Volume 1 Issue 9, September 2014

Editorial
Chauhan AR

Multiple Cesarean Section Scar Dehiscences
Samant PY, Parulekar SV.

Partial Mullerian Agenesis Presenting as Hematometra and Hematosalpinx
Narkhede HR, Pawar A, Mhatre JP, Mhatre PN.

Silent Rupture Of Left Horn Of Bicornuate Uterus And Fetal Survival
More V, Warke H, Mali K, Satia MN.

 Postmenopausal Ovarian Stromal Hyperplasia
Channawar S, Patil D, Pawde A, Chauhan AR.

Antiphospholipid Antibody Syndrome
Chakre S, Pardeshi S, Mayadeo NM, Shah S.

Cervical Lipoleiomyoma
Chaudhari J, Kothari K, Gupta AS, Dwivedi J.

Giant Borderline Mucinous Cystadenoma With Previous 3 Cesareans
Katke RD, Kiran U, Saraogi M, Sarode S, Thawal R.

Cervical Agenesis With Scar Endometriosis
Poonia S, Panchbudhe S, Satia MN.

Congenital Diaphragmatic Hernia
Kulkarni YS, Kakade AS, Maalavika A.

Editorial

Chauhan AR

Uterine rupture, a rare but catastrophic obstetric emergency, is defined as a complete or full- thickness disruption of all the uterine layers including the serosa; it may occur in an unscarred or scarred uterus. The commonest scar encountered is that of previous cesarean section (CS). Premonitory signs like maternal tachycardia, abnormal CTG, severe abdominal pain, scar tenderness, vaginal bleeding, hematuria, loss of station of presenting part and should clue the clinician to impending rupture. Rupture is associated with serious maternal hemorrhage, fetal distress or demise, and urgent laparotomy, all of which contribute to high maternal and perinatal morbidity and mortality. On the other hand, uterine dehiscence is an incomplete disruption of a preexisting uterine scar with intact uterine serosa. Dehiscence is more common than rupture, is often occult and outcomes are better as hemorrhage is less and the fetus, umbilical cord and placenta are contained within the uterus.
It bears remembering that though previous uterine scar is an important factor for rupture, majority of cases with scarred uteri result in dehiscence rather than rupture. In developed countries, uterine rupture is usually seen with trial of labor after previous lower segment cesarean section (LSCS). A WHO systematic review published in 2005 on the worldwide prevalence of uterine rupture and subsequent maternal morbidity and mortality found that in developed countries, prevalence of rupture with previous CS was around 1%, and in women without previous CS, prevalence was extremely rare, <1 per 10,000.  Rising primary cesarean section rates, and in response to this trend, rising rates of trial of labor after cesarean (TOLAC) are responsible for rupture; WHO recommends that efforts should focus on reduction in CS rates and careful monitoring of patients who opt for vaginal birth after CS (VBAC).
The scenario of uterine rupture is quite different in resource- poor settings, where the WHO review found that in least- developed countries (Nigeria, Ghana, Bangladesh), 75 % of cases of rupture were seen in unscarred uteri, with attendant high maternal mortality (1 to 13 %) and perinatal mortality (74 to 92 %). Studies from the Indian subcontinent have shown that prolonged obstructed labor and lack of access to operative delivery are still the main cause of uterine rupture, followed by scarred uterus. A ten year analysis of uterine rupture by Sahu from a teaching hospital (JIPMER Pondicherry) found majority (70%) in unbooked cases, with an equal distribution of rupture among scarred and unscarred uteri. In patients with unscarred uterus, cephalopelvic disproportion (CPD) followed by malpresentation was the leading cause. Postpartum hemorrhage was seen in 66% of cases and as many as 83% cases required blood transfusion. They reported a perinatal mortality of 94.07% and maternal mortality of 2.76%. These figures are probably representative of data from major referral centers in the country; they put a huge burden on existing healthcare facilities.
Risk factors for uterine rupture are well known and include number and type of previous CS: one previous LSCS (further classified as single or two-layer closure), one previous low vertical incision, previous classical CS or multiple previous CS; the risk of scar rupture increases to approximately 2% with 2 or more previous CS. Other uterine surgeries like myomectomy, congenital anomalies of the uterus, labor induction especially with prostaglandins, dystocia, CPD, instrumentation and trauma are also important risk factors. Conversely, longer inter-pregnancy interval after previous CS (> 2 years) allows the scar to recover its maximum tensile strength and reduces the risk of rupture.  Previous successful vaginal birth especially previous VBAC, is the single most important predictor of successful vaginal delivery.
Women with previous CS opting for VBAC versus elective repeat CS should be counseled in the antenatal period and a clear defined labor plan in accordance with current guidelines should be documented. VBAC should be considered in optimum clinical settings with facilities for close monitoring and resources for immediate LSCS and neonatal resuscitation. Patients should be informed about the high chance (>70 %) of successful VBAC after one previous LSCS and the low risk of rupture uterus (22 – 74/ 10,000). However they should also be informed that induced labor, especially with prostaglandins, carries a higher risk of uterine rupture.
A Cochrane review in 2013 on VBAC has met with some criticism as their conclusions are not precise. They state that “planned elective repeat cesarean section and planned VBAC for women with prior cesarean birth are both associated with benefits and harms”. They further state that as the data was gathered from non-randomised studies, randomized controlled trials were required and existing results should be interpreted with caution.
In the developing world, our decisions need to be tailored to the availability of facilities and the “repeat elective LSCS versus VBAC debate” should take a backseat to the prevention of obstructed labor. The WHO review recommends formulation of stringent guidelines to ensure that misoprostol is used in safe dosages for labor induction in resource –poor settings to prevent iatrogenic ruptures. Ensuring reduction of unwanted pregnancies, especially in women with high parity, and providing safe access to emergency obstetric care, should be our top priorities.
We are pleased to present our readers with the September issue of the journal, wherein two cases highlight the risk factors for rupture uterus. In cases with congenital anomalies, the uterine musculature varies in thickness in different parts of the uterus, and the abnormal portion tends to become thin as pregnancy advances; an extremely high incidence of rupture (8 %) has been reported. When pregnancy implants in a rudimentary horn, rupture usually occurs remote from term in the majority of cases.

Multiple Cesarean Section Scar Dehiscences

Author Information

Samant PY*, Parulekar SV**
(* Additional Professor, ** Professor and Head, Department of Obstetrics and Gynacology, Seth G.S. Medical College and K.E.M Hospital, Mumbai, India.)

Abstract

Scar dehiscence is not a unique finding at cesarean section. But dehiscence at more than one site at the same time in a case is extremely rare. We report an incidental finding of scar dehiscence at two sites at elective cesarean section at 38 weeks gestation in a registered antenatal patient with two previous cesarean sections.

