Volume 4 Issue 5, May 2017

Editorial
Gupta AS

Rare Case Of Aggressive Angiomyxoma Of Vault
Chaudhari HK, Parulekar SV.

“Skipping” Fallopian Tube - An Interesting Finding!
Pednekar R, Parulekar SV.

Traumatic Posterior Dislocation Of Urethra
Pednekar R, Valvi D, Warke HS.

Headache In Pregnancy- Innocuous Symptom To Warning Sign
Swati HV, Samant PY, Bhosle S, Pai K.

Tubo-Ovarian Abscess In a Sexually Naive Girl- A Presentation Of Genital Koch’s

Deshpande PS, Thosar MA, Gupta AS.

Primary Ovarian Abscess - An Unusual Case
Kumbhar P, Daigavane M, Kolhe A, Samant PY.

A Rare Variety Of Squamous Papilloma Of The Uterine Cervix In A Teenage Girl
Satia MN, Mali K, Kolhe A, Mandaokar V.

Puberty Menorrhagia Due To Glanzmann’s Thrombasthenia
Vaidya A, Hatkar PA, Satia MN, More V. 

Ewing’s Sarcoma In Pregnancy- a Rare Case

Shah S, Swaminathan G, Warke HS, Mayadeo NM.

Editorial

Gupta AS

Tuberculosis is a public health issue in India which is a high incidence country. Extrapulmonary tuberculosis besides involving the reproductive organs can also involve the gastro intestinal tract, meninges, brain, skin, bones, joints and almost any organ in the body.

Genital Tuberculosis is rampant in India. Estimated prevalance is about 20% though this may still be the tip of the iceberg as it is usually asymptomatic; diagnosis is difficult and many of the diagnosed cases may not be reported. It silently damages the reproductive potential of the woman. Most of the times it is incidentally detected  during an infertility work up in a young woman. Gynecologists may be the first consultants to see these women. 

Unlike pulmonary tuberculosis all extrapulmonary tuberculosis including genital Kochs are paucibacillary. Very few mycobacterium tuberculosis complex can be isolated from these sites making the diagnosis and identifying the causative organism more difficult. Besides being paucibacillary obtaining specimens for diagnosis usually requires invasive or operative procedures.

Gynecologists when they encounter or suspect genital tuberculosis should be well informed about the latest diagnostic modalities that should be used to clinch the diagnosis. They should also be aware of all the samples that need to be collected, proper method of transport to the laboratories, designated laboratories performing the tests and storage of the samples in case of delay in reaching the laboratory.

Multiple diagnostic modalities can be used for diagnosis of genital Kochs. Microbiological tests would include smear and staining for acid fast bacilli, culture for mycobacterium tuberculosis; MGIT (Mycobacterium Growth Inference Tube). Molecular tests  detect the nucleic acid of the organism. This would include the  cartridge based nucleic acid amplification (CBNAAT), Gene Xpert, TB PCR, linear assays. These tests will also detect the drug resistance to first line drugs mainly rifampicin and isoniazid. Specimens that should  be collected by the gynecologist when they suspect genital Kochs includes fluids and aspirates from the endometrial cavity, peritoneal cavity or washings, secretions from the vagina and the cervix. Quantity of the fluids should be as large as possible (5-10 ml) as the infection is paucibacillary. These fluids are sent in the Gene Xpert tubes that identifies the DNA of the organism and gives the report within a couple of hours. These fluids are also sent for culture (MGIT).  Tissue biopsies that are sent for culture should be sent in sterile normal saline and not in formalin. During transport to the laboratory they should remain moist and temperature should be at 4 -15°Celsius. If specimens cannot be transported to the laboratory within one hour, it is recommended to store them at 4°C. Tissue biopsies, endometrium for CBNAAT have to be prepared in the laboratory to free the  organism from the cellular debris. Contaminated specimens require rigorous decontamination procedures to eliminate the unwanted normal flora to prevent their overgrowth and consumption of the entire media before the TB bacilli even start to grow. 

All concerted efforts should be made to clinch the diagnosis. However, if the specimen sent does not have the bacilli ( paucibacillary  nature) the tests will be false negative. Hence the 2016 revised national tuberculosis control program (RNTCP ) guidelines permits the treatment of tuberculosis on clinical suspicion even if bacteriological or histopathological diagnosis cannot be established.

The May issue is ready for our esteemed readers and we hope that it is as informative as our previous issues.

Rare Case Of Aggressive Angiomyxoma Of Vault

Author information

Chaudhari HK*,Parulekar SV**.

( * Associate Professor, ** Professor and Head, Department of Obstetrics and Gynecology, Seth GS Medical College and KEM Hospital, Mumbai, India.)

Abstract

Aggressive angiomyxoma  is a slow-growing a rare mesenchymal  soft tissue tumor which arises in the pelvis and perineal region. It is not a common  type of tumor in this location.  Surgical removal  is the main treatment of aggressive angiomyxoma. Here, we present a case of a vaginal aggressive angiomyxoma in a 70 year old woman in which the diagnosis was only made after histological examination. The appearance,, presentation, diagnosis and treatment of this rare tumor are outlined.

Introduction

Aggressive angiomyxoma is a unusual deep soft tissue tumor occurring in the pelviperineal regions of young female patients.[1] As the name suggests, it may have a locally aggressive course and may exert pressure on adjacent organs. This slowly growing painless neoplasm was first described by Steeper and Rosa in 1983.[2] It is slow growing but problematic due to frequent local recurrence (30-72%). However metastasis are very uncommon. Misdiagnosis is very frequent problem. Diagnosis is mostly made on histopathology following surgical removal . We report a case of aggressive angiomyxoma in 70 years female presenting with a lesion resembling granulation tissue on vault of vagina following a vaginal hysterectomy done 11.5 years ago.

Case Report

A 70 year old woman, married for 50 years, para 2 living 2, presented with a complaint of something coming out per vaginum for 5-6 months. The symptom was aggravated by coughing, sneezing and lifting heavy weights. She had undergone a vaginal hysterectomy 11.5 years ago for a similar complaint. Operative notes of hysterectomy  were  not available. There was no history of any major medical or surgical illness in the past. There was no history of any bowel related symptoms. Her general and systemic examination revealed no abnormality. There was trunkal obesity. Speculum examination showed central transverse type moderate cystocele, mild central transverse type rectocele , lax perineum, and a granuloma of approximately 0.5 x .2 x .2  cm at right angle of vault. The Granuloma was soft and did not bleed to touch. The uterus was absent. There were no pelvic masses. Her Pap showed atrophic inflammatory with atypical epithelial cells. Results of tests for fitness for anesthesia were normal.

Figure 1. Gross appearance of the tumor.

An anterior colporrhaphy and a posterior colpoperineorraphy were done after excision of the Granuloma at the vault of vagina. She made an uneventful recovery. Her speculum and vaginal examination done agter 5 days revealed a healed vault and no pelvic abnormality.

Histopathological examination of the granulation tissue from the vaginal vault  showed a polypoidal tumor composed of bland spindled  and stellate cells  embedded in loose myxoid stroma. Numerous vessels were seen within the tumor which were dilated and showed extravasation of red blood cells. The stroma also showed numerous mixed inflammatory infiltrate. There was no evidence of necrosis, atypia or increased mitosis. A diagnosis of aggressive angiomyxoma of the vagina was made. At three monthly follow up examinations, the patient was found to be well, without any local recurrence or distant metastases.

Figure 2. Polypoidal tumour. (10X)

Figure 3. Tumor shows numerous dilated blood vessels ,inflammatory cells in myxoid stroma. (45X)

Figure 4. Dilated vessels ,neutrophils ,lymphocytes , few eosinophils in myxoid stroma. (100X)

Figure 5. Bland spindle cells are seen in myxoid stroma. (100X)

Discussion

Aggressive angiomyxoma of the genital region is a unusual condition that has been reported to show a peak incidence in young women in the third and fourth decades of life.[3-11] The tumor particularly involves the pelvis, often in the vagina, vulva, perineum or buttocks.[4] The tumor typically grows slowly and insidiously. It usually takes 2 months to 17 years for the patients to go to the hospital with a mass after the initial discovery. It may become larger and occupy the whole pelvic region and invade paravaginal and pararectal spaces displacing pelvic structures.[5,15] Gross examination shows it as a soft, bulky mass whose external surface is smooth and usually without a capsule. Some have finger-like projections into neighboring tissues. It has a gelatinous, homogeneous consistency. Focal areas of hemorrhage and congestion are seen on cut surface. It generally displaces rather than invades adjacent viscera, and is rarely damaging. Although invasions are rare, it can invade the bladder, bowel and pelvic bone.[3,6,7,8,11] Histologically, the tumor consists of a hypocellular population of small, to some extent stellate and spindle-shaped neoplastic cells with thin cytoplasmic processes.[3,5,10,15] The cells are scattered in a loose myxoid matrix composed of delicate curling collagen fibrils and hyaluronic acid, which gives the tumor a pale-pink color by eosin staining.[6,7,10] At the same time there is also an accompanying particularly vascular component, ranging from minute  capillary-like vessels to larger and thick-walled vessels with a distinct smooth muscle cells but with no evidence of anastomosis.[3,7,9-11] It takes long time to grow, but local recurrences of tumor are often seen (30-72% of the cases), sometimes even years later. Preoperative identification  is difficult due to rarity of the condition and an absence of gross characteristic features. It should be considered in every mass in the genital, perianal and pelvic region. Awareness  of the condition is essential to avoid inappropriate surgical treatment and wrong prognostication. Surgery is the approved treatment and it consists of a wide excision, but the importance of total destruction does not seem to be of crucial significance. The appropriateness of radiation therapy, embolization and/or hormonal treatment  has not been clearly proven yet but may become future another possibility. The term "aggressive" was chosen to give special importance to the neoplastic quality of the blood vessels, its locally infiltrative quality and the high danger of local recurrence, not to indicate its malignant nature.[12,13,8,10] However, there are reports of metastasized disorder  in the literature.[14,15] Hence meticulous follow up needs to be done over many years after initial surgical treatment, and local recurrence as well as distant metastases should be looked for.

