Volume 2 Issue 4, April 2015


Parulekar SV

Congenital malformations of the female genital tract have fascinated gynecologists owing to both their complexity and their effects on the menstrual and reproductive functions of the woman. Some of them are treatable very easily and the results are quite satisfactory, as in the case of an imperforate hymen. Others do not have good treatment, such as a bicornuate uterus. Some others have multiple forms of treatment, none of them perfect, as in the case of cervical atresia. Some of them have no treatment, as in the case of mullerian agenesis. Science is evolving, and new forms of treatment continue to be found for conditions which had no treatment in the past. A woman with mullerian agenesis could not have a baby in the past. It became possible with in vitro fertilization and embryo transfer to a surrogate mother. Now a uterine transplant has also become possible. While science is making progress, one would expect everyone to work in that direction. However gynecologists continue to work on aspects that one would expect to have been settled long ago. Classification of various malformations of the female genital tract was done quite well by Jones. Jones’ classification was the most basic and widely used one. We learned it as students three and a half decades ago, and used it for these many years thereafter. Division of the malformations into three groups: agenesis, lateral fusion defects (obstructive and non-obstructive or symmetrical and asymmetrical), and vertical canalization defects (obstructive and non-obstructive) was clear, informative, explained the embryological basis, and implied the type of treatment required. Despite availability of such good classification, a large number of classifications evolved, developers of each one finding deficiencies with the previous ones. Buttram et al, Jarcho, Fenton and Singh, American Fertility Society, Semmens, Oppelt et al, Acién, and European  Society  of  Human  Reproduction  and  Embryology produced their own classifications. In fact, American Fertility Society produced two of them, one for mullerian anomalies and one for uterovaginal anomalies. None of them could explain the need to change over from the established Jones’ classification convincingly. None of them was comprehensive, unambiguous, and easy to use. None of them made any impact on the mode of management of these cases or prognosis, because the treatment and prognosis would not vary depending on into which class and subclass a particular malformation was placed. In this issue we have a new classification of the malformations of the female genital tract, that is said to be comprehensive, unambiguous, precise, and easy to use. A clinical study is in progress comparing this classification to the older classifications. The results of that study and of other studies inspired by this article may decide if this classification gains wide acceptance.

Large Bilateral Mucinous Cystadenoma Of Ovary

Author Information

More V*, Warke H**, Mayadeo NM***, Satia MN***.
(* Assistant Professor, ** Associate Professor, *** Professor. Department of Obstetrics and Gynecology, Seth GS Medical College & KEM Hospital, Mumbai, India)


Mucinous cystadenoma accounts for 8-10% of all the epithelial ovarian tumors. Large benign ovarian tumors are rare due to general awareness and improved diagnostic technology. We report a case of a 30 year old woman who presented with a large ovarian cyst. Exploratory laparotomy with total abdominal hysterectomy and bilateral salphinogoophorectomy was performed. Final histopathology report was suggestive of bilateral mucinous cystadenoma of the ovary.


Mucinous cystadenoma is a benign ovarian tumor. It arises from the surface epithelium of the ovary. Mucinous cystadenoma accounts for 8-10% of all the epithelial ovarian tumors. Eighty percent of the mucinous tumors are benign and only ten percent of them are bilateral. They are commonly seen in the reproductive age. They may attain enormous size filling the entire abdomen and may cause pressure symptoms. Prognosis is good after surgical debulking.

Case Report

A 30 year old woman, married for 14 years but separated from husband for 12 years, came to the gynecology outpatient department with complaints of abdominal distension for 8 months and decreased appetite and breathlessness for 2 months. Ulltrasonography from a private hospital was suggestive of a large complex cystic mass of 20x16x19cm with mild free fluid in the abdomen with gross hydronephrosis and hydroureter.  There was no significant medical and surgical illness. On examination her general condition was fair and vital parameters were stable.  She was poorly built and nourished with body weight of 51 kg. Systemic examination revealed no abnormality. On abdominal examination there was a cystic mass filling the entire abdominal cavity extending up to the xiphisternum. There was another separate cystic mass or lobulation of the same cyst in right hypochondriac and subcoastal region extending laterally and posteriorly for around 15 cm. Computerized tomography of abdomen and pelvis showed two different cystic multiloculated masses occupying the entire abdominal cavity, gross hydronephrosis and hydroureter, with no evidence of ascites. Tumor markers including serum LDH, Ca 125, AFP, CEA, beta hCG were normal. Ca 19-9 was 1234.75 u/ml, which was markedly raised. Complete hemogram, blood sugars, liver function tests, renal function tests, urinalysis, chest radiograph and electrocardiogram were within normal limits. Preoperative ureteric stenting was done. Exploratory laparotomy was performed under epidural and spinal anesthesia. Abdomen was opened with a midline vertical incision which was extended10-12 cm above the umbilicus. Mild ascites was present. Peritoneal fluid was sent for cytology. Intraoperatively bilateral large ovarian cysts were noted, both well capsulated without any surface invasion. Left ovarian cyst measured 28x12x10 cm and right ovarian cyst measured 23x8x10 cm. Bilateral salphingoophorectomy was done and the specimen sent for frozen section. The left ovarian cyst weighed 10 kg and the right ovarian cyst weighed 7 kg. Frozen section report was inconclusive for malignancy. In view of bilateral large ovarian masses with ascites total abdominal hysterectomy and omentectomy were done. Liver, spleen and bowels were normal. Post operative course was uneventful. Final histopathology report was suggestive of a benign mucinous cystadenoma. Cytology of peritoneal fluid was negative for malignant cells. The ureteric stents were removed   postoperatively. Postoperatively the patient weighed 33 kg.

Figure 1: Black arrow shows right ovarian cyst, yellow arrow shows left ovarian cyst and green arrow shows uterus.


Ovarian tumors are divided into four major categories: epithelial tumors, germ cell tumors, sex-cord-stromal tumors and metastatic tumors. Mucinous cystadenomas are epithelial tumors which are cystic and have loculi lined with mucin secreting epithelium. Mucinous cystadenomas represent 8-10% of all epithelial ovarian tumors and 15% of all ovarian tumors.[1,2] The tumors are bilateral in only 10% of cases which was the case in our patient.[3] They are further divided into benign, borderline and malignant and have a tendency to attain huge sizes filling the entire abdominal cavity. In our case, both cysts were huge, left sided ovarian cyst measuring 28x12x10 cm weighing 10 kg and right sided ovarian cyst measuring 23x8x10 cm weighing 7 kg. On gross appearance, they are characterized by cysts of variable sizes without surface invasion. They are more common in reproductive age group.[4] Patients present with gradually increasing mass in the abdomen and dull aching pain. When the tumor is big enough to fill the entire abdomen, then it may lead to cardiorespiratory embarrassment or gastrointestinal symptoms. Ultrasonography, computerized tomography and magnetic resonance imaging help in evaluation of these tumors. Management of the ovarian cysts depends on the patient’s age, the size of the cyst and histopathology. In benign lesions, ovarian cystectomy or salphingooophorectomy is adequate.[3] But in our case both the cysts were huge destroying the entire ovarian tissue with presence of ascites and malignancy could not be ruled out on frozen section. Hence total abdominal hysterectomy with bilateral salphingo-oophorectomy with omentectomy was performed. Postoperatively the patient was counseled regarding occurrence of menopausal symptoms and the need of hormone replacement therapy.

