Original Article

The Cleft Palate-Craniofacial Journal

1-8
One-Year Treatment Outcome of Profile ª 2021, American Cleft Palate-
Craniofacial Association

Changes After Transcutaneous Maxillary Article reuse guidelines:

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DOI: 10.1177/10556656211005638
Distraction Osteogenesis in Growing journals.sagepub.com/home/cpc

Children With Cleft Lip and Palate

Wei-Ling Gao, DDS1 , Yi-Hao Lee, DDS1, Chi-Yu Tsai, DDS1,

Te-Ju Wu, DDS, MS1, Jui-Pin Lai, MD2, Shiu-Shiung Lin, DDS, MS1,
and Yu-Jen Chang, DDS, MS1

Abstract
Objective: To evaluate the long-term stability of LeFort I osteotomy followed by distraction osteogenesis with a transcutaneous
rigid external device for the treatment of severe maxillary hypoplasia in patients with cleft lip and palate.
Patients and Methods: Nine patients with cleft lip and palate underwent rigid external distraction after a LeFort I osteotomy for
maxillary advancement. Lateral cephalometric films were analyzed for assessment of treatment outcome and stability in 1 month,
6 months, and 1 year after distraction.
Results: Significant maxillary advancement was observed in the horizontal direction, with the anterior nasal spine (ANS) distance of
the maxilla increasing by an average of 20.5 + 5.1 mm after distraction. The ANS relapse rates in 6 months and 1 year were 8.7%
and 12.8%, respectively. The mean inclination of upper incisors to the palatal plane was almost unchanged (before: 109.8 + 6.6 ;
after: 108.9 + 7.5 ). The movement ratios at the nasal tip/ANS, soft tissue A point/A point, and the upper vermilion border/
upper incisor edge were 0.36:1, 0.72:1, and 0.83:1, respectively.
Conclusion: Considerable maxillary advancement was achieved with less change of incisors inclination after distraction. Moreover,
the relapse rate after 1 year was minimal. The concave facial profile was improved as well as the facial balance and aesthetics.

Keywords
cleft lip, cleft palate, rigid external device, transcutaneous maxillary distraction, long-term stability, soft tissue profile

Introduction demonstrated by Hierl and Hemprich in 1999 (Hierl & Hem-

Distraction osteogenesis (DO) was first applied by Codivilla in
1905 to lengthen the short femurs with the technique of gradual
distraction (Codivilla, 1905). Since then, this technique has
been widely utilized to treat uneven extremities. The first appli-
cation on craniofacial treatment was in 1992 when McCarthy
successfully lengthened the mandible by 18 to 24 mm in 4 chil-
dren with Pierre Robin sequences (McCarthy et al., 1992).
Nowadays, this treatment modality is also applied to treat
patients having cleft lip and palate with severe maxillary hypo-
plasia maxilla, which can only be managed by orthognathic
surgery, even after primary surgery by experienced cleft sur-
geons. In 1997, Polley and Figueroa first applied the DO with a
rigid external distraction (RED) device to treat patients with
cleft lip and palate (Polley & Figueroa, 1997). The combination
of the RED device with an intraoral skeletal anchorage was
prich, 1999). They carried out a midface DO by utilizing
intraoral miniplates and micromesh in treating an edentulous
adult patient with cleft lip and palate. Bone-borne RED is an
effective alternative method that can be applied at different
1
Department of Craniofacial Orthodontics, Department of Dentistry,
Kaohsiung Chang Gung Memorial Hospital and Chang Gung University
College of Medicine, Kaohsiung
2
Department of Plastic and Reconstructive Surgery, Kaohsiung Chang Gung
Memorial Hospital and Chang Gung University College of Medicine,
Kaohsiung
Corresponding Author:
Yu-Jen Chang, Department of Orthodontics, Kaohsiung Chang Gung Memorial
Hospital, No. 123, 3F, Ta Pei Road, Niaosong District 833, Kaohsiung.
Email: orthodontist.chang@gmail.com
2 The Cleft Palate-Craniofacial Journal XX(X)

