Volume 6 Number 5

Editorial
Gupta AS

Cranial Keratinization and Pigmentation Of Vaginal Epithelium Without vaginal Prolapse
Solanke S, Parulekar SV.

Massive Broad Ligament Ovarian Leiomyoma In A Post-Hysterectomized Woman
Pednekar R, Parulekar SV.

Peripartum Hysterectomy In A Case Of Post Cesarean Pyometra
Madge H, Shah R, Warke HS.

Ruptured Right Horn Pregnancy In A Bicornuate Uterus
Rane P, More V.

Postpartum Urinary Bladder Rupture
Malani K, Samant PY, Thakur H.

A Rare Case Of Sjogren's Syndrome In Pregnancy And Perinatal Outcome
Akalyaa K, Sreelatha S, Kusuma M V, Yashaswini  G, Ramaiah R.

Malignant Ovarian Tumor In A Teenage Girl
Bathija S, Hatkar P.

Surgical Adhesive Dressing induced Allergic Contact Dermatitis
Srinivasan N,  Desai G,  Hatkar P.


Remembering Past Greats: Ingemar Ingemarsson
Prasad M.

Editorial

Gupta AS

Pregnancy is not a disease. It is a physiological process, a period of anticipation, joy and expectation. However, sometimes in a moment a normal pregnancy becomes abnormal, or high risk and the outcome sometimes for the mother or the child or both becomes tragic. Usually it is expected that a low risk pregnancy would end in a normal delivery with a good maternal, and fetal outcome. Unnecessary or unwarranted operative delivery can be one of the causes of changing a normal pregnancy to an abnormal one.
The commonest operative delivery performed in women is cesarean section. Globally the incidence of cesarean sections is increasing in developed as well as the developing countries. The ideal rate as stated by WHO and the international societies should be between 10-15 %. However in reality they are hovering around 35-40 % and in certain setups may even be as high as 70%.  Many of these cesarean births are not warranted. Clinicians should not aim to attain the ideal or targeted rate but should evaluate need based or evidence based need for cesarean deliveries.
Cesarean section is associated with various immediate or late complications. Hemorrhage primary, reactionary or secondary, hollow viscus injuries to the urinary and the gastrointestinal tract, sepsis and need of re-laparotomy are the commonest complications encountered after cesarean section.
Intraoperative hemorrhage can occur due to various reasons like, uterine atony, tears, extensions of incisions, injuries to cervix or even the vagina in second stage sections with deeply engaged head where the incision may be placed near the cervico vaginal junction in thinned out tissues, bladder trauma or bleeding following adhesiolysis, coagulation disorders and disorders of placentation. Cause should be treated.
Postoperative or postpartum sepsis usually caused by cesarean section. Surgical site infections, endometritis are the commonest sites for infection and incidence of infection is higher in emergency cesarean sections. Evidence shows that antibiotic prophylaxis prior to skin incision, good hemostasis and sound surgical technique can reduce postoperative wound sepsis.
Hollow viscus injuries are serious complications of cesarean section and are best avoided or diagnosed during and repaired intraoperatively. Incidence of bladder injuries is about 0.1-0.13%. As the incidence of cesarean section is rising so also the incidence of repeat cesarean sections is increasing. Careful separation of the bladder from the lower uterine segment is warranted to prevent direct or ischemic bladder injuries. Incidence of major degrees of placenta previa alone or with focal or complete morbid adherence of placenta increases the risks of bladder injuries and also ureteric injuries significantly. Intraoperative diagnosis and confirmation sometimes by retrograde cystodistention with methylene blue and meticulous double layer closure with a delayed absorbable suture and continuous bladder decompression for 14 days along with urinary antiseptics results in good healing of the bladder injury. Disruption of the repair or non detection of the injury can present with oliguria, anuria, urinary incontinence, vesico-vaginal fistula, urinary ascites, deranged renal function or intra-abdominal bleed. Urologist consultation, imaging studies and re-exploration with repair and prolonged catheterization, breakdown of the repair and increased morbidity is the unfortunate outcome of this complication.
Ureteric injuries are rarer than vesical injuries and they are more likely to be missed intraoperatively and present at various times and have various presentations in the postoperative period. Ureter can get transected, ligated, kinked at the level of the uterine incision, at its insertion in the bladder during obstetric total hysterectomy in cases of morbidly adherent placenta or atonic PPH with placenta previa, or injured while repairing a bladder injury.
Bowel injuries is not a serious risk as the gravid uterus shields the intestines behind itself. A bowel loop trapped in adhesions due to previous surgeries may be injured while separating adhesions or while entering the peritoneal cavity. Bowel trauma should be diagnosed during surgery and repaired by surgeons promptly either by closing the injury or by resection and an ostomy. However, many of these injuries remain undetected during the intraoperative phase and presents with fecal peritonitis.
Obstetricians should be alert to prevent a low risk pregnancy or delivery accelerating into the high risk group and developing serious morbidity, or even mortality due to complications many of them being inadvertently iatrogenic.
The much awaited May issue of our journal is here for our most valued readers and it has besides a bouquet of cases one case of postpartum bladder injury which makes for interesting reading.

Cranial Keratinization and Pigmentation Of Vaginal Epithelium Without vaginal Prolapse

Author information

Solanke SS*, Parulekar SV**.
(* Second Year Resident, ** Professor and Head, Department of Obstetrics and Gynecology, Seth G S Medical College & K E M Hospital, Mumbai, India.)
Abstract
Fluctuations in the circulating level of estrogen and progesterone occurring during the menstrual cycle and in pregnancy influence the health of the genital epithelium. In addition, the menopause and subsequent estrogen deficiency have been implicated in the etiology of a number of urogenital complaints including incontinence, urgency and recurrent urinary tract infection. Here, we are presenting a case of keratinization and pigmentation of the posterior vaginal wall mucosa in postmenopausal woman without eversion of the posterior vagina.

Introduction
Normal vagina is a stratified nonkeratinized epithelium.[1.2] It has basal, parabasal, intermediate and superficial cell layers. Fluctuations in the circulating level of estrogen and progesterone occurring during the menstrual cycle and in pregnancy influence the health of the genital epithelium.[3] Urogenital problems in the elderly female population are experienced by one third of women from the age 50 years.[4] Atrophic changes within the bladder neck, urethra and vagina are mainly due to estrogen deficiency. Vaginal dryness and friction with undergarments is often followed by keratiniation and pigmentation of the of vaginal mucosa.[5]

Case Report

A 62 years old woman, married for 48 years came to the gynecological outpatient clinic with complaints of something coming out per vaginum for 2 years. It was reducible on its own. She also complained of dryness of vagina and hot flushes.  She had no bladder or bowel complaints, white discharge or dyspareunia. She had four full term vaginal deliveries and was menopausal for 12 years. She had been recently diagnosed with hypertension and had no other medical or surgical high risk factors. On examination she had third degree uterine prolapse with large cyctocoele but no rectocoele. On per speculum examination, cervix and vagina were healthy, but posterior vaginal wall showed 2x2 cm area of blackish pigmentation in continuity with perineal skin. Her investigations for fitness for anesthesia were within normal limit. She underwent vaginal hysterectomy with site specific cyctocoele repair and a perineorraphy. During surgery, the pigmented part of vagina was excised and vaginal mucosa  was sutured in continuous non interloking manner with polygalatin No.0. Postoperativly the vaginal mucosa healed well and vaginal vault was healthy. Patient made an uneventful recovery. Histopathological examination of the specimen showed keratinization and melanin pigmentation of the affected portion of the vagina that had been removed during surgery.

Figure 1. Pigmentation of the lower posterior vagina.

Figure 2. Postoperative appearance.

