Literature DB >> 16138897

Self-mutilation behaviour in Lesch-Nyhan syndrome.

R G E C Cauwels1, L C Martens.   

Abstract

Lesch-Nyhan syndrome (LNS), first described in 1964 by Lesch and Nyhan, is a rare X-linked genetic disorder involving (near) absence of the enzyme hypoxanthine-guanine phosphoribosyl transferase (HPRT). It occurs in 1:100,000 to 380,000 live births (1, 2). The deficiency of HPRT activity leads to an excessive uric acid production resulting in neurological, renal and musculoskeletal manifestations. Death usually occurs in the second or third decade from infection or renal failure. Clinical presentation is characterized by mental retardation, choreoathetosis, spasticity, hyperuricemia and cerebral palsy. A characteristic feature of LNS is the appearance of intractable self-injurious behaviour (SIB), usually in the form of severe lip and finger biting, gouging of eyes, face scratching and head banging requiring extreme management techniques such as the application of restraints and or extraction of teeth at an early age. In this case report a unique approach of SIB in LNS is presented.

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Year:  2005        PMID: 16138897     DOI: 10.1111/j.1600-0714.2005.00330.x

Source DB:  PubMed          Journal:  J Oral Pathol Med        ISSN: 0904-2512            Impact factor:   4.253


  4 in total

1.  Case report: A unique presentation of severe tongue biting in 10 month-old twins with a novel approach to management.

Authors:  A Suchak; M Mars
Journal:  Eur Arch Paediatr Dent       Date:  2010-06

2.  Lesch-Nyhan syndrome: The saga of metabolic abnormalities and self-injurious behavior.

Authors:  Nitesh Tewari; Vijay Prakash Mathur; Divesh Sardana; Kalpana Bansal
Journal:  Intractable Rare Dis Res       Date:  2017-02

3.  Oral Self-Mutilation in Lesch-Nyhan Syndrome: A Case Report.

Authors:  José Ferrão; Cristina Rodrigues Barros; Luísa Figueiredo; Ana Fernandes
Journal:  Cureus       Date:  2022-08-11

4.  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
  4 in total

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