Odontogenic Cutaneous Fistula: A Zombie Diagnosis.

BACKGROUND: Odontogenic cutaneous fistula (OCF) is a pathologic communication between the cutaneous surface of the face and oral cavity. Majority of oral cutaneous fistulas are reported to arise from chronic dental infection. Delay in treatment may result in chronic tissue injury as well as cosmetic deformities. Lesions are often misdiagnosed, leading to delayed management. Misdiagnosis may be the result of the variability of clinical morphological presentation and location of lesions compounded by the lack of knowledge that these lesions can have dental etiology. It is estimated that half of patients with OCF undergo multiple dermatologic surgical operations, antibiotic regimens, and other excisions and biopsies before a correct diagnosis is made. Herein, we present a systematic review to detail cases of odontogenic cutaneous lesions that had been previously misdiagnosed or managed inappropriately. In addition, we include a case report from our own institution. The aims are to demonstrate various presentations of OCF, increase awareness of plastic surgeons and oral maxillofacial surgeons to this pathological condition, and reinforce the importance of prompt diagnosis and treatment. METHODS: The study was conducted following the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) statement. A literature search was conducted in PubMed, Web of Science, and Cochrane databases from January 1, 2013 to July 24, 2023. The following search terms were utilized: "odontogenic cutaneous fistula" OR "odontogenic cutaneous sinus." RESULTS: Twenty-three articles published between 2013 and 2023 were included in this review. In addition, we report a case from our own institution. A total of 28 cases were included in the review. CONCLUSIONS: Diagnosis of OCF is challenging for numerous reasons. Misdiagnosis can lead to multiple trials of antibiotics and surgical procedures as well as reoccurrence of the lesion. Cases summarized highlight the importance of communication between oral maxillofacial surgery, plastic surgery, other medical subspecialists, and dentists in the evaluation of patients with head and neck lesions. Physicians should consider dental etiology in the differential diagnosis of orofacial skin lesions, even if teeth appear normal on oral examination.

Ovarian tumour-cutaneous fistula as a primary presentation of pelvic malakoplakia coexisting with a benign ovarian tumour mimicking advanced ovarian cancer.

Malakoplakia is a rare granulomatous, chronic inflammatory disease generally affecting the urogenital organs, though it can arise in other organs. The clinical manifestations of malakoplakia vary depending on the affected organ. The final diagnosis is confirmed by the presence of Michaelis-Gutmann bodies on pathology. This report describes a case of pelvic malakoplakia accompanied by an ovarian tumour-cutaneous fistula, initially misdiagnosed as advanced ovarian cancer invading the anterior abdominal wall with left pleural effusion based on imaging studies and increased serum carbohydrate antigen 19-9. The patient underwent left thoracentesis and fluid collection from the fistula tract for cytology, which showed no malignancy. She underwent primary debulking surgery, including removal of the fistula tract from anterior abdominal wall. Histopathological examination revealed malakoplakia coexisting with mucinous cystadenoma of the left ovary. For postoperative management, she received prolonged oral antibiotics for 6 months. There was no evidence of disease recurrence at the 24-month follow-up.

High-Resolution Ultrasound of Odontogenic Cutaneous Sinus Tract: An International Multicentric Experience and a Review of the Literature.

OBJECTIVES: To investigate the role of high-resolution ultrasound (HR-US) in the initial and differential diagnosis of the Odontogenic Cutaneous Sinus Tract (OCST) in a multicentric setting. METHODS: Skin HR-US examinations of OCSTs performed between January 2019 and June 2023 at different Institutions were retrospectively reviewed. Epidemiological and clinical data (age, gender, location of the skin lesion, causative tooth, and the clinical suspicion) as well as HR-US imaging findings (morphology and length of the sinus tract, Doppler signal, and cortical bone interruption of maxilla or mandible) were collected. US examinations were performed by expert radiologists using a high-performance US scanner, employing a high-frequency linear probe (15 MHz or higher frequencies). In only one patient the HR-US exam was integrated with strain elastography (SE). RESULTS: Sixteen patients were enrolled with a median age of 37.6 years (range 16-70 years). The most frequent clinical suspicion was epidermal cyst, while OCST was suspected in only two cases. In all cases, HR-US depicted the sinus tract as a nodular, triangular or "champignon-shaped" lesion in the subcutaneous layer, which continued with a slightly tortuous band structure, up to the focally interrupted cortical bone plate. Furthermore, color Doppler evaluation showed color signals around and/or within the lesion, expression of inflammation. On SE, the sinus tract showed a hard pattern, due to fibrous and granulomatous tissue. CONCLUSIONS: HR-US, thanks to its high spatial resolution, allows the evaluation of OCST, and play a crucial diagnostic role, mainly when the clinical suspicious is different.