Table 1. Patient Characteristics. d) was started after a latency period of 7 days after surgery. The
DO process lasted for around 1 month and was terminated when
Period of Period of
Age, distraction, maintain, A-Nv, OJ,
the desired skeletal relationship was achieved. The RED devices
No. Gender year days days mm mm were removed after an 8-week consolidation period.
Lateral cephalometric films were collected 1 week before (T0),
1 M 12 21 64 8.3 3.2 immediately 1-month after the termination of DO (T1), 6-month
2 F 12.5 46 64 12.4 4.7
after (T2), and 1-year after (T3) distraction. Selected skeletal,
3 M 11.2 33 85 12.8 16
4 F 11.5 22 76 10 3.7 dental, and soft tissue landmarks are listed in Appendix A. The
5 F 11.8 47 71 7.2 13.2 definitions of all the landmarks and reference lines (Appendix A;
6 F 11.3 23 66 11 4.6 Figure 1) followed those by Lin et al. (2012). The AudaxCeph
7 M 11.4 35 61 17 13.2 Empower VER5.2 was used to analyze changes among the above
8 M 11.2 21 83 8.5 7.7 4 stages. First, we set up an x-y coordinate system on the pre-
9 F 11.4 33 85 12.4 10.1 operative cephalogram by fabricating a line 7 below the
Mean 11.6 31.2 72.8 11.1 6.7
sella-nasion (S-N) plane as the x-axis, and we used the sella point
Abbreviations: F, female; M, male; OJ, overjet. as the original point. Then we drew a line perpendicular to the
x-axis by intersecting the line at the sella for the y-axis. We
superimposed the cephalometric films to record the changes
levels of skeletal fragmentation in various osteotomies includ- between each phases. Changes were assessed by applying linear
ing LeFort I, II, and III (Monaghan et al., 2002). In contrast to and angular parameters of skeletal and dental structures.
orthognathic surgery, the greatest advantage of DO is the abil- Measurements were taken by 1 examiner (Wei-Ling Gao).
ity to improve the growth of bony defects along with the simul-
taneous possibility of expansion of soft tissues (Rachmiel et al.,
1998). In the study by Loboa et al. (2004), the neoformation of Error Study
mesenchymal tissues was triggered by the distraction device in A cephalometric tracing and all measurement assessment were
a regular rhythm, which caused little trauma to the tissues and taken by 1 researcher. Reproducibility of the reference lines
activated new bone formation. was verified by comparing the angle between the S-N plane and
Swennen et al. (2001) reviewed 16 studies that discussed 7 below the S-N plane in all the cephalograms in 2 occasions
maxillary distraction with tooth-borne anchorage in with a 1-month interval. There was no statistical difference in
121 patients. The results revealed maxillary advancement of defining the reference lines among the cephalometric tracing.
1 to 17 mm, and although a relapse was noted in 65 patients, The method of evaluating the systematic and random errors
there was no clear data regarding the degree/rate of relapse. in the tracing process followed the study by Houston (1983),
In contrast, the bone-borne group demonstrated an average of and the tested null hypothesis showed that there was no differ-
11 mm advancement at the A point with good stabilities in ence between the first and the second measurements.
nongrowing patients. Further improvement for better adapta-
tion of the skeletal anchorage was subsequently introduced,
including a combination of RED with transcutaneous skeletal Results
anchorage to reduce the patients’ discomfort as well as to gain
better control of distraction. The average period of distraction was 31.2 + 10.3 days, and
To reduce tooth complications from tooth-borne RED, we the average maintenance period was 72.8 + 9.7 days. The
used a direct bony attachment for the advancement of the max- mean predistraction overjet was 6.7 + 4.8 mm, with a range
illa. This study aimed to evaluate the long-term stability of of 13.2 to 3.2 mm. At the end of the distraction, the mean
LeFort I osteotomy followed by DO with skeletal anchorage overjet was 4.6 + 2.6 mm, with a range of 2.1 to 8.4 mm,
via transcutaneous RED device for treating patients having which increased by an average of 13.5 + 4.9 mm (Table 1).
cleft lip and palate with severe maxillary hypoplasia. All skeletal and dental angular/linear changes between T1 and
T0 are shown in Table 2.

