Special Topic Fistula Incidence after Primary Cleft Palate Repair: A Systematic Review of the Literature Joseph T. Hardwicke, Ph.D., F.R.C.S.(Plast.) Gabriel Landini, Ph.D. Bruce M. Richard, M.S., F.R.C.S.(Plast.) Birmingham, United Kingdom
Background: The development of an oronasal fistula after primary cleft palate repair has a wide variation reported in the literature. The aim of this review is to identify the reported oronasal fistula incidence to provide a benchmark for surgical practice. Methods: A systematic review was undertaken to investigate the incidence of fistula. Multiple meta-analyses were performed to pool proportions of reported fistulae, in each data set corresponding to the continent of origin of the study, type of cleft, and techniques of cleft palate repair used. Results: A total of 9294 patients were included from 44 studies. The overall incidence of reported fistula was 8.6 percent (95 percent CI, 6.4 to 11.1 percent). There was no significant difference in the fistula incidence corresponding to the continent of origin of each study or the repair technique used. The incidence of fistula in cleft lip–cleft palate was 17.9 percent, which was significantly higher (p = 0.03) than in cases of cleft palate alone (5.4 percent). Conclusions: Palatal fistulae were more likely to occur in cases of combined cleft lip–cleft palate, compared with cleft palate alone. The authors would recommend the prospective examination and recording of all fistulae to a standardized classification scheme. (Plast. Reconstr. Surg. 134: 618e, 2014.) CLINICAL QUESTION/LEVEL OF EVIDENCE: Therapeutic, III.
T
he risk of development of an oronasal fistula after primary cleft palate repair is well known. The literature reports a wide variation in incidence, ranging from 0 to 77.8 percent,1,2 and the presence of a fistula is one of the important factors indicating the early outcomes of surgery.3 Palatal fistulae are located in the secondary palate connecting the oral and nasal cavities,4–6 including fistulae of the hard palate, the transition between hard and soft palates, and the soft palate.5–9 However, other authors also record fistulae of the primary palate, including linguoalveolar and labioalveolar fistulae,10–14 although this will include intentional, unrepaired fistulae. Some groups have attempted to standardize the nomenclature associated with palatal fistulae to reduce ambiguity in reporting.14,15 Rather than the actual classification, it may be used to report small, or asymptomatic fistulae that do not require surgical From the Department of Plastic Surgery, Birmingham Children’s Hospital NHS Foundation Trust; and the School of Dentistry, University of Birmingham. Received for publication January 6, 2014; accepted March 19, 2014. Copyright © 2014 by the American Society of Plastic Surgeons DOI: 10.1097/PRS.0000000000000548
618e
intervention, which may be deficient in the literature. The continuity of care from birth to adulthood may not be the standard of care worldwide, and the long-term follow-up may reveal the late presentation of fistulae.16 As such, the true incidence of oronasal fistula may be underreported in the literature. Postoperative oronasal fistula occurs because of a failure of normal palatal wound healing after surgical repair.11,17 It may be related to patient factors such as age at operation,4 type and extent of cleft,17 and associated syndromes.18 Operative factors such as experience of the operating surgeon,4,11 tension at the site of repair, bleeding, and infection19 have also been implicated. Fistulae of significant sizes can lead to nasal air escape, difficulty with articulation, and nasal regurgitation of food, all of which may require repair. With a wide range of surgical techniques, schedules for palate repair, and multiple postoperative management plans, there is little consensus and uniformity worldwide in the approach to this
Disclosure: The authors have no financial interest to declare in relation to the content of this article.
www.PRSJournal.com
Volume 134, Number 4 • Postoperative Palatal Fistulae problem. Current ongoing multicenter studies have set the objective to identify best practices.20 At present, there is little high-quality evidence available to determine the optimum conditions that prevent fistula formation. The aim of this review is to identify the reported oronasal fistula incidence in the literature, to interrogate the data for possible factors that may influence fistula development, and to provide a standard for surgical practice to be benchmarked against.
