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Volume 5 Issue 4, April 2018

Editorial - WHO Surgical Safety Checklist

Parulekar SV

Healthcare is broadly speaking preventive and curative. Therapeutic measures are medical surgical and medical. Both of these methods are likely to create complications through negligence. About 50% of the surgical cases which caused harm to the patient by surgery were due to preventable actions or unsafe surgical practices. Hence WHO created a document called 'WHO Surgical Safety Checklist'. Working groups of international specialists reviewed the literature and the experiences of clinicians, the results of which were used to develop this checklist by WHO in collaboration with the Harvard School of Public Health. Global implementation of this 19-item tool was recommended by WHO. The job done by WHO was indeed commendable. A good surgical team always has a checklist in mind, and it was so even before WHO brought out the document in 2009. But all teams or team members not always so careful, and a point or two can be left out. A checklist prevents such a mishap. Hence I am all in favor of a checklist. Our readers will remember that we have devoted an entire issue to checklists in department of Obstetrics and Gynecology of K E M Hospital, Mumbai, India. A surgical checklist was also included in it.

A critical review of the WHO checklist is in order nine years after the original document was published. There are three sections in the checklist - before induction of anesthesia, before skin incision, and before the patient leaves the operation theater (OT).

As per the checklist in its existing form, before induction of anesthesia, at least a nurse and anesthetist have to see that the patient confirms his/her identity, procedure, surgical site and consent. Actually the surgeon has to be the principal person, and all three of them have to be a part of the procedure. Marking the site is a little superfluous, as an error can be committed at this stage just as the stage of operating without prior marking. Check of anesthesia machine should also include disposable kits, drugs and anesthetic gases. Checking functionality of pulse oximeter alone is not adequate. All equipment needs to be checked, including mulipara monitors, electrocautery, special surgical instruments (based on nature of the operation, such as endoscopes, monitors, microscopes, and suction units). The estimation of expected blood loss is always a matter of dispute between anesthetists and surgeons when it has already occurred. Preoperative estimate needs to be determined during the planning stage of the operation and not before induction of anesthesia. It should be based on statistics and not opinion of the surgical team. The checklist should not be to see if the estimated figure is entered in the case paper, but to confirm that blood compatible with the patient's blood is available when the estimated blood loss is more than 500 ml.

Prior to making the skin incision, the checklist states all team members have introduced themselves by name and role. This is perhaps necessary when OT slots are given by prior booking to different teams. But it is superfluous when the team is fixed at least for the day, when all team members know one another. Confirming the patient's name at this stage is bordering on ridiculous, as anesthesia should never been given unless the patient has been identified first. Discovering that a wrong person lies anesthetized on the operation table implies that it is still OK to back out. This stage should never be reached.
Medicolegal negligence lawsuits cannot be prevented even if the patient is taken out of anesthesia. Checking how long the operation will last needs to be checked before administration of anesthesia, so that the anesthesiologist can plan accordingly. Checking it afetr induction of anesthesia serves no purpose. A nurse checking that all equipment required is available, properly functional and sterile at this stage is too late. She has to do it before the patient enters the OT. Recommendations for before patient leaves operating room also leave much to be desired. The nurse verbally confirming the name of the procedure after is complete does not help anyone. God forbid, but if it discovered at this stage that a wrong procedure has been performed, imagine the consequences. Completion of instrument, sponge and needle counts needs to be done before the patient's operative area is closed, such as the abdomen and thorax. If anything is found missing after closure of the surgical incision, one has to open the patient again. That not only increases the patient's morbidity but also will lead to litigation for negligence, which the surgical team will lose.

I hope WHO notes the need for a newer version of the original checklist. I also hope that it involves in this venture more surgeons and anesthetists rather than preventive medicine or public health specialists.

Successful Outcome In A Case With Previous Upper Segment Cesarean Section

Author Information


Bora A*, Prasad M**, Gupta AS***.
(* Second year Resident, ** Assistant Professor, *** Professor and Head, **** Professor, Department of Obstetrics and Gynecology, Seth GS Medical College and KEM Hospital, Mumbai, India.)

Abstract

The incidence of repeat cesarean sections has increased over the past few decades. One of the biggest risks is the risk of uterine rupture, making planning of an appropriate time for delivery of such a pregnancy a major decision. Such a case of previous two cesarean sections with history of an upper segment incision has been discussed here.

Introduction

The rate of cesarean delivery has increased over the past decade from 5% in 1970 to 32.3% in 2008 [1] and a major factor behind this increase is a decrease in the number of vainal births after cesarean section (VBAC). This has lead to scenario where we encounter a vast majority of patients with previous two cesarean sections, and the risks and management may vary in each individual case.

Case Report

A 25 year old G3P2L1 registered in the antenatal department of our hospital in view of a previous upper segment cesarean section. She had one living issue and one neonatal death, and was referred to our hospital for further care. She was antenatally registered in a private hospital at 6 weeks of gestation and was compliant to antenatal care. Hemoglobin was 12.9 gm%, thyroid profile was within normal limits, fasting and postprandial blood sugars were 90 mg/dl and 100 mg/dl respectively; there were no fetal malformations and placenta was posterior. 
Her first pregnancy was 1 year after marriage, a spontaneous conception and she was managed at a rural peripheral hospital. At 9 months of gestation, she had preterm premature rupture of membranes and cesarean section was performed. Documentation regarding this pregnancy was not available. Male child of approximately 3 kg was born and was well till 3 months of life, when the child succumbed to severe gastroenteritis, details of which was not known.
In her second pregnancy, the conception was spontaneous and approximately one year after the first pregnancy completion. Antenatal follow up details were not available. She denied being offered a trial of scar. She was delivered by cesarean section and detailed intraoperative notes were not available. However, there was a line diagram showing previous upper segment rupture and another showing the sacculation of membranes and intrauterine presence of the fetus (figures 1 and 2). Previous scar had ruptured, but placenta was not at the scar. Omental adhesions were present. However, there was no mention of type or location of incision taken in this procedure. This pregnancy was a live birth fortuitously as the rupture of the upper segment scar had not resulted in expulsion of fetus into the peritoneal cavity nor had it affected the placenta.  
At 35 weeks of gestation in her current pregnancy, she presented to the emergency department with the complaints of pain in abdomen. There was no leaking or vaginal bleeding. Pulse was 80 beats per minute, blood pressure was normal and systemic examination was unremarkable.  Uterus was approximately 36 weeks in size and no uterine activity was felt. There was no abdominal tenderness. Fetal heart sounds on doppler were 140 beats per minute, and non stress test was reactive. She was scheduled for a lower segment cesarean section after completion of steroid cover owing to her previous history of upper segment scar and its dehiscence in her previous pregnancy.
Intra operative, flimsy adhesions were seen between the anterior uterine wall and lower uterine segment. Two transverse scars were seen, separate from each other. One scar was seen in the lower uterine segment but slightly high up and one in the body of the uterus approximately 2 cm below the insertion of the fallopian tubes. The scars were thinned out but there was no sign of dehiscence.(figure 3) Fetus was delivered by a lower segment incision. Her post operative period was uneventful. She was discharged on day 4 post delivery.

Figure 1. Line diagram drawn by clinician operating second CS indicating previous upper segment ruptured incision.

Figure 2. Line diagram drawn by clinician operating second CS showing sacculation of the membrane (blue arrows) and intrauterine presence of the fetus. (lateral view)


Figure 3. Intraoperative image showing the previous healed transverse scar (white arrows) and the fibrous edge now incised of the previous lower segment scat (yellow arrows)


Figure 4. Intraoperative image after suturing lower segment incision. Upper incision in the middle part of the uterine body. (white arrow)

