Volume 4 Issue 8, August 2017

Editorial
Gupta AS

Sepsis Due To Uterine Scar Necrosis
Dalvi P, Chaudhary HK.

Peripheral Candidial Funisitis
Alfiya Bano, Parulekar SV.

A Rare Case On Ovotesticular Sex Disorder
Dhanawat J, Pai K, Dharmadhikari M, Samant PY.

Translocated IUCD In Peritoneal Cavity
Agrawal A, Samant PY, Swathi HV.

Ventricular Arrhythmia In Early Pregnancy
Nasare PS, Madhva Prasad S, Gupta AS.

Scar Endometriosis: a Rare Case
Shaikh A, Mayadeo MN, Warke HS.

Conservative Management Of Pyoperitoneum
Shetty A, Prasad M, Gupta AS.

Pregnancy With Takayasu Arteritis And Heart Disease

Venkateswaran S, Chauhan AR.

Laparoscopic Approach In Rare Case Of Isolated Fallopian Tube Torsion

Bhate A, Bhate M , Chitnis S.

Editorial

Gupta AS

In this modern era of antibiotics, knowledge and implementation of good clinical practices for asepsis during pregnancy and labor, we still see a significant number of post abortal or post partum women with major degrees of sepsis.

Sepsis remains a significant corner of the triad of maternal mortality. The tip of the this iceberg hides a large number of women with serious and varying degrees of morbidity caused by such sepsis. Many times after treatment women are left behind with lifelong serious sequalae like secondary infertility, ectopic pregnancies, chronic pelvic pain, subacute or chronic intestinal obstruction and others.

Puerperal or post abortal sepsis usually remains contained within the pelvic cavity. However, when the organism causing the infection is very virulent or the woman is immune compromised or labor has been prolonged, neglected or obstructed or operative interventions have been done then the severity of the sepsis is such that the infection spreads beyond the pelvic cavity involving the general peritoneal cavity leading to peritonitis and all clinical classical signs of peritonitis.

These women on presentation appear sick, have constitutional symptoms, tachycardia, fever, abdominal distension, guarding, tenderness, rebound tenderness, rigidity, foul smelling lochia, uterine tenderness and or sub involution of the uterus. Abdominal distension due to paralytic ileus adds on to the distension caused by collection of the pus or exudate. 

Early diagnosis by laboratory investigation and imaging modalities form the cornerstone for further definitive treatment which is essential to reduce morbidity, mortality and sequelae. Complete blood counts, high vaginal swabs for aerobic and anaerobic cultures, ultrasound of the pelvis and abdomen to see the extent, amount, type of collection, state of the uterus and adenxa, remnants of the placenta, products of conception in the endometrial cavity is required    

CT scan study  with contrast adds to the diagnostic accuracy of ultrasound. A diagnostic tap of the fluid is many times required to identify the causative micro organism. It is essential to differentiate between infection caused by mycobacterium or by other aerobic or anaerobic germs. Tuberculosis needs to be excluded as it flares up in the post abortal or post partum women.  

Infections caused by polymicrobial pathogens require broad spectrum antibiotics and myobacteria tuberculosis requires anti tubercular chemotherapy. The chemotherapies are not completely effective in the presence of pus. Drainage of the pus is mandated under cover of effective chemotherapy/ antibiotics. This can be done surgically or by intervention radiology. Surgery allows the drainage of all pus quickly, (though the pus can recollect) so allows the quick action of the appropriate antibiotics. Intraperitoneal drains are usually inserted to ensure continuous drainage and prevent recollection of the fresh exudate. However, in suspected cases of abdominal tuberculosis leaving drains is not advisable as fistulas may form. Placing a pigtail catheter through radiology is a viable alternative in patients who have high surgical morbidity, drainage is slow, catheter can get blocked, may not be able to drain out pus from all areas of the abdomen and reinsertion may be needed. The choice between the two options has to be judiciously selected by the treating doctor. Surgical option is always available if pigtail catheter fails to complete the drainage of the pus. 

Every obstetrician must aim to eliminate or drastically reduce the causes responsible for postpartum sepsis. Prompt diagnosis and vigorous treatment can reduce the morbidity and long term sequelae. 

I bring the August issue of our journal to our readers and hope that our August  readers find the case reports in this issue informative and educative.

Sepsis Due To Uterine Scar Necrosis

Author Information

Dalvi P*, Chaudhary HK**. 

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

Abstract

Puerperal sepsis after cesarean section may present with systemic manifestations. Post operative sepsis due to necrosis of lower segment cesarean section (LSCS) uterine scar is not very common. Here we are presenting a case of puerperal sepsis in a patient with lower uterine segment scar necrosis and dehiscence. An exploratory laparotomy was done and the necrotic tissue was excised following which broad spectrum antibiotics were continued.

Introduction

Any bacterial infection of the genital tract within 42 days post-delivery is termed as puerperal sepsis. Proper aseptic precautions can prevent puerperal sepsis.[1] Post operative necrosis of the LSCS uterine scar is a rare occurrence.[2] Sepsis continues to be among the leading causes of maternal mortality in India, the other significant causes being preeclampsia and obstetric hemorrhage. 

Case Report

A 30 year old, para 2 living 1, married since 4 years, day 9 of emergency LSCS done at a private hospital was referred to the emergency department with complaints of pain in the abdomen since 2 days and abdominal distention since 2 days. She had history of fever since 1 day and 3 episodes of vomiting. There was no history of any major medical or surgical illness in the past. She was a known case of hypothyroidism on tablet levothyroxine 50 µgm. There was no history of any bowel or urinary bladder related symptoms.

Her first LSCS had been done 1 year ago and the baby had died at 4 months of age, the exact cause was not known. The second LSCS had been done 9 days ago in view of previous LSCS at a private hospital and she delivered a female child of 2.7 kg. As per available operative notes the surgery was uneventful.

Her clinical examination revealed a pulse of 94 beats per minute, blood pressure was 130/ 80 mm of Hg, bilateral pedal edema grade 2 pitting was present. On abdominal examination, abdominal wall edema and gross ascites was present, Pfannensteil scar was healthy. On per speculum: cervix and vagina were healthy, no foul smelling discharge was noted. Vaginal examination revealed a midposed uterus but exact uterine size could not be assessed. A cystic mass was palpable in the left iliac fossa about 10 x 10 cm. 

Hemoglobin was 11.2 gm %, WBC count 21860/ cu mm and platelet count of 1 lakh/ cu mm. USG showed a large complex left ovarian cyst with internal echoes suggestive of dermoid cyst. Gross ascites with internal septations were noted. CT scan was done which showed a 10 cm left ovarian cyst, with mild hepatomegaly. Specks of air were seen at the LSCS scar site. Fecal loaded colon was noted up to rectum. No evidence of small bowel obstruction was present.

An exploratory laparotomy was planned in view of suspected peritonitis. A general surgery reference was taken to rule out intestinal obstruction. Intra operative findings revealed 1.5 to 2 liters of inflammatory fluid with pus flakes present in the pelvis and peritoneal cavity. Suture line over the uterus was unhealthy with necrotic tissue seen protruding from the right side of the uterine incision, for about 2 - 3 cm in length. Greyish white necrotic tissue present over the uterine incision suture line was excised. Left ovarian dermoid cyst was seen of 7 x 8 x 10 cm with presence of pus flakes around it. Left oophorectomy was done. About  2 liters of wash was given with warm normal saline. Hemostasis was checked and confirmed. Two intra-peritoneal drains were placed, first in the pouch of Douglas and second, anterior to the uterus. The abdomen was closed in layers. 

Figure 1. Necrotic tissue over the uterine suture line.

Figure 2. Necrotic tissue over the uterine suture line.

The patient was monitored strictly in the post operative period. Drains were removed on day 4 post operatively. Higher antibiotics were started and were continued for 7 days. The post operative period was otherwise uneventful. Pus culture showed enterobacter sensitive to piperacillin, which patient received along with metronidazole for 14 days. Excised necrotic tissue which was sent for histopathological examination showed multiple bits of fibrino- suppurative exudates with mixed dense inflammation, hemorrhage and fibrin. No viable tissue was seen.

Discussion

The incidence of puerperal sepsis has declined remarkably due to the strict maintenance of asepsis in labor ward and operation theaters. There have been various predisposing factors for puerperal infection. Sepsis is more commonly seen with LSCS as compared vaginal delivery. The duration of rupture of membranes, repeated vaginal examinations, anemia and prolonged labor pose a risk for puerperal sepsis in case of vaginal deliveries.

Most common organisms involved in puerperal sepsis are bacteria from the female genital tract. The focus of infection in case of sepsis following cesarean section is the infected surgical incision. Fever is an important presenting feature in puerperium.

Necrosis of the uterine scar may occur due to infection of the scar site. In the immediate postpartum period it may present as secondary hemorrhage and can also lead to peritonitis. A high index of suspicion may be needed and radiological modalities can be used for confirmation. 

