Literature DB >> 16138673

Botulinum toxin as a novel treatment for self-mutilation in Lesch-Nyhan syndrome.

Edward Dabrowski1, Sarah A Smathers, Curt S Ralstrom, Michael A Nigro, Jimmie P Leleszi.   

Abstract

Lesch-Nyhan syndrome (LNS) is an X-linked recessive disorder resulting from a deficiency of the metabolic enzyme hypozanthine-guanine phosphoribosyltransferase (HPRT). This syndrome presents with abnormal metabolic and neurological manifestations including hyperuricemia, mental retardation*, spastic cerebral palsy (CP), dystonia, and self-mutilation. The mechanism behind the severe self-mutilating behavior exhibited by patients with LNS is unknown and remains one of the greatest obstacles in providing care to these patients. This report describes a 10-year-old male child with confirmed LNS who was treated for self-mutilation of his hands, tongue, and lips with repeated botulinum toxin A (BTX-A) injections into the bilateral masseters. Our findings suggest that treatment with BTX-A affects both the central and peripheral nervous systems, resulting in reduced self-abusive behavior in this patient.

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Year:  2005        PMID: 16138673

Source DB:  PubMed          Journal:  Dev Med Child Neurol        ISSN: 0012-1622            Impact factor:   5.449


  11 in total

Review 1.  Lesch-Nyhan Syndrome: Models, Theories, and Therapies.

Authors:  Scott Bell; Ilaria Kolobova; Liam Crapper; Carl Ernst
Journal:  Mol Syndromol       Date:  2016-09-24

2.  Consequences of Delayed Dental Extraction in Lesch-Nyhan Disease.

Authors:  Emily M Goodman; Rosa J Torres; Juan G Puig; H A Jinnah
Journal:  Mov Disord Clin Pract       Date:  2014-09

Review 3.  Inborn errors of purine and pyrimidine metabolism.

Authors:  A Jurecka
Journal:  J Inherit Metab Dis       Date:  2009-03-15       Impact factor: 4.982

4.  Botulinum toxin type A in the healing of chronic lesion following bilateral spasticity of gluteus muscle.

Authors:  Emanuele Cigna; Michele Maruccia; Benedetta Fanelli; Nicolò Scuderi
Journal:  Int Wound J       Date:  2012-10-19       Impact factor: 3.315

5.  The differential diagnosis of Huntington's disease-like syndromes: 'red flags' for the clinician.

Authors:  Davide Martino; Maria Stamelou; Kailash P Bhatia
Journal:  J Neurol Neurosurg Psychiatry       Date:  2012-09-19       Impact factor: 10.154

6.  Kinetic and reaction pathway analysis in the application of botulinum toxin a for wound healing.

Authors:  Frank J Lebeda; Zygmunt F Dembek; Michael Adler
Journal:  J Toxicol       Date:  2011-11-24

Review 7.  Hypoxanthine-guanine phosophoribosyltransferase (HPRT) deficiency: Lesch-Nyhan syndrome.

Authors:  Rosa J Torres; Juan G Puig
Journal:  Orphanet J Rare Dis       Date:  2007-12-08       Impact factor: 4.123

8.  Oral Hemorrhage in a 3-year-old Boy Caused by Self-Mutilating Behavior.

Authors:  Jenny Lyn Y Uy; José Florencio F Lapeña
Journal:  Pak J Med Sci       Date:  2016 Nov-Dec       Impact factor: 1.088

9.  A modified intraoral resin mouthguard to prevent self-mutilations in lesch-nyhan patients.

Authors:  Giulia Ragazzini; Alessia Delucchi; Enrico Calcagno; Roberto Servetto; Gloria Denotti
Journal:  Int J Dent       Date:  2014-07-02

10.  Lesch-Nyhan Syndrome: Disorder of Self-mutilating Behavior.

Authors:  Prasad Jathar; Amey M Panse; Madhura Jathar; Pritesh N Gawali
Journal:  Int J Clin Pediatr Dent       Date:  2016-06-15
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