Endoscopic Suture With Chemocauterization: An Effective Treatment of Congenital Pyriform Sinus Fistula.

OBJECTIVE: Endoscopic cauterization is an effective method for treating pyriform sinus fistula (PSF). However, these approaches sometimes result in a higher failure rate. We present an effective technique utilizing suture combined with chemocauterization as first-line treatment in patients with PSF and evaluate the safety and efficacy of its use in 126 patients. STUDY DESIGN: Retrospective study. SETTING: Tertiary referral center. METHODS: Retrospective case review of patients treated between March 2012 and June 2021 at our institution with descriptive statistical analysis. RESULTS: A total of 126 patients with PSF were included in this study with a mean age of 14.7 years. There was no sex predilection. The majority of patients presented with a left-sided neck lesion (89.7%). Ten patients presented following prior attempts at the surgery of the PSF at another institution; 8 via open surgery and 2 following endoscopic CO2 laser cauterization; other patients only had a history of repeat incision and drainage or antibiotic treatment. The success rate of obliteration of the internal opening was 96.83% after a single treatment without complications. Following reoperation, a successful outcome was achieved in the remaining 4 patients. Length of stay ranged from 10 to 14 days. No recurrences occurred within 12 to 120 months followed-up. CONCLUSION: Endoscopic suture combined with chemocauterization is a safe and effective treatment of PSF. Surgery can be performed during the acute cervical inflammatory period without increased risk of complication or recurrence, however, patients found to have acute changes affecting the pyriform sinus should be treated with a staged surgery strategy.

Prenatal diagnosis of pyriform sinus fistula using ultrasonography by detecting the communication to the pharynx.

OBJECTIVE: Pyriform sinus fistula (PSF) is a congenital anomaly which originates from the pharyngeal pouch. PSF is initially recognized as a cyst around the fetal neck, but accurate prenatal diagnosis of the disease is challenging. We aimed to report the key findings and tips in accurately distinguishing PSF from other differential diagnosis by which enables detection of the communication of the nuchal cyst and the pharynx. CASE REPORT: We report a case in which we were able to diagnose PSF as early as 18 weeks of gestation with ultrasonography. We used epiglottis as a landmark, and detected an unilobular cyst arising from the pharynx. CONCLUSION: Ultrasonography is a powerful tool in prenatal diagnosis of PSF especially at early stage of pregnancy. By detecting the epiglottis, it can locate the communication of the nuchal cyst and the pharynx, and thereby enables an accurate diagnosis of PSF.

Piriform fossa sinus tract - a 15-year retrospective review with a focus on atypical neonatal presentations.

BACKGROUND: Third and fourth branchial anomalies are rare, accounting for less than 10% of all branchial anomalies. The piriform fossa sinus tract (PFST) typically presents with left-side suppurative thyroiditis, although it can present earlier in neonates as a non-inflamed cystic neck mass. PFST poses a considerable diagnostic challenge with variable clinical and imaging features, leading to long delays to definitive diagnosis and appropriate management. OBJECTIVE: To analyse the patterns of presentation and imaging findings in children with PFST, with a particular focus on neonatal presentation. MATERIALS AND METHODS: This was a retrospective review of the clinical presentation, imaging findings and management in 16 cases of PFST presenting to our tertiary children's hospital between 2003 and 2018. Cases were identified by medical records and picture archiving and communication system (PACS) search using relevant International Classification of Diseases (ICD)-10 coding. RESULTS: Age at presentation ranged from prenatal to 16 years, with a male-to-female ratio of 2:1. All patients presented with neck swelling. Thirteen patients (81%) had suppurative thyroiditis at initial presentation. Two patients had severe thyroiditis/mediastinitis that required intensive care unit admission. Three neonates presented with noninfected, asymptomatic large cystic neck masses; two of these were detected prenatally and misdiagnosed as lymphatic malformations with subsequent spontaneous clinical resolution that later represented with evidence of PFST. The PFST was on the left side in 15/16 (94%) patients. All patients had neck imaging before definitive diagnosis. Imaging studies included radiographs, ultrasound, computed tomography, magnetic resonance imaging and barium esophagram studies. No single modality was diagnostic of PFST in all patients. Seventy-five percent of patients had multimodal imaging before diagnosis. All PFSTs were confirmed by endoscopic visualisation. Management of PFST was by endoscopic cauterisation in 13 patients and open surgery in 2. One patient did not require surgical correction. CONCLUSION: Our study highlights the complex nature of PFST. The anomaly is uncommon, has variable clinical and imaging features and may have a lengthy, complicated course if not considered at initial presentation. An episode of suppurative thyroiditis in a child should prompt investigation for PFST. We describe atypical presentations with cystic masses in neonates that appear to resolve but represent later as typical clinical features of PFST.

