Volume 6 Number 2

Editorial
Gupta AS

Cesarean Scar Endometriosis
Bijapur S, Panchbudhe S, Parulekar SV.

A Case Of Advanced Glioma In Third Trimester Of Pregnancy
Solanke SS, Pednekar R, Parulekar SV.

A Rare Case Of Primary Abdominal Ectopic Pregnancy
Agrawal P, Honavar P, Samant P.

Episiotomy Granuloma Due To Nonabsorbable Suture Material
Gaikwad C, Prasad M, Gupta AS.

Post Traumatic Vaginal Stenosis
Saxena A, Koshewara P, Gupta AS.

Fibro-Epithelial Polyp Of The Vulva
Srinivasan N, S Panchbudhe S.


Ovarian Stimulation and Preeclampsia- Dual Causality Association with Subcapsular Hematoma?
Bharti S, Malani K, Yadav K, Samant P.

Cystadenofibroma Of Ovary
Ruhil MS, Dwivedi JS, Hatkar PA, Kothari K.


Remembering Past Greats: Ian Donald
Prasad M, Venkatesh S.

Editorial

Gupta AS

Liver disorders in pregnancy cause significant maternal and fetal morbidity and mortality. An obstetrician encounters 3 types of liver disorders while treating pregnant women. First are the incidental liver diseases that are caused by infective etiology like hepatitis A, B, C, E, Dengue hepatitis, and malaria. The second type of women are those already afflicted by chronic liver diseases or portal hypertension and have a coincidental pregnancy. There are five pregnancy related or pregnancy specific conditions of which three would require urgent intervention in the form of immediate delivery at the onset of the conditions. These five conditions are hyperemesis gravidarum that occurs in the first trimester, intra hepatic cholestasis of pregnancy, HELLP (hemolysis, elevated liver enzymes and low platelets), pre-eclamptic hepatic dysfunction, AFLP (acute fatty liver of pregnancy). The last three can prove catastrophic to the mother and child if the woman is not delivered immediately. I will be highlighting a few aspects of AFLP which has remained an under diagnosed condition.
AFLP is usually a condition of young primigravidas, in second half of their pregnancy who probably had a compensated hepatic mitochondrial fatty acid oxidation mechanism pre pregnancy. Decompensation of this process occurs in pregnancy probably due to the fetus being homo or heterozygous carrier of this autosomal recessive genetic disorder. Thus a compensated energy deficient woman decompensates when need for energy increases as in late pregnancy. Thus it can also be called as Mitochondrial hepatopathy. Prompt diagnosis and delivery can improve the maternal outcome as the placenta having the same genetic composition of the fetus increases the fatty acids (arachidonic acid) and its oxidative metabolites in the maternal circulation thus increasing load on the hepatic mitochondria.
Biochemical tests of liver are unaffected by pregnancy. Presence of jaundice, serum bilirubin levels usually very high or greater than 10 mg%, with aminotransferase levels moderately elevated, coagulopathy, abnormal serum creatinine levels, hypoglycemia (blood sugar less than 60 mg%), negative hepatic viral markers and blood smear negative for malarial parasite should make an obstetrician suspect AFLP. Clinical (Swansea) diagnostic criteria can be used to diagnose AFLP. Of the 14 parameters listed, presence of 6 in the absence of any other cause for liver failure are sufficient to aid early diagnosis of AFLP so as to start immediate, aggressive treatment (delivery). Abdominal pain, jaundice, vomiting, increased thirst or urination, encephalopathy, elevation of serum aminotransferases, creatinine, ammonia, white blood cell counts, uric acid levels, and prothrombin time; decreased blood sugar levels, presence of ascites/ bright liver on ultrasound and diagnostic liver biopsy showing diffuse or perivenular microvesicular steatosis are the 14 clinical parameters for diagnosing AFLP. These criteria have been validated against liver biopsy (gold standard). Liver biopsies are delayed due to coagulopathy. Prompt, early delivery after correction of coagulopathy can reduce maternal mortality to 10 % or less.
HELLP and pre-eclampstic liver dysfunction patients clinical presentation can overlap with AFLP and vice versa leading to difficulty in diagnosis. However, all three life threatening conditions have to be managed by urgent delivery. Multi disciplinary team approach, intensive care and urgent delivery can significantly improve obstetric outcomes.
An interesting care of pregnancy with in vitro fertilization appears in this issue who had hepatic dysfunction. The postulation of incipient pre pregnancy hepatic parenchymal insult due to ovarian hyperstimualtion leading to subsequent development of liver subcapsular hematoma makes for interesting reading.

Cesarean Scar Endometriosis

Author Information

Bijapur S*, Panchbudhe S**, Parulekar SV***.
(* Third Year Resident, ** Assistant Professor, *** Professor and Head, Department of Obstetrics and Gynecology, Seth G S Medical College & K E M Hospital, Mumbai, India.)

Abstract

Cesarean scar endometriosis is a rare presentation of extra pelvic endometriosis. Usually, it presents as a triad of underlying mass at the incision site, cyclical menstrual scar site pain and history of previous gynecological or obstetric surgery leading to the diagnosis. We report our experience in managing cesarean scar endometriosis and highlight the diagnosis and treatment options.

Introduction

Endometriosis at the site of previous surgery scar is increasing now-a-days, mainly due to increased rate of cesarean sections. Cesarean scar endometriosis (CSE) is an unusual manifestation of extra pelvic endometriosis, reported to be 0.03 – 0.45% in women undergoing cesarean sections.[1] Endometrial cells are implanted directly into the surgical site and can progress to endometriosis in optimal conditions. Here we discuss a case of scar endometriosis developing in the scar of previous cesarean section, 2 years after the primary surgery.

Case Report

A 36 year old woman, married for 18 years, para 1 living 1, with previous lower segment cesarean section (LSCS), presented to the outpatient department with complaints of lump and pain over the scar of previous LSCS for 1 year, the pain being dull aching and increasing during menstruation. She had undergone LSCS 2 years back, in view of meconium stained amniotic fluid. Operative notes showed that the procedure was uneventful. Thea patient also gave history of undergoing diagnostic hystero-laparoscopy 3 years back in view of primary infertility. Operative notes were not available. Her general and systemic examination findings were normal. On local examination, a healthy Pfannenstiel scar of LSCS was noted. There was a firm, minimally tender lump of around 2x3 cm close to the left angle of the Pfannenstiel scar, which was subcutaneous in origin. There was another 2X2 cm mass on the right side near the end of the scar. On speculum examination, her cervix and vagina were healthy, and bimanually uterus was anteverted, normal sized, with clear fornices. Ultrasonography done showed features suggestive of scar endometriosis, around 2.6X2 cm in the subcutaneous plane at the scar site in lower abdomen. Her investigations for fitness for anesthesia showed normal results. She was posted for excision of scar endometriosis.

Transverse incision was taken over the skin near the previous Pfannensteil scar and subcutaneous tissue reached. A firm, nodular mass of around 3X2 cm found near upper margin of left side of the incision. It was excised en-mass by sharp and blunt dissection, along with a small part of surrounding normal tissue to ensure complete removal and sent for histopathological examination. The mass extended into the rectus sheath, hence part of rectus sheath was also excised. Similar procedure was repeated on right side for nodule of 2X2 cm, found near the right end of scar. The cut section of the mass showed chocolate colored fluid. The defect in the rectus sheath was closed with a polypropylene mesh of 6X2 cm with intermittent sutures of No. 1-0 polypropylene and then subcutaneous tissue and skin were closed. Postoperative course was uneventful. Histopathology report was suggestive of endometriosis. The patient was offered injection leuprolide depot for 6 months for ovarian axis suppression.

Figure 1. Clinical appearance of the lesion. The extent of the endometriosis is shown by arrows.

Figure 2. Intraoperative appearance of the lesion.

Figure 3. Polypropylene mesh is being sutured to the edges of the defect in the rectus sheath.

Discussion

Endometriosis is defined as occurrence of functioning endometrial tissue, including glands and stroma outside the uterine cavity. It is most commonly seen on peritoneum, surface lining of the pelvic cavity, ovaries, posterior cul-de-sac, and uterosacral ligaments. In very few cases, implants of endometriosis can occur outside the pelvis, called as extra pelvic endometriosis. It is mainly seen in women of reproductive age.
The pathogenesis of scar endometriosis is believed to be the result of mechanical iatrogenic implantation, by direct inoculation of the abdominal fascia and/or subcutaneous tissue with endometrial cells during surgical procedures like cesarean sections, hysterectomies, tubal ligations, ovarian cystectomies and amniocenteses, which, stimulated by estrogen, become active and expand. The most prominent risk factor for the presence of endometriosis in scar tissue is previous history of obstetric surgical procedures.[3] 
The presence of hormone-sensitive tissue under the skin explains the clinical features including cyclic pain, swelling and blood-like brown leakage during menstruation (found only during surical excision, since the lesion usually does not communicate with outside). Pain, either cyclical or noncyclical, is the most common major symptom, reported by more than 80% of patients.[4] With respect to imaging, ultrasound is the most reliable imaging tool for the diagnosis of CSE, providing a differential diagnosis of incisional hernia, abscess, hematoma, cyst or lipoma in most cases.[5] It can also help reveal deep infiltration guiding the surgical excision. Computed tomography or magnetic resonance imaging are rarely needed. Fine needle aspiration cytology (FNAC) is a cost-effective, and accurate diagnosis tool to be included in the diagnostic tests.
Therapeutic management is essentially based on wide surgical excision, with clear margins and reconstruction of affected tissue and is the one overly recommended.[6] Medical treatment with hormone suppression has been tried but recurrence is common. Recently, there have been reports of the use of the gonadotrophin agonist (leuprolide acetate), found to provide only prompt improvement in symptoms with no change in the lesion size.[7] Follow up of endometriosis patients is important because of the chances of recurrence and possibility of malignancy. Using a good technique and taking proper care while performing a cesarean section may help prevent scar endometriosis.

