Volume 4 Issue 1, January 2017

Editorial
Parulekar SV

Endometrial Appearance During Myomectomy
Dalvi P, Parulekar SV.

MRKH Syndrome Variant With Meckel’s Diverticulum
Manjrekar VM, Parulekar SV.

Pregnancy - A Primary Cause Of Ischemic Stroke?
Shah A, Chauhan AR.

Necessary Myomectomy At Cesarean Section
Sneha V, Kale K, Chauhan AR.

Multiple Parasitic Leiomyomas After Laparoscopic Myomectomy: An Unusual Complication Of Power Morcellation
Shah NH, Kale K, Paranjpe S, Nagarkatti RS.

Atypical Posterior Uterine Rupture
Dharmadhikari M, Daigavane M, Samant PY, Chaudhari HK.

Spontaneous Hemoperitoneum Secondary To Warfarin Toxicity: Learning The Balancing Act
Bhosle S, Mhaskar P, Honavar P, Samant PY.

Large Degenerated Broad Ligament Fibroid
Bora A, Madhva Prasad S, Gupta AS.

Successful Maternal And Fetal Outcome In A Case Of Non Cirrhotic Portal Hypertension In Pregnancy
Sikarwar  R, Panchbudhe S,  Satia M.

Editorial

Parulekar SV

Medical errors may occur due to an error in judgment, lack of knowledge, inadequacy of training, ttempting techniques beyond one's ability, lack of concentration or negligence. These errors could be in diagnostic or therapeutic decision making or in carrying out the treatment in time or effectively. Therapeutic errors may be in medication or surgical procedures.These may result in mortality, serious or not so serious morbidity, or may be 'near misses' which could have resulted in serious injury to the patient but luckily did not. Medical errors are far more common than believed. According to one report these are said to be the third leading cause of death in US. The scenario is probably not very different in other countries. What varies is the degree to which such errors are reported. It is said that about one third patients admitted to hospitals experience errors in their management. It is also said that 90% of these errors go unreported. There is a tendency to hide such errors for reasons like fear of litigation, need for compensation, loss of face, damage to reputation and practice, criticism by peers, and disciplinary action by employers. Ethically it is mandatory that such errors be reported to the patients. But there is another reason for such reporting too. It creates awareness of such errors, which helps peers and administrators devise methods of minimizing or avoiding such errors. Errors may be  reported in internal audits and departmental meetings. However such information reaches very few individuals. The only known method that will disseminate the information to a large number of individuals involved with giving healthcare  is reporting in scientific journals. Such reports may be in the form case reports or results of analysis of mandatory reporting to governmental agencies. Confidentiality of patients is always maintained in journal reports, and hence the physician's fear of admission of an error may be reduced by that measure. This fear can be reduced further by giving legal protection to the person reporting the error, so that more people are encouraged to report errors. This will need a change in the legislature. That may or may not occur, at least not in the near future. In the meantime, we have started a new feature in JPGO under the category 'error'. We published one error in the December issue of the journal. It was widely appreciated. We propose to publish errors which are unique, perhaps the first such occurrences in the world literature. We are proud to say that JPGO is one of the very few journals which publish errors. We are sure that it will go a long way in advancing scientific knowledge and reducing the risk of occurrence of such errors in future.

Endometrial Appearance During Myomectomy

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Author Information

Dalvi P*, Parulekar SV**.

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

Abstract

Myomectomy is the process of removal of leiomyomas. In contrast to a hysterectomy it allows the uterus to be left in place and hence is a fertility preserving surgery. It is considered important that the endometrium is not opened during myomectomy, so as to avoid uterine rupture in a future pregnancy. We report a case where an open myomectomy was done for a large fundal and posterior, subserosal leiomyoma without opening the endometrium.

Introduction

Uterine leiomyomas often develop in the anterior and posterior walls since the bulk of the myometrium is located in the anterior and posterior walls of the uterus.[1]   Leiomyomas can also be located at other locations. The incision on the uterus is generally such that the scar would lie on the anterior surface of the uterus.[2,3,4] Irrespective of the site of the uterine incision, it is considered important that the endometrium is not opened during myomectomy, because that increases the risk of uterine rupture in a future pregnancy. Here we describe a case of a myomectomy done large fundal and posterior uterine wall leiomyoma lying in close proximity to the posterior endometrium. It is presented mainly for the gross appearance of the endometrium after the dissection.

Case Report

A 32-year-old woman, married for 7 years, para 1 living 1 abortion 1, came with complaints of polymenorrhagia for 7 months with cycles lasting 3 to 4 days every 15 to 20 days, associated with soakage of 4 pads per day and passage of clots. She had a history of dysmenorrhoea for 3 months and history of perceiving a mass per abdomen for 7 days  There was no history of any major medical or surgical illness in the past. She had a history of a lower segment cesarean section done 5 years ago, at the time of which she had received 2 units whole blood transfusion. There was no history of any bowel or urinary bladder related symptoms. Her clinical examination revealed a pulse of 96 beats per minute, blood pressure was 110/70 mm of Hg, and severe pallor. Abdominal examination showed a Pfannensteil scar and a 20 weeks’ size, firm, nontender mass arising from the pelvis. Per speculum examination showed normal findings, except that the cervix was drawn up. Bimanual pelvic examination showed the uterus enlarged to 20 weeks’ size, firm, globular. The lateral fornices were free. General and systemic examination revealed no abnormality. Her hemoglobin was 7.6 g/dl. Biochemical investigations for fitness for anesthesia showed normal results. Abdominopelvic ultrasonography showed uterine enlargement, with an intramural leiomyoma in the fundus and body of the uterus measuring 19.5x11.8x8.4 cm. The endometrium was distorted. Two units of packed cells were transfused. A laparotomy was performed. The anterior surface of the uterus was found to be directly adherent to the anterior abdominal wall at the scar site. These adhesions were released by sharp dissection. A large leiomyoma was found to be in the posterior wall of the uterus (figure 1). It was removed by Bonney’s hood operation. After enucleation of the leiomyoma, the endometrium was found bulging in the bed of the leiomyoma, giving rise to an “egg in a nest” appearance (figure 2). The bed of the leiomyoma was closed by interrupted sutures of No. 1 polyglactin, taking care not to injure the endometrium. The uterine incision was closed with sutures of No. 1-0 polyglactin as in Bonney’s hood operation. Estimated blood loss was about 650 ml. One unit blood transfusion was given. The patient made an uneventful recovery.

Figure 1.  Intraoperative findings showing a large fundal leiomyoma (black arrow) and a simple cyst in the left ovary (yellow arrow).

Figure 2. showing the endometrium pouting out of the leiomyoma bed (arrows) after the leiomyoma was enucleated.

Discussion

A posterior wall incision of the uterus uterus is generally avoided since the postoperative scar would be open to the peritoneal cavity and would form adhesions with bowel or omentum.[5,6]  An upper segment posterior uterine leiomyoma is usually managed by Bonney’s hood operation as was done in the above case.[7]  At the time of performing a myomectomy every attempt is made so as not to breach the endometrium. However in cases of leiomyomas lying in close proximity to the endometrium distorting the endometrium, it is quite likely to occur. Since there is no way of knowing exactly where the endometrium would be, extreme care needs to taken to identify it and avoid injuring it. This is important because any future pregnancy may be associated with risk of uterine rupture during pregnancy or labour in a patient with a prior history of myomectomy.[8] This case is presented mainly to show the appearance of the endometrium so that it can be identified by a gynecologist in training.  