In cases with post operative LSCS scar dehiscence exploratory laparotomy with repair has previously been considered to be the treatment, but due to the fragile nature of the infected tissue and excessive bleeding many patients may need hysterectomy.[4] Recently microsurgical laparoscopic repair have been reported.[3] In our case also, an exploratory laparotomy was done and the necrotic tissue was excised, no bleeding was encountered. There are reports wherein the scar dehiscence has been managed conservatively using only antibiotics for treating the infection and the uterine incision is allowed to heal by secondary intention.[1] 

Conclusion

Puerperal sepsis is a largely preventable cause of morbidity and mortality. Aseptic precautions will help to reduce the rate of sepsis and its complications. Prompt diagnosis will require a high index of suspicion and confirmation by using radiological tests.

References

1. Parulekar SV, Hira P. Post cesarean Anterior Pre peritoneal Abscess. JPGO 2015 Volume 2 Number 8. Available from: http://www.jpgo.org/2015/08/postcesarean-anterior-preperitoneal.html 
2. Mahajan D, Kang M, Sandhu MS, Jain V, Kalra N, Khandelwal N. Rare complications of cesarean scar. Indian J Radiol Imaging 2013; 23:258-61.
3. Donnez O, Jadoul P, Squifflet J, Donnez J. Laparoscopic repair of wide and deep uterine scar dehiscence after cesarean section. Fertil Steril. 2008; 89(4): 974-980.
4. Wagner MS, Bedard MJ. Post postpartum uterine wound dehiscence: a case report. J Obstet Gynaecol Can. 2006; 28(8): 713-5.

Citation

Dalvi P, Chaudhary HK. Sepsis Due To Uterine Scar Necrosis. JPGO 2017. Volume 4 No.8. Available from: http://www.jpgo.org/2017/08/sepsis-due-to-uterine-scar-necrosis.html

Peripheral Candidial Funisitis

Author Information

Alfiya Bano*, Parulekar SV**. 

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

Abstract

We present a case of necrotizing candidial funisitis. It was unusual in that there were no significant predisposing factors and no demonstrable source of infection. The lesions found were also different from the classical description.

Introduction

Acute funisitis and chorionic vasculitis are fetal inflammatory responses to an amniotic cavity chemotactic gradient. They can result either because of intraamniotic infection or “sterile” intraamniotic inflammation which occurs in absence of demonstrable microorganisms. The prevalence of chorioamnionitis is 3-5% of placentas delivered at term as compared to 94% of placentas delivered between 21 to 24 weeks of gestation. The rate is higher in patients with spontaneous labour, ruptured membranes, preterm labour and clinical chorioamnionitis. We present here an unusual case of peripheral candidial funisitis.

Case  Report

A 28 year old woman, married for 5 years, gravida 4 para 1 living 1 abortion 2, presented to the emergency room with 37 weeks of amenorrhea and a complaint of leaking per vagina for 3 hours and labor pains for 0.5 hour. There was no history of fever or foul smelling discharge per vagina. She had undergone an appendectomy 2.5 years ago. She had been hospitalized at 29 weeks of gestation for threatened  preterm labour and iron deficiency anemia. She had been managed with tocolysis and iron therapy. There was no other contributory past history. Her anomaly scan showed fetal dextrocardia and normal situs (intrathoracic and intraabdominal). Her general condition was fair and vital parameters were within normal limits. Her systemic examination revealed no abnormality. Obstetric examination showed a single fetus of size of 34 weeks in vertex presentation. Fetal heart rate was 142 bpm, regular. Her uterine activity was 1 in 10 minutes, each contraction lasting for 10 seconds. A speculum examination showed normal vagina and clear, odourless amniotic fluid. A high vaginal swab was sent for microbiologic studies, which subsequently showed no growth. Vaginal examination showed the cervix to be 1.5 cm dilated and 30% effaced. There was a free leak of clear amniotic fluid. Her investigations showed Hb of 10 g/dl, white blood cell (WBC) count of 20900/cmm,  platelet count of 260X109/L, C-reactive protein 2.57 mg/dL, negative VDRL, nonreactive HIV, nonreactive HBsAg, and normal thyroid, liver and renal function tests. She was treated with parenteral amoxycillin and clavulanate, gentamycin and metronidazole in view of high white cell count and premature rupture of membranes (PROM). She progressed in labor slowly and developed acute fetal distress after 5 hours and at 3 cm cervical dilatation. There were repeated variable decelerations up to 90 bpm from a baseline of 110 bpm. An emergency lower segment cesarean section was performed and a male baby weighing 2.24 kg was delivered. Its 1 and 5 minute Apgar scores were and 9 and 10 respectively. The liquor was clear. The entire umbilical cord showed white tubercles measuring 2-3 mm in diameter (figure 1). The placenta and cord were sent for histopathological examination. Parenteral antibiotics were continued postoperatively. The baby was sent to the neonatal intensive care unit. The patient’s condition remained stable. There was no fever or foul smelling lochia. WBC counts were 24500/cmm on day 2  and 12300/cmm on day 04. Investigations of the baby showed early onset sepsis (WBC counts 29500/cmm and immature neutrophil to total neutrophil count ratio > 0.4). Hence the baby was given intravenous antibiotics (ampicillin plus sulbactam and amikacin). Neonatal 2 D-echo showed dextrocardia, a small ostium secundum type atrial septal defect, multiple small ventricular septal defects, a tiny patent ductus arteriosus with left to right shunt. Blood culture showed no growth. Parenteral antibiotics were continued for 7 days. Parents took discharge against medical advice for the baby on day 7 of life. Histopathology report was received after 15 days from birth. It showed mild vasculitis, acute on chronic funisitis, acute chorioamnionitis with focal distal villous hypoplasia. The inflammation was located at the surface of the umbilical cord suggestive of peripheral funisitis. Special stains for fungi – gomori methenamine silver (GMS) stain and periodic acid-schiff (PAS) stain showed yeast and pseudohyphae of candida  in the umbilical cord. All efforts to contact the patient and inquire about the health of the baby failed.

Figure 1. Tubercle like lesions on umbilical cord.

Discussion

Acute inflammatory lesions of placenta and umbilibal cord consist of acute histologic chorioamnionitis, funisitis (inflammation of umbilical vein followed by arteries followed by Wharton’s jelly) and chorionic vasculitis.[1] The characteristic feature of acute chorioamniotis is diffuse neutrophilic infiltration.[2] It can result either because of intraamniotic infection or ‘sterile’ intra-amniotic inflammation which occurs in absence of demonstrable microorganisms.[3] In response to the infection, cytokines get released by endothelium, macrophages and mast cells. This creates a chemical gradient which attracts neutrophils. It may get augmented by some peptides released from the microorganisms. The prevalence of chorioamnionitis and funisitis is 3-5% of placentas delivered at term as compared to 94.4% of  placentas   delivered between 21 to 24 weeks of gestation.[4] The rate is higher in patients with spontaneous labor, ruptured membranes, preterm labour and clinical chorioamnionitis.[5,6,7] Pathways for microbiological invasion of amniotic fluid are ascending from lower genital tract (the commonest route), hematogenous, or iatrogenic (amniocentesis and fetoscopy).[8] The most common organisms found in the amniotic cavity are Mycoplasma genitalium, Ureaplasma species, Gardenerella vaginalis, Fusobacteria and Candida. Women who become pregnant with intrauterine contraceptive devices in situ are at high risk for infection with Candida albicans.[9,10] It can be polymicrobial (30% cases). Fetal response to microbial invasion is higher in preterm than term gestation (as microbial invasion is established before initiation of preterm labor). Hence the rate of neonatal sepsis is more in preterm babies.

The case presented was unusual in that there was no source of sepsis in the mother’s lower genital tract. There were no clinical symptoms suggestive of sepsis, and leucocytosis and raised levels of C-reactive protein were the only indicators of maternal sepsis. However, the lesions found in the umbilical cord were not due to bacterial infection but due to Candidial infection. Usually the lesions of candidial funisitis are seen as multiple, yellow or white, small plaques on the surface of the umbilical cord. In our case the lesions were rounded tubercles and not plaques. The placenta did not show any features of fungal infection. The mother remained well without any treatment for vaginal candidiasis, suggesting that those organisms must have been commensals. It was unfortunate that the parents took the baby home without treatment of the fungal infection and also of the cardiac lesions.