References

1. N. S. Nominato, L. F. V. S. Prates, I. Lauar, J. Morais, L. Maia, and S. Geber, “Caesarean section greatly increases risk of scar endometriosis,” European Journal of Obstetrics Gynecology and Reproductive Biology, vol. 152, no. 1, pp. 83–85, 2010.
2. M. Paşalega, C. Mirea, I. D. Vîlcea et al., “Parietal abdominal endometriosis following cesarean section,” Romanian Journal of Morphology and Embryology, vol. 52, no. 1, pp. 503–508, 2011.
3.  Leite GK, Carvalho LF, Korkes H, Guazzelli TF, Kenj G, Viana Ade T. Scar endometrioma following obstetric surgical incisions: retrospective study on 33 cases and review of the literature.
4. J. Zhang and X. Liu, “Clinicopathological features of endometriosis in abdominal wall—clinical analysis of 151 cases,” Clinical and Experimental Obstetrics and Gynecology, vol. 43, no. 3, pp. 379–383, 2016.
5. J.-H. J. Hensen, A. C. Van Breda Vriesman, and J. B. C. M. Puylaert, “Abdominal wall endometriosis: clinical presentation and imaging features with emphasis on sonography,” American Journal of Roentgenology, vol. 186, no. 3, pp. 616–620, 2006.
6. E. M. Oh, W. Lee, J. M. Kang, S. T. Choi, K. K. Kim, and W. K. Lee, “A surgeon’s perspective of abdominal wall endometriosis at a caesarean section incision: nine cases in a single institution,” Surgery Research and Practice, vol. 2014, Article ID 765372, 4 pages, 2014. 
7. Rivlin ME, Das SK, Patel RB, Meeks GR. Leuprolide acetate in the management of cesarean scar endometriosis.

Citation

Bijapur S, Panchbudhe S, Parulekar SV. Cesarean Scar Endometriosis. JPGO 2019. Vol 6 No. 2. Available from: https://www.jpgo.org/2019/01/cesarean-scar-endometriosis.html

A Case Of Advanced Glioma In Third Trimester Of Pregnancy

Author Information

Solanke SS*, Pednekar R**, Parulekar SV***.
(* Third Year Resident, ** Assistant Professor, *** Professor and Head, Department of Obstetrics and Gynecology, Seth G S Medical College & K E M Hospital, Mumbai, India.)

Abstract

Glioma presenting during pregnancy carries unique challenges to the patient, baby, family, and health care providers. We describe an unusual case of advanced glioma  that was diagnosed for the first time at 34 weeks of gestation. While her medical and obstestric management was associated with a significant ethical dilemma, the mother and fetus successfully survived through the neuorosurgery and the woman delivered a healthy baby spontaneously vaginally four days post decompression neurosurgery i.e. right frontal craniotomy with excision of right frontal glioma. The objective in presenting this particular case is to highlight management of advanced glioma  in pregnancy and determining mode of delivering a baby of a pregnant female with advanced glioma.

Introduction

Central nervous system diseases, including intracranial tumors, particularly malignant brain tumors and trauma, remain a leading cause of indirect maternal mortality.[1] Primary central nervous system tumors occur in about 6 in 100000 females, but are rare during pregnancy.[2] A study reported the incidence of maternal malignant brain tumors at 3.6 per 1 million live births.[3] The management of pregnant women with malignant brain tumors has not been well evaluated. The present study reports a case of a pregnant woman in the third trimester with a glioma. The patient underwent craniotomy for brain tumor resection under general anesthesia at 36 weeks of gestation and had a spontaneous by preterm vaginal delivery at 36 weeks and 4 days of gestation.

Case Report

A 34-year-old primigravida, married for 1year, with 34 weeks of gestation presented with a complaint of 5 episodes of giddiness with brief period of loss of consciousness over 7 days.  She was evaluated by her neurologist. Her magnetic resonance imaging (MRI) of the brain showed neoplastic etiology, i.e., diffuse infiltrative glioma described as 7X8.5 cm infiltrating mass involving left frontal lobe, left gangliocapsular region, genu and body of corpus callosum, insula and left superomedial temporal lobe with subfulcrine herniation and midline shift to right by 1.5 cm. After multidisciplinary consultation with the obstetrics, general medicine, neurosurgery, neuromedicine and anesthesiology departments, the initial decision was to delay the neurosurgery until after delivery. The patient and her family approved this strategy. The decision was made to treat the patient with dexamethasone and levetiracetam to control the seizures and accelerate fetal lung maturity. The patient’s antenatal profile investigations and anomaly scan result were within normal limit. However, her condition worsened. She showed signs of cerebral edema and increased intracranial pressure. She had five episodes of seizures in the ward which were controlled with intravenous phenytoin and dexamethasone.  She was completely eptonized with phenytoin 100 mg PO q8h and dexamethasone 4 mg q8h. Then a decision to perform left craniotomy with excision of glioma was made for decompression of the brain. Proposed neurosurgery was done under general anesthesia at 36.0 weeks of gestation in supine positon. Fetal heart rate (FHR) monitoring was done intraoperatively. FHR remained about 130 beats per minute (bpm) with no deccelerations. The patient recovered quickly from anesthesia without any neurological deficit and was extubated in the operating room. Few minutes after extubation, FHR baseline shifted to 110 bpm. Therefore continuous fetal heart rate monitoring was done for 4 more hours. FHR increased to 140 bpm 4 hours later. Non stress test done on second day of post operative period was reactive. 72 hours post surgery, the patient went into spontaneous labor. The labour progressed well. A live male child weighing 2.662 kg was delivered by preterm vaginal delivery. The baby cried immediately after birth. The neonate’s Apgar score at 1, 3, and 10 min was 9/10. Oxytocin (20 units) was administered i.v. to improve the uterine contraction. The histopathology report revealed an oligodendroglioma  WHO grade II. The patient was discharged 14 days after surgery. Adjuvant chemo-radiotherapy was planned 2 weeks after surgery. The mother remained in a good condition without any neurological deficit and the baby was kept with the mother and relatives from day one of life.

Figure 1. MRI of brain.

Discussion

Modern treatment has improved prognosis in glioma patients so that more young women with glioma present for management. [4,5]. Peeters et al found in a multicenter case series that there was tumor progression and clinical deterioration  of gliomas during pregnancy, and cesarean section did not improve the outcome in any way. [4] benefit of CS over vaginal delivery with respect to adverse events, in stable women at term. This was confirmed by other non biased studies too.[6,7,8] In a multicenter, prospective case-control study of 129 children, Amant et al found that there was no significant difference in cardiac and neurocognitive outcome with prenatal exposure to different cancers and their therapy. [9] Van Calsteren K et al reported that prenatal exposure to chemotherapy increased incidence of preterm labor and intrauterine fetal growth restriction.[10] The risk of development of congenital malformations was not reported to be increased. However the long term effects were not addressed in these reports. Further studies are required to study such effects. There are some unanswered questions in the management of pregnant women with gliomas. There is no consensus on issues like when a pregnancy should be discouraged, best schedules for monitoring such gravidas and their fetuses, schedules for safe administration of radio-  and chemotherapy to such patients and the best mode of delivery. There is also a need for multicentric registries of such tumors and their management and obstetric and patients' outcomes.
Our patient had a lucky outcome. The time of presentation was at a reasonably advanced gestational age, so that the childbirth had to be deferred only for a few weeks, postponing radio- and/or chemotherapy. Her surgery was successful and there were no neurological postoperative complications. She went into spontaneous labor, progressed and delivered vaginally without any mishap. We have presented this case so that the number of cases of gliomas in pregnancy increases and an analysis of a large number of such cases in future can help one draw meaningful conclusions.