Discussion

Proliferation and differentiation of cells in female reproductive organs, the oviduct, uterus and vagina, are regulated by endogenous estrogen.[6] Vaginal basal and parabasal epithelial cells show cytokeratins (K1, 4, 5 and 13), filaggrin, transglutaminases (-1 and -3) and caspase 14. But they do not normally form any keratin bundles as in skin.[7] Vaginal epithelium tends to get keratinized and pigmented with melanin when it is exposed to outside with vaginal wall prolapse of a long duration. It is usually seen in the most dependent position of the prolapsed vagina.[8]
The case presented was unusual in that there was no prolapse or eversion of the lower vagina. Thus the usual mechanism of development of keratinization and pigmentation of the vagina could not be operative. The only explanation possible is that the lower vagina was exposed because of laxity of the perineum due to past non-repaired obstetric second degree perineal tear. Even then the finding was unusual because such an occurrence is not normally seen in other women with a laxity of the perineum. The case is presented only as a curious phenomenon, and also to highlight the point that such epithelium needs to be excised in order to achieve sound epithelial healing postoperatively.

References
  1. Anderson DJ, Marathe J, Pudney J. The structure of the human vaginal stratum corneum and its role in immune defense. Am J Reprod Immunol. 2014;71(6):618-23.
  2. DeLancey JOL. Surgical Anatomy of the Female Pelvis. In: Jones HW, Rock J, editors. Te Linde's Operative Gynecology. 11th edition. Philadelphia: Lippincott Williams & Wilkins (LWW);2015. pp 100-101.
  3. Stenberg A, Heimer G, Ulmsten U, Cnattingius S. Prevalence of genitourinary and other climacteric symptoms in 61-year-old women. Maturitas 1996; 24: 31–36
  4. Bachmann GA, Nevadunsky NS. Diagnosis and treatment of atrophic vaginitis. Am Fam Physician2000; 61: 3090–3096
  5. Samsioe G. Urogenital aging—a hidden problem. Am J Obstet Gynecol 1998; 178: S245–S249
  6. Skala CE, Petry IB, Albrich SB, Puhl A, Naumann G, Koelbl H. The effect of hormonal status on the expression of estrogen and progesterone receptor in vaginal wall and periurethral tissue in urogynecological patients. Eur J Obstet Gynecol Reprod Biol 2010; 153: 99–103.
  7. Menon GK, Cleary GW, Lane ME. The structure and function of the stratum corneum. Int J Pharm. 2012;435:3–9.
  8. Zimmerman CW. Pelvic Organ Prolapse: Basic Principles. In: Jones HW, Rock J, editors. Te Linde's Operative Gynecology. 11th edition. Philadelphia: Lippincott Williams & Wilkins (LWW);2015. pp 827-846.
Acknowledgment

We thank Dr. Sana Bijapur for taking intraoperative photographs.

Citation

Solanke S, Parulekar SV. Cranial Keratinization and Pigmentation Of Vaginal Epithelium Without vaginal Prolapse. JPGO 2019. Vol 6 No. 5. Available from: https://www.jpgo.org/2019/05/cranial-keratinization-and-pigmentation.html

Massive Broad Ligament Ovarian Leiomyoma In A Post-Hysterectomized Woman

Author Information

Pednekar R*, Parulekar SV**.
(* Assistant Professor, ** Professor and Head, Obstetrics and Gynaecology, Seth G S Medical College and K E M Hospital, Mumbai, India.)

Abstract

We present to you a rare case of massive broad ligament ovarian leiomyoma in a 48 year old woman, para 4, who came with complaints of continuous pain in abdomen since 3 years, who also had undergone total abdominal hysterectomy 6 years back for abnormal uterine bleeding for uterine leiomyomas. While preoperative imaging gave differential diagnosis of broad ligament leiomyomas or ovarian tumor, her tumor marker CA125 was negative, the intra-operative findings were suggestive of ovarian origin, and histopathological findings confirmed a leiomyoma.

Introduction

Uterine leiomyoma is a common occurrence but extrauterine leiomyoma are rare and they pose a great diagnostic challenge due to their unusual location. Leiomyoma is benign tumor arising from smooth muscle cells. Technically, it can arise from any anatomical site which has smooth muscle cells in it. Extrauterine leiomyomas are usually seen originating from genitourinary tract (from the ovaries, fallopian tube, broad ligament, urinary bladder, urethra, vagina and vulva).[1,2].  Ovarian leiomyomas are even rarer. It is important to differentiate it from a malignant ovarian tumor which can be done by doing magnetic resonance imaging (MRI) and serum cancer antigens and observing intra-operative gross anatomical features of the tumor. We present to you a similar case of a huge broad ligament ovarian leiomyoma in a post-hysterectomized female.

Case Report

A 48 year old woman, para 4 living 4, with all full term normal deliveries, presented to our outpatient department with complaints of continuous abdominal pain for 3 years and occasional bleeding per rectum for 2 years. There were no other significant complaints. She had undergone total abdominal hysterectomy 6 years back for abnormal uterine bleeding for 12 weeks’ size uterus with leiomyomas. On per abdominal palpation there was 22 weeks’ size mass arising from pelvis, firm in consistency with restricted mobility. Per speculum examination revealed normal looking vagina. On bimannual examination, per abdominal findings were confirmed and a nodularity of the mass could be felt through the vault. The patient was also evaluated for bleeding per rectum and found out to have hemorrhoids. Transvaginal and transabdominal ultrasonography (USG) showed a large hypoechoic lesion arising from pelvis around 15x8 cm in size, possibly broad ligament fibroid or ovarian tumor. The ovaries could not be visualized separately. A contrast enhanced computerized tomography (CT) scan revealed a 1.5x15.5x16.5 cm ( APxTRxCC) sized mass lesion in the pelvis showing heterogeneous enhancement. Right ovary could not be seen separately. Arterial supply of the lesion appeared to be from branches of right gonadal artery and venous drainage into gonadal vein. Fat planes were compressed but maintained. Left ovary was normal, seen separately from the lesion. The mass was abutting right ureter without obstruction. The impression was of broad ligament leiomyoma. The serum cancer antigen CA125 level was in normal limit. Preoperatively right ureteric stenting was done and the patient was posted for exploratory laparotomy. The intra-abdominal examination showed a large mass of around 20 cm in diameter arising from pelvis, with firm consistency and irregular bosselated surface with many vessels running across it. Right Fallopian tube was stretched over the mass, right ovary could not be seen separately, suggesting an ovarian origin. The right ureter was seen running under the peritoneum on the right side of the tumor. The left fallopian tube and left ovary were seen adherent to bowel. Bowel loops were adherent to the mass posteriorly and urinary bladder was adherent anteriorly. The mass was freed from all the adherent structures by sharp and blunt dissection. The right ureter was freed from the mass. The ureteric integrity and course were confirmed on both the sides. The mass was separated out by clamping, cutting and ligating the feeding infundibular vessels. Pelvic peritoneum was closed with a suture of No. 1-0 polyglactin. The mass sent for histopathology examination which confirmed leiomyoma with hyaline change. Postoperative course of the patient was uneventful. The ureteric stent was subsequently removed on day 7.



Figure 1. Intra-operative findings of ovarian tumor (OT), with stretched out fallopian tube (FT) and  ovarian vessels (OV).

