J Stomatol Oral Maxillofac Surg 122 (2021) 13–17

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Original Article

Effectiveness of presurgical orthodontics in cleft lip and palate patients

with alveolar bone grafting: A systematic review
Li Ma 1,*, Yali Hou 1, Guijun Liu, Tianqi Zhang
Department of Stomatology, Shandong Provincial Hospital Affiliated to Shandong First Medical University, Jinan, Shandong Province 250021, China

A R T I C L E I N F O A B S T R A C T

Article history: Objective: Alveolar repair has become a routine part of treatment protocols for patients with non-
Received 8 July 2020 syndromic cleft lip and/or palate, but there is no clear conclusion of whether the presurgical orthodontic
Accepted 22 July 2020 treatment is necessary to alveolar bone grafting or not. The purpose was to determine the necessity of
Available online 15 August 2020
the presurgical orthodontics in cleft lip and palate patients.
Materials and methods: Electronic databases including PubMed, Ovid, Embase, Cochrane Library, Web of
Keywords: Knowledge, and China Biology Medicine disc (SinoMed) were searched. Only studies published in
Bone volume
English or Chinese were included. The last search was updated on 1 May 2020. 1225 articles remaining
Cleft lip
Cleft palate
after the exclusion of duplicates. Finally, there were 11 publications (five in English and six in Chinese)
Alveolar bone grafting eligible for systematic review according to the previously established inclusion and exclusion criteria. A
Orthodontics descriptive statistical method was used to present data. The methodological index for non-randomized
studies (MINORS) was used to determine the risk of bias.
Result: Eleven articles were included in this review, of which seven publications were retrospective study
and four articles were comparative study. The average success rate of reconstruction with the presurgical
orthodontic treatment was approximately 70–97%, while the success rate of the non-presurgical
orthodontics was 25–80%. The fixed and removable presurgical orthodontic methods were frequently
performed, rather than a single treatment model. The incidence of the postoperative complications
resulting from whether adopting the presurgical orthodontics was different from none to 75%.
Conclusion: There are a higher postoperative bone formation rate and a lower complication rate after ABG
with presurgical orthodontics. However, more studies with high methodological quality and with a
longer follow-up are needed to offer more safety for practitioners and patients regarding the surgical
method selected to repair the cleft alveolar.
C 2020 The Author(s). Published by Elsevier Masson SAS. This is an open access article under the CC BY-

NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).

1. Introduction different types of clinical presentations such as unilateral or

Non-syndromic cleft lip and/or cleft palate (NSCL/P) is one of
the commonest birth deformities. The incidence is approximately
1 in 1000 to 1 in 500 live births[1]. NSCL/P has a great impact on the
quality of life including esthetics, function, psychological impact,
dental development, and facial growth [1]. The alveolar cleft is
known as the developmental defect of bone in the alveolar process
of maxilla which accounts for 75% of the NSCL/P patients with
* Corresponding author. Department of Stomatology, Shandong Provincial
Hospital Affiliated to Shandong First Medical University, No.51, Weiliu Road,
Jinan, Shandong Province 250021, China.
E-mail address: mali3709@163.com (L. Ma).
1 well-established, ABG is not always successful. Long term success
The two authors contributed equally to this article.
bilateral and complete or incomplete. Alveolar clefts lead to a bone
defect in the alveolar process, discontinuity of maxillary arch, and
inhibition of midface growth in patients with NSCL/P [2]. The
alveolar repair has become a routine part of treatment protocols
for patients with NSCL/P. Its primary purpose is to restore the
function and continuity of the maxillary arch at the cleft site and to
repair the oronasal fistula and nasal deformity through providing
alar base support.
Alveolar bone grafting (ABG) using autogenous bone including
tibia, rib, calvarium, and mandible, has been studied and compared
[3]. The selection is often dependent on the surgeon’s experience
and preference, the bone volume needed, and even by donor site
morbidity [4]. Although ABG is popularity and its procedures are

https://doi.org/10.1016/j.jormas.2020.07.010
2468-7855/ C 2020 The Author(s). Published by Elsevier Masson SAS. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-

