INTRODUCTION
Among the congenital craniofacial malformations, cleft lip and palate (CLP) is the
most common and occurs in 1 of every 650 births in Brazil1. CLP may cause functional limitations in speech, difficulty eating and breathing,
and negative social and psychological consequences during adulthood.
The palate acts as an anatomical barrier that separates the oral cavity from the nasal
cavity. Together with other structures of the pharynx, it contributes to the function
of the velopharyngeal sphincter by assisting in speech and feeding. Without the normal
function of these structures, patients with cleft palate may develop changes such
as nasal air leak and food reflux through the nose2. Moreover, patients with CLP have social adaptation problems because of their physical
appearance.
CLP is classified according to the affected region into pre-foramen (lips and primary
palate), transforamen (primary and secondary palate), and post-foramen. Therefore,
the submucosal cleft should be identified in cases of bifid uvula, and cleft palate
size can be classified as narrow, normal, or wide.
The history of palatoplasty is long, dating from 17603, with significant progress in clinical research, and most techniques are based on
mobilization of the axial flaps supplied by the greater palatine artery. Von Langenbeck4 (1862) described the use of bilateral mucoperiosteal flaps without reconstruction
of the intravelar muscle or palatal stretching, leading to the development of the
push back techniques described by Veau (1931)5 and Wardill and Killner (1937)6,7.
Repair of the soft palate requires dissection of the palatal muscles and repositioning
of the levator veli palatini muscle with or without manipulation of the palatoglossal
and palatopharyngeal arches and tensor veli palatini muscle for reconstructing the
muscle sheath8. Braithwaite9 (1964) and Kriens10 (1969) defined “intravelar veloplasty” as the rotation and terminoterminal anastomosis
of the levator veli palatini muscle to the tensor veli palatini muscles to improve
the functional results of palatoplasty11. This technique was modified by Cutting et al.12 (1995) and Sommerlad et al.13 (2002). Furlow Junior14 (1986) described double-opposing Z-palatoplasty and found that it achieved good functional
results in the nasal and oral lining with lower rates of velopharyngeal insufficiency
(VFI).
Regardless of the technique used, the objectives of palatoplasty are to: 1) Stretch
the palate to minimize the incidence of VFI and promote adequate speech development;
2) Minimize the restriction of maxillary and alveolar growth; and 3) Prevent complications,
including oronasal fistulas (ONFs).
OBJECTIVE
To present the author’s experience and the incidence of ONF in 278 cases of primary
palatoplasty using the von Langenbeck technique associated with intravelar veloplasty.
METHODS
This retrospective study reviewed the medical records of all patients treated surgically
at the Mário Covas Treatment Center for Craniofacial Malformations, Guilherme Álvaro
Hospital, Santos, São Paulo, Brazil, between May 2010 and May 2018.
The inclusion criteria were diagnosis of cleft palate (CP) or CLP associated or not
with syndromes (excluding cases of cleft soft palate) and treated with primary palatoplasty
using the described technique performed by the same surgeon (the author).
A total of 278 records were selected, and the following data were obtained:
Diagnosis: right, left, or bilateral CLP corresponding to trans-foramen clefts; and
bilateral CP, corresponding to post-foramen clefts;
Sex (male/female)
Race (Caucasian, mixed, or Black);
Patient age (in months) at the time of primary palatoplasty;
Surgical stage of primary palatoplasty (one or two);
Clinical course with ONF (yes/no) during the 6-month follow-up period after the last
procedure.
The data collected over the 8-year study period (May 2010 to May 2018) were organized
and recorded in an Excel spreadsheet, and the data on the appearance of ONF were analyzed
in eight 1-year periods based on the date of the last surgery.
Phonation results were not included in the analysis because the objective of this
study was to evaluate the aesthetic results and the incidence of ONF.
RESULTS
A total of 278 primary palatoplasty procedures using the von Langenbeck technique
associated with intravelar veloplasty were evaluated, including 225 (80.9%) performed
in two surgical stages (soft palate first, followed by hard palate) and 53 (19.1%)
performed in one surgical stage.
The study population included 182 (65.5%) men and 96 (34.5%) women.
In preoperative diagnoses, the incidence of left and bilateral CLP was 26.3% and 27%,
respectively, while that of complete CP and right CLP was 37.4% and 7.6%, respectively.
A total of 157 patients (56.4%) were Caucasian, 107 (38.4%) were mixed, and 14 (5.04%)
were Black. The average age at the time of primary palatoplasty was 17.2 months.
Postoperative complications included total suture dehiscence (2 cases [0.7%]), postoperative
bleeding (4 cases [1.44%]), and infection (2 cases [0.72%]). There were no cases of
flap necrosis.
