AI Article Synopsis

  • Primary adrenal insufficiency (PAI) can be an early indicator of X-linked adrenoleukodystrophy (X-ALD), a rare genetic disorder with serious health impacts, including the possibility of severe neurological decline in affected boys.
  • An 11-year-old patient with PAI, initially thought to be due to his type 1 diabetes, later showed signs of advanced cerebral ALD (cALD) after 11 months, making him ineligible for a potentially life-saving hematopoietic cell transplant (HCT).
  • Early detection of X-ALD is crucial, as PAI typically appears before other neurological symptoms; thus, boys with PAI should be tested for X-ALD regardless of

Article Abstract

Primary adrenal insufficiency (PAI) is often the first clinical sign of X-linked adrenoleukodystrophy (X-ALD), a rare genetic disorder that can present with various clinical phenotypes. A subset of boys with X-ALD develop cerebral ALD (cALD), characterized by progressive central demyelination, neurocognitive decline, and ultimately death. Timely intervention with hematopoietic cell transplant (HCT) can be a life-saving therapy by stopping progression of cerebral disease. We report the case of an 11-year-old boy with type 1 diabetes mellitus who presented with PAI, growth delay, and symptoms of attention deficit hyperactivity disorder. Given his history of T1DM, his PAI was presumed to be autoimmune and he was started on hydrocortisone and fludrocortisone. Eleven months later brain magnetic resonance imaging revealed white matter hyperintensity consistent with advanced cALD. The degree of disease progression at the time of diagnosis rendered the patient ineligible for transplant and he has continued to experience progressive neurologic decline. Initial symptoms of cALD are often subtle but should not be overlooked, as early identification of X-ALD is critical to allow early intervention with lifesaving HCT. PAI typically presents prior to the onset of neurologic symptoms. All boys who present with PAI should undergo workup for X-ALD with plasma very long chain fatty acid testing, even in the setting of underlying autoimmune disease.

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Source
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC9017996PMC
http://dx.doi.org/10.1210/jendso/bvac039DOI Listing

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