References

1. Cantwell R, Clutton-Brock T, Cooper G, Dawson A, Drife J, Garrod D, et al. Saving Mothers' Lives: Reviewing maternal deaths to make motherhood safer: 2006-2008. The Eighth Report of the Confidential Enquiries into Maternal Deaths in the United Kingdom. BJOG. 2011 Mar;118 Suppl 1:1-203. doi: 10.1111/j.1471-0528.2010.02847.x.
2. Chang L, Looi-Lyons L, Bartosik L, Tindal S. Anesthesia for cesarean section in two patients with brain tumours. Can J Anaesth. 1999 Jan; 46(1):61-5.
3. Haas JF, Jänisch W and Staneczek W. Newly diagnosed primary intracranial  neoplasms  in  pregnant  women:  a population-based assessment. J Neurol Neurosurg Psychiat 1986;49:874-880.
4. Claus EB, Black PM. Survival rates and patterns of care for patients diagnosed with supratentorial low-grade gliomas: Data from the SEER program, 1973–2001. Cancer. 2005;106(6):1358–1363. doi: 10.1002/cncr.21733.
5. Peeters S, Pagès M, Gauchotte G, Miquel C, Cartalat-Carel S, Guillamo JS. Interactions between glioma and pregnancy: insight from a 52-case multicenter series. J Neurosurg. 2017;128:1–11.
6. Pallud J, Duffau H, Razak RA, Barbarino-Monnier P, Capelle L, Fontaine D et al. Influence of pregnancy in the behavior of diffuse gliomas: clinical cases of a French glioma study group. J Neurol. 2009;256(12):2014–2020. doi: 10.1007/s00415-009-5232-1.
7. Zwinkels H, Dörr J, Kloet F, Taphoorn MJB, Vecht CJ. Pregnancy in women with gliomas: a case-series and review of the literature. J Neurooncol. 2013;115:293–301. doi: 10.1007/s11060-013-1229-9.
8. Abd-Elsayed AA, Díaz-Gómez J, Barnett GH, et al. A case series discussing the anaesthetic management of pregnant patients with brain tumours. F1000Research. 2013;2(92):1–14.
9. Amant F, Vandenbroucke T, Verheecke M, Fumagalli M, Halaska MJ, Boere I, et al. Pediatric Outcome after Maternal Cancer Diagnosed during Pregnancy. International Network on Cancer, Infertility, and Pregnancy (INCIP). N Engl J Med. 2015 Nov 5; 373(19):1824-34.
10. Van Calsteren K, Heyns L, De Smet F, Van Eycken L, Gziri MM, Van Gemert Wet al. Cancer during pregnancy: an analysis of 215 patients emphasizing the obstetrical and the neonatal outcomes. J Clin Oncol. 2010 Feb 1; 28(4):683-9.

Acknowledgments

We thank Dr Chagla AS and Dr. Akshay Hadwalkar from department of neurosurgery for neurosurgical management of the patient.

Citation

Solanke SS, Pednekar R, Parulekar SV. A Case Of Advanced Glioma In Third Trimester Of Pregnancy. JPGO 2019. Vol 6 No. 2. Available from: https://www.jpgo.org/2019/01/a-case-of-advanced-glioma-in-third.html

A Rare Case Of Primary Abdominal Ectopic Pregnancy

Author Information

Agrawal P*, Honavar P**, Samant P***.
(* Junior Resident, ** Assistant Professor, *** Academic Professor, Department of Obstetrics and Gynecology, Seth G  S Medical College and KE M Hospital, Mumbai, India.)

Abstract

A patient with a history of 6 weeks amenorrhea, with urine pregnancy test positive and ultrasound scan suggestive of ectopic pregnancy in left adnexa with extensive hemo peritoneum, on laparotomy revealed a primary abdominal ectopic pregnancy. The presentation and management of the case has been described in the case report.

Introduction

Following fertilization and fallopian tube transit, the blastocyst normally implants in the uterine cavity. Implantation elsewhere is considered as ectopic pregnancy. The most common ectopic implantation site is the fallopian tube, though ectopic pregnancy is known to implant anywhere in the pelvis and abdomen.[1] One advanced abdominal pregnancy occurs for every 8099 hospital deliveries.[2] Here we discuss an interesting case of primary abdominal ectopic pregnancy.

Case Report

A 26 year old woman, with one previous vaginal delivery, came with complaints of 6 weeks amenorrhea, acute abdominal pain, vomiting and giddiness. On examination the pulse was 100 beats per minute, and blood pressure was 90/60 mm of Hg. Abdomen was tender and appeared distended. Per vaginal examination revealed bilateral cervical motion tenderness. Uterus was six weeks gestation size. Hemoglobin was 8.3 g/dl. Ultrasonography was suggestive of ruptured live ectopic pregnancy of 9 weeks in the left adnexa with hematoma of 350 cc. After consent, in view of ruptured ectopic pregnancy, exploratory laparotomy was done. Intraoperative 650 g blood clots were retrieved with approximately 1600 ml hemoperitoneum. Bilateral fallopian tubes and ovaries looked normal with no evidence of gestational sac (figure 1). Bladder was found densely adherent to fundus and anterior wall of the uterus (figure 2). Appendix was adherent to the right fallopian tube. Rent of about 4 x 3 cm was noted above the left uterosacral ligament (figure 3). Gestational sac was seen adhered to the posterior wall of the uterus near the left uterosacral ligament. A rent on the posterior wall of the uterus was seen after separation of the gestational sac which had bleeding base hence required repair of the rent. This extrauterine pregnancy was not communicating with the uterine /cervical cavity, both adnexa were normal and the gestation sac was deriving its blood supply from its area of implantation; posterior wall of the uterus near the left uterosacral ligament thus suggesting it to be a primary abdominal pregnancy. It also was not a tubal abortion as the tubes were healthy and the gestational sac was extracted from the posterior surface of uterus leaving behind a bleeding vascular surface. Ectopic gestation was removed and a Urologist was called for ureteric tracing due to its close proximity to the raw area and uterine rent repair was done. Uterine curettage that was done due to a thickened endometrium showed an Arias Stella reaction. As initial impression was that of a cervical pregnancy having ruptured intraperitoneally curettage was done. It was noted that there was no communication of the cervical canal with the peritoneal cavity. Four units of fresh frozen plasma and 5 units of packed cell volume were given during the operation. Histopathology of peritoneal contents showed products of conception.
She had a stormy post operative course with shock liver, consequent to intraoperative hypotension. She was administered albumin and liver safe antibiotics. Ascites caused wound dehiscence, but wound cultures did not grow any organisms. The wound healed with secondary intention. She made a gradual recovery and was discharged after 3 weeks with advice about barrier contraceptive.

Figure 1. Posterior aspect of uterus showing bilateral normal fallopian tubes and ovaries.

Figure 2. Anterior aspect of uterus showing densely adherent bladder to the uterine fundus and anterior wall.

Figure 3. Uterine site of implantation of ectopic gestation (black arrow) with ureteric tracing and isolation done (white arrow).

Discussion

Urine and serum beta-human chorionic gonadotropin (β-hCG) assays and transvaginal ultrasonography have made earlier diagnosis of ectopic pregnancy possible. Although a zygote can traverse the tube and implant primarily in the peritoneal cavity, most abdominal pregnancies are thought to follow early tubal rupture or abortion with reimplantation.[3]
Absence of an intrauterine gestational sac, absence of both an evidently dilated tube and a complex adnexal mass, a gestational cavity surrounded by loops of bowel and separated from them by peritoneum, a wide mobility similar to fluctuation of the sac, particularly evident with pressure of the transvaginal probe toward the posterior cul-de-sac, are the ultrasound diagnostic criteria of an early abdominal pregnancy.[4]
Systemic methotrexate therapy is a safe alternative to surgery in a stable patient with unruptured ectopic pregnancy.[5] Surgical management is the mainstay which may be done by laparotomy and includes removal of ectopic with careful assessment for removal of the placenta thus avoiding the possible torrential hemorrhage as the normal hemostatic mechanism of myometrial contraction is lacking.

Early abdominal pregnancy has similar risk factors to any ectopic pregnancy; these include a previous history of ectopic pregnancy or tubal surgery, endometriosis, history of pelvic inflammatory disease (PID) or current use of intrauterine device.[6] The patient in our case had a previous vaginal delivery. She gave no history of abdominal surgery or uterine curettage. She did not have any complaints or history suggestive of PID yet, intraoperative dense adhesions between urinary bladder and the anterior wall of uterus were seen. Also, appendix was found adherent to the right fallopian tube. Above findings cannot rule out a possibility of asymptomatic PID. Pelvic inflammation or increased vascularity could have created a nidus for implantation, thus explaining the probable cause of placentation into the abdominal cavity near the postero-inferior aspect of the uterus.
There was cervical motion tenderness on bimanual examination and the ultrasonography revealed left adnexal ectopic with significant hemoperitoneum. The above findings were consistent with the intraoperative findings. As the gestational sac was implanted just near the uterosacral ligament not involving the cervical canal or ureter, closure of rent formed after extraction of ectopic could be done satisfactorily. The cervical bottle neck could be felt during curettage.
Even if the incidence of abdominal pregnancy is low, early diagnosis and prompt treatment are essential to avert a catastrophic event. Hence, the treating clinicians must have high index of suspicion and carefully plan the management of all cases of ectopic accordingly.

Acknowledgement

We thank Dr. Priya Misar for clicking the intraoperative photos.