PATIENTS AND METHODS Data Sources We conducted a systematic literature review of publications in English of the following electronic databases: Cochrane Database of Systematic Reviews, Cochrane Central Register of Controlled Trials, MEDLINE, and Embase. The following key words were used: (cleft) AND (palate) AND (fistula) AND (repair OR palatoplasty). The date range was from January 1, 2000, to January 9, 2013. Study Selection Articles were included if they met the following criteria: Population: Human participants with nonsyndromic cleft palate who underwent primary cleft palate repair. Intervention: Case series, cohort studies, controlled trials, and randomized controlled trials of patients undergoing primary repair of cleft palate; all ages at intervention; all surgical repair techniques; prospective or retrospective data collection. Outcome: Number of reported oronasal fistulae as a proportion of the entire cohort. A fistula was defined as an unintentional connection between the oral and nasal mucosa of the secondary palate arising after primary cleft palate repair, which may be symptomatic or asymptomatic. Study selection was performed through two levels of screening. In the first level, abstracts were reviewed for the exclusion criteria (summarized in Table 1).1,2,5,9,13,17,21–57 In the second level screening, all articles filtered through the first level were read in their entirety and the same inclusion and exclusion criteria were applied. Only studies that successfully passed both levels of screening were included in our analysis. The present investigation is reported using the Meta-analysis Of
Observational Studies in Epidemiology guidelines for metaanalyses of observational studies.58 Assessment of Methodologic Quality The methodologic quality of randomized controlled trials was assessed using the Detsky scale.59 The methodologic quality of nonrandomized studies was assessed using the Methodological Index for Nonrandomized Studies60 instrument. We appraised each study and calculated a Detsky score from a maximum of 20, or a Methodological Index for Nonrandomized Studies score from a maximum of 16 for noncomparative studies and 24 for comparative studies. Studies that received at least 75 percent of the maximum Methodological Index for Nonrandomized Studies or Detsky score were considered to be high quality, which is consistent with previous research.60–63 Data Extraction We recorded data using Microsoft Excel (Microsoft, Corp., Redmond, Wash.). The extracted data are summarized in Table 1. Articles were included if a subgroup of patients fulfilling the exclusion criteria (e.g., Pierre Robin sequence) could be extracted from the reported cohort. The minimum length of follow-up was recorded as in the study by Becker and Hansson.24 Data Synthesis and Analysis We performed multiple meta-analyses to pool proportions of reported fistulae in each data set corresponding to the continent of origin of the study, type of cleft, and techniques of cleft palate repair used. We first transformed proportions by means of the Freeman-Tukey double arcsine method64,65 and then calculated the pooled proportions as the back-transform of the weighted mean of the transformed proportions, using a random effects model with a 95 percent confidence interval: we tested the significance of heterogeneity between studies using the Cochran Q test66 and selected the random effects model. We conducted a meta-regression based on a random effects logistic model for proportion of fistulae between each data set. Analysis of pooled proportions was performed and presented using StatsDirect (StatsDirect Ltd, Cheshire, United Kingdom), and IBM SPSS Statistics for Windows, Version 19.0 (IBM Corp., Armonk, N.Y.) was used for meta-regression. The rest of the data were summarized and reported in a descriptive manner. The threshold considered for statistical significance was p < 0.05.
