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Volume 5 Issue 1, January 2018

Editorial

Parulekar SV

We are happy that we have successfully completed four years of publication of the journal. We feel that it is a significant achievement, as all work is done by clinicians taking out time from their private time, spending their own money, and not taking help of any professional publishing house. We have managed to keep access to the journal free and we will keep it so in future too. A rising readership of the journal is reward enough for us. Our new year resolution is to try and make the journal even better and more useful to our readers.

Prior to invention of electronic fetal heart rate (FHR) monitors, intensive intermittent FHR auscultation was done. Electronic monitors made the process convenient. The principle underlying the process was that identification of changes in FHR helped diagnose fetal well being or its compromise early. This was expected to reduce fetal hypoxic morbidity and intrapartum deaths. In 1991 clinicians started questioning the usefulness of FHR monitoring in achieving the desired goals. There were debates and publications claiming it to be useful and not useful. Randomized controlled trials showed that intensive intermittent FHR auscultation acieved the same results as FHR monitoring. It was also shown that the latter increased rates of cesarean section and operative vaginal delivery. Though it reduced intrapartum fetal deaths, the incidence of cerebral palsy remained unchanged. It still continues to be used extensively because of its convenience and reduction in intrapartum fetal deaths and early onset neonatal convulsions. A drawback of electronic FHR monitors is that the false positive rate is quite high. Another drawback is possibility of a number of errors which can be due to machine malfunction or patient-related problems. A number of such errors have been described, and some of them cannot be overcome, except by adopting new technology. A number of such technologies are being evolved, such as the STAN monitor, the Monica AN24 monitor, the MindChild MERIDIAN monitor, and an FHR monitoring system based on radiofrequency technology. While such development is taking place, the older technology is by no means obsolete. Hence we have to live with the errors associated with it and try to circumvent those or eliminate them in newer models of the machines. It is interesting that newer errors are being reported even now. One such error is discussed in this issue of the journal. I hope it helps clinicians using electronic FHR monitors, and also the manufacturers who make the monitors.


Pubovesicocervical Fascial Leiomyoma

Author Information

Parulekar SV* Fernandes GC**.
(*Professor and Head, Department of Obstetrics and Gynecology, ** Associate Professor, Department of Pathology, Seth G S Medical College & KEM Hospital, Mumbai, India.)

Abstract

A leiomyoma is the most common pelvic tumor in women. It is found in about 20–30% of women above 35. Extrauterine leiomyomas are rare. A case of a leiomyoma in the pubovesicocervical fascia is reported here. The patient presented with a complaint of feeling a mass in the vagina on self examination, which was unusual. A leiomyoma in this location has not been reported before in the literature.

Introduction

A leiomyoma is the most common benign tumor in women.[1] The uterus is the most common site. Extrauterine sites include the broad ligaments, round ligaments, utero-ovarian ligaments and vagina.[2,3,4] A leiomyoma in the pubovesicocervical fascia has not been reported so far. Such a leiomyoma in a 34 year old parous woman is reported here.

Case Report

A 34 year old woman with two normal deliveries in the past, presented with a complaint of the presence of a mass that she could feel inside the vagina. There was no abnormal vaginal bleeding, leucorrhea, urinary disturbances, difficulty in defecation or dyspareunia. Her menstrual cycles were regular, every month and were associated with normal flow over 3-4 days. There was no dysmenorrhea. Her medical and surgical history was not contributory. Her general condition was fair and vital parameters were within normal limits. Her general and systemic examination findings were normal. Abdominal examination showed normal findings. A speculum examination showed a 2 cm long and 1.5 cm wide projection from the anterior vaginal wall, looking like portio vaginalis. A normal looking cervix was seen 1 cm behind it (figure 1). A bimanual pelvic examination showed the uterus to be of normal size and displaced posteriorly. A 5 cm long, 3 cm wide and 2 cm deep mass was felt between the urinary bladder in front and the uterus behind. It was nontender and firm in consistency. The lower end of the mass was projecting into the vagina. A diagnosis of pelvic extraperitoneal mass was made. A computed tomography (CT) of the abdomen and pelvis confirmed the presence of the mass between the bladder and the uterus (figure 2). There was no connection between the mass and the uterus or the urinary bladder. The kidneys and ureters were normal. Considering the possibility of malignancy or local recurrence of a benign tumor in that location after excision of the mass, the patient was referred to an oncology center. A biopsy was obtained there, which showed it to be a leiomyoma. Immunohistochemical study showed that the tumor cells were diffusely positive for smooth muscle actin (SMA) and desmin reinforcing smooth muscle differentiation. She was referred back to us for further treatment, with a statement that it was polyp from the cervix. A reference  was made to urologists, considering the risk of bladder injury during excision of the mass. The urologist diagnosed it to be a cervical polyp and assured of help during surgery, should the urinary bladder get injured. Her tests for fitness for anesthesia showed normal findings. Vaginal excision of the mass was made by cutting the epithelium over it and dissecting it sharply, safeguarding the urinary bladder and ureter carefully. A mass measuring 7x4x2 cm was excised. It had no connection with the urinary bladder, uterus or ureter. The cavity left behind was occluded with interrupted sutures of No. 1-0 polyglactin. Redundant part of vaginal mucosa was excised and vaginal epithelium was sutured with interrupted sutures of No. 1-0 polyglactin. The patient made an uneventful recovery. Histopathological examination of the mass showed features of a classic leiomyoma with hyalinization. There was no nuclear atypia, mitoses or necrosis. A diagnosis of a leiomyoma was made.


Figure 1. Appearance of the mass on speculum examination of the vagina.


Figure 2. CT scan of the abdomen and pelvis. The mass is seen between the uterus and the urinary bladder.


Figure 3. CT angiography showing vascularity of the mass.


Fig 4. Lieomyoma composed of smooth muscle arranged in fascicles and parallel bundles.
[H&E x 100]


Fig. 5. Smooth muscles cells arranged in interlacing fashion with a focus of hyalinization denoted by an arrow.  [H&E x 100]


Fig. 6. High power view of the spindle shaped smooth muscle cells with bland cigar-shaped nuclei. [H&E x 400]

Discussion

A leiomyoma is the most common pelvic tumor in women.[5,6] It is a benign smooth muscle tumor, usually arising from the myomatrial cells. Vaginal smooth muscle tumors are rare. Vaginal epitheloid leiomyoma is ever rarer.[7] It was a differential in the case presented, which was ruled out subsequently by histopathological examination. A true broad ligament leiomyoma arises from smooth muscle in the walls of blood vessels or from embryonic remnants of paramesonephric duct. Similar tissue exists in the pubovesicocervical fascia. Hence development of a leiomyoma in this location can be expected. But such a case has not been reported so far. Development of solid tumors in retroperitonal space or extraperitoneal space raises the suspicion of a sarcoma,  lymphoma, epithelial tumour, benign neurogenic tumor, lipoma, angiomyolipoma, fibromatosis and metastatic tumor from elsewhere. Many of these benign tumors have a tendency to spread locally and recur despite wide excision. In our case, a biopsy at a cancer institute showed it to be a leiomyoma. The diagnosis was confirmed by review of the slides in our institute. The diagnosis was confirmed on histopathology of the tumor after its surgical removal. An unusual feature of this tumor was that it was growing between the urinary bladder and the uterus in its upper part, and into the vaginal cavity under the anterior vagina in its lower part. A vaginal leiomyoma usually expands the vagina over it and grows downwards. Growth of the leiomyoma between the urinary bladder and the uterus in this case suggests that it was a tumor that developed between the urinary bladder and the uterus extraperitoneally and grew downwards, expanding the vagina over its lower end. It had not grown out from the uterus, the cervix or the urinary bladder, as there was no physical connection between any of these structures and the leiomyoma. It was surprising that the woman examined herself and felt the tumor. It was more surprising that the tumor was mistaken to be arising from the uterine cervix by oncologists and urologists. This emphasizes the fact that such tumors should be treated after joint consultation between gynecologists, urologists and oncologists. Imaging in the form of ultrasonography, computed tomography and magnetic resonance imaging avoids such errors in diagnosis and helps manage such cases better. The final diagnosis in all cases is by histopathological examination.[8] A needle aspiration biopsy is useful. In cases like the one presented here, it is easy as the mass is accessible vaginally and can be biopsied easily. Removal of the mass can cause inadvertent injury to the urinary bladder and/or the urethra. A cautious and meticulous technique helps avoid such mishaps.

Acknowledgment

I thank Dr. Sreshthha Mahanti for clinical photograph of the patient.