References

1. Panelli DM, Phillips CH, Brady PC. Incidence, diagnosis and management of tubal and nontubal ectopic pregnancies: a review. Fertility Research and Practice 2015;1(15):1-20.
2. Nunyalulendho ND, Einterz EM.  Advanced abdominal pregnancy: case report and review of 163 cases reported since 1946. Rural and Remote Health 2008;8:1087. Available: www.rrh.org.au/journal/article/1087
3. Cunningham FG. Ectopic pregnancy. Cunningham FG, Leveno KJ, Bloom SL, Spong CY, Dashe JS, Hoffman BL, et al. Ectopic pregnancy. In: Williams obstetrics. 24th ed. New York: McGraw-Hill Education; 2014.p. 388.
4. Gerli S, Rossetti D, Baiocchi G, Clerici G, Unfer V, Di Renzo GC. Early ultrasonographic diagnosis and laparoscopic treatment of abdominal pregnancy. Eur J Obstet Gynecol Reprod Biol 2004;113(1):103–5.
5. Talwar P, Sandeep K, Naredi N, Duggal BS, Jose T. Systemic methotrexate: An effective alternative to surgery for management of unruptured ectopic pregnancy. Medical Journal Armed Forces India 2013; 69(2):130-133.
6. Stuki D, Buss J. The ectopic pregnancy, a diagnostic and therapeutic challenge. J Med Life 2008; 1(1):40-8.

Citation

Agrawal P, Honavar P, Samant P. A Rare Case Of Primary Abdominal Ectopic Pregnancy. JPGO 2019. Volume 6 No.2. Available from: https://www.jpgo.org/2019/01/a-rare-case-of-primary-abdominal.html

Episiotomy Granuloma Due To Nonabsorbable Suture Material

Author Information

Gaikwad C*, Prasad M*, Gupta AS**.

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

Abstract

An episiotomy is to be sutured with absorbable sutures. If non-absorbable sutures are used, they can result in foreign body reaction, and other complication. A case of removal of linen sutures 6 months after delivery is being described here.

Introduction

Episiotomy is a very commonly performed surgical procedure. There are estimates that it may be the most commonly performed surgical procedure world over. Any complication of such a procedure bears significance to the obstetrician community. One such case is reported here.

Case Report

A 29 years old, Gravida 2, Para 2, Living 1, Stilborn 1 presented to us in the outpatient clinic with persistent yellowish vaginal discharge since 6 months. She was referred from a private center in view of suspected rectovaginal fistula. She had undergone a vaginal delivery at a primary health care center, outcome being a male stillborn due to birth asphyxia. Three days after delivery, she started complaining of foul-smelling discharge per vaginum for which she was admitted in a private hospital.  For puerperal sepsis with anemia, she was given parenteral antibiotics for 3 days and 1 unit packed red cells was transfused.  She was discharged on oral antibiotics, oral iron and antiseptic ointment for local application at the episiotomy site. After 4 months she again presented with vaginal discharge and persistent abdominal pain to the same clinic. She was investigated and was given clotrimazole vaginal pessary for 7 days. Oral fluconazole, azithromycin, secnidazole single dose combination was also given. Blood investigation reports were within normal limits. Due to persistent symptom of burning micturition, tablet Norfloxacin 400 mg BD was given for one week. X-ray abdomen and ultrasonography of the abdomen and pelvis were normal. After 10 days she was re-examined for persistent vaginal discharge, and due to suspected rent on the posterior vaginal wall, and suspicion of rectovaginal fistula, she was referred to our hospital. Pap smear was sent and it showed inflammatory smear. However, there were no problems with defecation.
General and systemic examination was within normal limits. Abdomen was soft, non-tender. On local examination, 3-4 linen sutures were seen on posterior vaginal mucosa, corresponding to the apex of the previously sutured episiotomy. (Figure 1) Few linen sutures were removed from skin at site of episiotomy. Per vaginal examination uterus was anteverted, and normal size. Few more linen sutures embedded within the mucosa were felt. Per rectal examination was done and the suture site was inspected to see for any rectovaginal fistula. No fecal material or gas bubbles could be expressed out at the suture site and no defect or puckering was felt within the rectal mucosa. For removal of the foreign body, she underwent a vaginal exploration. Episiotomy granuloma excision, removal of the foreign bodies and suturing of the mucosal defect was done.
Procedure was done under spinal anaesthesia.  Vaginal walls were retracted and the linen threads at episiotomy site were clearly visualized and were removed. (Figure 2) Granulation tissue on the vaginal mucosa along the line of the episiotomy was incised and raw bleeding mucosal edges were sutured to each other with polyglactin no 2-0 in continuous interlocking sutures. A mucosal defect with fibrous band was seen about 1cm proximal to the introital edge. (Figure 3) This was released vertically and the resultant edges were sutured. Hemostasis was achieved. She was relieved of her pain. She was reexamined 10 days and again after 4 weeks after the procedure, and she had no complaints.

Figure 1. Vaginal mucosa with linen suture in situ.

Figure 2. Removed linen suture.

Figure 3. Excised Fibrotic band in lower part of the vagina.

Discussion

Foreign bodies have the propensity to induce tissue reaction. The severity of reaction depends on the duration of the stay of the material inside the body. This can interfere with wound healing. A balance between strength of suture material to ensure healing as against adverse effect of chronic inflammation is critical.[1] In this case episiotomy was sutured with linen which is cause of granuloma formation. Unnecessary persistence of linen intra-abdominally causing problems have been described almost a century prior.[2]  Episiotomy is to be repaired only by absorbable sutures.[3] Multiple guidelines are available on the suture materials which are allowable for episiotomy, and non-absorbable sutures find no mention in them.[4,5]

Arguably, this case is similar to a retained foreign body, the foreign body here being a non-absorbable suture. All efforts should be taken to avoid such a scenario, since it can lead to potential medicolegal problems.[6]  Long term presence of foreign body in an episiotomy scar has been described by Lapinska et al. In that case, it was a surgical needle.[7] Our patient presented with repeated episodes of local genital tract infection in the first few months post delivery. This should have prompted a search for a foreign body, but since it was controlled with multiple courses of antibiotics, the diagnosis was delayed.

We have presented this case to highlight the fact that non-absorbable sutures are to be avoided in episiotomy suturing. This should be considered as a differential diagnosis in persistent vaginal discharge postdelivery

Conclusion

A case of non-absorbable suture material, removed remote from delivery period is described. Clinicians should be aware that even inadvertently, non-absorbable sutures should not be used for episiotomy suturing.

References

1. Rowan T. Non-Absorbable Sutures, Explained. 2018. Available from: http://knowledge.sourcebookmaterials.com/non-absorbable-sutures-explained
2. Mitchell AB. Persistence of silk and linen sutures. Br Med J. 1915 May 15; 1(2837): 870.
3. Kettle C, Dowswell T, Ismail KMK. Absorbable stitches for repair of episiotomy and tears at childbirth. 2010. Available from: https://www.cochrane.org/CD000006/PREG_absorbable-stitches-for-repair-of-episiotomy-and-tears-at-childbirth
4. Episiotomy and perineal repair guideline. (GL836). NHS. 2018. Available from:  http://www.royalberkshire.nhs.uk/Downloads/GPs/GP%20protocols%20and%20guidelines/Maternity%20Guidelines%20and%20Policies/Intrapartum/Episiotomy%20and%20perineal%20trauma%20repair_V7.1_GL836.pdf
5. Mullally A, Murphy DJ. Episiotomy.  Glob Libr. Women’s Med. 2011. Available from:  https://www.glowm.com/section_view/heading/Episiotomy/item/128
6. Sharma G, Bigelow JC. Retained foreign bodies: a serious threat in the Indian operation room. Ann Med Health Sci Res. 2014;4(1):30-7.
7. Lapińska S, Tokarska M, Emerich J, Sobol A. [A long-time presence of a foreign body in an episiotomy scar]. [Article in Polish] Ginekol Pol. 1998;69(9):714-6.

Citation

Gaikwad C, Prasad M, Gupta AS. Episiotomy Granuloma Due To Nonabsorbable Suture Material. JPGO 2019. Volume 6 No.2. Available from: https://www.jpgo.org/2019/01/episiotomy-granuloma-due-to.html

Post Traumatic Vaginal Stenosis

Author Information

Saxena A*, Koshewara P**, Gupta AS***.
(* Junior Resident, ** Senior Residnt, ** Professor, Department of Obstetrics and Gynecology, Seth G S Medical College and K E M Hospital, Mumbai, India.)

Abstract

Vaginal stricture in adults is not a common entity. In literature, vaginal stenosis and strictures can be multifactorial and there are different modalties used to tackle this problem. Patients diversely present with mild dyspareunia to severe dysmenorrhea and formation of hematocolpos. The biggest challenge is to prevent reformation of the strictures or re-stenosis of the vagina. Here we are presenting an unmarried, sexually active woman, a prior road traffic accident victim who presented with the complaint of apareunia and was managed surgically.

Introduction

Loss of elasticity or narrowing of the vagina is known as vaginal stenosis. Strictures can be congenital, due to trauma, infection, radiotherapy, idiopathic and other causes. These can present as dyspareunia, amenorrhea, dysmenorrhea or even abdominal masses. We are hereby presenting the case of a 24 years old, unmarried, sexually active woman with vaginal stricture, presenting to our OPD after 2 and a half years of a road traffic accident with complaints of apareunia.

