Abstract
Objective
To describe an experience with laryngeal oncocytic cystadenomas and review the published literature regarding this uncommon diagnosis.
Methods and results
A clinical review of patients presenting with cystic laryngeal masses in an urban academic medical center between January and December 2013 was performed. Three patients, two female and one male, with a mean age of 68 years, were diagnosed with oncocytic cystadenomata of the larynx. Major presenting symptoms included dysphonia, globus, and ipsilateral otalgia. Endoscopic examinations revealed a cystic structure arising from varied subsites of the larynx: laryngeal ventricle, aryepiglottic fold, and pre-epiglottic space. Cross-sectional radiographic imaging was obtained in each case. The patients were treated with transoral (CO 2 ) laser microsurgery (TLM). In all three cases, pathological analysis revealed oncocytic cystadenoma with clear margins.
Conclusions
Oncocytic cystadenoma is a rare entity of the larynx predominantly affecting elderly patients. Clinical presentation and imaging may suggest the diagnosis of an internal laryngocele. Complete excision is both diagnostic and therapeutic, and typically can be achieved using TLM. When clear margins are obtained, no adjuvant therapy is indicated. Although laryngoceles and malignancies are more commonly encountered, oncocytic cystadenomas should remain in the differential of cystic laryngeal masses.
1
Introduction
Cystic lesions of the larynx may be classified as neoplastic or nonneoplastic. Neoplastic cysts classically include cystadenomas, congenital cysts, traumatic cysts, vascular malformations, and hemangiomas. Nonneoplastic cysts include glandular cysts, amygdaloid cysts, and lymphatic cysts . Cystadenomas – also termed papillary cystadenomas, oncocytic adenomas, or, most commonly, oncocytic cystadenomas – are rare neoplastic lesions whose epithelial components resemble that of Warthin’s tumors (papillary cystadenoma lymphomatosum) found in the major salivary glands . Laryngeal oncocytic cystadenomas are typically considered separately from solid adenomas of the larynx . Oncocytes are hypertrophic epithelial cells with excessive mitochondria that impart a distinctive eosinophilic cytoplasm. Although individual cases of cystic adenomas arising in the larynx have been reported as early as 1891 , the first case series, comprising four such cases, was reported in 1944 . The most common laryngeal site affected is the supraglottis, and lesions usually present in the seventh or eighth decades of life . They are occasionally diffuse or multifocal , but more commonly present as single lesions causing hoarseness . If large enough, they may cause upper airway obstruction . While many agree that the etiology of oncocytic cystadenomas is primarily neoplastic , delayed hypersensitivity has been cited as an inciting factor, similar to what has been suggested for Warthin’s tumors . Optimal treatment involves surgical removal, either endoscopic or open laryngeal surgery .
2
Methods
This study received exemption status from the university medical institutional review board. Surgical databases were searched retrospectively for diagnosis of laryngeal mass from January 1 to December 31, 2013. Inclusion criteria required a histopathologic diagnosis of oncocytic cystadenoma involving any portion of the larynx. Identified patients were then reviewed for clinical, radiographic, and pathologic data.
Following the clinical study, a systematic literature review was carried out. Medline/PubMed was utilized to search the published English language manuscripts related to this topic. Boolean search terms were utilized to include key words of “larynx,” “laryngeal,” “cystadenoma,” and “oncocytic.” The results of the literature search have been included in the descriptive portions of this report.
2
Methods
This study received exemption status from the university medical institutional review board. Surgical databases were searched retrospectively for diagnosis of laryngeal mass from January 1 to December 31, 2013. Inclusion criteria required a histopathologic diagnosis of oncocytic cystadenoma involving any portion of the larynx. Identified patients were then reviewed for clinical, radiographic, and pathologic data.
Following the clinical study, a systematic literature review was carried out. Medline/PubMed was utilized to search the published English language manuscripts related to this topic. Boolean search terms were utilized to include key words of “larynx,” “laryngeal,” “cystadenoma,” and “oncocytic.” The results of the literature search have been included in the descriptive portions of this report.
3
Results
Three patients met inclusion criteria of diagnosis of laryngeal oncocytic cystadenoma. Two females and one male, with an average age at presentation of 68 years (range, 65 to 70) were treated within the review time period. In patients #1 and #2, presenting symptoms included globus and dysphonia; in #3, the only presenting symptom was severe unilateral otalgia. Consequently, the mass of patient #3 was identified by his otologist. Patient #1 denied a history of smoking; the remaining two patients reported prior tobacco use of 25 pack-years and 50 pack-years, respectively.
Preoperative video laryngostroboscopy was performed in all three patients to evaluate the symptoms and laryngeal function. In patient #1, a cyst was noted in the posterior aspect of the right vallecula, representing a pre-epiglottic space mass. In patient #2, a cystic mass obstructing the anterior aspect of the right ventricle was noted. In patient #3, only a fullness of the left aryepiglottic fold was noted.
Cross-sectional imaging (CT or MRI) was performed in all patients. Characteristic imaging findings of oncocytic cystadenomas were seen uniformly and are described within Fig. 1 .
