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CASE REPORT  
Year : 2011  |  Volume : 54  |  Issue : 3  |  Page : 572-573
Extrauterine adenomyoma with uterus like features: A rare entity presenting 17 years post hysterectomy


Department of Pathology, Rajiv Gandhi Cancer Institute And Research Center, Sector-5, Rohini, Delhi, India

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Date of Web Publication20-Sep-2011
 

   Abstract 

We report a case of a 47-year-old female (posthysterectomy) with bleeding per vaginam. Imaging studies showed a large abdomino-pelvic mass diagnosed as extrauterine adenomyoma with uterus-like features. This pathological entity is extremely uncommon with only few case reports available in the reported literature. This case is being highlighted for its rarity and to discuss the possible theories for origin of this uncommon condition.

Keywords: Adenomyoma, extrauterine, uterus-like mass

How to cite this article:
Khurana A, Mehta A, Sardana M. Extrauterine adenomyoma with uterus like features: A rare entity presenting 17 years post hysterectomy. Indian J Pathol Microbiol 2011;54:572-3

How to cite this URL:
Khurana A, Mehta A, Sardana M. Extrauterine adenomyoma with uterus like features: A rare entity presenting 17 years post hysterectomy. Indian J Pathol Microbiol [serial online] 2011 [cited 2020 Jun 3];54:572-3. Available from: http://www.ijpmonline.org/text.asp?2011/54/3/572/85096



   Introduction Top


Adenomyomas are benign tumors composed of smooth muscle and benign endometrium. [1] They are typically located in myometrium, but also noted in the cervix and the  Fallopian tube More Details. Extrauterine adenomyomas are rare, and those mimicking a uterus are further rarer. Due to the rarity of this entity, a definite histogenesis and origin of such extra uterine masses is still unknown. [2]


   Case Report Top


A 47-year-old female presented with complain of bleeding per vaginam, since 20 days prior to presentation. There was no associated pain or any other symptom. Her general physical examination was unremarkable. Patient had a past history of undergoing sub-total hysterectomy 17 years back for complains of multiple fibroids. Five years later she underwent an explorative laparotomy for removal of bilateral ovaries, which on histopathology revealed endometriotic tissue. On per abdomen examination there was a vague lump palpable in the epigastrium and the left lumbar region. Per speculum examination was unremarkable. Contrast-enhanced computerized tomography (CECT) revealed a large predominantly cystic mass in the abdomino-pelvic region with internal septae. The mass was crossing the midline [Figure 1]. At few places, cyst wall showed thickened solid areas. Findings were suggestive of a neoplastic pathology. Retroperitoneal lymph nodes were not enlarged. Serum CA125 levels were 363U/ml (normal range 0-35). A preoperative diagnosis of a gastrointestinal stromal tumor (GIST) and leiomyosarcoma was offered. Subsequently she underwent excision of the mass and the specimen was sent to our department. Gross examination revealed multiple peritoneal covered soft tissues, largest measuring 13 Χ 9 Χ 5 cm with irregular grey white surface. On cut section, multi-septate cystic spaces filled with serous fluid constituted majority of the specimen. Few thickened grey white solid areas with entrapped mucoid material were seen [Figure 2]. Multiple sections were given from the specimen. Microscopy revealed a thick muscular cyst wall, which was lined by endometrial glands with accompanying endometrial stroma [Figure 3]. Few foci showed cystic change in these glands. Nuclear anaplasia, increased mitoses or necrosis were absent. Broad papillae lined by tall columnar ciliated epithelium resembling fallopian tube were noted in few sections [Figure 4]. Background showed areas of hemorrhage, hemosiderin-laden macrophages and chronic inflammatory cells. In view of the past history of hysterectomy, a final diagnosis of extrauterine adenomyoma with uterus-like features was offered. Her preoperative period is uneventful.
Figure 1: CECT image showing a predominantly cystic mass with peripheral thickened solid areas in the abdomino-pelvic region

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Figure 2: Cut surface showing multiple cystic spaces with peripheral thickened grey white areas

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Figure 3: Benign endometrial glands surrounded by smooth muscle bundles (H and E, x200)

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Figure 4: Broad plicae-like structures lined by tall columnar epithelium. (H and E, x200). Inset showing ciliated epithelium (H and E, x400).

