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Reduction of microbleeds by immunosuppression in a patient with Aβ-related vascular inflammation.

Traschütz A, Tzaridis T, Penner AH, Kuchelmeister K, Urbach H, Hattingen E, Heneka MT - Neurol Neuroimmunol Neuroinflamm (2015)

Bottom Line: Under long-term immunosuppressive treatment, a reduced number of cortical micobleeds was observed on repeat MRIs because of both the prevention of new microbleeds and the clearance of those existing at baseline.This study provides Class IV evidence.This is a single observational study without controls.

View Article: PubMed Central - PubMed

Affiliation: Departments of Neurology (A.T., T.T., M.T.H.), Neuroradiology (A.-H.P., E.H.), and Neuropathology (K.K.), University of Bonn, Germany; Department of Neuroradiology (H.U.), University of Freiburg, Germany; and German Center for Neurodegenerative Disease (M.T.H.), Bonn, Germany.

ABSTRACT

Objective: To investigate whether the occurrence or clearance of microhemorrhages in cerebral amyloid angiopathy (CAA)-related vascular inflammation can be modified by immunosuppressive treatment.

Methods: Clinical and radiologic follow-up for more than 5 years of a patient with histopathologically confirmed CAA-related vascular inflammation treated with a prolonged and tapered regimen of IV cyclophosphamide and oral steroids.

Results: Under long-term immunosuppressive treatment, a reduced number of cortical micobleeds was observed on repeat MRIs because of both the prevention of new microbleeds and the clearance of those existing at baseline.

Conclusions: Sustained immunosuppression should be considered and systematically investigated as a treatment option for cortical microbleeds in CAA and related inflammatory phenotypes.

Classification of evidence: This study provides Class IV evidence. This is a single observational study without controls.

No MeSH data available.


Related in: MedlinePlus

Comparison of microbleeds before and after treatmentCoronal T2*-weighted image from May 2009 (left) shows multiple dotlike hypointense microbleeds mainly in the left temporal lobe. Although some lesions were still seen in 2014 (right, arrows), most of the microbleeds disappeared (arrowheads).
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Figure 3: Comparison of microbleeds before and after treatmentCoronal T2*-weighted image from May 2009 (left) shows multiple dotlike hypointense microbleeds mainly in the left temporal lobe. Although some lesions were still seen in 2014 (right, arrows), most of the microbleeds disappeared (arrowheads).

Mentions: After 2 treatments with cyclophosphamide 4 weeks apart, there was marked clinical improvement and the patient reported a regression of his tinnitus and partial improvement of the abovementioned hypesthesia. Delayed recall, which was shown to be mildly impaired in formal neuropsychological testing at baseline, was also restored. A follow-up MRI demonstrated near-complete resolution of T2/FLAIR hyperintensities (figure 1). Immunosuppressive treatment was continued, but oral prednisone was gradually tapered to a maintenance dose of 5 mg and the time intervals between cyclophosphamide pulses gradually increased to 3, 6, 9, and finally 12 months. In order to reduce the possible risk of secondary malignancies,1 pulse doses of cyclophosphamide were reduced to 375 mg/m2 in 2012 and to 200 mg/m2 in 2013 (table). During this treatment, the disease remained clinically and radiologically stable. Of note, there were no further microbleeds on repeat MRIs until July 2014, and the number of existing microbleeds was substantially reduced (figure 3).


Reduction of microbleeds by immunosuppression in a patient with Aβ-related vascular inflammation.

Traschütz A, Tzaridis T, Penner AH, Kuchelmeister K, Urbach H, Hattingen E, Heneka MT - Neurol Neuroimmunol Neuroinflamm (2015)

Comparison of microbleeds before and after treatmentCoronal T2*-weighted image from May 2009 (left) shows multiple dotlike hypointense microbleeds mainly in the left temporal lobe. Although some lesions were still seen in 2014 (right, arrows), most of the microbleeds disappeared (arrowheads).
© Copyright Policy - open-access
Related In: Results  -  Collection

License
Show All Figures
getmorefigures.php?uid=PMC4608757&req=5

Figure 3: Comparison of microbleeds before and after treatmentCoronal T2*-weighted image from May 2009 (left) shows multiple dotlike hypointense microbleeds mainly in the left temporal lobe. Although some lesions were still seen in 2014 (right, arrows), most of the microbleeds disappeared (arrowheads).
Mentions: After 2 treatments with cyclophosphamide 4 weeks apart, there was marked clinical improvement and the patient reported a regression of his tinnitus and partial improvement of the abovementioned hypesthesia. Delayed recall, which was shown to be mildly impaired in formal neuropsychological testing at baseline, was also restored. A follow-up MRI demonstrated near-complete resolution of T2/FLAIR hyperintensities (figure 1). Immunosuppressive treatment was continued, but oral prednisone was gradually tapered to a maintenance dose of 5 mg and the time intervals between cyclophosphamide pulses gradually increased to 3, 6, 9, and finally 12 months. In order to reduce the possible risk of secondary malignancies,1 pulse doses of cyclophosphamide were reduced to 375 mg/m2 in 2012 and to 200 mg/m2 in 2013 (table). During this treatment, the disease remained clinically and radiologically stable. Of note, there were no further microbleeds on repeat MRIs until July 2014, and the number of existing microbleeds was substantially reduced (figure 3).

Bottom Line: Under long-term immunosuppressive treatment, a reduced number of cortical micobleeds was observed on repeat MRIs because of both the prevention of new microbleeds and the clearance of those existing at baseline.This study provides Class IV evidence.This is a single observational study without controls.

View Article: PubMed Central - PubMed

Affiliation: Departments of Neurology (A.T., T.T., M.T.H.), Neuroradiology (A.-H.P., E.H.), and Neuropathology (K.K.), University of Bonn, Germany; Department of Neuroradiology (H.U.), University of Freiburg, Germany; and German Center for Neurodegenerative Disease (M.T.H.), Bonn, Germany.

ABSTRACT

Objective: To investigate whether the occurrence or clearance of microhemorrhages in cerebral amyloid angiopathy (CAA)-related vascular inflammation can be modified by immunosuppressive treatment.

Methods: Clinical and radiologic follow-up for more than 5 years of a patient with histopathologically confirmed CAA-related vascular inflammation treated with a prolonged and tapered regimen of IV cyclophosphamide and oral steroids.

Results: Under long-term immunosuppressive treatment, a reduced number of cortical micobleeds was observed on repeat MRIs because of both the prevention of new microbleeds and the clearance of those existing at baseline.

Conclusions: Sustained immunosuppression should be considered and systematically investigated as a treatment option for cortical microbleeds in CAA and related inflammatory phenotypes.

Classification of evidence: This study provides Class IV evidence. This is a single observational study without controls.

No MeSH data available.


Related in: MedlinePlus