Literature DB >> 19629744

Long-term needs of adult patients with organic acidaemias: outcome and prognostic factors.

E Martín-Hernández1, P J Lee, A Micciche, S Grunewald, R H Lachmann.   

Abstract

BACKGROUND: With improvements in the treatment of children with organic acidaemias (OA), the number surviving to adulthood is increasing. To plan appropriate services for their care it is important to know what their needs are.
OBJECTIVE: To describe the clinical and social problems affecting adult patients with OA. PATIENTS AND METHODS: We reviewed the medical records of 15 adult patients diagnosed with OA. Social attainment (housing, schooling and occupation) was analysed. Nutritional status was evaluated by body mass index (BMI) and laboratory studies. Neurological and visceral complications were noted. Cognitive outcome was evaluated by psychometric testing and/or educational attainment.
RESULTS: Seven had methylmalonic acidaemia (MMA), 4 isovaleric acidaemia (IVA) and 4 propionic acidaemia (PA). Ten were female, and median age was 23.5 years (range 18-48). All but three had late-onset disease. Two patients became pregnant during follow up. Four patients had obtained university degrees and were working. Three-quarters of the patients required some kind of social support. All had a good nutritional status. Height was normal in IVA and 3 PA patients. Osteoporosis was present in 2 out of 8 patients assessed. A variety of neurocognitive or visceral complications were seen in two-thirds of the patients. Metabolic decompensations were unusual.
CONCLUSIONS: The approach to adult patients with OA has to be multidisciplinary, with the clinician and dietician as the core of the team, but with the collaboration of clinical nurses specialists, social workers and other specialist services and the support of a biochemical and molecular laboratory.

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Year:  2009        PMID: 19629744     DOI: 10.1007/s10545-009-1191-12

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


  37 in total

1.  Potential relationship between genotype and clinical outcome in propionic acidaemia patients.

Authors:  C Pérez-Cerdá; B Merinero; P Rodríguez-Pombo; B Pérez; L R Desviat; S Muro; E Richard; M J García; J Gangoiti; P Ruiz Sala; P Sanz; P Briones; A Ribes; M Martínez-Pardo; J Campistol; M Pérez; R Lama; M L Murga; T Lema-Garrett; A Verdú; M Ugarte
Journal:  Eur J Hum Genet       Date:  2000-03       Impact factor: 4.246

2.  Propionic acidemia revisited: a workshop report.

Authors:  J O Sass; M Hofmann; D Skladal; E Mayatepek; B Schwahn; W Sperl
Journal:  Clin Pediatr (Phila)       Date:  2004 Nov-Dec       Impact factor: 1.168

3.  Propionic acidaemia with basal ganglia stroke: treatment of acute extrapyramidal symptoms with L-DOPA.

Authors:  A P Burlina; C Baracchini; C Carollo; A B Burlina
Journal:  J Inherit Metab Dis       Date:  2001-10       Impact factor: 4.982

4.  Long-term follow-up of 77 patients with isolated methylmalonic acidaemia.

Authors:  E R Baumgarter; C Viardot
Journal:  J Inherit Metab Dis       Date:  1995       Impact factor: 4.982

5.  Propionic acidaemia: clinical, biochemical and therapeutic aspects. Experience in 30 patients.

Authors:  W Lehnert; W Sperl; T Suormala; E R Baumgartner
Journal:  Eur J Pediatr       Date:  1994       Impact factor: 3.183

Review 6.  The adolescent with an inborn error of metabolism: medical issues and transition to adulthood.

Authors:  Gregory M Enns; Wendy Packman
Journal:  Adolesc Med       Date:  2002-06

7.  Neurologic nonmetabolic presentation of propionic acidemia.

Authors:  W L Nyhan; C Bay; E W Beyer; M Mazi
Journal:  Arch Neurol       Date:  1999-09

8.  Liver transplantation for methylmalonic acidaemia.

Authors:  W van't Hoff; P J McKiernan; R A Surtees; J V Leonard
Journal:  Eur J Pediatr       Date:  1999-12       Impact factor: 3.183

9.  Clinical outcome of long-term management of patients with vitamin B12-unresponsive methylmalonic acidemia.

Authors:  S B van der Meer; F Poggi; M Spada; J P Bonnefont; H Ogier; P Hubert; E Depondt; D Rapoport; D Rabier; C Charpentier
Journal:  J Pediatr       Date:  1994-12       Impact factor: 4.406

10.  The impact of screening for propionic and methylmalonic acidaemia.

Authors:  James V Leonard; Suresh Vijayaraghavan; John H Walter
Journal:  Eur J Pediatr       Date:  2003-10-30       Impact factor: 3.183

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1.  Nutritional and Pharmacological Management during Chemotherapy in a Patient with Propionic Acidaemia and Rhabdomyosarcoma Botryoides.

Authors:  E Martín-Hernández; P Quijada-Fraile; L Oliveros-Leal; Mt García-Silva; C Pérez-Cerdá; M Baro-Fernández; V Pérez-Alonso; Jl Vivanco
Journal:  JIMD Rep       Date:  2012-03-21

2.  Biochemical Monitoring and Management During Pregnancy in Patients with Isovaleric Acidaemia is Helpful to Prevent Metabolic Decompensation.

