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January - February 2008
Letters to the Editor
2.
Aldrich MS, Vanderzant CW, Alessi AG, Abou-Khalil B, Sackellares
JC. Ictal cortical blindness with permanent visual loss. Epilepsia
1989;30:116-20.
3.
Chung SJ, Lee JH, Lee SA, No YJ, Im JH, Lee MC. Co-occurrence
of seizure and chorea in a patient with nonketotic hyperglycemia.
Eur Neurol 2005;54:230-2.
4.
Hadjikoutis S, Sawhney IM. Occipital seizures presenting with
bilateral visual loss. Neurol India 2003;51:115-6.
5.
Bodis-Wollner I, Atkin A, Raab E, Wolkstein M. Visual association
cortex and vision in man: Pattern-evoked occipital potentials in a
blind boy. Science 1977;198:629-31.
6.
Sadeh M, Goldhammer Y, Kuritsky A. Postictal blindness in adults.
J Neurol Neurosurg Psychiatry 1983;46:566-9.
7.
Backhouse O, Leitch RJ, Thompson D, Kriss A, Charris D, Clayton
P, et al. A case of reversible blindness in maple syrup urine disease.
Br J Ophthalmol 1999;83:250-1.
8.
Paiboonpol S. Epilepsia partialis continua as a manifestation of
hyperglycemia. J Med Assoc Thai 2005;88:759-62.
9.
Lavin PJ. Hyperglycemic hemianopia: A reversible complication of
nonketotic hyperglycemia. Neurology 2005;65:616-9.
89
Figure 1: Ocular motility examination revealed mild limitation with
narrowing of the palpebral Þssure and globe retraction of the right eye
on abduction (A). However adduction of the right eye did not show
any limitation (B)
10. Harden CL, Rosenbaum DH, Daras M. Hyperglycemia presenting
with occipital seizures. Epilepsia 1991;32:215-20.
Inverse Duane’s retraction syndrome
following myocysticercosis
Dear Editor,
Duane’s retraction syndrome is a well-known congenital
ocular motility disorder.1 In the most common type of Duane’s
retraction syndrome, there is marked restriction of abduction,
with eyeball retraction and narrowing of the palpebral Þssure
on adduction. In inverse Duane’s syndrome, abduction of the
affected eye is possible to some extent. However, the globe
retraction occurs on abduction accompanied by narrowing
of the palpebral Þssure and pseudoptosis. We report a case of
inverse Duane’s syndrome due to myocysticercosis involving
the medial rectus muscle. To the best of our knowledge, based
on systematic search of English language literature, no similar
case has been reported before.
An 18-year-old lady presented to us with complaints of
diplopia on right gaze of 20 days duration. Visual acuity in each
eye was 20/20 without any refractive error. Cover test revealed
an esotropia of 8 prism diopters. Ocular motility examination
revealed mild limitation of abduction in the right eye with
normal adduction [Fig. 1]. There was 3 mm of globe retraction
and 3 mm of narrowing of the palpebral Þssure on abduction
in the right eye. Forced duction test revealed restriction of
abduction in the right eye. Conjunctival congestion was
observed near the medial rectus muscle insertion. Ultrasound
B-scan [Fig. 2] showed a grossly thickened medial rectus muscle
with a cystic lesion and a high reßective spot (scolex) within
suggestive of medial rectus muscle cysticercosis. The scolex
was not moving nor was there any evidence of calciÞcation.
The cyst was noted to be producing scleral indentation which
was conÞrmed on fundus examination. She was started on oral
albendazole 15 mg/Kg body weight and Prednisolone 1 mg/Kg
body weight. The diplopia resolved within two weeks and
Figure 2: Ultrasound B-scan of the right orbit, showing the presence of
a cystic lesion within the medial rectus muscle and a high reßective spot,
scolex, with the corresponding A-scan showing a spike corresponding
to the scolex.
her ocular movements were full without any globe retraction
on abduction. Ultrasound B-scan showed that the scolex had
disappeared. At last follow-up three months later, she was
asymptomatic.
Inverse Duane’s syndrome is very uncommon. This was
Þrst reported by Thomas Duane in 1976.2 They preferred the
name “pseudo-Duane’s retraction syndrome” as the etiology
of these cases is completely different from Duane’s retraction
syndrome. Four cases were noted following trauma and one
patient had orbital metastases. The primary abnormality in
inverse Duane’s syndrome lies in the medial rectus muscle.
It is most often due to trauma to the medial wall with
entrapment of the medial rectus muscle in the fractured lamina
papyracea. Pterygium has also been cited to be a cause of
inverse Duane’s syndrome.3 Congenital cases have also been
reported where medial rectus muscle shortening has been
suggested.4,5 Chatterjee et al. observed the presence of Þbrous
bands extending from the medial recti muscles to the medial
orbital wall.4 The basic cause of palpebral narrowing in Duane’s
90
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Indian Journal of Ophthalmology
retraction syndrome is miswiring, while in inverse Duane’s
syndrome it is restrictive.
