Annals of Indian Academy of Neurology
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   Table of Contents - Current issue
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January-March 2019
Volume 22 | Issue 1
Page Nos. 1-126

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ORATION  

Neurology practice in India: At crossroads p. 1
Sanjeev V Thomas
DOI:10.4103/aian.AIAN_507_18  
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VIEW POINT Top

Innovations in acute stroke reperfusion strategies Highly accessed article p. 6
Venugopalan Y Vishnu, MV Padma Srivastava
DOI:10.4103/aian.AIAN_263_18  
Vascular neurology is witnessing unprecedented innovations in the management of acute ischemic stroke, especially in reperfusion strategies. The emergence of mechanical thrombectomy with new generation devices has revolutionized the treatment of acute ischemic stroke with large vessel occlusion. The reperfusion strategies are evolving with the extension of the window period for thrombolysis and endovascular therapy through the concept of “tissue clock” in addition to the established “time clock.” The newer generation of thrombolytic drugs like tenecteplase are promising exciting times ahead in acute stroke care. In this “viewpoint,'” the evolution of reperfusion therapy in acute ischemic stroke will be discussed followed by recent innovations in reperfusion strategies.
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Anticancer drugs for Parkinson's disease: Is it a ray of hope or only hype? p. 13
Deepa Dash, Vinay Goyal
DOI:10.4103/aian.AIAN_177_18  
Parkinson's disease (PD) is a progressive neurodegenerative disorder characterized by the death of dopaminergic (DA) neurons in the substantia nigra. To develop therapeutic strategies to halt or slow the neurodegenerative process, it is imperative that we understand the pathogenesis of PD. With the current state of knowledge, multiple pathological pathways such as oxidative stress, inflammation due to microglial activation, apoptotic pathway activation via Abelson (c-Abl)tyrosine kinase enzyme, and DA toxins have been incriminated in causing DA neuronal death in PD. In the recent times, there is growing evidence of the role of c-Abl nonreceptor tyrosine kinase in the pathogenesis of PD. We give a short account of the potential of c-Abl inhibitors, the currently used anticancer drugs such as nilotinib in preventing the neurodegenerative process in PD.
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AIAN REVIEW Top

Vascular parkinsonism: A review on management updates p. 17
Thilini B Udagedara, Alahakoon Mudiyanselage Buddhika Dhananjalee Alahakoon, Inuka Kishara Goonaratna
DOI:10.4103/aian.AIAN_194_18  
“Vascular parkinsonism (VP)” is a condition which presents with the clinical features of parkinsonism that are presumably caused by cerebrovascular disease. It is classically described as symmetrical lower-body parkinsonism with gait unsteadiness and absence of tremors and is usually associated with pyramidal signs. Treatment for VP remains challenging as available data on the efficacy of current treatment options are contentious. VP is generally considered to be poorly responsive to levodopa, the most effective of the current treatment modalities for parkinsonism. However, there is evidence that some patients benefit from therapy with levodopa. This article reviews the place of levodopa in the treatment of VP.
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Propranolol: A 50-year historical perspective p. 21
AV Srinivasan
DOI:10.4103/aian.AIAN_201_18  
Propranolol is a beta-adrenergic receptor antagonist that was developed by the British scientist Sir James Black primarily for the treatment of angina pectoris, more than 50 years ago. It was not long before several other cardiovascular as well as noncardiovascular therapeutic uses of propranolol were discovered. Propranolol soon became a powerful tool for physicians in the treatment of numerous conditions such as hypertension, cardiac arrhythmias, myocardial infarction, migraine, portal hypertension, anxiety, essential tremors, hyperthyroidism, and pheochromocytoma. Owing to its action at multiple receptor sites, propranolol exerts several central and peripheral effects and is therefore useful in various conditions. Right from reduction in postmyocardial mortality to control of anxiety in performers, propranolol plays an important role in a plethora of medical conditions. Interestingly, even today, newer indications of this age-old drug are being discovered. Moreover, propranolol treatment has been found to be cost-effective when compared to other corresponding treatment options for individual indications. In this article, we attempt to recount the journey of propranolol right from its inception to the present day.
