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Editorial

Volume 1 Issue 1, January 2014

Editorial
Parulekar SV

Ovarian Failure with Uterine Artery Embolization
Mirchandani A, Parulekar SV.

 Hysterographic Assessment of Repair of Uterine Chronic Rupture
Sharma G, Samant PY, Parulekar SV.

 Ectopia Vesicae and Intrauterine Pregnancy
Mahajan J, Samant PY,  Parulekar SV.

 Free-Floating Intraperitoneal Mass
Gupta P, Parulekar SV.

 Genital Myiasis
Dhakne P, Gupta AS.

 Intramyometrial pregnancy
Kumari M, Gupta AS.

 Forgotten Vaginal Ring Pessary
Khadkikar R, Panchbudhe S, Channawar S, Chauhan AR.

 Difficult Vaginal Hysterectomy: A New Approach
Parulekar SV

Editorial

Parulekar SV
Editor-in-Chief

In a busy hospital practice, one gets to see a lot of patients. When the hospital is a tertiary level care center, like our center, one encounters a number of unusual cases. There is a lot to be learned from such cases, because their presentation and management are different from the descriptions in the standard textbooks. Unless these cases are documented, they tend to be forgotten, until one encounters a similar case again. Without documentation, knowledge of these cases is not available to others in the scientific community. Case reports need to be published so that these two deficiencies in learning are overcome. Then one day someone can review the literature and compile a series of all such cases, so that statistically significant conclusions can be drawn. Science makes progress in this way.
There is another deficiency in learning. Practice of conventional Gynecology and Obstetrics is based on textbook teaching and management algorithms. But new ideas are required for science to progress. Many people get new ideas. These ideas can be on anything – clinical tests, laboratory tests, instrumentation, or operative procedures. A few of these are brilliant. These thinkers need a platform to present their ideas, even before they go through the process of clinical studies, so that there can be brainstorming on them. The final version can then be put to critical evaluation in a scientific study.
Journal of Postgraduate Gynecology & Obstetrics is launched with these objectives in mind. We believe in free education for all, including publication of scientific content. Hence we have made this journal open source. Unlike many such journals on the net, it is totally free. There are no hidden charges, like processing fee, printing fee, color illustration charges etc. It is perhaps the only journal that readers can access for free and contributors can contribute to for free. This should help all those in the less privileged countries, though people in the privileged countries are also welcome to these resources.

Ovarian Failure with Uterine Artery Embolization

Author Information

Mirchandani AM*, Parulekar SV**, Samant PY***

(* Assistant Professor, ** Professor and Head of Department, *** Additional Professor
Department of Obstetrics and Gynaecology, Seth G.S. Medical College and K.E.M Hospital, Mumbai, India.)

Abstract
            Uterine artery embolization (UAE) has been performed to treat uterine leiomyomas conservatively. A case of premature ovarian failure resulting from UAE is presented and alternative treatment options are discussed.

Introduction
            Uterine leiomyomas can be treated medically or surgically. The use of UAE by interventional radiology is on the rise. However it may cause amenorrhea by causing ovarian and uterine damage. A case of 32 years old woman who developed secondary amenorrhea after UAE for uterine leiomyoma is presented and alternative treatment options are discussed.

Case Report
            A 32 years old nulliparous woman presented with secondary amenorrhea for 1 year. She had undergone UAE for uterine leiomyoma measuring 12 cm in diameter 4 years ago by another consultant. She continued to menstruate normally for another year, developed oligomenorrhea for 1 year, and then amenorrhoea for one year. Her general condition was fair and vital parameters were normal. Systemic examination revealed no abnormality. Abdominopelvic examination and pelvic ultrasonography revealed a small uterus with nonmeasurable endometrial thickness and an absence of uterine leiomyoma. Her serum FSH level was 96 mIU/ml. She had no evidence of other endocrine gland dysfunction clinically as well as on investigations. She was put on hormone replacement therapy and did well on it.

