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examination demonstrating histologic features of IBM in several different skeletal muscles, including the diaphragm.[5] The diaphragm is not a common muscle chosen for biopsy. In our case, it was biopsied only because the patient was undergoing an exploratory laparotomy to rule out neoplasia. Our patient did not have respiratory symptoms, supporting the conclusion by one that diaphragm abnormalities can be present in patients without respiratory symptoms; therefore, IBM may involve the diaphragm more often than is clinically recognized.[1] Although, there has been one described autopsy study, to the best of our knowledge, this is the first reported case of IBM with biopsy evidence of diaphragm involvement. Awareness of the clinicopathologic spectrum of IBM is paramount for optimal patient management and to avoid potentially hazardous procedures as in this case. Additional clinical and pathological studies are necessary to assess the true prevalence of such occurrences.
Sarah E. Martin, Dibson D. Gondim, Eyas M. Hattab Department of Pathology and Laboratory Medicine, Indiana University School of Medicine, Indianapolis, IN, USA E-mail: [email protected]
References 1. 2. 3. 4.
5.
Teixeira A, Cherin P, Demoule A, Levy-Soussan M, Straus C, Verin E, et al. Diaphragmatic dysfunction in patients with idiopathic inflammatory myopathies. Neuromuscul Disord 2005;15:32-9. Greenberg SA. Pathogenesis and therapy of inclusion body myositis. Curr Opin Neurol 2012;25:630-9. Voermans NC, Vaneker M, Hengstman GJ, ter Laak HJ, Zimmerman C, Schelhaas HJ, et al. Primary respiratory failure in inclusion body myositis. Neurology 2004;63:2191-2. Littleton ET, Man WD, Holton JL, Landon DN, Hanna MG, Polkey MI, et al. Human T cell leukaemia virus type I associated neuromuscular disease causing respiratory failure. J Neurol Neurosurg Psychiatry 2002;72:650-2. Inamori Y, Higuchi I, Inoue T, Sakiyama Y, Hashiguchi A, Higashi K, et al. Inclusion body myositis coexisting with hypertrophic cardiomyopathy: An autopsy study. Neuromuscul Disord 2012;22:747-54.
A case of frontal lobe infarct in Plasmodium vivax infection Sir, Malaria is a mosquito-borne infectious disease caused by protists of the genus Plasmodium. According World Health Organization (WHO) estimates, there were about 219 million cases of malaria in 2010 and an estimated 660,000 deaths.[1] In South-East Asia, the second most affected region in the world, India has the highest malaria burden (with an estimated 24 million cases/year).[1] Cerebral malaria due to Plasmodium vivax, although rare, has been reported from Indonesia, India and Papua New Guinea. Recently cases with multiple cerebral infarcts following P. vivax infection has been reported from Korea.[2] Here, we report a case of frontal lobe cerebral infarct in P. vivax infection. A 30-year-old female patient presented with the complaints of fever since 15 days and headache and altered sensorium since 1 day. Fever was intermittent and was associated with chills and rigor. At the other facility peripheral blood smear showed P. vivax rings and hemoglobin was 6.6 g/ dL, total leukocyte count was 11000/mm3 and platelet count was 1.66 lakhs/mm3. Neurological examination revealed neck rigidity and Kernig’s sign. The patient was put on intravenous artesunate and ceftriaxone; fluids, antipyretics and other supportive treatment were also given. On the next day repeat investigations revealed: Hemoglobin of 6.9 g/dL, mild deranged liver function test (serum glutamic oxaloacetic transaminase: 51 IU/L, serum glutamic-pyruvic transaminase: 46 IU/L) and slightly low serum protein (total protein - 6.19 g/dL and albumin - 2.5 g/dL). Ultra sound abdomen showed hepatomegaly and splenomegaly. A packed cell has been transfused for severe anemia management. Magnetic resonance imaging brain done the next day showed infarct involving right frontal lobe [Figure 1a-c].
