Volume 2 Issue 5, May 2015

Editorial
Gupta AS

Large Hematosalpinx Secondary To Unicornuate Noncommunicating Uterine Horn
Goel A, Walasangikar VB, Pawde A, Chauhan AR.

Retained nude Copper T
Puri J, Gupta AS.

Laparoscopic Repair of Hydrocele of Canal of Nuck
Borse M, Kotekar O, Prasad R.

Vaginal Epidermal Cyst In Pregnancy
Madhva Prasad S, Mhaske N, Kharat D, Fonseca MN.

Primary Tubal Gestational Choriocarcinoma Mimicking Tubal Ectopic Pregnancy
Mahajan N, Shende D, Shirodkar S.

A Giant Ovarian Cyst: A Rare Case Of Mucinous Cystadenocarcinoma Of The Ovary In A Teenage Girl
Rathod S,  Vijayalakshmi S.

Aneurysmal Aortoarteritis And Fetal Death
Dalvi P, Parulekar SV, Mirchandani AM.

Vesicophobia during vaginal hysterectomy- Opening Anterior Pouch Through Posterior Colpotomy
Tanksale SJ, Parulekar SV.

Hysterectomy For Cervical Atresia
Parulekar SV

Quiz April 2015

Editorial

Gupta AS

Cervical atresia is fortunately a rare congenital Mullerian anomaly with poor results after surgical corrections. Vaginal aplasia is associated in 50% of these cases. Young, adolescent girls with a functional uterus above an atretic or a dystretic cervix underwent repeated unsuccessful attempts at reconstruction of the cervix which eventually led to a hysterectomy. This was the fate of most of these teenagers almost a decade or two back. The problem was of constructing a patent cervical canal and maintaining its patency permanently. The reconstructed cervix would close down despite use of intracervical stents like Foley catheters as the cervical mucosa would not regenerate or could not be created.
Over the last 2 decades many successful attempts have been made to establish permanent patency of the functional uterus to the exterior. These reconstructive plastic procedures include excision of the atretic part of the cervix and directly anastomosing the uterus to the vagina, use of bio medical grafts like amniotic membrane, vaginal mucosa, porcine small intestine submucosa, patients' own colon and even full thickness skin graft. Stents used were Foley catheters, silicone stents and polytetrafluroethylene Gore-Tex stents.
Some patients have isolated cervical atresias but a patent vagina. The other category which poses greater management challenge is that of an adolescent with cervical atresia and vaginal aplasia. In such cases there is a need to not only establish patency of the uterus to the exterior but also perform a vaginoplasty to establish a functional vagina.
The various methods attempted to restore functional and reproductive capability of these individuals include cervical drilling and canalization procedures, uterovaginal anastomosis, uterovestibular anastomosis, uterocolonocovaginoplasty performed either through a standard laparotomy or laparoscopically. Anastomosis are preferred over canalization procedures  even though they are easy to perform as the incidence of re-stenosis is very high (40-60 % ) with drilling and canalization procedures. This results in multiple repeat surgeries, increased morbidity and eventually leads to a hysterectomy.
In patients with only cervical atresia, or associated vaginal aplasia successful restoration of menstrual, sexual and reproductive function have been reported in numerous studies by various authors by performing uterovaginal anastomosis or uterovestibular anastomosis. Restenosis, infections are real morbidities which still require hysterectomy. 
These procedures are usually performed in a semi-lithotomy position to allow concurrent abdominal and vaginal procedure. After these procedures patients have good vaginal depth for sexual function and pregnancies have been reported naturally or after use of assisted reproductive techniques. All patients who have delivered have delivered by elective cesarean section. No vaginal births have been reported. Since the procedure of uterovaginal anastomosis or uterovestibular anastomosis involves incising the uterine fundus elective prelabor cesarean section is safer for childbirth.
Reviewing the literature, studying the treatment options and noting the success rate of the uterovaginal anastomosis procedures it is heartening to see that an option for conservative treatment is a reality for the adolescent girl with cervical atresia.  Early diagnosis, desire to save the uterus, restore menstrual, sexual and reproductive function either spontaneous or with assisted reproductive techniques, opting for uterovaginal anastomosis as the 1st line of treatment can change the grim outlook for these adolescent girls who are standing at the brink of their womanhood and save them from a hysterectomy.

Large Hematosalpinx Secondary To Unicornuate Noncommunicating Uterine Horn

Author Information

Goel A*, Walasangikar VB*, Pawde A**, Chauhan AR***.
(*Second Year Resident, ** Fourth Year Resident, *** Additional Professor, Department of Obstetrics and Gynecology, Seth GS Medical College & KEM Hospital, Mumbai, India.)

Abstract

Unicornuate uterus with a rudimentary horn is the rare congenital anatomic anomaly of the female genital system (incidence of 1/100,000), causing many obstetric and gynecological complications.[1] We report a case of huge hematosalpinx secondary to unicornuate non-communicating horn presenting as lump in abdomen in a teenage girl.

Introduction

The reported incidence of congenital uterine anomalies is 0.5 – 1%.[2] Unicornuate uterus is classified as Type II mullerian duct anomaly by the American Fertility Society.[3] The rudimentary horn is connected to unicornuate uterus with a fibrous or fibromuscular band in 10-15% of cases.[4] The diagnosis of this anomaly is usually delayed, as it remains asymptomatic and initial symptoms are non-specific. It may be diagnosed due to complications such as hematometra, endometriosis, infertility and ectopic pregnancy.

Case Report

A 14 year old girl presented with dysmenorrhea for last 6 months and pain in abdomen for last 3 months, both of which had increased in intensity for last 2 months. The pain started a day prior to menses, continued during menses and was associated with vomiting. She also complained of a lump in the lower abdomen for last three months. She had attained menarche at the age of 11 years and her menstrual cycles were regular.
On examination, general and systemic examinations were normal. On abdominal examination a firm, non-tender, irregular and immobile mass arising from the pelvis in midline was palpable corresponding to 18-20 weeks’ uterine size. Speculum and vaginal examination was not done as the patient was unmarried and hymen was intact. The patient was not willing for rectal examination. Our clinical impression was ovarian cyst or more unlikely, a large endometrioma.
Ultrasound was suggestive of a uterine horn on the right side in pelvis with normal size, shape and normal endometrium, but with a displaced axis. Another uterus- like structure was seen on left side at a higher location suggestive of second horn with contents within the endometrial cavity with fluid levels and echoes suggestive of hematometra. This was seen to be draining into a large elongated tubular structure (20 x 15 x 7 cm) with partial septae with echoes within it, most likely representing hydro- or hematosalpinx. Bilateral ovaries were normal. Mild hydronephrosis and hydroureter of left kidney were seen; right kidney was normal. Her routine hematological and biochemical investigations were within normal limits. CA 125 level was not elevated indicating non endometriotic/ benign lesion. Magnetic resonance imaging (MRI) was suggestive of bicornuate uterus with obstructed left horn not communicating with the cervix resulting in hematometra and hematosalpinx. Left horn was displaced anterosuperiorly abutting the anterior abdominal wall in left iliac fossa and normal appearing right horn along with cervix was seen in situ.