Discussion
            The blood supply of the uterus is through uterine arteries (branches of anterior divisions of internal iliac arteries) and ovarian arteries (branches of abdominal aorta). The uterine artery and ovarian artery anastomose with each other in the mesosalpinx near the uterine cornu. Three types of anastomoses are identified.[1] In type I (21.7%) flow from the ovarian artery to the uterus is through anastomoses with the main uterine artery. In type II (3.9%) the ovarian artery supplies the uterus directly. In type III (6.6%) the major blood supply to the ovary is from the uterine artery. The type III cases are classified as high risk of ovarian damage by UAE.
            Uterine leiomyomas can be treated medically with GnRH analogues or mifepristone. However the results are not lasting. Surgical treatment modalities include hysterectomy, myomectomy, myolysis, laparoscopic uterine artery occlusion, uterine artery embolization, and magnetic resonance imaging-guided focused ultrasound surgery.[2] UAE is an effective treatment alternative for uterine leiomyomas (98.97%).[3] However there are reports of ovarian damage with UAE.[3,4,5] The rate of ovarian failure after UAE is 1.23%[3] to 14%[6], which is lower than the rate of loss of ovarian perfusion demonstrated by doppler studies before and after UAE (54% total loss and 35% partial loss).[7] This is due to recovery of ovarian circulation. In the case presented, the patient had normal menses for two years after UAE. This was followed by oligomenorrhea for 1 year and then amenorrhea for 1 more year. She had suffered both ovarian damage and uterine damage from UAE. However the effects were delayed by 2 years after UAE, which is unusual. It is possible that her ovarian reserve was low at the initiation of treatment and UAE further depleted it. As a result, she got ovarian failure, which got initiated after a year and completed after two years.
            Amenorrhea after UAE is both of ovarian and uterine origin. Uterine damage is said to occur due to excessive embolization.[8] Ovarian damage appears to be due to nontarget organ embolization and possibly irradiation. Damage by embolization can be prevented by selective coil embolization of a uterine artery-to-ovarian artery communication before UAE. [9]
            We recommend that myomectomy should be the primary form of treatment of uterine leiomyomas in young women. If facilities are available, magnetic resonance imaging-guided focused ultrasound surgery may be used to achieve myolysis. UAE should be reserved for women when hysterectomy is the only option and it is not acceptable to the patient.
References
  1. Razavi MK, Wolanske KA, Hwang GL, Sze DY, Kee ST, Dake MD. Angiographic classification of ovarian artery-to-uterine artery anastomoses: initial observations in uterine fibroid embolization. Radiology. 2002 Sep; 224(3):707-12.
  2. Levy BS.Modern management of uterine fibroids. Acta Obstet Gynecol Scand. 2008; 87(8):812-23.
  3. Guo WB, Yang JY, Chen W, Zhuang WQ. Amenorrhea after uterine fibroid embolization: a report of six cases. Ai Zheng. 2008; 27(10):1094-9.
  4. Stringer NH, Grant T, Park J, Oldham L. Ovarian failure after uterine artery embolization for treatment of myomas.  J Am Assoc Gynecol Laparosc. 2000 Aug; 7(3):395-400.
  5. Hehenkamp WJ, Volkers NA, Broekmans FJ, de Jong FH, Themmen AP, Birnie E, Reekers JA, Ankum WM. Loss of ovarian reserve after uterine artery embolization: a randomized comparison with hysterectomy. Hum Reprod. 2007 Jul; 22(7):1996-2005.
  6. Payne JF, Robboy SJ, Haney AF. Embolic microspheres within ovarian arterial vasculature after uterine artery embolization. Obstet Gynecol. 2002 Nov;100(5 Pt 1):883-6.
  7. Ryu RK, Chrisman HB, Omary RA, Miljkovic S, Nemcek AA Jr, Saker MB, Resnick S, Carr J, Vogelzang RL. The vascular impact of uterine artery embolization: prospective sonographic assessment of ovarian arterial circulation. J Vasc Interv Radiol. 2001 Sep; 12(9):1071-4.
  8. Pelage JP, Cazejust J, Pluot E, Le Dref O, Laurent A, Spies JB, Chagnon S, Lacombe P. Uterine fibroid vascularization and clinical relevance to uterine fibroid embolization. Radiographics. 2005 Oct; 25 Suppl 1:S99-117.
  9. Wolanske KA, Gordon RL, Wilson MW, Kerlan RK Jr, LaBerge JM, Jacoby AF. Coil embolization of a tuboovarian anastomosis before uterine artery embolization to prevent nontarget particle embolization of the ovary.  J Vasc Interv Radiol. 2003 Oct; 14(10):1333-8.
Citation
Mirchandani A, Parulekar SV. Ovarian Failure with Uterine Artery Embolization. JPGO 2014 Volume 1 Number 1 Available from: http://jpgyob.blogspot.in/2014/01/ovarian-failure-with-uterine-artery.html

Hysterographic Assessment of Repair of Uterine Chronic Rupture


Author Information
Sharma G*, Samant PY**, Parulekar SV***
(* Third Year Resident, ** Additional Professor, *** Professor and Head of Department
Department of Obstetrics and Gynaecology, Seth G.S. Medical College and K.E.M Hospital, Mumbai, India.)


