The first known PT was described in 1838 by Johannes Müller [4, 5]. In recognition of their benign behavioral characteristics, they were temporarily designated cystosarcoma phyllodes. Phyllodes tumors were first classified by the World Health Organization in 1982, and they are further divided into benign, borderline, and malignant types [6, 7].

It is estimated that less than 1% of all breast tumors are PTs, which are most prevalent in the breast [8]. The incidence of prostate PT is second only to that of breast cancer [9]. However, fewer than 100 cases of prostate PT have been reported [10]. In addition to the breast and prostate, primary PTs, such as the verumontanum [2] and seminal vesicles [10]. There is a very low incidence of PT in the urinary tract.

Tchrakian et al. first reported the unique PT case of a human bladder PT in 2018, in which a 54-year-old man underwent a partial cystectomy due to multiple recurrences [11]. During histopathological examination, an epithelio-stromal biphasic polypoid tumor was observed in the mucosal layer of the bladder dome. Neither heterologous components nor infiltrative growth was detected. In this case, it was classified as a benign/low-grade bladder tumor.

To the best of our knowledge, only one case of primary ureteric PT has been reported. A leaf-like pattern was observed in our patient, as was biphasic differentiation between the epithelial and stromal layers, as previously reported. We observed that the stromal cells in our patient were spindle shaped and stellate, which was in accordance with benign lesions, such as noninfiltrative borders, bland cytology, few mitotic figures, and low proliferation rates.

Bostwick DG et al. reviewed 23 prostate lobed tumors on the basis of five histological features, including cell size, cell atypia, the number of mitotic images per 10 high-power fields, the stromal-to-epithelial ratio and necrosis [12]. In the PT of the prostate, a lower level, a middle level, and a higher level are distinguished. It will be possible to observe recurrence rates and distant metastases among these three grade levels. Note that the histologic grade of prostate lobar tumors does not indicate the long-term prognosis but that the longer they are present, the more likely they are to be localized and metastasized distantly. Therefore, complete resection is necessary at first diagnosis.

There are few reports of primary lobe tumors in areas other than the breast and prostate and few reports of urinary diseases such as seminal vesicle glands. Primary lobe tumors of the ureter are extremely rare. A case of urothelial carcinoma with a terminal lobe tumor of the ureter was reported by Dang Linlin [3]. She reported that ductal carcinoma in situ occurred in conjunction with a lobe tumor. However, further research is needed regarding the adjuvant treatment of lobular tumors associated with ductal carcinoma in situ or invasive ductal carcinoma. Fortunately, our case was benign with low cell density, no atypia, no mitotic image, no sarcomatosis or invasive behavior, wild-type p53 expression and low proliferation.

In conclusion, It is extremely rare for benign phyllodes tumors to develop in the ureter with some of them showing morphologic overlap with fibroadenoma, suggesting that they may have a common origin. As phyllodes tumors, our case fit the conventional definition. Given the rare occurrence of lobe tumors in the ureter and bladder, no established standard of treatment exists and the prognosis is not clear [13]. It is believed that complete resection of the tumor reduces the risk of recurrence, but there is no specific prognosis, and further close follow-up is needed. There are also some limitations to our manuscript. In order to gather more information, we reviewed the pathological diagnosis of terminal ureteral lobular tumors in our department over the past ten years. Regrettably, only this case was found to be used as a reference by the pathologist. Morever, our follow-up time was too short to provide accurate information about this patient’s subsequent recurrence and metastasis. Further study is needed to establish the clinical significance of this manifestation.