ORIGINAL ARTICLE
A Cohort Study of Strabismus Rates Following Correction
of the Unicoronal Craniosynostosis Deformity:
Conventional Bilateral Fronto-Orbital Advancement
Versus Fronto-Orbital Distraction Osteogenesis
Ian C. Hoppe, MD and Jesse A. Taylor, MD
Background: The purpose of this prospective study is to compare
perioperative morbidity and strabismus rates between traditional
fronto-orbital advancement reconstruction (FOAR) and fronto-
orbital distraction osteogenesis (FODO) in unicoronal craniosynos-
tosis (UCS).
Method: A consecutive group of 15 patients undergoing FODO for
isolated UCS were compared to a contemporaneous group of 15
patients undergoing traditional FOAR for UCS. Patient age,
operative time, blood loss, blood replacement, technical details
of the surgery, length of stay, complications, and strabismus rates
were documented and compared statistically using chi-square and
Student t test with a significance value of 0.05.
Results: The 15 patients undergoing FODO were younger (6.3 and
9.8 months, P < 0.05), experienced less operative time for the initial
procedure (111 versus 190 minutes, P < 0.01), less blood loss (26%
versus 50% of total blood volume, P < 0.01), and less blood
replacement (40% versus 60% of total blood volume, P < 0.05).
One patient in the FODO group experienced a new-onset strabismus
postoperatively compared with 5 in the FOAR group (P < 0.05).
There were no complications requiring a return to the operating
room in either group.
Conclusions: Fronto-orbital distraction osteogenesis for the
treatment of isolated UCS is associated with a favorable
perioperative morbidity profile and a decreased incidence of
postoperative strabismus compared with traditional FOAR. These
From the Division of Plastic Surgery, The University of Pennsylvania and
the Children’s Hospital of Philadelphia, Philadelphia, PA.
Received March 3, 2021.
Accepted for publication April 3, 2021.
Address correspondence and reprint requests to Jesse A. Taylor, MD,
Division of Plastic Surgery, The University of Pennsylvania and the
Children’s Hospital of Philadelphia, 34th Street and Civic Center
Boulevard, Philadelphia, PA 19104; E-mail: taylorj5@email.chop.edu
This paper was presented in part at the 2018 American Society of Craniofa-
cial Surgeons annual meeting during the 2018 American Cleft Palate-
Craniofacial annual meeting in Pittsburgh, PA. It was the recipient of the
2018 Kawamoto Best Paper Award in recognition of the best craniofacial
paper presented at the meeting.
This study was approved by the Institutional Review Board of the Children’s
Hospital of Philadelphia
The authors report no conflicts of interest.
Supplemental digital contents are available for this article. Direct URL
citations appear in the printed text and are provided in the HTML and
PDF versions of this article on the journal’s Web site (www.jcraniofa-
cialsurgery.com).
Copyright # 2021 by Mutaz B. Habal, MD
ISSN: 1049-2275
DOI: 10.1097/SCS.0000000000007773
2362 The Journal of Craniofacial Surgery  Volume 32, Number 7, October 2021
Copyright © 2021 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
positive factors are tempered by the need for an additional
procedure for removal of the device and lack of long-term
outcomes data on the technique.
Key Words: Craniosynostoses, osteogenesis, strabismus
(J Craniofac Surg 2021;32: 2362–2365)
T he cranial deformity in unicoronal craniosynostosis (UCS) is
characterized by ipsilateral forehead and supraorbital retrusion,
brow elevation, and contralateral forehead bossing.1 During growth
of the skull, restriction at the sphenoid wing results in the charac-
teristic harlequin orbital deformity and translates these changes
through the cranial base to affect facial appearance in the form of
nasal root deviation towards the affected side and chin point
deviation towards the unaffected side.1 Patients with UCS demon-
strate a higher-than-normal rate of strabismus, likely due to a
combination of the altered orbital bony anatomy, altered attachment
of the trochlea, and altered position of the extra-ocular eye mus-
cles.2 In addition, a high incidence of postoperative strabismus
following traditional, open, fronto-orbital advancement with cranial
vault remodeling (FOAR) is well documented and thought to be due
to the same combination of factors interacting with the changes of
acute bony repositioning and re-draping of the soft tissues that
characterize FOAR.3
Our group has had a longstanding interest in the relationship
between strabismus and UCS,2 and we were quite interested in the
report by MacKinnon et al,4 demonstrating that earlier intervention
in the form of an endoscopic strip craniectomy and helmet therapy
was associated with a decreased rate of postoperative strabismus,
attributed to the gradual change in orbital position associated with
this technique as well as the earlier age of intervention before the
development of strabismus.
