Literature DB >> 14970743

Homozygous acute intermittent porphyria in a 7-year-old boy with massive excretions of porphyrins and porphyrin precursors.

J Hessels1, G Voortman, A van der Wagen, C van der Elzen, H Scheffer, F M J Zuijderhoudt.   

Abstract

A 7-year-old boy demonstrating hepatosplenomegaly, mild anaemia, mild mental retardation, yellow-brown teeth and dark red urine had excessively elevated levels of urinary delta-aminolevulinic acid, porphobilinogen and uroporphyrin. Furthermore hepta-, hexa-, penta- and copro(I)porphyrins were highly increased in urine. This pattern of porphyrin precursor and metabolite excretion is characteristic of acute intermittent porphyria. The decreased copro(III)/copro(I+III) ratio, normally not found in acute intermittent porphyria, is discussed. The porphobilinogen deaminase activity in red cells was decreased to 2-4%. Mutation analysis revealed a novel homozygous L81P mutation in exon 6 of the porphobilinogen deaminase gene. The father and mother, shown to be gene carriers of the same mutation, are asymptomatic and have normal urinary porphyrin precursor and metabolite excretion.

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Year:  2004        PMID: 14970743     DOI: 10.1023/B:BOLI.0000016613.75677.05

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


  12 in total

1.  Precision and accuracy of a HPLC method for measurement of fecal porphyrin concentrations.

Authors:  Floris M J Zuijderhoudt; Johannes S Kamphuis; Willem E Kluitenberg; Jenneke Dorresteijn-de Bok
Journal:  Clin Chem Lab Med       Date:  2002-10       Impact factor: 3.694

2.  [A CHILD WITH A SPECIAL FORM OF PORPHYRIA].

Authors:  V H DE VILLENEUVE; S K WADMAN; J M VAN DE HEUVEL
Journal:  Maandschr Kindergeneeskd       Date:  1964-12

3.  Homozygous acute intermittent porphyria: compound heterozygosity for adjacent base transitions in the same codon of the porphobilinogen deaminase gene.

Authors:  D H Llewellyn; S J Smyth; G H Elder; A C Hutchesson; J M Rattenbury; M F Smith
Journal:  Hum Genet       Date:  1992-04       Impact factor: 4.132

4.  Interdependence between degree of porphyrin excess and disease severity in congenital erythropoietic porphyria (Günther's disease).

Authors:  A G Freesemann; L K Bhutani; K Jacob; M O Doss
Journal:  Arch Dermatol Res       Date:  1997-04       Impact factor: 3.017

5.  On accuracy and precision of a HPLC method for measurement of urine porphyrin concentrations.

Authors:  F M Zuijderhoudt; S G Koehorst; W E Kluitenberg; J Dorresteijn-de Bok
Journal:  Clin Chem Lab Med       Date:  2000-03       Impact factor: 3.694

6.  Hydroxymethylbilane synthase: complete genomic sequence and amplifiable polymorphisms in the human gene.

Authors:  H W Yoo; C A Warner; C H Chen; R J Desnick
Journal:  Genomics       Date:  1993-01       Impact factor: 5.736

7.  A retrospective study of a patient with homozygous form of acute intermittent porphyria.

Authors:  G J Beukeveld; B G Wolthers; Y Nordmann; J C Deybach; B Grandchamp; S K Wadman
Journal:  J Inherit Metab Dis       Date:  1990       Impact factor: 4.982

Review 8.  Erythropoietic and hepatic porphyrias.

Authors:  U Gross; G F Hoffmann; M O Doss
Journal:  J Inherit Metab Dis       Date:  2000-11       Impact factor: 4.982

9.  Non-erythroid form of acute intermittent porphyria caused by promoter and frameshift mutations distant from the coding sequence of exon 1 of the HMBS gene.

Authors:  S D Whatley; A G Roberts; D H Llewellyn; C P Bennett; C Garrett; G H Elder
Journal:  Hum Genet       Date:  2000-09       Impact factor: 4.132

10.  Alternative transcription and splicing of the human porphobilinogen deaminase gene result either in tissue-specific or in housekeeping expression.

Authors:  S Chretien; A Dubart; D Beaupain; N Raich; B Grandchamp; J Rosa; M Goossens; P H Romeo
Journal:  Proc Natl Acad Sci U S A       Date:  1988-01       Impact factor: 11.205

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  9 in total

1.  Homozygous hydroxymethylbilane synthase knock-in mice provide pathogenic insights into the severe neurological impairments present in human homozygous dominant acute intermittent porphyria.

Authors:  Makiko Yasuda; Lin Gan; Brenden Chen; Chunli Yu; Jinglan Zhang; Miguel A Gama-Sosa; Daniela D Pollak; Stefanie Berger; John D Phillips; Winfried Edelmann; Robert J Desnick
Journal:  Hum Mol Genet       Date:  2019-06-01       Impact factor: 6.150

Review 2.  Acute Intermittent Porphyria in children: A case report and review of the literature.

Authors:  Manisha Balwani; Preeti Singh; Anju Seth; Ekta Malik Debnath; Hetanshi Naik; Dana Doheny; Brenden Chen; Makiko Yasuda; Robert J Desnick
Journal:  Mol Genet Metab       Date:  2016-10-15       Impact factor: 4.797

3.  Biochemical compared to molecular diagnosis in acute intermittent porphyria.

Authors:  U Grob; H Puy; K Jacob; J C Deybach; J Kremer; M O Doss
Journal:  J Inherit Metab Dis       Date:  2006-02       Impact factor: 4.982

Review 4.  The acute hepatic porphyrias.

Authors:  Bruce Wang
Journal:  Transl Gastroenterol Hepatol       Date:  2021-04-05

5.  Feline acute intermittent porphyria: a phenocopy masquerading as an erythropoietic porphyria due to dominant and recessive hydroxymethylbilane synthase mutations.

Authors:  Sonia Clavero; David F Bishop; Mark E Haskins; Urs Giger; Raili Kauppinen; Robert J Desnick
Journal:  Hum Mol Genet       Date:  2009-11-24       Impact factor: 6.150

Review 6.  Recent advances on porphyria genetics: Inheritance, penetrance & molecular heterogeneity, including new modifying/causative genes.

Authors:  Makiko Yasuda; Brenden Chen; Robert J Desnick
Journal:  Mol Genet Metab       Date:  2018-11-30       Impact factor: 4.797

7.  Acute intermittent porphyria in Argentina: an update.

Authors:  Gabriela Nora Cerbino; Esther Noemí Gerez; Laura Sabina Varela; Viviana Alicia Melito; Victoria Estela Parera; Alcira Batlle; María Victoria Rossetti
Journal:  Biomed Res Int       Date:  2015-05-17       Impact factor: 3.411

8.  Identification of Shell Colour Pigments in Marine Snails Clanculus pharaonius and C. margaritarius (Trochoidea; Gastropoda).

Authors:  S T Williams; S Ito; K Wakamatsu; T Goral; N P Edwards; R A Wogelius; T Henkel; L F C de Oliveira; L F Maia; S Strekopytov; T Jeffries; D I Speiser; J T Marsden
Journal:  PLoS One       Date:  2016-07-01       Impact factor: 3.240

9.  Porphyria: What Is It and Who Should Be Evaluated?

Authors:  Yonatan Edel; Rivka Mamet
Journal:  Rambam Maimonides Med J       Date:  2018-04-19
  9 in total

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