Major Articles

Consecutive superior oblique palsy after adjustable

suture spacer surgery for Brown syndrome:
incidence and predicting risk
Medha Sharma, MD,a Sarah MacKinnon, MSc, OC(C),a David Zurakowski, MS, PhD,c,d
and Linda R. Dagi, MDa,b

PURPOSE To determine the incidence of and to identify characteristics predicting significant superior
oblique palsy (SOP) after adjustable superior oblique suture spacer surgery for treatment of
Brown syndrome.
METHODS The medical records of patients treated for unilateral Brown syndrome with adjustable su-
ture spacers (2005-2016) were reviewed to identify possible association of age at surgery,
spacer length, surgeon performing procedure, severity of Brown syndrome, preoperative
hypotropia in primary position and affected side gaze, and reduction in Brown restriction
on postoperative superior oblique function. “Good” postoperative superior oblique func-
tion was defined as absence of hypertropia and diplopia in primary position and no more
than intermittent diplopia in downgaze comfortably fused with #4D base-down or head
tilt of \10 . Presence of postoperative hypertropia in primary position with increase in
downgaze met criteria for significant SOP. Postoperative Brown restriction of # 2 indi-
cated resolution of Brown syndrome.
RESULTS Median age at surgery was 59 months, interquartile range (IQR) was 32-82 months, and
median spacer length was 6 mm (range, 2–7 mm) for 19 included patients. Preoperative me-
dian hypotropia was 9D (IQR, 0D-12D) in primary position and 18D (IQR, 5D-22D) in
affected side gaze. Of 19 patients, 16 (84%) achieved sufficient resolution of Brown syn-
drome, but 6 (32%) developed significant SOP. Modest preoperative hypotropia in
affected side gaze was the only predictor of significant SOP (likelihood ratio test 5 7.11;
P 5 0.008). Logistic regression modeling enabled estimation of risk of significant SOP
based on preoperative side gaze hypotropia.
CONCLUSIONS Suture spacer surgery can result in significant SOP. Risk may be predicted by magnitude of
preoperative side gaze hypotropia. ( J AAPOS 2018;22:335-339)

omplete superior oblique tenotomy1,2 and

C
tenectomy3 effectively eliminate the mechanical
limitation in adduction and disfiguring misalign-
Author affiliations: aDepartment of Ophthalmology, Boston Children’s Hospital, Boston,
Massachusetts; bDepartment of Ophthalmology, Harvard Medical School, Boston,
Massachusetts; cDepartments of Anesthesia and Surgery, Boston Children’s Hospital, Boston,
Massachusetts; dDepartment of Anesthesia, Harvard Medical School, Boston, Massachusetts
This research was supported by the Children’s Hospital Ophthalmology Foundation
Chair fund held by Linda R Dagi, MD
Presented in part as a poster at the 2017 Meeting of the Association for Research in
Vision and Ophthalmology,Baltimore, Maryland, May 7-11, 2017.
Submitted January 12, 2018.
Revision accepted April 18, 2018.
Published online September 17, 2018.
Correspondence: Linda R. Dagi, MD, Boston Children’s Hospital, 300 Longwood
Avenue, Boston, MA 02115 (email: Linda.Dagi@childrens.harvard.edu).
Copyright Ó 2018, American Association for Pediatric Ophthalmology and
Strabismus. Published by Elsevier Inc. All rights reserved.
1091-8531/$36.00
https://doi.org/10.1016/j.jaapos.2018.04.017
ment in upgaze that characterize Brown syndrome, but
approximately 80% of patients thus treated develop iatro-
genic superior oblique palsy (SOP).1,4 Secondary diplopia
in primary and reading positions has sometimes eluded
remedy, even after surgical intervention.5 Alternative pro-
cedures that aim to reduce the risk of consecutive SOP by
enabling graded reduction in superior oblique tone have
been introduced.6-12
Adjustable suture spacer surgery, introduced by Suh and
colleagues,9,10 enables a graded lengthening of the superior
oblique tendon. With intraoperative adjustment of the
spacer length, the technique facilitates a near-matching
of superior oblique tone between the affected eye and the
normal eye while reducing Brown restriction.9,10 We
report our experience using suture spacer surgery to treat
Brown syndrome, including incidence of consecutive
SOP, and the results of logistic regression modeling for
factors predictive of this outcome.

