Oronasal Fistula: A Complication of Cleft Palate Surgery

Oronasal fistula is one of the disappointing expected outcomes of cleft palate surgery. It may follow palatoplasty for palatal clefting patients, which is defined as an abnormal communication between the oral and nasal cavities. More than one system has attempted its classification. It may be a small and a symptomatic or symptomatic fistula that requires correction. Its rate differs widely according to many factors. Both surgical and non-surgical methods (the pros and cons of each should be considered) are available for its correction, and there are adjunctive therapies to assist in improving the surgical outcome.

Reinforcing the Modified Double-Opposing Z-Plasty Approach Using the Pedicled Buccal Fat Flap as an Interpositional Layer for Cleft Palate Repair

Pedicled buccal fat flaps have been adopted in primary Furlow double-opposing Z-plasty palatoplasty to reduce oronasal fistula formation or to attenuate maxillary growth disturbance. We combined both goals in a single intervention. This study describes a series of 33 modified Furlow small double-opposing Z-plasty palatoplasties reinforced with a middle layer of pedicled buccal fat flaps between the oral and nasal layers for full coverage of the dissected palatal surfaces, with rapid mucosalization of lateral relaxing incisions and no dehiscence or fistula formation.

NK/T cell Lymphoma as a Rare Cause of an Oronasal Fistula

NK/T cell lymphoma is one of the most unique and rare forms of extranodal non-Hodgkin’s lymphoma, mostly derived from natural killer cell lineages and occasionally cytotoxic T cell lines. Due to the non-specific presentation of NK/T cell lymphoma such as nasal obstruction, nasal discharge, and epistaxis, diagnosis is often an issue and can be misleading. Oronasal fistula following a non-healing ulcer on the soft palate, can be one of the clinical presentations of NK/T cell lymphoma. Here, we are reporting a rare case of NK/T cell lymphoma in a 32-year-old gentleman who presented with an oronasal fistula post tissue biopsy for a non-healing ulcer over the soft palate. The tissue biopsy of the soft palate mass was revealed as NK/T cell lymphoma and was staged as Stage1b after computed tomographic imaging revealed a local tumour invasion without sign of nodal involvement and no metastasis. The patient eventually started with the SMILE Protocol and responded well up to the date. The ulcer healed but the fistula persisted. Primary closure was planned by the oromaxillofacial surgery team after completion of chemotherapy.

Speech Outcome Analysis after Primary Cleft Palate Repair: Interim Siriraj Hospital Audit

Objective: To evaluate the speech outcomes after primary cleft palate repair in a single tertiary medical institution of Thailand.Materials and Methods: A prospective cohort study was performed. Patients who had cleft palate with/without cleft lip and underwent primary cleft palate repair were included. Speech assessment was performed using the Pittsburgh weighted speech score (PWSS) by a speech-language pathologist.Results: Forty patients (21 males and 19 females) who underwent primary cleft palate repair at Siriraj Hospital were included. The median age at the time of speech evaluation was 7 years. The median age at primary cleft palate surgery was 12 months. The predominant cleft palate type was Veau 3 (47.5%). Oronasal fistula occurred 40%. Two-flap palatoplasty and intravelar veloplasty were the most common procedures. Median PWSS was 7, in which the competence velopharyngeal mechanism was found 5%, borderline competence 10%, borderline incompetence 32.5%, and incompetence velopharyngeal mechanism 52.5%. Among the velopharyngeal incompetence group, articulation disorder was the most common disorder with median score of 3. Besides, the median scores for hypo/hyper-nasality, nasal emission, phonation, and facial grimace disorder were 1, 2, 0 and 0, respectively. There was no statistically significant association between velopharyngeal incompetence and cleft types, age at primary surgery, type of operation, the width of cleft palate and prevalence of postoperative oronasal fistula or otitis media effusion.Conclusion: Velopharyngeal incompetence has been commonly identified after cleft palate repair in our institute. The articulation disorder is the most common characteristic.