References

1. Cramer, S.F., Patel, A. The frequency of uterine leiomyomas. Am J Clin Pathol. 1990;94:435–438.
2. Breech LL, Rock JA. Leiomyomata uteri and myomectomy. In Rock JA, Jones HW III, editors. Te Linde’s Operative Gynecology. 10th ed. New Delhi: Wolters Kluwer Health – Lippincott Williams & Wilkins 2008; pp. 687-726.
3. Guarnaccia MM, Rein MS. Traditional surgical approaches to uterine leiomyomas: abdominal myomectomy and hysterectomy. Clin Obstet Gynecol 2001;44:385-400.
4. Parulekar SV. Practical Gynecology and Obstetrics. 5th ed. Mumbai: Vora Medical Publications; 2011, pp345-8.
5. LaMorte AI, Lalwani S, Diamond MP. Morbidity associated with abdominal myomectomy. Obstet Gynecol 1993;82:897-900.
6. 6.6.Iverson  RE,  Chelmow  D,  Strohbehn  K, Waldman L, Evantash ED.  Relative  morbidity of abdominal  hysterectomy  and  myomectomy  for  management  of uterine leiomyomas. Obstet Gynecol 1996;88:415–419.
7. Tito Lopes T, Spirtos NM, Naik R, Monaghan JM. Bonney’s Gynaecological Surgery. 11th ed. Wiley Blackwell:2011; Pp 121-124.
8. Frishman, G.N., Jurema, M.W. Myomas and myomectomy. J Minim Invasive Gynecol. 2005;12:443–456 (quiz 57–58).
9. Schrinsky DC, Benson RC. Rupture of the pregnant uterus: a review. Obstet Gynecol Surv. 1978;33(4):217-32.

Citation

Dalvi P, Parulekar SV. Endometrial Appearance During Myomectomy. JPGO 2017. Volume 4 No. 1. Available from: http://www.jpgo.org/2017/01/endometrial-appearance-during-myomectomy.html

MRKH Syndrome Variant With Meckel’s Diverticulum

Author Information

Manjrekar VM*, Parulekar SV**.

(* Third Year Postgraduate student, **Professor and Head, Department of Obstetrics and Gynecology, Seth G. S. Medical College and K. E. M. Hospital, Mumbai, India.)

Abstract

Mullerian anomalies form a spectrum of developmental anomalies of the female reproductive system ranging from lateral and vertical fusion defects to partial or complete absence of reproductive  structures embryologically derived from these. Here we present a case of agenesis of the upper vagina and cervix with bilateral rudimentary horns in a bicornuate uterus with a well differentiated and functioning endometrium causing endometriosis and cyclical pain. There was an asymptomatic Meckel’s diverticulum too. The diagnostic dilemma and management options  of such cases are discussed..

Introduction

Mullerian anomalies of the female reproductive tract can range between 1 in 4000 to 10000. Meyer Rokitasky Kuster Hauser syndrome occurs in 1:4000 to 1:5000 women.[1] The diagnosis represents a challenge and clinical examination is the first step in arriving at a provisional diagnosis. The presentation is usually that of primary amenorrhoea, cryptomenorrhoea, dysmenorrhoea, chronic pelvic pain, dyspareunia and difficulty in sexual function. When there is absence or cervix and upper vagina there can be hematometra without hematocolpos. Meckel’s diverticulum occurs in 2% of population.[2]

Case Report

An eighteen-year-old unmarried girl came to the outpatient department with a history of primary amenorrhea and cyclical lower abdominal and lower back pain on fixed days of the month for 2 years. The pain was of spasmodic nature, lasting for 6-7 days every month. She had no other gynecological complaints. She had attained thelarche at the age of 12 years and pubarche at the age of 13 years. She had no sister. Her mother had attained menarche at the age of 13 years. There was no history of tuberculosis, excessive weight gain, hirsutism, galactorrhoea or headache and blurring of vision and no history of bowel or bladder complaints. On examination she was moderately built and nourished. Her vital parameters were in normal limits. There was no thyroid enlargement. Breast development and secondary sexual characters were Tanner’s stage V. There was no galactorrhea. Hirsutism was not seen.  There were no skeletal anomalies. Her abdomen was soft and external genitalia were normal in appearance. There was a blind pouch of vagina of 1cm depth. Uterus was not palpable per rectally and no other pelvic masses were felt. A provisional diagnosis of mullerian agenesis was made. Her ultrasonography showed an absence of internal genital organs. The kidneys were normal. Magnetic resonance imaging showed two rudimentary uterine horns in both the adnexae with adjacent ovaries showing follicles within. Both horns showed well differentiated endometrium and myometrium. The right horn measured 2.2x1.3x2.4 and the left horn is 2.1x2.1x2.3. Cervix and upper vagina were absent. Her investigations for fitness for anesthesia showed normal results. A laparoscopy was done under general anesthesia. It showed bilateral rudimentary uterine horns each about 2x3x3 cm, right one larger than the left, with normal attached fallopian tubes and ovaries. Cervix was not visualized but a communicating fibrous ridge was seen between the two horns. Few endometriotic spots were seen over the peritoneum covering the pouch of Douglas and bilateral uterosacral ligaments and bladder peritoneum (figure 1). In view of laparoscopy findings showing pelvic endometriosis, bilateral hematometra in bilateral rudimentary horns with functioning endometrium and cervical agenesis a decision for made to excise the two horns of a bicornuate uterus. An exploratory laparotomy was done and excision of bilateral horns was carried out.  The ovaries and fallopian tubes being normal were left behind. Maeckel’s diverticulum was identified as an incidental finding (figure 2). A surgical reference was taken. As the diverticulum had a broad base it was left undisturbed. The postoperative course was uneventful and she was discharged on 5th postoperative day. In view of the presence of pelvic endometriosis, 3.75 mg leuprolide depot was given intramuscularly once a month for three months. She was asymptomatic for three months subsequently.

Figure 1. Pelvic findings: left uterine horn (LH), right uterine horn (RH), and right ovary.

Figure 2. Meckel's diverticulum (arrows).

Discussion

MRKH syndroe can be associated with anomalies of the urinary system and skeletal system.[3] Association with gastrointestinal anomalies is rare. Its association with Meckel’s diverticulum has not been reported so far.[4,5] The presence of Meckel’s diverticulum in our case was diagnosed incidentally. The patient had never been symptomatic for it. 

The management of MRKH syndrome is creation of a neovagina when the patient is ready to be sexually active. If there is a functioning uterus, though rudimentary, the treatment cannot be delayed, as that can lead to endometriosis and its complications. If there is a functional cervix, the treatment is creation of a new vagina and anastomosis of the cervix to the apex of the new vagina. However, the absence of cervix makes this choice of treatment not desirable. Repeated efforts at creating a new cervical opening often fail, necessitating a hysterectomy at the end.[6,7]

In the case presented, there were two rudimentary uterine horns at the level of the pelvic brim. The cervix was absent, and the distance from the apex of a new vagina to great to be bridged despite mobilization of the horns. Hence a hysterectomy had to be performed. A vaginoplasty for permitting sexual function was planned at a later date .

Conclusion

Though conventional treatment of MRKH syndrome is creation of a new vagina for permitting sexual function, it needs to be modified if there is hematometra of the rudimentary iterine horns and pelvic endometriosis. Absence of functional cervix in the horns necessitates hysterectomy to prevent progressiveenlargement of hematometra and associated pain. One needs to look for not only urinary tract and skeletal anomalies, but also gastrointestinal anomalies, because only then one may be able to say with convition whether gastrointestinal anomalies are or are not associated with MRKS syndrome.

A case of primary amenorrhea when encountered needs complete and thorough evaluation keeping in mind functional and structural causes for it. A plan should be formulated after proper counseling of the patient and her relatives. A radiologist and a plastic/reconstructive surgeon should also be involved in the decision making process . renal and axial skeletal anomalies should always be looked for and kept in mind prior to performing any invasive procedure. The management of such conditions should be done with an individualized approach .

