Literature DB >> 28357186

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

Nitesh Tewari1, Vijay Prakash Mathur1, Divesh Sardana1, Kalpana Bansal1.   

Abstract

Lesch-Nyhan syndrome (LNS) is an X-linked recessive disorder of purine metabolism caused by a mutation in Xq26.2-q26.3 (OMIM 308000.0004). The presence of the diagnostic triad, i.e. signs of self-injurious behavior (SIB) and results of pedigree analysis and novel molecular biology & genetic testing, confirms the diagnosis of LNS. With a level of hypoxanthine guanine phosphoribosyl-transferase 1 (HPRT1) enzyme activity < 2%, patients develop neurological, neurocognitive, and neuromotor symptoms along with SIB. Described here is a case of 4-year-old boy who was diagnosed with LNS. The boy displayed SIB, i.e. biting of the lips and fingers, and he had cerebral venous sinus thrombosis caused by LNS.

Entities:  

Keywords:  Lesch-Nyhan disease; X-linked disorder; self-injurious behavior

Year:  2017        PMID: 28357186      PMCID: PMC5359358          DOI: 10.5582/irdr.2016.01076

Source DB:  PubMed          Journal:  Intractable Rare Dis Res        ISSN: 2186-3644


  12 in total

1.  Modes and patterns of self-mutilation in persons with Lesch-Nyhan disease.

Authors:  Kenneth L Robey; John F Reck; Karen D Giacomini; Gabor Barabas; Gary E Eddey
Journal:  Dev Med Child Neurol       Date:  2003-03       Impact factor: 5.449

2.  A FAMILIAL DISORDER OF URIC ACID METABOLISM AND CENTRAL NERVOUS SYSTEM FUNCTION.

Authors:  M LESCH; W L NYHAN
Journal:  Am J Med       Date:  1964-04       Impact factor: 4.965

3.  Recurrent thrombosis in a patient with Lesch-Nyhan syndrome.

Authors:  Irbaz Bin Riaz; Muhammad Husnain; Huthayfa Ateeli
Journal:  Am J Med       Date:  2014-02-13       Impact factor: 4.965

4.  Sudden death in Lesch-Nyhan disease.

Authors:  Vladimir Kostadinov Neychev; H A Jinnah
Journal:  Dev Med Child Neurol       Date:  2006-11       Impact factor: 5.449

5.  Self-mutilation behaviour in Lesch-Nyhan syndrome.

Authors:  R G E C Cauwels; L C Martens
Journal:  J Oral Pathol Med       Date:  2005-10       Impact factor: 4.253

6.  Oral self-mutilation by a 17-month-old child with Lesch-Nyhan syndrome.

Authors:  N Rashid; H Yusuf
Journal:  Int J Paediatr Dent       Date:  1997-06       Impact factor: 3.455

7.  Clinical severity in Lesch-Nyhan disease: the role of residual enzyme and compensatory pathways.

Authors:  Rong Fu; Diane Sutcliffe; Hong Zhao; Xinyi Huang; David J Schretlen; Steve Benkovic; H A Jinnah
Journal:  Mol Genet Metab       Date:  2014-11-08       Impact factor: 4.797

8.  Management of oral lesions in Lesch-Nyham syndrome.

Authors:  M Romero Maroto; C Ruiz Duque; G Vincent; I Garcia Recuero; A Romance
Journal:  J Clin Pediatr Dent       Date:  2014       Impact factor: 1.065

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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