Introduction

With rising trend and incidence of cesarean section combined with high parity and propensity to have unplanned and home deliveries; the risks to maternal and fetal life posed by scar dehiscence and rupture are very high. Scar dehiscence is defined as disruption of the uterine muscle with intact uterine serosa. [1] It is also called as Cesarean scar defect (CSD), and involves myometrial discontinuity at the site of a previous Cesarean section scar. Though the commonest etiological factor for dehiscence is a cesarean scar; other scars like those of myomectomy, metroplasty, accidental perforation during curettage are known to cause rupture or dehiscence. Myomectomy using cautery is also known to cause scar dehiscence and can pose risk of rupture [2]. With rising incidence of multiple cesarean sections, occurrence of multiple dehiscent windows is plausible.

Case Report

Thirty-four years old multigravida with previous two cesarean sections was admitted for elective cesarean section with tubal ligation after thirty-eight weeks. Her preoperative investigations were normal. Obstetric ultrasonography was unremarkable. She did not report any abdominal pain or backache before surgery.
Patient had a high transverse scar of previous surgeries, about two inches above the pubic hairline. The abdomen was opened transversely along the scar. On laparotomy, the lower segment was well formed. Uterovesical fold of peritoneum was opened and bladder was dissected down. Two subcentimeter foci of translucent myometrial windows one below the other were identified in the lower segment, with only a layer of visceral peritoneum each covering them. Bladder was densely adherent to the myometrium just below the lower window. Incision was taken to include the upper window. After delivering the baby, the edges were examined. Lower edge of the current incision as well as the myometrium between the two windows was extremely thin. Considering the risk of bladder injury, and the fact that the patient desired tubal ligation, repair the lower defect was not attempted. Scarred myometrium edges of the upper window were excised and incision was closed in single non interlocking layer with no 1 polyglactin. Tubal ligation was performed. The patient and her relatives were informed about the findings and need for caution in case they ever considered recanalization of fallopian tubes.


Figure1: Two scar defects (arrows) are seen one below the other on the lower segment.

Discussion

Generally dehiscence of Cesarean scar is detected at laparotomy and it is almost always asymptomatic. Scars of myomectomy [2], uterine perforation, and classical Cesarean section are known to cause symptomatic dehiscence. Pathologists have noted signs of inflammation like congested endometrium in the scar recess, lymphocytic infiltration, foreign body giant cell reaction and capillary dilatation; 2-15 years after cesarean section in hysterectomy specimens. [3] These are considered to be responsible for diverse complaints such as lower abdominal pain, dyspareunia, and dysmenorrhea. In nonpregnant state, uterine scar defects may be associated with intermenstrual bleeding in women with a previous cesarean delivery.[4] Wang et al found that width of the CSD was associated with post- menstrual spotting, dysmenorrhea, and chronic pelvic pain and that multiple Cesarean sections were seen to be associated with increased width and depth of the CSD.[5] Multiple Cesarean sections were also found to be associated with dysmenorrhea, independent of CSD width, but not with postmenstrual bleeding or chronic pelvic pain.[5] Scar dehiscence is a known complication associated with previous Cesarean section. Risk of uterine scar dehiscence or window is about 4% and that of scar rupture is 0.4 to 0.6% after previous lower segment Cesarean section. [6] Though in most cases scar dehiscence is an incidental finding in an asymptomatic parturient; this term implies possibility of clinically significant events and should be considered as such.[7] Use of electrocautery at myomectomy has been shown to be associated with scar dehiscence and rupture of uterus.[2] Risk of pregnancy-related uterine rupture attributable to laparoscopic myomectomy is 1%.[8] A single-layer closure is associated with increase in the risk of rupture compared with a double-layer closure.[9] Lower segment approximation excluding endometrium and using synthetic non locking sutures makes the scar more secure.[9,10] During closure of the uterine incision, excision of the previous scar prevents scar defect in the fibrosed suture line.
In a prospective observational study of 642 women with VBAC Rosenberg et al found that thickness of lower uterine segment at around 37 weeks of pregnancy was inversely proportional to the likelihood of detection of defects at physical examination at delivery.[11] Magnetic resonance imaging is considered superior to CT in evaluating complications at the incisional site.[12] Based on MR imaging, investigators have classified dehiscence into complete; where all the layers except peritoneum are separated and partial; where either innermost or outermost layers are separated but myometrium is intact.[13] In a study of 467 cases of VBAC, in 414 cases, Kaplan et al examined the scar transcervically and did not detect a single case of dehiscence. [14] Only indications for scar exploration are unexpected vaginal bleeding or hypovolemia after VBAC as per ACOG practice Bulletin on VBAC.[7] Excision of the previous scar and appropriate anatomical repair in continuous nonlocking suture with delayed absorbable synthetic material should be employed to reduce chance of multiple scar defects. Since scar defects are also associated with abnormal uterine bleeding and chronic pelvic pain, multiple scar defects may potentiate the problem.

References
  1. Landon MB, Hauth JC, Leveno KJ, Spong CY, Leindecker S, Varner MW, et al. Maternal and perinatal outcomes associated with a trial of labor after prior cesarean delivery. N Engl J Med 2004; 351:2581–9.
  2. Hasbargen Uwe et al. Uterine dehiscence in nullipara. Human reproduction, 2002;17:2180-2182 .
  3. Morris H. Surgical pathology of the lower uterine segment caesarean section scar: is the scar a source of clinical symptoms? Int J Gynecol Pathol. 1995;14:16-20.
  4. Van Horenbeeck A, Temmerman M, Dhont M. Cesarean scar defect: correlation between Cesarean section number, defect size, clinical symptoms and uterine position. Obstet Gynecol. 2003;102:1137-9.
  5. Wang C B, Chiu WW C, Lee C Y, Sun Y L, Lin Y H, Tseng C J. Cesarean scar defect: correlation between Cesarean section number, defect size, clinical symptoms and uterine position. Ultrasound Obstet Gynecol 2009; 34: 85-89.
  6. Thorkild F. Nielsen, Ulf Ljungblad, Henrik Hagberg. Rupture and dehiscence of cesarean section scar during pregnancy and delivery American J Obstet Gynecol 1989;160:569–573.
  7. ACOG Practice Bulletin No. 115 Vaginal Birth After Previous Cesarean Delivery August 2010, p 2.
  8. Dubuisson JB, Fauconnier A, Deffarges JV et al. Pregnancy outcome and deliveries following laparoscopic myomectomy. Hum Reprod. 2000;15: 869-73.
  9. Bujold E1, Bujold C, Hamilton EF, Harel F, Gauthier RJ. The impact of a single-layer or double-layer closure on uterine rupture. Am J Obstet Gynecol 2002;186:1326-30.
  10. Bérubé Laurie, Mariko Arial, Geneviève Gagnon, Normand Brassard, Amélie Boutin, Emmanuel Bujold, Factors Associated With Lower Uterine Segment Thickness Near Term in Women With Previous Caesarean Section. J Obstet Gynaecol Can 2011;33:581–587.
  11. Rozenberg P, Goffinet F, Phillippe HJ, Nisand I. Ultrasonographic measurement of lower uterine segment to assess risk of defects of scarred uterus. Lancet. 1996 Feb 3; 347(8997):281-4.
  12. Rivlin ME, Patel RB, Carroll CS, Morrison JC. Diagnostic imaging in uterine incisional necrosis/dehiscence complicating cesarean section. J Reprod Med 2005;50: 928–932.
  13. Maldjian C1, Milestone B, Schnall M, Smith R. MR appearance of uterine dehiscence in the post-cesarean section patient. J Comput Assist Tomogr. 1998;22(5): 738-41.
  14. Kaplan B, Royburt M, Peled Y, Hirsch M, Hod M, Ovadla Y, Neri A. Routine revision of uterine scar after prior cesarean section. Acta Obstetricia et Gynecologica Scandinavica 1994;73:473-475.
Citation

