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ORIGINAL ARTICLE

Endoscopic Versus Open Total Vault Reconstruction of


Sagittal Craniosynostosis
Sultan Z. Al-Shaqsi, MBChB, PhD, Nicole Wing Lam, Hon,y
Christopher R. Forrest, MD, MSc, FRCS(C), FACS,y and John H. Phillips, BSc, MA, MD, FRCSCy

Sagittal craniosynostosis is the most common form of congenital


cranial deformity. Surgical interventions are performed either open
C raniosynostosis is a spectrum of cranial deformities caused by
variable premature ossification of cranial sutures leading to
restriction in skull growth that could cause functional abnormali-
1,2
or endoscopic. Advancements in minimally invasive surgery have ties. Cranial development is a complex process and any disrup-
enabled the development of the endoscopic suturectomy technique. tion could lead to complex deformities. Surgical correction of such
This is contrasted to the traditional open cranial vault reconstruc- deformities is indicated for functional or aesthetic concerns. There
tion. There is a paucity of data comparing the head shape changes is a predictable pattern of head shape deformity depending on the
suture involved. Sagittal craniosynostosis is an abnormal elongated
from both techniques. This study aims to compare the morphologi-
shape of the skull secondary to premature fusion of sagittal suture.3
cal outcome of endoscopic suturectomy versus total cranial vault It is the commonest form of nonsyndromic craniosynostosis. It is
reconstruction. estimated to occur in 1 in 5000 live births with a 3 to 1 ratio of male
Methods: This is a retrospective comparative study involving 55 to female predilection.3,4
cases of sagittal craniosynostosis, 37 of which has open total cranial For children with a confirmed diagnosis of isolated sagittal
vault reconstruction and 18 had endoscopic suturectomy procedure. craniosynostosis, surgical correction is indicated for functional or
Preoperative and postoperative 3D photographs of both groups were morphological concerns.5 The surgical intervention involves
analyzed and compared. The change in correction between removing the abnormally fused suture and allowing the cranium
preoperative and postoperative state was measured against a to expand in order to accommodate the growing brain.6 Hence,
crowd-driven standard for acceptable head shape. cranial vault reshaping procedures have shown to have an impact on
the neurodevelopmental outcome including speech and language,
Results: Total cranial vault had higher percentage change between
visuospatial skills, memory, and attention.7–9 The optimal timing
pre and postoperative cranial index than endoscopic suturectomy and extent of suture release continues to be controversial.10,11
(14.7% versus 7.7%, P ¼ 0.003). However, both techniques were Traditionally, surgeons have thought that a wide suture release
able to achieve the minimum standard of 70% correction (TCV and a total cranial vault reshaping was required to allow the growing
107.5%, ES 100.4%, P ¼ 0.02). brain to expand and minimize restriction. Since the introduction of
Conclusion: Total cranial vault and endoscopic suturectomy are minimally invasive endoscopic suturectomies at an early age,
effective in correcting scaphocephaly among children with sagittal limited and targeted suturectomies have been shown to be as
craniosynostosis. Additionally, both techniques are able to achieve effective as total extensive release in terms of brain growth vol-
a percentage correction that exceeds the 70% benchmark ume.12 Therefore, the 2 broad categories of surgical intervention are
established by the lay public. either endoscopic suturectomy or total vault reshaping depending
on the age of diagnosis.
Total cranial vault remodeling (TCVR) is a well-established
Key Words: 3D, cranial vault, craniosynostosis, endoscopic, surgical technique for the management of craniosynostosis.9,10
outcome, reconstruction, sagittal, scaphocephaly The critical steps include removing the cranial bone with the affected
suture, reshaping the bone grafts and reorganizing the reshaped grafts
(J Craniofac Surg 2021;32: 915–919) in a way that ensures expansion of the cranial dimension in the most
constricted regions. The reshaped bony segments are stabilized by
means of internal fixation. Small areas of excess space are created by
this re-organizing procedure and the growing brain fills such space.13
From the Plastic and Reconstructive Surgery, University of Toronto, The Despite significant advances in surgical technique, fixation methods
Hospital for Sick Children, Ontario; and yThe Hospital for Sick Children, and anesthesia, total cranial vault reconstruction has a well-docu-
Toronto, Canada. mented relatively high rate of blood loss requiring transfusion,
Received May 17, 2020.
prolonged hospital, and intensive care stay.14–17
Accepted for publication October 27, 2020.
Address correspondence and reprint requests to Sultan Z. Al-Shaqsi, Endoscopic suturectomy, first reported in the literature in 1998,
MBChB, PhD, Plastic and Reconstructive Surgery, University of as advances in minimally invasive surgery continues to evolve.12,16
Toronto, The Hospital for Sick Children, 555 University Avenue, ON It provides a minimally invasive correction of craniosynostosis. It
M5G 1X8, Canada; E-mail: sultan.al-shaqsi@mail.utoronto.ca relies on the malleability of skull bones to expand and remodel after
The authors report no conflicts of interest. the procedure and therefore usually recommended for children
Supplemental digital contents are available for this article. Direct URL before the age of 6 months.18
citations appear in the printed text and are provided in the HTML and The 2 techniques have been extensively compared and con-
PDF versions of this article on the journal’s Web site (www.jcraniofa- trasted in the literature. Although total cranial vault reshaping is
cialsurgery.com). effective in correcting head shape deformity in sagittal craniosyn-
Copyright # 2020 by Mutaz B. Habal, MD
ISSN: 1049-2275 ostosis, it is associated with increased blood transfusion rates,
DOI: 10.1097/SCS.0000000000007307 length of operative time, and length of stay in hospital compared

