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CASE REPORT  
Year : 2021  |  Volume : 64  |  Issue : 4  |  Page : 806-809
Mesonephric adenocarcinoma in the uterine corpus: A case report and review of the literature


Department of Pathology, West China Second University Hospital, Sichuan University, Key Laboratory of Birth Defects and Related Diseases of Women and Children (Sichuan University), Ministry of Education, China

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Date of Submission01-Apr-2020
Date of Decision27-May-2020
Date of Acceptance22-Jun-2020
Date of Web Publication20-Oct-2021
 

   Abstract 


Mesonephric adenocarcinoma (MNA) is a rare malignancy arising from the mesonephric remnant of the female reproductive tract, typically found in the cervix. MNA is uncommon in the uterine corpus, only 33 cases have been described in the literature. A 55-year-old postmenopausal woman presented with pink vaginal discharge and bilateral hip pain for 2 months, with the help of histopathologic observation and immunohistochemical staining, a diagnosis of “MNA” was made. The tumor invaded the whole layer of myometrium without endometrium involvement, mesonephric remnants and hyperplasia of the mesonephric duct were also found at the periphery of the neoplasm. After the operation, the patient was treated with 3 cycles of chemotherapy. The patient was followed for 6 months with disease. Further experience to diagnose and cure this rare tumor is warranted.

Keywords: Diagnosis, mesonephric adenocarcinoma, uterine corpus

How to cite this article:
Tang X, Li L, He Y. Mesonephric adenocarcinoma in the uterine corpus: A case report and review of the literature. Indian J Pathol Microbiol 2021;64:806-9

How to cite this URL:
Tang X, Li L, He Y. Mesonephric adenocarcinoma in the uterine corpus: A case report and review of the literature. Indian J Pathol Microbiol [serial online] 2021 [cited 2021 Dec 7];64:806-9. Available from: https://www.ijpmonline.org/text.asp?2021/64/4/806/328552





   Introduction Top


Mesonephric adenocarcinoma (MNA) is a rare malignant tumor in the gynecologic tract, typically occurring in the lateral wall of the cervix.[1] MNA of the uterine corpus is extremely rare, only 33 cases reported in the English literature. In this study, we report one case of MNA in the uterine corpus with its clinicopathologic features, and relevant literatures are reviewed.


   Case History Top


A 55-year-old postmenopausal woman, presented with pink vaginal discharge and bilateral hip pain for 2 months, gynecological examination revealed a 3-cm polypoid mass on the right side of the vagina, cervical cytology revealed atypical glandular cells, transvaginal ultrasound examination showed uterine occupation, CT showed soft tissue mass in the posterior wall and the fundus of the uterus, bone destruction and soft tissue mass in the right ischium, scattered nodules in bilateral lung, metastatic tumors were considered, and a hysteroscopic curettage and vaginal biopsy were performed, the mass was diagnosed as adenocarcinoma. The patient underwent hysterectomy, bilateral salpingo-oophorectomy. Gross examination revealed a large nodular mass located in the posterior wall of the uterus [Figure 1]. The mass measured 11 × 7 × 7 cm in size. Cervix and bilateral adnexa were grossly normal. Morphologically, the tumor showed a variety of growth patterns, including tubulocystic, papillary, retiform, solid components, and a sarcomatous component, were typically seen [Figure 2]a and [Figure 2]b. A few adenoid structures were observed in the focal solid nests, and dense eosinophilic secretions were observed in the gland lumens. The tumor cells were relatively consistent, mild to moderate atypia, and mitotic counts were ranged from 1 to 8/10 high-power fields. In addition, a sarcomatous component composed of spindle-shaped cells was recognized, and there was a transition from the carcinomatous component to the sarcomatous component. Mesonephric remnants and hyperplasia of the mesonephric duct with atypical architectural and nuclear features were also found at the periphery of the neoplasm [Figure 2]c and [Figure 2]d. The tumor cells invaded the whole layer of myometrium and involved the right ovary, the entire endometrium was submitted for microscopic examination and showed normal proliferative endometrium. Immunohistochemical staining demonstrated that tumor cells were positive for TTF-1, PAX8, PAX2, and CD10 [Figure 3]a, [Figure 3]b, [Figure 3]c, a sarcomatous component were positive for Vimentin [Figure 3]d, and all the tumor cells were negative reactivity for GATA3, ER, PR, p16, HNF-1β, NapsinA and wild-type p53 immunostaining pattern, the Ki-67 proliferative index was about 80%. The pathological diagnosis was MNA with a sarcomatous component. After the operation, the patient was treated with 3 cycles of ifosfamide, cisplatin, bevacizumab and Pamidronate disodium. The patient was followed for 6 months with disease.
Figure 1: Gross findings of uterine mesonephric adenocarcinoma