References
  1. Kurman R. J., Ronnett B. M., Ellenson L. H. In: Blaustein's Pathology of the Female Genital Tract. 6th. Kurman R. J., Ronnett B. M., Ellenson L. H., editors. London, UK: Springer; 2011.pp 453-527.
  2. Stankova T., Ganovska A., Kovachev S. Vaginal leiomyoma after total abdominal hysterectomy—clinical case and review of literature. Akusherstvo i Ginekologiia. 2015;54(6):39–42.
  3. Bansal P., Garg D. A case of massive broad ligament leiomyoma imitating an ovarian tumour. Journal of Clinical and Diagnostic Research. 2014;8(3):136–137. doi: 10.7860/JCDR/2014/7642.4136.
  4. Ichigo S., Takagi H., Matsunami K., Murase T., Ikeda T., Imai A. A large ovarian leiomyoma discovered incidentally in a 76-year-old woman: case report. European Journal of Gynaecological Oncology. 2015;36(2):203–205. doi: 10.12892/ejgo2611.2015.
  5. Serden S. P., Brooks P. G. Treatment of abnormal uterine bleeding with the gynecologic resectoscope. Journal of Reproductive Medicine for the Obstetrician and Gynecologist. 1991;36(10):697–699.
  6. Baird D. D., Dunson D. B., Hill M. C., Cousins D., Schectman J. M. High cumulative incidence of uterine leiomyoma in black and white women: ultrasound evidence. American Journal of Obstetrics and Gynecology. 2003;188(1):100–107. doi: 10.1067/mob.2003.99.
  7. Tanaka Y, Nagasaka M, Takahashi M, Kobayashi M. Rare Epithelioid Leiomyoma of the Vagina Exhibiting a Pelvic Mass. Case Reports in Obstetrics and Gynecology. 2017;2017:2190135. doi:10.1155/2017/2190135.
  8. Strauss DC, Qureshi YA, Hayes AJ, et al. The role of core needle biopsy in the diagnosis of suspected soft tissue tumours. J Surg Oncol. 2010;102:523–529.
Citation

Parulekar SV, Fernandes GC. Pubovesicocervical Fascial Leiomyoma. JPGO 2018. Volume 5 No.1. Available from: http://www.jpgo.org/2018/01/pubovesicocervical-fascial-leiomyoma.html

Phantom Fetal Heart Sounds – A New Machine Error

Author Information

Parulekar SV

(Professor and Head, Department of Obstetrics and Gynecology, Seth G S Medical College & KEM Hospital, Mumbai, India.)

Abstract

Electronic fetal heart rate monitoring has been around for a long time. Technology has evolved rapidly, while the older technology also continues to be used effectively. Noninvasive monitoring using Doppler ultrasound based monitors is sensitive and is used widely. A number of errors are possible, either due to machine malfunction or patient-related factors. A new error due to machine malfunction is described here.

Introduction

Phillipe LeGaust first described fetal heart tones in the 17th century in his poetry in an ancient French dialect. He had actually picked it up from his friend Marsac who was a physician in Limousin, who gets credit for hearing the fetal heart sounds for the first time. Lejumeau Kergaradec, a French nobleman, suggested that auscultation could be of value in the diagnosis of pregnancy and twins, and also in determining fetal lie and presentation. Evory Kennedy of Dublin published a book entitled 'Observations on Obstetric Auscultation' in 1833.[1] He also described the funic souffle for the first time. DeLee-Hillis stethoscope used to auscultate fetal heart sounds was described by Hillis and DeLee independently.[2,3] Kilian first proposed that changes in the fetal heart rate might be used to diagnose fetal distress.[4] Von Winckel described in 1893 the criteria of fetal distress. Cremer, a German physiologist, used a galvanometer to obtain the first recording of FHR in 1906.[5] He recorded the FHR electronically for the first time, using of abdominal and intravaginal leads in 1906. Hammacher and Hewlett-Packard produced the first commercially available fetal monitor based on external tocography and phonocardiography in 1968. More recently Doppler ultrasound has been used extensively to provide FHR signals for monitoring fetal well being. A number of errors are associated with electronic FHR monitoring, either due to amchine malfunction or patient-related factors. A new machine error is described here.

Case Report

An fetal electronic heart rate (FHR) monitor was being used in the labor ward for intrapartum recording using a Doppler external transducer. The display on the LCD screen showed a normal pattern, with good beat-to-beat variability as well as long term variability. The audio signal of fetal heart sounds was also satisfactory. However the resident doctor using the machine noticed that the monitor continued to show a satisfactory FHR tracing and the audio signal continued as before even after the transducer was removed from the patient’s abdomen. A machine error was diagnosed. The occurrence of the error was documented on a video. Owing to an inability to display the video in a journal article, snapshots of the video obtained at different times are shown below.


Figure 1. Early part of the record.


Figure 2. A record obtained some time after the first one.


Figure 3. A record obtained much later.

In troubleshooting of the machine, it was switched off and then on. The error continued. The transducer was removed from the machine, at which point it promptly displayed an error that the transducer port was empty. The error resumed when the transducer was connected to the port. The transducer was substituted with another functioning transducer, and the error still remained. The transducer was put on another machine and it functioned properly, without any error. So a tentative diagnosis of an error in the motherboard (printed circuit board) of the machine was made. The machine was sent to the manufacturer for repairs. The diagnosis of damage to the motherboard was confirmed.

Discussion

It is not necessary to use a direct fetal ECG signal to record the FHR in the antenatal period and most often in intrapartum period. An instrument which functions on the Doppler principle is adequate for this purpose.[6-8] Ultrasonic beam passes through soft tissues and gets reflected partly when it encounters an interface of increased density. The reflected signal undergoes a frequency change called Doppler shift if the interface is moving. If the movement is away from the signal source, the frequency decreases and if it is toward the signal source, it increases. Older instruments and many of the newer instruments use multiple crystals in the transducer to transmit the signal and receive the reflected signals continuously. Newer machines use a pulsed Doppler in which the emission of ultrasound waves and the reception of the reflected waves are alternated. This decreases the amount and duration of fetal exposure to ultrasound energy. The rhythmic movement of the fetal heart produces Doppler shifts which enable recording of FHR and production of fetal heart sounds which can be heard.

Many errors can occur with this system. The signal is interefered with by maternal muscle movements, change of position or uterine contractions and fetal movements away from the transducer. Loss of alignment between the ultrasonic beam and moving surface of the fetal heart can result in loss of FHR signal and erroneous records. Sometimes there is halving of FHR when above 240 due to electronic logic used by the machine to produce a FHR that is in the range which is acceptable to the clinician. It also occurred in earlier machines when a large P wave was counted in addition to R wave.[9] If maternal aortic blood flow signal is strong, it may be picked up and recorded as FHR. This tends to occur also when the fetus is dead and only signal available for being picked up is that from the maternal aorta. There can be an apparent increase in short-term variability due to detection and recording of the highest point of the waveform, even if it may not appear at the same time in each successive waveform.[10] This error varying waveform amplitude problems is minimized or eliminated by the use of high-speed microprocessor integrated circuitry to achieve autocorrelation to FHR technology.[11]
A number of such technologies are being evolved, such as the STAN monitor, the Monica AN24 monitor, the MindChild MERIDIAN monitor, and an FHR monitoring system based on radiofrequency technology. The use of any new technology needs to be done with some caution. A failure to consider various sources of error during manufacture of the equipment and writing the code for function of the processor can result in serious errors. The case described here is the first report of such an error. It was due to a fault developed in the motherboard of the machine. Discussion of the electronics of the error is beyond the scope of this article. But if attention is not paid to the FHR recording by the clinician, then a record may be obtained which will be totally different from the actual FHR, giving false security and decisions based on that record may result in serious adverse consequences for the fetus. The resultant litigations against the clinician will prove very costly, though the fault lies with the manufacturer and the maintenance engineer. Compensations will be awarded to the patients because the clinician is expected to detect malfunction of the equipment he uses and get it repaired as soon as it is detected. Total reliance on machines is not justifiable, since auscultation of FHR is a time tested and proved method of FHR monitoring, and cannot be given up because a machine can do the job.

Conclusion

Though modern monitoring equipment is far superior to the human senses of hearing, seeing and touching, it should be used with caution. Electronic equipment can malfunction in ways that are diverse and not always easily noticeable. Periodic checking of the equipment by maintenance personnel and by the clinicians would help prevent morbidity and mortality for the patients and lawsuits for negligence by the clinicians.

