[A case of bilateral horizontal gaze ophthalmoplegia: The 1+1 syndrome].

B. Bourre, N. Collongues, M. Bouyon, J. Aupy, F. Blanc, C. Speeg, J. de Seze
Revue Neurologique. 2010-12-01; 166(12): 1028-1031
DOI: 10.1016/j.neurol.2010.06.005

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1. Rev Neurol (Paris). 2010 Dec;166(12):1028-31. doi: 10.1016/j.neurol.2010.06.005.
Epub 2010 Aug 21.

[A case of bilateral horizontal gaze ophthalmoplegia: The 1+1 syndrome].

[Article in French]

Bourre B(1), Collongues N, Bouyon M, Aupy J, Blanc F, Speeg C, de Seze J.

Author information:
(1)Département de neurologie, hôpital universitaire de Rouen, 1, rue de Germont,
76031 Rouen, France.

INTRODUCTION: Oculomotor deficiencies in multiple sclerosis (MS) are frequently
characterized by internuclear ophthalmpoplegia or isolated abduction or adduction
palsies. Complete unilateral conjugate gaze paralysis and the « one and a half »
syndrome are rare. Complete bilateral horizontal gaze paralysis has been
exceptionally reported.
OBSERVATION: Here, we describe an unusual oculomotor paralysis as a suspected
first event of MS. A 24-year-old woman with an uneventful medical history
presented for sudden onset of binocular diplopia. On examination, abduction and
adduction saccades were impossible, whereas vertical eye saccades and convergence
were normal. Oculocephalic reflex failed to improve horizontal eye movement. No
nystagmus and no other sign of brainstem dysfunction were observed. Visual acuity
was 4/10 in the right eye and 6/10 in the left eye. A sign of Marcus Gunn was
noted in the right eye. Blood samples and cerebrospinal fluid were normal, no
oligoclonal bands were detected. Visual evoked potentials were significantly
impaired in both eyes and argued for bilateral optic neuritis. Brain MRI scans
showed white matter T2-hypersignal abnormalities, which fulfill Barkhof criteria
for MS. A small symmetric lesion was noted in the posterior part of the medial
pontine tegmentum. As a first episode of MS was suspected, treatment with
methylprednisolone 1000 mg/d for 3 days was started, and was followed by complete
recovery of eye movements and visual acuity after 3 weeks.
DISCUSSION: To our knowledge, only two cases of complete horizontal bilateral
ophthalmoplegia have been reported in the literature. Both were associated with
peripheral facial nerve palsy as a first event in MS. In our case report, we
describe for the first time a complete bilateral horizontal ophthalmoplegia with
no other brainstem dysfunction. By analogy with the « one and a half » syndrome,
such complete horizontal gaze paralysis could be named a « one and one » syndrome
and seems to be specifically related to a first event of MS.

Copyright © 2010 Elsevier Masson SAS. All rights reserved.

DOI: 10.1016/j.neurol.2010.06.005
PMID: 20728911 [Indexed for MEDLINE]

Auteurs Bordeaux Neurocampus