  1. Vizza F, Galati GM, Corrade G, Atlante M, Infante C, Sbiroli C. Voluminous mucinous cystadenoma of the ovary in a 13-year-old girl. J Ped Adoles Gynecol. 2005: 18(6):419-422.
  2. Mittal S, Gupta N, Sharma A, Dadhwal V. Laprascopic management of a large recurrent benign mucinous cystadenoma of the ovary. Arch Gynecol Obstet. 2008; 277(4): 379-380.
  3. Alobaid AS. Mucinous cystadenoma of the ovary in a 12-year-old girl. Saudi Med J. 2008;29(1):126-128.
  4. Ioffe OB, Simsir A, Silverberg SG. In: Practical Gynaecologic Oncology. Berek JS, Hacker NF, editor. Lippincott Williams and Wilkins Company; 2000. Pathology; pp. 213-214.

More V, Warke H, Mayadeo NM, Satia MN. Large Bilateral Mucinous Cystadenoma Of Ovary. JPGO 2015. Volume 2 No. 4. Available from: http://www.jpgo.org/2015/04/large-bilateral-mucinous-cystadenoma-of.html

Cesarean Scar Ectopic Pregnancy: Diagnostic and Therapeutic Dilemmas

Author Information

Jagtap V*, Samant P**, Valvi D***, Parulekar SV****.
(*Second year Resident, **Additional Professor, ***Assistant Professor, **** Professor and Head. Department of Obstetrics and Gynecology, Seth G. S. Medical college and KEM hospital, Mumbai, India)


Though considered rare, the incidence of cesarean scar ectopic pregnancy (CSEP) is rising with increasing incidence of cesarean sections. Five fold increase is reported in ten years from year 2000. Even with good transvaginal ultrasound (TVS), the pregnancy may be misdiagnosed as cervical ectopic. We report a case of CSEP with failed medical management, later managed with surgical excision.


Incidence of CSEP in the literature is around 1:1800 to 1:2000 and increasing due to liberal use of cesarean section.[1,2,3] CSEP poses a therapeutic challenge and risk in women desirous of child bearing. Suction evacuation (S&E) can pose dual threat of hemorrhage and perforation. Options like sonographically guided S&E and laparoscopic management are heavily dependent on the surgeon’s skill. Medical method is limited by embryonic age and Beta Human Chorionic Gonadotropin (b HCG) titers and prolonged time of observation for titer decline. Failure of resolution warrants rescue surgical intervention.[4]

Case Report

27 years old, gravida 2, para 1 with previous cesarean section and two months’ amenorrhea was referred with painless vaginal bleeding for 2 days and sonography suggesting cervical ectopic pregnancy. She was hemodynamically stable. On speculum examination the external os was closed, there was no bleeding and the cervix was unremarkable. Bimanual examination was deferred so as not to disturb cervical ectopic pregnancy. Her TVS prior to admission showed gestational sac of diameter 2.6 cm with live embryo corresponding to 7.6 weeks in the cervical canal. Internal os was reported open. Biochemical tests for fitness for anesthesia were normal. TVS at our center confirmed a live pregnancy at the cesarean scar site, while the cervix was normal. Her serum b HCG on admission was- 81616 mIU/ml. The patient was counseled about therapeutic options. She opted for multidose methotrexate (MTX) which was given with alternate day folinic acid rescue.
Her serial b HCG values were as follows.

Serum b HCG mIU/ml

TVS on day18 showed 9.6 weeks’ viable fetus.  Magnetic Resonance Imaging (MRI) was performed. It showed 4.1x 4.7x3.7 cm gestational sac implanted within anterior part of lower segment of uterus, with no myometrial tissue between the sac and the bladder. Uterine fundus was empty. Endometrial thickness was 1.7 cm. Based on MRI and TVS reports, and a rise in serum b HCG level despite treatment, a laparotomy was undertaken. The uterus was bulky. The anterior wall of the isthmus was distended and vascular. The isthmic area was transversely incised away from the bladder, the sac and placenta were removed, uterus was curetted retrogradely, the scar was excised and edges were sutured. The products were sent for histopathological examination. Distance of external os from the incision was 3.5 cm. The patient made an uneventful recovery. Histopathology of the specimen confirmed the diagnosis of cesarean scar ectopic pregnancy.

Figure1. Transvaginal ultrasonogram suggestive of cervical ectopic pregnancy.

Figure 2. MRI image of scar ectopic pregnancy with absent myometrium between the sac and the bladder.

Figure 3. Distended vascular anterior wall of the isthmus.

Figure 4. Normal undistended posterior cervical wall


Rotas et al defined CSEP as the one located outside the uterine cavity with myometrium and fibrous scar tissue surrounding it completely.[2] Vial et al divided the scar implantations in two groups based on their growth either in cervico-isthmic space or into the deeper myometrium and proposed criteria for the same.[5] Our case fits in the second category which grew into the muscle and was likely to rupture. Tsai et al proposed clubbing early lower segment aberrant implantations as ‘Low-lying-implantation ectopic pregnancy’ for ultrasound diagnosis as there was no significant difference in operation time, blood loss and success in their cohort of forty two cases.[6] The most common presentation is painless vaginal bleeding and hemorrhage from uterine rupture can be fatal. In a review of more than seven hundred cases of CSEP Timor-Tritsch et al found that diagnosis was missed in more than 13% cases. [7] MRI is helpful in making a diagnosis if TVS is inconclusive.[8] Medical management has been done with local or systemic MTX therapy or Local embryocide injection like potassium chloride.[7] With advanced gestational age and serum b HCG levels in excess of 5000 mIU/ml, systemic MTX alone is not effective and intrasac MTX or other additional therapy is required.[9] Curettage has been found to have the highest complication rate.[7] Jurkovic et al reported 71% success with medical management in early SCEPs.[3] Intrasac instillation of potassium chloride or MTX had the least complication rate in the series reviewed by Timor-Tritsch et al.[7] Hysterectomy is the last resort in cases complicated with life threatening hemorrhage. In conclusion apparent cervical ectopic pregnancy may actually be a scar ectopic implantation in cases of previous cesarean section and careful review is required to avoid scar rupture.
  1. Wu X, Xue X, Wu X, Lin R, Yuan Y, Wang Q, Xu C, He Y, Hu W. Combined laparoscopy and hysteroscopy vs. uterine  curettage in the uterine artery embolization-based  management of cesarean scar  pregnancy: a cohort study: Int J Clin Exp Med 2014;7(9):2793-2803.
  2. Rotas MA, Haberman S, Levgur M, Cesarean scar ectopic pregnancies, etiology, diagnosis and management, Obstet Gynecol 2006;107:1373-81.
  3. Jurkovic D, Hillaby K, Woelfer B, Lawrence A, Salim R, Elson CJ. First-trimester diagnosis and management of pregnancies implanted into the lower uterine segment cesarean section scar. Ultrasound Obstet Gynecol 2003;21:220-227.
  4. Timor-Tritsch IE, Monteagudo A, Santos R, Tsymbal T, Pineda G, Arslan AA. The diagnosis, treatment, and follow-up of cesarean scar pregnancy. Am J Obstet Gynecol 2012; 207:44. e1-13.
  5. Vial Y, Petignat P, Hohlfeld P. Pregnancy in a caesarean scar. Ultrasound Obstet Gynecol 2000:16:592-3.
  6. Tsai SW, Huang KH, Ou YC, Hsu TY, Wang CB, Chang MS, Li KH, Kung FT, Low-lying-implantation ectopic pregnancy: a cluster of cesarean scar, cervico-isthmus, and cervical ectopic pregnancies in the first trimester. Taiwan J Obstet Gynecol. 2013 Dec;52(4):505-11.
  7. Timor-Tritsch IE, Monteagudo A. Unforeseen consequences of the increasing rate of cesarean deliveries: early placenta accreta and cesarean section scar pregnancy; a review. Am J Obstet Gynecol 2012;207:14-29.
  8. Wu R, Klein MA, Mahboob S, Gupta M, Katz DS. Magnetic Resonance Imaging as an Adjunct to Ultrasound in Evaluating Cesarean Scar Ectopic Pregnancy. J Clin Imag Sci 2013;3:1-5. 
  9. Ash A, Smith A, Maxwell D. Caesarean scar pregnancy. BJOG 2007; 114:253–263.