Materials and Methods

Patients
In this study, all the 9 patients with cleft lip and palate received
primary lip/palate repair and alveolar bone graft in Kaohsiung
Chung Gung Memorial Hospital. However, they developed a
severe class III skeletal pattern with significant midface hypo-
plasia that required DO. All patients (4 boys, 5 girls; average
age: 11.6 + 0.4 years old; range: 11.2-12.5 years old) underwent
LeFort I osteotomy and RED device installation by the same
surgeon (Table 1 and Figure 1). Distraction (at a rate of 1 mm/
Skeletal Change (T1–T0)
The initial skeletal discrepancy (A-Nv) of the maxilla in the
horizontal direction was 11.1 + 3.0 mm, with a range of
7.2 to 17.0 mm. The mean increase was 19.9 + 7.0 mm
at the end of the distraction. The maxilla showed significant
advancement in the horizontal direction and moved inferiorly.
The position changes of landmark in ANS and A point were
significant: ANS (horizontal: 20.5 + 5.1 mm; vertical:
1.8 + 5.5 mm) and A point (horizontal: 20.4 + 5.0 mm;
vertical: 3.6 + 8.6 mm).
Gao et al
Figure 1. Eleven-year-old girl (case no. 4) with complete bilateral left cleft lip and palate and maxillary deficient facial profile was treated with
maxillary lefort I osteotomy and distraction osteogenesis to improve facial profile (A and B) before surgery; (C and D) during distraction; (E and
F) immediately after distraction; (G and H) after 1-year review; and (I) cephalometric landmarks and reference lines selected in this study. (The
definitions of landmarks are listed in Appendix A).
The mandible showed mild advancement in the horizontal
direction, although the vertical position varied from case to
case. The average records of position changes of mandible
were: B point (horizontal: 2.7 + 1.9 mm in absolute value;
vertical: 2.1 + 5.6 mm in absolute value) and Pog (horizon-
tal: 2.7 + 2.9 mm in absolute value; vertical: 0.4 + 5.0 mm
in absolute value). The distraction moved the maxilla for-
ward, and the direction of distraction induced a clockwise
rotation of the mandible (in the downward and backward
directions).
Dental Change (T1–T0)
The upper incisor edge showed great anterior advancement
(horizontal: 17.4 + 5.1 mm; vertical: 4.4 + 5.5 mm). Mean-
while, the upper molar showed similar anterior movement as
that of the incisors (horizontal: 13.6 + 7.5 mm; vertical: 3.5 +
4.1 mm), indicating a minimal side effect over the dental
anchorage. Furthermore, the inclination of the upper incisors
to the palatal plane showed little change after distraction
(T0: 109.8 + 6.6 ; T1: 108.9 + 7.5 ), which indicates min-
imal dental side effect over the frontal dentition.
Relapse Rate
After 6 months, ANS showed a horizontal relapse by 1.8 +
2.4 mm and an inferior change by 0.4 + 3.2 mm on aver-
age. The relapse rate was 8.7% in the horizontal direction
3
and an increase of 24.1% in the vertical direction. A point
showed a backward change by 2.3 + 2.0 mm on average
and a superior change by 1.3 + 3.7 mm. The relapse rate
was 11.2% in the horizontal direction and 35.5% in the
vertical direction. After 12 months, ANS showed a horizon-
tal relapse by 2.6 + 2.3 mm and a superior change by 2.2
+ 5.6 mm on average. The relapse rate was 12.8% in the
horizontal direction and an increase of 22.2% in the vertical
direction. A point showed a backward change by 2.1 + 3.0 mm
on average and a superior change by 0.9 + 4.1 mm. The
relapse rate was 10.1% in the horizontal direction and 23.9%
in the vertical direction (Table 3).
Correlation Between Soft Tissue and Hard Tissue
In the maxilla, the correlation coefficient ranged from 0.22 to
0.46 on the horizontal plane and 0.12 to 0.34 on the vertical
plane. The forward movements of the soft and hard tissues were
correlated, but there was no significant correlation (0.12  r 
0.46). The ratio for the horizontal movement at the level of the
nasal tip/ANS, the soft A point/A point, and the upper vermi-
lion border/upper incisor edge was 0.36:1, 0.72:1, and 0.83:1,
respectively (Table 4).
Discussion
Distraction osteogenesis is a surgical procedure that is easy to
control. It can activate a regeneration process and start a
4 The Cleft Palate-Craniofacial Journal XX(X)