Discussion

Uterine leiomyoma is the most common benign tumor of female genital tract. Its true prevalence is underestimated. According to recent longitudinal studies, the estimated lifetime risk of leiomyoma in a woman over the age of 45 years is more than 60%, and is more common in blacks than in whites. The cause of leiomyoma remains unclear and its biology poorly understood. The key regulating factors in growth of a leiomyoma are ovarian steroids- oestrogen and progestogen, growth factors and angiogenesis, and the process of apoptosis. Black race, heredity, nulliparity, obesity, polycystic ovary syndrome, diabetes and hypertension are associated with increased risk, and there is emerging evidence that familial predisposition to leiomyomas is associated with a distinct pattern of clinical and molecular features compared with leiomyomas in families without this prevalence.[3] In addition, unusual growth patterns may be seen, including benign metastasizing leiomyoma, disseminated peritoneal leiomyomatosis, intravenous leiomyomatosis, parasitic leiomyoma, and retroperitoneal growth. In the presence of such a pattern, a synchronous uterine leiomyoma or a previous hysterectomy for removal of a primary uterine tumor may be indicative of the diagnosis.[3]
Ovarian leiomyomas are rare benign tumors comprising of 0.5%–1% of all benign ovarian tumors.[6] They are mostly unilateral, incidently found, a few millimeters in diameter, and are asymptomatic. They occur in premenopausal women.[7,8] Ovarian leiomyomas can be primary, secondary or parasitic in origin. Primary ovarian leiomyomas originate from ovarian tissues, including the intraovarian blood vessels, smooth muscle fibers, or similar tissues within the ovarian stroma and tunica albuginea.[4-6] Secondary leiomyomas can occur from intravenous leiomyomatosis or from leiomyomatosis peritonealis disseminata.[1] Parasitic ovarian leiomyomas are extraovarian in origin like from uterine pedunculated leiomyoma which later gets attached to the ovary.[4] The incidence of primary ovarian leiomyomas is low.[4] In the present case, considering that the patient had undergone a hysterectomy for uterine leiomyomas 6 years back, the possibility of a broad ligament leiomyoma could not be ruled out, the genetic tendency for development of leiomyomas being as before hysterectomy.
Because ovarian leiomyoma are the rare tumors of the ovary, they are not suspected preoperatively or intraoperatively. They can be easily misdiagnosed as either ovarian malignant tumors or ovarian fibroma/thecoma or uterine pedunculated leiomyoma or broad ligament leiomyoma.[4,9]
Imaging methods include USG, CT, and MRI. It is also important to differentiate from malignant ovarian tumor by doing tumor markers such as CA125. But confirmatory diagnosis can only be made after histopathological examination and immunohistochemistry.[10] A possibility of leiomyosarcoma should also be ruled out.[7] Whenever it is highly suspicious of malignancy a frozen section of the tumor can also be helpful to formulate the surgical management plan.
Our case was unusual in that the ovarian tumor had grown between the two leaves of the broad ligament, while most of the ovarian tumors grow into the peritoneal cavity. Development of such a tumor is further unusual in that it is not often reported after a hysterectomy. Since there is no uterus to impede its growth, such a mass is likely to grow across the midline and press upon structures on both sides of midline, and the urinary bladder anteriorly. Our patient had a globular tumor in midline, to which the bladder was adherent anteriorly.
The surgical approach will be dependent on age of the patient, menopausal status, and wishes for fertility. For patients nearing menopause or postmenopausal women hysterectomy with bilateral salpingo-oopherectomy can be done. If the tumor is unilateral, unilateral salpingo-oopherectomy can be done considering the benign nature of the tumor. In young women where fertility needs to preserved ovary can be preserved by enucleating only the leiomyoma.[11,12] In our case, since the woman had undergone hysterectomy and the tumor was unilateral, we undertook unilateral salpingo-oopherectomy. 

Conclusion

It is important to consider differential diagnosis of ovarian leiomyoma in a case of solid adnexal mass. Because of its rarity and imaging similarities to most commonly occurring uterine pedunculated leiomyoma, it is difficult to diagnose it preoperatively. But accurate intra-operative observations and histopathological examination of the tumor confirms the diagnosis. Immunohistochemistry for actin and desmin will also add to the diagnosis.

References
  1. Fasih N, Alampady K. Shanbhogue P, Macdonald DB, Fraser-Hill MA, Papadatos D, et al. Leiomyomas beyond the uterus: unusual locations, rare manifestations. Radiographics. 2008 Nov-Dec;28(7):1931-48. doi: 10.1148/rg.287085095
  2. S. Cavkaytar, A. Karaer & T. Ozbagi. Primary ovarian leiomyoma in a postmenopausal woman, Journal of Obstetrics and Gynaecology 2010;30:7, 746-747.
  3. Okolo S. Incidence, aetiology and epidemiology of uterine fibroids. Best Pract Res Clin Obstet Gynaecol. 2008;22(4):571-88.
  4. Koo Y, Cho Y, Kim J, Lee J, Kim M, Joo-Myung Kim J, et al. Ovarian leiomyoma as a potential cause of compromised fertility. Fertility and Sterility , Volume 95, Issue 3, 1120.e11 -1120.e14.
  5. Rajabi P, Hani M, Bagheri M, Mirzadeh F. Large ovarian leiomyoma in young woman. Adv Biomed Res. 2014;3:88.
  6. Agrawal R, Kumar M, Agrawal L, Agrawal KK. A huge primary ovarian leiomyoma with degenerative changes: an unusual. J Clin Diagn Res. 2013;7(6):1152-4.
  7. Tomas D, Lenicek T, Tuckar N, Puljiz Z, Ledinsky M, Kruslin B. Primary ovarian leiomyoma associated with endometriotic cyst presenting with symptoms of acute appendicitis: A case report. Diagn Pathol. 2009;4:25.
  8. Blue NR, Felix JC, Jaque J. Primary ovarian leiomyoma in a premenarchal adolescent: first reported case. J Pediatr Adolesc Gynecol 2014 Aug 1;27(4): e87-8.
  9. Usta U, Karadag N, TÜrkmen E, Haltas H. Primary leiomyoma of the ovary. Balkan Med J. 2006;23:39–42.
  10. Taskin MI, Ozturk E, Yildirim F, Ozdemir N, Inceboz U. Primary ovarian leiomyoma: A case report. International journal of surgery case reports. 2014;5:665-8.
  11. Wei C, LilicN, Shorter N, Garrow E. Primary ovarian leiomyoma : a rare cause of ovarian tumor in adolescence. J Pediatr Adolesc Gynecol2008;21(1):33-6.
  12. Ozcimen EE, Oktem M, Zeyneloglu HB, Ozdemir BH, Kuscu E. Primary leiomyoma of the ovary in a young woman: Literature review and report of a case. Eur J Gynaecol Oncol. 2006;27: 310-2.
Citation

Pednekar R, Parulekar SV. Massive Broad Ligament Ovarian Leiomyoma In A Post-Hysterectomized Woman. JPGO Vol 6 No. 5. Available from: https://www.jpgo.org/2019/05/massive-broad-ligament-ovarian.html

Peripartum Hysterectomy In A Case Of Post Cesarean Pyometra

Author Information

Madge H*, Shah R**, Warke HS***.
(* Senior Resident, ** Assistant Professor, *** Associate Professor, Department of Obstetrics and Gynecology, Seth G S Medical College and K E M Hospital, Mumbai, India.)

Abstract

Peripartum hysterectomy is commonly performed for obstetric hemorrhage and adherent placenta. We report a case of post cesarean pyometra and uterine necrosis who underwent hysterectomy wherein the presenting features were superficial wound gape and vaginal discharge.

Introduction

Uterine scar dehiscence can complicate cesarean section with complications like post partum hemorrhage, endomyometritis, localized/ generalized peritonitis, and sepsis.[1] Patients may need to undergo exploration and hysterectomy to treat sepsis. Peripartum hysterectomy is defined as hysterectomy performed after birth until 1 month after delivery.[2, 3] The incidence of emergency peripartum hysterectomy ranges from 0.2 to 5.09 per 1000 deliveries. The median of its incidence is 0.61 per 1000 deliveries.[4] We present a case of post cesarean pyometra and scar dehiscence presenting as superficial abdominal wound gape. Patient was taken up for exploratory laparotomy and the findings of uterine necrosis and purulent collection in the abdomen ultimately warranted a peripartum hysterectomy.

Case Report

A 20 year old para 1 woman was referred with post cesarean wound gape and vaginal discharge from post-operative (post-op) day 7 not responding to antibiotics. She had undergone cesarean section 13 days ago at a private hospital for anhydramnios and rupture of membranes for greater than 12 hours. On examination she was conscious, oriented, afebrile, pulse was 90/min., blood pressure was 110/70 mm of Hg; cardiovascular and respiratory system examinations were normal. Abdominal examination revealed uterus of 16-18 weeks, contracted. Superficial skin gape with subcuticular sutures and purulent foul smelling discharge was seen from the suture line, slough was present at the base of the wound but the rectus sheath beneath was intact. Per speculum and per vaginal examination revealed hot inflamed vagina with copious foul smelling discharge.
Investigations showed hemoglobin of 9.1 gm/dl, leucocyte count of 14,100/dl, platelet count of 2.88 lakhs/µl, normal liver and renal function tests. A 30 cc organized collection was noted in the pelvis on sonography. Contrast-enhanced CT scan of abdomen and pelvis showed pyometra with wound dehiscence, 21x21 mm collection tracking out from the uterus, multiple collections in the abdominal cavity upto the right subhepatic region were reported.
She was started on broad spectrum parenteral antibiotics (piperacillin-tazobactam, metronidazole and gentamycin). She developed fever and leucocyte count rose to 18,600/dl. Decision for emergency exploratory laparotomy was taken.  Intra-operatively, uterus was found to be soft with shaggy necrotic incision line and wound rupture of the  entire suture line. Purulent intrauterine collection of about 50 cc was noted. Collection was also in the abdomen along the paracolic gutters reaching upto the inferior surface of the liver (about 150 cc). 