Abstract
Spontaneous rupture of uterus in a primigravida prior to onset of labor is extremely rare. The uterus needs to be conserved in such cases, if possible. The future obstetric outcome is influenced by quality of the scar  We report hysterographic findings in a case of spontaneous chronic upper segment rupture in 26 year old primigravida with 30 weeks of gestation.
Introduction
Poor healing of a uterine scar may be associated with complications in future pregnancies like scar pregnancy, morbidly adherent placenta, scar dehiscence or rupture.[1]  The healing may be related to the site of the rupture (upper segment versus lower segment), the type of rupture (acute versus chronic), the type of suture material, and/or the suturing technique. A defective scar increases the risk of recurrent rupture. Though the value of a hysterography in assessment of the integrity of uterine lower segment scars is unproved, it may be useful in assessment of upper segment scars in between pregnancies. An unusual case of healing of such an injury is presented.
Case Report
A 30 year old woman, married for 12 years, primigravida presented with 7.5 months of amenorrhea and complaints of watery vaginal discharge and pain in abdomen for 10 days. She had undergone diagnostic hysteroscopy and laparoscopy one year ago for evaluation of infertility. Operative details of the procedure were not available. On examination her vital parameters were normal. She had severe pallor. There was no tenderness, guarding or rigidity on abdominal examination. Uterine fundal height was of 30 weeks of gestation. The fetus was in left acromioanterior oblique lie. Clear amniotic fluid was seen to be draining from the cervix which was uneffaced and closed, Her hemoglobin was 6.2 g/dL, white cell count and C reactive protein levels normal. Ultrasonography showed a viable fetus of 25 weeks of gestation and estimated weight of 700 g in an oblique lie. It had an encephalocoele. Amniotic fluid index was zero. There was no evidence of peritoneal fluid collection. Four units of packed cells were transfused to correct the anemia. White cell count and C reactive protein levels were estimated every day. In view of prolonged rupture of membranes, an oblique lie, and rising levels of C reactive protein, a hysterotomy was performed under spinal anesthesia. A live female fetus weighing 650 g was delivered through a lower segment transverse incision. A small portion of the placenta was delivered out of the uterine incision. A 5 cm diameter defect was noted in the left fundal area of the uterus. The placenta and membranes were seen extruding from the defect. The uterine wall was thinned around the defect, and its edges were fibrotic and not bleeding. The sigmoid, transverse colon and a loop of small bowel were adherent to the posterior wall of the uterus below the defect. The placenta was adherent to the sigmoid colon. The adhesions between the uterus and bowel were released with the assistance of surgeons. A small part of the placenta was left attached to the sigmoid colon because it could not be separated safely. Small serosal tears in the sigmoid colon were repaired. The uterine rent was repaired with a continuous suture of polygalactin no. 1 excluding the decidua. The primary uterine incision was closed similarly. The patient was transfused with 4 units of packed cells and 4 units of fresh frozen plasma. She made an uneventful recovery.
The couple was counseled regarding the future risk of rupture of uterus at scar site and increased risk of maternal and perinatal morbidity and mortality. However the couple was keen on having another pregnancy. So a hysterography was performed after 6 months to assess healing of the scar. It showed multiple pockets of the dye in the myometrium at the site of the scar, and an intravasation of the dye (figure 1). There was no defect in the myometrium at the scar site.
Figure 1. Hysterogram: there are multiple pockets of the dye in the myometrium (solid arrows) and venous intravasation of the dye (hollow arrow).
Discussion
The incidence of uterine rupture increases in a case of scarred uterus depending on the type and site of incision or injury. There is a 32% risk of uterine rupture in a patient with prior upper segment rupture in comparison to 6% risk in a patient with lower segment rupture.[2,3] Rupture of unscarred uterus is associated with higher parity, prolonged labor, obstetric maneuvers (internal podalic version, manual removal of placenta, and instrumental delivery), inherent weakness of myometrium, disorders of collagen matrix (Ehler’s Danlos type IV), uterine anomalies, and placental abruption.[4] A spontaneous rupture of the uterus in a nulliparous woman is unusual, but may be seen if there has been uterine injury or surgery in the past. Hysterography can be used to evaluate integrity of scar. It may show invasion of the myometrium by the scar, pockets of the dye in the myometrium, and intravasation of the dye. The differential diagnoses of this appearance include prominent cervical glands, result of past myomectomy or curettage, uterine diverticula, and focal adenomyosis.[5,6]
In the case presented, the rupture of the uterus might have been due to uterine injury at the time of hysteroscopy and laparoscopy. It had been a slow rupture, so that the placenta got extruded and implanted on the sigmoid colon nearby. That also explained why she did not have features of internal hemorrhage and a hemoperitoneum at laparotomy. Multiple pockets of the dye in the myometrium at the site of the scar indicate ingrowth of the endometium into the myometrium as in adenomyosis, related to deficient endometrium locally. Venous intravasation was due to deficient endometrium. The thinning of the uterine wall locally and the radiographic appearance of the scar indicate a high risk of a rupture of the uterus in a future pregnancy, possibly at an even earlier gestational age.
References
  1. Naji O, Abdallah Y, Bij De Vaate AJ, Smith A, et al. Standardized approach for imaging and measuring Cesarean section scars using ultrasonography. Ultrasound Obstet Gynecol 2012;39:252–259.
  2. Reyes-Ceja L; Cabrera R; Insfran E; Herrera-Lasso F.  Pregnancy Following Previous Uterine Rupture: Study of 19 Patients. Obstetrics & Gynecology 1969;34:387-389.
  3. Ritchie EH: Pregnancy after rupture of the pregnant uterus: A report of 36 pregnancies and a study of cases reported since 1932. J Obstet Gynaecol Br Commonw 1971;78:642-648.
  4.  Turner M.J: Uterine rupture. Best Pract Res Clin Obstet Gynaecol 2002;16:69-79.
  5. Ahmadi F, Torbati L, Akhbari F, Shahrzad G. Appearance of uterine scar due to previous caesarean section on hysterosalpingography: various shapes, locations and sizes. Iranian Journal of Radiology. 2013;10:103-10. 
  6. Fabres C, Aviles G, De La Jara C, et al. The cesarean delivery scar pouch: clinical implications and diagnostic correlation between transvaginal sonography and hysteroscopy. J Ultrasound Med 2003;22:695-700


Citation
Sharma G, Samant PY, Parulekar SV. Hysterographic Assessment of Repair of Uterine Chronic Rupture. JPGO 2014 Volume 1 Number 1 Available from: http://jpgyob.blogspot.in/2014/01/hysterographic-assessment-of-repair-of.html

