Pyriform Sinus Fistula in the Fetus and Neonate: A Systematic Review of Published Cases

Abstract

Purpose: To provide recommendations for the diagnosis and treatment of pyriform sinus fistula (PSF) in the fetus and neonate through a systematic review of the published literature. Methods: PubMed and Embase (1968-2019) were searched, and additional publications were obtained by searching the references by hand. The two reviewers assessed all papers and extracted the following variables: demographics, clinical features, diagnostic tests, interventions, and prognoses. Results: Forty-two papers were included, comprising a total of 158 cases. PSF presented almost exclusively on the left side (95.56%). Patients usually presented with a neck mass (100%) and respiratory distress (43.18%). The false-negative rate (FNR) of prenatal ultrasonography (US) was significantly higher than that of prenatal magnetic resonance imaging (MRI) (P < 0.01). For the diagnosis of PSF in neonates, computerized tomography (CT) and MRI were the most accurate diagnostic modalities. Ex utero intrapartum treatment (EXIT) was performed during delivery in 6 patients (26.09%). Among 135 patients with a reported date of definitive surgery, 117 (86.67%) underwent surgery during the neonatal period. Complications after definitive surgery appeared in 5 patients (3.16%), and all of them recovered spontaneously within 3 months. Furthermore, recurrence occurred in 4 patients (2.53%). Conclusion: In fetal cases with PSF suspected by US, MRI is necessary to confirm the diagnosis. During the neonatal period, patients with PSF typically present with a neck mass and respiratory distress, and CT/MRI appears to be the preferred diagnostic method. Definitive surgery is effective for treating neonatal PSF, with a low complication rate and low recurrence rate.

Hoarseness as the first symptom in a patient with acute suppurative thyroiditis secondary to a pyriform sinus fistula: A case report

“…The initial symptoms of PSF include neck mass with or without abscess, acute thyroiditis, and thyroid lesion [1][2][3]. In some cases, particularly in neonates, a neck mass compressing the surrounding structure, resulting in dyspnea, has been reported [2,4]. However, hoarseness, as the rst symptom of PSF, has not yet been reported.…”

“…Dyspnea may appear secondary to lesion compression due to a neck mass [2,4]. However, hoarseness has not been reported as the rst symptom of a PSF.…”

Multimodal imaging of congenital pyriform fossa fistula in children

“…Most pediatric patients suffer from long-term repeated infection or multiple abscess incisions and drainage because of delay in diagnosis. Because of the limitations of the scanning area, the non-specific presentation, and the less-experienced operator, the accuracy of ultrasound (US) is lower than that of CT and MRI ( 3 , 4 ). The study summarized the imaging manifestations of CT, barium esophagography, and MRI of pediatric CPSF in different periods, so as to provide the reference for clinical diagnosis and treatment of pediatric CPSF as soon as possible.…”

A safe and effective surgical method for complex pyriform sinus fistula

“…The typical presentations of lesions in pyriform sinus fistula are recurrent low‐neck abscesses, acute suppurative thyroiditis, and neck masses 14–16 . Approximately, 90% of these sinus tracts are situated on the left side of the neck, which may be due to embryonic development 17–19 . An internal fistula is usually located at the tip or base of the pyriform fossa 20,21 .…”

“… 14 , 15 , 16 Approximately, 90% of these sinus tracts are situated on the left side of the neck, which may be due to embryonic development. 17 , 18 , 19 An internal fistula is usually located at the tip or base of the pyriform fossa. 20 , 21 Generally, the pyriform sinus fistula passes through the larynx near the cricothyroid joint and then partially passes through or terminates at the upper level of the lateral lobe of the thyroid.…”