Journal of AAPOS 335

336 Sharma et al
Methods
Records of patients with Brown syndrome seen at Boston Chil-
dren’s Hospital from 2005 to 2016 were retrospectively reviewed
to identify patients treated with suture spacer surgery. Patients
with simultaneous vertical rectus or inferior oblique muscle sur-
gery, bilateral Brown syndrome, or an orbital mass resulting in
pseudo-Brown restriction were excluded. Institutional Review
Board approval was obtained from Boston Children’s Hospital,
and the study complied with the US Health Insurance Portability
and Accountability Act of 1996 and with the principles of the
Declaration of Helsinki.
Indications for surgery were anomalous head posture to main-
tain fusion, history of recent loss of anomalous head posture with
loss of fusion, or persistent complaints of diplopia or downshoot
in adduction. The diagnosis was confirmed with intraoperative
forced ductions.13
Surgical technique was as described by Suh and colleagues.9,10
The final length of each 5-0 Poly (ethylene terephthalate) spacer
(Mersilene; Ethicon, Somerville, NJ) was determined by the
results of intraoperative forced ductions13 and exaggerated trac-
tion testing of the operated superior oblique.14 The goal of adjust-
ment was to create a spacer length that left residual superior
oblique tone in the affected eye equal to that in the unoperated
eye by the exaggerated traction test14 while enabling adequate
reduction in Brown restriction (based on forced ductions).13 If
the intraoperative exaggerated traction test suggested excessive
superior oblique tone, the length was slightly increased. If the su-
perior oblique tone was inadequate, the length was reduced. The
exaggerated traction test was repeated to determine whether bilat-
erally symmetric superior oblique tone had been achieved; spacer
length was further adjusted if necessary.
The following data were extracted from the medical record: age
at surgery (in months), length of spacer (in mm), surgeon per-
forming the procedure, and immediate preoperative and final
sensorimotor status. Sensorimotor data analyzed were as follows:
pre- and postoperative alignment by alternate prism and cover
test in primary position, affected side gaze, and reading position
(downgaze); severity of Brown restriction (graded 1 to 5); su-
perior oblique ductions (graded 0 to 4); inferior oblique overac-
tion (graded 0 to 14); head posture (by goniometry); binocular
fusion (by Worth 4-dot test); and stereopsis (Randot Stereotest;
Stereo Optical, Chicago, IL). Hypertropia was denoted with a
plus sign; hypotropia, with a minus sign. Reduction in Brown re-
striction was calculated as the difference between pre- and post-
operative Brown restriction.
Data for the final outcome visit were extracted from the last
sensorimotor evaluation available. If additional strabismus sur-
gery related to Brown syndrome, SOP, or any vertical misalign-
ment was performed, the final outcome visit was deemed to be
the last performed prior to this additional surgery. Length of
postoperative follow-up (in months) was time from the primary
surgery to the final outcome examination.
Our primary outcome measure, superior oblique function, was
classified as either “good” or consistent with “significant SOP.”
Good superior oblique function required absence of manifest hy-
pertropia in primary position and no more than intermittent
Volume 22 Number 5 / October 2018
diplopia in downgaze comfortably fused with #4D base-down
prism or torticollis of \10 . Postoperative hypertropia in primary
position, increasing in downgaze, qualified as significant SOP.
Postoperative Brown restriction of # 2 was deemed sufficient
resolution of Brown syndrome.
We assessed the effect of spacer length on resolution of Brown
syndrome as well as the possible association of spacer length, age
at surgery, operating surgeon, severity of preoperative Brown lim-
itation, preoperative hypotropia in primary position and affected
side gaze, and reduction of Brown restriction on final postopera-
tive superior oblique function.
Statistical Analysis
The Spearman r rank order correlation coefficient was used to
measure the association of spacer length and preoperative Brown
restriction and was also used to measure the association of severity
of preoperative Brown restriction, change in Brown restriction,
operating surgeon, spacer length, and preoperative hypotropia
in primary position and contralateral side gaze versus designation
of postoperative superior oblique function as good or significantly