In our Unit, correction of the UCS deformity has traditionally
been achieved by a bilateral FOAR.5 This procedure has the longest
documented track record for safety and efficacy, but may be
associated with significant blood loss necessitating transfusion,
comparatively long anesthetic times, and a variably high incidence
of extraocular muscle dysfunction postoperatively.3,6 It has been
shown that treatment of UCS with fronto-orbital distraction osteo-
genesis (FODO) can effect changes in the cranial base and is
associated with a decreased operative time and hospital length of
stay.7,8 By not detaching the bone flap from the dura, there may be
decreased intraoperative blood loss and subsequent need for blood
product replacement. In addition, the gradual process of distraction
may allow the extraocular muscles and trochlea time to accommo-
date to their new position, possibly affecting rates of strabismus.
The purpose of this study is to compare perioperative morbidity
and strabismus rates between traditional FOAR and FODO.
The Journal of Craniofacial Surgery  Volume 32, Number 7, October 2021
METHODS
The authors’ Institutional Review Board-approved, prospective, cra-
niofacial registry was queried from July 2012 to September 2017 for
all patients having undergone correction of isolated UCS by the senior
author (JAT). Exclusion criteria were multi-suture craniosynostosis
and follow-up with both ophthalmology and plastic surgery of less
than 1 year. The parents of children presenting with nonsyndromic,
UCS were explained the risks and benefits of FODO and FOAR and
were allowed to choose between the 2 procedures; thus, we did not
randomize patients to either treatment group. 15 consecutive patients
who underwent correction via FODO were included and compared to
the most recent 15 patients who underwent FOAR for UCS. Com-
parison was performed with regards to demographics, intraoperative
details, perioperative morbidity, and rates of strabismus.
Intraoperative data collected included length of surgery, blood
loss, and blood replacement. Perioperative details collected included
intensive care unit length of stay and total length of stay. Specific
attention was given to preoperative and postoperative ophthalmology
examinations to determine rates of strabismus and further delineate
the types of extraocular muscular dysfunction. For the distraction
group, information regarding duration and magnitude of distraction
as well as length of time in consolidation was collected.
Surgical Techniques
The senior authors’ (JAT) technique for FODO has been
described previously.8,9 Briefly, a coronal incision is performed
and all osteotomies that would be completed for an FOAR are
performed, except for the transverse osteotomy that separates the
bandeau from the frontal bone, without elevation of the frontal bone
flap off of the dura overlying the frontal lobe. The osteotomy of the
sphenoid wing and orbital roof is performed through a ‘‘pterional
window,’’ described by Choi et al,7 with protection of the intracra-
nial and orbital contents. The contralateral supraorbital and fore-
head osteotomies are performed in a ‘‘perforated’’ fashion with an
ultrasonic scalpel where the forehead deformity transitions. In
addition, the affected orbit undergoes radial osteotomies with an
ultrasonic scalpel to facilitate horizontal ‘‘unfurling’’ of the orbit
during distraction (Fig. 1A-B).
Distraction is started on postoperative day 2 at a rate of 1 mm per
day. The endpoint of distraction is based on a slight overcorrection
of the supraorbital region.
The technique for traditional FOAR consists of a classic bandeau
extending into the unaffected orbit with a bifrontal craniotomy and
subsequent reshaping and replacement of the free bone grafts
secured with resorbable plates and screws (Fig. 1C–E).