Samant PY,  Parulekar SV. Multiple Cesarean Section Scar Dehiscences. JPGO 2014 Volume 1 Number 9 Available from: http://www.jpgo.org/2014/09/multiple-cesarean-section-scar.html

Partial Mullerian Agenesis Presenting as Hematometra and Hematosalpinx

Author Information

Hemraj R Narkhede*, Amol Pawar**, Jyoti P Mhatre***, Pravin N Mhatre****.
(* Assistant professor, ** Associate Professor, **** Professor. Department of Obstetrics and Gynecology, Seth G S Medical College & Nowrosjee Wadia Maternity Hospital, Parel, Mumbai, India. *** Senior Consultant, Kedar Hospital, Mazgaon, Mumbai, India.)

Abstract

A rare case of Mullerian agenesis presented with acute abdomen. hematometra, hematosalpix and bowel obstruction. We performed extirpative operation to save the life of patient. Vaginoplasty was performed in the same sitting to drain peritoneal cavity and for coital function. Type and time of presentation of Mullerian agenesis make these cases challenging and individualization of management on case to case basis is needed.

Introduction

Absent vagina is a rare Mullerian anomaly. The incidence of Mullerian anomaly reported is between 0.1-3.5%.[1,,2,3,4] Vaginal agenesis has a reported incidence of 1 in 5000 women.[5] The clinical presentation varies in each case depending on the type of anomaly, age at presentation and in relation to menarche. The surgical management of each case is challenging and should be individualised on case to case basis. Common presentation of vaginal agenesis is primary amenorrhea. Mullerian agenesis is second most common cause of primary amenorrhea in adolesence.[6] Here we report a case of partial Mullerian agenesis involving absence of vagina and cervix, due to its rare occurrence and challenging surgical management.

Case report

A 17 year unmarried girl presented with acute abdomen, pelvic mass, fever with rapid thready pulse and mild hypotension The pain in abdomen which was initially cyclical had turned into continuous pain since one year. The patient was having vomiting and colicky abdominal pain with gradual distension of lower abdomen. She had a tense, tender mass corresponding to 30 weeks of pregnacy. Bowel sounds were absent. The secondary sexual characters were well developed with absence of menarche. On investigation she was found to be anemic with raised counts. The patient was kept nil by mouth with Ryle’s tube aspiration and started with injectable antibiotic. Ultrasonography showed a large mass measuring 14×13×12 cm with multiple septa and fluid and dilated bowels. Magnetic resonance imaging (MRI) was done for detailed anatomic evaluation (figures 1, 2, 3). It showed septate uterus with two complete separate uterine cavities, larger on right side with hematometra. Both fallopian tube were thickened and diffusedly dilated from cornu till fimbrial end with large hematosalpinx. Vagina was not seen. The diagnosis of hematometra, hematosalpinx with subacute intestinal obstruction was made.


Figure 1. MRI of abdomen and pelvis, longitudinal section showing extent of hematometra and right fallopian tube.


Figure 2. MRI of abdomen and pelvis, transverse section showing hematometra.


Figure 3. MRI of abdomen and pelvis, transverse sections showing showing both ovaries (arrows).

Exploratory laparotomy was done through Pfannenstiel incision. Hemoperitoneum along with foul smelling pus was drained. (figure 4). There was large hematometra with hematosalpinx, bilateral chocolate cysts and multiple dense adhesions with bowels in pouch of Douglas. The uterus was bicornuate with right horn enlarged as compared to the left horn. Uterus and both fallopian tubes were gangrenous in appearance. The bowel adhesions were separated with utmost care. A decision was taken to perform an extirpative surgery, to remove both the horns of the uterus along with the tubes and right ovary. Left ovary was preserved. Vaginoplasty was done in same sitting with a combined procedure of anterior peritoneum pull through and posteriorly implanting of freeze dried amnion graft.


Figure 4. Intraoperative findings and post-operative specimen.

Prophylactic nasogastric drainage was done. Postoperative period was uneventful with the patient being discharged on day 5 of surgery. The patient came for regular follow-up during which period she was trained for regular self vaginal dilatation using a glass dilator. She married post one year of surgery leading a satisfactory conjugal relationship.

Discussion

A vaginal atresia results when caudal portion mesonephric duct fails to canalise and fuse with urogenital sinus. This caudal portion of vagina is replaced by fibrous tissue. In 1998 American society for reproductive medicine classified the Mullerian anomalies.[7] Vaginal atresia is type 1 anomaly. In considering management of Mullerian agenesis, the age of patient and psychological implication for the patients’s condition are of paramount importance. Accurate delineation of anatomy is a must before proceeding for surgical management. In this case presence of hematometra, hematosalpinx with acute abdomen and sub-acute intestinal obstruction presented with challenge for diagnosis and management of Mullerian agenesis. The reason for delayed presentation may be due to neglecting of symptoms of persistent pelvic pain. The absence of menstrual cycle by 17 years of age should have encouraged the patient to seek a medical opinion. Pelvic MRI is the preferred modality when there is a suspicion of any Mullerian anomaly.[8] Secondary bacterial infection leads to the development of pyosalpinx and pyometra. Chocolate cyst and pelvic adhesion may be due implantation endometriosis leading to intestinal obstruction. We thought of uterine conservative procedure before start of surgery. However intraoperative gangrenous appearance of hemtometra and hematosalpinx prevented any such approach. Hence we peformed hysterectomy and bilateral salpingectomy and right oophorectomy. Left ovary was preserved. An early diagnosis and management of obstructive anomalies could have prevented such radical surgery. Vaginoplasty was performed in the same sitting so as to provide drainage and to give her coital function. We dissected the vaginal space from below and mobilized peritoneal flap from above. Due to adhesions in pouch of Douglas, partial mobilization of the peritoneum was possible anteriorly. Posteriorly freeze dried amnion graft was placed. In follow-ups we encouraged for vaginal dilatation. She is married and has normal sexual life after an year of surgery.

