Literature DB >> 19631922

Cell encapsulation as a potential nondietary therapy for maternal phenylketonuria.

Donna A Santillan1, Mark K Santillan, Stephen K Hunter.   

Abstract

OBJECTIVE: The objective of this work was to determine whether cells overexpressing phenylalanine (Phe) hydroxylase (PAH) can significantly reduce Phe in vitro for potential use as a therapy for preventing maternal phenylketonuria. STUDY
DESIGN: Human 293T and WRL68 cell lines were transiently and stably transfected to overexpress PAH. Cells were encapsulated within microspheres of sodium alginate. Timed measurements of Phe in media were performed using tandem mass spectrometry.
RESULTS: Both nonencapsulated and encapsulated transiently transfected cells overexpressing PAH significantly reduced the Phe concentration in media by approximately 50% in comparison to mock-transfected cells. Cell line clones stably expressing PAH significantly decreased the Phe concentration in the media by up to 85% compared with media alone.
CONCLUSION: Both unencapsulated and encapsulated cells overexpressing PAH significantly reduce Phe in vitro. Studies using phenylketonuria model mice will be important in determining the ability of our therapy to prevent the teratogenic effects of elevated maternal Phe in maternal phenylketonuria.

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Year:  2009        PMID: 19631922      PMCID: PMC8928171          DOI: 10.1016/j.ajog.2009.05.035

Source DB:  PubMed          Journal:  Am J Obstet Gynecol        ISSN: 0002-9378            Impact factor:   8.661


  41 in total

1.  The enzymatic conversion of phenylalanine to tyrosine.

Authors:  S UDENFRIEND; J R COOPER
Journal:  J Biol Chem       Date:  1952-02       Impact factor: 5.157

Review 2.  Maternal phenylketonuria: a metabolic teratogen.

Authors:  H L Levy; M Ghavami
Journal:  Teratology       Date:  1996-03

3.  Administration-route and gender-independent long-term therapeutic correction of phenylketonuria (PKU) in a mouse model by recombinant adeno-associated virus 8 pseudotyped vector-mediated gene transfer.

Authors:  Z Ding; P Georgiev; B Thöny
Journal:  Gene Ther       Date:  2006-04       Impact factor: 5.250

4.  Research design, organization, and sample characteristics of the Maternal PKU Collaborative Study.

Authors:  Richard Koch; Colleen Azen; Eva Friedman; William Hanley; Harvey Levy; Reuben Matalon; Bobbye Rouse; Friedrich Trefz; Jiaping Ning; Felix de la Cruz
Journal:  Pediatrics       Date:  2003-12       Impact factor: 7.124

5.  Polydisperse dextran as a diffusing test solute to study the membrane permeability of alginate polylysine microcapsules.

Authors:  V Coromili; T M Chang
Journal:  Biomater Artif Cells Immobilization Biotechnol       Date:  1993

6.  Encapsulated beta-islet cells as a bioartificial pancreas to treat insulin-dependent diabetes during pregnancy.

Authors:  S K Hunter; Y Wang; C P Weiner; J Niebyl
Journal:  Am J Obstet Gynecol       Date:  1997-10       Impact factor: 8.661

7.  Promotion of neovascularization around hollow fiber bioartificial organs using biologically active substances.

Authors:  S K Hunter; J M Kao; Y Wang; J A Benda; V G Rodgers
Journal:  ASAIO J       Date:  1999 Jan-Feb       Impact factor: 2.872

8.  Long-term enzymatic and phenotypic correction in the phenylketonuria mouse model by adeno-associated virus vector-mediated gene transfer.

Authors:  Hyun-Jeong Oh; Eun-Sook Park; Seongman Kang; Inho Jo; Sung-Chul Jung
Journal:  Pediatr Res       Date:  2004-06-04       Impact factor: 3.756

9.  How practical are recommendations for dietary control in phenylketonuria?

Authors:  J H Walter; F J White; S K Hall; A MacDonald; G Rylance; A Boneh; D E Francis; G J Shortland; M Schmidt; A Vail
Journal:  Lancet       Date:  2002-07-06       Impact factor: 79.321

10.  Maternal phenylketonuria and hyperphenylalaninemia. An international survey of the outcome of untreated and treated pregnancies.

Authors:  R R Lenke; H L Levy
Journal:  N Engl J Med       Date:  1980-11-20       Impact factor: 91.245

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