Volume 4 Issue 10, October 2017

Editorial
Parulekar SV

Uterosacral Shelf For Cystocele Repair
Parulekar SV

Laparoscopic Management Of Para-Ovarian Cyst Torsion With 20-Week Pregnancy
Bhate A, Joshi D, Chitnis S.

Deranged Liver Enzymes In A Case Of Diabetes In Pregnancy:  Diagnostic Dilemma
Saxena A, Thunga C, Madhva Prasad S, Gupta AS.

Lateral Approach To Hemihematometra
Shetty A, Madhva Prasad S, Gupta AS.

An Unusual Case Of Placenta Increta
Mehta N, Honavar P, Parulekar SV.

Laparoscopic Management Of Non Communicating Rudimentary Horn Pregnancy
Shah NH, Deshmukh M, Somani A, Paranjpe SH.

Successful Management Of H1N1 Infection Complicating A Case Of Twin Pregnancy With Eclampsia
Kambhampati L, More V.

Pregnancy In A Post Renal Transplant Patient
Shah S, Warke H, Mayadeo N.

Rare Case Of Stickler's Syndrome
Dhanawat J, Uppal M, Samant PY.

Editorial

Parulekar SV

Genital prolapse was known to Hippocrates and Galen, in whose writings it finds a place. The exact pathophysiology of the condition was not known then. But it was known that it caused a significant deterioration of the quality of life. Hence attempts were made to repair it. The earliest attempts were simple. Pessaries were used as trusses to control the symptoms of prolapse in the middle of the nineteenth century. Earliest surgical attempts included suturing the opposite side labia together in the midline, and removing a part of the vagina to narrow its width. First operation on the anterior vaginal wall was done by Heming in 1831. Modern techniques of colporrhaphy were being used and reported in the early part of the 20th century. Howard Kelly from Baltimore promoted the plication of pubovesicocervical fascia in the midline for repair of a cystocele. It was assumed that endopelvic fascial support structures got damaged during childbirth causing vaginal wall prolapse. Kelly’s plication of the fascia was based on that concept. This concept remained strong almost throughout the 20th century. A major breakthrough came with identification and classification of fascial defects as proximal, distal, central, and lateral by Richardson et al from Georgia. It was found that the endopelvic fascia invested and supported all the pelvic organs. It connected the pelvic organs to the retropubic fascia, fascia on the lateral pelvic walls (obturator fascia, in which a condensation was present to form the arcus tendineus fasciae pelvis), and pelvic floor (formed by the levatores ani). It was condensed to form uterosacral and cardinal ligaments which were connected to the pericervical ring. DeLancey further showed that the proximal one third of the vagina was suspended by the uterosacral ligaments, the middle one third was supported by lateral attachments to the pelvic side wall at the arcus tendineus fasciae pelvis and the distal one third of the anterior vagina was fused with the urogenital diaphragm. Damage to the fascial attachments to the middle one third of vagina resulted in paravaginal defects. Damage to the fascial attachments between the vagina and the uterosacral-cardinal ligament complexes resulted in central transverse defects. These concepts have become very important in site-specific repairs. It is now found that the plication procedure for repair of a cystocele bunches the endopelvic connective tissue in the midline, which places more stress on paravaginal defects by pulling the detached edge of the fascia away from the pelvic side wall and aggravates the prolapse. As a result, the failure rates are unacceptably high, and there are functional disturbances of micturition too. In more recent times, owing to perhaps a desire to keep doing something new or perhaps due to a marketing strategy of manufacturers of synthetic meshes, there has been growing interest in using meshes under the vagina to provide support to it. Subsequently there have been reports of mesh erosion and other problems and a trend towards not using meshes. Perhaps litigations by patients discouraged doctors. Manufacturers inserted a statement warning about the complications of meshes and asking the doctors to use the product at their own discretion. Perhaps it removed the responsibility from them to the doctors, which discouraged the use of meshes even further. In the meantime, we have been using site specific repair with excellent results and did not have to resort to the use of meshes. In this issue of the journal, we have a report of an innovative operation in which the uterosacral ligament pedicles were placed under the bladder base to support it. If one thinks about it, one realizes it is one form of a site specific repair, since the end result is bladder fascia being attached to the uterosacral ligaments. I hope the readers benefit from this article and enjoy this issue like the previous ones.

Uterosacral Shelf For Cystocele Repair

Innovation

Author Information

Parulekar SV

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

Abstract

Approximately 11% of women will undergo pelvic reconstructive surgery for pelvic organ prolapse and/or urinary incontinence during their lifetime. Cystocele in women is one of the common problems associated with uterovaginal prolapse. It occurs when the fascia between urinary bladder and vagina is weakened allowing the bladder to herniate into the vagina. While usually not lifethreatening, cystocele is often associated with deterioration in quality of life and may contribute to bladder and sexual dysfunction. It was conventionally repaired by plication of the pubovesicocervical fascia from the right and left sides in the midline. The next step in its evolution was site specific repair, in which the type of defect in the fascia was repair instead of plicating the fascia indiscriminately. The third step was use of biological and synthetic meshes. However the meshes have their own problems and complications. A novel approach of using the uterosacral ligaments to perform a cystocele repair after a vaginal hysterectomy is described here.

Introduction

Genital prolapse in women was known to Hippocrates and Galen. Heming first operated on the anterior vaginal wall in 1831. Howard Kelly of Baltimore championed the concept of plication of pubovesicocervical fascia in the midline for repair of a cystocele.[1] In 1909, George R. White of Georgia published an account of cystocele repair using a transvaginal paravaginal approach.[1,2] A. Cullen Richardson and associates of Georgia developed the concept of classifying fascial defects as proximal, distal, central, and lateral.[2,3] This encouraged gynecologists to identify and repair each vaginal defect and to return support attachments to their original anatomic location. Emphasis was focused on he hernial nature of prolapse and led to the abandonment of absorbable suture in favor of permanent suture in repairs.[4-8] The results of site specific repair of a cystocele are far superior to those of plication of the fascia.[9-12] Postoperative complications and urinary disturbances are also much less. Subsequently the trend has been moving towards use of biological or synthetic meshes for the repair. Meshes behave differently in the subvaginal space than in the abdominal wall. Infection, erosion and extrusion rates are higher and so is the risk of litigation. We prefer a site specific repair to the use of a mesh. In case the fascia is too weak to be repaired adequately to provide a satisfactory repair, the uterosacral ligaments may be used to provide the required support to the urinary bladder. A novel approach of using the uterosacral ligaments to perform a cystocele repair after a vaginal hysterectomy is described here.

