Features Associated With High Risk of Recurrence in Saccular Laryngeal Cysts: A Systematic Review and Qualitative Synthesis

Abstract

Introduction: Saccular cysts are rare benign anomalies of the larynx (1.5% of laryngeal anomalies) determined by dilatation of the laryngeal saccule. They can be congenital or acquired. Clinical manifestation depends on the site of the cyst and the age of the patient. Children frequently have stridor and dyspnea, while adults usually complain of dysphonia.The therapeutic approach can be surgical or nonsurgical (needle aspiration or drainage).However, guidelines on the management of this disease are lacking in the literature.This systematic literature review was conducted through an interferential analysis with the aim of detecting the association between clinical, diagnostic, and operative factors and the recurrence of saccular cysts.

Materials and methods: This systematic literature review was evolved following the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) 2020 checklist statement Papers published up to August 2023 containing relevant clinical data were searched in Embase/PubMed, Web of Science, Scopus, and Cochrane database. Full texts lacking information on the patient's data, clinical, radiological, and/or histological diagnosis of saccular cysts of the larynx were excluded. Articles with follow-up inferior to 6 months and not reporting any treatment of the cyst were excluded, too. Data concerning signs and symptoms, cyst characteristics, and the type of treatment were described, and the risk of recurrence was studied in association with these features. A qualitative synthesis of the results was performed.

Results: Ninety-eight articles were screened. One hundred two patients were involved in the analysis. The articles with low or intermediate risk of bias were 22 (8 case reports and 14 case series) and they were included in the statistical analysis. Median age was 25 years (range 0-81). Saccular cysts were congenital (46/102, 45%) of cases acquired (56/102, 55%). Relying on the available data, all patients were symptomatic, with stridor as the main symptom (59%). Surgical approach was performed in 85% of cases, while drainage or needle aspiration of the cyst was the treatment of choice in the remaining 25% of cases. Twenty-four percent of patients had recurrence of the disease within 3 months. Median age at recurrence was 9 years. Cysts characteristics, preoperative management, and type of treatment were associated with an increased risk of disease recurrence (P < 0.05).

Conclusions: Saccular cysts are rare congenital or acquired laryngeal diseases. Data regarding their management and factors associated with the risk of recurrence are still limited. Congenital cysts treated conservatively have a higher risk of recurrence than acquired cysts treated by open surgical techniques or by microlaryngoscopy. A better knowledge of factors associated with a higher risk of recurrence could help choose the best treatment.

Keywords: Laryngeal; Laryngeal cysts; Saccular cysts; Voice.