A New Report of Combined Central and Peripheral Demyelination: A Case Report

Alshamrani, Foziah; AlYami, Rawan S; Alghanimi, Ibrahim; Alajaji, Reef; Alkhaldi, Modhi; Al-Amri, Abdulla

doi:10.3389/fneur.2021.730129

Cited by 6 publications

(12 citation statements)

References 15 publications

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“…Though CIDP is a disorder of the peripheral nervous system resulting from the deterioration of the myelin sheath, sporadic case reports of combined central and peripheral demyelination (CCPD) have been reported. CCPD is a large term that was proposed to describe a situation associated with demyelinating lesions in both the central and peripheral systems (CNS and PNS) ( 51 , 52 ). The results of our study prove that, in typical CIDP, there is also brain involvement, and metabolic changes may be detected even in the normal-appearing precentral gyri.…”

Section: Discussionmentioning

confidence: 99%

Central nervous system involvement in chronic inflammatory demyelinating polyradiculoneuropathy—MRS and DTI study

Dziadkowiak,

Koszewicz,

Podgórski

et al. 2024

Front. Neurol.

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ObjectiveThe current research aimed to analyze the alterations within the motor cortex and pyramidal pathways and their association with the degree of damage within the peripheral nerve fibers in patients with chronic inflammatory demyelinating polyradiculoneuropathy (CIDP). To achieve that goal, we investigated the microstructural changes within the pyramidal white matter tracts using diffusion tensor imaging (DTI) parameters, evaluated metabolic alterations in both precentral gyri using magnetic resonance spectroscopy (MRS) ratios, and correlated them with the neurographic findings in patients with CIDP.MethodsThe spectroscopic ratios of NAA/Cr, Cho/Cr, and mI/Cr from both precentral gyri and the values of fractional anisotropy (FA), axial diffusivity (AD), and mean diffusivity (MD) from both of the corticospinal tracts were correlated with the results of neurological and neurographic findings. The comparison of DTI parameters between the patients and controls was performed using Student’s t-test or the Mann–Whitney U test. Due to the lack of normal distribution of most variables, Spearman’s Rho rank coefficient was used to test all correlations. All analyses were performed at a significant level of alpha = 0.05 using STATISTICA 13.3.ResultsCompared to the control group (CG), the patient group showed significantly lower ratios of NAA/Cr (1.66 ± 0.11 vs. 1.61 ± 0.15; p = 0.022), higher ratios of ml/Cr in the right precentral gyrus (0.57 ± 0.15 vs. 0.61 ± 0.08; p = 0.005), and higher levels of Cho/Cr within the left precentral gyrus (0.83 ± 0.09 vs. 0.88 ± 0.14, p = 0.012). The DTI parameters of MD from the right CST and AD from the right and left CSTs showed a strong positive correlation (0.52–0.53) with the sural sensory nerve action potential (SNAP) latency of the right sural nerve. There were no other significant correlations between other DTI and MRS parameters and neurographic results.SignificanceIn our study, significant metabolic alterations were found in the precentral gyri in patients with CIDP without clinical symptoms of central nervous system involvement. The revealed changes reflected neuronal loss or dysfunction, myelin degradation, and increased gliosis. Our results suggest coexisting CNS damage in these patients and may provide a new insight into the still unknown pathomechanism of CIDP.

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Section: Discussionmentioning

confidence: 99%

Central nervous system involvement in chronic inflammatory demyelinating polyradiculoneuropathy—MRS and DTI study

Dziadkowiak,

Koszewicz,

Podgórski

et al. 2024

Front. Neurol.

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“…Multiple studies describing patients diagnosed with MS with a concurrent peripheral nervous system (PNS) lesion have been found. [9][10][11][12][14][15][16][17][18][19] Retrospective studies indicate that trigeminal involvement due to MS demyelination accounts for a small percentage of patients, ranging from 2.8 to 6.8%. 9,11 This prevalence is further supported by a recent systematic review, which confirms a value of $6%.…”

Section: Discussionmentioning

confidence: 99%

“…We searched on PubMed with the following search words: “Multiple sclerosis,” “Combined Central and Peripheral Demyelination,” “Trigeminal Nerve,” “Central Nervous System,” and “Magnetic Resonance Imaging.” Multiple studies describing patients diagnosed with MS with a concurrent peripheral nervous system (PNS) lesion have been found. 9 10 11 12 14 15 16 17 18 19…”

Section: Discussionmentioning

confidence: 99%

“…Although documented cases of combined central and peripheral demyelination exist, our understanding of this phenomenon is limited to case reports or small case series. [10][11][12][17][18][19] Further studies are necessary to elucidate the occurrence of PNS lesions in MS and provide deeper insights into their underlying pathogenic mechanisms.…”

Section: Conflict Of Interestmentioning

confidence: 99%

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A Rare Occurrence of Demyelinating Lesions of Bilateral Trigeminal Nerves: An Atypical Presentation of Pediatric Multiple Sclerosis

Manfrini,

Falcioni,

Cavassa

et al. 2023

Journal of Pediatric Neurology

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Pontine trigeminal root entry zone is a typical, although uncommon, location for multiple sclerosis (MS) lesions to occur. Here, we present a 17-year-old girl with nausea, vomiting, and vertigo. Neurological examination was consistent with central nystagmus, positive Romberg's test, and left-sided hyperreflexia. Baseline magnetic resonance imaging fulfilled McDonald 2017 criteria for MS and showed T2-hyperintense and T1-hypointense bilateral demyelinating lesions at the intramedullary portion of the trigeminal root of the fifth nerve, with no contrast enhancement or restricted diffusion. Bilateral intrapontine trigeminal involvement is a rare finding in MS, as well as the combined central and peripheral demyelination. Furthermore, very limited information and cases have been described in pediatric patients.

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“…Under CCPD, demyelinating changes occur simultaneously or successively in the peripheral nervous system and the central nervous system, which is rare in the clinic. CCPD is a pedigree disease and cannot be considered a simple combination of MS and CIDP [10][11][12][13][14][15]. At present, there is no unified diagnostic standard for CCPD.…”

Section: Introductionmentioning

confidence: 99%

Untitled

2022

J. Multiscale Neurosci.

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We report the diagnostic process of a rare patient with a combined central and peripheral demyelinating disease with antibodies against paranodal protein neurofascin 186 (anti-NF186)-the patient presented with weakness in both lower limbs. The clinical process and evidence are similar to subacute combined degeneration. However, thoracic spinal MRI shows T1 equal signal, and T2 high signal lesions in thoracic vertebrae 5-7 mainly involve white matter. The Aquaporin 4 antibody of the central nervous system indicates the disease is of an optic neuromyelitis pedigree. The antibodies against paranodal protein neurofascin 186 indicate demyelinating peripheral nerve pathogenesis. The final diagnosis was a combined central and peripheral demyelinating disease based on the results of various examinations. The patient's condition improved after methylprednisolone therapy and intravenous immunoglobulin.

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A New Report of Combined Central and Peripheral Demyelination: A Case Report

Cited by 6 publications

References 15 publications

Central nervous system involvement in chronic inflammatory demyelinating polyradiculoneuropathy—MRS and DTI study

Central nervous system involvement in chronic inflammatory demyelinating polyradiculoneuropathy—MRS and DTI study

A Rare Occurrence of Demyelinating Lesions of Bilateral Trigeminal Nerves: An Atypical Presentation of Pediatric Multiple Sclerosis

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