Abstract
BackgroundSialadenoma papilliferum (SP) is an extremely rare benign neoplasm of salivary glands. To explore and define the clinicopathological features of SP, we retrospectively analyzed 89 cases previously reported and five new cases.MethodsThe clinical features, histopathology, immunohistochemistry and molecular analysis of our cases were further performed and the related literatures were reviewed and analyzed.ResultsCombining 89 cases from the literature with our cases, the hard palate was the most common locations for SP. However, two of our cases were rarely located in the esophageal mucosa. Among all cases, the male gender was more affected, with the average age and median age of 61.8 and 62 years, respectively. Conventional histomorphologically, SP was characterized by complex papillary structures with a biphasic growth pattern of exophytic squamous component and endophytic glandular component. The glandular structures were lined by a double layer of epithelium composed of flattened or cuboidal basal cells and a cuboidal or columnar luminal cells formed papillary infoldings into the ductal lumina. Immunohistochemically, the luminal epithelial configurations showed strong expression of CK7 along the luminal cell membrane, while the basal myoepithelia displayed strong nuclear p63 expression. In both the glandular and squamous tumour components showed BRAF V600E-positive immunostaining and BRAF V600E mutation.ConclusionFor the first time, we have comprehensively aggregated and analyzed 90 cases sialadenoma papilliferum from almost all previous publications, and further explored the clinicopathological features of SP; concordantly, this study demonstrated that SP shows a papillomatous growth pattern with exophytic and endophytic proliferation of ductal epithelium composed of double-layered cells harboring BRAF V600E mutation. Additionly, adequate treatment for SP is surgical excision, with a favorable prognosis in patients.
Highlights
Sialadenoma papilliferum (SP) is an extremely rare benign neoplasm of salivary glands
Patients were followed for 6 to 22 months, and their prognosis was good without recurrence and worsening progression
Treatment and follow up Our patients were followed for 6 to 22 months, and their prognosis was good without recurrence and worsening
Summary
Sialadenoma papilliferum (SP) is an extremely rare benign neoplasm of salivary glands. Sialadenoma papilliferum (SP) is a rare benign neoplasm [1,2,3], estimated to account for less than 1% of all minor salivary gland tumours and 3–5% of head and neck tumours [2,3,4,5,6,7] It was described initially in 1969 by Abrams and Finck, because of its histomorphology closely resembling that of the syringocystadenoma papilliferum of cutaneous adnexal origin, and a total of 90 cases were reported by 2021 [2,3,4,5, 8]. Given the rareness of SP and difficulty in distinguishing it from other malignant tumours, the experience gained from the present cases and thorough analysis of medical literature may be useful for pathologists and clinicians on the correct diagnosis of this disease
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