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Volume 3 Issue 11, November 2016

Editorial

Gupta AS

Pelvic masses detected late in pregnancy or in labor create diagnostic as well as management conflicts. These masses can be from gynecologic or non gynecologic organs. Uterine masses like leiomyomas, ovarian lesions like functional or neoplastic cysts (benign or malignant), paraovarian cysts or hydrosalpinx originate from the female reproductive organs whereas tumors form the bowel, bladder, ureters, muscles, bony pelvis are non gynecologic masses. Large fecoliths or bladder calculi can also masquerade as a pelvic mass. 
Large pelvic masses irrespective of the organ of origin if they lie below the presenting part in late pregnancy or at start of labor will prevent the descent of the presenting part and will lead to obstructed labor if undetected or neglected.
The occurrence of obstructed labor due to pelvic masses in recent times has reduced due to early detection and follow up or treatment in pregnancy with widespread practice of women registering early in pregnancy, having pelvic examinations and undergoing if not multiple then at least one ultrasonography in pregnancy. Presently, asymptomatic pelvic masses are detected due to almost ubiquitous use of ultrasonography and due to the increased sensitivity available with ultrasound technology. Previously, in the pre ultrasound period, many of these masses were detected either after symptoms developed or else during cesarean section or in the puerperium.
Most of the adnexal masses are ovarian cysts usually benign and functional. Simple cysts less than 5 cm in size usually resolve spontaneously in the second trimester of pregnancy and need only ultrasonography follow up. Complex ovarain cysts or even simple cysts larger than 5 cm in size do resolve spontaneously in about 70% of the women. If these cysts do not resolve spontaneously then surgical treatment may have to be considered. The first half of the pregnancy or upto 22 to 24 weeks of gestation is the best time recommended for surgery in the pregnant woman. Early surgery may be unnecessary as most of these lesions resolve. Delay in elective surgical treatment may result in an emergency procedure for indications like torsion. Approximately 5% of the adnexal masses are malignant. Doppler studies may be able to indicate its malignant nature. 
Once diagnosed, these masses need close followup to chart the most optimal treatment plan. In many cases surgery may be deferred for unresolved pelvic masses till cesarean section (not done for the pelvic mass) or till puerperium. Sometimes these masses increase in size like fibroids. Solid tumors like subserosal, pedunculated fibroids, or fibroids situated in the lower segment, and ovarian cysts may fill the pelvis and prevent engagement of the presenting part. Similarly large vesical calculi or impacted fecoliths in the rectum and the sigmoid colon reduce the pelvic diameters and either prevent engagement of the presenting part or cause obstructed labor. 
Though the number of women seeking antenatal care is increasing there is still a significant number of gravid women who present in labor or are delivered by untrained birth attendants. In early labor and with a detailed clinical examination and availability of ultrasound a provisional diagnosis is usually reached and appropriate treatment can be carried out without increased maternal and perinatal morbidity. However, when such a gravida presents in advanced labor showing all signs of obstructed labor; diagnosis becomes difficult but not impossible as many times the clinical findings are distorted. Ultrasonography, magnetic resonance imaging if available may provide the diagnosis. Delay in treatment can increase the maternal and perinatal morbidity and mortality. Chances of uterine rupture, ischemic necrosis of the bladder leading to fistula formation are real risks. Obstetrician has to anticipate difficulties during cesarean delivery due to the distorted anatomy and be careful to avoid hollow viscus injury.
This November issue of JPGO brings a couple of rare cases. One of these is a large vesical calculus diagnosed for the first time in labor. I hope our esteemed readers enjoy the collection of cases in this issue.

Subseptate Uterus, Double Cervix With Longitudinal Vaginal Septum: A Rare Case Report

Author Information

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

Abstract

Müllerian anomalies represent a vast range of structural abnormalities resulting from the improper development of the embryological müllerian ducts. Use of magnetic resonance imaging is very helpful to diagnose such anomalies. A nulligravida was diagnosed to have a septate uterus with double cervix and a vagina with partial longitudinal vaginal septum.  A rare developmental version is a septate uterus with cervical duplication and a complete longitudinal vaginal septum. The existence of this anomaly challenges classical müllerian developmental theory.

Introduction

Congenital uterine anomalies in the population are estimated to have an incidence of 0.001-10%.[1] A rare developmental anomaly is a septate uterus with cervical duplication and a complete longitudinal vaginal septum. The presence of this malformation challenges classical müllerian developmental theory. Fusion defects of the müllerian ducts have been described by the American Fertility Society.[2] But, septate uterus with cervical duplication and longitudinal vaginal septum is not detailed by this classification. This paper outlines description of such a case. Only six  such cases have been reported in the world literature.[1] This report should help understand the embryogenesis of the female genital tract better and also improve the existing classification systems.

Case Report

A 22-year-old woman, married for 4 years, presented with a complaint of oligomenorrhea and primary infertility. She reported menarche at age 13. Her past menstrual cycles were regular. Gynecological examination revealed a phenotypic female, breast development of Tanner stage IV, normal external genitalia, a longitudinal vaginal septum  and two normal appearing cervices (figure 1). A 3D ultrasound was suggestive of septate uterus (figure2). There were no anomalies of the urinary tract. Her reports of investigations for fitness for anesthesia were normal. Hysteroscopy showed incomplete uterine septum in the uterine cavity. The continuity of the uterine cavity below the lower edge of the septum and above the internal of each of the cervix was demonstrated by a curious phenomenon of the uterine distension fluid spurting out of the right cervix when a hysteroscope was passed into the left cervix and distension medium (Ringer's lactate) was injected under pressure (figure 3). It was visualized by passing a uterine sound through the right side cervix and visualizing it through the left cervix using the hysteroscope, from below the lower edge of the cervix.. Laparoscopy revealed a single uterus with broad fundus (figure 4) and bilateral hydrosalpinx (figure 5). The ovaries were normal.  The patient's postoperative course was uneventful.


Figure 1: 3D ultrasound  suggestive of septate uterus


Figure 2: View of double cervix with longitudinal vaginal septum.


Figure 3. Hysteroscopy fluid spurting out of the right cervix when hysteroscope was passed through the left cervix. 


Figure 4: Laparoscopic view of uterus.


Figure 5: Left hydrosalpoinx.

Discussion

This interesting müllerian anomaly of a partial uterine septum, dual cervices and longitudinal vaginal septum cannot be explained by the commonly used classification of anomalies suggested by Buttram.[3] Review of the literature reveals this to be an uncommonly reported anomaly.[4,5] These anomalies are contradictory with the theory of linear caudal to cephalad müllerian fusion as described by Crosby and Hill in 1962.[3] Specifically, this theory supports that uterine development is the result of the fusion of the müllerian ducts during the embryonic life in an early stage of development, starting at the most caudal aspect known as the müllerian tubercle and moving in a cranial direction. Septal resorption was thought to follow shortly thereafter, starting at any point of fusion and going in either or both directions. The case reported here cannot be made clear by this theory, because a dual vagina-cervix complex expresses failure of caudad fusion while a septate uterus indicates normal cephalad fusion with unsuccessful septal resorption. The present case is explained better by the hypothesis suggested by Musset et al. and advocated by McBean and Brumsted when they described one more similar anomaly.[4] Musset put forward an alternative, three-stage series of process in which the medial aspects of the müllerian ducts begin to fuse in the middle and continue in both the cephalad as well as caudal directions simultaneously. This is then accompanied by fast cellular proliferation between the ducts, configuring the uterine corpus and cervix, and septal resorption, all of which occur in either directions simultaneously. According to this supposition, the dual vagina/cervix complex could be described by failed fusion of the müllerian ducts in the caudal direction starting towards  the uterine isthmus. The septate uterus plus vagina could then be explained by complete failure of normal septal resorption after  normal fusion. This anomaly is classified by Toaff as a Type 1A uterine communication and further approves Musset's theory of embryologic development.[5] Parulekar’s EAC classification of congenital malformations of the female genital tract covers this anomaly.[6] Its description is as follows: FAR-hydrosalpinx,  FAL-hydrosalpinx, UtR1-5 cm Fn NoObst, UtR2-5 mm Fn NoObst, UtL1-5 cm Fn NoObst, UtL2-5 mm Fn NoObst, Ut longitudinal septum 4 cm, CxR1 1 cm Fn NoObst, CxR2 1.5 cm Fn NoObst, CxL1 1 cm Fn NoObst, CxL2 1.5 cm Fn NoObst, VgR 9 cm NoObst, VgL 9 cm NoObst, Vg longitudinal septum.