Access this article online Quick Response Code:
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a
b
PMID: *** DOI: 10.4103/0028-3886.128313
Received: 23-01-2014 Review completed: 31-01-2014 Accepted: 24-01-2014
Neurology India | Jan-Feb 2014 | Vol 62 | Issue 1
c Figure 1: (a) T1-weighted magnetic resonance (MR) image showing right frontal lobe infarct. (b) T2-weighted MR image showing right frontal lobe infarct. (c) Diffusion weighted MR showing right frontal lobe infarct
67
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Cerebrospinal fluid examination was normal. Patient improved on antimalarial therapy and was discharged on day-5. P. vivax accounts for nearly half of all malaria infections and is now recognized as a cause of complications and death. WHO defines cerebral malaria as unarousable coma in a patient with P. falciparum asexual parasitemia after other causes of encephalopathy have been excluded. Almost all patients with cerebral malaria present with fever, rigors and/or chills. Altered sensorium might be present from the outset or might develop slowly over a period of several days. Signs of irritability, restlessness or psychotic behavior can be the initial manifestations of cerebral involvement. Neck stiffness is occasionally encountered, but other signs of meningism are uncommon in our experience. In patients with P. vivax infections behavior changes, altered sensorium, seizures, cerebellar manifestations and ataxia, hemiparesis, aphasia, psychosis, acute inflammatory demylienating polyneuropathy and late bilateral facial paralysis have been described[3] and some of the cases may develop multi-organ involvement. Two mechanisms have been proposed for the neurological complications in malaria. The mechanical theory [4] suggests that cerebral capillary and venules occlusion by parasitized erythrocytes is caused by direct action of the parasite on the erythrocyte, distorting its morphology, altering the elasticity and plasticity and changing its surface properties. By the impaired capillaries flow and adhesion to the endothelium and other blood cells (forming aggregates, “rosettes”) obstruction supervenes. This results in thrombosis, anoxia and infarct and tissue necrosis. In more serious cases, further endothelial damage produces an increase in capillary permeability and even hemorrhages. According to the humoral[4] theory the action of non-specific vasoactive inflammatory substances producing changes in the capillary permeability results in compromisation of blood flow, impaired tissue perfusion and cellular hypoxia. Among the mediators tumor necrosis factor alpha and interleukin-2 are the important mediators in the inflammatory process. This theory holds that the more severe cases of malaria, especially cerebral malaria, are the result of an exacerbated inflammatory response of the host. Only single case of cerebral infarct has been reported as a manifestation of P. vivax infection until date.[2] Our patient was young with no known risk factors for ischemic stroke developed a frontal lobe cerebral infarct during the acute phase of P. vivax malarial infection. We feel the cerebral infarction is related to P. vivax infection however the exact mechanisms are not clear. P. vivax malaria is considered
68
to be benign infection, However, it can be complicated by thrombocytopenia, cerebral malaria and acute renal, hepatic and pulmonary dysfunctions and death.[5]
O. P. Jatav, Ashish Singhal, Ajay Pal Singh Department of General Medicine, Gajra Raja Medical College, Gwalior, Madhya Pradesh, India E-mail: [email protected]
References 1. 2. 3. 4. 5.
WHO World Malaria Report 2012 FACT SHEET. Available from: http:// www.who.int/malaria. [Last accessed on 2013 Mar 19, 7 pm IST]. Young-Kyoung J, Minn YK, Soo-Jin C, Kwon KH. Multiple cerebral infarcts following acute Plasmodium vivax infection. Korean J Stroke 2012;14:149-51. Garg RK, Karak B, Misra S. Neurological manifestations of malaria: An update. Neurol India 1999;47:85-91. Leopoldino JF, Fukujima MM, Gabbai AA. Malaria and stroke. Case report. Arq Neuropsiquiatr 1999;57:1024-6. Limaye CS, Londhey VA, Nabar ST. The study of complications of vivax malaria in comparison with falciparum malaria in Mumbai. J Assoc Physicians India 2012;60:15-8. Access this article online Quick Response Code:
Website: www.neurologyindia.com PMID: *** DOI: 10.4103/0028-3886.128314
Received: 01-11-2013 Review completed: 07-11-2013 Accepted: 02-02-2014
Polymyositis with myotonia in a patient with common variable immunodeficiency Sir, Common variable immune deficiency (CVID) is a primary immunodeficiency disorder characterized by hypogammaglobulinemia, recurrent bacterial infections and a high prevalence of autoimmune diseases, as well as neoplasia[1,2] and usually present with symptoms in the second to fourth decade of life.[3] CVID is caused by intrinsic B or T cell defects,[1] but the exact pathogenesis of this condition is poorly understood. Until date only one case of CIVD with inclusion body myositis,[2,3] and two cases with polymyositis (PM)[4] have been reported, hence this case report.