Table 2. Cephalometric Measurement of Distraction (T1–T0) parallel-fibered bone commences. The duration of the distrac-
Changes. tion phase depends on the degree and severity of the skeletal
T0 mean + T1 mean + T1–T0 mean
deformity. However, the rhythm and rate of distraction are
Variables SD, mm SD, mm + SD, mm P more critical compared with the duration. A distraction rate
of 1 mm/d is a common standard used in most studies; hence,
Skeletal we used the same criterion in all our patients and obtained good
***
SNA,  72.3 + 7.6 93.0 + 7.9 23.2 + 9.0
results. The best rhythm for DO is a continuous steady-state
SNB,  71.3 + 7.6 76.9 + 6.4 3.0 + 4.7 .09
ANB,  10.3 + 3.9 10.3 + 5.2 20.3 + 4.9 *** separation of the bone fragments (Sultan, 2016). After the dis-
A-Nv, mm 11.1 + 3.0 17.2 + 7.6 19.9 + 7.0 *** traction bone length is acceptable, the activation of distraction
***
OJ, mm 6.7 + 4.9 4.6 + 2.4 13.5 + 4.9 will be terminated. Subsequently, the consolidation phase
**
OB, mm 3.7 + 4.7 8.3 + 3.0 2.1 + 4.4 begins, wherein the bone and extensive amounts of osteoid
***
ANS (X), mm 51.8 + 5.8 70.6 + 4.6 20.5 + 5.1 undergo mineralization and eventual remodeling.
ANS (Y), mm 47.8 + 4.1 48.5 + 7.6 1.8 + 5.5 .354 Consolidation is a period after the end of the distraction when
***
A(X), mm 51.6 + 5.6 69.4 + 5.2 20.4 + 5.0
the mobile fragments are stabilized in the preplanned position.
A(Y), mm 43.3 + 3.8 47.3 + 9.3 3.6 + 8.6 .239
B(X), mm 63.8 + 8.3 65.9 + 7.5 2.7 + 1.9 ** The distractor is kept to be passive, and it combines with the
B(Y), mm 76.0 + 7.4 74.5 + 9.0 2.1 + 5.6 .303 whole rigid fixation device to act as a “retainer.” This period
*
Pog(X), mm 65.3 + 10.0 67.8 + 9.4 2.7 + 2.9 varies from 8 to 12 weeks to allow mineralization of callus to
Pog(Y), mm 87.9 + 7.5 90.1 + 10.6 0.4 + 5.0 .821 occur across osseous gap. The consolidation phase is essential in
Dental bone healing and should be long enough to ensure the sufficient
***
U1i(X), mm 53.3 + 7.9 70.7 + 7.5 17.4 + 5.1 reunification of the bony fragments. It is logical that the longer
U1i(Y), mm 61.4 + 3.8 64.9 + 6.3 4.4 + 5.5 .349
phase of consolidation may guarantee the less relapse potential
U1-SN,  101.3 + 9.5 95.5 + 7.0 6.9 + 11.6 .114
U1-PP,  109.8 + 6.6 108.9 + 7.5 0.9 + 6.3 .633 of bony fragments. However, a longer consolidation phase may
L1-Md,  69.8 + 8.1 77.3 + 7.6 5.9 + 4.3 .232 also lead to some complications, such as mucosal dehiscence,
Soft tissue infection, fracture, pin-track ulcers and associated discomfort,
*
Pn0 (X), mm 77.9 + 5.3 85.4 + 3.7 7.6 + 3.5 and displacement of the transported segment (Natu et al., 2014).
Pn0 (Y), mm 34.0 + 6.3 29.9 + 5.3 4.0 + 5.7 .342 To balance the outcome stability and potential complications
**
Sn0 (X), mm 67.0 + 6.8 80.1 + 3.2 12.9 + 4.5 during the consolidation phase, all of our cases had an 8-week
Sn0 (Y), mm 45.5 + 3.4 41.9 + 4.5 2.7 + 4.3 .453
*** consolidation period. It is also indicated in our study that the
A0 (X), mm 67.2 + 6.5 82.1 + 3.3 14.7 + 4.7
A0 (Y), mm 50.7 + 4.4 49.1 + 5.6 1.0 + 3.9 .659 amount of bony relapse should be taken into account and applied
Ls0 (X), mm 70.7 + 7.0 85.2 + 4.4 14.4 + 4.0 *** for overcorrection during distraction period to achieve the
Ls0 (Y), mm 56.9 + 5.3 58.0 + 6.8 2.0 + 5.0 .765 desired result. The further relapsed condition of longer follow-
up should be investigated in the future.
Abbreviations: OB, overbite; OJ, overjet; SD, standard deviaition; T0, before
treatment; T1, after distraction; T1–T0, pre- and postdistraction changes; (X),
The first historical use of RED in the skeletal anchorage was
horizontal change; (Y), vertical change. performed in an adult female having cleft lip and palate with a
*P value <0.05. 20 mm horizontal advancement and a 13 mm movement infer-
**P value <0.005. iorly (Hierl & Hemprich, 1999). However, no long-term
***P value <0.001.
follow-up was conducted. The average sagittal advancements
in our study were 20.5 mm in ANS and 20.4 mm in A point.
Our results were similar to those of previous studies but
mechanical enhancement of biological responses to the injured revealed greater distraction range than those reported in previ-
tissues and, consequently, lead to new bone formation. Distrac- ous studies either with dental anchorage or with skeletal ancho-
tion osteogenesis contains 3 phases: latency, distraction, and rage. Cheung and Chua reviewed 26 papers regarding
consolidation (Ai-Aql et al., 2008). The initial trauma brought maxillary distraction in 276 patients with cleft in 2006. Most
by surgery requires a latency phase so that the initiation of of the results on maxillary horizontal advancement were only
primary healing can occur. This period generally lasts for in the range of 5 to 9 mm (Cheung & Chua, 2006).
3 to 5 days. If the latency period is longer than 10 to 14 days, The direction of the distracted maxilla in our study was
a premature bone union tends to make DO difficult. During the managed to gain a backward rotation of the mandible so that
distraction phase, tensile forces are applied to the callus base in their concave class III profile could be simultaneously
a rhythm and at a particular rate. When the callus is stretched, a improved. Our goal for the RED treatment was not only to
central fibrous zone called the “fibrous interzone” forms, obtain significant improvement of the maxillary deficiency,
which is a zone full of chondrocyte-like cells, fibroblasts, and which is the main problem in most patients with cleft lip and
oval cells (the intermediate cells between fibroblasts and chon- cleft palate (CLCP), but also by controlling the direction of
drocytes in morphology; Vauhkonen et al., 1990; Aronson, distraction to achieve the effect of a clockwise rotation of the
1994; Sato et al., 1998; Sultan, 2016). In the distraction phase, mandible. The common treatments in patients with skeletal
the process of distraction activates the transportation of bone class III malocclusion include growth modification, dentoal-
fragments, and the formation of a new, immature, woven, and veolar compensation (camouflage), and orthognathic surgery
Gao et al 5