Case Report

Twenty-four years old, unmarried woman had visited our out patient department (OPD) with the chief complain of apareunia due to vaginal stricture after two and a half years, following a road traffic accident.
In December 2015, she met with a road traffic accident and had suffered multiple injuries, hence was referred to our hospital for further management. She had suffered multiple crush injuries. The right knee and femur had multiple fractures. There was avulsion of the urethra, vagina and rectum. The posterior wall of vagina was hanging loosely. She was hemodynamically unstable.
She was examined in the emergency services department. On examination, per abdomen, the urinary bladder was distended up to the umbilicus. On local examination, urethral opening could not be seen due to lacerations around the urethra, posterior vaginal wall was hanging loosely and the perineal body was torn. The lower one third of vagina had a laceration, which connected it to the rectum. The left lateral wall of the vagina had a laceration exposing the left ischiorectal fossa. The posterior wall of the vagina was severely damaged. But due to the hemodynamic instability, she needed life saving surgeries first. Emergency open cystostomy and a diversion sigmoid colostomy were done. Since the patient was not stable hemodynamically at that time, vaginal repair could not be done immediately.

She was lost to follow up with us for 2.5 years, until she came to the gynecology OPD with the above complaints. She told us that after approximately 6 months of amenorrhea, she had spontaneously started menstruating again. She had no menstrual complains. Her only complaint was of apareunia which wasn’t the case before she had met with the accident.
On examination in the OPD, her vital parameters were stable and systemic examination was within normal limit. Inspection of the abdomen showed scars of previous surgeries and it was soft and non tender on palpation. On local examination of perineum, a scar was seen in the perineal area, extending upto the left side of the anus towards the left ischiorectal fossa. Speculum could not be inserted. Digital examination revealed a tight ring of scarring in the posterior lower end of vagina, 1 cm above the introitus. Only tip of the index finger could be inserted through the ring.  On per rectal examination, the examining finger did not indent the scar tissue anteriorly. Uterus was normal size and fornices were clear.
She was admitted for release of introital strictures in the post menstrual phase. Her routine investigations were within normal limits and ultrasonography of abdomen and pelvis was within normal limit.
On the day of surgery, a written valid informed consent was taken. Under spinal anesthesia, vaginal introitus appeared very narrow. On per vaginal examination a fibrous band of tissue was felt mid vaginally, on the posterior wall, extending from 4’o clock to 8’o clock position. The fibrous band was held with an Allis forceps in full thickness at 6’o clock position and transversely cut along its entire length. Upper vaginal mucosal edge was undermined to release some vaginal rugae to prevent tension on the suture line. The two mucosal edges were approximated to cover the raw area created, with suture material Polyglactin 910 (2-0), with simple interrupted sutures. The raw vaginal mucosa was thus epithelialized. The final introital diameter was approximately 3 cm.

Figure 1. Vaginal stricture prior to surgery.

Figure 2. End result after excision of the stricture.

She tolerated the procedure well and was discharged on the same day. She was asked to follow up after 1 week and to apply topical estrogen over the vaginal mucosa twice daily. A fortnight later, she came to our OPD for follow up and local examination was done. There was no evidence of restenosis or fibrosis. Vaginal mucosa was healthy. The suture site had healed completely. She was reassured that she could now resume with sexual intercourse.

Discussion

Vaginal stenosis can occur primarily or can occur secondary to many causes with varied presentations
R. Ganapathy evaluated secondary repair of the perineum following perineal trauma at childbirth and stated that a few patients developed superficial dyspareunia after perineal repair surgery.[1] Fowler reported a case of a 26 year old female patient who sustained a pelvic fracture at the age of 10. She later followed up in the gynecology department as she was unable to have sexual intercourse. She was diagnosed with vaginal stenosis. She was menstruating regularly but through a fistula in the distal vaginal vault. When investigated, radiographic and dye studies showed almost heterotopic ossification and complete vaginal obstruction. Vaginal injuries though rare, require a high index of suspicion.[2]

Vaginal stenosis does not always occur secondary to injury. It has also been described in women secondary to radiotherapy. Sato et al have reported a case of a 26 year old patient who had been given chemotherapy for acute lymphoblastic leukemia.[3] She presented with secondary amenorrhea and cyclic abdominal pain. She was diagnosed with vaginal stenosis due to adhesion of vaginal wall. The adhesion could have been caused due to vaginal inflammation induced by anticancer agents or due to poor estrogenization of the vaginal mucosa as a result of gonadotropin-releasing hormone agonist therapy. The latter was given for ovarian protection during chemotherapy. Since the incidence of such chemotherapy and protection with gonadotropin releasing hormone has increased, keen observation and adequate intervention would be important.

Although our patient was managed surgically other modalities for treatment of secondary vaginal strictures and stenosis have been tried. Betalli et al have reported a case of a 14-year-old girl who was admitted for recurrent abdominal pain.[4] The symptoms had begun at menarche (since 3 months) and had increased in intensity every month. She had been given chemotherapy at the age of 2 years for rhabdomyosarcoma. Hematocolpos, hematometra and hematosalpinx were diagnosed. Acute symptoms were partially relieved due to drainage with a needle through a vaginoscope. Magnetic resonance imaging (MRI) was done to exclude the recurrence of tumor. It also confirmed that a hematocolpos was still present. Vaginoscopy revealed the presence of a partially obstructing stenosis of the vaginal inlet which was about 1 cm in length. Dilators were used to widen the stricture and a stent was left in situ which was removed after 5 days. Due to persistence a second dilatation was performed after a month and Mitomycin-c was applied topically. After 1 month, endoscopic visualization demonstrated an improvement of the stricture but there was persistence of a submucosal fibrous ring in the vaginal lumen. The application of mitomycin-C was repeated at 3 and 7 months. Further improvement of the stricture was seen.
Yanai described a 25-year-old patient who presented with complains of secondary amenorrhea and dyspareunia. She suffered from vaginal outflow obstruction which presented as secondary amenorrhea. She had undergone bone marrow transplantation (BMT) for acute myelocytic leukemia, which caused ovarian failure. Oral mucositis as a result of chronic graft versus host (GVH) reaction also occurred. She was treated with hormone replacement therapy for 3 years and had regular menses which gradually ceased and were associated with dyspareunia and abdominal cramps. Abdominal ultrasonography was suggestive of hematocolpos. Sonography guided adhesiolysis of  dense vaginal obstruction allowed free drainage. Histopathology report confirmed a graft-versus-host reaction. When patients have graft-versus-host disease post BMT, they may develop vaginal stricture. After the procedure, high dose estrogen therapy (combined later with progesterone), was initiated and repeated vaginal dilatations were performed. Hormone induced menstruation reoccurred. Dyspareunia gradually disappeared.[5]

The best time of perineal repair in any patient is at the time of primary pelvic injury to restore anatomy. But as our patient was severely injured at the time of presentation and there were other life saving surgeries that needed to be performed, vaginal repair could not be done primarily. After that, the patient was lost to follow up for two years, till she came back with complains of inability to have coitus, which wasn’t the case prior to the accident.
If discovered incidentally, it is important to consider the age of the patient when the surgery should be performed. If there are no menstrual complaints or complaints of dysmenorrhea and the patient is sexually inactive, it is advisable to wait till the patient desires to become sexually active or is planning to conceive, since the chances of restenosis are very high.
In case, surgical or medical management is carried out in a sexually inactive patient, she has to be explained about regular use of dilators to prevent restenosis.

Conclusion

In our patient, since the fibrous tissue could be removed in totality, rugorosity of the vagina could be released and mucosal approximation was complete we believe that the chances of restenosis are less.
In conclusion, vaginal stenosis secondary to trauma is a rare case and active management of primary pelvic injury can prevent these from occurring. Active management of such cases in the post operative period is also very important to prevent restenosis with the use of dilators or topical creams or regular sexual intercourse. Regular follow ups are essential too, to detect and prevent restenosis.

References

1. Ganapathy R, Bardis NS, Lamont RF. Secondary repair of the perineum following childbirth. Journal of Obstetrics and Gynaecology. 2008; 28(6):608-613.
2. Fowler JA, Goodman GP, Evans JM, Schober JM. Long-term Vaginal Sequelae Secondary to Pediatric Pelvic Fracture. Journal of Pediatric and Adolescent Gynecology. 2009; 22(1):e15-19.
3. Sato M, Harada M, Oishi H, Wada-Hiraike O, Hirata T, Nagasaka K, et al. Vaginal stenosis after gonadotropin-releasing hormone agonist therapy during treatment for acute lymphoblastic Leukemia. Journal of Lower Genital Tract Disease. 2016; 20(2):e11-13.
4. Betalli P, De Corti F, Minucci D, Mazzarotto R, Meneghini L, Bisogno G, et al. Successful topical treatment with mitomycin-C in a female with post-brachytherapy vaginal stricture. Pediatric Blood Cancer. 2008; 51(4):550-552.
5. Yanai N, Shufaro Y , Or R, Meirow D. Vaginal outflow tract obstruction by graft-versus-host reaction. Bone Marrow Transplantation. 1999; 24(7);811–812.


Citation

Saxena A, Koshewara P, Gupta AS. Post Traumatic Vaginal Stenosis. JPGO 2019. Volume 6 No.2. Available from: https://www.jpgo.org/2019/01/post-traumatic-vaginal-stenosis.html

Fibro-Epithelial Polyp Of The Vulva

Author Information

Srinivasan N*, Panchbudhe S**
(* Junior Resident, ** Assistant Professor, Department of Obstetrics & Gynecology, Seth G S Medical College and K E M Hospital, Mumbai, India.)