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ORIGINAL ARTICLES Top

Newer antiepileptic drugs discontinuation due to adverse effects: An observational study p. 27
Mehdi Golpayegani, Farhad Salari, Kurosh Gharagozli
DOI:10.4103/aian.AIAN_25_18  
Aims: Antiepileptic drugs are the main therapy for epilepsy. However, the incidence of adverse effects (AEs) results in treatment discontinuation. The aim of this study is evaluating the factors involved in discontinuation of antiepileptic drugs. Settings and Design: We studied 2797 epileptic patients who consumed levetiracetam (LEV), oxcarbazepine (OXC), topiramate (TPM), zonisamide (ZNS), rufinamide, and lacosamide to evaluate the discontinuation because of AEs. Statistical Analysis Used: Data were analyzed using descriptive statistics and Chi-square test. Results: This study showed the rate of discontinuation due to adverse reactions as follows: TPM (7.10%), OXC (4.5%), ZNS (1.8%), and LEV (1.6%) (Chi-square analysis, P < 0.0001). Our study also showed that 1.35% of the patients did not continue the therapy because of subjective experiences of the AEs. Furthermore, neurologic complications in TPM, skin rashes in OXC, and patients' subjective experiences in LEV prescription were the main reasons for nonadherence due to a AEs. Conclusions: AEs in newer antiepileptic drugs are extremely prevalent. Our observation revealed that skin rashes and paresthesia were the most probable causes of treatment discontinuation because of AEs.
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Risk factors of recurrence of febrile seizures in children in a tertiary care hospital in Kanpur: A one year follow up study p. 31
Navneet Kumar, Tanu Midha, Yashwant Kumar Rao
DOI:10.4103/aian.AIAN_472_17  
Background: Febrile seizures are commonly seen in children and about one-third of the children develop a recurrence of febrile seizures. Objectives: The main objective is to study the risk factors associated with recurrence of febrile seizures in Indian children. Methods: This prospective, longitudinal study was carried out in the Department of Pediatrics, GSVM Medical College, Kanpur. All children, 6 months to 5 years of age, attending the department from February 2015 to January 2016 presenting with first febrile seizures were included in the study and followed up for recurrence. Results: Of 528 children, 174 (32.9%) had recurrence and 354 (67.1%) had a single episode of febrile seizures. Recurrence was more in children <18 months (41.3%) as compared to children ≥18 months (24.1%). Children with temperature 101°F during the seizure had a recurrence rate of 52.5% while recurrence was seen in only 17.2% in children with temperature ≥105°F. There was a significant declining trend of recurrence with increase in temperature. Recurrence was significantly more common in children with a family history of febrile seizures (45.5%) as compared to those without family history (27.8%). Multiple logistic regression analysis revealed that younger age at onset of first seizure, lower temperature during the seizure, brief duration between the onset of fever and the initial seizure, and family history of febrile seizures were risk factors significantly associated with recurrence of febrile seizures in children. Conclusion: Younger age at first seizure, short duration of fever before the onset of first febrile seizure, lower temperature at onset, and family history of febrile seizures are risk factors of recurrence of febrile seizures in children.
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Practice trends in treating central nervous system tuberculosis and outcomes at a tertiary care hospital: A cohort study of 244 cases p. 37
Vinay Goyal, Arunmozhimaran Elavarasi, Abhishek , Garima Shukla, Madhuri Behari
DOI:10.4103/aian.AIAN_70_18  
Introduction: Tubercular meningitis (TBM) is a common cause of chronic meningitis in India; however, there is a paucity of literature on optimum duration and choice of drug therapy. Materials and Methods: This was an ambispective cohort study. Results: Two hundred and forty-four patients of central nervous system tuberculosis (CNS TB) who were seronegative for HIV were studied of whom 198 had TBM and 46 patients had tuberculoma without meningitis. Before completion of treatment, 84% of TBM patients underwent imaging. There was no difference in disability or mortality in patients, who were treated with various drug regimens in terms of duration of therapy or number of drugs at initiation of treatment. However when patients developed new complications, adding more drugs improved survival. Prolonging corticosteroid administration in patients with nonsatisfactory improvement at 8 weeks was not associated with prevention of disability. Conclusions: CNS TB is treated by neurologists and physicians in India, as per their experience due to different recommendations in various guidelines. There is a tendency to decide when to stop treatment based on neuroimaging given the fear of poor outcomes associated with recurrence of the disease. The duration of treatment or choice of drugs at the start of treatment did not affect disability.