Table 3. Cephalometric Measurement of Treatment Stability.

T2–T1 T3–T1
T1 T2 T3
Mean + SD, Mean + SD, Mean + SD, Mean + SD, Relapse Mean + SD, Relapse
Variables mm mm mm mm rate P mm rate P
Skeletal
*** ***
SNA 93.0 + 9.8 91.7 + 9.0 92.3 + 9.0 1.9 + 1.9 0.081 3.5 + 3.3 0.151
SNB 76.9 + 25.3 76.4 + 6.3 85.0 + 6.3 0.0 + 1.2 0.000 1.00 0.3 + 4.2 0.111 1.00
** **
ANB 10.3 + 5.2 8.2 + 4.9 7.3 + 4.9 1.9 + 1.7 0.093 3.2 + 1.9 0.156
A-Nv 17.2 + 27.5 13.7 + 8.8 7.6 + 8.8 2.9 + 4.8 0.146 .202 3.0 + 5.0 0.151 .202
U1-SN 95.5 + 7.3 94.3 + 8.7 98.2 + 8.7 2.5 + 6.5 0.361 .310 1.8 + 3.5 0.257 .310
L1-Md 77.3 + 28.4 75.2 + 8.1 90.5 + 8.1 1.4 + 4.4 0.244 .389 4.2 + 7.4 0.717 .389
** ***
OJ 4.6 + 2.6 3.1 + 2.6 3.0 + 2.6 2.3 + 2.0 0.171 3.0 + 1.2 0.225
OB 8.3 + 20.2 9.1 + 1.5 1.4 + 1.5 1.3 + 2.4 0.592 .364 0.3 + 3.7 0.156 .834
ANS(X) 70.6 + 7.5 68.6 + 5.4 71.3 + 5.4 1.8 + 2.4 0.087 .067 2.6 + 2.3 0.128 .067
ANS(Y) 48.5 + 25.4 47.9 + 5.0 43.6 + 5.0 0.4 + 3.4 0.241 .730 2.2 + 5.6 1.222 .730
** **
A(X) 69.4 + 8.8 67.7 + 6.1 70.7 + 6.1 2.3 + 2.0 0.112 2.7 + 2.5 0.101
A(Y) 47.3 + 9.3 45.6 + 6.1 49.7 + 6.1 1.3 + 3.7 0.355 .627 0.9 + 4.1 0.239 .627
B(X) 65.9 + 8.1 66.0 + 8.5 64.1 + 8.5 0.9 + 2.0 0.086 .504 0.6 + 4.4 0.061 .744
B(Y) 74.5 + 13.2 73.1 + 8.7 83.0 + 8.7 0.4 + 3.9 0.188 .788 3.8 + 5.1 1.823 .261
Pog(X) 67.8 + 13.0 68.2 + 9.5 65.1 + 9.5 0.5 + 3.0 0.119 .713 0.7 + 4.5 0.175 .713
Pog(Y) 90.1 + 14.4 89.0 + 10.0 97.8 + 10.0 0.2 + 4.5 0.586 .889 2.6 + 5.3 6.557 .569
Abbreviations: OB, overbite; OJ, overjet; SD, standard deviation; T1, after distraction; T2, 6-month follow-up; T3, 1-year follow-up; T2–T1, relapse amount and
relapse rate after 6 months; T3–T1, relapse amount and relapse rate after 1 year; (X), horizontal change; (Y), vertical change.
**P value <0.005.
***P value <0.001.