Discussion

VBAC is contraindicated in women with previous classical cesarean scar due to the high risk of uterine rupture. Our patient had one prior upper segment cesarean section and another prior lower segment cesarean section. This patient was not offered trial of labor in this pregnancy (third pregnancy) as diagrammatic details of her second pregnancy were available. Whether the patient was offered trial of labor during her second pregnancy is not clearly known. Intra operative finding showed two incisions on the uterus, both transverse one in the middle part of the uterine body and one in the lower segment as seen in figure 3. In the second cesarean birth the diagrammatic notes document the upper transverse scar rupture. It seems that the fetus was delivered not through this rupture but from a fresh lower segment scar (referral note had a mention of the same) through which we performed the current cesarean birth. However, since the upper segment scar had ruptured in the second pregnancy it is difficult to contemplate why the treating surgeon did not deliver that fetus through the ruptured upper segment scar but placed a fresh lower segment incision. This was confirmed by the intraoperative findings (figure 3). Figure 2 indicates that the fetus in the second pregnancy was a breech presentation. Probably an attempt to deliver the breech through the ruptured transverse upper segment incision may not only have resulted in difficulties, but trauma to the fetus and extension of the ruptured segment may have been the cause for not delivering the fetus through the rupture site. The first delivery through a transverse incision in the middle part of the body must have also resulted in difficulty in delivery of the fetus as neither the head nor the buttocks would be available for grasping unless the fetus was in the transverse lie. However, details of the same are not available and this is mere speculation. Usually the incision for a classical cesarean section is vertical on the uterine body. The presence of the transverse scar in the middle part of the upper segment probably suggests that the 1st surgeon was not adequately trained in obstetrics to identify the lower segment and instead of placing this transverse incision in the lower segment placed it in the middle of the upper segment. A catastrophic mistake would be taking a repeat upper segment incision. 
However, the recent RCOG guidelines state that patient with two or more prior lower segment cesarean deliveries must be counseled by a senior obstetrician and may be offered VBAC; only after specific points of discussion which include risk of uterine rupture, maternal morbidity and individual likelihood of successful VBAC (like giving a history of prior vaginal delivery) have been taken into account. It has also been opined that such labor should be undertaken only in a center with suitable expertise and immediate possible recourse to surgical delivery. However, this recommendation is grade C.[1]  The ACOG guidelines have also mentioned somewhat similar guidelines.[2,3] However, previous classical or “T” incision, prior uterine rupture, extensive transfundal uterine surgery are not considered as candidates for VBAC. Patients with a previous low vertical uterine incision are not found to be at an higher risk for uterine rupture as compared to the ones with a previous low transverse uterine incision.[4]
It is to be highlighted that above recommendations are only for those whose previous scars are well documented. Our case highlights the fact that documentation of details of prior operative procedures, and knowledge of a prior scar is very important before deciding the route of delivery. Absence of documentation of the first surgical procedure was the most probable cause of the rupture seen in the second pregnancy. In this patient's second pregnancy, the clinician was probably unaware of the higher incision. Many a times the patient or the relatives are unable to narrate the entire sequence of events or the exact details of a prior condition or a prior surgery. In such cases, previous documents, papers amount to a valuable source of information. The first time a patient presents to a doctor, meticulous history taking is mandatory. Reviewing the details in every visit, with the intent of eliciting any extra information is also recommended. Documentation of the second delivery surgery notes allowed us to deliver her before term (at 35 weeks of gestation age) so as to circumvent repeat scar rupture of the upper segment.
Some situations where transverse fundal uterine incisions were used successfully have found mention in literature. Taga et al conducted a study involving seven cases of ELBW infants delivered through transverse fundal incisions; wherein the placenta was covering the entire anterior wall.[5] A similar case has been published by Kotsuji et al, where a transverse fundal incision was used in a case of placenta previa to reduce the intraoperative blood loss.[6]
Classical teaching mandates taking of the incision in the lower uterine segment by identifying the junction of the uterovesical fold, dissecting the bladder away, and incising below the part which is the junction of the upper and lower segment. An incision at the upper part of the lower segment has been shown to reduce blood loss, enhance uterine retraction, predispose to fewer complications, is easier to repair, precludes bladder adhesion to the suture line and reduce operation time.[7]
However, when such different methods (incisions) are used, there should be meticulous documentation; which will aid the clinician treating the next pregnancy.
In women after two lower segment cesarean scars, scheduling an elective cesarean section at around 39 weeks compared to at around 38 weeks is associated with an higher risk for maternal adverse outcome with no apparent advantage in terms of neonatal outcome.[8]
To conclude, there was no dilemma in this pregnancy (third) in scheduling the patient for a cesarean section due to previous operative findings. However, the dilemma in the cases where no intraoperative details are available is highlighted. These clinical decisions should be modified on individual presentations.

References
  1. RCOG Green Top Guideline No 45. Birth after Previous cesarean birth. Available at:  www.rcog.org.uk/globalassets/documents/guidelines/gtg_45.pdf 
  2. ACOG 2017 Guidelines: Who is a Candidate for a Vaginal Birth after Cesarean Section? ObG Project. Available from : https://www.obgproject.com/2017/08/09/candidate-vaginal-birth-cesarean-section/
  3. American College of Obstetricians and Gynecologists. ACOG Practice bulletin no. 115: Vaginal birth after previous cesarean delivery. Obstet Gynecol. 2010 Aug;116(2 Pt 1):450-63
  4. Shipp TD, Zelop CM, Repke JT, Cohen A, Caughey AB, Lieberman E. Intrapartum uterine rupture and dehiscence in patients with prior lower uterine segment vertical and transverse incisions. Obstet Gynecol. 1999; 94(5 Pt 1):735–40.
  5. Taga A, Kondoh E, Hamanishi J, Kawasaki K, Fujita K, Mogami H, et al. Transverse fundal uterine incision for delivery of extremely low birth-weight infants. J Matern Neonatal Med. 2014; 27(12):1285–7.
  6. Kotsuji F, Nishijima K, Kurokawa T, Yoshida Y, Sekiya T, Banzai M, et al. Transverse uterine fundal incision for placenta praevia with accreta, involving the entire anterior uterine wall: a case series. BJOG. 2013; 120(9):1144–9.
  7. Shao Y, Pradhan M. Higher Incision at Upper Part of Lower Segment Cesarean Section. J Nepal Med Assoc. 2017 52(194):764–70.
  8. Melamed N, Hadar E, Keidar L, Peled Y, Wiznitzer A, Yogev Y. Timing of planned repeat cesarean delivery after two or more previous cesarean sections – Risk for unplanned cesarean delivery and pregnancy outcome. J Matern Neonatal Med. 2014; 27(5):431–8.
Citation

Bora A, Prasad M, Gupta AS. Successful Outcome In A Case With Previous Upper Segment Cesarean Section. JPGO 2018. Volume 5 No.4. Available from: http://www.jpgo.org/2018/04/successful-outcome-in-case-with.html

Abdominal Site Specific Anterior Colporrhaphy

Author Information

(Parulekar SV. Professor and Head, Department of Obstetrics and Gynecology, Seth G S Medical College & K E M Hospital, Mumbai, India.)

Abstract

An anterior colporrhaphy is conventionally done by the vaginal route, as it is convenient and more effective than when done by an abdominal route. But if the patient requires and abdominal hysterectomy for any reason, the anterior colporrhaphy can be done abdominally too. The old technique of excision of a triangular portion of the anterior vagina and closure of the defect is now replaced by a site specific repair, as it is more scientific, more effective, and associated with no disturbance of urinary function as compared to the older technique. One such case is presented here.

Introduction

A cystocele usually begins where the pubovesicocervical fascia between the urinary bladder and vagina tears at its attachment to the uterosacral-cardinal ligament complex (central transverse type) or white line (lateral type). A cystocele is best repaired vaginally. However if the patient needs an abdominal hysterectomy, an abdominal hysterectomy can be done after the hysterectomy. The repair involved excision of a triangular portion of the anterior vagina and closure of the defect. With advent of site specific repair of a cystocel, the principles used in vaginal colporrhaphy are followed in abdominal colporrhaphy too. The technique of an abdominal anterior colporrhaphy in a case of  a central transverse type of cystocele is presented.

Technique

After abdominal hysterectomy, the urinary bladder is dissected off the anterior vagina, up to the lower limit of the cystocele. A teraingular flap of vagina is excised, its base along tha antior cut edge of the vaginal vault, and apex at the lower limit of the cystocele, such that after approximation of the oblique cut edges, the anterior vagina will be re-formed without any redundancy. These edges of the vagina are sutured with sutures of No. 1-0 polyglactin. For an central transverse type of cystocele, the upper edge of the pubovesicocervical fascia (PVCF) is sutured to the uterosacral ligaments, its respective half to the respective side ligament, with interrupted sutures of No. 1-0 polypropylene. The two uterosacral ligaments are approximated in the midline to remove any slack in them and are sutured to the centre of the vault of the vagina, to suspend it and prevent a posthysterectomy vault prolapse. The vault of the vagina is closed with a suture of No. 1-0 polyglactin passed through the anterior and posterior edges.


Figure 1. After an abdominal hysterectomy, the bladder has been dissected downward from the front of the anterior vagina, up to the level of the lower limit of the cystocele. The cut anterior edge of the vault of the vagina is held with Allis' forceps and redundant part of the anterior vagina is being cut.


Figure 2. A triangular portion of the anterior vagina is excised, the apex of the triangle at the lower limit of the cystocele.


Figure 3. lateral edges of the defect in the anterior vagina are approximated with sutures of No. 1-0 polyglactin.


Figure 4. Closure of the anterior vaginal edges is almost complete.