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Pattern of idiopathic intracranial hypertension in Indian population p. 47
Asutosh Pal, Prasenjit Sengupta, Debsadhan Biswas, Chetana Sen, Avik Mukherjee, Sandip Pal
DOI:10.4103/aian.AIAN_116_18  
Objective: To evaluate the clinical and demographic profile, laboratory parameters and outcomes of Idiopathic Intracranial hypertension (IIH) patients from Indian subpopulation. Materials and method: We did a prospective study on patients who fulfilled the revised diagnostic criteria for Primary pseudotumor cerebri syndrome in adults and children, proposed by Friedman et al in 2013. All patients were examined for BMI, papilloedema, extraocular muscle movement, opening CSF pressure and underwent MRI, MR venography of brain and perimetry. Patients were followed up for a minimum period of 6 months, upto 2 years, with the outcomes monitored being visual acuity, visual field, headache, diplopia and optic disc changes. Results: We evaluated 33 patients (31 female and 2 male). 25 patients had BMI less than 25. Commonest clinical presentation was headache. 7 patients showed normal CSF opening pressure. The most common MRI finding was flattening of posterior aspect of globe and was found in 90.90% (30). 25 patients showed either unilateral or bilateral transverse sinus stenosis. Most common finding in perimetry was enlarged blind spot. 4 patients recovered spontaneously and rests were treated with acetazolamide (1gm/day). All showed favorable outcome when followed up. Conclusion: Obesity may not be a dominant risk factor for development of IIH in the Indian subcontinent. Non obese IIH have better prognosis and tend to have a good response to medical management alone.
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Refining the clinical features of serotonin syndrome: A prospective observational study of 45 patients p. 52
Sanjay Prakash, Chaturbhuj Rathore, Kaushik Kumar Rana, Ankit Dave, Hemant Joshi, Jay Patel
DOI:10.4103/aian.AIAN_344_18  
Introduction: Serotonin syndrome (SS) is a drug-induced clinical syndrome that results from the excess intrasynaptic concentration of serotonin. Prospective observations are limited for SS. Methods: We prospectively recruited 45 consecutive adult patients (>18 years) fulfilling the Hunter's criteria for SS. All patients were subjected to a detailed clinical history and examinations. Patients were subjected to appropriate investigations to find out the other causes. The causation of SS to serotonergic drugs was assessed according to Naranjo adverse drug reaction probability scale. Results: The mean age was 37.3 years (range: 18–59 years). Sixty-two percent of patients were male. There were 15 different underlying clinical syndromes for which serotonergic drugs were started. Psychiatry conditions (36%) and cough/respiratory tract infection (16%) were the two most common clinical conditions for starting serotonergic drugs. We noted 49 different symptoms and physical signs. Overall, tremor (78%) and dizziness (47%) were the two most common symptoms. Headache (16%) and dizziness (16%) were the two most common initial (or first) symptoms. However, gait difficulty and febrile encephalopathy were the two most common reasons to visit the hospital. We noted 18 different drugs causing SS. Thirty-eight percent of patients received single serotonergic agent antidepressants, pain medicines and cough syrups were other important drugs causing SS. Conclusions: This study represents the largest clinic-based study on SS. SS is not rare in clinical practice. However, various aspects of this syndrome are still to be determined. All patients on serotonergic drugs should be physically examined for the presence of SS on the development of any new symptom.
