Volume 2 Issue 10, October 2015

Editorial
Parulekar SV

Limb Body Wall Complex.
Dhanawat J, Parulekar SV.

Giant Ovarian Cyst.
Wagh P, Samant PY, Parulekar SV

Successful Outcome Of Pregnancy In A Patient Of Aortoarteritis.
Mehta V, Maurya N, Gupta AS.

Successful Expectant Management Of Retained Placenta In A Patient With Multiple Fibroids
Saxena N, Tiwari N, Amin K, Chauhan AR.

 Invasive Gestational Trophoblastic Neoplasm: A Rare Interesting Case Managed Successfully With Chemotherapy.
Madhu kumari, Chavan NN, Changede P, Gupta S.

Anterior Abdominal Wall Adhesions Mimicking Intramyometrial Collection
Pawde A, Chauhan AR, Mayadeo NM.

 Atypical Prsentation Of A Cervical Fibroid In Pregnancy. 
Joshi A, Mhaske N, Kharat D, Fonseca MN.

 A Twisted Ovarian Mucinous Cystadenoma Complicating Pregnancy: Case Report
Verma K

 Post PICC Line Thrombosis In a Severely Anemic Post Abortal Patient.
Desai DV, Karve N, Gupta AS.

 Announcement

Editorial

Parulekar SV

Ovarian cancer is often asymptomatic until it is quite advanced. Thus it is more dangerous than other gynecologic cancers because it is diagnosed at a more advanced stage than others. There are no good and easy screening methods for its early detection. If its prevention is at all possible, it would be ideal from the women's point of view, the lifetime risk of developing ovarian cancer being 1-2%. Recently there has been quite a bit of interest in the role of salpingectomy in reducing the risk of development of ovarian cancers. It has been shown that both bilateral tubal ligation and bilateral salpingectomy before the age of 35 years reduces the risk of development of ovarian cancer by up to 50%. The benefit is greater with salpingectomy than with tubal ligation. The cancers prevented are mainly the endometrioid cancer. Significant benefit is also seen with serous epithelial cancers. There is no protection from the development of borderline cancers. The mechanism underlying this protection is not understood very clearly.


The process of ovulation causes surface injury to the ovary. If the process of healing of that injury is not proper, there is a possibility of mutations which could lead to the development of cancer. Gonadotropins could be involved in the development of the malignant change. Inflammatory agents and infection that reach the ovary by ascending route from the lower genital tract through the uterus and the fallopian tubes could also be causative agents. The process of tubal interruption would prevent such agents and endometrial cells reaching the ovary and protect it from development of cancer. Interruption of the tube could be associated with reduced blood flow from anastomosis between uterine and ovarian arteries to the ovary, reducing the amount of gonadotropins reaching the ovary and thus reduce the risk of development of ovarian cancer. It is also likely that the cancer, especially the serous type, originates in the adjacent fallopian tube and spreads to the ovary. Removal of the fallopian tube would afford protection from ovarian cancer, but not simple ligation. For women who desire permanent contraception, salpingectomy rather than tubal ligation should be offered. But that may be associated with higher morbidity and deterioration of ovarian function due to reduced blood supply. If a woman has a high risk of ovarian cancer, prophylactic removal of ovaries after completion of the childbearing function should be combined with salpingectomy, if the woman so desires.

Limb Body Wall Complex

Author Information

Dhanawat J*, Parulekar SV**.
(* First Year Resident, ** Professor and Head, Department of Obstetrics and Gynecology, Seth G S Medical College & KEM Hospital, Mumbai, India.)

Abstract

Limb body wall complex (LBWC), also called as body stalk complex or cyllosomas is a rare polymalformative fetal malformation syndrome of uncertain etiology, which results in malformations of the head, heart, lung, diaphragm, kidney, gonads or limbs. We present such a case who presented at 31 weeks 3 days of gestation with absent anterior abdominal wall and absent left lower limb.

Introduction

The limb body wall complex is probably heterogenous. Possible mechanisms of its development include disruption of germ disc, vascular system, and amnion. It mainly includes abdominal wall defect and a wide variety of limb anomalies. It can be associated with other visceral anomalies. Two phenotypes have been described, placento-cranial type showing craniofacial defects and amniotic bands and placento-abdominal adhesion type with urogenital anomalies, atresia and abdominal placental attachment with persistence of the extraembryonic coelom.[1] In LBWC, there’s no sex predilection, or known recurrence risk. Karyotype study normal.[2] The case presented here was a primigravida with a fetus of 31 weeks 3 days of gestation with absent anterior abdominal wall and absent left lower limb.

Case Report

A 22 years old female, married since 2 years, primigravida with  31 weeks 3 days of amenorrhea,  presented with failure of abdominal enlargement as expected. She did not have any past medical or surgical illness. On examination, per abdomen, the uterine size was of 24 weeks. External ballottement positive. Fetal heart sounds were absent. On per vaginal examination, the cervix was closed and uneffaced. Abdominal ultrasonography showed intrauterine fetal demise. There was a single lower limb and exomphalos major. Tablet misoprostol was administered in a dose of 800 micrograms vaginally. She delivered a fetus in breech presentation. The fetus had exomphalos major, absent left lower limb, external male genitalia, and well formed eyes and ears. It weighed 556 g. Her past ultrasonographic (USG) scan showed a fetus in breech presentation with normal cardiac activity, AFI of 5, and no obvious congenital anomalies. A subsequent USG scan performed before presenting to us showed absent cardiac activity and reversal of flow in uterine artery. Placental biopsy revealed infarct with single umbilical artery and single umbilical vein.


Figure 1. Fetal appearance: there is a single (right) lower limb and ruptured exomphalos major.


Figure 2. Babygram showing single (right) lower limb.

Discussion

Congenital malformations of the ventral abdominal wall occurs in many forms, ranging from exomphalous to gastroschisis to more complexes such as Pentalogy of Cantrell and LBWC. LBWC includes an abdominal wall defect and is associated with various visceral and limb anomalies. The diagnostic criteria most commonly quoted are those given by Van allen et al in 1987.[3] The presence of two of the following three is required: exencephaly/encephalocele with facial defects, thoraco and/or abdominoschisis, and limb defects. LBWC results from defect in early embryonic folding process. Recurrence in next pregnancy is low. There is no sex predilection.[1] Familial recurrence of one sibling with LBWC  and another with amniotic band syndrome[4], and an occurrence of two siblings born with LBWC[5] have been reported suggesting genetic origin. LBWC should be differentiated from other entities because treatment will vary accordingly. Differential diagnosis includes abdominal wall defects such as gastroschisis, omphalocele , ectopia cordis, amniotic band syndrome, cloacal dystrophy and urachal cyst. They can be differentiated on the basis of site. Ectopia cordis is typically located at the anterior aspect of thorax, omphalocele and gastroschisis are localized to the umbilical and paraumbilical areas. Cloacal dystrophy, urachal cyst involve the lower abdominal wall. Exomphalous is usually seen in trisomies 13, 18, and 21. Pathogenic theories that have been proposed include early amnion rupture theory (exogenic theory)[6], vascular disruption theory (endogenous theory,[3] and embryonic dysgenesis or germ disc defect.[7] Certain drugs, tobacco, alcohol and some other unkown environmental factors are few teratogenic causes leading to LBWC.[5] Cocaine teratogenecity is also associated with LBWC (Viscarello et al 1992).[8] We could not identify any cause in the case presented. Early detection of the condition (prior to 20 completed weeks of gestation) can help the patient make a choice of medical termination of pregnancy.

References
  1. Russo R, D'Armiento M, Angrisani P, Vecchione R. Limb body wall complex: a critical review and a nosological proposal. Am J Med Genet 1993;47:893-900.
  2. S. Saritha, Gouri, Sumangala. Limb body wall complex or body stalk complex or cyllosomas: a case report.  Int J Res Med Sci. 2013 May;1(2):132-137.
  3. Van Allen MI, Curry C, Gallagher L. Limb body wall complex: I. Pathogenesis. Am J Med Genet 1987;28:529-48.
  4. Levy R, Lacombe D, Rougier Y, Camus E: Limb body wall complex and amniotic band sequence in sibs.  Am J Med Genet 2007;143A:2682-2687. 
  5. Luehr B, Lipsett J, Quinlivan JA: Limb-body wall complex: A case series.  J Matern Fetal Neonatal Med 2002, 12:132-137.
  6. Torpin R. Amniochorionic mesoblastic fibrous strings and amnionic bands: Associated constricting fetal malformations or fetal death. Am J Obstet Gynecol. 1965;91:65–75. 
  7. Streeter GL. Focal deficiency in fetal tissues and their relation to intrauterine amputation. Conntrib Embryol 1930;33:41-49.
  8. Viscarello RR, Ferguson DD, Nores J, Hobbins JC. Limb-body wall complex associated with cocaine abuse: further evidence of cocaine's teratogenicity. Obstet Gynecol 1992;80(3 Pt 2):523-6.
Citation

Dhanawat J, Parulekar SV. Limb Body Wall Complex. JPGO 2015. Volume 2 Number 10. Available from: http://www.jpgo.org/2015/10/giant-ovarian-cyst.html

Giant Ovarian Cyst

Image
Author information

Wagh P*, Samant PY**, Parulekar SV***
(* Senior resident, ** Additional Professor, *** Professor and Head, Department of obstetrics and gynecology, Seth G S Medical college and KEM Hospital, Mumbai, India.)

