Treat       Date:  2006-12       Impact factor: 2.570

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