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   Discussion Top


The first uterus-like mass was described by Cozzutto [3] in 1981. Since then only 15 such cases have been published in the current literature. [2] Majority of extrauterine cases were of ovarian origin. [1],[4] Rare cases originating from broad ligament and mesentery have been reported. [4],[5],[6] Age of presentation was variable. The gross findings of previously reported cases consistently showed a solid cystic mass; similar findings were noted in the present case. [1] Various hypotheses put forward for the occurrence of this rare phenomenon include mullerian duct anomaly, a metaplastic change or a heterotropia. [6],[7] Pueblitz and his co-authors [8] believed that a congenital malformation, possibly caused by a defect in fusion of mullerian ducts, should be considered in pathogenesis of this condition. The sub-celomic mesenchyme theory supports the opinion that the sub-peritoneal mesenchymal cells retain the ability of duplicating mullerian structures, rather than these being embryological defects. [1],[5] As in this case, the patient had past history of endometriosis for which she underwent surgery, we propose the mullerian regenerative theory as the likely cause for origin of the uterus-like peritoneal mass. Further the presence of fallopian tube like mucosa in the indexed case, hints at mullerian metaplasia as the possible pathogenesis. The present case is being highlighted for extreme rarity and its clinico-radiological findings simulating a pelvic malignancy.

 
   References Top

1.Redman R, Wilkinson EJ, Massoll NA. Uterine-like Mass with Features of an Extrauterine Adenomyoma presenting 22 years after Total abdominal hysterectomy-Bilateral salpingo-oopherectomy. Arch Pathol Lab Med 2005;129:1041-3.  Back to cited text no. 1
[PUBMED]  [FULLTEXT]  
2.Menn KA, Qin L, Fedoriw GD, Gillette ME, McCarthy S. Uterus-like mass : M0 RI appearance of a very rare entity. J Magn Reson Imaging 2007;26:162-4.  Back to cited text no. 2
[PUBMED]  [FULLTEXT]  
3.Cozzutto C. Uterus like mass replacing ovary. Arch Pathol Lab Med 1981;105:508-11.  Back to cited text no. 3
[PUBMED]    
4.Horie Y, Kato M. Uterus-like mass of the small bowel mesentery. Pathol Int 2000;50:76-80.  Back to cited text no. 4
[PUBMED]  [FULLTEXT]  
5.Ahmed AA, Swan RW, Owen A, Kraus FT, Patrick F. Uterus-like mass arising in the broad ligament : A0 metaplasia or mullerian duct anamoly? Int J Gynecol Pathol 1997;16:279-81.  Back to cited text no. 5
[PUBMED]    
6.Liang YJ, Hao Q, Wu YZ, Wu B. Fertil Steril 2010;94:1347.  Back to cited text no. 6
    
7.Kaufman Y, Lam A. The pelvic uterus-like mass- a primary or secondary mullerian system anamoly? J Minim Invasive Gynecol 2008;15:494-7.  Back to cited text no. 7
[PUBMED]  [FULLTEXT]  
8.Pueblitz-Peredo S, Luevano-Flores E, Rincon-Taracena R, Ochoa- Carrillo FJ. Uterus-like mass of the ovary : E0 ndometriosis or congenital malformation. Arch Pathol Lab Med 1985;109:361-4.  Back to cited text no. 8
    

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Correspondence Address:
Anuj Khurana
Department of Pathology, 98, SFS Flats Phase-4, Ashok Vihar, Delhi - 110 052
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0377-4929.85096

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    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]

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