Figure 1. T2 weighted coronal MRI image showing unicornuate uterus in communication with hematosalpinx.


Figure 2. T1 weighted axial MRI image showing unicornuate uterus and hematosalpinx,


Figure 3. T2 weighted coronal MRI image showing unicornuate uterus and hematosalpinx.

On exploratory laparotomy, a unicornuate uterus was seen in continuation with a normal functional cervix on the right side. Right fallopian tube and ovary were normal. Left rudimentary horn was seen connected to the uterus with a fibromuscular band. Left fallopian tube was retort shaped, distended, tense, 16 x 10 x 8 cm in size, lying anterosuperior to the rudimentary horn with agglutinated fimbrial end. It was class [FaL 16 cm - UtR 5 cm Fn NoObst - UtL1 3 cm Fn Obst (left hematosalpinx)] by the new EAC classification of congenital malformations of the female genital Tract.[5]


Figure.4 Intraoperative image showing unicornuate uterus, fibromuscular band, rudimentary horn in communication with left hematosalpinx with agglutinated fimbrial end and left ovary.

Excision of left rudimentary non-communicating horn with salpingectomy was done: left round ligament was clamped, cut and transfixed; fibromuscular band was clamped, cut and transfixed using polyglactin No.1. A window was created in the broad ligament and mesosalphinx was dissected by sharp dissection. Left ovary was separated from the rudimentary horn by sharp dissection and cauterization. Left ovary with its vascular pedicle was conserved. Successive clamps were applied in the meso-salpinx to remove the left rudimentary horn with hematosalphinx. Extra-peritonization of broad ligament stump and left round ligament was done.


Figure 5. intraoperative image showing unicornuate uterus and conserved left ovary.

Post-operatively the patient recovered uneventfully and resumed normal cycles and without dysmenorrhea. Histopathology report confirmed a rudimentary horn showing pre-pubertal changes with left sided hematosalpinx.

Discussion

The differential diagnosis of acute abdominal pain in adolescent girls should include mullerian anomalies. Although the incidence is low, evaluation and treatment should be performed expeditiously as the conditions can pose risk to life or future fertility. Although hematosalpinx is not common in teenage years, its prompt treatment is needed.
Physical examination may not diagnose these anomalies accurately; imaging modalities such as ultrasound, Hysterosalpingography HSG and MRI are important tools for diagnosis. Typically, HSG films show a deviated banana-shaped cavity with a single fallopian tube, Hematosalpinx  is also diagnosed accurately with HSG. Rudimentary uterine horn in association with a unicornuate uterus is best diagnosed by sonography.[6] MRI is useful for early diagnosis.
As unicornuate uterus with functional uterine horn is associated with complications such as endometriosis, infertility, ectopic pregnancy; excision of a cavitary rudimentary horn is indicated.[7] Whereas, excision of nonfunctioning horn (solid, with no functional endometrium) is not routinely recommended, as no adverse effects are reported. Salpingectomy or salpingo-oophorectomy on the side with the rudimentary horn, however, has been suggested to prevent ectopic pregnancy in women with a unicornuate uterus.
Hematosalpinx in unicornuate uterus secondary to backflow from an obstructed horn leading to endometriosis as well as adenomyosis is known. However, occurrence of a large hematosalpinx like in the case presented here is rare due to limited capacity of the fallopian tube to distend as well as its communication with the peritoneal cavity. Such huge hematosalpinges tend to undergo torsion, though this was not seen in our case. Exploratory laparotomy with de-torsion  of hematosalpinx  may help to conserve the tube. However in our case, large size as well as agglutinated fimbriae warranted salpingectomy along with excision of non-communicating rudimentary horn. 

References
  1. Atmaca R, Germen AT, Burak F, Kafkasli A. Acute abdomen in a case with non-communicating rudimentary horn and unicornuate uterus. JSLS 2005; 9(2): 235–237.
  2. Khanna V, D'Souza J, Tiwari SS, Sharma RK, Shrivastava AK. Unicornuate uterus with an obstructed rudimentary horn: A report of two cases with an imaging perspective Medical Journal Armed Forces India. 2013; 69(1): 78–82.
  3. The American Fertility Society. The American Fertility Society classifications of adnexal adhesions, distal tubal occlusion, tubal occlusion secondary to tubal ligation, tubal pregnancies, Müllerian anomalies and intrauterine adhesions. Fert Ster. 1988; 49:944–955.
  4. Goel P, Aggarwal A, Devi K, Takkar N, Saha PK, Huria A, Unicornuate uterus with non-communicating rudimentary horn – different clinical presentations, Journal of Obstetric Gynaecology India 2005;55: 155-158.
  5. Parulekar SV. Classification Of Congenital Malformations Of The Female Genital Tract. JPGO 2015. Volume 2 No. 4, Available from: http://www.jpgo.org/2015/04/eac-classification-of-congenital.html
  6. Szkodziak P, Woźniak S, Czuczwar P, Paszkowski T, Milart P, Wozniakowska E, Szlichtyng W. Usefulness of three dimensional transvaginal ultrasonography and hysterosalpingography in diagnosing uterine anomalies. Ginekol Pol. 2014 May; 85(5):354-9.
  7. Fujimoto VY, Klein NA, Miller PB, Late-onset hematometra and hematosalpinx in a woman with a non-communicating uterine horn  A case report.  J Reprod Med 1998;43(5):465-7. 
Citation

Goel A, Walasangikar VB, Pawde A, Chauhan AR. Large Hematosalpinx Secondary To Unicornuate Noncommunicating Uterine Horn. JPGO 2015. Volume 2 No. 5. Available from: http://www.jpgo.org/2015/05/large-hematosalpinx-secondary-to.html



Retained Nude Copper-T

Author Information

Puri J *, Gupta AS**.
(* Second Year Resident, ** Professor, Department of Obstetrics and Gynecology, Seth GS Medical College & KEM Hospital, Mumbai, India)
                        
Abstract

Intra Uterine Contraceptive Device (IUCD) is most commonly used for long-term contraception. Long-standing in utero Copper T has been found to cause endometrial changes, abnormal uterine bleeding (AUB), complications like perforation, migration into peritoneal cavity and resultant pelvic infections. Our patient is a case of menorrhagia, which when examined, was found to have a nude IUCD, forgotten in utero for 22 years.