References

1. Pacora P, Chaiworapongsa T, Maymon E, Kim YM, Gomez R, Yoon BH, et al. Funisitis and chorionic vasculitis: the histological counterpart of the fetal inflammatory response syndrome. J Matern Fetal Neonatal Med.2002;11(1):18-25.
2. Redline RW, Faye-Petersen O, Heller D, Qureshi F, Savell V, Vogler C, Amniotic infection syndrome: nosology and reproducibility of placental reaction patterns. Pediatr Dev Pathol.2003;6(5):435-48.
3. Romero R,Miranda J, Chaiworapongsa T,Chaemsaithong P,Martinez A,et al.Clinical chorioamniotis at term I:microbiology of amniotic cavity using cultivation and molecular techniques.J Perinat Med.2015;43(1):19-36.
4. Russell P. Inflammatory lesions of the human placenta: Clinical significance of acute chorioamnionitis. Am J Diagn Gynecol Obstet. 1979;2:127–37.
5. Seong HS, Lee SE, Kang JH, Romero R, Yoon BH. The frequency of microbial invasion of the amniotic cavity and histologic chorioamnionitis in women at term with intact membranes in the presence or absence of labor.Am J Obstet Gynecol. 2008;199(4):375 e1–5
6. Park HS, Romero R, Lee SM, Park CW, Jun JK, Yoon BH. Histologic chorioamnionitis is more common after spontaneous labor than after induced labor at term. Placenta. 2010;31(9):792–5. 
7. Lee SM, Lee KA, Kim SM, Park CW, Yoon BH. The risk of intra-amniotic infection, inflammation and histologic chorioamnionitis in term pregnant women with intact membranes and labor. Placenta. 2011;32(7):516–21.
8. Romero R, Mazor M. Infection and preterm labor. Clin Obstet Gynecol. 1988;31(3):553–84.
9. Romero R, Reece EA, Duff GW, Coultrip L, Hobbins JC. Prenatal diagnosis of Candida albicans chorioamnionitis. Am J Perinatol. 1985;2(2):121–2.
10. Qureshi F, Jacques SM, Bendon RW, Faye-Peterson OM, Heifetz SA, Redline R, Sander CM. Candida funisitis: A clinicopathologic study of 32 cases. Pediatr Dev Pathol 1998;1(2):118-24.

Citation

Alfiya Bano, Parulekar SV. Peripheral Candidial Funisitis. JPGO 2017. Volume 4 No.8. Available from: http://www.jpgo.org/2017/08/peripheral-candidial-funisitis.html

A Rare Case On Ovotesticular Sex Disorder

Author Information

Dhanawat J*, Pai K**, Dharmadhikari M*, Samant PY***.

(*Third year resident, **Assistant Professor, ***Professor and Unit Head , Department of Obstetrics and Gynecology, KEM Hospital, Parel, Mumbai, India)

Abstract

Ovotesticular disorders are a rare variety of complaints. In these disorders, ovarian tissue and testicular tissue are present in the same individual irrespective of the genotype. We are presenting such a case where a 19 year old with a genotype of 46XX (female), with gonads having ovarian and testicular tissue was raised as a male by parents since childhood.

Introduction

Ovotesticular disorders are rare disorders. Karyotype associated with these disorders usually are 46XX. However, 46XY, and mosaics like 46XX/46XY, 45X/46XY all have been documented. These are grouped together in the ‘Disorders of Sex Development’ (DSD).

Case Report

A 19 year old person reared, as a male, uneducated, farmer by occupation belonging to a low socio-economic background presented with complaints of gynecomastia and cyclical hematuria. This individual was born at home, and the delivery was conducted without any birth attendant.  Though the parents noticed ambiguous genitalia at birth, no medical advice was sought.  On examination sexual maturity rating (SMR) was Pubic hair (P)5; Breast (B)5. Patient had a phallus of 4 cm in length, with no urethral opening. Single small perineal opening was present. No gonads were palpable. Hormonal studies were sent and all values were normal for a female genotype. Karyotype was 46XX. Ultrasound (USG) of the abdomen and pelvis was suggestive of normal uterus, and presence of bilateral gonads, most likely to be ovaries. Cystourethrogram showed no evidence of fistula. Magnetic resonance imaging (MRI) showed presence of mullerian structures. It showed normal vagina, normal bladder, membranous urethra opening into penile urethra of length 4.7 cm and a rudimentary external visible penis was seen. No structures similar to testes in morphology or signal intensity was seen.

Figure 1. MRI of genito-urinary structures. Red arrow shows the uterus, blue arrow head is the urinary bladder, yellow arrow is the vagina and the lavender arrow is the phallus.

Patient followed up after 1 year with complaints of acute abdominal pain in the emergency room. USG showed torsion of the right gonad. Patient underwent emergency right gonadectomy for the same. Left gonad was adherent to the sigmoid colon and hence was left untouched. Histopathology examination of the gonad showed ovarian tissue consisting of stroma with primordial follicles and testicular tissue consisting of seminiferous tubules. Hence, diagnosis of 46XX ovo-testicular disorder was made. Patient further underwent hysterectomy with left gonadectomy a year after the first surgery and bilateral mastectomy six weeks after  hysterectomy. The patient will undergo reconstruction of a penis along with hormonal treatment with testosterone.  

Discussion

The term ‘Disorders of Sex Development’ (DSD) was given to conditions where disharmony between chromosomal, anatomical and gonadal sex exist.[1] 

Ovotesticular disorders are rare disorders, accounting for less than 10% of the developmental sexual disorders, with genital ambiguity occurring in 1 in 4500 births.[2]  Most common karyotype associated with these disorders are 46XX, uncommon being 46XY, mosaics like 46XX/46XY, 45X/46XY. Molecular basis of these disorders are not known but autosomal recessive cases have been reported. They are classified on histopathological examination as lateral when testis is on one side and ovary on the other; unilateral when ovotestis is on one side and testis or ovary is on the other side, bilateral when both side ovotestis is present.[3] A study done by Krob et al, found that the most common type of gonad was ovotestis (44.4%), followed by ovary (21%) and testis (12.5%). Ovarian tissue was most commonly found on the left side and testis on the right for unknown reasons.[4]  Gonadal tumors in these patients occur in 2.6 % of the cases, the testicular component more likely to get malignant. Dysgerminomas, seminomas, gonadoblastomas, yolk sac carcinomas have all been reported.[5]    

These disorders have varied clinical presentations. Most present with ambiguous genitalia with various degrees of virilisation, though normal female genitalia may be present rarely. Rarely individuals present in puberty with secondary sexual characters contradicting their sex. Approach to such patients is very challenging. Matsui et al reported 8 patients, wherein one of them was diagnosed 7 years after the first visit.[5] It’s a social and medical challenge, as proper assignment of sex, medical, surgical and psychosocial interventions are needed. Involvement of the neonatologist, obstetrician, psychologist, urologist, endocrinologist are needed. After determining the sex of the individual, surgery with biopsy to determine the gonads is done. Accordingly, contralateral gonad is removed and reconstruction of external genitalia is done.  Having one on one parental discussion about the future prospects of such patients on issues like fertility, their gender assignment, the mental acceptance of the condition by the child, it’s effects on his or her mental status is of utmost importance.

  

Conclusion

Approach to ambiguous genitalia involves meticulous history taking, thorough examination, with multiple imaging studies for genito-urinary anatomy, and karyotyping for genetic sex. Various surgical  approaches may be required. Realization that these surgeries are necessary but may not be 100 % successful in helping the individual lead a proper sexual life, and fertility could be compromised mandates one on one discussion. Understanding the psychosocial aspect of such patients help in better treatment.

References

1. Hughes IA, Houk C, Ahmed SF, Lee PA, LWPES Consensus Group, EPSE Consensus Group. Consensus statement on management of intersex disorders. Arch Dis Child 2006;91(7): 554-563 
2. Josso N, Audi L, Shaw G. Regional Variations in the Management of Testicular or Ovotesticular Disorders of Sex Development. Sex Dev 2011;5(5): 225-234  
3. Hughes I. Disorders of sex differentiation. In Kronenberg HM, Melmed S, Polonsky K, Larsen PR ,editors. Williams Text Book Of Endocrinology. 11th ed. Massachusetts: Elesvier – Saunders  2007; pp. 804-805.
4. Krob G, Braun A, Kuhnle U. True hermaphroditism: geographical distribution, clinical findings, chromosomes and gonadal histology. Eur J Pediatr. 1994;153(1):2-10.
5. Matsui F, Shimada K, Matsumoto F, Itesako T, Nara K, Ida S, et al. Long-term outcome of ovotesticular disorder of sex development: a single center experience. Int J Urol. 2011;18(3):231-6.

Citation

Dhanawat J, Pai K, Dharmadhikari M, Samant PY. A Rare Case On Ovotesticular Sex Disorder. JPGO 2017. Volume 4 No.8. Available from: http://www.jpgo.org/2017/08/a-rare-case-on-ovotesticular-sex.html

Translocated IUCD In Peritoneal Cavity

Author Information 

Agrawal A*, Samant PY**, Swathi HV***

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

Abstract

Finding of intrauterine contraceptive device (IUCD) incidentally during abdominal and pelvic surgery from colon, peritoneal cavity, pouch of Douglas and bladder have been reported in many cases. Usually this is the sequel of uterine perforation and IUCD being displaced. In some cases, IUCD can be detected incidentally during investigation and in some cases directly during surgery. Here we report a case of an incidentally detected IUCD in the pouch of Douglas during vaginal hysterectomy.

Introduction

IUCD is commonly used for birth spacing and longer acting contraceptive methods. In spite of all the advantages it also has some complications which need to be explained to the clients before insertion.  Complications include infection, irregular bleeding, chronic pelvic pain, infertility and ectopic pregnancy. There may be some serious complications like perforation of the uterus and translocation into adjacent structures. Though rare; they may be life threatening leading to perforation of the colon and even fistula formation, but in certain cases translocation may be asymptomatic.

