Figure 1: The uterine cut surface with cyst formation without relation to the endometrium.
Figure 2: Multiple cysts of variable sizes lined with benign epithelium are seen (H&E x40).
Many extragenital locations have been reported in the literature such as peritoneum, subperitoneal tissue, colon, appendix, umbilicus, and lymph nodes[2,4]. The examples of tumor-like cystic endosalpingiosis were localized to the female pelvis and most of those cases were reported to be derived from the serosal surface of the uterus and the ovary[1,6-11]. The tumor-like cystic endosalpingiosis was located in a subserosal location with intramural involvement in only six cases previously published in the literatüre[1,5,12-14]. The case presented here was different from the cases reported before with its feature of purely uterineintramural cystic tumor-like endosalpingiosis without a serosal component as published by Çil et al.[15] (Table I).
Tumor-like cystic endosalpingiosis usually mimics other lesions such as leiomyoma, serosal cyst etc.[1,9]. The lesion mimicked leiomyoma in the current case as well. The differential diagnosis of endosalpingiosis includes cystic tumor-like lesions involving the uterus such as cystic adenomyosis with tubal metaplasia, adenomatoid tumor, and peritoneal inclusion cyst[5,6,8,12,13]. Absence of endometrial stroma differentiates cystic adenomyosis from endosalpingiosis. Cystic adenomatoid tumors may present as a uterine cystic mass. They have multiple small ovoid or slit-like spaces lined with flattened and cuboidal non-tubal type epithelial cells admixed with smooth muscle fibers[5,8]. They are usually located on the subserosal area of the female genital tract, sometimes with a myometrial invasion.
However, there was no evidence of serosal involvement in our case and immunohistochemistry would be helpful in the differential diagnosis otherwise.
The etiology and the histogenesis of cystic endosalpingiosis is not clear, but may be related to inflammatory conditions, previous adnexal surgery or peritoneal mesothelial metaplasia depending on the growth factor stimuli or steroid hormones[13]. There is some evidence that it might be a secondary müllerian system pathology with mullerianosis; such as lymphadenectomy specimens resected due to ovarian serous carcinoma usually containing endosalpingiosis, endosalpingiosis cases almost always being accompanied by a serous cystic neoplasm, and urinary bladder endosalpingiosis[1,16]. Our case had follicular salpingitis that might support the idea of a possible relationship between an inflammatory process and this entity.
In conclusion, endosalpingiosis rarely presents with such large cystic changes and striking macroscopic findings. Awareness of the existence of this rare lesion will prevent an incorrect diagnosis.
1) Clement PB, Young RH: Florid cystic endosalpingiosis with
tumor like manifestations: Am J Surg Pathol 1999, 23;166-175 [ Özet ]
2) McCluggage WG, Clements WD: Endosalpingiosis of the colon
and appendix: Histopathology 2001, 39:645-646 [ Özet ]
3) Ong NCS, Maher PJ, Pyman J M, Gordon S, Readman, E:
Endosalpingiosis, an unrecognized condition: Report and
literature review. Gynecol Surg 2004, 1:11-14
4) Papavramidis TS, Sapalidis K, Michalopoulos N,
Karayannopoulou G, Cheva A, Papavramidis ST: Umblical
endosalpingiosis: A case report: J of Med Case Reports 2010,
4:287 [ Özet ]
5) Shim SH, Kim HS, Jo M, Chang SH, Kwak JE: Florid cystic
endosalpingiosis of the uterus: A case report. Korean J of Pathol
2008, 42:189-191
6) Rosenberg P, Nappi L, Santoro A, Bufo P, Greco P: Pelvic masslike
florid cystic endosalpingiosis of the uterus: A case report
and a review of the literature. Arch Gynecol Obstet 2011, 283:
519-523 [ Özet ]
7) Heatley MK, Russell P: Florid cystic endosalpingiosis of the
uterus. J Clin Pathol 2001, 54:399-400 [ Özet ]
8) Chang Y, Tsai EM, Yang C: Multilobular cyst as endesalpingiosis
of uterine serosa: A case report. J Med Sci 2003, 19:38-41 [ Özet ]
9) Taneja S, Sidhu R, Khurana A: MRI apperance of florid cystic
endosalpingiosis of the uterus: A case report. Korean J Radiol
2010, 11:476-479 [ Özet ]
10) Bryce RL, Barbatis C, Charnock M: Endosalpingiosis in
pregnancy. Case report. Br J Obstet Gynaecol 1982, 89:166-168 [ Özet ]
11) Lee SN, Cho MS, Kim SC: Tumor-like multilocular cystic
endesalpingiosis of the uterine serosa: Possible clinical and
radiologic misinterpreted. Acta Obstet Gynecol Scand 2005,
84:98-99 [ Özet ]
12) Fukunaga M: Tumor-like cystic endosalpingiosis of the uterus
with florid epithelial proliferation. APMIS 2004, 112:45-48 [ Özet ]
13) Kajo K, Zúbor P, Macháleková K, Plank L, Visnovskỳ J: Tumorlike
manifestation of endesalpingiosis in uterus: A case report.
Pathology Research and Practice 2005, 201:527-530 [ Özet ]
14) Youssef AH, Ganesan R, Rollason TP: Florid cystic
endosalpingiosis of the uterus. Histopathology 2006, 49:546-547 [ Özet ]
15) Cil AP, Atasoy P, Kara SA: Myometrial involvement of tumorlike
cystic endosalpingiosis: A rare entity. Ultrasound Obstet
Gynecol 2008, 32:106-110 [ Özet ]
16) Young RH, Clement PB. Müllerianosis of the urinary bladder.
Mod Pathol 1996, 9:731-737 [ Özet ]