Operative Technique

1. While performing the vaginal hysterectomy, the uterosacral ligaments are clamped, cut and ligated quite close to the uterus, so that maximum possible lengths of the ligaments is made available for use during anterior colporrhaphy. One end of a ligature on each ligament is kept long and held with a hemostat.
2. Epinephrine in saline (1:300000) is infiltrated under the vaginal mucosa overlying the cystocele.
3. A midline incision is made in the anterior vagina overlying the cystocele.
4. A flap of the anterior vagina are raised on each side by sharp dissection, separating the vagina from the fascia overlying the urinary bladder.
5. The dissection is carried out laterally up to the white lines on the two sides.
6. Hemostasis is achieved. This is important, as a failure to do so may result in formation of a hematoma between the urinary bladder and the uterosacral ligaments fixed below it. Infection in the hematoma would result in formation of an abscess and its complications.
7. The uterosacral ligaments are fixed under the urinary bladder.
8. Two sutures of No. 1 polyglactin are passed through the terminal cut part of each ligament. They are then passed through the vagina lateral to the urethra. When the sutures are tied on each side, the ligaments get fixed under the urinary bladder.
9. The gap between the two uterosacral ligaments is closed with interrupted sutures of No. 1-0 polypropylene passed through adjacent edges of the ligaments.
10. Lateral edge of each uterosacral ligament is sutured to the white line of that side with interrupted sutures of No. 1-0 polypropylene.
11. Redundant part of the vaginal mucosa is excised. The vaginal edges are sutured to each other in midline with interrupted sutures of No. 1-0 polyglactin.
12. The center of the vault of the vagina is suspended from the uterosacral ligaments in midline with a suture of No. 1-0 polyglactin.
13. The transverse edge of the anterior vagina is sutured to the transverse edge of the posterior vagina with interrupted sutures of No. 1-0 polyglactin.

Figure 1. Anterior colporrhaphy dissection has been done. The urinary bladder (UB), left uterosacral ligament (LUSL) and the right uterosacral ligament (RUSL) are seen.

Figure 2. No. 1 polyglactin ligature on the RUSL is threaded on a curved needle.

Figure 3. The cystocele is reduced by pressure of a finger.

Figure 4. The RUSL ligament is drawn forward along the right side of the urethra.

Figure 5. The needle is passed through the right flap of the vaginal mucosa, from inside out, at the level a little posterior to the external urinary meatus.

Figure 6. Another suture of No. 1 polyglactin suture is passed through the terminal part of the RUSL and tied.

Figure 7. The suture is passed through the right flap of the vaginal mucosa, from inside out, 5 mm away from the first suture.

Figure 8. The LUSL ligament (arrows) is drawn forward along the left side of the urethra.

Figure 9. No. 1 polyglactin ligature on the LUSL is threaded on a curved needle.

Figure 10. The needle is passed through the left flap of the vaginal mucosa, from inside out, at the same level as the first suture on the right side.

Figure 11. Another suture of No. 1 polyglactin suture is passed through the terminal part of the LUSL and tied. It is passed through the left flap of the vaginal mucosa, from inside out, 5 mm away from the previous suture.

Figure 12. The two sutures on the left side are tied on the external surface of the left flap of vaginal mucosa. The sutures on the right side are tied similarly.

Figure 13. The RUSL (black arrows) and LUSL (white arrows) are seen to be drawn forwards under the base of the urinary bladder.

Figure 14. The two uterosacral ligaments are approximated in the midline with interrupted sutures of No. 1-0 polypropylene.

Figure 15. Approximation of the two uterosacral ligaments in the midline is complete.

Figure 16. The gap between the LUSL and left lateral pelvic wall is demonstrated by passing a finger between the two.

Figure 17. The left edge of the LUSL is sutured to the left white line with interrupted sutures of No. 1-0 polypropylene.

Figure 18. The right edge of the RUSL is sutured to the right white line with interrupted sutures of No. 1-0 polypropylene.

Figure 19. The end result: the two uterosacral ligaments have been sutured to each other in midline, and each one has been sutured to the white line of the respective side laterally.

Discussion

Pelvic endopelvic fascia is fibroelastic connective tissue matrix with varying amounts of smooth muscle. It covers all the pelvic organs It fills the space between the pelvic diaphragm, the pelvic sidewall, and the visceral peritoneum. The endopelvic fascia covering the obturator internus is called as the obturator fascia. It is well defined in the arcus tendineus fascia pelvis (white line). It is condensed to form the pubocervical ligaments, which are attached to the under surface of the superior pubic ramus, the arcus tendineus fascia pelvis and the pericervical ring. This insertion is continuous with and forms part of the pericervical ring. The pubovesicocervical fascia is attached to the pubic tubercles, the pubic arch, the urogenital diaphragm, the white line, the pericervical ring and the cardinal ligaments. It is continuous with the fascia over the urinary bladder and the vagina. Continuity of the fascia, the uterosacral-cardinal ligament complex and vesical fascia is essential for keeping the bladder supported.[1,2] If the fascia gets detached from the white line, a lateral cystocele develops.[9] If it gets separated from the pericervical ring and the uterosacral-cardinal cardinal ligament complex, a central transverse type of cystocele develops.[9] Identification of these defects is easy clinically and during anterior colporrhaphy.