Conclusion

Single subseptate uterus with two cervices and complete vaginal septum is a rare congenital malformation of the female genital tract. It is best explained by  Musset’s theory of embrryonic development of the female genital tract. It is covered completely by EAC classification of congenital malformations of the female genital tract.

Acknowledments

We thank Dr Neha Agrawal for taking photographs in figures 1, 2, 4, and 5) and Dr Vibhav Manjrekar for figure 3.

References
  1. Wai CY, Zekam N, Sanz LE. Septate uterus with double cervix and longitudinal vaginal septum. A case report, J Reprod Med. 2001 Jun;46(6):613-7.
  2. Barbanti S, Chiamulera N, Botelho B. Vaginal Septoplasty in Septate Uterus with Double Cervix. Case Reports in Obstetrics and Gynecology Volume 2014 (2014). Available from: https://www.hindawi.com/journals/criog/2014/367360/
  3. Buttram VC Jr, Gibbons WE. Müllerian anomalies: A proposed classification (an analysis of 144 cases). Fertil Steril 1979;32:40-46.
  4. McBean, J.H., Brumsted, J.R. Septate uterus with cervical duplication: a rare malformation. Fertil Steril 1994;62:415-17.
  5. Toaff ME, Lev-Toaff AS, Toaff R. Communicating uteri: Review and classification with introduction of two previously unreported cases. Fertil Steril 1984;41:661-79.
  6. Parulekar SV. Classification Of Congenital Malformations Of The Female Genital Tract. JPGO 2015. Volume 2 No. 4, Available from: http://www.jpgo.org/2015/04/eac-classification-of-congenital.html
Citation

Chaudhari HK,  Parulekar SV. Subseptate Uterus, Double Cervix With Longitudinal Vaginal Septum: A Rare Case Report. JPGO 2016. Volume 3 No. 11. Available from: http://www.jpgo.org/2016/11/subseptate-uterus-double-cervix-with.html

Intestinal Malrotation In A Case Of Bicornuate Bicollis Uterus

Author Information

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

Abstract

Congenital malformations of the female genital tract may be seen in approximately 5 to 7 percent of the population. They are known to be associated with various other anomalies such as renal, musculoskeletal, gastrointestinal etc. We report an unusual association of an intestinal malrotation with the sigmoid colon on the right side in a case of bicornuate bicollis uterus with a longitudinal vaginal septum.

Introduction

Müllerian anomalies include various malformations in the female genital tract. They may be associated with renal and anorectal malformations, which occur sporadically, without any familial tendency.[1] Of these anomalies, uterus didelphys due to a failure of fusion of müllerian ducts, is seen in about 11%.[2] In 75% cases of didelphic uterus a longitudinal vaginal septum is seen.[3] Renal anomalies are seen in 20% patients with uterus didelphys, with renal agenesis being the most common anomaly. We report the case of a patient  uterus didelphys  with  intestinal malrotation.

Case Report

A 23 year old primigravida , married for 2 years, was scheduled for an elective cesarean section for breech presentation at term. The breech presentation was persistent in her third trimester. There was no medical or surgical illness in the past. There was no history of any bowel or urinary bladder related symptoms. Her clinical examination revealed a pulse of 86 beats per minute, blood pressure was 110/70mm of Hg. General and systemic examination revealed no abnormality. Abdominal examination showed uterine size of 38 weeks, breech presentation, and normal fetal heart sounds. A speculum examination showed complete longitudinal vaginal septum and two cervices. A vaginal examination showed a complete longitudinal vaginal septum and both the cervices uneffaced and closed. Ultrasonography (USG) showed a bicornuate uterus, a single live intra uterine gestation in the right horn of the uterus in breech presentation, and no anomalies of maternal urinary tract. Investigations for fitness for anesthesia showed normal findings. An elective lower segment caesarean section was performed. The patient delivered a male child of 2.3 kg with APGAR 9/10. Intraoperative findings revealed pregnancy in the right horn of a bicornuate bicollis uterus. Incidentally it was also found that the sigmoid and the appendix were on maternal right side. There was no intestinal obstruction. The rest of the anatomy was normal.  Post operative period was uneventful and the patient made a complete recovery.


Figure 1. Intraoperative findings: two horns of the uterus are seen from posterior aspect, the medial end of the suture in the lower segment incision of the right horn is seen (yellow arrow). The sigmoid colon is seen on the right side (black arrow).


Figure 2. Intraoperative findings: two horns of the uterus are seen from posterior aspect, the appendix is seen behind the right horn (black arrow).

Discussion

The uterus, fallopian tube, cervix and upper two thirds of the vagina develop from a pair of mullerian ducts while the lower third of the vagina develops separately from the urogenital sinus. The co-existence of müllerian anomalies with renal anomalies ipsilateral to the side of obstruction occurs due to arrest in embryological development at the eighth week of gestation, which may affect both paramesonephric and mesonephric ducts. Renal anomalies like ipsilateral renal agenesis,  renal dysplasia, double collecting system and ectopic ureter are known to be  associated with didelphys uterus.[4] It is also associated with duplication of inferior vena cava, high-riding aortic bifurcation, malposition of ovary and malrotation of intestine.[5] Uterus didelphys is a part of OHVIRA syndrome (obstructed hemivagina, ipsilateral renal anomaly).[6] Malrotation may occur alone or associated with other anomalies like atresia of small bowel. During normal abdominal development, the gut herniates out from the abdominal cavity, where it undergoes a 270 degrees counterclockwise rotation around the superior mesenteric vessels. Following this rotation, the bowel returns to the abdominal cavity, the duodenojejunal loop getting fixed to the left of the midline and the cecum getting fixed in the right lower quadrant. Intestinal nonrotation or incomplete rotation is any variation in this rotation and fixation of the gastrointestinal tract during development. Intestinal rotation and fixation can be interrupted during fetal development at different locations leading to various presentations of disease.[5] Stage I of midgut rotation occurs between 5-10 weeks' gestation, in which the physiologic herniation of the bowel takes place. Nonrotation is the result of arrest in development at stage I. As the gut lengthens without rotation, the mesentery forms a narrow base predisposing to to volvulus. Stage II is at 10 weeks  of gestation, in which the bowel returns to the abdominal cavity. Incomplete rotation is due to arrest in stage II. It is likely to result in duodenal obstruction by compression of its third portio, or due to internal herniations. Stage III is from 11 weeks of gestation to term, in which the cecum descends to the right lower quadrant and the mesenteries get fixed.
This case is interesting because of the association of malformations of the genital tract and gastrointestinal tract. The incomplete rotation of the bowel made the patient vulnerable to intestinal obstruction. She was counseled accoridngly and advised to retain the operative notes carefully and report to a medical center in case she developed constipation and vomiting associated with abdominal distension at any time in future. No operative treatment was given for the incomplete rotation because none was warranted.

References
  1. Edmonds DK: Rokitansky syndrome and other Mullerian anomalies. In: Balen AH, Creighton SM, Davies MC et al. (eds.), Paediatric and adolescent gynaecology. Cambridge, England: Cambridge University Press, 2004. pp 267–74.
  2. Nahum GG: Uterine anomalies. How common are they, and what is their distribution among types? J Reprod Med 1998;43: 877–87.
  3. Buttram VC Jr: Müllerian anomalies and their management. Fertil Steril 1983; 40(2):159–63.
  4. Coskun A, Okur N, Ozdemir O, Kiran G. Uterus didelphys with an obstructed unilateral vagina by a transverse vaginal septum associated with ipsilateral renal agenesis, duplication of inferior vena cava, high-riding aortic bifurcation and intestinal malrotation. Fertil Steril 2008;90:2006.
  5. Madureira AJ, Mariz CM, Bernardes JC, Ramos IM. Uterus didelphys with obstructing hemivaginal septum and ipsilateral renal agenesis. Radiology 2006;239:602e6.
Citation

Dalvi P, Parulekar SV. Intestinal Malrotation In A Case Of Bicornuate Bicollis Uterus. JPGO 2016. Volume 3 No. 11. Available from: http://www.jpgo.org/2016/11/intestinal-malrotation-in-case-of.html

Obstetric Hysterectomy: A Life Saving Measure For Puerperal Sepsis

Author Information

Shah S*, Pardeshi S**, Warke H***, Mayadeo N****.
(* Second year resident, ** Assistant Professor, *** Associate Professor, **** Professor. Department of Obstetrics and Gynecology, Seth GS Medical College & KEM hospital, Mumbai, India.)

