INTRODUCTION
Among the complications after primary palatoplasty, one of the most discussed in the
literature, is the formation of oronasal fistulas1-6. In the literature, it is observed that the occurrence of palate fistula is widely
variable, ranging from authors who report the absence of these complications (0%)
to those that indicate a 78% occurrence of palate fistula after primary palatoplasty7,8. The wide variation in the occurrence of fistula reflects the diversity of protocols
for primary surgical correction of the clef lip and palate. However, it can also be
related to the lack of consensus regarding terminology and the classification of fistula
according to what is reported by professionals from different areas of health9.
The terminology found in the literature to classify fistula is quite varied. Studies
report that fistulas located on the primary palate (anterior to the incisive foramen),
for example, can be called palate fistula, anterior palate fistula, labial alveolar
fistula, lingual alveolar fistula and also vestibular fistula5,10-12. Fistulas located on the secondary palate (or posterior to the incisive foramen),
in turn, can also be called palate fistulas, observing variation in terminology according
to the affected region (hard palate fistula, transition fistula between the hard palate
and soft palate, soft palate fistula)2,3,5,12,13.
Fistula classification systems involving a systematic approach to document the occurrence
and location of the fistula have been described in the international literature4,14. An acceptable level of agreement between evaluators in identifying fistula, however,
can be challenging to achieve even when a standardized protocol is implemented12. Characterizing aspects where there is a lack of consensus among evaluators during
fistula identification is essential for the development of a standardized classification
protocol, and that it can be implemented in Brazilian craniofacial services in order
to provide a systematic documentation of the results of primary palatoplasty11.
OBJECTIVE
This study aimed to verify the consensus among plastic surgery (PC) and Speech-Language
Pathology (SLP) professionals regarding the occurrence of fistula in the same craniofacial
service.
METHODS
The study was approved by the institution’s Human Research Ethics Committee under
number 1,337,917. This study involved an analysis of the presence and location of
fistulas in data in the medical records of 466 patients. The medical records studied
belonged to patients with unilateral cleft lip and palate, without syndromes or associated
malformations, of both sexes, who underwent primary palatoplasty in a single stage
by the surgical techniques of von Langenbeck or Furlow. The primary palatoplasties
of the studied group were performed between 1996 and 2004. The reports of the occurrence
of fistula of interest for this study were obtained during the clinical evaluation
and recorded in the post-surgical evaluation protocols in the patients’ medical records,
as is routinely performed at the research institution in the areas of PC and SLP.
For this study, records of fistula up to three years after primary palatoplasty were
included. (by the PC area, by the SLP area or by both areas).
In this work, dehiscence records (partial or total) were treated as a fistula. The
survey of data contained in the post-surgical evaluation forms of plastic surgery
gave rise to the registration of the occurrence of fistula by the PC area. However,
the survey of the data contained in the speech therapy assessment protocol gave rise
to the registration of the occurrence of fistula by the area of the SLP.
Clinical record of fistula by plastic surgery
The post-surgical evaluation protocol in the area of plastic surgery, in force during
the period studied, was applied by the plastic surgeon who performed a face-to-face
assessment based on an oral inspection of the areas of the hard and soft palate after
primary palatoplasty. The oral inspection was performed with the use of a flashlight
to illuminate the evaluated area and a spatula to lower the tongue and allow visualization
of the entire soft palate. When observing on the palate a region suggestive of false
fistula (false-bottom) or hidden fistula, the diagnostic tests performed by the professional
included: a) lighting with the flashlight to check the projection of the light in
the nasal area; b) palpation/manipulation of the irregular area of the palate, seeking
to verify false-bottom in recesses of tissue; c) use of air injection in the area
(using dental equipment) to check the passage of air to the nostrils through the patient’s
report or observation of bubble.