References

1. The American Fertility Society classifications of adnexal adhesions, distal tubal occlusion, tubal occlusion secondary to tubal ligation, tubal pregnancies, müllerian anomalies and intrauterine adhesions. Fertil Steril. 1988 Jun. 49(6):944-55.
2. Dumper J, Mackenzie S, Mitchell P, Sutherland F, Quan ML, Mew D. Complications of Meckel's diverticula in adults. Can J Surg. 2006 Oct; 49(5): 353–357.
3. Raybaud C, Richard O, Arzim M, David M. Mayer-Rokitansky-Küster-Hauser syndrome: associated pathologies. Arch Pediatr. 2001;8:1209-1213.
4. Leijonmarck CE, Bonman-Sandelin K, Frisell J, Raf L. Meckel's diverticulum in the adult. Br J Surg 1986;73: 146-9.
5. Park JJ, Wolff BG, Tollefson MK, Walsh EE, Larson DR. Meckel diverticulum: the Mayo Clinic experience with 1476 patients (1950-2002). Ann Surg 2005;241: 529-33.
6. Rock JA, Reeves LA, Retto H, Baramki TA, Zacur HA, Jones HW., Jr. Success following vaginal creation for Mullerian agenesis. Fertil Steril. 1983;39:809–813.
7. Roberts CP, Haber MJ, Rock JA. Vaginal creation for mullerian agenesis. Am J Obstet Gynecol. 2001;185:1349–52; discussion 1352-3. doi: 10.1067/mob.2001.119075.

Citation

Manjrekar VM, Parulekar SV. MRKH Syndrome Variant With Meckel’s Diverticulum JPGO 2017. Volume 4 No. 1. Available from: http://www.jpgo.org/2017/01/mrkh-syndrome-variant-with-meckels.html

Pregnancy - A Primary Cause Of Ischemic Stroke?

Author Information

Shah A*, Chauhan AR**.

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

Abstract

Hemiparesis is unilateral paresis i.e. weakness of the entire right or left side of the body. Ischemic strokes are rare in pregnancy and may present in a setting of common causes like smoking or hypertension, or rarer causes such as postpartum cardiomyopathy, coagulopathies or vasculopathy. A 1955 study in Minnesota USA reported an incidence of 3.5 ischemic strokes per 100000 population.[1] We present a case of sudden onset hemiparesis without any previous medical illness in a pregnant patient, where other causes of hemiparesis were ruled out.

Case Report

26 years, married since 6 years, G3P2L2, 32 weeks of gestation, presented to the emergency room of our tertiary care center with sudden onset right-sided weakness and headache. She had no history of vomiting, altered sensorium, convulsions or loss of consciousness. Past medical and surgical history was not contributory. Her previous obstetric history was not significant; she had previous two uneventful pregnancies with full term normal deliveries.
On examination, the patient was conscious and oriented. Her pulse was 98 beats per minute and blood pressure was126/ 86 mm of Hg. Obstetric evaluation was unremarkable. Eye movements were normal and there was no evident facial asymmetry. Fundus examination was also normal; there was no evidence of papilledema. There were no signs and symptoms suggestive of arthritis, skin changes (malar rash) or renal involvement. Thorough neurological examination revealed:

	Right	Left
Power Upper Limb	3	5
Power Lower Limb	3	5
Tone	Normal	Normal
Plantar Reflex	Increased	Decreased

Patient was admitted and investigated. Carotid artery Doppler examination revealed normal findings. However magnetic resonance imaging (MRI) of the brain was suggestive of acute non-hemorrhagic infarct in the left posterior communicating artery territory (PCA). MR angiography revealed occlusion of the left PCA along with an aberrant right subclavian artery. Left PCA was not visualized beyond the P2 segment.
To rule out possible causes of stroke in young adults like autoimmune factors, atherosclerosis, hematological and cardiac disorders, the patient was fully investigated. Lipid profile, anti nuclear antibody (ANA), anti double stranded DNA (dsDNA), anti cardiolipin antibody (ACLA), anti phospholipid antibody (APLA), β2 glycoprotein and homocysteine levels were advised; all were negative except ANA which was weakly positive (1:80) +4, fine speckled + cytoplasmic. Complement studies (C3, C4) were also normal. Neurology and rheumatology opinions were sought; they advised medical line of management with no neurological intervention; patient was started on tab aspirin 150 mg and tab atorvastatin 40 mg daily along with daily physiotherapy. Though lipid profile was normal, atorvastatin was initiated empirically as a plaque stabilizer. Thorough cardiac evaluation was also done including 2D- echocardiography which was normal with an ejection fraction of 60 %. Routine antenatal investigations and USG were normal. During the course of her admission, fetal heart sounds were regularly auscultated and there was no adverse pregnancy event.
The patient recovered spontaneously over the course of two weeks and was discharged with power of 5/5. She followed up regularly in the antenatal clinic and had no complaints. She presented at 38 weeks’ of gestation in active labor, and had an uneventful full term normal delivery. Postnatal neurological examination revealed no abnormality. Mother and baby were discharged on the fourth postnatal day.

Discussion

Stroke associated with pregnancy is a recognized entity, wherein pregnancy itself may be responsible for the increased incidence. Stroke related to pregnancy is associated with significant morbidity and mortality. The incidence during pregnancy was reported to be 5 infarctions per 100,000 pregnancies in Glasgow, Scotland.[2] Although the risk of pregnancy-related stroke is reported to be higher among women aged 35 years and older, ischemic subtype has been associated with a younger maternal age.[3] Pregnancy specific causes of ischemic stroke include preeclampsia, eclampsia, amniotic fluid embolism, peripartum cardiomyopathy, postpartum cerebral angiopathy and choriocarcinoma.[4] A study by Bremme concluded that most arterial strokes present in the third trimester and puerperium.[5] The physiological adaptations in the cardiovascular and coagulation systems in pregnancy, which are more significant around delivery and in the peurperium, are likely to contribute to the increased risk of an ischemic stroke.[4] Due to the hypercoagulable state in pregnancy, the risk of venous stasis and consequently of thromboembolic events increases. Modifications of hemostatic factors during pregnancy are: a) an increase in procoagulant factors like Fibrinogen (Factor I), von Willebrand factor, Factors V, VII, VIII, IX, X, XII, XIII, fibrinolytic factors like plasminogen activator inhibitor 1 and 2 (PAI-1, PAI-2); b) decrease in coagulation inhibitors like protein S; c) increase in other factors like prothrombin fragment 1+2, thrombin-antithrombin complex and D-dimer.[6]
In the case presented above the patient spontaneously developed hemiparesis. As per the investigations no significant cause could be found. ANA 1:80 titer is not significant enough to cause cerebral infarction; all other reports were within normal limits. Thus in the above case we can consider pregnancy itself to be an independent principal factor and the primary cause of ischemic stroke.[6]

References

1. Wiebers DO, Whisnant JP. The incidence of stroke among pregnant women in Rochester, Minn, 1955 through 1979. JAMA. 1985; 254(21): 3055–3057. 
2. Cross JN, Castro PO, Jennett WB. Cerebral strokes associated with pregnancy and the puerperium. BMJ. 1968; 3: 214–218.
3. Kittner SJ, Stern BJ, Feeser BR, Hebel R, Nagey DA, Buchholz DW, et al. Pregnancy and the risk of stroke. N Engl J Med. 1996; 335(11): 768–774.
4. Bremme KA. Haemostatic changes in pregnancy. Best Pract Res Clin Haematol. 2003; 16(2): 153–16.
5. Jaigobin C, Silver FL. Stroke and Pregnancy. Stroke. 2000; 31(12): 2948–2951.
6. Martin PJ, Enevoldson TP, Humphrey PR. Causes of ischemic stroke in the young. Postgrad Med J.1997; 73(885): 8–16.

