Sclerosing Polycystic Adenosis of the Parotid Gland: A Systematic Review and Report of 2 New Cases

Abstract

We sought to evaluate clinical-epidemiologic aspects, pathologic characteristics, and treatment of sclerosing polycystic adenosis (SPA) of the parotid gland and to report 2 new cases. We conducted a systematic review following PRISMA (Preferred Reporting Items for Systematic Reviews and Meta-Analyses) guidelines for studies published in or before June 2016. The inclusion criteria were as follows: final diagnosis of SPA affecting the parotid gland and articles published in Spanish or English. The exclusion criteria were as follows: unclear diagnosis after pathologic analysis and patients affected by other conditions in the major salivary glands. We added 2 new cases. Our review ultimately included 21 articles and 45 cases. The mean patient age was 41years (range, 7-84years). The female-to-male ratio was 2:1. In all patients the tumor showed progressive growth. The evolution range was 7days to 11years. In 17.7% of cases, the tumor was associated with pain. The average tumor size was 30.3mm (range, 12-60mm). Fine needle aspiration biopsy (FNAB) was unable to diagnose 30 cases. Benign tumors comprised 58% of tumors (18), whereas malignancy was present in 25.8% (8). Thirty-five tumors were well circumscribed. There were 8 multinodular or lobed cases. The tumor was described as encapsulated in 1 case, partially encapsulated or pseudo-encapsulated in 16, and not encapsulated in 12. Five cases showed different degrees of degenerative changes, ranging from dysplasia to invasive carcinoma. All cases underwent surgical treatment. Superficial parotidectomy with preservation of the facial nerve was performed in 22 cases (61.1%), total parotidectomy in 8 (22.2%), and tumor enucleation in 6(16.6%). The mean follow-up period was 51.5months (range, 5-276months). Of documented cases, 74.2% had 2 or more years of follow-up. Recurrences occurred in 6 cases (16.6%). We consider SPA a benign tumor with progressive growth, which is occasionally painful. It occurs around age 40years and occurs more often in female patients. Fine needle aspiration biopsy does not provide an adequate preoperative diagnosis. Recurrences are infrequent. Follow-up should be customized according to the pathologic findings. Malignant transformation may occur. Superficial parotidectomy with facial nerve preservation is the treatment of choice.