References

1. Carta G, Parisse V, Accurti V, Sollima L, Di Stefano L, D'Alfonso A, et al. Aggressive angiomyxoma of the vaginal wall at the initial stage: a case report. Eur J Gynaecol Oncol. 2012;33(6):669-71.
2. Dahiya M, Singhal C, Bansal S, Jindal U. Aggressive angiomyxoma of vagina mimicking genital prolapse: a case report and review of literature. Int J Reprod Contracept Obstet Gynecol. 2015 Aug;4(4):1231-1233.
3. 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;283(5):1145-8.
4. Foust-Wright C, Allen A, Shobeiri SA. Periurethral aggressive angiomyxoma: a case report. Int Urogynecol J. 2013;24(5): 877-80.
5. Wang Q, Zhao M, Lin X, Zhong W, Gao Y. Aggressive angiomyxoma of the vulva: intra-operative pathological diagnosis is useful in deciding the scope of surgery and reducing recurrence. Acta Chir Belg. 2012;112(1):79-84.
6. Haldar K, Martinek IE, Kehoe S. Aggressive angiomyxoma: a case series and literature review. Eur J Surg Oncol. 2010;36(4):335-9.
7. Dierickx I, Deraedt K, Poppe W, Verguts J. Aggressive angiomyxoma of the vulva: a case report and review of literature. Arch Gynecol Obstet. 2008;277(6):483-7.
8. Smith HO, Worrell RV, Smith AY, Dorin MH, Rosenberg RD, Bartow SA. Aggressive angiomyxoma of the female pelvis and perineum: review of the literature. Gynecol Oncol. 1991;42(1):79-85.
9. Fetsch JF, Laskin WB, Lefkowitz M, Kindblom LG, Meis-Kindblom JM. Aggressive angiomyxoma: a clinicopathologic study of 29 female patients. Cancer. 1996;78(1):79-90.
10. Behranwala KA, Thomas JM. Aggressive angiomyxoma: a distinct clinical entity. Eur J Surg Oncol. 2003;29(7):559-63.
11. Cinel L, Taner D, Nabaei SM, Dogan M. Aggressive angiomyxoma of the vagina. Report of a distinctive type gynecologic soft tissue neoplasm. Acta Obstet Gynecol Scand. 2000;79(3):232-3.
12. Steeper TA, Rosai J. Aggressive angiomyxoma of the female pelvis and perineum. Report of nine cases of a distinctive type of gynecologic soft-tissue neoplasm. Am J Surg Pathol. 1983;7(5):463-75.
13. Dierickx I, Deraedt K, Poppe W, Verguts J. Aggressive angiomyxoma of the vulva: a case report and review of literature. Arch Gynecol Obstet. 2008;277(6):483-7.
14. Siassi RM, Papadopoulos T, Matzel KE. Metastasizing aggressive angiomyxoma. N Engl J Med. 1992;341(23):1772.
15. Blandamura S, Cruz J, Faure Vergara L, Machado Puerto I, Ninfo V. Aggressive angiomyxoma: a second case of metastasis with patient's death. Hum Pathol. 2003;34(10):1072-4.

Citation

Chaudhari HK, Parulekar SV. Unruptured Pregnancy In Rudimentary Horn Of Unicornuate Uterus. JPGO 2017. Volume 4 No. 5. Available from: http://www.jpgo.org/2017/05/rare-case-of-aggressive-angiomyxoma-of.html

“Skipping” Fallopian Tube - An Interesting Finding!

Author Information

Pednekar R*, Parulekar SV(* Assistant Professor, **Professor and Head, Department of Obstetrics and Gynecology, Seth G. S. Medical College and KEM Hospital, Mumbai, India.)

Abstract

This is an interesting finding where we came across a fallopian tube going and adhering to the other side’s fallopian tube in such a way that the loop so formed is like a “skipping rope” moving anterior and posterior to the uterine fundus. This is the first case of this type in the world literature.

Introduction

Infertility due to fallopian tubal adhesions may be due to pelvic inflammatory disease, past pelvic surgery or endometriosis.[1] The fallopian tubes may be adherent to adjacent structures like the ovaries, broad ligament, uterus, bowel and/or omentum. Adhesion of one fallopian tube to the other forming a skipping-rope-like structure has not been described in the literature so far. We present such a case in which the two tubes were adherent to each other to form a half loop that could be swung to the back and front of the uterus freely.

Case Report

A 33 years old woman, married for 9 yeas, para 1, death 1 with no living issue, presented to us for reversal of interval tubal ligation done 6 years ago. Her menstrual cycles were regular, every 28- days, with bleeding for 3-4 days. She had a normal delivery 8 years ago, and an interval sterilization operation done by minilaparotomy 4 years ago. Baby died at 7 years of age , 1 yr back due to jaundice. Details of the operation were not available. There was no significant medical or surgical illness in the past. There was no history suggestive of pelvic inflammatory disease or tuberculosis. There were no bowel or bladder complaints. She had a normal vaginal delivery at home 8 yrs back. On examination her vital parameters were normal. General and systemic examination revealed no abnormality. Abdominal examination showed a 2 cm long suprapubic vertical midline scar. There was no tenderness, guarding or rigidity. Per speculum examination was normal. Per vaginally uterus was found to be of normal size, anteverted, smooth, firm, mobile, non-tender. Bilateral fornices were free. Reports of investigations for fitness for anesthesia were normal. Her husband’s semen analysis was normal. Her pelvic ultrasonography revealed no abnormality. Her hysterosalpingography showed  partial opacification of both the fallopian tubes with intravasation of contrast, suggestive of bilateral midsegment tubal block.

Figure 1. Hysterosalpingography. Site of tubal blocks (yellow arrows), intravasation of the dye (pink arrows).

A laparoscopy was performed, with a plan to perform a tubal reconstruction surgey if the fallopian tubes were found to be salvageable. The uterus and both the ovaries were found to be normal. The left fallopian tube showed a small hydrosalpinx, the lateral part of that tube being adherent  to bowel. The right fallopian tube was found adherent to the medial third of left fallopian tube by its fimbrial end and the loop so formed was freely mobile over the uterine fundus. Actually it was found to be in the uterovesical pouch of peritoneum when the laparoscope was inserted into the peritoneal cavity and the uterus was anterverted with a uterine manipulator inserted into the uterocervical canal. It could be moved freely anteriorly and posteriorly. There were minimal omental adhesions to the side wall. No other significant finding noted.

Figure 2. Laparoscopy findings: site of adhesion of the right fallopian tubal fimbriae to the left fallopian tube (arrow).

Figure 3. Laparoscopy findings: loop formed by adherent fallopian tubes (arrows).

With these findings, the decision of tubal recanalization was abandoned as left fallopian tube had mild hydrosalpinx with fimbrial adhesions to bowel and the fimbria of right  fallopian tube was damaged too due to adhesions to other tube thus leading to poor prognosis for pregnancy in the future, had the recanalization and repair been attempted. Only the fimbria of the right fallopian tube was separated from left tube with the help of bipolar electrocautery and scissors to release the loop. The patient made an uneventful recovery. She was counseled to undergo in vitro fertilization and embryo transfer.

Discussion

Fallopian tube is around 8 to 14 cm long tubular structure extending from uterine cornu, one on each side, into the broad ligament, which is then called mesosalpinx. The fallopian tube is divided into an interstitial portion, isthmus, ampulla, infundibulum and fimbrial extremity. Congenital fusion of fimbrial ends is not possible as the development of fallopian tubes is from paramesonephric ducts which fuse medially in the pelvis to form the uterus, whereas the lateral free portions develop into fallopian tubes. Therefore the fusion of fimbrial region to each other has to be acquired secondary to either previous surgery, pelvic inflammatory disorder or endometriosis.[1,2,3] Endometriosis has been described to cause adhesion of the ovaries to each other behind the uterine corpus.[4] There is no description of adhesion of the fallopian tubes to form a skipping-rope-like structure in the literature, due either to endometriosis or any other cause. In our case the patient had no signs and symptoms or examination and operative findings which would suggest either pelvic inflammatory disease or endometriosis. She only had prior interval tubal ligation by open method which is the only inciting factor leading to such type of adhesions and owing to the longer length of the tube which had given rise to “a skipping rope” type of loop with that much mobility; which, we thought, was interesting to note. A tubal reconstructive surgery was not done in view of extensive tubal damage, including formation of hydrosalpinx, loss of fimbriae.[5,6,7,8] We released the right fallopian tube from the left one surgically so as to prevent internal herniation and intestinal obstruction in future.