Case Report

A 44 year old woman presented to gynecology outpatient department with irregular menstrual cycles and heavy bleeding for 6 months. She bled every 15-20 days and the  bleeding lasted for 7-8 days with passage of clots but no pain. She was P4 L4 with all normal deliveries. Her last child birth was 12 years ago. She didn’t give any history of contraception use or sterilization operation. She didn’t have any significant medical or surgical illness. On examination, her vital parameters were stable, there was no thyroid swelling, systemic examination was normal. Abdomen was soft with no tenderness or mass. On pelvic examination uterus was 10-12 weeks in size with multiple small fibroids.  Ultrasonography revealed bulky uterus with multiple fibroids in the anterior wall and on the fundus. Cervical and endometrial aspiration cytology were normal. She was advised vaginal hysterectomy. Hysterectomy was uneventful but there was a foreign body located in the peritoneal cavity which was retrieved. It was found to be an IUCD (Cu 7) without strings and was nude without any copper wire wound to it. There was no visible scar on the uterine walls. On recovery, the she was asked about IUCD insertion; she was not aware of this IUCD insertion and had not experienced any remarkable pelvic pain or gastrointestinal symptoms in the past. IUCD was removed and her post-operative period was uneventful.

Figure 1. Uterus with Copper 7.

Discussion 

Risk of IUCD perforation may vary from 1.3-1.8/ 1000.[1,2] IUCD should be placed with proper skill and caution to prevent this complication. IUCD can be placed post abortion, post placental, post partum, interval after 6 weeks of delivery or at any time between the 7th to 10th day of the menstrual cycle. Risk of perforation and displacement is slightly higher when it is placed immediately after delivery, as the uterus is soft, involuting and has thinning of the uterine wall during the postpartum period.[3] Early presentation is usually symptomatic with pain in abdomen while late presentation is asymptomatic and they present with missing thread or pregnancy.[4,5]

According to Balci et al diagnosis of missing IUCD can be made with pelvic ultrasonography (USG) as the first diagnostic modality. Pelvic radiography (X Ray lateral view with uterine sound) can also be done when diagnosis can’t be made by USG.[5] 

Tarus and Kaufman in their study found that abdominal sonography and computerized tomography (CT) scan were helpful in locating IUCD.[6] X-ray and USG may be helpful but CT scan provides clear information regarding IUCD location in relation to other viscera.

Management of translocated IUCD will depend on clinical judgment and symptoms of the patient. Usually endoscopic management is preferred. Mosley et al in 2012 in their review regarding removal of missing IUCD stated that majority of the displaced IUCD were copper IUCD or Lippes loop.[1] Out of 129 cases of displaced IUCD in 120 cases laparoscopic removal was done and in 20 of 120 cases laparoscopy had to be converted to open method. The approach may be decided depending upon the pre- operative evaluation and location of IUCD prior to surgery.[4,6] In cases with dense adhesion of bowel and urogenital organs to IUCD laparotomy is preferred. Laparoscopy is now been the preferred mode as it is associated with less tissue handling, lesser adhesion formation, shorter hospital stays and rapid recovery.[1,2] In our case probably adhesions did not form as the Cu 7 was nude and there was no infection.

Cases have been reported with IUD displaced into the bladder, colon, peritoneum, appendix, omentum, adnexa leading to many complications like peritonitis, fistula formation, obstruction, and tubo-ovarian abscess.[7] Cystoscopy, sigmoidoscopy and colonoscopy may be of help in retrieving IUCD in cases with IUCD in bladder or colon respectively.[8] 

Conclusion

Timing and technique of IUCD placement is important in preventing complication. IUCD should be placed after proper counseling and awareness so that if the thread is missing, she can come for follow up. Translocation into abdominal cavity though very rare, is still a life threatening complication.

References

1. Mosley FR, Shahi N, Kurer MA. Elective Surgical Removal of Migrated Intrauterine Contraceptive Devices From Within the Peritoneal Cavity: A Comparison Between Open and Laparoscopic Removal. JSLS 2012;16:236–41.
2. Arslan A, Kanat-Pektas M, Yesilyurt H, Bilge U. Colon penetration by a copper intrauterine device: a case report with literature review. Arch Gynecol Obstet. 2009;279(3):395-7.
3. Katara AN, Chandiramani VA, Pandya SM, Nair NS. Migra-tion of intrauterine contraceptive device into the appendix. Indian J Surg. 2004;66:179–180.
4. Weerasekera A, Wijesinghe P, Nugaduwa N. Sigmoid colocolic fistula caused by intrauterine device migration: a case report. Journal of Medical Case Reports.2014;8:81. 
5. Balci O, Mahmoud AS, Capar M, Colakoglu MC. Diagnosis and management of intra-abdominal, mislocated intrauterine devices. Arch Gynecol Obstet. 2010;281(6):1019 –1022.
6. Taras AR, Kaufman JA. Laparoscopic retrieval of intrauterine device perforating the sigmoid colon. JSLS. 2010; 14(3): 453–455.
7. Mederos R, Humaran L, Minervini D. Surgical removal of an intrauterine device perforating the sigmoid colon: a case report. Int J Surg. 2008;6(6):e60-2. 
8. Medina TM, Hill DA, DeJesus S, Hoover F. IUD removal with colonoscopy: a case report. J Reprod Med. 2005;50(7):547-9.

Citation

Agrawal A, Samant PY, Swathi HV. Translocated IUCD In Peritoneal Cavity. JPGO 2017. Volume 4 No.8. Available from: http://www.jpgo.org/2017/08/translocated-iucd-in-peritoneal-cavity.html

Ventricular Arrhythmia In Early Pregnancy

Author Information 

Nasare PS*, Madhva Prasad S**, Gupta AS***

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

Abstract

Cardiac arrhythmias are among the most common cardiac complication encountered during pregnancy. Pregnancy may act as a trigger for exacerbation of pre-existing arrhythmias, whereas some arrhythmias may manifest for the first time. We present a case of new onset ventricular tachycardia in a 30 year old pregnant woman having one previous uneventful full term normal vaginal delivery. Difficulties while administering antiarrhythmic treatment in a pregnant woman and limitations for using diagnostic imaging modalities are discussed. Medical termination of pregnancy was performed to save mothers’ life considering high recurrence rate of ventricular tachycardia (VT).           

Introduction

Severe arrhythmias requiring aggressive or invasive therapies are rare. A combination of hormonal, hemodynamic and autonomic changes is a likely cause of arrhythmias in pregnancy. Little data is available on the efficacy or safety of antiarrhythmic drugs in a pregnant woman with arrhythmias. 

Case Report

A 30 year old woman married since 5 years gravida 2 para 1 living 1 with 3 months pregnancy presented to the antenatal outpatient department with complaints of palpitation, breathlessness and tremors in the hands for approximately three months. She was apparently well till around 3 months prior when she noticed these symptoms which were sudden in onset. Such episodes used to occur daily, one or two episodes a day and used to last for not more than 15 minutes. However, these symptoms subsided immediately upon taking rest; were not persistent and since it did not interfere with her daily activities, she did not seek any particular treatment for the same.   

During this time, she also experienced amenorrhea and was confirmed to be pregnant on dip stick testing, following which she experienced emesis, which was however not excessive. At 12 weeks 4 days of gestation by menstrual dates she presented to the outpatient department. 

On examination, she was stable, afebrile with a blood pressure of 100/70 mm of Hg. There was no pallor, edema or icterus. Abdominal examination was unremarkable and vaginal examination found a soft, pregnant uterus of 12 weeks size with internal os closed. Respiratory and neurological systems were unremarkable. However, the pulse was 136 beats per minute on the radial artery, irregular, with intermittent missed beats, but bilaterally symmetrical and palpable in all peripheral pulses, with no radio femoral delay. Pulse was rechecked after rest. Findings were the same.

First differential diagnosis of tachycardia was thyrotoxicosis (due to possible hydatidiform mole) and urgent ultrasonography (USG) was done. However it was suggestive of a single live intrauterine gestation of 12.4 weeks.

She was immediately admitted to emergency medical services. Electrocardiography showed ventricular tachycardia (heart rate of 220 beats per minute), right bundle branch block and atrioventricular dissociation (figure 1). Injection verapamil 50 mg was given intravenously. Tachycardia did not subside, and heart rate remained 220 beats per minute. Repeat dose of injection verapamil was given slowly over 15 minutes intravenously. She developed broad complex tachycardia, blood pressure dropped and became unrecordable and peripheral pulses became feeble. Direct cardio version shock of 200 Joules was given. However her persistent tachycardia settled only after 2 repeat shocks one of 200 J, another of 300 J and slow intravenous injection of amiodarone 150 mg. After these intensive measures, she was revived and normal sinus rhythm was achieved. A two-dimensional echocardiography was suggestive of moderate to severe mitral regurgitation and moderate tricuspid regurgitation, moderate pulmonary hypertension, thick and fleshy anterior mitral leaflets and restricted mobility of posterior mitral leaflet. She continued to have monomorphic ventricular tachycardia (figure 2) and was maintained on tablet amiodarone 200 mg and tablet labetalol 200 mg both twice a day.