Authors:  D D J Habets; N C Schaper; H Rogozinski; F J van Spronsen; M van Rijn; J Bierau; J A Bakker
Journal:  JIMD Rep       Date:  2011-09-27

Review 3.  Organic acidurias in adults: late complications and management.

Authors:  Ali Tunç Tuncel; Nikolas Boy; Marina A Morath; Friederike Hörster; Ulrike Mütze; Stefan Kölker
Journal:  J Inherit Metab Dis       Date:  2018-01-15       Impact factor: 4.982

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Authors:  Jonathan D Picker; Christopher A Walsh
Journal:  Ann Neurol       Date:  2013-09       Impact factor: 10.422

5.  Long-term renal outcome in methylmalonic acidemia in adolescents and adults.

Authors:  Myriam Dao; Jean-Baptiste Arnoux; Frank Bienaimé; Anaïs Brassier; François Brazier; Jean-François Benoist; Clément Pontoizeau; Chris Ottolenghi; Pauline Krug; Olivia Boyer; Pascale de Lonlay; Aude Servais
Journal:  Orphanet J Rare Dis       Date:  2021-05-13       Impact factor: 4.123

6.  Guidelines for the diagnosis and management of methylmalonic acidaemia and propionic acidaemia: First revision.

Authors:  Patrick Forny; Friederike Hörster; Diana Ballhausen; Anupam Chakrapani; Kimberly A Chapman; Carlo Dionisi-Vici; Marjorie Dixon; Sarah C Grünert; Stephanie Grunewald; Goknur Haliloglu; Michel Hochuli; Tomas Honzik; Daniela Karall; Diego Martinelli; Femke Molema; Jörn Oliver Sass; Sabine Scholl-Bürgi; Galit Tal; Monique Williams; Martina Huemer; Matthias R Baumgartner
Journal:  J Inherit Metab Dis       Date:  2021-03-09       Impact factor: 4.750

7.  Delineating the spectrum of impairments, disabilities, and rehabilitation needs in methylmalonic acidemia (MMA).

Authors:  Yiouli P Ktena; Scott M Paul; Natalie S Hauser; Jennifer L Sloan; Andrea Gropman; Irini Manoli; Charles P Venditti
Journal:  Am J Med Genet A       Date:  2015-05-10       Impact factor: 2.578

8.  Clinical and neurocognitive outcome in symptomatic isovaleric acidemia.

Authors:  Sarah C Grünert; Udo Wendel; Martin Lindner; Michael Leichsenring; K Otfried Schwab; Jerry Vockley; Willy Lehnert; Regina Ensenauer
Journal:  Orphanet J Rare Dis       Date:  2012-01-25       Impact factor: 4.123

Review 9.  Proposed guidelines for the diagnosis and management of methylmalonic and propionic acidemia.

Authors:  Matthias R Baumgartner; Friederike Hörster; Carlo Dionisi-Vici; Goknur Haliloglu; Daniela Karall; Kimberly A Chapman; Martina Huemer; Michel Hochuli; Murielle Assoun; Diana Ballhausen; Alberto Burlina; Brian Fowler; Sarah C Grünert; Stephanie Grünewald; Tomas Honzik; Begoña Merinero; Celia Pérez-Cerdá; Sabine Scholl-Bürgi; Flemming Skovby; Frits Wijburg; Anita MacDonald; Diego Martinelli; Jörn Oliver Sass; Vassili Valayannopoulos; Anupam Chakrapani
Journal:  Orphanet J Rare Dis       Date:  2014-09-02       Impact factor: 4.123

10.  Dietary practices in propionic acidemia: A European survey.

Authors:  A Daly; A Pinto; S Evans; M F Almeida; M Assoun; A Belanger-Quintana; S M Bernabei; S Bollhalder; D Cassiman; H Champion; H Chan; J Dalmau; F de Boer; C de Laet; A de Meyer; A Desloovere; A Dianin; M Dixon; K Dokoupil; S Dubois; F Eyskens; A Faria; I Fasan; E Favre; F Feillet; A Fekete; G Gallo; C Gingell; J Gribben; K Kaalund Hansen; N M Ter Horst; C Jankowski; R Janssen-Regelink; I Jones; C Jouault; G E Kahrs; I L Kok; A Kowalik; C Laguerre; S Le Verge; R Lilje; C Maddalon; D Mayr; U Meyer; A Micciche; U Och; M Robert; J C Rocha; H Rogozinski; C Rohde; K Ross; I Saruggia; A Schlune; K Singleton; E Sjoqvist; R Skeath; L H Stolen; A Terry; C Timmer; L Tomlinson; A Tooke; K Vande Kerckhove; E van Dam; T van den Hurk; L van der Ploeg; M van Driessche; M van Rijn; A van Wegberg; C Vasconcelos; H Vestergaard; I Vitoria; D Webster; F J White; L White; H Zweers; A MacDonald
Journal:  Mol Genet Metab Rep       Date:  2017-10-03
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