Neisseria flavescens Infection in Atypical Multiple Vallecular Cysts

Abstract

A 53 year-old female patient presented with symptoms of intermittent odynophagia and halitosis persisting for 2 months. Physical examination revealed bilateral lingual tonsillar cysts and multiple vallecular cysts with clear fluid. Laboratory studies were unremarkable. The patient underwent surgery, during which uncapping of the multiple vallecular cysts was performed, and multiple microbiological samples and biopsies were taken. After surgery, the patient had complete resolution of all her symptoms. Pathological results demonstrated lymphoepithelial cysts. Microbiological tests demonstrated an infection by Neisseria flavescens, which is a non-pathogenic commensal of the oropharynx, and has never been described as causing agent of infected vallecular cysts.

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Introduction

Multiple vallecular cysts composed of lymphoepithelial tissue are uncommon, and have rarely been described in the literature. These are rare benign cysts, assumed to be caused by inflammation, irritation, or trauma, leading to obstruction and dilatation of mucous glands or lingual tonsillar crypts [2]. They occur most often in the vallecular region and lingual aspect of the epiglottis. Neisseria flavescens is non-pathogenic gram negative diplococcic, commensal of the oropharynx, which has rarely been associated with infectious processes, and has not been described as a causative agent of infected vallecular cysts.

Rare Case Report

A 53 year-old female patient, with controlled hypercholesterolemia and hypothyroidism, presented with symptoms of intermittent odynophagia and halitosis persisting for 2 months. Physical examination revealed bilateral lingual tonsillar cysts, and multiple vallecular cysts (~8) containing clear fluid (Fig. 1). Due to the patient’s persisting complaints, the patient was operated. During surgery, multiple biopsies from the lingual tonsillar cysts were obtained, and uncapping of the all vallecular cysts was performed, and fluid drained from these cysts was sent to bacteriology (Fig. 2). Post-operatively, the patient was treated with intravenous amoxicillin/clavulanate and steroids, and was discharged without further complications. At the post-operative visit, the patient was satisfied, stating complete resolution of symptoms. Pathological results demonstrated cystic lesions surrounded by reactive lymphoid tissue, compatible with lymphoepithelial cysts (Fig. 3). Microbiological tests were equivocal and suspicious for N. cinerea. The samples were sent for bacterial PCR, which demonstrated an infection by N. flavescens.

Fig. 1.

Multiple vallecular cysts demonstrated during fiber-optic laryngoscopy in office. Epiglottis (white arrow)

Fig. 2.

Uncapping of multiple vallecular cysts

Fig. 3.

Histological demonstration of vallecular cyst. Demonstrating intact vallecular mucosa (dashed black arrow) covering dense lymphoid tissue, with two germinal centers (white arrow). The cystic cavity, covered by non-keratinizing stratified squamous epithelium (black arrow). (Hematoxylin & Eosin ×20)

Discussion

Multiple infected vallecular cysts have rarely been described in the literature. Their incidence, etiology and the causative organisms implicated are usually unknown. Vallecular cysts, also known as epiglottic mucus retention cysts, are classified as ductal, retention, or lymphoepithelial cysts [1]. They are assumed to be caused by inflammation, irritation, or trauma, leading to obstruction and dilatation of mucous gland or lingual tonsillar crypt [1]. Lymphoepithelial cysts are lined by squamous epithelium with underlying lymphoid follicles. They occur almost exclusively in the vallecula, epiglottis, and pyriform fossa [6]. These cysts are commonly diagnosed in children with symptoms of upper airway obstruction. In adults, an association has been established between infected vallecular cysts and severe supraglottic infection including epiglottitis [2], leading to increased risk for upper airway obstruction [10]. This patient did not present with upper airway obstruction, but complained of odynophagia and halitosis, which could be explained by the infected cysts. The recommended treatment for vallecular cyst is transoral surgical removal. Surgical methods include simple drainage, uncapping, or marsupialization. Instrumentation includes forceps, cautery, CO₂ laser, and even Da Vinci robotic surgical system [7]. The patient’s microbiological results demonstrated infection by Neisseria, and it was only after PCR results that the exact type of infection was identified. N. flavescens is a gram negative diplococcic, generally considered as a non-pathogenic commensal of the oropharynx, which is rarely associated with infectious processes. Most of N. flavescens infected patients have severe underlying diseases, for example, immunodeficiency and diabetes [4]. In the literature, there are only a few references regarding pathogenic N. flavescens. It is described as causing infective endocarditis [8], septicemia with meningitis [3, 9], necrotizing pneumonia and empyema [5], but no reference has been found describing these bacteria as causing vallecular cysts. To our knowledge, this is the first report of N. flavescens in the upper airway, in an otherwise healthy patient.

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Conflicts of interest

None.