Cardiac magnetic resonance imaging (MRI) suggested inflammatory cardiomyopathy with mediastinal lymphadenopathy. Differential diagnosis was infiltrative cardiomyopathy which was considered to be granulomatous (sarcoidosis) or infective (tuberculous) myocarditis. (Figure 3) 

Rheumatology opinion was taken, and it was advised to consider invasive testing (myocardial biopsy) for confirmation of probable sarcoidosis. To assess feasibility of myocardial biopsy, contrast enhanced computerized tomography of the chest was done. However, it showed sub-centimeter reactive lymph nodes in pretracheal and prevascular region, largest measuring 5mm. (figure 4) It also showed moderate cardiomegaly with dilated left ventricles and infiltrates. (figure 5) In view of possible difficulty in cardiac biopsy involving sub-centimeter nodes and that the patient was pregnant, the apt diagnostic procedure (which could have confirmed the diagnosis) had to be postponed. In view of this, she was started on immunosuppressant medication (tablet prednisolone 50 mg daily). 

A medical termination of pregnancy in view of grave risk to the mother was recommended as another episode of ventricular tachycardia could lead to sudden maternal death. 

After stabilization of the patient and almost a week of no further episodes of decompensation of the patient, due consent was obtained and medical termination of pregnancy was done. Three doses of 400 mcg tablet misoprostol per vaginum 4 hours apart were required for expulsion of a female abortus of 74 grams, after which emergency check curettage was done. She did not develop any further episodes of ventricular tachycardia. She was discharged from the intensive cardiac care unit (ICCU) on tablet amiodarone 200 mg BD, tablet labetalol 200 mg BD, tablet prednisolone 50 mg OD in tapering doses. Mediastinal lymph node biopsy was attempted but in view of extremely small lymph nodes it was not possible and she was discharged on same drugs and was advised to follow up in the event of recurrence of symptoms.  

Figure 1. ECG showing ventricular tachycardia approximately 300 beats per minute with right axis deviation

Figure 2. ECG showing ventricular tachycardia approximately 200 beats per minute with left axis deviation 

Figure 3. Contrast enhanced MRI images showing edema of apical septum mid anterior wall and anterolateral segment and patchy areas of mid myocardial and epicardial enhancement of mid anterior wall, mid lateral wall mid septum. (arrow pointing to apical septum)

Figure 4. Axial CECT chest showing pretracheal lymph node (arrow)

Figure 5. Axial CECT showing mild dilated left ventricle. (arrow)

Discussion

“Tachyarrhythmias” refer to non-sustained or sustained forms of tachycardia originating from myocardial foci or re-entrant circuits. The definition of tachycardia are rhythms that produce a ventricular rate > 100 beats per minute.[1] These are possibly explained due to hormonal imbalances of pregnancy, which can predispose to tachyarrythmias. It can also occur in patients with history of congenital heart disease.[2] Our patient had structural heart disease (mitral regurgitation), as confirmed by two dimensional echocardiography. Whether this lesion was a congenital lesion is not clear because no prior medical records were available. However ventricular tachycardia has been reported in pregnant woman without structural heart disease also. Though not clearly explained, it is postulated that abrupt changes in venous return could lead to acute changes in the blood volume predisposing to arrhythmias.[3] Arrhythmias in pregnancies are otherwise uncommon. In a study done by Lee et al, atrial fibrillation was noted in around 60 among 100000 pregnancies.[4] Ventricular fibrillation in general population is estimated to occur at a rate of less than 2 per 100000. Incidence is increased with adolescence and has a male predominance. The intriguing fact about ventricular tachycardia is that majority of the patients die at presentation in the emergency service department. However, our patient was hemodynamically stable at presentation.[5]

Very similar to what occurred in our patient, broad complex tachycardia and collapse has been described in pregnancy, but however in late gestation.[6] The common anti-arrhythmic agents used in the first line management of ventricular tachycardias include sotalol, procainamide, amiodarone and verapamil. In this case, verapamil was used. Soon upon administration of verapamil, there occurred hemodynamic collapse. Such a side effect is well documented and was not entirely unexpected. Soon after this, our patient required direct cardioversion. Though concerns regarding fetal arrhythmias and precipitation of preterm labor exist, cardioversion when indicated should be performed with no hesitation, since it is life saving for the mother.[7]

Amiodarone is a known cause for fetal hypothyroidism and is discouraged during pregnancy. But in our patient, in the interest of maternal safety, it had to be administered. [8]

Our patient was investigated for cause of ventricular tachycardia and cardiac MRI was suggestive of myocarditis. Myocarditis can be because of viral etiology such as coxsackie virus or immunopathogenesis involving cardiac myosin. These etiologies may be subclinical. Endomyocardial biopsy is mandatory to diagnose the etiology of myocarditis.[9] Myocardial biopsy gives conclusive diagnosis, but was not done in our patient due to her pregnant state.

Cardiac MRI in our patient suggested cardiac sarcoidosis. Though FDG- positron emission tomography appears to have better sensitivity than MR imaging, the latter is feasible in pregnancy and suggested the diagnosis.[10] Cardiac sarcoidosis causing ventricular tachycardia is a well established but rare entity. The exact pathogenesis is not clearly understood and though catheter ablation is best treatment, the VT recurrence free survival was only 37% in one year.[11]

Because of this high recurrence rate, MTP was advised given the grave risk to physical health of the mother if pregnancy was continued. This case is presented to highlight the rare occurrence of ventricular arrhythmia in pregnancy,  presentation of ventricular arrhythmia without mortality, successful stabilization of the patient with pharmacological and electrical cardioversion and a medical condition necessitating medical termination of pregnancy. The multidisciplinary management of a high risk pregnancy in a tertiary care set up is highlighted. 

References

1. Marchlinski F. The Tachyarrythmias. In: Longo DL, Fauci AS, Kasper DL, Hauser SL, Jameson JL, Loscalzo J, Eds. Harrison’s Principles of Internal Medicine, 18th ed. New York: Mc Graw Hill 2012; pp.1489.
2. Enderlin EA, Khaled KT, Oke L, Madmani M, Paydak H. Management of tachyarrhythmia during pregnancy. Turk Kardiyol Dern Ars. 2017 Mar; 45(2):189–96.
3. Ferreira NS, Barros TL, Gismondi RA. Supine Frequent Ventricular Extrasystoles in a Pregnant Woman without Structural Heart Disease. Case Rep Med [Internet]. 2016 ; 2016:1–3. Available from: http://www.ncbi.nlm.nih.gov/pubmed/27642300 
4. Lee M-S, Chen W, Zhang Z, Duan L, Ng A, Spencer HT, et al. Atrial Fibrillation and Atrial Flutter in Pregnant Women—A Population‐Based Study. J Am Heart Assoc. 2016; 5(4):e003182.
5. Tseng WC, Wu MH, Chen HC, Kao FY, Huang SK. Ventricular Fibrillation in a General Population - A National Database Study. Circ J. 2016; 80(11):2310–6.
6. Sengupta A, Slater TA, Sainsbury PA. The investigation and management of broad complex tachycardia and ventricular standstill presenting in pregnancy: A case report. Obstet Med. 2014; 7(3):131–4. 
7. Enriquez AD, Economy KE, Tedrow UB. Contemporary management of arrhythmias during pregnancy. Circ Arrhythm Electrophysiol. 2014;7(5):961-7
8. Vianna FSZ, Schuler-Faccini L, Weber-Schondorfer. Heart and Blood medicaitons. In: Schaefer C, Peters PWJ, Miller RK, eds. Drugs During Pregnancy and Lactation. Treatment Options and Risk assessment. 3rd edition. Amsterdam: Elsevier 2015; Pg 211. 
9. Rose NR. Viral myocarditis. Curr Opin Rheumatol. 2016; 28(4):383-9.  
10. Sharma S. Cardiac imaging in myocardial sarcoidosis and other cardiomyopathies. Curr Opin Pulm Med. 2009; 15(5):507–12. 
11. Kumar S, Barbhaiya C, Nagashima K, Choi EK, Epstein LM, John RM, et al.Ventricular tachycardia in cardiac sarcoidosis: characterization of ventricular substrate and outcomes of catheter ablation. Circ Arrhythm Electrophysiol. 2015;8(1):87-93.

Citation

Nasare PS, Madhva Prasad S, Gupta AS. Ventricular Arrhythmia In Early Pregnancy. JPGO 2017. Volume 4 No.8. Available from: http://www.jpgo.org/2017/08/ventricular-arrhythmia-in-early.html

Scar Endometriosis: A Rare Case

Author Information 

Shaikh A*, Mayadeo MN**, Warke HS***.

(* Second year Resident, ** Professor and Head of the Unit, *** Associate Professor Department of Obstetrics and Gynecology, Seth GS Medical College and KEM Hospital, Mumbai, India.)

Abstract

Endometriosis is defined as presence of endometrium like stroma and glands outside the uterine cavity. There are many causes of scar endometriosis. A common theory is the direct implantation of endometrial tissue in surgical scars. Here we present a case of a 30 year old female presenting with scar endometriosis after 3 years of tubal ligation. 