If the endopelvic fascia is too weak, a site specific repair does not often work because there is no specific defect located at any particular site, but all the fascia is weak. In such situation, if the uterosacral-cardinal ligaments are thick, they can be used to support the urinary bladder. In the new operation described here, the uterosacral ligaments are carried forward in the same direction in which they are present normally. That becomes possible because they are lengthened due to genital prolapse. Since they originate from the same endopelvic fascia, being its condensed parts, they can be used effectively for the repair of the cystocele. When they are joined to each other in the midline and fixed under the urinary bladder, they provide a strong and wide shelf for the urinary bladder to rest on. There are gaps between the sides of the ligaments and the obturator fascia, and if these are not occluded, the patient may present with uni- or bilateral cystocele. These gaps are closed by suturing the lateral edges of the uterosacral ligaments to the white lines of the respective sides. This results in reestablishment of continuity of fascia between the white lines laterally and uterosacral ligaments posteriorly. Provision of such a strong support with endogenous tissue does away with the need for synthetic meshes and possibility of complications associated with their use.

References

1. Milley PS, Nichols DH. The relationship between the pubocervical ligaments and the urogenital diaphragm in the human female. Anat Rec. 1969;163:433.
2. Richardson AC, Lyon JB, Williams NL. A new look at pelvic relaxation. Am J Obstet Gynecol 1976;126:568.
3. Richardson AC. Female pelvic floor support defects. Int Urogynecol J Pelvic Floor Dysfunct I 996;7(5):241.
4. DeLancey JO. Pelvic organ prolapse: clinical management and scientific foundations. Clin Obstet Gynecol 1993;36:895.
5. Johnston SL, Low JA. Anterior colporrhaphy. In Drutz HP, Herschorn S, Diamant NE. Eds Female Pelvic Medicine and Reconstructive Pelvic Surgery. 1st edition. London. Springer. 2017. pp 329-337.
6. Karram MM. Vaginal operations for prolapse. In: Baggish MS, Karram MM, eds. Atlas of Anatomy and Gynecologic Surgery. Philadelphia: WB Saunders, 2001:394.
7. Weber AM, Walters MD. Anterior vaginal prolapse: review of anatomy and techniques of surgical repair. Obstet Gynecol 1997;89:311.
8. Kovac SR, Stubbs JTF. Repair of the Anterior Segment. in  Advances in Reconstructive Vaginal Surgery. Kovac SR; Zimmerman CW. eds.  Lippincott Williams & Wilkins. 1st Edition. 2007. pp 470-551.
9. Zimmerman CW. New concepts in restoration of the anterior vaginal compartment. Operative Techniques in Gynecologic Surgery 2001;6:116.
10. Link G, van Dooren IM, Wieringa NM. The extended reconstruction of the pubocervical layer appears superior  to  the  simple  plication  of the  bladder adventitia concerning anterior colporrhaphy: a  description of two techniques  in  an  observational  retrospective analysis. Gynecol Obstet Invest 2011;72(4):274-280.
11. Morse AN, O'dell KK, Howard AE, Baker SP, Aronson MP, Young SB. Midline anterior repair alone vs anterior repair plus vaginal paravaginal repair: a comparison of anatomic and quality of life outcomes. Int Urogynecol J Pelvic Floor Dysfunction. 2007;18:245-9.
12. Hosni MM, El-Feky AEH, Agur WI, Khater EM. Evaluation of three different surgical approaches in repairing paravaginal support defects: a comparative trial. Arch Gynecol Obstet 2013;288(6):1341–1348.

Citation

Parulekar SV. Uterosacral Shelf For Cystocele Repair. JPGO 2017. Volume 4 No.10. Available from: http://www.jpgo.org/2017/10/uterosacral-shelf-for-cystocele-repair.html

Laparoscopic Management Of Para-Ovarian Cyst Torsion With 20-Week Pregnancy

Author Information 

Bhate A*, Joshi D** , Chitnis S***.

(* Director, Shubhdeep Nursing Home, Andheri, Mumbai, ** Consultant, Leela Joshi Hospital, Bhiwandi, Thane, *** Consultant, Department of Obstetrics & Gynaecology, MaxCure Superspeciality, Jogeshwari, Mumbai, India.)

Abstract 

Adnexal torsion (AT) in pregnancy is a rare condition which presents as an acute abdomen. The diagnosis may be missed because of accompanying non-specific signs and symptoms. Diagnostic laparoscopy followed by detorsion and cystectomy, offer fair chances of saving the adnexa as well as facilitate early recovery. We present a case of acute torsion of a large para-ovarian cyst which was treated by laparoscopy, where both ovary and fallopian tube could be preserved.

Introduction

Adnexal torsion remains a rare emergency which eludes diagnosis. It must be considered as a differential diagnosis in pregnant patients presenting with acute pelvic pain.[1] It has similar presentation in pregnant and non-pregnant women, although underlying pathology may be different. There has been an observation that majority of cases in pregnancy are associated with functional cysts.[2] Torsion during pregnancy occurs primarily in the first or the second trimester.[3] Torsion in pregnancy is increasingly being reported due to liberal use of ultrasound in pregnancy, which also happens to be the most valuable tool in diagnosis of torsion. Earlier, all cases were treated by laparotomy and adnexectomy. This now has been popularly replaced by laparoscopy due to its associated safety, faster recovery and minimal risk to the fetus.

Case Report

A 23 year old primigravida visited antenatal clinic for the first time with 5 months of amenorrhea associated with pain in abdomen. Pain was dull aching in nature, non-radiating and aggravated by activity. She had no other complaints such as vomiting or fever. She had conceived spontaneously and had an uneventful pregnancy so far. She appeared pale but had stable vital parameters. Uterine height corresponded to 24 weeks of gestation. She was admitted and started on intravenous isoxsuprine and micronized progesterone. Investigations revealed iron deficiency anemia and leucocytosis (hemoglobin of 7.6 g/dL and white blood cell count of 12,300/cmm). Her liver and renal function tests, urine routine and quadruple markers were normal. Ultrasound scan for anomalies was done. It showed a 19-20 weeks’ fetus in changing lie with prominent renal pelvis. A simple cyst of 10.4x7.2 cm was noted in the left adnexa with scanty flow in pedicle. Bilateral ovaries were seen separately and were normal in size. She had not undergone any investigations in the past, thereby the duration of adnexal mass could not be determined. Diagnosis of left adnexal torsion was made and decision for performing a laparoscopy was taken. 