Table 4. Soft Tissue-to-Hard Tissue Ratio After Distraction and increased to 2.6 + 2.3 mm after 1 year on average. For the
Correlation Between Soft Tissue and Hard Tissue Movement. A point, the 6-month relapse was 2.3 + 2.0 mm, and this
increased to 2.7 + 2.5 mm after 2 years. The 1-year relapse
Variables Soft tissue: hard tissue Correlation coefficient
rates in the horizontal direction were 8.7% and 11.2% in ANS
Prn: ANS(X) 0.36:1 0.22 and A point, respectively. The 1-year relapse rates were 12.8%
Prn: ANS(Y) 2.2:1 0.16 and 13.3% in ANS and A point, respectively. These results are
A0 : A point(X) 0.72:1 0.36 similar to those reported by Harada et al. (2001; 12% relapse
A0 : A point(Y) 0.29:1 0.12
Ls0 : U1i(X) 0.83:1 0.46
rate after 6 months; use of skeletal anchorage). The vertical
Ls0 : U1i(Y) 0.57:1 0.35 movements by distraction in this study were 1.8 mm in ANS
and 3.6 mm in A point (both inferiorly), indicating that the
Abbreviations: (X), horizontal change; (Y), vertical change. maxilla was moved in a counterclockwise direction. The result
showed the greater stability in our study compared to the study
(Rabie et al., 2008). The concept of RED includes orthognathic by Painatt et al. (2017), which revealed a 38% skeletal relapse
surgery and growth modification. Applying DO in preadoles- in the adults of their study. The patients in our study were all
cence children with CLCP may reduce the future need to growing children (11.2-12.5 years old) with a better blood
undergo further surgical procedures. In our study, we will supply compared to adults, and an improved ability of tissue
closely review the enrolled patients regarding their growth and repair may be the reason for better postdistraction stability.
the demand for orthognathic surgery. Hence, we suggest that patients with CLCP should undergo
Aksu et al. (2010) used a dental anchorage for tooth-borne DO in the preadolescent period if possible.
maxillary distraction in adult patients with cleft lip and palate. The upper lip advanced significantly with DO, and the soft-
After the distraction, there was a significant sagittal movement to-hard tissue ratio was 0.36:1 in the nasal tip to ANS and
of the upper incisors, revealing a 4.5 mm labial advancement. 0.73:1 in soft tissue A point to skeletal A point on average.
This result shows that the traction force may cause undesirable These ratios were considerably lower than those reported in
labial tipping of the anterior teeth. The inclination of the inci- previous studies. Most of these studies have reported that
sors to the palatal plane in our study showed little or no change patients with LeFort I advancement showed ratios of soft-to-
immediately after distraction. Hence, skeletal anchorage can hard tissue changes between 0.17 and 0.36:1 for nasal tip
help to avoid dentition side effects. change to maxillary forward movement and 0.29 to 0.62:1 for
Our study showed that after 6 months, the ANS showed a the upper lip to upper incisor movement. The results varied due
relapse in the horizontal direction of 1.8 + 2.4 mm, and this to multiple factors, such as age, patient variation (with clefts or
6 The Cleft Palate-Craniofacial Journal XX(X)
not, unilateral or bilateral), surgical methods, and treatment
intervention.
Our results were lower than those reported by Wen-Ching
Ko et al. (2000), which revealed ratios of the nasal tip to ANS
to be 0.53:1, 0.96:1 for soft A point to skeletal A point, and
0.8:1 for the upper vermilion border/upper incisor level. The
results may be stratified by age. The mean age in our study was
11.6 + 0.4 years, which was younger than that recorded in the
previous study (5.2-25.7 years in the study by Ko et al.). In the
study by Ko et al., the results varied for different ages; they
speculated that younger patients showed poor correlation
between hard and soft tissues. This may result from the less
developed soft tissues, with more fat and fewer muscles in
children. Moreover, a different soft tissue response in the result
may be attributed to the presence of scars and the difference in
type and severity of the cleft lip and palate. In our study, the
sagittal movement ratio of nasal tip/ANS had a negative correla-
tion. This result was different from that of Ko et al., which
revealed a 0.495 correlation coefficient. The skin tension
increased after distraction. The more the extent of distraction,
the less the expression of the advancement of the nasal tip due
to the progressive increasing scarred soft tissue tension.
In the study by Suhr and Kreusch (2004), common chal-
lenges in using DO included the choice of different devices,
the determination of the vector of distraction, the osteotomy
site and area, and the patient’s cooperation. Some studies
showed that the probability of complications was 22.8% (Dou-
glas, 2000). In our study, although the direction of distraction
was not proper at the initial stage and showed inappropriate
skeletal changes in the vertical and horizontal directions in