paretic. Preoperative versus postoperative change in Brown re-
striction was compared between patients, according to SOP clas-
sification, using the nonparametric Mann-Whitney U test.
Change in Brown restriction was summarized by the median
and range. Two-tailed values of P \ 0.05 were considered statis-
tically significant. Statistical analysis was performed using IBM
SPSS version 23.0 (IBM Corporation, Armonk, NY).
Multivariable logistic regression analysis was used to determine
independent factors affecting postoperative superior oblique
function, including length of spacer, operating surgeon, age, pre-
operative measurements of Brown restriction and hypotropia
(primary position and contralateral side gaze), and pre- to postop-
erative change in Brown restriction while adjusting for patient age
as a covariate in the model.15
Results
Of 21 patients treated for unilateral Brown syndrome with
superior oblique suture spacers, 19 (11 females) met inclu-
sion criteria. (One was excluded because an orbital tumor
caused pseudo-Brown; the other, because vertical rectus
surgery was performed along with spacer surgery.) All cases
were performed by one of 5 attending surgeons. Median
age at surgery was 59 months (IQR, 32-82 months). Me-
dian follow-up was 19 months (IQR, 3-42 months).
Of the 19 patients, 16 (84%) had sufficient resolution of
Brown syndrome, and no additional surgery was performed
to reduce Brown restriction; 13 of 19 retained good supe-
rior oblique function, but 6 of 19 (32%) demonstrated sig-
nificant SOP.
There was no relation between postoperative superior
oblique function and the surgeon performing the proced-
ure (P 5 0.39 [c2]). Univariate analysis demonstrated no
association of length of spacer (P 5 0.152), preoperative
vertical deviation in primary position (P 5 0.151), severity
of preoperative Brown restriction (P 5 0.831), and change
in Brown restriction (P 5 0.152) with postoperative
Journal of AAPOS
Volume 22 Number 5 / October 2018
FIG 1. Preoperative hypotropia in affected side gaze versus risk of sig-
nificant SOP. Less hypotropia in affected side gaze confers a predict-
ably greater risk of significant SOP. Logistic regression model:
likelihood ratio test, 7.11; P 5 0.008.
superior oblique function. An association of age at time of
surgery (P 5 0.017) and preoperative alignment in affected
side gaze (P 5 0.005) was noted by univariate analysis.
Multivariable logistic regression analysis demonstrated
that irrespective of age, preoperative vertical deviation in
contralateral gaze was the only predictor of postoperative
superior oblique function (likelihood ratio test, 7.11;
P 5 0.008). A logistic regression model based on our
data demonstrates the predictive nature of hypotropia in
side gaze (P 5 0008; see Figure 1).
Median follow-up for patients was longer for those with
good superior oblique function (median, 25 months [IQR,
8-91 months]) than for those with significant SOP (median,
3 months [IQR 2-24 months]; P 5 0.046).
Detailed sensorimotor and demographic information by
patient is provided in eTable 1; eTable 2 provides summa-
rized preoperative sensorimotor characteristics, spacer
length, Brown restriction and length of follow-up for the
entire group, and postoperative superior oblique function
(good vs significant SOP).
At the time of surgery 15 of 19 patients had documented
fusion or stereopsis. Three patients without documented
fusion had evidence of prior fusion based on a history of
torticollis lost over time. Seventeen patients demonstrated
fusion and a wide range of stereopsis after suture spacer
surgery, 4 with ocular torticollis. The 2 who lost fusion
were among the 6 who had developed significant SOP;
one was diplopic, and the other developed suppression—
both declined additional surgery. The remaining 4 of the
6 who had demonstrated significant postoperative SOP
and ocular torticollis underwent additional strabismus sur-
gery with successful conversion back to good superior ob-
lique function, fusion, and stereopsis. In 2 of the 4,
adhesions between the superior oblique and adjacent supe-
rior rectus or sclera were noted intraoperatively, and there
Journal of AAPOS
Sharma et al 337
was improvement in infraduction after scar tissue removal.
In both cases, simultaneous ipsilateral inferior oblique sur-
gery was performed because the mechanical limitation of
infraduction noted at the time of reoperation did not
explain the degree of infraduction deficit documented clin-