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Correlation between heart rate variability and bladder sensations during filling and voiding phase of urodynamic study in patients with myelopathy p. 61
Tenzil Gomez, Anupam Gupta, UK Rashmi Krishnan, Anirban Chakraborty, Talakad Narasappa Sathyaprabha, K Thennarasu
DOI:10.4103/aian.AIAN_453_17  
Objective: The objective of this study was to correlation between heart rate variability (HRV) and bladder sensations during filling and voiding phase of urodynamic study (UDS) in patients with myelopathy. Materials and Methods: Myelopathy patients (traumatic and nontraumatic) within 6 months of illness were included in the study. Demographic data, etiopathological diagnosis, and urinary complaints were noted. UDS was performed and simultaneous HRV calculated at each event of filling and voiding phase by recording and calculating standard deviation of normal-to-normal (NN) interval (SDNN), root mean square of successive differences, total power (TP), average heart rate, high frequency (HF), low-frequency (LF) ratio, and data analyzed. Results: The study included 30 patients (23 males) with a mean age of 31.2 years (range 18–60 years, standard deviation 11.6). The mean of LF in normalized units showed an increase from 43.6 ± 14.1 at baseline to 48.9 ± 17.4 at strong desire to void (SDV) and at urgency to 44.1 ± 14.5. HF at baseline 40.4 ± 14.1 reduced to 36.4 ± 12.8 at SDV and rose at urgency to 41.2 ± 13.2. LF/HF at baseline was 1.3 ± 0.8, which increased to 1.6 ± 1.1 at SDV and reduced at urgency to 1.2 ± 0.6. Significant change in mean value was seen in TP (P = 0.01) and SDNN (P = 0.009) at first desire to void. Significant positive trend was seen in TP (P = 0.048) and SDNN (P = 0.042) during filling. Conclusion: Comparison of HRV measures failed to show significant rise in sympathetic or parasympathetic component in myelopathy patients during UDS and requires more critical evaluation.
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Conglomerate ring-enhancing lesions are common in solitary neurocysticercosis and do not always suggest neurotuberculosis p. 67
Ajay Garg, Khush Preet Kaur, Leve Joseph Devaranjan Sebastian, Shailesh B Gaikwad, Rohit Bhatia, Mamta Bhushan Singh, Achal Srivastava, Ravindra Mohan Pandey
DOI:10.4103/aian.AIAN_221_18  
Background and Purpose: Differentiating between neurocysticercosis (NCC) and neurotuberculosis has serious therapeutic implications and this distinction relies heavily on neuroimaging. Few case reports discuss the conglomeration of ring-enhancing lesions (RELs) in patients with solitary NCC. The aim of our study is to describe the imaging findings of conglomerate RELs in a cohort of patients with solitary NCC, emphasizing the frequency of conglomeration. Materials and Methods: This retrospective study included 100 patients with solitary NCC. Two neuroradiologists analyzed contrast-enhanced computed tomography (CT) images regarding morphology, enhancement pattern, location, number of lesions, and degree of perilesional edema. The solitary lesions were classified as solitary discrete RELs (SD-RELs) when a well-defined lesion was seen and solitary conglomerate RELs (SC-RELs) when two or more ring lesions or ring/rings plus disc lesions were present contiguously. Follow-up CT scans were evaluated for the resolution of lesions and surrounding edema. Results: Out of 100 patients, 42 were SD-RELs and 58 were SC-RELs. No statistically significant difference was found between both groups in terms of age of presentation, clinical presentation, lesion size and location, and degree of perilesional edema. Larger lesions (>10 mm) were more likely to show scolex and were associated with greater degree of edema in both subgroups. During follow-up, 13 patients had new lesions (SD-RELs-5, SC-RELs-8). In SD-RELs, follow-up lesions were in the same location in four patients and new location in one; and in SC-RELs, lesions were in the same location in seven and in new location in one case. Conclusion: Conglomeration of RELs is a common finding in patients with solitary NCC.