Introduction

Endometriosis is defined as presence of endometrial-like stroma and glands outside the uterine cavity.[1] The name ‘endometriosis’ was given by Rokitansky in 1860. It is a rare entity. It is reported in 0.03-1.08 % of the women following obstetric or gynecological surgeries. Post salpingectomy endometriosis is documented in 20-50% of the tubes which are examined after ligation. It occurs in 8-15 % of the women of the reproductive age group. It usually occurs in region such as abdominal wall, ovaries, cul de sac, uterine ligaments, pelvic peritoneum, bowel and rectovaginal septum. It can also be present in unusual places like CNS, thorax, urinary tract, gastrointestinal tract and in cutaneous tissues.[2] There are many causes of scar endometriosis. A common theory is the direct implantation of endometrial tissue in scars during the operation which commonly follows obstetric and gynecology surgeries.[3]

Case Report

A 30 year old G4P3L3MTP1 with mini laparotomy tubal ligation done 3 years back at a tertiary medical care center came to the outpatient department with chief complaints of cyclical pain at tubectomy scar site since 2 years. She also had complaints of bloating sensation during menses. She did not have any previous history of endometriosis. Her general examination was normal. On local examination there was presence of 4 x 3 cm size nodule at the tubectomy scar site. The nodule was tender and immobile. Fine needle aspiration cytology (FNAC) showed presence of endometriosis. Ultrasonography (USG) of the abdominal wall was suggestive of 4 x 3 cm size scar granuloma with no abnormalities of the uterus and ovaries. Small vertical incision was taken over the nodule of about 3 cm in size. There was presence of fibrous tissue which was excised by sharp dissection and sent for histopathology. Post-operative stay in the ward was uneventful and she was discharged on day 5 of the procedure. Histopathology examination of the tissue showed fibrous tissue with few scattered endometrial glands and endometrial stroma.

Figure 1. The scar endometriosis tissue being excised at the previous tubectomy scar site

Figure 2. The scar endometriosis tissue after excision

Discussion

Scar endometriosis usually follows previous abdominal surgery including hysterectomy (10 %), cesarean section (16 %) and tubal ligation(3.3 %). During minilaparotomy tubal ligation, endometrial tissue might be seeded into the wound and under hormonal influence these cells proliferate. Post salpingectomy endometriosis is usually noted at the tip of the stump, 1-4 years after a tubal ligation. The chances of endometriosis detection are higher if electrocautery has been used in tubectomy, if the proximal stump is short and the post ligation interval is long. Care for its judicious use can be taken to avoid this complication. The differential diagnosis includes hernia, hematoma, suture granuloma, lipoma, abscess, sebaceous cysts, neoplastic tissue and metastatic carcinoma.[4] The etiology is said to be the direct implantation of endometrial tissue. Under hormonal influence these cells may proliferate or the surrounding tissue may undergo metaplasia which leads to scar endometriosis. The time interval between the surgery and the presentation of scar endometriosis may differ from 3 months to 10 years.[5] Ultrasonography is the most common imaging modality used to detect endometriosis. CT scan or MRI may also be used for deep pelvic endometriosis. FNAC aids in accurate diagnosis especially in enlarged masses. Histology confirms the diagnosis of scar endometriosis. There is presence of endometrial glands, stroma and hemosiderin pigments on histopathology.[6] Local wide excision of scar endometriotic tissue with at least 1 cm margin free of the pathology left behind is the treatment of choice for scar endometriosis. This reduces the recurrence rate. Peritoneum is usually not opened in all the cases of scar endometriosis due to discrepancy in its etiology. Oral contraceptives, progestational and androgenic agents have been used in the treatment of scar endometriosis. Hormonal suppression is partially effective and surgical excision of the scar is the definitive treatment.[5]

Conclusion

Scar endometriosis is a rare and uncommon diagnosis. One should maintain a high level of suspicion if a woman presents with cyclical pain with a painful swelling at an abdominal incisional site, most commonly after any pelvic surgery. In such instances scar endometriosis should be considered as an important differential diagnosis. 

References

1. Goel P, Sood SS, Dalal A; Romilla. Cesarean scar endometriosis--report of two cases. Indian J Med Sci. 2005;59(11):495-8.
2. Wolf GC, Singh KB. Cesarean scar endometriosis: a review. Obstet Gynecol Surv. 1989;44(2):89-95.
3. K. Al-Jabri. Endometriosis at caesarian section scar. Oman Medical Journal. 2009;24,294-95.
4. Blanco RG, Parithivel VS, Shah AK, Gumbs MA, Schein M, Gerst PH. Abdominal wall endometriomas. Am J Surg. 2003;185(6):596-8.
5. Sunanda N, Asma F. Tubectomy scar endometriosis-a rare case report. International Journal of Contemporary medical research 2016;3(10): 2957-8.
6. Rao LCV, Sumalatha B, Swathi V. Scar endometriosis: A case series and review of literature. Int J Sci Stud 2015;3(4):180-183.

Citation

Shaikh A, Mayadeo MN, Warke HS. Scar Endometriosis: a Rare Case. JPGO 2017. Volume 4 No.8. Available from: http://www.jpgo.org/2017/08/scar-endometriosis-rare-case.html

Conservative Management Of Pyoperitoneum

Author Information 

Shetty A**, Prasad M** , Gupta AS*** 

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

Abstract 

Pyoperitoneum following spontaneous abortion is an uncommon entity. We present a case of gross pyoperitoneum with ruptured pyosalpingx following spontaneous abortion with retained products of conception with septicemia managed with pig tail catheter drainage and intravenous antibiotics. 

Introduction

Septic abortion begins as endometritis and after involving the endometrium; the infected retained products of conception can cause parametritis and even peritonitis. Bacteremia and sepsis can occur at any stage of septic abortion. Pelvic inflammatory disease (PID) is a common sequel of septic abortion. 

Case Report

A 20 year old gravida 2 para 1 living 1 abortion 1 presented to emergency room with acute onset abdominal pain, abdominal distension, fever and malaise. After a 3 month period of preceding amenorrhea (pregnancy confirmed by urine pregnancy kit), she had a spontaneous abortion at home. She denied any history of intrauterine manipulation. She presented 3 weeks after this abortion. The symptoms started gradually after the abortion and progressively increased. Following this, she presented to a hospital and underwent examination and ultrasonography (USG). Though this report was not available to us, she was told that USG did not show any evidence of retained products of conception. However, in view of persistence of symptoms, she presented to our hospital. Though main complaints were abdominal pain and distension, she did not have any bowel related symptoms. She had tachycardia with a pulse rate of 120 beats/ minute, blood pressure of 100/60 mm of Hg and she was very pale. Cardiovascular system examination was unremarkable apart from tachycardia and respiratory system was normal. Abdomen showed guarding, tenderness and distension. Bowel sounds were present. Per speculum examination showed foul smelling, purulent discharge from the cervix and no local lesions were visualized. On vaginal examination size of the uterus could not be made out, cervical motion tenderness was present, os was closed and there was no bleeding. Swab was collected and she was started on empirical antibiotics in view of white blood cell count of 19200/ mcL with neutrophilia of 80 %. Lymphocyte count was 20%. She was seronegative. Two units of packed red cells were transfused in view of hemoglobin of 3 gm% after administration of parenteral frusemide. Ultrasonography (USG) was suggestive of continuous tubular structure arising from left side of the uterus crossing over to the right side with echoes and gross ascites. Features were suggestive of acute pelvic inflammatory disease with pyosalpinx and pyoperitoneum. Serum creatinine was 1.7 mg/dl. CT scan confirmed the findings of USG and also identified retained products of conception measuring around 2x2 cm.  She was not seropositive . In view of sepsis and poor general condition of the patient, decision of insertion of a pigtail catheter was taken. A decision for exploratory laparotomy in the event of non-improvement was was the next plan of action. Pigtail catheter was inserted under ultrasonographic guidance in the right iliac fossa under all aseptic precautions. About 1800 ml of foul smelling pus was drained and sample was sent for aerobic, and mycobacterial culture. Hemoglobin increased and creatinine normalized. After stabilization and under antibiotic cover dilatation and curettage was done and retained products of conception were removed. Specimen was sent for culture (aerobic and anaerobic). Review ultrasonography showed 30 ml residual collection with pyosalpinx. Five days later, a second pigtail catheter had to be inserted to drain a recollected 800 ml pus from the right paracolic gutter.  Bacterial culture of the pus grew E.coli sensitive to amikacin and parenteral amikacin in therapeutic dose was given for 14 days. Mycobacterial and anaerobic cultures were negative. She improved gradually, no further collection occurred, pigtail catheter was removed and she was discharged on day 17 

Figure 1. CT scan image. Arrow shows the pyoperitoneum

Figure 2. Xray abdomen showing the Pigtail in situ (Arrow)

Discussion

Peritonitis is inflammation of the serosal membrane lining the abdominal cavity and therein contained organs. In the current era where induced abortions are liberalized and  medically supervised, the occurrence of post-abortion infections has reduced.  Maternal death is infrequent in such conditions. However, about 2 percent of women with miscarriage develop pelvic infection and sepsis.[1] CDC has given clinical criteria of an inflammatory syndrome occurring within 2 to 3 weeks after spontaneous or induced abortion. [2]

Multiple organisms are implicated in the pathogenesis of PID. These include Neisseria gonorrhoeae, gardnerella vaginalis, haemophilus influenzae, and anaerobes such as peptococcus and bacteroides species. As polymicrobial growth is most common we used piperacillin tazobactum for gram positive organisms, metronidazole for anaerobic and amikacin for gram negative organism as an empirical formulation.[2] 

PID may produce tubo-ovarian abscess (TOA) and rupture can result in peritonitis. Our patient also had a pyoperitoneum that extended upto the perihepatic region. Life threatening complication of acute rupture of a TOA may result in diffuse peritonitis and necessitate urgent abdominal surgery.[3] However, in our patient such a measure was not undertaken as she had severe anemia, was hemodynamically stable and her condition was not acute. Patients in whom surgery is deferred, computerized tomography must be done to exclude bowel injuries. Prior to insertion and drainage with a pigtail catheter it is essential to confirm that the collection is homogeneous and not loculated. Blockage of the catheter placed may require manipulation or sometimes change of the catheter. Also, large or recurrent infected collections may require multiple catheter placements.[4]

Intrauterine retention of products of conception associated with post-abortion endometritis must be evacuated either by medical or surgical methods. [5] The curettage material obtained in our case did not grow any organism. Studies have demonstrated curettage to have better outcome as compared to conservative management in patient with acute kidney injury.[6]

Studies have demonstrated risk of bowel obstruction due to adhesions, however there are no comparative studies to compare the incidences between surgical exploration and conservative management with a pigtail catheter.