References
  1. Kennedy E. Observations on Obstetric Auscultation. Hodges and Smith, Dublin, 1833.
  2. Hillis DS. Attachment for the stethoscope. JAMA 1917;68:910.
  3. DeLee JB. Ein nues stethoskop für die Geburtshilfe besonders geeignet. Zentralbl Gynaekol 1922;46:1688.
  4. Goodlin R: History of fetal monitoring. Am J Obstet Gynecol 1979;133:325.
  5. Whitfield CR. Foetal heart rate monitoring--present lessons and future developments. The Ulster Medical Journal. 1966;35(1):75-82.
  6. American College of Obstetricians and Gynecologists, authors. ACOG Practice Bulletin No. 106: Intrapartum fetal heart rate monitoring: nomenclature, interpretation, and general management principles. Obstet Gynecol. 2009;114:192–202.
  7. The American College of Obstetricians and Gynecologists. Fetal heart rate monitoring during labor. 2001. Available from http://www.acog.org/publications/patient_education/bp015.cfm
  8. Chen P. Fetal heart monitoring. Department of Obstetrics & Gynecology, University of Pennsylvania Medical Center. 2004. Available from: http://pennhealth.com/ency/article/003405.htm
  9. Stampalija, T., Signaroldi, M., Mastroianni, C., Rosti, E., Signorelli, V., Casati, D., & Ferrazzi, E. M. (2011). Fetal and maternal heart rate confusion during intra-partum monitoring: comparison of trans-abdominal fetal electrocardiogram and Doppler telemetry. The Journal of Maternal-Fetal & Neonatal Medicine, 25(8), 1517–1520.
  10. Murata Y, Martin C: Systolic time intervals of the fetal cardiac cycle. Obstet Gynecol 1974;44:224.
  11. Fukushima T, Flores CA, Hon EH, et al.: Limitations of autocorrelation in fetal heart rate monitoring. Am J Obstet Gynecol 1985;153:685.
Citation

Parulekar SV. Phantom Fetal Heart Sounds – A New Mmachine Error. JPGO 2018. Volume 5 No.1. Available from: http://www.jpgo.org/2018/01/phantom-fetal-heart-sounds-new-machine.html

Challenging Case Of Pregnancy Associated Cardiomyopathy Of Multifactorial Origin

Author Information

Jagtap S*, Samant PY**, Yadav K***.
(* Senior Resident, ** Professor, *** First Year Resident. Department of Obstetrics and Gynecology, Seth GS Medical College and KEM Hospital, Mumbai, India.)

Abstract
We present a rare case of cardiomyopathy of probable multifactorial origin who presented with severe hypertension and was detected to have hypothyroidism. The additional burden of endocrine and hypertensive disorder added to the complexity of management. Though it was  diagnosed as peripartum cardiomyopathy, a review of diagnosis was required. 

Introduction

Peripartum cardiomyopathy (PPCM), is a disorder causing cardiac failure. It typically affects pregnant women during a period of one month prior to delivery to five months post partum.  Clinical presentations have been found ranging from thromboembolism to cardiac failure and maternal mortality. It has a tendency to recur in subsequent pregnancies and maternal mortality is considered to be as high as 25 to 50 %. Association of hypertension of uncertain duration and hypothyroidism creates diagnostic dilemma, and add to maternal and fetal morbidity. Multidisciplinary approach aids safe management. 

Case Report

A 30 year old primigravida with 32 weeks and 5 days gestation was referred by a peripheral hospital to our tertiary care center in view of pre-eclampsia. 
The patient had complaints of right-sided chest pain since afternoon of the day of admission and breathlessness for 3 days. There were no other complaints like palpitation, or dizziness or loss of consciousness. She also reported bilateral pedal edema since 7th month of pregnancy. There were no complaints of headache, vomiting, epigastric pain, or blurring of vision. She did not reveal history of any other major medical or surgical conditions in the past. She gave no positive family history. She was taking treatment from a private practitioner since first trimester. She was detected to be hypertensive in the 5th month of pregnancy, and was started on tablet labetalol 100 mg twice daily, but she discontinued treatment after a month. 
On examination her general condition was fair. There was no pallor. She was afebrile, there was no remarkable tachycardia. Blood pressure was 110/70 mm of Hg (on antihypertensive medication). No abnormality was detected on auscultation. Bilateral pedal and vulval edema was present. There was no pallor/ icterus/ cyanosis/ clubbing or lymphadenopathy. Deep tendon jerks were normal.
On abdominal examination, uterus was 26-28 weeks in size, relaxed with a single fetus in vertex presentation and fetal heart sounds were regular at 130 beats per minute. Vulval edema was present. On internal examination cervix was long, posterior, uneffaced. Os was closed. 
Her Hemoglobin was 11.5 gm%. Urine albumin was 3+. PT was within normal limits and INR was 0.93. Serum electrolytes were normal. TSH was advised which was found to be 8.09 IU/ml. Renal and liver function tests were normal. Coagulation screen was normal. Renal ultrasound was normal. Screening for APLA syndrome, ANA, ds DNA was negative. Fundoscopy revealed normal findings. Electrocardiogram was reported to have a normal sinus rhythm, normal axis and T wave inversion in leads V2 to V6. Troponin T test was negative. Echocardiogram was indicative of dilated cardiomyopathy with global hypokinesia and moderate mitral regurgitation with ejection fraction of 45 %. On cardiologists advise oral Furosemide and potassium supplementation were added and antihypertensives were continued . 
In view of hyponatremia that was diagnosed as hypervolemic hyponatremia, fluid restriction was advised. Moist oxygen by mask was started for breathlessness. Endocrinologist started her on Levothyroxine 50 mcg/ day. Parenteral betamethasone was given for fetal lung maturity and parenteral ceftriaxone and gentamycin were also started in therapeutic doses. Labetalol, and furosemide were continued.                                                                                                                                                      
Induction of labor was done as blood pressure rose to 170/110 mm Hg. Epidural labor analgesia was given. She delivered a preterm baby weighing1.24 kg who had an Apgar score of 9/10. She was reviewed by cardiologists on day 2 of delivery. Post delivery echocardiogram revealed ejection fraction of 30%. She was continued on bed rest and the same medical management. On day 3 of delivery her SGOT was 134 U/L, alkaline phosphatase was high at 253 IU/L. Observation and avoidance of hypotension/ hypertension, hepatotoxic drugs, hypoglycemia and sepsis was advised. She recovered in a week and was discharged on diuretics, antihypertensives and thyroid supplements. She and her family were counseled about risk of recurrence in subsequent pregnancies and barrier contraception was advised.

Discussion

Peripartum cardiomyopathy is called diagnosis of exclusion where all other conditions causing cardiac failure are ruled out. A met analysis by Bello et al found pooled prevalence PIH of 22% in PPCM as compared to 5% in normal pregnancy and prevalence of hypertension during pregnancy of 37% in PPCM.[1] Our patient was hypertensive since the fifth month of pregnancy and a treatment defaulter.
Johnson Coyle and colleagues stated that myocardial infarction, sepsis, severe pregnancy induced hypertension, structural cardiac diseases and other factors that may cause cardiomyopathy must be ruled out before labeling a case as PPCM.[2] Both hypo and hyperthyroidism are considered as causative factors in cardiomyopathy though mechanisms differ. Hypothyroidism causes low output cardiomyopathy by affecting alfa myosin chains and affecting ionic channels.[3] Our patient was detected to have hypothyroidism which could have contributed to her cardiomyopathy.
Besides the diagnostic dilemma; the challenge was to deliver her safely. After delivery deranged liver function required careful monitoring and management.
Epidural analgesia and anesthesia help in PPCM cases as those permit gradual induction of anesthesia. Also there is minimal fluctuation in hemodynamic parameters.[4] Epidural induced sympathectomy improves cardiac condition during labor.[5] Multidisciplinary management and epidural analgesia helped our patient go through labor smoothly. Our patient had mild hepatic derangement, which recovered over a few days. Hepatic congestion and cellular failure induced by cardiac failure is reported in cases of PPCM. Fussell reported a case of fulminant hepatic failure that recovered after treating previously undiagnosed peripartum cardiomyopathy.[6] 

Conclusion

It is important to consider and investigate inflammatory, metabolic, cardiological and other factors in cases of peripartum cardiomyopathy so that accurate treatment is initiated and chance of recurrence in subsequent pregnancies reduce. For safe transition through induced or spontaneous labor epidural analgesia is a safe intervention.

