Lateral Sinus Thrombosis Associated With Zoster
Sine Herpete
James Chan, MD, Richard T. Bergstrom, MD, Donald C. Lanza, MD, and
John G. Oas, MD
Herpes zoster results from reactivation of the varicella zoster virus (VZV). Zoster sine herpete
(ZSH) is an uncommon manifestation of VZV infection and presents with similar symptoms
but without the vesicular rash. We describe an unusual case of lateral sinus thrombosis (LST)
that developed during the clinical course of ZSH in the C2 distribution. A 55-year-old woman
presented with a 3-day history of left temporal and postauricular pain, nausea, vomiting, and
mild photophobia. She denied otalgia, otorrhea, and hearing loss. Examination revealed
hyperesthesia in the left C2 nerve root distribution without evidence of herpetic rash. A
computed tomography scan showed minimal fluid in the left mastoid cavity (not mastoiditis)
and thrombus within the left lateral and sigmoid dural sinus. Magnetic resonance imaging
and magnetic resonance angiogram confirmed these findings. Laboratory studies revealed
elevated neurotrophic immunoglobulin G levels to VZV. Hypercoagulable studies were
normal. She was subsequently treated with Neurontin, acyclovir, and anticoagulation. Her
symptoms improved, and she was discharged 3 days later. LST is generally a complication
of middle ear infection. Nonseptic LST, however, may result from dehydration, oral contraceptive use, coagulopathy, or thyroid disease. This unusual case raises the suspicion that
thrombosis resulted from VZV associated thrombophlebitis in the ipsilateral cerebral venous
sinuses along the second cervical nerve root distribution. A high index of suspicion is
necessary in such cases so that a different treatment course can be identified and antiviral
medication initiated promptly.
(Am J Otolaryngol 2004;25:357-360. © 2004 Elsevier Inc. All rights reserved.)

Zoster sine herpete (ZSH) is a recognized
clinical variant of herpes zoster and presents
with painful neuralgia, without vesicular lesions, in a dermatomal distribution. Herpes
zoster infections involving the cerebral vasculature have been described and confirmed by
postmortem examinations.1 Anatomic and
histologic studies show neuronal innervation
of cerebral vasculature by cervical nerves.2,3
Furthermore, pontine infarction as a result of
reactivation of the zoster virus within brainstem vasculature has been described.4,5 Our
aim is to report an unusual case of lateral
sinus thrombosis associated with cervical

From the Department of Otolaryngology and Communicative Disorders, The Cleveland Clinic Foundation,
Cleveland, OH.
Presented at the American Academy of Otolaryngology–Head and Neck Surgery Annual Meeting, September 21-24, 2003, Orlando, FL.
Address correspondence to: John G. Oas, MD, The
Cleveland Clinic Foundation, A71, 9500 Euclid Avenue,
Cleveland, OH 44195. E-mail: oasj@ccf.org.
© 2004 Elsevier Inc. All rights reserved.
0196-0709/$ - see front matter
doi:10.1016/j.amjoto.2004.03.007

ZSH and provide a pertinent review of the
literature to support this diagnosis. Recognizing this rare entity can lead to initiation of
appropriate therapy in a timely manner.
CASE REPORT
A 55-year-old woman presented with a
3-day history of left temporal and postauricular pain, nausea, vomiting, and mild photophobia. She denied otalgia, otorrhea, and
hearing loss. Examination revealed hyperesthesia in the left C2 nerve root distribution
without evidence of herpetic rash. A computed tomography (CT) scan showed minimal
fluid in the left mastoid cavity (not mastoiditis) and thrombus within the left lateral and
sigmoid dural sinus (Figs 1 and 2A and B).
Magnetic resonance imaging (MRI) and magnetic resonance angiogram confirmed these
findings (Figs 3A and B). Laboratory studies
revealed elevated neurotrophic immunoglobulin G levels to varicella zoster virus (VZV).
Hypercoagulable studies were normal. She
was subsequently treated with Neurontin,

American Journal of Otolaryngology, Vol 25, No 5 (September-October), 2004: pp 357-360

357

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CHAN ET AL

Cerebral Vascular Infarction Associated
With Herpes Zoster
Several authors have suggested that cervical
herpes zoster can lead to cerebral vascular
infarction. Patrick et al4 reported a patient
with cervical herpes zoster infection followed
by pontine infarction. They proposed a mechanism whereby varicella zoster was reactivated within the dorsal root ganglia along
cervical nerve pathways to the vertebrobasilar
system, which led to angiitis and infarction

Fig 1. Axial CT scan with contrast showing minimal
fluid in the left mastoid cavity.

acyclovir, and anticoagulation. Her symptoms
improved, and she was discharged 3 days
later.
DISCUSSION
The present case is unusual in that the patient developed idiopathic lateral sinus
thrombosis with unilateral C2 dermatomal
hypesthesia and dysthesia ipsilateral to the
lateral sinus thrombosis. However, cutaneous
herpetic lesions were absent. Serologic studies confirmed reactivation of VZV. After thorough evaluation for other causes, the constellation of findings suggests that the lateral
sinus thrombosis is associated with ZSH.
ZSH
Herpes zoster (shingles) is characterized by
cutaneous or mucosal herpetic lesions distributed in a unilateral segmental dermatome.
Painful neuralgia is typically present. It is
thought to be the result of reactivation of VZV
within the dorsal root ganglia. A clinical variant is ZSH, which is also caused by reactivation of VZV leading to radicular pain but
without the accompanying rash. A number of
clinical studies support the existence of this
syndrome.6-8

Fig 2. Coronal CT scan showing minimal mucosal
thickening and opacification of air cells in the (A) left
mastoid as compared with the (B) right.

