How to make an accurate diagnosis of fetal pyriform sinus fistula in utero: experience at a single medical center in mainland China

Liu, Zequn; Han, Jin Hee; Fu, Fang; Liu, H.S; He, Qiuming; Zhong, Wei; Wang, Hongying; Yang, Xin; Li, Dong-Zhi; Liao, Can

doi:10.1016/j.ejogrb.2018.05.039

Cited by 8 publications

(10 citation statements)

References 28 publications

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“…However, the diagnosis of fetal PSF remains a great challenge for radiologists due to the small cyst size. This review revealed 23 cases with a prenatal diagnosis of PSF (6,7,9,24,32,54). PSF-related neck abnormality is often identified in the second or third trimester.…”

Section: Discussionmentioning

confidence: 99%

“…Inconsistent results found by the two reviewers were further assessed and discussed to reach a consensus. Figure 1 illustrates the detailed flowchart of the search strategy, and 42 studies were included in this review (6,(9)(10)(11).…”

Section: Registration and Search Strategymentioning

confidence: 99%

“…In contrast, neck masses and respiratory problems are the main presentations in neonates with PSF, and tracheal compression may lead to apnea. Therefore, the diagnosis of PSF in the fetus and neonate has been widely acknowledged as a growing concern (6)(7)(8)(9)(10). In addition to the clinical presentations, the preferred imaging test between children and neonates is also quite different (11).…”

Section: Introductionmentioning

confidence: 99%

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

Chen

Sheng

et al. 2020

Front. Pediatr.

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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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Section: Discussionmentioning

confidence: 99%

Section: Registration and Search Strategymentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

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

Chen

Sheng

et al. 2020

Front. Pediatr.

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“…Recently, prenatal diagnosis of pyriform sinus fistula has been made based on the findings of ultrasonography and magnetic resonance imaging (MRI). And EXIT procedure was applied in some cases 5,6,14 . PSF should be suspected in newborns with a cervical mass and in children with recurrent neck infection, especially on the left side.…”

Section: Discussionmentioning

confidence: 99%

Differences in the diagnosis and management of pyriform sinus fistula between newborns and children

Sheng

et al. 2019

Sci Rep

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Pyriform sinus fistula (PSF) is a rare congenital entity. We hypothesized that clinical features, diagnosis and treatment may differ between newborns and children. 190 patients diagnosed with PSF were divided into two groups: neonatal (n = 15) and childhood (n = 175). The medical records including demographic and clinical data were retrospectively analyzed. There were 102 boys and 88 girls in this study. Most patients presented as a left-sided neck lesion. A neck mass, with or without infection and respiratory distress, was the common finding in newborn patients. Prenatal diagnosis was made in three cases. However, in childhood group, initial symptoms were neck abscess (78.8%), acute thyroiditis (11.4%), neck mass (6.9%), and thyroid lesion (2.8%). The presence of a cervical mass with air pocket showed on CT image was thought to be the pathognomonic finding of neonatal PSF. The diagnosis was usually established by barium esophagography in older children. Delayed accurate diagnosis was detected in both groups. The median time from onset to diagnosis was 22 months and 1 year respectively. Endoscopic-assisted open surgery was performed successfully in all patients, with good outcomes in majority cases of both groups (93.3% and 95.4%). Recurrence was developed in 5 patients. PSF should be suspected in newborns with cervical mass and in children with recurrent neck infection, especially on the left side. Early diagnosis and treatment might avoid repeated surgical procedures. Complete resection of the cyst and fistula in non-infected state is essential for good outcomes.

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“…The preliminary diagnosis of CSPF by preoperative imaging examinations is important. Insufficient understanding of the clinical and imaging manifestations may easily lead to misdiagnosis and delay the subsequent treatment (8)(9)(10). The clinical symptoms and imaging findings of CPSF in neonates differ from those in children or adults (11).…”

Section: Introductionmentioning

confidence: 99%

Imaging Findings in Neonates With Congenital Pyriform Sinus Fistula: A Retrospective Study of 45 Cases

Zhao

Yao

et al. 2021

Front. Pediatr.

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Background: Congenital pyriform sinus fistula (CPSF) is a rare branchial cleft deformity. The characteristics and management of CPSF in neonates are different from those in children or adults, and a comprehensive understanding of the imaging features of neonatal CPSF can facilitate its preoperative diagnosis. Thus, the aim of this study was to summarize the ultrasonography (US) and CT imaging findings of CPSF in neonates.Methods: Forty-five full-term neonates with CPSF, confirmed by pathology after surgical resection from January 2012 to October 2020, were included in this retrospective study. All patients underwent preoperative cervical US and contrast-enhanced CT examinations, and the imaging findings were analyzed.Results: Forty-six cervical cystic masses were found in 45 neonates, including one case with bilateral lesions, three cases with lesions on the right side, and 41 cases on the left side. Both US and CT detected neck abnormality among all cases, while the diagnostic accuracy of US (15/46, 32.6%) was lower than that of CT (42/46, 91.3%). Moreover, CT showed significantly higher detection rates of intralesional air bubbles, involvement of the ipsilateral thyroid, deviation of the airway, and expansion into the mediastinal and retropharyngeal space compared with the US. As the age increased, it was more likely to present some features including the absence of air-containing, thick cyst wall, and poorly defined border (ρ <0.05).Conclusion: CPSF in the neonates showed distinctive imaging findings on contrast-enhanced CT scan, which provides important supplementary information for the diagnosis of CPSF after the initial US examination.

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How to make an accurate diagnosis of fetal pyriform sinus fistula in utero: experience at a single medical center in mainland China

Cited by 8 publications

References 28 publications

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

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

Differences in the diagnosis and management of pyriform sinus fistula between newborns and children

Imaging Findings in Neonates With Congenital Pyriform Sinus Fistula: A Retrospective Study of 45 Cases

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