References
  1. Bello N, Rendon I. Arany Z. The relationship between pre-eclampsia and peripartum cardiomyopathy: a systematic review and meta-analysis. J Am Coll Cardiol. 2013;62(18):1715-1723. 
  2. Johnson-Coyle L, Jensen L, Sobey A. Peripartum Cardiomyopathy: Review and Practice Guidelines. Am J Crit Care 2012;21(2): 89-98
  3. Patel H, Madanieh R, Kosmas CE, Vatti SK, Vittorio TJ. Reversible Cardiomyopathies. Clin Med Insights Cardiol. 2015;9(Suppl 2):7-14.
  4. Dutt A, Agarwal A, Chatterji R, Ahmed F.  Anesthetic management for caesarean section in a case of peripartum cardiomyopathy. Anesth Essays Res. 2013; 7(2): 273–275.
  5. Bhakta P, Biswas BK, Banerjee B. Peripartum Cardiomyopathy: Review of the Literature. Yonsei Med J. 2007;48(5):731-47.
  6. Fussell KM, Awad JA, Ware LB. Case of fulminant hepatic failure due to unrecognized peripartum cardiomyopathy. Crit Care Med. 2005;33(4):891-3.

Citation

Jagtap S, Samant PY, Yadav K. Challenging Case Of Pregnancy Associated Cardiomyopathy Of Multifactorial Origin. JPGO 2018. Volume 5 No.1. Available from: http://www.jpgo.org/2018/01/challenging-case-of-pregnancy.html

Infertility Due To Genital Tuberculosis

Author Information

Shetty A*, Pardeshi S**, Gupta AS***
(* Third year Resident, ** Assistant Professor, *** Professor, Department of Obstetrics and Gynecology, Seth GS Medical College and KEM Hospital, Mumbai, India..)

Abstract

Genital tuberculosis is one of the important cause of infertility in India. Apart from concerns of drug toxicity, this may be a contributing factor in the increasing incidence of multi-drug resistant tuberculosis. Female genital tuberculosis (FGTB) is an important cause of significant morbidity, short term and long term sequelae especially infertility. We present a case of confirmed genital tuberculosis and a discussion on the various tests that are available for diagnosis of FGTB.

Introduction

Genitourinary Tuberculosis (GUTB) is the second most common extra pulmonary manifestation of tuberculosis (TB). Lack of an accurate diagnostic test has led to rampant use of anti-tubercular treatment in infertile women.[1] FGTB is an important cause of significant morbidity, short term and long term sequelae especially infertility; the incidence of which varies from 3 to 16 % cases in India.[2] An isolated involvement of genital organs is reported in 5-30 % of the cases.[3] Disease usually severely affects the fallopian tubes leading to infertility. Diagnosis is usually difficult because of the asymptomatic nature of the disease or atypical clinical presentation. [4]

Case Report

A twenty-six-year-old woman with a prior 1 abortion came with the chief complaints of oligomenorrhea of 3 years duration and secondary amenorrhea of 1 year duration for evaluation of infertility. She had no history of tuberculosis, thyroid dysfunction or galactorrhea. Her cycles previously have been regular, with moderate amount of flow. She also gave history of missed abortion a year back, no curettage was done. Her general and systemic examinations were normal.  On local speculum examination, cervix had an erosion that did not bleed on touch, vagina was healthy, uterus was normal in size, anteverted, and fornices were normal. Urine pregnancy test was negative. Serological, biochemical, and hormonal profile were advised. Progesterone challenge test with oral medroxyprogesterone acetate was given. She had no withdrawal bleeding with progesterone, and was advised estrogen challenge test. She received conjugated equine estradiol for 21 days but had no withdrawal bleeding. Her hormonal profile that included TSH, serum LH and FSH levels were all within normal limits. Semen analysis was within normal limits. Ultrasonography showed sub endometrial calcification with endometrial thickness of 3.6 mm. A colposcopy,  diagnostic hystero-laparoscopy and endometrial curettage was performed. Colposcopy was negative. On hysteroscopy right ostia was visualized, left ostia was not visualised and left uterine wall was irregular with multiple transverse bands of adhesions. On laparoscopy bilateral tubes were rigid, and both ovaries were obscured due to dense adhesions. Multiple tubercles were seen. Peritoneal and endometrial samples were collected and sent for smear, staining and isolation of AFB (acid fast bacilli), Gene Xpert, MGIT (mycobacterium growth indicator tube), TB PCR (tuberculosis polymerase chain reaction) and histopathology. MGIT showed growth of mycobacterium tuberculosis complex. Endometrial PCR was negative. Histopathology showed evidence of necrotising granulomatous endometritis possibly of tuberculous etiology. She was started on anti-tuberculous therapy (AKT). After 6 months of AKT she remained amenorrhoeic. She was started on cyclical conjugated equine estrogens in the dose of 1.25 mg twice daily for 21 days. She had no withdrawal bleeding even after 2 cycles of estrogen therapy. A second look hysteroscopy showed an irregular left uterine wall was with multiple transverse bands of adhesions. Curettage was done and endometrial samples were send for repeat Kochs’ cultures and histopathology to assess the need for further continuation of anti-tuberculous treatment. There was no evidence of tuberculosis on histopathology. She was counseled regarding IVF embryo transfer, surrogacy and adoption options. 

Discussion

Female genital tuberculosis is a common cause of infertility. It usually has mild clinical picture and usually detected with high index of clinical suspicion. Incidence of female genital tuberculosis is 27% worldwide.[5] Incidence is increased to about 58% in endemic country like India.[6] About 27 % of patients with genital tuberculosis present with menstrual irregularities.[7]
Four clinical criteria are established by Klein et al.[8] These are 1) There are irregular calcifications in adnexal area. 2) Tubal obstruction in zone of transition between isthmus and ampulla. 3) Multiple constrictions in tube. 4) Endometrial adhesions or obliteration in absence of history of curettage.
In study conducted by Kapoor et al it is reported that only 25 % of the cases of tuberculous endometritis are detected on histopathology as involvement of endometrium is usually focal and due to cyclical shedding of endometrium, evidence of tuberculosis may not be picked up in all cycles.[9] However in our case histopathology showed evidence of tuberculosis. 
Polymerase chain reaction (PCR) test in the endometrial tissue has been used for identification of tuberculous bacilli. Endometrial scrapped samples are sent in normal saline. The DNA samples are amplified using primer. MGIT test works on the principle that fluorescent compound in the broth is sensitive to the dissolved oxygen which is released by the actively respiring tuberculous bacteria. This fluorescence can be recorded by automated instruments. This test can also be used to detect culture and sensitivity pattern of the bacilli. 
In our case mycobacterium growth indicator tube (MGIT) showed growth of mycobacterium tuberculosis but TB PCR was negative, probably because of the sampling error that occurs as there is only a focal area of endometrial lesions or bacterial load is less as tuberculosis has a paucibacillary nature. Moreover, endometrial samples are always admixed with blood and presence of inhibitors can also be possibility for false negative results. A negative PCR results may be misleading, so when GTB is clinically suspected and PCR tests are negative further testing should be gone to confirm the diagnosis.[10] 
In study conducted by Sharma et al, starting of anti-tuberculosis therapy in early disease improved the clinical outcome whereas advanced disease had poorer outcomes.[11] 
Infertility management for these patients is usually multidrug anti-tuberculous therapy. In case of presence of synechiae, hysteroscopy with adhesiolysis and with insertion of intrauterine device for short periods is recommended. Conjugated sstrogens can be used for rapid growth of endometrium. Spontaneous pregnancy after the diagnosis of genital tuberculosis is rare and if it occurs it usually results is an abortion or ectopic pregnancy. Frydman et al reported about 25% pregnancy rate per transfer in tuberculous infertility.[12]  In vitro fertilisation presents the only treatment for tubal and possibly endometrial tubercular infertility and improves the chances of fertility in what was earlier considered a desperate situation.

Conclusion

In this era of multi drug resistant tuberculosis it is essential for all clinicians especially those treating extra pulmonary tuberculosis to ensure that all diagnostic modalities that are available for diagnosing pulmonary tuberculosis and used to confirm extra pulmonary tuberculosis like genital Kochs’.