Phase II Clinical Trial of Intravenous Levothyroxine to Mitigate Pharyngocutaneous Fistula in Euthyroid Patients Undergoing Salvage Laryngectomy.

PURPOSE: Patients undergoing head and neck cancer surgery after prior radiation or chemoradiation are at high risk for wound complications. Hypothyroidism is a known risk factor for wound complications, especially fistulae after salvage total laryngectomy. The purpose of this phase II clinical trial is to investigate the effect of perioperative intravenous levothyroxine supplementation on wound complications in patients undergoing salvage total laryngectomy. PATIENTS AND METHODS: Euthyroid patients previously treated with radiation/chemoradiation undergoing total laryngectomy were prospectively recruited (n = 72). Postoperatively, intravenous levothyroxine was administered at a weight-based dose (1.3 mcg/kg/d) and transitioned to enteral dosing on day 7. Free T3, T4, and thyroid-stimulating hormones were collected, and dosing was adjusted accordingly. The primary endpoints were rates of fistula formation and fistula requiring reoperation, compared with matched historic controls. All patients were monitored for adverse effects. RESULTS: The rate of postoperative hypothyroidism was 21% compared with 49% in a matched historic cohort. The rate of fistula formation was 18.1%, whereas the rate of fistula requiring reoperation was 4.2%, significantly lower than rates in our historic cohort (34.6% and 14.8%, respectively; P = 0.02 and 0.01). Postoperative hypothyroidism and recurrent clinical stage predicted fistula requiring reoperation in multivariate analysis; other acute phase reactants were not predictive. There were no observed adverse events related to levothyroxine supplementation. CONCLUSIONS: Postoperative intravenous levothyroxine supplementation reduced rates of acute hypothyroidism, fistula formation, and fistula requiring reoperation in patients undergoing salvage total laryngectomy without adverse effects. Intravenous levothyroxine is a viable strategy to reduce wound complications in this high-risk patient population.

Stapler versus conventional pharyngeal repair after total laryngectomy: a randomized clinical trial.

OBJECTIVES: The aim of the current study was to evaluate the functional outcomes of stapler pharyngeal closure after total laryngectomy by the incidence of PCT and assessment of swallowing after surgery. In addition, the study aimed to evaluate the oncological outcomes in terms of patients' survival rates. METHODS: This randomized clinical trial was conducted on 58 patients with advanced laryngeal carcinoma who underwent total laryngectomy. Patients were randomly assigned to two groups according to the method of pharyngeal repair after laryngectomy: manual closure group (n = 28), and stapler group (n = 30). Functional and oncological outcomes were assessed and compared. RESULTS: The incidence of pharyngocutaneous fistula was significantly less in the stapler group. Additionally, operative time was significantly shorter and swallowing function was better in the stapler group compared to the manual group. There was no statistically significant difference between groups regarding survival rates. CONCLUSION: The stapler is a reliable method for pharyngeal closure after total laryngectomy if the limits of its indications regarding the primary tumor are considered. Stapler closure decreases the incidence of PCF and decreases the surgical time. Good swallowing outcomes are achieved without compromising the oncological outcomes.

A 16-Year-Old Girl with Sinonasal Cutaneous Fistula Following Excision and Radiotherapy for Rhabdomyosarcoma Requiring Reconstructive Surgery Using an Expanded Forehead Flap.

