Case report
A case of relapsing–remitting facial palsy and ipsilateral brachial plexopathy caused by HSV-1

https://doi.org/10.1016/j.jcv.2016.03.003Get rights and content

Highlights

  • To our knowledge, this is the first documentation of relapsing-remitting facial and brachial plexus neuritis caused by recurrent reactivation of HSV-1.

  • The recurrent multifocal peripheral neuronal involvement argues for an immune-mediated polyneuropathy, but the stereotype neurological symptoms confined to the same anatomical areas during each episode, and with short latency of HSV-1 reactivation, favor an infectious neuropathy.

  • Mannose-binding lectin deficiency (MBL) is associated with recurrent infections, but the role in patients with recurrent HSV-1 reactivation is still not known.

ABSTRACT

The etiologies of Bell’s palsy and brachial neuritis remain uncertain, and the conditions rarely co-occur or reoccur. Here we present a woman in her twenties who had several relapsing-remitting episodes with left-sided facial palsy and brachial neuropathy. The episodes always started with painful left-sided oral blisters. Repeat PCRs HSV-1 DNA from oral vesicular lesions were positive. Extensive screening did not reveal any other underlying cause. Findings on MRI T2-weighted brachial plexus STIR images, using a 3.0-Tesla scanner during an episode, were compatible with brachial plexus neuritis. Except a mannose-binding lectin deficiency, a congenital complement deficiency that is frequently found in the general Caucasian population, no other immunodeficiency was demonstrated in our patient. In vitro resistance to acyclovir was tested negative, but despite prophylactic treatment with the drug in high doses, relapses recurred. To our knowledge, this is the first ever reported documentation of relapsing-remitting facial and brachial plexus neuritis caused by HSV-1.

Section snippets

Why this case is important?

Over the years, several explanations for Bell’s palsy (BP) have been given, but the etiology remains uncertain. The reason may be that there is no common etiological factor [1], but a common etiological pathogenesis. Both viral and autoimmune mechanisms have been suggested [2]. Herpes simplex virus type 1 (HSV-1) was postulated as the causative agent over 40 years ago [3], but direct evidence for HSV-1 infection in patients with BP is still lacking. Likewise, brachial plexus neuritis (BPN) is

Case description

A 24-year-old Caucasian woman with polycystic ovary syndrome (PCOS) experienced in mid-August 2012 painful blisters on her tongue (middle lateral part) and inside her mouth (in the posterior part, outside the lower row of teeth in the mandibular gingivae) on the left side. A week later she woke up with an ipsilateral facial palsy accompanied with ear pain, facial numbness, and altered taste on the left side of the tongue. On August 27 she was hospitalized due to slight pain in her anterior left

Other similar and contrasting cases in the literature

There is quite sound evidence that VZV may cause both unilateral peripheral facial nerve palsy [2] and BPN [6], but we found only three cases of BP associated with documented herpes simplex gingivostomatitis in the litterature [7], [8], [9]. In a case series presented by Vahlne et al., one of the patients had overt herpes labialis during the BP [10]. Ghonmin reported two cases with bilateral BP following a recent presumed herpetic gingivostomatitis, but viral conformation was not documented [11]

Discussion

To our knowledge, this is the first reported association between HSV-1 reactivation and facial palsy combined with brachial plexopathy. Documented recurrent intraoral HSV is uncommon. The underlying predisposition to frequent reactivations is unknown, but low cell-mediated immunity probably plays an important role [13], and reduced MBL-mediated complement activation increases susceptibility to viral infections [14], including herpes [15], [16]. The patient’s PCOS may also be of importance.

Ethics

The patient read the text and gave informed consent to publishing it.

Ethical approval

Ethical approval was given by the patient safety deputy, reference number 12/2015.

Conflicts of interest

The authors have nothing to disclose.

Acknowledgment

This research received no specific grant from any funding agency in the public, commercial, or not-for-profit sectors.

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