nc-nd/4.0/).
14 L. Ma et al. / J Stomatol Oral Maxillofac Surg 122 (2021) 13–17
rates using the Bergland scale [5] from periapical and occlusal
radiographs vary from 67% to 95%. Determination of the possible
factors on the failures of ABG that affect the quality of grafting can
help the oral and maxillofacial surgeon to maximize favorable
outcomes. Therefore, to improve the long-term success rates of
ABG, in many Craniofacial Centers, presurgical orthodontic
treatment is approved as a common practice to improve the
alignment of inclined and rotated central incisor adjacent to the
alveolar bone cleft [6]. The presurgical orthodontics plays an
important role in correcting misaligned central incisors or
repositioning displaced maxillary alveolar segments and allows
the oral and maxillofacial surgeon better access for placement of
the graft and closure of the soft tissue [6]. Besides, the higher
success rate was found in cases of orthodontic space closure than
in cases of space openings, suggesting the importance of
presurgical orthodontics [7]. However, Manosudprasit et al. [8]
indicated that the success of grafting is not related to orthodontic
preparation. An explanation of this finding was provided by Long
et al. [9] who found a low correlation between cleft width and bond
graft adequacy. Furthermore, Aurouze et al. [10] found that the size
of presurgical cleft defects appears to no correlate with the success
of the secondary alveolar bone graft. Therefore, they concluded
that the presurgical orthodontic treatment before bone graft
appears to have little or no impact on the success of the ABG.
To explore whether it is necessary to perform the presurgical
orthodontic treatment before ABG or not, we systematically
reviewed retrospective and prospective studies that evaluated the
bone volume and the incidence of postoperative complications
after ABG with or without the presurgical orthodontics. The
present systematic review could answer the following questions:
1. Whether it is required for the preoperative orthodontic
treatment before ABG.
2. Whether the presurgical orthodontic treatment can significant-
ly improve the success rate of postoperative bone grafting and
reduce the occurrence of postoperative complications.
2. Material and methods
Preferred Reporting Items for Systematic Reviews and Meta-
Analyses (PRISMA) guidelines were used in this study [11].
2.1. Study characteristics
P: at least 10 NSCL/P patients
I: alveolar bone grafting with presurgical orthodontics
C: not required
O: bone volume and complications
2.2. Inclusion criteria
1. studies performed in humans with no restrictions on age,
gender, or ethnicity;
2. unilateral and/or bilateral cleft alveolar;
3. follow-up time larger than 6 months;
4. publications limited to English or Chinese.
2.3. Exclusion criteria
The exclusion criteria were as follows: case reports, systematic
reviews, patients with syndromes, or with any other comorbidities;
studies with follow-up time less than 6 months; duplicated
publications; experimental studies.
2.4. Search strategy
We searched PubMed, Ovid, Embase, Cochrane Library, Web of
Knowledge, and China Biology Medicine disc (SinoMed) indepen-
dently using the search terms: ((((((((((((cleft lip) OR cleft palate)
OR Harelip) OR ‘‘Cleft Lip’’[Mesh]) OR ‘‘Cleft Palate’’[Mesh])) AND
alveolar)) OR alveolus cleft) OR alveolar cleft)) AND (((((bone
grafting) OR bone graft) OR ‘‘Bone Transplantation’’[Mesh]) OR
Bone Transplantation) OR ‘‘Alveolar Bone Grafting’’[Mesh])) AND
(((orthodontics OR orthodontics[mesh]) OR ‘‘Orthodontics, Cor-
rective’’[mesh])). Only studies published in English or Chinese
were included. The last search was updated on 1 May 2020.
2.5. Study selection
The search process was conducted by two authors (Ma L. and
Hou Y.L.), and a third reviewer (Liu G.J.) was consulted under a
consensus was not achieved between the two first reviewers. Titles
and abstracts were screened by these reviewers and potentially
eligible papers were read fully to assess their eligibility. Clinical
studies were sought, including randomized or non-randomized
clinical trials and prospective or retrospective observational
studies. Data were extracted from those records and then retrieved
for detailed text evaluation. These procedures were conducted by
the first and second authors. The following information was
collected: study type, number, and age of patients, gender, follow-
up time, donor site, cleft types, graft types, postoperative bone
volume, and complications.
2.6. Outcomes
The primary outcome was the postoperative bone volume in
cleft alveolar patients after ABG with presurgical orthodontics.
Secondary outcomes were complications of recipient sites for
alveolar cleft reconstruction; dehiscence and/or infection; peri-
odontal problems; oronasal fistula.
2.7. Statistical analysis
A descriptive statistical method was used to present data.
Based on the evaluation criteria for bone formation after bone
grafting in the articles, we drafted the rules as follows: the bone
volume at six months above 50% after bone grafting were judged
as successful bone grafts, below 50% failures were judged bone
graft. Simultaneously, we also performed a descriptive statistical
analysis of complications in the donor and recipient areas after
bone grafting.
2.8. Methodological quality and risk of bias assessments
To determine the risk of bias, and, consequently, the quality of
the included studies, the methodological index for non-random-
ized studies (MINORS) was used [11]. MINORS is a valid instrument
designed to assess the methodological quality of non-randomized
surgical studies, whether comparative or non-comparative.
MINORS contained 12 items, the first eight being specifically for
non-comparative studies. Every item was given three different
grades. Zero represents the item content was not reported; one
indicates the item content reported but inadequate; two represent
the item content was reported and adequate, and the ideal global
score for comparative studies was equal to 24, and 16 for non-
comparative studies.
 L. Ma et al. / J Stomatol Oral Maxillofac Surg 122 (2021) 13–17 15