Conclusion

Our patient improved symptomatically with the minimally invasive procedures. This case is being presented to highlight the fact that minimally invasive procedures can be as effective as surgical exploration in appropriate cases of acute severe PID.

References

1. Cunningham FG. Abortion. In: Cunningham FG,  Leveno KJ,  Bloom SL,  Spong CY,  Dashe JS, Hoffman BL et al, eds. Williams Obstetrics 24th ed. Mc Graw Hill. New York. 2014;350-8.
2. Pelvic Inflammatory Disease (PID). 2015 Sexually Transmitted Diseases Treatment Guidelines [Internet]. Available from: https://www.cdc.gov/std/tg2015/pid.htm
3. Thakur HS, Madhva Prasad S, Gupta AS. Postpartum Tuberculous Pyoperitoneum. JPGO 2016;3(7) Available from: http://www.jpgo.org/2016/07/postpartum-tuberculous-pyoperitoneum.htm
4. Percutaneous Abscess Drainage [Internet]. Available from: https://www.radiolmogyinfo.org/en/info.cfm?pg=percabscessd
5. Lamy C, Mezan de Malartic C, Perdriolle E, Gauchotte E, Villeroy-de-Galhau S, Delaporte MO,  et al. Management of post-abortum infections. J Gyncol Obstet Biol Reprod. 2012; 41(8):904–12. 
6. Singhal PC, Kher VK, Dhall GI, Mehta RL, Visweswaran RK, Nampoory MR, et al.  Conservative vs. surgical management of septic abortion with renal failure. Int J Gynaeco Obstet. 1982;20(3):189–94.

Citation

Shetty A, Prasad M, Gupta AS. Conservative Management Of Pyoperitoneum. JPGO 2017. Volume 4 No.8. Available from: http://www.jpgo.org/2017/08/conservative-management-of-pyoperitoneum.html

Pregnancy With Takayasu Arteritis And Heart Disease

Author Information

Venkateswaran S*, Chauhan AR**

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

Abstract

Takayasu aortoarteritis (TA) is a condition that causes narrowing, occlusion and aneurysm of medium and large arteries. Pregnancies in such cases are associated with severe life threatening systemic complications, and hence require super-specialty management and future contraception counseling. We present a case of a 30 year old primigravida with rheumatic heart disease with severe aortic stenosis in a diagnosed case of TA. Pregnancy was continued with multidisciplinary care and the patient delivered by a lower segment cesarean section at term for obstetric indication.

Introduction

Takayasu aortoarteritis (TA) is a chronic idiopathic large vessel vasculitis. It primarily involves the aorta and its branches, coronaries and pulmonary arteries. It is also known commonly as “young female arteritis” owing to the demographic distribution; and “pulseless disease” as it causes progressive inflammation, narrowing, scarring and abnormal ballooning within the aorta and its major branches. It shares some histologic features with giant cell arteritis, which is the other major large-vessel vasculitis. Affected individuals are usually women in reproductive age group and of Asian origin. The incidence is 2.6 cases per million population per year.[1] TA was earlier suspected to be an infection, later identified as an autoimmune condition. Genetic factors may also have an important role.[2] The inflammatory process has been thought to be initiated by cell mediated immunity. Pro-inflammatory cytokines like tumor necrosis factor (TNF)-alpha, interferon (IFN) gamma and interleukins are associated with granuloma formation on large vessel walls.[3] Successful management during pregnancy requires a multidisciplinary approach with obstetrician, cardiologist and rheumatologist because of multiple systemic complications associated with the disease.[4]

Case Report

A 30 year old primigravida presented at 26 weeks of gestation to our OPD. She was a diagnosed case of TA since the last 10 years. Rheumatic heart disease with aortic regurgitation (AR) had been diagnosed 6 years ago; aortic valve replacement with bioprosthetic valve (Epic® St. Jude’s valve) was done within a year of diagnosing AR. She had no complaints and was regularly following up with a cardiologist. She was on oral prednisolone 20 mg, metoprolol 50 mg and diuretic (furesemide 40 mg), all once daily. A repeat echocardiogram was done after she conceived for evaluation of persistently raised blood pressure in both upper limbs; and it was suggestive of severe restenosis of the aortic valve and type 2 diastolic dysfunction. Patient was then referred to us for further multidisciplinary management. 

On examination her general condition was fair. She was afebrile and had no recent history of acute febrile illness. Her radial pulse was 80 beats/ minute and of good volume in the left radial artery but was felt feebly in the right radial artery. Blood pressure in the left brachial artery was 165/ 80 mm Hg and right brachial artery was 178/ 80 mm Hg. On cardiovascular auscultation, both heart sounds were heard normally and there was a pansystolic murmur. Respiratory system was normal. There was no neurological or vascular deficit in any limbs. Per abdomen, uterus was 24 - 26 weeks’ size, fetal heart sounds were heard and regular, uterus was relaxed. Per vaginally os was closed and uneffaced.

Complete hemogram was normal with a hemoglobin of 12.5 gm %, adequate total leucocyte and platelet counts. Her plasma sugars, liver, renal and thyroid function tests were within normal limits. Her CRP level was 21.7 mg/ dL. All autoimmune markers (ANA, dsDNA, anti ß2 glycoprotein, lupus anticoagulant, anti-phospholipid antibody) were negative. Fetal malformation scan was normal.

Cardiologist repeated a 2D echocardiogram which showed very severe aortic stenosis with a valve area of 0.6 square cm, mild mitral regurgitation, mild tricuspid regurgitation and mild pulmonary hypertension. Shortening of second stage of labor was advised due to severe degenerative stenosis of the bioprosthetic aortic valve. She was asked to continue all medicines. Rheumatologist advised to continue prednisolone 20 mg daily for 6 weeks, followed by tapering of dose by 2.5 mg every 15 days. Tablet labetalol 100 mg twice daily and nifedipine 20 mg twice daily were started and were continued throughout pregnancy.

Her blood pressure was closely monitored and she was watched for development of preeclampsia. Obstetric ultrasound with Doppler studies were done serially (weekly) for early detection of intrauterine growth restriction. At 37 weeks of gestation, she had prelabour rupture of membranes with poor Bishop score, and was unwilling for induction of labor. Hence emergency lower segment cesarean section was done under spinal anesthesia. She delivered a healthy female neonate of 2.5 kg birth weight with Apgar score of 9/10. The LSCS was uneventful with 600 ml of blood loss. In the immediate postoperative period she developed acute breathlessness due to severe hypovolemia and pulmonary edema. She was then intubated on he operation table itself and shifted to cardiac intensive care unit for observation. Diuretics and inotrope support were started and she was extubated within 24 hours. She had a gradual recovery was shifted to the ward after 5 days. She started breastfeeding and was discharged after cardiac and rheumatologic opinion and advised to continue antihypertensives and steroids in tapering dose. 

At cardiac follow up post-delivery, she has been advised a re-do aortic valve replacement due to severe aortic stenosis, restricted aortic valve movement and hypokinesia at the left ventricular base.

Discussion

More than 150 pregnancies in women with TA have been published. Most common complication seen is hypertension (in around 30% pregnancies) with 20% developing preeclampsia. Myocardial infarction, aortic aneurysm and dissection, pulmonary embolism and such other serious maternal complications have been reported. Intrauterine growth restriction due to poor placental perfusion is seen in 20% pregnancies, and intrauterine fetal demise in 8% pregnancies.[4] In uncomplicated cases of TA in pregnancy, the entire duration of pregnancy and delivery are usually uneventful. In our patient, due to severe aortic stenosis as a consequence of degeneration of the biprosthetic valve, patient decompensated in the peripartum period. Newer bioprosthetic valves have a longer life than older ones, ranging from 8 to 20 years. The main advantage is that anticoagulation is not required; however in younger patients they are not preferred mainly due to the need for repeat surgery.With suitable anticoagulation, mechanical valves are preferred in young patients < 40 years of age, especially in low resource settings where repeat surgery may be difficult.[5] Decision for cesarean section or trial of normal labor is up to the treating obstetrician.