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A series of biopsy-proven patients with immunoglobulin G4-related neurological disease p. 73
Yareeda Sireesha, Megha S Uppin, Shridhar Ganti, Rajesh Alugolu, Vijaya Saradhi Mudumba, Suchanda Bhattacharjee, Mathukumalli L Neeharika, Jogendra Bastia, Meena Angamuthu Kanikannan
DOI:10.4103/aian.AIAN_283_18  
Aim: To study the clinical presentation, radiological findings, and therapy responsiveness of patients with biopsy-proven immunoglobulin G4 (IgG4)-related neurological disease. Methods: The study was conducted between January 2016 and March 2018 from the Department of Neurology and Pathology of Nizam's Institute of Medical Sciences. Patients with neurological symptoms and biopsy suggestive of IgG4-related disease (IgG4-RD) were included. These patients were studied for their demographic pattern and clinical presentation. The presence of serological markers such as vasculitic profile and IgG4 levels was analyzed. Radiological findings were studied in detail. Therapeutic agents used and the response to therapy were assessed. Results: There were six cases with IgG4-related neurological disease which were all hypertrophic pachymeningitis. The age ranged from 35 to 64 (mean = 46) years. The clinical presentation was acute in one, subacute in two, and chronic in three patients. The most common presenting symptom was headache (4), followed by gait and/or urinary disturbances (2), paraparesis (1), and diplopia (1). IgG4 levels were elevated in 50% of them. Pseudotumor-like mass and sinovenous thrombosis, not described previously, were seen in one patient. All the patients were treated with oral or intravenous steroid. Rituximab was given in three patients; azathioprine was the steroid-sparing agent in one patient. Those with acute/subacute onset of presentation had an excellent response to steroids. All the patients with a chronic duration of their symptoms received empirical anti-tuberculous therapy before a definitive diagnosis of Ig G4-RD was made. Conclusions: The characterization of patients with IgG4-related neurological disease based on the understanding of the clinical spectrum increases the confidence in the clinician to resort to early immunosuppression, thereby having prognostic implications.
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Parkinson's disease in intensive care unit: An observational study of frequencies, causes, and outcomes p. 79
Gunchan Paul, Birinder Singh Paul, Parshotum Lal Gautam, G Singh, S Kaushal
DOI:10.4103/aian.AIAN_44_18  
Objective: To analyze the frequency, causes, and outcomes of admission to the Intensive Care Unit (ICU) among Parkinson's disease (PD) population so that preventive measures can be developed. Methods: We prospectively observed patients with diagnosis of PD admitted to ICU from January 2014 to December 2016. Based on etiology for hospital admission, they were divided into two groups – related to PD (further divided into direct or indirect) or not associated with PD at all. Etiology for hospitalization was determined from history and investigational data. The primary outcome was death or discharge from the hospital. Factors contributing to ICU admission were analyzed by comparing these patients with a cohort of 50 PD patients admitted to the neurology ward during the same study period. All values were expressed as mean (standard deviation) and percentages using SPSS version 16.0. Results: Fifty-three (36%) out of a total of 146 patients required ICU admission. Most common causes leading to admission in decreasing order of frequency were fever (34%), delirium (16%), falls (12%), encephalopathy (8%), gastrointestinal emergencies (6%); while direct disease-related severe dyskinesias were seen only in two patients (4%). 13.7% needed mechanical ventilation and mean duration of ventilation was 5.94 days with mortality rate of 20%. Significant factors predicting ICU admission, and thus, poor outcomes were age >65 years, history of previous admission within the last 12 months, delirium, and hypoalbuminemia. There was no significant association between the incidence of ICU admission and duration of disease or severity of the disease. Conclusions: Poor outcome in PD patients is due to systemic causes, hence multidisciplinary teamwork may improve outcome in these patients.
