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2021 Does The Timing of 1-Stage Palatoplasty With Radical Muscle Dissection Effect Long-Term Midface Growth A Single-Center Retrospective Analysis
2021 Does The Timing of 1-Stage Palatoplasty With Radical Muscle Dissection Effect Long-Term Midface Growth A Single-Center Retrospective Analysis
2021 Does The Timing of 1-Stage Palatoplasty With Radical Muscle Dissection Effect Long-Term Midface Growth A Single-Center Retrospective Analysis
Vikram Shetty, MDS, DNB, MBBS1, Nanda Kishore Patteta, BDS, MDS2,
Anirudh Yadav, BDS, MDS3 , Devyani Bahl, BDS3, and Hermann F. Sailer4
Abstract
Objective: To evaluate the long-term effect of timing of 1-stage palatoplasty on midfacial growth in patients with cleft lip and palate (CLP).
Design: Retrospective observational cohort study.
Study Setting: Institutional hospital.
Patients: One hundred twelve patients with CLP who underwent palatoplasty and were divided into 3 groups: group I: operated
between 9 and 11 months; group II: operated between 18 and 20 months; and group III: operated between 21 and 24 months.
Interventions: All patients underwent von Langenbeck palatoplasty technique, which was converted to a Bardach 2-flap technique in
case of any technical difficulties. The patients were followed up between 8 and 9 years when they reported for secondary alveolar
bone grafting. Postsurgical cephalometric and dental casts measurements were taken for midfacial growth analysis.
Main Outcome Measures: The cephalometric measures were analyzed for midfacial growth and compared within the groups.
Results: Statistically significant difference (P < .01) was found on comparing the cephalometric parameters such as sella–nasion–A
point angle (SNA), A point–nasion–B point angle (ANB), n toperpendicular to point A (N-perpA), condylon to point A (Co-A),
anterior nasal spine to posterior nasal spine (ANS-PNS), nasion to Anterior nasal spine (N-ANS), nasion to menton (N Me), and
witts appraisal (Witt (AO-BO)) in group I when compared to both group II and group III patients, implying deficient midfacial
growth in group I. No statistical difference was found in the cephalometric values between group II and group III. Group II had
better cephalometric measurements than group III, showing better growth in group II than group III. Overall, there was less
incidence of midfacial hypoplasia in patients treated between 18 and 20 months (group II).
Conclusion: We conclude that palatal closure carried out at 18 to 20 months and 21 to 24 months is associated with better
midfacial growth when compared to closure at 9 to 11 months. The best time to operate would be between 18 and 20 months to
avoid speech disturbances. Midfacial growth can be greatly influenced by the timing of 1-stage palatoplasty.
Keywords
von Langenbeck palatoplasty, Bardach 2-flap palatoplasty, midfacial hypoplasia
1
Nitte Meenakshi Institute of Craniofacial Surgery, NITTE Deemed to be
University, Mangalore, India
Introduction 2
Department of Cleft and Craniofacial Orthodontics, NITTE Meenakshi
Cleft lip and palate (CLP) are one of the most common con- Institute of Craniofacial Surgery, NITTE Deemed to be University,
Mangalore, India
genital anomalies affecting 1:700 live births, and isolated cleft 3
Department of Oral and Maxillofacial Surgery, A.B. Shetty Memorial Institute
palate has an incidence of 1:2500 live births with a higher of Dental Sciences, NITTE Deemed to be University, Mangalore, India
female gender predilection (Mahajan et al., 2018). Patients 4
Chairman, Klinik Professor Sailer, Zurich, Switzerland
with clefts are born with a challenging deformity that requires
multiple surgical interventions in order to achieve functional and Corresponding Author:
Anirudh Yadav, Department of Oral and Maxillofacial Surgery, A.B. Shetty
esthetic harmony. The goal of the surgical repair of the palate is to Memorial Institute of Dental Sciences, NITTE Deemed to be University,
provide an anatomic separation of the oral cavity from the nasal Mangalore 575018, India.
