Chen T, Chen J, Sheng Q, Zhu L, Lv Z. Pyriform Sinus Fistula in the Fetus and Neonate: A Systematic Review of Published Cases.
Front Pediatr 2020;
8:502. [PMID:
32984214 PMCID:
PMC7477109 DOI:
10.3389/fped.2020.00502]
[Citation(s) in RCA: 1] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 04/23/2020] [Accepted: 07/16/2020] [Indexed: 12/02/2022] Open
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.
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