The Journal of Craniofacial Surgery  Volume 32, Number 3, May 2021 915
Copyright © 2021 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
Al-Shaqsi et al The Journal of Craniofacial Surgery  Volume 32, Number 3, May 2021

to endoscopic interventions such as endoscopic suturec- the 3D photos, the head surface was selected and transferred as the
tomies.14,15,19,20 Hence it is usually associated with higher overall Wavefront Object (.obj) file extension format into the Canfield
cost than endoscopic procedures. Studies have estimated the aver- Mirror database (Canfield; Fairfield, NJ). All 3D photographs were
age cost difference between endoscopic and total vault to be then oriented in the Frankfort horizontal plane by rotating and
between CD $11,603 to $31,744 per case.21,22 Endoscopic proce- translating it such that the right tragion and right lower orbitale were
dures are shown to be less invasive, as they are associated with situated on the horizontal plane, eyes leveled, and the cranium
lower complication rate, shorter hospital stay, and have a faster bisected on the sagittal plane. Once orientation is established,
recovery time than total cranial vault reshaping.19 However, for standard anthropometric lengths which include glabella to opistho-
endoscopic release to be effective, it has to be performed while skull cranion (g-op), most anterior point on the cranium to the opistho-
bones are still malleable, which is before 6 months of age.9,10,23 If cranion (ap-op), left to right euryon (eu-eu) were calculated. All
performed after 6 months, the effectiveness of endoscopic release is measurements were executed by the same investigator. Cranial
reduced, and total vault reshaping is usually required. Index was calculated from the (eu-eu) divided by (ap-op)  100.26
Public perception of head shape is an essential outcome measure
that is usually underestimated.24 Studies have shown that children Statistical Analysis
with cranial deformities are perceived ‘‘differently’’ by the public.25 Anonymous extracted data variables were entered into a pre-
Such issues are important to investigate to mitigate and reduce populated Microsoft Excel (Microsoft Corporation, Redmond,
social stigma associated with cranial deformities. A recent crowd WA). Descriptive analysis was used to present the study results
sourcing study has shown that the public considers at least 70% and patient demographics. Subsequently, the main outcome mea-
correction in sagittal craniosynostosis is needed in order for the sure (change in head shape) was analyzed by comparing the
public to consider the head shape as ‘‘normal.’’24 Studies have percentage change and absolute change in Cranial Index between
focused on comparing the perioperative differences and outcomes the preoperative and postoperative 3D photographs through a
between the 2 techniques. There is currently a paucity in evidence Pearson Student t test analysis. A percentage correction to normal
comparing the long-term outcome of the 2 techniques (ie, endo- cranial index was determined for each patient, adjusting for age and
scopic versus open total vault reconstruction). A study by Le et in sex. An alpha of 0.05 was used to determine statistical significance.
2014 looked at the long-term head shape correction between 13 All data were de-identified and Statistical analysis was performed
patients who underwent open vault reconstruction compared to 6 using Statistical Package for the Social Sciences (SPS, IBM Corp,
cases of endoscopic suturectomy indicated equivalence head shape Armonk, NY). Approval for this study was obtained from the
correction.20 This study is limited by small numbers of cases and Hospital for Sick Children ethics board. This study was conducted
lack of benchmark in which the outcome was measured against. in accordance with Helsinki principles of medical research.27
Therefore, this article presents a relatively larger sample of patients
and presents a validated benchmark correction percentage in which
the two techniques can be measured against. RESULTS
There were 55 sagittal craniosynostosis patients that were identified
for this study. Eighteen patients had undergone endoscopic suture-