Figure 5. The longitudinal suture line in the anterior vagina is seen.


Figure 6. PVCF between the anterior vagina and the urinary bladder is drawn up.


Figure 7. A suture of No. 2-0 polypolypropylene is passed through PVCF on the left of midline.


Figure 8. The suture passed through PVCF is passed through left uterosacral ligament (LUSL) and tied.


Figure 9. A suture of No. 2-0 polypolypropylene is passed through PVCF on the right of midline.


Figure 10. The suture passed through PVCF is passed through right uterosacral ligament (RUSL) and tied.


Figure 11. A suture of No. 2-0 polypolypropylene is passed through PVCF in the midline, LUSL and RUSL, approximating the two uterosacral ligaments in midline to each other and PVCF.


Figure 12. Fascial defect in midline between folded portions of the PVCF is closed with a simple suture of No. 2-0 polypolypropylene, to prevnt formation of cystocele through the defect in future.


Figure 13. Two uterosacral ligaments are approximated in midline with a simple suture of No. 2-0 polypolypropylene, to remove any slack in them and improve support to the vault of the vagina.


Figure 14. Anterior colporrhaphy and vault suspension are complete. The open vault of vagina is seen (arrows).

Figure 15. The vault of vagina has been sutured with No. 1-0 polyglactin suture.

Discussion

Uterovaginal prolapse affects up to 50% of women.[1] About 11% of all women are likely to require repair of such a prolapse.[1] with a lifetime risk of undergoing surgery for POP or incontinence of 11% .[1] The anterior vagina and bladder base are separated by PVCF, which is attached to the uterosacral-cardinal ligament complex in the center and the white line over the obturator internus fascia laterally. It connects the vagia to the bladder and urethra in front and supports them. If it tears from its attachment(s) a cystocele forms. Tear from the ligament complex produces a central transverse type of defect, as was seen in the case presented. Tear from the white line results in a lateral type of cystocele, which can be uni- or bilateral.[2-4] Such tears occur as a result of childbirth trauma.[5] 

Conventioanl repair of a cystocele involved plication of the PVCF in the midline, irrespective of the type of defect present. It was not scientific, as it did not address the pathology. Hence it was more likely to fail.[6] It also disturbed urinary physiology and produced postoperative urinary symptoms. It has largely been replaced by a site specific repair.[7-10] It is convenient to perform the operation vaginally, as that is the direction in which the vagina passes with prolapse. It is also more likely to be complete due to ease of access.[11] But sometimes an abdominal colporrhaphy has to be done, as when an abdominal hysterectomy is necessary. To perform a colporrhaphy vaginally first would entail traction on sutures and bleeding during abdominal hysterectomy. To perform vaginal colporrhaphy after an abdominal hysterectomy is inconvenient as the patient needs to be repositioned, and a different set of instruments is required. An abdominal colporrhaphy is a suitable option in such cases. The old technique of excision of a triangular part of the anterior vagina and approximation of the cut edges is now replaced by a site specific anterior colporrhaphy.[12] The technique gives satisfying results as shown in the illustrations in this article.[13]

Acknowledgment

I thank Dr Maitreyee Parulekar for taking intraoperative pictures.

References
  1. Olsen AL, Smith VJ, Bergstrom JO, Colling JC, Clark AL. Epidemiology of surgically managed pelvic organ prolapse and urinary incontinence. Obstet Gynecol. 1997;89(4):501–506. doi: 10.1016/S0029-7844(97)00058-6. 
  2. Summers A, Winkel LA, Hussain HK, Delancey JO. The relationship between anterior and apical compartment support. Am J Obstet Gynecol. 2006;194:1438–1443. doi: 10.1016/j.ajog.2006.01.057.
  3. Rooney K, Kenton K, Mueller ER, FitzGerald MP, Brubaker L. Advanced anterior vaginal wall prolapse is highly correlated with apical prolapse. Am J Obstet Gynecol. 2006;95:1837–1840. doi: 10.1016/j.ajog.2006.06.065.
  4. DeLancey JO. Structural support of the urethra as it relates to stress urinary incontinence: the hammock hypothesis. Am J Obstet Gynecol. 1994;170:1713–1720.
  5. Tegerstedt G, Miedel A, Maehle-Schmidt M, Nyren O, Hammarstrom M. Obstetric risk factors for symptomatic prolapse: a population-based approach. Am J Obstet Gynecol. 2006;194:75–81. doi: 10.1016/j.ajog.2005.06.086.
  6. Whiteside JL, Weber AM, Meyn LA, Walters MD. Risk factors for prolapse recurrence after vaginal repair. Am J Obstet Gynecol. 2004;191:1533–1538. doi: 10.1016/j.ajog.2004.06.109.
  7. Mallipeddi PK, Steele AC, Kohli N, Karram MM. Anatomic and functional outcome of vaginal paravaginal repair in the correction of anterior vaginal wall prolapse. Int Urogynecol J. 2001;12:83–88. doi: 10.1007/s001920170070.
  8. Jelovsek JE, Maher C, Barber MD. Pelvic organ prolapse. Lancet. 2007;369:1027–1038. doi: 10.1016/S0140-6736(07)60462-0.
  9. Young SB, Daman JJ, Bony LG. Vaginal paravaginal repair: one-year outcomes. Am J Obstet Gynecol. 2001;185:1360–1366. doi: 10.1067/mob.2001.119073. [PubMed] [Cross Ref]
  10. Viana R, Colaco J, Vieira A, Goncalves V, Retto H. Cystocele–vaginal approach to repairing paravaginal fascial defects. Int Urogynecol J. 2006;17:621–623.
  11. Roovers JP, van der Vaart CH, van der Bom JG, et al. A randomised controlled trial comparing abdominal and vaginal prolapse surgery: effects on urogenital function. BJOG 2004; 111:50.
  12. Weber AM, Walters MD, Piedmonte MR, Ballard LA. Anterior colporrhaphy: a randomized trial of three surgical techniques. Am J Obstet Gynecol 2001; 185:1299.
  13. Maher C, Baessler K. Surgical management of anterior vaginal wall prolapse: an evidence based literature review. Int Urogynecol J Pelvic Floor Dysfunct 2006; 17:195.
Citation

Parulekar SV. Abdominal Site Specific Anterior Colporrhaphy. JPGO Vol 5 No. 4. Available from: http://www.jpgo.org/2018/04/abdominal-site-specific-anterior.html

Lipid Infiltration Of Fallopian Tube

Author Information

Abstract

The human fallopian tubal wall has three serosa on the outside, smooth muscular layer in the middle and mucosa on the outside. The serosa is peritoneum. The mucosa is of columnar and ciliated epithelium. There is no fat in the fallopian tube. A case is presented here showing fat under the serosa of both the fallopian tubes in the distal part. This is the first case report of this condition in the world literature.

Introduction

The human fallopian tube has mesothelium on the outside (serosa), smooth muscle in the middle, and columna, ciliated epithelim lining the inside (mucosa). It does not normally contain any fat. Ectopic tissues tissues may be found in the fallopian tube at times, but fat is not one of them. A patient with primary infertility was found to have fat in the distal part of each fallopian tube during laparoscopy.

Case Report

A 23 year old woman presented for evaluation and management for primary infertility. She has been married for 2 years, and the couple had not used any contraception during the 2 years of cohabitation. Her menstrual cycles were every 28-30 days, regular, and associated with moderate flow. Her medical, surgical, sexual, and personal history was not contributory. Her general and systemic examination revealed no abnormality. Bimanual pelvic examination showed an anteverted, normal sized uterus, and no abnormality. Her husband's semen analysis showed normal findings. Her fasting and postpradndial plasma sugar levels, liver function tests, renal function tests, serum TSH were normal. Her ovulation study by ultrasonography showed spontaneous ovulation. A laparoscopy was performed, which showed normal uterus, ovaries, urinary bladder, bowel, and peritoneum. The fallopian tubes were normal in size and shape, but had fat under the serosa in their distal one-third portions. Chromopertubation showed that they were patent.


Figure 1. Left fallopian tube and ovary.


Figure 2. Left fallopian tube and ovary.

Discussion

The human fallopian tubal wall has three serosa on the outside, smooth muscular layer in the middle and mucosa on the outside. The serosa is peritoneum. The mucosa is of columnar and ciliated epithelium. Fallopian tube does not contain any fat.[1] Ectopic tissue like  hilus cells may occur in the fallopian tube and paratubal tissue.[2,3] Adrenal cortical tissue has been found in the broad ligament.[4]  Ectopic pancreatic tissue has also been reported in the fallopian tube.[5] However presence of fat has not been reported in the tube, except as a tumor (lipoma).[6-8] However the distribution of fat was uniform and the surface was smooth in our case, which ruled out a lipoma. Hence a biopsy was not done. This case is reported to bring to notice an anatomical variation, and also to make readers aware that it is not a disease, when such a finding is seen coincidentally.

