Neonatal respiratory distress due to a third or fourth branchial pouch sinus: A systematic review.

OBJECTIVES: Third and fourth branchial pouch sinuses can be rare causes of respiratory distress in neonates. An overview of this distinct clinical entity is missing in literature. To aid clinicians in recognizing and adequately treating this unique entity, we conducted a systematic review to discuss patient characteristics, diagnostic considerations and treatment strategy. METHODS: MEDLINE and EMBASE were searched from inception to December 29th, 2020. Original studies concerning patients with respiratory symptoms as a result of a third or fourth branchial pouch sinus, as confirmed with rigid endoscopy, videofluoroscopy or during surgery. RESULTS: Thirty-nine studies describing 56 patients (66% male, aged 0-30 days) were analyzed. Symptoms included cervical mass (76.8%), stridor (55.4%), dyspnea (35.7%) and cyanosis (17.9%) due to a third (39.3%) or fourth (60.7%) branchial pouch sinus. Intubation was performed before treatment in 31.3%. The piriform sinus opening was identified with rigid endoscopy in 81.1%. Surgery was the treatment of choice in the majority of patients (85.7%), with a success rate of 100% and a complication rate of 10.7%. Endoscopic cauterization was successful in 40% and endoscopic cauterization followed by sclerotherapy was successful 100%, with no complications. CONCLUSION: Third or fourth branchial pouch sinuses can lead to respiratory distress in neonates. It is important to recognize this distinct clinical picture for adequate diagnosis and treatment. Rigid endoscopy is indicated to demonstrate an opening in the piriform sinus and provides the opportunity to directly perform treatment with endoscopic cauterization. If this is insufficient to relief respiratory symptoms due to a persistent cyst, sclerotherapy or surgical excision should be considered.

MRI in Children With Pyriform Sinus Fistula.

BACKGROUND: A variety of imaging modalities have been described for the diagnosis of congenital pyriform sinus fistula (CPSF). To date, there have been few MRI reports. PURPOSE: To evaluate MRI findings of CPSF and interobserver reliability. STUDY TYPE: Retrospective. POPULATION: In all, 115 patients aged 23 days to 15.4 years at operation. FIELD STRENGTH/SEQUENCE: 3.0T/axial T1 -weighted image (T1 WI)-SPIR, axial T2 WI, axial T2 WI-STIR, coronal T2 WI-SPIR, diffusion-weighted imaging (DWI), axial and coronal gadolinium-enhanced T1 WI-SPIR. ASSESSMENT: For each patient, the medical records, including demographics, clinical manifestations, and MRI findings were reviewed. All the MRI studies were interpreted by three radiologists independently. STATISTICAL TEST: Kendall's W test was made to determine the interobserver reliability of three reviewers for MRI findings. RESULTS: CPSF occurred on the left side in 104 (90.4%) patients and on the right side in 11 (9.6%) patients. The male-to-female ratio was 59:56. The age at first episode varied from birth to 12.3 years. There was one neonate patient, who presented with a unilocular cystic mass in the left neck. A tunnel-like lesion between the pyriform fossa and the upper pole of the thyroid gland, T2 high signal behind the cricothyroid joint, thyroid gland involvement, deep neck abscesses or masses were noted in 46 (40%), 93 (80.9%), 96 (83.5%), and 36 (31.3%) patients, respectively. There was excellent interobserver reliability for all the MRI findings, ranging from 0.84 to 1.00. DATA CONCLUSION: The sinus tract presenting with a tunnel-like lesion goes behind the cricothyroid joint in most cases. For patients presenting with acute suppurative thyroiditis (AST) or neck infection with thyroid gland involvement, the presence of T2 high signal behind the cricothyroid joint highly suggests the diagnosis of CPSF. MRI is a reliable method for the diagnosis of CPSF. LEVEL OF EVIDENCE: 4 TECHNICAL EFFICACY STAGE: 2.

Fourth branchial anomalies: Predictive factors of therapeutic success.