Data were recorded in person by the plastic surgeon in the post-surgical evaluation
protocol for plastic surgery, including the following observations: registration of
the method for closing the anterior palate and the soft palate (relaxing incisions;
vomerian flap, pharyngeal flap, others); transoperative complications (flap fraying,
suture under tension; review of hemostasis and others) and postoperative complications
(fistula or dehiscence; infection and others). In cases of fistula, the evaluation
protocol also requested an indication of the affected area and the drawing of the
occurrence in a diagram of the palate (as shown in the evaluation form). From the
post-surgical evaluation protocol for the PC area, therefore, the presence and location
of the fistula in the palate for the present study are identified.
Clinical record of fistula by Speech-Language Pathology
The speech therapy evaluation protocol, in effect during the period studied, was applied
by a speech therapist on the same day that the post-surgical PC evaluation protocol
was applied. For inclusion in the study, therefore, all patients had their PC and
SLP assessments performed in person on the same day, independently for each area.
During the evaluation, the speech therapist performed an inspection of the areas of
the hard and soft palate using a flashlight to illuminate the evaluated area and a
spatula to lower the tongue in order to visualize the entire soft palate, including
the uvula. In cases of identification of an area suggestive of a false or hidden fistula,
the Speech-Language Pathologist referred the patient for diagnostic testing of lighting,
palpation, and/or air injection by the plastic surgeon.
The data were recorded in person by the Speech-Language Pathologist. The evaluation
protocol included the following information regarding the fistula: absent; vestibular
on the right; vestibular on the left; on the hard palate; on the soft palate; in the
transition region from the hard palate to the soft palate. In the SLP area assessment
protocol; therefore, the registration of the presence and location of the fistula
was identified for the present study.
Gold standard classification for the occurrence of fistula (GSF)
Once the data of the protocols of the areas of the PC and the SLP were collected,
there was a divergence in the records between the areas, and it was decided to apply
a gold standard assessment of the occurrence of fistula (GSF). For the GSF assessment,
a fistula was defined as a failure of healing or a rupture of the suture, observed
after primary repair of the palate. That is, after an attempt to repair the tissues
in the area of the cleft palate, the unwanted opening of the sutures occurred.
The GSF evaluation was performed by a single professional in the field of plastic
surgery, with over 30 years of clinical experience in surgical correction of the palate
and in the post-palatoplasty evaluation. The GSF was not performed in person and was
based on the analysis of all records documented in the medical records about fistula
and also on the analysis of photographic images of the fistula, when existing. It
should be noted, however, that at the time of the post-surgical evaluation of the
studied cases, the photographic records of the fistulas, although indicated, were
not made in a standardized way and were not obtained for all cases. The photographs
in the institutional collection were taken by the institution’s photographer and not
by professionals from the areas of PC and SLP, and these images were used in a complementary
way to the survey of fistula records in the medical record.
In the medical record, all existing documentary protocols were consulted where the
occurrence of fistula could be registered. That is, in addition to the consultation
carried out in the PC and SLP protocols (objects of this work), documentation from
other areas (nursing, pediatrics and dentistry) was analyzed. For the analysis of
the photographs, the plastic surgery professional who performed the GSF evaluation
used the incisive foramen (IF) as an anatomical landmark1 and grouped the fistulas according to their location concerning the IF, indicating,
therefore, if the occurrences were before or after the IF.
The findings obtained from the analysis of all documentary records in the medical
record plus the findings from the analysis of existing photographs were combined to
establish the gold standard assessment of the occurrence of fistula (GSF). GSF was
the tool used as a reference for interpreting the findings recorded individually in
the protocols of the areas of PC and SLP, allowing researchers to corroborate and
compare the findings of this study. According to the GSF, 466 patients were grouped
into four categories regarding the existence and location of a fistula after primary
palatoplasty, including Group 1: patients who did not present a fistula (N = 302;
65%); Group 2: patients with fistulas located in the area anterior to the incisive
foramen (N = 91, 20%); Group 3: patients with fistulas located in the area posterior
to the incisive foramen (N = 43; 9%); Group 4: patients with fistulas covering the
area before and after the incisive foramen (N = 30; 6%). That is, while the majority
of patients did not have a fistula (65%), a total of 164 (35%) patients had some type
of fistula on the palate, as indicated in the GSF. Of the 164 fistulas identified
in the GSF assessment, 78 (17%) occurred in patients who received the Furlow procedure,
and 86 (18%) occurred in patients who received the von Langenbeck procedure. However,
it should be noted that data on the transversal amplitude of the fissure, on the surgical
technique, and the surgeon in primary palatoplasty was not the object of this study.