BACKGROUND Sinonasal rhabdomyosarcoma (RMS) is a rare malignancy in children and adolescents. It is aggressive and locally invasive, and can require local postoperative radiotherapy. This report presents the case of a 16-year-old girl with a sinonasal-cutaneous fistula following excision and radiotherapy for rhabdomyosarcoma, which required reconstructive surgery using an expanded forehead flap. CASE REPORT We report the case of a16-year-old girl who was referred to our clinic with sinonasal-cutaneous fistula. Prior to presentation at our department, she presented with bilateral intermittent nasal congestion 3 years ago. At a local hospital, orbital computed tomography and nasal endoscopic biopsy revealed an embryonal rhabdomyosarcoma (ERMS). One month later, skull base tumor resection, nasal cavity and sinus tumor resection, and low-temperature plasma ablation were performed at a local hospital. Two weeks after the operation, the patient received intensity-modulated radiation therapy for a total of 50 Gy. Chemotherapy started 15 days after radiotherapy, using a vincristine, dactinomycin, and cyclophosphamide (VAC) regimen. Approximately 1 month later, an ulcer appeared at the nasal root and the lesion gradually expanded. The patient was referred to our hospital due to the defect. Firstly, a tissue expander was implanted at the forehead for 7 months. Then, the skin around the defect was trimmed and forehead flap was separated to repair the lining and external skin. The flap survived well 1-year after the operation. CONCLUSIONS This report highlights the challenges of post-radiation reconstructive surgery and describes how an expanded forehead flap can achieve an acceptable cosmetic outcome in a patient with a sinonasal-cutaneous fistula.

Fistulising skin metastases in Crohn's disease: a case report and review of the literature.

BACKGROUND: Metastatic Crohn's disease is a rare disorder characterized by various granulomatous skin lesions that occur independently of gastrointestinal tract involvement. However, currently there is no standardized care or specific treatment. Therapeutic approaches include immunosuppressive agents, such as corticosteroids, azathioprine, and monoclonal antibodies targeting inflammatory cytokines like tumor necrosis factor (TNF). CASE PRESENTATION: We present a case of a 29-year-old western European woman with significant blind ending abdominal subcutaneous fistulas and abscesses, who sought evaluation in the dermatology department. Histological examination revealed multiple epithelioid cell granulomas. There was no evidence of infectious or rheumatologic diseases such as sarcoidosis. The tentative diagnosis was metastatic Crohn's disease, which was not related to an intestinal manifestation of the disease. The patient responded to infliximab but had to discontinue it due to an allergic reaction. Subsequent adalimumab treatment failed to induce clinical remission; thus, therapy was switched to ustekinumab, resulting in a positive response. Written informed consent for publication of their clinical details and clinical images was obtained from the patient. For our study more than 1600 publications were screened for cases of metastatic Crohn's disease on PubMed database. 59 case reports with 171 patients were included in the analysis and evaluated for localization, diagnostic and therapeutic approaches, and complications and were summarized in this review. CONCLUSION: The successful ustekinumab treatment of a patient with metastatic Crohn's disease underscores the potential of this minimally investigated therapeutic option, highlighting the need for future treatment guidelines given the increasing prevalence of such cases.

Infrahyoid muscle flap as augmentation of pharyngeal repair after total laryngectomy.

INTRODUCTION: To assess the usage of contralateral infrahyoid muscles as onlay flap in prevention of pharyngo-cutaneous fistula (PCF) in total laryngectomy (TL) patients with high risk factors of PCF development. METHODS: This study included 10 patients who suffered from advanced laryngeal carcinoma with presence of risk factors for development of PCF. We added an enforcement muscular layer during neo-pharynx wall repair, the infrahyoid muscle flap of the contralateral side of the tumour origin as an onlay muscular flap to cover the anastomotic site for healing augmentation. Patients were followed up for PCF development where the PCF incidence was calculated. RESULTS: One patient died from congestive heart failure in the 21th post-operative day so, the actual PCF rate in TL patients with high risk factors of PCF development in our study was 11.11% (1 out of 9 cases). CONCLUSIONS: Infrahyoid muscle flap may have a role in preventing PCF after TL in patients with high risk factors of PCF development in this case series study to be further assessed in other studies to justify its role.

Reconstruction for Salvage Laryngectomy With Limited Pharyngectomy.