Citation

Shah A, Chauhan AR. Pregnancy The Primary Cause Of Ischemic Stroke. JPGO 2017. Volume 4 No. 1. Available from: http://www.jpgo.org/2017/01/pregnancy-primary-cause-of-ischemic.html

Necessary Myomectomy At Cesarean Section

Author Information

Sneha V*, Kale K**, Chauhan AR***.

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

Abstract

There has always been much reluctance among surgeons to perform myomectomy during cesarean section (CS) because of the theoretical risk of massive hemorrhage and postoperative morbidity. However, in recent times, better patient selection, better hemostatic measures and postoperative care have led to more surgeons attempting myomectomy at CS. Here we report a case of a large lower segment fibroid obstructing the delivery of the fetus, necessitating myomectomy at CS.

Introduction

Leiomyomata (fibroids) are benign smooth muscle cell tumors of the uterus and female pelvis. The incidence of fibroids during pregnancy is estimated to be 0.1 to 12.5%.[1] Most often they do not pose any problem during pregnancy; however, around 10 to 30% of pregnant women are symptomatic and have pain and pressure symptoms due to complications like torsion or red degeneration. There could also be intrapartum complications like obstructed labor, postpartum hemorrhage and subinvolution of the uterus. [1] With increase in maternal age at pregnancy, the incidence of fibroids encountered during cesarean section is also on the rise and poses a management challenge to the surgeon.

Case Report

A 36-year-old primigravida conceived after infertility treatment, was referred to our tertiary care center at 33 weeks of gestation in view of ultrasonography (USG) showing severe oligohydramnios with intrauterine growth restriction (IUGR) and large lower uterine segment intramural fibroid of 8 x 10 cm. Patient had undergone a cervical cerclage at 20 weeks of gestation in view of short cervical length.

All routine antenatal investigations were normal. On examination, her pulse was 86 beats per minute; her BP was 120/80 mm Hg. On abdominal examination, uterus was smaller than expected for her gestation age and was only 28 weeks’ size; fetus was in cephalic presentation, with normal fetal heart sounds. The fibroid was palpable anteriorly on the left side, approximately 8 cm in size; it was non-tender. On per vaginal examination, cervical os was closed and uneffaced, knot of encerclage was in place, and pelvis was adequate for the fetus.

USG performed at our center showed severe oligohydramnios (amniotic fluid index of 0-1) with IUGR and absent diastolic flow on obstetric Doppler. There was an intramural fibroid of 8 x 10 cm in the anterolateral wall on the left side.

In view of these findings and history of infertility with large lower segment fibroid, she was scheduled for an elective lower segment CS. Cesarean myomectomy was anticipated hence patient and her relatives were counseled about the same and consent was taken. Adequate blood was cross matched and kept ready.

Patient was operated under regional anesthesia; abdomen was opened through a vertical midline incision till the uterus was reached. In situ, there was evidence of a intramural fibroid of approximately 8 -10 cm diameter at the junction of the upper and lower uterine segment with the uterovesical fold of peritoneum running midway over the fibroid. The uterovesical fold was dissected and the bladder was mobilized inferiorly. The lower edge of the fibroid was palpated, and a transverse incision was made below the lower margin of the fibroid in the lower segment. Amniotomy was done; however, the fetal head which was small could not be delivered due to obstruction by the fibroid. Hence, the decision for cesarean myomectomy was taken. Going through the upper margin of the uterine incision, the fibroid was rapidly dissected and enucleated by sharp and blunt dissection in the usual manner, as seen in Figures 1 and 2.

Figure 1. Fibroid being enucleated. A – The fibroid, B – Upper edge of uterine incision.

Figure 2. Uterine and fibroid cavities after myomectomy. A – Myoma bed. B - Uterine cavity. C – Upper edge of uterine incision. D – Lower edge of uterine incision.

Once the myomectomy was performed, a live male child of 1.14 kg was easily delivered. The newborn had an Apgar score of 9 and required neonatal intensive care unit admission. Base of the myoma was sutured in three layers, as seen in Figure 3, followed by suturing of uterine incision and closure of abdomen as usual in layers. The patient tolerated the procedure well and postoperative course was uneventful. There was no postpartum hemorrhage or infective complications. Blood transfusion was not required. She was discharged from our center on postoperative day 5. On further follow up at day 15, there was no evidence of subinvolution or secondary postpartum hemorrhage.

Figure 3. Base of the fibroid sutured. A – Sutured myoma bed. B - Upper edge of the incision. C - Lower edge of the incision.

Discussion

Fibroids encountered during cesarean section pose a therapeutic dilemma. Myomectomy has traditionally been discouraged during cesarean section due to fear of excessive bleeding from an obstetric uterus. However, recently, many case reports and studies in literature have reliably shown that cesarean myomectomy in properly selected patients and in experienced hands, is a relatively safe procedure. Studies have demonstrated no significant increase in the rates of intraoperative and postpartum hemorrhage, blood transfusion, infections, duration of surgery and length of hospital stay in patients undergoing cesarean myomectomy as compared to those undergoing cesarean section alone. [2, 3] Several techniques have been described for achieving hemostasis during cesarean myomectomy. These include- three layered suturing of the base of the fibroid as was done in our case, application of tourniquet at the base of the broad ligament and the infundibulopelvic ligament, bilateral uterine artery ligation, prophylactic high dose oxytocin infusion in intra and postoperative periods or a combination of these methods.[4]

Most important factors influencing decision of myomectomy during CS include type, location and to some extent, size of the fibroid. Large anterior wall fibroids obstructing delivery of baby necessitate myomectomy, as was evident in our case. Some authors have advocated routine removal of all anterior wall uterine fibroids during cesarean section.[5] Fibroids which are subserous, pedunculated, those causing difficulty in closure of uterine incision and those with unusual intraoperative appearance are the ones that are most frequently removed during cesarean myomectomy.[4] As far as the size is concerned, fibroids as small as 1 cm to as large as 40 cm have been successfully removed during cesarean section in reported cases, the average size being 6– 10 cm.[3,6]

The main advantage of cesarean myomectomy is its cost effectiveness and avoidance of a future operation which is important especially in low-resource settings. [4] This is applicable to many parts of our country where patients are not likely to follow up and there are limited healthcare facilities. Studies have also confirmed no significant adverse effect of cesarean myomectomy on future fertility and/or subsequent pregnancy outcome.[7] Cesarean myomectomy when performed through the lower uterine segment, increases the chances of vaginal birth after cesarean section (VBAC) in subsequent pregnancies. When compared with interval myomectomy, the scar integrity following cesarean myomectomy has been shown to be better (assessed with ultrasound/ at the time of repeat cesarean section in subsequent pregnancies). [4, 8]

Conclusion

Thus, cesarean myomectomy in selected cases and in experienced hands may be considered safe and effective. By avoiding a future surgery and additional costs on healthcare system, it may be especially relevant in low resource settings like India. However, it should be noted that most studies published in literature are retrospective in nature and results from more extensive, randomized controlled trials are awaited before cesarean myomectomy is routinely recommended.

References

1. Cooper NP, Okolo S. Fibroids in pregnancy – common but poorly understood. Obstet Gynecol Surv. 2005; 60(2):132-8.
2. Park BJ, Kim YW. Safety of cesarean myomectomy. J Obstet Gynaecol Res. 2009; 35(5): 906-11.
3. Kwon DH, Song JE, Yoon KR, Lee KY. The safety of cesarean myomectomy in women with large myomas. Obstet Gynecol Sci. 2014; 57(5): 367-72.
4. Awoleke JO. Myomectomy during caesarean birth in fibroid-endemic, low-resource settings. Obstet Gynecol Int. 2013; 2013: 520834.
5. Kwawukume EY. Myomectomy during cesarean section. Int J Gynaecol Obstet. 2002; 76(2):183–4.
6. Ma PC, Juan YC, Wang ID, Chen CH, Liu WM, Jeng CJ. A huge leiomyoma subjected to a myomectomy during a cesarean section. Taiwan J Obstet Gynecol. 2010; 49(2): 220-2.
7. Adesiyun AG, Ojabo A, Durosinlorun-Mohammed A. Fertility and obstetric outcome after caesarean myomectomy. J Obstet Gynaecol. 2008; 28(7): 710-712.
8. Cobellis L, Messalli EM, Stradella L, Pecori E, Gioino E, De Lucia E et al. Myomectomy during cesarean section and outside pregnancy. Different outcomes of scars. Minerva Ginecologica. 2002; 54(6):483-6.