Figure 1. Devitalised uterus with purulent discharge.


Figure 2. Necrotic uterus with scar dehiscence.

Omentum was adherent to the right lateral abdominal wall, uterine cornua and right fallopian tube. Sigmoid colon was adherent to posterior surface of the uterus. Small bowel loops were matted. She underwent hysterectomy in view of these findings of uterine necrosis and abdominal sepsis. Surgery was performed by a multidisciplinary team of senior obstetricians, general surgeons and urosurgeons. Two packed red blood cell volumes were transfused intraoperatively. She was monitored post operatively in anesthetic intensive care unit for 2 days and thereafter in the ward.
Post-operatively antibiotics were changed according to the culture sensitivity report. Injection Vancomycin was started in response to wound swab that grew Enterococcus faecium and injection Meropenem was started in response to growth of sensitive Enterobacter aerogenes. She recovered uneventfully with no fever spikes and leucocyte count normalizing by post-op day 6. Suture removal was done on day 14, her wound had healed completely and she was discharged. On follow-up after 6 weeks she was asymptomatic with healthy suture line and no discharge.

Discussion

Wound gape and discharge is a common complication post cesarean section. However, suspecting postpartum pyometra and peritonitis/ sepsis as an underlying complication in non healing wounds requires a vigilant eye and high degree of suspicion. Incidence of uterine scar dehiscence is around 0.6 % worldwide.[5] The condition may further be complicated by uterine necrosis. Investigative modalities such as Magnetic Resonance Imaging (MRI) or pelvic Computed Tomography (CT) may aid in forming the diagnosis.[6]
Exploratory laparotomy is warranted in such cases. Emergency re-exploration post cesarean section is seen in about 0.315 % of patients. Of these 64.81 % undergo hysterectomy.[7] Though the most common cause for hysterectomy is post-partum hemorrhage (51.85 %), puerperal sepsis and wound dehiscence lead to hysterectomy in 12.96 % cases as treatment of choice in 6 % of patients with postoperative uterine wound sepsis and necrosis.[7] Peripartum hysterectomy is performed as treatment of choice in 6% of patients with postoperative uterine wound sepsis and necrosis.[8] Total or sub-total hysterectomy was also observed as the definitive treatment in a study by Nigam A et al wherein conservative measures of broad spectrum antibiotics and abdominal lavage failed to heal an infected subinvoluted uterus which was found to be necrotic later on.[6]

Conclusion

Uterine and abdominal involvement in the septic process should be kept in mind in non healing wounds. Timely management of rupture of membranes, strict aseptic techniques intra-operatively and adequate antibiotic cover should be followed at the primary treating hospital itself. CT/ MRI are helpful in arriving at a diagnosis, however an exploratory laparotomy based on clinical findings should never be delayed. While conservative measures like lavage may help in localized infections, peritoneal sepsis is best managed with hysterectomy.

References
  1. Bharatam KK, Sivaraja PK, Abineshwar NJ, Thiagarajand V, Thiagarajane DA, Bodduluri S, et al. The tip of the iceberg: Post cesarean wound dehiscence presenting as abdominal wound sepsis. Int J Surg Case Rep. 2015;9:69-71.
  2. Sakse A, Weber T, Nickelsen C, Secher NJ . Peripartum hysterectomy in Denmark 1995-2004. Acta Obstet Gynecol Scand. 2007;86(12):1472-1475.
  3. Bodelon C, Bernabe-Ortiz A, Schiff MA, Reed SD. Factors associated with peripartum hysterectomy. Obstet Gynecol. 2009;114(1):115-123.
  4. de la Cruz CZ, Thompson EL, O’Rourke K, Nembhard WN. Cesarean section and the risk of emergency peripartum hysterectomy in high-income countries: a systematic review. Arch Gynecol Obstet. 2015;292(6):1201-1215.
  5. Diaz SD, Jones JE, Seryakov M, Mann WJ. Uterine rupture and dehiscence: ten-year review and case-control study. South Med J. 2002;95(4):431-5
  6. Nigam A, Gupta N, Elahi AA, Jairajpuri ZS, Batra S. Delayed uterine necrosis: Rare cause of nonhealing wound. J Clin Diagnostic Res. 2016 Dec;10(12):QD01-QD02.
  7. Akther R, Hossain T, Rashid M. Relaparotomy After Cesarean Delivery: A Prospective Study. J Dhaka Med Coll. 2011;20(1):57-62.
  8. Kwame-aryee R, Kwakye A, Seffah JD. Peripartum hysterectomies at the Korle-Bu teaching hospital: a review of 182 consecutive cases. Ghana Med J. 2007;41(3):133-138.
Citation

Madge H, Shah R, Warke HS. Peripartum Hysterectomy In a Case Of Post Cesarean Pyometra. JPGO 2019. Vol 6 No. 5. Available from:  https://www.jpgo.org/2019/05/peripartum-hysterectomy-in-case-of-post.html

Ruptured Right Horn Pregnancy In A Bicornuate Uterus

Author Information

Rane P*, More V*.
(* Senior Resident, ** Assistant Professor, Department Of Obstetrics and Gynecology, Seth G S Medical College and K E M Hospital, Mumbai, India.)

Abstract

Malformed uterus like a bicornuate uterus with pregnancy fails to expand as pregnancy advances resulting in a catastrophic event like rupture of the pregnant horn leading to maternal morbidity as well as mortality. This is a case of a woman with bicornuate uterus and 16 weeks of gestation with a ruptured right horn of uterus. Patient was referred from a peripheral hospital in view of ruptured ectopic pregnancy. At our hospital exploratory laparotomy with excision of the ruptured right horn of uterus with repair of the uterine defect was done.

Introduction

Incidence of uterine congenital anomalies due to Mullerian defects in normal fertile population is 3.2 %. Bicornuate uterus accounts for 5 % of all Mullerian anomalies.[1] Uterine anomalies have been classified according to American Fertility Society (now American Society for Reproductive Medicine). It divides uterine anomalies into seven groups but does not include vaginal and some combined anomalies. European societies like European Society of Human Reproduction and Embryology (ESHRE) and European Society for Gynecological Endoscopy (ESGE) have introduced a classification based on the anatomy of female genital tract that includes malformations of the uterus, cervix and vagina.[2] Bicornuate uterus is associated with obstetric complications like recurrent pregnancy loss, preterm labor, fetal malpresentations, intrauterine growth restriction, increased risk of operative intervention including cesarean section. This is a case of bicornuate uterus with rupture of right pregnant horn of uterus, classified as U4aC0V0 according to ESHRE/ESGE classification of female genital tract anomalies.[3]

Case Report

A 23 year old primigravida with 16 weeks 2 days of spontaneous conception was referred from a peripheral hospital with ultrasonography (USG) suggestive of ruptured right adnexal ectopic pregnancy with live fetus in the abdominal cavity and gross hemoperitoneum. She had presented with complaints of abdominal pain, giddiness and history of fall at home. She was advised urgent USG and was referred to our hospital for further management. She came to the emergency room with complaints of pain in abdomen and giddiness. She was a known case of bronchial asthma since 10 years but was not on any medication with no recent acute exacerbation. She had no history of surgeries or menstrual complaints. On examination, she was conscious and well oriented. She was afebrile but pale, had a pulse of 100 per minute and blood pressure of 90/60 mm of Hg. Cardiovascular and respiratory systems were within normal limits. On abdominal examination, abdomen was uniformly distended with tenderness, rigidity and dull note all over the abdomen. On speculum examination, cervix and vagina looked healthy with minimal bleeding through the os. On, bimanual examination uterine size could not be assessed. Marked right forniceal fullness and tenderness was noted whereas left fornix appeared to be free.
On investigation, her hemoglobin was 7 gm/dl. One unit of packed cells was transfused and an emergency exploratory laparotomy was done. On laparotomy around 1000 ml hemoperitoneum and 100 grams of blood clots were noted. Bicornuate uterus with rupture of right horn with placenta in situ and fetus lying in the peritoneal cavity was noted. Fetus and placenta were delivered out. The pregnant right horn of uterus was found communicating with the left horn. Bilateral fallopian tubes and ovaries appeared normal. Excision of the ruptured right horn followed by repair of the uterine defect was done in two layers using polyglactin 1 in simple interrupted technique. Patient tolerated the procedure and anesthesia well. Post-operative course was uneventful. On discharge, advice regarding contraception and early registration in the next pregnancy was given. She was explained about the risk of rupture and need for cesarean section in the next pregnancy.[4] Histopathology report confirmed the diagnosis of a ruptured uterine horn.