Conclusion

The vagaries in Mullerian agenesis, both in type and time of presentation make these cases challenging. The treatment protocool cannot be generalised but is based on individual clinical scenario. This case report highlights and confirms the same.

References
  1. Marcal L, Nothaft MA, Coelho F, Volpato R, Iyer R. Mullerian duct anomalies: MR imaging. Abdom Imaging. Dec 2011; 36(6):756-64. .
  2. Bermejo C, Martínez PT, Cantarero R, Diaz D, Pedregosa JP, Barrón E, et al. Three-dimensional ultrasound in the diagnosis of Müllerian duct anomalies and concordance with magnetic resonance imaging. Ultrasound Obstet Gynecol 2010; 35(5):593-601. 
  3. Steinmetz GP. Formation of artificial vagina. West J Surg 1940; 48:169-3.
  4. Strassmann EO. Operations for double uterus and endometrial atresia. Clin Obstet Gynecol. 1961; 4:240.
  5. Rock JA. Surgery for anomalies of the mullerian ducts. In: Tompson JD, Rock JA, eds. TeLind's Operative Gynecology. 9th ed. Philadelphia, Pa: JB Lippincott Williams & Wilkins; 2003:705.
  6. Petrozza JC, Gray MR, Davis AJ, Reindollar RH. Congenital absence of the uterus and vagina is not commonly transmitted as a dominant genetic trait: outcomes of surrogate pregnancies. Fertil Steril 1997; 67(2):387-9. 
  7. The American Fertility Society classifications of adnexal adhesions, distal tubal occlusion, tubal occlusion secondary to tubal ligation, tubal pregnancies, Mullerian anomalies and intrauterine adhesions. Fertil Steril. Jun 1988; 49(6):944-55.
  8. Marten K, Vosshenrich R, Funke M, Obenauer S, Baum F, Grabbe E: MRI in the evaluation of Mullerian duct anomalies. Clin Imaging 2003, 27:346–350.

Citation

Narkhede HR, Pawar A, Mhatre JP, Mhatre PN. Partial Mullerian agenesis presenting as Hematometra and Hematosalpinx. JPGO 2014 Volume 1 Number 9. Available from: http://www.jpgo.org/2014/09/partial-mullerian-agenesis-presenting.html

Silent Rupture Of Left Horn Of Bicornuate Uterus And Fetal Survival

Author Information

More Vibha*, Himangi Warke**, Mali Kimaya*, Satia MN***.
(* Assistant Professor, ** Associate Professor, *** Professor. Department of Obstetrics and Gynecology, Seth GS Medical College & KEM Hospital, Mumbai, India)

Abstract

We report a case of pregnancy with silent rupture at 32 weeks of gestation in a left horn of bicornuate uterus which continued till 35 weeks, followed by delivery of a live fetus.  The diagnosis of bicornuate uterus was missed in early weeks of gestation on ultrasonography (USG) and later was misdiagnosed as abdominal pregnancy. Preoperative diagnosis with USG and magnetic resonance imaging (MRI at our institute confirmed the diagnosis of pregnancy in the left horn of uterus. A live fetus was delivered by cesarean section followed by excision of the left uterine horn.

Introduction

Congenital malformation of uterus is due to abnormal fusion of mullerian ducts or failure of absorption of the septum. Mullerian duct anomalies are estimated to occur in 0.4%.[1] They can be diagnosed at different stages of life. Abortion, preterm delivery, malpresentation, fetal growth restriction are more commonly seen in women who have bicornuate uterus. Non invasive imaging modalities like USG and MRI helps in diagnosis of bicornuate uterus in early weeks of gestation.

Case Report

A 30 year old woman, married for 15 years, gravida 2 para 1 with 33 weeks of pregnancy was referred from a private hospital in view of USG suggestive of an abdominal pregnancy with hemoperitoneum. She had full term normal delivery 9 years ago. Her early USG at 17 weeks and 32 weeks showed normal intrauterine pregnancy. At 32 weeks of gestation she was admitted in a private hospital in view threatened preterm labor. USG done at that time showed an extrauterine abdominal pregnancy with hemoperitoneum for which she was referred to our tertiary care center. On examination, her general condition was fair, vital parameters were normal. Abdominal examination revealed pregnancy of 32 weeks in breech presentation, normal fetal heart sounds and relaxed uterus. USG at our center showed a bicornuate uterus with pregnancy in the left uterine horn with no hemoperitoneum. MRI also showed an intrauterine pregnancy in the left horn. She was hospitalized. An elective lower segment casarean section at term was planned for her in view of breech presentation with bicornuate uterus. But she went into spontaneous labor three weeks later and an emergency lower segment cesarean section was performed. Intraoperatively there was around 50 ml of hemoperitoneum (altered brown colored) with clots in the pouch of Douglas. Pregnancy was seen in the left horn of uterus. Right horn of uterus, right fallopian tube and ovary were normal. A curvilinear incision was made on the lower part of the left uterine horn and a fetus weihing 2.3 kg in breech presentation was delivered. The uterine wall of left horn was extremely thin (2- 3 mm). There was a rent of around 1 cm noted 8 cm from the upper margin of the left uterine horn near the left ovary. It was sealed by blood clots and omentum suggestive of an old rupture, which had probably occurred at 32 weeks when she was admitted with threatened preterm labor. There were dense adhesions seen between the posterior surface of left uterine horn, left ovary and omentum. Placenta was morbidly adherent and was removed piecemeal. Left uterine horn was atonic and hence adhesiolysis followed by excision of left uterine horn with left salpingoophorectomy was done. She was transfused with 1 unit of whole blood. Postoperative course was uneventful.


Figure 1. Left rudimentary horn with fetus (black arrow), right horn (yellow arrow) and right fallopian tube (blue arrow).


Figure 2. Left rudimentary horn after delivery of baby (black arrow) with omental adhesions (green arrow), right horn (yellow arrow).


Figure 3. right horn after excision of rudimentary horn.


Figure 4. Excised left horn.