619e
Plastic and Reconstructive Surgery • October 2014 Table 1. Inclusion and Exclusion Criteria Applied to the Screened Articles and Data Selected for Extraction Inclusion Criteria Population
Patients undergoing primary cleft palate repair Nonsyndromic cleft palate
Intervention
All types of cleft palate Human participants Randomized and nonrandomized studies; noncomparative studies; case series Unique cohort with oronasal fistulae previously unreported in the literature Study cohort of >20 patients English language literature All surgical repair techniques and schedules
Outcome
Prospective or retrospective data collection Patients with unintentional postoperative oronasal fistulae
Exclusion Criteria Any secondary cleft surgery (e.g., fistula repair, revision palatoplasty)* Diagnosis of cleft palate as part of a sequence or syndrome* Other craniofacial clefts* Nonhuman studies Single case reports; review articles Cohort with oronasal fistulae previously reported in the literature* Study cohort of
Background: The development of an oronasal fistula after primary cleft palate repair has a wide variation reported in the literature. The aim of this review is to identify the reported oronasal fistula incidence to provide a benchmark for surgical practice. Methods: A systematic review was undertaken to investigate the incidence of fistula. Multiple meta-analyses were performed to pool proportions of reported fistulae, in each data set corresponding to the continent of origin of the study, type of cleft, and techniques of cleft palate repair used. Results: A total of 9294 patients were included from 44 studies. The overall incidence of reported fistula was 8.6 percent (95 percent CI, 6.4 to 11.1 percent). There was no significant difference in the fistula incidence corresponding to the continent of origin of each study or the repair technique used. The incidence of fistula in cleft lip–cleft palate was 17.9 percent, which was significantly higher (p = 0.03) than in cases of cleft palate alone (5.4 percent). Conclusions: Palatal fistulae were more likely to occur in cases of combined cleft lip–cleft palate, compared with cleft palate alone. The authors would recommend the prospective examination and recording of all fistulae to a standardized classification scheme. (Plast. Reconstr. Surg. 134: 618e, 2014.) CLINICAL QUESTION/LEVEL OF EVIDENCE: Therapeutic, III.
T
he risk of development of an oronasal fistula after primary cleft palate repair is well known. The literature reports a wide variation in incidence, ranging from 0 to 77.8 percent,1,2 and the presence of a fistula is one of the important factors indicating the early outcomes of surgery.3 Palatal fistulae are located in the secondary palate connecting the oral and nasal cavities,4–6 including fistulae of the hard palate, the transition between hard and soft palates, and the soft palate.5–9 However, other authors also record fistulae of the primary palate, including linguoalveolar and labioalveolar fistulae,10–14 although this will include intentional, unrepaired fistulae. Some groups have attempted to standardize the nomenclature associated with palatal fistulae to reduce ambiguity in reporting.14,15 Rather than the actual classification, it may be used to report small, or asymptomatic fistulae that do not require surgical From the Department of Plastic Surgery, Birmingham Children’s Hospital NHS Foundation Trust; and the School of Dentistry, University of Birmingham. Received for publication January 6, 2014; accepted March 19, 2014. Copyright © 2014 by the American Society of Plastic Surgeons DOI: 10.1097/PRS.0000000000000548
618e
intervention, which may be deficient in the literature. The continuity of care from birth to adulthood may not be the standard of care worldwide, and the long-term follow-up may reveal the late presentation of fistulae.16 As such, the true incidence of oronasal fistula may be underreported in the literature. Postoperative oronasal fistula occurs because of a failure of normal palatal wound healing after surgical repair.11,17 It may be related to patient factors such as age at operation,4 type and extent of cleft,17 and associated syndromes.18 Operative factors such as experience of the operating surgeon,4,11 tension at the site of repair, bleeding, and infection19 have also been implicated. Fistulae of significant sizes can lead to nasal air escape, difficulty with articulation, and nasal regurgitation of food, all of which may require repair. With a wide range of surgical techniques, schedules for palate repair, and multiple postoperative management plans, there is little consensus and uniformity worldwide in the approach to this
Disclosure: The authors have no financial interest to declare in relation to the content of this article.
www.PRSJournal.com
Volume 134, Number 4 • Postoperative Palatal Fistulae problem. Current ongoing multicenter studies have set the objective to identify best practices.20 At present, there is little high-quality evidence available to determine the optimum conditions that prevent fistula formation. The aim of this review is to identify the reported oronasal fistula incidence in the literature, to interrogate the data for possible factors that may influence fistula development, and to provide a standard for surgical practice to be benchmarked against.
