Journal of Clinical Neuroscience xxx (2017) xxxxxx

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Journal of Clinical Neuroscience

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Case report

One and a half syndrome following penetrating head injury: Case report
Mathew R. Voisin a,, Sanskriti Sasikumar b, Jeremy H. Russell a, Gelareh Zadeh a
a
Department of Neurosurgery, Toronto Western Hospital, University of Toronto, Toronto, Ontario, Canada
b
Department of Neurology, Toronto Western Hospital, University of Toronto, Toronto, Ontario, Canada

a r t i c l e i n f o a b s t r a c t

Article history: The authors report a case of a 22-year-old otherwise healthy female who presented following a head
Received 17 December 2016 injury during a bar altercation, with no associated loss of consciousness and an unknown mechanism
Accepted 5 March 2017 of injury. Examination revealed an isolated 1 cm laceration on the right upper eyelid, superior to her
Available online xxxx
medial canthus. She experienced diplopia on right horizontal gaze due to a left internuclear ophthalmo-
plegia (INO) with an associated left conjugate horizontal gaze palsy, collectively described as a left one-
Keywords: and-a-half syndrome. CT and MRI demonstrated evidence of a deep penetrating injury above the right
One and a half syndrome
medial canthus, traversing the ethmoid and sphenoid sinuses, the dorsum sella, narrowly missing the
Endoscopy
Brainstem
basilar artery, penetrating the pons, and extending to the floor of the contralateral fourth ventricle.
Head trauma The patient was diagnosed with multiple sinus fractures, lesions in her left paramedian pontine reticular
Transsphenoidal formation (PPRF) and medial longitudinal fasciculus (MLF), and progressive pneumocephalus. She under-
went a transsphenoidal endoscopic repair via a vascularized mucosal flap without complication.
Postoperatively, the patients pneumocephalus resolved and her conjugate gaze markedly improved;
however, minimal diplopia remained. This case demonstrates the importance of the clinical exam, and
its benefit in localizing imaging findings and guiding treatment.
2017 Elsevier Ltd. All rights reserved.

1. Introduction consciousness during an altercation and developed new onset

One-and-a-half syndrome is a rare condition characterized by
conjugate lateral gaze palsy in one direction, and an internuclear
ophthalmoplegia (INO) in the ipsilateral eye [1,2]. The two principal
manifestations of orbital one-and-a-half syndrome involve the ipsi-
lateral medial longitudinal fasciculus (MLF), and either an ipsilateral
abducens nucleus lesion or the ipsilateral paramedian pontine retic-
ular formation (PPRF) [2,3]. Previous work has described multiple
causes for the condition including multiple sclerosis (MS), brainstem
tumors, strokes, or arteriovenous malformations [25]. The following
case report describes a young female who presented with a left-sided
one-and-a-half syndrome following penetrating head trauma with a
CSF leak. This is the first case report of a traumatic one-and-a-half
syndrome, and demonstrates the interplay between the physical
examination and imaging in proper diagnosis and treatment.
2. Case report
A 22-year-old otherwise healthy female presented to the emer-
gency department after she sustained a head injury without loss of
Corresponding author at: Toronto Western Hospital, 399 Bathurst Street,
Toronto, Ontario M5T 2S8, Canada.
E-mail address: mvoisin@qmed.ca (M.R. Voisin).
diplopia worse on right lateral gaze. Inspection of the wound site
revealed an isolated 1 cm laceration on the upper eyelid above
the right medial canthus. On examination, she demonstrated a
conjugate horizontal gaze palsy of the left eye in both directions
and impaired adduction of the right eye. She also had signs and
symptoms of a CSF leak from her right naris.
A plain CT head scan along with high-resolution skull base imag-
ing demonstrated pneumocephalus with multiple right parame-
dian central skull base fractures extending through the right
lamina papyracea, adjacent anterior and posterior ethmoid air cells,
inferior sphenoid sinus and finally dorsum sella (Fig. 1). MRI
demonstrated evidence of a deep penetrating injury entering the
right medial canthus, traversing the right medial conus and bony
margins seen on CT, exiting the clivus adjacent to the basilar artery,
crossing midline and penetrating the left pons before extending to
the floor of the fourth ventricle on the contralateral side (Fig. 1). A
single line drawn along the tract of the injury was approximately
12 cm with no evidence of retained foreign material.
A typical transsphenoidal approach was used and the sphenoid
sinus was opened bilaterally with a blood stained CSF pool noted
on the right hand side (Fig. 2). The sphenoid sinus was stripped
of mucosa clarifying the bony defect (Fig. 2). A septal flap was
placed to cover the defect and repair the CSF leak.

http://dx.doi.org/10.1016/j.jocn.2017.03.008
0967-5868/ 2017 Elsevier Ltd. All rights reserved.

