Neuromyelitis optica spectrum disorders in South of Tunisia: A rare entity with low seroprevalence of anti-aquaporin 4 autoantibodies

Mejdoub, Sabrina; Feki, S.; Dammak, Mariem; Farhat, Nouha; Hdiji, Olfa; Boukthir, S.; Hachicha, H.; Mhiri, Chokri; Masmoudi, Hatem

doi:10.1016/j.neurol.2019.08.009

Cited by 7 publications

(13 citation statements)

References 45 publications

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“…The NMO-to-MS ratio is lower in North African countries (9:67, 36 64:503, 42 20:330, 37 8:877 38 ) and higher in studied populations from sub-Saharan Africa (2:0 29 , 4:3 41 , 95:2 43 ).…”

Section: Demographic Characteristicsmentioning

confidence: 75%

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Neuromyelitis Optica Spectrum Disorders in Africa

Musubire

Derdelinckx

Reynders

et al. 2021

Neurol Neuroimmunol Neuroinflamm

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Background and ObjectivesNeuromyelitis optica (NMO) is a CNS inflammatory disease that predominantly affects the optic nerves and the spinal cord. It is more frequent in Asian and African populations than in European ones. Data on epidemiology, clinical presentation, additional investigations, and treatment in the African continent are scarce. We aim to (1) collect and analyze published data on neuromyelitis optica spectrum disorder (NMOSD), (2) indicate challenges in the diagnosis and management, and (3) discuss opportunities for future research, education, and policy making, specifically on the African continent.MethodsA systematic review was performed in January 2021 with the search terms “Neuromyelitis optica and Africa,” “Devic Disease and Africa,” and “NMOSD and Africa.” We included all study types except case reports, correspondence, or conference abstracts on NMO or NMOSD. Extracted data included study design, country, study period, demographic and clinical characteristics, results of paraclinical investigations, and outcome. Data analysis was performed with descriptive statistics.ResultsWe retrieved a total of 79 records, of which 19 were included. Ten of 54 African countries reported a total of 410 cases. Almost half of them were from North African countries. The mean age at diagnosis was 33 years (range 7–88 years), and 75% were female. Transverse myelitis followed by optic neuritis were the most frequent symptoms at the time of presentation. One hundred nineteen patients experienced at least 1 previous relapse, and 106 had a relapsing course after diagnosis. Relapses were treated with IV methylprednisolone. Azathioprine and steroids were used most often as maintenance treatments. Outcomes were rarely described.DiscussionThe majority of studies on NMOSD from the African continent are retrospective, and most countries do not report any data. Our systemic review shows that data derived from patients living in Africa correspond well to what has been previously published in meta-analyses on patients of African ancestry with NMOSD who live outside of Africa, except for a younger age at onset and a lower proportion of females. We advocate for systematic data collection to adequately capture and monitor the burden of NMOSD, for expansion of research efforts and facilities to perform fundamental and clinical research, and for improved access to health care including diagnostics, treatments, and rehabilitation services for people affected by NMOSD in the African continent.

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“…The NMO-to-MS ratio is lower in North African countries (9:67, 36 64:503, 42 20:330, 37 8:877 38 ) and higher in studied populations from sub-Saharan Africa (2:0 29 , 4:3 41 , 95:2 43 ).…”

Section: Demographic Characteristicsmentioning

confidence: 75%

“…37,41,42,44,47,48,[50][51][52] Visual evoked potentials (VEP) were abnormal in the majority of the patients in whom they were performed. 36,38,39,44,48,51,52 Optical coherence tomography was mentioned only in 1 study and was abnormal in a patient with recurrent ON. 38…”

Section: Clinical Presentation and Comorbiditiesmentioning

confidence: 99%

Neuromyelitis Optica Spectrum Disorders in Africa

Musubire

Derdelinckx

Reynders

et al. 2021

Neurol Neuroimmunol Neuroinflamm

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“…The diagnosis of NMOSD was made in 7 seropositive patients and 2 seronegative patients by testing for anti-AQP4 antibodies in 170 consecutive Tunisian patients with inflammatory disease of the central nervous system and optic cord involvement. This resulted in a seroprevalence of 77.7% in the NMOSD subgroup [ 7 ]. Li et al's studies found that during NMOSD, microglia could be activated by interleukin- (IL-) 6 and type I interferons (IFN-Is), leading to signal transducer and activator of transcription (STAT) activation.…”