Discussion

Uterus during embryogenesis is formed by fusion of two mullerian ducts. Failure of fusion of the two mullerian ducts results in separate uterine horns (bicornuate uterus). Bicornuate uetrus can be bicornis bicollis or uterus bicornis unicollis. The two uterine horns in uterus bicornis unicollis may be equal but rudimentary, or equal and well developed, or one horn may be less developed than the other. Bicornuate uterus accounts for 25% of all müllerian anomalies. Bicornuate uterus is type four according to the American Fertility Society classification of müllerian anomalies. In majority, bicornuate uterus is undiagnosed. In others, it may be accidentally diagnosed during investigation of infertility or repeated abortion or during dilatation and curettage or cesarean section. Abortion, preterm delivery, malpresentations, IUGR are more commonly seen in women who have bicornuate uterus. There is increased incidence of cesarean delivery (82% as reported by Heinonen).[2] Diagnosis of uterine anomalies on two dimensional USG is difficult.[3] Three dimensional USG is an excellent method to evaluate these malformations.[4] MRI delineates duct anomalies and their extent and its accuracy reported is 100%.[5,6] Rupture in such cases is due to inability of malformed uterus to expand.[7] There is also increased risk of postpartum hemorrhage in cases with uterine anomalies as seen in our case, which required excision of the horn.[8]

References

  1. Grimbizis G. F., Camus M., Tarlatzis B. C., Bontis J. N., Dervoey P. Clinical implications of uterine malformation and hysterscopictreatment results. Human Reproduction update. 2001;7(2): 161-174. 
  2. Heinonen P. K. Uterus didelphys: a report of 26 cases. Eur J Obstet Gynecol Reprod Biol.1984;15(5):345-50.
  3. Nicolini U, Bellotti M, Bonazzi B, Zamberletti D, Candiani GB. Can ultrasound be used to screen uterine malformations? Fertil Steril 1987;47:89-93.
  4. Fedele L, Dorta M, Brioschi D, Massari C, Candiani GB. Magnetic resonance evaluation of double uteri. Obstet Gynecol 1989;74:844-47.
  5. Pellerito JS, McCarthy SM, Doyle MB, Glickman MG, DeCherney AH. Diagnosis of uterine anomalies: Relative accuracy of MR imaging, endovaginal sonography, and hysterosalpingography. Radiology 1992;183(3):795-800.
  6. Whitehouse DB. Post-partum haemorrhage from uterus pseudo-didelphys. BJOG: An International Journal of Obstetrics & Gynaecology, 1955;62: 109-110.

Citation

More V, Warke H, Mali K, Satia MN. Silent Rupture of Gravid Rudimentary Horn and Fetal Survival. JPGO 2014 Volume 1 Number 9 Available from:  http://www.jpgo.org/2014/09/silent-rupture-of-gravid-rudimentary.html

Postmenopausal Ovarian Stromal Hyperplasia

Author Information

Sarita Channawar*, Devendra Patil**, Anuya Pawde***, A. R. Chauhan***
(* Assistant Professor, ** Third tear Resident, *** Senior Registrar, **** Additional Professor. Department of Obstetrics and Gynecology, Seth G.S. Medical College and K.E.M Hospital, Mumbai, India.)

Abstract

Ovarian stromal hyperplasia (OSH) and hyperthecosis (OHT) are rare causes of virilization in postmenopausal women. A 60 year old postmenopausal woman with virilization due to bilateral ovarian stromal hyperplasia is discussed here.

Introduction

Hirsutism in women may be defined as excess thick (terminal) hair growth in body parts where normally there is no presence of terminal hair. It is an early sign of hyperandrogenism. The causes of hirsuitism in postmenopausal women can be many, like ovarian stromal hyperplasia, ovarian hyperthecosis, ovarian tumors, adrenal tumors, Cushing’s syndrome and insulin resistance syndrome. Ovarian stromal hyperplasia and hyperthecosis are very rare causes of hyperandrogenism. The serum testosterone levels show moderate elevation resulting into virilisation. This case report highlights ovarian stromal hyperplasia as a cause hyperandrogenism and virilization in a postmenopausal woman.

Case Report

A 60 year old parous woman, postmenopausal since ten years, presented with a history of worsening hirsutism (increasing facial hair) since 1-2 months, and excessive weight gain since 1 year (BMI of 34.57). She was a known case of diabetes mellitus and hypertension on treatment since 10 years and 2 years respectively, and had undergone cholecystectomy 10 years ago. On general examination, she had facial mooning and excessive facial hair. Abdominal examination revealed central obesity but no palpable lump. Local genital examination showed mild clitoromegaly; per speculum examination showed healthy cervix and vagina; on bimanual examination the uterus was normal in size with no palpable adnexal mass.


Figure: 1 Hirsuitism


Figure: 2 Clitoromegaly

The patient was managed jointly with an endocrinologist. Her investigations were directed at finding the source of androgen excess and ruling out Cushing’s syndrome. The serum concentration of testosterone (25.88 ng/ml) and dehydroepiandrosterone sulphate (DHEAS - 54.22 ug/ml) were significantly elevated, while level of 17-hydroxy-progesterone (15.68 ng/ dl), aldosterone ( 29.13 ng/dl), androstenedione (26.03 ng/dl), deoxycortisol (33.85 ng/ml), cortisol (12.9 ug/ml), corticosterone (159.74 ng/ml), FSH 45 mIU/ml, LH 13.78 mIU/ml (postmenopausal) were normal. Pelvic ultrasonography showed normal uterus with bulky ovaries without follicles, vascularity or any solid cystic lesion. Computerized tomography (CT) of the abdomen and pelvis was suggestive of bilateral adrenal hyperplasia, with small cortical cyst in left kidney and bilateral ovaries bulky for age. The extent of adrenal hyperplasia was considered normal for her postmenopausal age. The basal level of serum cortisol (22.31), oral dexamethasone suppression cortisol (0.97) and basal ACTH (33.7) were normal. So adrenal cause of hyperandrogenism was ruled out and provisional diagnosis of ovarian nonmalignant cause of peripheral androgen excess was made. The plan of management was surgery with bilateral oophorectomy for removal of the source of excess androgen; accordingly she underwent total abdominal hysterectomy with bilateral salpingo-oophorectomy. Intraoperatively, the uterus was smaller than normal size, but both ovaries were bulky and smooth. Her postoperative course was uneventful and she was discharged on postoperative day 5, at which time her repeat serum testosterone level was 0.9 ng/ml, within normal range. The histopathology report was suggestive of bilateral ovarian stromal hyperplasia with no evidence of malignancy.