Ectopia Vesicae and Intrauterine Pregnancy


Author Information

Mahajan JS*, Samant PY**,  Parulekar SV***
(* Third Year Resident, ** Additional Professor, *** Professor and Head of Department
Department of Obstetrics and Gynaecology, Seth G.S. Medical College and K.E.M Hospital, Mumbai, India.)
Abstract
A rare case of ectopia vesicae and intrauterine pregnancy in a 25 years old, unmarried woman is presented. A medical termination of pregnancy was performed by dilatation and evacuation.
Introduction
Ectopia vesicae is a congenital anomaly in which there is failure of development of lower anterior abdominal wall. It occurs in 1 in 25000-50,000 people, the incidence in males being twice  as in females. It is seven times more common in babies conceived by in vitro fertilization. [1] The lower anterior abdominal wall and the anterior wall of the bladder are absent. The posterior bladder wall and the ureteric orifices are exposed. The bladder neck and urethra are poorly defined. The pubic symphysis  is widely separated.[2,3] There are reports of intrauterine pregnancy  and successful deliveries in patients with uncorrected ectopia vesicae. Cesarean section  at term gestation has always been preferred in such cases. A case of ectopia vesicae in a primigravida with 11 weeks of gestation managed by medical termination of pregnancy is presented.
Case report
 A 25  years  old unmarried woman presented with chief complaints of  amenorrhea for  3      months, pain in abdomen and generalized weakness for 1 week. She had an ultrasonographic scan showing an intrauterine gestation of 11 weeks. She gave a history of urinary incontinence from childhood and only spotting monthly from the orifice below the bladder from the age of puberty at the age of 14 years. There was a history of interfemoral sexual intercourse. On  abdominal examination, the umbilicus was absent. The open bladder base was at the lower end of the abdominal wall. An  opening was  present in the area of the mons pubis which was extremely tender to digital palpation,. The clitoris was absent, the labia minora were widely separated, and the pubic rami were absent. On per rectal examination, a bulky uterus was palpable anterior to rectum. One finger examination was done through the introital orifice. It was extremely painful. The uterus could be felt posteriorly, but its size could not be determined. The cervix  could not be felt.
Figure. 1. Ectopia vesicae. The pubic bones and labia minora  appear to be parts of lower abdominal wall. Pubic hair is seen on either side of midline. Introitus appears to be very small.

Her serum beta-hCG level was 97218 mIU/ml. Ultrasonography showed an intrauterine fetus of 11 weeks of gestation. Magnetic resonance imaging showed a single intrauterine gestation. The connection between the gravid uterus and the external orifice could not be defined.  It also could not be determined if the opening present on mons was vaginal or urethral.
Examination under general anesthesia showed that the  introit opening led to a cavity which had a normal appearing cervix. It was dilated to 9.5 mm. The products of conception were evacuated with ovum forceps. A blunt curettage was done. The patient made an uneventful recovery. She was given contraceptive advice and referred to a urologist for repair of the ectopia vesicae.
Discussion
 Ectopia vesicae or bladder extrophy occurs due to a failure of development of the lower anterior abdominal wall. That leaves the posterior wall of the bladder bulging like soft red swelling with multiple sites of excoriation on it. The condition should be surgically treated in childhood. A neglect may lead to its persistence in adult life, with increased risk of  ascending infection as openings of ureters are on the surface, and of development of malignancy in the exposed mucus membranes. In a combined series of 22 patients, there were 32 pregnancies, of which two aborted spontaneously and two were terminated medically. There were 27 live births and one intrauterine death of twins.[4] There were two case reports of continuation of the pregnancy to the third trimester and successful obstetric outcome was obtained by caesarean section.[5]  In the case presented, the pregnancy termination was performed for social reason (out of wedlock pregnancy) rather than ectopia vesicae.
References
1.      Wood  HM,  Trock  BJ,  Gearhart  JP:  In  vitro  fertilization  and  the cloacal-bladder exstrophy-epispadias complex: Is there an association? J Urol 2003;69:1512-1515.
2.      Gearhart JP, Ben-Chaim J, Jeffs RD, Sanders RC. Criteria for the prenatal diagnosis of classic bladder exstrophy. Obstet Gynecol 1994; 85:961-964.
3.      Woodhouse CRJ. The gynaecology of exstrophy. BJU International 1999; 83(S3):34-38.
4.      Woodhouse CRJ. Long term results of bladder exstrophy. In Gearhart JP, Mathews R (editors). The Exstrophy-Epispadias Complex: Research Concepts and Clinical Applications. First Edition. New York.  Kluwer Academic/Plenum Publishers. 1999; p 182.
5.      Mandal A, Chaudhuri S, Manna SS, Jana SK, et al. Successful pregnancy outcome in a woman with untreated ectopia vesicae: A case report and review of literature. J Obstet Gynecol Res 2013;39:868–871.