Endoscopic coblation treatment for congenital pyriform sinus fistula in children.

ABSTRACT: Congenital pyriform sinus fistula (CPSF) is a very rare branchial apparatus malformation. Traditional open surgery for fistulectomy might fail to excise the lesion completely, leading to continual recurrence. Herein, we report our experience of endoscopic coblation technique for treatment of CPSF in children.To observe the clinical efficacy of endoscopic coblation treatment of CPSF in children, especially for those in acute infection stage.Retrospective case series with 54 patients (including 20 cases in acute infection stage and 34 cases in non infection stage) who were diagnosed with CPSF between October 2017 to November 2019, all patients were treated with endoscopic coblation to close the piriform fossa fistula, neck abscess incision and drainage performed simultaneously for acute infection stage cases. Data collected including age of diagnosis, presenting symptoms, diagnostic methods, prior and subsequent treatments, length of hospitalization, and recurrence were analyzed.Of the 20 cases in acute infection stage, there were 3 children with transient vocal cord paresis all of which resolved with 1 month. Four children of the 34 cases in non infection stage appeared reddish swelling of the neck on the 4th, 5th, 6th, and 7th days after coblation and then underwent abscess incision and drainage. All cases experienced no recurrence, vocal cord paralysis, pharyngeal fistula and massive hemorrhage after their first endoscopic coblation of the sinus tract in the follow up of 3 to 28 months.Endoscopic coblation is an effective and safe approach for children with CPSF, neck abscess incision and drainage could be performed simultaneously in acute infection stage. We advocate using this minimally invasive technique as first line of treatment for CPSF.

Absceso retrofaríngeo por fístula congénita del seno piriforme en una adolescente / Retropharyngeal abscess due to a congenital pyriform sinus fistula in a teenager

Arch Argent Pediatr 2020;118(1):e81-e84 / e81Presentación de casos clínicosRESUMENLas fístulas del seno piriforme son anomalías poco frecuentes de los arcos branquiales. La forma de presentación más común en los niños y los adolescentes es la tiroiditis aguda supurada y/o los abscesos cervicales laterales recurrentes. Sin embargo, las fístulas se pueden manifestar de forma atípica. La rareza de esta patología y la presentación clínica atípica pueden demorar el diagnóstico, lo que aumenta el riesgo de infecciones recurrentes y complicaciones.Se presenta el caso inusual de una adolescente de 13 años con absceso retrofaríngeo debido a una fístula congénita del seno piriforme, tratada de forma exitosa mediante electrocauterización endoscópica.

Pyriform sinus fistulas are rare anomalies of the branchial arches. The most common form of presentation in children and adolescents is acute suppurative thyroiditis and/or recurrent lateral cervical abscesses. However, fistulas can manifest atypically. The rarity of this pathology and the atypical clinical presentation can delay the diagnosis increasing the risk of recurrent infections and complications.We present the unusual case of a 13-year-old teenager with retropharyngeal abscess due to a congenital pyriform sinus fistula successfully treated by endoscopic electrocautery

Congenital pyriform sinus fistula presenting as a neck abscess in a newborn: A case report.