References

1. Serafini P, Batzofin J. Diagnosis of female infertility: A comprehensive approach. J Reprod Med. 1989;34:29–40.
2. Weström L, Joesoef R, Reynolds G, Hagdu A, Thompson SE. Pelvic inflammatory disease and fertility. A cohort study of 1,844 women with laparoscopically verified disease and 657 control women with normal laparoscopic results. Sex Transm Dis. 1992 Jul-Aug;19(4):185–192.
3. Patil M. Assessing tubal damage. J Hum Reprod Sci 2009;2:2–11.
4. Dunne C, Nakhuda G, Bedaiwy MA. Erosion of bilateral "kissing" ovaries into one large endometrioma. J Obstet Gynaecol Can. 2014 Oct;36(10):855-6.
5. Honoré GM, Holden AE, Schenken RS. Pathophysiology and management of proximal tubal blockage. Fertil Steril. 1999;71:785–795.
6. Greenhill J. Present status of plastic operations on the fallopian tubes. Am J Obstet Gynecol. 1956;71:516–559.
7. Dubuisson JB, Chapron C, Nos C, et al. Sterilization reversal: fertility results. Hum Reprod. 1995;10:1145–1151.
8. Yoon TK, Sung HR, Kang HG, et al. Laparoscopic tubal anastomosis: fertility outcome in 202 cases. Fertil Steril. 1999;72:1121–1126.

Citation

Pednekar R, Parulekar SV. “Skipping” Fallopian Tube - An Interesting Finding! JPGO 2017. Volume 4 No. 5. Available from: http://www.jpgo.org/2017/05/skipping-fallopian-tube-interesting.html

Traumatic Posterior Dislocation Of Urethra

Author Information

Pednekar R*, Valvi D*, Warke HS**.

(* Assistant Professor, ** Associate Professor, Department of Obstetrics and Gynecology, Seth G. S. Medical College and KEM Hospital, Mumbai, India.)

Abstract

We present a case of an unusual periurethral tear due to obstetric trauma which dislocated the urethra posteriorly, thus making it difficult to catheterize the patient. The patient was a second gravida and had come in labor to receiving room with head on perineum already. In spite of making an episiotomy to decrease the birth trauma and attempt at controlled delivery of the fetal head, a periurethral tear and a small perineal tear occurred, which subsequently had to be sutured in the operation theatre.

Introduction

Trauma to the maternal lower genital tract during childbirth should be preventable with due care. Unfortunately it continues to occur, because of deficiencies in administration of healthcare, and sometimes the patients presenting too late even when adequate healthcare facilities are available. Injuries to the perineum, vagina, and cervix are much more common than those to the area anterior to the vaginal opening, i.e. lateral to the urethra and around clitoris. We present an unusual case in which there was disruption of epithelium all around the urethra resulting in posterior dislocation of the distal urethra and external urethral meatus.

Case Report

A 26 years old, gravida 2 para 1 living 1, previous full term vaginal delivery, with term gestation, came in labor with head on perineum and delivered a male child of 3.13 kg in vertex presentation. The baby cried immediately after birth and handed over to a neonatologist. Placenta and membranes were delivered intact and completely. The mother had no other high risk factor, her past medical and surgical history was non significant. She had a full term normal vaginal delivery of a male baby weighing 3 kg. On examination, her vital parameters were normal. General and systemic examination revealed no abnormality. On local examination there was a tear of around 3 cm extending from external urethral meatus to clitoris anteriorly. It was bleeding. The external urinary meatus was dislocated posteriorly. The width of the raw area was 2 cm.

Figure 1. Injury to the area anterior to the external urethral meatus (white arrows). Vaginal opening (green arrow) Foley’s catheter (FC) are marked.

Figure 2. The part of the torn portion near the external urethral meatus is sutured.

Figure 3. The torn portion anterior to the external urethral meatus is sutured almost completely. The part near the clitoris is being sutured.

Figure 4. The end result of the repair.

Also there was around 0.5 cm of tear in the epithelium of the vestibule posterior to the external urinary meatus. Thus the external urethral meatus was in the middle of a raw surface without any overlying epithelium. Cervical tracing was normal. Anal sphincters were intact. Episiotomy edges were intact. There was a small first degree perineal tear in the midline. The patient was shifted to operation theatre for vaginal exploration. She was given spinal anaesthesia. A size 16 the urethral meatus was located and Foley’s catheter was passed through the urethra into the urinary bladder.  The tear was sutured with interrupted sutures of No. 3.0 polyglactin, first anterior and then posterior to the external urinary meatus. All dead space underneath was occluded while placing the sutures. Patient withstood procedure well and stable. Postoperatively the Foley’s catheter was kept in situ for a week. Patient recovered well.

Discussion

The lower two third part of the urethra lies immediately above the anterior vaginal wall opening into external urethral meatus which lies in the midline of the vestibule, 1 to 1.5 cm below the public arch just below the clitoris and just above the vaginal opening.[1] Lacerations of the vaginal and perineum are classified as first to fourth degree perineal tears. First degree involves the fourchette, perineal skin and vaginal mucosa . Periurethral tears are also included in this which bleed profusely. Second degree involves , in addition,  fascia and perineal body muscles. Third degree involves anal sphincters and A fourth degree extends through rectal mucosa.[1] Risk factors may include nulliparity, second stage arrest of labour, mid or low forceps, use of local anaesthetics and Asian race.[2] Timing of episiotomy is also important, if performed too late, lacerations will not be prevented. Typically, episiotomy is given with crowning of the presenting part, when 3-4 cm of head diameter is visible during a contraction.[1] Anterior tears involving the urethra and labia are more common in women in whom episiotomy is not given.[1] According to Carrolli and Mignini, the incidence of anterior perineal injury was lower in the routine-use episiotomy group.[3] Such tears also occur when modified Ritgen’s maneuver is not performed properly and premature extension of the fetal head is allowed to occur during childbirth.[1] Positioning of the legs during labour is also important. There should not be too wide separation of legs or one leg should not be higher than the other. This exerts pulling forces on the perineum and may increase the chances of perineal injury.[1] Laine et al suggest that slow delivery of the head while advising the parturient not to push may reduce the perineal laceration.[4]

In our patient the various above mentioned factors such as unnecessary pushing, too late episiotomy and sudden delivery of the head, came into play giving rise to anterior perineal injury. What is interesting in this case is that there was epithelial disruption all around the external urethral meatus, so that the meatus got dislocated posteriorly. Such an injury is very rare. Proper repair of such an injury is essential in order to restore normal position of the dislocated urethra and prevent misdirection of the urinary stream in future.

References

1. Mikolajczyk RT, Zhang J, Troendle J, Chan L. Risk Factors for Birth Canal Lacerations in Primiparous Women. Am J Perinatol. 2008 May; 25(5): 259–264.
2. Combs CA, Robertson PA, Laros RK Jr. Risk factors for third degree and fourth degree perineal lacerations in forceps and vacuum delivery. Am J Obstet Gynecol. 1990 Jul;163(1 Pt 1):100-4.
3. Carroli G, Mignini L: Episiotomy for vaginal birth. Cochrane Database Syst Rev 1:CD000081,  2009.
4. Laine K, Pirhonen T, Rolland R, Pirhonen J. Decreasing the incidence of anal sphincter tears during delivery. Obstet Gynecol 2008 May;111(5):1053-7.

Citation

Pednekar R, Valvi D, Warke HS. Traumatic Posterior Dislocation Of Urethra. JPGO 2017. Volume 4 No. 5. Available from: http://www.jpgo.org/2017/05/traumatic-posterior-dislocation-of.html

Headache In Pregnancy- Innocuous Symptom To Warning Sign

Author Information

Swati HV*, Samant PY**, Bhosle S***, Pai K, ****

(* First year resident, ** Professor, *** Second Year Resident, **** Assistant Professor, Department of Obstetrics and Gynecology, KEM Hospital, Mumbai, India)

Abstract

A 21 year old primigravida at 36 weeks and 2 days of gestation presented to emergency with severe intractable headache. She was evaluated to rule out all pathological causes of headache as she was not responding to basic conservative measures like relaxation therapy, paracetamol, hydration for benign headaches. Neurophysician finally diagnosed her to have developed new onset migraine after noting the imaging studies to be normal. Here we discuss the spectrum of causes of headache in pregnancy, which can just be a innocuous symptom to a warning sign of a serious underlying pathology. 