cavity and the creation of a functional velopharyngeal mechanism Email: anirudhyadav6469@gmail.com
2 The Cleft Palate-Craniofacial Journal XX(X)
without causing growth restriction of the maxilla. Ideally, surgical Exclusion Criteria
repair of the palate should provide for normal hearing and speech,
1. Patients having undergone osteotomies, distraction
uninhibited growth of maxilla, and an ideal occlusion relationship
osteogenesis, fixed orthodontic therapy, or bone
and without hindering psychosocial development (Kuijpers-
grafting.
Jagtman & Long, 2000; Pereira et al., 2011).
2. Patients with isolated cleft palate.
Primary surgical reconstructions in patients with CLP result
3. Patients with severe systemic conditions affecting
in scar tissue formation at the operated site. Ultimately, this
growth.
causes static and dynamic alterations that, in association with
4. Patients with incomplete intraoperative records, irregu-
the cleft itself, have negative consequences on maxillary
lar follow-up.
growth and development, thus affecting the whole maxillofa-
cial complex of the patient (Ross 1987; Rohrich et al., 2000; A minimum follow-up of 8 years was used to assess maxillary
Friede, 2007; Stein et al., 2007). According to previous studies, growth. When the patients presented for follow-up at 8 to 9 years
the restriction of maxillary growth does not depend on the of age, a lateral cephalogram was taken, study casts were made,
genetic predisposition associated with the presence of the cleft photographs taken, and a detailed history was documented.
but is rather a consequence of the primary surgical repair (Mars Eight to 9 years is taken as the age for assessment of max-
& Houston, 1990; Capelozza et al., 1996; Shetye & Evans, 2006). illary growth in these patients prior to the secondary alveolar
Nevertheless, the timing of palate repair, the ability of the bone grafting (SABG) procedure. The SABG involves further
surgeon, the width of the cleft, and the surgical technique have dissection of the mucosa up to the piriform; hence, it can fur-
an impact on the results and also interfere with the growth and ther attribute to restricted maxillary growth. Therefore, as we
development of the facial structures involved. are concerned with establishing an ideal age for palatoplasty,
Understandably, the surgery differs from center to center we have taken this age as the point of assessment.
and surgeon to surgeon. Cleft palate repair is usually performed When the patients reported at the said age, we assessed the
at approximately 9 to 18 months of age for speech development following clinical parameters with the help of clinical photo-
(Ahmed & Sondos, 2012). graphs and dental casts:
This study aimed to determine the ideal age for the surgical
repair of cleft palate and assess the impact of palatal repair on Upper incisor visibility (repose and smile)
the maxillary growth. Intercanine width
Intermolar width
Bardach 2-flap technique in situations in which technical dif- Table 1. Distribution of the Patients Based on Surgical Technique.
ficulties (tension across the cleft) were encountered.
Surgical technique
Lignocaine (1:120 000) was used for the greater palatine and
incisive nerve blocks and for hydrodissection infiltration. The Von Langenbeck Bardach Total
incision for the von Langenbeck technique was carried out
Count 16 23 39
along the cleft margin with the mucoperiosteal flap remaining Group I % within group 41.0 59.0 100.0
pedicled anteriorly and posteriorly. The lateral releasing inci- % within surgical 42.1 31.1 34.8
sion was placed, leaving a cuff of marginal mucosa lateral to technique
the alveolus. The releasing incision was extended till the pos- Count 13 24 37
terior margin of the maxillary tuberosity without curving lat- Group II % within group 35.1 64.9 100.0
erally behind the tuberosity. The mucoperiosteal flap was % within surgical 34.2 32.4 33.0
elevated and mobilized around the greater palatine neurovas- technique
Count 9 27 36
cular bundle, after which the posterior margin of the hard palate Group III % within group 25.0 75.0 100.0
was identified and freed off the abnormal muscle attachments. % within surgical 23.7 36.5 32.1
Radical muscle dissection was carried out, exposing the levator technique
muscles, and the tensor muscles were elevated off the hamulus Count 38 74 112
without fracturing it. The nasal layer was elevated off the sep- Total % within group 33.9 66.1 100.0
tum. Z-plasty of the nasal layer was carried out in the soft % within surgical 100.0 100.0 100.0
palate area, allowing for a pushback of the soft palate muscles. technique
After the nasal mucosa and the soft palate muscles were closed
in layers, the oral mucoperiosteal flaps are brought together.