ically. In addition, the clinically noted infraduction deficit
was primarily in adduction. Two other patients with surgi-
cally treated SOP had no evidence of mechanical restric-
tion of infraduction. One was successfully treated with a
5 mm temporal tuck of the superior oblique rather than
replacement of the spacer, because this was technically
easier to perform. The second was treated with an ipsilat-
eral inferior oblique recession.
Discussion
Our review of the effects of adjustable suture spacer surgery
for treatment of Brown syndrome found that whereas 84%
of patients had substantial resolution of Brown syndrome,
32% developed significant SOP. Modest preoperative hy-
potropia in the affected side gaze was the only identified
risk factor for SOP. Fortunately, it is readily quantified
by the surgeon prior to intervention.
Spacer surgery was developed with the expectation that
graded, intraoperative adjustment based primarily on
equalizing tension in the treated superior oblique with
the superior oblique of the contralateral eye would sub-
stantially reduce the risk of symptomatic SOP. Although
adjustable spacers do reduce this risk, any procedure that
elongates or recesses the superior oblique tendon will
affect superior oblique function. The procedure appears
to be particularly poorly tolerated in patients with minimal
preoperative hypotropia in side gaze. In contrast, signifi-
cant hypotropia in side gaze may indicate a coupling of su-
perior oblique overaction with Brown restriction and be
logically associated with a reduced risk of consecutive SOP.
For example, patients 16, 18, and 19 had long spacers
placed in the setting of minimal hypotropia in primary po-
sition and side gaze but had persistent preoperative torti-
collis and constant complaints of diplopia. For these
patients, adjustable suture spacer surgery performed with
intraoperative confirmation of “sufficient” post-spacer su-
perior oblique tone gave false hope of preventing SOP.
Some might advocate avoiding surgery in these symptom-
atic but mildly affected patients because of the risk of SOP.
Several authors have reported spontaneous resolution or
improvement in 10% to 89% patients with congenital
Brown syndrome over time.16-20 Given the potential for
spontaneous resolution of Brown syndrome and the high
risk of SOP associated with minimal preoperative
hypotropia in affected side gaze, conservative
management would have been preferable for these
patients. The original publication on suture spacers
mentions checking fundus torsion in cases where
interpretation of the exaggerated traction test is uncertain
or if preoperative torsional symptoms are significant.9 A
338 Sharma et al
handy alternative based on preplaced corneal markings can
provide this information as well.21 Whether SOP could
have been avoided in patients with minimal hypotropia in
affected side gaze by monitoring intraoperative change in
ocular torsion rather than the results of exaggerated trac-
tion and forced duction testing remains outside the pur-
view of this study, because sufficient data were not
available on this characteristic.
According to the logistic regression model based on
our data, a patient with 6D of preoperative hypotropia
in affected side-gaze has an 80% chance of developing
clinically significant SOP after suture spacer surgery as
currently performed. In contrast, a patient with 19D of
preoperative hypotropia in affected side gaze has a
90% chance of retaining good postoperative superior ob-
lique function. Patients with .16D of hypotropia in
affected side gaze have $80% estimated probability of
retaining good postoperative superior oblique function.
Based on this data, we recommend suture spacer surgery
when preoperative hypotropia in affected side gaze
is . 16D and the patient complains of frequent diplopia
or psychosocial stress related to the misalignment.
Future modifications in the spacer technique or our abil-
ity to better predict residual postoperative superior obli-
que function might alter this recommendation. All
patients, regardless of severity of their Brown syndrome,
should be informed that there is the possibility of spon-
taneous resolution over time.16-20
It is notable that the group of patients who developed
significant SOP had shorter follow-up than the group
with good postoperative function. Thus, evolution toward
significant SOP did not appear to be the natural outcome of
longer follow-up.
Although our incidence of superior oblique palsy after
spacer placement might seem greater than has been previ-
