• Users Online: 53
  • Home
  • Print this page
  • Email this page
Home About us Editorial board Ahead of print Current issue Search Archives Submit article Instructions Subscribe Contacts Login 


 
 Table of Contents  
CASE REPORT
Year : 2018  |  Volume : 13  |  Issue : 2  |  Page : 111-113

Cotyledonoid dissecting leiomyoma of the uterus


Department of Obstetrics and Gynaecology, Christian Medical College, Ludhiana, Punjab, India

Date of Web Publication23-Nov-2018

Correspondence Address:
Dr. Arti Gupta Tuli
Associate Professor, Department of Obstetrics and Gynaecology, Christian Medical College, Ludhiana - 141 008, Punjab
India
Login to access the Email id

Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jdmimsu.jdmimsu_65_17

Rights and Permissions
  Abstract 


This is a rare case of Cotyledonoid Dissecting Leiomyoma (CDL) in a 50 year old lady. She was evaluated for heavy menstrual bleeding and congestive dysmenorrhea. Her ultrasonogram pelvis and MRI pelvis revealed a large cystic space occupying lesion in pelvis situated in pouch of douglas measuring 7x4 cm with peripherally arranged hemorrhagic cyst, largest measuring 3.3X2.7 cm likely to be of left ovarian origin. She was taken up for Laparoscopy proceed laparotomy which revealed normal uterus with bilateral fallopian tubes and ovaries. A 10x10 cm exophytic, fleshy, serosal, polypoidal mass originating from posterolateral wall of uterus giving an impression of malignancy was noted. Total abdominal hysterectomy with bilateral salpino-oopherectomy was done and specimen sent for histopathological examination. HPE report showed it to be CDL which was benign.

Keywords: Cotyledonoid, dissecting, leiomyoma, uterus


How to cite this article:
Tuli AG, Goyal S. Cotyledonoid dissecting leiomyoma of the uterus. J Datta Meghe Inst Med Sci Univ 2018;13:111-3

How to cite this URL:
Tuli AG, Goyal S. Cotyledonoid dissecting leiomyoma of the uterus. J Datta Meghe Inst Med Sci Univ [serial online] 2018 [cited 2018 Dec 16];13:111-3. Available from: http://www.journaldmims.com/text.asp?2018/13/2/111/246014




  Introduction Top


Leiomyoma is the most common tumor of the genital tract.[1] Uterine leiomyoma is classified into two major categories – conventional leiomyoma which are symmetrically expansible, rounded, and circumscribed and uterine smooth muscle tumors with unusual growth patterns.[2] Due to its striking resemblance to cotyledons of placenta and its dissecting nature, it is named as cotyledonoid dissecting leiomyoma (CDL). It was previously also known as Sternberg tumor or grape-like leiomyoma.[2],[3] It is important to recognize this tumor since it may be confused with a malignant neoplasm because of its bizarre macroscopic appearance, but it has a benign histology and biological behavior.[3] To date, only 41 cases of CDLs have been reported in literature.[3] Here, we emphasize that clinicians should be aware of this rare entity to prevent unnecessary aggressive treatment due to its warning gross morphology.


  Case Report Top


We report a rare case of CDL in a 50-year-old female. She was para 2, living 2 (P2L2) with previous lower segment cesarean section. She presented with heavy menstrual bleeding and congestive dysmenorrhea for the past 4 months. On abdominal examination, no mass was palpable; on pelvic examination, uterus felt bulky, a large irregular mass felt in pouch of Douglas pushing the uterus anteriorly. Ultrasonography showed uterus bulky, 11.5 cm × 3.6 cm in size. Left lateral wall of uterus showed evidence of a heterogeneous mass of 7 cm × 4 cm. Mass showed increased vascularity. Margins of mass not well defined. Endometrial thickness was 5mm. Right ovary showed evidence of a retention cyst 3.3 cm. Left ovary enlarged measures 10.6 cm × 4.9 cm. Left ovary adherent to left side of uterus showed a cyst of 3.1 cm with fine internal echoes (? endometriotic cyst). Magnetic resonance imaging pelvis showed uterus of 10.8 cm × 4.4 cm size. Endometrial cavity was normal. Junctional zone was normal. Well-defined space-occupying lesion appearing hypointense on T2W images seen in the right lateral wall of uterus in intramural location measuring approximately 2.8 cm × 2.4 cm. Similar smaller space-occupying lesion was seen in posterior myometrium measuring approximately 1.4 cm × 1.3 cm. Right ovary showed two simple unilocular cysts measuring 2.6 cm × 2.5 cm and 3.1 cm × 2.1 cm. A large cystic space-occupying lesion was seen in pelvis situated in pouch of Douglas measuring 7 cm × 4 cm with peripherally arranged hemorrhagic cyst largest measuring 3.3 cm × 2.7 cm likely to be left ovarian origin leading to ovarian enlargement. Uterus displaced anteriorly by the lesion. No enlarged lymph nodes or ascites were present. Tumor markers were CA-125-67U/ml and HE4-55 pmol/L. Other relevant laboratory investigations were within normal range. The patient was taken up for diagnostic laparoscopy to look for the nature of mass (benign/malignant). On laparoscopy, uterus was of normal size, pushed anteriorly by a big mass in the POD which was variegated, congested, and with irregular margins. Both ovaries and tubes were almost normal. Origin of mass could not be made out. The appearance of the mass was felt to be malignant, so plan for laparotomy followed by total abdominal hysterectomy with bilateral salpingo-oophorectomy was made. Laparotomy findings were uterus normal size. Bilateral tubes and ovaries were normal. A 10 cm × 10 cm exophytic, fleshy, serosal, polypoidal mass originating from posterolateral wall of uterus giving an impression of malignancy was noted. Cut section of uterus was normal. Macroscopically, the entire specimen including tumor, uterus, and adnexa weighed 450 g. The serosal surface showed an irregularly contoured exophytic mass measuring 11.8 cm × 10.5 cm × 5.5 cm, arising from the lower left lateral posterior uterine wall. The mass was bosselated, partly cystic and large part of the mass had poorly defined borders with the myometrium in the lower uterine wall. Cut surface demonstrated firm nodules of grayish white tissue and multiple thin-walled cysts. The myometrium showed trabeculations and focal cystic areas. Histopathologically, tumor had two components – a cotyledonoid extrauterine component and an intramural component and the former was intramural in origin. The lesion consisted of a proliferation of smooth muscle cells with intra- and extrauterine tumor component with hyalinization. The tumor was composed of interlacing fascicles, whorls, and bundles of bland smooth muscle cells. No increase in cellularity, mitosis, or atypia was seen. Areas of perinodular hydropic degeneration were also evident. Myometrial component showed tumor nodules with a dissecting pattern within adjacent uterine smooth muscle layer. Immunohistochemistry showed smooth muscle actin, h-caldesmon: positive in tumor cells (tumor of smooth muscle) Ki-67 Proliferating index: low (2%), CD34: negative in tumor cells, highlights the blood vessels (intravascular component was excluded), and CD10: negative in tumor cells. A diagnosis of CDL was made according to the macroscopic, microscopic, and immunophenotype appearance of the tumor [Figure 1], [Figure 2], [Figure 3], [Figure 4].
Figure 1: Macroscopic findings – the resected uterus shows a congested exophytic bosselated mass attached to the posterior surface (anterior Views)

