Literature DB >> 20823030

Experience with hyperphenylalaninemia in a developing country: unusual clinical manifestations and a novel gene mutation.

Pascale E Karam1, Rose T Daher, Lisbeth B Moller, Mohamad A Mikati.   

Abstract

We report our experience in a cohort of patients with hyperphenylalaninemia in a tertiary care referral center in Lebanon. Forty-one sequential patients were studied: 34 classical phenylketonuria (PKU), 3 hyperphenylalaninemia (non-PKU), and 4 biopterin metabolism defects. The majority of cases were clinically diagnosed at variable ages with variable neurological outcomes. Only 29.3% were detected by neonatal screening. Two unusual cases were observed in the context of inadequate treatment in 1 and delayed therapy in the other: a newborn with PKU developed severe keratomalacia; and a 5-year-old girl with dihydropteridine reductase deficiency due to a novel mutation identified in the quinoid dihydropteridine reductase gene developed Lennox-Gastaut syndrome and white matter changes with periventricular cysts. Part of our experience parallels that in the West. However, the clinical manifestations observed in our patients emphasize the importance of a national newborn screening program with efficient management of diagnosed cases.

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Year:  2010        PMID: 20823030     DOI: 10.1177/0883073810375116

Source DB:  PubMed          Journal:  J Child Neurol        ISSN: 0883-0738            Impact factor:   1.987


  2 in total

1.  Phenylketonuria screening and management in southeastern Europe - survey results from 11 countries.

Authors:  Mojca Zerjav Tansek; Urh Groselj; Natalija Angelkova; Dana Anton; Ivo Baric; Maja Djordjevic; Lindita Grimci; Maria Ivanova; Adil Kadam; Vjosa Kotori; Hajrija Maksic; Oana Marginean; Otilia Margineanu; Olivera Miljanovic; Florentina Moldovanu; Mariana Muresan; Michaela Nanu; Mira Samardzic; Vladimir Sarnavka; Aleksei Savov; Maja Stojiljkovic; Biljana Suzic; Radka Tincheva; Husref Tahirovic; Alma Toromanovic; Natalia Usurelu; Tadej Battelino
Journal:  Orphanet J Rare Dis       Date:  2015-05-30       Impact factor: 4.123

Review 2.  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

  2 in total

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