Alveolar Cleft Reconstruction Using Double Iliac Corticocancellous Bone Plates Grafting Technique in Mixed Dentition Phase

Objective The primary goal of maxillary alveolar cleft reconstruction in patients with cleft lip/palate at the phase of mixed dentition is to build bone in the cleft area which in turn allows closure of the oronasal fistula, establishes arch continuity, and improve maxillary stability. This study aimed to evaluate the double iliac corticocancellous bone plates grafting technique for initial alveolar cleft grafting. Design This prospective study was conducted on 12 consecutive patients with unilateral complete alveolar cleft and previous cleft lip and palate corrective surgery. Intervention For all patients, the iliac crest graft was harvested and cut into 2 cortical bone plates and adapted labially and palatally. Both plates were fixed with screws then the gap between the 2 plates was filled with cancellous bone. The grafted side was compared to the contralateral side 9 months postoperatively regarding labio-palatal alveolar width and bone density, while the 9 months postoperative graft height was compared to the immediate postoperative height using computed tomography scans. Results All grafted sides showed similarity to the contralateral sides regarding both alveolar width and bone density with minimal crestal bone resorption. Conclusion The double iliac corticocancellous bone plate grafting technique seems to be a reliable method for alveolar cleft reconstruction with adequate bone quality and contour.

Predictors of Success After Furlow Palatoplasty for Repair of Cleft Palate: Does Syndrome Matter?

Objective To determine predictors of success following Veau 1 and 2 cleft palate repair in patients with and without syndromes. Study Design Retrospective review of prospectively collected data. Setting Tertiary care children’s hospital. Methods All children <18 months of age undergoing Furlow palatoplasty for Veau 1 and 2 cleft repair between 2000 and 2014 with postoperative perceptual speech assessment (PSA). Results In total, 368 consecutive patients were identified; 95 were excluded, resulting in 273 patients. Median age at surgery was 13.0 months (interquartile range [IQR], 11-15 months) with postoperative PSA at a median of 32.3 months (IQR, 26.3-44.5 months). Fifty patients (18.3%) had syndrome diagnosis; 59 patients (21.6%) had nonsyndromic Robin sequence. Velopharyngeal insufficiency (VPI) occurred in 27 patients (10.5%); 13 underwent secondary speech surgery. Cleft-related speech errors occurred in 46 patients (17.6%). Non-cleft-related speech errors occurred in 155 patients (59.6%) and reduced intelligibility in 127 patients (47.9%). Oronasal fistula occurred in 23 patients (8.8%) and was exclusive to Veau 2 clefts. In multivariate analysis, age >13 months at palatoplasty demonstrated a 6-fold higher rate of VPI (hazard ratio [HR], 6.64; P < .01), worse speech outcomes (HR, 6.04; P < .01; HR, 1.60; P < .01; HR, 1.57; P = .02), and greater speech therapy utilization (HR, 2.18; P < .01). Conclusion VPI occurred in 10% of patients undergoing Furlow palatoplasty repair of Veau 1 or 2 clefts. Age <13 months at palatoplasty was associated with improved speech outcomes and lower VPI incidence (2.8% vs 16.2%). Syndromic diagnosis was associated with noncleft speech errors and reduced intelligibility on univariate analysis but not velopharyngeal function after palatoplasty.

Local delivery of FTY720 induces neutrophil activation through chemokine signaling in an oronasal fistula model

