http://www.ncbi.nlm.nih.gov/pmc/articles/PMC2994358/?tool=pubmed
JRSM Short Rep. 2010 November; 1(6): 56.
Published online 2010 November 25. doi: 10.1258/shorts.2010.010080.
PMCID: PMC2994358
Copyright © 2010 Royal Society of Medicine Press
A case of Lyme neuroborreliosis with bilateral recurrent laryngeal nerve
palsy
M Furtner,1 M Patjas,2 B Frauscher,1 E Schmutzhard,1 and J Willeit1
1Department of Neurology, Innsbruck Medical University, Innsbruck, Austria
2Department of Hearing, Voice, and Speech Disorders, Innsbruck Medical
University, Austria Correspondence to: Martin Furtner. E-mail:
martin.furtner@i-med.ac.at
This is an open-access article distributed under the terms of the Creative
Commons Attribution License
(http://creativecommons.org/licenses/by-nc/2.0/), which permits
non-commercial use, distribution and reproduction in any medium, provided
the original work is properly cited.
We report a case of neuroborreliosis with severe bilateral recurrent nerve
palsy, which had to be treated at an intensive care unit because of acute
respiratory distress.
Case history
A 66-year-old woman was transferred to our facility from a local hospital.
Her only remarkable prior condition was diabetes mellitus, for which she
was
on oral medication. She did not recall a tick bite or any skin changes
during the last months.
During the past 4 weeks, she had suffered severe lumbosacral and
cervicobrachial pain with multisegmental radiation (visual analog scale
8–10). Pain had responded insufficiently to non-steroidal analgesics and
opioids. Computed tomography (CT) scans of the cervical and lumbar spine
had
been unremarkable except for moderate spondylarthrosis. She had been
hospitalized for the past 4 days due to a severe bout of pain radiating
into
both legs, with nocturnal accentuation. During the 24 hours prior to
admission to our facility, leg weakness rapidly emerged and resulted in an
inability to stand, while pain had improved.
On admission (day 1), the patient presented with distally accentuated
paresis and slight muscular atrophy in arms and legs. Cranial nerves were
unremarkable, as were phonation and swallowing ability. Deep tendon
reflexes
were absent in the lower and reduced in the upper extremities.
She was unable to walk unaided, sensation in both hands and feet being
diminished.
Radicular or cord compression was ruled out by magnetic resonance (MR)
tomography of the entire spine.
Cerebrospinal fluid (CSF) analysis showed 57 leukocytes/?L (normal 0–4;
mainly activated lymphocytes, several plasma cells), elevated protein
content (103 mg/dL; normal 15–45) and IgG serum/liquor index 1.80 (normal
<0.50) but normal glucose concentration. CSF serology was positive for
Borrelia burgdorferi IgG and IgM (enzyme-linked immunosorbent assay [ELISA]
and immunoblot) but negative for other neurotropic agents. Numerous
oligoclonal bands were found. In the serum, B. burgdorferi IgG and IgM
antibodies were detectable as well.
Parameters for autoimmune diseases were negative.
A diagnosis of Lyme polyradiculitis was made and ceftriaxone 2 g IV once
daily was started. Despite treatment, the patient's condition further
worsened. On day 2, she developed asymmetrical tetraparesis (right arm
power
grade [PG] 3–4; left arm PG 4; right leg PG 3, left leg PG 2) and
incomplete
loss of sensation in all extremities. Electromyography confirmed a
generalized axonal neuropathy with reduced compound muscle action potential
amplitudes and loss of F waves. The most striking new symptom was dysphonia
with a soft and hoarse voice. Over the following
24 hours, the speech abnormality progressed to aphonia. Simultaneously,
dyspnoea developed and ambient-air peripheral oxygen saturation rapidly
decreased from 98% to 80%. Laryngoscopy on day 2 demonstrated bilateral
vocal cord paralysis, with both vocal cords fixed in a paramedian position
(online video). She was monitored at the neurological intensive care unit
(ICU) from days 3 to 6, and received nasal oxygen. Intubation as well as
tracheostomy were discussed, but did not become necessary.
Differential diagnosis regarding the clinical worsening at this point
included polyneuritis cranialis (Miller-Fisher syndrome), which could be
ruled out by negative antiganglioside antibodies, and brainstem
encephalitis, which was discarded after a normal contrast-enhanced brain MR
tomography.
Another CSF sample was taken on day 19. Abnormalities had partly resolved
(40 leukocytes/?L, protein 72 mg/dL, and IgG index 0.90); serology showed
positive B. burgdorferi IgG and borderline-positive IgM (ELISA and
immunoblot). CSF/serum Borrelia IgG index from this CSF sample was markedly
elevated at 7.5 (normal, <1.3).
Ceftriaxone was administered for three weeks. During this period, the
patient gradually recovered, and recurrent nerve function normalized.
Residual weakness (hands, PG 4; legs, PG 4-5) and slightly reduced
sensation
in both hands and feet persisted. The patient was admitted to a
rehabilitation facility. At follow-up after 6 months, no residual weakness,
in particular, no laryngeal abnormalities were found anymore.
Neuropathic pain mainly affecting the right leg was present and required
treatment with gabapentin.