Introduction

Intra Uterine Contraceptive Device is the most common method of reversible contraception worldwide. An estimated 150 million women use IUCDs worldwide. It is the second most common method of contraception after female sterilization. Government of India advocated the use of Cu-T-200B for 4 years during the time period of 1975 to 2002. Cu-T-200B should either be replaced or removed not later than 4 years from the date of insertion. Cu-T-380A is being supplied in the Indian states and Union Territories since 2003-2004 under National Family Welfare Program. Approximately 13.6% of couples use IUCD as the selected method of contraception.[1]

Case Report

A 48 year old female, gravida 4, para 4, living 4, had come to the gynecology outpatient department (OPD) with a complaint of menorrhagia for the last 6 cycles. On obtaining history, patient gave the history of having an Intra Uterine Contraceptive Device (IUCD) inserted after her last childbirth about 22 years back. She had developed similar complaints of menorrhagia one year after the IUCD insertion. She had gone to a private practitioner who removed the IUCD. However, on redirect questioning it was found that the IUCD was not shown to the patient after its removal. The complaints of the patient had subsided. The patient also gave a history of not using any contraception after the Copper-T was removed. It was after 22 years that patient had developed symptoms of menorrhagia again, six months back, when she had gone to a private practitioner again, who started her on Tab. Norethisterone. She took the medication for about two months and then stopped taking it on her own. She relapsed with menorrhagia, for which she came to our Gynecology OPD. On examination, her abdomen was soft, without any guarding, tenderness or rigidity. On speculum examination bleeding through cervical os was present, black colored threads were seen coming through the cervical os. The cervix and vagina were healthy. On bimanual pelvic examination, a normal sized, anteverted, mobile, nontender uterus was palpated, there was no forniceal masses or tenderness. The vagina and the cervix were then swabbed with Povidone-Iodine and the threads were pulled with a sterile straight hemostat. There was no resistance, and painless extraction of an IUCD was done. There was a forgotten intrauterine IUCD in the patient for 22 years, which looked like as is shown in Figure 1.


Figure 1: Nude Copper-T 200B.

There was no copper left on the polyethylene base of the Copper-T. The patient was shown the retained IUCD. The patient was admitted in the ward for injectable Tranexemic acid therapy. Her Hb was 6.6 g/dl. She improved symptomatically after Inj. Tranexemic acid IV therapy and was discharged on Tab. Ferrous Sulphate 200 mg tds. She did not have any menorrhagia subsequently.

Discussion

In 1965, Lippes’ loop was introduced as the IUCD, under National Family Welfare Programme. Copper T 200B was introduced in 1975, which was replaced with Copper T 380A in 2002,[2] as Copper T 380A was found more efficacious and safer for longer duration of use as compared to Copper T 200B.[3] The fragmentation of copper was reduced because of addition of Silver in Copper T 380A without affecting its dissolution. Our case is a patient of forgotten IUCD for 22 years, which was Copper T 200B, which was used around 22 years back. Copper T 200B has 120 mg of copper, wound around the vertical limb of the IUD in the form of a 0.25 mm diameter copper wire. The exposed surface area of copper is 200 mm2. The copper from the IUD is released at the rate of 50 mcg per day. The copper in the IUCD should get dissolved in 6.57 years by that calculation.[4] USFDA approved lifespan of Copper-T 200B is 4 years.[5] The patient had not used any contraception even after the completion of days of use of the Copper T 200B. Luckily the patient did not conceive even after expiration of the usage time of Copper T 200B probably because of Copper absorption even after the expiry date, as the amount of copper left could not be estimated, or because of the copper T was acting as an inert IUD, and also contributory factor of declining fertility in an aging woman. IUCD can cause complications like menorrhagia, pelvic pain, pelvic infection, perforation of uterus and hence translocation of IUD. Longstanding IUCDs have been reported to wander in the peritoneal cavity into the Pouch Of Douglas, perforating rectum, appendix and urinary bladder, causing bladder stones.[6] Long-standing IUDs also cause endometrial changes. A study shows the endometrial changes like interstitial hemorrhage in 72% of IUCD users versus 65% in control; glandular hyperplasia in 6% and atrophy in 5% of IUCD users in the study was observed with no incidence of either glandular hyperplasia or atrophy in the controls.[7] Asynchronism between the cyclical phase and histologic phase of endometrium by 4 days in IUCD users was found to be 61%. This asynchronism is found to have antifertility action in IUCD users. Hyperplasia is mostly found in women with IUVDs inserted for more than 5 years.[5]

To ensure the acceptance of IUCDs as method of contraception by many more women, health care providers should take measures in satisfactorily counseling women about the procedure of insertion, to encourage them to ask questions, and answer them. Regular follow-up data sheet should be maintained. The patient should be called for regular follow-up at 1st then 3rd month for the IUCD thread check[1] and yearly thereafter, as the chances of expulsion are maximum during the first year.[8] The patient should be shown the Copper T that was there inside at the time of removal, when it’s removed, which in our case the patient was not shown. Either the older Copper T was not removed or a fresh Copper T was inserted for which a fresh consent was not obtained, either verbal or written. Each time the patient comes up with a fresh complaint or before starting her on a new treatment, she should be examined, unlike our patient who was started on T. Norethisterone without examination. Examination would have diagnosed the cause of her AUB. Such clinical practices should be condemned. Luckily, in this case, the patient had no serious complications like perforation of the uterus or any other vital organs.