She was transfused with 1 unit of packed cells prior to surgery. Peritoneal access was gained with supraumbilical port and lateral ports under general anesthesia. Port placement was higher than usual in order to prevent injury to the uterus. Upon entry into the abdomen, a left para-ovarian cyst with single twist of its pedicle was noted. Fallopian tube was stretched over the cyst. The left ovary was normal. Right ovary and fallopian tube were normal. Pedicle was derotated and cyst was enucleated. Cyst contents (clear straw colored fluid) were drained. It was a simple cyst with no solid component. The excised cyst wall was sent for histopathology. There was no compromise to ipsilateral fallopian tube and ovary. Post-operatively, the patient recovered uneventfully with no obstetric complications. 

Figure 1. Gravid uterus (U) and the paraovarian cyst (C).

Figure 2. Gravid uterus (U) and the paraovarian cyst (C), Ovary (O), Tube (T).

Figure 3. Retracting the gravid uterus.

Figure 4. Incision for enucleating the cyst.

Discussion

Adnexal masses may be found in 0.19-8.8% of all pregnancies.[4] Adnexal torsion is an emergency which warrants early diagnosis and treatment to prevent complications. A low incidence of 1-5 per 10000 pregnancies makes it a diagnostic dilemma.[5] Incidence of adnexal torsion rises considerably in pregnancies achieved after assisted reproductive techniques.[6] Medial torsion while considering infundibulopelvic ligament as the reference point has an overall higher incidence. In pregnancy however, lateral torsion is not uncommon and may be seen in as high as 35.6% women.[7] Symptoms are commonly non-specific, with pelvic pain being the commonest complaint. The patient may also have nausea, vomiting and fever. White blood cell count is frequently elevated. Outcome of pregnancy is variable and depends on gestational age.[8] 

Ultrasound is the most commonly used diagnostic tool. Presence of symptoms (nausea and vomiting), elevated leukocytes and ovarian cyst > 5 cm in diameter are indicative of torsion and aid in diagnosis. Magnetic resonance imaging may be used to aid diagnosis in doubtful cases.

Several approaches have been used for surgical intervention in adnexal torsion, e.g., laparotomy (Pfannenstiel, midline vertical incision or para-rectal incision).[9] Laparoscopy is a reasonably safe technique for establishing diagnosis and treatment in the same sitting.[4] It may be carried out in any trimester with minimum risk to mother and fetus, although peritoneal entry may be problematic in advanced gestation.[10] Entry through Palmar’s space, located in the mid clavicular line 3 cm below the coastal margin, is a suitable alternative in such cases.[11] This approach is especially useful in cases where problems are anticipated in umbilical entry, as in presence of large fibroid, peritoneal adhesions and as in our case an enlarged gravid uterus. Entry may be made by Veress’ needle or small sized trocar and cannula. Our patient presented with pain in abdomen. She was admitted and swiftly investigated to reveal a large adnexal cyst with reduced flow in the pedicle along with anemia and leukocytosis. Peritoneal access during laparoscopy was a concern, hence supraumbilical port was placed 3-4 cm above the usual location. Lateral ports were placed under vision. 

Conclusion

Adnexal torsion in pregnancy is rare and clinical diagnosis is difficult due to vague symptoms and signs. Doppler ultrasound is a useful tool to diagnose torsion by detecting absence or presence of vascularity in the pedicle. It is also helpful in assessing whether the patient needs adnexectomy.[12] Laparoscopy can be safely used for treatment of adnexal torsion with good surgical and obstetric outcomes. It also has the advantage of less discomfort and early recovery.[8] In advanced gestation entry through Palmar’s point or open laparoscopy may be preferred in order to avoid penetrative injuries. Early surgical intervention in adnexal torsion is the key to prevent loss of vascularity in adnexae. Adnexectomy can be prevented by timely treatment of torsion.

References

1. Bouguizane S, Bibi H, Farhat Y, Dhifallah S, Darraji F, Hidar S, et al. [Adnexal torsion: a report of 135 cases]. J Gynecol Obstet Biol Reprod (Paris). France; 2003 Oct;32(6):535–40. 
2. Melcer Y, Sarig-Meth T, Maymon R, Pansky M, Vaknin Z, Smorgick N. Similar But Different: A Comparison of Adnexal Torsion in Pediatric, Adolescent,  and Pregnant and Reproductive-Age Women. J Womens Health (Larchmt). 2016;25(4):391–6. 
3. Fouedjio JH, Fouogue JT, Fouelifack FY, Nangue C, Sando Z, Mbu RE. [Torsion of uterine appendages during pregnancy: report of a case at Yaounde Central Hospital, Cameroon]. Pan Afr Med J. Uganda; 2014;17:39. 
4. Cavaco-Gomes J, Moreira CJ, Rocha A, Mota R, Paiva V, Costa A. Investigation and Management of Adnexal Masses in Pregnancy. Scientifica (Cairo).2016;2016:3012802.
5. Bras R, Braga J, Tome A, Ferreira H. Adnexal Torsion in the First Trimester of Pregnancy: Diagnosis, Laparoscopic Management, and Review of the Literature. Surg Technol Int.2017;30:210–4. 
6. Hasson J, Tsafrir Z, Azem F, Bar-On S, Almog B, Mashiach R, et al. Comparison of adnexal torsion between pregnant and nonpregnant women. Am J Obstet Gynecol.  2010 ;202(6):536.e1-6. 
7. Krissi H, Hiersch L, Aviram A, Ashwal E, Goldschmit C, Peled Y. Factors Affecting Adnexal Torsion Direction: A Retrospective Cohort Study. Gynecol Obstet Invest.  2016;81(5):405–10. 
8. Chang S-D, Yen C-F, Lo L-M, Lee C-L, Liang C-C. Surgical intervention for maternal ovarian torsion in pregnancy. Taiwan J Obstet Gynecol.2011;50(4):458–62.
9. Basaranoglu S, Agacayak E, Tune SY, Icen MS, Turgut A, Peker N, et al. Clinical experience in pregnancies complicated by adnexal torsion. Clin Exp Obstet Gynecol. 2016;43(3):345–9. 
10. Weiner E, Mizrachi Y, Keidar R, Kerner R, Golan A, Sagiv R. Laparoscopic surgery performed in advanced pregnancy compared to early pregnancy. Arch Gynecol Obstet. 2015;292(5):1063–8. 
11. Ramcharan P, Flynn M, Berkshire Grant G. You have to prove yourself all the time: People with learning disability. In: Grant G, Goward P, Richardson M and Paul Ramcharan P editors. Learning Disability: A life cycle approach. 2nd ed. England. McGraw -Hill  Education. Open University Press. 2010; p. 311–28. 
12. Arena S, Canonico S, Luzi G, Epicoco G, Brusco GF, Affronti G. Ovarian torsion in in vitro fertilization-induced twin pregnancy: combination of  Doppler ultrasound and laparoscopy in diagnosis and treatment can quickly solve the case. Fertil Steril 2009;92(4):1496(e9-13).