Fig.1. Bicornuate uterus with right ruptured uterine horn.

Discussion

Development of the female urogenital tract is a complex and multifactorial process. The uterus and upper part of vagina develop from two paired Mullerian ducts. Congenital malformations occur if there is agenesis of one or two ducts or absence of fusion or reabsorption of septum between the ducts. Defect may be partial or total and may affect one or multiple areas of the genitourinary tract.[5]
Uterine anomalies have been classified according to The American Fertility Society (now The American Society for Reproductive Medicine) and European societies like European Society of Human Reproduction and Embryology (ESHRE) and European Society for Gynecological Endoscopy (ESGE).[2] EAC (Embryological aberration, Anatomical abnormalities and Clinical features) classification is a recent classification for congenital malformations of female genital tract. As per EAC, this case can be classified as UtR 12 cm Fn No Obs, UtL 6 cm Fn No Obs.[6]
USG is a useful tool in the diagnosis of such cases, but it is only 26 % sensitive and sensitivity further decreases as pregnancy advances.[7] In the past majority of the cases were diagnosed after rupture of the rudimentary horn. However, with advances in imaging techniques like 3 or 4-dimensional ultrasound and MRI, diagnosis can be made before rupture. Incidence of rupture in an anomalous uterus is 8-10 % as a malformed uterus is not able to expand as pregnancy advances.[8,9] But there are several reports of different clinical presentations of pregnancy in a bicornuate uterus ranging from rupture in early gestation to successful outcomes with live pregnancy reaching term.[10,11]
USG in early gestation helps in the diagnosis of uterine anomalies. High index of suspicion is required to diagnose Mullerian anomalies in pregnancy, so that catastrophic events like rupture of pregnant horn of uterus is prevented as well as treated at the earliest, preventing maternal morbidity and mortality. Treatment in such patients include exploratory laparotomy with excision of the pregnant horn and reconstruction of uterine defect.

References
  1. Simon C, Martinez L, Pardo F, Tortajada M, Pellicer A. Mullerian defects in women with normal reproductive outcome. Fertil Steril. 1991;56(6):1192-93.
  2. The American Fertility Society. The American Fertility Society classification of adnexal adhesions, distal tubal occlusion, tubal occlusion secondary to tubal ligation, tubal pregnancies, Mullerian anomalies and intrauterine adhesions. Fertil Steril. 1988, 49(6):944-55.
  3. Grimbizis GF, Gordts S, Di Spiezio Sardo A, Brucker S, De Angelis C, Gergolet M et al. The ESHRE/ESGE consensus on the classification of female genital tract congenital anomalies. Hum Reprod. 2013 Aug;28(8):2032-44.
  4. Maruthini D, Sharma V. A case of live birth after uterine reconstruction for recurrent cornual ectopic pregnancy following IVF treatment. Case Reports in Obstetrics and Gynecology. Volume 2013. Available from: https://doi.org/10.1155/2013/625261
  5. Spencer TE, Dunlap KA, Filant J. Comparative developmental biology of uterus: insights into mechanisms and developmental disruption. Mol Cell Endocrinol 2012 May 6;354(1-2):34-53.
  6. Parulekar SV. EAC Classification of Congenital Malformation of The Female Genital Tract. JPGO 2015. Volume 2 No.4. Available from: http://www.jpgo.org/2015/04/eac-classification-of-congenital.html
  7. Jayasinghe Y, Rane A, Stalewkski H, Grover S. The presentation and early diagnosis of the rudimentary uterine horn. Obstet Gynecol. June 2005;105(6):1456-67.
  8. Walsh CA, Baxi LV. Rupture of primigravida uterus: a review of the literature. Obstet Gynecol Surv. 2007 May;62(5):327-34.
  9. Ravasia DJ, Brain PH, Pollard JK. Incidence of uterine rupture among women with mullerian duct anomalies who attempt vaginal birth after caesarean delivery. Am J Obstet Gynecol 1999 Oct;181(4):877-81.
  10. Kore S, Pandole A, Akolekar R, Vaidya N, Ambiye VR. Rupture of left horn of bicornuate uterus at twenty weeks of gestation. J Postgrad Med. 2000;46(1):39-40.
  11. Adeyemi AS, Atanda OO, Adekunle AD. Successful pregnancy in one horn of a bicornuate uterus. Ann Afr Med. 2013 Oct-Dec;12(4):252-4.
Citation

Rane P, More V. Ruptured Right Horn Pregnancy In A Bicornuate Uterus: A Case Report. JPGO 2019. Volume 6. No. 5. Available from: https://www.jpgo.org/2019/05/ruptured-right-horn-pregnancy-in.html

Postpartum Urinary Bladder Rupture

Author Information

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

Abstract

Bladder and uterine injury during instrumental delivery is a known complication but isolated intraperitoneal bladder rupture following instrumentation is rare. Etiology cannot be established if historical sequence is unclear and bladder injury may be blamed on the instrumentation which in all probability was without complications. We present a case of isolated spontaneous postpartum bladder rupture without uterine rupture because of its rarity.

Introduction

Assisted vaginal delivery is application of art of obstetrics in stressful conditions. The application outcome depends more on judgment, training and experience of obstetrician. The rate of operative vaginal delivery broadly varies worldwide and is commonly about 10-12 % of all deliveries.[1,2] Though forceps application is associated with genital tract as well as bladder trauma, isolated injury to bladder after instrumental delivery is rare. The sequence of events requires close scrutiny before bladder rupture is attributed to instrumentation. Spontaneous puerperal bladder rupture may occur if bladder emptying does not occur due to painful episiotomy and spasm; or unanticipated pressure necrosis due to prolonged pressure of compression by fetal head against symphysis pubis.

Case Report

A 31 year old para 1 living 1 delivered a child of 2.6 kg via outlet forceps for fetal indication at a tertiary care center. She was discharged uneventfully after 3 days. On the 7th postpartum day, she had multiple low grade fever spikes along with complaints of abdominal distension, pain, constipation, burning micturition and incomplete urinary evacuation. She followed up in the previous center and was referred to our hospital in view of deranged renal function tests, requiring super specialty management. On admission, she had stable vital parameters. Bowel sounds were sluggish, there was suprapubic tenderness, and it was dull on percussion. The fundus of the uterus could not be felt well on account of distension and tenderness. Vaginal examination revealed closed internal os, healthy lochia and a healing episiotomy wound.  She was kept nil by mouth, started on intravenous fluids, and empirical antibiotics. Urethral catheter (PUC) was inserted and 1.5 liters of clear urine was drained. In her serological and biochemical investigations, her renal function tests were deranged with serum creatinine levels of 3.8 mg%, blood urea nitrogen levels of 65.3 mg%, and blood urea level of 139 mg%. Tests for malaria, leptospirosis and dengue were negative. Hemoglobin (Hb) was 8.9 gm% and WBC Count was 14,600/cmm, C reactive protein was 158 mg/L. She was assessed by a nephrologist. Intake output charting, urine routine, microscopy, urine culture and antibiotic sensitivity and ultrasonography (USG) for kidney, ureters, and bladder were advised. The urologist advised continuous bladder drainage, full bladder and post void USG, non-contrast CT ( NCCT) with intravesical contrast (CT cystogram).  USG  was suggestive of mild bilateral hydronephrosis with mild jejunal dilatation. NCCT scan (with intravesical contrast, no intravenous contrast used due to deranged renal function tests) was suggestive of mild ascites with multiple pockets of collection with diffuse omental and mesenteric fat stranding. Postpartum bulky uterus and normal adnexae were noted. Final diagnosis was paralytic ileus with cystitis and acute kidney injury. Urine culture grew Klebsiella pneumoniae sensitive to Piperacillin tazobactum (Piptaz) / Amikacin/ and Imipenem. She was started on injection Piptaz (4.5 mg), Injection Metronidazole (100 c.c.), and was gradually shifted to oral fluids after assessing her bowel sounds. She recovered clinically, passed flatus and stools. The renal function gradually improved. Foley’s catheter was removed after 4 days.  Catheter drained adequate clear urine on each of these 4 days. She was able to pass urine with no urinary complaints. She was discharged at a creatinine level of 0.7 mg%.  She was referred from a private hospital to our center again on day 20 postpartum, with recurrence of fever spike, pain in abdomen, inability to pass urine and pyuria. Contrast enhanced CT scan done in private hospital showed loculated peripheral enhancing fluid collection continuous with bladder suggestive of intraperitoneal bladder rupture.