In cases where there was significant tension across the cleft Note that we did not include patients between 12 and
defect, which was often observed at the junction of the hard and 17 months as we did not have any patients in this interim age
soft palate regions, the procedure was modified to a Bardach group. According to the first protocol at our institute, patients
2-flap technique by incising behind the anterior alveolus, thus were operated between 9 and 11 months of age. The revised
mobilizing the flaps from the anterior end. It was observed that protocol at our institute follows the Zurich protocol timing and
the conversion was more commonly needed where the cleft hence we operated patients between 18 and 20 months.
defect was around 11 mm or more. Further mobilization was
achieved by undermining the palate mucosa at the posterior
Results
edge of the lateral releasing incision by incising the periosteum
and cauterizing the fine lesser palatine vessel allowing us to A total of 112 patients with CLP were evaluated, consisting of
stretch the greater palatine vessel out of the foramen without 8 to 9 years of age. The mean age of the patients getting oper-
damaging it. It may be necessary to carefully dissect the peri- ated for palatoplasty of groups I to III were 9.77 + 1.724,
osteal fibers around the neurovascular bundle to facilitate its 18.00 + 0.001, and 19.86 + 0.798 months, respectively.
unimpeded stretch out of the foramen. The periosteum poster- Table 1 presents the distribution of patients based on the sur-
olateral to the maxillary tuberosity was elevated by releasing the gical technique (von Langenbeck and Bardach 2-flap tech-
lateral fibrous attachments without tearing the overlying mucosa. nique). Table 2 provides comparisons of descriptive
statistics for all the cephalometric measurements among the
groups. All the cephalometric values had a statistically sig-
Assessment nificant difference (P < .05) when group I was compared with
group II and group III. However, no statistically significant
The cephalometric results obtained on follow-up were com- difference was found on comparing the parameters between
pared within the 3 groups to assess the maxillary growth. Also, group II and group III.
measurements were recorded on dental casts and the values On assessing the MHB scoring index on dental casts, those
compared to the MHB index to assess the craniofacial growth. patients in group I had a mean score of 4.25 + 0.23, group II
The scoring was done as 3 to þ1 from palatal to labial over- had a mean score of 1.42 + 0.06, and group III had a mean
lap for the incisors and from 0 to 2 from palatal to labial score of 1.82 + 0.14.
overlap for canines, premolars, and molars. The representative images of the patients in the 3 groups are
shown in Figures 1 to 3).