Citation

Bhate A, Joshi D, Chitnis S. Laparoscopic Management Of Para-Ovarian Cyst Torsion With 20-Week Pregnancy.  JPGO 2017. Volume 4 No. 10. Available from: http://www.jpgo.org/2017/10/laparoscopic-management-of-para-ovarian.html

Deranged Liver Enzymes In A Case Of Diabetes In Pregnancy: Diagnostic Dilemma

Author Information

Saxena A*, Thunga C**, Madhva Prasad S***, Gupta AS****.

(* First year Resident, ** Senior Resident, *** Assistant Professor, **** Professor, Department of Obstetrics and Gynecology, Seth GS Medical College and KEM Hospital, Mumbai.)

Abstract

A case of a pregnancy with diabetes mellitus and incidental finding of deranged liver enzymes and the diagnostic dilemma associated with such a condition is presented here. A brief discussion of the possibilities is presented.

Introduction

Diabetes in pregnancy can be classified as those known to have diabetes prior to pregnancy, pre gestational or overt diabetes; and those diagnosed during pregnancy, gestational diabetes mellitus. Classification of non pregnant individuals with diabetes is based on the presumed etiopathogenesis and pathological manifestations, broadly as Type 1 diabetes with absolute insulin deficiency and Type 2 diabetes with insulin resistance. Most of the gestational diabetic women are likely to have Type 2 diabetes which had been undiagnosed till pregnancy.[1]

Concern is mainly due to the complications associated with diabetes in pregnancy, both maternal and fetal morbidity and mortality.  Most of them present with deranged blood sugar levels and no symptoms. Some present with complications like ketoacidosis, hypoglycemia, hypertension, infections, pre eclampsia, diabetic nephropathy, neuropathy, and or retinopathy. There are certain unusual manifestations like this case that we are presenting. There was an incidental finding of asymptomatic deranged liver enzymes in a pregnant woman who had diabetes and  no other cause could be found for the same.

Case Report

Twenty nine years old woman, married since 12 years, gravida 4 para 2 abortion 1 living 1, presented to the outpatient department at 31 weeks of gestation. Her pregnancy was complicated by preeclampsia; and the first living issue was a was 3 kg male child, born from a full term lower segment cesarean section, who lived well but died at 4 months of age due to pneumonia. Her second child was a female born from a full term normal delivery, 2.5 kg at birth and is now 8 years old. After this she had a spontaneous abortion at 3 months of amenorrhea, followed by a check curettage.  There was no history of raised blood sugars in any of the previous pregnancies. Her both parents were diabetic.

In this pregnancy, she had registered elsewhere, and presented at 31 weeks with abnormal fasting blood sugars and was admitted for the same.  On examination, she was conscious, oriented, with normal pulse, blood pressure, cardiovascular and respiratory system examinations. On local examination uterus was 30 weeks in size with a live fetus in cephalic presentation and closed cervical os. She had a report of fasting blood sugars measuring 108 mg %. Oral glucose tolerance test was done with 75 gms glucose and the values (all in mg %)  were 216 (fasting), 307 (1 hour), 322 mg (2 hour) and 288 (3 hours).

Endocrinologists advised diabetic diet with self monitoring of blood glucose 6 times a day, and injection Human Insulin [R] 10-10-10-0 U and Human Insulin [N] 22-0-0-26 U, subcutaneously was started. She was given injectable steroids for lung maturation (dexamethasone 6 mg 4 doses 12 hours apart) and insulin values were adjusted accordingly. 

Incidentally, abnormal liver function tests (LFTs) were detected. Ultrasound abdomen was done which was suggestive of normal liver echotexture with no focal lesions but the gall bladder was distended with sludge. All viral markers of hepatitis and HIV were negative. She gave no history suggestive of any liver disorder such as icterus, itching, nausea, vomiting, oedema, or abnormal bleeding. Upon further questioning, she reported use of some homeopathic medications for 6 months that had been discontinued for the last 2 months. Gastroenterologists opined to avoid hepatotoxic drugs and monitor liver function tests every 3rd day.  

However, in view of increasing trends of liver enzymes (as shown in table 1), a decision to terminate pregnancy was taken (at 30weeks 3 days of gestation). Her coagulation profile was checked and was normal. She was taken up for LSCS in view of breech presentation with worsening of liver function tests. She delivered a female child of 1.862 kg, with Apgar score 0f 9/10, was admitted to NICU where she initially required continuous positive airway pressure ventilation and was gradually weaned off. Injection insulin was discontinued on postpartum day 3 and converted to tablet metformin 500 mg 2-1-2 at discharge. Wound check was healthy, daily blood sugars and liver function tests were monitored (as shown in the table 1). She was discharged on day 5 and was asked to follow up in OPD after 15 days for suture removal and her LFT reports. On 19th post operative day when she came to OPD for follow up her LFTs had come to normal. Her recent fasting sugars were 150 mg% and she was taking medications for the same.