References
  1. Chaturvedi GC, IUCD Reference Manual for Medical Officers. Family Planning division, Ministry of health and family Welfare. Government of India July 2007. 2nd ed. Government Of India, Nirman Bhavan, New Delhi, 110011. Available from- http://cfw.tg.nic.in/pdf/Guidelines_IUCD_MOs.pdf
  2. World Contraceptive Use- 2005, Director, Population Division, Department of Economic and Social affairs. United Nations, New York. NY 10017. Available from- http://www.un.org/esa/population/publications/contraceptive2005/WCU2005.htm
  3. Sharma RS, Rajalakshmi M, Jeyraj DA, Current Status of Fertility Control Methods in India. Journal of Biosciences. November 2001; Volume 26 Number 4: 391-405. Available from- http://www.ias.ac.in/jbiosci/nov2001/391.pdf
  4. Gupta AS, Parulekar SV. Bare Retained IUD’s. Bombay Hospital Journal. 1998; Volume 40 Number 2: 293-295
  5. Chaudhuri SK. Practice of fertility control- A comprehensive approach. 7th ed. New Delhi Elsevier Publications; 2008: 90-119
  6. Nigam A, Biswas R, Mishra A. Misplaced Intrauterine Contraceptive Device: an enigma. Open Access Journal of Contraception. 2011; 2: 1-3
  7. Lee YB, Kim CS, Ro JY, Kwak HM, Song CH. Long term effects of IUD on Human Endometrium- Histologic, histochemical and ultrastructural studies. Yonsei Medical Journal. 1983; Volume 24 Number 2: 141-148
  8. Kaneshiro B, Aeby T. Long-term safety, efficacy, and patient acceptability of the intra-uterine Copper T- 380A contraceptive device. International Journal of Women’s health. 2010; 2: 211-220.
Citation

Puri J, Gupta AS. Retained nude Copper T. JPGO 2015. Volume 2 No. 5. Available from: http://www.jpgo.org/2015/05/retained-nude-copper-t.html

Concurrent Laparoscopic Myomectomy and Repair of Hydrocele of Canal of Nuck

Author Information

Borse M*, Kotekar O**, Prasad R***.
(* Consultant Obstetrician and Gynecologist , Deenanath Mangeshkar Hospital, Pune
** Consultant Obstetrician and Gynaecologist, Dr. Saroj Tamboli Memorial Hospital, Alibaug; *** Clinical attache to Dr. Mahindra Borse; India.)

Abstract

Hydrocele of canal of Nuck in an adult female is a rare entity arising from a persistent fold of peritoneum. Here we describe a case of an hour-glass shaped hydrocele in a 32 year old multiparous lady with a 20-week size fibroid uterus. Following laparoscopic myomectomy the hydrocele was excised and the deep inguinal ring was repaired with purse-string polypropylene sutures.

Introduction

Hydrocele of canal of Nuck is a rare entity. It arises from a persistent fold of peritoneum (canal of Nuck), analogous to the processus vaginalis in the male that failed to fuse and involute in early extra – uterine life. Though the condition is most often seen in young girls with an incidence of 1%[1], it has been reported in older women. The hydrocele can be small and asymptomatic, or grow to form a large multiloculated cyst[2,3,4] or could be associated with an indirect inguinal hernia.  Here, we report a case of a large fibroid uterus with an associated hydrocele.

Case Report

A 32 year old woman, para 2 living 2, presented with complaints of menorrhagia since 6 months and a gradually increasing lump in the lower abdomen and left groin since one year. Her menstrual cycles had increased in duration from 3 to 10-12 days over the last 6 months, with heavy flow and passage of clots. She began to feel a firm lump in her lower abdomen one year ago, which had gradually increased in size and was presently as big as a fist. At about the same time, she felt an additional mass in her left groin, which also increased in size over the period of a year. On examination, her general condition was fair except for mild pallor. On local examination, a 4*4 cm flocculant, nontender, irreducible swelling was noted in the left inguinal region.


Figure 1. Inguinal component of the hydrocele (arrows).

There was no impulse generated over the mass when the patient was asked to cough. The contralateral inguinal region was normal. Per abdomen, a 20-week size firm, immobile mass was palpable arising from the pelvis. On per speculum examination, the cervix and vagina were found to be healthy. The vaginal examination confirmed the presence of a 20-week size uterus with normal adnexae. A provisional diagnosis of fibroid uterus with left sided hydrocele of canal of Nuck was made. On ultrasonography a 12x10x10 cm anterior wall fibroid was visualized. However, the inguinal area had not been imaged. After correction of anemia, she underwent a laparoscopic myomectomy with high ligation of the canal of Nuck on the left side and tubal ligation. Intraoperatively, the uterus was enlarged with an anterior wall fibroid of about 10x10x10 cm and a 2x3 cm cyst was seen stretching the left deep inguinal ring. This was the intra–abdominal portion of the hydrocele. The trans - illumination test was positive. The fallopian tubes and ovaries on both sides were normal.


Figure 2 – Intraabdominal portion of the hydrocele (black arrow), enucleated leiomyoma (yellow arrow)..

The peritoneum over the cyst was incised and drained to permit visualization of the canal. The cyst wall was dissected off the round ligament and surrounding connective tissue and sent for histopathological examination.


Figure 3 – Opening of the overlying peritoneum

The stretched deep inguinal ring was then repaired using a 1-0 polypropylene suture taken in a purse string fashion, incorporating the round ligament into the stich, and thus obliterating the opening. At this point, the bulge above the labia majora disappeared, even when the pneumoperitoneal pressure was raised to 25 mm of Hg.  A second stitch was taken to ensure adequate closure. The overlying peritoneum was closed. The right deep inguinal ring was found to be normal.


Figure 4: Purse-string suture obliterating the deep inguinal ring (arrow).

The histolopathological report of the cyst wall showed a mesothelial cell lining along with fibrocollagenous and fibrofatty tissue. She made an uneventful recovery and at her 3-month follow up visit the she was cured.

Discussion

The canal of Nuck is a fold of peritoneum that traverses the inguinal canal and ends at the labium majus which begins to fuse during the 7th month of fetal life and occludes completely to form a fibrous cord by the 1st year of extra-uterine life. If the fold remains patent, it can form a hydrocele (due to accumulation of fluid) or a hernia (due to abnormal protrusion of viscous). There is a 1% incidence of hydrocele of canal of Nuck in children, but its incidence in the adult female is unclear because of its rarity.[1] The mesothelial cells that line the canal secrete fluid that is reabsorbed through venous channels or the lymphatics.[2] Inflammation, trauma or blockage of lymphatic channels could lead to an imbalance in the rate of secretion and absorption, resulting in a hydrocele, though in most cases the occurrence is idiopathic.[2] In our case, the probable etiology could be blockage of lymphatic channels due to pressure from the large fibroid as the hydrocele first manifested when the fibroid began to grow. Hydrocele of canal of Nuck could be encysted (present along the track of descent), have a persistent communication with the peritoneal cavity or be shaped like an hourglass. In our case it was the third type with both abdominal and inguinal components constricting at the deep inguinal ring.