Table 1
Included studies assessing the effectiveness of repair of the alveolar cleft, and extracted data.

Author Year Type of study No. of patients Gender Type of cleft Age at time of
surgery (years)

Jia YL, et al. 2004 Retrospective 16 9 M/7 F 10 UI/6 BI 8–22

Giudice G, et al. 2007 Retrospective 30 Not reported Not reported 9–11
Qu YQ, et al. 2009 Retrospective 18 13 M/5 F Not reported 8–12
Wang HY. 2011 Retrospective 80 54 M/26 F Not reported 6.5–18.6
Manosudprasit M, et al. 2011 Retrospective 101 46 M/55 F 101 UI 6–34
Liu L. 2014 Retrospective 200 164 M/36 F 90 UI/110 BI 9–23
Wang LH, et al. 2015 Retrospective 80 46 M/34 F Not reported 5.5–17.5
Liao YF, et al. 2015 Retrospective 77 49 M/28 F 77 UI 9–12
Chang CS, et al. 2016 Prospective 22 Not reported 22 UI 9–10
Wang F, et al. 2017 Retrospective 50 31 M/19 F Not reported 3–11
Datana S, et al. 2018 Prospective 30 Not reported 30 UI 9–13
Jia YL, et al. 2004 Retrospective 16 9 M/7 F 10 UI/6 BI 8–22

M: male, F: female, Un: unilateral, Bi: bilateral.

3. Result Kappa was calculated and showed a consensus rate between

The search produced 2086 results, and there were 1225 arti-
cles remaining after the exclusion of duplicates. Of these,
1205 publications were excluded after reading the title and
abstract. The 12 remaining articles were read fully and one paper
was excluded for the unclear division of groups. Finally, there
were 11 publications (five in English and six in Chinese) (Table 1)
eligible for systematic review according to the previously
established inclusion and exclusion criteria. Nine of the
11 articles were retrospective study and two were a prospective
study, and then underwent a risk of bias assessment. Cohen’s
reviewers of 93.7%. Details regarding the selection process are
shown in Fig. 1.
3.1. Quality assessment
Of the 11 articles that underwent quality assessment, there
were 4 [6–8,12] publications of comparative studies and 8 [13–
19]of non-comparative studies. Results for comparative studies
ranged from 16 to 19 (Table 2), and results for non-comparative
studies are from 11 to 14 (Table 3). The quality of these studies is
medium to high. Neither of the papers was able to make

Fig. 1. Flowchart of the study selection process.