Table 1: Pattern of platelet counts, liver function tests and fasting blood sugars from antenatal to postpartum period during her hospital stay 

	On admission	6 days     prior	3 days prior	1day prior	Day Of LSCS	Day 1 Post op	Day 2 Post op	Day 4 Post op	Day 19 Post op
Hb  (gm%)	10.2		9.1	8.5	8.8	10.1	11.3	10.8
Platelet Count (cmm)	1.15 lakh		1.2 lakh	1.02 lakh	1.1 lakh	1.4 lakh	1.4 lakh	1.6 lakh
Alkaline Phosphatase	536	571	450	427	420	-	-	396	298
SGOT (IU/ml)	195	213	307	317	248	332	76	55	31
SGPT (IU/ml)	136	166	277	299	257	341	211	114	14
FBS (mg%)	274	228	267	197	153	140	132	136

Discussion

Liver has a role in maintaining normal glucose homeostasis. In our patient, both liver enzyme derangement and abnormal blood sugar values were detected at approximately the same time. Whether one preceded the other remote from the time of detection is not known. In such a scenario, any of the possibilities could have existed. That is, liver disease as a consequence of diabetes, diabetes as a complication of liver disorder or liver disorder coincidental with diabetes. This association may explain the pathogenesis behind diabetes and abnormal liver enzymes, or it could be purely coincidental.

One study done in 2011 found an association between insulin resistance and aminotransferases, in the absence of detectable steatosis by ultrasonography. This study indicated that in diabetes, even mild steatosis is sufficient to mediate the derangement of aminotransferases.[2]

The presentation of non-alcoholic steatohepatitis can vary from asymptomatic elevated liver enzymes to full blown fibrosis and nodular regeneration. Hence they can be considered as a cause for chronically elevated liver enzymes in asymptomatic diabetic patients, particularly if they are obese and have hyperlipidemia.[3] This could be one of the differential diagnosis in our case. Our patient had BMI of 31.8 kg/m2 but lipid values were not checked.

Ultrasound abdomen showed a normal liver echotexture with no focal lesions and a distended gall bladder with sludge.  No specific correlation between  pregnancy an non alcoholic steatohepatitis have been reported.  Weight loss upto 10%, good control of blood glucose levels and addition of ursodeoxycholic acid can help in normalizing the liver enzymes in such conditions.[4, 5]

Biliary diseases and diabetes have an association, obesity being a common factor. Although in pregnancy, lithogenicity or the cholesterol saturation index of bile increases due to hyperestrogenemia, the biliary sludge that appears in pregnancy is known to resolve post partum and rarely results in gall stones. Our patient had obesity and gall bladder sludge. However, gallstone disease in pregnancy is extremely rare.[6]

Some studies have shown rare association between the use of oral hypoglycemics and liver injury, mostly with use of sulphonylureas, causing chronic hepatitis and necroinflammatory changes.[6] This possibility is less probable in our case as she was diagnosed with diabetes during antenatal visit and was not on oral hypoglycemics earlier. However, this is relevant in patients who may present with diabetes and raised liver enzymes.[7]

Though we cannot comment on the role of homeopathic medications that she had consumed prior to the ANC visit, literature reports a study of toxic hepatitis or drug induced liver injury in Type 2 diabetic patients treated with Gymnema sylvestre. This is a plant which is known to be a potent antidiabetic, widely used in Ayurveda and Homeopathy medications.[8]

A study done by Frazer and associates found an association between chronic hepatitis C and impaired glucose tolerance, and reported that diabetes is found to be more frequent in patients with hepatitis C than in general population.[9] Our patient was negative for viral markers of hepatitis. 

Autoimmune hepatitis is another progressive liver disease that can manifest at any time of life, including pregnancy and postpartum. Pregnancy is a period of immune tolerance hence the disease activity of autoimmune conditions attenuates during antenatal period but worsens during postpartum period. Hence post pregnancy is a better time to evaluate presence of autoantibodies.[10] This is not applicable to our study as her LFTs normalized after delivery.

Considering the liver diseases unique to pregnancy, HELLP syndrome which is a complication occurring as a sequel to severe pre eclampsia must be considered here. It is also known that there is a high risk for a diabetic mother to develop chronic or gestational hypertension,  specially pre eclampsia. A study by Yanit and colleagues in 2012, reported that pre eclampsia developed three or four times more often in women with overt diabetes.[11]

Another possible explanation for our case could be atypical HELLP syndrome with normal blood pressure in a diabetic pregnant woman. HELLP syndrome comprises of hemolysis, elevated liver enzymes and low platelet count, as a complication in a patient with severe pre eclampsia. Literature study reveals some atypical presentations of this condition where no other etiology could be considered.[12] After delivery the liver enzymes level began to reduce, with improvement in haemoglobin levels and platelet count. 

To conclude, diabetes in pregnancy and liver disease could coexist in a patient with various manifestations and possible conditions are discussed above. The condition is discussed because sometimes decisions to terminate pregnancy need to be taken even without a specific diagnosis, and many a time extensive evaluation in postpartum period may be required for accurate diagnosis.  