Importance: Closure technique for optimization of postoperative and functional outcomes following salvage laryngectomy remains an area of debate among head and neck surgeons. Objective: To investigate the association of salvage laryngectomy closure technique with early postoperative and functional outcomes. Design, Setting, and Participants: This retrospective cohort study included patients from 17 academic, tertiary care centers who underwent total laryngectomy with no or limited pharyngectomy after completing a course of definitive radiotherapy or chemoradiotherapy with curative intent between January 2011 and December 2016. Patients with defects not amenable to primary closure were excluded. Data were analyzed from February 14, 2021, to January 29, 2024. Exposures: Total laryngectomy with and without limited pharyngectomy, reconstructed by primary mucosal closure (PC), regional closure (RC), or free tissue transfer (FTT). Main Outcomes and Measures: Patients were stratified on the basis of the pharyngeal closure technique. Perioperative and long-term functional outcomes were evaluated with bivariate analyses. A multivariable regression model adjusted for historical risk factors for pharyngocutaneous fistula (PCF) was used to assess risk associated with closure technique. Relative risks (RRs) with 95% CIs were determined. Results: The study included 309 patients (256 [82.8%] male; mean age, 64.7 [range, 58.0-72.0] years). Defects were reconstructed as follows: FTT (161 patients [52.1%]), RC (64 [20.7%]), and PC (84 [27.2%]). A PCF was noted in 36 of 161 patients in the FTT group (22.4%), 25 of 64 in the RC group (39.1%), and 29 of 84 in the PC group (34.5%). On multivariable analysis, patients undergoing PC or RC had a higher risk of PCF compared with those undergoing FTT (PC: RR, 2.2 [95% CI, 1.1-4.4]; RC: RR, 2.5 [95% CI, 1.3-4.8]). Undergoing FTT was associated with a clinically meaningful reduction in risk of PCF (RR, 0.6; 95% CI, 0.4-0.9; number needed to treat, 7). Subgroup analysis comparing inset techniques for the RC group showed a higher risk of PCF associated with PC (RR, 1.8; 95% CI, 1.1-3.0) and predominately pectoralis myofascial flap with onlay technique (RR, 1.9; 95% CI, 1.2-3.2), but there was no association of pectoralis myocutaneous flap with cutaneous paddle interposition with PCF (RR, 1.2; 95% CI, 0.5-2.8) compared with FTT with cutaneous inset. There were no clinically significant differences in functional outcomes between the groups. Conclusion and Relevance: In this study of patients with limited pharyngeal defects, interpositional fasciocutaneous closure technique was associated with reduced risk of PCF in the salvage setting, which is most commonly achieved by FTT in academic practices. Closure technique was not associated with functional outcomes at 1 and 2 years postoperatively.

Vesicocutaneous fistula at site of closed suprapubic tube tract as the first presenting sign of giant bladder stone.

Bladder stones represent approximately 5% of all cases of urolithiasis and are typically identified and managed long before causing irreversible renal injury. We present a case of a man in his 40s with a prior history of a gunshot wound to the abdomen who presented with leakage from a previously healed suprapubic tube tract and was found to have a giant bladder stone with a resulting renal injury. He subsequently underwent a combined open cystolithotomy and vesicocutaneous fistulotomy during his hospitalisation, which helped to improve his renal function. In addition to there being few reported cases of bladder stones >10 cm, this represents the first report in the literature of an associated decompressive 'pop-off' mechanism through a fistulised tract.

Mapping the risk factors, pathogens, and antibiotic of pharyngocutaneous fistula in patients after neck open surgery.