METHODOLOGY ctomy and 37 patients had undergone total cranial vault reconstruc-
tion. Males constituted 81.5% (n ¼ 44) of the total sample size. In the
Clinical Materials and Methods endoscopic group, males constituted 82.4% (n ¼ 14) and in the total
A retrospective review was performed on all children diagnosed cranial vault reconstruction group, males constituted 81.1% (n ¼ 30).
with sagittal craniosynostosis and underwent either endoscopic The mean age of presentation was 10.0 ( 16.3) months, 2.9 ( 2.8)
suturectomy or total cranial vault reconstruction at the Hospital months for endoscopic and 13.4 ( 19.2) months for total cranial
for Sick Children, Toronto, Canada between 2009 and 2019. vault reconstruction (P ¼ 0.01). The mean age of operation was 12.3
Patients for this study were recruited from a departmental database ( 16.9) months, 3.6 ( 2.7) months for the endoscopic group, and
that collects clinical and 3D photographs of all craniofacial patients. 16.6 ( 19.2) months for the total cranial vault group (P ¼ 0.02). The
The inclusion criteria for this study group was defined as mean age for follow-up in the endoscopic group was 5.5 ( 3.7)
patients who were clinically diagnosed with sagittal craniosynos- months, whereas in the total cranial vault reconstruction group the
tosis and had 3D photographs taken to document their preoperative mean age of follow up was 20.7 ( 20.3) months. Despite corrected
and postoperative head shape following either endoscopic suture- cranial index values following surgery in our patient group, in the
ctomy or total cranial vault reconstruction performed by craniofa- total cranial vault reconstruction group, 10.8% (n ¼ 4) of patients had
cial surgeons at the Hospital for Sick Children. Children without pre to undergo titanium mesh addition to compensate for soft spots and
or postoperative 3D photographs were excluded. There were 55 2.7% (n ¼ 1) underwent secondary surgical intervention. From our
identified patients who met the study inclusion criteria of which 18 patient group, 7.2% (n ¼ 4) of patients were syndromic. This
patients had undergone endoscopic craniectomy and 37 patients had included Jacobsen syndrome, Xia-Gibbs Syndrome, and FGFR2
undergone total cranial vault reconstruction. Mutation from the total cranial vault reconstruction cohort and
Chart review of all cases was conducted, and variables were Xp22.33p22.32 deletion from the endoscopic craniectomy group.
extracted. Variables including demographics, clinical presentation, All patients were clinically diagnosed with isolated sagittal cranio-
timing of surgical intervention, surgical technique, date of 3D synostosis through the use of computed topography.
photographs, and medical and surgical history were extracted from Supplemental Table, http://links.lww.com/SCS/C133 one
medical records. describes the main outcome measured in this article. Cranial index
For each case a detailed analysis of the preoperative and was compared between the 2 treatment groups pre- and postopera-
postoperative 3D photographs was done using a standardized tively. This study found a significant difference (P ¼ 0.02) in
method which was previously published.33 All 3D photographs measured cranial index between endoscopic suturectomy
were captured by a trained technician using 3dMD Face System (74.1  4.4) and total vault reconstruction group (79.3  10.0)
(3dMD; Atlanta, GA). A single tightly fitted nylon swimming cap postoperatively. Both surgical groups displayed successful surgical
was used on the subject’s head to flatten the hair to provide better expansion as an increase in cranial index was observed among both
resolution of the cranial vault shape. After technical refinement of surgical groups. However, on average total vault reconstruction

916 # 2021 Mutaz B. Habal, MD

Copyright © 2021 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
The Journal of Craniofacial Surgery  Volume 32, Number 3, May 2021Which Technique Leads to Greater Change in Head Shape?

FIGURE 1. Preoperative photos of a female child with sagittal craniosynostosis FIGURE 3. Preoperative photos of a female child with sagittal craniosynostosis
at 6 months of age. at 4 months of age.