Abstract

Fibroepithelial polyps are mesenchymal lesions that are site specific and usually found in the vulvovaginal area of females in their late reproductive ages. These tumors are usually small and are seen only during routine gynecological examination. Although the tumor is benign, it closely resembles some malignant vulvar lesions, therefore accurate histopathologic diagnosis is required. We present a case of a 24 year old female with a giant fibroepithelial stromal polyp of the vulva.

Introduction

Fibroepithelial polyps are site specific mesenchymal lesions that are and usually found in the vulvovaginal area of females in their late reproductive ages. They are mostly found in the vagina and rarely they are found in extra-genital sites. These are hormone sensitive tumors, they are most commonly seen in pregnancy and grow larger in size in pregnancy. They are also seen in pre-menopausal females on hormone replacement therapy. Typically these lesions do not grow large and are discovered during routine gynecologic examination. These tumors can have varied appearance, can be sessile, pedunculated, or polypoidal. Patients may present with complaints of bleeding, discharge and general discomfort over the area.

Case Report

A 24 year old nulliparus female; married since 6 months, admitted with complaints of a mass that had been growing on her left labium. Following a brief physical examination, verification was made of a left labial mass. She first noticed a marble-sized “bump” on her left labium about six months earlier. The mass gradually increased in size over last six months until its current size on presentation. She was on second day of her cycle and had no complaint of nausea, vomiting or other constitutional symptoms. There was no significant medical or surgical history. She had no history of any sexually transmitted diseases or gynecological surgery. She had regular menstrual cycles, 30 days cycle intervals and 4 to 6 day cycle lengths with moderate flow. She was sexually active since last 6 months, and complained of pain, dyspareunia at entry. She had no history of usage of any contraceptives or hormonal pills.
Her physical exam was remarkable only for a 5-6 cm, non-tender, lemon-sized, ulcerating pedunculated mass (red arrow) extending from the lower end of the left labium majus. On touch, lesion appeared solid but soft in consistency and was compressible upon application of gentle pressure. The mass was free from all sides, and had a 2-3 cm thick pedicle (blue arrow) that connected the mass with the lower edge of the left labium majus. The stalk was vascular with an ulcer over the serosal surface of the polyp.  There was no change in the size of the mass with Valsalva maneuver.
Transvaginal ultrasound showed normal anatomy of the uterus and ovaries but also described a broad-based encapsulated soft tissue mass arising from the left labium at its lower end. Her all routine investigations were within normal limits and she was tested seronegative for HIV/ HbsAg/ Ani-HCV/ RPR. She was posted for labial mass excision under short general anesthesia after taking written, valid and informed consent from her and her relatives. Intra-operative photography consent was also obtained. Semilunar incisions were placed on anterior and posterior aspects of the pedicle, joined with each other and deepened until the pedicle was cut off, separating the mass from the labium. Hemostasis is achieved with polyglactin no. 1 simple interrupted sutures placed over the labial muscle bed (Green arrow) and skin was approximated with No. 2-0 monofilament nylon vertical mattress sutures. She tolerated the procedure and anesthesia well. The pathology report suggested fibroepithelial stromal polyp and she was advised to return to her routine gynecology check up for continued surveillance.

The labial mass was globular, pedunculated, soft in consistency, measuring 5.0x3.5x2.0 cm On cutting opening the specimen, whitish pink glistening surface was seen (orange arrow). Skin over the stalk was dark brown in color, wrinkled and showed healed ulcers. Skin on the mass proper was pale and showed ulcers in various stages of healing with absence of myxoid areas. Microscopic evaluation of the lesion in our case revealed polypoidal lesion lined by keratinized stratified squamous epithelium. Focally, it was ulcerated and replaced by fibrino-suppurative exudates. Sub-epithelial tissue showed dense collagenization, mixed inflammation and ectatic blood vessels while deeper tissue was loose and showed scattered spindle and few stellate cells. Blood vessels of various calibre were also seen with few blood vessels thickened and dense lympho-plasmacytic infiltrate were seen along with perivascular inflammation suggestive of Fibro-epithelial polyp of the vulva.

Figure 1. Photo showing the polypoidal mass (red arrow) attached to the lower edge of left labium majus by a pedicle (blue arrow).

Figure 2. Photo showing free movement of the polyp without any adhesion to other parts of the genitalia.

Figure 3. Interrupted sutures taken on the vulvar polyp bed after resection of the mass (green arrow).

Figure 4. Cut section of the polyp showing glistening pinkish white tissue (orange arrow).

Discussion

Fibro-epithelial polyps of the vulva have different sizes and appearance. The margins of these tumors coalesce with healthy tissue and are thick-walled with a central core. Malignancy should be excluded in every diagnosis of fibroepithelial vulval polyp. Sarcomas have similar characteristics like that of fibroepithelial polyp and to differentiate between them, microscopic examination is essential for final diagnosis.[1]
The most characteristic microscopic feature of a fibroepithelial stromal polyp is the presence of stellate and multinucleate stromal cells which are identified near the epithelial-stromal interface.[2] The mesenchymal cells of the polyp can also stain positive for actin, desmin, vimentin, estrogen and progesterone receptors.
Sarcomas are distinguished from the most malignant looking fibroepithelial polyps as sarcomas have identifiable tumor margins, cellular homogeneous distribution and absence of stellate and multinucleate stromal cells near the epithelial-stromal interface. Microscopic evaluation of the lesion in our case revealed polypoidal lesion lined by keratinised stratified squamous epithelium. Focally, it was ulcerated and replaced by fibrino-suppurative exudates. Sub-epithelial tissue showed dense collagenisation, mixed inflammation and ectatic blood vessels while deeper tissue was loose and showed scattered spindle and few stellate cells. Blood vessels of various calibre were also seen with few blood vessels thickened and dense lympho-plasmacytic infiltrate were seen along with perivascular inflammation suggestive of fibro-epithelial polyp of the vulva.
Absence of myxoid areas rules out possibility of aggressive vulval angiomyxomas.
As an adjunct to microscopic evaluation, imaging is also important in the diagnosis of fibroepithelial polyps. It evaluates blood supply and flow and determines the origin and extent of the tumor. Although CT and MRI may be used, they are not as cost effective or widely available as ultrasonography (USG).[3] Therefore, USG is the first line imaging tool. USG also is fast, helps in real time determination of capacity for exploration and ability to visualize the entire lesion in a single image. The other differential diagnosis is botryoid embryonal rhabdomyosarcoma but these tumors are found in young pre-pubertal girls. There is absence of characteristic hypercellular subepithelial layer and there are specific immunohistochemical markers for skeletal muscle differentiation. These polyps can recur rarely, especially if they are not completely removed.[4] Literature reports a case of growth of a giant cell fibroblastoma arising at the site of a previously excised stromal polyp.[5] Due to this all patients with diagnosed fibroepithelial polyps should be followed up regularly. Fibro epithelial polyps though benign, most often resemble serious and malignant growths, as a result, histopathological evaluation is essential to rule out malignancy.

Acknowledgments

We thank Dr. Mona Agnihotri, Assistant Professor in Histopathology, Seth GS Medical College and K E M Hospital for her assistance in understanding the histopathology of the fibroepithelial stromal polyp. We thank Dr. Ashwini Desai (Third year obstetric and gynecology resident) for the intra-operative photos of our case.

References

1. Nucci MR. Mesenchymal Lesions. In Nucci MR, Olivia E. editors. Gynecologic Pathology: A Volume in the series Foundations in Diagnostic Pathology. 1st ed. China: Elsevier Churchill Livingstone 2009;31–32.
2. Bozgeyik Z, Kocakoc E, Koc M, Ferda Dagli A. Giant fibroepithelial stromal polyp of the vulva: extended field-of-view ultrasound and computed tomographic findings. Ultrasound Obstet Gynecol. 2007;30(5):791-2.
3. Orosz Z, Lehoczky O, Szoke J, Pulay T. Recurrent giant fibroepithelial stromal polyp of the vulva associated with congenital lymphedema. Gynecol Oncol. 2005;98(1):168-71.
4. Ostör AG, Fortune DW, Riley CB. Fibroepithelial polyps with atypical stromal cells (pseudosarcoma botryoides) of vulva and vagina. A report of 13 cases. Int J Gynecol Pathol. 1988;7(4):351-60.
5. Han X, Shen T, Rojas-Espaillat LA, Hernandez E. Giant cell fibroblastoma of the vulva at the site of a previous fibroepithelial stromal polyp: a case report. J Low Genit Tract Dis. 2007;11(2):112-7.

Citation

Srinivasan N, S Panchbudhe S. Fibro-Epithelial Polyp Of The Vulva. JPGO 2019. Volume 6 No.2. Available from: https://www.jpgo.org/2019/01/fibro-epithelial-polyp-of-vulva.html

Ovarian Stimulation And Preeclampsia- Dual Causality Association with Subcapsular Hematoma?

Author Information

Bharti S*, Malani K*, Yadav K*, Samant P**.
(* Junior Resident, ** Academic Professor, Department of Obstetrics and Gynecology, Seth G S Medical College and K E M Hospital, Mumbai, India.)