Abstract

Giant ovarian tumors may distort the anatomy and pose operative difficulties. We present an image study  of a huge ovarian tumor which  had  grown into the broad ligament leaves and distorted the pelvic organ relationship.

Introduction

Usually large ovarian cysts, displace the uterus to opposite side or downwards . They may also cause displacement of ureters changing their course which increases the risk of ureteric injuries during surgeries. Such masses may also cause compression of the ureters causing hydroureters and hydronephrosis . Obstruction of pelvic lymphatics by such masses may cause edema of the lower limbs.
In our case,  the large cyst had grown into the leaves of broad ligament hence the uterus and fallopian tube were stretched upwards.  The relationship of pelvic organs was distorted which posed challenge during removal of the ovarian mass and hysterectomy. Herewith we share the intra-operative images and how challenges were overcome.

Case Report

A 45 years old, morbidly obese woman weighing 102 kg presented with abdominal distension for one and a half month, associated with dull abdominal pain and breathlessness on and off .
Her abdomen was non tender and over distended. A large cystic mass was felt arising from the pelvis, extending up to the epigastrium. On bimanual examination, the cervix was high up in anterior fornix, a cystic non-tender mass was felt above it,  while the uterine size could not be assessed due to obesity. Her Ca 125 levels were 26 u/ml. Ascitic fluid tapped in private hospital showed ADA -63 u/l and no abnormal cells. Computerised Tomography reported a  34x25x22 cm sized ovarian multiloculated cystic lesion suggestive of  mucinous or serous cystadenoma. There was no comment on organ relationship distortion.

On laparotomy, a large cyst of the right ovary seen occupying most of the abdominal cavity, it had grown into the broad ligament. A normal sized uterus was seen over the right anterior broad ligament at a level of 2 inches below the level of umbilicus. The right fallopian tube was stretched over the anterior and superior surface of the mass, the fimbriae fanned out over a distance of 3 cm. The left ovary and fallopian tube were normal. The cyst: approximately 35x40x35 cm, smooth surface, large cystic component and nearly 5x 8x 8 cm solid component at the base on right side.


Figure 1: Arrow showing uterus stretched over the anterior wall of the cyst.


Figure 2: Black arrows showing fimbrial end of right fallopian tube, yellow arrow showing the uterus.


Figure 3: White arrow showing left ovary, yellow arrow showing the left fallopian tube, black arrow showing uterus.

The right broad ligament was opened by division of the right round ligament. The leaves of the broad ligament were cut and dissected off the surface of the cyst, which was then removed along with the right fallopian tube. The cyst contained 14.5 L of fluid, which was serous in most of the part, and mucinous in one loculus. Total abdominal hysterectomy and left salpingo-oophorectomy were performed. Both the ureters were found to be normal in their position and size. Frozen section of the cyst showed a cystic mucinous benign neoplasm.


Figure 4. Inside of cut section of the cyst

Discussion

A large ovarian cyst usually expands the fallopian tube over its surface.[1,2,3] It usually displaces the uterus to the opposite side and either downwards or upwards.[4] What was most unusual in the case presented was the location of the uterus on the anterior surface of the ovarian tumor, 5 cm below the umbilicus. Another unusual feature was the fimbrial end of the fallopian tube stretched over a length of 3 cm. Careful identification of these structures as well as other structures like the ureters and the infundibulopelvic ligament is important in successful surgical management of the case and preventing injury to the ureter.

Acknowledgment

We thank Dr Rashmi Prasad for taking the operative pictures.

References
  1. Menahem S, Shvartzman P. Giant ovarian cyst mimicking ascites. J Fam Pract 1994;39:479–481.
  2. Farinetti A, Butazzi A, Tazzioli G, Saviano L, Saviano M. Giant ovarian cyst. A case weighing 23 kg. Literature review. Minerva Chir 2003;58:261–265. 
  3. Hart WR. Mucinous tumors of the ovary: a review. Int J Gynecol Pathol 2005;24:4–25.
  4. Zanini P, Cavalca A, Benatti E, Drei B. Benign giant ovarian cystadenoma. Description of a clinical case. Minerva Ginecol 1996;48:215–219.
Citation

Wagh P, Samant PY, Parulekar SV. Giant Ovarian Cyst. JPGO 2015. Volume 2 Number 10. Available from: http://www.jpgo.org/2015/10/giant-ovarian-cyst.html

Successful Outcome Of Pregnancy In A Patient Of Aortoarteritis

Author Information

Mehta V*, Maurya N**, Gupta AS***.
(* Second Year Resident, ** Senior resident, *** Professor. Department of Obstetrics & Gynecology, Seth G.S.Medical College & KEM Hospital, Mumbai, India.)

Abstract

Aortoarteritis synonymous with Takayasu`s arteritis is also known as ‘Pulseless disease or Aortic arch syndrome’. It is a rare disease causing inflammation, scarring and narrowing of large vessels with formation of aneurysms. We are reporting a patient of aortoarteritis in pregnancy who presented to us at 16 weeks of gestational age with severe hypertension (210/120 mm Hg). Considering her medical condition she was planned for Medical Termination of Pregnancy (MTP). However, second trimester MTP was not performed as she was not fit to undertake that procedure. Consequently her pregnancy continued. A successful outcome was achieved with optimal management of high blood pressure and no change in the disease status.

Introduction

Aortoarteritis is chronic inflammation of large vessels, specially aorta and its branches. It was first described by a Japanese ophthalmologist Mikito Takayasu in 1908 by reporting association between absences of upper extremity pulse with formation of retinal arteriovenous anastomosis.[1] Aortoarteritis is a rare disease with higher prevalence in Asian and Japanese population. It commonly involves aortic arch in Japanese population whereas in Asian population it commonly involves thoracic or abdominal part of aorta. This disease is common in younger people with higher prevalence in second and third decade of life. So it is not uncommon to encounter it during pregnancy. It is a common cause of severe hypertension in pregnancy. Surgical procedures for stenotic lesions reduce the complications of pregnancy and improves fetal outcome. It requires a team approach of cardiologist, rheumatologist and an obstetrician.

Case Report

A 32 year old, married, G2P1L1 with previous lower segment cesarean section (LSCS) previously performed for postdatism with fetal distress presented to us at 16 weeks of gestation with blood pressure of 230/110 mm of Hg in right arm and 140/100 mm of Hg in left arm. Her right radial, bilateral femoral and bilateral dorsalis pedis pulsations were almost imperceptible. The patient's condition was diagnosed a year back in 2014 as she had uncontrolled high blood pressure. It was diagnosed by coronary angiography, which showed 80% proximal stenosis of the left anterior descending branch and left circumflex branch (non dominant) of left coronary artery with normal right coronary artery (dominant) as seen in figure 1.


Figure 1 Coronary angiography.