Statistical Analysis
The reliability of the single measurement was calculated using
Discussion
the Dalberg formula of error method. SPSS statistical program Growth is one of the most important areas of study in CLP care
was used for calculations of mean values. among other associated outcome variables. Growth is usually
The nonparametric test was done using the Kruskal-Wallis measured based on the degree of vertical maxillary growth,
test and post hoc Mann-Whitney U test. horizontal protrusion, transverse arch restriction, and
4 The Cleft Palate-Craniofacial Journal XX(X)
95% CI
Dependent variable (cephalometric parameter) (I) Group (J) Group Mean difference (I-J) P Lower bound Upper bound
a
SNA (degrees) Group I Group II 6.205 .001 7.94 4.47
Group III 5.055a .001 6.80 3.31
Group II Group I 6.205a .001 4.47 7.94
Group III 1.150 .349 0.62 2.92
ANB (degrees) Group I Group II 3.493a .001 4.37 2.62
Group III 2.940a .001 3.82 2.06
Group II Group I 3.493a .001 2.62 4.37
Group III 0.553 .405 0.34 1.44
N-perpA (mm) Group I Group II 3.694a .001 4.67 2.71
Group III 3.000a .001 3.99 2.01
Group II Group I 3.694a .001 2.71 4.67
Group III 0.694 .282 0.31 1.69
Co-A (mm) Group I Group II 16.272a .001 18.90 13.65
Group III 13.739a .001 16.38 11.09
Group II Group I 16.272a .001 13.65 18.90
Group III 2.533 .070 0.15 5.21
ANS-PNS (mm) Group I Group II 7.484a .001 9.36 5.60
Group III 5.968a .001 7.86 4.07
Group II Group I 7.484a .001 5.60 9.36
Group III 1.516a .172 0.40 3.43
Witt (AO-BO) Group I Group II 2.790a .001 3.53 2.05
Group III 2.412 .001 3.16 1.67
Group II Group I 2.790a .001 2.05 3.53
Group III 0.378a .677 0.38 1.13
N-ANS (mm) Group I Group II 4.207a .001 6.20 2.22
Group III 3.096 .001 5.10 1.09
Group II Group I 4.207a .001 2.22 6.20
Group III 1.110a .559 0.92 3.14
N-Me (mm) Group I Group II 5.698a .001 8.34 3.06
Group III 3.951 .001 6.61 1.29
Group II Group I 5.698a .001 3.06 8.34
Group III 1.747a .352 0.94 4.44
Abbreviations: ANB, A point–nasion–B point angle; ANS-PNS, anterior nasal spine to posterior nasal spine; AO-BO, witts appraisal; Co-A, condylon to point A;
N-ANS, nasion to Anterior nasal spine; N-Me, nasion to menton; N-perpA, A- n to perpendicular to point A; SNA, sella–nasion–A point angle.
a
p < 0.05.
occlusion. It is generally accepted that the surgical scarring (3-8 years) has been known to cause severe articulation errors
caused by primary surgical correction of CLP and other interven- (Owman et al., 1998).
tions contributes greatly to midface growth restriction, resulting Thus, a delayed 1-stage cleft palate repair should be planned
in class III patterns (Chait et al., 2002; Campbell et al., 2010). at an age not too late to cause articulation errors while not being
Early palatal surgery before 1 year of age leads to retarda- too early to cause growth impairment. In this study, patients
tion of the growth of the maxilla due to insufficiency of the soft were divided into 3 groups based on the age of repair of the
tissue to cover the defect. This lack of soft tissue causes scar- cleft palate. The patients were operated with a 1-stage palato-
ring, leading to a higher risk of developing velopharyngeal plasty using the von Langenbeck technique with radical muscle
insufficiency, ultimately leading to improper speech. To coun- dissection and pushback as the first choice to minimize the palatal
ter these speech development problems, the best time to do the scarring (Pigott et al., 2002). However, in situations where tech-
surgery is 10 to 20 months of age (Langford et al., 2003). nical difficulties (increased tension across a wide cleft palate)
One-stage cleft palate repair remains the most common pro- were encountered, the procedure was converted to the Bardach
tocol in North America (Kim et al., 2002). Early soft palate 2-flap technique. Table 1 shows that a high number of patients
repair at around 1 year of age using the 2-stage palatal repair were converted to the Bardach 2-flap technique as most of the
has been reported to have more favorable anteroposterior patients with cleft palate encountered in our center showed the
growth outcomes due to the hard palate mucosa being cleft width to be more than 11 mm. Also, the increase in the
untouched at this age (Liao et al., 2010). Additionally, the conversion rates in subsequent groups (groups I through III)
abnormal palatal anatomy arising from late hard palatal repair shows that as the age at which palatoplasty is carried out
6 The Cleft Palate-Craniofacial Journal XX(X)
Figure 4. Timeline for sequence of palatoplasty protocol followed at NITTE Meenakshi Institute of Craniofacial Surgery.