ONF occurred in 61 patients (21.94%) during the study period, and the incidence decreased
progressively. The incidence of ONF in the first 1-year period (May 2010 to May 2011)
and the last 1-year period (May 2017 to May 2018) was 25.00% and 18.75%, respectively
(Table 1).
Table 1 - Incidence of oronasal fistula (ONF) among during 8-year study period since May 2010
and May 2018.
Period |
Patients |
Fistula |
% |
May 2010 - May 2011 |
4 |
1 |
25.00 |
May 2011 - May 2012 |
7 |
2 |
28.57 |
May 2012 - May 2013 |
36 |
9 |
25.00 |
May 2013 - May 2014 |
35 |
9 |
25.71 |
May 2014 - May 2015 |
47 |
11 |
23.40 |
May 2015 - May 2016 |
49 |
10 |
20.41 |
May 2016 - May 2017 |
52 |
10 |
10.23 |
May 2017 - May 2018 |
48 |
9 |
18.75 |
Total |
278 |
61 |
21.94 |
Table 1 - Incidence of oronasal fistula (ONF) among during 8-year study period since May 2010
and May 2018.
Data on ONF for the primary and secondary palates were included in the study, although
their site of occurrence was not determined.
Surgical technique
Patients undergoing primary palatoplasty are treated surgically at 6–18 months of
age depending on the ease of follow-up. In cases of trans-foramen clefts, primary
cheiloplasty is performed in patients older than 3 months and primary palatoplasty
can be performed 6 months later using the von Langenbeck technique with or without
a vomer flap. Cheiloplasty was not performed together with primary palatoplasty in
this population.
CP type was classified as narrow, normal, or wide; in narrow clefts, the procedure
was performed in one surgical stage, while in normal or wide clefts, it was performed
in two stages (soft palate first, hard palate 6 months later).
All patients underwent surgery while under general anesthesia and orotracheal intubation
under direct vision. A Dingman mouth gag was positioned after adequate visual inspection
(Figure 1).
Figure 1 - Preoperative aspect of a bilateral transforamen cleft.
Figure 1 - Preoperative aspect of a bilateral transforamen cleft.
The edges of the cleft were demarcated on the soft palate, which was infiltrated with
2% lidocaine combined with a vasoconstrictor (1: 200,000) for topical anesthesia.
An incision was made on the edges, and the plane of the oral and nasal mucosa was
dissected to release the anterior insertion of the levator veli palatini muscle bilaterally
and, if necessary, the insertion of the tensor veli palatini muscle. The nasal mucosa
was closed and the cleft uvula was repaired. The levator veli palatini muscle was
closed and sutured with U-shaped Vicryl® 4-0 sutures, while the oral mucosa was sutured with U-shaped Vicryl® 5-0 sutures.
Relaxing incisions and vomer flaps were demarcated in the second surgical stage (Figure 2), and the incision sites and hard palate were infiltrated with 2% lidocaine combined
with a vasoconstrictor (1: 200,000) for topical anesthesia.
Figure 2 - Preoperative markings.
Figure 2 - Preoperative markings.
Bilateral relaxing incisions were made on the medial hard palate at the alveolar crest
according to the von Langenbeck procedure.
The bilateral mucoperiosteal flaps were elevated, the pedicle of the greater palatal
artery was identified, and the nasal mucosa of the hard palate was dissected (Figure 3).
Figure 3 - Visualization of the pedicle of the greater palatal artery after elevation of the
bilateral mucoperiosteal flaps.
Figure 3 - Visualization of the pedicle of the greater palatal artery after elevation of the
bilateral mucoperiosteal flaps.
In wide and normal clefts, unilateral or bilateral mucoperiosteal flaps of the vomer
were elevated to close the nasal lining (Figures 4 and 5).
Figure 4 - Elevation of the vomer flaps.
Figure 4 - Elevation of the vomer flaps.
Figure 5 - Suture of the nasal lining in a patient with a postforamen cleft.
Figure 5 - Suture of the nasal lining in a patient with a postforamen cleft.
The mucoperiosteal flaps were closed, and the oral mucosa was sutured with U-shaped
Vicryl 5-0 sutures (Figures 6 and 7).
Figure 6 - Posterior rotation of the levator veli palatini muscle.
Figure 6 - Posterior rotation of the levator veli palatini muscle.
Figure 7 - Intravelar veloplasty.
Figure 7 - Intravelar veloplasty.
Hemostatic dressings remained at the site of the relaxing incisions only in cases
in which the open area was large because they did not impair healing, although they
had no demonstrable benefit (Figures 8 and 9).
