Clinical efficacy and safety of intrathecal methotrexate in the treatment of Balo’s concentric sclerosis: a case report
Deniz YUKSEL, Hale Celik, Ozge Kucur, Kader Oguz
Introduction: Balo’s concentric sclerosis (BCS) is a rare, progressive variant of multiple sclerosis characterized by concentric rings of demyelinated white matter and relatively preserved myelin alternating with each other. Intense immunosuppressive treatment may be indicated in patients with aggressive disease. Method: Childhood onset BCS which benefited from intrathecal (IT) methotrexate (MTX) is presented. Results: Case: A previously healthy 5 year-old boy was admitted with weakness of left arm/leg and aphasia. All laboratory tests were normal. Cerebrospinal fluid (CSF) oligoclonal bands were type 2 negative. Magnetic Resonance Imaging (MRI) showed multiple tumefactive and multi-layered demyelinating lesions (Figure1). Intravenous (IV) immunoglobulin, IV+oral steroids, plasma exchange were given sequentially. The left hemiplegia recovered partially, but right hemiplegia developed on the 7 days later. Immunosuppressive therapy with azathioprine and rituximab were given, but the disease progressed radiologically (Figure 2). IT MTX was started with 12,5 mg via lumbar puncture and repeated every two months. No side effects were observed in the patient. After 1 year of ITMTX treatment, his left hemiplegia had fully and right hemiplegia partially recovered, and he started to speak few words. Radiological remission was also observed (Figure 3). Treatment plan is to continue with azathioprine for approximately 1 more year. Conclusion: The safety and the tolerability of long-term therapy with ITMTX in patients with treatment-resistant, progressive forms of MS were reported previously. There was evidence of disease stabilization in our patient and long-term ITMTX appears as a safe therapeutic option in advanced BCS.
Keywords: Balo’s concentric sclerosis, intrathecal methotrexate, multiple sclerosis, aggressive disease, childhood
Deniz YUKSEL
University of Health Sciences Turkey, Dr Sami Ulus Maternity and Child Health and Diseases Research and Training Hospital
Turkey
Hale Celik
University of Health Sciences Turkey, Dr Sami Ulus Maternity and Child Health and Diseases Research and Training Hospital
Turkey
Ozge Kucur
Tekirdağ İsmail Fehmi Cumalıoğlu City Hospital
Turkey
Kader Oguz
Hacettepe University Faculty of Medicine
Turkey
Introduction: Balo’s concentric sclerosis (BCS) is a rare, progressive variant of multiple sclerosis characterized by concentric rings of demyelinated white matter and relatively preserved myelin alternating with each other. Intense immunosuppressive treatment may be indicated in patients with aggressive disease. Method: Childhood onset BCS which benefited from intrathecal (IT) methotrexate (MTX) is presented. Results: Case: A previously healthy 5 year-old boy was admitted with weakness of left arm/leg and aphasia. All laboratory tests were normal. Cerebrospinal fluid (CSF) oligoclonal bands were type 2 negative. Magnetic Resonance Imaging (MRI) showed multiple tumefactive and multi-layered demyelinating lesions (Figure1). Intravenous (IV) immunoglobulin, IV+oral steroids, plasma exchange were given sequentially. The left hemiplegia recovered partially, but right hemiplegia developed on the 7 days later. Immunosuppressive therapy with azathioprine and rituximab were given, but the disease progressed radiologically (Figure 2). IT MTX was started with 12,5 mg via lumbar puncture and repeated every two months. No side effects were observed in the patient. After 1 year of ITMTX treatment, his left hemiplegia had fully and right hemiplegia partially recovered, and he started to speak few words. Radiological remission was also observed (Figure 3). Treatment plan is to continue with azathioprine for approximately 1 more year. Conclusion: The safety and the tolerability of long-term therapy with ITMTX in patients with treatment-resistant, progressive forms of MS were reported previously. There was evidence of disease stabilization in our patient and long-term ITMTX appears as a safe therapeutic option in advanced BCS.
Keywords: Balo’s concentric sclerosis, intrathecal methotrexate, multiple sclerosis, aggressive disease, childhood
Deniz YUKSEL
University of Health Sciences Turkey, Dr Sami Ulus Maternity and Child Health and Diseases Research and Training Hospital
Turkey
Hale Celik
University of Health Sciences Turkey, Dr Sami Ulus Maternity and Child Health and Diseases Research and Training Hospital
Turkey
Ozge Kucur
Tekirdağ İsmail Fehmi Cumalıoğlu City Hospital
Turkey
Kader Oguz
Hacettepe University Faculty of Medicine
Turkey
Deniz YUKSEL
University of Health Sciences Turkey, Dr Sami Ulus Maternity and Child Health and Diseases Research and Training Hospital Turkey
University of Health Sciences Turkey, Dr Sami Ulus Maternity and Child Health and Diseases Research and Training Hospital Turkey