Discussion

Ovarian stromal hyperplasia (OSH) is the nodular or diffuse proliferation of ovarian stroma, and ovarian hyperthecosis (OHT) is stromal proliferation with luteinised stromal cells. Both OSH and OHT are non-neoplastic pathologies involving both ovaries, causing excess androgen production, and usually seen in postmenopausal females. OSH and OHT are seen in postmenopausal women but these histopathological conditions rarely cause elevation in testosterone and virilisation. A wide range of clinical manifestations like hirsutism, virilization, abnormal menses, obesity, hypertension and insulin resistance are seen.[1] Mild diffuse bilateral hyperplasia is found in one third of perimenopausal and postmenopausal women. The case presented here is a postmenopausal patient with markedly elevated testosterone levels and signs of virilization secondary to ovarian stromal hyperplasia.
The cause of significantly increased androgen secretion in postmenopausal patients with OSH and OHT is increased gonadotropin secretion which causes stimulation of gonadal cells to produce testosterone and androstenedione. Androgens are aromatized to estrogens by granulosa cells in premenopausal women, but this does not occur to the same degree in postmenopausal women, leading to predominance of androgens.[2] The increase in estrogen production may result in increased risk of endometrial hyperplasia and carcinoma, especially in postmenopausal women.[3] It is also necessary to rule out ovarian and adrenal tumors in postmenopausal woman with hirsutism and marked hyper-androgenism.
Androgen levels are important for diagnosis; testosterone and DHEA-S should be measured first and are usually found high for postmenopausal age.[4] If serum total testosterone level is >150 ng/ml, imaging of adrenals and ovaries must be done. A transvaginal pelvic ultrasound and CT scan or magnetic resonance imaging are very useful for diagnosis.[5] If not diagnosed on these modalities, selective ovarian venous sampling has been described.[6] Once the diagnosis is made using history, clinical examination, laboratory parameters and imaging, the patient should undergo bilateral oophorectomy to remove the cause of excess androgens and to reverse virilization.[7]

Conclusion

Hyperandrogenism in postmenopausal women is a diagnostic challenge. The causes of postmenopausal virilization may be associated with adrenal or ovarian androgen-secreting tumors or with benign conditions. The development of virilization can be progressive (characteristic of benign causes), or rapid (characteristic of malignant tumors), and detailed clinical history is critical to differentiate these conditions. Imaging techniques do not always reveal the cause of hyperandrogenism, in which case adrenal and/or ovarian venous sampling, though difficult, may be used. The cases of postmenopausal severe and acute onset virilization, where Cushing’s syndrome is ruled out and adrenal imaging is normal, should be treated with bilateral oophorectomy. This approach is necessary to avoid further delay in definitive treatment.

References
  1. Rousset P, Gompel A, Christin-Maitre S, Pugeat M, Hugol D, Ghossain MA, Buy JN. Ovarian hyperthecosis on grayscale and colour Doppler ultrasound, Ultrasound Obstet Gynaecol 2008; 32(5):694-9.
  2. Goldman JM, Kapadia LJ. Postgrad Med J. Virilization in a postmenopausal woman due to ovarian stromal hyperthecosis Postgrad Med J 1991;67(785): 304-306.
  3. Nagamani M, Hannigan EV, Dinh TV, Stuart CA. Hyperinsulinemia and stromal luteinization of the ovaries in postmenopausal women with endometrial cancer. J Clin Endocrinol Metab 1988;67(1):144-8.
  4. Martin KA, Chang RJ, Ehrmann DA, Ibanez L, Lobo RA, Rosenfield RL, et al. Evaluation and treatment of hirsuitism in premenopausal women; an endocrine society clinical practise guideline. J Clin Endocrinol Metab 2008;93(4):1105-20.
  5. Rothman MS, Wierman ME. How should postmenopausal androgen excess be evaluated? Clin Endocrinol 2011;75(2):160-4.
  6. Levens ED, Whitcomb BW, Csokmay JM, Nieman LK. Selective venous sampling for androgen producing pathology. Clin Endocrinology. 2009;70(4):606-614.
  7. Alpanes M, Gonzalez-Casbas JM, Sanchez J, Pian H, Escobar-Morreale HF. Management of postmenopausal virilization. J Clin Endocrinol Metab 2012;97(8):2584-8.
Author Information

Channawar S, Patil D, Pawde A, Chauhan AR. Postmenopausal Ovarian Stromal Hyperplasia. JPGO 2014 Volume 1 Issue 9. Available from : http://www.jpgo.org/2014/09/postmenopausal-ovarian-stromal.html

Antiphospholipid Antibody Syndrome

Author Information

Chakre Shila*, Pardeshi Sachin*, Mayadeo NM**, Shah S.
(* Assistant Professor, ** Professor, *** Third Year Resident. Department of Obstetrics and Gynecology, Seth G.S. Medical College and K.E.M Hospital, Mumbai, India.)

Abstract

Antiphospholipid  antibody syndrome in obstetric is clinically defined by  repeated unexplained abortion before 10th weeks, unexplained fetal loss at or after 10th week, premature birth before 34 weeks due to preeclampsia.[1] In Antiphospholipid antibody syndrome placental mediated complications such as mild to severe pregnancy induced hypertension, intrauterine growth restriction, intrauterine fetal demise, missed abortion, antepartum hemorrhage and threatened abortion are high and hence the rate of cesarean section is also high.[2] Antiphospholipid  antibody syndrome  management is based on combined medical-obstetric high risk management and treatment with aspirin and injection heparin but still there are refractory cases to treatment in which there are obstetric complications. We present a case report of different obstetric outcomes in a same patient with two separate pregnancy, one year apart in a treated Antiphospholipid antibody syndrome.

Introduction

Antiphospholipid antibody syndrome is defined by the presence of pregnancy morbidity and thromboembolic complications in presence of persistently increased titers of Antiphospholipid antibodies.[3] There are subtypes of antibodies in Antiphospholipid antibody syndrome. These are antibody against lupus anticoagulant, anticardiolipin antibody and anti β2 glycoprotein-1 antibodies. In Antiphospholipid antibody syndrome, pathogenesis for thrombosis is binding of Antiphospholipid antibodies to endothelial cells which stimulate an up regulation of adhesion molecule increasing leucocyte adhesions and thus thrombosis.[2] Anticardiolipin antibodies interfere with anticoagulation pathway. Lupus anticoagulant lead to thromboembolic event rather than clinical bleeding.[3]