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Determinants of outcome in convulsive status epilepticus in adults: An ambispective study from central India p. 84
Raunak Dani, Ajoy Sodani, Kapil Telang, Richa Nigam
DOI:10.4103/aian.AIAN_466_17  
Background: The determinants of the outcome in adult convulsive status epilepticus(CSE), also the implication of the value of mean arterial blood pressure (MAP), and random blood sugar at admission on the outcome are not clear. Objectives: The objective of this study is to look for the determinants of unfavorable outcome in CSE. Materials and Methods: Ambispectively gathered data from 55 patients, treated consecutively with identical protocol during January 2010–December 2016, were analyzed. The demographic and clinical variables were identified and correlated with outcome in each individual. Results: There were 65.45% males and 34.55% females. Favorable outcome (conscious and discharged) was seen in 63.6%, unfavorable (death 14.5%, absent cortical functions 10.9%, and inability to wean-off anesthetic agents 10.9%). The parameters associated with unfavorable outcome were female gender (odds ratio [OR]: 1.45), MAP ≤80 mmHg (OR: 2.57), time to first medical attention >5 h (OR: 127.8), and time to control clinical seizures >3.5 h (OR: 7.87). Almost 44.2% of patients with SE severity score >2 had unfavorable outcome (sensitivity 75% and specificity 45.7%). New scoring system, the CSE outcome score (CSEOS, developed by combining the predictors associated with higher odds of poor outcome), predicted the poor outcome with the sensitivity and specificity of 90% and 54.29%, respectively. Discussion and Conclusion: Low MAP and delay of >3.5 h in treatment initiation or seizure control are the key determinants of poor outcome in CSE. With the incorporation of CSEOS, we believe that our findings can be helpful in the process of clinical decision-making and prognostication of patients with CSE.
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SHORT COMMUNICATIONS Top

Prospective hospital-based clinical and electrophysiological evaluation of acute organophosphate poisoning p. 91
Karkal Ravishankar Naik, Aralikatte Onkarappa Saroja, Nagabushan Hesarur, Rekha Satish Patil
DOI:10.4103/aian.AIAN_137_18  
Introduction: Acute organophosphate (OP) poisoning is one of the most common poisoning causing significant morbidity and mortality in developing countries. Acute cholinergic manifestations predominate with many patients requiring intensive care management and ventilator support. Nerve conduction studies including repetitive nerve stimulation can evaluate the altered neuromuscular transmission and peripheral nerve function by OPs. Objective: To evaluate the electrophysiological abnormalities in patients with acute OP poisoning and correlate with clinical status. Materials and Methods: Patients with acute OP poisoning admitted from August 2016 to August 2017 were prospectively studied. Nerve conduction studies including phrenic nerve conduction were performed within 24 h of admission. Repetitive nerve stimulation was performed at 3 and 30 Hz. Nerve conduction findings were compared with data from age-matched healthy controls. Results: Thirty patients were included (18 men and 12 women) in the study. Their age ranged from 16 to 47 years (30 ± 9.2). The first assessment revealed a mild reduction of compound muscle action potential (CMAP) amplitude and reduced F-wave persistence. Eleven patients had repetitive CMAPs suggesting cholinergic excess. Seven among the 11 patients requiring mechanical ventilation had decrement–increment response with 30 Hz stimulation and reduced diaphragmatic CMAP amplitude (P = 0.02). Conclusion: The presence of repetitive CMAPs, decrement–increment response to tetanic stimulation and reduced diaphragmatic CMAP amplitude in OP poisoning patients correlate with neuromuscular paralysis and need for mechanical ventilation.