She then underwent percutaneous transluminal coronary angioplasty. Post procedure within a month she again developed severe hypertension for which angiography was done. It showed significant luminal narrowing in descending thoracic aorta with diffuse luminal narrowing in bilateral common, external and internal iliac arteries, bilateral common femoral artery, superficial and popliteal arteries. ACR 1990 criteria were used to categorize this patient as a case of aortoarteritis. She was then referred to our institute for further management. On presentation she was 16 weeks pregnant. Her serological, biochemical profile were normal. CRP was 10.6 ng/dl and ESR after one hour was 40. ANA, ds DNA, cANCA and pANCA were negative. She was referred to us for her obstetric condition and consideration for a second trimester MTP, but MTP was not performed as her hypertension was not adequately controlled within the stipulated time for a MTP. The pregnancy was continued and the patient was under close and regular vigilance of the physician and the obstetrician. She remained well controlled on daily polytherapy comprising of oral sustained release Nifidepine 30 mg 6 hourly, extended release of Metoprolol  50 mg twice a day, Prazosin 5 mg twice a day, and Hydrochlorthiazide 12.5 mg twice a day. Patient was not on corticosteroids as her hypertension was controlled and the disease was not in the active phase. However, at 36.5 gestational weeks her blood pressure recording was 240/100 mm Hg in right arm and 140/100 mm Hg in left arm. She had no proteinuria and no premonitary symptoms of pre-eclampsia. Serological and biochemical profile were also normal. She was admitted, induction of labor and VBAC was planned. Ophthalmic examination excluded any hypertensive retinopathy. Pre-induction mechanical cervical ripening with Foley’s balloon catheter was performed. Cervical ripening was followed by infusion of oxytocin (low dose oxytocin regimen) for induction of labor. Intra partum maternal monitoring was done clinically with the mercury sphygmomanometer and the fetal monitoring was done by intermittent auscultation, fetal doppler and electronic fetal monitoring. However induction of labor failed and late fetal heart rate deceleration's indicative of utero- placental insufficiency was seen on intra-partum electronic fetal monitoring.  An emergency LSCS was performed under controlled general anesthesia under close supervision by the cardiologist and the physician.  Despite the continuation of all her antenatal antihypertensive agents  her blood pressure after induction of anesthesia was dangerously high.  Parenteral labetalol in a single dose of 140 mg was administered immediately after induction of anaesthesia. Male, appropriate for gestational age (AGA) neonate of 2.326 Kg with Apgar score of 4/10, 8/10, 9/10 was delivered. The neonate was observed in neonatal intensive care unit and the patient was monitored in the anesthesia intensive care unit for 24 hours. The patient had an uneventful post-operative course and was discharged on 6th postoperative day. Her blood pressure remained at 150/80 mm Hg in right arm and 100/60 mm Hg in left arm on all four antihypertensive agents. She was advised to follow up with the cardiologist.

Discussion 

Aortoarteritis is found in both the genders. Female to male ratio is about 1.58:1 in India and in Japan.[2] Diagnosis is made by physical examination including difference in blood pressure (BP) in both arms of about 30 mm Hg, asymmetrical pulse, absent pulse and presence of a bruit (94%), bounding pulse as a result of poststenotic dilatation, hypertension (74%) resulting from renal vascular involvement, hypertensive retinopathy, claudication, carotodynia, visual loss, stroke, and heart failure (28%).[3] Causes of hypertension in aortoarteritis include renal artery stenosis, coarctation of aorta, and involvement of baroreceptor and loss of elasticity of aorta. Mainstay of treatment is glucocorticoids (Prednisolone). If the condition remains uncontrolled then Cyclophosphamide, Methotrexate and Azathioprine can also be used. Arteriography is commonly used in the diagnosis of this condition but because of need of contrast and use of radiation, it is not safe in pregnancy and renal disease patients. Magnetic resonance imaging is preferred in such conditions as it can detect the inflammation and diagnose the disease earlier, but it is not able to detect lesions in distal subclavian artery and common carotid artery.[4] High resolution doppler ultrasonography is useful in such condition but has a limitation of less visualization of aorta.[5] The course of the disease remains unaffected during pregnancy.[5,6,7] However, there are increased chances of developing preeclampsia, eclampsia, placental abruption, intrauterine fetal growth restricition and death.  Vaginal delivery is preferred at term.[8] Change in systolic blood pressure during the second stage of labor should be kept in mind and use of instrumental delivery should be considered to cut short the second stage of labor. Poor outcome of disease occurs mainly in severe hypertension with involvement of abdominal aorta. Wong etal showed that abdominal aorta involvement is directly related to fetal weight.[9] There are no reported direct maternal deaths in these cases. Almost 83.9% women deliver a healthy, mature newborn, an incidence of 8.2% of stillbirths and 19.7% of growth restricted neonates have been reported in literature.[10]  In our case in spite of involvement of the abdominal aorta utero-placental circulation was not compromised and an AGA neonate was born. BP should be monitored in all 4 limbs with central aortic pressure monitoring during intrapartum period to prevent hypertensive complications and convulsions. Cesarean section is done only for obstetric indications.

Conclusion

Perinatal outcome in pregnancy in aortoarteritis is satisfactory but it requires close monitoring of the mother and fetus. Pregnancy does not seem to induce the disease activity. Perinatal and maternal death can be avoided by appropriate and prompt management and with co-ordination of obstetrician, cardiologist and rheumatologist.

References
  1. Hom C. Pediatric Takayasu arteritis. July,2013. Available at http://emedicine.medscape.com/article/1007566-overview
  2. Sharma BK, Jain S. A possible role of sex distribution in determining distribution of lesions in Takayasu’s arteritis. Int J Cardiol 1998;66910) Suppl 1: S81-4
  3. Choudhari KR, Mayadeo N. Aortoarteritis in pregnancy. Bombay Hospital Journal. 2002 Jul;44(3):444-5
  4.  Yamada I, Numano F, Suzuki S. Takayasu arteritis, evaluation with MR imaging. Radiology 1993 Jul;188(1):89-94.
  5. Ishikawa K, Matsura S. Occlusive thormboaortopathy and pregnancy – clinical course and management of 33 patients and deliveries. Am J Cardiology 1982;50:1293–300.
  6. Matsumura A, Moriwaki R, Numano F. Pregnancy in Takayasu arteritis from the view of internal medicine. Heart Vessel Suppl. 1992;7:20–4.
  7. Seo P. Pregnancy and vasculitis. Rheum Dis Clin North Am. 2007;33:299–317.
  8. Papantioniou N, Katsoulis I, Papageorgiou I, Antsaklis A. Takayasu arteritis in pregnancy: safe management options in antenatal care. Case report. Fetal Diagn Ther. 2007;22:449-51.
  9. Wong VCW, Wang RYC, Tse TF. Pregnancy and Takayasu’s arteritis. Am J Med 1983;75:597-601.
  10. Hauenstein E, Frank H, Bauer JS, Schneider K.T.M.,Fischer T. Takayasu’s arteritis in pregnancy: review of literature and discussion. J Perinat Med 2010;38:55–62.
Citation

Mehta V, Maurya N, Gupta AS. Successful Outcome Of Pregnancy In A Patient Of Aortoarteritis. JPGO 2015. Volume 2 Number 10. Available from: http://www.jpgo.org/2015/10/successful-outcome-of-pregnancy-in.html

Successful Expectant Management Of Retained Placenta In A Patient With Multiple Fibroids

Author Information

 Saxena N*, Tiwari N**, Amin K***, Chauhan AR****.
(* Previous ** Third Year Resident, *** Assisstant Professor, **** Additional Professor. Department of Obstetrics and Gynaecology, Seth GS Medical College and KEM Hospital, Mumbai, India.)

 Abstract

 There is an underestimation of the prevalence of uterine fibroids in pregnancy, however the complications are primarily attributed to their presence. We report a case of a 33 year old G2P0A1 who went into spontaneous labour and expelled the fetus at 26 weeks of gestation, however the trapped placenta failed to deliver even after attempting its manual removal under general anaesthesia due to mechanical obstruction by large fibroids. Successful expectant management of the retained placenta was done using multi dose regimen of methotrexate and serial monitoring of β-HCG levels; patient subsequently underwent abdominal myomectomy.

 Introduction

 Most pregnancies are unaffected by the presence of fibroids and in a majority, small fibroids are incidentally seen intraoperatively during cesarean section. Complications such as pain, vaginal bleeding, placental abruption, preterm labor, intra uterine growth restriction and retained placenta are mainly attributed to large fibroids more than 5 cm in diameter, especially submucosal and intramural fibroids.[1] We observed that administration of methotrexate resulted in decreased uteroplacental blood flow and placental size, eventually resulting in disappearance of the entire placenta.[2]

