Literature DB >> 23250513

Treatment of acute decompensation of maple syrup urine disease in adult patients with a new parenteral amino-acid mixture.

A Servais1, J B Arnoux, C Lamy, A Hummel, N Vittoz, I Katerinis, V Bazzaoui, S Dubois, C Broissand, M C Husson, M P Berleur, D Rabier, C Ottolenghi, V Valayannopoulos, P de Lonlay.   

Abstract

BACKGROUND: Acute decompensation of maple syrup urine disease (MSUD) is usually treated by enteral feeding with an amino-acid mixture without leucine (Leu), valine or isoleucine. However, its administration is ineffective in cases of gastric intolerance and some adult patients refuse enteral feeding via a nasogastric tube. We developed a new parenteral amino-acid mixture for patients with MSUD.
METHODS: Seventeen decompensation episodes in four adult patients with MSUD treated with a parenteral amino-acid mixture (group P) were compared to 18 previous episodes in the same patients treated by enteral feeding (group E).
RESULTS: The mean Leu concentration at presentation was similar in the groups P and E (1196.9 μmol/L and 1212.2 μmol/L, respectively). The mean decrease in the Leu concentration during the first 3 days of hospitalisation was significantly higher in group P than group E (p = 0.0026); there were no side effects. The mean duration of hospitalisation was similar (4 vs. 4.5 days, p = NS). No patient in group P deteriorated whereas one patient in group E required dialysis.
CONCLUSION: This new parenteral amino-acid mixture is safe and allows efficient Leu concentration decrease during acute MSUD decompensation episodes in adults. Its use avoids the need for nasogastric tube insertion.

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Year:  2012        PMID: 23250513     DOI: 10.1007/s10545-012-9570-2

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


  6 in total

1.  Branched-chain amino acid-free parenteral nutrition in the treatment of acute metabolic decompensation in patients with maple syrup urine disease.

Authors:  G T Berry; R Heidenreich; P Kaplan; F Levine; A Mazur; M J Palmieri; M Yudkoff; S Segal
Journal:  N Engl J Med       Date:  1991-01-17       Impact factor: 91.245

2.  Kinetic modeling of plasma leucine levels during continuous venovenous extracorporeal removal therapy in neonates with maple syrup urine disease.

Authors:  Philippe Jouvet; Philippe Hubert; Jean Marie Saudubray; Daniel Rabier; Nguyen K Man
Journal:  Pediatr Res       Date:  2005-07-07       Impact factor: 3.756

3.  Kinetics of neutral amino acid transport across the blood-brain barrier.

Authors:  Q R Smith; S Momma; M Aoyagi; S I Rapoport
Journal:  J Neurochem       Date:  1987-11       Impact factor: 5.372

4.  Treatment of the acute crisis in maple syrup urine disease.

Authors:  W L Nyhan; M Rice-Kelts; J Klein; B A Barshop
Journal:  Arch Pediatr Adolesc Med       Date:  1998-06

5.  Classical maple syrup urine disease and brain development: principles of management and formula design.

Authors:  Kevin A Strauss; Bridget Wardley; Donna Robinson; Christine Hendrickson; Nicholas L Rider; Erik G Puffenberger; Diana Shellmer; Diana Shelmer; Ann B Moser; D Holmes Morton
Journal:  Mol Genet Metab       Date:  2010-01-12       Impact factor: 4.797

6.  Total parenteral nutrition therapy of toxic maple syrup urine disease.

Authors:  I Townsend; D S Kerr
Journal:  Am J Clin Nutr       Date:  1982-08       Impact factor: 7.045

  6 in total
  6 in total

1.  Intravenous administration of a branched-chain amino-acid-free solution in children and adults with acute decompensation of maple syrup urine disease: a prospective multicentre observational study.

Authors:  Jean-Meidi Alili; Marie-Pierre Berleur; Marie-Caroline Husson; Karine Mention; Manuel Schiff; Jean-Baptiste Arnoux; Anaïs Brassier; Anne-Sophie Guemman; Coraline Grisel; Sandrine Dubois; Marie-Thérèse Abi-Wardé; Christine Broissand; Aude Servais; Myriam Dao; Pascale de Lonlay
Journal:  Orphanet J Rare Dis       Date:  2022-05-16       Impact factor: 4.303

2.  Real-world management of maple syrup urine disease (MSUD) metabolic decompensations with branched chain amino acid-free formulas in France and Germany: A retrospective observational study.

Authors:  Pascale de Lonlay; Roland Posset; Ulrike Mütze; Karine Mention; Delphine Lamireau; Manuel Schiff; Aude Servais; Jean Baptiste Arnoux; Anaïs Brassier; Myriam Dao; Claire Douillard; Chris Ottolenghi; Clément Pontoizeau; Federica Miotto; Jeannie Le Mouhaër
Journal:  JIMD Rep       Date:  2021-03-06

Review 3.  Maple syrup urine disease: mechanisms and management.

Authors:  Patrick R Blackburn; Jennifer M Gass; Filippo Pinto E Vairo; Kristen M Farnham; Herjot K Atwal; Sarah Macklin; Eric W Klee; Paldeep S Atwal
Journal:  Appl Clin Genet       Date:  2017-09-06

4.  Enteral tube feeding in patients receiving dietary treatment for metabolic diseases: A retrospective analysis in a large French cohort.

Authors:  Claire-Marine Bérat; Célina Roda; Anais Brassier; Juliette Bouchereau; Camille Wicker; Aude Servais; Sandrine Dubois; Murielle Assoun; Claire Belloche; Valérie Barbier; Virginie Leboeuf; François M Petit; Pauline Gaignard; Elise Lebigot; Pierre-Jean Bérat; Clément Pontoizeau; Guy Touati; Cécile Talbotec; Florence Campeotto; Chris Ottolenghi; Jean-Baptiste Arnoux; Pascale de Lonlay Pascale
Journal:  Mol Genet Metab Rep       Date:  2021-01-05

5.  Maple syrup urine disease decompensation misdiagnosed as a psychotic event.

Authors:  Tomoyasu Higashimoto; Matthew T Whitehead; Erin MacLeod; Danielle Starin; Debra S Regier
Journal:  Mol Genet Metab Rep       Date:  2022-06-18

6.  Intravenous branched-chain amino-acid-free solution for the treatment of metabolic decompensation episodes in Spanish pediatric patients with maple syrup urine disease.

Authors:  Paula Sánchez-Pintos; Silvia Meavilla; María Goretti López-Ramos; Ángeles García-Cazorla; Maria L Couce
Journal:  Front Pediatr       Date:  2022-08-15       Impact factor: 3.569

  6 in total

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