Literature DB >> 8344008

Delay in diagnosis of X-linked adrenoleukodystrophy.

B M van Geel1, J Assies, E B Haverkort, P G Barth, R J Wanders, R B Schutgens, A Keyser, C P Zwetsloot.   

Abstract

In 16 consecutive patients with clinically suspected and biochemically proven X-linked adrenoleukodystrophy (X-ALD), total delay (interval between onset of symptoms and diagnosis) and specialist delay (interval between referral to a specialist and diagnosis) were determined. All patients previously were unaware of the existence of X-ALD in their families. From the time of onset of symptoms attributable to this disease until diagnosis, mean total delay was 9.9 (range 1-33) years and mean specialist delay was 8.4 (range 0-33) years. Three patients who presented with adrenocortical insufficiency had mean total and specialist delays of 17.3 (range 9-33) years. Five patients with an initial diagnosis of multiple sclerosis had mean total and specialist delays of 12.8 (range 5-25) and 11.2 (range 1-23) years, respectively. In 12 patients with adrenomyeloneuropathy--the second most frequent phenotype of X-ALD--mean total delay was 11.0 (range 2-33) years and mean specialist delay 9.1 (range 0-33) years. Since 1981 X-ALD can be reliably diagnosed on the basis of elevated levels of very long chain fatty acids in plasma and/or cultured fibroblasts. The delays therefore must have been due to the unfamiliarity with the clinical manifestations and diagnostic possibilities of this disease. Once X-ALD is diagnosed, dietary treatment and/or bone marrow transplantation may be considered. Genetic counseling should be performed, and screening of other family members is essential for the early identification of affected relatives.

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Year:  1993        PMID: 8344008     DOI: 10.1016/0303-8467(93)90004-z

Source DB:  PubMed          Journal:  Clin Neurol Neurosurg        ISSN: 0303-8467            Impact factor:   1.876


  4 in total

Review 1.  X linked adrenoleukodystrophy: clinical presentation, diagnosis, and therapy.

Authors:  B M van Geel; J Assies; R J Wanders; P G Barth
Journal:  J Neurol Neurosurg Psychiatry       Date:  1997-07       Impact factor: 10.154

Review 2.  The Landscape of Hematopoietic Stem Cell Transplant and Gene Therapy for X-Linked Adrenoleukodystrophy.

Authors:  Eric J Mallack; Bela Turk; Helena Yan; Florian S Eichler
Journal:  Curr Treat Options Neurol       Date:  2019-11-25       Impact factor: 3.598

3.  MRI surveillance of boys with X-linked adrenoleukodystrophy identified by newborn screening: Meta-analysis and consensus guidelines.

Authors:  Eric J Mallack; Bela R Turk; Helena Yan; Carrie Price; Michelle Demetres; Ann B Moser; Catherine Becker; Kim Hollandsworth; Laura Adang; Adeline Vanderver; Keith Van Haren; Maura Ruzhnikov; Joanne Kurtzberg; Gustavo Maegawa; Paul J Orchard; Troy C Lund; Gerald V Raymond; Molly Regelmann; Joseph J Orsini; Elisa Seeger; Stephan Kemp; Florian Eichler; Ali Fatemi
Journal:  J Inherit Metab Dis       Date:  2021-01-09       Impact factor: 4.982

4.  Newborn Screening for X-Linked Adrenoleukodystrophy: Review of Data and Outcomes in Pennsylvania.

Authors:  Jessica R C Priestley; Laura A Adang; Sarah Drewes Williams; Uta Lichter-Konecki; Caitlin Menello; Nicole M Engelhardt; James C DiPerna; Brenda DiBoscio; Rebecca C Ahrens-Nicklas; Andrew C Edmondson; Francis Jeshira Reynoso Santos; Can Ficicioglu
Journal:  Int J Neonatal Screen       Date:  2022-03-23
  4 in total

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