RATIONALE: Congenital pyriform sinus fistula (CPSF) is a branchial abnormality originating from the third or fourth branchial pouch and is an important cause of anterior cervical abscess in children. Here we present a case of neck abscess in a newborn that was diagnosed as CPSF. PATIENT CONCERNS: A male infant with a birth weight of 3660 g was admitted to hospital 25 minutes after birth after discovery of a cystic mass with extensive skin swelling in the left side of the neck. B-mode ultrasonography of the left neck showed an anterior cervical cystic mass of indeterminate nature. DIAGNOSIS: Congenital pyriform sinus fistula. INTERVENTIONS: The neck abscess was incised and drained under general anesthesia. Examination under suspension laryngoscopy revealed a pyriform sinus fistula. Laser cauterization was performed simultaneously. The wound was dressed and anti-inflammatory treatment was provided. OUTCOMES: The neck wound healed uneventfully. After 3 months, the fistula was confirmed to be closed by laryngoscopy under general anesthesia. No recurrence was detected during 9 months of follow-up. LESSONS: CPSF should be strongly suspected in a patient with an unexplained neck abscess or recurrent acute suppurative thyroiditis, especially on the left side.

Spontaneous closure of branchial sinus of the pyriform fossa.

Management of third and fourth branchial cleft anomalies are similar. These anomalies should be suspected in a child with recurrent low-anterior neck abscess. Investigations in the form of cross-sectional studies and examination of the pharynx under anaesthesia will facilitate diagnosis and resolution of abscess. Spontaneous closure of the pyriform sinus can occur following conservative management with antibiotic treatment and abscess drainage. This emphasise the role of second-look prior to implementing endoscopic cauterisation or surgery.

Endoscopic Cauterization with Pneumatic Distension for Piriform Fossa Sinus Tracts.

INTRODUCTION: Piriform fossa sinus tracts (PFSTs) are a cause of recurrent neck infections in the pediatric population. Conventional management required open resection, but over the last years minimally invasive approaches have been reported in an attempt to endoscopically obliterate the PFST, using different methods such as electrocautery, laser, trichloroacetic acid, or silver nitrate. MATERIALS AND METHODS: We undertook a retrospective review of the medical records of 12 children (aged 4 months to 14 years) with PFSTs treated with endoscopic sclerosis with diathermy (ESD) between 2010 and 2016 at a tertiary care children's hospital. We also present a technical modification of ESD, using continuous infusion of airflow through the gastroscopy, to distend the piriform sinus and facilitate its recognition. PFST obliteration was performed using diathermy through a guide wire. RESULTS: Clinical presentation of the 12 affected children included neck tumor (7 [58%]), neck abscesses (4 [33%]), and thyroiditis (5 [41%]). All lesions occurred on the left side. All patients underwent both ultrasonography and barium esophagography (the latter being positive only in 50%). Two patients were treated with ESD after the open approach had failed. There was no procedure-related morbidity. One patient had a recurrence (positive barium swallow without symptoms). The success rate of this procedure in our series was 91% with one attempt and 100% with two attempts. CONCLUSION: In our experience, treatment of PFST with ESD is a reproducible, noninvasive, and an effective option. ESD could be considered a primary approach and also for revision after open surgery has failed in these patients.

Bilateral Piriform sinus fistulas: a case study and review of management options.

BACKGROUND: Piriform sinus fistulas occur due to developmental abnormalities of the third and fourth branchial arches, and almost always occur unilaterally. They generally present as recurrent abscesses in the anterior-inferior neck, with concurrent thyroiditis. They have conventionally been managed with complete removal of the sinus tract, and thyroidectomy if required; however, endoscopic approaches have been increasingly favored. Herein we describe a case of bilateral piriform sinus fistulas, and present a review of the literature concerning their endoscopic management. CASE PRESENTATION: Our patient was determined to have bilateral piriform sinus fistulas based on computer tomography, magnetic resonance imaging and microlaryngoscopy. We performed electrocauterization of the proximal fistula tracts, followed by injection of fibrin sealent. Our patient has not had a recurrence in the ten months since his procedure. There were no complications. Twenty-three articles describing an endoscopic approach to these fistulas were identified through PubMed, and a search through the references of related articles was completed. CONCLUSION: Of one hundred and ninety-five patient cases we reviewed, an endoscopic procedure success rate of 82% and complication rate of 5.6% was determined. Piriform sinus fistulas that occur bilaterally are a rare congenital abnormality of the neck. Endoscopic approaches are an acceptable alternative option to open procedures, with similar success and a lower rate of complications.