technique has significantly higher percentage change in calculated unit (ICU) stay.14,30 However, the degree of correction and the subtle
CI compared to the endoscopic group (TCVR ¼ 14.72  14.95, differences in head shape between the 2 techniques have not been
ES ¼ 7.75  5.99, P ¼ 0.03). This study found that both procedures investigated against a standardized outcome in studies and hence the
meet and exceed the benchmark correction of 70% in that on objective of this project. Hence, this study is unique in that it measures
average the endoscopic provides about 100.4% correction in cranial the effectiveness of TCVR versus ES against a publicly generated
deformity and open total vault provides on average 107.5%. There- crowds standard that reflects the acceptability of head shape by
fore, the mean difference in percentage correction between open laypersons.24
total cranial vault reconstruction and endoscopic suturectomy is As expected, this series showed that children with sagittal cra-
estimated to be 7% (P ¼ 0.02). (Supplemental Table 1, http:// niosynostosis who undergo TCVR are older than those who undergo
links.lww.com/SCS/C133) ES. This is similar to other published literature and reflects an
An example of sagittal craniosynostosis in a female child is inherent selection bias as the 2 procedures are indicated for different
shown in Figure 1. This has been surgically corrected by total age-groups of children. Endoscopic repair can only be performed,
cranial vault reconstruction at the age of 10 months and the whereas the skull bones are still amenable to molding likely within
immediate postoperative results at the age of 12 months are shown 4 months of age.9 A recent study by Melin et al found that of 88 cases,
in Figure 2. In contrast an example of a female child with sagittal ES were generally younger than those who underwent TCVR (3.8
craniosynostosis is shown in Figure 3. This patient underwent an versus 14.0 months).17 Although our study represents endoscopic
endoscopic suturectomy at the age of 5 months and helmet therapy cases for sagittal craniosynostosis, the study by Melin et al represents
thereafter. The postoperative results at the age of 12 months are all types of synostosis and found that type of craniosynostosis is not a
shown in Figure 4. factor in average age at the time of the procedure.17
The main finding from our study is that endoscopic suturectomy
DISCUSSION and open total vault reconstruction were both effective in correcting
Sagittal craniosynostosis is arguably the most common cranial vault the preoperative cranial deformity accounted for by cranial index
deformity that craniofacial surgeons deal with.28 Indications for measures (P < 0.05). This finding is consistent with published
surgical intervention in sagittal craniosynostosis children are based literature. A study by Le et al reviewed 46 cases of sagittal
on whether any functional deficits have been detected and whether the craniosynostosis, 13 open total vault reconstruction, and 33 endo-
aesthetic deformity warrants a surgical correction. Both endoscopic scopic with a mean follow-up of 97 months.20 The average age at
suturectomy with subsequent molding helmeting or open total vault surgery for the open group was 5.1 months compared to 3.3 for the
reconstruction have been shown to be equally effective.29 Despite endoscopic group.20 They found no significant difference in post-
their effectiveness, both techniques incur a significant health care cost operative length, width, Cranial Index, and cranial volume mea-
and have well documented complications such as bleeding requiring surements between the 2 groups reinforcing that both procedures
blood transfusion leading to prolonged hospital and intensive care seem to be equally effective in correcting the cranial deformity of

FIGURE 2. Postoperative photos of the child in Figure 1 at age 12 months after FIGURE 4. Postoperative photos of the child in Figure 3 at age 12 months after
undergoing total cranial vault reconstruction. undergoing endoscopic suturectomy and helmet therapy.

# 2021 Mutaz B. Habal, MD 917


Copyright © 2021 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
Al-Shaqsi et al The Journal of Craniofacial Surgery  Volume 32, Number 3, May 2021

sagittal craniosynostosis.20 Moreover, this study highlights that surgical techniques are offered to children with sagittal craniosyn-
unlike some reports that suggest surgery causes diminished cranial ostosis. This study showed that both techniques are effective in
growth by inducing synostosis in other cranial sutures, in this series correcting the head shape. More importantly, both techniques
the cranial volume is within normal limits for age and sex.15,20 confer significant improvement in head shape that is considered
The protocol for helmet therapy at our institution entails use for ‘‘normal’’ and meets the benchmark correction set by lay people.
4 to 6 months or until the patient is 12 months of age. The helmet is Based on current available evidence, endoscopic repair seems more
worn for 23 hours of the day and adjustments are made every 3 to 4 cost-effective and as equivalent to total cranial vault reconstruction.
weeks. Despite concerns regarding patient tolerance and skin It is still important to appreciate that total vault reconstruction is
irritation as complications of helmet therapy, this study indicates indicated for older children or where the deformity mandates a
that helmet therapy following endoscopic suturectomy enables wider surgical exposure to correct the deformity or where mallea-
comparable corrective results to TCVR methods.31–33 Until further bility of the skull bones cannot be harnessed with endoscopic repair.
studies are conducted to indicate the inefficacy of helmet therapy, Future research could address whether lay people are able to
our institution recommends the use of orthotic helmets following distinguish between the outcome of the 2 techniques.
ES among sagittal synostosis patients.
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918 # 2021 Mutaz B. Habal, MD

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