Case Report

A 28 years old gravida 7, abortion 5, IUFD1, presented to antenatal OPD, with one and half month of amenorrhea with urine pregnancy test positive. Patient previous records were checked. She had consecutive five missed abortion before 10 weeks of gestation. She was evaluated for recurrent pregnancy loss. When she was pregnant for 6th time, 2 years back her thrombophilia profile showed lupus anticoagulant positive. During that pregnancy, she was started on low dose aspirin 75 mg OD and low molecular weight heparin 40 mg subcutaneous daily in prophylactic doses as there was no history of thrombotic events. Patient was monitored throughout pregnancy for Antiphospholipid antibody syndrome related complications. There was no complication noticed. At around 35 weeks of gestation there was sudden intrauterine fetal demise despite of being on treatment. The cause of intrauterine fetal demise was not explained. Patient delivered vaginally, 2.6 kg, female baby. Placenta was sent for histopathology which revealed no obvious placental thrombosis. Patient was advised to repeat lupus anticoagulant after 6 weeks which again was positive. Patient conceived seventh time after one year. Again she was started on low dose aspirin 75mg OD and low molecular weight injection heparin 40 mg subcutaneous 24 hourly from 8 weeks of gestation after cardiac activity was documented on ultrasonography. She was monitored again for Antiphospholipid antibody syndrome complications by daily fetal kick count, non stress test and obstetric Doppler. Obstetric Doppler at 32 weeks of gestation was suggestive of mild uteroplacental but no fetoplacental insufficiency. Patient was followed up with Doppler after four weeks which was showing the same changes.
Patient delivered by cesarean section in view of precious pregnancy with baby weight of 2.7 kg, male child. Patient was discharged and was advised to continue low molecular weight heparin for 6 weeks at dose of 40 mg subcutaneous 24 hourly. The postpartum period was uneventful.

Discussion

Antiphospholipid antibodies causes activation of endothelial cells, monocytes and platelets causing overproduction of tissue factor, thromboxane A2 and complement activation.[2] All these lead to change in hemostatic system during normal pregnancy which results in hypercoagulable state. This leads to thrombosis and then to pregnancy complications associated with Antiphospholipid antibody syndrome. Antiphospholipid antibody trigger inflammatory response leading to trophoblast damage and so abnormal placentation.[4] Abnormal placentation which leads to  obstetric complication such as recurrent miscarriage, preterm delivery, oligohydramnios, prematurity, intrauterine growth restriction, fetal distress, fetal or neonatal thrombosis, preeclampsia, eclampsia, HELLP syndrome, arterial or venous thrombosis and placental insufficiency.[2] Obstetric Antiphospholipid antibody syndrome without thrombosis is currently managed by giving low dose aspirin and heparin.[5] Unfractionated heparin and low molecular heparin act by binding to antithrombin, to catalyze the molecule binding and altering the activity of serine protease procoagulants. There are more adverse outcomes with antibody against lupus anticoagulant.[6] Anticardiolipin antibody is associated with clinical bleeding. In our patient lupus anticoagulant was positive which might have lead to sudden intrauterine death even on treatment with low molecular heparin. There is continued occurrence of late pregnancy complications like preeclampsia, intrauterine growth restriction, prematurity and intrauterine fetal  demise despite treatment which suggest the need for strict monitoring of the disease and treatment-related complications throughout pregnancy.[7] In this pregnancy there was only uteroplacental insufficiency on low molecular weight heparin.
Lastly to conclude that severity of obstetric complications and thrombosis can be reduced by giving thromboprophylaxis with heparin, a close watch should be kept in the late pregnancy to prevent adverse outcomes though on heparin treatment. There might be a need of more dose effective regimen of heparin treatment.

References

1. Ruiz-Irastorza G, Crowther M, Branch W, Khamashta MA. Antiphospholipid syndrome.
Lancet 2010;376(9751):1498-1509.
2. Fosca A. F. Di Prima, Oriana Valenti, Entela Hyseni, Elsa Giorgio4, Marianna Faraci, Eliana Renda, Roberta De Domenico, Santo Monte. Antiphospholipid Syndrome during pregnancy: the state of the art. Journal of Prenatal Medicine 2011;5(2):41-53.
3. Henry Namme Luma, Marie-Solange Doualla, Elvis Temfack, Servais Albert Fiacre, Eloumou Bagnaka, Emmanuella Wankie Mankaa, Dobgima Fofung. The antiphospholipid antibody syndrome: a case report.International Medical Case Reports Journal 2012:563–67.
4. Stone S, Khamashta MA, Poston L. Placentation, Antiphospholipid syndrome and pregnancy outcome. Lupus 2001; 10: 67-74.
5. Sylvie Bouvier, E´ va Cochery-Nouvellon, Ge´ raldine Lavigne-Lissalde, E´ rick Mercier, Tess Marchetti, Jean-Pierre Balducchi, Pierre Mar`es and Jean-Christophe Gris. Comparative incidence of pregnancy outcomes in treated obstetric antiphospholipid syndrome: the NOH-APS observational study. Blood 2014;123(3):404-413.
6. Galli M, Luciani D, Bertolini G, Barbui T. Lupus anticoagulants are stronger risk factors for thrombosis than anticardiolipin antibodies in the Antiphospholipid syndrome: a systematic review of the literature.Blood 2003; 101: 1827-32.
7. Dadhwal V, Sharma AK, Deka D, Gupta B, Mittal S. The obstetric outcome following treatment in a cohort of patients with Antiphospholipid antibody syndrome in a tertiary care center. Journal of Postgraduate Medicine January 2011; 57:16-19.

Citation

Chakre S, Pardeshi S, Mayadeo NM, Shah S.  Antiphospholipid Antibody Syndrome. JPGO 2014 Volume 1 Number 9 Available from: http://www.jpgo.org/2014/09/antiphospholipid-antibody-syndrome.html

Cervical Lipoleiomyoma

Author Information

Jayashri Chaudhari*, Kanchan Kothari**, Alka Gupta***, Jyotsna Dwivedi****
(* Assistant Professor, ** Associate Professor, Department of Pathology, *** Professor, **** Second year Resident, Department of Obstetrics and Gynecology. Seth G.S. Medical College and K.E.M Hospital, Mumbai, India.)

Abstract

Lipoleiomyoma’s are fatty tumors of the uterus which are not commonly seen. Cervical lipoleiomyoma’s are even rarer then uterine lipoleiomyoma. We present an interesting case of a large cervical leiomyoma that not only was challenging to remove surgically but also presented a histopathological surprise when bundles of smooth muscle were seen to be interspersed with lobules of mature adipocytes.

Introduction

Fatty tumours primary to the uterus are very uncommon .The incidence varies from 0.03% to 0.2%. Lipoleiomyoma is a benign soft tissue tumor. It is more commonnly seen at extrauterine sites  either within the abdominal cavity or in the retroperitoneum. It can also befound in the muscular fascia and the subcutis.. [1] In the uterus, lipoleiomyomas are more common in the corpus [2] and are very rare in cervix with less than 15 cases of cervical lipoleiomyomata reported in literature. [2, 3, 4, 5, 6, 7, 8] We present a case of cervical lipoleiomyoma clinically mimicking malignancy.

