Infratentorial Involvement of Acute Disseminated Encephalomyelitis in a Postpartum Woman Gokcen Coban, MD, Fuldem Y. Donmez, MD, and Muhtesem A. Agıldere Abstract: Acute disseminated encephalomyelitis (ADEM) is an autoimmune inflammatory central nervous system disorder. The pathogenesis is not understood clearly and must be inves- tigated extensively. A 32-year-old woman was admitted with severe nausea, fever, vomiting, bilateral lower extremity weak- ness, and urinary incontinence. She had epidural anesthesia for labor 1 week ago. There was no history of vaccination or in- fection recently. Absence of oligoclonal bants, monophasic na- ture, evidence of cerebellar and spinal cord lesions, and dramatic response to intravenous steroid treatment suggested ADEM. We aimed to draw attention to the presence of ADEM in post- partum period after epidural anesthesia. Key Words: ADEM, diagnosis, epidural anesthesia, labor, MRI (Neurosurg Q 2013;23:213–215) A cute disseminated encephalomyelitis (ADEM) is an autoimmune inflammatory central nervous system (CNS) disorder, which usually occurs after an infection or vaccination. 1–3 However, to date, ADEM after epidural anesthesia and labor has not been reported. Unfortu- nately, the etiology of ADEM is still obscure and there are no biological markers to identify ADEM. Therefore, diagnosis is made on the basis of history, physical ex- amination, magnetic resonance imaging (MRI) of the brain and the spinal cord and by ruling out other diseases in the differential diagnosis such as multiple sclerosis. We report a 32-year-old woman, as the first presentation of ADEM in the postpartum period, with infratentorial involvement of the CNS diagnosed with MRI. Our ob- jective was to raise the awareness of the radiologist and neurologist regarding this difficult-to-diagnose disease in the presence of coexistent clinical, laboratory, and imag- ing findings. CASE REPORT A 32-year-old woman was admitted to our emergency room with severe nausea, fever, vomiting, bilateral lower extremity weakness, and urinary incontinence. There was no history of vaccination, hepatitis, seizure, visual disturbance, or any infection recently. The only clinical note was that she has had epidural anesthesia for labor 1 week ago. She was awake and oriented. On neurological examination, there was hypo- aesthesia under T6 level and vibration sensory loss of the lower extremity. Blood tests were unremarkable. Cerebrospinal fluid (CSF) and blood cultures were neg- ative. No oligoclonal band was detected. Serum antiaquaporin 4 (AQP4) antibody was negative. The tests carried out for human immunodeficiency virus, purified protein derivate, C-reactive protein, antinuclear antibody, and rheumatoid factor were all negative. On CSF examination, protein and glucose levels were 40 and 74 mg/dL, respectively. CSF virology study for cytome- galovirus, Epstein-Barr virus, influenza A and B, parainfluenza types, herpes simplex virus, Echo virus, chlamydia, rota virus, and adeno virus were negative. Brain MRI showed high signal intensity areas at cerebellar vermis adjacent to fourth ventricle on T2 and fluid attenuated inversion recovery images (Fig. 1). Cervical and thoracic spinal MRI showed hyperintense lesions in the spinal cord between C4- C5 (Fig. 2), T3-T7, and T11 levels (Fig. 3). After contrast ad- ministration, none of the lesions showed enhancement and there was no restriction on diffusion-weighted imaging. She was treated with intravenous (IV) methyl pre- dnisolone infusion (1000 mg/d) for 5 days, followed by oral steroids. On the third day of IV steroid therapy she had pro- gressive improvement and fully recovered on the ninth day. On the 12th month of her follow-up, she was fine and did not have any complaints. DISCUSSION The pathogenesis of ADEM is not understood clearly and must be investigated extensively. The absence of viral and bacterial antigens in the CNS is nearly uni- versal. The existing proofs suggest that ADEM results from a transient autoimmune response towards myelin or other self-antigens, via molecular mimicry or by non- specific activation of autoreactive T-cell clones. 4 ADEM usually occurs after an infection, most commonly mea- sles, mumps, influenza A or B, herpes simplex virus, ru- bella, mycoplasma, chlamydia, 1–3 or after a vaccination. 5 Furthermore, after IV and oral therapy with herbal ex- tracts have been reported. 6,7 Other than the case reports published in the literature, there is no definite etiological factor. As our patient lacked any of the above etiological factors, and the only clinical status was the delivery of her baby 1 week ago, our case might be the first documented case of postpartum ADEM. Neuroimaging is the most important supporter in diagnosing ADEM. The superiority of MRI has been From the Department of Radiology, Baskent University Faculty of Medicine, Selcuklu, Konya, Turkey. The authors declare no conflict of interest. Reprints: Gokcen Coban, MD, Department of Radiology, Baskent Uni- versity Faculty of Medicine, Hocacihan mah. Saray cad. No 1, Selcuklu, 42080 Konya/Turkey (e-mail: drgokcencoban@gmail.com). Copyright r 2013 by Lippincott Williams & Wilkins CASE REPORT Neurosurg Q Volume 23, Number 3, August 2013 www.neurosurgery-quarterly.com | 213