References 
  1. Mahajan N, Naidu P, Kaur SD. Insight into the diagnosis and management of subclinical genital tuberculosis in women with infertility. J Hum Reprod Sci.2016;9(3):135.
  2. Sharma JB, Kriplani A, Sharma E, Sharma S, Dharmendra S, Kumar S, et al. Multi drug resistant female genital tuberculosis: A preliminary report. Eur J Obstet Gynecol Reprod Biol. 2017 Mar;210:108-115.
  3. Yadav S, Singh P, Hemal A, Kumar R. Genital tuberculosis: current status of diagnosis and management. Transl Androl Urol 2017;6(2):222–33.
  4. Shrivastava G, Bajpai T, Bhatambare GS, Patel KB. Genital tuberculosis: Comparative study of the diagnostic modalities. J Hum Reprod Sci 2014;7(1):30–3. 
  5. Golden MP, Vikram HR. Extrapulmonary tuberculosis: an overview. Am Fam Physician. 2005;72(9):1761-8.
  6. Dam P, Shirazee HH, Goswami SK, Ghosh S, Ganesh A, Chaudhury K, et al. Role of latent genital tuberculosis in repeated IVF failure in the Indian Clinical setting. Gynecol Obstet Invest 2006; 61(4): 223-227.
  7. Chakrabarti AK, Sen S, Banerjee A, Roy K. Female genital tuberculosis- a retrospective study. Ind J Tub 1998; 45: 101-3.
  8. Klein TA, Richmond JA, Mishell DR Jr. Pelvic tuberculosis. Obstet Gynecol. 1976;48(1):99-104.
  9. Kapoor N, Pawar S, Sirakova TD, Deb C, Warren WL, Kolattukudy PE.Human granuloma in vitro model, for TB dormancy and resuscitation. PLoS One. 2013;8(1):e53657.
  10. Thangappah RB, Paramasivan CN, Narayanan S. Evaluating PCR, culture & histopathology in the diagnosis of female genital tuberculosis. Indian J Med Res. 2011 ;134:40-6.
  11. Sharma BJ,   Sneha J, Singh UB, Kumar S, Kumar RK, Singh N,et al. Effect of Antitubercular Therapy on Endometrial Function in Infertile Women with Female Genital Tuberculosis. Infect Disord Drug Targets. 2016;16(2):101-8.
  12. Frydman R, Eibschitz I, Belaisch-Allart JC, Hazout A, Hamou JE. In vitro fertilization in tuberculous infertility. J In Vitro Fert Embryo Transf. 1985;2(4):184-9.

Citation

Shetty A, Pardeshi S, Gupta AS. Infertility Due To Genital Tuberculosis. JPGO 2018. Volume 5 No.1. Available from: http://www.jpgo.org/2018/01/infertility-due-to-genital-tuberculosis.html

Use of Endoknife in Laparoscopy- An Innovation

Innovation
Author Information

Shah NH*, Somani A**, Deshmukh M**, Paranjpe S***
(* Consulting Gynecologist/Obstetrician & Hon. Endosopic Surgeon Wadia Hospital & Railway Hospital, Byculla, ** Consulting Gynecologist/Obstetrician Vardann hospital, *** Director: Velankar Hospital & Paranjpe Maternity Home, Chembur)

Abstract

Tissue retrieval in laparoscopy is a challenge despite various available techniques. Each technique has its own merits and demerits.The choice of tissue retrieval depends on the type and size of specimen to be delivered.We present here our technique of debulking of a large uterus with use of endoknife.With this technique large specimen can be debulked and retrieved vaginally in hysterectomy.

Introduction

Laparoscopy is one of the most preferred ways for hysterectomy due to its merits such as minimal incision, less duration of hospital stay and less post operative pain. Delivery of large specimen post hysterectomy is a challenge. Tissue retrieval techniques should adhere to principals of laparoscopy. Adhering to the principles of minimal access surgery, one has to retrieve the specimen from the abdominal cavity in such a way so as to not cause any infection, or tumor implantation or spillage. Tissue retrieval in laparoscopy can be done by following techniques; removal through trocar/ trocar site, culdotomy, enlarging the  incision, mini-laparotomy, or morcellation. Bulky specimens can be debulked vaginally by coring technique and retrieved vaginally. Power morcellation is used to debulk larger specimens and retrieve through side port. Power morcellation, if not done in an endo bag, causes spinning of tissue during debulking leading to  micro spillage.

Case Report

A 45 year old woman was posted for total laparoscopic hysterectomy in view of menorrhagia with multiple uterine fibroids. Ultrasonography showed multiple fibroid uterus with a well defined hypoechoic mass arising from the posterior wall of yhe uterus measuring 6x7x5 cm. Clinically uterus was around 20 weeks in size. Upon insertion of the laparoscope, a large uterus with multiple fibroids and a large posterior wall fibroid was seen. Hysterectomy was completed with routine steps, after which it was not possible to retrieve the specimen through the colpotomy incision.
Thus to debulk the large uterus in such a way so as to remove it through the colpotomy incision, an ENDOKNIFE was used. The idea was to make the large globular uterus into an elongated strip which could come out though the colpotomy incision. Thus as shown in figure 2, the endoknife was used to cut the uterus starting from above the cervix in circular manner so as to make it into a elongated strip. This enabled it to come out easily as an elongated structure.


Figure 1. Endoknife 


Figure 2. Showing the cutting path of the specimen


Figure 3. Specimen held with tenaculum and endoknife cutting from other side port


Figure 4. Final specimen after removing through the colpotomy incision

Discussion

Tissue retrieval in endoscopy is a challenge especially for bulky masses. Tissue retrieval should adhere to the principles of minimal invasive surgery. One method of tissue retrieval is to enlarge one of the side port incisions into a mini laparotomy and remove the specimen, but this is against the principles of laparoscopy.[1] The retrieval for large uterus or big fibroid is done by power morcellation. Power morcellation causes tissue spinning and thus leads to micro spillage. Power morcellator is an expensive instrument and requires energy source. Endoknife is manually operated, thus making it cost effective. Endoknife is a good alternative for low resource setting. Endoknife can be introduced from a 5mm side port. 
“The Orange peel technique” that Harrith Hasson has described is a technique in which scissors are used to peel off the tissue in the same way as one would peel off an orange. Before the availability of morcellators, scissors were used to have the same effect as that of a morcellator by making the tissue elongated and removing from the trocar cannula.[2] The long, thin strips then can be easily extracted through the trocar cannula. Unfortunately, laparoscopic scissors are too small or inadequate to incise a large specimen. Thus the use of an endoknife.
Further morcellator cannot be used in cases of suspected malignancy where tissues not only can be embedded in the surrounding areas but also tissue may be lost for histopathological diagnosis.[3]
Thus, tissue can be held with a tenaculum and debulked using an endoknife and the specimen is retrieved either vaginally, or from a colpotomy incision or even through the trocar cannula. The endoknife instrument has various positions namely 0, 1, 2 in which the blade is completely inside, half outside or completely outside. During insertion the blade is kept completely inside to avoid injury to surrounding structures. Endoknife does not cause any spinning of tissues, so reduces chances of tissue embedding and spillage. 
Also during the myomectomy, while not removing the uterus, if one prefers posterior colpotomy rather than morcellation, endoknife can be used to debulk and make an elongated structure so as bring out the specimen through the small posterior colpotomy incision.  

Conclusion

Tissue retrieval is an important issue in minimal access surgery. Various methods are being currently used and new technologies are being developed to make this procedure safe and effective for the patient. Thus as seen in this case, endoknife can be safely used as a cost effective method to remove the specimen.

References
  1. Schellpfeffer MA. A novel laparoscopic tissue retrieval device. JSLS 2011;15(4):527-32
  2. Hasson HM, Rotman C, Rana N, Sistos F, Dmowski WP. Laparoscopic myomectomy. Obstet Gynecol 1992; 80(5): 884-8.
  3. Pillai R, Yoong W. Posterior colpotomy revisited: A forgotten route for retrieving larger benign ovarian lesions following laparoscopic excision. Arch Gynecol Obstet 2010;281(4):609-11.

Citation

Shah NH, Somani A, Deshmukh M, Paranjpe S. Use of Endoknife in Laparoscopy- An Innovation. JPGO 2018. Volume 5 No.1. Available from: http://www.jpgo.org/2018/01/use-of-endoknife-in-laparoscopy.html

Laparoscopic Excision Of A Huge Ovarian Cyst In A Teenager

Author Information
Shah NH*, Shah R**, Kshirsagar S**
(*Director, **Fellow in Gynecology endoscopy, Vardann Multispeciality Hospital)

Abstract

Ovarian cysts were conventionally managed by laparotomy or minilaparotomy incisions. Laparoscopic management of ovarian cysts is gaining importance due to the various benefits and minimal invasive approach, especially in younger patients. Here we present a case of an 18 year old girl with a 20×20×15 cm size ovarian cyst treated laparoscopically.

Introduction

Ovarian cysts are common and symptomatic if large in size and pose a risk of rupture or torsion. Although studies have compared small to medium size cysts managed by laparotomy, minilaparotomy or laparoscopy, there aren’t many similar cases reported for large cysts.[1] Managing large ovarian cysts, extending supraumbilically, poses a challenge in terms of trocar entry and specimen retrieval. Below case presented is such of a 32 weeks size simple ovarian cyst operated by endoscopy. 