Jagtap V, Samant P, Valvi D, Parulekar SV. Cesarean Scar Ectopic Pregnancy: Diagnostic and Therapeutic Dilemmas. JPGO 2015. Volume 2 No. 4. Available from: http://www.jpgo.org/2015/04/cesarean-scar-ectopic-pregnancy.html

Low Grade Endometrial Stromal Sarcoma: Unusual Gross Appearance

Author Information

Ansari M*, Parulekar SV**.
(*Assistant Professor, ** Professor and Head, Department of Obstetrics and Gynecology, Seth GS Medical College & KEM Hospital, Mumbai, India.)


Low grade endometrial stromal sarcoma is not a very common tumor of the uterus. Clinically it is mistaken for a leiomyoma of the uterus. Its gross appearance may be suggestive of its diagnosis. We present a case in which its gross appearance was quite unusual.


Low grade endometrial stromal sarcoma is often mistaken for a leiomyoma of the uterus on clinical examination and on inspection of the external surface of the uterus during a laparotomy. The gross appearance of its cut surface may be suggestive of its diagnosis. But usually the specimen is not opened during the laparotomy and the diagnosis may be missed. The diagnosis is usually made after histopathological examination of the specimen. We present a case in which the gross appearance of the external surface of the uterus with a low grade endometrial stromal sarcoma was quite unusual.

Clinical Features

A 44 year old married woman, para 2 abortion 4, presented with menorrhagia for six months. Her past menstrual cycles were regular, and the flow was moderate. Her past, medical and surgical history were not contributory. General and systemic examination findings were normal, except for moderate anemia. Abdominal and bimanual pelvic examination showed the uterus was enlarged to 18 weeks’ size, globular, and with irregular upper surface. A diagnosis of uterine leiomyoma was made. Ultrasonography showed a well defined 10x8x8.5 cm sized mass with heterogenous echotexture in the uterine fundus with minimal vascularity, suggestive of a non-pedunculated leiomyoma. There was another 4.8x3.7x3.5cm sized lesion with similar appearance in the anterior wall of the uterus. Pap smear and endometrial aspiration cytology did not show any evidence suggestive of a malignancy. After correction of her anemia with transfusion of packed red cells and confirming that she was fit to undergo an operation, an exploratory laparotomy was performed. The findings are shown in figure 1. An abdominal hysterectomy and bilateral salpingo-oophorectomy was done. The patient made an uneventful recovery.

Figure 1. The uterus is enlarged. Three nodules with smooth external surface are seen over the uterine fundus anteriorly. A large vein is seen between two of these nodules.

Gross examination showed a uterus measuring 18x9x8 cm, with three small firm sessile masses measuring 3x2x2 cm on the anterior fundal wall. There was a large engorged vein running across anterior wall in between two of these masses. Cut surface showed involvement of the entire thickness of the myometrium by the tumor including the fundal nodules, but the serosa was intact. The mass extended into the endometrial cavity. Histopathology of the tumor showed tongues and sheets of neoplastic endometrial stromal cells in the full thickness of the myometrium. The serosa was intact. The tumor cells were oval shaped with basophilic mildly atypical nuclei and scanty cytoplasm. There was a prominent delicate arborizing vasculature throughout the tumor. Whorling of the neoplastic cells was seen around the arterioles. Two or three mitoses were seen per 10 HPF. There was no necrosis. A diagnosis of low grade endometrial stromal sarcoma was made.


Low grade endometrial stromal sarcoma is not a very common tumor of the uterus. Clinically it is mistaken for a leiomyoma of the uterus. Its gross appearance may be suggestive of its diagnosis. It may be seen as a submucous polyp, or an intramyometrial mass. The classical gross appearance of an intramyometrial mass is either a single nodule, multiple solid-cystic masses, or a poorly demarcated solid-cystic lesion.[1] About 50% cases are well circumscribed. Cut surface of the uterus shows worm-like masses penetrating the myometrium diffusely. Extrauterine extension is seen in 30% of cases. The diagnosis is usually made after histopathological examination.[2,3] Most of the tumors involve the endometrium too.[4]
Our case was unusual in that the tumor had grown as nodules on the uterine surface, but the serosa of these nodules was intact. A large vein between two of these nodules suggested vascularity, that might be due to underlying malignancy, though surface veins are known to occur with uterine subserous leiomyomas too.[5] Such an appearance has not been described in the literature before.


In such cases of unusual presentation of leiomyoma, one should consider possibility of endometrial stromal sarcoma, even though endometrial aspiration cytology or histopathology of curetted material does not show any malignancy. 

  1. Pekindil G, Tuncyurek O, Orguc S, Inceboz U, Kandiloglu AR, Caglar H. A case of endometrial stromal sarcoma with curvilinear calcification. Gynecologic Oncology 2005, 98:318-321.
  2. Mesia AF, Demopoulos I. Effects of leuprolide acetate on low-grade endometrial stromal sarcoma. Am J Obstet Gynecol 2000;182:1140-1141.
  3. Husseiny GE, Bareedy NA, Mourad WA, Mohamed G, Shoukri M, Subhi J. Prognostic factors and treatment modalities in uterine sarcoma. Am J Clin Oncol 2002, 25:256-260.
  4. Berkowitz RS, Goldstein DP. Uterine cancer. In Berek JS, Hacker NE, editors. Practical Gynecologic Oncology. 4th ed. Philadelphia Williams & Wilkins; 2005:431-432.
  5. Mirchandani A, Parulekar SV, Dalvi P. Hemoperitoneum from Ruptured Leiomyoma. JPGO 2014 Volume 1 Number 10. Available from: http://www.jpgo.org/2014/10/hemoperitoneum-from-ruptured-leiomyoma.html

Ansari M, Parulekar SV. Low Grade Endometrial Stromal Sarcoma: Unusual Gross Appearance. JPGO 2015. Volume 2 No. 4. Available from: http://www.jpgo.org/2015/04/low-grade-endometrial-stromal-sarcoma.html

Wandering Fimbria Of A Fallopian Tube


Raut DP *, Parulekar SV**, Valvi D***.
(* First Year Resident, ** Professor and Head, ***Assistant Professor, Department of Obstetrics and Gynecology, Seth GS Medical College & KEM Hospital, Mumbai, India.)