Introduction

Headache is amongst one of the most common discomforts in pregnancy. It can occur at any time during pregnancy; more commonly during first and third trimester. Commonest causes in first trimester include deprivation of sleep, dehydration, hypoglycemia, and stress. However most of the times headache will be secondary to an underlying pathology like pre eclampsia, TB meningitis, or reversible cerebral vasoconstriction syndrome (RCVS).

Case Report

A 21 year old primigravida with 36 weeks and 2 days of gestation was referred to our tertiary hospital with severe and intractable headache for evaluation. Headache was sudden in onset, in frontal and temporal region, throbbing in nature, non radiating, not associated with any pre monitory symptoms of preeclampsia, seizure episode, injury to head, loss of consciousness weakness of limbs, slurring of speech, bowel and bladder disturbances, fever, neck rigidity, and/ or photo phobia. She had 5 to 6 episodes of vomiting. On examination, her blood pressure was 120/80 mm Hg, urine albumin was absent, and knee jerks were normal. On abdominal examination uterus was 36 weeks in size and fetus was in the longitudinal lie with vertex presentation. Neurological examination showed no neurological deficits & cranial nerve examination was normal. She was initially admitted in medical intensive care unit for evaluation of headache. Fundoscopy was done and papilledema was ruled out. As patient gave history of vomiting, she was given prophylactic antibiotics as empirical treatment of meningitis and parenteral mannitol. Lumbar puncture and electrocardiogram were not done.

Ophthalmologists excluded refractive errors as a cause of headache. Urgent neurology reference was sought. They advised magnetic resonance imaging (MRI) of the brain without contrast and venography to rule out cortical venous thrombosis. It revealed no abnormality.

Symptomatic treatment was given for headache and vomiting in the form of paracetamol and ondensetron. Hydration was given with normal saline infusion. In spite of the treatment patient had no improvement in headache. Senior nuerophysician advised MR angiogram to rule out reversible cerebral vasoconstriction syndrome (RCVS) (Figure 1) Neurophysicians diagnosed her as a case of new onset migraine. As the headache was not relieved, decision to induce the patient was taken as the therapeutic agents that were advised were relatively unsafe in pregnant women like selective serotonin reuptake inhibitors, beta blockers, ergometrine derivatives. However she went into spontaneous onset of labor. She delivered a healthy male child weighing 2.8 kg. There was significant improvement in headache postpartum and did not require any medication for her headache. She was discharged on 4th postpartum day with prescription of NSAID if headache recurred and to follow up with neurology.

Figure 1. Normal MR Angiogram study

Figure 2. T2-weighted images with flair showing normal ventricles

Figure 3. MR venogram showing normal cortical and sagittal sinus.

Discussion

Headache is encountered commonly in pregnancy and can range from primary headaches, which are benign in nature to headache secondary to serious underlying pathology. Headaches in pregnancy are classified into benign and pathological, benign type including migraine, tension type headache, cluster headaches, analgesic-overuse headaches, and others. Pathological headaches can be due to a vascular event (hemorrhage or thrombosis) or raised intracranial pressure (ICP) such as in brain tumors and normal pressure hydrocephalus.[1] Headache marks the most common red flag sign of preeclampsia which requires active intervention. It is occipital, continuous, dull aching pain. Preeclampsia is linked to increased vascular reactivity, increased vasoconstrictors, endothelial damage and platelet hyperaggregation. Migraine is a common type of headache characterized by its unilaterality and pulsatility. The incidence of migraine is highest during the reproductive years in women.[2] Headaches in migraine are following the stimulation of trigeminal sensory neurons and the perivascular nerve endings, which is due to vasodilatation of extracranial and meningeal blood vessels.  [3] Migraine headache is influenced by estrogens. Increase in estrogen relieves the symptoms and decreased levels worsen the symptoms. As many as 10% of headaches initially present or are detected for the first time during pregnancy.[4]Migraine during pregnancy is difficult to diagnose as it needs to be differentiated from pre-eclampsia and other grave disorders like vascular disease, stroke, pseudotumor cerebri, hemorrhage, tumors, and infection. Reversible Cerebral Vasoconstriction Syndrome (RCVS) encompasses a diverse group of conditions, including hypertensive encephalopathy, and vasculopathies associated with pregnancy and postpartum (postpartum angiopathy). RCVS is characterized by sudden onset of a severe (thunderclap) headache. Interestingly, approximately 15% of patients with RCVS have a history of migraine headaches.[5]

Key examinations used to evaluate headache in pregnancy includes recording blood pressure, and urinalysis for proteinuria. Fundoscopy to see for signs of raised intracranial pressure should be done. Plantar responses, cranial nerves, gait assessment, tone, power, reflexes and coordination of all four limbs should be assessed. Neuroimaging might be considered in the evaluation of women with headaches that are of new onset or different from their usual headaches. In most patients with established migraine, however, neuroimaging is not usually necessary. Evidence-based guidelines issued by the American Academy of Neurology suggest that neuroimaging should be considered in the following patients with non acute headache: patients with an unexplained abnormal finding on neurological examination; patients with atypical headache features or headaches that do not fit in the strict definition of migraine or other primary headache disorders (or who have an additional risk factor, such as immune deficiency); and patients with sudden severe headache.[6] As single exposure to CT scan is within the limit of acceptable radiation (0.05Gy), a head CT scan is sufficient when neuroimaging is considered necessary. MRI is considered when a posterior fossa lesion or cerebrospinal fluid leak is suspected. MR angiography and MR venography are indicated when an arterial or venous lesion (for example, cerebral venous thrombosis) is considered in the differential diagnosis.

Non-pharmacological treatments, including biofeedback-assisted relaxation, hydration, bed rest, and a reduction in work or home responsibilities are usually enough to manage primary benign headache during pregnancy. Biofeedback methods combined with the occasional use of over-the-counter analgesics (for example, acetaminophen) are sufficient to control mild headaches in pregnancy and are safe to use during pregnancy. Additional pharmacological methods are required in approximately 25% of women for management of intractable headache during pregnancy.[7] β-blockers, calcium channel blockers, and selective serotonin reuptake inhibitors (SSRIs) are the additional therapeutic drugs available for treatment of migraine. Daily migraine prophylactic dose should be considered in women who have at least three or four severe migraine attacks per month throughout pregnancy. Use of the β-blocker propranolol increases risk of intrauterine growth restriction during pregnancy. Metoprolol and labetalol are alternatives to propranolol during pregnancy; however, with all of these agents, close ultrasound monitoring of fetal growth is essential, particularly throughout the third trimester.

Calcium channel blockers are effective for the management of migraines as well as to treat hypertensive disorders during pregnancy. Short-acting forms of these drugs can, however, lead to a sudden drop in maternal blood pressure and decreased uteroplacental perfusion, especially among hypertensive patients.

The SSRIs, especially paroxetine, have been associated with an increased risk of congenital heart defects, neonatal pulmonary hypertension and autism.[8] However, they have to be used with caution as most of the literature concerning the association between SSRIs and congenital anomalies is inconsistent.

Though our patient was diagnosed with migraine, the fact that she was better immediately postpartum without any therapy goes against the diagnosis.

It is important to note that pregnant woman with a simple symptom of headache can be at a high risk of myocardial infarction.[9]

Conclusions

Most headaches in pregnancy are benign and are due to primary headache disorders and can be treated with only reassurance and without any pharmacologic agents. A high index of suspicion for serious underlying pathology is necessary if atypical features are identified on history and/or clinical examination especially if a woman presents with a headache for the first time in her pregnancy so as to prevent significant morbidity and mortality.

References

1. Headache Classification Subcommittee of the International Headache Society (IHS) The International Classification of Headache Disorders, 3nd edition,(ICHD-IIRI). Cephalalgia 2013;33(9):629–808. http://www.ihs-headache.org/ichd-guidelines.
2. Revell K, Morrish P. Headaches in pregnancy. The Obstetrician & Gynaecologist 2014;16(3):179–84 
3. Cunningham FG. Neurological disorders. In: Cunningham FG, Leveno KJ, Bloom SL, Spong CY, Dashe JS, Hoffman BL, et al. editors. 24 th ed. Williams Obstetrics. New York: McGraw-Hill Education; 2014. p. 1187-89. 
4. Melhado EM, Maciel JA Jr, Guerreiro CA ;Headache during gestation: evaluation of 1101 women. Can J Neurol Sci 2007;34(2): 187-192. 
5. Calabrese LH, Dodick DW, Schwedt TJ, Singhal AB. Narrative review: reversible cerebral vasoconstriction syndromes.Ann Intern Med. 2007;146(1):34-44.
6. Frishberg BM, Rosenberg JH, Matchar DB, McCrory DC, Pietrzak MP, Rozen TD, et al.,Evidence-Based Guidelines in the Primary Care Setting: Neuroimaging in Patients with Nonacute Headache. The U.S. Headache Consortium.2005;3-18. http://tools.aan.com/professionals/practice/pdfs/gl0088.pdf.
7. Loder E. Migraine in pregnancy. Semin Neurol. 2007; 27(5), 425-433
8. Boukhris T., Sheehy O., Mottron L, Bérard A. Antidepressant Use During Pregnancy and the Risk of Autism Spectrum Disorder in Children. JAMA Pediatr. 2016;170(2):117-24.
9. Wabnitz A, Bushnell C.Migraine, cardiovascular disease, and stroke during pregnancy: systematic review of the literature. Cephalalgia. 2015;35(2):132-9.

