Five weeks after the last plasma exchange

Five weeks after the last plasma exchange. to reduce clinical relapses and brain volume loss in people with multiple sclerosis (MS).1,2 Dosing requires at least two intravenous courses at 12-month intervals.1,2 After each dose there is depletion of both T and B lymphocytes, yet data indicate that B cells recover more quickly than T cells, putting patients at risk for other autoimmune disorders, such as thyroid disease and immune thrombocytopenia.3 Recently, there is a growing body of literature suggesting increased disease activity after alemtuzumab induction associated with B-cell recovery.4C8 Of the 16 cases report to date, most were treated with fingolimod before alemtuzumab treatment.4C6,8 Of those treated for increased disease activity, most received rituximab.4,5,7 We describe an adult patient who presented with vision loss, cognitive decline, and altered gait who was stabilized with only steroid and extended plasma exchange therapy, without concomitant use of rituximab. The patient consented to the publication of the material contained herein. Case Presentation A 33-year-old woman was diagnosed at age 16 years as having relapsing-remitting MS based on clinical and magnetic resonance imaging (MRI) criteria. She had been taking several different disease-modifying therapies, including interferon beta-1a, interferon beta-1b, and natalizumab. Natalizumab use was discontinued due to a positive JC virus titer (JCV index, 0.73). Four months later she was admitted to the hospital for a multifocal relapse involving dysarthria, declining mobility, and cognitive impairment for several weeks. Examination was notable for trouble with following commands, word-finding difficulty, dysarthria, and paraparesis. The MRI performed before alemtuzumab use showed several new T2 lesions and areas of gadolinium enhancement (Figure 1A and Figure S1 [A1 and A2], which is published in the online version of this article at ijmsc.org). She received her first alemtuzumab infusion during the hospitalization. The preinfusion lymphocyte count was 1.24 109/L (reference range, 1.50C7.50 109/L). Approximately 6 weeks after hospitalization she was referred to our clinic, where her examination findings returned to baseline levels (visual acuity: 20/30 OD and 20/20 OS, no dysarthria or word-finding difficulty, and ambulatory without assistance). Follow-up brain MRI 3 months later showed no new lesions and resolution of the gadolinium-enhancing lesions (Figure 1B and Figure S1 [B1 and B2]). Laboratory investigations showed an elevated thyrotropin level (5.28 mIU/L) and a lymphocyte count of 0.87 109/L. Open in a separate window Figure 1. Serial T1-weighted brain magnetic resonance images of the patient after gadolinium administration A, Before alemtuzumab infusion. There are multiple gadolinium-enhancing lesions (arrowheads). B, After alemtuzumab infusion (4.5 months). There are no gadolinium-enhancing lesions. C, Disease activation with relapse. There is gadolinium enhancement of the optic nerves and chiasm (arrowhead). D, Madecassoside Madecassoside Recovery. After treatment with steroids and plasma exchange. The optic chiasm shows no gadolinium enhancement (arrowhead). E, Stabilization. Five weeks after the last plasma exchange. The optic chiasm shows no gadolinium enhancement (arrowhead). One month later the patient presented with bilateral vision loss and altered gait (ataxia and began using a walker). Examination showed Madecassoside a visual acuity of 20/100 C 1 at 1 ft OD and finger counting (one of three responses correct) OS along with worsening paraparesis. Urgent MRI showed multiple new gadolinium-enhancing lesions, including in the optic chiasm and nerves (Figure 1C and Figure S1 [C1CC4]), deep white matter, posterior fossa, and upper cervical spinal cord. Treatment was initiated with 5 days of intravenous methylprednisolone (Solu-Medrol; Pfizer, New York, NY) (1000 mg/d), followed by a 14-day prednisone taper. Her lymphocyte count was 0.77 109/L, and flow cytometry showed a normal number of Madecassoside B cells (0.33 109/L; reference range, 0.079C0.36 109/L) and a decreased absolute T-cell count (0.089 109/L; reference range, 0.53C2.19 109/L). Seven days later her gait improved (narrowed stance and ability to walk a few steps without Madecassoside assistance), as did her visual acuity (20/80 OD and 20/160 OS). Repeated brain MRI 7 weeks later showed reduced gadolinium enhancement of the optic chiasm and fewer gadolinium-enhancing lesions (not shown). Shortly after MRI, the patient started to develop worsening cognitive decrease and vision. She was treated with 5 days of oral prednisone (1250 mg/d) followed by hospitalization with 3 days of intravenous Solu-Medrol Rabbit Polyclonal to MP68 (1000 mg/d). Plasma exchange was initiated 4 weeks later on because of incomplete improvement of neurologic status. The patient received nine plasma exchanges over 6 weeks..