Case Report

An 18 year old unmarried nulligravid female presented with dull aching pain and lump in abdomen progressing over two years. She had attained menarche at 14 years age and had regular cycles. There were no bowel or bladder disturbances, no acute excruciating pain or vomiting, no fever, loss of appetite or loss of weight or family history of ovarian or breast cancer. Per abdomen a 36 weeks size mass was palpated which was cystic, non-tender and had side to side mobility. Trans abdominal ultrasound reveled a 20×20×15 cm size simple ovarian cyst. CA-125 levels were 14 IU/ml. Provisional diagnosis was of a benign ovarian cyst.


Figure 1.  Per abdomen 36 weeks' size cyst.

She was taken for surgery under general anesthesia. A supraumbilical 10 mm trocar was inserted by direct entry technique and two 6 mm accessory ports were taken under vision on the left lower and upper abdomen respectively. Intraoperative findings showed a large left ovarian cyst with a smooth regular outer surface reaching up to the under surface of the liver with the left fallopian tube stretched over its surface. There was no ascites or adhesions. The uterus and the right adnexa appeared normal.


Figure 2. Cyst reaching up to the liver.

Cyst was punctured and 6.5 liters clear fluid was aspirated under vision while preventing spillage. No septae or solid areas were encountered. Thin cyst wall remained without any identifiable ovarian tissue at the hilum. A unilateral salpingo-ovariectomy was done by lifting up the cyst wall and coagulating its base and ovarian and infundibulopelvic ligaments using bipolar cautery and cutting with harmonic scalpel. Specimen was retrieved through the left lower port after enlarging it to 1.5 cm. Peritoneal irrigation was done with 2 liters normal saline and hemostasis was confirmed. The procedure lasted 70 minutes. Postoperative condition of the was good and she was discharged on day 2. Histopathology reported benign serous cystadenoma.


Figure 3. 6.5 liters clear fluid aspirate.


Figure 4. Decompressed cyst.


Figure 5. Salpingo-ovariectomy using harmonic scissors.


Figure 6. Specimen of cyst wall and fallopian tube.


Figure 7. Final laparoscopic view.


Figure 8. Post-operative abdomen view showing port sites.

Discussion

Ovarian cysts are common pelvic masses in patients of fertile age (27.5% incidence), most are benign, serous cystadenoma being the most common variety (40.2%).[2)] Large ovarian tumors (> 10 cm) are more common in the extremes of the reproductive age.[3]
A rapid increase in size, a poor general condition of patient or a positive family history prompts towards malignancy. Cases should be evaluated further by an abdominopelvic imaging and serum tumor markers before planning the surgery.[3]
The earliest study by Salem et al in 2002 mentioned 15 large ovarian cysts managed laparoscopically through the Palmer point entry, cyst puncture and conventional specimen retrieval.[4] Study by Eltabbakh et al in 2008 of 33 large benign cysts resulted in two cases being converted to laparotomy. Unilateral salpingo-ovariectomy was done in 16 cases and bilateral in 4, cystectomy in 2 and hysterectomy with bilateral adnexectomy in 9 cases. Cysts were removed vaginally after aspiration through vagina in 11 cases, by a lower quadrant incision in 5 and through the umbilical incision in 15 cases.[5]
A similar case described by Rabbani et al used Veress intraumbilical needle to first aspirate the cyst followed by reinsertion and insufflation.[6] Whereas 5 cases reported in 2013 used open method for trocar entry and aspiration under vision followed by oophorectomy.[7]
Two technical difficulties for the endoscopist are inadvertent rupture and specimen retrieval. Entry can be facilitated by direct trocar entry or Veress insufflation. This can be intraumbilical, supraumbilical, via Palmer’s point or Lee Huang point or an open blunt trocar entry by Hasson’s technique. A Cochrane review update of 2015 found no advantage of either technique over the other.[8]
In multivariate analysis of epithelial ovarian cancer intraoperative spillage demonstrated no adverse effect on prognosis. Nevertheless, spillage should be avoided and thorough wash should be given.[9] Endobag extraction is the best method accepted unanimously although these are retrospective studies.[10] In a large study conducted by Lim S et al in 2012 for large malignant ovarian cysts treated laparoscopically in 81 patients, it was found that tumor size was not a limiting factor even in malignancies.[11]
A review of 20 articles encompassing 852 cases by Eltabbakh in 2016 estimated complication rates as 1.9 %. Laparoscopic conversion was in 3.9 %  cases. Borderline ovarian tumors and ovarian cancers were  2.5 %  and 3.1 % respectively.[12]
The most important aspect of surgical management by laparoscopy is patient selection. With considerable care and expertise laparoscopy can be defined as a gold standard for large ovarian masses.

References
  1. Yuen PM, Yu KM, Yip SK, Lau WC, Rogers MS, Chang A. A randomized prospective study of laparoscopy and laparotomy in the management of benign ovarian masses. Am J Obstet Gynecol. 1997;177(1):109–14.
  2. Pudasaini S, Lakhey M, Hirachand S, Akhter J, Thapa B. A study of ovarian cyst in a tertiary hospital of Kathmandu valley. Nepal Med Coll J. 2011;13(1):39–41.
  3. Shindholimath V V, Jyoti SG, Patil K V, Ammanagi AS. Laparoscopic management of large ovarian cysts at a rural hospital. J Gynecol Endosc Surg. 2009 Jul;1(2):94–7.
  4. Salem HA. Laparoscopic excision of large ovarian cysts. J Obstet Gynaecol Res. 2002 Dec;28(6):290–4.
  5. Eltabbakh GH, Charboneau AM, Eltabbakh NG. Laparoscopic surgery for large benign ovarian cysts. Gynecol Oncol. 2008 Jan;108(1):72–6.
  6. Rabbani I, Wynn JS, Hickling DJ. Laparoscopic excision of a large ovarian cyst. Gynecol Surgery 2007;4(3):225–7.
  7. Alobaid A, Memon A, Alobaid S, Aldakhil L. Laparoscopic management of huge ovarian cysts. Obstet Gynecol Int. 2013;2013:380854.
  8. Ahmad G, Gent D, Henderson D, O’Flynn H, Phillips K, Watson A. Laparoscopic entry techniques. Cochrane Database Syst Rev. 2015 Aug 31;8:CD006583.
  9. Dembo AJ, Davy M, Stenwig AE, Berle EJ, Bush RS, Kjorstad K. Prognostic factors in patients with stage I epithelial ovarian cancer. Obstet Gynecol. 1990;75(2):263–73.
  10. Duggal BS, Tarneja P, Sharma RK, Rath SK, Wadhwa RD, VSM. Laparoscopic Management of Adnexal Masses. Med J Armed Forces India. 2004;60(1):28-30.
  11. Lim S, Lee KB, Chon SJ, Park CY. Is tumor size the limiting factor in a laparoscopic management for large ovarian cysts? Arch Gynecol Obstet. 2012;286(5):1227-32.
  12. Eltabbakh G. Laparoscopic Surgery for Large Ovarian Cysts-Review. Trends Gynecol Oncol  2016; 1(3): 109.
Citation

Shah NH, Shah R, Kshirsagar S. Laparoscopic Excision Of A Huge Ovarian Cyst In A Teenager. JPGO 2018. Volume 5 No.1. Available from:http://www.jpgo.org/2018/01/laparoscopic-excision-of-huge-ovarian.html

Couvelaire Uterus Due To Chronic Abruption With a Successful Pregnancy Outcome

Author Information

Deshpande PS*, Honavar PU**, Gupta AS***
(* Third Year Resident, ** Assistant Professor, *** Professor, Department of Obstetrics and Gynecology,Seth GS Medical College and KEM Hospital, Mumbai, India.)

Abstract

Abruptio placentae is one of the dreaded cause of antepartum hemorrhage complicating 0.5-1 % or 1 in 200 pregnancies. The symptoms of abruption can mimic preterm labor or the process if chronic can be asymptomatic thus making clinical judgment the mainstay in diagnosis of majority of the cases.
Here we present a case of chronic abruption who presented as a case of threatened preterm labor. A routine ultrasonography (USG) detected a retroplacental clot and finally intraoperatively couvelaire uterus was seen confirming the diagnosis. 