The data presented below include the percentages of occurrence of fistula reported
by the areas of PC, ST, and GSF. The concordance between the findings was verified
with Kappa statistics.
RESULTS
When analyzing the data obtained in the records of the PC area, it was observed that
the surgeons reported that 275 patients (59%) did not present fistulas, 117 patients
(25%) presented some type of fistula and 74 patients (16%) did not present records
about the presence or absence of fistula (no data). Of the 117 patients who had fistula
reported by the PC, 48 patients (10%) did not indicate the location (incomplete data),
31 patients (7%) had fistulas in the region before the IF, 32 patients (8%) had a
fistula in the region after IF and six patients (1%) presented fistulas involving
both regions, before and after IF.
The same analysis was done for the records in the area of the SLP, observing that
the speech therapists reported 295 patients (63%) without fistulas and 171 patients
(37%) with some type of fistula. Of the 171 patients who presented a fistula, 69 patients
(15%) did not indicate the location (incomplete data), 73 patients (15.6%) were fistulas
in the region before the IF, 27 patients (6%) were fistulas in the posterior region
at IF, and two patients (0.4%) were fistulas involving the two regions, anterior and
posterior to IF.
As shown in Table 1, the gold standard assessment of the occurrence of fistula (GSF) identified fistula
in 164 patients (35% of the cases studied), differing from the area of speech therapy
and plastic surgery, which reported 171 (37%) and 117 (25%) cases with fistula, respectively.
That is, when the findings reported in the specific protocol for plastic surgery were
considered, the occurrence of fistula was 10% less than the occurrence observed in
GSF. Furthermore, when considering the findings reported in the specific speech therapy
protocol, the occurrence of fistula was 2% higher than the occurrence observed in
GSF.
Table 1 - Presence and location of fistulas in the records of professionals in plastic surgery
(PC), Speech-Language Pathology (SLP), and in the gold standard assessment of the
occurrence of fistula (GSF).
| N=466 |
Without
Fistula
|
Total Fistula |
Fistula anterior
to the IF
|
Fistula posterior
to the IF
|
Anterior and
posterior IF
|
Incomplete
Data
|
No Data |
| PC |
275 (59%) |
117 (25%) |
31 (6%) |
32 (7%) |
6 (2%) |
48 (10%) |
74 (16%) |
| SLP |
295 (63%) |
171 (37%) |
73 (16%) |
27 (5%) |
2 (1%) |
69 (15%) |
0 |
| GSF |
302 (65%) |
164 (35%) |
91 (20%) |
43 (9%) |
30 (6%) |
0 |
0 |
Table 1 - Presence and location of fistulas in the records of professionals in plastic surgery
(PC), Speech-Language Pathology (SLP), and in the gold standard assessment of the
occurrence of fistula (GSF).
It was also observed that the indication of the location of the fistula (before or
after the IF) was not possible for 48 cases evaluated by the PC due to incomplete
data. That is, 10% of the fistulas identified by the PC did not indicate the place
of occurrence (incomplete data). Also, analyzing the findings of PC, it was found
that 74 patients (16%) had no record regarding the absence or presence of fistula
(no data). The data collected in the SLP protocols, in turn, did not allow the identification
of the location of the fistula for 69 (15%) cases due to incomplete data. There was
no case without information about the presence or absence of a fistula in the evaluation
of the SLP.