Abstract

Puerperal sepsis is one of the major cause of maternal mortality among the developing countries as well as worldwide. In modern obstetrics with increasing number of hospital deliveries, deliveries by trained personnel, introduction of aseptic measures and availability of higher antibiotics the incidence of maternal mortality due to puerperal sepsis has greatly reduced. Here we are presenting a rare case of puerperal sepsis following a failed attempt of manual removal of placenta which then warranted an obstetric hysterectomy.

Introduction

Puerperal sepsis is defined as infection of the genital tract that occurs during the time period between the onset of labor or rupture of membranes, and the 42nd postpartum day with any of the following two criteria:
a. Fever (temperature greater than 38.0o C or 100.4o C).
b. Pelvic pain
c. Abnormal vaginal discharge
d. Sub involution of the uterus (< 2 cm per day during the first 8 days).
Usually symptoms are seen after 24 hours and up to 10 days. Most common infection is that of the uterus and its surrounding areas, hence it is also known as postpartum metritis. 
Puerperal sepsis if diagnosed at the right time is treatable. Major complications can be prevented, but when it goes unrecognized for a long period of time, it may lead to maternal mortality and those salvaged are left with irreversible morbidity.[1]

Case Report

A 22 year old primipara was referred to our tertiary care center in view of puerperal sepsis. She gave history of delivery of a dead fetus at home 19 days ago, following which she had excessive vaginal bleeding and was taken to a private hospital in an unconscious state. Manual removal of the placenta for retained placenta was done there under general anesthesia. She was discharged on day 5. She developed fever of 1030 C on day 14 of manual removal of placenta and went to another private hospital from where she was referred to our hospital in view of retained placenta with puerperal sepsis. On examination she was conscious and oriented in time, place and person but pallor was present. She was febrile. Her temperature was 38 degree C, pulse rate was 110/ min and blood pressure was 110/70 mm of Hg. On abdominal examination uterus was 14-16 weeks in size, deviated to the right side and there was tenderness in bilateral iliac fossae. On speculum examination there was copious amount of purulent foul smelling discharge extruding through the cervix. On vaginal examination uterine size was 14-16 weeks, it was deviated to the right side, and with significant fullness in the pouch of Douglas. Her hemoglobin was 7 gm%, total leukocyte count was 29,400/cmm for which broad spectrum antibiotics were started and she was transfused with one unit of packed cells. DIC profile was within normal limits. Tests for malarial parasites, leptospirosis and dengue NSV1 antigen were negative. Ultrasound of the pelvis showed a bulky uterus with a hypoechoeic lesion within the uterus of size of 7 x 5 x 3 cm suggestive of retained placenta. There was no evidence of adherent placenta. A 9 x 8 x 6 cm sized hypoechoeic lesion with septae was seen in the pouch of Douglas having volume of 216 cc. Magnetic resonance imaging was suggestive of a retained placenta within the fundus in a puerperal uterus with a large recto-uterine pouch collection measuring approximately 4 x 6.7 x 9 cm. The collection appeared to be traversing superiorly till L 5 vertebra. Another collection of size 1.7 x 3.7 x 3.5 cm along the right posterosuperior aspect of the uterine fundus was noted. Decision to perform an exploratory laparotomy was taken with consent for obstetric hysterectomy if required. Intraoperatively frank pus of about 300 ml was found in the peritoneal cavity. Parietal peritoneum was adherent to the bowel. Uterus was about 14-16 weeks size with a rent in the right cornual region measuring about 2 x 2 cm in size. It had necrosed edges. Bilateral ovaries were normal and covered with a thick layer of pus. Decision to perform obstetric hysterectomy was taken in view of the ragged perforation in the cornual region that was friable and suturing would have been difficult. Following obstetric hysterectomy a peritoneal drain was inserted in the peritoneal cavity.


Figure 1. Pus in the peritoneal cavity              


Figure 2. Uterus with pus flakes and necrotic edge of the rent in its wall.

After surgery the cut section of the uterus showed retained placental tissue, with uterine perforation in right superior quadrant near the cornual region. It had a necrotic and friable edge. 


Figure 3. Uterine rent in right cornual region.                                                


Figure 4. Retained placental tissue in the uterine cavity

She was started on parenteral broad spectrum antibiotics- meropenem, metronidazole and gentamycin after surgery. Culture sensitivity report of the drained pus was awaited. Peritoneal drain drained pus for 4-5 days. Culture sensitivity report showed methicillin resistant staphylococcus aureus (MRSA) sensitive to linezolid for which oral linezolid was given in therapeutic doses for a period of 14 days. On postoperative day 5 wound check was done which showed minimal serous discharge and peritoneal drain was removed. Wound swab was sent for culture sensitivity, which did not grow any organisms on culture. Suture removal was done on day 19 of obstetric hysterectomy. Postoperative course was uneventful and she was discharged on day 20.

Discussion

Maternal sepsis (10.7%) is the third leading cause of maternal mortality after hemorrhage (27.1%) and hypertension (14%) as per statistical analysis by WHO in 2015.[2]
The most important associated risk factor for uterine infection is the route of delivery. A study conducted showed that most incidents of sepsis resulted following home vaginal deliveries.[3] Most common factor associated with puerperal sepsis is anemia.[1,4,5] Other predisposing factors are home delivery, unhygienic means of delivery, retained products of conception, septic abortion, repeated vaginal examinations and prolonged rupture of membranes. Puerperal sepsis most commonly includes uterine infections such as endometritis, endomyometritis and endoparametritis. 
A high index of suspicion for uterine perforation should be present whenever any patient who has undergone any intrauterine procedure, presents with symptoms of vascular or visceral injury 2-3 weeks following surgery.[6] Women with delayed diagnosis of uterine perforation may typically present with signs and symptoms of associated complication such as sepsis and anemia. The cause of complications should be further evaluated so as to confirm suspected uterine perforation. Though uterine perforation cannot be confirmed or excluded with any imaging studies, febrile anemic patients presenting with pelvic mass or tenderness in the pelvic region should have an ultrasound done as it is the test of choice for evaluation.[6]
Management strategy includes blood & blood product transfusion, antibiotic therapy, intravenous fluids & surgery wherever indicated. Uterus should be inspected for any perforation if abdominal exploration is performed. In absence of any visible defect previous uterine perforation cannot be excluded, since small defects might close spontaneously. Uterine perforation may weaken the uterine wall and raises concerns for an increased risk of uterine rupture during a subsequent pregnancy.[7-9] Group B streptococcus, E. coli and staphylococcus aureus especially MRSA are the most common causative agents of postoperative infections and puerperal sepsis.[1, 4] Importance of timely surgery and obstetric hysterectomy if indicated along with broad spectrum antibiotics is most effective in reducing maternal morbidity and preventing maternal mortality.[10]

References
  1. Madhudas C, Khurshid F, Sirichand P. Maternal morbidity and mortality associated with puerperal sepsis. JLUMHS. 2011;10(03):121-123.
  2. Say L, Chou D, Gemmill A, Tunçalp Ö, Moller AB, Daniels J,et al. Global causes of maternal death: a WHO systematic analysis. Lancet Global Health. 2014;2(6): e323-e333.
  3. Ahmed MI, Alsammani MA, Babiker RA. Puerperal sepsis in a rural hospital in Sudan. Mater Sociomed. 2013;25(1): 19-22.
  4. Bhanap PL, Sakhre A: A Clinical Study of Puerperal Sepsis. Global research analysis. 2013;2(9): 126-127.
  5. Qadri S, Sharma K, Siddiqui B,Ehs A. Microbial Profile in Females with Puerperal Sepsis: A Major Threat to Women s Health:Study at tertiary health care centre.  Int. J. Curr. Microbiol. App. Sci (2015) Special Issue-1: 248-255
  6. Leibner EC. Delayed presentation of uterine perforation. Ann Emerg Med. 1995 Nov;26(5):643-6.
  7. Sentilhes L, Sergent F, Berthier A, Catala L, Descamps P, Marpeau L. Uterine rupture following operative hysteroscopy [article in French]. Gynecol Obstet Fertil 2006;34:1064–70.
  8. Sentilhes L, Sergent F, Roman H, Verspyck E, Marpeau L.Late complications of operative hysteroscopy: predicting patients at risk of uterine rupture during subsequent pregnancy. Eur J Obstet Gynecol Reprod Biol. 2005;120(2):134-8
  9. Kerimis P, Zolti M, Sinwany G, Mashiach S, Carp H. Uterine rupture after hysteroscopic resection of uterine septum. Fertil Steril. 2002;77(3):618-20.
  10. Rivlin ME, Hunt JA. Surgical management of diffuse peritonitis complicating obstetric/gynecologic infections. Obstet Gynecol. 1986;67(5):652-6.
Citation

Shah S, Pardeshi S, Warke HS, Mayadeo NM. Obstetric Hysterectomy: A Life Saving Measure For Puerperal Sepsis. JPGO 2016. Volume 3 No. 11. Available from: http://www.jpgo.org/2016/11/obstetric-hysterectomy-life-saving.html

Krukenberg Tumor: Non Hodgkin Lymphoma

Author Information

Thosar MA*, Shetty A**, Gupta AS***.
(* Assistant Professor, ** Second year resident, ***  Professor. Department of Obstetrics and Gynecology, Seth GS Medical College & KEM hospital, Mumbai, India.)