Citation

Sneha V, Kale K, Chauhan AR. Necessary Myomectomy At Cesarean Section. JPGO 2017. Volume 4 No. 1. Available from: http://www.jpgo.org/2017/01/necessary-myomectomy-at-cesarean-section.html

Multiple Parasitic Leiomyomas After Laparoscopic Myomectomy: An Unusual Complication Of Power Morcellation

Author Information

Shah NH*, Kale K**, Paranjpe S**, Nagarkatti RS*** 

(* Director, Vardann Hospital: Centre of Excellence in Laparoscopy and IVF, Mumbai, ** Ex-fellow, Vardann Hospital: Centre of excellence in laparoscopy and IVF, Mumbai, *** Director, Ashirwad Hospital, Mumbai, India.)

Abstract

Previous laparoscopic myomectomy with use of a power morcellator is being identified as an important risk factor for development of parasitic leiomyomas. With increasing use of laparoscopy, the incidence of this rare complication is on the rise. We present a 28 year old nulliparous female who came 3 years after a laparoscopic myomectomy with a large left adnexal mass, mimicking a pedunculated exophytic uterine fibroid. On laparoscopy, however, a large parasitic mass was found in pouch of Douglas, separate from the uterus, along with many scattered masses all of which were carefully removed; histopathological examination confirmed leiomyomas.  This case report is presented to increase the awareness of this rare entity among laparoscopic surgeons which will allow them to take appropriate measures to prevent its occurrence.

Introduction

Parasitic leiomyoma is a rare benign variant of uterine leiomyoma which has lost its connection to the uterus and is dependent on another organ or tissue for its main blood supply.[1] In recent years, there have been increasing reports of parasitic leiomyomas developing after laparoscopic hysterectomies and myomectomies involving the use of electric power morcellator, believed to arise from the small tissue fragments that are left behind after morcellation.[1-3] Literature on these ‘iatrogenic parasitic myomas’ is limited to case reports and few small case series. This case report is an attempt to further elaborate on the etiopathogenesis, clinical presentation, diagnosis and management of this rare complication and to contribute to the growing body of literature.

Case Report

A 28 year old nulligravida, married since 5 years with history of primary infertility presented to the outpatient department with complaints of recent onset mild to moderate dull aching pain in lower abdomen. Her menstrual cycles were regular with average blood flow. There was no history of leucorrhea, fever, associated urinary or gastrointestinal complaints. Patient had undergone a laparoscopic myomectomy three years ago for a right lateral wall large intramural fibroid (8 x 6cm). Electric power morcellator was used to retrieve the fibroid and histopathology had confirmed benign leiomyoma. Post operatively, patient had an uneventful recovery.

General and systemic examination revealed no significant abnormality. Per abdomen and per speculum examination were within normal limits. On per vaginal examination, a firm mass with restricted mobility, approximately 6 cm in size was noted in the posterior and left fornix. On trans-abdominal ultrasonography, there was a 7 x 6 cm left adnexal lesion, predominantly hypoechoic with central cystic change. Findings were confirmed on transvaginal sonography (TVS) and a diagnosis of pedunculated exophytic uterine fibroid with central degenerative changes was made. No other masses or lesions were identified.

After obtaining informed written consent, laparoscopy was performed with three ports: one primary supra umbilical port (10mm) and two accessory 6 mm ports on left side. At laparoscopy, multiple masses (total seven in number) with sizes ranging from 1 to 5 cm, grossly resembling leiomyomas, were seen attached to the anterior abdominal wall (Figure 1A),  omentum (Figure 1B) and the peritoneal adhesion bands (Figure 1C and D).

Figure 1 (A)  Parasitic leiomyoma attached to anterior abdominal wall. (B) Arrow showing the same parasitic leiomyoma with its pedicle attached to anterior abdominal wall. Circle showing parasitic myomas attached to the omentum. (C) and (D) Circles showing parasitic myomas attached to the peritoneal adhesion bands.

A large (approximately 6.5 cm) multilobulated mass was found in the pouch of Douglas, separate from the uterus and lateral to the left uterosacral ligament. The mass was firmly attached to the posterior leaf of broad ligament and was deriving its blood supply from left uterine artery (Figure 2A and B). Uterus appeared normal without any evidence of leiomyoma.

Figure 2 (A) Arrow showing parasitic leiomyoma in pouch of Douglas. Left ovary is seen separately. (B) Leiomyoma after removal from pouch of Douglas.

All the masses were carefully dissected off their respective attachments using bipolar electrocautery and scissors. Retrieval was achieved by morcellation of the masses in an endobag to avoid scattering of particles into the peritoneal cavity. Abdominal cavity was irrigated with around 3 liters of warm normal saline to ensure no particle was left behind. There were no intra/post operative complications. Diagnosis of parasitic leiomyoma was established after histopathologic confirmation. Patient was discharged on the third post operative day and is currently under treatment for infertility.

Discussion

Parasitic leiomyomas were first described by Kelly and Cullen in 1909. They were thought to be spontaneously arising from pedunculated subserosal fibroids which have outgrown their uterine blood supply and have become dependent on other organs and tissues for nourishment.[3] In recent years, it is postulated that seeding of peritoneal cavity with multiple small fragments of uterine and/or myoma tissue during morcellation may lead to implantation of these fragments over abdomino-pelvic organs, omentum, peritoneum etc. which develop into parasitic myomas, also termed as ‘iatrogenic parasitic myomas’.[1-3] Molecular genetic analyses have also confirmed that these iatrogenic parasitic myomas originate from the same leiomyomas of the uterus that were resected at the primary surgery.[4]

The true incidence of iatrogenic parasitic myomas developing after laparoscopic morcellation procedures is unknown. Leren [5] has reported an incidence of 0.12%, whereas Cucinella[6] reported a prevalence of 0.9%. Many patients with iatrogenic parasitic myomas remain asymptomatic; others present with non specific symptoms such as abdominal pain, mass sensation, deep dyspareunia, abdominal distension.[1, 2] Depending on the site of occurrence, they may present with constipation, dyschezia (sigmoid colon); urinary frequency (bladder); or right lower quadrant pain (appendix).[3] Narsimhulu reported a case of torsion of an iatrogenic parasitic myoma presenting with acute abdomen.[7]  Dan reported a case of iatrogenic parasitic myoma presenting with generalized peritonitis and sepsis.[8] The only symptom attributable to parasitic fibroids in our patient was mild to moderate lower abdominal pain.

In the present case, the patient presented 3 years after the initial surgery. In various studies, the time interval between the initial laparoscopic surgery and the detection of parasitic myomas at second surgery ranges from 2- 108 months. This variability could be attributed to differences in circulating hormone levels and receptor status of the myomas which affect onset of clinical symptoms and potential growth patterns.[1, 2]

The location of iatrogenic parasitic myomas is variable, but are more commonly located in the pelvis which is the dependent part of the abdomen.[7] They are also found in various other sites such as port entry site in the anterior abdominal wall, peritoneum, omentum, bowel, mesentery, appendix, bladder, rectus abdominis muscle, or subcutaneous tissue.[1,2] These are also the sites where small fragments of tissue formed during morcellation are likely to lodge and be missed. 