Click here to view
Figure 2: Note the bulbous protrusion of tumor (arrow) reminiscent of the maternal surface of placenta (posterior view)

Click here to view
Figure 3: Microscopy – cotylenoid leiomyoma (arrow) with dissecting component (asterisk)

Click here to view
Figure 4: Immunohistochemistry – low MIB-1 labeling index

Click here to view



  Discussion Top


CDL or Sternberg tumor was first described in 1996 by Roth et al.[4] It can be presented in a wide age range between 23 and 65 years of age (mean 40.3 years).[1] The most common presenting symptoms are abnormal uterine bleeding and pelvic mass,[1] just as our current case. The impression of malignancy can be attributed to the rarity and bizarre appearance of the tumor.[1],[2],[4] CDL on microscopy shows characteristic perinodular hydropic degeneration,[1] so it has to be differentiated from leiomyoma showing this change. The exophytic bulbous smooth muscle component, grossly resembling placental tissue protrudes from the uterine surface. The exophytic mass is usually in continuity with an intramural component dissecting the surrounding myometrium.[1] Intraoperative impression can be of a malignant neoplasm, especially leiomyosarcoma due to infiltrative growth pattern and softer consistency.[1] On microscopy, there is no atypia or increase in mitosis and tumor has low Ki-67 proliferative index.[1] Grossly as they resemble malignant tumors and differential diagnosis might be problematic, both gynecologic surgeons and pathologists should be well aware of these leiomyoma variants. Cotyledonoid leiomyoma has a distinctive gross appearance which usually raises the suspicion of sarcoma. Intraoperative frozen section is a helpful procedure to avoid overtreatment of such cases.[5]


  Conclusion Top


CDL is very rare benign uterine tumor. Intraoperatively, the worrisome appearance of gross specimen is often mistaken for malignancy leading to overtreatment. If awareness of this entity is there unnecessary surgical procedure (TAH+BSO) can be avoided in reproductive age group.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Acknowledgment

The authors would like to thank Dr. Prateek Ladha for helping in preparing the case report.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Ersöz S, Turgutalp H, Mungan S, Güvendı G, Güven S. Cotyledonoid leiomyoma of uterus: A case report. Turk Patoloji Derg 2011;27:257-60.  Back to cited text no. 1
    
2.
El-Agwany AS. Sternberg tumour of the uterus (Cotyledonoid uterine leiomyoma): A rare benign uterine smooth muscle tumour with suspicious appearance. Surg J 2013;8:4-5.  Back to cited text no. 2
    
3.
Smith CC, Gold MA, Wile G, Fadare O. Cotyledonoid dissecting leiomyoma of the uterus: A review of clinical, pathological, and radiological features. Int J Surg Pathol 2012;20:330-41.  Back to cited text no. 3
    
4.
Roth LM, Reed RJ, Sternberg WH. Cotyledonoid dissecting leiomyoma of the uterus. The sternberg tumor. Am J Surg Pathol 1996;20:1455-61.  Back to cited text no. 4
    
5.
Saeed AS, Hanaa B, Faisal AS, Najla AM. Cotyledonoid dissecting leiomyoma of the uterus: A case report of a benign uterine tumor with sarcomalike gross appearance and review of literature. Int J Gynecol Pathol 2006;25:262-7.  Back to cited text no. 5
    


    Figures

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



 

Top
 
 
  Search
 
Similar in PUBMED
   Search Pubmed for
   Search in Google Scholar for
 Related articles
Access Statistics
Email Alert *
Add to My List *
* Registration required (free)

 
  In this article
Abstract
Introduction
Case Report
Discussion
Conclusion
References
Article Figures

 Article Access Statistics
    Viewed51    
    Printed4    
    Emailed0    
    PDF Downloaded10    
    Comments [Add]    

Recommend this journal


[TAG2]
[TAG3]
[TAG4]