Citation
Mahajan J, Samant PY,  Parulekar SV. Ectopia  vesicae  and   intrauterine  pregnancy. JPGO 2014 Volume 1 Number 1 Available from: http://jpgyob.blogspot.in/2014/01/ectopia-vesicae-and-intrauterine.html

Free-Floating Intraperitoneal Mass


Author Information

Gupta P*, Parulekar SV**
(* Second Year Resident, ** Professor and Head of Department
Department of Obstetrics and Gynaecology, Seth G.S. Medical College and K.E.M Hospital, Mumbai, India.)
Abstract
A free-floating intraperitoneal mass is extremely rare, almost always discovered incidentally.
We present an anusual case of such a mass found incidentally during vaginal hysterectomy and vaginal wall prolapse repair. Pathological examination of the mass showed it to be a serous cystadenofibroma with autolytic changes. The origin of the mass was most probably one of the ovaries.
Introduction
Free-floating intraperitoneal masses are extremely rare. These abnormalities can be of unknown origin or can arise from various organs, most common being ovary due to autoamputation  following torsion or an appendix following infarction, an organised fat necrosis or an intra abdominal tumor. These are almost always found incidentally during surgery like an  exploration for an acute abdomen or abdominal distension or while investigating an unrelated disease. Ours is a case of a free-floating intraperitoneal mass discovered incidentally during vaginal hysterectomy of a 40 years old perimeopausal woman with uterine leiomyoma and vaginal wall prolapse. Both the ovaries were found to be normal. Pathological examination of the mass showed it to be a serous cystadenofibroma with autolytic changes. This is the first case of a free-floating intraperitoneal ovarian cyst in presence of normal ovaries.
Case report
A 40 years perimenopausal woman presented with complaints of menorrhagia and something coming out vaginally for four months. There was no history  of any episode of an acute pain in the abdomen. Her past medical and surgical history was not contributory. Her general condition was fair. General and systemic examination revealed no abnormality. Gynecological examination showed a uterus of 6 weeks’ size, moderate cystocele and moderate rectocele. Her hemogram and biochemical tests were normal. Abdominopelvic ultrasonography revealed a leiomyoma measuring 2 cm in diameter in the anterior wall of the uterus. There was no mass or free fluid. A vaginal hysterectomy with anterior colporrhaphy and posterior colpoperineorrhaphy was done under spinal anesthesia. After clamping, cutting and ligating the uterine vessels, a mass was noticed posterior and superior to the uterine fundus. After clamping, cutting and ligating the cornual structures on both the sides and removal of the uterus, the mass was found to be free-floating in the peritoneal cavity. It was removed easily. It was 8.0×4.5×5.0 cm in size, soft, and cystic (figure 1). Both the ovaries were found to be normal and in their normal positions. The patient made an uneventful recovery. Pathological examination showed a cystic mass with 20 ml of pultaceous material in it, suggestive of autolytic changes. Multiple nodulopapillary exascences were identified . Morphologically it resembled of  serous cystadenofibroma. Histopathological examination was uninformative due to autolytic changes in the mass.
Figure 1. Free-floating intraperitoneal mass.
Figure 2. View of pelvic cavity after vaginal hysterectomy. Both the ovaries (arrows) are normal and seen in normal positions.
Discussion
Free floating intraperitoneal masses are exceedingly rare masses. Such a mass may be the result of an auto amputation of an ovary (normal or with a cyst or tumor) following its torsion,[1,2] a mass from the appendix separated after auto amputation, infarcted appendices epiploicae, a hydatid cyst,[3] a mass from the rectum,[4] or organised fat necrosis or hematoma.[5] Such patients would have a history of an acute abdomen in the past, which resolved with conservative treatment. Most of these masses are detected incidentally at the time of an abdominal operation for some other indication. The concerned organ like ovary or appendix is absent in such cases. The case presented was unusual in that the structure of the mass was that of an ovarian serous cystadenofibroma, but both the ovaries were normal. Probably it was a tumor which had developed at a pole of ovary, the part between the tumor and the rest of the ovary being constricted due to past ovulations and scarring. This must have been the site of torsion of the mass, which caused necrosis of the connection and the separation of the mass. The rest of the ovary remained behind and survived. Another possibility is that it arose from ectopic ovarian tissue of embryological origin, a supernumerary ovary.[6,7] It is not possible to determine which of the two was the source of the mass. Irrespective of the source, all loose free-floating masses in the peritoneal cavity should be removed, as they can get infected.
References
1.      Currarino G, Rutledge JC. Ovarian torsion and amputation resulting in partially calcified, pedunculated cystic mass. Pediatric Radiology. 1989;19(6-7):395–399.
2.      Koike Y, Inoue M, Uchida K, Kawamoto A, et al. Ovarian autoamputation in a neonate: a case report with literature review.  Surgery International 2009;25(7):655-8.
3.      Çiftçi İ, Yılmaz H.Hepatic Hydatid Cyst and Intraperitoneal Free Hydatid Cyst. Eur J Gen Med 2012;9(Suppl 1):50-52.
4.      Takada1 A, Moriya1 Y, Muramatsu Y, Sagae T. A Case of Giant Peritoneal Loose Bodies Mimicking Calcified Leiomyoma Originating from the Rectum. Japanese Journal of Clinical Oncology 1998;28:441-442.
5.      Murat FJL, Gettman MT. Free-floating organized fat necrosis: rare presentation of pelvic mass managed with laparoscopic techniques. Urology. 2004;63(1):176–177.
6.      Litos MG, Furara S, Chin K Supernumerary ovary: a case report and literature review. J Obstet Gynaecol 2003, 23(3):325-7.
7.      Whitaker C, Tawfik O, Weed JC Jr: Serous cystadenoma arising in an ectopic ovary. Kans Med 1997, 98(2):24-6.

Citation
Gupta P, Parulekar SV. Free-Floating Intraperitoneal Mass. JPGO 2014 Volume 1 Number 1 Available from: http://jpgyob.blogspot.in/2014/01/free-floating-intraperitoneal-mass.html

Genital Myiasis


Author Information
Dhakne P*, Gupta AS**
(* Third Year Resident, ** Professor
Department of Obstetrics and Gynaecology, Seth G.S. Medical College and K.E.M Hospital, Mumbai, India.)