Evaluation of endoscopic coblation treatment for obliteration of congenital pyriform sinus fistula.

BACKGROUND: Congenital pyriform sinus fistula is a very rare branchial apparatus malformation. Conventional open surgery for fistulectomy might fail to excise the lesion completely, leading to continual recurrence. The aim of this study was to provide an innovative endoscopic coblation technique for patients with pyriform sinus fistula and evaluate its intermediate-term effectiveness. METHODS: Retrospective case series with 112 patients (age range 3-36 years) between 2013 and 2016 and underwent endoscopic coblation of the sinus fistula. Data collected including patient demographics, presenting symptoms, diagnostic methods, prior and subsequent treatments, length of hospital stay, and recurrence were analyzed. RESULTS: Of the 112 cases, there were no postoperative complications observed except temporary reddish swelling in three patients. Four cases were lost to follow-up. Of 108 patients, 106 experienced no recurrence after their first endoscopic coblation of the sinus tract. The remaining two patients with recurrence in the follow-up were ultimately treated with recoblation without complications or further recurrences. Also, Endoscopic coblation can be used to treat seven patients with recurrence who had experienced open resection. In our series, median follow-up period was 1.5 years. CONCLUSION: Endoscopic coblation is an effective approach for most patients. We advocate using this minimally invasive technique as first line of treatment for pyriform sinus fistula.

Estenosis de la apertura piriforme y síndrome de incisivo central único: casos clínicos / Piriform aperture stenosis and solitary median maxillary central incisor: clinical cases

El síndrome incisivo central único es un trastorno que involucra anomalías de la línea media. Se puede presentar con dificultad respiratoria poniendo en peligro la vida del recién nacido, debido a malformaciones nasales. Estas malformaciones incluyen atresia de coanas y la estenosis de la apertura del orificio piriforme. Debe pensarse en esta última en todo recién nacido con estridor y dificultad respiratoria de grado variable, asociado a la dificultad de pasar una sonda a través de la región anterior de las fosas nasales. El diagnóstico se confirma por tomografía computarizada del macizo craneofacial y el examen nasofibroscópico. La conducta terapéutica dependerá de la gravedad de los síntomas, pero en general si es que no hay compromiso respiratorio severo, se prefiere una conducta expectante, y esperar el crecimiento craneofacial del niño, para aumentar permeabilidad nasal. Presentamos dos casos clínicos.

Solitary median maxillary central incisor syndrome is a rare disorder involving midline abnormalities. It may present with life threatening respiratory distress in the neonate secondary to nasal malformations. These include choanal atresia and pyriform aperture stenosis. The last to be thought in any newborn with stridor and respiratory distress associated with the difficulty of passing a tube through the anterior region of the nostrils. The diagnosis is confirmed by craniofacial CT scan and nasolaryngoscope evaluation. The therapeutic approach depends on the severity of symptoms.

Can we make an accurate diagnosis of pyriform sinus fistula by fetal MRI?