The agreement and disagreement between the GSF, PC, and SLP findings were presented
in percentages and compared with Kappa statistics. The data reported in Table 2 indicate that the findings regarding the identification and location of fistula reported
in the medical records by plastic surgeons agreed with the GSF assessment in 59.7%
of the cases. In comparison, the records of speech therapists agreed with the GSF
assessment in 79.6% % of cases. According to the Kappa statistic, there was a regular
agreement between the GSF and the reports in the PC area (r = 0.32) and a substantial
agreement between the GSF and the reports in the SLP area (r = 0.63).
Table 2 - Percentage of agreement and disagreement between the findings of plastic surgery (PC),
Speech-Language Pathology (SLP), and the gold standard assessment of the occurrence
of fistula (GSF).
| Areas GSF |
Plastic surgery (r=0,32) |
Speech-Language Pathology (r=0,63) |
| PC Agree |
PC Disagree |
SLP Agree |
SLP Disagree |
| Without Fistula (N=302) |
233 (77.2%) |
69 (22.8%) |
285 (94.4%) |
17 (5.6%) |
| Fistula anterior to the IF (N=91) |
19 (20.9%) |
72 (79.1%) |
65 (71.4%) |
26 (28.6%) |
| Fistula posterior to the IF (N=43) |
21 (48.8%) |
22 (51.2%) |
20 (46.5%) |
23 (53.5%) |
| Anterior and posterior IF (N=30) |
5 (16.7%) |
25 (83.3%) |
1 (3.3%) |
29 (96.6%) |
| Total |
278 (59.7%) |
188 (40.3%) |
371 (79.6 %) |
95 (20.4%) |
Table 2 - Percentage of agreement and disagreement between the findings of plastic surgery (PC),
Speech-Language Pathology (SLP), and the gold standard assessment of the occurrence
of fistula (GSF).
It is noted that the highest percentage of agreement between professionals in the
areas of PC and SLP with GSF, was observed for cases where there was no fistula, with
77.2% for the PC area and 94.4% for the SLP area. The highest percentage of disagreement
with the GSF was for the group of fistulas that involved both the anterior and posterior
areas of the IF, 83.3% for the PC area, and 96.6% for the SLP area.
DISCUSSION
The data in the present study reflect disagreement between the areas of plastic surgery
and speech therapy, in the same craniofacial center, when reporting the occurrence
and location of a fistula after primary palatoplasty. The difficulty in interpreting
data from medical records and the lack of information regarding the occurrence or
location of a fistula justified these findings and was mentioned in other studies
4,5,10,11,13,15,16.
Comparing the findings about the occurrence of fistula observed in this study with
the findings reported in the literature is a complex task, since, according to the
area of PC, a total of 25% of the 466 patients had a fistula, and according to the
area of the SLP a total of 37% had a fistula. The GSF evaluation, in turn, suggested
that a total of 35% of the cases had a fistula. This disagreement can be explained
both by the lack of data and by the incomplete data aggravated by the disagreement
as to the terminology to indicate the location of the fistula.
Since these are patients with transforamenal cleft, it was expected that the highest
incidence of fistula would occur in the pre-foramen incisor region, which was documented
in this study by the SLP area and the GSF assessment. The area of PC, in turn, does
not always consider the pre-foramen fistula to be a surgical complication, since the
“intentional fistula” may be the result of a clinical decision taken at the time of
surgery depending on the width of the cleft palate and the surgical procedure performed,
which justifies, in part, the absence of data for 74 patients observed in the protocols
completed by the PC area. It should be noted, even when the pre-foramen fistula affects
only the vestibular region, the inspection exam and intraoral photographs may not
be sufficient for the correct identification of this type of fistula. A standardized,
systematic, and consecutive documentation of the results of the surgeries, therefore,
must be established by the interdisciplinary team, as it is essential and necessary
for the identification of surgical complications.