Citation

Swati HV, Samant PY, Bhosle S, Pai K. Headache In Pregnancy- Innocuous Symptom To Warning Sign. JPGO 2017. Volume 4 No.5. Available from: http://www.jpgo.org/2017/05/headache-in-pregnancy-innocuous-symptom.html

Tubo-Ovarian Abscess In a Sexually Naive Girl- A Presentation Of Genital Koch’s

Author Information

Deshpande PS*, Thosar MA**, Gupta AS***.

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

Abstract 

A tubo-ovarian mass/ abscess is a mass involving the ovaries, fallopian tubes and sometimes the adjacent pelvic organs (bladder/ bowel). One of the most common causes of a tubo-ovarian mass in a reproductive age group is usually a sequelae of sexually transmitted diseases.[1] However here we present a case of a tubo-ovarian mass that on exploration turned out to be a case of tubercular tubo-ovarian abscess in an unmarried girl.

Introduction 

Tubo-ovarain masses can arise from congenital malformations, infections or tumors. In the reproductive age group, tubo-ovarian abscesses are usually a complication of pelvic inflammatory disease (PID). The incidence of PID in the age group of 15-39 years is 10-13% with a peak of 20% in the 20-24 years age group.[2] One third of the patients with PID may develop a tubo-ovarian abscess.[3] 

Case Report

A 20 year old unmarried girl with history of surgery for bladder outlet obstruction in neonatal period presented with complaints of acute onset lower abdominal pain. Pain was persistent, gradually increasing in intensity over one month but was not associated with menstruation. There were no menstrual complaints. There was occasional history of vaginal white discharge. There was no history of fever or urinary complaints. On examination midline scar of previous surgery was seen and vague minimal tenderness and an ill defined mass was felt in the lower abdomen. Ultrasonography (USG) revealed two right ovarian complex cysts measuring 7.6 x 5.2 cm and another of 4.3 x 3.1 cm. Both cysts showed dense internal echoes without septations or mural nodules. Vascularity was preserved. Bladder showed mild diffuse thickening of its walls with no vesical calculus or mass. Provisional diagnosis on USG was endometriotic cyst.

She was also recently diagnosed as being HbsAg positive and she was not on any treatment for the same. She had no history of jaundice or blood transfusions. There was no history of pulmonary tuberculosis in the past or history of Koch’s contact. X-Ray chest was done which was normal. All laboratory investigations and tumor markers were done which were within the normal range. 

She underwent exploratory laparotomy. Intraoperatively adhesions were noted in the pelvis extending from the uterus to the anterior abdominal wall and lateral pelvic walls. Uterus visualized after adhesiolysis was normal in size and shape. The right adnexa showed 4x5 cm thick walled ovarian cyst containing caseous material with fallopian tube was stretched across it. Cystectomy was done and specimen was sent for histopathology. Caseous fluid that was aspirated was sent for MGIT (mycobacterium growth inference tube), and aerobic culture and sensitivity, GeneXpert, AFB (acid fast bacilli) smear and staining, cytology and for TB-PCR. Cyst wall and a full thickness ovarian biopsy was sent for histopathology. No tubercles were seen in the pelvis. No endometriotic spots were seen. Left ovary was normal though left tube also had caseous collection. Bowel and upper abdomen was normal. Ovarian reconstruction was done and the abdomen was closed in layers. Postoperatively she recovered well and had no complaints. GeneXpert was negative for mycobacterium tuberculosis, on TB-PCR there was no mycobacterium genus detected, AFB staining was negative and culture sensitivity showed no growth. Histopathology of the cyst wall showed dense lymphocytic infiltration. Though all tests for tuberculosis were negative first line antikochs drugs (AKT) were started in view of intraoperative findings and the histopathology finding of dense lymphocytic infiltration.

Figure 1. Ovarian Abscess (OA). Babcock’s forceps are holding the round ligament and the ipsilateral fallopian tube.

Figure 2. Aspiration of the ovarian abscess. Syringe has the aspirated caseous material.

Figure 3. Peeling of the thick abscess wall (black arrow) from the ovarian bed (blue arrow).

Discussion

Tubo ovarian masses comprises a broad spectrum of masses right from pelvic endometriosis, hydrosalpinx, hemorrhagic cysts, hematomas, malignancies to complex diverticular abscesses or appendicular masses. The clinical signs and imaging may not be definitive to diagnose the etiology and many times the diagnosis is done with intra-operative findings and histopathology.

A tubo-ovarian abscess usually arises from local spread of ascending infection affecting the upper genital and adjacent pelvic organs or rarely hematogenous spread. The risk factors for a tubo-ovarian abscess are history of PID, multiple sexual partners and age between 15 and 40 years.[2] In the study conducted by Habboub, 20% of patients were adolescent or young adults, 18% were not sexually active and 11% patients had recent pelvic surgery/intervention.[4] Rarely  in unmarried girls they occur as a result of blood borne or genitourinary infection or may mimic ovarian tumors.[5,6] Other causes reported are manifestations of Crohn’s disease, recurrent urinary tract infection,[7] or infection by rare organisms.[1] Up to 95% of cases present with abdominal pain, while other complaints are palpable adnexal mass, cervical motion tenderness, fevers or vaginal discharge.[4] Our patient only had abdominal pain with no overt signs or symptoms of abdominal abscess. 

In India the prevalence of genital tuberculosis in female population is nearly 12-20 % and in 20 % of these cases ovaries are affected.[8] Thus many of the inflammatory conditions affecting the ovaries, fallopian tubes and endometrium can be attributed to tuberculosis.

Although definitive diagnosis of genital tuberculosis requires definitive histopathological confirmation but this can be proven in only 11% of cases. Hence RNTCP Guidelines 2016 allows use of other less sensitive tests or high clinical suspicion for provisional diagnosis and starting treatment of genital TB.[9] The tests suggested are AFB staining, culture, PCR and histopathology. All of these are 100% specific however their sensitivity is 24%, 48%, 87% and 88% respectively. AFB staining has also a high false negative rate of 75% followed by 51% for culture and 12 % for PCR.[10] Thus in our case though the histopathological diagnosis and other tests were not confirmatory, we started AKT due to high clinical suspicion.

Hence in a high prevalence setting like India, a tubo-ovarian mass occurring in a sexually naive girl should generate a high degree of suspicion for genital tuberculosis.

Conclusion

One of the most common diseases affecting the genital tract in a high prevalence region like India is genitourinary tuberculosis. Sensitivity of diagnostic tests available limit the diagnosis of this condition. However id there is high clinical suspicion, signs and symptoms and clinical findings suggestive of tuberculosis, treatment should not be delayed for the same.

References

1. Gensheimer WG, Reddy SY, Mulconry M, Greves C. Abiotrophia/Granulicatella tubo-ovarian abscess in an adolescent virginal female. J Pediatr Adolesc Gynecol. 2010;23(1):e9-12.
2. Westrom L. Incidence, prevalence and trends of acute pelvic inflammatory disease and its consequences in industrialized countries. Am J Obstet Gynecol. 1980;138(7 Pt 2):880-92.
3. Shephard SM. Pelvic Inflammatory Disease. 2017. Available at emedicice.medscape.com/article/256448-overview#a3.
4. Habboub AY. Middlemore Hospital experience with tubo-ovarian abscesses:an observational retrospective study. Int J Womens Health. 2016; 8: 325–340.
5. Ashrafganjooei T,Harirchi I,Iravanlo G.Tubo-ovarian abscess in a virgin girl. Iran J Reprod Med. 2011 Summer; 9(3): 247–250.
6. Dogan E, Altunyurt S, Altindag T, Onvural A. Tubo-ovarian abscess mimicking ovarian tumor in a sexually inactive girl. JJ Pediatr Adolesc Gynecol 2004 Oct;17(5):351-2
7. Hartmann KA, Lerand SJ,Jay MS.Tubo-ovarian abscess in virginal adolescents:exposure of the underlying etiology. J Pediatr Adolesc Gynecol. 2009;22(3):e13-6.
8. Abulut S,Arikanoglu Z,Basbug M.Tubercular tubo-ovarian cystic mass mimicking acute appendicitis: a case report. J Med Case Reports. 2011; 5: 363.
9. Technical and Operational Guidelines for TB Contol in India.  Chapter 3. Case Finding and diagnostic strategy. New Delhi, India. Central TB Division. Directorate General of Health Services. Ministry of Health and Family Welfare. 2016.
10. Yadav SK, Jha NK, Sinha DK, Singla H, Kumar S, Yadav J, et al. Evaluation of the role of ascitic fluid polymerase chain reaction targeting IS6110 of Mycobacterium tuberculosis in the diagnosis of tuberculous intestinal obstruction. Annals of Tropical Medicine and Public Health. 2015;8(5):198-201.