Pregnancy complications, including pregnancy loss and preterm birth, are higher among women with all forms of vasculitis.[6] Pregnant women without active Takayasu arteritis have a low risk of developing a cardiovascular event. For women with chronic hypertension, it is important to note the development of preeclampsia, fetal growth restriction and abruption.[7]

Blood pressure can increase significantly during labor. In the presence of subclavian stenosis, blood pressure measurements may be inaccurate by an arm cuff. Internal blood pressure monitoring may be required in these cases. Elective cesarean section may be prudent for these patients. Anesthesia in cases of TA is challenging as uncontrolled hypertension and end-organ dysfunction need to be avoided. General as well as regional anesthesia may be used; newer reports suggest the safety of low-dose spinal anesthesia with close monitoring.[8] 

Conclusion

Thus it is seen that TA is a common condition in women of reproductive age group, these pregnancies should be regarded as extremely high-risk and given adequate multidisciplinary care. Combined efforts of the obstetrician, cardiologist and rheumatologist can result in successful maternal and perinatal outcome.

References

1. Johnston SL, Lock RJ, Gompels MM. Takayasu arteritis: a review. J Clin Pathol. 2002; 55(7): 481-6.
2. Ogino H, Matsuda H, Minatoya K, Sasaki H, Tanaka H, Matsumura Y et al. Overview of late outcome of medical and surgical treatment for Takayasu arteritis. Circulation. 2008; 118(25): 2738-47.
3. Goel R, Kabeerdoss J, Ram B, Prakash JAJ, Babji S, Nair A et al.  Serum Cytokine Profile in Asian Indian Patients with Takayasu Arteritis and its Association with Disease Activity. The Open Rheumatol J. 2017; 11: 23-9.
4. Hauenstein E, Frank H, Bauer JS, Schneider KT, Fischer T. Takayasu’s arteritis in pregnancy: review of literature and discussion. J Perinat Med. 2010; 38(1): 55-62.
5. Choudhary SK, Talwar S, Airan B. Choice of prosthetic heart valve in a developing country. Heart Asia. 2016; 8(1): 65-72.
6. Machen L, Clowse ME. Vasculitis and Pregnancy. Rheum Dis Clin North Am. 2017;43(2): 239-47.
7. Tanaka H, Tanaka K, Kamiya C, Iwanaga N, Yoshimatsu J. Analysis of pregnancies in women with Takayasu arteritis: complication of Takayasu arteritis involving obstetric or cardiovascular events. J Obstet Gynaecol Res. 2014; 40(9): 2031-6.
8. Dutta B, Pandey R, Darlong V, Garg R. Low-dose spinal anaesthesia for a parturient with Takayasu's arteritis undergoing emergency caesarean section. Singapore Med J. 2010; 51(6): e111-3.

Citation

Venkateswaran S, Chauhan AR. Pregnancy With Takayasu Arteritis And Heart Disease.  JPGO 2017. Volume 4 No.8. Available from: http://www.jpgo.org/2017/08/pregnancy-with-takayasu-arteritis-and.html

Laparoscopic Approach In Rare Case Of Isolated Fallopian Tube Torsion

Author Information

Bhate A*, Bhate M** , Chitnis S*** 

(* Director, ** Consultant,  Shubhdeep Nursing Home, Andheri(West), Mumbai. *** Consultant, Department of Obstetrics & Gynaecology, MaxCure Superspeciality, Jogeshwari (East), Mumbai)

Abstract 

Fallopian tube torsion is a rare cause of acute abdomen. It may masquerade as ovarian or cyst torsion, posing a diagnostic challenge. Diagnosis is made only intra-operatively during laparoscopy or laparotomy. We present a case of isolated fallopian tube torsion presenting as repeated episodes of acute pain in abdomen, eventually requiring salpingectomy.

Introduction

Fallopian tube torsion is a rare cause of acute abdomen. It may be associated with adjacent cysts, hydrosalpinx, pregnancy or past pelvic surgery.[1] Timely diagnosis and early operative intervention can prevent salpingectomy and preserve fertility. Ultrasound and Doppler studies should be done in all cases of acute abdomen presenting in emergency.[2] In selected cases where patient is stable, MRI may be done. Signs such as whirlpool sign and plicae tubaliae are frequently seen and may help in clinching the diagnosis.[3]

Case Report

A 36 year old multipara woman presented to emergency with acute severe pain in right flank & right groin and nausea. An urgent ultrasound revealed normal uterus and bilateral polycystic ovaries. A 5.3x3.1x4.3 cm multicystic lesion, was seen separate from ovaries. No abnormal vascularity or calcification was seen. Tumor markers and beta hCG were normal. She was put on intravenous analgesics and spasmolytics. After her pain subsided, she was discharged on oral NSAIDs. She returned to emergency after one month with similar acute pain with guarding and rigidity. She was admitted and a CT scan was done, which showed a 4x3 cm benign right ovarian cyst. Decision for diagnostic followed by operative laparoscopy, if required was taken. On pre-operative evaluation, she was stable with pulse of 90 beats/ min, and blood pressure of 100/70 mm of Hg. She had no co-morbidities. Her pre-operative investigations revealed no abnormality. 

Peritoneal access was gained by supra umbilical and lateral ports. Isolated torsion of right fallopian tube with 3 twists and terminal hydrosalpinx was noted. Left fallopian tube, uterus and bilateral ovaries were normal. Right salpingectomy was done. There were no intra-operative complications and she recovered uneventfully. She was discharged on day 4 of surgery.

Figure 1. Torsion of right fallopian tube with 3 twists and a terminal hydrosalpinx

Figure 2. Final intra-operative picture after right salpingectomy

Discussion

Isolated fallopian tube torsion is a very rare finding with an incidence of 1 in 15,00,000 females. It is more common on right side.[4] Most of the cases reported worldwide are in women of reproductive age group.[5] Infrequently, it may be seen in pre-pubescent girls or post-menopausal women. At present, there are no clinical features, laboratory or imaging features pathognomonic of this condition. Acute pain in abdomen with gastrointestinal symptoms may be presenting features. Rarely, it may present as primary infertility in adult women without any acute symptoms.[6] Patient may have positive peritoneal signs, tachycardia, and or leukocytosis.[7] Preoperative ultrasound usually shows heterogeneous cystic adnexal mass with variable free fluid. Early laparoscopy is the gold standard for diagnosis and management.[1]

In our case, endoscopic intervention was delayed as patient had pain relief after spasmolytic administration. When she came back to emergency with same complaints as her previous episode, she was admitted under physician’s care. When cause of pain could not be ascertained, gynecology reference was made and decision for diagnostic followed by operative laparoscopy, if required was taken. Imaging studies had not shown any evidence of torsion and diagnosis was made intra-operative. Owing to necrotic changes and completion of child-bearing, salpingectomy was done.

Isolated fallopian tube torsion is an intraoperative finding and cannot be diagnosed clinically. Hence, it is important that early laparoscopy be planned in cases with acute abdomen in which diagnosis cannot be ascertained clinically and radiologically. An early surgical intervention can aid in diagnosis, treatment and prevention of complications. It becomes more important when patient is a young woman, where preservation of fertility is a concern.

References

1. Krissi H, Shalev J, Bar-Hava I, Langer R, Herman A, Kaplan B. Fallopian tube torsion: laparoscopic evaluation and treatment of a rare gynecological entity. J Am Board Fam Pract 2001;14(4):274-277.
2. Gunal YD, Bahadir GB, Boybey O, Cil AP, Aslana MK. A rare cause of acute abdominal pain in children: Isolated tubal torsion; a case  series.Turk J Emerg Med. 2017 Jun; 17(2): 73–76. 
3. Sakuragi M, Kido A, Himoto Y, Onishi Y, Togashi K. MRI findings of isolated tubal torsions: case series of 12 patients: MRI findings suggesting isolated tubal torsions, correlating with surgical findings. Clin Imaging.2017;41:28–32.
4. Macedo M, Kim B, Khoury R, Narkiewicz L. A rare case of right lower quadrant abdominal pain. Am J Emerg Med.2017;35(4):668. 
5. Toyoshima M, Mori H, Kudo K, Yodogawa Y, Sato K, Kudo T, et al. Isolated torsion of the fallopian tube in a menopausal woman and a pre-pubertal girl: two case reports. J Med Case Rep. 2015;9:258. 
6. Murphy EM, Pereira N, Melnick AP, Spandorfer SD. Spontaneous bilateral torsion of fallopian tubes presenting as primary infertility. Womens Health (Lond). 2016; 12(3): 297–301.
7. Wong SW, Suen SH, Lao T, Chung KH. Isolated fallopian tube torsion: a series of six cases. Acta Obstet Gynecol Scand. 2010;89(10):1354-6.

Citation

Bhate A, Bhate M , Chitnis S. Laparoscopic Approach In Rare Case Of Isolated Fallopian Tube Torsion. JPGO 2017. Volume 4 No.8. Available from: http://www.jpgo.org/2017/08/laparoscopic-approach-in-rare-case-of.html