16 L. Ma et al. / J Stomatol Oral Maxillofac Surg 122 (2021) 13–17

Table 2 orthodontics. Among the specific data that can be retrieved, the
Methodological index for non-randomized studies (MINORS) results for compara-
average success rate of ABG with presurgical orthodontics was
tive studies.
approximately 83.9%, while the success rate of the patients
Author Year 1 2 3 4 5 6 7 8 9 10 11 12 Score without presurgical orthodontics was about 54.9%. In a quantita-
Giudice G, et al. 2007 2 2 2 2 0 2 2 0 1 2 1 2 16 tive study [6], the bone volume of the patients with presurgical
Manosudprasit M, et al. 2011 2 2 2 2 0 2 2 1 1 1 1 2 16 orthodontics versus without presurgical orthodontics was
Liu L. 2014 2 2 2 2 1 2 2 1 1 2 2 2 19 0.81  0.26 cm3 versus 0.59  0.22 cm3. After 6 months of repairing
Datana S, et al. 2018 2 2 2 1 0 2 2 1 2 2 2 2 18
alveolar segments, ABG with the presurgical orthodontics is superior
1. — a clearly stated aim, 2. — inclusion of consecutive patients, 3. — prospective to the non-presurgical orthodontics in terms of bone density, bone
data collection, 4. — endpoints appropriate to the aim of the study, 5. —unbiased
width, and bone volume.
assessment of the study endpoint, 6. — follow-up period appropriate to the aim of
the study, 7. — loss to follow up less than 5%, 8. — prospective calculation of the
study size, 9. — an adequate control group, 10. — contemporary groups, 11. — 3.4. Presurgical orthodontic methods
baseline equivalence of groups, 12. — adequate statistical analyses; 0 — not
reported, 1 — reported but inadequate, 2 — reported and adequate; ideal global The presurgical orthodontics methods for patients with the
score for comparative studies = 24.
alveolar cleft can be divided into removable appliances and fixed
appliances. The purpose of the correction is to expand the
maxillary dental arch, restore the twisted central incisors, align
the dentition, and provide bone conditions for alveolar bone
Table 3
Methodological index for non-randomized studies (MINORS) results for non- grafting. Of all the included studies, only one study [18] used a
comparative studies. removable appliance, and necessarily, added lip arches and tongue
springs during the treatment. The remaining studies used both
Author Year 1 2 3 4 5 6 7 8 Score
removable and/or fixed appliances which are mostly square wire
Jia YL, et al. 2004 2 2 2 2 0 2 2 0 12 bow or straight wire bow technique.
Qu YQ, et al. 2009 2 2 2 2 0 2 2 0 12
Wang HY. 2011 2 2 2 2 1 2 2 1 14
Wang LH, et al. 2015 2 2 2 1 0 2 2 1 12
3.5. Complications
Liao YF, et al. 2015 2 2 2 2 1 2 2 1 14
Chang CS, et al. 2016 2 2 2 2 1 2 2 1 14 Postoperative complications of bone graft surgery could be
Wang F, et al. 2017 2 1 2 1 1 1 2 1 11 artificially considered as short-term symptoms and long-term
1. — a clearly stated aim, 2. — inclusion of consecutive patients, 3. — prospective symptoms. The former contained wound dehiscence and wound
data collection, 4. — endpoints appropriate to the aim of the study, 5. —unbiased infection. Long-term complications are frequently the formation of
assessment of the study endpoint, 6. — follow-up period appropriate to the aim of periodontal pockets, gingival attachment loss, re-formation of
the study, 7. — loss to follow up less than 5%, 8. — prospective calculation of the
study size; 0 — not reported, 1 — reported but inadequate, 2 — reported and
oronasal fistula, and severe resorption of the transplanted bone. In
adequate; ideal global score for non-comparative studies = 16. the included studies, there were four studies gave explicit result on
complications of the patients after ABG with the presurgical
orthodontics. The complications include oronasal fistula, peri-
odontal problems, and infection. The incidence of the oronasal
prospective calculations of the scale of the study, nor did they fistula was reported in two studies, of which, one study indicating
report a full and fair assessment of the endpoints of the study. no oronasal fistula was found, and the other study showing 26.8%
patients were found the oronasal fistula after ABG with or without
3.2. Patients the presurgical orthodontics. Periodontal pockets larger than
3 mm were found in patients after ABG without the presurgical
The eligible studies included 700 patients with the operation orthodontics, the incidence was approximately 75%. Besides, only
time from 6 to 34 years. Of the 11 publications, 6 [6–8, 12, 13, two studies indicated that the infections were found after ABG
19]articles (466 patients) gave a clear diagnosis of whether the with or without the presurgical orthodontics, the incidences
alveolar cleft was unilateral or bilateral, of which 330 were respectively were 2.9% and 22%.
unilateral cleft alveolar and 136 were bilateral cleft alveolar. All
patients were followed for at least 6 months, with a scope of
follow-up time was from 6 months to 6 years. Three [6,14,19] of the 4. Discussion
included publications (82 patients) did not indicate special
numbers of men and women participated in the studies, of the Alveolar bone grafting (ABG) is an important part of treatment
other studies, there were 412 patients were male (66.7%) and protocols for patients with NSCL/P. The goals of ABG have both