Abstract

A 65 year old woman presented to Out patient department (OPD) with uterovaginal prolapse for 2 years, reducible initially but irreducible since 3 months and associated with foul smelling discharge for a fortnight. Third degree prolapse with necrotic ulcerated areas and numerous maggots moving in and out of the ulcerated prolapsed mass were seen on local examination. The patient was managed conservatively with removal of maggots with 100% turpentine oil and culture sensitive antibiotics. As the ulcer failed to show any signs of healing & prolapse remained irreducible, ulcer margin was biopsied. Biopsy showed squamous cell carcinoma cervix.

Introduction

Myiasis is derived from the Greek word ‘Myia’, meaning ‘fly’.[1]  Myiasis is the infestation by dipterous larva (maggots) of various fly species which feed on a living host or necrotic tissue. Cutaneous, nasopharyngeal, ophthalmic myiasis are common. Genital myiasis is a rare condition. We are reporting a rare case of genital myiasis in a woman of irreducible utero-vaginal prolapse with squamous cell carcinoma of cervix.

Case Report

A 65 years old multiparous, postmenopausal woman came in OPD with complaints of something coming out of vagina for 2 years, reducible initially which became irreducible for 3 months and associated with foul smelling discharge for 15 days. The patient belonged to poor socioeconomic condition, living alone and working as a daily wage worker in a paddy farm. On examination her general condition was fair. She was afebrile, and besides being mildly anemic her general and systemic examinations were within normal limits. There was no local lymphadenopathy. Her abdomen was soft, neither tender nor guarded. On local examination, third degree uterovaginal prolapse was seen. External os could not be easily identified. Prolapsed mass of cervix and vagina of approximately 9 cm x 7 cm was seen out of the introitus as shown in Figure 1.

Figure 1: Large ulcerative squamous cell carcinoma in a prolapsed cervix with maggots’ infestation. Solid white arrow indicates a maggot. Small white arrow shows limits of the ulcerated area.

It was irreducible. There were multiple necrotic ulcers over the prolapsed mass associated with foul smelling discharge and numerous maggots were seen moving in and out of this ulcerated tissue. Uterine size could not be ascertained. Bilateral parametrium felt indurated , but rectal mucosa was free on rectal examination. Her haemoglobin was 9 gm/dL,  white blood cell count 10,000/mm3, HIV and VDRL negative, urinalysis normal, renal and liver function tests normal. No growth was seen on urine culture. Ultrasonography of  the abdomen and pelvis was normal. Microbiologic study of the ulcer showed a growth of E. coli and Klebsiella sensitive to levofloxacin. She was treated with levofloxacin orally 400 mg qd for 14 days. The prolapsed mass was cleaned daily with normal saline and maggots were removed after application of 100% turpentine oil to the wound. Deep seated maggots would swarm up after irrigation of the ulcer with turpentine due to irritation and were then washed away with saline. Daily glycerine-MgSO4 dressing was applied over lateral and posterior aspect of the cervix. Glycerine and acriflavin dressing was applied over the ulcerated area. At the end of the 7th day the wound was free of maggots. However, the ulcer showed no signs of healing and neither the prolapse could be reduced. The cervix remained edematous. The margins of the ulcer were thick and indurated. A biopsy was taken from margins of the ulcer. It revealed a squamous cell carcinoma. The parametrium was indurated bilaterally indicative of FIGO stage IIb of carcinoma cervix. The patient was referred to oncology center for radiotherapy. Unfortunately, the patient was then lost for follow up.

Discussion

‘Myiasis’ was the name given in 1840, by Reverend Frederick William Hope to diseases resulting from dipterous larvae.[2] Though the term myiasis was first used in 1840, such conditions have been recorded even in ancient times.  Chief surgeon, Ambriose Pare, to King Henry III and King Charles IX observed that maggots often infested open wounds.[3]  Myiasis is most commonly reported from subtropical, tropical, and warm temperate regions even though it is globally distributed. Larvae may infect and feed dead, dying, necrotic or living tissue in various sites, like the skin, eyes, ears, stomach, intestinal tract, or genitourinary sites.[4] They may invade open injuries and wounds, lesions or unbroken skin. Some enter the body through body orifices like nose or ears. In present case there was genital infestation.
Genital myiasis in a woman is usually associated with poor general health and hygiene, restricted mobility, and ulcerating lesions.[5] The fetid odor possibly attracts the female flies who lay eggs in existing lesions.[6] Eggs hatch into the larvae. Larvae invade the tissues. As the larvae are photophobic they deeply penetrate the tissue by their sharp oral hooks and anchoring intersegmental spines and feed themselves on living tissues. Pupa emerges within 1-2 days and matures into an adult after 6 days.[7] The larvae secrete a toxin resulting in inflammation and it prevents tissue healing.[8] Myiasis is more likely to be associated with  sexually transmitted diseases and in immunocompromized patients.[9] Urogenital myiasis  can be external or internal based on the anatomical site.[10] External urogenital myiasis is common in women. The practices of not wearing undergarments, cervical carcinoma, or sexually transmitted diseases are conditions which predispose women to external myiasis. In women clitoris, vulva, urethra, vaginal cavity, and uterus have been affected and all reported cases have been associated with uterine prolapse. Internal urogenital myiasis is rare and it occurs when the larvae reach internal genitourinary organs. Burning micturition, loin pain, vaginal bleeding are some of the complaints of these patients. Symptoms subside after the expulsion of maggots.
The above case was an interesting case of external genital myiasis. This woman was from low socioeconomic strata. Her hygiene was poor and she did not wear any undergarment. She would squat to work in the paddy fields dressed in a sari which most likely allowed the adult fly to gain access to the prolapsed cervix; and lay eggs. The hot and humid climate of India facilitated their hatching and cervical ulcer got infested with maggots. Carcinoma of the cervix provided sufficient necrotic debris for the larvae to feed on and multiply. Literature search gave only one article of genital myiasis associated with prolapse and vaginal malignancy.[11]   
The treatment is simple and involves usage of antilarval measures (turpentine oil or mixture of turpentine oil and chloroform) followed by the removal of the larvae. Other agents like ether, chloroform, or surgical debridement have been used for removal of the larvae.[12] In the present case we used 100% turpentine oil, local antiseptics and antimicrobial agents along with culture specific antibiotics. Turpentine oil creates an anaerobic environment forcing the maggots to wriggle out to the surface and expel out or they can be washed away.[10] Though our treatment made the wound clean, it was not healing and we suspected malignancy, which was confirmed on biopsy.
 