Abstract

Primary non-Hodgkin lymphoma (NHL) rarely involves gynecological tract and is not considered in the differential diagnosis of bilateral ovarian masses. We present a case with bilateral ovarian masses who presented in emergency with intestinal perforation. On histopathology it was diagnosed as non-Hodgkin lymphoma.

Introduction

NHL involving gynecological tract is very rare and is generally not considered in the differential diagnosis of bilateral ovarian tumors. Though rare, ovarian involvement is most often found amongst genital tract. Up to 35% of primary ovarian tumors are bilateral and are similar to Krukenberg tumor.[1] Herewith we present a case of ovarian NHL that was diagnosed during laparotomy that was done for intestinal perforation. It supports the same Krukenberg concept of spread of NHL to ovary.

Case Report

A 23 year old unmarried girl was referred to the emergency of our tertiary care hospital with complaints of pain in abdomen and recurrent vomiting since three days. She had no significant past medical or surgical history except appendicectomy four years back. She had no menstrual complaints, and her menstrual cycles were regular.
On presentation she had no pallor, pulse rate was 128/ minute, blood pressure was in the normal range and at presentation patient was afebrile. There were no palpable cervical or axillary lymph nodes. Central venous pressure (CVP) was 8 cm of water. On abdominal examination, she had guarding, rigidity as well as rebound tenderness. Bowel sounds were absent.
On investigation hemoglobin was 11.4 gm%, total leucocyte count was 14500/ ml; on differential counts neutrophilia was present. Liver and renal function serological tests were normal. The patient had a computed tomography (CT) scan done before referral. It was suggestive of thickening and loss of gastric folds, collapsed small bowel with multiple air fluid levels as well as free air in the abdomen. Many enlarged celiac, perigastric, portal, aorto-caval and mesenteric lymph nodes were seen. There was copious free fluid in the abdomen suggestive of perforative peritonitis. Imaging also showed bilateral ovarian masses right greater than the left. They were solid, and homogeneous. Tumor markers for ovarian malignancy were sent.


Figure 1. Radiograph of the abdomen in standing position showing gas under the diaphragm and multiple air fluid levels.


Figure 2. CT scan pelvis. A is right ovarian mass and B is left ovarian mass.

An emergent exploratory laparotomy was performed for perforative peritonitis. On opening the abdomen there was a transverse intestinal perforation of 2.5 cm about 150 cm proximal to the ileocecal junction (ICJ). There were multiple impending perforation sites over the ileal segment of 10-12 cm. The base of the perforation was necrotic. Adjacent omentum was thickened. Celiac, perigastric and mesenteric lymph nodes were enlarged. Bowel segment was resected and a double barrel ileostomy was performed by the surgeons.


Figure 3. Resected segment of ileum. (A is the intestinal perforation. B, C and D are  impending perforation sites).


Figure 4. Bilateral ovarian masses. (A is deposit of tumor on the left ovary. B is right ovary replaced by tumor. C is the uterus).

There were bilateral ovarian masses, red fleshy, firm in consistency with an irregular surface. Right ovary was completely replaced with mass of 6×7 cm along with capsular breach. There was a deposit of similar mass of 3×3 cm on the surface of the left ovary. Right side salpingo-oophorectomy was done and wedge resection of the left ovary was performed. 
She developed massive intra abdominal hemorrhage on day 2 requiring re-exploration to control the bleeding. The tumor marker report that came subsequently showed elevated Lactate dehydrogenase (LDH) (1231 U/lit) levels. She developed disseminated intravascular coagulopathy and sepsis and succumbed on day 7 despite multiple transfusions and replacement of clotting factors. The relatives did not consent for an autopsy.
Final histopathology report showed the right ovary with features of non-Hodgkin lymphoma, wedge resection of other side ovary was normal. Bowel had ischemic necrosis and perforation, though no malignancy was identified.

Discussion

Lymphoma rarely occurs in the female genital tract. The incidence of primary lymphomas in the genital tract is reported to be 0.2 to 1.1% of the total lymphomas.[2,3] Secondary involvement is more common than primary and is estimated to be 7% of total lymphoma cases.[4] In only up to 35% of cases, primary ovarian lymphoma is bilateral while bilateral involvement was more often in secondary ovarian lymphomas.[1] Involvement of the uterus and ovaries by lymphoma is usually found at the time of autopsy performed after death due to advanced disease, suggesting metastasis of the disease to these organs similar to the Krukenberg concept of spread.[5] 
Though rare, the ovary is the most common organ in the gynecological tract to be involved by NHL. Malignant lymphoid precursors may present as a primary ovarian neoplasm or as a secondary dissemination from a distant disease. The median age of presentation in primary malignant ovarian lymphomas is 45 years while for secondary malignant ovarian lymphomas median age of presentation is 33 years.[1,5] Only Burkitt’s lymphoma presents at an early age (median age of 20 years).[6]
Presentation of primary or secondary ovarian lymphoma can be variable, with accidental discovery on radiological scanning, to overt genital bleeding or abdominal symptoms like swelling, distension and pain. Liang et al. suggested a strong correlation of gastrointestinal tract with ovarian lymphoma similar to our case report.[7] Christopoulos reported a case with NHL involving stomach and ovary and found a similar correlation. He also proposed to extend Krukenberg concept to include ovarian NHL.[8]
In 33 years single institute experience of genital NHL, Ahmad et al. found radiological imaging modalities were unable to differentiate between lymphoma from other malignancies.[9] Primary ovarian lymphoma was found to distort the internal structure of the ovary and often presented with a large ovarian mass. 
To classify NHL involving genital tract the World Health Organization classification is used which depends on incisional or excisional biopsies for diagnosis. Fine needle aspiration biopsies fail to demonstrate required cell-to-cell architecture for accurate diagnosis.[1] Diffuse large B-cell NHL is the most common type of NHL involving the female genital tract. Though primary gonadal disease evaluation reveals more advanced spread (FIGO stage 2–4), Vang et al. have reported better treatment outcome compared to secondary disease.[10] With systemic chemotherapy, up to 75% patients with localized disease were found to have better long-term survival and hence is recommended.[5] The survival in ovarian NHL and extra nodal NHL is similar with chemotherapy.[11] In cases of secondary involvement of the ovary, survival depends on the type of the lymphoma as well as other prognostic indices like age, tumor stage, the number of involved sites, performance status, and lactate dehydrogenase (LDH) levels. Due to the rarity of the condition no standard treatment protocols exist, still surgical cytoreduction followed by combination chemotherapy is used most commonly.
In our case due to multiple lymph node involvement in the celiac axis and small bowel ischemic ulcers, we believe ovarian mass was secondary to an undiagnosed primary in the stomach or the small bowel similar to a Krukenberg tumor. We postulate that metastatic tumor cells obliterated the mesenteric blood vessels leading to ischemic necrosis and small bowel perforations as the resected segment of the ileum was negative for malignancy. Unfortunately, we could not locate the primary as relatives did not consent for a pathological autopsy. Had the primary been diagnosed it would have lent support to the Krukenberg concept of spread to include ovarian NHL.[8] With this case report we want to emphasize that while considering differential diagnosis of bilateral ovarian masses one should be vigilant, and even rare causes like NHL should be considered. To achieve good outcomes multimodality treatment should be planned for these patients.

