Hematopoietic cell transplantation does not prevent myelopathy in X-linked adrenoleukodystrophy: a retrospective study

J Inherit Metab Dis. 2015 Mar;38(2):359-61. doi: 10.1007/s10545-014-9797-1. Epub 2014 Dec 9.

Abstract

Background: X-linked adrenoleukodystrophy (X-ALD) is a peroxisomal metabolic disorder. Male patients develop adrenocortical insufficiency (80 % before 18 years), a chronic myelopathy (adrenomyeloneuropathy (AMN); all in adulthood), or progressive cerebral demyelination (cerebral ALD; 40 % before 18 years). Cerebral ALD is treated with haematopoetic cell transplantation (HCT). It is unknown if AMN still develops in patients with X-ALD that underwent HCT for cerebral ALD in childhood.

Patients and methods: A retrospective observational study was performed by selecting all adult patients with X-ALD in our cohort that underwent HCT in childhood.

Results: This retrospective study found that three out of five patients in our cohort who underwent HCT in childhood developed signs of myelopathy in adulthood.

Conclusion: These data suggest that HCT for cerebral ALD in childhood does not prevent the onset of AMN in X-ALD in adulthood.

Publication types

  • Observational Study

MeSH terms

  • Adolescent
  • Adrenoleukodystrophy / complications
  • Adrenoleukodystrophy / diagnosis
  • Adrenoleukodystrophy / surgery*
  • Adult
  • Age Factors
  • Child
  • Child, Preschool
  • Disease Progression
  • Hematopoietic Stem Cell Transplantation*
  • Humans
  • Male
  • Retrospective Studies
  • Spinal Cord Diseases / diagnosis
  • Spinal Cord Diseases / etiology*
  • Time Factors
  • Treatment Outcome
  • Young Adult