The task force known as “Task Force Beyond Eurocleft”17, reports the importance of multicentric studies (national and international) to document
the results of the management of cleft lip and palate, so that scientific evidence
can be established to substantiate the use of treatment protocols with acceptable
results worldwide. The absence of a standardized and validated protocol for documenting
complications after primary surgeries in cleft lip and palate in Brazil, in some way,
justifies the existence of conflicting reports. Also, it makes the performance of
comparative studies between different craniofacial centers a complicated task.
The gold standard assessment of the occurrence of fistula used as a tool to compare
the findings of this study should be considered with caution, since, in addition to
not being an assessment performed in person, it considered data from various areas
of the medical record (in addition to the PC and ST) combined with the photo analysis
findings. It should also be noted that intraoral photographs were not obtained for
all cases (with a fistula and also without a fistula). That is, all 466 cases should
have intraoral photographic images obtained using a standardized protocol and with
quality control. The lack of a single terminology and a standardized post-surgical
evaluation protocol and used in consensus by the areas of PC and ST, therefore, justifies
the divergence found in this study.
In addition to the methodological limitations concerning photographic images that
were not obtained for all 466 cases and the fact that the GSF assessment was not performed
in person, it is also noted that information on the transversal width of the fissure
and the surgeon in the primary palatoplasty was not the subject of this study.
Regardless of the limitations existing in the methodology implemented in the present
study, the present data showed the lack of consensus regarding the reports of fistula,
suggesting that a systematic and adequate documentation of the results of the primary
palatoplasty will only be possible based on a reliable record of the clinical findings
registered interprofessionally, in person and also with photographic images registration.
It is suggested, therefore, the need to establish and validate a fistula classification
protocol, which can be applied both during the face-to-face oral inspection and from
the analysis of intraoral photographs (which must be obtained for all cases: with
and without fistula).
CONCLUSION
In this study, there was a discrepancy between the areas of speech therapy and plastic
surgery regarding the occurrence and location of the fistula after primary palatoplasty
in the same craniofacial center. The data point to the need for adjustments in the
evaluation protocols that take into account the terminology and location of the fistula.
It is suggested to be essential to create and implement a standardized fistula classification
system, which can be used effectively, consecutively and systematically, by craniofacial
teams, in order to provide multicenter studies that can establish scientific evidence
of the results of the treatment of the cleft lip and palate.
COLLABORATIONS
|
MFJ
|
Analysis and/or data interpretation, conception and design study, data curation, final
manuscript approval, formal analysis, methodology, project administration, writing
- original draft preparation, writing - review & editing.
|
|
GAP
|
Analysis and/or data interpretation, data curation, final manuscript approval, writing
- original draft preparation, writing - review & editing.
|
|
TVSB
|
Analysis and/or data interpretation, data curation, final manuscript approval, writing
- original draft preparation, writing - review & editing.
|
|
HLAS
|
Analysis and/or data interpretation, data curation, final manuscript approval, methodology,
writing - review & editing.
|
|
JCRD
|
Analysis and/or data interpretation, conception and design study, final manuscript
approval, formal analysis, project administration, supervision, writing - original
draft preparation, writing - review & editing.
|
REFERENCES
1. Williams WN, Seagle MB, Pegoraro-Krook MI, Souza TV, Garla L, Silva ML, et al. Prospective
clinical trial comparing outcome measures between Furlow and von Langenbeck palatoplasties
for UCLP. Ann Plast Surg. 2011 Feb;66(2):154-63.
2. Deshpande GS, Campbell A, Jagtap R, Restrepo C, Dobie H, Keenan HT, et al. Early complications
after cleft palate repair: a multivariate statistical analysis of 709 patients. J
Craniofac Surg. 2014 Sep;25(5):1614-8.
3. Hardwicke JT, Landini G, Richard BM. Fístula incidence after primary cleft palate
repair: a systematic review of the literature. Plast Reconstr Surg. 2014 Oct;134(4):618e-27e.
4. Smith DM, Vecchione L, Jiang S, Ford M, Deleyiannis FW, Haralam MA et al. The Pittsburgh
fistula classification system: a standardized scheme for the description of palatal
fistulas. Cleft Palate Craniofac J. 2007 Nov;44(6):590-4.