AIM: To review the fetal magnetic resonance imaging (MRI) features of pyriform sinus fistula (PSF) and to compare them with the postnatal clinical and surgical findings. The relevant medical literature is also discussed. MATERIALS AND METHODS: The location, shape, signal, effects of adjacent structures and in utero changes detected on fetal MRI were reviewed in three cases of PSF. The patient's respiratory status at birth, the application of ex utero intrapartum therapy (EXIT), infectious complications in the neonatal period, and the surgical and pathological findings were also reviewed and discussed. The study consisted of three pregnant women between 18 and 38 years of age, each with a single fetus. Fetal MRI was performed at 26 and 38 weeks of gestation in case 1, at 34 weeks of gestation in case 2, and at 34 and 38 weeks of gestation in case 3. Postnatal clinical follow-up extended from 9 months to 3 years. RESULTS: All lesions were well-circumscribed unilobular cysts with slightly thickened walls at the neck area. Case 1 was irregular in shape, and the other two cases were oval and extended longitudinally along the neck. The fluid contents showed signals consistent with amniotic fluid. Close contact with the thyroid gland was demonstrated to be a fairly characteristic feature that could be recognised on fetal MRI. The in utero follow-up detected slight changes in the signal, wall characteristics, contour, and airway deviation. Case 1 was bilaterally affected, causing it to be initially mistaken as macro-cystic lymphangioma. The diagnosis was corrected with the aid of postnatal imaging; however, a prenatal diagnosis was correctly made in cases 2 and 3. Airway obstruction was not observed in any of the three cases, and thus, EXIT was not applied. The fetal MRI findings were in good agreement with the postnatal clinical and surgical findings. In addition to the higher spatial delineation provided by fetal MRI, as described in the literature, the observation of close contact with the thyroid gland and a flattened deformity of the thyroid gland are other characteristic features. CONCLUSION: Fetal MRI is useful for displaying characteristics of PSF that can enable an accurate prenatal diagnosis.

The natural course of congenital nasal pyriform aperture stenosis.

OBJECTIVES/HYPOTHESIS: Patients with congenital nasal pyriform aperture stenosis (CNPAS) may become less symptomatic with age. Therefore, we aimed to develop a growth curve of the pyriform aperture so that a more comprehensive plan can be designed for CNPAS patients who show little response to conservative treatment. STUDY DESIGN: A single-institution study, retrospective review of CNPAS patients during the period November 1997 to December 2014. METHODS: We measured the distances between the bilateral nasal processes of the maxilla (interprocess distance [IPD]) on three-dimensional computed tomography images and then divided the patients into five different age groups. A growth curve of the pyriform aperture was then constructed based on the distance-age relationship. RESULTS: Fifty-four IPD measurements were included. The mean IPD was 3.57 mm in neonates < 1 month old, 4.08 mm in infants aged 1 to 3 months, 5.19 mm in the 4-month to 11-month age group, 6.61 mm in the 12-month to 36-month age group, and 9.20 mm in children > 36 months of age. We found that the cubic curve was the most appropriate growth curve, and that growth tended to be slower from 3.5 years to 6 years of age. CONCLUSIONS: The growth curve of the pyriform aperture in children with CNPAS developed in this study can aid in treatment planning and predict clinical outcome of CNPAS patients. Although CNPAS patients may become less symptomatic with age, when the observed IPD falls progressively farther from the curve, more aggressive intervention should be considered, such as changing the management strategy from observation to conservative treatment or from conservative treatment to surgery. LEVEL OF EVIDENCE: 4 Laryngoscope, 126:2399-2402, 2016.

Recurrent neck lesions secondary to pyriform sinus fistula.

Recurrent neck lesions associated with third or fourth branchial arch fistula are much less common than those of second arch and usually present with acute suppurative thyroiditis or neck abscess. Our aim is to describe clinical features, management and treatment outcomes of 64 cases of congenital pyriform sinus fistula (PSF). Medical record of these 64 patients (33 males, 31 females) treated at the First Affiliated Hospital of Zhengzhou University from 2011 to 2014 were reviewed. The patients comprised 33 males and 31 females, and their ages ranged from 18 months to 47 years (median 10 years, mean 12.7 years). Neck abscess and recurrent infection was the mode of presentation in 37 cases (57.8 %), 4 patients (6.3 %) presented with acute suppurative thyroiditis, neck mass was the mode of presentation in 17 cases (26.6 %), 2 patients (3.1 %) presented with neck mass with respiratory distress, and cutaneous discharging fistula was the mode of presentation in 1 cases (1.6 %). The remaining 3 patients (4.7 %) presented with cutaneous discharging fistula with neck infection. Investigations performed include barium swallow, CT scan, and ultrasound which were useful in delineating PSF tract preoperatively. Barium swallow was taken as the gold standard for diagnosis. Our patients were treated by fistulectomy with hemithyroidectomy, fistulectomy, fistulectomy with endoscopic electric cauterization, endoscopic electric cauterization or endoscopic coblation cauterization, respectively. Histopathologic examination of the surgical specimens revealed that they were lined with ciliated epithelium, stratified cuboid epithelium with chronic inflammatory cell infiltration and fibrosis. Voice hoarseness occurred after operation in seven patients, but disappeared 1 week later. PSF recurred in 6 patients, 4 of them were cured by a successful re-excision. One patient was cured by successful endoscopic electric cauterization. The other 1 has remained asymptomatic for 5 months. In our series, mean follow-up period was 13.3 months and median follow-up period was 12.5 months (range 2-40 months). Presence of congenital PSF should be suspected when intra-thyroidal abscess formation occurs as the gland is resistant to infection. Strong clinical suspicion, barium swallow study, CT scan and ultrasound are the key to diagnosis. Both fistulectomy with hemithyroidectomy and endoscopic treatment have comparable success rate. Endoscopic coblation cauterization may prove a useful and equally effective method of treatment for PSF in future.