Hydatid cyst involving the extraocular muscles has also been
reported. Kiratli et al. reported a case with involvement of the
medial rectus muscle.6 However, there was no ocular motility
restriction. On the B scan the hydatid cyst is usually seen as
a multilobulated cystic lesion in contrast to the presence of a
smooth lesion with a scolex, in cases with cysticercosis. While
cysticercosis is quite common in the extraocular muscles,
hydatid cysts involving the extraocular muscles are very
unlikely.6
Pollard et al. reviewed cases of orbital myositis leading to
acute rectus muscle palsy and observed that all patients with
medial rectus muscle involvement had exotropia while patients
with lateral rectus involvement had esotropia.7 It is likely that
cases where there is paralysis of the medial rectus muscle
manifest with exotropia, while our case which had a restrictive
problem, had esotropia.
Our patient presented with features suggestive of inverse
Duane’s syndrome. We suspected cysticercosis as it is common
in our setting. Ultrasound B-scan revealed the cyst with a
high reßective scolex within, characteristic of cysticercosis. In
addition the cyst was noted to be producing scleral indentation,
which has been described before.8 A high index of suspicion for
myocysticercosis should be entertained in all cases presenting
with sudden onset of inflammatory signs and unusual
ocular motility features. A computed tomogram scan and/or
ultrasound examination should be performed to conÞrm the
diagnosis in these cases.
Ramesh Murthy, MD, FRCS
From the Strabismus and Pediatric Ophthalmology, Oculoplasty and
Ocular Oncology Service, LV Prasad Eye Institute, Hyderabad, India.
E-mail: rameshmurthy@lvpei.org
References
Vol. 56 No. 1
Spontaneous late-onset comitant
acute nonaccommodative esotropia in
children
Dear Editor,
We read the article by Kothari,1 with great interest. The author has
described the clinical characteristics of spontaneous, late-onset
comitant acute nonaccommodative esotropia in children. We
appreciate that the author has drawn attention to this relatively
uncommon clinical entity. We would like to put forward a
different view regarding neuroimaging in such cases.
Many intracranial lesions present with comitant
nonaccommodative esotropia without neurological deficit
in early stage.2 Sometimes acute-onset nonaccommodative
esotropia is the only presenting sign of intracranial neoplasm.
There are many studies in the literature which state that
intracranial lesions may present with acute-onset comitant
esotropia without any neurological deÞcit or other signs.2-4
In patients with acute esotropia with corpus callosum
gliomas, clinical signs are absent in the early part of the disease.
Also, tumors involving the corpus callosum give rise to no
distinctive signs.3 Posterior fossa lesions are typically known
to present with only acute-onset comitant esotropia with no
other systemic signs such as papilledema.2
Acute comitant esotropia has also been described in patients
with Arnold Chiari malformation even prior to onset of typical
down beating nystagmus.4 It is important in these cases of
acquired esotropia to rule out the possibility of intracranial
tumor, Arnold Chiari malformation or other neurological
abnormalities.
Although most children with this form of esotropia are
otherwise healthy, central nervous system lesions must be
considred.5
Missing a brain tumor, however rare, in a child presenting
with acquired comitant nonaccommodative esotropia will
be an act of negligence. So we feel that neuroimaging should
be done in any patient presenting with acquired comitant
nonaccommodative esotropia.
1.
Duane A. Congenital deÞciency of abduction, associated with
impairment of adduction, retraction movements, contractions of
the palpebral Þssure and oblique movements of the eye, 1905. Arch
Ophthalmol 196;114:1255-7.
2.
Duane TD, Schatz NJ, Caputo AR. Pseudo-Duane’s retraction
syndrome. Trans Am Ophthalmol Soc 1976;74:122-32.
3.
Khan AO. Inverse globe retraction syndrome complicating
recurrent pterygium. Br J Ophthalmol 2005;89:640-1.
4.
Chatterjee PK, Bhunia J, Bhattacharya I. Bilateral Inverse Duane’s
retraction syndrome: A case report. Indian J Ophthalmol
1991;39:183-5.
5.
Lew H, Lee JB, Kim HS, Han SH. A case of congenital inverse
Duane’s retraction syndrome. Yonsei Med J 2000;41:155-8.
6.
Kiratli H, Bilgiç S, Oztürkmen C, Aydin O. Intramuscular hydatid
cyst of the medial rectus muscle. Am J Ophthalmol 2003;135:98-9.
References
7.
Pollard ZF. Acute rectus muscle palsy in children as a result of
orbital myositis. J Pediatr 1996;128:230-3.
1.
8.
Agrawal S, Agrawal J, Agrawal TP. Orbital cysticercosis-associated
scleral indentation presenting with pseudo-retinal detachment. Am
J Ophthalmol 2004;137:1153-5.
Uday V Naik, DNB; Sumita Agarkar, MS;
Meenakshi Swaminathan, MS; TS Surendran, DO, M. Phil
Pediatric Ophthalmology Department, Sankara Nethralaya, 18,
College Road, Chennai - 600 006, India.
E-mail: naikuv@rediffmail.com
2.
Kothari M. Clinical characteristics of spontaneous late-onset
comitant acute nonaccommodative esotropia in children. Indian J
Ophthalmol 2007;55:117-20.
Williams AS, Hoyt CS. Acute comitant esotropia in children with
brain tumors. Arch Ophthalmol 1989;107:376-8.