References
  1. Zhao XY, Hong XN, Cao JN, Leaw SJ, Guo Y, Li ZT, et al. Clinical features and treatment outcomes of 14 cases of primary ovarian non-Hodgkin’s lymphoma: a single-center experience. Med Oncol. 2011;28(4):1559–64.
  2. Freeman C, Berg JW, Cutler SJ. Occurrence and prognosis of extranodal lymphomas. Cancer. 1972;29(1):252–60. 
  3. Chorlton I, Norris HJ, King FM. Malignant reticuloendothelial disease involving the ovary as a primary manifestation: a series of 19 lymphomas and 1 granulocytic sarcoma. Cancer. 1974;34(2):397–407. 
  4. Woodruff JD, Noli Castillord, Novak ER. Lymphoma of the ovary. A study of 35 cases from the Ovarian Tumor Registry of the American Gynecological Society. Am J Obstet Gynecol. 1963;85:912–8. 
  5. Monterroso V, Jaffe ES, Merino MJ, Medeiros LJ. Malignant lymphomas involving the ovary. A clinicopathologic analysis of 39 cases. Am J Surg Pathol. 1993;17(2):154–70. 
  6. Lagoo AS, Robboy SJ. Lymphoma of the Female Genital Tract: Current Status: Int J Gynecol Pathol. 2006;25(1):1–21. 
  7. Liang R, Chiu E, Loke SL. Non-Hodgkin’s lymphomas involving the female genital tract. Hematol Oncol. 1990;8(5):295–9. 
  8. Christopoulos C, Skopeliti M, Rotas E, Mavridis A, Savva S, Papavassiliou E. Non-Hodgkin lymphoma involving stomach and ovary: extending the Krukenberg tumor concept. Ann Hematol. 2010;89(1):101–2. 
  9. Ahmad AK, Hui P, Litkouhi B, Azodi M, Rutherford T, McCarthy S, et al. Institutional review of primary non-hodgkin lymphoma of the female genital tract: a 33-year experience. Int J Gynecol Cancer. 2014;24(7):1250–5.
  10. Vang R, Medeiros LJ, Warnke RA, Higgins JP, Deavers MT. Ovarian non-Hodgkin’s lymphoma: a clinicopathologic study of eight primary cases. Mod Pathol. 2001;14(11):1093–9. 
  11. Dimopoulos MA, Daliani D, Pugh W, Gershenson D, Cabanillas F, Sarris AH. Primary ovarian non-Hodgkin’s lymphoma: outcome after treatment with combination chemotherapy. Gynecol Oncol. 1997;64(3):446–50.
Citation

Thosar MA, Shetty A, Gupta AS. Krukenberg tumor: Non Hodgkin Lymphoma. JPGO 2016. Volume 3 No. 11. Available from: http://www.jpgo.org/2016/11/krukenberg-tumor-non-hodgkin-lymphoma.html

Ruptured Granulosa Cell tumor Of The Ovary

Author Information

Satia MN*, Mali K**, Ganapathi T***.
(* Professor, ** Assistant Professor, *** Third Year Resident. Department of Obstetrics and Gynecology, Seth G S Medical College & KEM Hospital, Mumbai, India.)

Abstract

Acute abdomen with hemoperitoneum in childbearing age group is usually due to obstetrical complications but its extremely rare for a woman with gynecological conditions. We report a rare case of ruptured granulosa tumor of the ovary. She presented with acute abdomen due to hemoperitoneum. Ruptured ovarian tumor should be one of the differential diagnosis as ovarian tumors are known to occur at any age and clinicians should remain alert and aware of such a presentation. Anticipation of these problems and availability of facilities for frozen section would be of utmost importance as it would avoid incomplete surgeries. 

Introduction

Granulosa cell tumors account for approximately 4.3% of all ovarian cancers.[1] Granulosa cell tumors are functional ovarian tumors and hence are associated with endocrine manifestations. Approximately three fourth of the tumors are associated with hyperestrogenism but some may be virilizing. These tumors are rarely bilateral (< 5 %) and have low malignant potential (3-14 %) with high recurrence rate.[2] Symptoms depend on degree of hormonal activity and age of the patient. Clinicians should be aware of occurrence of granulosa cell tumors at any age and their propensity to rupture as in our patient who presented with acute abdomen and hemoperitoneum. Acute abdomen with hemoperitoneum is a common obstetric emergency, but a very rare entity in gynecologic conditions. It is most commonly seen in non accident related hospital admissions.[2] Gynecological conditions causing acute abdominal pain are ovarian torsion and ruptured corpus luteum hematoma in the child bearing age group. Our patient presented with hemoperitoneum and history of amenorrhea mimicking ruptured ectopic pregnancy or a ruptured corpus luteum cyst.
Case Report 

A 35 year old multiparous woman came to emergency room with severe abdominal pain and  giddiness. On examination her general condition was fair, she was pale with clinical hemoglobin of 6 gm%, pulse of 110/minute and blood pressure of 90/60 mm of Hg. Abdominal examination was suggestive of signs of acute abdomen with guarding, tenderness, rigidity. She was markedly obese. During pelvic examination there was tenderness in both the fornices with bogginess, more in the right fornix. Her urine pregnancy test (UPT) was negative, but colpopuncture was positive. Sonography examination revealed 500 ml of hemoperitoneum and hence the clinical impression of ruptured corpus luteum cyst was made. Decision of immediate exploratory laparotomy was taken. Intra operative findings revealed 500 cc of blood stained fluid along with clots weighing 200 gms. An actively bleeding large mass of 10x8 cms arising from the right ovary was observed. Uterus along with both fallopian tubes were normal and her left ovary was normal.


Figure 1. Ruptured 10 x 8 cm size ovarian cyst.


Figure 2. Ovarian cyst surface with normal uterus (U).

Right salpingo-oophorectomy was performed and the specimen was sent for histopathological examination. Histopathology report diagnosed a well differentiated adult type granulosa cell tumor of the right ovary.


Figure 3. Intact capsule (black arrow) seen in the histopathology slide indicating benign nature of the lesion. 


Figure 4.  Adult type granulosa cells (black arrow) with few areas of hemorrhage (yellow arrow) seen. 

Post operative period was uneventful and she was referred to the oncology center for further evaluation where the histopathology slides were reviewed. Immunohistochemistry was positive for MIC-2 (gene product that is a cell surface antigen), AE/AE-3 (screening marker antibody used for epithelial differentiation) and inhibin. Histopathology reported it to be a  sex cord stromal; granulosa cell tumor. She was advised to follow up at 6 monthly intervals.

Discussion

Granulosa stromal cell tumors are a type of sex cord stromal cell tumors (SCST). These tumors constitute 4.3% of all ovarian neoplasms.[1] They are differentiated histologically by WHO.[3] These tumors can occur in any age group that is 1 year to 90 years of age but they are most common in 50-55 years of age.[4] The chances of hemoperitoneum with granulosa cell tumor has been reported in 5 to 20% of cases.[3] Our patient presented with amenorrhea and acute onset of pain in abdomen. It is reported that tumor rupture occurred in 17.6% of patients prior to surgical intervention. However, in diagnosed cases of adult granulosa cell tumor patients’ have not presented with acute abdomen.[5]
Other causes of hemoperitoneum may include patients on anticoagulant therapy or patients having platelet function defects or rarely retrograde bleeding due to cervical stenosis.[6] Diagnosis is confirmed on laparotomy as in the present case where hemoperitoneum and active bleeding ovarian mass was noticed. Anticipation of these problems and timely actions like frozen sections of the suspicious lesions will help in preventing incomplete surgeries requiring repeat procedures. Clinicians should be alert to the possibility of propensity of granulosa cell tumors rupturing and causing hemoperitoneum. Twenty  percent of emergency surgical services visits are by the elderly out of which 3-4 % are for acute abdominal pain. Mortality is 6-8 times high in older patients with abdominal pain as compared to younger patients. About one-half to two-thirds of these patients need to be admitted for medical line of treatment, while one-third require surgical intervention.[7]. Management of granulosa tumors depends not only on the stage of the tumor but also the age of the patient. In adolescent or child bearing age group patients presenting with early stage disease, unilateral salpingo-oophorectomy and appropriate surgical staging is preferred in an attempt to preserve fertility. In fifth and sixth decade of life surgery consists of a total abdominal hysterectomy along with removal of both tubes and the ovaries and surgical staging which is the most important prognostic factor. The overall survival with stage I and stage II disease is 99%. Patients with large tumor size (≥ 10-15 cm), stage IC, poorly differentiated tumor, high mitotic index or tumor rupture are said to be high risk and  adjuvant chemotherapy should be advised in view of increased risk of relapse.[8] Patients with low risk stage I tumor should be kept on observation.[9] In patients with recurrence or stage III- IV granulosa cell tumors postoperative chemotherapy is recommended but the survival benefit is still not known due to paucity of results and lack of randomized trials, so no treatment has been standardized.[10] The lack of specific markers in the identification of granulosa cell tumors has lead to development of a panel of antibodies to identify inhibin, calretinin, epithelial membrane antigen, and low molecular weight keratin which is expressed on tumor cells, through immunohistochemistry. Immunohistochemistry not only helps in diagnosis but also is useful in administration of targeted chemotherapy against these tumors.[11]

Conclusion

Rupture of ovarian tumors are one of the rare causes of acute abdomen and in women presenting with acute abdomen with non obstetric causes, a high index of suspicion for malignancy should always be maintained. With good clinical suspicion and proper management patients can be given good quality care.