Because of their rarity and non specific symptoms, diagnosis of parasitic myomas is mostly incidental at the time of surgery for other indications.[3] Ultrasonography is a useful imaging modality for diagnosis. Trans-vaginal ultrasonography (TVS) is of great help in case of parasitic myomas in the cul de sac and broad ligament, as it permits clear visual distinction between the mass and the uterus and ovaries. In our case, however, TVS could not identify the myoma separately, probably due to a low index of suspicion. MRI seems particularly valuable in the diagnosis of parasitic myomas due to its superb contrast resolution and multiplanar capabilities and hence should be done in cases with high index of suspicion. [9]

Dilemmas exist regarding the treatment of iatrogenic parasitic myomas. According to few authors, surgical treatment is mandatory in symptomatic cases, in cases with progressive growth of lesions and asymptomatic myomas identified incidentally during surgical procedures for other indications.[6] However, there have been reports of parasitic myomas requiring extensive and complicated surgeries, especially where they are firmly attached to bowel or mesentery leading to intestinal perforation.[5, 8] Medical treatment in the form of intramuscular gonadotropin releasing hormone has been tried by Kumar et al. which relieved symptoms but was not effective in reducing the size of the parasitic myoma.[10] Thus, further research is necessary to determine the optimum line of management of iatrogenic parasitic myomas.

For prevention of this rare complication of tissue morcellation during laparoscopic surgeries, several authors insist on identification and complete removal of all tissue fragments from the abdomino-pelvic cavity, altering the patient position from Trendelenburg to reverse Trendelenburg and copious irrigation of abdomen and pelvis with normal saline.[2-8] Morcellating the tissue in an endobag should be considered as this may prevent the scattering of small fragments.[7] In the present case too, morcellation was carried out in an endobag followed by irrigation of abdominopelvic cavity to ensure that no fragment was left behind.

Conclusion

The present case reinforces the belief that prior laparoscopic uterine surgery with tissue morcellation using power morcellator is a risk factor for development of parasitic myomas. Clinicians should be aware of this entity and all measures necessary to prevent this rare complication must be taken during initial surgery.

References

1. Erenel H, Temizkan O, Mathyk BA, Karataş S. Parasitic myoma after laparoscopic surgery: a mini-review. J Turk Ger Gynecol Assoc 2015; 16: 181-6.
2. Huang PS, Chang WC, Huang SC. Iatrogenic parasitic myoma: a case report and review of the literature. Taiwan J Obstet Gynecol 2014; 53(3): 392-6.
3. Kho KA, Nezhat C. Parasitic myomas. Obstet Gynecol 2009;114(3): 611–5.
4. Miyake T, Enomoto T, Ueda Y, Ikuma K, Morii E, Matsuzaki S et al. A case of disseminated peritoneal leiomyomatosis developing after laparoscope-assisted myomectomy. Gynecol Obstet Invest 2009; 67(2): 96-102.
5. Leren V, Langebrekke A, Qvigstad E. Parasitic leiomyomas after laparoscopic surgery with morcellation. Acta Obstet Gynecol Scand 2012; 91(10): 1233-6.
6. Cucinella G, Granese R, Calagna G, Somigliana E, Perino A. Parasitic myomas after laparoscopic surgery: an emerging complication in the use of morcellator? Description of four cases. Fertil Steril 2011; 96(2): e90-6.
7. Narsimhulu DM, Eugene E, Sumit S. Torsion of an iatrogenic parasitic fibroid related to power morcellation for specimen retrieval. J Turk Ger Gynecol Assoc 2015; 16(4): 259-62.
8. Dan D, Harnanan D, Hariharan S, Maharaj R, Hosein I, Naraynsingh V. Extrauterine leiomyomata presenting with sepsis requiring hemicolectomy. Rev Bras Ginecol Obstet  2012; 34(6): 285-9.
9. Fasih N, Prasad Shanbhogue AK, Macdonald DB, et al. Leiomyomas beyond the uterus: unusual locations, rare manifestations. Radiographics 2008; 28(7):1931–48.
10. Kumar S, Sharma JB, Verma D, Gupta P, Roy KK, Malhotra N. Disseminated peritoneal leiomyomatosis: an unusual complication of laparoscopic myomectomy. Arch Gynecol Obstet 2008; 278(1): 93-5.

Citation

Shah NH, Kale K, Paranjpe S, Nagarkatti RS. Multiple Parasitic Leiomyomas After Laparoscopic Myomectomy: An Unusual Complication Of Power Morcellation. JPGO 2017. Volume 4 No. 1. Available from: http://www.jpgo.org/2017/01/multiple-parasitic-leiomyomas-after.html

Atypical Posterior Uterine Rupture

Author Information

Dharmadhikari M*, Daigavane M**, Samant PY***, Chaudhari HK***. 

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

Abstract

Vaginal birth after caesarean section (VBAC) is well accepted with proper patient selection. Uterine rupture during VBAC is one of the most dreadful complications. Rupture can occur at a site away from that of previous scar. If there are no prominent signs and the rupture goes undetected, it may have catastrophic consequences. A high index of suspicion is of paramount importance. Fortunately , such cases of atypical uterine rupture are extremely rare. We present a case of atypical uterine rupture of the posterior uterine wall, incidentally detected during cesarean section for fetal distress.

Introduction

Uterine rupture is a known complication of vaginal birth after cesarean section (VBAC) with a rate of 0.22-0.74%, with serious maternal and perinatal outcomes. It is conventionally believed to be due to compromised integrity of the scarred muscle. The incidence of rupture during VBAC is the lowest with spontaneous onset of labor as compared to induction or augmentation of labor. Prostaglandins used for cervical ripening  and oxytocin for induction and augmentation of labor increase the risk of uterine rupture by 2-3 fold. Uterine rupture in an unscarred uterus occurs in around once in 10,000 deliveries.[1] Atypical uterine rupture occurring at a site away from the previous scar is extremely rare.

Case Report

A 25 year old second gravida with previous one LSCS with 40 weeks and 4 days gestation was admitted in early labor. The patient had regular antenatal visits in this pregnancy. Previous section was done in view of anhydramnios at a private hospital at term gestation 3 years ago. There was no other high risk factor. Patient did not give any other relevant history, nor was there documentation of any other obstetric intervention. Mode of delivery with risks and benefits associated with VBAC were discussed with her and the partner. They had opted for vaginal delivery. Ultrasonography had revealed an upper segment placenta. Liquor was adequate. Estimated baby weight was approximately 2.5 kg.

The patient had a normal labor progression and was fully dilated without oxytocin. However, after full dilatation of cervix for one hour, the patient developed fetal distress with late decelerations up to 80 beats per minute. Her vital parameters were stable. There was no scar tenderness. On vaginal examination os was fully dilated, fully effaced with vertex presentation, station was at 0 and had not receded, occiput was at 9 o’ clock, membranes were absent, liquor was clear. Decision for emergency LSCS in view of fetal distress in second stage of labor was taken. 

Surgery commenced under spinal anesthesia. On laparotomy, a small amount of blood was noticed anterior to uterovesical peritoneum. Scar of previous LSCS was intact. Baby’s Apgar score at 1 min and 5 min was 9/10. Placenta and membranes were removed and the uterus was closed in situ. After closure, blood was seen pooling in the paracolic gutters. Hence the uterus was exteriorized and a posterior full thickness oblique uterine rupture of 6 cm in lower uterine segment was seen extending to the cervico-vaginal junction on the left side with active bleeding. The lacerated left uterine artery was seen bleeding. The rupture was repaired in one layer with polygalactic acid suture in continuous locking stitch. Left uterine artery was ligated and hemostasis was achieved. The ureters were traced and found to be away from sutures.