206 were female (33.3%). functional and esthetic purposes; the former include the closure of
the nasolabial fistula, providing bone support to allow the eruption
3.3. Bone volume of teeth in the cleft site, creation of support for the nasal ala and a
continuous maxillary dental arch. The latter include augmentation
Of the 11 publications, ten [7,8,12–19] studies qualitatively, of the pyriform region and the creation of a cosmetically pleasing
and one [6] study quantitatively evaluated the changes of the bone dental arch form and position of the teeth [14]. During the
volume at least after 6 months of operation. According to the treatment protocols, the orthodontists form an integral part of the
amount of osteogenesis, it is divided into four grades: grade I cleft care team. The orthodontic treatment for children with the
osteogenesis is 75–100%, grade II 50–75%, grade III 25–50%, and alveolar cleft should aim to provide a dentition that functions well
grade IV 0–25%. It is generally deemed that the grade I and II results and is capable of lifetime maintenance by routine oral hygiene and
are successful bone grafting, and grade III and IV results are bone dental care [20]. Therefore, the importance of the orthodontist in
graft failure. According to the statistical results of osteogenic planning, preparations, and follow-up around the ABG procedure is
volume in the literature, after 6months of ABG with presurgical also widely recognized. Successful ABG necessitates a joint
orthodontics, 70–97% of patients can obtain more than 50% of the orthodontic and surgical involvement pre-, peri-, and postopera-
bone volume and 25–80% of the patients without presurgical tively.
 L. Ma et al. / J Stomatol Oral Maxillofac Surg 122 (2021) 13–17
Although the treatment protocols and the treatment methods
were popularity and well-established, ABG is not always success-
ful. Success rates vary from 67% to 95%. The best results are
achieved if the graft is undertaken before the canine eruption.
Long-term success rates of between 72% and 95% are reported for
bone grafts before the canine eruption, whereas figures for bone
grafts after canine eruption lie between 67% and 91%. However,
higher grafting success was found in cases of presurgical
orthodontic treatment than in cases of non-presurgical orthodon-
tics, suggesting the importance of presurgical orthodontics. In the
included studies, the patients with presurgical orthodontics, 70% to
97% of patients can obtain successful ABG, and only 25% to 80% of
the patients can achieve successful ABG without presurgical
orthodontics. Therefore, presurgical orthodontic treatment could
provide a higher success rate for patients with ABG.
Despite its importance of the presurgical orthodontics for the
patients with the alveolar cleft, the presurgical orthodontics
methods were not consistent. In the included studies, only one
study [18] used a removable appliance, and the remaining studies
used both removable and/or fixed appliances. However, the
preoperative orthodontic methods in the included studies were
outmoded.
Secondary bone grafting is an important step in alveolar
deformity reconstruction in cleft lip and palate patients. In
addition to the benefits in providing bone support for tooth
eruption, secondary bone grafting can eliminate the oronasal
fistulae, improve oral hygiene by separating the oral and nasal
cavities, rebuild the morphology of hypoplastic pyriform aperture
and the soft tissue of the nasal base support, and stabilize the
maxillary arch, thus creating a sufficient volume of bone to allow
dental movement and subsequent residual space closure in the
cleft region [21]. Many patients with alveolar cleft have benefited
from successful secondary alveolar bone grafting, but some
disadvantages remain, such as infection, wound dehiscence
donor-site complications, and graft resorption. After 6 months
with alveolar bone grafting with the presurgical orthodontics, 70–
97% of patients can obtain more than 50% of bone volume, which is
significantly higher than reconstruction without the presurgical
orthodontics.
Regarding statistical analysis, it was not possible to perform a
meta-analysis because only three of the 11 eligible were the
prospective studies, and the other types of publications were
retrospective studies. Furthermore, the authors used different
controls and assessment methods, which made it impractical to
perform any comparisons or to perform statistical analysis. This
situation reveals the need for future researches to perform with the
highest methodological rigor to increase the level of scientific
evidence and to reduce the risk of bias.
5. Conclusion
According to the best available evidence, there is a higher
postoperative bone formation rate and a lower complication rate
after ABG with the presurgical orthodontics. However, there is a
clear need for studies with high methodological quality and with a
longer follow-up to find scientific evidence not yet verified,
offering more safety for practitioners and patients regarding the
ABG with or without a presurgical orthodontic method for the
patients with the cleft alveolar.
17
Source of support
None.
Funding
None.
Conflict of Interest declaration
The authors have no conflicts.
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