 Case Report

 A 33 year old P1L0A1 was referred to our emergency department on day 2 of preterm vaginal delivery with postpartum hemorrhage and retained placenta. She had registered antenatally at 24 weeks of gestation at a peripheral hospital. Normal intrauterine pregnancy along with multiple uterine fibroids was diagnosed on routine antenatal ultrasonography. She went into spontaneous inevitable preterm labour at 26 weeks of gestation and delivered a male baby of 800 g. The baby died immediately after birth. A decision for manual removal of placenta was taken when the placenta failed to separate and deliver spontaneously 30 minutes after delivery. However, under general anesthesia, the placenta could not be removed due to mechanical obstruction from the fibroids. At this time, the patient had a mild postpartum hemorrhage, hence the vagina was packed and the patient was referred to our tertiary care centre for further management.
On admission, her general condition was fair, vital parameters were normal, there was no tachycardia or pallor. Abdominal examination revealed uterus corresponding to 22 weeks' size, well contracted. On speculum examination, the vaginal pack was removed; the cervical os was partially open, but there was no evidence of placental tissue seen through cervical os nor was there any active bleeding.
All routine blood investigations and coagulation profile were normal (hemoglobin was 13.1 g/dl, INR was 1.08), β-HCG was 3395 mIU/L, and liver and renal function tests were normal. Ultrasound of pelvis revealed a well-defined hyperechoic lesion in upper uterine segment suggestive of retained placenta, with a volume of approximately 200 ml, along with multiple uterine fibroids in the anterior and right lateral walls measuring 7.5 x 7 cm and 6.8 x 5.8 cm respectively. Magnetic resonance imaging (MRI) revealed multiple large intramural fibroids in antero-inferior and postero-lateral wall measuring approximately 10 x 8 x 8 cm and 7.5 x 5.3 x 5.7 cm respectively. Placental tissue of size 8.3 x 4 x 5 cm was seen attached to the posterior wall in the fundal area without any evidence of myometrial invasion.
In view of stable general condition and the above reports, a decision to manage the patient conservatively was taken. She received alternate day regimen of 3 doses of methotrexate (1 mg/kg body weight) and 3 doses of leucovorin (0.1 mg/kg body weight) and was discharged after 7 days. The patient was followed up regularly with serial measurements of serum β-HCG and placental volume, which fell rapidly initially and gradually disappeared till β-HCG levels fell to less than 2 mIU/L and no evidence of placenta seen on follow up ultrasonography (USG) after 6 months. However, 6 months postpartum, the uterus was approximately 22 weeks size, firm, and mobile. In view of these findings and her previous poor obstetric outcome, abdominal myomectomy was done. Three large fibroids were enucleated, the largest of those indenting the endometrial cavity which was opened during myomectomy. Further hospital course was uneventful and the patient was discharged.

Figure 1. Uterus with multiple (3) fibroids.

Figure 2. After enucleation of fibroids.

 Discussion

 Fibroids have been documented to be a common cause of retained placenta.[3] However, other independent risk factors for retained placenta include preterm delivery owing to a smaller placenta with a thin cord, history of retained placenta in the previous pregnancy and history of abortions in the past.. [4] It is associated with many complications like bleeding, infection and intrauterine adhesions, thereby increasing the overall rate of maternal morbidity and mortality in developing countries.[5] Diagnosis is made when the placenta fails to deliver after 30 minutes of delivery of baby. However USG and MRI are frequently used imaging modalities for confirmation. We report a combination of factors that caused placenta to be trapped in our case - inevitable preterm labor where after the fetus was spontaneously expelled, the cervix closed down promptly, extreme prematurity with thin cord and small placenta, and location of the fibroids which obstructed the tract. In fact if this patient had reached term, it is unlikely that she would have delivered vaginally due to the location of the fibroids and distortion of the cavity.

 There are a variety of treatment options available nowadays, however conservative line of management offers the potential to preserve future fertility in women of child bearing age.
Methotrexate has emerged with promising results to cure persistent retained placental tissue. It acts on the dividing trophoblastic cells of the placental tissue and also reduces the neovascularization thus resulting in reduction and resorption of the retained placental tissue over a course of time. However serial monitoring with serum β-HCG levels and periodic USG examination for placental volume are required during the treatment.

 Other treatment modalities include internal iliac artery ligation, uterine artery embolization and cesarean hysterectomy. Literature has also documented studies regarding the efficacy of carbetocin, intra umbilical administration of oxytocin and use of prostaglandins; however the data regarding the safety profile is limited.[6] However, abdominal myomectomy in such patients offers the potential for future fertility as fibroids were considered to the be the main culprit for their poor obstetric outcome.

References
  1. Ouyang DW, Economy KE. Obstetric complications of fibroids. Obstet Gynecol clin North Am. 2006 Mar; 33(1):153-69.
  2. Lin K et al. Methotrexate management for placenta accreta: a prospective study. Arch Gynecol Obstet . 2015 Jun; 291(6):1259-647.
  3. Lee D, Johnson J. Hysterotomy for retained placenta in a septate uterus: A case Report. Case Rep Obstet Gynecol 2012. 2012 594140.
  4. Deckers EA, Stamm CA, Naake VL, Dunn TS, McFee JG. Hysterotomy for retained placenta in a term angular pregnancy. A case report. J Reprod Med. 2000;45:153–5.
  5. S U Mbamara, Abc Daniyan, Ejenobo Osaro, I C Mbah. Myomectomy for retained placenta due to incarcerated fibroid mass. Ann Med Health Sci Res 2015 Mar-Apr; 5(2):148-51
  6. Amr K Elfayomy. Carbetocin versus intra-umbilical oxytocin in the management of retained placenta: A randomized clinical study.J Obstet Gynaecol Res 2015 Aug; 41(8):1207-13.
Citation

 Saxena N, Tiwari N, Amin K, Chauhan AR.Successful Expectant Management Of Retained Placenta In A Patient With Multiple Fibroids. JPGO 2015. Volume 2 Number 10. Available from: http://www.jpgo.org/2015/10/successful-expectant-management-of.html

Invasive Gestational Trophoblastic Neoplasm: A Rare Interesting Case Managed Successfully With Chemotherapy

Author Information

* Madhu kumari, ** Chavan NN, *** Changede P, **** Gupta S.
(* Speciality medical officer, ** Additional Professor, *** Assistant professor, **** Registrar, Department of Obstetrics & Gynaecology, LTMM College & General Hospital, Mumbai, India.)

Abstract

Gestational trophoblastic neoplasms (GTN) are rare tumors that contribute to less than 1% of all gynecological malignancies. Invasive mole is a type of GTN. This is a case of an invasive mole of the uterus, which developed following a molar pregnancy evacuation. It was diagnosed by persistent raised level of β-HCG, transvaginal ultrasonography and color Doppler study. The tumor was treated successfully with chemotherapy.

Introduction

Gestational trophoblastic disease (GTD) is group of disorders related to pregnancy arising from abnormal placental trophoblast cells. It is of two types: pre-malignant conditions and malignant gestational trophoblastic neoplasia. Pre-malignant conditions include partial and complete hydatidiform moles. Gestational trophoblastic neoplasia includes: invasive mole, choriocarcinoma (CC) and placental site trophoblastic tumor (PSTT).[1] Invasive moles are localized GTN.[2] Diagnosis is done by persistent raised β-HCG and imaging. The imaging of choice is ultrasonography and color Doppler study. MRI helps in diagnosis of the spread of tumor. With early diagnosis the cure rate is high with chemotherapy.[3]

Case Report

A 26 years old patient married since two years, gravida 1 abortion 1, came to our tertiary care hospital outpatient department with complaints of three month of amenorrhea, pain in abdomen and bleeding per vagina since five days. The patient was apparently alright six months back when she missed her period for which urine pregnancy test was done and was tested positive. Ultrasonography of pelvis was suggestive of hydatidiform mole. She underwent suction evacuation in Rajasthan for the same. β-HCG level was not done.
Two month later a repeat check curettage was done in a private hospital in view of  persistent bleeding with β-HCG level 5,000miu/ml and ultrasonography of pelvis suggestive of persistent hydatidiform mole.
The patient followed up with serial serum β-HCG level and ultrasonography. β-HCG showed gradual rise in level up to 14,000 miu/ml. Ultrasonography was suggestive of development of left adnexal mass with multiple cysts with increased vascularity. Ultrasonography showed gradual increase in size of adnexal mass up to 5 x 3 x 2 cm inseparable from uterus and left ovary.
The patient came to our tertiary care hospital OPD with above said reports. On her examination vital parameters were normal, per abdomen - soft, per speculum - minimal bleeding present, cervix and vagina healthy, per vaginum – uterus of normal size, anteverted, cystic swelling of 4 × 4 cm in left adnexa was felt and 2 × 3 cm small cystic mass was felt in right adnexa. Both the masses were non-tender and mobile. A gestational trophoblastic neoplasm was suspected. Tumor marker, ultrasonography with colour Doppler and MRI pelvis were done. All were suggestive of invasive mole with left adnexal involvement without distant metastasis. WHO prognostic score was suggestive of low risk. An opinion of a medical oncologist was taken, based on which she was started on chemotherapy with two cycles of injection methotrexate 1 mg/kg on alternate day with injection leucoverin 0.1 mg/Kg. The treatment was started with monitoring of total blood cell count, liver function tests and renal function tests. Serial monitoring of serum β-HCG level was done.  Post second cycle β-HCG fell to undetectable level after 2 month. Regular follow up with monitoring of β-HCG level was done which showed no further increase.




Figures 1. Invasive gestational trophoblastic neoplasm, all showing involvement of uterus and left adnexa (red arrow).