Acknowledgment

I thank Dr Sarika Solanke for taking the intraoperative pictures.

References
  1. Junqueira’s Basic Histology text and Atlas.  Mescher LA Editor. 13th edition McGraw Hill New York 2013 p. 461.
  2. Palomaki JF, Blair OM. Hilus cell rest of the fallopian tube. A case report. Obstet Gynecol 1971;37:60–62.
  3. Honore LH, O’Hara KE. Ovarian hilus cell heterotopia. Obstet Gynecol 1979;53:461–464.
  4. Falls JL. Accessory adrenal cortex in the broad ligament. Incidence and functional significance. Cancer 1955;8:143–150.
  5. Mason TE, Quagliarello JR. Ectopic pancreas in the fallopian tube. Report of a first case. Obstet Gynecol 1976;48:S70–S73.
  6. Krutsay M. Lipoma of the Fallopian tube. Magy Onkol. 2008 Mar;52(1):71-3. doi: 10.1556/MOnkol.52.2008.1.10.
  7. Daro, A. F. (1935). Lipoma of the fallopian tube. American Journal of Obstetrics and Gynecology, 30(5), 695.
  8. Tóth F, Hegyi J. Lipoma of the fallopian tube associated with dermoid cyst and endometriosis. Orv Hetil. 1971 Apr 18;112(16):927-8.

Citation

Parulekar SV. Lipid Infiltration Of Fallopian Tube. JPGO Vol 5 No. 4. Available from: http://www.jpgo.org/2018/04/lipid-infiltration-of-fallopian-tube.html

Successful Vaginal Delivery After Hip And Knee Arthroplasty

Author Information

Nasare P*, Joshi A**, Gupta AS***
(* Second year Resident, ** Senior Resident, *** Professor, Department of Obstetrics and Gynecology, Seth G S Medical College and K E M Hospital, Mumbai, India.)

Abstract

An increasing number of young patients are undergoing hip arthroplasty for a variety of causes. The mode of delivery and management of such patients remains a challenge and has to be individualized. This case report discusses the mode of delivery in a multigravida patient with history of unilateral total hip arthroplasty and contralateral knee arthroplasty.

Introduction

Total hip and knee arthroplasty is one of the most successful orthopedic procedures performed today with minimal postoperative complications and restores maximum range of motion in a diseased joint. Pregnant women with total hip and/ or knee arthroplasty who do not have any obstetric indication for cesarean section should be given a vaginal trial depending upon the range of motion at the hip as well as the knee joint, feasibility for lithotomy position, condition of the prosthesis and severity of the symptoms. This will decrease the morbidity related to cesarean section.

Case Report

A 34 year old gravida 2, para 1, living 1, was referred for antenatal registration to our hospital at 39 weeks of gestation in view of previous history of hip and knee joint replacement. She was registered in a private hospital at 9 weeks of gestation and had regular antenatal visits during which she received calcium, iron and folic acid supplements. She had undergone right knee arthroplasty 3 years back in view of osteoarthritis. Left hip arthroplasty and left distal femur open reduction and internal fixation were also done 2 and a half years back for osteoarthritis. She did not have any difficulty while walking, but had difficult while climbing stairs and needed help for the same. Her gait did not fit into any specific described abnormal gait in literature. Since there was some difficulty anticipated during vaginal delivery, an orthopedic consultation was sought. As she was able to perform flexion and extension movements at the repaired hip and knee joint, orthopedic consultant opined that vaginal delivery was not contraindicated, but special caution should be exercised to avoid hyperflexion at the hip joint. Hence it was decided to give vaginal trial and consider cesarean section only for an obstetric indication.
She came to the emergency one week later in spontaneous labor. General examination was within normal limits. Cardio-respiratory systems were within normal limits. On abdominal examination uterus was full term with a vertex presentation that was 3/5th palpable. Fetal heart sounds were regular with a rate of 140 beats per minute. She was having two uterine contractions each lasting for twenty seconds in a 10-minute period. On vaginal examination the cervix was 2 centimeter dilated and 40 % effaced, vertex was at station minus 2, and membranes were present. She progressed spontaneously. During second stage of labor she was given a position in which both her legs were flexed to approximately 70 degrees at the hip joint and knees were flexed to 30 degrees with padded stirrups to support.  A small perineal tear was noticed which was sutured with chromic catgut no.’0’in the same position which was used for delivery. She delivered a healthy baby weighing 2.5 kilograms with Apgar of 9/10 A post-delivery radiograph for bilateral hip and knee was done, no dislocation or dislodgement of the prosthesis was identified (figures 1, 2, 3).


Figure 1. Radiograph showing total left hip arthroplasty (acetabular and femoral implant) in situ before delivery.

Figure 2. Radiograph showing total left hip arthroplasty (acetabular and femoral implant) in situ after delivery and no dislocation identified.

 Figure 3. Radiograph showing antero-posterior and lateral view of right knee joint prosthesis in situ and no dislocation identified.

Discussion

The pelvis is an articulation of the two symmetrical hip bones (innominate bones) and sacrum. It consists of three joints the sacroiliac joint, pubic symphysis and hip joint. The hip joint is an articulation of the acetabulum (the part over the pelvic bone) and the head of the femur. The hip joint which has a wide range of movements, is susceptible to restrictions due to a number of causes, a common etiology being osteoarthritis. Women in the reproductive age group can develop end stage arthritis of the hip due to developmental hip dysplasia, osteonecrosis, juvenile inflammatory arthritis and trauma. Some of them may require total hip arthroplasty. It is imperative that any problems affecting the hip joint would affect the pelvis.
Our patient underwent the arthroplasty due to osteoarthritis of the hip and knee. The prevalence of hip and knee osteoarthritis worldwide is approximately 7.4 % and 12.2 % respectively. The incidence is slightly more in women than in men (6.7 %).[1] About 2.2 % of patients who undergo total hip arthroplasty are women of reproductive age group and 14 % of them need hip arthroplasty in view of inflammatory rheumatic disease.[2] Although osteoarthritis is a degenerative disorder, a rising trend in younger age group is noted due to increased incidence of obesity, lower bone mineral density and vascular diseases.[3] Multiparity also has been associated with increased risk of symptomatic osteoarthritis especially of the knee joint.[4]
From a biomechanical point of view, neither hip nor knee joint replacement is a contraindication for pregnancy or for vaginal delivery. Pregnancy and delivery does not have any negative effects on the joints in terms of risk of dislocation, loosening, fracture, or revision surgery.[5] The mode of delivery in patients with history of arthroplasty has to be individualized depending upon the obstetric risk factors. Arthroplasty of the hip and/ or knee joint is not an absolute indication for cesarean section. These patients can be given a vaginal trial as long as the patient can adopt a lithotomy position.
Lithotomy position is the most commonly employed position for childbirth and instrumental delivery. There are three subtypes of lithotomy position; high lithotomy, standard lithotomy and low lithotomy. In a standard lithotomy position the patients’ legs are separated from the midline into 30 to 45 degrees of abduction and the hips are flexed until the thighs are angled between 80 and 100 degrees. Knee flexion required is 70 to 90 degrees. Patient's legs are placed into stirrups, with the knees bent and the lower legs are parallel to the plane of the torso. In high lithotomy position angulations at hip and knee joints are more acute whereas more obtuse in low lithotomy position.[6] In our case the range of motion at knee and hip joints after arthroplasty remained unaffected. Hence during delivery low lithotomy position could be given.
In total hip arthroplasty, the diseased articular surfaces are replaced with synthetic materials (like metal implants), thus relieving pain and improving joint kinematics and function. Metal implants used for arthroplasty corrode and release metal ions such as cobalt and chromium, which may cause systemic toxicity.[7] However there was no study addressing the issue of adequate time interval between joint replacement surgery and pregnancy.
Nepruez et al have opined that a minimum of 1 year duration is required for regaining maximum range of movements post arthroplasty.[8]  Hence pregnancy is better avoided till maximum joint function is regained so as to avoid the additional stress of pregnancy. In our case, the patient conceived after two years of the arthroplasty. She did not have any significant orthopedic problems in the antenatal period.