increases, the width across the cleft at the junction of the hard and Although the GOSLON yardstick has been accepted as the
soft palate also increases. Shaw et al. (2001) stated that palate gold standard to assess the craniofacial growth of patients with
repair is a nightmare as the age of the patient increases. Though cleft, it can be used in patients after the complete eruption of
the patients’ maximum age in our study did not exceed 24 months, permanent dentition (Mars et al., 1987). However, here we
we noticed a gradual increase in difficulty with progressive age, have used the MHB index to assess the craniofacial growth
like increased chances of bleeding and the amount of dissection of our patients with cleft as it can be used effectively in the
required to achieve tension-free closure. mixed dentition stage (Mossey et al., 2003), with a high corre-
Our study assessed long-term growth outcomes in 112 patients lation to GOSLON yardstick as validated in literature by var-
with unilateral CLP, 8 years after palate surgery, by measuring 8 ious studies (Almuhizi et al., 2016; Yakob et al., 2018). The
cephalometric parameters. The additional use of ANB and Witt mean scores achieved from the MHB index for groups I to III
appraisal in this study helped compare the midface growth in were 4.25, 1.4, and 1.82, respectively. Group I, where patients
relation to the mandible. Table 2 compares the midface growth who were operated between 9 and 11 months, had poorer scores
between the 3 groups. Our findings indicate that the growth mea- (mean ¼ 4.25) when compared to group II (mean ¼ 1.4) and
sured across all 8 cephalometric parameters showed no signifi- group III (mean ¼1.82), where patients were operated between
cant difference between group II and group III. However, patients 18 to 20 months and 21 to 24 months, respectively. This jus-
in group I showed a significant restriction in growth measured tifies the need for a midface advancement surgery later in the
across all the parameters compared to group II and group III. life for group I patients rather than the other 2 groups based on
Needless to say, the normal midface growth observed in the assessment by this index.
groups II and III consolidated our hypothesis that cleft width The assessment of growth in our study was carried out at 8 to
(which also increased with the age of surgery) does not play a 9 years of age before alveolar bone grafting was carried out.
role in growth restriction, provided the technique can be mod- While the ideal age to measure growth would be after the
ified with regard to the requirements of individual patients. pubertal growth spurt, the mucosal dissection over the septum
Hence according to our study, the main parameter that and alveolus during bone grafting procedures may in itself
affects long-term midface growth is the timing of surgery. As cause restriction of growth, thus causing a bias. On the other
a single operator conducted all surgeries, the effect of operator hand, it has been shown that the maximal restriction of growth
variations in surgical technique on growth could not be due to the primary cleft procedures would be apparent by
assessed. In our study, the ideal age for carrying out 1-stage 9 years of age. Therefore, growth assessment at 8 to 9 years,
palatoplasty would be 18 to 24 months if growth would be the as carried out in this article, would offer substantial insight into
only criterion. However, as the production of speech is a major the effect of timing of the palatoplasty procedure on midface
determinant of the success of palate surgery, late intervention growth. However, postpubertal assessment at a later stage will
may lead to errors in articulation and may cause hypernasality offer more value to the literature.
due to inadequacy in the velopharyngeal valve. Although this It would also be pertinent to mention that the constant
study does not measure speech outcomes, it has been proven ongoing evaluation of surgical protocols and their effects on
that speech deteriorates when palate surgery is postponed midface growth formed the basis of the 3 groups of this study.
beyond 20 months of age (Langford et al., 2003). Hence, Groups I and II signified the changes in the cleft palate proto-
patients operated in group II would possibly show the maxi- cols followed in our center, the timeline of which is shown in
mum combined benefit in terms of speech and growth Figure 4. Group III consisted of patients who presented late for
parameters. surgery.
Shetty et al 7