Citation

Deshpande PS, Thosar MA, Gupta AS. Tubo-Ovarian Abscess In a Sexually Naive Girl- A Presentation Of Genital Koch’s. JPGO 2017. Volume 4 No.5. Available from: http://www.jpgo.org/2017/05/tubo-ovarian-abscess-in-sexually-naive.html

Primary Ovarian Abscess - An Unusual Case

Author Information

Kumbhar P*,  Daigavane M**, Kolhe A***, Samant PY****.

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

Abstract 

Most of the primary ovarian abscesses are unexpected findings at laparotomy for tuboovarian mass or endometrioma. Clinical signs and symptoms of ovarian abscess are nonspecific and tubal involvement cannot be ruled out. Our case was detected on pelvic ultrasonography during investigation for recurrent fever when all other causes were ruled out. Neither microscopy nor culture showed microorganisms. TB PCR was negative.

Introduction 

Primary sterile ovarian abscess is a rare phenomenon. Clinical presentation ranges from asymptomatic abscess to diffuse peritonitis. The longer the delay in operative treatment, greater is the morbidity and mortality. Modern imaging modalities including ultrasound (USG) and magnetic resonance imaging (MRI) may help in making an early diagnosis. We describe an interesting case of a 36 year old parous woman with low grade fever and occasional pain in abdomen with ovarian abscess.

Case Report

A 36 year old woman came to medicine OPD with complains of fever since 15 days, with occasional pain in abdomen. There were no complaints of menstrual irregularities, dysmenorrhea and vaginal discharge. There were no bowel and bladder complaints. She had no history suggestive of past medical or surgical illnesses. She had one cesarean section. She did not report any contraception use. Her vital parameters were normal except for low grade fever. There were no specific abdominal signs.  Empirical treatment with antimalarials and broad spectrum antibiotics was given.  Complete blood count was suggestive of neutrophilic leucocytosis. Investigations including malarial antigen, leptospirosis and dengue antigen antibody tests, Widal and urine culture were negative. Abdominopelvic sonography done for abdominal pain was suggestive of left hemorrhagic ovarian cyst with pyosalpinx. She was referred to gynecology department. On abdominal examination there was no guarding tenderness and rigidity, a soft cystic mass could be palpated. On vaginal examination mass of about 8 cms in diameter was palpated posterolateral to the uterus on the right side. The uterus was normal in size. Computerized tomography scan (CT scan) showed cystic lesion in the pouch of Douglas of size 9x8 cm arising from the right ovary and causing mild ipsilateral hydro ureter, and hydronephrosis due to mass effect. The ipsilateral fallopian tube and contralateral tube and ovary were normal. There was no lymphadenopathy or ascites. In her tumor markers alfa feto protein was 1.03 ng/ml, carcinoembryogenic antigen was 0.83 ng/ml, LDH was 146 U/L and CA125 was 87.0 U/ml. She underwent exploratory laparotomy. Intraoperative findings showed a cystic thick walled ovarian mass of 10x12 cm on the right side adherent to the rectosigmoid and posterior wall of the uterus, obliterating the pouch of Douglas. Right fallopian tube was normal in caliber. Left fallopian tube and ovary were normal. The ovarian mass was separated from rectosigmoid and posterior wall of uterus by blunt and sharp dissection and oophorectomy was done.  Intraabdominal drain in pelvis was kept due to oozing. Postoperatively patient received one unit of blood. Postoperative  course was uneventful. Drain was removed on day 3 after surgery. Aspirated pus was sent for culture and TB PCR which came out to be negative. The culture was negative for microorganisms after 48 hours. Microscopy showed pus cells with no organisms or granulomas.  She was discharged on the 6th postoperative day. Sutures were removed on day 14 and the wound was healthy.

Figure 1. Ovarian abscess mass adherent to surrounding structures on exploratory laparotomy

Figure 2.  Ovarian abscess mass after removal. 

Figure 3.  Histopathology of ovarian abscess. Black arrow shows the cyst wall. Orange arrow the blood vessels and Blue arrow shows the inflammatory neutrophilic infiltrate.

Discussion

Ovarian abscess was first described by Aitken in 1869. Since then various cases of such abscesses have been documented. 

It is classified as

• Primary abscess occurs due to disruption of ovarian capsule, which is seen in ovulation or surgical intervention, which gives bacteria access to ovarian stroma. It can also occur due to hematogenus and lymphatic spread.

• Secondary abscess occurs due to spread of infection from surrounding structures. It may be associated with tuboovarian abscess, salpingitis, pelvic inflammatory disease or Gastro intestinal (GI) infections (diverticulitis and appendicitis).[1]

Ovarian abscesses are rare and for this reason their etiology remains poorly understood. 

Wetchler and Dunn [2] considered three possibilities to explain infection of the ovary.

1. The entry of bacteria into the parenchyma when the ovarian capsule is punctured.

2. Contamination via the blood stream.

3. Contamination via the lymphatic system. 

The most common mechanism is considered to be breach or alteration of the ovarian capsule at the time of ovulation, or by penetration during surgery or surgical procedures. The type of bacterium, its virulence, number of bacteria inoculated, spread through devitalized tissue and whether the infection occurred secondary to direct contamination at surgery, determines the clinical presentation, its severity and time of presentation (early or late). This complication has been reported to occur after vaginal hysterectomy, ovarian cystectomy, and cesarean section. Furthermore, transvaginal or percutaneous needle aspiration of an endometrioma have been considered in the causation of this rare phenomenon.[3] Ultrasonically guided vaginal oocyte collection is also a cause for ovarian abscess.[4] In our case the etiology of primary ovarian abscess remains unknown, it is possible that lymphatic spread occurred from some infective focus that was responsible for fever with which the patient presented. It is plausible that broad spectrum antibiotics sterilized the pus.

Primary ovarian abscess is mainly caused by organisms like salmonella, tuberculosis and actinomycoses Israeli which is associated with intrauterine contraceptive device users.[5,6] In our case aspirated pus was sent for culture and TB PCR which came out to be negative. TB PCR is considered to have high sensitivity.[7] There was neutrophilic infiltrate as shown in the photograph and the patient had neutrophilic leucocytosis, hence tuberculosis was ruled out. Hence tests like Mycobacteria growth indicator tube (MGIT), smear for AFB, Gene Xpert investigations were not done. In our case patient received broad spectrum antibiotics for seven days which probably sterilized the pus. 

Diagnosis of unruptured primary ovarian abscess is difficult because of variable clinical presentation and may remain silent before it causes pelvic pain, fever, pelvic tenderness or mass. Ultrasound at times can miss an ovarian abscess. MRI and CT scan may be helpful for confirmation of diagnosis

Treatment is surgical with unilateral salpingooophorectomy or oophorectomy may be performed under broad spectrum antibiotic coverage followed by specific antibiotics according to culture report.[8]  In this case, oophorectomy was done.  If the ovarian abscess ruptures, the clinical picture is much the same as that of a rupture of tubo-ovarian abscess, with abdominal distension, tachycardia, rebound tenderness, ileus and sometimes septic shock which may increase morbidity and mortality.

Conclusion

The prognosis after surgical management of a large unruptured primary ovarian abscess is good. In absence of tubal involvement, oophorectomy should be done.

References

1. Koehn RC. Ovarian abscess complicating pregnancy. US Armed Forces Med J. 1957; 8 (11):1664-9.
2. Wetchler SJ and Dunn LJ: Ovarian abscess. Report of a case and review of the literature. Obstet Gynecol Surv 40:476- 485,1985.
3. Romero B, Aibar L, Navarro LM, Fontes J, Calderón M-A, Mozas J. Pelvic abscess after oocyte retrieval in women with endometriosis: A case series. Iran J Reprod Med. 2013; 11(8): 677–680.
4. Aragona C, Mohamed MA, Espinola MS, Linari A, Pecorini F, Micara G. Clinical complications after transvaginal oocyte retrieval in 7,098 IVF cycles. Fertil Steril. 2011;95(1):293-4.
5. Goh WC, Beh ST, Chern B, Yap LK. A Three Year Review on Surgical Treatment of Tubo- Ovarian Abscess. Med J Malaysia. 2002; 57(3): 292-297.
6. Tohya T, Yoshimura T, Onoda C. Unilateral ovarian abscess caused by Salmonella. Infect Dis Obstet Gynecol 2003;11:217–219
7. Tan MF, Ng WC, Chan  SH, Tan WC. Comparative usefulness of PCR in the detection of mycobacterium tuberculosis in different clinical specimens. J. Med. Microbio- Vol 1997;46:164-169.
8. Gumanga SK, Kolbila DZ. Chronic Unruptured Primary Ovarian Abscess: A Case Report. Open Science Journal of Clinical Medicine 2015; 3(3): 83-85.