This paper presents incidental finding of spontaneously detached fimbria of fallopian tube implanted over the right uterosacral ligament in 32 year old woman   during  laparoscopic tubal ligation. This is the first case of this occurrence in the English world literature. Possible etiology of this condition is discussed.


Fallopian tube is attached to the cornu of the uterus, one on each side. It lies in a free fold of the broad ligament called the mesosalpinx. Its lateral or fimbrial end is free, below which lie the ovarian vessels which pass into the infundibulopelvic ligament. Detachment of a part or whole of the fallopian tube may be during embryological development, due to a number of causes in the childhood or adult life, or as a result of surgery. Except the surgically detached tube, others do not survive and are resorbed. We present a case in which there was spontaneous detachment of the fallopian tube, followed by its reimplantation over the uterosacral ligament of the same side. This is the first case of this occurrence in the English world literature.

Case Report

A 32 year woman, married for 12 years, Gravida 4 Para 3 Living 3 came to outpatient department for a medical termination of pregnancy and a sterilization operation. There was a history of a failure of male barrier contraception. There was no history any past medical or surgical illness, abdominal trauma, pelvic tuberculosis or tuberculosis in the past. She had not undergone any abdominal surgery in the past. Her three deliveries had been normal and uneventful. No abnormality was detected on general and systemic examination. Abdominal examination showed normal findings. Bimanual pelvic examination showed a retroverted uterus of 8 weeks’ size. All fornices were clear. Result of all investigations required for fitness for anesthesia were normal. A medical termination o pregnancy was done by rapid cervical dilatation and manual vacuum aspiration. Laparoscopy was performed to do tubal ligation by silastic band application. The uterine size, shape and surface were normal. Both  ovaries and the left fallopian tube were normal. The right fallopian tube ended blindly at the ampullary- infundibular junction. Normal-appearing fimbria were found attached firmly to the area lateral to the uterine end of the right uterosacral ligament (figure 1 and 2). No other abnormality detected. One silastic band was applied to each fallopian tube in the isthmic area. The right fimbria were left undisturbed. The patient tolerated the procedure well. Postoperative course is uneventful.

Figure 1. Laparoscopic view of the pelvic cavity. The fimbria of the right fallopian tube are pointed out by arrows.

Figure 2. Close up view of the detached fimbria of the right fallopian tube attached o the pelvic peritoneum.


The commonest cause of absence of a part or whole of a fallopian tube is iatrogenic – surgical removal for sterilization or disease like an ectopic pregnancy. Such a condition may also be due to a tubal ectopic pregnancy which undergoes resorption after local rupture. Torsion of a fallopian tube may occur due to presence of a hydrosalpinx, pelvic tumor, pelvic infection, or ectopic pregnancy causing a hematosalpinx.[1,2,3,4,5,6,7] Sometimes only the fallopian tube is lost, while at other times the ovary is also lost. The woman may have experienced acute abdominal pain in the past, which subsided over a few days with pain killers and observation. In such cases the condition comes to light much later when a laparoscopy or laparotomy is done for any unrelated condition. Developmental anomalies of part  of the mesonephric and paramesonephric ducts may be responsible for congenital absence of a part of the fallopian tube.[8,9,10] Detachment of a part or whole of the fallopian tube, followed by its reattachment to another part of the pelvis is not known to occur.
In our case, the right tube was found to be blocked at the ampullary-infundibular junction. The fimbria were separate from the fallopian tube and were attached firmly to the peritoneum lateral to the uterine end of the right uterosacral ligament. The most likely explanation of this occurrence is that the tube must have undergone torsion due to a hematosalpinx, and got adherent by its fimbrial end to the pelvic peritoneum lateral to the uterine end of the right uterosacral ligament, in order to receive blood supply. The twisted portion must have undergone necrosis, and the medial part of the tube must have receded to its original position, while the fimbria remained adherent. We left he spontaneously detached and reimplanted fimbria were left undisturbed as their removal would serve no purpose, and leaving them behind would not lead to any complication.
We have presented this case to make others aware of this condition, so that they can diagnose it when they encounter such a case themselves.

  1. Furui T, Imai A, Yokoyama Y, Tamaya T. Hematosalpinx and torsion of the fallopian tube in a virgin girl. Gynecol Obstet Invest 1993;35:123-5.
  2. Sorem KA, Bengtson JM, Walsh B. Isolated fallopian tube torsion presenting in labor. A case report. J Reprod Med 1991;36:763-4.
  3. Gupta A, Parulekar SV. Silent autoamputation of an adnexa. Bom Hosp J 1996;38(4):900-02.
  4. Rezvani M, Shaaban AM Fallopian tube disease in the Non pregnant Patient .Radio graphics. 2011;31:527–48.
  5. Bernardus RE, Van der Slikke JW, Roex AJ, Dijkhuizen GH, Stolk JG. Torsion of the fallopian tube: some considerations on its etiology. Obstet Gynecol 1984;64:675-8.
  6. Maynard SR, Peipert JF, Brody JM. Tubal torsion appearing as acute pelvic inflammatory disease. J Am Assoc Gynecol Laparosc 1996;3:431-3.
  7. Hansen OH. Isolated torsion of the Fallopian tube. Acta Obstet Gynecol Scand 1970;49:3-6.
  8. Paternoster DM, Costantini W, Uglietti A, Vasile C, Bocconi L. Congenital or torsion-induced absence of Fallopian tubes. Two case reports. Minerva Ginecol. 1998 May;50(5):191-4.
  9. A case of congenital unilateral partial absence of fallopian tube. Yazawa H1, Yabe M, Endo S, Hayashi S.Department of Obstetrics and Gynecology, Fukushima Red Cross Hospital, Fukushima, Japan. ikyoku12@fukushima-med-jrc.jp; Fukushima J Med Sci. 2010;56(1):44-9.
  10. Congenital absence of fallopian tube and ovary. Eustace DL Department of Obstetrics and Gynaecology, St Thomas's Hospital, London, UK. Eur J Obstet  Gynecol  Reprod Biol. 1992 Sep 23;46(2-3):157-9.

Raut DP, Parulekar SV, Valvi D. Wandering Fimbria Of A Fallopian Tube. JPGO 2015. Volume 2 No. 4. Available from: http://www.jpgo.org/2015/04/wandering-fimbria-of-fallopian-tube.html

Successful Obstetric Outcome in a Case of Uterus Bicornis Partial Bicollis

Author information

Tanksale SJ*, Parulekar SV**,  Samant PY***.
(* Fourth Year Resident, ** Professor, *** Associate Professor, **** Assistant Professor. Department of Obstetrics and Gynecology, Seth GS Medical College & KEM Hospital, Mumbai, India.)