Conclusion

Normal vaginal delivery can be conducted in patients with hip and/ or knee arthroplasty. These joints replacements do not classify as an indication for cesarean section. Also the functionality of the joint and longevity of the prosthesis is not affected by vaginal delivery.[9]

References
  1. Quintana JM, Arostegui I, Escobar A, Azkarate J, Goenaga JI, Lafuente I. Prevalence of knee and hip osteoarthritis and the appropriateness of joint replacement in an older population. Arch Intern Med . 2008;168(14):1576-84.
  2. Ostensen M. [Hip prostheses in women of fertile age. Consequences for sexuality and reproduction]. Journal of the Norwegian Medical Association: 1993; 113(12):1483–5.
  3. Litwic A, Edwards MH, Dennison EM, Cooper C. Epidemiology and burden of osteoarthritis. Br Med Bull. 2013;105:185–99.
  4. Wise BL, Niu J, Zhang Y, Felson DT, Bradley LA, Segal N, et al. The association of parity with osteoarthritis and knee replacement in the multicenter osteoarthritis study. Osteoarthritis Cartilage. 2013;21(12):1849–54.
  5. Sierra RJ, Trousdale RT, Cabanela ME. Pregnancy and childbirth after total hip arthroplasty. J Bone Joint Surg Br 2005 ;87(1):21–4.
  6. Warner MA, Warner DO, Harper CM, Schroeder DR, Maxson PM, R.N., M.S.Lower Extremity Neuropathies Associated with Lithotomy Positions. Anesthesiology.2000;93:938-942
  7. Maffulli N, Del Buono A, Denaro V. Hip artroplasty: A transient reason not to be pregnant. Surgeon. 2012;10(6): 347–9.
  8. Neuprez A, Neuprez AH, Kaux J-F, Kurth W, Daniel C, Thirion T, et al. Early Clinically Relevant Improvement in Quality of Life and Clinical Outcomes 1 Year Postsurgery in Patients with Knee and Hip Joint Arthroplasties. Cartilage.2017;00(0):1-13.
  9. McDowell CM, Lachiewicz PF. Pregnancy after total hip arthroplasty. J Bone Joint Surg Am. 2001;83–A(10):1490–4.
Citation

Nasare P, Joshi A, Gupta AS. Successful Vaginal Delivery After Hip And Knee Arthroplasty. JPGO 2018. Volume 5 No.4. Available from: http://www.jpgo.org/2018/04/successful-vaginal-delivery-after-hip.html

Pregnancy In An Operated Case Of Pheochromocytoma

Author information

Kumar M*, Mali KA**, Warke HS***
(* Junior Resident, ** Assistant Professor, *** Associate Professor, Department of Gynecology and Obstetrics, Seth GS Medical College and KEM Hospital, Mumbai, India.)

Abstract

Pheochromocytoma is a tumor of the chromaffin cells of the adrenal gland. Due to associated high maternal morbidity, it warrants early detection and proper treatment. The successful pregnancy outcome in a patient who had been operated for pheochromocytoma in the past is described here. 

Introduction

Pheochromocytoma is rare during pregnancy. It occurs with a frequency of 2-7 per one lakh pregnant women. Clinical manifestations include hypertension, sometimes resembling preeclampsia. Biochemical measurements of catecholamines and their metabolites is necessary to establish the diagnosis during pregnancy. 

Case Report

A 32 year old primigravida was referred at 35 weeks of gestation to the emergency department at our tertiary care centre with severe oligohydramnios and a loop of cord around the neck. There were no complaints of pain in abdomen/ leaking or bleeding per vaginum. She was perceiving good fetal movements. 
Eight years before, she had undergone an open abdominal procedure for pheochromocytoma. The histopathology report was suggestive of a 6.5 x 5 x 3 cm pheochromocytoma with central infarction. However, she had not been following up with a physician or endocrinologist for this condition in the recent past. She was admitted for evaluation. 
She had been registered at a private hospital where all antenatal investigations were found to be normal except deranged blood sugars. For an oral glucose tolerance test with values 132 gm %, 165 gm %, 170 gm % and 165 gm %, she had been started on diabetic diet at around 26 weeks of gestation.
Upon admission, her blood pressure was 120/ 80 mm Hg, and there was no albuminuria or any features suggestive of preeclampsia. Uterus was corresponding to weeks of gestation and fetus was live in vertex presentation. 
 Haemoglobin was 10.9 gm %, platelets 1,87,000/mm3 and white blood cell count was 11,000/mm3. All other investigations were normal. Non stress test was reactive. Ultrasonography showed 35 weeks of gestation with amniotic fluid index of 1 cm with normal doppler flow studies. Fetal survey showed mild left pelvicalyceal system fullness.
Endocrinologist opinion was taken in view of deranged sugars in a previously operated case of pheochromocytoma. She was started on tablet amlodipine 5 mg once daily due to the possibility of occurrence of hypertension. Blood glucose level monitoring was advised and due to raised sugar levels, tablet metformin 500 mg per day was started initially which was further increased to 500 mg twice a day. Due to persistent raised blood sugar values, injection insulin (regular) 2-0-0-2 units and insulin (normal) 4-0-0-4 units was started which was further increased to regular 4-0-0-4 units and 8-0-0-8 units.
Ultrasound abdomen and pelvis showed no remnants of pheochromocytoma. Plasma free normetanephrine levels were done which was within normal limits and any possibility of remnants of pheochromocytoma was ruled out.  NST done biweekly was reactive and weekly doppler revealed increasing amniotic fluid with normal doppler flows.  
Endocrinologists advised cesarean section to be the mode of delivery and to avoid a vaginal trial of labor so as to prevent hypertensive crisis, as a possibility of a small residual tumor had not been ruled out since imaging could not be done due to gravid state. She underwent elective lower segment cesarean section at 38 weeks of gestation under epidural anaesthesia. She delivered a male baby of 2.93 kg with Apgar score of 9/ 10. The procedure was uneventful. Intraoperatively, blood pressure remained normal.  In the postoperative period her blood sugar monitoring was done and gradually insulin and metformin were stopped. She had an uneventful postoperative course and was discharged on day 10. Amlodipine was continued and she was asked to follow up with endocrinologist. 
Discussion

Pheochromocytoma is a rare tumor of the chromaffin cells that cause increased levels of catecholamines and is known to increase morbidity and mortality. Maternal and fetal survival depends a lot on an early diagnosis, appropriate medical therapy and correct timing of surgery and delivery. Despite being an uncommon condition recognising it timely in a pregnant hypertensive woman is extremely important. For the biochemical diagnosis, plasma or urine metanephrines are test of first choice.[1] For reliable diagnosis magnetic resonance imaging is the investigation of choice with sensitivity of 90 %.[2] 
If a tumor is diagnosed in the first 24 weeks of gestation it should be treated by laparoscopic adrenalectomy after 10-14 days of medical therapy similar to that in the nonpregnant state. Preoperatively alpha and beta blockers need to be administered to control blood pressure and to prevent intraoperative crises. 
If the tumor is diagnosed in the third trimester the patient should be managed until the fetus is viable using the same drug regimen. Cesarean section is preferred over vaginal birth which is possibly associated with higher mortality. Our patient underwent a cesarean section, as advised by the endocrinologists, though she was an operated case of pheochromocytoma in the past. 
Some patients with pheochromocytoma have unknown genetic predisposition and are asymptomatic. Approximately 20% cases remain undiagnosed until labor.[3] The high levels of maternal mortality are related to complications of a hypertensive crises like stroke, cardiac arrest, pulmonary oedema. Fetal morbidity may be due to placental abruption, fetal hypoxia and intrauterine growth restriction.
The classical triad of symptoms for pheochromocytoma consist of headache, palpitation and sweating.[4] Other symptoms such as flushing and tremors may be present. Sudden occurrence of symptoms, significant variations in blood pressure, and orthostatic hypotension are features. Our patient was asymptomatic during her pregnancy, and her blood pressure remained normal. 
Plasma and urinary catecholamines are not increased in normal pregnancies and can be used as a screening test.[5]  In our case it was used as a test to look for recurrence, which was normal. Even in pre-eclampsia, plasma catecholamines are only slightly elevated. Maternal catecholamines do not cross the placental barrier.[6]
Being a rare condition, there are no prospective studies regarding mode of delivery in pheochromocytoma.  Vaginal deliveries appear to have a higher maternal mortality (31%) than cesarean section (19%) mainly due to hypertensive crisis. Therefore, cesarean section has been recommended as the preferred mode of delivery.[7] Similar advise was given by our endocrinologists and our patient underwent cesarean section, under epidural anesthesia.
She was advised to follow up with endocrinologists regularly. Patients with pheochromocytoma are at risk of developing tumor recurrences or new tumors, even after successful resection of primary tumor.[8] Regular monitoring of plasma metanephrines is the main method of follow-up, which our patient was not undergoing regularly, but underwent upon admission during this pregnancy. Since the disease is mostly sporadic, no specific follow up of the neonate is required.