The differential diagnoses for inguinal masses in women of reproductive age include hernia, lymphadenopathy, haematoma.[3,5,6] lipoma, epidermal inclusion cyst, round ligament cyst, ganglionic cyst, abscess, varicosity of the round ligament[7] and rarely endometriosis.[8] The presence of an irreducible, flocculant, transilluminant mass without a cough impulse or tenderness or signs of inflammation on it points to the diagnosis of a hydrocele. It can be confirmed on ultrasound which often shows a well-defined hypoechoic or anechoic mass in the inguinal region with posterior enhancement.[6,9] The most common complication is an associated hernia. Rare ones include hydrocele/ hernia of the contralateral side and infection.[10]

A report by Counsellor and Black on the histology of the hydrocele cyst wall in 17 cases showed flattened mesothelial cells and fibrous tissue along with occasional smooth muscle cells and blood vessels. [11] Our report was congruent with these findings. The standard recommended treatment for this condition is excision of the hydrocele sac and high ligation of the canal of Nuck.[12] This can be done through a groin incision or laparoscopically[13] - the latter has the advantages of a shorter operating lime, less post–operative pain and minimal scarring. If there is an associated hernia, a Trans–abdominal pre–peritoneal (TAPP) or Total extra–peritoneal procedure (TEP) may be required.[14] However, for an isolated hydrocele, a TAPP or TEP is not justified due to complications of the mesh like adhesions, peritoneal migration of the mesh, infection, bowel obstruction and scarring.[15]

Conclusion

Women, who in addition to their benign gynaecological condition, have a hydrocele of canal of Nuck, can be adequately treated by laparoscopic excision, high ligation of canal, and repair of the deep inguinal ring in the same sitting.

References
  1. Huang CS, Luo CC, Chao HC, Chu SM, Yu YJ, Yen JB. The presentation of asymptomatic palpable movable mass in female inguinal hernia. Eur J Pediatr. 2003;162:493–5.
  2. Stickel W, Manner M. Female Hydrocele (Cyst of the Canal of Nuck). J Ultrasound Med. 2004;23:429–432.
  3. Block RE. Hydrocele of the canal of Nuck: a report of five cases. Obstet Gynecol. 1975;45:464–6.
  4. Dawam D, Kanu P. Giant hydrocele of the canal of Nuck. Br J Urol. 1998; 81(4): 636.
  5. Schneider CA, Festa S, Spillert CR, Bruce CJ, Lazaro EJ. Hydrocele of the canal of Nuck. NJ Med. 1994;91:37–8.
  6. Anderson CC, Broadie TA, Mackey JE, Kopecky KK. Hydrocele of the canal of Nuck: ultrasound appearance. Am Surg. 1995;61:959–1.
  7. Oh SN, Jung SE, Rha SE, Lim GY, Ku YM, Byun JY, et al. Sonography of various cystic masses of the female groin. J Ultrasound Med. 2007;26:1735–42.
  8. Bagul A, Jones S, Dundas S, Aly EH. Endometriosis in the canal of Nuck hydrocele: an unusual presentation. Int J Surg Case Rep. 2011; 2: 288–9.
  9. Manjunatha YC, Beeregowda YC, Bhaskaran A. Hydrocele of the canal of Nuck: Imaging findings. Acta Radiol Short Rep. 2012;1:12–5.
  10. Ameh EA, Garba ES. Infected hydrocoele of the canal of Nuck in a neonate. Urol Int. 2003;71:226–7.
  11. Counseller VS, Black BM. Hydrocele of the canal of Nuck: Report of seventeen cases. Ann Surg. 1941;113: 625–30.
  12. De Meulder F, Wojciechowski M, Hubens G, Ramet J. Female hydrocele of the canal of Nuck: a case report. Eur J Pediatr. 2006 Mar; 165(3):193-4.
  13. Qureshi NJ, Krishnaswamy L. Laparoscopic excision of cyst of canal of Nuck. J Minim Access Surg. 2014 Apr-Jun; 10(2): 87–9.
  14. Toshifumi M, Takao H, Teijiro H, Nobuhide K, Shoji H, Hiroyuki O. Laparoscopic diagnosis and treatment of a hydrocele of the canal of Nuck extending in the retroperitoneal space: A case report. Int J Surg Case Rep. 2014; 5(11): 861–4.
  15. Yen C, Wang C,  Chang P, Lee C , Soong Y. Concomitant closure of patent canal of Nuck during laparoscopic surgery: case report. Hum. Reprod. 2001;16(2): 357-9.
Citation

Borse M, Kotekar O, Prasad R. Laparoscopic Repair of Hydrocele of Canal of Nuck. JPGO 2015. Volume 2 No. 5. Available from: http://www.jpgo.org/2015/05/concurrent-laparoscopic-myomectomy-and.html

Vaginal Epidermal Cyst In Pregnancy

Author Information

Madhva Prasad S*, Mhaske N**, Kharat D***, Fonseca MN****.
(* Senior Resident, ** Second Year Resident, *** Assistant Professor, **** Additional Professor. Department of Obstetrics/Gynecology, LTMMC and LTMGH, Mumbai, India.)

Abstract

A 22 year old with 32 weeks gestation presented with sudden onset, gradually progressing cystic swelling from vagina. Successful surgical resection was done. Histopathological examination confirmed it as epidermal cyst.

Introduction:

Cystic lesions of vagina are uncommon and are usually incidental findings. A rare presentation in pregnancy is reported here.