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Figure 2: The tumor represented mainly as a tubulocystic pattern (a), a sarcomatous component composed of spindle-shaped cells was recognized (b), mesonephric remnants and hyperplasia of the mesonephric duct were found at the periphery of the neoplasm (c and d)

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Figure 3: The tumor cells were positive for PAX8 (a), TTF-1 (b) and CD10 (c), the sarcomatous component was positive for Vim (d)

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   Discussion Top


MNA is a rare malignancy arising from the mesonephric remnant of the female reproductive tract.[2] MNA is uncommon in the uterine corpus, to our knowledge, only 33 cases (including ours) have been reported in English literature [Table 1]. There are two main theories about the origin of uterine corpus MNA, one theory is that it arises from mesonephric remnants, the other is that a Müllerian origin based on the lack of mesonephric remnants in the uterine corpus, and endometrial involvement in almost all reported cases.[3] Thus the term “mesonephric-like Müllerian adenocarcinoma” has been proposed for MNA arising from uterine corpus, as well as rare cases of MNA from ovary.[4] But this theory is still controversial, it is unknown whether these neoplasms in the uterine corpus represent mesonephric-derived adenocarcinomas or just mesonephric-like endometrial adenocarcinomas.[5] In our case, Mesonephric remnants and hyperplasia of the mesonephric duct are found at the periphery of the neoplasm, and the tumor is located deep in the myometrium and without endometrial involvement, So we believe that the tumor originated from mesonephric remnants.
Table 1: Reported cases of uterine corpus MNA

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The clinical symptoms are not specific, included abnormal vaginal bleeding, lower abdominal pain and presence of an abdominal mass.[6] Patients ages range from 33 to 81, with the majority of patients in the fifth and sixth decades of life.[3] Because of its localization, it is rarely discovered on PAP smear. According to the literature reported, uterine corpus MNA always develop metastatic disease, the most common site of metastasis is the lung, the reason for frequent pulmonary involvement of uterine corpus MNA is unclear, hematogenous spread is one of the possible mechanism, as vascular invasion is seen in the peritumoral areas.[7] In our case, we also found lymphovascular invasion in the peritumoral areas. Based on the CT scan, we concluded that the tumor had metastasized and involved bone and lung.

Because of its rarity and morphologic diversity, diagnosis of MNA can be challenging, it is made for the most part on biopsy or hysterectomy specimens. Morphologically, MNA of the uterine corpus is identical to other areas MNA. The tumors exhibit diverse architectural patterns, with various combinations of tubular, glandular, papillary, retiform, glomeruloid, sex cord-like, and comedonecrosis-like patterns.[7] It often has a biphasic variant, it can be either pure adenocarcinomas or adenocarcinomas that are associated with a sarcomatous component. The pure adenocarcinomas can be confused with endometrial carcinoma arising from Müllerian epithelium, including endometrioid adenocarcinoma, serous adenocarcinoma, especially clear cell carcinoma. Clear cell carcinomas of the uterine show varying degrees of cystic, papillary, and solid patterns, it may also exhibit prominent tubules filled with eosinophilic hyaline material, but MNA often consist of tubular glands that varied in size and lined by one to several layers of columnar cells, lack clear cells, eosinophilic cells, and hobnail-shaped cells, Moreover, in our case, the tumor cells were negative for HNF-1β and NapsinA, that can be differentiated from clear cell carcinoma. The sarcomatous component was easily misdiagnosed as a malignant mixed Müllerian tumor (MMMTs) or undifferentiated carcinoma in the biopsy specimen. However, MMMTs often occur in older patients, presented as an endometrial polyp and usually filled the entire endometrial cavity, many invade the myometrium but some are confined to polyps. MMMTs are composed of an admixture of histologically malignant epithelial and mesenchymal components, approximately half the cases demonstrate a homologous stromal component, most importantly, the uterine MMMTs lacked mesonephric hyperplasia.

