Acute disseminated encephalomyelitis, a rare post-malaria neurological complication: Case report and review of the literature

Carreira, Joana; Casella, Maria Isabel; Ascenção, Bianca Branco; Luís, Nuno; Gonçalves, Ana Catarina; Brito, Ana Paula Santos Oliveira; Sá, Joana; Parreira, Mário; Lopes, Delfim; Poças, José

doi:10.1016/j.tmaid.2018.03.005

Cited by 13 publications

(12 citation statements)

References 26 publications

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“…PMNS after P. falciparum infections are the most frequently reported [ 4 ]. Noteworthy, most of the PMNS presenting as ADEM occurred after non-falciparum malaria [ 6 ], but the reason for this association is not clear.…”

Section: Discussionmentioning

confidence: 99%

“…Auto-reacting T-cells could be triggered by a certain grade of molecular mimicry and nonspecific activation, leading to autoimmune response toward CNS antigens [ 6 , 15 ]. This mechanism is also a well-known prerequisite of classical AIE, AIDP and ADEM.…”

Section: Discussionmentioning

confidence: 99%

“…For example, ADEM-like and PMNS-like patterns share multiple overlapping features, such as multifocal white matter lesions, at times spontaneous recovery, and good response to immune-suppressive therapy. To this extent, imaging is possibly more significant for detecting ADEM-like forms rather than PMNS-like ones [ 6 ]. Furthermore, both DCA and AIDP may occur as part of CM as well as in post-malaria illness.…”

Section: Introductionmentioning

confidence: 99%

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Clinical presentation and immunological features of Post-Malaria Neurologic Syndrome: a case report and review of literature

Castaldo

Tascini

Siega

et al. 2020

Malar J

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Background Malaria still represents a major health threat, in terms of both morbidity and mortality. Complications of malaria present a diversified clinical spectrum, with neurological involvement leading to the most serious related-conditions. The authors recently encountered a case of a 60-year old Italian man presenting with confusion, language disturbances and Parkinson-like syndrome 3 weeks after complete remission from severe Plasmodium falciparum cerebral malaria. Chemical and microbiological analysis revealed aseptic meningitis, diffuse encephalitis and abnormal immune-activation. Re-infection and recrudescence of infection were excluded. Further analysis excluded paraneoplastic and autoimmune causes of encephalitis. A diagnosis of Post-Malaria Neurological Syndrome (PMNS) was finally formulated and successfully treated with high dose of steroids. Methods A systematic research of current literature related to PMNS was performed. Results 151 cases of PMNS were included, the majority of which occurred after severe P. falciparum infections. Four main clinical pattern were identified: 37% of the cases presented as “classical” PMNS, 36% presented as delayed cerebellar ataxia (DCA), 18% resembled acute inflammatory demyelinating polyneuropathy (AIDP), and 8% presented as acute disseminated encephalomyelitis (ADEM)-like form. Differentiation between different forms was not always simple, as clinical and radiological findings frequently overlap. Overall, in almost all of the tested cases, cerebrospinal fluid was found pathological; EEG revealed nonspecific encephalopathy in 30% of classical PMNS and 67% ADEM; imaging tests were found abnormal in 92% of ADEM-like forms. Pathogenesis remains unclear. An autoimmune mechanism is the most corroborated pathogenic hypothesis. Overall, the majority of PMNS cases revert without specific treatment. In most severe forms, high dose steroids, intravenous immunoglobulins, and plasmapheresis have been shown to improve symptoms. Conclusions PMNS is a disabling complication of malaria. The overall incidence is not known, due to frequent misdiagnosis and under-reporting. Pathogenesis is not also fully understood, but rapid response to immune-modulating treatment along with similarities to auto-immune neurological disease, strongly support a dysregulated immunological genesis of this condition. The lack of randomized controlled studies regarding therapeutic approaches is a major unmet need in this setting. A systematic collection of all the PMNS cases would be desirable, in order to increase awareness of this rare condition and to prospectively investigate the most appropriate management.