References

1. Feig DS, Palda VA. Type 2 diabetes in pregnancy: a growing concern.Lancet. 2002;359(9318):1690-2.
2. Esteghamati A, Noshad S, Khalilzadeh O, Khalili M, Zandieh A, Nakhjavani M. Insulin resistance is independently associated with liver aminotransferases in diabetic patients without ultrasound signs of nonalcoholic fatty liver disease. Metab Syndr Relat Disord. 2011;9(2):111-7. 
3. Sheth SG, Gordon FD, Chopra S. Nonalcoholic steatohepatitis. Ann Intern Med. 1997; 126(2):137-45.
4. Palmer M, Schaffner F. Effect of weight reduction on hepatic abnormalities in overweight patients. Gastroenterology. 1990;99(5):1408-13.
5. Laurin J, Lindor KD, Crippin JS, Gossard A, Gores GJ, Ludwig J, et al. Ursodeoxycholic acid or clofibrate in the treatment of non-alcohol-induced steatohepatitis: a pilot study.Hepatology. 1996;23(6):1464-7.
6. Kolbeinsson HM, Hardardottir H, Birgisson G, Moller PH. Gallstone disease during pregnancy at Landspitali University Hospital 1990-2010. Laeknabladid. 2016; 102(12):538-542.
7. Sola D, Rossi L, Schianca GP, Maffioli P, Bigliocca M, Mella R, et al. Sulfonylureas and their use in clinical practice. Arch Med Sci. 2015; 11(4): 840–848.
8. Shiyovich A, Sztarkier I, Nesher L. Toxic hepatitis induced by Gymnema sylvestre, a natural remedy for type 2 diabetes mellitus. Am J Med Sci 2010;340 (6): 514-7.
9. Fraser GM, Harman I, Meller N, Niv Y, Porath A. Diabetes mellitus is associated with chronic hepatitis C but not chronic hepatitis B infection. Isr J Med Sci 1996; 32(7):526-30.
10. Mehta V, Prasad M, Gupta AS. Autoimmune Hepatitis in Pregnancy. JPGO 2016;3(8). Available from: http://www.jpgo.org/2016/08/autoimmune-hepatitis-in-pregnancy.htm
11. Yanit KE, Snowden JM, Cheng YM, Caughey AB. The impact of chronic hypertension and pregestational diabetes on pregnancy outcomes Am J Obstet Gynecol 2012; 207(4): 333
12. Garrido MF, Carvajal JA. Normotensive HELLP syndrome: report of one case. Rev Med Chil. 2013; 141(11):1470-4.

Citation

Saxena A, Thunga C, Madhva Prasad S, Gupta AS. Deranged Liver Enzymes In A Case Of Diabetes In Pregnancy:  Diagnostic Dilemma.  JPGO 2017. Volume 4 No.10. Available from: http://www.jpgo.org/2017/10/deranged-liver-enzymes-in-case-of.html

Lateral Approach To Hemihematometra

Innovation

Author Information

Shetty A*, Madhva Prasad S**, Gupta AS***.

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

Abstract

Developmental anomalies of the Müllerian duct system represent some of the most fascinating disorders that obstetricians and gynecologists encounter. We present to you a case in which lateral approach was used to reach the collection and for excision of a non-communicating functional uterine horn. 

Introduction

Müllerian duct anomalies may produce reproductive failure like abortion and preterm birth, or obstetric problems like malpresentation, retained placenta, or they may be asymptomatic. Uterus with a non-communicating functional horn is a type of Mullerian anomaly and is a rare cause of dysmenorrhea. Here, we present such a case and discuss the management strategies. Reaching and excising such a functional but a noncommunicating uterine horn can be challenging at times but excision is warranted to improve the symptomatology. Chances of occurrence of endometriosis are reduced and future fertility outcomes are likely to improve significantly.

Case Report

A 14 year old unmarried girl, who had attained menarche a year prior, presented with severe incapacitating dysmenorrhea ever since onset of menses.  Cycle length was 28 days with 5 days flow. It was accompanied by severe pain, the height of which was during the menstrual flow. She was evaluated outside and found to have obstructed right functional non-communicating uterine horn and a left functional normal horn and no other medical or surgical illness. 

Her general and and detailed systemic examination were within normal limits. Abdomen was soft, non-tender and no masses were felt. Per rectal examination showed midline globular tender right horn of the uterus with what appeared as a distinct palpable depression; across which a left side horn was felt. No adnexa was felt through the right or the left fornix. Impression was obstructed right functional non-communicating horn with left functional normal horn. 

Ultrasonography (figure 1) showed uterus anteverted normal size and bicornuate.  Left cornual endometrial echo could be traced up to cervical canal with continuity of the lumen. Right cornua was not connected to the cervical canal but it ended bluntly. Organised blood echoes were seen in this endometrial cavity. 

MRI showed bicornuate uterus with right non-communicating horn, hyperintense collection in the endometrial cavity (figure 2) with right fallopian tube distended with hyperintense fluid suggestive of a hydrosalpinx. Left horn was normal. She was planned for an exploratory laparotomy with right salpingectomy and excision of right obstructed functional non-communicating horn with metroplasty. Intraoperatively, bilateral ovaries were normal. Left fallopian tube was otherwise normal but had attachment at top of the uterus. There was no uterine fundus above the insertion of the fallopian tubes. Uterine contour was normal on external examination. A faint bulge was seen over the right cornuo-fundal region on careful examination. Upon palpation, a firm globular swelling was felt in the right cornuo-fundal region; with doubtful communication to the cervical canal. However, cervix was single and appeared to be in normal communication with the left hemi-uterus. Right cornual end of the tube had a 1 cm atretic segment with doubtful connectivity to uterine cavity.  Hydrosalpinx was present from cornual end just distal to the atretic segment; extending close to fimbrial end but stopping short of fimbrial end with another atretic segment. (figure 3).

Salpingectomy was done carefully ensuring that the ovarian blood supply was not compromised. Needle passed from anterior and posterior surfaces into the presumptive area of hematometra, could not reach the collection. Here, an innovative procedure suggested by Dr. S.V. Parulekar was used. Needle was passed into the hematometra through a lateral approach, between the right round ligament and the right  utero-ovarian ligaments. Old collected blood about 2 cc was aspirated out. (figure 4). Diagnosis of hemi-hematometra was confirmed. With the needle as the guide a stab incision was taken and deepened (figure 5) and endometrial cavity was visualized. Lateral part of endometrium showed the tubal ostia, confirming endometrial cavity and tubal attachment. (figure 6). Gentle blunt probing was done and the extent of the horn was assessed in all directions; it was found to be around 2x2x2 centimeter. The endometrium, along with a few millimeter of myometrium was excised using cautery taking care not to excise deep into the tissue (figure 7) so as not to damage the septum between the 2 horns.  Cauterization was immediately stopped when the lateral part of the wall of the functioning uterine cavity was identified.  Excised tissue was sent for histopathological examination. After ensuring that no redundant endometrium was left in the right horn, small sub-centimeter pieces of gel foam were placed; and myometrial bed was approximated in consecutive layers from below upwards by taking continuous interlocking sutures with polyglactin No 1-0. By this, the functional cavity of the redundant obstructed right horn of the uterus was made non functional and was obliterated. So as to leave behind a single functional cavity of the uterus with communication to the cervix (figure 8). Postoperative course was uneventful and she menstruated subsequently and did not experience any severe excruciating dysmenorrhea..  

