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Letter to the Editor
Rapidly progressive dementia with generalized myoclonus in an adult: Do not forget subacute sclerosing panencephalitis
Demencia rápidamente progresiva con mioclonías generalizadas en un adulto: no olvides la panencefalitis esclerosante subaguda
R. Ghosha, S. Dubeyb, A. Mukherjeea, J. Benito-Leónc,d,e,
Corresponding author
jbenitol67@gmail.com

Corresponding author.
a Department of General Medicine, Burdwan Medical College & Hospital, Burdwan, West Bengal, India
b Department of Neuromedicine, Bangur Institute of Neurosciences, Kolkata, India
c Department of Neurology, University Hospital “12 de Octubre”, Madrid, Spain
d Department of Neurology, Centro de Investigación Biomédica en Red sobre Enfermedades Neurodegenerativas (CIBERNED), Madrid, Spain
e Department of Medicine, Complutense University, Madrid, Spain
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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Persistence of measles infection in children and young adults can rarely lead to a fatal progressive neurodegenerative disorder known as subacute sclerosing panencephalitis &#40;SSPE&#41;&#46;<a class="elsevierStyleCrossRefs" href="#bib0065"><span class="elsevierStyleSup">1&#8211;4</span></a> Amongst the other known neurological manifestations of measles infection&#44; SSPE is the rarest and most severe&#46;<a class="elsevierStyleCrossRefs" href="#bib0065"><span class="elsevierStyleSup">1&#8211;4</span></a> However&#44; measles infection is amenable to prevention by highly effective live vaccination&#46;<a class="elsevierStyleCrossRefs" href="#bib0065"><span class="elsevierStyleSup">1&#8211;5</span></a> The latency period between measles infection and development of clinical features of SSPE is variable and can be up to 2&#8211;3 decades&#46;<a class="elsevierStyleCrossRefs" href="#bib0065"><span class="elsevierStyleSup">1&#8211;4</span></a> Earlier affection and intrafamilial cases of SSPE have a shorter latency&#46;<a class="elsevierStyleCrossRefs" href="#bib0065"><span class="elsevierStyleSup">1&#8211;4</span></a> The clinical course is characterized by subacute to chronic progressive cognitive impairment&#44; behavioral abnormalities&#44; movement disorders &#40;especially myoclonus&#41;&#44; seizures&#44; visual disturbances&#44; and ataxia&#46;<a class="elsevierStyleCrossRefs" href="#bib0065"><span class="elsevierStyleSup">1&#8211;3</span></a> Although few reports of spontaneous remissions of SSPE have been documented&#44;<a class="elsevierStyleCrossRefs" href="#bib0090"><span class="elsevierStyleSup">6&#44;7</span></a> this disease is considered universally fatal&#46; Diagnosis of SSPE is difficult in developed countries because of extreme rarity of the condition and atypical and non-specific initial presentation because the sufferers come rather early in the course of disease&#46;<a class="elsevierStyleCrossRefs" href="#bib0065"><span class="elsevierStyleSup">1&#8211;3&#44;8</span></a> On the contrary&#44; in underdeveloped&#47;developing countries&#44; the delay in diagnosis occurs due to low health-related literacy in general population&#44; significant lack of knowledge and training of primary care physicians regarding these disorders&#44; inadequate health-seeking behavior of the general population&#44; and inadequate investigational backup in peripheral health care setups &#46;<a class="elsevierStyleCrossRefs" href="#bib0065"><span class="elsevierStyleSup">1&#8211;3&#44;8</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">We herein report a case of SSPE in a 36-year-old previously healthy man with questionable vaccination status in childhood who presented with rapidly progressive early onset dementia and subcortical myoclonus&#46; Neuroimaging revealed altered signals involving bilateral thalamus and brainstem widening the list of differential diagnoses&#59; however&#44; clinical history&#44; examination findings&#44; and high anti-measles IgG titers in serum and cerebrospinal fluid &#40;CSF&#41; clinched the diagnosis&#44; which was supported by classical Radermecker&#39;s complex in electroencephalography &#40;EEG&#41;&#46;<a class="elsevierStyleCrossRef" href="#bib0105"><span class="elsevierStyleSup">9</span></a></p><p id="par0015" class="elsevierStylePara elsevierViewall">A 36-year-old previously healthy man from a middle socio-economic family of rural West Bengal &#40;India&#41; was brought to the out-patient department with complaints of rapidly progressive behavioral abnormalities &#40;inattentiveness&#44; forgetfulness&#44; mood disturbances&#44; aggressive behavior&#44; lack of interest in surroundings&#44; calculation difficulties&#44; difficulties in recognizing relatives and common objects&#44; and loosing ways during navigation&#41; and visual difficulties for last 3&#8211;4 months&#46; The family members also gave history of objects falling from his hands unknowingly&#44; and gradually evolving abnormal involuntary jerky &#8220;shock-like&#8221; movements involving all the limbs and torso for the last 2 months&#46; Past medical history and history of long-term drug exposure or addiction in any form were unyielding&#46; The family members revealed that one of his brothers also had succumbed to a similar illness within 2 years of developing similar symptoms at the age of 18 years&#46; On enquiry&#44; his mother described an episode of &#8220;fever with confluent rash&#8221; with spontaneous recovery in all her children&#44; in their childhood&#44; over a period of 2 weeks suggestive of measles&#46; Neurological examination revealed multi-domain cognitive impairments with subcortical myoclonus&#46;</p><p id="par0020" class="elsevierStylePara elsevierViewall">Brain magnetic resonance imaging &#40;MRI&#41; showed non-enhancing