Estimated blood loss was 2800 ml. Intraoperatively 3 units of packed cells and 4 units of fresh frozen plasma were transfused. Intraperitoneal drain was placed and abdomen was closed after confirming hemostasis. Per speculum examination was done after the procedure to rule out any unnoticed vaginal extension of uterine tear. Anterior cervical lip was seen. Posterior lip was pulled up due to suturing. A centimeter long posterior vaginal wall laceration was noted away from the cervix, which was sutured. The patient was stable in the postoperative period. Later on further enquiry, she revealed history of dilatation and curettage for incomplete abortion 2 years before this pregnancy. She was discharged on day 7 with a 2.6 kg male child.

Figure 1: Posterior uterine rupture.

                           

Figure 2: Repair of posterior uterine rupture.

Discussion

In a scarred uterus, the vast majority of uterine dehiscence and ruptures involve the previous uterine scar due to fibrosis and lack of elasticity. In some cases, the posterior wall of the uterus may be excessively stretched and thinned due to the rigid anterior uterine scar that prevents equal stretching, and may cause atypical uterine rupture of healthy tissue. The site of uterine rupture in such situations is unpredictable.[2] This is comparable to sacculation  in which a part of the posterior uterine wall softens and bulges like an aneurysm allowing growth of the fetus into the abdomen with increased risk of uterine rupture.

Prior unrecognized uterine perforation during dilatation and curettage or during insertion of IUCD, may magnify the risk for atypical uterine rupture especially in the setting of excessive uterine distension as myometrium is focally weakened. Previous curettage and myometrial injury were probably the cause of rupture in our case; however patient had not revealed history of curettage.

Factors which predispose to uterine rupture during VBAC are induction of labor, augmentation of labor, multiparity, uterine anomalies, endometritis, prolonged labor, incoordinate action, fetal malpresentation or malposition, very early repeat cesarean section (less than 2 years) and morbid adherence of placenta. [1]

In cases of posterior uterine rupture in patients of VBAC published between 1997 and 2007 [3–7]  a case of previous twin pregnancy was included.[7] It is possible that uterine hyperdistension predisposes weak thinned out musculature to atypical uterine rupture. Hawe et al and. Figueroa et al reported two cases of posterior uterine rupture with the use of prostaglandin for induction of labor.[3,5] However in our case there was no use of oxytocics or prostaglandins. Singhal et al and Majumdar et al attributed atypical uterine rupture to fetal malposition and malpresentation with prolonged labor.[6,7] Malposition may alter the distribution of the contractile forces in labor and may cause  dystocia; while certain malpresentations can cause incoordinate uterine action and obstruction, which may also precipitate atypical uterine rupture.[2]

The first sign of posterior uterine rupture in our case was an abnormal cardiotocogram (CTG) with persistent fetal bradycardia and  arrest of labor; however there was no suspicion of rupture. CTG abnormalities are associated with 55–87% of uterine ruptures.[8] Other recognizable signs of uterine rupture include receding of the presenting part and inefficient contractility.[1] In our  case maternal vital parameters were stable despite massive uterine rupture, which may have been due to the presenting part compressing the rupture site and in turn acting as a temporary tamponade with active bleeding occurring only after delivery of the baby.

Conclusion

Posterior uterine rupture may occur without conventional signs like uterine tenderness.  Detailed elicitation of obstetric history, close monitoring during labor and a high index of suspicion for uterine rupture especially in cases  with uterine overdistension, previous operative interventions on the uterus, fetal malpresentation go a long way in ensuring good maternal and perinatal outcome.

References

1. RCOG Green Top 45. Birth After Previous Cesarean Section. Green RCOG.2007.
2. Navaratnam K, Ulaganathan P, Akhtar MA, Sharma SD, Davies MG. Posterior uterine rupture causing fetal expulsion into the abdominal cavity: a rare case of neonatal survival. Case Reports in Obstetrics and Gynecology. 2011; Article ID 426127. Hindawi Publishing Corporation. doi:10.1155/2011/426127
3. Hawe JA, Olah KS. Posterior uterine rupture in a patient with a lower segment caesarean section scar complicating prostaglandin induction of labour. BJOG.1997; 104(7): 857–8. 
4. Wang PH, Yuan CC, Chao HT, Yang MJ, Ng HT. Posterior uterine wall rupture during labour. Hum Reprod. 2000; 15(5): 1198–9. 
5. Figueroa R, Garry D, Mackenzie AP. Posterior uterine rupture in a woman with a previous cesarean delivery. Journal of Maternal- Fetal and Neonatal Medicine. 2003; 14(2):130–1. 
6. Singhal SR, Agarwal U, Sangwan K, Khosla A, Singhal S. Intrapartum posterior uterine wall rupture in lower segment cesarean section scarred uterus. Acta Obstet Gynecol  Scand. 2005; 84 (2):196–7. 
7. Majumdar S, Warren R, Ifaturoti O. Fetal survival following posterior uterine wall rupture during labour with intact previous caesarean section scar. Arch Gynecol Obstet. 2007; 276 (5): 537–40.
8. Guise JM, McDonagh MS, Osterweil P, Nygren P, Chan BK, Helfand M. Systematic review of the incidence and consequences of uterine rupture in women with previous caesarean section. BMJ. 2004; 329(7456):19–25.

Citation

Dharmadhikari M, Daigavane M, Samant PY, Chaudhari HK. Atypical Posterior Uterine Rupture. JPGO 2017. Volume 4 No. 1. Available from: http://www.jpgo.org/2017/01/atypical-posterior-uterine-rupture.html

Spontaneous Hemoperitoneum Secondary To Warfarin Toxicity: Learning The Balancing Act

Author Information

Bhosle S*, Mhaskar P**, Honavar P***, Samant PY****.

(*Second Year Resident, **Senior Registrar, ***Assistant Professor, ***Additional Professor, Department of Obstetrics and Gynecology, Seth G.S. Medical College and K.E.M. Hospital, Mumbai, India).

Abstract

Spontaneous hemoperitoneum is presence of intra-abdominal hemorrhage from non-traumatic cause. Warfarin is a known entity to cause hemoperitoneum which can be life threatening. Warfarin, a coumarin derivative, is a commonly used and safe drug in the prevention of thromboembolic events; its toxicity is dose dependent. Management of unexpected bleeding poses a difficulty in controlling International Normalized Ratio (INR) while avoiding thrombosis. This is a case report of spontaneous hemoperitoneum secondary to warfarin toxicity in a case of rheumatic heart disease with choked valve.

Introduction

Warfarin is used in conditions like mechanical heart valves, atrial fibrillation, embolic events, cerebral ischemic events and venous thromboembolism. Each condition though requires a different level of anticoagulation. It is life saving in cases of choked heart valve. With warfarin toxicity, spontaneous hemoperitoneum can occur or there can be bleeding in the retroperitoneal space.[1] INR level above therapeutic range is the most important risk factor for warfarin- induced major bleeding, independent of the indication for therapy, with the risk increasing with INR > 3.[2] Here we present a case of intraperitoneal hemorrhage due to warfarin toxicity with choked valve adding to difficulties in the management.

Case Report

24 year old patient with history of two spontaneous abortions presented to us with vomiting and acute abdominal pain. She had undergone mitral valve replacement for severe mitral stenosis 2 years ago and was on warfarin 5 mg once a day since then.

In her recent pregnancy, she had been admitted in cardiac ICU for thrombosed mitral valve in first trimester. Her INR then was 1.42. She was started on unfractionated heparin and inj. streptokinase. In the course of this admission, she aborted spontaneously. She remained admitted for nearly 45 days. Her INR was maintained around 3. Echocardiogram showed adequate movement of the affected valve leaflets. The patient was discharged on tab warfarin 5 mg when she was stable. 