Amongst the Mullerian duct anomalies,  a bicornuate uterus is a common anomaly. Bicornuate uterus has two Uterine cavities with single cervix. In this case report,  we have a patient with Bicornuate uterus with single cervix but with partial fusion defect. The previous missed abortion was in one horn and next pregnancy in other horn.


Uterine malformation occur as a result of fusion defect in Mullerian duct development during embryogenesis. Bicornuate uterus is one of the common Mullerian duct anomaly. The fusion process of upper part of Mullerian duct is affected and as a result caudal part is fused normally whereas the cephalic part is bifurcated. Mullerian duct anomalies are associated with miscarriage, preterm labor and malpresentations. Pregnancies in a bicornuate uterus are therefore considered as high-risk due to their association with poor reproductive outcome and require extra monitoring.

Case Report

A 28 year old gravida 3 with no living issues with previous one neonatal death and one medical termination of pregnancy (MTP) was registered at our hospital at 22 weeks. She had one preterm delivery 3 years ago. That baby died in neonatal period. She had undergone one first trimester MTP 2 years ago, which had not been successful. So she was subjected to a repeat procedure at a municipal peripheral hospital, which was abandoned due t creation of a false passage, and she was referred to this hospital. She was found to have the missed abortion in the right horn of a bicornuate uterus. An attempt to enter the horn under laparoscopic control failed, a false passage was created between the two horns, and a hysterotomy had to be performed to remove the dead fetus. Two years later she conceived again and registered with us at 22 weeks. She had an ultrasonography (USG) report of 19 weeks gestation with pregnancy in left horn of a bicornuate uterus. Both the kidneys were normal. She had regular follow up in the antenatal clinic. At 33 weeks of gestation, she was admitted with preterm labor and complaints of leaking per vaginum. There was no evidence of leaking per vaginum and preterm labor was controlled with nifedipine. She was given dexamethasone for fetal lung maturity. USG showed evidence of intrauterine growth restriction (IUGR) of he fetus. She was observed in antenatal ward with fetal surveillance. At 35 weeks, USG and obstetric Doppler was performed which showed IUGR with fetal parameters corresponding to 29 weeks and fetoplacental insufficiency. An emergency lower segment cesarean section was performed, delivering the baby weighing 1800 g through a lower segment transverse incision taken on the left horn. The right horn measured 10 cm in length, was of normal shape, and did not show any scar or adhesions (figure 1). The separation of the two horns was up to the level of middle of the cervix, such that there was a single external os, and a separate internal os for each horn. The APGAR scores of the baby were of 9 at 1 and 5 minutes.

Figure 1. Operative findings during cesarean section.


Antenatal diagnosis of a mullerian anomaly is important so as to ensure appropriate management. A study by Hua et al. found that the presence of a maternal uterine anomaly detected at the time of anatomic survey is associated with an increased risk of preterm birth, preterm premature rupture of membranes, breech presentation, and cesarean section.[1]  American Congress of Obstetrics and Gynecology guidelines for management of IUGR recommend serial biomery by USG for monitoring growth of the fetus in pregnancies complicated by maternal mullerian anomalies, to screen for IUGR.[2] Raga et al found that the chances of having a term pregnancy were 60%, with a take-home baby rate of 62.5%.[3] These percentages disagree with previous reports on a poor reproductive outcome associated with a bicornuate uterus. There are some reports which found the poorest prognosis when the defect was partial rather than complete. Bicornuate uterus has single cervix and two uterine cavities. In this case, there was single external os and a separate internal os for each horn. This could explain the failed attempt to evacuate uterine cavity in previous missed abortion. Behavior of such a cervix during labor has not been reported in literature.


When a mullerian anomaly is identified, the woman should be counseled about reproductive prognosis and adverse pregnancy outcome. Though management guidelines exist for most of the mullerian anomalies, some cases require individualized management.

  1. Hua M, Odibo AO, Longman RE, Macones GA, Roehl KA, Cahill AG. Congenital uterine anomalies and adverse pregnancy outcomes. Am J Obstet Gynecol 2011;205:558.e1-5.
  2. American Congress of Obstetrics and Gynecology Committee on Practice Bulletins-
  3. Obstetrics. ACOG practice bulletin: intrauterine growth restriction. Obstet Gyncol 2000;
  4. 95(Suppl):1-12
  5. Raga F, Bauset C, Remohi J, Bonilla-Musoles F, Simon C, Pellicer A. Reproductive impact of congenital Mullerian anomalies. Human Reproduction 1997;12(10):2277–2281.

Tanksale SJ, Parulekar SV,  Samant PY. Successful Obstetric Outcome in a Case of Uterus Bicornis Partial Bicollis. JPGO 2015. Volume 2 No. 4. Available from: http://www.jpgo.org/2015/04/successful-obstetric-outcome-in-case-of.html


Author Information

Agrawal S*, Parulekar SV**
(* Second Year Resident, ** Professor and Head, Department of Obstetrics and Gynecology, Seth G.S Medical College and KEM Hospital, Mumbai, India.)


Phocomelia is an uncommon congenital malformation of the fetus, after the ban on the use of thalidomide during pregnancy.


The word phocomelia is derived from the Greek words ‘phoco’ meaning seal and ‘melia’ meaning limb. Phocomelia is a very rare congenital disorder involving the upper, lower or all limbs. It is characterized by extremely shortened or even absent proximal and mid portions of the limbs. The fingers may be fused together. In extreme cases the hands and feet may be directly attached to the trunk (tetraphocomelia). Its causes include intrauterine exposure to thalidomide, autosomal recessive inherited condition or sporadic change in the gene, Roberts syndrome, Grebe syndrome, von Voss-Cherstvoy syndrome, and Schinzel-phocomelia syndrome. We present a case of second trimester abortion in which the fetus had phocomelia.

Case Report

A 30 year old woman, married for 10 years (non consanguineous), gravida 5, para 2, living 2, spontaneous abortion 3 with 5 months of amenorrhea, was referred from a private maternity home in view of ultrasonography suggestive of intrauterine fetal demise of 18 weeks and with a cystic hygroma at the neck. She was treated with tablet misoprostol 800 μg per vaginally for termination of pregnancy at the private nursing home, but was later on referred to our hospital in view of platelet count of 95000/cubic mm. Her obstetric history was as follows.
G1: female child of 8 years,full term normal delivery at home.
G2: female child of 6 years,full term normal delivery at home.
G3: spontaneous abortion at 2 months amenorrhea, check curettage done.
G4: spontaneous abortion at 2 and 1/2 months amenorrhea, check curettage done.
G5: spontaneous abortion at 3 months amenorrhea,check curettage done
G6: present pregnancy
Her hemoglobin was 14 g/dl, white cell count 16,000/cmm, platelet count 136000/cmm, blood urea nitrogen 8.7 mg%, serum creatinine 0.7 mg%, fasting blood sugar 97 mg%, post lunch blood sugar 105 mg%, VDRL negative, aPTT-31.5 sec(T), 30 sec(C), lupus Anticoagulant/anti cardiolipin antibodies absent, T3-101, T4-10.2, and TSH 4.4 μIU/ml. Endocrinological reference was done. No intervention was advised for elevation of TSH. Abdominopelvic ultrasonography showed intrauterine fetal death at 18 weeks and a cystic hygroma. The process of termination continued and patient aborted with three more doses of tablet misoprostol 400 μg every 4 hours. The abortus was found to have short limbs, protuberant abdomen, and neck swelling (figure 1). Its sex could not be determined. It weighed 145 g. The placenta weighed 185 g. Fetal biopsy and karyotyping could not be done as relatives were not willing for the same. A clinical diagnosis of phocomelia was made. Placental histopathology showed chorioamnionitis.