5. Winters R, Carter JM, Givens V, St Hilaire H. Persistent oro-nasal fístula after primary
cleft palate repair: minimizing the rate via a standardized protocol. Int J Pediatr
Otorhinolaryngol. 2014 Jan;78(1):132-4.
6. Muzaffar AR, Byrd HS, Rohrich RJ, Johns DF, LeBlanc D, Beran SJ, et al. Incidence
of cleft palate fístula: an institutional experience with two-stage palatal repair.
Plast Reconstr Surg. 2001 Nov;108(6):1515-8.
7. Xu JH, Chen H, Tan WQ, Lin J, Wu WH. The square flap method for cleft palate repair.
Cleft Palate Craniofac J. 2007 Nov;44(6):579-84.
8. Mak SY, Wong WH, Or CK, Poon AM. Incidence and cluster occurrence of palatal fistula
after furlow palatoplasty by a single surgeon. Ann Plast Surg. 2006 Jul;57(1):55-9.
9. Bykowski MR, Naran S, Winger DG, Losee JE. The rate of oronasal fistula following
primary cleft palate surgery: a meta-analysis. Cleft Palate Craniofac J. 2015 Jul;52(4):e81-7.
10. Eberlinc A, Koželj V. Incidence of residual oronasal fistulas: a 20-year experience.
Cleft Palate Craniofac J. 2012 Nov;49(6):643-8.
11. Passos VAB, Carrara CFC, Dalben GS, Costa B, Gomide MR. Prevalence, cause, and location
of palatal fistula in operated complete unilateral cleft lip and palate: retrospective
study. Cleft Palate Craniofac J. 2014 Mar;51(2):158-64.
12. Richardson S, Agni NA. Palatal fistulae: a comprehensive classification and difficulty
index. J Maxillofac Oral Surg. 2014 Sep;13(3):305-9.
13. Diah E, Lo LJ, Yun C, Wang R, Wahyuni LK, Chen YR. Cleft oronasal fistula: a review
of treatment results and a surgical management algorithm proposal. Chang Gung Med
J. 2007 Nov/Dec;30(6):529-37.
14. Fayyaz GQ, Gill NA, Ishaq I, Aslam M, Chaudry A, Ganatra MA, et al. Pakistan comprehensive
fistula classification: a novel scheme and algorithm for management of palatal fístula/dehiscence.
Plast Reconstr Surg. 2019 Jan;143(1):140e-51e.
15. Phua YS, Chalain T. Incidence of oronasal fistulae and velopharyngeal insufficiency
after cleft palate repair: an audit of 211 children born between 1990 and 2004. Cleft
Palate Craniofac J. 2008 Mar;45(2):172-8.
16. Ahmed MK, Maganzini AL, Marantz PR, Rousso JJ. Risk of persistent palatal fistula
in patients with cleft palate. JAMA Facial Plast Surg. 2015 Mar/Apr;17(2):126-30.
17. Semb G. International Confederation for cleft lip and palate and related craniofacial
anomalies task force report: beyond Eurocleft. Cleft Palate Craniofac J. 2014 Nov;51(6):e146-55.
1. Hospital de Reabilitação de Anomalias Craniofaciais, Universidade de São Paulo,
Bauru, SP, Brazil.
2. Universidade de São Paulo, Bauru, SP, Brazil.
*Corresponding author: Jeniffer de Cassia Rillo Dutka Rua Silvio Marchione, nº3-20 - Vila Nova Cidade Universitária, Bauru, SP, Brazil
Zip Code: 17012-900 E-mail: jdutka@usp.br
Article received: June 27, 2019.
Article accepted: February 29, 2020.
Conflicts of interest: none.
Institution: Hospital de Reabilitação de Anomalias Craniofaciais, Universidade de
São Paulo, Bauru, SP, Brazil.