Conclusion
We are reporting this case as genital myiasis is rare and it is still rarer when associated with advanced malignancy. Cleaning the external wound with turpentine clears the maggots from the wound, prevents extensive tissue destruction and allows wounds to heal. But when despite being free of maggots the wounds do not heal a possibility of underlying malignancy should be considered and evaluated. 
 
References
  1. Burns T, Breathnach S, Cox N, Griffiths C. Diseases caused by arthropods and other noxious animals. In:Rook's Textbook of Dermatology. Vol 2. 7th ed. Malden, MA: Blackwell Publishing; 2004:33.8 - 11.
  2. John, David and Petri, William. Markell and Voge’s Medical Parasitology: 9th Edition. Missouri: Saunders Elsevier, 2006. p. 328-334.
  3. Sherman, RA, Hall, MJR, and Thomas, S. “Medicinal Maggots: An ancient remedy for some contemporary afflictions.” Annual Review of Entomology 45 (2000): 55-81.
  4. Ockenhouse, Christian, Samlaska, Curt, Benson, Paul, Roberts, Lyman, Eliasson, Arn, Malane, Susan, and Menich, Mark. “Cutaneous myiasis caused by the African tumbu fly.” Archives of Dermatology 126 (1990):199-202.
  5. Meinhardt W. Urogenital myiasis caused by scuttle Flu larvae (Diptera:Phoridae). Br J Urol 1989;64:547-8.
  6. Passos MR, Carvalho AV, Dutra AL, Goulart Fiho RA, Barreto NA, Salles RS, et al. Vulvar Myiasis. Dis Obstet Gynecol 1998;6:69-71.
  7. Ramalingam S, Nurulhuda A, Bee LH. Urogenital Myiasis by chrysomya bezziana (Diptera: Calliphoridae) in peninsular Malaysia. Southeast Asian J Trop Med Public Health 1980;11:405-7.
  8. Kersten RC, Shoukrey NM, Tabara KF. Myiasis. Opthalmology 1986; 93:1228-32.
  9. Singh I, Gathwala G, Yadav SP, Wig U. Ocular myiasis. Indian Pediatr 1991; 28: 1524-5.
  10. Fabio Francesconi and Omar Lupi. Myiasis. Clin Microbiol Rev. 2012 January; 25(1): 79–105.
  11. Baidya J. A rare case of genital myiasis in a woman with genital prolapse and malignancy and review of the literature. Ann Trop Med Public Health [serial online] 2009 [cited 2013 Dec 10];2:29-30. Available from  http://www.atmph.org/text.asp?2009/2/1/29/64274
  12. Lebwohl MG, Heymann WR, Berth-Jones J, Coulson I. Myiasis. In: Treatment of Skin Diseases. Comprehensive Therapeutic Strategies. 2nd ed. Elesevier-Mosby; 2006:420-421.
Citation
Dhakne P, Gupta AS. Genital Myiasis. JPGO 2014 Volume 1 Number 1 Available from: http://jpgyob.blogspot.in/2014/01/genital-myiasis.html 

Intramyometrial pregnancy


Author Information
Kumari M*, Gupta AS**
(* Third Year Resident, ** Professor
Department of Obstetrics and Gynaecology, Seth G.S. Medical College and K.E.M Hospital, Mumbai, India.)
Abstract
An interesting case of  intramyometrial pregnancy managed conservatively with methotrexate is reported. Patients condition was not diagnosed for 9 months. She underwent multiple Dilatation and curettage procedures for retained products of conception and was again misdiagnosed as a case of arterio-venous malformation for which she was referred to us; a clinical suspicion combined with a MRI confirmed the suspicion of an intramyometrial pregnancy. As the patient was young and desired childbearing and was averse to further surgical management she was successfully managed by methotrexate.
Introduction
Intramyometrial pregnancy or intramural pregnancy is a rare type of ectopic pregnancy and is very difficult to diagnose. Its exact incidence is unknown due to its rare occurrence and the fact that only isolated case reports are  documented  till date. The fertilized ovum or the conceptus implants in the myometrium and the gestational sac is located within the uterine wall wholly surrounded by the myometrium and unconnected to the uterine cavity, fallopian tube, or round ligament.[1]