Case Report

A 22 year old primigravida was on regular antenatal follow up. At 32 weeks of gestation, she complained of mass per vaginum. She reported discomfort with walking and increased vaginal pressure. Symptoms were present since 2 weeks and were gradual in progression. There was no history of fever, cough or pain in abdomen.
She was afebrile, and systemic examination was unremarkable. Obstetric examination revealed a live intrauterine gestation of around 32 weeks.
On local examination, a single, smooth, fluctuant 4x4 cm cystic swelling was seen arising from the right postero-lateral wall of vagina, distending the introitus. There was minimal tenderness, but no redness or discharge.  HIV/HBsAg/HCV were negative. Blood sugars were normal. Complete blood counts including white blood cell counts were within normal limits. Tablet cefixime 250 mg twice a day for one week. Persistence of the cyst and increase in symptoms, necessitated surgical removal. Cyst removal was done under intravenous sedation. Patient was taken in lithotomy position (figure 1) and bladder emptied. Base of the cyst was identified around 4 cm above the introitus, which was clamped and cut. The stump was ligated with delayed absorbable sutures of polygalactin no 1. Cyst fluid was aspirated and sent for microscopy and culture. Cyst wall sent for histopathological examination Patient was discharged on postoperative day 4.
Cyst fluid microscopy showed no malignant cells and culture was negative. Histopathological examination revealed epidermal cyst (figure 2).
On follow up after 2 weeks, the sutures were intact and no recurrence or induration was felt at the surgical site.  Patient went into spontaneous labor. In the second stage of labor, upon crowning, left mediolateral episiotomy was given, with no difficulty. She delivered a healthy male child of 2700 grams. There was no perineal tear. Day 1 post delivery examination photograph is shown. (figure 3) Patient was followed up 1 month post delivery. The episiotomy site and the surgical site had healed well.


Figure 1: Arrow showing vaginal cyst distending introitus (72 dpi resolution).


Figure 2: (96 dpi resolution) A: Stratified squamous epithelium; B: Fibro-muscular cyst wall; C: Focal inflammatory infiltrate.

Discussion

Benign cystic lesions of the vagina present a spectrum, from small asymptomatic lesions to large ones. These may be of embryological origin, ectopic tissue or urological abnormalities.[1]
While most are asymptomatic, many are incidentally discovered during gynecological examination.  Histologically, they can be Mullerian cysts (30%), Bartholin's duct cysts (27.5%), epidermal cysts (25%), Gartner's duct cysts(12.5%) and endometrioid cysts (2.5%) [2] While the occurrence of vaginal cystic lesions, in general, has been described as rare, [3, 4] a literature search reveals that the number of cases reported in pregnancy are few. While conservative management can be done, such a method of management entitles close follow up, to ensure that the cyst is managed appropriately by drainage during early labour.[5,6] When drainage is done during antenatal period, [7] the patient should be followed up regularly due to the possibility of recurrence. Persistent symptoms may necessitate excision during pregnancy, [8] as in the case reported here. Vaginal fibrosis and difficulty during episiotomy are potential problems, which were not encountered in this patient.  Surgical management may be deferred to the puerperal period when the size of the cyst is relatively constant and is only minimally symptomatic [9] It is important not to overlook these cysts since the rare possibilities of malignancy [10,11,12] of  and  rectovaginal fistulae exist. [13] This case is being reported for its rarity and to add to the knowledge about this entity.

References
  1. Eilber KS, Raz S. Benign cystic lesions of the vagina: a literature review. J Urol . 2003 Sep; 170(3):717–22
  2. Kondi-Pafiti A, Grapsa D, Papakonstantinou K, Kairi-Vassilatou E, Xasiakos D. Vaginal cysts: A common pathological entity revisited. Clin Exp Obstet Gynecol 2008 Jan; 35(1): 41–4 .
  3. Jayaprakash S, Lakshmidevi M, Kumar SG. A rare case of posterior vaginal wall cyst. BMJ Case Rep [Internet]. 2011; Published online 2011 Jul4.  doi:  10.1136/bcr.02.2011.3804.
  4. Rao A, Rao B, Kurian MJ, Pai RR. Two rare presentations of epidermal cyst. J Clin Diagn Res. 2014 Oct; 8(10): OD 1–3.
  5. Arumugam A, Kumar G, Si L, Vijayananthan A. Gartner duct cyst in pregnancy presenting as a prolapsing pelvic mass. Biomed Imaging Interv J [Internet]. 2007 Oct;3(4):e46. PMID: 3097688
  6. Lallar M, Nandal R, Sharma D, Shastri S. Large posterior vaginal cyst in pregnancy. BMJ Case Rep [Internet]. 2015 Jan ;2015; Published online 2015 Jan 20. doi: 10.1136/bcr-2014-208874. 
  7. Hasbargen U, Hillemanns P, Scheidler J, Kimmig R, Hepp H. [Paravaginal abscess in pregnancy]. Zentralbl Gynakol [Internet]. 2001 Oct [ cited 2015Mar 18] ;123 (10):595–8. [ Article in German] Available from: http://www.ncbi.nlm.nih.gov/pubmed/11753817 
  8. Pereira N, Guilfoil DS. Excision of an enlarging vaginal epidermal inclusion cyst during pregnancy: a case report. J Low Genit Tract Dis. 2012 Jul; 16(3):322–4.
  9. Goldberg Y, Lavie O, Mandel R, Auslender R. Imaging of an atypical large perineal cyst diagnosed during pregnancy. Ultrasound Obstet Gynecol . 2012 Dec; 40(6):721–3.
  10. Němejcová K, Dundr P, Povýšil C, Sláma J. Primary vaginal squamous cell carcinoma arising in a squamous inclusion cyst: Case report. Cesk Patol [Internet]. 2012 Jan [cited 2015 Mar 18];48(3):153–5. [ Article in Czech] Available from: http://www.ncbi.nlm.nih.gov/pubmed/23057430
  11. Zhao J, Xiang Y, Zhao D, Ren T, Feng F, Wan X. Isolated epithelioid trophoblastic tumor of the vagina: a case report and review of the literature. Onco Targets Ther. 2013 Jan ;6:1523–6.
  12. Dahiya K, Jain S, Duhan N, Nanda S, Kundu P. Aggressive angiomyxoma of vulva and vagina: a series of three cases and review of literature. Arch Gynecol Obstet. 2011 May; 283(5):1145–8.
  13. Nasser HA, Mendes VM, Zein F, Tanios BY, Berjaoui T. Complicated rectovaginal fistula secondary to Bartholin’s cyst infection. J Obstet Gynaecol Res. 2014 Apr ;40(4):1141–4.
Citation

Vaginal Epidermal Cyst In Pregnancy. Madhva Prasad S, Mhaske N, Kharat D, Fonseca MN. JPGO 2015. Volume 2 No. 5. Available from: http://www.jpgo.org/2015/05/vaginal-epidermal-cyst-in-pregnancy.html

Primary Tubal Gestational Choriocarcinoma Mimicking Tubal Ectopic Pregnancy

Author Information

Mahajan N, Shende D, Shirodkar S.
(* Assistant professor, ** Ex-Resident, *** Professor. Department of Obstetrics and Gynecology, B.Y.L. Nair Hospital and charitable trust, Mumbai Central, Mumbai, India.)