Figure 1. Sketch drawn by sonologist to depict the pathology.

Figure 2. MRI image with blue arrow pointing to distended uterine horn and red arrow pointing to normal uterine cavity.

Figure 3. Intraoperative findings showing hydrosalpinx and slight right sided uterine bulge.

Figure 4. Laterally inserted needle aspirating blood confirming hematometra.

Figure 5. Proceeding with lateral dissection.

Figure 6. Right sided functional horn excision and coring of endometrium. Blue arrow pointing out to coagulated myometrium, green arrow is pointing out the left horn bulging through the septum between the two horns.

Figure 7. Cavity sutured.

Figure 8. Final view after suturing of serosa. 

Discussion

Surgical procedure designates the correct diagnosis of type of Mullerian anomaly. In our case, the MRI was suggestive of bicornuate uterus with a non-communicating horn. However, intraoperative features, which is the gold standard, showed it to be a septate uterus with a partial septum running from top of the uterus to the right wall, dividing the uterine cavity unequally resulting in non-communication with the cervical canal on the right side. The functional endometrium thus trapped within the smaller cavity caused the severe dysmenorrhea and made her seek medical help soon after menarche.

Terminology and classification of Mullerian anomalies continues to be imperfect, and our patient would probably not fit into any of the classes in the American Fertility Society classification, the VCUAM classification or the Clinical Embryological classification. It would fit into a rather broad U6 (Unclassified Malformations) in the ESHRE classification.[1] The EAC classification is one which may help in correct scientific communication of this malformation, the main advantage being that functionality is also taken into account;[2] and not just the morphological features, as in most other classifications. This patient fits into an unclassified Mullerian anomaly.  

When there is no communication between the functional horn and its main functional endometrial cavity or the cervix, symptomatology of severe dysmenorrhea, hematometra and hematosalpinx is expected.[3]

In our case, the symptomatology was seen and hematometra was observed; but hematosalpinx was not observed. This is due to atretic segments of fallopian tube in the cornual end. Probably due to this, she presented at a much earlier time than otherwise expected. Endometriosis was also not seen, though it is expected in patients with non-communicating functional horns with canalized fallopian tubes due to retrograde menstruation. 

Many different methods of imaging can be used for confirmation of type of Mullerian abnormality. Ultrasonography is a well-accepted modality; however, 3 dimensional ultrasonography is the best and performs well in comparison to gold standard MRI.[4]   As the collection was small we could not reach it anteriorly or from the posterior or superior aspect. However as the myometrium is thinnest in the lateral wall at the cornua the lateral approach worked by a lateral thought process. Though not entirely similar, when other techniques are not feasible, a lateral approach can be considered in surgery of the uterus, as described for myomectomy procedures.[5]

Excision of the horn and coring out of the endometrium was done. Our method also ensured that the resulting lateral wall of the functional horn did not weaken as the majority of the myometrium was retained after coring of the endometrium and closed so as to strengthen the inner septum. As described, this was achieved by open technique. If surgical expertise is available, laparoscopic/ hysteroscopic excision can be considered. But whether the outcomes are better, is not yet clear.[6,7] We present this case so that readers remember this lateral approach when faced with dilemma during surgery and restrict excision to bare minimum.

Acknowledgement

Dr. S.V. Parulekar, Professor and Head for innovating the unique lateral approach to reach the collection inside the hemiuterus of a septate uterus .

References

1. Grimbizis GF, Campo R. Clinical approach for the classification of congenital uterine malformations. Gynecol Surg. 2012; 9(2):119–29.
2. Parulekar SV. EAC Classification Of Congenital Malformations Of The Female Genital Tract. JPGO 2015;2(4). Available from: http://www.jpgo.org/2015/04/eac-classification-of-congenital.html
3. Woźniakowska E, Stępniak A, Czuczwar P, Milart P, Paszkowski T. Secondary dysmenorrhea due to a, non-communicating functional uterine horn. Ginekol Pol. 2017;88(7):404–5. 
4. Ergenoglu AM, Sahin Ç, Şimşek D, Akdemir A, Yeniel AÖ, Yerli H et al. Comparison of three-dimensional ultrasound and magnetic resonance imaging diagnosis in surgically proven Müllerian duct anomaly cases. Eur J Obstet Gynecol Reprod Biol. 2016 Feb;197:22-6.  
5. Parulekar SV. Myomectomy: Lateral Extraperitoneal Approach. JPGO 2014; 1(2). Available from: http://www.jpgo.org/2014/02/myomectomy-lateral-extraperitoneal.html
6. Dwivedi R, Perera K, Eedarapalli P. Non-Communicating inactive rudimentary  horn of the uterus presenting with dysmenorrhoea—a case report of successful laparoscopic excision. Open J Obstet Gynecol [Internet]. 2011;1:213–6. Available from: http://www.scirp.org/journal/ojog/
7. Akdemir A, Ergenoglu AM, Yeniel AÖ, Sendag F, Karadadaş N. Coring-type laparoscopic resection of a cavitated non-communicating horn under hysteroscopic assistance. J Obstet Gynaecol Res. 2014;40(7):1950–4.

Citation

Shetty A, Madhva Prasad S, Gupta AS. Lateral Approach To Hemihematometra. JPGO 2017. Volume 4 No.10. Available from: http://www.jpgo.org/2017/10/lateral-approach-to-hemihematometra.html