Figure 8 - Immediate postoperative period of palatoplasty using a vomer flap and the von Langenbeck
technique associated with intravelar veloplasty in a patient with a bilateral transforamen
cleft.
Figure 8 - Immediate postoperative period of palatoplasty using a vomer flap and the von Langenbeck
technique associated with intravelar veloplasty in a patient with a bilateral transforamen
cleft.
Figure 9 - Aspect of the cleft immediately after surgery.
Figure 9 - Aspect of the cleft immediately after surgery.
The steps mentioned above were performed simultaneously in cases of primary palatoplasty
in a single surgical step.
DISCUSSION
The controversial aspects of palatoplasty include the ideal age at the time of primary
surgery to interfere as little as possible with facial growth and allow adequate speech
development.
CLP affects facial bone growth, allowing the development of trends and different protocols
for its repair as well as surgical repair in one or two surgical stages15,16 without affecting mandibular growth17.
There is no consensus on the ideal age for primary palatoplasty. In our protocol,
this surgery was performed at the age of 6–18 months in one stage or alternatively
one stage for narrow clefts and two stages for normal and wide clefts.
Despite being the oldest technique, von Langenbeck’s palatoplasty is still used and
a good option for wide and incomplete clefts because it is simple and facilitates
dissection18,19. Palatoplasty combined with repair of the nasal lining and muscle sheath is safe
and has a low rate of ONF20. The occurrence of ONF depends on patient age21, cleft type and extent22, association with syndromes23, surgeon experience, and factors that affect the surgical outcome including suture
tension, bleeding, and infection20.
The incidence of ONF depends on surgical timing, and the primary surgery can be delayed
because of the limited access to health services as demonstrated in our sample by
the significant difference in patient age at the time of the first surgery. Surgeon
experience also plays a fundamental role given that the author’s learning curve improved
over time (Figure 10). The sample was evaluated in eight 1-year periods from May 2010 to May 2018. The
incidence of ONF in the first and last periods was 25% and 18.75%, respectively, and
the average incidence throughout the study period was 21.94%, which agrees with data
in the literature. It should be noted that all ONFs present at 6 months postoperative
were identified, including those located anterior to the incisive foramen given that
some studies disregarded them.
Figure 10 - Curva shows incidence which decreased progressively a complication as ONF among during
8-year study.
Figure 10 - Curva shows incidence which decreased progressively a complication as ONF among during
8-year study.
The reported incidence of ONF is 0–60%, and the incidence in Brazil is 15.3%24.
ONF can be classified as symptomatic or non-symptomatic; the former does not always
require surgical management. However, in this case series, all ONFs located posterior
to the incisor foramen were operated upon at 6 months after definitive surgery using
different techniques according to their size and location because their description
is beyond the scope of this study.
CONCLUSIONS
Primary palatoplasty using the von Langenbeck technique associated with intravelar
veloplasty was reproducible in our service when performed in one or two surgical stages.
Although the incidence of ONF was higher than that reported in the literature, this
surgical procedure is considered safe when the learning curve is reached and improves
the aesthetics of CP.
COLLABORATIONS
MRM
|
Analysis and/or data interpretation, Conception and design study, Data Curation, Final
manuscript approval, Formal Analysis, Funding Acquisition, Methodology, Project Administration,
Realization of operations and/or trials, Resources, Supervision, Writing - Review
& Editing
|
CGM
|
Analysis and/or data interpretation, Conception and design study, Data Curation, Formal
Analysis, Investigation, Methodology, Project Administration, Realization of operations
and/or trials, Writing - Original Draft Preparation
|
LG
|
Conception and design study, Methodology, Realization of operations and/or trials,
Writing - Original Draft Preparation
|
ACC
|
Analysis and/or data interpretation, Conception and design study, Methodology, Realization
of operations and/or trials
|
AOE
|
Analysis and/or data interpretation, Data Curation, Formal Analysis, Investigation,
Realization of operations and/or trials, Writing - Original Draft Preparation
|
OS
|
Conceptualization, Final manuscript approval, Methodology, Project Administration,
Resources, Supervision, Validation
|
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1. Serviço de Cirurgia Plástica Osvaldo Saldanha, Universidade Metropolitana de Santos,
Santos, SP, Brazil.
Corresponding author: Carlos Goyeneche Montoya Avenida Ana Costa, 146, Conj. 1201, Santos, SP, Brazil. Zip Code: 11060-002. E-mail:
carlosgoye.m@gmail.com
Article received: February 22, 2019.
Article accepted: October 21, 2019.
Conflicts of interest: none.