Sclerosing polycystic adenosis of the submandibular gland: Two case reports

BACKGROUND Sclerosing polycystic adenosis(SPA)is a rare disease of salivary glands,similar to fibrocystic disease of the breast.It occurs over a wide age range and exhibits a slight female preference.Most SPA cases have occurred in the parotid gland.The exact nature of SPA is unclear,but its tumor nature has recently been proposed.Although SPA has a good prognosis after adequate surgery,atypical lesions might occur,ranging from mild dysplasia to carcinoma in situ in some cases.To the best of our knowledge,only five cases of SPA in the submandibular gland have been reported to date.Here,we present two new cases of SPA involving the submandibular gland.CASE SUMMARY A 50-year-old woman and a 52-year-old woman were referred to Tongji Hospital in Wuhan,China,with complaints of moderate pain,recurrent swelling,and a mass in the submandibular area.After admission,the two cases of the submandibular mass were examined physically.The boundary of the submandibular tumor was clear,and the range of motion was good.After preoperative examinations,surgery was performed on a selective basis.Postoperative histopathological examination revealed a well-defined mass with acinar structures,ducts,or cystic dilated glands of various sizes scattered in a large number of proliferative sclerosing stroma.There were flat and cuboidal cells,and eosinophils in the duct epithelium.There was also a eosinophilic substance in the lumen of dilated cysts.No atypical epithelial hyperplasia,invasive growth,or carcinoma in situ was found.Based on the above findings,the mass was diagnosed as SPA.Both patients have remained asymptomatic and no recurrence or distant metastasis had occurred by the 7-mo and 5-year follow-up,respectively.CONCLUSIONSPA is a rare disease of the salivary gland. Even though it has a good prognosisafter adequate surgery, atypical lesions may occur from mild dysplasia tocarcinoma in situ. However, no recurrence, distant metastasis, or mortality hasbeen reported for submandibular gland SPA. Clinicians and pathologists shouldbe familiar with the characteristics of SPA in the submandibular gland to avoidmisdiagnosis and overtreatment.

Development of vesicovaginal fistula caused by vaginal adenosis: A case report

Introduction: Vaginal adenosis is one of the rare diseases of the vagina, and almost all patients are asymptomatic. We report a case of spontaneous vaginal adenosis, which caused vesicovaginal fistula. Case Presentation: Our patient was a 25-year-old Japanese woman. She was admitted to our hospital, and her chief complaint was continuous urine flow from the vagina. We found a tumor with a vesicovaginal fistula in her vagina. Subsequent cytological analysis of vaginal smears showed a normal vaginal mucosa. The patient underwent tumor resection and a fistula patch. Pathological diagnosis was adenosis of the vagina, even though the patient had no known history of intrauterine diethylstilbestrol exposure or Müllerian developmental abnormalities. Conclusion: The clinical course of our case was a malignant tumor, which invasively bored a hole in the vaginal wall forming a vesicovaginal fistula, even though it was a benign lesion. Therefore, overzealous treatment should be avoided in this case.