Discussion

An invasive mole occurs commonly after the evacuation of GTD. Edematous chorionic villi with trophoblastic proliferation are its characteristic. It can invade into the myometrium of the uterus or to adjacent structures.[4] Invasive mole usually presents with vaginal bleeding, an enlarged uterus and high urinary or serum β-HCG level. It mostly occurs following the evacuation of a molar pregnancy. Choriocarcinoma can occur after a hydatidiform mole or even after a normal pregnancy. Choriocarcinoma usually occurs after interval of more than six months. β-HCG levels are much higher than in invasive mole.[5] Persistent raised level of β-HCG, should suggest GTN after evacuation of a complete mole.
The cancer committee of the international federation of gynecologists and obstetricians (FIGO) has established the regression guidelines for the diagnosis of post molar gestational trophoblastic neoplasia.[6]
Ultrasonography (USG) is one of the important tools in diagnosis of suspected GTN.[7] On USG an invasive hydatidiform mole, a placental site trophoblastic tumor, and choriocarcinoma typically exhibit a heterogeneous, hyperechoic, solid mass with cystic vascular spaces, located within the myometrium.[7,8] Color doppler imaging helps in the assessment of neovascularisation in these tumors. A combination of both biochemical findings and USG appearances helps to differentiate between these.[9] Invasive mole can rarely metastases to the lungs. Metastases are more common with choriocarcinoma.[10] The absence of metastases helps to rule out choriocarcinoma.
Management of an invasive mole includes treatment with chemotherapy as well as continuous monitoring of β-HCG. Methotrexate is helpful in treatment of most non-metastatic and low risk cases.[11,12] Follow up of Patients with GTN should be with weekly quantitative β-HCG levels until the values are normal for three consecutive weeks, then should be monitored monthly for 12 months.

Conclusion

Gestational trophoblastic disease (GTD) is a group of disorders related to pregnancy arising from abnormal placental trophoblast cells. Early diagnosis in cases of persistent raised level of β-HCG helps in successful treatment with chemotherapy.

References
  1. Ngan HY, Bender H, Benedet JL, Jones H, MontruccoliGC, Pecorelli S; FIGO Committee on Gynecologic Oncology. Gestational trophoblastic neoplasia, FIGO 2000 staging and classification. Int J Gynaecol Obstet 200383 Suppl1: 175-177.
  2. Hammond CB. Gestational trophoblastic neoplasms. In: Scott JR, DiSaia PJ, Spellacy WN (eds). Danforth’s Ob178 Kavitha Nair et al Invasive mole of the uterus – a rare case diagnosed by ultrasound: a case report obsstetrics and Gynecology, 8th ed. Philadelphia: LippincottWilliams& Wilkins 1999: 927-937.
  3. Tie W, Tajnert K, Plavsic SK. Ultrasound imaging of gestational trophoblastic disease. DSJUOG 2013; 7: 105-112.
  4. Soper JT, Mutch DG, Schink JC; American College of Obstetricians and Gynaecologists. Diagnosis and treatment of gestational trophoblastic disease: ACOG Practice Bulletin No.53. GynecolOncol 2004; 93: 575-585.
  5. Maeda K, Kurjak A, Varga G, Honemeyer U. Trophoblastic diseases. DSJUOG 2012; 6: 27-42.
  6. Goldstein DP, Berkowitz RS. Current management of gestational trophoblastic neoplasia. HematolOncolClin North Am 2012; 26: 111-131.
  7. Jauniaux E. Ultrasound diagnosis and follow up of gestational trophoblastic disease. Ultrasound ObstetGynecol1998; 11: 367-377.
  8. Zhou Q, Lei XY, Xie Q, Cardoza JD. Sonographic and Doppler imaging in the diagnosis and treatment of gestational trophoblastic disease: a 12-year experience. J Ultrasound Med 2005; 24: 15-24.
  9. Timmerman D, Wauters J, Van Calenbergh S, et al. Color Doppler imaging is a valuable tool for the diagnosis and management of uterine vascular malformations. Ultrasound ObstetGynecol 2003; 21: 570-577.
  10. Smith HO, Kohorn E, Cole LA. Choriocarcinoma and gestational trophoblastic disease. Obstetrics andGynaecology Clin North Am 2005; 32: 661-684.
  11. May T, Goldstein DP, Berkowitz RS. Current chemo therapeutic management of patients with gestational trophoblastic neoplasia. Chemother Res Pract 2011; 2011: 806256.
  12. Homeslay HD. Single agent therapy for non-metastatic andlow risk gestational trophoblastic disease. J.Reprod.Med 1998:43; 69-74
Citation

Madhu kumari, Chavan NN, Changede P, Gupta S. Invasive Gestational Trophoblastic Neoplasm: A Rare Interesting Case Managed Successfully With Chemotherapy. JPGO 2015. Volume 2 Number 10. Available from: http://www.jpgo.org/2015/10/invasive-gestational-trophoblastic.html

Anterior Abdominal Wall Adhesions Mimicking Intramyometrial Collection

Author Information

Pawde A*, Chauhan AR**, Mayadeo NM***.
(* Senior Registrar, ** Additional Professor, *** Professor, Department of Obstetrics and Gynecology, Seth G S Medical College and KEM Hospital, Mumbai, India.)

Abstract

With increasing rates of cesarean section (CS), complications in cases of previous CS such as dense adhesions and adhesions between bladder and uterus are on the rise. These may be identified on speculum and vaginal examination, or by ultrasound. We report a case of dense anterior abdominal wall adhesions which was misdiagnosed as a large intramyometrial collection in a first trimester termination of pregnancy which was abandoned due to hemorrhage.

Introduction

Adhesions are a common complication after any abdominal surgery. They may present depending upon their location and severity. in cases of previous lower segment cesarean section (LSCS), adhesions have been reported between posterior surface of urinary bladder and uterus.[1] The anterior abdominal wall may also be densely adherent to either the bladder or the uterus itself. Bladder is pulled up due to dense adhesions, and there may be acute anteversion of the uterus leading to change the cervico-uterine angle. These adhesions may be asymptomatic or patients may have chronic pelvic pain; more importantly they pose difficulty in subsequent surgeries.

Case Report

G3 P2 L2 with 12 weeks’ gestation with previous LSCS was referred to our tertiary care hospital 6 hours after attempted first trimester abortion at a peripheral hospital with history of torrential intraoperative bleeding. The referral note stated that the procedure had been abandoned in view of profuse bleeding while dilating the cervix with metal dilators. She did not have any other complaints such as abdominal pain or hematuria. On examination, her vital parameters were normal; abdomen was soft and non-tender, without any obvious lump or signs of perforation. On speculum examination cervix and vagina were healthy, the cervix was high-up and pulled suprapubically; there was no bleeding coming through cervix. On vaginal examination uterus was non- tender, corresponding to 12 weeks' size, and a firm lump was felt anterior to the uterus with restricted mobility. Sonography reported a large intramyometrial collection (7 x 8 cm) with vascularity, with intact serosa suggestive of perforation in the region of the lower uterine segment. MRI confirmed the sonography report. β HCG was 70,000 mIU/ l. Other biochemical investigations were normal.
Laparotomy was performed: abdomen was opened through the previous Pfannenstiel incision. On inspection, the uterus was acutely anteverted and pulled up, approximately 6 to 8 weeks’ size, with thick bands of adhesions extending from anterior surface of the uterus to the anterior abdominal wall. This gave the appearance of a large sealed off “collection” which was reported on USG and MRI. No obvious hematoma or collection was noted over the anterior uterine wall. Sharp dissection and cautery helped to separate the dense bands. With great difficulty, the region of the previous scar was reached, which was deeply situated; probably the original incision was supracervical. The bladder was densely adherent below it. An extremely small contained collection and perforation in the anterior uterine wall at this level was noted and after evacuation of the products of conception, uterus was sutured with delayed absorbable sutures of polyglactin 910 No 1. Integrity of the bladder was checked with retrograde instillation of methylene blue dye and bladder was found to be intact. Patient recovered uneventfully.


Figure 1. Adhesions between uterus and the anterior abdominal wall.

Discussion

This case highlights the problems both of MTP and laparotomy in patients of previous cesarean section, as adhesions are a common complication. Thorough examination prior to the MTP may have demonstrated a pulled up cervix or puckering or dimpling i.e. tightly held vaginal mucosa at the bladder, which would have allowed the surgeon to anticipate the possibility of adhesions between uterus, bladder and anterior abdominal wall.[2] Care must be exercised prior to MTP in cases of previous LSCS; USG prior to MTP may have diagnosed adhesions or discrepancy between the clinical estimation of uterine size (12 weeks) and actual gestational age. Technique of first trimester MTP in such patients especially to prevent perforation and other complications needs emphasis. If forceful dilation is attempted in an acutely anteverted uterus it may lead to perforation, hence should be avoided. During dilatation, straightening of the utero- cervical angle by traction on cervix with the vulsellum is a simple step which helps to align the canal and prevent perforation.[3] Methods such as pre-operative cervical ripening with misoprostol and use of manual vacuum aspiration syringe make dilatation easier and procedure less traumatic, respectively. Hence evacuation is simpler and associated with fewer complications, namely perforation and bleeding.[4] MTP was abandoned in this case due to bleeding; this usually signifies perforation or incompleteness of the procedure. USG if available in the operation theatre may have helped.[5]
On ultrasound examination, adhesions are visible in tissue with same density as uterine muscle, reported as horn or beaking of uterus.[6] Rarely as in our case do these adhesions form thick bands which appeared as serosa on ultrasound and MRI. Exploratory laparotomy helped to correctly identify the adhesions. When such adhesions are encountered during a surgery which requires separation of bladder from the uterus, sharp dissection should be performed to prevent inadvertent injury to bladder and integrity of bladder should be confirmed whenever in doubt; it should be tackled at the same sitting.[1,4]

Conclusion

Previous caesarean section may be associated with varied degrees of adhesions and may pose difficulty in subsequent procedures. These should be anticipated with thorough preoperative examination and tackled cautiously.