PURPOSE: Current literature lacks consensus on risk factors for pharyngocutaneous fistula (PCF), and empirical antibiotic guidelines for PCF are limited. The aim of this study was to reduce the incidence of PCF and improve antibiotic treatment efficacy for patients with PCF after open neck surgery by analyzing their clinical characteristics, pathogenic bacteria, and antibiotic susceptibility. METHODS: This study was a 13-year single-center retrospective cohort study, including 699 patients who underwent open neck surgery for laryngeal and hypopharyngeal cancer. Univariate and multivariate logistic regression analyses were conducted to identify the risk factors associated with the occurrence of PCF after surgery. The microbial species causing PCF were analyzed, and the antibiotic sensitivity of the top three pathogens was assessed. Venn diagrams were used to illustrate the antibiotics that exhibited 100% sensitivity against all three identified pathogens. RESULTS: The incidence of PCF after open neck surgery was 8%. Logistic univariate and multivariate analyses revealed that flap reconstruction (OR = 3.62, 95% CI [2.02-6.52]), history of preoperative radiotherapy (OR = 2.01, 95% CI [1.31-2.73]), significant postoperative bleeding (OR = 1.79, 95% CI [1.11-2.69]), and history of diabetes (OR = 1.34, 95% CI [1.29-2.46]) were significantly associated with PCF occurrence. Among the 38 cases of PCF patients, the top three identified pathogens were Pseudomonas aeruginosa, Escherichia coli, and Enterobacter cloacae. The antibiotics cefepime, meropenem, ticarcillin/clavulanic acid, and cefoperazone/sulbactam showed 100% sensitivity against these top three pathogens. CONCLUSION: Special attention should be given to patients undergoing open neck surgery, especially those with intraoperative flap reconstruction, a history of preoperative radiotherapy, postoperative bleeding, or diabetes. Strengthening monitoring and care is crucial in preventing the occurrence of PCF. According to antibiotic usage guidelines and considering the distribution of pathogens in PCF patients, empirical antibiotic treatment with cefoperazone/sulbactam or ticarcillin/clavulanic acid is recommended prior to obtaining susceptibility test results.

Risk factors and healing factors for pharyngocutaneous fistula after total laryngectomy for laryngeal cancer: An epidemiological study.

To analyse the risk factors and healing factors of pharyngocutaneous fistula (PCF) in patients with laryngeal cancer after total laryngectomy, and to explore the relevant epidemiology. A retrospective analysis was conducted on laryngeal cancer patients who underwent total laryngectomy in our hospital from January 2010 to December 2022. The 349 patients included in the study were divided into a PCF group of 79 and a non-PCF group of 270. Perform one-way analysis of variance and multivariate logistic analysis on various data of patients included in the statistics, and analyse the risk factors and healing factors of PCF. Smoking, history of radiation therapy for laryngeal cancer, history of chemotherapy for laryngeal cancer, tumour location (larynx, pharynx, oesophagus), preoperative albumin, postoperative proteinaemia, <99 haemoglobin, postoperative haemoglobin, postoperative C-reactive protein (CRP) level are the risk factors for PCF. Also, radiation therapy and postoperative proteinaemia were the main reasons for preventing PCF healing. Smoking history, laryngeal cancer, radiation therapy, albumin, haemoglobin and CRP are risk factors for postoperative PCF after total laryngectomy, while radiation therapy and postoperative hypoalbuminaemia are key factors affecting PCF healing.

Impact of stapler versus manual closure techniques on pharyngocutaneous fistula development post-total laryngectomy: A systematic review and meta-analysis.

Pharyngocutaneous fistula (PCF) is a major complication after total laryngectomy, with significant morbidity and mortality. Whether mechanical stapler closure of the pharynx reduces fistula rates compared to hand-sewn techniques remains unclear. We conducted an updated systematic review and meta-analysis to clarify this question. Five databases were systematically searched from inception through November 2023 for studies comparing stapler versus suture closure for fistula outcomes after laryngectomy. Odds ratios (OR) were pooled using random-effects models and fixed-effects models. Subgroup and sensitivity analyses were performed. Risk of bias was appraised using NHLBI tools. Nine studies with 803 patients were included. Mechanical closure significantly reduced fistula incidence versus suture closure (OR = 0.57, 95% CI 0.34-0.95, p = 0.03). Subgroup analysis found that stapling's protective effect varied by patient age, country/region, linear stapler size and female percentage. Stapling reduced fistula odds by 80% in the Turkey subgroup (OR = 0.20, 95% CI 0.09-0.50) but showed no benefit in other regions. Patients <60 years showed an 84% fistula reduction with stapling (OR = 0.17, 95% CI 0.06-0.45), whereas older subgroups did not. Linear stapler size of 60 mm significantly reduced fistula occurrence while 75 mm did not. There was no evidence that low female percentage mitigated stapling benefits. Mechanical stapler closure after total laryngectomy meaningfully reduces the likelihood of postoperative PCF formation compared to hand-sewn closure, especially for patients younger than 60 years old. The absolute risk reduction supports its utility to prevent this complication.