bilaterally symmetrical altered intensity lesions&#44; hyperintense on T2-weighted imaging and FLAIR sequences&#44; involving thalamus and brainstem &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46; EEG revealed periodic discharges suggestive of Radermecker complexes &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46; Contrast enhanced MR angiography and MR venography excluded common vascular insults&#46; Accordingly&#44; relevant tests were done&#44; which excluded metabolic disorders &#40;blood glucose&#44; thyroid&#44; liver and renal function tests&#44; electrolytes&#44; vitamin B1 and B12&#44; arterial blood gas analysis&#44; serum lactate&#44; CSF lactate&#44; lactate&#47;pyruvate ratio&#44; and anti-thyroperoxidase&#47;thyroglobulin antibody were normal&#41;&#44; toxic encephalopathy &#40;toxicology screen&#41;&#44; acute infectious encephalopathies &#40;neuro-viruses panel&#44; including SARS-CoV-2 and HIV 1&#44; 2&#44; were negative&#41;&#46; Autoimmune&#47;paraneoplastic encephalitis panel was negative&#46; CSF study revealed lymphocyte-predominant pleocytosis &#40;cell count&#58; 25&#47;&#956;l&#41;&#44; increased total protein &#40;108<span class="elsevierStyleHsp" style=""></span>mg&#47;dL&#41;&#44; and raised IgG index&#46; CSF and serum were tested for anti-measles IgG antibody titers and they came out to be extremely high confirming the diagnosis of SSPE further &#40;fulfillment of Dyken&#39;s criteria&#41;&#46;<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">8</span></a></p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0025" class="elsevierStylePara elsevierViewall">Quite expectedly&#44; resource-constrained countries with a high burden of measles encounter a higher incidence of SSPE&#46;<a class="elsevierStyleCrossRefs" href="#bib0065"><span class="elsevierStyleSup">1&#8211;3</span></a> Besides&#44; significant number of cases remain undiagnosed&#47;misdiagnosed and unreported&#46;<a class="elsevierStyleCrossRefs" href="#bib0065"><span class="elsevierStyleSup">1&#8211;3&#44;8</span></a> However&#44; measles vaccine has a preventive effect against SSPE by simply protecting against measles&#46;<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">5</span></a> Countries with high vaccination gap and recent outbreaks is associated with higher incidence of SSPE&#46;<a class="elsevierStyleCrossRefs" href="#bib0065"><span class="elsevierStyleSup">1&#8211;5</span></a> Provided the potential lethality of long-term infection of measles&#44; SSPE should be diagnosed early and reported&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">Of note is that MRI patterns of SSPE may mimic metabolic disorders&#46;<a class="elsevierStyleCrossRef" href="#bib0110"><span class="elsevierStyleSup">10</span></a> Symmetrical thalamic and brainstem involvement without affection of periventricular sub-cortical white matter and cortex is extremely rare in SSPE&#46;<a class="elsevierStyleCrossRefs" href="#bib0110"><span class="elsevierStyleSup">10&#8211;12</span></a> However&#44; neuroimaging findings of SSPE have typically a dynamic and evolving course&#46; And in this case&#44; though the cortical involvement was not evident on conventional imaging&#44; clinical picture suggested involvement of fronto-parietal and occipital cortices &#40;inattentiveness&#44; forgetfulness&#44; mood disturbances&#44; aggressive behavior&#44; apathy&#44; dyscalculia&#44; prosopagnosia&#44; and visual object agnosia&#41;&#46; Cortical and sub-cortical white matter intensity changes are expected to be visualized in the follow-up scans&#46; Symmetrical thalamic involvement made it difficult for us to diagnose the case as similar neuroimaging findings can be observed in many other neurological disorders&#46;<a class="elsevierStyleCrossRef" href="#bib0125"><span class="elsevierStyleSup">13</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">In closing&#44; SSPE is almost universally fatal and even symptomatic treatment does not help much leading to a disastrous end of a young productive life&#46; Measles vaccination is the only definite way to prevent SSPE until date hence all children and eligible adolescents should get it without hesitancy&#46; SSPE should be kept in the list of differential diagnoses&#44; while dealing a case of subacute-to-acute onset rapidly evolving cognitive decline with myoclonic jerks&#46;</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Ethics statement</span><p id="par0040" class="elsevierStylePara elsevierViewall">Informed written consent was obtained from the patient involved in this study&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Disclosures</span><p id="par0045" class="elsevierStylePara elsevierViewall">R&#46; Ghosh reports no disclosures relevant to the manuscript&#46;</p><p id="par0050" class="elsevierStylePara elsevierViewall">S&#46; Dubey reports no disclosures relevant to the manuscript&#46;</p><p id="par0055" class="elsevierStylePara elsevierViewall">A &#46;K&#46; Mukherjee reports no disclosures relevant to the manuscript&#46;</p><p id="par0060" class="elsevierStylePara elsevierViewall">J&#46; Benito-Le&#243;n reports no disclosures relevant to the manuscript&#46;</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Funding</span><p id="par0065" class="elsevierStylePara elsevierViewall">None&#46;</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0020">Conflict of interest</span><p id="par0070" class="elsevierStylePara elsevierViewall">The authors have no conflict of interest&#46;</p></span></span>"
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es en pt

¿Es usted profesional sanitario apto para prescribir o dispensar medicamentos?

Are you a health professional able to prescribe or dispense drugs?

Você é um profissional de saúde habilitado a prescrever ou dispensar medicamentos