References
  1. Sbarra M, Boyd M, Dardarian T S. Complications due to adhesion formation following cesarean sections: A review of deliveries in three cases. Fertility Sterility 2009; 92 (1): 394.e13-e16.
  2. Sheth S, Hajari A. The Place of Vaginal Hysterectomy. Glob. Libr. Women’s Med. (ISSN: 1756-2228) 2010; DOI 10.3843/GLOWM.10465.
  3. Jonathan D B, Sherlock D J, Atkinson A L. Use of an Intubating Stylet as a Guide to Complete Uterine Curettage Complicated by Uterine Perforation. Case Reports in Obstetrics and Gynecology 2013, Article ID 195383. http://dx.doi.org/10.1155/2013/195383.
  4. Davey A K, Maher P J. Surgical adhesions: a timely update, a great challenge for the future. J Minim Invasive Gynecol 2007; 14: 15-22.
  5. Elsayed M A. Routine ultrasound guided evacuation of first trimester missed abortion versus blind evacuation. Middle East Fertility Society Journal 2014;19(3):171-75.
  6. Walid MS, Heaton RL. Uterine peaking-sonographic sign of vesico-uterine adhesion. Ger Med Sci. 2011; 9:1612-16.
Citation

Pawde A, Chauhan AR, Mayadeo NM. Anterior Abdominal Wall Adhesions Mimicking Intramyometrial Collection. JPGO 2015. Volume 2 Number 10. Available from: http://www.jpgo.org/2015/10/anterior-abdominal-wall-adhesions.html

Atypical Prsentation Of A Cervical Fibroid In Pregnancy

Author Information

Joshi A*, Mhaske N**, Kharat D***, Fonseca MN****.
(*2nd year post graduate student, ** 3rd year post graduate student, *** Assistant Professor, **** Additional Professor, Department of Obstetrics/Gynecology, LTMMC and LTMGH, Mumbai, India.)

Abstract

Cervical fibroids are a rare occurrence. The prevalence of fibroids during pregnancy, irrespective of their site, has been reported to be 1-4% and can present as a diagnostic dilemma. Cervical leiomyomas may be confused with chronic uterine inversion, procidentia or cervical carcinoma. A prolapsed cervical mass simulating a uterine inversion, with a high beta hCG causing a diagnostic dilemma is presented here.

Introduction

A protruding cervical mass in a woman of reproductive age group encompasses a wide range of diagnostic dilemmas for the gynecologist. The differential diagnosis includes procidentia, uterine inversion, cervical fibroid or cervical malignancy. A good clinical acumen, careful examination and meticulous histopathological analysis is of paramount importance in such cases, and all possible diagnostic and interventional approaches should be explored. A cervical fibroid with pregnancy is an uncommon entity and may pose additional challenges due to atypical presentation.

Case Report

A 33 year old primipara, married since 15 years, presented to our tertiary care center, with chief complaint of something coming out per vaginum since 2 months. Her previous menstrual cycles were irregular and she reported menstrual-like bleeding, starting 15 days prior and she was bleeding continuously since then. On examination, her pulse rate 110 per minute, blood pressure 90/60 mm Hg, with severe pallor, but no icterus or edema.  Cardiovascular and respiratory system examination were within normal limits. There was no abdominal guarding, rigidity or tenderness. Local examination showed a large fleshy prolapsed mass measuring approximately 8x8 cm, soft in consistency, with irregular contour (figure 1). On per vaginal examination the uterus was 14 weeks' size with cervical rim felt beyond the mass with marked tenderness on deep palpation.
Laboratory investigations revealed severe anemia. Blood sugars, liver and renal function tests, coagulation profile, thyroid function test were within normal limits. Urine pregnancy test was equivocal. Therefore we decided to send beta-HCG and surprisingly, beta-HCG was 2,23,700 IU/ml. Patient was admitted and transfused 2 units of packed red cells and IV antibiotics were given. A computerized tomography abdomen with pelvis revealed 8x6x13 cm cervical fibroid with endometrial thickness of 2.4 cm. Repeat beta-HCG after 48 hours was 307000 IU/ml. A decision for examination under anesthesia with dilatation and curettage was taken with the following differential diagnosis in mind: cervical fibroid with incomplete abortion, chronic uterine inversion, gestational trophoblastic neoplasia with cervical growth.
On examination under anesthesia, a large polypoidal, 8x8 cm pedunculated fleshy mass arising from 9’o clock position of the cervix was noted, it could be extruded out from the vagina, and uterine sound could be easily negotiated (figure 2). A cervical polypectomy with dilatation and curettage was done and specimens sent for histopathology examination. Histopathology of mass revealed few irregular glands with complex architecture with minimal nuclear atypia. No evidence of mitosis or necrosis. Surprisingly, the features were described to be suggestive of “adenoma malignum” and immunohistochemistry was advised. The endometrial curettage specimen revealed products of conception. Repeat beta-HCG done after one week was 123000. Immunohistochemistry (sent to a referral oncological laboratory) report of the cervical mass showed mucinous glandular epithelium negative for CEA, CDX2 and CK20, but positive for CK7, while Ki-67 index was upto 1%.There was no cytologic atypia. The histopathological diagnosis was revised to one of adenomyoma of cervix, without any features suggestive of malignancy.  pproximately two weeks later, patient presented with profuse bleeding per vaginum, for which dilatation and curettage was performed and histopathology revealed products of conception.


Figure 1. Mass coming out per vaginum (arrow).


Figure 2. Origin of the mass from the cervix (arrow).


Figure 3. Pedicle of the mass on the cervix (arrow) after the procedure.

Discussion

Leiomyomas are the most common pelvic tumors among women of reproductive age group. Cervical leiomyomas contribute to 1-2% of leiomyomas.[1] Pregnancy associated with cervical fibroid is a rare occurrence. It may present with heavy bleeding, pain, anemia and generalized discomfort.  Cervical fibroids in pregnancy cause obstructed labor unless very well taken up into the lower segment. Infection in a fibroid can occur postpartum more commonly following an abortion.
Cervical fibroids in pregnancy pose a distinctive challenge. Management options depend on the patient's general condition and gestational age at the time of presentation and future fertility desire.[2]
Minimal deviation adenocarcinoma (MDA), also known as adenoma malignum of the uterine cervix, is a rare entity, which may be associated with Peutz-Jeghers syndrome. MDA appears as multilocular lesions with solid components that extend from the endocervical glands to the deep cervical stroma. Accurate diagnosis of MDA is important because of early dissemination into peritoneal cavity, and necessitates aggressive management.[3,4]
MDA was an opinion of the pathologist of our center, which was ultimately ruled out upon immuno-histochemistry. It is important to note that this rare entity, has a very high inter-observer variability in its reporting 5, mainly because this entity can closely mimic other pathologies.[4,6]

Immunohistochemistry refers to the process of detecting antigens (e.g., proteins) in cells of a tissue section by exploiting the principle of antibodies binding specifically to antigens in biological tissues. The pseudocapsule of the fibroid expresses proteins like collagen and laminin IV, used as specific markers. The fibroid development is postulated to progress due to angiogenic factors and markers specific to those are proposed for management options. Thus, although immunohistochemistry is in its primitive stages as regards management of fibroids, it shows a promising future.[7,8]
Despite the varied modes of presentation of fibroids, very rarely do they need immune-histochemistry for their final confirmation. In this particular case, the diagnosis could be ascertained only after this special investigation, and hence this case is presented here.

