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Intensity of MRIGadoliniumEnhancement inCerebralAdrenoleukodystrophy:A Biomarker forInflammation andPredictor of OutcomefollowingTransplantation in… Weston P. MillerLuiz Fernando Alves LimaMantovaniJohn G. Muzic2015 год

Intensity of MRI Gadolinium Enhancement in Cerebral Adrenoleukodystrophy: A Biomarker for Inflammation and Predictor of Outcome following Transplantation in Higher Risk Patients
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Аннотация: Outcomes following hematopoietic stem cell transplantation for higher risk childhood-onset cerebral adrenoleukodystrophy are variable. We explored whether a brain MR imaging gadolinium intensity scoring system improves prediction of neurologic outcome.We developed a 4-point scale of gadolinium intensity relative to the choroid plexus: 0 = no enhancement; 1 = hypointense; 2 = isointense; 3 = hyperintense. The interobserver concordance of the scale was assessed on 30 randomly chosen studies. Scores were generated for 64 evaluable patients and compared with CSF chitotriosidase levels, a known inflammatory marker correlating with outcomes following transplantation. For 25 evaluable higher risk patients (Loes ≥10), the gadolinium intensity score was compared with longer term posttransplantation clinical change.The gadolinium intensity scoring system showed good interobserver reproducibility (κ = 0.72). Of 64 evaluable boys, the score positively correlated with average concomitant CSF chitotriosidase activity in nanograms/milliliter/hour: 0: 2717, n = 5; 1: 3218, n = 13; 2: 6497, n = 23; and 3: 12,030, n = 23 (P < .01). For 25 evaluable higher risk patients, more intense pretransplantation brain MR imaging gadolinium enhancement predicted greater average loss on the adrenoleukodystrophy neurologic function scale following transplantation: 0/1: adrenoleukodystrophy neurologic function scale score difference = 4.3, n = 7; 2/3: adrenoleukodystrophy neurologic function scale score difference = 10.4, n = 18 (P = .05).Gadolinium enhancement intensity on brain MR imaging can be scored simply and reproducibly for cerebral adrenoleukodystrophy. The enhancement score significantly correlates with chitotriosidase. In boys with higher risk cerebral disease (Loes ≥10), the enhancement score itself predicts neurologic outcome following treatment. Such data may help guide treatment decisions for clinicians and families.
Год издания: 2015
Авторы: Weston P. Miller, Luiz Fernando Alves Lima Mantovani, John G. Muzic, Jeffrey Rykken, Rakhee Gawande, Troy C. Lund, Ryan Shanley, Gerald V. Raymond, Paul J. Orchard, David Nascene
Издательство: American Society of Neuroradiology
Источник: American Journal of Neuroradiology
Ключевые слова: Peroxisome Proliferator-Activated Receptors, Adenosine and Purinergic Signaling, Infectious Encephalopathies and Encephalitis
Другие ссылки: American Journal of Neuroradiology (PDF)
American Journal of Neuroradiology (HTML)
PubMed Central (HTML)
Europe PMC (PubMed Central) (PDF)
Europe PMC (PubMed Central) (HTML)
PubMed (HTML)