Introduction

Abruptio placentae is defined as the premature separation of partial or complete placenta after 20 weeks of gestation any time before the delivery of the fetus. Abruption can either be revealed or concealed with bleeding seen per vaginum or as a retroplacental clot respectively. According to the severity of the presentation, abruption can be acute or chronic with the diagnosis of chronic made, as in our case, by USG.  As the symptoms of chronic abruption are very non-specific, good clinical judgment and diligent fetal monitoring is needed to prevent the maternal and fetal morbidity and mortality that may result otherwise.

Case Report

A 27 year old G4A3 at 34 weeks of gestation with bad obstetric history was referred to our hospital as a case of threatened preterm labor with breech presentation. On admission, her general condition was fair and vital parameters were stable with a pulse of 86/ min. and BP of 120/80 mm of Hg. There was no pallor, icterus or pedal edema. She was complaining of intermittent abdominal pain,  but there was no vaginal bleeding and she was perceiving fetal movements well. On per abdominal examination she was 34 weeks, but the  presentation could not be made out. Uterus felt a little tense but was not tender and there were no uterine contractions. On per vaginum examination os was closed, posterior and tubular. There was no show or bleeding.
She was already on oral Aspirin and intramuscular depot progesterone support in view of bad obstetric history from the first trimester of her pregnancy. She also gave history of admission 5 days back in a tertiary care center in view of decreased fetal movements. She was given complete dose of parenteral steroids for fetal lung maturity. Her serological and biochemical investigations were normal. Her USG was suggestive of single, live gestation of 32 weeks, a fundo-posterior placenta, no retroplacental clots and the fetus in transverse lie. She took discharge against medical advice and presented 3 days later again with pain in abdomen to a primary health care facility who referred her to our tertiary care hospital.
USG was done for lie and presentation and it showed a single, live gestation of 32 weeks and a fundo-posterior placenta with a 11.3x6.4x11cm heterogeneously hyperechoic lesion suggestive of large retroplacental hematoma. Urgent non stress test was done and a non reactive pattern was seen with poor beat to beat variability but without any accelerations or deceleration's. In the investigations that were done, her hemoglobin was 11 gm%, platelet count was1.3 lakhs/ cc, prothrombin time was 12.5/13.1, and INR was 0.91. Her creatinine was 0.8 mg/dl and SGOT and SGPT were 32 and 41 U/L respectively. Thus, as biochemical parameters and coagulation profile was normal she was taken up for emergency lower segment cesarean section. On entering the peritoneal cavity almost the entire external surface of the uterus both anteriorly and posteriorly appeared bluish black suggestive of couvelaire uterus. (Figure.1,2)


Figure 1: Anterior surface of the couvelaire uterus.


Figure 2: Posterior surface of the couvelaire uterus.

She delivered a female child of 1.9 kg with an Apgar of 9/10. Placenta was posterior and around 630 gm of clots were seen from behind the placenta (concealed abruption). 40% of the placental surface was separated. They were old clots and the placental surface that was seen looked normal. No hematoma was seen extending into the broad ligament. Bilateral tubes and ovaries were normal. There was no hemoperitoneum. Uterine incision was closed and intravenous oxytocin was given (20 units in 500 ml Ringer Lactate). Uterus was well contracted. Hemostasis was achieved and abdomen was closed. Post operatively one unit of whole blood was transfused and rest of her recovery was uneventful.

Discussion

Abruption commonly presents with acute symptoms of abdominal pain and vaginal bleeding, however, 10-20 % of the cases present with only preterm labor with no or scanty vaginal bleeding.[1] The classical triad of abdominal pain, vaginal bleeding and uterine hypertonus is present in only 10 % cases of abruption.[2]  Although acute and chronic abruption has not been defined explicitly, sudden and > 50 % separation of the placenta leads to sudden fetal death. However, if the process is chronic and self limiting, the fetus might survive as compensatory mechanisms act.[3]
In our case, as it was a couvelaire uterus, resulting in increased uterine tone the presentation could not be delineated and so luckily, she was sent for a USG to confirm lie and presentation that picked up the retroplacental clot. 40% of the placenta had separated and a clot was seen thus the process might have started 5 days back when she got admitted as a case of threatened preterm. It went undiagnosed at that time and as the symptoms subsided she took discharge against medical advice. The process restarted and then as she developed pain again she came back to the hospital.  As it was a chronic process, the feto-placental insufficiency set in slowly and hence the kick count (which is a subjective parameter) was not affected but the non stress test was non reactive. Thus, in cases of chronic abruption, the patient can present with non-specific symptoms, with a normal coagulation profile and stable vital parameters. In cases of concealed abruption, abdominal pain rather than vaginal bleeding is ominous as large surface area of the placenta may be separated resulting in underlying fetal distress as it was seen in our patient.[4]
If chronic abruption develops in the second trimester it leads to the development of Chronic Abruption Oligohydramnios Sequence (CAOS) in 60% patients.[5] This sequence is characterized by vaginal bleeding, oligohydramnios and intact membranes. However in our patient the abruption was seen at a later gestational age and there was no evidence of any recent vaginal bleeding or oligohydramnios. In our case as the abruption happened in 3rd trimester, and timely steroid doses were administered the neonatal outcome was good. 
The symptoms of abruption also depend on the location of the placenta. In a retrospective study done by Suzuki comparing the outcomes of abruption in anteriorly versus posteriorly implanted placentas, it has been observed that vaginal bleeding, abdominal pain and non- reassuring fetal heart status; all are less seen with posteriorly located placentas. Thus, posteriorly placed placentas can be silent killers. Also, the need for blood transfusion, chances of DIC and blood loss are less in posterior placental abruptions’ signifying that they are less severe.[6] 
Couvelaire uterus or uteroplacental apoplexy is a severe form of abruption where there is widespread extravasation of blood into the uterine musculature and beneath the serosa. Incidences of hemoperitoneum caused due to abruption with seepage of blood through the fallopian tube have also been reported.[2] Couvelaire uterus can only be diagnosed intraoperatively and confirmed on histology, hence its true incidence is not known. However, it complicates 5% of the cases of abruption. A study of 17 cases of couvelaire uterus in severe abruption by Cheng et al stated that the risk of post partum hemorrhage, hemorrhagic shock, disseminated intravascular coagulation, intrauterine fetal demise and neonatal asphyxia and demise is more with couvelaire uterus and abruption versus only abruption.[6]  In olden days couvelaire uterus mandated hysterectomy, however now it is well known that the condition resolves on its own.

Conclusion

Thus, chronic abruption is usually a radiological diagnosis and needs prompt delivery of the baby. This silent condition can be a catastrophe in waiting, if prompt diagnosis and delivery and not done as hypoxic injury to the fetus or decompensation of the mother can occur.

References
  1. Ananth CV, Kinzler WL. Placental abruption: Clinical features and diagnosis. Available from: https://www.uptodate.com/contents/placental-abruption-clinical-features-and-diagnosis.
  2. Bertholdt C, Vincent-Rohfritsch A, Tsatsaris V, Goffinet F. Placental Abruption Revealed by Hemoperitoneum: A Case Report. Am J Perinatol Rep 2016;6(4):e424-e426. 
  3. Deering SH. Abruptio Placentae. Available from: https://emedicine.medscape.com/article/252810-overview#a3.
  4. Mei Y, Lin Y. Clinical significance of primary symptoms in women with placental abruption. J Matern Fetal Neonatal Med. 2017; Jul 6:1-4.
  5. Elliott JP, Gilpin B, Strong TH Jr, Finberg HJ. Chronic Abruption- Oligohydramnios Sequence. The Journal of reproductive medicine.1998; 43(5):418-422.
  6. Nagayama C and Suzuki S. Influence of placental position on outcome in patients with placental abruption. J Nippon Med Sch. 2006;73(60):351-353.

Citation

Deshpande PS, Honavar PU, Gupta AS. Couvelaire Uterus Due To Chronic Abruption With a Successful Pregnancy Outcome. JPGO 2018. Volume 5 No.1. Available from: http://www.jpgo.org/2018/01/couvelaire-uterus-due-to-chronic.html

Borderline Mucinous Cystadenoma Of Ovary

Author Information

Jain N*, Chaudhari HK**, Mali K***
(* Second year resident, ** Associate professor, *** Assistant professor. Department of Obstetrics and Gynecology, Seth G.S. Medical college and KEM hospital, Mumbai, India.)

Abstract

Among ovarian tumors epithelial cell tumor are the most common tumors. Among epithelial tumors serous type is the most common tumor. Less common types are mucinous, clear cell, endometroid, transitional cell (Brenner), and undifferentiated tumors. Mucinous tumors are either benign, borderline, or malignant. Borderline tumor is of low malignant potential. Here we report a case of borderline mucinous cystadenoma in a 28 year old female para3, living3, one ectopic pregnancy, who had pain in abdomen since 5 months. She presented to us in the outpatient department (OPD) and was managed successfully by exploratory laparotomy with left salpino-oophorectomy followed by total abdominal hysterectomy and omentectomy.