References
  1. Ayhan A, Salman MC, Velipasaoglu M, Sakinci M, Yuce K. Prognostic factors in adult granulosa cell tumors of the ovary: a retrospective analysis of 80 cases. J Gynecol Oncol. 2009;20(3):158–63.
  2. Hastings RS, Powers RD. Abdominal pain in the ED: a 35 year retrospective. Am J Emerg Med. 2011;29(7):711-6.
  3. Chada-Ajwani S. Ovarian Granulosa cell tumors. Histopathology, immunopathology and prognosis. Druk: Krips Repro Meppel;1987:11-32.
  4. Vani BR, Geethamala K, Geetha RL, and Srinivasa MV. Granulosa cell tumor of ovary: A clinicopathological study of four cases with brief review of literature. J Midlife Health. 2014; 5(3): 135–138.
  5. Taira Y, Hirakawa M, Nagayama C, Ikemiyagi K, Touma T, Tokashiki M. Successful treatment of adult-type granulosa cell tumor of the ovary by palliative radiotherapy.J Obstet Gynaecol Res. 2012;38(2):461-5. 
  6. Habek D, Habek JC, Barbir A, Barbir M. Ruptured ovarian granulosa cell tumors as a cause of the acute abdomen. Arch Gynecol Obstet. 2003;267(3):175-6.
  7. Sanson TG, O'Keefe KP. Evaluation of abdominal pain in the elderly. Emerg Med Clin North Am. 1996;14(3):615-27.
  8. Kottarathil VD, Antony MA, Nair IR, Pavithran K. Recent advances in granulosa cell tumor ovary: a review. Indian J Surg Oncol. 2013;4(1):37-47.
  9. Khosla D, Dimri K, Pandey AK, Mahajan R, Trehan R. Ovarian granulosa cell tumor: Clinical features, treatment, outcome, and prognostic factors. N Am J Med Sci. 2014; 6(3): 133–138.
  10. Segal R, DePetrillo AD, Thomas G: Clinical review of adult granulosa cell tumors of the ovary. Gynecol Oncol 1995; 56(3): 338-44
  11. Nofech-Mozes S, Ismiil N, Dubé V, Saad RS, Khalifa MA, Moshkin O, et al. Immunohistochemical characterization of primary and recurrent adult granulosa cell tumors. Int J Gynecol Pathol. 2012;31(1):80-90.
Citation

Satia MN, Mali K, Ganapathi T. Ruptured Granulosa Cell tumor of Ovary. JPGO 2016. Volume 3 No. 11. Available from: http://www.jpgo.org/2016/11/ruptured-granulosa-cell-tumor-of-ovary.html

A Bladder Stone Obstructing Labor

Author information

Salunkhe RY
(Practicing obstetrician and gynecologist, Chalisgaon, Maharashtra, India)

Abstract

Bladder stone is rare in pregnancy. Previously totally asymptomatic bladder stone causing obstructed labor is very rare. I present a case of obstructed labor due to a big bladder stone. Cesarean section was performed. The stone was not removed Intraoperatively.

Case Report 

A 24 year old unregistered, full term primigravida, came with complaint of minimal abdominal pain. On per abdominal examination the uterus was full term and irritable, the presentation was cephalic, and the fetal heart sounds were normal. On per vaginal examination a 10 x 10 cm hard, immobile, non tender mass was found, arising from the right anterolateral pelvic wall. The cervix was 2 cm dilated, 60% effaced and was pushed to the left posterolateral side. Fetal head was high up and the mass was preventing the head from entering the pelvis. The membranes were intact. Ultrasonography (USG) showed 10x8 cm sized bladder stone. CBC and serum creatinine were within normal limit. HIV, and HbsAg were negative. The patient was advised cesarean section. She went home and came on the next day with increased pain in abdomen. Her examination showed the cervix was 3 cm dilated and thick. There was minimal caput, station was high, and membranes were absent. Emergency cesarean section was done with a urosurgeon stand by. Healthy male baby was delivered. Diagnosis of bladder stone was confirmed intraoperatively. The stone was not removed. It was hard, mobile, 12x 12 cm in size inside the bladder (figures 1 and 2). Postoperative course was uneventful.


Figure 1. Vesical calculus lifted out of the pelvis.


Figure 2 Vesical stone in the pelvis.

Discussion

Previously totally asymptomatic bladder stone obstructing labor has been a very rarely reported entity. Bladder stones are mostly formed of calcium phosphate. Congenital and acquired diverticuli of the urinary bladder may be a reason for stasis and development of calculi. Contrary to past teaching, a low-calcium diet promotes stone formation. There is no evidence to state that pregnancy increases the risk of stone formation. Prevention can be done with hydration and diet low in sodium and protein. Dysuria, frequency, hesitancy, and terminal hematuria are symptoms of vesicle calculus. High index of suspicion is needed for diagnosis of a bladder stone. In general during pregnancy urinary obstruction, recurrent severe infection, intractable suprapubic pain and heavy bleeding are indications for stone removal. The early diagnosis and appropriate surgical management of a large vesical calculus prevent complications like recurrent urinary tract infections and obstructed labor.[1] The differential diagnosis of hard mass felt on vaginal examination and obstructing labor is broad ligament fibroid, carcinoma cervix or carcinoma of rectum invading the parametrium, a giant fecolith, ovarian tumor, hydatid cyst, and a lesion from lateral bony pelvic wall. In my case diagnosis was confirmed by ultrasonography, but radiography can also be a good diagnostic tool. Otherwise in other suspicious cases CT scan (not recommended in pregnancy) and MRI can also be helpful according to availability. Rarely PET scan can be done. If the patient is first seen in labor it may be possible to displace the calculus above the presenting part and allow spontaneous vaginal delivery.[2] But if this is difficult, cesarean section should be performed. In my case displacement of the stone above the presenting part was not possible so I decided to do cesarean section. I kept a urosurgeon as standby. Intraoperatively there was no difficulty in delivery of the baby. The stone was not removed simultaneously as there was high chance of fistula formation as there is marked congestion with hypertrophy of the muscles and elastic tissue of the bladder wall.[3] Particularly in a nulligravida woman in late pregnancy in whom the presenting part often engages before labor, the entire base of the bladder is pushed forward and upward, converting the normal convex surface into a concavity. As a result, difficulties in diagnostic and therapeutic procedures are greatly increased. In addition the pressure of the presenting part impairs the drainage of the blood and lymph from the bladder base, often rendering the area edematous, easily traumatized, and probably more susceptible to infection.[4] The stone can be removed less invasively by cystolithotripsy later on. Unfortunately patient did not come for follow up. So further management of the bladder stone could not be done.

Conclusion

Vesicle calculi, if symptomatic during pregnancy should be treated to prevent unnecessary cesarean section due to obstructed labor. If it presents during labor and stone is mobile, manipulation with hand and pushing it above the presenting part can prevent obstructed labor and allow normal delivery. Cesarean should be done in case of obstructed labor. Stone removal during cesarean should be avoided as it is associated with high rate of infection and fistula formation.

Acknowledgement

I would like to thank Dr Sanjay Chavhan, consultant obstretrician and gynecologist, Dr Satyajit Purnapatre, practicing urosurgeon, and Dr Sweta Ajay Wable, anesthetist to manage this patient.

References
  1. Pricilla RA, David KV, Venkatesan S, Benjamin SJ. Early Diagnosis of a Large Vesical Calculus Complicating Pregnancy. J Family Med Prim Care. 2013; 2(1): 88–89.
  2. van Coeverden de Groot HA. Obstructed labour caused by a vesical calculus: case report. SJOG.1964;38:46–47.
  3. Penning SR, Cohen B, Tewari D, Curran M, Weber P. Pregnancy complicated by vesical calculus and vesicocutaneous fistula. Am J Obstet Gynecol. 1997;176(3):728–9.
  4. Cunningham FG. Maternal Physiology. In Cunningham FG, Leveno KJ, Bloom SL, Hauth JC, Rouse DJ, Spong CY, editors. Williams Obstetrics. 23rd edition. New York: McGraw-Hill Education 2010; pp 125.
Citation

Salunkhe RY. A Bladder Stone Obstructing Labor. JPGO 2016. Volume 3 No. 11. Available from: http://www.jpgo.org/2016/11/a-bladder-stone-obstructing-labor.html

A Rare Case Of Hystero-Ileal Fistula

Author Information

Satia MN*, Ganapathi T**, Mali K***
(* Professor, ** Third Year Resident, *** Assistant Professor. Department of Obstetrics and Gynecology, Seth G S Medical College & KEM Hospital, Mumbai, India.)