RESULTS
Sixteen patients (12 female, 4 male) underwent FODO for the
treatment of UCS during the study period, and 1 was excluded
due to multi-suture involvement (UCS þ sagittal). The remaining
15 patients were compared to the last 15 patients (11 female, 4 male)
who underwent traditional FOAR treatment of UCS during the same
period. The mean age of initial surgery was 6.3 months for the
FODO group and 9.8 months for the traditional FOAR group
(P < 0.05). Characteristics of the included patients are presented
in Supplementary Digital Content, Table 1, http://links.lww.com/
SCS/C732. Perioperative data is presented in Supplementary Digi-
tal Content, Table 2, http://links.lww.com/SCS/C732. Three com-
plications occurred in the FODO group. Two patients experienced
fracture of a distractor baseplate during the consolidation phase
with a very slight amount of relapse noted in one and none in the
other. There were 2 postoperative complications in the conventional
FOAR group: 1 wound infection that was treated with oral anti-
biotics and 1 delayed plate exposure 6 months postoperatively,
# 2021 Mutaz B. Habal, MD
Copyright © 2021 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
A Cohort Study of Strabismus Rates
FIGURE 1. (A) Anterior view of intraoperative appearance following
osteotomies and placement of distractor for right fronto-orbital distraction.
Note the contralateral perforating osteotomies and the radial osteotomies of the
affected orbit to facilitate unfurling of the orbit during the distraction process.
(B) Lateral view of postoperative appearance following osteotomies for right
fronto-orbital distraction and placement of distractor. (C) Anterior view of
markings for traditional 2/3 fronto-orbital advancement. (D) Superior view of
intraoperative appearance following advancement of fronto-orbital bandeau
with supraorbital strut in place. (E) Anterior view of intraoperative appearance
following replacement of the frontal bone to the advanced fronto-orbital
bandeau.
which was treated with local wound care followed by removal
of the plate in the office.
Mean distance distracted in the distraction group was 35 þ/–
8 mm with an average of 55 þ/– 24 days in consolidation. The
average age at distractor removal was 8.9 þ/– 3.6 months. The
removal procedure took an average of 34 þ/– 13 minutes of operative
time with an average blood loss of 19 þ/– 14 cc. The length of stay
associated with removal was on average 24 hours. The Whitaker scale
was used to grade the 1-year postoperative results. In the FODO
group, 13 patients were a Whitaker Grade 1 and 2 patients were a
Whitaker Grade 2. In the FOAR group, 13 patients were a Whitaker
Grade 1 and 2 patients were a Whitaker Grade 2.
The development of new-onset postoperative strabismus was
more common in those patients undergoing FOAR (Supplementary
Digital Content, Table 3, http://links.lww.com/SCS/C732). Five
(33%) patients in the FOAR group experienced a new diagnosis
of strabismus postoperatively, compared to only 1 (7%) patient in
the FODO group (P < 0.05). The most common finding was some
degree of esotropia in the eye contralateral to the side of synostosis.
One patient in each group required postoperative surgery for the
correction of strabismus. The other 4 patients with new-onset
strabismus in the FOAR group were treated with full-time glasses
(2), periodic eye patching with atropine drops (1), and periodic
patching (1). Detailed ophthalmologic findings are presented in
Supplementary Digital Content, Table 4, http://links.lww.com/
SCS/C732.
DISCUSSION
In this study, we report a difference in postoperative strabismus
rates with FODO as compared to FOAR with reasonable periopera-
tive morbidity and aesthetic results with both techniques in the
short-term. The rate of postoperative strabismus in traditional
FOAR treatment of UCS is variably high, and in our series it
was 33%, somewhere in the middle of the range that has been
reported.3 In contrast, strabismus only occurred in 1 patient in the
FODO group for a rate of postoperative strabismus of 7%.
The definitive answer as to what causes variably high rates of
strabismus in UCS is unknown, and is likely due to a combination of
the altered orbital bony anatomy, altered attachment of the trochlea,
and altered position of the extra-ocular eye muscles.2 In addition, a
high incidence of postoperative strabismus following traditional,
2363
Hoppe and Taylor The Journal of Craniofacial Surgery
open, FOAR is well documented and thought to be due to the same
combination of factors interacting with the changes of acute bony
repositioning and re-draping of the soft tissues that characterize
FOAR.3
Although it is not possible with available data to define causal-
ity, several factors may provide an explanation for the different
rates of strabismus between FOAR and FODO. It has been shown
that an earlier age of surgery for the correction of UCS results in a
decreased rate of ophthalmologic complications postoperatively.10
In our study, FODO was performed at an average of 3 months earlier
than FOAR. We typically choose to perform traditional FOAR for
UCS at an older age due to lower published rates of long-term
fronto-orbital retrusion and temporal hollowing.11 Simply changing
the timing of FOAR from an average of 23 months to less than
6 months has been shown to dramatically decrease the rate of
resultant strabismus.10 It is possible that the findings of this study
are due to an earlier age of intervention, rather than differing
techniques. An earlier intervention may prevent the development
of strabismus that often accompanies this deformity.