some cases, it was not difficult to control and adjust the desir-
able direction of distraction because the device is uncompli-
cated in changing the direction of the tracing wire. However,
this can cause scars in the wire exit. In the study by Yu and
Woo (2016), although they tried to reduce the size and visibi-
lity of the pin site scars in the alar crease, the scars were still
prominent and became more distinguished if infection
occurred. In our study, the scar is not prominent because of the
good resilience in young children. Even if patients care about
the dark deposit, laser resurfacing surgery can be applied since
it is not an uncommon or complicated treatment.
The use of a transcutaneous device is not rare for distraction
in mandibular advancement. Infection is one of the most com-
mon complications during distraction. Indeed, one of our
patients was infected during the consolidation phase. But after
proper medication therapy, the procedure of distraction went
well and showed good results. The biggest benefit of a trans-
cutaneous device was the reduction of discomfort for the
patient, especially on the lips. In addition, the direction of
distraction was easy to operate and maintain than traditional
distraction through the mucosa. The previous study (Balaji &
Balaji, 2018) did not demonstrate any significant difference
between internal and external distraction devices in either the
horizontal or vertical amount of distraction in maxilla. How-
ever, we found that transcutaneous external distraction device
allowed clinicians to better control the directions of distraction
in maxilla 3-dimensionally (yaw, roll, and pitch; Pelo et al.,
2007). Patients could also easily activate the device according
to instruction. Our patients also reported little influence in their
chewing function or daily activity during the consolidation
phase since they did not wear any tooth-borne or bone-borne
devices inside their mouth. External distraction device did tend
to increase certain social and physical hardship, which may not
be observed in those internal distraction device patients (Yor-
ikatsu et al., 2019). Therefore, our patients were surgically
treated during summer vocation so that their physical and psy-
chosocial distress could be minimized. Moreover, the daily
intraoral activation of internal distraction device could be more
difficult for patients to handle. Another advantage of our trans-
cutaneous external distraction device is much easier for clin-
icians to monitor the distraction process of distraction during
both activation and consolidation phases. Any defects of the
external distraction device can be immediately detected.
The imaging tool applied in this study was lateral cephalo-
metric analysis, which revealed the changes of treatment in
sagittal and vertical planes. Investigation of potential error dur-
ing landmarks identification and parameter measurement was
carried out according to Houston’s method (Houston, 1983).
The result of our error study indicated both the systemic and
random errors did not cause any impact to our result. However,
this 2-dimensional (2D) imaging tool is limited in explanation of
3-dimensional (3D) changes of surgical result, especially in
those patients requiring asymmetrical correction. The patient
enrolled in our study did not present any significant asymmetry
in their original dentoskeletal discrepancy. Therefore, there was
no asymmetric bony positioning applied during the active dis-
traction. Nevertheless, 3D landmarks other than those located in
the median plane of the anterior bony contour and soft tissue
profile cannot be assessed in 2D cephalometric imaging. The
other issues such as 3D airway changes, soft tissue envelope
expansion, or growth assessment of craniofacial structures asso-
ciated with DO can be much more reliable to apply 3D imaging
(Durão et al., 2013). Computed tomography or cone-beam com-
puted tomography scan has been utilized in simulation/naviga-
tion protocol for orthognathic planning (Chang et al., 2020).
This protocol has not been applied in growing patients requiring
DO. There are 2 reasons: high radiation exposure and potential
image noise caused by radiation scattering due to the metal
components of DO device. Both issues require setting up a new
relevant protocol in the future, such as application of nonmetal-
lic materials or redesign the fitting position of the retractor.
Hence, we believe that our results are of importance in reflecting
distraction and postdistraction effects.
Another limitation of our study, however, is the small sam-
ple size, which is similar to the previous studies. Patients with
CLCP are not common in Taiwan. From 1994 to 2013, the
annual incidence rate of CLCP was 1.48 ‰ in Taiwan (Chang
et al., 2016). Nevertheless, irrespective of the amount of dis-
traction and the relapse rate, similar results were obtained in all
of our patients, indicating the reliability of the method of dis-
traction used.
Gao et al 7