Abstract

Abortion is an important health concern in women but increasingly governed by the patriarchal interest which curbs the freedom of women who seek abortion as a right.  Illegal unsafe abortion remains an alternative for the poor, widow and the unmarried. These unsafe abortions pose a great danger to the life of the patient. Our present case is an illustration of complication of abortion resulting in uterine perforation, bowel injury and  hystero-ileal fistula. In our case per rectal examination was normal, there were no signs of injury to the perineum yet the patient was passing feces through the vagina.

Introduction 

 As per WHO one woman in every 8 minute dies in developing nations due to morbidity arising from unsafe or  illegal abortion, making it one of the most important cause of maternal mortality.[1] Five million women suffer injury as a result of morbidity arising from unsafe or  illegal abortion. The risk of bleeding requiring transfusion following an abortion is less than 1 in 1000,  risk of uterine perforation is 1-4 in 1000 and risk of cervical trauma to the external os is 1 in 100.[2] During suction and evacuation the surgeon is operating blindly, by sense of feel. If manipulation of the surgical instrument is done carelessly or forcibly, uterus and even her bladder or bowel can be perforated. Failure to diagnose uterine perforation may lead to major complications & these patients usually present to the emergency surgical unit with increased abdominal pain, bleeding and fever. The presence of pain during rectal examination and bleeding through rectum makes the diagnosis of post abortion bowel injury almost certain. In post abortion patients with abdominal pain beyond the pelvic area, suspect perforation and evaluation should include upright radiographs, pelvic sonography & computed tomography (CT) scan. If suspicion is high, diagnostic laparoscopy should be done.

Case Report

A 20 year old unmarried woman came to the emergency surgical unit with complaints of passage of feces through the vagina since 10 days. She had 2 months of amenorrhea with history of fever associated with giddiness for which she was given intravenous fluids along with multivitamin injection by a general practitioner following which she was referred to a higher center for medical termination of pregnancy on request. When she came to our hospital she gave history of a spontaneous abortion of 2 months gestation following an accidental fall 20-25 days ago. She denied history of medical termination of pregnancy or any instrumentation done for the same. She had history of fever associated with giddiness. On admission, she was febrile, had tachycardia with a pulse of 110/ minute and blood pressure of 100/70 mm of Hg. On abdominal examination, there was mild distension, tenderness in the hypogastrium and feeble peristalsis. On speculum examination, cervix could not be visualized due to semisolid fecal matter persistently filling the vagina. There was no bleeding. On vaginal examination, the uterus was soft, about 10 weeks in size, os was admitting tip of the finger, feces was flowing out from the os. There was no palpable rent in the vagina. On rectal examination, rectal mucosa was intact and there was no evidence of fourth degree perineal tear. A clinical impression of utero-intestinal fistula was made and sonography was advised which revealed a normal uterus and adnexa without any evidence of free fluid in the peritoneal cavity. In view of the inconclusive findings of sonography, CT scan of the abdomen and pelvis with anal contrast was advised. The CT scan was suggestive of a communication between the ileum and the uterus along with dilatation of the proximal bowel loops, with bowel adhered to the fundus of the uterus and no plane of cleavage between them. The contrast was seen flowing through the bowel into the uterus and filling the vagina. An emergency exploratory laparotomy was performed jointly by surgeons and gynecologists. On entering the peritoneal cavity small bowel loops were found to be dilated and adhered to the fundus of the uterus. There was minimal free fluid in the peritoneal cavity and no evidence of peritonitis. There was no fecal material in the peritoneal cavity. Adhesiolysis was done to release the bowel loops. On examination, 3 perforations of about 1.5 cm each, 7-10 cms apart were seen in the ileum. Loops of ileum was adherent to the uterus and the perforations were communicating with the uterine cavity via a rent of 3x3 cm in the uterine fundus.
Figure 1. Red arrow showing uterine rent of 3x3 cm and black arrow showing ileal perforation.
Figure 2. Black arrow showing the uterine rent of 3x3 cm  at the level of fundus of the uterus.
She probably concealed the history of instrumentation for abortion, but intra operative findings were more in favor of a probable unsafe abortion by an untrained person leading to bowel injury and fistula formation. Bilateral fallopian tubes and ovaries were healthy. The uterine cavity was flushed with normal saline and all fecal material was suctioned out. No products of conception were found. The uterine rent was closed in a simple interrupted manner using polyglactin No. 1 sutures. Hemostasis was checked at the suture site. Resection anastomosis of the ileum  with double barrel ileostomy was done. Postoperatively she was stable. She was put on higher antibiotics. She was started on oral fluids after 72 hours. She was discharged with the temporary stoma and was asked to follow up for stoma removal after 6-8weeks.

Discussion

Around 10 women out of 100 undergoing medical termination of pregnancy develop immediate complications postoperatively. One-fifth (2%) of these complications are  major  complications.[3] Major risks and complications of abortion are described extensively in the medical literature. Every year 21.6 million unsafe abortions are estimated to take place, mainly in the developing countries resulting in the death of approximately 47000 women.[4] The MTP act was enacted mainly to reduce illegal unsafe abortions and to reduce the complications leading to morbidity and mortality.[5] Immediate complications of unsafe abortion like hemorrhage, sepsis, injury to the uterus, bladder or bowel, shock and death  can occur.[6]  MTP done by unskilled and untrained persons without  knowledge and aseptic precautions lead to complications right from visceral injuries, genital fistulas, septicaemia to death.[7] If major blood vessel is injured during perforation patients may present with hypovolemic shock. Bowel injury can occur along with uterine perforation. If initially unrecognized, patients present with nausea, and vomiting, pain in abdomen with distension & sometimes fever. Monitoring vital parameters is essential for patients with post abortion complications. Increasing fever could be a sign of progressive infection, low blood pressure along with tachycardia are the signs of severe hemorrhage or septic shock.[8] Patients with uterine perforation that are not diagnosed during the procedure usually present to the emergency services with increased abdominal pain, bleeding and fever. Minimal suprapubic tenderness is common in the post abortion period. Severe tenderness in the abdomen strongly indicates instrumental injury complications like perforation, bowel injury or bladder injury. Silent abdomen is a sign of peritonitis. Vaginal examination will help in assessing the amount of bleeding, source of bleeding whether uterine, cervical or vaginal. A digital rectal examination is recommended in patients in whom the bowel may have been injured. Tenderness on rectal examination and bleeding through rectum suggests possible bowel injury. In such patients thorough evaluation by sonography and contrast enhanced CT scans  help in reaching an accurate diagnosis and planning the surgery. Depending on the site of perforation, number of perforations and the vascularity of the bowel segment, resection anastomosis with temporary stoma is done. In the postoperative period bleeding through vagina is checked and patient is called for stoma removal after 3 months. Stoma care is taught to the patient and relatives.

Conclusion

Unsafe abortions pose a life threatening situation & a threat to social development. It is a major public health problem especially in the developing world. Early diagnosis and intervention might provide better outcome. Therefore prompt referral to tertiary care centers as & when required and safe abortion services by trained & efficient personnel  will go a long way in decreasing the complications. 