Purpose Cleft palate repair surgeries lack a regenerative reconstructive option and, in many cases, develop complications including oronasal fistula (ONF). Our group has developed a novel murine phenocopy of ONF to study the oral cavity wound healing program. Using this model, our team previously identified that delivery of FTY720 on a nanofiber scaffold had a unique immunomodulatory effect directing macrophages and monocytes into a pro-regenerative state during ONF healing. Here, the objective of this study was to determine the effects of local biomaterial-based FTY720 delivery in the ONF model on the early bulk gene expression and neutrophil phenotypic response within the regenerating tissue. Methods Using a mouse model of ONF formation, a palate defect was created and was treated with FTY720 nanofiber scaffolds or (blank) vehicle control nanofibers. At 1 and 3 days post-implantation, ONF oral mucosal tissue from the defect region was collected for RNA sequencing analysis or flow cytometry. For the RNA-seq expression profiling, intracellular pathways were assessed using the KEGG Pathway database and Gene Ontology (GO) Terms enrichment interactive graph. To assess the effects of FTY720 on different neutrophil subpopulations, flow cytometry data was analyzed using pseudotime analysis based on Spanning-tree Progression Analysis of Density-normalized Events (SPADE). Results RNA sequencing analysis of palate mucosa injured tissue identified 669 genes that were differentially expressed (DE) during the first 3 days of ONF wound healing after local delivery of FTY720, including multiple genes in the sphingolipid signaling pathway. Evaluation of the DE genes at the KEGG Pathway database also identified the inflammatory immune response pathways (chemokine signaling, cytokine-cytokine receptor interaction, and leukocyte transendothelial migration), and the Gene Ontology enrichment analysis identified neutrophil chemotaxis and migration terms. SPADE dendrograms of CD11b+Ly6G+ neutrophils at both day 1 and day 3 post-injury showed significantly distinct subpopulations of neutrophils in oral mucosal defect tissue from the FTY720 scaffold treatment group compared to the vehicle control group (blank). Increased expression of CD88 and Vav1, among other genes, were found and staining of the ONF area demonstrated increased VAV1 staining in FTY720‐treated healing oral mucosa. Conclusion Treatment of oral mucosal defects using FTY720 scaffolds is a promising new immunotherapy to improve healing outcomes and reducing ONF formation during cleft palate surgical repair. Local delivery of FTY720 nanofiber scaffolds during ONF healing significantly shifted early gene transcription associated with immune cell recruitment and modulation of the immune microenvironment results in distinct neutrophil subpopulations in the oral mucosal defect tissue that provides a critical shift toward pro-regenerative immune signaling.

Is Risk of Secondary Surgery for Oronasal Fistula Following Primary Cleft Palate Repair Associated With Hospital Case Volume and Cost-to-Charge Ratio?

Objective: This study assesses the association between risk of secondary surgery for oronasal fistula following primary cleft palate repair and 2 hospital characteristics—cost-to-charge ratio (RCC) and case volume of cleft palate repair. Design: Retrospective cohort study. Setting: This study utilized the Pediatric Health Information System (PHIS) database, which consists of clinical and resource-utilization data from >49 hospitals in the United States. Patients and Participants: Patients undergoing primary cleft palate repair from 2004 to 2009 were abstracted from the PHIS database and followed up for oronasal fistula repair between 2004 and 2015. Main Outcome Measure(s): The primary outcome measure was whether patients underwent oronasal fistula repair after primary cleft palate repair. Results: Among 5745 patients from 45 institutions whom met inclusion criteria, 166 (3%) underwent oronasal fistula repair within 6 to 11 years of primary cleft palate repair. Primary palatoplasty at high-RCC facilities was associated with a higher rate of subsequent oronasal fistula repair (odds ratio [OR] = 1.84 [1.32-2.56], adjusted odds ratio [AOR] = 1.81 [1.28-2.59]; P ≤ .001). Likelihood of surgery for oronasal fistula was independent of hospital case volume (OR = 0.83 [0.61-1.13], P = .233; AOR = 0.86 [0.62-1.20], P = .386). Patients with complete unilateral or bilateral cleft palate were more likely to receive oronasal fistula closure compared to those with unilateral-incomplete cleft palate (AOR = 2.09 [1.27-3.56], P = .005; AOR = 3.14 [1.80-5.58], P < .001). Conclusions: Subsequent need for oronasal fistula repair, while independent of hospital case volume for cleft palate repair, increased with increasing hospital RCC. Our study also corroborates complete cleft palate and cleft lip as risk factors for oronasal fistula.