Figure 1. CT scan of the abdomen and pelvis. White arrow points to bowel and omental adhesions causing a mass to form around the bladder. Black arrow points to intraperitoneal dye.

She was received in surgical emergency unit with catheter in situ, she was afebrile with stable vital parameters. There was abdominal distension (bowel sounds were present) with suprapubic tenderness. Involuting uterus with closed os was noted. Episiotomy wound had gaped superficially. She was kept nil by mouth, started on intravenous fluids. In her biochemical reports the Hb was 9.5gm%, WBC was 8900/ cmm, serum creatinine was 0.5 mg%, Blood urea nitrogen was 5.2 mg%. Serum electrolytes were within normal limits. USG was suggestive of 83 cc of thick septated collection in the right paracolic gutter extending into inter bowel space suggestive of bladder rupture. CECT showed hypo dense collection of 5 x 2.4 cm with enhancing walls and a track extending from pouch of Douglas to sub hepatic region, clumped bowel loops in left iliac fossa with collection around them. Distended bladder showed a breach of 1.9 cm in left anterio-superior border with spillage in left iliac fossa. Exploratory laparotomy with bladder repair was advised by urologist. Intraoperatively, an abscess cavity formed by small bowel, sigmoid colon, uterus and bladder fundus with approximately 30 c.c. of pus was evident. Cavity was opened, with sharp dissection. Uterus was found intact. A 2 x 1 cm accidental bowel serosal tear during dissection was identified and sutured. Rest of small bowel and sigmoid colon were normal. A 4 x 5 cm of rupture site with irregular inflamed margins was seen on the bladder dome. Its edges were freshened. Bilateral ureteric orifices were cannulated and brought out via 20 Fr urethral catheter. Bladder fundus was sutured with polyglactin 2-0 in two layers after placing 16 Fr. suprapubic catheter. Methylene blue test was done to rule out leak. Abdominal drain was kept in pelvis before closure to facilitate drainage of any pelvic collection. Episiotomy wound needed only dressing. She recovered uneventfully and was discharged with catheters in situ. At follow up, her ureteric and suprapubic catheters were removed after normal cystogram. This was followed by per urethral catheter removal after the 21st post operative day. Episiotomy wound healed by secondary intention.

Discussion
 
An isolated intraperitoneal bladder rupture following vaginal delivery is rare.[3,4] The term “spontaneous rupture of urinary bladder- SRUB” was coined by Sisk and Wear.[5] In obstetric patients, bladder rupture is often accompanied by uterine rupture. In obstetrics, isolated bladder rupture is observed in very few cases. Sustained pressure from fetal head against bladder during forceful uterine contractions can lead to pressure necrosis of the weakest part that is bladder dome and bladder rupture. Other factors include large baby weight, prolonged duration of the second stage, history of previous pelvic surgery, bladder diverticula and neuropathic bladder. Bladder rupture is likely during labor if it is not emptied before instrumentation. If the rupture occurs in spite of regular drainage; it is likely to have occurred postpartum. Unnoticed urinary retention or incomplete evacuation during puerperium due to pain of perineal repair causing retention may lead to subsequent bladder rupture.[6] Bladder rupture in our case is most likely to have occurred postpartum since the patient gave history of being catheterized before forceps application and had no urinary complaints at discharge. We feel that our patient had a neuropathic bladder which must have overdistended again on discharge and ruptured spontaneously at some point at home. Already her urine had infection (Klebseilla had grown) and on Day 20 pyuria was also seen suggestive of infection. Patients usually present with anuria/ oliguria/ haematuria and abdominal pain. Urinary ascites is not seen as frequently because there is rapid absorption of urine from peritoneal cavity.
The choice of imaging is CT cystogram. Inadequate filling with dye, obstruction by omentum, clot or surrounding organ could temporarily block small bladder rent leading to false negative CECT.[7] High index of suspicion and prompt intervention are required if a patient presents post-delivery with acute urinary retention and ascites.

Conclusion

Presence of acute renal failure, bowel ileus and new onset ascites in a postpartum patient with uneventful immediate postpartum course should always raise a suspicion of bladder rupture and prompt the treating physician to confirm the diagnosis. One should be cautious before incriminating the instrumentation for the same.

References
  1. Hubena Z, Workneh A, Siraneh Y. Prevalence and Outcome of Operative Vaginal Delivery among Mothers Who Gave Birth at Jimma University Medical Center, Southwest Ethiopia. Journal of Pregnancy. 2018; Article ID 7423475:12 pages. Available from: https://www.hindawi.com/journals/jp/2018/7423475/
  2. RCOG Green–top Guideline No. 26. Operative Vaginal Delivery. 2011; pages 2-19.  Available from: https://www.rcog.org.uk/globalassets/documents/guidelines/gtg_26.pdf
  3. Kibel AS, Staskin DR, Grigoriev VE. Intraperitoneal bladder rupture after normal vaginal delivery. J Urol. 1995;153(3 Pt 1):725-7.
  4. Baheti VH, Wagaskar VG, Patwardhan SK. Missed Iatrogenic Bladder Rupture Following Normal Vaginal Delivery. J Clin Diagn Res. 2015;9(10):PD01-2.
  5. Sawalmeh H, Al-Ozaibi L, Hussein A, Al-Badri F. Spontaneous rupture of the urinary bladder (SRUB); A case report and review of literature. Int J Surg Case Rep. 2015;16:116-8.
  6. Png KS, Chong YL, Ng CK. Two cases of intraperitoneal bladder rupture following vaginal delivery. Singapore Med J. 2008;49(11):e327–29.
  7. Qiao P, Tian D, Bao Q. Delayed diagnosis of spontaneous bladder rupture: a rare case report. BMC Womens Health. 2018;18:124.
Citation

Malani K, Samant PY, Thakur H. Postpartum Urinary Bladder Rupture. JPGO 2019. Vol 6 No. 5. Available from: https://www.jpgo.org/2019/05/postpartum-urinary-bladder-rupture.html

A Rare Case Of Sjogren's Syndrome In Pregnancy And Perinatal Outcome

Author Information

Akalyaa K*, Sreelatha S**, Kusuma M V*** ,Yashaswini  G *, Ramaiah R**.
(* Junior Resident, ** Professor, *** Assistant Professor. Department of Obstetrics and Gynecology, ESIMC PGIMSR, Bangalore, India.)
                 
Abstract

Sjogren's syndrome is an autoimmune disease with a high prevalence of anti SSA (anti Ro) and anti SSB (anti la) antibodies. Pregnancy associated with such disorders have a very high clinical impact on both the mother as well as the fetus. Sjogren's syndrome is one of the most common autoimmune disease with a prevalence between 0.1 and 4.8 % affecting women in the fourth decade of life.