Case Report
  A 26 years old gravid 2, abortion 1 was referred to us in February 2013  in view of uterine arteriovenous malformation. She complained of pain in abdomen and 6 weeks of amenorrhea. Her menstrual cycles had been irregular for preceding 9 months.  She had undergone a blunt curettage in April 2012 after a spontaneous abortion. Curettings were not sent for histopathological examination. Dilatation and curettage was again done in July 2012 as she continued to bleed irregularly, ultrasonography suggested retained products of conception and serum β-HCG level was 25,000 mIU/ml.  The procedure was repeated by another consultant. Histopathological report showed no chorionic villi. Her complaints persisted. A pelvic ultrasonography in December 2012 showed uterine arterio-venous malformation and she was then referred to us. Her vital parameters were within normal limits. General and systemic examination showed no abnormality. She was mildly anaemic. Her abdomen was soft. Vaginal examination showed no pelvic tenderness or masses. The uterus was of 12 weeks size, fundus bulky, soft, and anteverted. There was no active bleeding. Ultrasonography showed retained products of conception. Serum β-HCG value was 25,900 mIU/ml. Her haematological indices, serum chemistry, and chest radiograph were all within normal parameters. With a diagnosis of an incomplete abortion, a blunt curettage was done. In view of scanty curettage sample, an intramyometrial pregnancy was suspected. MRI was done. It showed a well defined rounded lesion seen in anterior myometrium measuring  3.36x3.23x2.9 cm size in anteroposterior, transverse and longitudinal dimensions. Multiple cystic spaces are seen within it with foci of haemorrhage. Post contrast imaging revealed peripheral enhancement of the lesion with homogenous enhancement of the rest of the lesion. Endometrial cavity was empty and not connected to the this mass. Engorged parametrial vessels were seen on the left side. Both ovaries were normal. These findings were suggestive of  an  intramyometrial pregnancy with cystic changes and haemorrhage inside. Histopathological study of the curetting’s did not show any chorionic villi.
Figure 1. MRI of the uterus. C: is the empty endometrial cavity; IM:  the intramural pregnancy in its Gestational sac.
In view of stable condition of patient and patient and her husband’s desire for future childbearing and her aversion to further surgery conservative management was started. Methotrexate 50mg/m 2 was administered intramuscularly. Her serial serum β-HCG  values fell progressively reaching less than1.2 mIU/ml at end of 3 months. Ultrasonography at that time showed calcification of lesion which had decreased in size  to 2x2 cm . Her regular menstrual cycles resumed.
Discussion
An intramyometrial pregnancy is associated with factors causing trauma to the endometrium disrupting its continuity thus technically creating a path for the conceptus to gain entry into the myometrium for implantation. Such trauma occurs in a caesarean section , curettage, misplaced IUD, adenomyosis, placenta accreta, and necrosis of a uterine leiomyma after uterine artery embolization.[2, 3, 4] It may follow in vitro fertilisation.[5] Usually the diagnosis of intramural pregnancy cannot be made until uterine rupture  occurs, which usually occurres between about 11 and 30 weeks of  gestation. An intramural pregnancy is difficult to be  seen on  ultrasonography and  is easily confused with  a degenerating myoma , products of conception or congenital uterine anomaly.[3,5] In a hemodynamically stable patient who wants a future pregnancy, this condition  can be managed conservatively by local administration of KCL or Methotrexate or by systemic methotrexate either in single dose or as multiple dose regimen. [2,6,7,8] A  nonradical  surgical management in form of wedge resection or enucleation with myometrial reconstruction can be performed too. A patient with uterine rupture and hemodyamic instability requires a hysterectomy.

References
1.      Ginsberg K, Quereshi F, Thomas M, Snowman B. Intramural ectopic pregnancy implanting in adenomyosis. Fertil Steril 1989;51:354–6.
2.      Lu HF, Sheu BC, Shih JC, Chang YL, et al. Intramural ectopic pregnancy sonographic picture and its relation with adenomyosis. Acta Obstet Gynecol Scand 1997;76:886–9.
3.      Hsieh YY, Chang CC, Tsai HD, Yeh LS, et al. Intramural pregnancy with negative [beta]-hCG. J Reprod Med 1998;43:468–70.
4.      Bazin F, Compton BC. Intramural pregnancy. Am J Obstet Gynecol 1957;73:1141–2.
5.      Cava EF, Russell WM. Intramural pregnancy with uterine rupture. Am J Obstet Gynecol 1978;131:214–6.
6.      Fait G, Goyert G, Sundareson A, Pickens A Jr. Intramural pregnancy with fetal survival: Case history and discussion of etiologic factors. Obstet Gynecol 1987;70:472–4.
7.      Hamilton CJCM, Legarth JL, Jaroudi KA. Intramural pregnancy after in vitro fertilization and embryo transfer. Fertil Steril 1992;57:215–7.
8.      Neiger R, Weldon K, Means N. Intramural pregnancy in a cesarean section scar. J Reprod Med 1998;43:999–1001.

Citation
Kumari M, Gupta AS. Intramyometrial pregnancy. JPGO 2014 Volume 1 Number 1 Available from: http://jpgyob.blogspot.in/2014/01/intramyometrial-pregnancy.html