Abstract

Subcapsular liver hematoma associated with severe preeclampsia is extremely rare but potentially life threatening complication of pregnancy. It is almost exclusively associated with severe preeclampsia or with HELLP (hemolysis, elevated liver enzymes, and low platelet count) syndrome. We present a case of subcapsular liver hematoma with severe preeclampsia in a 28 year old multigravida with bad obstetric history who had an in-vitro fertilization (IVF) conception. We support the hypothesis that there might be a link between ovarian hyperstimulation and liver dysfunction.

Introduction

Subcapsular hematoma of liver has been reported in less than 2 % of pregnancies complicated by severe preeclampsia and HELLP syndrome but is associated with significant maternal and perinatal morbidity and mortality.[1] It may present as right upper quadrant, epigastric or shoulder pain, abdominal distention, nausea and vomiting. It can lead to life threatening complications such as disseminated intravascular coagulation, acute liver failure, acute renal failure and hemorrhage due to hepatic rupture. Conservative management of subcapsular hematoma is safe in hemodynamically stable patient. In case of rupture of the Glisson’s capsule surgical intervention may be required. It is also proposed that vascular compromise of liver parenchyma caused by ovarian stimulation numerous times may be compounded by HELLP syndrome and may result in subcapsular hematoma of the liver.[2]

Case Report

A 28 year old multigravida with past history of two ectopic conceptions and one abortion, with 28 weeks IVF conception was referred from a peripheral hospital with severe preeclampsia and complaints of right hypochondriac pain for 2 days. She had conceived in her first assisted conception cycle. Cycle had been stimulated with HMG, and a single embryo was transferred. Her ultrasonography (USG) done in private hospital showed a subdiaphragmatic hematoma in addition to a singleton live pregnancy of 28 weeks and 2 days. She had been detected to have hypertension 5 days earlier and was started on tablet labetalol 100 mg thrice a day. On admission, she was pale, and had bilateral pedal edema. Respiratory and cardiovascular examination was unremarkable. Her B.P was 150/94 mm of Hg with 2+ grade of proteinuria. Hemoglobin was 7.3 g/dl, white blood cell count was 13,400/cmm, Platelets were1.44 Lac/cmm and INR was 1.1. Liver function was deranged. Serum total bilirubin was 2.1 mg/dl, SGOT was 356.78 IU/l and SGPT was 652.14 IU/l. Coagulation profile was normal, viral markers were negative. USG done in our institute was suggestive of increasing size of the right sub diaphragmatic collection from 250 cc to 450 cc. MRI (magnetic resonance imaging) was done. It was suggestive of a large unruptured subcapsular hematoma of 6.5x6x5.5 cm in size.

Figure 1.  MRI image of subcapsular hematoma shown by Black arrows.

She was administered two units of packed red cells. Steroid coverage was given for enhancing lung maturity. On the 2nd day, SGOT was 1500 IU/l, SGPT was 1 000 IU/l and INR was 0.95. Gastroenterology surgeons recommended conservative management for subcapsular hematoma and monitoring of liver functions and coagulation profile. Labor was induced after informed high-risk consent and confirming adequate blood and blood product availability
Pre-induction ripening of cervix was done by Foley’s catheter. Due to inadequate ripening, vaginal Misoprostol was used for induction. Injection MgSO4 was started by Zuspan regimen in view of bilateral deep tendon reflexes being brisk. She delivered a male baby of 920 grams vaginally with APGAR score of 4/10 at 5 minutes. Postpartum USG was done to quantify the volume of hematoma which was 650 cc.
Liver function tests (LFT’s) done on postpartum day 3 showed marginal improvement with SGOT 538 IU/l, SGPT 634 IU/l, and INR 0.96. On day 4 postpartum, her SGOT was 131 IU/l and SGPT was 325 IU/l. Subcapsular hematoma was managed conservatively. Serial USG on postpartum day 8 was suggestive of decrease in size of hematoma from 650 to 400 cc and at the end of 3 weeks postpartum organized hematoma with septae was noted. She was discharged in a stable condition on antihypertensive medication. Baby expired on day 10 of life due to intraventricular hemorrhage with severe perinatal asphyxia. She was advised to consider adoption, as spontaneous conception was not possible due to previous bilateral salpingectomy.

Discussion

Subcapsular liver hematoma is accumulation of blood between the capsule of Glisson and the liver parenchyma. It was first described in association with pregnancy by Abercombie in 1844.[3] Rosen and colleagues have quoted a speculation that the initial event in the pathogenesis of subcapsular hematoma is fibrin deposition in the liver sinusoids that is followed by thrombosis, and hepatic hemorrhage that then dissects through the capsule. [4] Histopathological examination of the livers’ with subcapsular hematomas have shown hemorrhage and necrosis in hepatic parenchyma, periportal necrosis and microaneurysms. [3] Subcapsular liver hematoma is mostly associated with preeclampsia/ eclampsia and HELLP syndrome, presenting in second or third trimester with 30 % occurring postpartum within 48 hours of delivery. Some case reports and reviews exist in which upto 14 % patients with liver hematoma had no clear diagnosis of preeclampsia or HELLP syndrome.[5] In the investigations, severe anemia, markedly elevated transaminase enzymes, thrombocytopenia, and abnormal coagulation profiles are noted.  The hematoma can be diagnosed by USG, CT scan, and/or MRI. It requires prompt delivery.[6]
Giugliano and colleagues have described a case of near fatal liver dysfunction with ruptured liver hematoma in an IVF pregnancy complicated by hypertension.[2] They propose that multiple cycles of controlled ovarian stimulation or resultant hyperstimulation in IVF pregnancy may cause silent liver dysfunction due to inflammatory mediators and it may be worsened by HELLP syndrome due to preeclampsia. Different mediators like renin-angiotensin or interleukin 6 are found to increase in ovarian hyperstimulated cycles. These mediators are likely to cause microvascular thrombosis leading to liver parenchymal ischemia and thus adversely affecting liver function.[7] Hepatic injury due to HELLP may be due to endothelial dysfunction. All these together can cause the patient to present with DIC, hypervolemia, liver ischemia, and subcapsular hematoma.[8] In a large retrospective cohort study of 1,357 pregnancies conceived by assisted reproductive technology, preeclampsia has been found to be more common.[9]
The index pregnancy was the first IVF conception of our patient. She presented with signs and symptoms of preeclampsia, markedly elevated serum aminotransferase, raised bilirubin, and low hemoglobin. MRI confirmed the diagnosis of a large subcapsular hematoma of the liver. Though we could not establish occurrence of ovarian hyperstimulation in the early pregnancy in our case; it is an interesting hypothesis for occurrence of subcapsular hematoma at a later gestation with preeclampsia. The incidence of recurrent hepatic capsular rupture is quoted to be less than 2%.[10]
Management in hemodynamically stable patient is essentially conservative and includes intensive fluid replacement, blood and fresh frozen plasma transfusion, monitoring of liver function test, serial scan of hematoma by USG or CT scan until resolution of liver hematoma. Decision to terminate pregnancy vaginally should be considered and cesarean delivery should be reserved for obstetric indications only. Hemodynamic instability requires exploration. Bleeding surfaces are packed with collagen sponges and perihepatic space is drained. In some cases, percutaneous hepatic artery embolization could be an alternative procedure for control of hemorrhage. Liver transplantation may be needed in cases of acute liver failure.[6] In our patient, hemodynamic status was not compromised. We followed non-invasive conservative management. Since high estrogen is a potential cause for liver dysfunction, non hormonal contraception would probably be the safest advise for such patients.

Conclusion

Early diagnosis and intervention decreases morbidity and mortality in a case of severe pre-eclampsia with subcapsular hepatic hematoma and deranged liver function tests. More studies are required to explore the possible synergistic link between multiple consecutive ovarian stimulation cycles and HELLP syndrome in preeclampsia affecting liver histology and function.

References

1. Manas KJ, Welsh JD, Rankin RA, Miller DD. Hepatic hemorrhage without rupture in preeclampsia. New England Journal of Medicine1985;312(7):424–426.
2. Giugliano E, Cagnazzo E, Pansini G , Vesce F, Marci R. Ovarian stimulation and liver dysfunction: Is a clinical relationship possible? A case of hepatic failure after repeated cycles of ovarian stimulation. Clin Exp Reprod Med 2013;40(1):38-41
3. Karateke A, Silfeler D, Karateke F, Kurt R, Guler A, and Kartal I. “HELLP Syndrome Complicated by Subcapsular Hematoma of Liver: A Case Report and Review of the Literature,” Case Reports in Obstetrics and Gynecology. 2014;Article ID 585672: 3 pages.
4. Rosen SA,  Merchant SH,  VanderJagt TJ,  Crookston KP.  Spontaneous Subcapsular Liver Hematoma Associated With Pregnancy. Archives of Pathology & Laboratory Medicine.2003;127(12):1639-1640.
5. Schwartz ML, Lien JM. Spontaneous liver hematoma in pregnancy not clearly associated with preeclampsia: a case presentation and literature review. Am J Obstet Gynecol. 1997;176(6):1328-32.
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7. Borgaonkar MR, Marshall JK. Marked elevation of serum transaminases may be associated with ovarian hyperstimulation syndrome. Am J Gastroenterol. 1999;94(11):3373.
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Citation

Bharti S, Malani K, Yadav K, Samant P. Ovarian Stimulation And Preeclampsia- Dual Causality Association with Subcapsular Hematoma? JPGO 2019. Volume 6 No.2. Available from:  https://www.jpgo.org/2019/01/ovarian-stimulation-and-preeclampsia.html

Cystadenofibroma Of Ovary

Author Information

Ruhil MS*, Dwivedi JS**, Hatkar PA***, Kothari K****.
(* Junior Resident, ** Assistant Professor, *** Associate Professor , Department of Obstetrics and Gynecology, **** Assistant professor, Department of Pathology, Seth G S Medical College and K E M Hospital, Mumbai, India.)