One significant difference between FOAR and FODO is acute
versus gradual repositioning of bone and soft tissue. In FODO
mediated reconstruction of UCS, subperiosteal stripping is per-
formed of the periorbita, but no movement of the bone is performed
acutely. Although disinsertion of the trochlea still occurs with
FODO as a result of this subperiosteal stripping there is no acute
movement of its bony insertion. Instead, the periorbita are gradually
expanded during the distraction process, which may allow for a
more gradual reorientation of the extraocular muscles.
The data presented in this study supports a favorable periopera-
tive profile for the correction of UCS with both treatment modali-
ties. FODO’s shorter operative time, even with addition of the
second procedure for device removal, represents an advantage
given the recent trend towards decreasing anesthetic exposure
during the first several years of life.12 It was also associated with
less estimated blood loss, and lower blood product transfusion with
a trend toward a decrease in hospital stay. Fronto-orbital distraction
osteogenesis mediated reconstruction allows surgery to be per-
formed at an earlier age which may be beneficial in order to
ameliorate issues associated with increased intracranial pressure,
including the poorly understood effects it may have on neuropsy-
chological outcomes.13– 17 Other advantages of distraction are well
understood including greater degree of movement and greater
stability of the advanced segment than with traditional single-stage
movements.18,19 Perhaps the most advantageous aspect of distrac-
tion is the gradual expansion of the soft tissue envelope surrounding
the bony segment. This soft tissue envelope ultimately restricts the
amount of advancement that may be achieved in traditional bony
movements. It has been shown in UCS that overcorrection of the
deformity is associated with an improved long-term aesthetic
outcome.20 Therefore it should be possible to perform a greater
degree of overcorrection with FODO, potentially resulting in an
improved long-term aesthetic outcome. The same gradual length-
ening of skin and fat may be transmitted to the trochlea and extra-
ocular eye muscles, thereby leading to lower rates of strabismus,
though causality cannot be concluded from this study.
A study performed by Choi et al7 found that FODO in the
treatment of UCS has the potential to correct the torsion seen in the
cranial base. Specifically, computed tomography scans obtained
postoperatively demonstrated a decreased cranial index of asym-
metry as measured from the cranial base. This represents an
important finding as correction of the cranial base may translate
to correction of the sphenoid wing deformity, which conveys the
deformities seen in the facial skeleton in UCS. This could poten-
tially decrease the rate of relapse/recurrence seen with traditional
FOAR methods of UCS correction.
2364
Copyright © 2021 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
 Volume 32, Number 7, October 2021
Our study does present several limitations. Our series represents
a rather small cohort of patients with UCS. This is limited by the
rarity of this disease as well as the limited experience we have with
this new treatment modality. Since the first report of our group’s
experience with this technique8 we have accrued enough patients to
show statistical significance with regards to operative time, blood
loss, and blood replacement. We have also demonstrated a
decreased rate of new-onset strabismus in FODO. We do not have
long-term results with regards to intracranial morphology and
aesthetic outcomes. The senior author’s (JAT) practice is to perform
a computed tomography scan only when clinically indicated in
order to minimize patients’ exposure to ionizing radiation. As a
result, we do not have routine postoperative imaging that would
provide meaningful measures of outcomes. Our aesthetic evaluation
is limited to our relatively short-term follow-up and the Whitaker
rating scale, which is inherently subjective in nature. Finally,
ophthalmologic outcomes are measured at limited intervals and
over a relatively short period of time, representing a poor evaluation
of the effect either of these surgical procedures has on long-term
extraocular muscle function. That said, most studies evaluating
strabismus rates in UCS have found strabismus to be present within
1 year of surgery.2 –4,6,7
CONCLUSIONS
Fronto-orbital distraction osteogenesis for the treatment of isolated
UCS is associated with a favorable perioperative morbidity profile
and decreased incidence of postoperative strabismus compared with
traditional FOAR in this single surgeon series. It is unclear whether
these findings are a result of the technique itself or a simply an
earlier age at intervention. The promise of FODO is tempered by the
need for an additional procedure for removal of the device and lack
of long-term follow-up data on the stability of the advancement.
Further investigation is warranted on both of these fronts.
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Relaxing by meditation.

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