ously reported, none of the prior studies on spacers, sili-
cone expanders, or superior oblique lengthening have
provided the detailed sensorimotor analysis in downgaze
in addition to primary position that identify less severe
manifestations of SOP included in our series, and some
have reported results only in populations of patients with
more severe Brown syndrome.6,12,13,22,23
Wright2 described his experience with silicone spacers in
15 patients with Brown syndrome. Of 15 patients, 6 devel-
oped hypertropia in primary position, consistent with su-
perior oblique paresis. Stager and colleagues14 used
silicone expanders on 19 patients with Brown syndrome
and reported overcorrection with significant SOP in 4 pa-
tients; the incidence of milder SOP was not noted.
Awad and colleagues22 investigated the use of large supe-
rior oblique tendon silicone expanders for patients with se-
vere Brown syndrome and primary position hypotropia
ranging from 20D to 40D. Even in this severely affected
population, 3 of 12 patients demonstrated hyperphoria in
primary position after 1 year; no data were provided on
motility in downgaze.
Volume 22 Number 5 / October 2018
Moghadam and colleagues24 described split-tendon
lengthening of the superior oblique tendon to treat Brown
syndrome. Their cohort all had very severe Brown syn-
drome, with 10D to 25D hypotropia in primary position.
The magnitude of preoperative primary position hypotro-
pia would likely have prevented consecutive SOP, regard-
less of the graded treatment used.
Suh and colleagues9 reported 1 of 3 treated patients with
SOP. In the longer-term study,10 4 of 13 patients devel-
oped hypertropia in contralateral side gaze, likely associ-
ated with SOP. Yazdian and colleagues25 reported 2
overcorrections in 25 patients treated for Brown syndrome
with suture spacers. No definition of overcorrection was
offered, and only mean pre- and postoperative primary po-
sition vertical alignments were provided.
Awadein and Gawdat,23 using a modified suture spacer
technique,26 reported no cases of SOP in the suture spacer
group; however, the range of preoperative hypotropia in
affected side gaze in their series (19.4D  8.9D) was similar
to that in our patients who retained good superior oblique
function (21D  11D).
The most surprising outcome from our series was the
lack of demonstrated association of spacer length with
severity of preoperative Brown restriction or with the
development of postoperative SOP. This lack of correla-
tion has been noted previously10,23 and may result from
a number of causes. First, cases from 5 attending
surgeons were included in this study. Preoperative
grading of severity of Brown restriction did not always
correlate with severity based on the more objective
measure of hypotropia in primary position and affected
side gaze; approach to grading Brown restriction was
likely inconsistent between attending surgeons.
Although the technique of spacer placement and
methodology guiding choice of spacer length were the
same, intraoperative judgment regarding final choice of
spacer length and differences in technique used to
designate the length of spacer placed may have
contributed to an inability to identify anticipated
associations. Finally, as some variants of Brown
syndrome reduce in severity with exaggerated forced
ductions, surgical manipulation might, itself, have
modified the Brown restriction intraoperatively in some
cases. In such cases, a shorter-than-anticipated spacer
might have been sufficient to resolve the residual limita-
tion left after surgical manipulation.
This study is limited by its retrospective nature and by
the fact that the surgeries were performed by 5 different
surgeons. Moreover, the modest number of patients may
have confounded our ability to identify all characteristics
associated with consecutive SOP. The natural history of
our studied cohort, had no spacer surgery been per-
formed, remains unknown. Also, our definitions of out-
comes, while consistent with our clinical experience, are
unique to this study. Patients with good postoperative su-
perior oblique function were pleased with their outcomes
Journal of AAPOS
Volume 22 Number 5 / October 2018
and had no reason to consider further surgery. Those with
significant SOP were symptomatic and benefited from
additional surgery.
Acknowledgments
The authors acknowledge the editorial assistance by Jane Patrick.
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339.e1 Sharma et al Volume 22 Number 5 / October 2018