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Rapid processing of perfusion and diffusion for ischemic strokes in the extended time window: An Indian experience p. 96
Devashish Vyas, Vikram Bohra, Vivek Karan, Vikram Huded
DOI:10.4103/aian.AIAN_142_18  
Background: Endovascular treatment of acute ischemic stroke with large-vessel occlusion is the standard of care now. Initially restricted to 6 h after onset, the treatment can now be offered to selected patients up to 24 h based on clinical and imaging criteria. Objective: Perfusion imaging can help in identifying patients who may benefit from endovascular treatment in the extended time window. Manual analysis of perfusion images is time and skill intensive. Rapid processing of perfusion and diffusion (RAPID) is an automated image analysis system that analyzes perfusion maps. We report our initial experience of using this system in selection of patients for endovascular stroke treatment. Methods: All patients who presented with acute stroke underwent baseline imaging with computed tomography (CT) and CT angiogram or magnetic resonance imaging (MRI) and MR angiogram. Patients presenting between 6 and 24 h after onset underwent perfusion imaging, which was analyzed by RAPID. The results were used to select the patients who then underwent mechanical thrombectomy. Results: RAPID results identifying ischemic core and hypoperfused tissue were available within 5 min in each of the three cases. At 3 months, all patients showed improvement in the modified Rankin Scale. Conclusion: In extended time windows, RAPID provides a fast and reliable estimate of salvageable brain tissue to help select patients for endovascular treatment.
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IMAGES IN NEUROLOGY Top

Neurosarcoidosis as a cause of longitudinally extensive myelitis: Neuroimaging clues for diagnosis p. 100
Ajay Asranna, Aneesh Mohimen, Roopa Rajan, Muralidharan Nair
DOI:10.4103/aian.AIAN_162_18  
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CASE REPORTS Top

Anti-NMDAR encephalitis in association with herpes simplex virus and viral and bacterial zoonoses p. 102
Stefanie Kar Yan Hung, Fu Liong Hiew, Shanthi Viswanathan
DOI:10.4103/aian.AIAN_232_18  
Multiple co-infections can predispose a patient to autoimmune encephalitis. Out of thirty cases of N-methyl-D-aspartate receptor (NMDAR) encephalitis seen at a single tertiary referral center, only two cases of co-infection with NMDAR encephalitis were identified. One of these cases was highly interesting due to the presence of more than one co-infections along with the presence of cortical dysfunction, seizures, and orofacial dyskinesias at the onset in a male in the absence of tumors, which was refractory to initial treatment.
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Hyperacute paraplegia and neurovascular (immuno vasculotoxic) catastrophe of nicolau syndrome: Primum non nocere p. 104
Bhaskara P Shelley, Prasanth Prasad, Malla M Manjunath, Shrijeet Chakraborti
DOI:10.4103/aian.AIAN_298_18  
A case of Nicolau syndrome (NS) in a 36-year-old adult taking an unusual and devastating hyperacute irreversible paraplegia after an intramuscular injection of benzathine penicillin as a part of routine chemoprophylaxis of her rheumatic heart disease is reported. Although this syndrome is a considerably rare, iatrogenic and underappreciated dermatologic entity, we reiterate in this report, its extracutaneous systemic potential for a catastrophic neurovascular phenomenon and morbidity as well as its possible preventive measures. The apoplectiform onset of T10 flaccid areflexic paraplegia, with the cutaneous hallmark of “embolia cutis medicamentosa” was corroborated by magnetic resonance imaging evidence of centromedullary complete cord involvement from T10 to conus medullaris. Combination therapy with pulse methylprednisolone, low-molecular-weight heparin, and pentoxifylline infusion proved unsuccessful. The skin biopsy and direct immunofluorescence revealed features were consistent with NS with overlap features of leukocytoclastic vasculitis, hitherto not reported. The literature of this preventable and iatrogenic disorder is reviewed, and plausible etiology is discussed.
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Eosinophilic meningitis: Report of three cases from a hilly area in Kerala p. 108
Bindu Varghese, Murali Krishna Menon, Thara Pratap, A Moheen, Jomy Mathew, TP Vijayan, MP Tomy, Muhammed Jasim Abdul Jalal
DOI:10.4103/aian.AIAN_254_18  
Normal cerebrospinal fluid (CSF) does not contain eosinophils. The presence of >10 eosinophils/μL in CSF or at least 10% eosinophils in total CSF leukocyte count confirms eosinophilic meningitis. We present three patients with eosinophilic meningitis from the same locality with peripheral eosinophilia.