References
  1. Kamra HT, Dantkale SS, Birla K, Sakinlawar PW, Narkhede RR. Myxoid  leiomyoma of cervix. J Clin Diagn Res. 2013 Dec;7(12):2956–7.
  2. Gandhi AC, Dugad HI, Shah Y. A rare presentation of cervical fibroid in pregnancy. Ann Afr Med. 2014 Jan 1;13(2):88–90.
  3. Mowat A, Land R. Adenoma malignum presenting as urinary incontinence. Int Urogynecol J. 2014 Feb 27; 25(9):1287–9.
  4. Zhou H. Diagnostic challenges in minimal deviation adenocarcinoma of the uterine cervix: A report of two cases and review of the literature. Mol Clin Oncol. 2013 Jul; 1(5):833–8.
  5. Tsuda H, Mikami Y, Kaku T, Akiyama F, Hasegawa T, Okada S, et al. Interobserver variation in the diagnosis of adenoma malignum (minimal deviation adenocarcinoma) of the uterine cervix. Pathol Int. 2003 Jul; 53(7):440–9. 
  6. Mikami Y, Hata S, Fujiwara K, Imajo Y, Kohno I, Manabe T. Florid endocervical glandular hyperplasia with intestinal and pyloric gland metaplasia: worrisome benign mimic of “adenoma malignum”. Gynecol Oncol. 1999 Sep; 74(3):504–11. 
  7. Tinelli A, Malvasi A, Cavallotti C, Dell’Edera D, Tsin DA, Stark M, et al. The management of fibroids based on immunohistochemical studies of their pseudocapsules. Expert Opin Ther Targets. 2011 Nov;15(11):1241–7. 
  8. Tal R, Segars JH. The role of angiogenic factors in fibroid pathogenesis: potential implications for future therapy. Hum Reprod Update. Jan;20(2):194–216. 
Citation

Joshi A, Mhaske N, Kharat D, Fonseca MN. Atypical Prsentation Of A Cervical Fibroid In Pregnancy. JPGO 2015. Volume 2 Number 10. Available from: http://www.jpgo.org/2015/10/atypical-prsentation-of-cervical.html

A Twisted Ovarian Mucinous Cystadenoma Complicating Pregnancy: Case Report

Author Information

Verma K.
(Assistant Professor, Department of Obstetrics & Gynecology, S.P. Medical College and Associate Group of P.B.M. Hospital, Bikaner Rajasthan, India.)

Abstract

Mucinous cystadenoma is the second most common type of benign epithelial ovarian tumor. It is uncommon during pregnancy. In this case I report a 35 year old fifth gravida with 12 weeks of pregnancy presenting with three month amenorrhea and severe pain in abdomen and vomiting since two days. She had a twisted mucinous cystadenoma of right ovary. Right salpingo- oophorectomy was done. She came in follow up with 18 weeks of healthy viable pregnancy that was confirmed by USG. Although surgical intervention is a well-accepted choice in such an acute condition but abdominal surgery in a pregnant state always carries some risk to mother and unborn fetus, so before management appropriate counsellng of patient should be done as well as risks involved with surgery must be taken into consideration.

Introduction

The incidence of adnexal masses in pregnancy varies from 2% to 10%[1] While incidence of ovarian torsion in pregnancy is 5%.[2] The most common histological diagnosis of surgically treated adnexal masses was reported to be dermoid cyst followed by cystadenoma then functional cysts and endometrioma.[4] Mucinous cystadenoma is a surface epithelial tumor of ovary. Most of them are benign, lobulated with smooth wall. They comprise 28% of adnexal masses found during pregnancy. The most frequent and serious complication of benign ovarian cysts during pregnancy is adnexal torsion. Incidence of adnexal torsion is 5% during pregnancy. Torsion is most common in first trimester and then get less frequent as pregnancy advances. Patients undergoing emergency surgery because of torsion or hemorrhage are at the greatest risk of spontaneous abortion or premature delivery compared to elective surgery, which must be delayed till 2nd trimester. Here, I report a rare occurrence of torsion of large mucinous cystadenoma in first trimester of pregnancy.

Case Report

A 35 year old women gravida 5 para 2 abortus 2 presented to our gynecology emergency with 3 month amenorrhea and pain in abdomen with vomiting for two days. She had 2 alive children delivered vaginally followed by 2 spontaneous abortions at 2 and 2.5 months of gestational age. In this pregnancy she did not took any antenatal treatment. Her past medical and surgical history was insignificant. On examination she had moderate grade of pallor. She was cold and clammy, her pulse was 120/minute and blood pressure was 90/70 mm Hg. On per abdominal examination there was moderate distension with generalized tenderness and rigidity. On per vaginal examination cervix was soft, os closed, exact size of uterus was not made out because of distension.
Differential diagnosis of ruptured ectopic pregnancy or torsion of ovarian cyst was made. Ultrasonography (USG) showed a single live intrauterine pregnancy, corresponding to 11 weeks and 3 days maturity. A heteroechoic lesion is noted in pelvis arising from adnexa, measuring 13.9 cm x 8.6 cm. Both ovaries were not separately seen. Moderate to gross hemoperitoneum was seen.
Decision of emergency laparotomy was taken with proper counseling of the patient and her relatives. During laparotomy moderate hemoperitoeum was found with a large right unilocular cystic mass of 12x10 cm, twisted around its pedicle. The uterus was 12-14 weeks' size and left tube and ovary were healthy. Right salpingo-ophorectomy was done followed by peritoneal washing. Her post-operative course was uneventful. She was discharged home after obstetric USG confirmed a viable pregnancy of 12 weeks. The histology report showed a benign mucinous cystadenoma of the ovary. Patient came for follow up, currently having 18 weeks of viable pregnancy.


Figure 1 Intraoperative picture showing uterus (green arrow) with right adnexal mass.


Figure 2 Showing macroscopic appearance of the mass.
Management depend upon the symptoms, gestational age, size and character of the cysts. If the mass is unilateral unilocular and less than 6 cm in diameter, observation is recommended. If it is larger than 6 cm, solid, bilateral, persists into the second trimester or become symptomatic then a surgical intervention is required. If the ovarian cyst is diagnosed in the first trimester it is better to wait until 16 weeks' gestation when the pregnancy is more secure and the cyst may resolve spontaneously by that time. If torsion or rupture of ovarian mass found or any features suggestive of malignancy are present, then emergency surgery is the treatment of choice irrespective of the period of gestation.[9] It is concluded that ovarian cyst in pregnancy must be followed up properly due to possibility of adverse effect of cyst on pregnancy. Though torsion is uncommon in pregnancy it should be kept in mind for making differential diagnosis of acute abdominal pain in pregnancy. Early diagnosis and appropriate intervention are associated with favorable fetomaternal outcome.

References
  1. Schwartz N, Timor-Tritsch IE, Wang E. Adnexal masses in pregnancy. Clinical obstetrics and gynecology. Dec 2009;52(4):570-585.
  2. Giuntoli RL, 2nd, Vang RS, Bristow RE. Evaluation and management of adnexal masses during pregnancy. Clinical obstetrics and gynecology. Sep 2006;49(3):492-505.
  3. Boulay R, Podczaski E. Ovarian cancer complicating pregnancy. Obstetrics and gynecology clinics of North America. Jun 1998;25(2):385-399.
  4. Sherard GB, 3rd, Hodson CA, Williams HJ, Semer DA, Hadi HA, Tait DL. Adnexal masses and pregnancy: a 12-year experience. American journal of obstetrics and gynecology. Aug 2003;189(2):358-362.
  5. Vizza E, Galati GM, Corrado G, Atlante M, Infante C, Sbiroli C. Voluminous mucinous cystadenoma of the ovary in a 13-year-old girl. Journal of pediatric and adolescent gynecology. Dec 2005;18(6):419-422.
  6. Tugrul S, Pekin O, Ayvaci H, Tarhan N, Uludogan M. Giant benign mucinous cystadenoma growing during pregnancy: a case report. Clinical and experimental obstetrics & gynecology. 2007;34(2):126-127.
  7. Chiou SY, Lev-Toaff AS, Masuda E, Feld RI, Bergin D. Adnexal torsion: new clinical and imaging observations by sonography, computed tomography, and magnetic resonance imaging. Journal of ultrasound in medicine : official journal of the American Institute of Ultrasound in Medicine. Oct 2007;26(10):1289-1301.
  8. Lee CH, Raman S, Sivanesaratnam V. Torsion of ovarian tumors: a clinicopathological study. International journal of gynaecology and obstetrics: the official organ of the International Federation of Gynaecology and Obstetrics. Jan 1989;28(1):21-25.
  9. Leiserowitz GS. Managing ovarian masses during pregnancy. Obstetrical & gynecological survey. Jul 2006;61(7):463-470.
Citation

Verma K. A Twisted Ovarian Mucinous Cystadenoma Complicating Pregnancy: Case Report.
JPGO 2015. Volume 2 Number 10. Available from: http://www.jpgo.org/2015/10/a-twisted-ovarian-mucinous-cystadenoma.html

Post PICC Line Thrombosis In a Severely Anemic Post Abortal Patient

Author Information

Desai DV*, Karve N**, Gupta AS***.
(* First Year Resident, ** Fourth Year Resident, *** Professor. Department of Obstetrics & Gynecology, Seth G.S.Medical College & KEM Hospital, Mumbai, India.)