Conclusion

To conclude, pregnancy with a pheochromocytoma is a rare but dangerous combination. The prognosis of pregnant patients with a pheochromocytoma has improved and appropriate management at a tertiary care centre with a multidisciplinary approach involving expert obstetricians, endocrinologists and anesthetists is preferred. 

References
  1. Oishi S, Sato T. Pheochromocytoma in pregnancy: A review of Japanese literature. Endocrine Journal 1994; 41(3): 219-25
  2. Schenker JG, Granat M. Pheochromocytoma and pregnancy- an updated appraisal. Australian N Z J Obstet Gynaec. 1982; 22(1):1-10
  3. Dean RE. Pheochromocytoma and Pregnancy. Obstetrics and Gynaecology 1958; 11: 35-42
  4. Zuspan FP. Urinary excretion of epinephrine and norepinephrine during pregnancy. Journal of clinical endocrinology and Metabolism; 1970; 30(3): 357-360
  5. Ahlawat SK, Jain S, Kumari S, Varma S, Sharma BK. Pheochromocytoma associated with pregnancy: case report and review of literature. Obstetrical and Gynaecological Survey 1999; 54(11): 728-737.
  6. Oliva R, Angelos P, Kaplan E, Bakris. Pheochromocytoma in pregnancy: a case series and review. Hypertension 2010; 55(3):600-6
  7. Sarathi V, Lila AR, Bandgar TR, Menon PS, Shah NS. Pheochromocytoma and pregnancy: a rare but dangerous combination. Endocrine practice. 2010; 16(2): 300-9
  8. Amar L, Fassnacht M, Gimenez-Roqueplo AP, Januszewicz A, Prejbisz A, Timmers H. Long-term postoperative follow-up in patients with apparently benign pheochromocytoma and paraganglioma. Horm Metab Res.2012 May;4(5):385-9.

Citation

Kumar M, Mali KA, Warke HS. Pregnancy In An Operated Case Of Pheochromocytoma. JPGO 2018. Volume 5 No.4. Available from:http://www.jpgo.org/2018/04/pregnancy-in-operated-case-of.html

Pseudocyesis: A Tale Of Phantom Twin Pregnancy

Author Information

Ostwal P*, Balsarkar GD**, Mahale N***
(* Senior Resident, ** Professor, *** Junior Resident, Nowrosjee Wadia Maternity Hospital, Mumbai, India.)

Abstract

Pseudocyesis is a psychosomatic condition in which the patient displays signs and symptoms of being pregnant in absence of actual pregnancy. It may be secondary to severe psychological distress to the woman owing to socio-cultural factors. In this paper, the authors describe one such case.

Introduction

In our society, childbirth is considered as a requirement for validation of marriage and generational continuity. Infertility puts undue psychological pressure on the woman involved. This may lead to manifestation of pseudocyesis or phantom pregnancy. These patients exhibit all the signs and symptoms of pregnancy except for the actual presence of pregnancy. Such patients are psychologically volatile and the woman as well as the family needs proper counseling. This situation requires a different approach by the clinician and awareness of the phenomenon goes a long way in preventing consequent complications. It is therefore incumbent on the treating obstetrician to keep pseudocyesis as one of the possibilities when such a patient presents. This phenomenon has incited comment time and again, and a few reports can be found in psychiatry literature. The authors wish to bring it to the notice of fellow obstetricians to be aware of its possibility in similar cases.

Case Report

A 33 year old illiterate woman presented to us with complaints of severe lower abdominal pain since 6 hours, (which she insisted was labor pain) and bleeding per vaginum. She gave history of amenorrhea since 8 months. She had once visited a local doctor to get urine pregnancy test and per-vaginum examination done.  She was not given any assurances about pregnancy but was strongly convinced that she was pregnant with twins. She recounted experiences of repeated tiredness, nausea, vomiting, progressive abdominal enlargement, breast enlargement and fetal movements. 
On presentation, her general examination revealed normal vital parameters, and abdominal and vaginal examination also confirmed a non-pregnant status. When interviewed, she revealed her stressful relationship with husband and in-laws. She had been unsuccessfully undergoing treatment for infertility for ten years and due to the social pressures and financial burden, there was disharmony in marital life leading to domestic violence. A missed period had convinced her and the family members of her being pregnant.
 After being confronted with the truth, she became hysterical and was in state of denial, and the husband became abusive. After counseling, both calmed down and the woman was discharged with an advice to follow up for further counseling with a psychologist.

Discussion

Derived from the Greek roots, ‘pseudo’ (meaning false) and ‘kyeis’ (meaning pregnancy), pseudocyesis has also been called as phantom pregnancy, spurious pregnancy, hysterical pregnancy, meteorism, phantom tumor and tympanites, amongst others.[1] Earliest descriptions are found in a 300 BC text by Hippocrates. Since then there have been multiple records surfacing about similar incidences, mostly in western world. It describes a clinical syndrome in which a non-pregnant woman manifests a strong conviction of being pregnant associated with signs and symptoms mimicking the experience of being pregnant.[2,3]
Diagnostic and Statistical Manual-5 (DSM-5) has classified pseudocyesis under “Other Specified Somatic Symptom and Related disorders”, as opposed to a delusional disorder (wherein the manifestations are only psychological symptoms, not physical) or malingering (wherein the patient feigns symptoms consciously to gain advantage).[4]
Most of these cases generally belong to the age group of 20-44 years and 80 % of them are married,[5] which was the case in our patient too. The time interval upto which the symptoms may last vary from few weeks to 8- 9 months or longer.[6] In modern era with radiological facilities, demonstration of absentia of fetus and placenta leads to conclusive diagnosis.[7] The strength of conviction of our patient in her pregnant status is evident by the fact that she came with 8 months amenorrhea.
The causative factors involved in pseudocyesis are still unclear. The psychological factors that can have major impact etiologically are very strong fear factor, or strong desire or wish to be pregnant, which stems from socio-cultural pressures leading to physical manifestations as a stress response. This goes on to explain the occurrence of the condition in settings of second or third marriages or infertility, as was the case with our patient.[8] Our patient came from a low socioeconomic strata, a finding which was echoed in various studies.[9] In majority of previously reported cases, pseudocyesis occurred in the backdrop of some pre-existing psychiatric illness though this was not the case with our patient.
In clinical obstetric practice, it has become extremely important to document properly the clinical findings and confirm them with investigations. In an increasingly litigious society like ours, the prevalent socio-cultural milieu and conditions like pseudocyesis spawn fertile grounds for mistrust and litigation.

Conclusions
  1. It is important for obstetricians to be aware of the phenomenon of pseudocyesis and keep it as a differential diagnosis in those with no physical or investigational evidence of pregnancy. These patients should be counselled adequately and require proper psychological profiling to reach the root cause of the disorder and remedial measures to prevent further problems.
References
  1. Kulcsar DD. Pseudocyesis. Can Med Assoc J. 1951; 64(4): 305–308
  2. Brockington I. Obstetric and gynaecological conditions associated with psychiatric disorder. In Gelder MG, Lopez-Ibor JJ, Andreasen N, editors. New Oxford Textbook of Psychiatry. 1st ed. Oxford: Oxford University Press 2000; pp. 1195–209
  3. Apefel RJ, Mazor MD. Psychiatry and Reproductive Medicine. In Kaplan HI, Sadock BJ, editors. Comprehensive Textbook of Psychiatry. 5th ed. Baltimore: Williams & Wilkins 1989; pp. 1332-35.
  4. American Psychiatric Association. Diagnostic and Statistical Manual of Mental Disorders. 5th ed. Arlington: APA publishing; 2013. 
  5. Paulman PM, Sadat A. Pseudocyesis. J Fam Pract. 1990; 30(5):575–6
  6. Brown E, Barglow P. Pseudocyesis - a paradigm for psychophysiological interactions. Arch Gen Psychiatry. 1971; 24(3):221–9
  7. Small GW. Pseudocyesis: An overview. Can J Psychiatry. 1986; 31(5):452–7
  8. Steinberg A, Pastor N, Winheld EB, Segal HI, Shechter FR, Colton NH. Psychoendocrine relationships in pseudocyesis. Psychosomat Med. 1946; 8:176–9
  9. Dafallah SE. Pseudocyesis and infertility. Saudi Med J. 2004; 25(7):964–5.

Citation

Ostwal P, Balsarkar GD, Mahale N. Pseudocyesis: A Tale Of Phantom Twin Pregnancy. JPGO 2018. Volume 5 No.4. Available from: http://www.jpgo.org/2018/04/pseudocyesis-tale-of-phantom-twin.html

A Rare Case Of Sealed Off Ruptured First Trimester Interstitial Ectopic Pregnancy

Author Information

Bhate A*, Bandukwalla V**, Chitnis S***
(* Director, Shubhdeep Nursing Home, **Consultant, Saket Maternity & Nursing Home *** Consultant, Department of Obstetrics & Gynaecology, AllCure Superspeciality, Mumbai, India.)