A panel of immunohistochemical stainings, including GATA3, PAX8, and CD10, may be useful in the differential diagnosis, these markers seem unique for MNA, and are not seen in other kinds of carcinomas.[8],[9] MNA showed wild-type p53, patchy p16 and negative reactivity for hormonal receptors. GATA3 has recently been shown to be a sensitive and specific marker of mesonephric epithelia, it can be used to distinguish MNA from other carcinomas of the gynecologic tract.[9] However, in the reported literatures, not all MNA express GATA3 positively, even when GATA3 is positive, it remain some differences in the level of expression.[1],[5] Some research demonstrate that TTF-1 can be positive in mesonephric carcinomas.[5] In our report, the tumor cells were negative for ER, PR, P16, HNF-1β, NapsinA, and showed wild-type p53, this can be distinguish it from endometrial carcinoma, furthermore, the tumor cells were positive for TTF-1, PAX8, PAX2, and CD10, although GATA3 was negative, the diagnosis of MNA was made. Pors et al. found the inverse pattern of staining between GATA3 and TTF1 in MNA, it meant in areas of tumor that were positive for GATA3 were completely negative for TTF1, and vice versa, they suggest that GATA3 staining should be performed first because of its higher sensitivity in a suspected case of mesonephric neoplasm, if negative, due to the inverse staining pattern between GATA3 and TTF1, TTF-1 staining could be attempted.[10]

Surgical resection is the main treatment, and some cases are received adjuvant chemotherapy and radiation. The prognosis of uterine MNA is difficult to estimate but based on the limited cases have reported that the tumor often carry a worse prognosis, and the relation of the sarcomatous component with the prognosis is not yet clear. Hence, more cases are needed to reliably estimate the behavior of these rare tumors.

Because of the rarity of this tumor, the etiology and precursor lesions remain unclear. Considering its high metastatic potential and frequent pulmonary involvement, further experience to diagnose and cure this rare tumor is warranted.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Kolin DL, Costigan DC, Dong F, Nucci MR, Howitt BE. A combined morphologic and molecular approach to retrospectively identify KRAS-mutated mesonephric-like adenocarcinomas of the endometrium. Am J Surg Pathol 2019;43:389-98.  Back to cited text no. 1
    
2.
Bagué S, Rodríguez IM, Prat J. Malignant mesonephric tumors of the female genital tract: A clinicopathologic study of 9 cases. Am J Surg Pathol 2004;28:601-7.  Back to cited text no. 2
    
3.
Zhang L, Cai Z, Ambelil M, Conyers J, Zhu H. Mesonephric adenocarcinoma of the uterine corpus: Report of 2 cases and review of the literature. Int J Gynecol Pathol 2019;38:224-9.  Back to cited text no. 3
    
4.
McFarland M, Quick CM, McCluggage WG. Hormone receptor-negative, thyroid transcription factor 1-positive uterine and ovarian adenocarcinomas: Report of a series of mesonephric-like adenocarcinomas. Histopathology 2016;68:1013-20.  Back to cited text no. 4
    
5.
Liang Y, Shi H, Zhu X, Yu M, Zhang X. Mesonephric adenocarcinoma of the uterine corpus: A report on 2 cases with comparison to its cervical counterpart. Int J Gynecol Pathol 2020. [online ahead of print]. doi: 10.1097/PGP.0000000000000639.  Back to cited text no. 5
    
6.
Zhao J, Liu C, Qi J, Qu P. Mesonephric carcinoma of the uterine corpus: A report of two cases. Oncol Lett 2016;11:335-9.  Back to cited text no. 6
    
7.
Na K, Kim HS. Clinicopathologic and molecular characteristics of mesonephric adenocarcinoma arising from the uterine body. Am J Surg Pathol 2019;43:12-25.  Back to cited text no. 7
    
8.
Ordi J, Nogales FF, Palacin A, Márquez M, Pahisa J, Vanrell JA, et al. Mesonephric adenocarcinoma of the uterine corpus: CD10 expression as evidence of mesonephric differentiation. Am J Surg Pathol 2001;25:1540-5.  Back to cited text no. 8
    
9.
Howitt BE, Emori MM, Drapkin R, Gaspar C, Barletta JA, Nucci MR, et al. GATA3 is a sensitive and specific marker of benign and malignant mesonephric lesions in the lower female genital tract. Am J Surg Pathol 2015;39:1411-9.  Back to cited text no. 9
    
10.
Pors J, Cheng A, Leo JM, Kinloch MA, Gilks B, Hoang L. A comparison of GATA3, TTF1, CD10, and calretinin in identifying mesonephric and mesonephric-like carcinomas of the gynecologic tract. Am J Surg Pathol 2018;42:1596-606.  Back to cited text no. 10
    

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Correspondence Address:
Yanmei He
No. 20, Section 3, South Renmin Road, Chengdu - 610 041
China
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/IJPM.IJPM_298_20

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