Introduction

Ovarian cancer is third most common cancer in women after cervical and breast cancer.  Ovarian tumors are classified on the basis of histology. Epithelial ovarian tumor is most common; in 90 % cases.[1] According to histological type; 75-80% of epithelial cancers are serous. Less common types are endometrioid (10%), mucinous (5%), clear cell (5%), transitional cell (Brenner <1%), and undifferentiated carcinoma <1%.  Borderline mucinous  tumors of low malignant potential are difficult to identify, detailed study of multiple sections are needed. Well differentiated mucinous epithelium may be seen immediately adjacent to a poorly differentiated focus. These cystic ovarian tumors have loculi lined with mucin secreting epithelium. The lining epithelial cells contain intracytoplasmic mucin and resemble those of endocervix and pylorus or intestine. They may reach enormous size, filling the entire abdominal cavity. Management of borderline mucinous tumor is surgical removal.

Case Report

A 28 year old woman, married since 8 years, para 3 living 3, one ectopic pregnancy came to OPD with complaints of pain and abdominal distension since 5 months. There was no history of any other medical or surgical illness. Menstrual cycles were regular. She had all 3 normal deliveries. She had an ultrasonography (USG) report on presentation that showed a right renal calculus, and a left adnexal teratoma extending to right side. Uterus was anteverted normal size, right ovary was normal in size, left ovary was about 18x10x14 cm sized, a heteroechoic soft tissue mass was noted and in the midline there were hypoechoic necrotic areas and areas of calcification. This lesion showed internal vascularity. Left ovary was not seen separately .
On examination general and systemic examination were normal. Per abdomen examination showed a 28 weeks size mass arising from the pelvis. There was no guarding, rigidity, or tenderness. On per speculum examination cervix was hypertrophied and deviated to the right side. On  vaginal examination a mass of 28 weeks, arising from the pelvis was felt. Mass was felt in the left fornix, extending upto the abdomen. Uterus was retroverted, and its size could not be assessed. Ultrasonography was repeated. It was suggestive of a multiloculated cystic mass about 16x13x17 cm in size probably arising from the left ovary, left ovary separately not visualized suggestive of a mucinous cystadenoma. Right ovary and uterus were normal in size and appearance. Ultrasonography of  kidney, ureter and  bladder was within normal limits. CT scan showed  left ovarian mass with multiple cysts, some cysts showing hemorrhage. It measured 21x12x19 cm. Few calcification's were present. It was well defined, smooth mass sited on the uterus. Uterus and right ovary were normal. No lymphadenopathy and ascites were seen. CT scan findings were suggestive of left ovarian cystic neoplasm with hemorrhagic cyst.  Tumor marker serum alpha fetoprotein was 1.75 ng/ml, and serum CA125 was 56.2 units/ml.  Serological, biochemical and coagulation profile were within normal limits.
On staging laparotomy approximately 20x20x6.5 cm solid cystic mass arising from the left ovary was seen. Left ovarian mass was removed and sent for frozen section, which was suggestive of borderline mucinous neoplasm. Exploratory laparotomy with left salpingo-oophorectomy followed by total abdominal hysterectomy with omentectomy was  done. Specimen was sent for histopathology. On day 5 patient was discharged.
As per the histopathology report grossly the left ovarian mass was of  20x20x 6.5 cm size  containing solid cystic areas. Cut section showed thick mucinous brownish colored fluid, approximately 500 ml. Cyst was largely solid with few cystic areas. Solid areas were yellow. No solid area in its wall, and wall was smooth. Multiple sections studied showed a multilocular cystic tumor with complex papillary and glandular architecture. The lining epithelium showed mucinous epithelium (intestinal type). The cells showed high N:C ratio and prominent nucleolus. Few goblet cells were also seen. Areas of necrosis mixed with hemorrhage, fibrin and mucin were seen, foamy macrophages and mixed inflammatory cells were also present. No invasion was seen. Endometrium was poorly preserved, myometrium was unremarkable. Cervix showed papillary endocervicitis and squamous metaplasia. Omentum had  mucinous areas of dense neutrophilic infiltrates and congested blood vessels, no tumor cells  were seen.(figure 1 and 2)
Histopathological impression was of a borderline mucinous neoplasm of  the  left ovary.


Figure 1.  Histopathology picture of Borderline mucinous cystadenoma. (Low Power)
Red arrow shows complex papillary and glandular architecture. The lining epithelium shows mucinous epithelium (intestinal type).


Figure 2. Histopathology picture of Borderline mucinous cystadenoma. (High power ). Black arrow shows the cells showed high N: C ratio. Green arrow shows prominent nucleolus with few goblet cells. Red arrow shows complex papillary and glandular architecture. The lining epithelium shows mucinous epithelium (intestinal type). No invasion is seen.

Discussion

Taylor first described as other type tumor that was different from benign as well as malignant epithelial ovarian tumor.[2] WHO assigned name “borderline” in 1973 to those tumors having morphological criteria with absence of stromal invasion.[3] Presently, borderline ovarian tumors are staged according to international federation of gynecology and obstetrics (FIGO) classification of ovarian cancer. Mucinous tumor represent about 8 to 10 % of epithelial tumor.[1] Mucinous tumors are divided into three types; benign, borderline, and malignant. Borderline tumors remain confined to the ovary for long periods, occur predominantly in premenopausal women  between 30 to 50 years of age and are associated with very good prognosis.[3]  Principle treatment of borderline tumor is surgical resection of the primary tumor. There is no evidence that adjuvant chemotherapy and radiotherapy improve survival. If frozen section determine the histology of borderline and patient is  premenopausal and desires to preserve ovarian function in that case conservative surgery like  unilateral oophorectomy or cystectomy may be done.[4] As per Ayhan et al., borderline ovarian tumor can safely be treated with conservative surgery.[5] As per Guvenal et al’s study, 3% recurrence rate was seen in patients who underwent radical surgery whereas 8.3% recurrence rate occurred in patients who underwent conservative surgery.[6] As per Boran et al. study no recurrence were reported in patients who underwent radical surgery, whereas the recurrence rate was 6.5 % in patients who underwent conservative surgery.[7]

Conclusion

Borderline tumors are considered to be precursors of low-grade ovarian cancers. Accurate diagnosis and staging facilitate optimal patient management. If conservation of fertility is not required then radical surgery is preferable.

Acknowledgement

We thank Dr. Mona Agnihotri, pathologist at KEM hospital for histopathology report and histopathological images.

References
  1. Scully RE, Young RH, Celment PB. Tumours of the ovary, maldeveloped gonads, fallopian tube and braod ligament. In Rosai J, Sobin LH. editors. Atlas of Tumor Pathology, 1st edition. Washington, D.C.: Armed Forces Institute of Pathology 1998;3rd series. fascicle 23: pg 81.
  2. Taylor HC Jr. Malignant and semi malignant tumours of the ovary. Surg Gynecol 1929; 48:204–230.
  3. Gershenson DM. Clinical management potential tumours of low malignancy. Best Pract Res Clin Obstet Gynaecol. 2002;16(4):513–527.
  4. Berek JS, Friedlander ML, Hacker NF. Epithelial ovarian, fallopian tube and peritoneum. In Berek JS,Hacker NF, editors. Berek and Hacker’s Gynaecologic Oncology. 6th edition.Philadelphia: Wolters Kluwer Health – Lippincott Williams & Wilkins 2015; pg. 443-508.
  5. Ayhan A, Guvendag GES, Guven S, Kucukali T. Recurrence and prognostic factors in borderline ovarian tumors. Gynecol Oncol. 2005;98(3):439–445.
  6. Guvenal T, Dursun P, Hasdemir PS, Hanhan M, Guven S, Yetimalar H, et al. Effect of surgical staging on 539 patients with borderline ovarian tumors: a Turkish Gynecologic Oncology Group study. Gynecol Oncol. 2013;131(3):546–550.
  7. Boran N, Cil AP, Tulunay G, Ozturkoglu E, Koc S, Bulbul D, et al. Fertility and recurrence results of conservative surgery for borderline ovarian tumors. Gynecol Oncol. 2005;97(3):845-51.
Citation

Jain N, Chaudhari HK, Mali K. Borderline Mucinous Cystadenoma Of Ovary. JPGO 2018. Volume 5 No.1. Available from:http://www.jpgo.org/2018/01/borderline-mucinous-cystadenoma-of-ovary.html