Abstract

Pregnancy in the interstitial part of a fallopian tube is a rare and life threatening obstetric emergency. It is associated with uterine rupture and significantly high maternal morbidity. We present a case of ruptured first trimester interstitial ectopic pregnancy which was sealed off by bowel, thus preventing hemorrhage. The case was managed laparoscopically, excising a thin portion of myometrium along with the ectopic mass, followed by cornual repair.

Introduction

Interstitial pregnancy is defined as implantation of embryo in interstitium, the proximal most part of fallopian tube. It is a rare entity with poor prognosis if not diagnosed and treated in a timely manner. Ectopic pregnancy has an incidence of 11 per 1000 pregnancies. Interstitial pregnancy accounts for 2-4 % of all cases but 20 % of ectopic related mortality.[1] This is explained by high rates of rupture and hemorrhage. Risk factors are similar to tubal ectopic pregnancy, including PID, use of intrauterine device, Mullerian anomalies, use of ART and previous pelvic surgery.[2] Ipsilateral salpingectomy is a unique risk factor exclusive to interstitial ectopic.[3] Interstitial pregnancy is usually diagnosed between 6 to 10 weeks. A combination of history, examination and investigations aids an early detection. Transvaginal ultrasound, serial β human chorionic gonadotropin and progesterone measurements help in accurate diagnosis.[4] Several modalities are available for the management of interstitial ectopic (observation, medical management, hysteroscopy guided suction, cornual wedge resection and hysterectomy). In addition, laparoscopy is a safe and effective method. It is not contraindicated even in cases with rupture.[5]

Case Report

A 30 year old woman presented with 2 months amenorrhea, positive pregnancy test and pain in abdomen. She had no bleeding or syncopal attacks. She had undergone cesarean delivery 2 years back and the current pregnancy was a spontaneous conception. She had no history of pelvic inflammatory disease, intrauterine contraceptive device use or pelvic surgery. On examination, pulse and blood pressure were normal. There was no abdominal distension, but there was tenderness in right iliac fossa. Vaginal examination revealed a bulky uterus with ill-defined fullness in left fornix. Ultrasound showed empty uterine cavity and a heterogenous mass adjoining the uterus, partially surrounded by myometrium. Both ovaries were seen separately and there was no hemoperitoneum. Hemoglobin was 9.8 gm % and other investigations were within normal limits. She was taken for laparoscopic resection of ectopic mass with salpingectomy for a provisional diagnosis of tubal ectopic pregnancy.
Peritoneal access was gained by supraumbilical port and 2 lateral ports. Uterus was bulky with normal fallopian tubes and ovary on left side. Omentum and large bowel were adherent to right cornua, fallopian tube and ovary were not visualized. Dissection revealed a ruptured interstitial ectopic sealed off by omentum and bowel. Conceptus was removed and sent for histopathological examination, which confirmed the diagnosis (Figure 1). Surrounding myometrium was infiltrated with diluted vasopressin, and a small portion was excised. Right salpingectomy was done. The myometrium was repaired with interrupted sutures with vicryl 1. (figure 2) She had an uneventful post-operative recovery.


Figure 1. Intra-operative view showing site of rupture and extruded conceptus after separation of adhesions.


Figure 2. Right salpingectomy and myometrial repair was done.

Discussion

Non-tubal ectopic pregnancies account for only 10 % of all ectopic pregnancies but contribute majorly to morbidity and mortality. Interstitial pregnancy itself is very rare, roughly 2 % of all ectopic pregnancies. However, it has a higher contribution to mortality due to increased risk of rupture in highly vascularized myometrium.[6] Our patient likely had a small sized rupture with slow hemorrhage, which resulted in its sealing off by omentum before significant bleeding could occur.
Diagnosis is usually made by a transvaginal ultrasound, which is an important investigation in such cases. It is based on ultrasound criteria set by Bourdel.[7] 
a. Gestational sac (GS) placed eccentrically with respect to the uterine sagittal axis.
b. Absence of double decidual sac sign around the GS.
c. Myometrial layer less than 5 mm around the GS.
d. Interstitial line sign (presence of an echogenic line that joins the endometrial cavity to GS in the cornual region)
In cases where TVS is inconclusive, MRI may be done for confirmation. In our patient, diagnosis of tubal ectopic was made on ultrasound and MRI was not done.
Several modalities of treatment have been studied for interstitial pregnancy. The management should be tailored according to individual patients. Our patient was symptomatic, hence a surgical procedure was done for her. 
The management of an interstitial ectopic pregnancy parallels that of other tubal ectopic pregnancies, in general.[8] A study by Hunt et al from showed 87% success rate with single or multi-dose methotrexate in non-tubal ectopic.[9] Case reports by Macaes and Sanchez Contreras also demonstrate safety of local Methotrexate injection.[10,11] Laparoscopy may be undertaken as primary method of treatment in pregnancy of unknown location, when patient desires sterilization surgery or when patient has acute abdomen. It is also used as secondary treatment in failure of medical therapy.[9] Myometrium may be repaired by double purse string,[12] figure of eight or interrupted sutures. Laparotomy and cornual wedge resection was earlier used as first choice surgery, however, it is rarely required now due to superior visualization by endoscopy. In unruptured interstitial pregnancy, hysteroscopic visualization of gestational sac followed by suction and evacuation under ultrasound or laparoscopic guidance can be considered. There is risk of uterine rupture and hemorrhage in this procedure and need for conversion to laparoscopy or laparotomy must be discussed with the patient.

Conclusion

Interstitial pregnancy is a rare obstetric emergency. Transvaginal ultrasound is a crucial tool in early diagnosis. Management should be tailored as per individual case. In unruptured interstitial pregnancy in a stable patient, laparoscopy can be a safe and effective technique. 

References 
  1. Kahramanoglu I, Mammadov Z, Turan H, Urer A, Tuten A. Management options for interstitial ectopic pregnancies: A case series. Pak J Med Sci. 2017 33(2): 476–482.
  2. Brennan DF. Ectopic Pregnancy-Part I: Clinical and Laboratory Diagnosis. Acad Emerg Med 1995; 2(12):1081–9. 
  3. Simpson J, Alford C, Miller A. Interstitial pregnancy following homolateral salpingectomy. Am J Obstet Gynaecol. 1961;(82):1173–9. 
  4. Murray H, Baakdah H, Bardell T, Tulandi T. Diagnosis and treatment of ectopic pregnancy. Canad Med Assoc J. 2005; 173(8):905-12.
  5. Grimbizis GF, Tsalikis T, Mikos T, Zepiridis L, Athanasiadis A, Tarlatzis BC, et al. Case report: laparoscopic treatment of a ruptured interstitial pregnancy. Reprod Biomed Online. 2004; 9(4):447–51. 
  6. Surbone A, Cottier O, Vial Y, Francini K, Hohlfeld P, Achtari C. Interstitial pregnancies’ diagnosis and management: an eleven cases series. Swiss Med Wkly. 2013; 143: w13736. 
  7. Bourdel N, Roman H, Gallot D, Lenglet Y, Dieu V, Juillard D, et al. Interstitial Pregnancy. Ultrasonographic diagnosis and contribution of MRI. A case report. Gynecol Obstet Fertil. 2007; 35(2):121–4. 
  8. Verma U, English D, Brookfield K. Conservative management of nontubal ectopic pregnancies. Fertil Steril. 2011; 96(6):1391-5
  9. Hunt SP, Talmor A, Vollenhoven B. Management of non-tubal ectopic pregnancies at a large tertiary hospital. Reprod Biomed Online. 2016; 33(1):79–84. 
  10. Macaes A, Fernandes S, Rodrigues C, Branco M. Interstitial ectopic pregnancy managed with local methotrexate. BMJ Case Rep. 2015; pii: bcr2015212563. PMID: 26452744 
  11. Sanchez CJ, Hernandez VLE, Villalobos CMA, Risco CRJ. Conservative treatment of interstitial ectopic pregnancy and endometriosis with laparoscopic methotrexate and potassium chloride and complementary management with nasal nafarelin. Report of a case and review of the literature. Ginecol Obstet Mex. 2000; 68:35–8. 
  12. Guan J, Yu X. Laparoscopic Double Purse-String Suture Technique and Its Fertility Outcome in Interstitial Pregnancy: A Review of 15 Cases. J Minim Invasive Gynecol. 2015; 22(6S): S144. 

