Literature DB >> 18425437

Serum prolactin as a tool for the follow-up of treated DHPR-deficient patients.

D Concolino1, G Muzzi, M Rapsomaniki, M T Moricca, M G Pascale, P Strisciuglio.   

Abstract

Deficiency of dihydropteridine reductase causes a variant form of phenylketonuria associated with a devastating neurological disease characterized by mental retardation, hypokinesis and other features relating to basal ganglia disorder. Hyperphenylalaninaemias with tetrahydrobiopterin deficiency make up about 1-3% of all hyperphenylalaninaemias. We describe three patients from Calabria, a southern region of Italy, who have a dihydropteridine reductase deficiency, caused by the same mutation (p.L14P) also found in the nearby region of Sicily. We report the evolution of clinical and biochemical data during the treatment of these patients where we used prolactin serum determination to adapt the specific therapy. This report suggests that serum prolactin levels can be a good biomarker for optimal dosage of hydroxylated precursors in long-term treatment monitoring.

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Year:  2008        PMID: 18425437     DOI: 10.1007/s10545-007-0788-3

Source DB:  PubMed          Journal:  J Inherit Metab Dis        ISSN: 0141-8955            Impact factor:   4.982


  15 in total

1.  Genotype-phenotype correlation in dihydropteridine reductase deficiency.

Authors:  L de Sanctis; C Alliaudi; M Spada; R Farrugia; R Cerone; G Biasucci; C Meli; E Zammarchi; T Coskun; N Blau; A Ponzone; I Dianzani
Journal:  J Inherit Metab Dis       Date:  2000-06       Impact factor: 4.982

2.  Molecular analysis of dihydropteridine reductase deficiency: identification of two novel mutations in Japanese patients.

Authors:  H Ikeda; Y Matsubara; H Mikami; S Kure; M Owada; T Gough; P M Smooker; M Dobbs; H H Dahl; R G Cotton; K Narisawa
Journal:  Hum Genet       Date:  1997-10       Impact factor: 4.132

3.  Monitoring treatment in tetrahydrobiopterin deficiency by serum prolactin.

Authors:  M Spada; S Ferraris; G B Ferrero; M Sartore; C Lanza; F Perfetto; L de Sanctis; C Dompé; N Blau; A Ponzone
Journal:  J Inherit Metab Dis       Date:  1996       Impact factor: 4.982

4.  DHPR deficiency in Italy.

Authors:  A Ponzone; V Ricca; S Ferraris; O Guardamagna; G Bracco; S Pagliardini; F Levis; M Giovannini; E Riva; R Longhi
Journal:  J Pediatr       Date:  1984-12       Impact factor: 4.406

5.  Structure and expression of human dihydropteridine reductase.

Authors:  J Lockyer; R G Cook; S Milstien; S Kaufman; S L Woo; F D Ledley
Journal:  Proc Natl Acad Sci U S A       Date:  1987-05       Impact factor: 11.205

Review 6.  Dihydropteridine reductase deficiency in man: from biology to treatment.

Authors:  Alberto Ponzone; Marco Spada; Silvio Ferraris; Irma Dianzani; Luisa de Sanctis
Journal:  Med Res Rev       Date:  2004-03       Impact factor: 12.944

7.  Atypical (mild) forms of dihydropteridine reductase deficiency: neurochemical evaluation and mutation detection.

Authors:  N Blau; C W Heizmann; W Sperl; G C Korenke; G F Hoffmann; P M Smooker; R G Cotton
Journal:  Pediatr Res       Date:  1992-12       Impact factor: 3.756

8.  Malignant hyperphenylalaninemia--clinical features, biochemical findings, and experience with administration of biopterins.

Authors:  D M Danks; P Schlesinger; F Firgaira; R G Cotton; B M Watson; H Rembold; G Hennings
Journal:  Pediatr Res       Date:  1979-10       Impact factor: 3.756

9.  Identification and in vitro expression of mutations causing dihydropteridine reductase deficiency.

Authors:  P M Smooker; D W Howells; R G Cotton
Journal:  Biochemistry       Date:  1993-06-29       Impact factor: 3.162

Review 10.  Molecular basis of dihydropteridine reductase deficiency.

Authors:  P M Smooker; R G Cotton
Journal:  Hum Mutat       Date:  1995       Impact factor: 4.878

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  4 in total

1.  An international survey of patients with tetrahydrobiopterin deficiencies presenting with hyperphenylalaninaemia.

Authors:  Thomas Opladen; Georg F Hoffmann; Nenad Blau
Journal:  J Inherit Metab Dis       Date:  2012-06-23       Impact factor: 4.982

Review 2.  Disorders of biopterin metabolism.

Authors:  Nicola Longo
Journal:  J Inherit Metab Dis       Date:  2009-02-09       Impact factor: 4.982

Review 3.  Genetics of monoamine neurotransmitter disorders.

Authors:  Wai-Kwan Siu
Journal:  Transl Pediatr       Date:  2015-04

Review 4.  Consensus guideline for the diagnosis and treatment of tetrahydrobiopterin (BH4) deficiencies.

Authors:  Thomas Opladen; Eduardo López-Laso; Elisenda Cortès-Saladelafont; Toni S Pearson; H Serap Sivri; Yilmaz Yildiz; Birgit Assmann; Manju A Kurian; Vincenzo Leuzzi; Simon Heales; Simon Pope; Francesco Porta; Angeles García-Cazorla; Tomáš Honzík; Roser Pons; Luc Regal; Helly Goez; Rafael Artuch; Georg F Hoffmann; Gabriella Horvath; Beat Thöny; Sabine Scholl-Bürgi; Alberto Burlina; Marcel M Verbeek; Mario Mastrangelo; Jennifer Friedman; Tessa Wassenberg; Kathrin Jeltsch; Jan Kulhánek; Oya Kuseyri Hübschmann
Journal:  Orphanet J Rare Dis       Date:  2020-05-26       Impact factor: 4.123

  4 in total

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