SINUS THROMBOSIS ASSOCIATED WITH ZOSTER

359

staining within the media of the cerebral arteries. Their results provide supporting evidence that the vasculopathy after shingles results from varicella zoster reactivation within
the cerebral artery leading to in situ thrombosis.1
Anatomic Support of Neural Innervation
of Dural Sinuses
Detailed immunohistochemical studies provide evidence that dural sinuses are innervated by nerve fibers. Barrett et al2 used
a highly specific fluorescence microscopic
technique to show the localization of neurotransmitters in nerve cell bodies and their
terminals within cerebral blood vessels. Investigators have also shown that the upper
cervical nerve roots invest the vertebrobasillar
system. Anatomic and histologic studies in
cats, by Saito and Moskowitz,3 show that cervical nerves innervate the vasculature of the
circle of Willis. It is therefore possible that the
relationship between the cutaneous symptoms and the dural sinus thromboses is a
result of retrograde viral migration from the
dorsal root ganglia via cervical nerve roots.
Lateral Sinus Thrombosis Associated
With ZSH

Fig 3. (A) Coronal MRI with and without gadolinium
and (B) MRA showing occlusion of the lateral sinus with
thrombus formation.

of the posterior circulation. Willeit and
Schmutzard5 described a patient with cervical
herpes zoster and delayed brainstem infarction and suggested that reactivation of VZV
induced cerebral arteritis.5
Pathological Evidence of Herpes Zoster
Pathological studies have shown large-vessel vasculopathy after herpes zoster infection.
Eidelberg et al1 presented the postmortem examination and brain biopsy of 2 patients with
thrombotic occlusions after segmental herpes
zoster infection. Varicella zoster viral antigens
were also detected by immunoperoxidase

Otolaryngologists are often involved in the
care of patients with lateral sinus thrombosis
(LST). It is usually the result of a complication
of otitis media and mastoiditis. This case
shows the importance of a thorough physical
examination. Although the MRI study revealed fluid in the mastoids, our clinical examination was inconsistent with mastoiditis.
Serological data provided further evidence of
reactivation of the varicella virus. Based on
the constellation of symptoms and findings,
we hypothesize that ZSH is the etiology of the
LST in our patient. Recognition of this condition can lead to implementation of appropriate therapy in a timely fashion, including the
use of antiviral medication and anticoagulation.
CONCLUSION
LST is generally a complication of middle
ear infection. Nonseptic LST, however, may

360

result from dehydration, oral contraceptive
use, coagulopathy, or thyroid disease. This
unusual case raises the suspicion that thrombosis resulted from VZV-associated thrombophlebitis in the ipsilateral cerebral venous sinuses along the second cervical nerve root
distribution. A high index of suspicion is necessary in such cases so that a different treatment course can be identified and antiviral
medication initiated promptly.
REFERENCES
1. Eidelberg D, Sortel A, Horoupian DS, et al: Thrombotic cerebral vasculopathy associated with herpes zoster. Ann Neurol 19:7-14, 1986

CHAN ET AL

2. Barrett RE, Fraser RAR, Stein BM: A fluorescence
histochemical survey of monaminergic innervation of cerebral blood vessels in primate and humans. Trans Am
Neurol Assoc 96:39-45, 1971
3. Saito K, Moskowitz MA: Contributions from the
upper cervical dorsal roots and trigeminal ganglia to the
feline circle of Willis. Stroke 20:524-526, 1989
4. Patrick JT, Russell E, Meyer J, et al: Cervical (C2)
herpes zoster infection followed by pontine infarction.
J Neuroimaging 5:192-193, 1995
5. Willeit J, Schmutzhard E: Cervical herpes zoster and
delayed brainstem infarction. Clin Neurol Neurosurg 93:
245-247, 1991
6. Fox RJ, Galetta SL, Mahalingam R, et al: Acute,
chronic, and recurrent varicella zoster virus neuropathy
without zoster rash. Neurology 57:351-354, 2001
7. Gilden DH, Dueland AN, Devlin ME, et al: Varicellazoster virus reactivation without rash. J Infect Dis 166:
S30-34, 1996 (suppl 1)
8. Gilden DH, Wright RR, Schneck SA, et al: Zoster sine
herpete, a clinical variant. Ann Neurol 35:530-533, 1994