Citation

Bhate A, Bandukwalla V, Chitnis S. A Rare Case Of Sealed Off Ruptured First Trimester Interstitial Ectopic Pregnancy. JPGO 2018. Volume 5 No.4. Available from: http://www.jpgo.org/2018/04/a-rare-case-of-sealed-off-ruptured.html

Secondary Sjogren’s Syndrome In Pregnancy

Author Information 

Yadav K*, Samant PY**, Thakur HS***
(* Junior Resident, ** Additional Professor, *** Assistant Professor, Department of Gynecology and Obstetrics, Seth G S Medical College and K E M Hospital, Mumbai, India.)

Abstract

We present a case of a 30 year old woman in her second pregnancy with secondary Sjogren’s syndrome associated with systemic lupus erythematosus (SLE), rheumatoid arthritis and right internal jugular vein thrombosis. This overlap is a rare phenomenon seen in pregnancy as the syndrome is mostly seen in the late reproductive age.

Introduction

Sjogren’s syndrome is a chronic inflammatory disorder. It is characterised by lymphocytic infiltration of exocrine glands. Patients with the syndrome present with xerostomia, xerophthalmia, parotid gland enlargement affecting quality of life. Prevalence rate of the disease is between 0.1 % to 4.8 %.[1] Primary Sjogren’s syndrome occurs in absence of another underlying rheumatic disorder. Secondary sjogren syndrome is associated with underlying rheumatic disorder such as SLE, rheumatoid arthritis or scleroderma. Sjogren’s syndrome predominantly occurs in women.  
Sjogren’s syndrome may have associated antiphospholipid syndrome (APS) which is a hypercoagulable state that may cause arteriovenous thrombosis and/ or thrombocytopenia resulting in a series of thrombotic clinical symptoms. In pregnancy APS can cause miscarriage, premature birth, and still birth. A multidisciplinary approach can achieve successful maternal and fetal outcome. 

Case Report

A 30 year old second gravida, was transferred to our institute at 22 weeks of gestation with severe multiple small joint pains and swelling of right lower limb for 2 months. She was diagnosed with rheumatoid arthritis two years prior.  Around one year prior to presentation, she had right internal jugular vein thrombosis extending to superior vena cava and was on treatment for same with warfarin, prednisolone and immunosuppressive agents; hydroxy chloroquine and azathioprine. In the first pregnancy, she had a termination of pregnancy at 16 weeks for single umbilical artery in the fetus and a positive screen for aneuploidy.  
In a private hospital, during the first trimester of present pregnancy, warfarin was changed to low molecular weight heparin 0.4 mg subcutaneously. At her presentation here, she was admitted by rheumatologists. Investigations revealed positive anticardiolipin antibody immunoglobulin G (aCLA Ig G), anti- Sjogren’s syndrome-related antigen A (SSA) positivity. Diagnosis of secondary Sjogren’s syndrome was made according to American College of Rheumatology criteria as she was SSA positive and ANA positive with a titre of  1:320.[2] There was no xerostomia, xerophthalmia or parotid swelling. Prednisolone was stopped and tab dexamethasone 4 mg was started to prevent fetal adrenal insufficiency[3] and 75 mg aspirin. LMWH was continued. Right lower limb venous Doppler was indicative of varicose veins and conservative treatment with elastic stockings and limb elevation were advised. Her obstetric evaluation was normal. Obstetric sonography was normal and corresponded to 23 weeks. Fetal echocardiogram was normal and congenital heart block which can occur due to positive SSA antibody. After the pain and swelling subsided, she was discharged and advised antenatal check-up. 
She was monitored for preeclampsia, anemia or antepartum haemorrhage and intrauterine growth restriction (IUGR). She was admitted at 36 weeks gestation for fetoplacental insufficiency and asymmetrical IUGR. Non-stress test (NST) was reactive. Her haemoglobin was 12.9 gm %, WBC-16,900/ cu mm, platelet count 2.2 lakh/ cu mm, INR-1.02, aPTT-27.3 (control 29) , fasting blood sugar 101mg/ dl, postprandial blood sugar 114 mg/ dl and TSH was 3.93 IU/ ml. She went into spontaneous labor and heparin was stopped. She delivered uneventfully vaginally with baby weight of 2.2 kg with Apgar score of  9/ 10. Tablet warfarin was restarted postpartum and she was discharged on day 9 once INR reached desired level.  Contraception counselling was done and she chose barrier method of contraception. She was asked to follow up in cardiology and rheumatology clinics.

Discussion 

Sjogren’s syndrome overlapping SLE and rheumatoid arthritis in pregnancy is associated with maternal and neonatal complications. There is high incidence of poor fetal outcome hence prenatal counselling and antenatal care play a major role. Such pregnancies require multidisciplinary approach with rheumatologist, obstetrician and paediatrician. 
Several studies have demonstrated increased rate of spontaneous abortion, intrauterine growth restriction, preterm labour and fetal loss. Most dreaded complication of pregnancy in patient with Sjogren’s syndrome is increased risk of delivering baby with complete heart block (CHB) especially if she is anti SSA positive.[1] Hence frequent surveillance by serial echocardiograms has been advised. In our case, fetal 2D-Echo at 24 weeks was normal. The reported prevalence of CHB with anti SSA positive woman is 1-2 % with recurrence rate approximately 10 times higher.[1] 
Maternal treatment with dexamethasone or betamethasone reduces the antibody mediated inflammatory damage hence improves fetal outcomes.[3] But this treatment may increase the risk of infection, osteoporosis and glucose intolerance while in fetus it may cause IUGR, oligohydramnios and possibly adrenal suppression. Alternative therapies include plasmapheresis, intravenous immunoglobulins and beta sympathomimetics. Our patient was given dexamethasone from 22 weeks of gestation and delivered at near term with IUGR. 
Young women with Sjogren’s syndrome often have systemic and severe disease compared with patient with primary Sjogren’s syndrome which occur in later stage of life.[4] Our patient was 30 years old with Sjogren’s syndrome with SLE and rheumatoid arthritis. Relative risk of fetal loss is 2.7 in primary Sjogren’s syndrome and 2.2 in SLE which may be attributed to secondary Sjogren’s syndrome. Brucato et al found that anti-Ro positive SLE women had higher rate of spontaneous abortion (ie) 18% as compared to 5.6% with anti Ro negative SLE women. There is significantly higher rate of recurrent pregnancy loss in patients with anti-Ro non SLE positive women which was 23.7%.[5] Long acting progesterone intrauterine device (IUD) or subdermal implants are preferred and depot medroxyprogesterone acetate (DMPA) should be avoided in patient on steroids or patients at risk of osteoporosis.[6]
Various studies have proven that general outcome of the pregnancies in these pregnant women with autoimmune   disorders can be excellent with multidisciplinary team approach is adopted. 

Conclusion

Women with Sjogren’s syndrome are likely to have more complications during pregnancy with increased risk of poor fetal outcome. Women with these disorders should undergo preconceptional counselling and these high risk pregnancies should be optimally managed by multidisciplinary approach involving obstetrician, rheumatologist and neonatologist.  
  
References 
  1. Gupta S, Gupta N. Sjogren syndrome and pregnancy. A literature review. Perm J 2017; 21: 16047. doi: 10.7812/TPP/16-047. 
  2. Bowman SJ, Fox RI. Classification criteria for Sjogren's syndrome: nothing ever stands still! Ann Rheum Dis. 2014; 73(1): 1–2. 
  3. Tegethoff M, Pryce C, Meinlschmidt G. Effects of intrauterine exposure to synthetic glucocorticoids on fetal,newborn, and infant hypothalamic-pituitary- adrenal axis function in humans: A systematic review. Endocr Rev.2009; 30:753-89. 
  4. Hussein SZ, Jacobsson LHT, Lindquist PG, Theander E. Pregnancy and fetal outcome in women with primary Sjogren’s syndrome compared with women in the general population: a nested case control study. Rheumatology 2011; 50:1612-7 
  5. Brucato A, Cimaz R, Caproli R, Ramoni V, Buyon J. Pregnancy outcomes in patients with autoimmune diseases and anti RO/SSA antibodies. Clin Rev Allergy Immunol. 2011; 40(1):27-41.
  6. Sammaritano LR. Contraception in patients with systemic lupus erythematosus and antiphospholipid syndrome. Lupus. 2014; 23(12):1242-5.

Citation

Yadav K, Samant PY, Thakur HS. Secondary Sjogren’s Syndrome in Pregnancy. JPGO 2018. Volume 5 No.4. Available from: http://www.jpgo.org/2018/04/secondary-sjogrens-syndrome-in-pregnancy.html