Abstract

Gestational Choriocarcinoma (GCC) can arise from normal term pregnancy, hydatidiform moles, non-molar miscarriages and ectopic pregnancies. In our case, a 31 year old woman gravida 2 para 1 with irregular vaginal bleeding and pain in lower abdomen was operated for ruptured ectopic pregnancy. At laparotomy 8x6x6 cm purple coloured hematosalphinx like mass was present. Histopathological examination confirmed choriocarcinoma for which she received four cycles of injection Methotrexate. Tubal GCCs are rare but should be considered, particularly if there is a firm mass of ectopic pregnancy.

Introduction

Tubal gestational choriocarcinoma is found in only 0.76% of gestational trophoblastic disease (GTD) which is extremely rare.[1] Very few cases have been reported in the literature about GCC of the fallopian tube. We present a case report of primary choriocarcinoma of the fallopian tube without metastases, treated successfully with surgery and chemotherapy.

Case Report

A 31 year old woman G2 P1L1 was admitted with irregular vaginal bleeding since 3 months and pain in lower abdomen since 7 days. On examination patient was stable and bimanual examination revealed 6 weeks size uterus with tender firm mass measuring 6x5 cm in right fornix. Urine pregnancy test was positive. A transvaginal-sonography showed a well-defined hypoechoic mass measuring 6.4x4.6 cm in right tubo-ovarian region with free fluid in Pouch of Douglas. At laparotomy 8x6x6 cm purple coloured hematosalphinx like mass (figure 1) was present on right side along with 500 ml hemoperitoneum. Right salphingectomy was done. Left side fallopian tube, bilateral ovaries and uterus were normal. Her β-hCG was 4, 37,238mIU/m. Histopathology report was suggestive of choriocarcinoma of fallopian tube. Histopathological examination (figure 2) showed tubal lining at one end and presence of plexiform network of cytotrophoblast and syncytiotrophoblast, with large areas of necrosis and haemorrhage. Extensive sampling of the tubal mass did not reveal any chorionic villi. There was invasion of smooth muscle of the fallopian tube but extension to the serosal surface was not seen. Her chest X-ray and CT scan revealed no signs of metastasis. She received four cycles of injection Methotrexate following which her β-hCG was 2.59mIU/ml. Her one year follow up was uneventful.


Figure 1: Gross photograph of fallopian tube GCC.


Figure 2: Histopathology of GCC.

Discussion

The clinical features of tubal GCC include bleeding, pain and an adnexal mass, which were present in our patient. Half of the cases present with a sudden onset of pain and bleeding. They are often mistaken for ectopic pregnancies until histological diagnosis is made. GCC usually metastasizes to the vagina and lungs, followed by brain, liver and kidneys. Epidemiology of fallopian tube GCC is not yet proved due to limited literature.[2] Tubal choriocarcinoma is mostly gestational, rarely can it be non-gestational. The gestational type arises from tubal pregnancy or metastasis from a uterine choriocarcinoma, whereas the non-gestational type which is extremely rare arises from an aberrant meiosis of maternal genotype. The clinical course of this patient was 3-4 months. This choriocarcinoma could have arisen from a complete hydatidiform mole in the fallopian tube that developed rapidly as there was no evidence of gestational components in the pathological specimen. In our case, the GCC was unlikely to be a metastasis from previous normal pregnancy as her last childbirth was 3 years back and it was unlikely that undetected choriocarcinoma had remained dormant for 3 years. In view of the likelihood that the tubal choriocarcinoma followed an ectopic molar gestation without any metastasis, her modified FIGO/WHO Prognostic score was stage I:4 (2 points for β-hCG and 2 points for size of mass).
Before the advent of modern chemotherapy, GCC carried an almost 90% mortality rate. Some of these deaths were secondary to hemorrhage but the majority were attributable to usually distant metastasis. Choriocarcinoma of the fallopian tube is highly chemosensitive and offers favorable prognosis. Chemotherapy using single or combination drugs is given in most patients following surgery with excellent response similar to our case.[3]
Genotyping and DNA polymorphism analysis can discriminate between gestational and non-gestational choriocarcinoma.[4] In our case genotyping p56kIP2 immunostaining and STR analysis was not done due to economic constraints. Most choriocarcinomas are now treated with combination chemotherapy without distinguishing the gestational from the non-gestational type.[5] Our patient responded very well to single agent chemotherapy as her follow up to 1 year was without any relapse.

Tubal GCCs are rare but should be considered, particularly if there is a firm mass of ectopic pregnancy. Also all ectopic pregnancies with mass of more than 5 cm diameter should have high level of suspicion of GCC, as baseline β-hCG level is crucial in the management of tubal GCC. Histopathological examination of specimen and follow up of patient with report is important part in proper diagnosis and treatment of any patient.

References
  1. Muto MG, Lage JM, Berkowitz RS, Goldstein DP, Bernstein MR. Gestationaltrophoblastic disease of the fallopian tube. J Reprod Med. 1991;36(1):57-60.
  2. Ubayasiri K, Hancock B, Duncan T. A case of primary choriocarcinoma of thefallopian tube. J Obstet Gynaecol. 2010;30(8):881-3.
  3. Gálvez CR, Fernández VC, de Los Reyes JM, Jaén MM, Teruel RG. Primary tubalchoriocarcinoma. Int J Gynecol Cancer. 2004;14(5):1040-4.
  4. Popiolek DA, Yee H, Mittal K, Chiriboga L, Prinz MK, Caragine TA, BudimlijaZM. Multiplex short tandem repeat DNA analysis confirms the accuracy of p57 (KIP2) immunostaining in the diagnosis of complete hydatidiform mole. Hum Pathol. 2006;37(11):1426-34.
  5. Namba A, Nakagawa S, Nakamura N, TakazawaY,Kugu K, Tsutsumi O, Taketani Ovarian choriocarcinoma arising from partial mole as evidenced bydeoxyribonucleic acid microsatellite analysis. Obstet Gynecol. 2003;102:991-4
  6. Nakayama M, Namba A, Yasuda M, Hara M, Ishihara O, Itakura A. Gestational choriocarcinoma of Fallopian tube diagnosed with a combination of p57KIPimmunostaining and short tandem repeat analysis: case report. J ObstetGynaecolRes. 2011;37(10):1493-6.
Citation

Mahajan N, Shende D, Shirodkar S. Primary Tubal Gestational Choriocarcinoma Mimicking Tubal Ectopic Pregnancy. JPGO 2015. Volume 2 No. 5. Available from: http://www.jpgo.org/2015/05/primary-tubal-gestational.html

A Giant Ovarian Cyst: A Rare Case Of Mucinous Cystadenocarcinoma Of The Ovary In A Teenage Girl

Author Information

Rathod S*,  Vijayalakshmi S**.
(* Assistant Professor, ** Professor & Head of Department of Obstetrics and Gynaecology, Adichunchanagiri Institute of Medical Sciences, B. G. Nagar, Nagamangala, Mandya.)