Figure 1. Fetus with phocomelia.

Figure 2. Babygram, anteroposterior view.

Figure 3. Babygram, lateral view.


Phocomelia can be caused by intrauterine exposure to thalidomide. In addition, there might be club feet, polydactyly, facial abnormalities, microphthalmia, congenital cardiac anomalies, and intrauterine fetal growth restriction.[1] It was seen mainly from 1957 to 1961, when thalidomide was used to treat nausea and vomiting in pregnancy.[2] The risk of embryopathy due to exposure to thalidomide in the first trimester is 10-50%. However the use of this drug in pregnancy is contraindicated now, and cases of phocomelia due to thalidomide are not seen in modern times. The drug is used for erythema nodosum leprosum, and accidental ingestion of the drug during pregnancy is a possibility. Our patient did not have any history of ingestion of this drug, neither intentionally nor accidentally.
Roberts Syndrome is inherited as an autosomal recessive condition.[3,4] It has intrauterine growth restriction, asymmetrical limb reduction, silvery blonde and sparse scalp hair, microcephaly, hypertelorism, exophthalmos, congenital cataracts, coloboma, hypoplastic nasal alae, midfacial cleft, facial haemangioma, bilateral cleft lip or palate, dysplastic ears, brain malformations, renal malformations, renal dysplasia, cardiac anomalies, genital malformations, and hypogonadism. Our patient had no family history of phocomelia, and the fetus did not have any of the malformations visible externally. A necropsy was not permitted by the parents, so that internal organ anomalies could not be checked.
Grebe syndrome is inherited recessively, caused by a mutation in the gene encoding cartilage-derived morphogenetic protein-1.[5] The axial skeleton is normal, the limbs are severely shortened and deformed (humeri and femora relatively normal, the radii/ulnae and tibiae/fibulae were short and deformed), fused carpal and tarsal bones, many metacarpal and metatarsal bones absent. Our case did not have this syndrome, as the femora and humeri were not normal.
Von Voss-Cherstvoy syndrome is an early lethal multiple congenital anomaly syndrome characterized by phocomelia of upper limbs, variable brain anomalies, encephalocele, vertebral and ribs anomalies, urogenital abnormalities, and thrombocytopenia.[6] Our case had affected lower limbs and did not show any other features of the syndrome.
Schinzel-phocomelia syndrome (limb/pelvis-hypoplasia/aplasia syndrome) shows limb defects due to hypo- or aplasia of long bones unusual facies, pelvic defects, dislocation of hip, hypoplasia of iliac bone, aplasia of pubic bones, thoracic deformity, and genitourinary anomalies.[7] Our case did not show these features.
Phocomelia may be seen in families, the inheritance being autosomal recessive. There was no family history of this disorder in the case presented. It can occur sporadically due to gene mutation. This appears to be the case in our patient.


  1. Lee A, Kratochwil A, Deutinger J et-al. Three-dimensional ultrasound in diagnosing phocomelia. Ultrasound Obstet Gynecol. 1995;5 (4): 238-40.
  2. Strömland K, Miller MT. Thalidomide embryopathy: revisited 27 years later. Acta Ophthalmol (Copenh). 1993;71 (2): 238-45.
  3. Ahmed AA, Imrie S, Duncan R, Tolmie J. Roberts syndrome: facial dysmorphology in a mildly affected case. Clinical dysmorphology 2009;18:236-7.
  4. Goh ES, Li C, Horsburgh S et-al. The Roberts syndrome/SC phocomelia spectrum--a case report of an adult with review of the literature. Am. J. Med. Genet. A. 2010;152A (2): 472-8.
  5. Costa T, Ramsby G, Cassia F, Peters K, Soares J, Correa J, et al. Grebe syndrome: Clinical and radiographic findings in affected individuals and heterozygous carriers. American Journal of Medical Genetics 1998;75:523–529.
  6. Lubinsky MS, Kahler SG, Speer IE, Hoyme HE, Kirillova IA, Lurie IW. von Voss-Cherstvoy syndrome: a variable perinatally lethal syndrome of multiple congenital anomalies. Am J Med Genet 1994;52(3):272-8.
  7. Chitayat D1, Stalker HJ, Vekemans M, Delneste D, Azouz EM. Phocomelia, oligodactyly, and acrania: the Schinzel-Phocomelia syndrome. Am J Med Genet 1993;45(3):297-9.

Agrawal S, Parulekar SV. Phocomelia. JPGO 2015. Volume 2 No. 3. Available from: http://www.jpgo.org/2015/04/phocomelia.html

Hysterotomy For Evacuation Of Products Of Conception In A Bicornuate Uterus

Author Information

Acharya VV, Parulekar SV, Samant PY, Mirchandani AM
(* Ex Fourth Year Resident, ** Professor and Head, *** Additional Professor, **** Assistant Professor. Department of Obstetrics and Gynecology, Seth GS Medical College & KEM Hospital, Mumbai, India)


Mullerian anomalies increase the potential for complications during pregnancy termination. We present an unusual complication of abortion in a patient with a uterus bicornis unicollis.
A laparoscopy was performed, but the pregnant horn could not be entered under laparoscopic guidance since the axis of the right horn was at an angle of 80-90° with that of the cervical canal. Laparotomy and hysterotomy had to be performed in order to complete the abortion.


The fallopian tubes, uterus, the uterine cervix, and the superior part of the vagina develop from the mullerian ducts. The ovaries and lower third of the vagina have different embryological origins derived from germ cells that migrate from the primitive yolk sac and the sinovaginal bulb, respectively.[1] A number of malformations can occur with disruption of this system. They range from agenesis of the uterus and vagina, their duplication, to minor abnormalities of the uterine cavity. Müllerian malformations are often associated with urinary system and axial skeletal anomalies. In fact, mullerian anomalies are often first detected during evaluation of the patients for the associated conditions. These malformations can affect the reproductive outcome adversely, causing abortions and preterm deliveries, especially in women with unicornuate, bicornuate, didelphys and septate uteruses.[2] Later in pregnancy, unsuspected uterine malformations may present as intrauterine fetal growth restriction due to abnormal placentation, or abnormal fetal positioning related to mechanical factors in the shape of the uterine cavity. Labor, delivery, and third stage problems may occur due to incoordinate uterine muscular activity. Another problem associated with such malformations is difficulty in surgical abortion. We present here a case of such difficulty wile performing rapid cervical dilatation and evacuation of the products of conception.