Section: Introductionmentioning

confidence: 99%

[Retracted] Correlation of IgH‐CDR3 Immune Repertoire Diversity Test in Peripheral Blood of Neuromyelitis Spectrum Diseases

Peng

Deng

2022

BioMed Research International

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Neuroretinitis spectrum disorder (NMOSD) is generally regarded as an acute or subacute inflammatory demyelinating disease of the central nervous system, mainly involving the optic nerve and spinal cord, mediated by humoral immunity. To address these questions, this work established an immunomic library of the heavy chain complementarity determinant 3 (gHI-CDR3) of peripheral blood lymphocyte B cell receptors. The library was established in patients with a spectrum of neurosyphilis-retinitis disorders. Six NMOSD patients and six healthy volunteers were recruited, and the NMOSD group was divided into an early-onset group and a stable group according to treatment conditions. The IgH-CDR3 gene fragment cultured in vitro was amplified by multiplex PCR technology, and the gene was sequenced by the second-generation high-throughput sequencing technology, and the statistical analysis was carried out by the method without reference. The quantity, type, and diversity of IgH-CDR3 in peripheral blood B lymphocytes of NMOSD patients were significantly lower than those of normal group ( P > 0.05 ); the variation of IgH-CDR3 sequence in the initial stage of treatment was higher than that in the stable stage ( P > 0.05 ); the replication frequency of the characteristic gene “CASSICLGSGCGGYYYGMDVW” was significantly increased in patients at the initial stage of NMOSD treatment ( P < 0.05 ). The conclusion was that the gene expression and gene expression analysis of NMOSD patients could accurately judge the condition of NMOSD patients, evaluate their efficacy, and provide new molecular targets and new theoretical basis for clinical application.

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“…Variations in prevalence have been described among different geographic areas and ethnicities [ 1 ]. Prevalence estimates range from 0.34 to 10/100,000 in adults [ 1 ].The frequency of NMOSD diagnosis was 5.3 % in 170 Tunisian patients with a context of optic neuritis (ON) and/or myelitis [ 2 ]. Six core clinical characteristics are identified in NMOSD, including optic neuritis, acute myelitis (mainly longitudinally extensive transverse myelitis, LETM), area postrema syndrome, acute brainstem, and diencephalic or cerebral syndromes.…”

Section: Introductionmentioning

confidence: 99%

Neuromyelitis optica and concomitant pulmonary tuberculosis: a case report

et al. 2021

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Background Concomitant diagnosis of neuromyelitis optica spectrum disease and pulmonary tuberculosis has rarely been reported. Case report We report a case involving a young Tunisian male patient who developed dry cough followed, 2 months later, by weakness in the lower limbs. The findings of central nervous system imaging and anti-aquaporin-4 antibody positivity were compatible with the diagnosis of neuromyelitis optica spectrum disease. Constellation of the clinical and the typical radiological pulmonary findings in our patient, coming from an endemic region, allowed the diagnosis of pulmonary tuberculosis, although sputum smear examination for acid-fast bacilli and cultures was negative. The patient received anti-tuberculous polytherapy associated with immunomodulation, consisting of methylprednisolone and intravenous immunoglobulins. Pulmonary infection symptoms initially improved but with no motor recovery. The patient suddenly died at home 4 months after the onset of the first symptoms. Current data regarding the clinical presentation of this underreported concomitant or associated condition, the possible pathophysiological mechanisms, and the therapeutic options were reviewed. Conclusions This case underscores the necessity to understand the exact mechanism of these coincident entities and to clarify the best immunomodulatory choice since immunosuppression targeting neuromyelitis optica spectrum disease can lead to dissemination of pulmonary tuberculosis.

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Neuromyelitis optica spectrum disorders in South of Tunisia: A rare entity with low seroprevalence of anti-aquaporin 4 autoantibodies

Cited by 7 publications

References 45 publications

Neuromyelitis Optica Spectrum Disorders in Africa

Neuromyelitis Optica Spectrum Disorders in Africa

[Retracted] Correlation of IgH‐CDR3 Immune Repertoire Diversity Test in Peripheral Blood of Neuromyelitis Spectrum Diseases

Neuromyelitis optica and concomitant pulmonary tuberculosis: a case report

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