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

Elia Manfrini; Ludovica Falcioni; Vanna Cavassa; Eleonora Cocco; Stefano Sotgiu; Luca Saba

doi:10.1055/s-0043-1772574

Journal of Pediatric Neurology, Table of Contents

Journal of Pediatric Neurology 2023; 21(06): 445-449
DOI: 10.1055/s-0043-1772574

Case Report

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

Authors

Elia Manfrini

¹Department of Radiology, University of Cagliari, Cagliari, Italy
Ludovica Falcioni

²Unit of Child and Adolescent Neuropsychiatry, Department of Biomedical Sciences, University of Cagliari, Cagliari, Italy

³Child Neuropsychiatry Unit, Department of Medicine, Surgery and Pharmacy, University Hospital of Sassari, Sassari, Italy
Vanna Cavassa

²Unit of Child and Adolescent Neuropsychiatry, Department of Biomedical Sciences, University of Cagliari, Cagliari, Italy

³Child Neuropsychiatry Unit, Department of Medicine, Surgery and Pharmacy, University Hospital of Sassari, Sassari, Italy
Eleonora Cocco

⁴Department of Medical Sciences and Public Health, Multiple Sclerosis Center, Binaghi Hospital, University of Cagliari, Cagliari, Italy
Stefano Sotgiu

³Child Neuropsychiatry Unit, Department of Medicine, Surgery and Pharmacy, University Hospital of Sassari, Sassari, Italy
Luca Saba

¹Department of Radiology, University of Cagliari, Cagliari, Italy

Abstract

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.

Keywords

magnetic resonance imaging - multiple sclerosis - combined central and peripheral demyelination - child - trigeminal nerve

Full Text

References

References
1 Wallin MT, Culpepper WJ, Nichols E. et al; GBD 2016 Multiple Sclerosis Collaborators. Global, regional, and national burden of multiple sclerosis 1990-2016: a systematic analysis for the Global Burden of Disease Study 2016. Lancet Neurol 2019; 18 (03) 269-285
2 Yan K, Balijepalli C, Desai K, Gullapalli L, Druyts E. Epidemiology of pediatric multiple sclerosis: a systematic literature review and meta-analysis. Mult Scler Relat Disord 2020; 44: 102260
3 Frahm N, Peters M, Bätzing J. et al. Prevalence of pediatric multiple sclerosis in Germany: a nationwide population-based analysis. Eur J Neurol 2021; 28 (09) 3173-3176
4 Boesen MS, Jensen PEH, Born AP. et al. Incidence of pediatric neuromyelitis optica spectrum disorder and myelin oligodendrocyte glycoprotein antibody-associated disease in Denmark 2008‒2018: a nationwide, population-based cohort study. Mult Scler Relat Disord 2019; 33: 162-167
5 Marrie RA, O'Mahony J, Maxwell C. et al; Canadian Pediatric Demyelinating Disease Network. Incidence and prevalence of MS in children: a population-based study in Ontario, Canada. Neurology 2018; 91 (17) e1579-e1590
6 Dilokthornsakul P, Valuck RJ, Nair KV, Corboy JR, Allen RR, Campbell JD. Multiple sclerosis prevalence in the United States commercially insured population. Neurology 2016; 86 (11) 1014-1021
7 Dell'Avvento S, Sotgiu MA, Manca S, Sotgiu G, Sotgiu S. Epidemiology of multiple sclerosis in the pediatric population of Sardinia, Italy. Eur J Pediatr 2016; 175 (01) 19-29
8 Thompson AJ, Banwell BL, Barkhof F. et al. Diagnosis of multiple sclerosis: 2017 revisions of the McDonald criteria. Lancet Neurol 2018; 17 (02) 162-173
9 Nakashima I, Fujihara K, Kimpara T, Okita N, Takase S, Itoyama Y. Linear pontine trigeminal root lesions in multiple sclerosis: clinical and magnetic resonance imaging studies in 5 cases. Arch Neurol 2001; 58 (01) 101-104
10 da Silva CJ, da Rocha AJ, Mendes MF, Maia Jr AC, Braga FT, Tilbery CP. Trigeminal involvement in multiple sclerosis: magnetic resonance imaging findings with clinical correlation in a series of patients. Mult Scler 2005; 11 (03) 282-285
11 van der Meijs AH, Tan IL, Barkhof F. Incidence of enhancement of the trigeminal nerve on MRI in patients with multiple sclerosis. Mult Scler 2002; 8 (01) 64-67
12 Gass A, Kitchen N, MacManus DG, Moseley IF, Hennerici MG, Miller DH. Trigeminal neuralgia in patients with multiple sclerosis: lesion localization with magnetic resonance imaging. Neurology 1997; 49 (04) 1142-1144
13 Filippi M, Preziosa P, Banwell BL. et al. Assessment of lesions on magnetic resonance imaging in multiple sclerosis: practical guidelines. Brain 2019; 142 (07) 1858-1875
14 Noory N, Smilkov EA, Frederiksen JL. et al. Neurovascular contact plays no role in trigeminal neuralgia secondary to multiple sclerosis. Cephalalgia 2021; 41 (05) 593-603
15 Oudejans E, Luchicchi A, Strijbis EMM, Geurts JJG, van Dam AM. Is MS affecting the CNS only? Lessons from clinic to myelin pathophysiology. Neurol Neuroimmunol Neuroinflamm 2020; 8 (01) e914
16 Zakrzewska JM, Wu J, Brathwaite TSL. A systematic review of the management of trigeminal neuralgia in patients with multiple sclerosis. World Neurosurg 2018; 111: 291-306
17 Cortese A, Franciotta D, Alfonsi E. et al. Combined central and peripheral demyelination: Clinical features, diagnostic findings, and treatment. J Neurol Sci 2016; 363: 182-187
18 Ogata H, Matsuse D, Yamasaki R. et al. A nationwide survey of combined central and peripheral demyelination in Japan. J Neurol Neurosurg Psychiatry 2016; 87 (01) 29-36
19 Alshamrani F, Alyami R, Alghanimi I, Alajaji R, Alkhaldi M, Alamri A. A new report of combined central and peripheral demyelination: a case report. Front Neurol 2021; 12: 730129