Citation

Kumbhar P, Daigavane M, Kolhe A, Samant PY. Primary Ovarian Abscess - An Unusual Case. JPGO 2017. Volume 4 No.5. Available from: http://www.jpgo.org/2017/05/primary-ovarian-abscess-unusual-case.html

A Rare Variety Of Squamous Papilloma Of The Uterine Cervix In A Teenage Girl

Author Information

Satia MN*, Mali K**, Kolhe A***, Mandaokar V****

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

Abstract 

Squamous papilloma of uterine cervix is a rare and benign tumor generally observed in teenagers and young women. The prognosis is typically excellent with treatment, since these are benign tumors. We report one such case in a 19 year old girl with a verrucous  mass at the vulva with a foul smelling discharge. It was excised and histopathology was suggestive of  papillary exuberant immature metaplasia (PIM). The human papilloma virus (HPV) 18 genotype testing was positive. 

Introduction 

A squamous cell benign papilloma most commonly arises from the stratified squamous epithelium of either the skin, lips, cervix, vagina or anal canal.[1]  Squamous papillomas are slow-growing and benign tumors quite uncommon in the uterine cervix. The common locations are vulva and vagina. Human papilloma virus (HPV) infection is a major cause of these lesions though most HPV infections do not cause cancer. However there are a number of other conditions that cause papillomas; however, in many cases the cause is unknown. Estimated rate of recurrence is 60 to 66% and malignant transformation is seen in 30 to 40% of cases.[2] A thorough physical examination along with Pap smear & colposcopic directed biopsy is mandatory for diagnostic evaluation because this benign tumor may be confused for a low-grade squamous intraepithelial lesion (LSIL) which is   a pre malignant condition or condyloma acuminatum of the cervix or it may resemble cervical cancer. Differential diagnosis should include condyloma acuminatum, and focal epithelial hyperplasia and differentiation is done accurately by microscopic examination only. Antigens of the HPV types 6 and 11 are identified in approximately 50% of cases of squamous cell papillomas by the immunoperoxidase stains. There is no evidence that papillomas are pre malignant. Patient can be completely cured when a meticulous surgical excision is done to remove the tumor completely.  Follow-up care with regular screening is important and should be encouraged. The prognosis of squamous papilloma of uterine cervix is generally excellent with appropriate treatment since it is a benign tumor. 

Case Report

A 19 year old girl presented to the gynecology out patient department (OPD) of a tertiary care center with complaints of purulent vaginal discharge since last 6 months with itching around the vulva since then. Initially on inquiry she did not give history of sexual exposure, however later she agreed for the same and gave a history of sexual exposure for the past 2 years. On clinical examination her vital parameters were stable & on local examination she had verrucous lesions at the vulva with purulent and foul smelling discharge around them as shown in figure 1.

Figure 1. Verrucous growth seen at the introitus.

  

As she denied any history of sexual intercourse a speculum and bimanual vaginal  examination was not done. A clinical impression of condylomata accuminata was made & she was referred to skin OPD where she was examined and a biopsy was taken. Histopathology report was suggestive of syringocystadenoma papilliformis which is an apocrine sweat gland tumor and she was referred back to us. After preoperative evaluation, VDRL and HIV testing which were negative an examination under anesthesia (EUA) was done. On EUA the mass appeared as a polypoidal growth from the posterior lip of the cervix. It was clamped cut and ligated with polyglactin suture (figure 2). Electrocautery of the bleeding points on the posterior lip of the cervix was done. 

Figure 2. Excision of the verrucous lesion of the cervix.

Post operative period was uneventful.  She remained asymptomatic. Histopathology report was endocervical polyp with exuberant squamous metaplasia with papillary hyperplasia. (figure 3)

Figure 3. Histopathology picture shows high power and low power microscopy showing squamous metaplasia.

She was advised HPV testing to find out the strain. Genetic testing of the tissue which was sent for biopsy was done which came positive for high risk HPV 18. She has been explained & reassured about the prognosis and the need for regular annual follow up and Pap smear.  

Discussion

Human Papilloma viruses belong to the family of Papova viruses. These viruses promote cell proliferation and results in the development of benign papillomatous lesions of the genital tract, upper respiratory tract, cutaneous lesions of the skin and at other sites. There are more than 70 different types of HPV viruses and they are identified by molecular hybridization of DNA extracted from warty lesions from various sites. Each virus type has a definite site of infection. HPV infections can be spread inadvertantly by people who remain symptom free despite having the HPV infection. Mode of transmission remains sexual contact. These infected persons do not develop any lesions or warts nor does it progress to cancer. They may remain symptom free always. The causative viruses for these lesions are HPV 6 and 11 and HPV types 16,18, 31 and 33 are often associated with neoplastic transformation.[3] HPV DNA genotyping is required to determine whether the patient is infected with HPV. Whether the body's immune system can permanently clear the HPV infection is not clear. In  some cases patients may test positive for HPV infection and then the test may come negative for months to years, only to have a positive test result many years later. It is difficult to identify whether the infection is due to latent viral infection or is due to reinfection of the virus. A papilloma is a benign epithelial tumor which grows outwards as finger-like projecions. It shares some histologic and cytologic features with squamous metaplasia (SM), atypical immature squamous metaplasia (AIM), high-grade squamous intraepithelial neoplasia (HSIL) and papillary squamous cell carcinoma (PSC). PIM has been suggested to be a subset of condyloma associated with low-risk type human papilloma virus but HPV genotyping of the tissue sample was reported as HPV 18 though the etiologic role and biologic behavior of the disease are still elusive. The diagnosis can be difficult with cytological characteristic. The lesions appear histologically as an exophytic mass with epidermal hyperplasia, hyperkeratosis but without basement membrane disruption.[4] 

Conclusion

In a number of cases, body's immune system defeats a HPV infection before it creates warts. The warts may look like flat lesion or cauliflower like lesions. Repeated infection with certain HPV strains can cause precancerous lesions over time. If not treated, these lesions can become cancerous. Hence, it is important for women to have regular Pap tests, which can detect precancerous changes in the cervix that might lead to cancer. Hence current guidelines recommend that women between 21 to 29 years of age should have a Pap test every three years. Women between 30 to 65 years of age are advised to continue having a Pap test every three years, or every five years.

References

1. New Zealand Dermatological Society. Squamous Cell Papilloma. New Zealand Dermatological Society. 2007.
2. Chu QD, Vezeridis MP, Libbey NP, Wanebo HJ. Giant condyloma acuminatum (Buschke-Lowenstein tumor) of the anorectal and perianal regions. Analysis of 42 cases. Dis Colon Rectum. 1994;37(9):950-7.
3. Boshart M, zur Hausen H. Human papillomaviruses in Buschke-Löwenstein tumors: physical state of the DNA and identification of a tandem duplication in the noncoding region of a human papillomavirus 6 subtype. J Virol. 1986;58(3):963-6.
4. Ciobanu AM, Popa C, Marcu M, Ciobanu CF. Psychotic depression due to giant condyloma Buschke–Löwenstein tumors.  Rom J Morphol Embryol 2014, 55(1):189–195.

Citation

Satia MN, Mali K, Kolhe A, Mandaokar V. A Rare Variety Of Squamous Papilloma Of The Uterine Cervix In A Teenage Girl. JPGO 2017. Volume 4 No.5. Available from: http://www.jpgo.org/2017/05/a-rare-variety-of-squamous-papilloma-of.html

Puberty Menorrhagia Due To Glanzmann’s Thrombasthenia

Author Information

Vaidya A*, Hatkar PA**, Satia MN***, More V****.

(* Second year Resident, ** Associate Professor, **** Professor, *** Assistant Professor, Department of Obstetrics and Gynecology, Seth GS Medical College and KEM Hospital, Mumbai, India.)

  

Abstract 

Glanzmann's thrombasthenia is a rare autosomal recessive bleeding diathesis.[1, 2] The platelet glycoprotein (GP) IIb/ IIIa complex is either absent or dysfunctional which causes a defect in platelet aggregation and hence the coagulation cascade. We are reporting an interesting case of conservative management done in a patient with puberty menorrhagia suffering with a variant of Glanzmann’s thrombasthenia. 

Introduction

Glanzmann's thrombasthenia has been named after Dr. Eduard Glanzmann, a Swiss pediatrician. It is a platelet function disorder with defective clot retraction due to a dysfunctional aggregation of glycoprotein IIb/ IIIa. The incidence is 1 in 1 million.[3] It causes prolonged bleeding time and poor platelet aggregation that results in bleeding manifestations. Patients commonly present with episodes of bleeding from external orifices (epistaxis, menorrhagia, less commonly hematemesis, malena and hematuria) as well as generalized rash over the body (purpura, petechial hemorrhages, or gingival bleeding). Menorrhagia is seen in most female patients from the time of menarche and is a serious concern as they have heavy bleeding requiring transfusion of blood and blood products.

Case Report

A 13 year old unmarried girl presented to the emergency ward of our department at a tertiary care hospital with complaints of heavy bleeding per vaginum along with passage of clots during her 1st menstrual cycle. She was referred from a primary health center in view of her menorrhagia with severe anemia. She was a known case of Glanzmann thrombasthenia which was diagnosed when she was 8 years old during screening as her younger brother had died of the same disease. There was no history of consanguinity in her parents. She had history of epistaxis and easy bruising on and off since childhood. She came with complaints of soakage of 8-10 pads/ day for almost 10 days. Bleeding was not controlled with intravenous (IV) tranexamic acid that was started at the primary health center. 