Systematic Review of Endoscopic Obliteration Techniques for Managing Congenital Piriform Fossa Sinus Tracts in Children.

BACKGROUND: Piriform fossa sinus tracts (PFSTs) are a recognized cause of recurrent deep neck infections in the pediatric population. Conventional management has historically required open resection, but over recent years minimally invasive endoscopic approaches to obliterate the pharyngeal opening of the sinus have been performed in many centers. However, there is a lack of clear evidence regarding the success rate and safety of these approaches. OBJECTIVE: To determine the success rate of endoscopic management of PFST through a systematic review of the existing literature. DATA SOURCES: MEDLINE (1964-2014) and bibliographies of identified papers. REVIEW METHODS: Two authors independently reviewed 170 abstracts and identified relevant studies for full-text review. Data were independently extracted from those studies, and the Oxford Centre for Evidence-Based Medicine guidelines were used to classify the level of evidence. RESULTS: Thirteen studies met the inclusion criteria, comprising a total of 84 patients. All included studies were evidence level 4 (case series). Various methods of obliterating the PFST were described: electrocautery (n = 39), laser (n = 19), trichloroacetic acid (n = 19), silver nitrate (n = 4), combination of silver nitrate and laser (n = 2), and fibrin glue (n = 1). The success rate for endoscopic management of PFST was 89.3% overall (90.5% in primary cases and 85.7% in revision cases). The only adverse event reported was temporary vocal cord immobility in 2.4% (n = 2) of cases. CONCLUSION: Endoscopic management of pediatric PFST appears to be safe and effective, as a primary option and for revision after open surgery.

A Review Over the Past 15 Years of the Management of the Internal Piriform Apex Sinus Tract of a Branchial Pouch Anomaly and Case Description.

OBJECTIVES: Literature review of treating the piriform apex sinus tract through microlaryngoscopy and a case description. REVIEW METHODS: Fourteen papers were identified in PubMed using the search criteria of piriform sinus fistula, microlaryngoscopic repair, and endoscopy. Institutional Review Board approval was obtained. RESULTS: One hundred forty-five cases including ours were available for review, with 182 procedures. Sixty-two cases were male, 73 female, and 10 genders were not reported. Multiple treatment options were used, including electrocautery, chemocautery, mass excision, fibrin glue, lasers, suture closure, or combination of stated modalities. Of the 182 procedures, 147 procedures were performed endoscopically. There were 37 recurrences (25%). These patients either underwent a repeat endoscopic procedure or an open excision. One hundred and ten (75%) endoscopic procedures were successful. CONCLUSIONS: Piriform sinus tract anomalies often present as a mass and recurrent neck infections. This review reveals that treating the internal piriform sinus opening alone can be successful. This procedure has low morbidity, short operative time, and high success. We advocate this approach first with a combined open/laryngoscopic approach for failed cases. To our knowledge, our technique of CO2laser ablation of the tract followed by suture closure has not been previously described. We believe this to be the first comprehensive review of this topic and the largest series of cases included in a single report.