Please cite this article in press as: Voisin MR et al. One and a half syndrome following penetrating head injury: Case report. J Clin Neurosci (2017), http://
dx.doi.org/10.1016/j.jocn.2017.03.008
2 Case report / Journal of Clinical Neuroscience xxx (2017) xxxxxx

Fig. 3. Two month post-operative (A) MRI FLAIR and (B) T2 demonstrating a more
clearly defined penetrating injury tract.

hyperintensity along the penetrating injury tract (Fig. 3) better

defined than previous imaging with no new acute findings.

3. Discussion

Fig. 1. Preoperative high-resolution skull base CT and MRI images demonstrating a
deep penetrating injury. (A) Axial CT with right ethmoid sinus anterior wall fracture
and fluid collection within anterior and posterior ethmoid air cells as well as right
sphenoid sinus. (B) Axial CT slice superior to A with arrows showing bony breach in
posterior sphenoid sinus/clivus. (C) Sagittal T1 MRI image with hypointense tract
extending through the pons to the floor of the fourth ventricle. (D) Axial FLAIR MRI
image with tract edema traversing the pons and crossing midline and extending to
the contralateral pontine tegmentum.
Postoperatively the CSF leak resolved and her left conjugate
gaze palsy improved, however, her left INO remained. At two
month follow up her left horizontal conjugate gaze had markedly
improved, and her left eye adduction was approaching full range
of movement, leading to minimal diplopia with nystagmus in her
right eye on right lateral gaze. Repeat MRI demonstrated a T2
One and a half syndrome is a condition popularized by Miller
Fisher, in 1967, to describe a horizontal gaze palsy accompanied
by an ipsilateral INO sparing convergence [1]. The first anatomical
description of the condition was by Freeman in 1943 and Wall and
Wray in 1983 reported a summary of 49 cases of the condition
with MS and stroke being the two most common causes [2]. One
and a half syndrome can manifest in two ways: either affecting
the ipsilateral medial longitudinal fasciculus (MLF) and abducens
nucleus, or the ipsilateral MLF and the paramedian pontine reticu-
lar formation (PPRF) [6,7]. Although the current literature
describes multiple etiologies for the cause of this syndrome, the
anatomical underpinnings of the syndrome remain the same.
Our patient experienced a penetrating stab injury with a tract
that extended through the pons and terminated at the contralat-
eral fourth ventricle. The left pontine tegmentum and ipsilateral

Fig. 2. Intraoperative images taken endoscopically demonstrating a (A) CSF leak within the right sphenoid sinus after the anterior sphenoid wall removal and (B) the original
bony defect seen after stripping sphenoid mucosa.

Please cite this article in press as: Voisin MR et al. One and a half syndrome following penetrating head injury: Case report. J Clin Neurosci (2017), http://
dx.doi.org/10.1016/j.jocn.2017.03.008
 Case report / Journal of Clinical Neuroscience xxx (2017) xxxxxx
MLF and PPRF demonstrated increased signal intensity on T2 and
FLAIR and reduced signal on T1 MRI sequences along the tract in
the absence of a foreign body. This case report is the first to
describe a traumatic cause of one-and-a-half syndrome due to
direct injury to the pontine tegmentum from a penetrating head
injury, and illustrates the importance of clinico-radiological
findings in patient workup and management.
Sources of support
None. No conflicts of interest to report.
Funding
This research did not receive any specific grant from funding
agencies in the public, commercial, or not-for-profit sectors.
Consent
Consent was obtained from the patient for the potential
publication of the following images and manuscript.
Please cite this article in press as: Voisin MR et al. One and a half syndrome following penetrating head injury: Case report. J Clin Neurosci (2017), http://
dx.doi.org/10.1016/j.jocn.2017.03.008
3
Disclosures
No authors have any disclosures relevant to the published work
and no conflicts of interest to declare.
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