Case Report
A 29 year old woman with 8 weeks of amenorrhea and features suggestive of an incomplete abortion was referred to our centre from a peripheral hospital after two failed attempts at surgical evacuation of the uterine contents. This was her second conception. In the past, she had a previous preterm vaginal delivery at 8 months of gestation, followed by neonatal demise due to extreme prematurity. In the current conception, she had undergone dilatation and curettage at a private nursing home following an ultrasonography (USG) report of an early embryonic foetal demise. But she continued to have bleeding per vaginum. A repeat ultrasonography at a Municipal Hospital revealed retained products of conception for which a repeat curettage was advised. At the time of curettage, the suspicion of creation of a false passage led to abandonment of the procedure. She was explained the need for exploratory laparotomy but she declined and was hence referred to our centre for further management. Her per vaginal examination revealed a six weeks sized uterus deviated to the right. Another USG done at our center raised the possibility of a bicornuate uterus with retained products of conception in the right horn. Medical method of termination with misoprostol was not considered due to the history of failed previous curettage and the chance of rupture of an anomalous uterus.[3] The woman was explained the need for performing the curettage under laparoscopic guidance to avoid any false passage or perforation. Consent was also taken for a hysterotomy if the attempt at laparoscopically guided uterine evacuation failed. After appropriate preoperative work up and keeping adequate blood ready, laparoscopy was performed under general anaesthesia. The finding of a bicornuate uterus with pregnancy in the right horn was confirmed. Under laparoscopic guidance, a few attempts at dilatation of the endocervical canal to enter into the right horn were made but they led only into a false passage, which opened between the two horns under the uterovesical fold of the peritoneum. The axis of the right horn of the uterus was 80-90° with that of the cervical canal. Hence a uterine sound, a cervical dilator, and even a cervical probe could not be negotiated into the horn. Finally, a hysterotomy was performed (figure 1) and the gestational sac was delivered en sac from the right horn. A gentle curettage was done through the hysterotomy incision. Then a retrograde dilatation of the endocervical canal was done, passing cervical dilators down the cervical anal passed through the uterine incision and guided by two fingers placed behind the cervix (figure 2). The uterine incision was closed with a continuous stitch of No. 1 polyglactin. The patient made an uneventful recovery. At the time of discharge, the possibility of operative intervention in future pregnancy in view of hysterotomy done during this conception was explained.

Figure 1. Hysterotomy of the right gravid horn. The gestational sac is being extruded through the uterine incision.

Figure 2. Retrograde cervical dilatation. A cervical dilator is seen exiting from the cervix, passed downward through the hysterotomy incision.


Mullerian anomalies increase the potential for complications during pregnancy termination.[4] Standard procedures of surgical abortion can fail in some of these cases. If the angle between the two horns is wide, an instrument like a uterine sound, cervical probe or dilator passed up through the cervical canal may not enter the uterine cavity early in pregnancy. That would made surgical abortion impossible, or might cause uterine perforation. A hysterotomy should be reserved for such extremely difficult cases when all possible conservative techniques fail. Such a difficulty of alignment of the axis of the uterine horn and that of the cervical canal is usually not encountered during childbirth, owing to globular enlargement of the gravid horn, which comes into line with the cervical canal and the other horn gets displaced to one side. The cervix gets drawn up and effaces and dilated due to uterine contractions, and the problem of nonalignment of the axes of the uterine cavity and cervical canal gets solved.


Conventional approach to surgical abortion can fail in cases of uterine anomaly. A hysterotomy should be reserved for the most difficult and rare instances of such failed abortion.

  1. Chandler TM, Machan LS, Cooperberg PL, Harris AC, Chang SD. Mullerian duct anomalies: from diagnosis to intervention. The British journal of radiology. 2009;82(984):1034–42.
  2. Chan YY, Jayaprakasan K, Tan A, Thornton JG, Coomarasamy A, Raine-Fenning NJ. Reproductive outcomes in women with congenital uterine anomalies: a systematic review. Ultrasound in obstetrics gynecology the official journal of the International Society of Ultrasound in Obstetrics and Gynecology. 2011;38(4):371–82.
  3. Van Der Veen NM, Brouns JFGM, Doornbos JP, Van Wijngaarden WJ. Misoprostol and termination of pregnancy: is there a need for ultrasound screening in a general population to assess the risk for adverse outcome in cases of uterine anomaly? Archives of Gynecology and Obstetrics. Springer-Verlag; 2011;283(1):1–5.
  4. Lazenby GB, Huang C, Rahall AM, Fogelson NS. Pregnancy termination via laparotomy in a woman with bicornuate uterus. Contraception. Elsevier Inc.; 2007. p. 241–3. 

Acharya VV, Parulekar SV, Samant PY, Mirchandani AM. Hysterotomy for evacuation of products of conception in a bicornuate uterus. JPGO 2015. Volume 2 No. 4. Available from: http://www.jpgo.org/2015/04/hysterotomy-for-evacuation-of-products.html

Exceptionally Large Adenomyosis

Author Information

Singhal N*, Thakur H**, Gupta AS***.

(First Year Resident, ** Assistant Professor, *** Professor. Department of Obstetrics and Gynecology, Seth GS Medical College & KEM Hospital, Mumbai, India.)


Adenomyosis is characterized by uterine enlargement due to ectopic rests of endometrium located deep within the myometrium. It usually presents with pelvic pain, dysmenorrhea and menorrhagia. We present a case of adenomyosis presenting with menorrhagia where the uterine size was equal to a 16 weeks size on abdominal and pelvic examination. Cases have previously reported uterine sizes up to 12 weeks.


Adenomyosis is a benign condition which usually affects women in their forties and fifties. The incidence of the adenomyosis ranges from 20%-60% depending on histologic criteria and the degree of sectioning.[1]  According to one study nearly 90% cases are seen in parous women.[2] It is a clinical diagnosis which can be confirmed by histopathology report. It can have a significant impact on the lives of those affected with the disorder due to complications ranging from pelvic pain to menorrhagia to potential adverse fertility consequence.