PATIENTS AND METHODS Data Sources We conducted a systematic literature review of publications in English of the following electronic databases: Cochrane Database of Systematic Reviews, Cochrane Central Register of Controlled Trials, MEDLINE, and Embase. The following key words were used: (cleft) AND (palate) AND (fistula) AND (repair OR palatoplasty). The date range was from January 1, 2000, to January 9, 2013. Study Selection Articles were included if they met the following criteria: Population: Human participants with nonsyndromic cleft palate who underwent primary cleft palate repair. Intervention: Case series, cohort studies, controlled trials, and randomized controlled trials of patients undergoing primary repair of cleft palate; all ages at intervention; all surgical repair techniques; prospective or retrospective data collection. Outcome: Number of reported oronasal fistulae as a proportion of the entire cohort. A fistula was defined as an unintentional connection between the oral and nasal mucosa of the secondary palate arising after primary cleft palate repair, which may be symptomatic or asymptomatic. Study selection was performed through two levels of screening. In the first level, abstracts were reviewed for the exclusion criteria (summarized in Table 1).1,2,5,9,13,17,21–57 In the second level screening, all articles filtered through the first level were read in their entirety and the same inclusion and exclusion criteria were applied. Only studies that successfully passed both levels of screening were included in our analysis. The present investigation is reported using the Meta-analysis Of
Observational Studies in Epidemiology guidelines for metaanalyses of observational studies.58 Assessment of Methodologic Quality The methodologic quality of randomized controlled trials was assessed using the Detsky scale.59 The methodologic quality of nonrandomized studies was assessed using the Methodological Index for Nonrandomized Studies60 instrument. We appraised each study and calculated a Detsky score from a maximum of 20, or a Methodological Index for Nonrandomized Studies score from a maximum of 16 for noncomparative studies and 24 for comparative studies. Studies that received at least 75 percent of the maximum Methodological Index for Nonrandomized Studies or Detsky score were considered to be high quality, which is consistent with previous research.60–63 Data Extraction We recorded data using Microsoft Excel (Microsoft, Corp., Redmond, Wash.). The extracted data are summarized in Table 1. Articles were included if a subgroup of patients fulfilling the exclusion criteria (e.g., Pierre Robin sequence) could be extracted from the reported cohort. The minimum length of follow-up was recorded as in the study by Becker and Hansson.24 Data Synthesis and Analysis We performed multiple meta-analyses to pool proportions of reported fistulae in each data set corresponding to the continent of origin of the study, type of cleft, and techniques of cleft palate repair used. We first transformed proportions by means of the Freeman-Tukey double arcsine method64,65 and then calculated the pooled proportions as the back-transform of the weighted mean of the transformed proportions, using a random effects model with a 95 percent confidence interval: we tested the significance of heterogeneity between studies using the Cochran Q test66 and selected the random effects model. We conducted a meta-regression based on a random effects logistic model for proportion of fistulae between each data set. Analysis of pooled proportions was performed and presented using StatsDirect (StatsDirect Ltd, Cheshire, United Kingdom), and IBM SPSS Statistics for Windows, Version 19.0 (IBM Corp., Armonk, N.Y.) was used for meta-regression. The rest of the data were summarized and reported in a descriptive manner. The threshold considered for statistical significance was p < 0.05.
619e
Plastic and Reconstructive Surgery • October 2014 Table 1. Inclusion and Exclusion Criteria Applied to the Screened Articles and Data Selected for Extraction Inclusion Criteria Population
Patients undergoing primary cleft palate repair Nonsyndromic cleft palate
Intervention
All types of cleft palate Human participants Randomized and nonrandomized studies; noncomparative studies; case series Unique cohort with oronasal fistulae previously unreported in the literature Study cohort of >20 patients English language literature All surgical repair techniques and schedules
Outcome
Prospective or retrospective data collection Patients with unintentional postoperative oronasal fistulae
Exclusion Criteria Any secondary cleft surgery (e.g., fistula repair, revision palatoplasty)* Diagnosis of cleft palate as part of a sequence or syndrome* Other craniofacial clefts* Nonhuman studies Single case reports; review articles Cohort with oronasal fistulae previously reported in the literature* Study cohort of