Abstract

Thrombosis of deep neck veins is a rare complication associated with peripheral vein catheterization albeit with a high risk of adverse outcome. We report a case of internal jugular vein thrombosis in a severely anemic patient with peripheral intravenous central catheter (PICC) line inserted for monitoring central venous pressure.

Introduction

In the year 1856 Virchow published the landmark study on the etiopathogenesis of pulmonary thromboembolism, which later lead to the consensus on the high risk factors for thrombosis in blood vessels, now known as the Virchow’s Triad; namely- hypercoagulablility, endothelial injury and hemodynamic changes.[1] Obstetric patients are privy to all three of these factors which subjects them to a high risk of thrombosis throughout their antenatal and postpartum period. Pregnancy itself is a hypercoagulable state, a natural mechanism to reduce the total blood loss at the time of delivery. The shearing forces of the fetal head over the pelvic tissues, often times causes pelvic vessel thrombosis due to local trauma. Hyperdynamic circulatory changes, physiological anemia of pregnancy and stasis of blood in lower limb veins adds to the probability of thrombosis and subsequent embolism. Anemia causes turbulence of blood flow in vessels and thus adds to the risk of thrombosis. At our institute we cater to high-risk obstetric patients, a large proportion of which are anemic. We report a case of internal jugular vein thrombosis in a severely anemic patient with peripheral intravenous catheter line inserted for monitoring central venous pressure during process of abortion.

Case Report

A 32 year old gravida 4, para 3, living 3 with 15 weeks' twin gestation came with threatened abortion and a history of vaginal bleeding for the last 15 days. All her previous pregnancies were vaginal home deliveries. She had no previous medical or surgical history suggestive of any risk of hypercoagulability. On examination she had severe pallor and tachycardia - pulse 100 bpm. the uterine size was of 18 weeks of gestation. Blood mixed liquor was demonstrated to be leaking from the cervical os. Ultrasonography showed twin live gestation of 19+6 weeks and 19 weeks with anhydramnios. Laboratory investigations showed Hb - 4.8 g/dl and normal DIC profile. She was started on parenteral ceftriaxone, metronidazole and gentamicin. Peripheral intravenous central catheter (PICC), size 14-16 Fr was inserted in the right median basal vein for central venous pressure (CVP) monitoring after written and informed consent by a trained resident doctor under full aseptic precautions. There was no difficulty in its insertion and it was performed in a single attempt with free flow of blood from it. Ringer's lactate infusion was slowly given initially. It was secured by two linen sutures and strapped by an elastic adhesive tape. She was then transfused 2 units of packed red blood cells (PRBCs) under strict monitoring on two consecutive days from the PICC. In between the PICC was flushed with normal saline. No antibiotics or oxytocin infusion was administered from that PICC.
Decision was taken for induction of abortion. After written and informed consent, induction of abortion was done with PGE1. A tablet of 400 μg PGE1 was inserted vaginally every 4 hourly till a total dose of 2000 μg was administered. As the internal os had opened PGE1 was sequentially followed by infusion of oxytocin 20 IU in 500 ml Ringer’s lactate. The patient aborted on 3rd day after induction. Emergency check curettage was done with 4th unit of PRBCs on flow.
Strict monitoring of CVP was done via the PICC line. The column moved well with inspiration and was not suggestive of any partial or complete block of the PICC. The patient complained of pain, swelling and difficulty in neck movements 30 hours after the check curettage. She never had fever. Prompt ultrasonography (USG) with Doppler of the neck showed right internal jugular vein thrombosis extending from the right retro-mandibular region till its opening into the right subclavian vein, a length of approximately 6 cm, the left side being normal. Neck radiograph was within normal limits. PICC was then immediately removed on day 6 after insertion.
Hematologist was consulted and the patient was started on unfractionated Heparin 5000 IU S.C. 6 hourly and warfarin 5 mg H.S. Warfarin dose was adjusted with daily monitoring of INR and heparin was discontinued when PT-INR was above 2. Thrombophilia evaluation was deferred for 6 weeks on the hematologist’s advice. Patient was discharged on warfarin 7.5 mg H.S. She was advised to continue it for 6 months. The couple was counseled to use barrier contraception and avoid pregnancy for 6 months.

Discussion

Internal jugular vein thrombosis is a serious event with fatal outcome.[2] PICC’s have become popular for patients requiring long term intravenous therapy due to less catheter related serious complications due to improvements in catheter designs (flexible non-thrombogenic silicone catheter).[3] Complications of the PICC include occlusion (7%), rupture (1.6%), accidental withdrawal (2.4%), infection (2.4%), pulmonary embolism, sepsis with septic emboli, and intracranial extension of thrombus.[4] There is also a risk of cardiac perforation and arrhythmia if tip of PICC gets advanced.[1]

Guideline for Peripheral intravenous line insertion[5]

Only competent staff should do the procedure. PICC line should be inserted in an area where asepsis can be maintained. All sterile techniques should be implemented with good record keeping and documentation. Post procedure modified chest radiograph should be taken with visualization of catheter along the total arm length across the axillary and subclavian veins. Preferable location of the tip of the PICC is in the distal 1/3rd of the superior vena cava (SVC) near its entry into the right atrium. USG guided insertions are recommended in case of difficult insertions. Basilic vein (8 mm) is the most preferred vein as it is less tortuous. Sterile transparent semi-permeable self adhesive dressings should be used to prevent complications. The syringe recommended to flush the PICC should be a 10 ml syringe as smaller syringes used for flushing can generate a higher pressure that can result in damage to the vessel wall. The last 1/2 ml of flush solution should be under positive pressure to prevent the back flow of blood from the catheter tip so as to prevent occlusion by clot formation. Presence of tachycardia, tachypnea, hypotension, pain, trismus, raised central venous pressure should raise a suspicion of a complication related to these PICC. Management consists of anticoagulants, dose optimization with PT-INR monitoring, prophylactic parenteral antibiotics, Superior vena cava (SVC) filters in case of failure of medical line of management. Certain precautions are recommended for insertion of the PICC line. Proper indication, asepsis, use of local anesthesia and post procedure radiography for confirmation of correct placement are desirable. Regular flushing of the cap at the end of the line decreases tip site infection. To prevent phlebitis, some clinicians add heparin and hydrocortisone to the solution or nitroglycerine patch over the catheter site. It is imperative that peripheral catheter sites be inspected regularly and catheter sites changed if evidence of phlebitis develops.
In our case, the PICC was inserted by a trained resident doctor. However, the PICC should have been removed immediately after the check curettage or after the completion of the transfusion of the 4th  PRBC as the patient did not need any further CVP monitoring. Catheterization for prolonged duration can cause endothelial damage of the vessel walls and initiate the formation of a thrombus. Proper implementation of the guidelines, prompt removal when the need for the PICC ceases and a high degree of suspicion of complications should be followed by all clinicians who have patients requiring PICC monitoring.

References
  1. Silver D, Vouyouka A. Management of venous thromboembolism. In Baker R J, Fischer JE (ed); Master of surgery ;4th edition; Philadelphia, Lippincott Williams & Wilkins, A Wolter Kluver, 2001; Volume 2; pg. 2199
  2. Schroeder RA, Barbeito A, Bar-Yosef S, Mark, JB. Cardiovascular monitoring. In Miller RD (ed); Eriksson LI, Fleisher LA, Wiener-Kronish JP, Young WL. (Associate Editors). Miller's Anesthesia. 7th edition. Philadelphia, Churchill Livingstone,Elsevier; 2010; Volume 1, pg 1287-1289.
  3. Weissman C. Nutrition and Metabolic Control. In Miller RD (ed); Eriksson LI, Fleisher LA, Wiener-Kronish JP, Young WL. (Associate Editors). Miller's Anesthesia.7th edition. Philadelphia, Churchill Livingstone, Elsevier; 2010; Volume 2, pg. 2947.
  4. Vidal V, Muller C, Jacquier A, Giorgi R, Le Corroller T, Gaubert J, etal. Prospective evaluation of PICC line related complications. J Radiol. 2008 Apr; 89 (4) :495-8.
  5. Guideline for Peripherally Inserted Central Venous Catheters (PICC). Queensland Government. Centre for Healthcare Related Infection Surveillance and Prevention & Tuberculosis Control Version 2 – March 2013; 1-13 Available at: https://www.health.qld.gov.au/qhpolicy/docs/gdl/qh-gdl-321-6-1.pdf
Citation

Desai DV, Karve N, Gupta AS.Post PICC Line Thrombosis In a Severely Anemic Post Abortal Patient. JPGO 2015. Volume 2 Number 10. Available from: http://www.jpgo.org/2015/09/post-picc-line-thrombosis-in-severely.html