Horizontal pharyngeal closure during total laryngectomy reduces rates of pharyngocutaneous fistula.

PURPOSE: Pharyngocutaneous fistula (PCF) is a possible complication following total laryngectomy (TL), with a mean incidence of 17%. We intended to investigate the effect of pharyngeal closure type during TL on the prevention of PCF. METHODS: We retrospectively reviewed patients that underwent TL with a horizontal pharyngeal closure over a 10-year period. The frequency of PCF clinically, dysphagia, total oral diet, postoperative dilatation of the neopharynx and voice problems were tabulated. RESULTS: Seventy-seven subjects underwent TL due to laryngeal tumor without pharyngeal extension. Of them, 45 underwent a salvage TL. PCF occurred in 1/77 subjects. The rest of the subjects (76/77) did not develop a PCF, neither in the early nor in the late postoperative phase. All subjects (15/77) that underwent implantation of a voice prosthesis were satisfied with their voice. No subject complained about dysphagia. Every subject achieved total oral diet. CONCLUSION: The horizontal pharyngeal closure is a safe pharyngeal closure technique during TL, reduces PCF rates (< 2%), results in excellent voice rehabilitation and swallowing function, and can also be used during salvage TL instead of a major pectoral flap. This type of closure should be used only in selected patients with laryngeal disease without pharyngeal extension.

Modified PATIO technique for urethrocutaneous fistula after hypospadias repair: Experience from a tertiary referral hospital.

INTRODUCTION: Urethrocutaneous fistula (UCF) is a common complication after hypospadias repair with an incidence of 5-10%. Several techniques are described for its repair: small UCFs are frequently corrected by isolation, excision, and closure with apposition of a protective second layer. In 2008 Malone described the PATIO technique: the fistula tract is turned inside out in the urethral lumen preventing contact with passing urine without direct urethral sutures. OBJECTIVE: Aim of our study is to present our outcomes using a modified version of the PATIO technique, with a more reproducible isolation of the tract and without its fixation at the urethral meatus. STUDY DESIGN: We retrospectively reviewed all cases of UCFs corrected with a modified PATIO technique at our center between 2016 and 2020. Data collected from electronical clinical notes were age at UCF closure, location of UCF, presence of meatal stenosis and clinical outcomes. Data are presented as median and IQR. RESULTS: In the study period we performed 425 urethroplasties for distal and mid penile hypospadias. The incidence of UCFs was 7% (30/425) and 25 patients underwent UCF correction with modified PATIO. Median age at repair was 4.5 years (IQR: 2.5-6.2). At a median follow-up of 3 years (IQR: 2-4) recurrence was observed in 5 cases out of 24 with one patient who was lost at follow-up (20.8%). One case was corrected successfully with re-do modified PATIO technique, while 4 are awaiting repair. One cases was lost at follow-up. UFC-recurrence was homogeneously distributed along the study period. DISCUSSION: Risk factors for UCF recurrence are mostly the type of hypospadias, neo-urethral length, and quality of the urethral plate. Among the many existing techniques, we propose a modified version of Malone's PATIO repair. We believe that the use of four stay-suture to isolate the fistula allows a well-defined dissection of the tract along its surface, compared to the use of a single stay-suture. In our experience, there is no need to keep and fix the traction on the fistula tract to the urethral meatus, probably reflecting the efficacy of the fistula closure during the introflection, which is then maintained without traction. Limitations to our study include the retrospective nature of the review, the small sample size of the cohort and the absence of control groups. CONCLUSIONS: Our results appear consistent with literature regarding the efficacy of PATIO principles in treating UCF. Modified PATIO seem to be particularly reproducible, showing encouraging results.

Utero-cutaneous fistula following caesarean section.

A multiparous woman in her 20s, presented to the clinic with bleeding from caesarean scar site. After clinical examination and radiological evaluation with ultrasound and MR fistulogram, the diagnosis was utero-cutaneous fistula. Complete resection of fistulous tract with uterine defect closure and supportive omental patch placement was done after an intraoperative demonstration of the utero-cutaneous fistula by injecting methylene blue dye. Utero-cutaneous fistula is a rare complication after caesarean section. Cyclical bleeding from an abnormal opening in previous caesarean scar site is the classical presentation of this condition.