Case Report

A 40 year old patient G4P3L2A1 came in the outpatient clinic in January 2013 with a complaint of heavy vaginal bleeding since 2 monthly cycles. Detailed history was taken and general examination was done. On abdominal examination the uterus was not palpable. On speculum examination cervix and vagina were healthy and bleeding was present. On vaginal examination uterus was 8 to 10 weeks in size, anteverted and fornices were free. The patient was advised to undergo a hysterectomy. But she was not willing for surgical management. Her hemogram and other investigations like serum TSH, liver and renal function tests, coagulation profile, and prolactin were within normal limits. Her PAP smear was inflammatory and endometrial aspirate showed no malignant cells. Ultrasonography of the pelvis showed endometrial hyperplasia (endometrial thickness 13 mm) but uterus was normal in size (dimensions of the uterus were not mentioned). In view of abnormal uterine bleeding and a normal endometrial cytology and patient not willing for surgical management, she was started on oral ormeloxifene 30 mg biweekly for 3 months and then once a week for the next 3 months. However, as her symptoms were not relieved an emergency therapeutic dilation and curettage (D&C) was done in April 2013.  At that time the uterus measured 12x8x7 cm on ultrasonography (USG) and uterocervical length measured with the uterine sound during D&C was 10 cm. Patient was continued on ormeloxifene  as her histopathology report showed proliferative changes in the endometrium with no hyperplasia or atypia. She responded to the 6 month course of this medication. She presented again with similar complaints in November 2013 and a second course was started in November 2013 since patient had responded to ormeloxifene and she was still not willing for a surgical management by hysterectomy. She followed up irregularly but continued her drug till May 2014. She then had 3 months of amenorrhea. Patient had another episode of menorrhagia in November 14 in which she bled continuously for one month. It failed to respond to parenteral tranexamic acid. On abdominal examination uterus was now 14-16 weeks in size. On speculum examination the cervix and vagina were healthy. On vaginal examination the uterus was 16 weeks in size, anteverted and fundus was broad. Ultrasound (USG) of the pelvis showed an enlarged uterus (18.8 x 6.2 x 8.6 cms) with thickened endometrium (15.4 mm). A D&C was done in view of the above USG findings. UCL was 15 cm and histopathology report was suggestive of bleeding endometrium. No atypia or hyperplasia was detected. As the medical therapy had failed and the uterine size had increased from 8 to 10 weeks to 16 weeks (dimensions of 8.6x5.9x4.9 cm to 18.8x6.2x8.6 cm) in 21 months while taking two 6 month courses of ormeloxifene, a total abdominal hysterectomy was performed on 4th December 2014.  In intraoperative findings uterus was 16 weeks in size. On gross examination of the uterus, it was 14 x 6 x 4 cm in size as shown in Figure 1.

Figure 1. Enlarged adenomyotic uterus

On cut section the endometrium appeared thick and disordered. Myometrium was hypertrophied and trabeculated, measuring 4 cm in thickness, suggestive of adenomyosis. A submucous leiomyomatous polyp was found at the isthmus measuring 1x1x1 cm (figure 2).

Figure 2: Cut surface showing the thickened endometrium, myometrial hypertrophy and submucous leiomyomatous polyp.

The uterus weighed 680 gm. (normal size of the uterus is 60 g).[3]) On microscopic examination islands of endometrial glands were present within the myometrial tissue which confirmed the diagnosis of adenomyosis. Endometrium was in proliferative phase and submucous trabecular leiomyomatous polyp with no evidence of malignancy was seen.


Adenomyosis usually presents with dysmenorrhea, menorrhagia, and chronic pelvic pain in multiparous women in the fourth or fifth decade of life. Many studies show link between uterine trauma like prior pregnancy or surgery (which lead to disruption between endometrium and myometrium) and adenomyosis. There is also a causal link with various hormones like estrogen and progesterone, which play a role in development and maintenance of adenomyosis. Myometrial hyperplasia is caused by the ectopic endometrial glands which makes the myometrium dysfunctional and inefficient in contracting and stopping the bleeding .Various imaging modalities like transvaginalUSG and magnetic resonance imaging (MRI) are nowadays used for diagnosing adenomyosis.[4] Treatment depends upon the severity of the symptoms and the age of the patient. Mild symptoms can be controlled by NSAIDs, GnRH agonists, hormonal contraceptives, selective estrogen receptor modulator (SERM) like ormeloxifene.[5] In our study the cause of the abnormal uterine bleeding may be the missed out submucous leiomyoma on USG. A hysteroscopy couldn’t be done because the patient always presented in emergency with bleeding. Ormeloxifene controlled the menorrhagia in both the treatment cycles probably by its action on the estrogen receptors on the endometrium. However, while on treatment the uterine size increased. Ormeloxifene’s anti estrogen action on endometrial estrogen receptors is known to prevent proliferation of the endometrium, promote decidualization of the endometrium and  helps in controlling uterine bleeding.[6] Action of the SERM ormelofexine on the myometrium is not reported. Myometrium expresses two types of estrogen receptors α and β. Uterine enlargement and proliferation is regulated by the α receptors.[7] We postulate that ormeloxifene has an undetected action on the myometrial receptors as the uterine size increased from 8-10 weeks to 16 weeks and the uterocervical length increased from 10 cm to 15 cm and myometrial hypertrophy developed while the patient was on the drug ormeloxifene. This enlargement in the uterine size increased the surface area for bleeding, myometrial adenomyosis prevented adequate myometrial contractility and the presence of the undiagnosed endometrial polyp added to the severity of her symptoms. The drug's effects on myometrial receptors should be studied as this may adversely affect the treatment of these patients with abnormal uterine bleeding.


This case is interesting as the adenomyosis was larger in size than other reported cases. It would be interesting to know whether ormeloxifene acted differentially on endometrium and myometrium. It acted like estrogenic antagonists on endometrium and estrogenic agonists on the myometrium.

  1. Hesla JS, Rock JA. Endometriosis. In Rock JA, Jones HW III. Te Linde’s Operative Gynecology. 10th ed. New Delhi: Wolters Kluver Health (India) Pvt. Ltd. -Lippincott Williams & Wilkins 2009; pp 470-473.
  2. Rapkin AJ, Howe CN. Pelvic Pain and Dysmenorrhea. In Berek JS. Berek & Novak’s Gynecology. 14th ed. New Delhi: Wolters Kluwer Health (India) Pvt. Ltd. - Lippincott Williams and Wilkins 2007; pp 520-521.
  3. Padubidri VJ, Daftary SN. Anatomy. In Padubidri VJ,  Daftary SN. Howkins & Bourne Shaw’s Textbook of Gynecology.15th edition. New Delhi: Reed Elsevier India private limited 2011. pp 6.
  4. Bazot M, Cortez A, Darai E, Rouger J, Chopier J, Antonie J, et al. Ultrasonography compared with magnetic resonance imaging for the diagnosis of adenomyosis: correlation with histopathology. Human Reproduction 2001;16(11):2427-2433.
  5. Benagiano G, Brosens I, Carrara S. Adenomyosis: new knowledge is generating new treatment strategies. Women’s Health 2009;5(3):297-311.
  6. Ormeloxifene information from DrugsUpdate. Available from: http://www.drugsupdate.com/generic/view/367/Ormeloxifene
  7. Blesson CS, Masironi B, Sahlin L. Effects of selective estrogen receptor agonists on estrogen receptor expression in the uterus of ovariectomized rats. OJMIP (Open Journal of Molecular and Integrative Physiology). 2012;2(2) May Available from: http://www.scirp.org/journal/PaperInformation.aspx?PaperID=19377#.VQFCfvmUcQM

Singhal N, Thakur H, Gupta AS. Exceptionally Large Adenomyosis. JPGO 2015. Volume 2 No. 4, Available from: http://www.jpgo.org/2015/04/exceptionally-large-adenomyosis.html