Introduction

Sjogren’s syndrome (SS) is an autoimmune disease and is characterized by lymphocytic infiltration of lacrimal and salivary glands resulting in sicca symptoms. It can present either alone, that is primary Sjogren's syndrome (pSS), or can be associated with an underlying connective tissue disease, most commonly rheumatoid arthritis or systemic lupus erythematosus which is called as secondary Sjogren's syndrome. Sjogren's syndrome manifests from mild glandular manifestations to severe extra glandular disease like vasculitis, neuropathy and glomerulonephritis. The effect of autoimmune disease on pregnancy differs according to the maternal disease activity, severity of organ damage, antibody profile, and drug treatment.[1] Pregnancy complications due to the autoantibodies like anti-Ro/SSA and anti-La/SSB in the maternal serum causes neonatal lupus and congenital heart block (CHB), intrauterine growth restriction and low birth weight babies. The incidence of neonatal lupus in an offspring born to a mother with anti-Ro/SSA antibodies is 12 % , but it can be as high as more than 20 % if the mother has given birth to a child with neonatal lupus or CHB before. The diagnosis of Sjogren's syndrome is challenging due to varied presentation of symptoms and thus large number of patients remain undiagnosed. Therefore preconceptional counseling is very important in such patients.

Case Report

An unbooked primigravida with term gestation and with IUGR was admitted to our hospital  with labor pains. On examination she was normotensive, pulse rate was normal, with no pallor or pedal edema. Uterus was 32 weeks in size with IUGR.  Uterus was contracting with frequency of 1 to 2 contractions in 10 minutes, each lasting for 20 seconds. Presentation was cephalic and fetal heart rate was 140 beats per minute. On per vaginal exam, cervix was soft, 1 cm dilated, bag of membranes were present, and vertex was at -3 station. She underwent an emergency cesarean section in view of non reassuring NST. She delivered a single, live, male baby with birth weight of 1.8 kg. On postoperative day 2, she developed purpuric rashes over the lower limbs and deviation of angle of mouth to right side with left sided facial palsy for which she was evaluated and was found to have Primary Sjogren's syndrome as ANA profile with anti Ro and anti la antibodies were positive. Her ECG and 2D Echo were normal. Her rashes subsided by post operative day 7 and the facial palsy improved with physiotherapy. She was stable and was discharged on day 10.

Discussion

Sjogren's syndrome is the second most common autoimmune disease after rheumatoid arthritis, with the prevalence between 0.1 % and 4.8 % in various group of populations.[1] It occurs at any age but mainly affects women in the fourth decade of life.[2] The prevailing recent social trend toward late marriages and advanced maternal age increases the disease impact on pregnancy. Patient may present with generalized symptoms such as fatigue, dry mouth and eyes.[3] Sjogren's syndrome may present with extra glandular features such as isolated leucopenia, urticaria, irritable bowel syndrome, vasculitis, arthralgia, chronic non-deforming non-erosive polyarthritis, psychiatric symptoms, bladder symptoms, auditory,  vestibular and dental problems. Vascular manifestations are palpable purpura, erythematous rash, urticaria, photosensitivity, erythema nodosum, Raynaud’s phenomenon and gangrene. Neurological manifestations are mononeuritis multiplex, symmetrical polyneuropathy, carpal tunnel syndrome, trigeminal neuropathy, recurrent seizures, aseptic meningitis, and chronic immune demyelinating polyneuropathy.[4] Marker’s used to diagnose Sjogren's syndrome are antinuclear antibodies (most frequent), anti SSA (anti-Ro; most specific), anti SSB (anti-La), and cryoglobulins and hypocomplementemia (prognostic marker).[5] These antibodies cross the placenta by the beginning of 12 weeks of gestation and cause fetal cellular damage by macrophage activity, myocarditis and arrhythmia.[6] Fetal surveillance is done by serial echocardiograms and obstetric sonograms between 16 to 20 weeks of gestation. The goals of management includes early diagnosis and treatment of congenital heart block to improve the fetal outcome.[7] The pregnancy outcome is excellent with the use of antenatal corticosteriods such as dexamethasone or betamethasone, which can reduce the antibody-mediated inflammatory damage of nodal tissue.[8] Dexamethasone reverses carditis and incomplete heart block and also improves fetal hemodynamics and hence it is recommended in recent or incomplete heart block, or in cardiac failure.[9]

Conclusion

Women with Sjogren's syndrome must undergo prenatal counseling explaining all the risks and complications, medications that are contraindicated in pregnancy, whether the patient is in the best condition to get pregnant according to underlying disease activity and the need to control the disease well before conception. These high-risk pregnancies can be optimally managed by a multidisciplinary approach involving a high-risk obstetrician, a rheumatologist, and a pediatrician. High degree of suspicion and proactive approach can pick up the disease at the earliest.

References
  1. Gupta S, Gupta N. Sjögren syndrome and pregnancy: A literature review. Perm J 2017;21:16-047
  2. Hussein SZ, Jacobsson LT, Lindquist PG, Theander E. Pregnancy and fetal outcome in women with primary Sjogren's syndrome compared with women in the general population: a nested case-control study. Rheumatology (Oxford). 2011;50(9):1612-7.
  3. Goules AV, Skopouli FN. Prognostic factors and survival. In: Ramos-Casals M, Stone JH, Moutsopoulos HM, editors. Sjögren’s syndrome: diagnosis and therapeutics. 1st ed. London, United Kingdom: Springer 2012; pp. 129-48.
  4. Pulukool S, Jeyaseelan L, Scofield RH,  Danda D. Clinical Characteristics and Outcome of Primary Sjogren’s Syndrome: A Large Asian Indian Cohort. Open Rheumatol J. 2015; 9: 36–45.
  5. Brito-Zerón P, Ramos-Casals M, Bove A, Sentis J, Font J. Predicting adverse outcomes in primary Sjogren’s syndrome: identification of prognostic factors. Rheumatology (Oxford) 2007;46(8):1359-62.
  6. Brucato A, Cimaz R, Caporali R, Ramoni V, Buyon J. Pregnancy outcomes in patients with autoimmune diseases and anti-Ro/SSA antibodies. Clin Rev Allergy Immunol. 2011;40(1):27-41.
  7. Rosenthal D, Druzin M, Chin C, Dubin A. A new therapeutic approach to the fetus with congenital complete heart block: preemptive, targeted therapy with dexamethasone. Obstet Gynecol 1998;92(4 Pt 2):689-91.
  8. Saleeb S, Copel J, Friedman D, Buyon JP. Comparison of treatment with fluorinated glucocorticoids to the natural history of autoantibody-associated congenital heart block: retrospective review of the research registry for neonatal lupus. Arthritis Rheum. 1999;42(11):2335-45.
  9. Buyon J,  Roubey R,  Swersky S,  Pompeo L,  Parke A,  Baxi L, et al. Complete congenital heart block: risk of occurrence and therapeutic approach to prevention. J Rheumatol. 1988;15(7):1104-8.
Citation

Akalyaa K, Sreelatha S, Kusuma M V, Yashaswini  G, Ramaiah R. A Rare Case Of Sjogren's Syndrome In Pregnancy And Perinatal Outcome. JPGO 2019. Vol 6 No. 5. Available from:  https://www.jpgo.org/2019/05/a-rare-case-of-sjogrens-syndrome-in.html

Malignant Ovarian Tumor In A Teenage Girl

Author Information

Bathija S*, Hatkar P**.
(*First year resident, ** Associate Professor, Department of Obstetrics and Gynecology, Seth G S Medical College & K E M Hospital, Mumbai, India.)
Abstract
Ovarian mucinous tumors in adolescence are rare with less than fifty cases reported in literature. Majority of them belong to the benign or borderline epithelial tumor type with only twelve cases of cystadenocarcinoma reported till date. Here we discuss a case of a 17 year old girl admitted to our hospital with pain and distension of abdomen. She underwent ultrasonography (USG) and computed tomography (CT) of the pelvis. Both modalities revealed a large ovarian cyst. The management of such cases is tricky as fertility sparing approach is desired. However, left salpingo-oopherectomy was done in view of suspected ovarian mucinous cystadenocarcinoma on frozen section. The case is presented for its rarity and unique presentation.
Introduction
Epithelial ovarian tumors are not common in childhood and adolescence. They comprise 15-20 % of cases with malignant lesions being rare in this age group. Ovarian mucinous carcinomas have a distinct biological behaviour and clinical course compared with serous carcinoma subtypes.[1] Primary ovarian mucinous carcinoma is much less common than previously thought (6-25 %) and probably accounts for only 2.4 % of ovarian mucinous  cancers.[2,3] The rest are reclassified as mucinous intraepithelial carcinoma, borderline tumor or metastatic disease from a non-ovarian origin (stomach, colon, pancreas, breast, appendix, uterine cervix).[4] Common features of primary ovarian mucinous carcinoma include young age at the time of diagnosis, well-differentiated histology (typically within one ovary) and rare extraovarian involvement.[1,5] All these clinicopathological characteristics suggest that the management of ovarian mucinous carcinoma should be different to that for ovarian serous carcinoma. However, more evidence is required to consolidate these findings. We present a rare case of a 17 year old female with primary mucinous ovarian cystadenocarcinoma.
Case Report