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Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

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Clinical presentation and immunological features of Post-Malaria Neurologic Syndrome: a case report and review of literature

Castaldo

Tascini

Siega

et al. 2020

Malar J

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“…4 The PMNS with normal neuroimaging as in our case was reported by Zambito et al 11 The MRI brain has been found to be unremarkable in most of the patients who had non-ADEM-like presentation and bilateral cerebral white matter lesions in patients with ADEM. 12 Rarely abnormal signal changes in the brain stem, thalamus, corona radiata, internal capsule, cerebellum, and even in the spinal cord have been reported.…”

Section: Discussionmentioning

confidence: 99%

Case Report: An Adolescent Girl with Isolated Neuropsychiatric Features and Apparent Post-Malaria Neurological Syndrome

Panda

Sharawat

Panda³

2020

The American Journal of Tropical Medicine and Hygiene

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The post-malaria neurological syndrome (PMNS) is an unusual and relatively underreported complication of malaria, which usually occurs after the resolution of acute febrile illness and the patient is free from parasitemia. The clinical spectrum of the PMNS varies from acute-onset cerebellar ataxia to significant encephalopathy with focal deficits resembling acute disseminated encephalomyelitis. Uncommon presentations of PMNS include Guillain-Barre syndrome, postural tremor, or even isolated neuropsychiatric features. Although in a significant proportion of PMNS cases clinical resolution occurs with conservative treatment only, corticosteroids have been used in an attempt to hasten recoveries. Here, we present a case of a 12-year-old girl with acute onset, isolated neuropsychiatric features, following Plasmodium falciparum malaria. Neuroimaging, clinical examination, and cerebrospinal fluid studies were within normal limits. The child recovered completely after treatment with methylprednisolone pulse therapy. This case report illustrates the need for creating awareness about this uncommon complication of malaria. In view of the uncommon complication, early diagnosis and prompt treatment might help in the early resolution of symptoms.

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“…Viral triggers commonly associated with ADEM include in uenza virus, enterovirus, measles, mumps, rubella, varicella-zoster virus, Epstein-Barr virus, cytomegalovirus, hepatitis A and B and coxsackievirus (19,20). Parasitic infections, such as malaria or toxoplasmosis, have also been recently reported (21,22). Moreover, several bacterial triggers can occasionally play a role, including M. Pneumoniae, C. Pneumoniae, H. In uenzae, Salmonella, Borrelia burgdorferi, Leptospira, Legionella, Rickettsia, Campylobacter and β-hemolytic Streptococcus (19,(23)(24)(25).…”

Section: Introductionmentioning

confidence: 99%

Acute Disseminated Encephal Omyelitis (ADEM) After Pneumococcal Meningitis In A Child

Foiadelli¹,

Trabatti²,

Bassanese³

et al. 2020

Preprint

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Background: Acute disseminated encephalomyelitis (ADEM) is an inflammatory demyelinating disorder of the central nervous system (CNS), whose pathogenesis is still debated. It occurs more frequently in children, and the first neurological symptoms usually appear between 2 and 4 weeks after a trigger event, such as an infection or vaccination. Several viral agents have been related to the development of ADEM, while bacteria and parasites are less frequently involved. Severe Streptococcus pneumoniae infection has been rarely described as a trigger for CNS demyelination in particularly predisposed subjects.Case presentation: A 10 year old girl was evaluated for headache, fever and vomit. CSF analysis revealed pleocytosis and presence of S. pneumoniae antigen, and proper antibiotic therapy for bacterial meningitis was started, with rapid improvement. A breach in the right frontal bone, due to a car accident occurring the previous year, was considered the gateway for pneumococcal infection. Three days after admission, the girl developed drowsiness, altered speech and left hemiparesis. Brain MRI showed multiple T2-hyperintense bilateral lesions in the supratentorial white matter, and ADEM was diagnosed. Considering the underlying bacterial meningitis, intravenous immunoglobulins were preferred to steroid therapy, and the patient progressively recovered. However, due to recurrence of encephalopathic symptoms after 11 days, high-dose intravenous steroid therapy was performed. The neurological outcome was favourable, with complete regression of the white matter lesions after 4 months and absence of relapses over a follow-up period of 2 years.Conclusions: Occurrence of ADEM following pneumococcal meningitis is rare, and very few cases have been described in children. It should be suspected in case of persistence, recurrence or onset of new symptoms despite adequate antimicrobial treatment, relying on brain MRI for a thorough differential diagnosis. High level of surveillance is mandatory in patients with predisposing factors to invasive pneumococcal disease. In some cases, acute demyelination can occur few days after the onset of meningitis; pathogenetic mechanisms are not yet fully clarified and the choice of the correct therapeutic approach can be challenging.

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Acute disseminated encephalomyelitis, a rare post-malaria neurological complication: Case report and review of the literature

Cited by 13 publications

References 26 publications

Clinical presentation and immunological features of Post-Malaria Neurologic Syndrome: a case report and review of literature

Clinical presentation and immunological features of Post-Malaria Neurologic Syndrome: a case report and review of literature

Case Report: An Adolescent Girl with Isolated Neuropsychiatric Features and Apparent Post-Malaria Neurological Syndrome

Acute Disseminated Encephal Omyelitis (ADEM) After Pneumococcal Meningitis In A Child

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