Patient presented to us on the day after discharge with acute pain in abdomen and pelvis. There was no vaginal bleeding, vomiting, foul discharge. She was afebrile, had tachycardia, her blood pressure was 100/60 mm Hg. Marked pallor was noted. Abdominal examination revealed guarding and tenderness with slight abdominal distension. Ultrasonography (USG) showed ill-defined non homogenous adnexal mass of about 5 cm diameter. Computerised axial tomography scan (CT scan) of abdomen and pelvis revealed ill-defined collection in pelvis suggestive of hemoperitoneum. Her investigations are listed in the table below. After admission, cardiologists omitted warfarin and started Inj. heparin 25,000 units in 500 ml of normal saline, 1000 units/ hour at the rate of 20 ml/ hour.

DAY	1	2	3	5	7
Hemoglobin  (Hb)	8.4	6	8.1	3.8	8.5
Prothrombin time (PT)	41.2	62	37.4	63.9	29.6
International normalized ratio (INR)	3.61	5.54	3.23	6.45	2.47
PCV transfusion	1 unit	2 unit	1 unit	4 units	1 unit
Blood products		4 FFP 4 cryoprecipitate		8 FFP 4 cryoprecipitate 4 platelets

When her INR increased to 6.45 and abdominal girth increased significantly, cardiology reference was urgently re-sought and they advised correction of the same with transfusion of fresh frozen plasma (FFPs) and blood. She was kept nil by mouth, was transfused with 4 units packed cell volume and FFPs, electrolyte imbalance was corrected over 4 - 5 days. Her hemoglobin increased to 8.5, INR reduced to 2.47, abdominal girth reduced. Patient was started on tab warfarin 2 mg daily after 7 days of admission with inj heparin 25000 unit / day. Heparin was omitted completely when her INR reached therapeutic range. On the day of discharge her hemoglobin was 10.8 and INR 2.06. She was symptomatically better and the hemoperitoneum had resolved.

Discussion

Pregnancy itself is a hypercoagulable state. It is an adaptive mechanism for preventing postpartum bleeding.[3] The risk of thrombosis or embolism increases when combined with additional underlying hypercoagulable states. Pregnancy changes the plasma levels of many clotting factors, such as fibrinogen. Thrombin levels increase. Protein S, an anticoagulant, decreases.[4] However, protein C and antithrombin III, , the other major anticoagulants, remain constant. Increased levels of  plasminogen activator inhibitor-1 (PAI-1) and plasminogen activator inhibitor-2 (PAI-2), synthesized from the placenta can cause impaired fibrinolysis.[5] Mechanical valves carry the risk of valve thrombosis which is increased during pregnancy. In a large review, this risk was 3.9 % with oral anticoagulants (OACs) throughout pregnancy, 9.2% when heparin was used in the first trimester and OACs in the second and third trimester, and 33% with heparin use throughout pregnancy.[6] The type and location of the prosthetic valve and other factors like history of prior thromboembolic event, atrial fibrillation, prosthesis in mitral position, and multiple prosthetic valves, determine the risk of valve thrombosis and thromboembolic events. Spontaneous abortion is the most frequent fetal complication associated with pregnancy in women with mechanical heart valves (28%).[7] Warfarin and heparin both carry this risk. 

Warfarin is a coumarin oral anticoagulant used for prophylaxis as well as treatment of thromboembolism. Warfarin treatment depends upon interaction between physiological, environmental and genetic factors.[8] Cyp2C9 liver enzyme is involved in warfarin metabolism. Two common allelic variants associated with reduced enzyme activity are CYP2C9*2 and CYP2C9*3.  Persons who inherit one or two copies of CYP2C9*2 or CYP2C9*3 are more sensitive to warfarin as compared to others and they would require lower doses of warfarin and are at a greater risk of hemorrhage with warfarin initiation.[9] Vitamin K epoxide reductase complex subunit-1 (VKORC 1) is the site of action of warfarin. It is the major allele in Asian populations, and may be a contributing factor for lower warfarin dosing requirements often observed in Indian patients. Inherited differences in VKORC 1 affect warfarin efficacy. Warfarin also inhibits synthesis of natural anticoagulants proteins C and S thus results in high rates of bleeding. Warfarin has a narrow therapeutic index, hence physicians prescribing this drug have to adjust the dose in such a way that it achieves effective thrombus prevention for whatever indication it is prescribed but also avoids fatal dose related toxicity.

In our case, spontaneous intraperitoneal hemorrhage caused intestinal irritation and thus vomiting and abdominal pain. To diagnose hemoperitoneum, USG and CT scan can be done. CT scan plays an important role as it is a sensitive method defining both site and extent of hemorrhage. In our case of anticoagulant induced hemorrhage, nasogastric decompression, bowel rest, correction of electrolyte disturbances, blood transfusion for anemia correction and correction of coagulopathy with FFP, was our treatment strategy. This patient being a case of heart disease (with mechanical valve replacement) had the critical need of ongoing anticoagulation hence heparin was given as a temporary measure, and to be safe oral anticoagulation was continued after resolution of hematoma with regular monitoring of INR. To prevent similar catastrophic events, regular systemic examination and monitoring of INR is required and should be kept in the range of 2.0 - 3.5 for optimum outcome and lesser morbidity.[10]

Conclusion

Warfarin related toxicity can be treated by withholding the drug and transfusion of FFP and PCV and readjustment of dose. The only way to prevent such an incident is careful monitoring of INR to prevent over-dosage, drug interactions and regular follow up of patient with INR report. It is desirable to do genetic mutation study of above two genes before starting warfarin to avoid toxicity. The science of balancing the patient symptomatology and maintaining INR within the target range, continuing anticoagulation for the indication by switching over to heparin followed by resuming warfarin once favorable, is a challenging job and was a lesson learned.

References

1. Schattner A, Kozack  N, Friedman J. Idiopathic Spontaneous hemoperitoneum, Arch Intern Med. 2001;161(7):1009 -10.
2. Schulman S, Beyth RJ, Kearon C, Levine MN. Hemorrhagic complications of anticoagulant and thrombolytic treatment: American College of Chest Physicians Evidence-Based Clinical Practice Guidelines (8th edition). Chest 2008;133(6 Suppl): 257S-298S.
3. Gresele P, Fuster V, Lopez JA, Page CP, Vermylen J. (Eds). Platelets in hematologic and cardiovascular disorders: A clinical handbook. 2008; Cambridge University Press, UK. 
4. de Boer K, ten Cate JW, Sturk A, Borm JJ, Treffers PE. Enhanced thrombin generation in normal and hypertensive pregnancy. Am J Obstet Gynecol 1989;160 (1):95–100.  
5. Brenner B. Hemostatic changes in pregnancy. Thromb Res 2004;114(5-6): 409-14.
6. Chan WS, Anand S ,Ginsberg JS. Anticoagulation of pregnant women with mechanical heart valves: a systemativ review of the literature. Arch Intern Med 2000;160(2):191-6.
7. Ashour ZA, Shawky HAF, Hussein MH. Outcome of pregnancy in women with mechanical valves. Texas Heart Institute Journal. 2000; 27(3): 240-5.
8. Herman D, Locatelli I, Grabnar I, Peternel P, Stegner M, Mrhar A et al. Influence of CYP2C9 polymorphisms, demographic factors and concomitant drug therapy on warfarin metabolism and maintenance dose. Pharmacogenomics J. 2005; 5(3):193-202.
9. Redman AR. Implications of cytochrome P450 2C9 polymorphism on warfarin metabolism and dosing. Pharmacotherapy 2001; 21(2): 235-42. 
10. Lacy CF, Armstrong LL, Goldman M, Lance LL. (Eds.) Drug information handbook 2001-2002. 9th ed. Cleveland: Lexy-Comp, 2001, pp1287-1291.

Citation

Bhosle S, Mhaskar P, Honavar P, Samant PY. Spontaneous Hemoperitoneum Secondary To Warfarin Toxicity: Learning The Balancing Act. JPGO 2017. Volume 4 No. 1. Available from: http://www.jpgo.org/2017/01/spontaneous-hemoperitoneum-secondary-to.html