On examination she was markedly pale, her vital parameters were stable and there was no evidence of organomegaly on per abdomen findings. She was started on tablet medroxyprogesterone acetate (MPA) 10 mg tid, and IV tranexamic acid 500 mg tid. Both were continued for 3 days. Blood investigations like hemoglobin, coagulation profile, liver and renal function tests were sent and she was sent to hematologist for opinion where she was admitted. She had an initial hemoglobin level of 5.6 gm % for which she was given 2 units of blood transfusion. Repeat hemoglobin was 8.5 gm % which dropped to 3.5 gm % as her menorrhagia continued over a period of 3-4 days. She was given multiple blood transfusions for the same and serial monitoring of hemoglobin was done. In spite of this her menorrhagia persisted and finally the patient was started on combined oral contraceptive pills and injectable ethamsylate. In spite of oral contraceptive pills in the dose of 2 tablets tid for 5-6 days her heavy bleeding continued & therefore she was given injection testosterone enanthate 50 mg IM daily for 4 doses over 48 hours and Novo 7. Novo 7 which is Factor VII was given in the dose of 90 mcg/ kg. Single dose of 2500 mcg (with a half-life of 3 hours) was given IV every 3 hours for 5 days upto a total dose of 50 mg (20 doses) as advised by the hematologist. Since that failed to control bleeding,  injection Leuprolide acetate 3.75 mg, was given with a view to completely suppress the hypothalamo-pituitary ovarian axis. Leuprolide acetate was actually contraindicated because of her age and premature suppression of HPO axis but it was used as a life saving medication. She was soaking around 8-10 pads/ day which was a subjective assessment but bleeding stopped completely in 2 days after starting injection Leuprolide. Her general condition improved & hemoglobin of 7.7g/dL was achieved at the time of discharge. Total of 25 blood transfusions, along with parenteral albumin transfusion for hypoproteinaemia along with the above mentioned medical management one after the other over 19 days were given. She was discharged and was advised to start on tablet tranexamic acid 500 mg tid from day 1 of her next menses and to follow up in emergency ward if bleeding is not controlled on oral tranexamic acid.

Discussion

Glanzmann’s thrombasthenia (GT) is a genetic disorder which is autosomal recessive most commonly seen in patients who give positive history of consanguineous marriages. Heterozygotes are asymptomatic with normal platelet function tests. The glycoprotein complex IIb/ IIIa binds to fibrinogen when activated in normal coagulation pathways and bridges the platelets in the presence of calcium to form platelet aggregates which are responsible for the clotting mechanism. In patients with GT this complex is deficient and hence the pathology. In spite of normal platelet counts patient exhibits bleeding manifestations. Three groups of this disease are found. Type I have less than 5 % of the normal GP IIb/ IIIa complex and clot retraction is absent. Type II have 5-20 % of the normal complex and clot retraction is impaired. Type III are variants who have normal receptor levels but defective receptor function.[5] Our patient was a case of Type I GT as diagnosed by flow cytometry.

The severity of bleeding is independent of the type of the disease. Carriers or heterozygotes have normal hemostatic function. Epistaxis, gingival bleeding, purpura and menorrhagia in women are recurrent features seen in Glanzmann’s thrombasthenia. Bleeding time is prolonged. Normal platelet count, morphology and a normal coagulation profile is usually seen on laboratory evaluation. These patients have abnormal clot retraction time and when the test is performed using collagen and ADP epinephrine then platelet aggregation test results are abnormal due to dependence of these agents on fibrinogen. Fibrinogen attachment to the platelets is essential for these factors to cause aggregation. Platelet aggregation occurs in response to ristocetin because of its independence from fibrinogen.[6] Receptor assay using flow cytometry can be done to detect the presence of the GP IIb-IIIa complex, GP IIb (CD 41), GP IIIa, (CD 61) and fibrinogen by using monoclonal antibodies. The same method is used to detect the carrier status of family members with this disorder.[7] The goal of treatment for patients with factor VII deficiency is to control bleeding episodes. Recommended treatment is platelet transfusions. However, about 15-30 % of patients do not respond to these transfusions and this may be because they have developed antibodies to GP IIb-IIIa and/ or HLA antibodies. They have also developed presently or in the past refractoriness to platelet transfusions. The management of patients with GT is challenging and requires multidisciplinary approach. If the bleeding is serious then vigorous treatment should be initiated to reduce further life threatening complications. Thus the step wise management is an initial trial of anti-fibrinolytics (tranexamic acid or ethamsylate) followed by hormonal support (high dose progestins or combined oral contraceptive pills) for maintenance. Blood and blood products are administered to correct anemia.

Every transfusion increases the risk of alloimmunization. Repeated platelet transfusions lead to platelet refractoriness by formation of platelet glycoprotein antibodies and Human Leucocyte Antigen (HLA) immunization. This increases the susceptibility to a massive hemorrhage.[8] All patients need to be vaccinated against Hepatitis B and medications affecting platelet function should be avoided. Such cases benefit from newer modalities of treatment such as Factor VIIa therapy. Factor VIIa therapy was initially used for treatment of Hemophilia but now has been successfully used to treat hemorrhage from bleeding disorders such as Glanzmann Thrombasthenia.[9, 10] Patients with Glanzmann's thrombasthenia have antibodies to GP IIb-IIIa and/ or HLA along with past or present refractoriness to platelet transfusions. Factor VIIa (Novo7) is essential for the treatment of bleeding episodes and for the prevention of bleeding during surgery or invasive procedures. The recommended dosage is 90 mcg/ kg; range is 80- 12 mcg/ kg by intravenous injection every 2 hours; range is 1.5- 2.5 hours. To secure hemostasis at least three doses should be administered. Factor VIIa has been used with good effectiveness in Glanzmann's thrombasthenia patients. It has low risk of transmission of hepatitis virus or retroviruses as it is free of human plasma and protein (albumin). If the patient has already developed alloimmunization the only treatment option remaining is plasmapheresis.

As far as management is concerned, it is advisable to avoid intramuscular injections as well as NSAIDS in these patients. Platelet transfusion should be given only if bleeding is life threatening and not for minor petechiae.

Conclusion

Glanzmann’s thrombasthenia patients do well with necessary supportive care. Spontaneous bleeding is not common. Post traumatic bleeding can occur, which can be serious and even life threatening. The need for immediate and appropriate individualized medical measures forms the mainstay of management in rare coagulation disorders like Glanzmann thrombasthenia.

References

1. Pittman MA, Graham JB. Glanzmann's thrombopathy: an autosomal recessive trait in one family. Am J Med Sci. 1964;247:293-303. 
2. Glanzmann E. Hereditaire Hamorrhagische thrombasthenic. Ein Beitrag Zur Pathologie der Blutplattchen. Jahrbuch Kinderheilkde. 1918;88:1-42. 
3. Poon MC. Clinical use of recombinant human activated factor VII (rFVIIa) in the prevention and treatment of bleeding episodes in patients with Glanzmann’s thrombasthenia. Vasc Health Risk Manag. 2007;3(5):655-64. 
4. George JN, Caen JP, Nurden AT. Glanzmann's thrombasthenia: the spectrum of clinical disease. Blood. 1990;75(7):1383-95. 
5. Bisch FC, Bowen KJ, Hanson BS, Kudryk VL, Billman MA. Dental considerations for a Glanzmann’s thrombasthenia patient: case report. J Periodontol. 1996;67(5):536-40. 
6. Fernandes GM, de Melo RM, Plens G, Pontes EM, Silva MM, da Rocha JC. Surgical and clinical management of a patient with Glanzmann’s thrombasthenia: a case report. Quintessence Int. 2004;35(8):617-20. 
7. Sharp WJ, Khanduri UD, Christie BS. Rapid heterozygote detection in Glanzmann's thrombasthenia.Br J Haematol. 1998;101(1):66-9. 
8. Kantarci A, Cebeci I, Firatli E, Atamer T, Tuncer O. Periodontal management of Glanzmann’s thrombasthenia: report of 3 cases. J Periodontol. 1996;67(8):816-20.  
9. Franchini M, Zaffanello M, Veneri D. Recombinant factor VIIa. An update on its clinical use. Thromb Haemost. 2005;93(6):1027-35. 
10. Balci YI,  Karabulut A, Kabukcu S, Sari I, Keskin A. Intensive Menstrual Bleeding Successfully Treated With Recombinant Factor VIIa in Glanzmann Thrombasthenia. Clinical and Applied Thrombosis/Hemostasis.2011;17(4):320-2.

Citation

Vaidya A, Hatkar PA, Satia MN, More V. Puberty Menorrhagia Due To Glanzmann’s Thrombasthenia. JPGO 2017. Volume 4 No.5. Available from: http://www.jpgo.org/2017/05/puberty-menorrhagia-due-to-glanzmanns.html