References 
  1. Gole G, Santpur U, Kaul R. Severe Intraabdominal Trauma In Illegal Abortion: A Case Report. The Internet Journal of Gynecology and Obstetrics. 2012;16(3). 
  2. Summary of recommendations. Care of women requesting induced abortion. Evidence-based clinical guidelines number 7. Royal College of Obstetricians and Gynaecologists. London: RCOG Press. Nov 2011;pg.9
  3. Joint study of the Royal College of General Practitioners and the Royal College of Obstetricians and Gynaecologists. Induced abortion operations and their early sequelae. J R Coll Gen Pract. 1985;35(273):175–180.
  4. Shah I, Ahman E. Unsafe abortion in 2008: global and regional levels and trends. Reprod Health Matters. 2010;18(36):90-101. 
  5. Patra AP, Rayamane AP, Shaha KK, Kundargi PA, Mohanty MK, Das S. Practice of Illegal Abortion in India – With Reference to a Case Report. Ind J Forensic Med Pathol 2013;6(4):29-35.
  6. Haddad LB, Nour NM. Unsafe abortion: unnecessary maternal mortality. Rev Obstet Gynecol. 2009;2(2):122-6.
  7. Nayak PK, Mitra S, Padma A, Agrawal S. Late presentation of unsafe abortion after 5 years of procedure.Case Rep Obstet Gynecol.2014;2014:456017.
  8. Malhotra N, Sirsam S, Inamdar SA. A Case of Illegal Septic Abortion Leading to Maternal Mortality. JSAFOG. 2010;2(2):149–51.
Citation

Satia MN, Ganapathi T, Mali K. A Rare Case Of Hystero-Ileal Fistula. JPGO 2016. Volume 3 No. 11. Available from: http://www.jpgo.org/2016/11/a-rare-case-of-hystero-ileal-fistula.html

Pregnancy With Addison’s Disease

Author Information

Patil S*, Dhokia T**, Chauhan AR***.
(* Ex Fourth Year Resident, ** Third Year Resident, *** Professor, Department of Obstetrics and Gynecology, Seth GS Medical College and KEM. Hospital, Mumbai, India.)

Abstract

Addison’s disease or primary adrenal insufficiency is one of the rare endocrine disorders in which the adrenal glands are unable to produce enough steroid hormones.  Addison’s disease in pregnancy is even more rare. Addisonian crisis can occur following stressful conditions such as labor, surgery, infections and puerperium. We present a case of Addison’s disease with pregnancy and its successful outcome by multidisciplinary approach.

Introduction

Primary adrenocortical insufficiency or Addison’s disease is an endocrine disorder; majority of cases are due to autoimmune adrenalitis.[1] Untreated primary adrenocortical insufficiency results in subfertility.[2] Addisonian crisis is a rare and life-threatening event in pregnant women, and occurs due to stressful conditions such as hyperemesis gravidarum, labor, puerperium, infection or surgical interventions.[3] Patients with this disease in pregnancy can have symptoms such as weakness, fatigability, weight loss, myalgia, fever, anorexia, nausea, vomiting and mental irritability. While complications of Addison’s disease in pregnancy include postural hypotension, hyponatremia, hyperkalemia, pigmentary changes, these are seen less frequently due to the availability of steroid therapy. Addison’s disease was associated with a high maternal mortality rate before steroid treatment was available; the mortality has decreased from 45% in 1930, to 7% during 1948-1955, to 0.7% by the year 2000. Serious fetal complications such as intrauterine death are also reported.[4]

Case Report

Mrs. NS, 25 years old primigravida with 33 weeks of gestation, a diagnosed case of Addison’s disease was referred to us for tertiary care. Patient presented with complaints of headache and blurring of vision since one week and pain in abdomen since last 2 days. Patient had history of irregular use of steroids. In 2010, she was prescribed topical steroids for a skin lesion which she used for 6 months. However the treatment did not help and her skin lesions increased, for which she was investigated; serum cortisol level was less than 0.2 µg/ dl (normal range 5 - 25 µg/ dl). Subsequent testing of serum cortisol was done in 2012, 2013, 2015; all levels reported were less than 0.2 µ/ dl. Thus a diagnosis of Addison’s disease was made. Thereafter patient was put on tablet prednisolone 5 mg daily. In 2013, during a course of treatment with steroids, patient developed cushingoid features such as striae all over the body, humped back, generalized anasarca and weight gain. At this time, she stopped treatment on her own. In 2015, she developed Bell’s palsy and was diagnosed to have raised intracranial tension and papilledema. Patient was managed conservatively and restarted on tablet prednisolone 5 mg. While on treatment with prednisolone she conceived in January 2016 and again discontinued her steroidal treatment. 
On presentation to us, she had raised blood pressure (150/ 100 mm Hg) and urine albumin 3 + by dipstick method. Patient was started on tablet labetelol 200 mg twice daily and tablet nifedepine 10 mg three times a day. Ophthalmology and endocrinology references were taken. There was no abnormality found on fundoscopy. Endocrinologist advised fresh 8 am serum cortisol, which was 17.21 µg/ dl, hence steroids were not required. Other investigations and fetal surveillance were normal. Meanwhile patient complained of persistent headache and blurring of vision; in view of associated persistent hypertension and albuminuria despite about a week of treatment, decision to terminate pregnancy in view of preeclampsia was taken.  Induction of labor was done at 34 weeks with PGE2 gel. She delivered vaginally a healthy baby of 2.274 kg. Peri delivery steroids were not given. She did not develop adrenal crisis during intrapartum and postpartum period. Mother and baby were discharged after a week of observation.

Discussion

In 1950, Brent studied 39 cases of Addison’s disease with pregnancy. He stated that symptoms of Addison’s disease may mask those of pregnancy and vice versa. Patients require constant observation, careful supervision, need for hospitalization especially in first trimester of pregnancy. Spontaneous delivery at term has best results.[5] Trainer PJ in 2002 stated that adrenal insufficiency can be difficult to diagnose during pregnancy, however once the diagnosis is made there is usually a successful outcome with careful monitoring, steroidal dose adjustment and parenteral steroidal cover during labor.[6] Quinkler et al in 2012 observed that pregnant women with Addison’s disease mostly do not need to adjust the dose of glucocorticoids in first and second trimester but dose needs to be increased in third trimester due to increased free cortisol during this period. The mineralo-corticoid dose also needs to increase during third trimester due to anti mineralocorticoid action of progesterone.[7]  
Early morning plasma cortisol levels < 3 µ/ dL confirm adrenal insufficiency while a cortisol > 19 µ/dL excludes the diagnosis in stable pregnant patients. The patient in this case report had cortisol of 17.21 µ/ dL hence adrenal insufficiency cannot be excluded in spite of normal serum cortisol levels.[8, 9] Plasma cortisol level may fall in the normal range during pregnancy due to increase in cortisol binding globulin concentration in second and third trimester as probably happened in our patient.[10] 
The stress of pain, physical exertion, hemorrhage and surgical intervention in labor, the physiological diuresis and withdrawal of adrenal hormones from the fetal adrenals in puerperium can lead to an Addisonian crisis. To prevent this, parenteral cortisone should be administered during labor.  Thus, careful monitoring and treatment with steroids in patients with Addison’s disease and pregnancy especially during first trimester, labor and in first week of puerperium results in successful outcome of pregnancy.

Conclusion

Pregnancy with Addison’s disease may present with normal serum cortisol levels; however these cases should be carefully observed for Addison’s crisis during labor and other stress conditions.

References
  1. Erichsen MM, Husebye ES, Michelsen TM, Dahl AA, Løvås K. Sexuality and fertility in women with Addison's disease. J Clin Endocrinol Metab. 2010;95(9):4354-60.
  2. Oelkers W. Adrenal insufficiency. N Engl J Med 1996;335(16):1206-1212.  
  3. Albert E, Dalaker K, Jorde R, Berge LN. Addison’s disease and pregnancy. Acta Obstet Gynecol Scand. 1989;68(2):185-87.
  4. Ambrosi B, Barbetta L, Morricone L. Diagnosis and management of Addison’s disease during pregnancy. J Endocrinol Invest. 2003;26(7):698-702.
  5. Brent F. Addison’s disease and pregnancy. Am J Surg. 1950;79(5):645-52.
  6. Trainer PJ. Corticosteroids and Pregnancy. Semin Reprod Med. 2000;20(4):375-80.
  7. Quinkler M, Hahner S. What is the best long-term management strategy for patients with primary adrenal insufficiency? Clin Endocrinol (Oxf). 2012;76(1):21–25
  8. Mckenna DS, Wittber GM, Nagaraja HN, Samuels P. The effects of repeat doses of antenatal corticosteroids on maternal adrenal function. Am J Obstet Gynecol. 2000; 183(3):669-73. 
  9. Grinspoon SK, Biller BM.  Clinical review 62: Laboratory assessment of adrenal insufficiency. J Clin Endocrinol Metab. 1994;79(4):923-31.
  10. Nolten WE, Lindheimer MD, Oparil S, Ehrlich EN. Desoxycorticosterone in normal pregnancy. I. Sequential studies of the secretory patterns of desoxycorticosterone, aldosterone and cortisol. Am J Obstet Gynecol 1978;132(4):414-20.
Citation

Patil S, Dhokia T,  Chauhan AR. Pregnancy with Addison’s Disease. JPGO 2016. Volume 3 No. 10. Available from: http://www.jpgo.org/2016/11/pregnancy-with-addisons-disease.html