Case Report: Vagal Nerve Neuritis Associated with Pulmonary Melioidosis Provides Potential Insights into the Pathophysiology of Neuromelioidosis

Rosie J. Matthews Department of Medicine, Cairns Hospital, Cairns, Australia;

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Simon Smith Department of Medicine, Cairns Hospital, Cairns, Australia;

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Ian Wilson Department of Medicine, Cairns Hospital, Cairns, Australia;

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Richard Tjahjono Department of Surgery, Cairns Hospital, Cairns, Australia;
Sydney Medical School, University of Sydney, Sydney, Australia;

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Soon Young Department of Radiology, Cairns Hospital, Cairns, Australia;

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Josh Hanson Department of Medicine, Cairns Hospital, Cairns, Australia;
The Kirby Institute, University of New South Wales, Sydney, Australia

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ABSTRACT.

Encephalomyelitis is the most frequent manifestation of neuromelioidosis in Australia. It is hypothesized that Burkholderia pseudomallei causes encephalomyelitis after entering the brain directly, if complicating a scalp infection, or after traveling to the brain within peripheral or cranial nerves. A 76-year-old man presented with fever, dysphonia, and hiccups. Chest imaging demonstrated extensive bilateral pneumonia with mediastinal lymphadenopathy, blood cultures isolated B. pseudomallei, and nasendoscopy confirmed a left vocal cord palsy. Magnetic resonance imaging identified no intracranial abnormality but demonstrated an enlarged, enhancing left vagus nerve, consistent with neuritis. We hypothesize that B. pseudomallei invaded the vagus nerve in the thorax, was traveling proximally—involving the left recurrent laryngeal nerve and causing the left vocal cord palsy, but had not yet reached the brainstem. Given the frequency of pneumonia in cases of melioidosis, the vagus nerve may represent an alternative, and indeed common, route for B. pseudomallei to enter the brainstem in cases of melioidosis-related encephalomyelitis.

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Author Notes

Address correspondence to Josh Hanson, The Kirby Institute, University of New South Wales, Level 6, Wallace Wurth Building, High St., Sydney, Kensington NSW 2052, Australia. E-mail: jhanson@kirby.unsw.edu.au

Authors’ addresses: Rosie J. Matthews, Simon Smith, and Ian Wilson, Department of Medicine, Cairns Hospital, Cairns, Australia, E-mails: rosie.matthews@health.qld.gov.au; simon.smith2@health.qld.gov.au; ian.wilson@health.qld.gov.au. Richard Tjahjono, Department of Surgery, Cairns Hospital, Cairns, Australia, and Sydney Medical School, University of Sydney, Sydney, Australia, E-mail: richardtjahjono@gmail.com. Soon Young, Department of Radiology, Cairns Hospital, Cairns, Australia, E-mail: soon.young@health.qld.gov.au. Josh Hanson, Department of Medicine, Cairns Hospital, Cairns, Australia, and The Kirby Institute, University of New South Wales, Sydney, Australia, E-mail: jhanson@kirby.unsw.edu.au.

  • 1.

    Gassiep I , Armstrong M , Norton R , 2020. Human melioidosis. Clin Microbiol Rev 33: e00006e00019.

  • 2.

    Currie BJ et al., 2021. The Darwin Prospective Melioidosis Study: a 30-year prospective, observational investigation. Lancet Infect Dis 21: 17371746.

  • 3.

    Stewart JD , Smith S , Binotto E , McBride WJ , Currie BJ , Hanson J , 2017. The epidemiology and clinical features of melioidosis in Far North Queensland: implications for patient management. PLoS Negl Trop Dis 11: e0005411.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 4.

    Gora H , Hasan T , Smith S , Wilson I , Mayo M , Woerle C , Webb JR , Currie BJ , Hanson J , Meumann EM , 2022. Melioidosis of the central nervous system; impact of the bimABm allele on patient presentation and outcome. Clin Infect Dis (in press). doi: 10.1093/cid/ciac111.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 5.

    Smith S , Hanson J , Currie BJ , 2018. Melioidosis: an Australian perspective. Trop Med Infect Dis 3: 27.

  • 6.

    Currie BJ , Fisher DA , Howard DM , Burrow JN , 2000. Neurological melioidosis. Acta Trop 74: 145151.

  • 7.

    McLeod C , Morris PS , Bauert PA , Kilburn CJ , Ward LM , Baird RW , Currie BJ , 2015. Clinical presentation and medical management of melioidosis in children: a 24-year prospective study in the Northern Territory of Australia and review of the literature. Clin Infect Dis 60: 2126.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 8.

    Liu PJ , Chen YS , Lin HH , Ni WF , Hsieh TH , Chen HT , Chen YL , 2013. Induction of mouse melioidosis with meningitis by CD11b+ phagocytic cells harboring intracellular B. pseudomallei as a Trojan horse. PLoS Negl Trop Dis 7: e2363.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 9.

    St John JA et al., 2014. Burkholderia pseudomallei penetrates the brain via destruction of the olfactory and trigeminal nerves: implications for the pathogenesis of neurological melioidosis. MBio 5: e00025.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 10.

    St John JA , Walkden H , Nazareth L , Beagley KW , Ulett GC , Batzloff MR , Beacham IR , Ekberg JA , 2016. Burkholderia pseudomallei rapidly infects the brain stem and spinal cord via the trigeminal nerve after intranasal inoculation. Infect Immun 84: 26812688.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 11.

    Lim WK , Gurdeep GS , Norain K , 2001. Melioidosis of the head and neck. Med J Malaysia 56: 471477.

  • 12.

    Mohanty S , Sarkar S , Mishra B , 2020. Melioidosis of the head and neck: a case series from Eastern India. Infect Dis Rep 12: 3645.

  • 13.

    Gadsby BA , Oaten A , Davies P , Simpson G , Vincent S , 2022. Mediastinal melioidosis: a case series from far North Queensland. Respirol Case Rep 10: e01017.

  • 14.

    Davies P , Smith S , Wilcox R , Stewart JD , Davis TJ , McKenna K , Hanson J , 2022. Examination of the independent contribution of rheumatic heart disease and congestive cardiac failure to the development and outcome of melioidosis in Far North Queensland, tropical Australia. PLoS Negl Trop Dis 16: e0010604.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 15.

    Berthoud HR , Neuhuber WL , 2000. Functional and chemical anatomy of the afferent vagal system. Auton Neurosci 85: 117.

  • 16.

    Canning BJ , Spina D , 2009. Sensory nerves and airway irritability. Handb Exp Pharmacol 194: 139183.

  • 17.

    Weijs TJ , Ruurda JP , Luyer MD , Nieuwenhuijzen GA , van Hillegersberg R , Bleys RL , 2015. Topography and extent of pulmonary vagus nerve supply with respect to transthoracic oesophagectomy. J Anat 227: 431439.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 18.

    Hanson J , Smith S , 2019. High rates of premature and potentially preventable death among patients surviving melioidosis in tropical Australia. Am J Trop Med Hyg 101: 328331.

    • PubMed
    • Search Google Scholar
    • Export Citation
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