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Recurrent encephalopathy with spinal cord involvement: An atypical manifestation of Aicardi–Goutières syndrome p. 111
Debopam Samanta, Raghu Ramakrishnaiah
DOI:10.4103/aian.AIAN_12_18  
Aicardi–Goutières syndrome (AGS) is a rare, genetic inflammatory disease due to mutations in any of the seven genes discovered to date (TREX1, RNASEH2A, RNASEH2B, RNASEH2C, SAMHD1, ADAR, and IFIH1). Clinical onset is seen most commonly in utero or in infancy; irritability, feeding difficulties, jitteriness, microcephaly, abnormal movements, seizures, bone marrow suppression, and liver dysfunction are seen either during the neonatal age group or within the first few months of life with abrupt onset of neurologic regression and slowing of head growth. Diffusely abnormal white matters with swelling of frontal or temporal lobes, cerebral atrophy, and intracranial calcification are typical neuroradiologic abnormalities. However, ADAR mutation, a recently discovered AGS gene, can cause late-onset acute or subacute onset of severe dystonia and features of bilateral striatal necrosis on neuroimaging, in the absence of other typical features of AGS. We report a detailed description of a 5-year-old boy who had a recurrent encephalopathic presentation in the setting of infection. Magnetic resonance imaging (MRI) of brain revealed prominent and fairly symmetrical signal abnormalities in the cerebellar peduncles, thalamus, midbrain, and pons. His throat swab was positive for influenza B, and he was initially diagnosed with influenza encephalopathy. He had a recurrence after 18 months of his initial presentation, and his brain MRI showed extensive areas of signal abnormality similar to, but more extensive than, his previous scan. Extensive spinal cord swelling was also seen. His chronic skin finding was recognized as dyschromatosis symmetrica hereditaria (DSH), and genetic testing revealed compound heterozygous mutations of ADAR gene – causative for AGS. This is the first presentation of recurrent acute encephalopathy in the setting of documented ADAR mutation with the longest interval documented between two acute presentations. This is also the first documentation of extensive spinal cord involvement, which will expand its phenotype. This case also highlights the importance of early identification of DSH, a subtle but characteristic skin lesion of ADAR mutations, for prompt diagnosis of this rare condition.
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CLINICAL SIGN Top

Unilateral vertical pendular nystagmus in multiple sclerosis: A distinctive neuro-ophthalmological sign p. 116
Jeenendra Prakash Singhvi, Amit Shankar Singh
DOI:10.4103/aian.AIAN_289_18  
Lesions in multiple sclerosis can involve brain, optic nerve and spinal cord. We here report a patient of multiple sclerosis, who had unilateral vertical pendular nystagmus, because of simultaneous involvement of optic nerve and asymmetric brain stem lesions. This specific combination is rarely seen in other disorders, therefore can be considered as a distinctive neuro-opthalmoloical sign of multiple sclerosis.
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LETTERS TO THE EDITOR Top

Transient left alien hand syndrome after left anterior cerebral artery infarction p. 118
Mansoor C Abdulla, Rejeesh Saseendran
DOI:10.4103/aian.AIAN_143_18  
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D-bifunctional protein deficiency: A case report of a Turkish child p. 119
Faruk Incecik, Neslihan O Mungan
DOI:10.4103/aian.AIAN_273_18  
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Leucine-Rich glioma-inactivated protein 1 antibody-positive limbic encephalitis in a patient with adenocarcinoma of prostate: A case report p. 121
Devaraddi Navalli, Narayan R Mutalik, G Jayalakshmi
DOI:10.4103/aian.AIAN_377_18  
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Intravenous immunoglobulin for severe protracted pediatric Guillain–Barre syndrome: Is single dose adequate? p. 123
Priyanka Madaan, Prashant Jauhari, NM Shruthi, Biswaroop Chakrabarty, Sheffali Gulati
DOI:10.4103/aian.AIAN_100_18  
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A case of relapsing-remitting tumefactive demyelination p. 124
Ankur Wadhwa, Atri Chatterjee, Neera Chaudhry, Sanghamitra Laskar, Shishir Chandan
DOI:10.4103/aian.AIAN_375_18  
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