Conclusions relapse rate after 1 year. Further, we recommend that

patients with CLCP with severe skeletal discrepancies
In this study, distraction using RED devices with skeletal
should undergo distraction using RED devices in the pre-
anchorage in children with cleft lip and palate revealed
adolescent period.
significant maxilla advancement and showed a reasonable

Appendix A

Table A1. Definitions of Landmarks (Houston, 1983; Lin et al., 2012).

Skeletal and dental landmarks

N (nasion) The most anterior point on the frontonasal suture

S (sella)
Or (orbitale)
Po (porion)
Cd (condylion)
ANS (anterior nasal spine)
PNS (posterior nasal spine)
Point A (subspinale)
U1i (incisor superius)
U1a (apex of upper incisor)
L1i (incisor inferius)
L1a (apex of lower incisor)
Point B (supramentale)
Pog (pogonion)
Gn (gnathion)
Me (menton)
Go (gonion)
Ar (Articulare)
Soft tissue landmarks
PRN (pronasale)
Soft A0 point
Ls0
The center point of the hypophysial fossa
The lowest point of the average on the right and left orbital margins
The uppermost, outermost point on the bony external auditory meatus
The most superior posterior point on the head of the mandibular condyle
The tip of the anterior nasal spine
The tip of the posterior nasal spine
The deepest midline point in the curved bony outline from the base to the alveolar process of the maxilla
The tip of the crown of the most anterior maxillary central incisor
The root apex of the most anterior maxillary central incisor
The tip of the crown of the most anterior mandibular central incisor
The root apex of the most anterior mandibular central incisor
The most posterior point on the outer contour of the mandibular process in the median plane
The most anterior point of the bony chin in the median plane
The most inferior point on the mandibular symphysis in the midline
The most caudal point in the outline of the symphysis.
The most posterior inferior point on the angle of the mandible
The intersection of the posterior border of the neck of the mandibular condyle and the lower margin of the
posterior cranial base
The most anterior, inferior point on the nose tip, which is intersected by a tangent line connecting with the chin
profile
The most posterior point on the concavity between the upper lip and nose
The most anterior point on the convexity of the upper lip

Authors’ Note ORCID iD

The data sets supporting the conclusions of this article are included
within the article. Written informed consent was obtained from all the
patients before enrollment in the study. This study has been carried out
in accordance with The Code of Ethics of the World Medical Asso-
ciation (Declaration of Helsinki) and has been approved by the Insti-
tutional Review Board of the Chang Gung Medical Foundation (IRB
No: 202000531B0).
Acknowledgments
We would like to thank the Biostatistics Center, Kaohsiung Chang
Gung Memorial Hospital, for their assistance with all statistical
analyses.
Declaration of Conflicting Interests
The author(s) declared no potential conflicts of interest with respect to
the research, authorship, and/or publication of this article.
Funding
The author(s) received no financial support for the research, author-
ship, and/or publication of this article.
Wei-Ling Gao, DDS https://orcid.org/0000-0002-3194-5758
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