Abstract 

Ovarian cystadenofibroma is a rare benign ovarian tumor. It contains both epithelial and fibrous stromal components. The cystadenofibroma on imaging has often a complex appearance. On imaging, it is visualized as cystic to solid masses.  This may also resemble a malignant tumor. We describe here the case of a 48-year-old woman with an ovarian cystic mass that was diagnosed as a benign cystadenofibroma of the ovary on histopathology report. The patient underwent successful total abdominal hysterectomy with bilateral salpingoophorectomy.

Introduction

Surface epithelial-stromal tumors are the most common tumors of the ovary. Majority of these occur in women between fourth to sixth decades of life. Benign ovarian serous tumors account for approximately 16% of all ovarian epithelial neoplasm. Approximately 30-50% of them are bilateral. The fibroblastic stromal component is unduly prominent in some serous tumors and appears grossly as white, solid, nodular foci in an otherwise typical cystic neoplasm. These can be classified into benign; adenofibroma and cystadenofibroma, borderline, and malignant adenofibrocarcinoma and cystadenofibrocarcinoma. Cystadenofibromas are one of the rare ovarian benign tumors. These constitute 1.7% of all benign ovarian tumors.[1] These are usually seen in women of age between 15-65 years.[2] They can composed of serous, mucinous, endometrioid, and transitional epithelium. They are bilateral in 15% of the cases.[1]

Case Report

A 48 year old woman, para 3 living 3, came to gynecology outpatient clinic with a chief complaint of right sided pain in the abdomen for 7-8 months. The pain got aggravated in the last two months. The pain was associated with abnormal uterine bleeding and was cyclical in nature. Her menstrual cycle length was 30 days with last menstrual period four weeks ago, increased and it was associated with passage of clots, and she had secondary congestive dysmenorrhea. She had no significant family history. She was examined in the outpatient gynecology clinic. On general examination, her general condition was fair, temperature was 37ºC, pulse was 78 beats/minute and the blood pressure was 160/90 mm of Hg. Per abdomen examination revealed a palpable mass of around 24 weeks in size which was firm in consistency. Vaginal examination showed a healthy cervix, vagina, and a bulky, minimally tender uterus and a soft, mobile right adnexal mass of approximately 20 cm in diameter. After admission, medicine faculty was consulted for evaluation and management of hypertension. She was started on tablet Telmisartan 40 mg with Hydrochlorothiazide 12.5 mg OD and salt restricted diet. Ophthalmology evaluation was done and there was no evidence of hypertensive retinopathy. Blood investigations were sent. Her hemoglobin was 11gg/dl, leucocytes 8000/cmm, platelets 2.0 lac/cmm, TSH was 1.1 mIU/L, fasting and postprandial blood sugar levels were within normal limits. Urine routine and microscopy was within normal limits. Tumor markers were done. Serum LDH was 290 U/L, CEA levels were 4.7 ng/ml, βHCG was < 0.13 mIU/ml, CA 125 was 39.6 units/ml. Pelvic ultrasound (USG) showed a 19x19x11 cm large adnexal mass on the right side, right ovary could not be separately identified, suggesting a possible ovarian origin. The mass had a cystic component with thick septae and also a solid component without definite evidence of posterior acoustic shadowing to suggest calcification. Minimal intralesional vascularity was identified. There was no free or loculated fluid in the pelvis. Contrast enhanced computerized tomography (CT) was suggestive of 19x12x20 cm complex cystic mass lesion arising from the right adnexa. The lesion was predominantly anterior to the urinary bladder with two loculi within; one of the loculi had hyperdense content within. No calcification, ascites or lymphadenopathy was seen. Features were suggestive of a possible ovarian chocolate cyst. She underwent an exploratory laparotomy. The uterus, fallopian tubes and right ovary were normal. was normal. There was a 18x15 cm ovarian cyst on the left side. There were no adhesions. A total abdominal hysterectomy with bilateral salpingo-oophorectomy was performed. Uterus, cervix, bilateral fallopian tube and ovaries were sent for histopathological examination. On histopathological examination, endometrium was proliferative, and myometrium was adenomyotic.  Cervical epithelium showed immature squamous metaplasic changes. Bilateral fallopian tubes were unremarkable. Left ovarian cyst showed ovarian cyst lined by single layered cuboidal epithelium. Many polypoidal structures with thick fibrous cores lined by single layer of cuboidal epithelium were seen. No complex papillary projections, nuclear atypical, or invasion was seen. Findings were suggestive of left ovarian serous cystadenofibroma.

Figure 1. Histopathology image of ovarian serous cystadenofibroma. Black arrow shows papillae and yellow arrow shows stroma.

Discussion

Ovarian cystadenofibroma is a relatively rare benign tumor as compared to ovarian cystadenoma. The actual incidence of ovarian cystadenofibromas is not known.   Primary ovarian cystadenofibromas are usually encountered in women of age group between 15 and 65 years. These tumors account for approximately 1.7 % of all benign ovarian neoplasms.[2] Ovarian cystadenofibroma is a type of surface epithelial tumor. All   the subtypes of these tumors exhibit a fibrous stroma in variable amounts. When the   stroma occupies a greater proportion than the cystic portion, the suffix “fibroma” is added, as in “serous adenofibroma.” The presence of more than 1 cyst over 1 cm in diameter warrants the use of the prefix “cyst” as in “cystadenofibroma.” These tumors are   classified according to the epithelial cell types.
Various types of these tumors are; serous, endometrioid, mucinous, clear cell, and   mixed categories. These tumors are classified as benign, borderline, or malignant, according to the degree of epithelial proliferation and its relation to the stromal components. Most of the reported ovarian cystadenofibromas were benign.[1] There is a strong suspicion of ovarian malignancy when a patient presents with an ovarian cystic mass with solid components. Ovarian cystadenofibromas are notable from   imaging modalities because they often have solid components and thus mimic malignant neoplasm. Some investigators have reported that this type of tumor can be diagnosed as malignant based on imaging alone.[3] Moreover, these tumors may have the gross appearance of a malignant tumor at the time of surgery. A frozen-section diagnosis is helpful in many of these cases because a correct diagnosis of cystadenofibroma in the intraoperative period can save the patient from unnecessary extensive surgery. Therefore, recognition of this tumor by surgeons is of utmost importance. However in this case as the CT scan report was suggestive of chocolate cyst, frozen section wasn’t done. When imaged by ultrasonography or CT, ovarian cystadenofibroma may appear as a complex solid and cystic mass and difficult to differentiate from malignancy. Thus, MRI may be an essential modality for diagnosing this tumor, especially when the characteristic “black sponge” appearance is observed on MRI. The “black sponge” appearance reflects dense fibrous stromal proliferation with scattered small cystic glandular structures on the T2-weighted images. It was considered to be characteristic of the solid components of cystadenofibromas.[4] The sensitivity and specificity of MRI in differentiating benign and malignant tumors are 93.10% and 83.33% respectively.[5] Outwater et al. reported that the dense fibrous tissue of the solid component of cystadenofibroma showed very low signal intensity on T2-weighted images, which are similar to a skeletal muscle.[6] Cystadenofibrocarcinoma, the malignant form, has a solid portion with strong enhancement and higher signal intensity compared to benign cystadenofibromas. Usually, the risk of malignancy increases with an increasing solid tissue portion and thicker septa and contrast enhancement usually facilitates differentiation between benign and malignant lesions. In this case MRI wasn’t done as patient was not affording the same. Ovarian cystadenofibroma being a rare tumor, its occurrence may be difficult to detect. Therefore, ovarian cystadenofibroma is one of the tumors subject to differential diagnosis in which preoperative diagnosis by MRI is essential. In addition, frozen sections may be helpful in the intraoperative assessment of ovarian masses to rule out malignancy and provide guidance for appropriate surgical management. In our case since patient already had menorrhagia with suspected adenomyosis and a large cystic solid ovarian neoplasm, the decision for a hysterectomy with bilateral salpingo oophorectomy was taken preoperatively. Hence we did not consider doing a frozen section on the neoplasm though it would have been better as further staging laparotomy if needed could have been performed.


References

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Citation

Ruhil MS, Dwivedi JS, Hatkar PA, Kothari K. Cystadenofibroma Of Ovary. JPGO 2019. Volume 6 No.2. Available from: https://www.jpgo.org/2019/01/cystadenofibroma-of-ovary.html