J. Inher. Metab. Dis. t4 (1991) 400-402
© SSIEM and KluwerAcademicPublishers. Printed in the Netherlands
Short Communication
Seven-Year Experience of a Reference
Laboratory for Detection of Inborn Errors of
Metabolism in Brazil
R. GIUGLIANI, J. C. DUTRA, M. L. BARTH, C. S. DUTRA-FILHO,
S. L. GOLDENFUM and M. WAJNER
Medical Genetic Unit, Clinical Hospital of Porto Alegre, Rua Ramiro Barcelos 2350,
Porto Alegre RS 90210, Brazil
Inborn errors of metabolism (IEM) are a heterogeneous group of more than 300
genetic defects related to the synthesis, metabolism, transport and storage of
biochemical compounds. The large number of specific biochemical techniques required
for their detection and diagnosis and the rarity of each disorder makes advisable the
creation of regional reference diagnostic laboratories. We now present the results of
our investigations performed in high risk patients at the Regional Laboratory for
Inborn Errors of Metabolism, Porto Alegre, Brazil, during a 7-year period.
MATERIALS AND METHODS
We studied 3749 patients referred to our laboratory from 1982 to 1989. The main
clinical indications for referral were: neuromotor delay/mental retardation (1088),
seizures (489) and hepatomegaly and/or splenomegaly (196). All patients were initially
submitted to screening tests consisting of qualitative tests on urine (Benedict's, ferric
chloride, dinitrophenylhydrazine, nitrosonaphthol, cyanide-nitroprusside, toluidine
blue, acetyl-trimethylammonium chloride, p-nitroaniline, and Ehrlich tests) and paper
amino acid chromatography of blood and urine. When at least one test showed a
positive or doubtful result, the patient was recalled and the investigation continued
through more specific methods, consisting of chromatographic analysis of urine
(glycosaminoglycans, oligosaccharides, sialyloligosaccharides and carbohydrates) and
quantitative and/or qualitative assays of urine and/or blood metabolites (oxalic acid,
sialic acid, glycosaminoglycans, porphobilinogen, amino acids and organic acids). If
there was a suspicion of an IEM undetectable by the screening tests based on clinical,
radiological and/or laboratory notes, the patient was then submitted to a focussed
investigation through specific diagnostic methods.
RESULTS
Investigations have been completed on 3161 patients so far, and a metabolic defect
was identified in 251 cases (7.9%) (Table 1). The most frequently diagnosed
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