Neurology India | Jan-Feb 2014 | Vol 62 | Issue 1
Copyright of Neurology India is the property of Medknow Publications & Media Pvt. Ltd. and its content may not be copied or emailed to multiple sites or posted to a listserv without the copyright holder's express written permission. However, users may print, download, or email articles for individual use.
examination demonstrating histologic features of IBM in several different skeletal muscles, including the diaphragm.[5] The diaphragm is not a common muscle chosen for biopsy. In our case, it was biopsied only because the patient was undergoing an exploratory laparotomy to rule out neoplasia. Our patient did not have respiratory symptoms, supporting the conclusion by one that diaphragm abnormalities can be present in patients without respiratory symptoms; therefore, IBM may involve the diaphragm more often than is clinically recognized.[1] Although, there has been one described autopsy study, to the best of our knowledge, this is the first reported case of IBM with biopsy evidence of diaphragm involvement. Awareness of the clinicopathologic spectrum of IBM is paramount for optimal patient management and to avoid potentially hazardous procedures as in this case. Additional clinical and pathological studies are necessary to assess the true prevalence of such occurrences.
Sarah E. Martin, Dibson D. Gondim, Eyas M. Hattab Department of Pathology and Laboratory Medicine, Indiana University School of Medicine, Indianapolis, IN, USA E-mail: [email protected]
References 1. 2. 3. 4.
5.
Teixeira A, Cherin P, Demoule A, Levy-Soussan M, Straus C, Verin E, et al. Diaphragmatic dysfunction in patients with idiopathic inflammatory myopathies. Neuromuscul Disord 2005;15:32-9. Greenberg SA. Pathogenesis and therapy of inclusion body myositis. Curr Opin Neurol 2012;25:630-9. Voermans NC, Vaneker M, Hengstman GJ, ter Laak HJ, Zimmerman C, Schelhaas HJ, et al. Primary respiratory failure in inclusion body myositis. Neurology 2004;63:2191-2. Littleton ET, Man WD, Holton JL, Landon DN, Hanna MG, Polkey MI, et al. Human T cell leukaemia virus type I associated neuromuscular disease causing respiratory failure. J Neurol Neurosurg Psychiatry 2002;72:650-2. Inamori Y, Higuchi I, Inoue T, Sakiyama Y, Hashiguchi A, Higashi K, et al. Inclusion body myositis coexisting with hypertrophic cardiomyopathy: An autopsy study. Neuromuscul Disord 2012;22:747-54.
A case of frontal lobe infarct in Plasmodium vivax infection Sir, Malaria is a mosquito-borne infectious disease caused by protists of the genus Plasmodium. According World Health Organization (WHO) estimates, there were about 219 million cases of malaria in 2010 and an estimated 660,000 deaths.[1] In South-East Asia, the second most affected region in the world, India has the highest malaria burden (with an estimated 24 million cases/year).[1] Cerebral malaria due to Plasmodium vivax, although rare, has been reported from Indonesia, India and Papua New Guinea. Recently cases with multiple cerebral infarcts following P. vivax infection has been reported from Korea.[2] Here, we report a case of frontal lobe cerebral infarct in P. vivax infection. A 30-year-old female patient presented with the complaints of fever since 15 days and headache and altered sensorium since 1 day. Fever was intermittent and was associated with chills and rigor. At the other facility peripheral blood smear showed P. vivax rings and hemoglobin was 6.6 g/ dL, total leukocyte count was 11000/mm3 and platelet count was 1.66 lakhs/mm3. Neurological examination revealed neck rigidity and Kernig’s sign. The patient was put on intravenous artesunate and ceftriaxone; fluids, antipyretics and other supportive treatment were also given. On the next day repeat investigations revealed: Hemoglobin of 6.9 g/dL, mild deranged liver function test (serum glutamic oxaloacetic transaminase: 51 IU/L, serum glutamic-pyruvic transaminase: 46 IU/L) and slightly low serum protein (total protein - 6.19 g/dL and albumin - 2.5 g/dL). Ultra sound abdomen showed hepatomegaly and splenomegaly. A packed cell has been transfused for severe anemia management. Magnetic resonance imaging brain done the next day showed infarct involving right frontal lobe [Figure 1a-c].