Case Report

A  40 years old woman, having completed her family  presented  with complaints of something coming out of vagina since 6 months and hesitancy in passing urine since 2 months. Patient had undergone surgery for fibroid uterus 3-4 years back, and another for cervical polyp 2 years back. Operative details were not available.  There was no history of fever, pain in abdomen, or burning micturition or any other urinary complaints. On examination, her abdomen was soft. Speculum examination showed a large, non ulcerated mass. The mass appeared to be a fibroid growth.  On bimanual vaginal examination the whole anterior part of cervix was replaced with the fibroid.  Cervical rim or os could not be felt. Decision for total abdominal hysterectomy was taken. All preoperative serological, biochemical, radiological investigations  were within normal limits. After written, valid consent and peri operative parenteral antibiotics patients’ abdomen was opened by midline, infraumblical incision after administering spinal anesthesia. Intraoperatively there was difficulty in delivering out the uterus due to large posterior cervical fibroid growing downwards. Another small intramural fundal fibroid was also noted.  Total size of the uterus was around 12 weeks. Bilateral adnexa were normal. The upper pedicles of the uterus (round ligaments, cornual pedicles) could be divided and ligated easily. Bladder was low and not elevated by the cervical fibroid.Anterior fold of peritoneum could be identified and incised and the bladder was reflected off the isthmus of the cervix and the bladder was retracted.  Bilateral uterine vascular pedicles were divided and ligated. Supra cervical hysterectomy done and the cervical stump was then  held with long Allis forceps. The cervix was broad, and its lower end could not be felt. The whole cervix posteriorly, left laterally, anteriorly was replaced with the fibroid. Only a small area in the right anterior part did not have the fibroid extension. The cervix was debulked by bisecting it with an electrocautery and the fibroid was reached and enucleated from within its mucosal surface. The vagina was opened anteriorly on the right sided and a retrograde Maingot’s clamp was applied to the left angle of the vagina and then to the left uterosacral ligament. Both these pedicles were then divided and ligated. Fibroid was handed over to the nurse and sent for frozen section to exclude sarcoma changes as the gross examination of the fibroid did not show a whorled appearance or standard degenerative changes. Right vaginal angle was held with Allis forceps and divided and ligated. The bisected stump of the cervix on the right side was clamped laterally with Maingot’s clamp and divided and ligated and removed. The remaining surgery was completed by standard method and the patient was subsequently discharged on the 5th post operative day.  Frozen section was suggestive of degenerated cervical fibroid leiomyomatous polyp.


Figure 1: Gross photograph showing large cervical whitish firm mass with few cystic areas.

On microscopy the mass was covered by stratified squamous epithelium. The underlying tissue shows fascicles and bundles of smooth muscle with interspersed lobules of mature adipocytes. Few dilated endocervical glands were seen below the squamous epithelium and focally in the periphery amidst the tumor. There was no proliferation of endocervical glands There was no atypia or mitosis. The histological diagnosis was lipoleiomyoma of cervix. Sections from the myometrium showed two small subserosal leiomyomata.


Figure 2: (HE 400X) Fascicles of smooth muscle with intermixed mature adipocyte.

Discussion

Uterine lipoleimyomas are rare tumors and represent less than 0.2 % of benign neoplasms [6].  Most of them are reported in uterine corpus . A large study of 50 uterine lipoleiomyoma revealed that the mean patient age was 54 and  mean  tumor size was 4.6 cm. Forty-three (83%) tumors were located in the uterine corpus, and 7 (13%) were in the cervix.[8]. The immunohistochemical findings suggest a complex histogenesis for these tumors. They may develop either due to direct transformation of smooth muscle cells into adipocytes by means of progressive intracellular storage of lipids or from the immature mesenchymal cells surrounding the vessels [6 ] The consistent finding of chromosomes 12 and 14 on different derivatives in these tumor indicates that the t(12;14) was a primary event and immunohistochemical studies showed that HMGI-C was aberrantly expressed in this tumor indicating that uterine lipoleiomyomas have a pathogenetic origin similar to that of typical leiomyomas.[9] Lipomatous component of these tumors can be picked up by CT scan or MRI. [7] They behave like the usual leiomyomas and no recurrence or fatality due to tumor is reported [8]

Conclusion

Lipomatous tumors of the uterus are rare and that to cervix is very rare, only less than 15 cases of cervical lipoleiomyomas are reported. We present a rare case of cervical lipoleiomyoma mimicking malignancy. 

References

1.      Kumar S, Garg S, Rana P, Hasija S, Kataria SP, Sen R.  Lipoleiomyoma of Uterus: Uncommon Incidental Finding.  Gynecol Obstet 2013;3(2). Available from: http://omicsonline.org/lipoleiomyoma-of-uterus-uncommon-incidental-finding-2161-0932.1000145.pdf
2.      Walid MS, Heaten RL Case report of a cervical lipoleiomyoma with an incidentally discovered ovarian granulosa cell tumor – imaging and minimal – invasive surgical procedure GMS Ger Med Sci 2010; 8. Available from: http://www.egms.de/static/en/journals/gms/2010-8/000115.shtml#Abstract
3.      Terada T. Huge lipoleiomyoma of the uterine cervix. Arch Gynecol Obstet. 2011; 283(5):1169-71.
4.      Terada T. Giant Subserosal Lipoleiomyomas of the Uterine Cervix and Corpus: A Report of 2 Cases. Appl Immunohistochem Mol Morphol.  2012 Jan 26. [Epub ahead of print].
5.      Volpe R, Canzonieri V, Gloghini A, Carbone A. Lipoleiomyoma with metaplastic cartilage" (benign mesenchymoma) of the uterine cervix. Pathol Res Pract 1992; 188(6):799-801.
6.      Bolat F, Kayaselçuk F, Canpolat T, Erkanli S, TUNCER I. Histogenesis Of Lipomatous Component In Uterine Lipoleiomyomas Turkish Journal of Pathology 2007;23(2):82-86
7.      Fagouri H, Hafidi MR, Guelzim K, Hakimi I, Kouach J, Moussaoui DR, Dehayni M. Lipoleiomyoma Of The Uterine Cervix (About An Observation) International Journal Of Scientific & Technology Research 2014; 3 ( 3):449-450.
8.      Wang X, Kumar D, Seidman JD..Uterine lipoleiomyomas: a clinicopathologic study of 50 cases. Int J Gynecol Pathol 2006;25(3):239-42.
9.      Pedeutour F, Quade BJ, Sornberger K, Tallini G, Ligon AH, Weremowicz S, Morton CC. Dysregulation of HMGIC in a uterine lipoleiomyoma with a complex rearrangement including chromosomes 7, 12, and 14. Genes Chromosomes Cancer. 2000; 27(2):209-15

Citation

Chaudhari J, Kothari K, Gupta AS, Dwivedi J. Cervical Lipoleiomyoma. JPGO 2014 Volume 1 Number 9 Available from: http://www.jpgo.org/2014/09/cervical-lipoleiomyoma.html