PURPOSE: The purpose of this work was to determine the epidemiology and the predictive factors of success of the surgical management of fourth branchial anomalies. METHODS: This is a multicentric retrospective review from 1998 to 2016 of patients who presented with an endoscopically-confirmed fourth branchial pouch anomaly. Data were analyzed according to sex, age, clinical features, number of recurrences, treatment modalities (endoscopic and/or cervicotomy), post-operative complications and follow-up. RESULTS: Fifty-two children have been included. The average age at diagnosis was 4.5 years. Among them, 73.1% were female, 11.4% were neonatal forms; 94.2% of lesions were left-sided; 75% of patients presented a cervical abscess as first symptom, and 7.7% of children presented with dyspnea. Average time between first symptoms and management was 9.5 months. Management was endoscopic in 73.1% of patients (laser in 84.2%, coagulation in 15.8%) with about a third of recurrence after one procedure. Overall success of endoscopic procedures reached 84.2%. A cervical open surgery was performed in 26.9% as first line treatment. Overall success of cervicotomy reached 85.7%. No complications of endoscopic surgery have been identified. There were 35.7% complications of cervicotomy (2 recurrent nerve palsy, 2 keloid scars, 1 pharyngostoma). An association was proved between recurrences and initial abscess (OR = 2.44), and with age between 3 and 5 (OR = 4). CONCLUSION: Endoscopic treatments appear to be effective in first line approach in the management of fourth branchial anomalies, offering an excellent efficiency with rare complications. We identified two risk factors of recurrence: age between 3 and 5 years old and history of cervical abscesses. LEVEL OF EVIDENCE: IV.

KTP laser assisted endoscopic tissue fibrin glue biocauterization for congenital pyriform sinus fistula in children.

OBJECTIVE: This study aims to assess the efficacy of a novel endoscopic management for congenital pyriform sinus fistula (CPSF) using potassium titanyl phosphate (KTP) laser assisted endoscopic tissue fibrin glue biocauterization in children. METHOD: From 2010 to 2014, a total of 5 children with recurrent or acute suppurative thyroiditis or neck abscess secondary to CPSF were enrolled retrospectively in this study. RESULTS: Mean age at the first time of endoscopic biocauterization was 6.2 ± 0.7 (5-7) years. The barium swallow study detected a fistula in four cases. Endoscopy identified an internal opening at the pyriform sinus in all cases with four on the left side and one on the right side. All patients underwent KTP laser assisted endoscopic tissue fibrin glue biocauterization as treatment for CPSF. Only one case required the second endoscopic procedure due to fluctuation of symptoms. Post-endoscopic follow-up duration of these patients was 24.6 ± 11.6 (7-36) months. Neither complications nor recurrences were noted during follow-up in all patients. CONCLUSIONS: For children presenting with repeated acute suppurative thyroiditis or neck infections, clinicians should highly suspect the possibility of CPSF. Endoscopy should be performed not only to confirm the diagnosis but also could be served as an initial treatment modality of biocauterization by KTP laser and tissue fibrin glue, which was demonstrated as a less invasive, safe, and effective method in children.

Surgery versus endoscopic cauterization in patients with third or fourth branchial pouch sinuses: A systematic review.

OBJECTIVES: To systematically review the current literature on treatment of third and fourth branchial pouch sinuses with endoscopic cauterization, including chemocauterization and electrocauterization, in comparison to surgical treatment. DATA SOURCES: PubMed, Embase, and the Cochrane Library. REVIEW METHODS: We conducted a systematic search. Studies reporting original study data were included. After assessing the directness of evidence and risk of bias, studies with a low directness of evidence or a high risk of bias were excluded from analysis. Cumulative success rates after initial and recurrent treatments were calculated for both methods. A meta-analysis was conducted comparing the success rate of electrocauterization and surgery. RESULTS: A total of 2,263 articles were retrieved, of which seven retrospective and one prospective article were eligible for analysis. The cumulative success rate after primary treatment with cauterization ranged from 66.7% to 100%, and ranged from 77.8% to 100% after a second cauterization. The cumulative success rate after the first surgical treatment ranged from 50% to 100% and was 100% after the second surgical attempt. Meta-analysis on electrocauterization showed a nonsignificant risk ratio of 1.35 (95% confidence interval: 0.78-2.33). CONCLUSIONS: The effectiveness of cauterization in preventing recurrence seems to be comparable to surgical treatment. However, we suggest endoscopic cauterization as the treatment of choice for third and fourth branchial pouch sinuses because of the lower morbidity rate.