Access this article online Quick Response Code:
Website: www.neurologyindia.com
a
b
PMID: *** DOI: 10.4103/0028-3886.128313
Received: 23-01-2014 Review completed: 31-01-2014 Accepted: 24-01-2014
Neurology India | Jan-Feb 2014 | Vol 62 | Issue 1
c Figure 1: (a) T1-weighted magnetic resonance (MR) image showing right frontal lobe infarct. (b) T2-weighted MR image showing right frontal lobe infarct. (c) Diffusion weighted MR showing right frontal lobe infarct
67
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Cerebrospinal fluid examination was normal. Patient improved on antimalarial therapy and was discharged on day-5. P. vivax accounts for nearly half of all malaria infections and is now recognized as a cause of complications and death. WHO defines cerebral malaria as unarousable coma in a patient with P. falciparum asexual parasitemia after other causes of encephalopathy have been excluded. Almost all patients with cerebral malaria present with fever, rigors and/or chills. Altered sensorium might be present from the outset or might develop slowly over a period of several days. Signs of irritability, restlessness or psychotic behavior can be the initial manifestations of cerebral involvement. Neck stiffness is occasionally encountered, but other signs of meningism are uncommon in our experience. In patients with P. vivax infections behavior changes, altered sensorium, seizures, cerebellar manifestations and ataxia, hemiparesis, aphasia, psychosis, acute inflammatory demylienating polyneuropathy and late bilateral facial paralysis have been described[3] and some of the cases may develop multi-organ involvement. Two mechanisms have been proposed for the neurological complications in malaria. The mechanical theory [4] suggests that cerebral capillary and venules occlusion by parasitized erythrocytes is caused by direct action of the parasite on the erythrocyte, distorting its morphology, altering the elasticity and plasticity and changing its surface properties. By the impaired capillaries flow and adhesion to the endothelium and other blood cells (forming aggregates, “rosettes”) obstruction supervenes. This results in thrombosis, anoxia and infarct and tissue necrosis. In more serious cases, further endothelial damage produces an increase in capillary permeability and even hemorrhages. According to the humoral[4] theory the action of non-specific vasoactive inflammatory substances producing changes in the capillary permeability results in compromisation of blood flow, impaired tissue perfusion and cellular hypoxia. Among the mediators tumor necrosis factor alpha and interleukin-2 are the important mediators in the inflammatory process. This theory holds that the more severe cases of malaria, especially cerebral malaria, are the result of an exacerbated inflammatory response of the host. Only single case of cerebral infarct has been reported as a manifestation of P. vivax infection until date.[2] Our patient was young with no known risk factors for ischemic stroke developed a frontal lobe cerebral infarct during the acute phase of P. vivax malarial infection. We feel the cerebral infarction is related to P. vivax infection however the exact mechanisms are not clear. P. vivax malaria is considered
68
to be benign infection, However, it can be complicated by thrombocytopenia, cerebral malaria and acute renal, hepatic and pulmonary dysfunctions and death.[5]
O. P. Jatav, Ashish Singhal, Ajay Pal Singh Department of General Medicine, Gajra Raja Medical College, Gwalior, Madhya Pradesh, India E-mail: [email protected]
References 1. 2. 3. 4. 5.
WHO World Malaria Report 2012 FACT SHEET. Available from: http:// www.who.int/malaria. [Last accessed on 2013 Mar 19, 7 pm IST]. Young-Kyoung J, Minn YK, Soo-Jin C, Kwon KH. Multiple cerebral infarcts following acute Plasmodium vivax infection. Korean J Stroke 2012;14:149-51. Garg RK, Karak B, Misra S. Neurological manifestations of malaria: An update. Neurol India 1999;47:85-91. Leopoldino JF, Fukujima MM, Gabbai AA. Malaria and stroke. Case report. Arq Neuropsiquiatr 1999;57:1024-6. Limaye CS, Londhey VA, Nabar ST. The study of complications of vivax malaria in comparison with falciparum malaria in Mumbai. J Assoc Physicians India 2012;60:15-8. Access this article online Quick Response Code:
Website: www.neurologyindia.com PMID: *** DOI: 10.4103/0028-3886.128314
Received: 01-11-2013 Review completed: 07-11-2013 Accepted: 02-02-2014
Polymyositis with myotonia in a patient with common variable immunodeficiency Sir, Common variable immune deficiency (CVID) is a primary immunodeficiency disorder characterized by hypogammaglobulinemia, recurrent bacterial infections and a high prevalence of autoimmune diseases, as well as neoplasia[1,2] and usually present with symptoms in the second to fourth decade of life.[3] CVID is caused by intrinsic B or T cell defects,[1] but the exact pathogenesis of this condition is poorly understood. Until date only one case of CIVD with inclusion body myositis,[2,3] and two cases with polymyositis (PM)[4] have been reported, hence this case report.
Neurology India | Jan-Feb 2014 | Vol 62 | Issue 1
Copyright of Neurology India is the property of Medknow Publications & Media Pvt. Ltd. and its content may not be copied or emailed to multiple sites or posted to a listserv without the copyright holder's express written permission. However, users may print, download, or email articles for individual use.