Discussion
Lyme borreliosis is a tick-borne infectious disease caused by Borrelia
burgdorferi. It involves many organs (skin, heart, joints) and causes a
variety of neurological manifestations. Meningo-polyradiculoneuritis
(Bannwarth syndrome) with severe radicular pain and/or motor symptoms
represents the typical pattern in the acute stage. These signs and symptoms
take 4–6 weeks to develop, and as in our patient, may be bilateral.
However,
with antibiotic treatment, prognosis usually is benign, and progression to
tetraplegia is an infrequent event. The combination of radiculitis and
cranial neuritis is common (about 60% of patients). Cranial neuritides
mainly affect the facial nerve (in up to 60% of all neuroborreliosis
patients, bilateral in 40%), and the abducens nerve in 10%.1–3 Less
frequently, all other cranial nerves (except the olfactory) may be
affected.
Recurrent nerve palsy is extremely rare, with only six reported occurrences
(all unilateral), having caused a sore throat or hoarseness, but no
dyspnea.
Prognosis was good after antibiotic treatment in all cases.4–7 Our patient
presented with severe pain and tetraparesis due to Lyme polyradiculitis,
while recurrent nerve palsy occurred later. This case requires attention
because it is the first with bilateral vocal cord paralysis due to
affection
of both recurrent nerves. Accordingly, critical narrowing of the glottis
(online video) and dyspnoea ensued.
Clinical worsening was rapid, and intubation seemed imminent. Despite
immediate treatment with ceftriaxone, signs and symptoms initially
worsened.
There are relatively few reports on therapy efficacy or latency of response
in Lyme disease.8 In most cases, however, amelioration of symptoms,
especially pain, is seen promptly after antibiotic therapy. As in our
patient, response may sometimes be delayed for a few days.
It is interesting that in all six previously reported cases, recurrent
nerve
paralysis was an isolated symptom. A survey covering otolaryngologic
aspects
of Lyme disease found that 4.9% of patients reported hoarseness. There is
no
mention, however, if this was associated with vocal cord dysfunction.9 The
relatively long duration (four weeks) of sensory radicular symptoms before
the rapid occurrence of tetraparesis and vocal cord paralysis within 72
hours deserves comment. In the literature, variable onset of paresis has
been reported, which may indeed occur after several weeks of sensory
symptoms.2,3 Summing up, this report intends to raise awareness that Lyme
disease may cause bilateral recurrent nerve palsy with respiratory
dysfunction.
Prognosis may be favorable upon expeditious antibiotic treatment despite
severe initial deficits.
Comment on the online video
The examiner (PM) is telling the patient to phonate (say 'hee'). There is
bilateral vocal cord paralysis, with both vocal cords fixed in a paramedian
position. The video can be seen at
http://shortreports.rsmjournals.com/cgi/content/full/shorts.2010.010080/DC1.
DECLARATIONS
Competing interests
None declared
Funding
None
Ethical approval
Written informed consent to publication has been obtained from the patient
or next of kin
Guarantor
MF
Contributorship
All authors were responsible for patient care; MF was responsible for
writing the article; MP was responsible for the online video; BF, ES and JW
were responsible for the critical reading of the manuscript
Acknowledgements
The online video is provided with permission by M Zorowka (Department of
Hearing, Voice and Speech Disorders, Innsbruck Medical University)
Reviewer
Lionel Martzolff
References
1. Schmutzhard E, Stanek G, Pohl P Polyneuritis cranialis associated with
Borrelia burgdorferi. J Neurol Neurosurg Psychiatry 1985;48:1182–4 [PMC
free
article] [PubMed] 2. Steere AC Lyme disease. N Engl J Med 1989;32:586–96
[PubMed] 3. Coyle PK, Schutzer SE Neurologic aspects of Lyme disease. Med
Clin North Am 2002;86:261–84 [PubMed] 4. Karosi T, Rácz T, Szekanecz E,
Toth
A, Sziklai I Recurrent laryngeal nerve paralysis due to subclinical Lyme
borreliosis. J Laryngol Otol
2010;124:336–8 [PubMed]
5. Martzolff L, Bouhala M, Dukic R, et al. [Recurrent nerve palsy due to
Lyme disease: report of two cases]. Rev Med Interne 2010;31:229–31 [PubMed]
6. Neuschaefer-Rube C, Haase G, Angerstein W, Kremer B [Unilateral
recurrent
nerve paralysis in suspected Lyme borreliosis]. HNO 1995;43:188–90 [PubMed]
7. Schroeter V, Belz GG, Blenk H Paralysis of recurrent laryngeal nerve in
Lyme disease. Lancet 1988;2:1245. [PubMed] 8. Halperin JJ, Shapiro ED,
Logigian E, et al. ; Quality Standards Subcommittee of the American Academy
of Neurology. Practice parameter:
treatment of nervous system Lyme disease (an evidence-based review):
report of the Quality Standards Subcommittee of the American Academy of
Neurology. Neurology 2007;69:91–102 [PubMed] 9. Moscatello AL, Worden DL,
Nadelman RB, Wormser G, Lucente F Otolaryngologic aspects of Lyme disease.
Laryngoscope 1991;101:592–5 [PubMed]
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