eTable 1. Summary of demographic and sensorimotor characteristics

Age at Pre-op vertical deviation, PD (IQR) Brown
Post-op SO surgery, mos Spacer length, Follow-up,
function (IQR) mm (IQR) Primary Side gaze Pre-op Post-op mos (IQR)
All (n 5 19) 59 (32-82) 6 (4-7) 9 ( 12 to 0) 18 ( 22 to 5) 4 ( 4 to 3) 2 ( 2 to 4) 19 (3-42)
Good SO function 41 (29-63) 5 (4-6) 10 ( 16 to 3) 20 ( 25 to 18) 4 ( 4 to 3) 2 ( 2 to 1) 25 (8-91)
(n 5 13)
Significant SOP 75 (66-192) 6.75 (5-7) 2 ( 10 to 0) 0 ( 13 to 0) 4 ( 5 to 3) 0 (0 to 2) 3 (2-24)
(n 5 6)
IQR, interquartile range; PD, prism diopter; Post-op, postoperative; Pre-op, preoperative; SO, superior oblique; SOP, superior oblique palsy.

Journal of AAPOS
Journal of AAPOS

Volume 22 Number 5 / October 2018

eTable 2. Preoperative and final postoperative sensorimotor data for patients with Brown syndrome treated with intraoperative adjustable suture spacera,b
Vertical deviation Vertical deviation
primary affected side Vertical deviation
Length of Sufficient
distance, PD gaze, PD downgaze, PD Fusion/stereopsis Brown limitation
Age, spacer, Indication for Follow-up, Brown Post-op SO
No. mos Eye mm surgery Pre-op Post-op Pre-op Post-op Pre-op Post-op Pre-op Post-op Pre-op Post-op mos resolution function
1 59 L 4 torticollis, then 10 2 20 4 10 2H No Yes (3000) 3 1.5 25 Yes Good
loss of fusion
2 32 L 4 torticollis 8 3 14 5 3 5H Yes Yes 4 3 7 No Good
3 31 R 4 torticollis 18 0 25 0 0 0 Yes (340) Yes (200) 4 3.5 19 No Good
4 26 R 6 torticollis 10 0 23 0 0 0 Yes (550) Yes (550) 4 3 2 No Good
5 16 R 6 torticollis NA 0 NA 2 NA 0 NA Yes (25) 3 1 97 Yes Good
6 66 R 6 loss of fusion 16 10 20 10 20 8 No Yes (800) 3 1 16 Yes Good
7 57 L 7 loss of fusion 4 0 NA 0 NA 0 No Yes (3000) 4 1 85 Yes Good
8 32 L 7 torticollis 25 0 33 NA 8 NA Yes Yes (20) 4 2 8 Yes Good
9 41 L 3.5 torticollis 10 2H 18 2H 0 2H Yes (400) Yes (40) 4 0 120 Yes Good
10 96 L 6 torticollis1 0 3H 20 2H 0 4H Yes (70) Yes (100) 4.5 2 121 Yes Good
11 642 R 5 torticollis 14 1H 28 2H 0 3H Yes (70) Yes (50) 4 0 26 Yes Good
12 19 L 5 torticollis 2 4 10 NA 0 0 Yes Yes (70) 4 2 8 Yes Good
13 46 R 4 torticollis 6 0 18 2H 5 5H Yes (100) Yes (200) 2 2 42 Yes Good
14 82 L 7 torticollis 10 10 HT 20 8 HT 0 14 HT Yes (50) No - diplopia 4 2 3 Yes sig SOP
15 67 R 2 torticollis 11 6 HT 10 4 HT 3 16 HT Yes (100) No 2 0 (11 OAIO) 28 Yes sig SOP
16 85 L 6 increasing 0 20 HT 0 35 HT 0 25 HT Yes (100) Yes (30) 4 0 (12 OAIO) 23 Yes sig SOP
diplopia
17 63 L 7 torticollis, 4 10 HT 6 25 HT 0 23 HT Yes (100) Yes (3000/25)c 5 0 (13 OAIO) 2 Yes sig SOP
followed by
suppression
18 69 L 6.5 torticollis1 0 2 HT 0 5 HT 2 20 HT Yes (50) Yes (70) 3.5 0 0 Yes sig SOP
19 513 L 7 torticollis, 0 10 HT 6 23 HT 0 25 HT Yes (20) Yes (3000) 3 1 3 Yes sig SOP
diplopia1
H, hyperphoria; HT, hypertropia; NA, not available; Pre op, preoperative; Post op, postoperative; PD, prism diopter; SO, superior oblique; SOP, superior oblique palsy; Sig, significant.
a

Sharma et al
Negative sign indicates hypotropia.
b
Patients with torticollis but an “orthotropic” status in primary position were presumed to have had either a very small hypertropia in true primary position not documented or discomfort with
diplopia noted with minimal elevation prompting the maintenance of ocular torticollis to minimize symptomatic diplopia.
c
Improved stereopsis after reoperation.

339.e2