Abstract

Mucinous cystadenomas more often occur in the fourth and fifth decades, This report concerns an unsuspected giant mucinous ovarian cystadenocarcinoma  in a 19-year-old girl with a huge abdominal enlargement. She presented with  pain in abdomen and  abdominal mass of 34 weeks size. Ultrasound revealed  multiloculated  large ovarian cyst, Staging laparotomy and left side salphingoophorectomy was done. The histopathology examination revealed mucinous cystadenoma with well differentiated carcinoma of Grade Ia. Compared to serous type, mucinous cytadenocarinoma, in stage 1 is usually confined to ovary and survival rate is more, and if mucinous cystadenocarcinoma presents  in advanced  stages  the  prognosis  is  poor.
Introduction

Mucinous cystadenomas have origins from inclusions and invaginations of the ovarian celomic epithelium and persistence of Müllerian cells, or from Wolffian epithelium.[1] They often occur in the fourth and fifth decades, accounting for 25% of the ovarian tumors, 5% are bilateral and 15% are malignant .  These tumour can be benign, borderline, and malignant tumours. Also these tumour can be primary and secondary metastic tumour.  On ultrasound the mucinous ovarian tumour are thick wall, multiloculated, and  of variable size. Histologically, mucinous cystadenoma is lined by tall columnar non-ciliated epithelial cells with apical mucin and basal nuclei.   Here is a rare case report of  huge  mucinous  cystadenocarcinoma weighing about 4.5 kg  in a teenage  girl.

Case Report

A 19 year old girl presented with history of pain in abdomen to our outpatient clinic. There was no history of menstrual irregularities. There was no history of  loss of appetite or weight.  She was a known case of rheumatoid arthritis from the past 2 years and she was on irregular treatment.  Clinically, her abdomen was uniformly distended up to 34 weeks’ size. She was unaware of  mass in her abdomen.


Figure 1. Ultrasonography of abdomen and pelvis showing the mass.

Ultrasound  revealed multiloculated large ovarian cyst,  measuring of 35 X 20 cm with internal echoes and septum thickness of 4.5 mm. (figure 1). Color Doppler scan revealed regular vascular branching and flow. Computed Tomography scan revealed same findings as ultrasound and there was no evidence of paraaortic lymphadenopathy . The serum CA-125 (36.70 IU/mL) was mildly elevated, whereas alpha fetoprotein (0.74 ng/mL)  and  lactic dehydrogenase (176 IU/mL) were normal.
Staging laparotomy  and left sided salphingoophorectomy was done. Intraoperatively a left sided huge ovarian mass of variable consistency with intact capsule, with no surface growth was found.  There was no evidence of ascites or metastasis to peritoneum or paraaortic or pelvic  lymph nodes. Biopsy was taken from the surface of the right ovary. Macroscopically the ovarian mass measured 35 x 20 cm, weighed 4.5 kg. On cut section the mass was multiloculated with mucinous fluid of 4 liters with some solid components  (figure 2).


Figure 2. Gross appearance of the tumor.

The histopathology examination revealed mucinous cystadenoma with well differentiated carcinoma of Grade Ia (figure 3). Right sided ovarian sample was normal. Peritoneal wash cytology gave no evidence of  malignant cells. Postoperative period was uneventful. Patient showed no fresh symptoms or signs of recurrence  on  regular follow-up.


Figure 3. Histopathological appearance of the tumor

Discussion

In ovarian masses of any age group, the first thing is to confirm whether it is malignant or not, if found malignant then to find whether it is a primary or a secondary involvement. Lee and Young classified tumors as metastatic or primary tumor based on 12 parameters.[2] Metastatic tumors pathologically had a diameter less than 13 cm, were bilateral, and had surface involvement, hilar involvement and nodularity. These tumors were associated with infiltrative patterns of invasion, signet ring cells, small glands/tubules and single neoplastic cells. Primary mucinous carcinomas of the ovary were usually larger, unilateral, had an expansile growth pattern with complex papillae and necrotic luminal debris. The primary treatment for early stage mucinous neoplasm is surgical (total abdominal hysterectomy, bilateral salpingo-oophorectomy, and surgical staging as with serous tumors). In patients who have undergone a thorough staging laparotomy and in whom there is no evidence of spread beyond the ovary, the uterus and contralateral ovary can be retained in women who wish to preserve fertility. In 2004, Hess et al stated that advanced stage mucinous ovarian cancer had a worse outcome than women with non mucinous type.[3]

References
  1. Gorgone S, Minniti C, Ilaqua A, Barbuscia M: Giant mucinous cystadenoma in a young patient. A case report. G Chir 2008, 29:42–44.
  2. Lee KR, Young RH. The distinction between primary and metastatic mucinous carcinomas of the ovary: gross and histologic findings in 50 cases. Am J Surg Pathol. 2003; 27:281-292.
  3. Hess V, A'Hern R, Nasiri N, King DM, Blake PR, Barton DP, Shepherd JH, Ind T, Bridges J, Harrington K. Mucinous epithelial ovarian cancer: a separate entity requiring specific treatment. J Clin Oncol. 2004;22:1040-1044.
Citation

Rathod S,  Vijayalakshmi S. A Giant Ovarian Cyst: A Rare Case Of Mucinous Cystadenocarcinoma Of The Ovary In A Teenage Girl. JPGO 2015. Volume 2 No. 5. Available from: http://www.jpgo.org/2015/05/a-giant-ovarian-cyst-rare-case-of.html