Contributions
Abstract: EP1587
Type: Poster Sessions
Abstract Category: Therapy - Immunomodulation/Immunosuppression
Background: tumefactive demyelinating lesions may be a diagnostic and therapeutic challenge [1], especially in pediatric patients [2]. Alemtuzumab showed efficacy in a fulminant form of multiple sclerosis (MS) [3].
Method: case report of an adolescent with tumefactive relapsing MS who underwent a sustained remission after alemtuzumab therapy.
Results: in February 2015, a 16 years old male, without remarkable previous clinical history, had a subacute sensorimotor right hemiparesis associated with headache and vomiting. Brain MRI showed five lesions (including corpus callosum) with ring-shaped enhancement, one with a tumor-like appearance with pronounced perilesional oedema. Spinal cord MRI, CSF, evoked potentials, autoantibody screening (including anti-NMO and anti-MOG) were normal. He was treated with an intravenous (iv) methylprednisolone course (ineffective), then with two courses of plasma exchange (partial recovery), finally with two Rituximab courses 15 days apart, with slow clinical improvement. In April 2015 brain MRI showed partial regression of one lesion, but worsening of the pseudotumoral one, associated with a further deterioration of symptoms. In May 2015 a second CSF analysis was negative, as well as the work up for lymphoproliferative diseases (total body CT, testicles ultrasound, eye fundus examination). In May 2015 cyclophosphamide was started (9 courses monthly), without a significant remission of disease activity. In July 2015 brain MRI showed inflammatory activity in almost all lesions with increased left fronto-temporal perilesional oedema. The patient presented marked dysarthria that was treated with iv methylprednisolone, with only partial recovery. In autumn 2015, problems of memory and attention arose. In February 2016 brain MRI showed a further reduction of posterior lesions, but new, large pseudotumoral bi-frontal contrast-enhancing lesions appeared. Brain biopsy confirmed the diagnosis of inflammatory demyelinating disease compatible with MS, excluding other mimickers (neoplasm, lymphoma, vasculitis, ADEM or infection). Alemtuzumab was then started in March 2016, with a sustained clinical improvement and complete remission of any activity at subsequent brain MRI. No adverse events were observed.
Conclusions: alemtuzumab led to a long-term remission in a young patient with tumefactive relapsing MS, confirmed by neuropathological examination. Alemtuzumab should be considered early in the therapeutic algorithm of these forms.
Disclosure: Dr. D. Baroncini received travel grants from Genzyme, Merck and Biogen for participation at national and international congresses; he received speaking honoraria from Sanofi and Novartis, and personal compensation from Almirall for scientific publication.
Dr. P. Annovazzi received honoraria for lecturing and participation in advisory boards, and/or travel expenses for attending congresses and meetings from Merck, Biogen, Teva, Sanofi-Genzyme, Mylan, Almirall, Roche and Novartis.
Dr. C. Guaschino received travel grants from Sanofi, Genzyme and Teva for participation at national and international congresses.
Dr. L. Saraceno has no disclosures.
Dr. G. Minonzio has no disclosures.
Dr. A. Ghezzi received fees for consultant and speaking activities from Merck, Novartis, Biogen, Genzyme, Teva, Roche, Almirall, Mylan.
Prof. Comi has received compensation for consulting services with the following companies: Novartis, Teva, Sanofi, Genzyme, Merck, Biogen, Excemed, Roche, Almirall, Chugai, Receptos, Forward Pharma and compensation for speaking activities from Novartis, Teva, Sanofi, Genzyme, Merk, Biogen, Excemed, and Roche.
Dr. C. Stadelmann has received speaker´s honoraria from Novartis-Pharma and Bayer-Schering AG.
Dr. M. Zaffaroni has received honoraria for lecturing or participation in advisory boards, and financial support for attending congresses from Almirall, Biogen Idec, Genzyme, Merck Serono, Novartis, and Teva
Abstract: EP1587
Type: Poster Sessions
Abstract Category: Therapy - Immunomodulation/Immunosuppression
Background: tumefactive demyelinating lesions may be a diagnostic and therapeutic challenge [1], especially in pediatric patients [2]. Alemtuzumab showed efficacy in a fulminant form of multiple sclerosis (MS) [3].
Method: case report of an adolescent with tumefactive relapsing MS who underwent a sustained remission after alemtuzumab therapy.
Results: in February 2015, a 16 years old male, without remarkable previous clinical history, had a subacute sensorimotor right hemiparesis associated with headache and vomiting. Brain MRI showed five lesions (including corpus callosum) with ring-shaped enhancement, one with a tumor-like appearance with pronounced perilesional oedema. Spinal cord MRI, CSF, evoked potentials, autoantibody screening (including anti-NMO and anti-MOG) were normal. He was treated with an intravenous (iv) methylprednisolone course (ineffective), then with two courses of plasma exchange (partial recovery), finally with two Rituximab courses 15 days apart, with slow clinical improvement. In April 2015 brain MRI showed partial regression of one lesion, but worsening of the pseudotumoral one, associated with a further deterioration of symptoms. In May 2015 a second CSF analysis was negative, as well as the work up for lymphoproliferative diseases (total body CT, testicles ultrasound, eye fundus examination). In May 2015 cyclophosphamide was started (9 courses monthly), without a significant remission of disease activity. In July 2015 brain MRI showed inflammatory activity in almost all lesions with increased left fronto-temporal perilesional oedema. The patient presented marked dysarthria that was treated with iv methylprednisolone, with only partial recovery. In autumn 2015, problems of memory and attention arose. In February 2016 brain MRI showed a further reduction of posterior lesions, but new, large pseudotumoral bi-frontal contrast-enhancing lesions appeared. Brain biopsy confirmed the diagnosis of inflammatory demyelinating disease compatible with MS, excluding other mimickers (neoplasm, lymphoma, vasculitis, ADEM or infection). Alemtuzumab was then started in March 2016, with a sustained clinical improvement and complete remission of any activity at subsequent brain MRI. No adverse events were observed.
Conclusions: alemtuzumab led to a long-term remission in a young patient with tumefactive relapsing MS, confirmed by neuropathological examination. Alemtuzumab should be considered early in the therapeutic algorithm of these forms.
Disclosure: Dr. D. Baroncini received travel grants from Genzyme, Merck and Biogen for participation at national and international congresses; he received speaking honoraria from Sanofi and Novartis, and personal compensation from Almirall for scientific publication.
Dr. P. Annovazzi received honoraria for lecturing and participation in advisory boards, and/or travel expenses for attending congresses and meetings from Merck, Biogen, Teva, Sanofi-Genzyme, Mylan, Almirall, Roche and Novartis.
Dr. C. Guaschino received travel grants from Sanofi, Genzyme and Teva for participation at national and international congresses.
Dr. L. Saraceno has no disclosures.
Dr. G. Minonzio has no disclosures.
Dr. A. Ghezzi received fees for consultant and speaking activities from Merck, Novartis, Biogen, Genzyme, Teva, Roche, Almirall, Mylan.
Prof. Comi has received compensation for consulting services with the following companies: Novartis, Teva, Sanofi, Genzyme, Merck, Biogen, Excemed, Roche, Almirall, Chugai, Receptos, Forward Pharma and compensation for speaking activities from Novartis, Teva, Sanofi, Genzyme, Merk, Biogen, Excemed, and Roche.
Dr. C. Stadelmann has received speaker´s honoraria from Novartis-Pharma and Bayer-Schering AG.
Dr. M. Zaffaroni has received honoraria for lecturing or participation in advisory boards, and financial support for attending congresses from Almirall, Biogen Idec, Genzyme, Merck Serono, Novartis, and Teva