Literature DB >> 18797062

Homocystinuria due to cystathionine beta synthase deficiency.

T Narayana Rao1, K Radhakrishna, T S Mohana Rao, P Guruprasad, Kamal Ahmed.   

Abstract

A two year-old male child presented with cutis marmorata congenita universalis, brittle hair, mild mental retardation, and finger spasms. Biochemical findings include increased levels of homocysteine in the blood-106.62 micromol/L (normal levels: 5.90-16 micromol/L). Biochemical tests such as the silver nitroprusside and nitroprusside tests were positive suggesting homocystinuria. The patient was treated with oral pyridoxine therapy for three months. The child responded well to this therapy and the muscle spasms as well as skin manifestations such as cutis marmorata subsided. The treatment is being continued; the case is reported here because of its rarity. Homocysteinuria arising due to cystathionine beta-synthase (CBS) deficiency is an autosomal recessive disorder of methionine metabolism that produces increased levels of urinary homocysteine and methionine It manifests itself in vascular, central nervous system, cutaneous, and connective tissue disturbances and phenotypically resembles Marfan's syndrome. Skin manifestations include malar flush, thin hair, and cutis reticulata / marmorata.

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Year:  2008        PMID: 18797062     DOI: 10.4103/0378-6323.42916

Source DB:  PubMed          Journal:  Indian J Dermatol Venereol Leprol        ISSN: 0378-6323            Impact factor:   2.545


  4 in total

Review 1.  Cystathionine β-synthase deficiency: Of mice and men.

Authors:  Warren D Kruger
Journal:  Mol Genet Metab       Date:  2017-05-19       Impact factor: 4.797

Review 2.  Oxidative Stress in Homocystinuria Due to Cystathionine ß-Synthase Deficiency: Findings in Patients and in Animal Models.

Authors:  Jéssica Lamberty Faverzani; Tatiane Grazieli Hammerschmidt; Angela Sitta; Marion Deon; Moacir Wajner; Carmen Regla Vargas
Journal:  Cell Mol Neurobiol       Date:  2017-03-03       Impact factor: 5.046

3.  A Case Report of Homocystinuria With Dystonia and Stroke.

Authors:  Arezoo Rezazadeh; Shahram Oveisgharan; Gholamali Shahidi; Reza Naghdi
Journal:  Child Neurol Open       Date:  2014-08-26

4.  Homocystinuria with Stroke and Positive Familial History.

Authors:  Ali Mazaheri; Neda Mostofizadeh; Mahin Hashemipour
Journal:  Adv Biomed Res       Date:  2017-10-25
  4 in total

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