A 17 year old girl presented to the gynecology outpatient department with complaints of abdominal distension which was insidious in onset associated with lower abdominal pain for the past 3 months. There was no significant medical or surgical history. She had no complaints of weight loss, loss of appetite or menstrual complaints. On examination, her general condition was fair, she was thin built and weighed around 49 kg. Her general physical examination was unremarkable. On abdominal inspection, there was generalised distension and the skin over the abdomen was tense and shiny. On palpation, a cystic mass was felt arising from the pelvis and extending till the epigastrium and on both sides upto iliac fossae occupying the whole of the abdomen. The mass was firm in consistency with no appreciable solid areas. Per vaginal examination revealed a firm cystic mass with lower extent of the mass that caused forniceal fullness. Tumor markers were evaluated and showed elevated CA 125 level of 131 U/L. β-human chorionic gonadotropin, alpha feto protein, carcino embryonic antigen and lactate dehydrogenase were within normal limits. Mantoux test was negative. Complete hemogram, liver function test and coagulation profile were found normal. USG revealed right ovarian 25x15 cm unilocular cyst with one solid component measuring approximately 4x2 cm without vascularity. There was evidence of bilateral ureteric compression and bilateral hydronephrosis, right more than left with no ascites. Contrast enhanced CT scan of the abdomen and pelvis revealed a well-defined 25x5x23 cm cyst having few non-enhancing septa with foci of calcification within. Right ovary showed multiple follicles and left ovary was not seen separately. The mass was displacing the retroperitoneal structures posteriorly and hydronephrosis with hydroureter in upper one third of the right ureter was confirmed. There was no evidence of ascites or lymphadenopathy. Urinary bladder, visualised vessels, bones and sections of chest were normal. CT scan impression was that of a mature cystic teratoma of left ovary or a mesenteric cyst. Urological examination was done and DJ stenting of the right ureter was performed for the treatment of hydronephrosis. After obtaining a valid consent, she was posted for exploratory laparotomy with left ovarian cystectomy SOS left salphingo-oophorectomy. A vertical midline skin incision was taken. A left ovarian cyst measuring 25×20 cm was noted. [Fig. 1] Left fallopian tube was found stretched over the cyst. There was no evidence of ascites. The cyst was punctured inadvertently while opening the abdomen. There was minimal spillage of cyst fluid into the abdominal cavity. About six liters of mucinous fluid was aspirated from the cyst, which was sent for cytology examination for presence of malignant cells. The left ovarian complex cyst wall was excised and sent for frozen section. It showed presence of two solid areas both approximately 4x3cm. On the right side, a simple ovarian cyst approximately 3 cm in diameter was found. Right sided ovarian cystectomy and ovarian reconstruction was done. The cyst fluid was sent for cytology and cyst wall was sent for frozen section. Frozen section report of the left ovarian cyst showed atypical proliferative mucinous neoplasm suspicious of mucinous carcinoma and cytology report did not show malignant cells. Right ovarian cyst showed features of a benign cyst. Left salpingo-oophorectomy was done. There was no evident lymphadenopathy and uterus, right fallopian tube, pouch of Douglas, under surface of liver, omentum and peritoneum were grossly unremarkable. However, omental biopsy was done to rule out micrometastasis after noting frozen section report and counseling the parents and taking appropriate consent.
Final Histopathology report:
  1. Left ovarian cyst showed atypical proliferative mucinous neoplasm with intraepithelial carcinoma.
  2. Right ovarian cyst: benign nature of cyst.
  3. Omentum: mild lymphohistiocytic infiltrate. No evidence of metastasis.
This confirmed the diagnosis of mucinous cystadenocarcinoma of left ovary with no metastasis.


Fig.1. Left ovarian cyst.


Fig.2. Image after left salphingo-oophorectomy was done.

Discussion
Mucinous ovarian tumors are rare in children less than 15 years of age with less than fifty cases reported in world literature. [6,7,8] Majority of these are benign or borderline tumors with frequency of carcinoma still rarer with only twelve cases reported. However, distinction between primary and metastatic tumors is vital to this age group since the prognosis and management of these tumors vary drastically. This distinction can be attained by a combined analysis of pathological and clinical features but it may be problematic if the extraovarian primary carcinoma is inconspicuous. Consequently, a considerable proportion of ovarian metastatic mucinous carcinomas are misdiagnosed as primary tumors.[9] Accurate diagnosis therefore becomes a precondition for the optional treatment of primary ovarian mucinous carcinoma. A majority of these patients present with vague symptoms that are initially ignored by them. This often leads to advancement of disease before the lesion gets diagnosed.  Furthermore, it is believed that childhood tumors are far more aggressive than their adult counterparts and progress to advanced disease despite treatment.[7]
Tumor markers and radiology in collaboration serve as a vital tool in the diagnosis of ovarian cancers.[10] CA 125 has been widely used as a marker for epithelial ovarian tumors however its utility is debatable. Although elevated serum CA 125 levels (>35 U/mL) have been found in more than 90% of advanced ovarian cancer patients, only 50% of patients with stage I disease have elevated levels.[11] Furthermore, CA 125 is also raised in approximately 1% of healthy individuals, in patients of liver cirrhosis, endometriosis, first-trimester pregnancy, pelvic inflammatory disease, pancreatitis and in other malignancies of lung, breast and pancreas.[12] Therefore, its raised levels must always be interpreted with caution and in conjunction with symptomatology and radiology. It is believed that if levels are initially raised at the time of detection they can be used as a marker for identifying residual or recurrent disease later at follow up.[8] Sometimes huge masses can create confusion as to the exact source of origin even on radiology. In this case too, the huge intra-abdominal mass on USG seemed to have originated from the right side, but a contrast enhanced CT suggested it to be a left ovarian cyst or mesenteric cyst. Elevated CA 125 levels in adjunct with the computed tomography scan report lead us to believe that it was an ovarian mass probably epithelial in origin and exploratory laparotomy was performed. The intraoperative examination of the other ovary and surrounding structures is crucial as it can often lead to escalation of the stage of the tumor if macroscopic deposits are visualized during surgery. Though the adult staging protocols dictate mandatory lymph node dissections and biopsies of peritoneal surfaces; these procedures are often omitted in pediatric and adolescent cases unless gross metastatic disease is present. Some surgeons perform a prophylactic wedge biopsy of the uninvolved ovary at the time of initial debulking surgery or biopsy from grossly suspicious foci [3]; but in our patient, the uterus, right fallopian tube, pouch of Douglas were unremarkable and the right ovary was grossly normal with a cyst of approximately 3 cm diameter. Thus, only left ovarian cyst wall and right ovarian cyst wall were sent for frozen section and no additional tissue samples were sent. When compared with primary ovarian serous carcinoma, primary ovarian mucinous carcinoma was more commonly associated with younger age, unilateral involvement of the ovary, unilateral tumour size >10 cm, less-advanced FIGO tumour stage (83% are stage 1), lower histological grade, higher completion rate of optimal primary surgery, more frequent use of unilateral salpingo-oophorectomy, lower volume of ascites and lower preoperative CA125 levels.[1] Our patient too had similar findings. Since on CT scan and intraoperatively no evidence of metastasis was seen unilateral salpingo-oopherectomy with omental biopsy was performed in view of frozen section report suggestive of mucinous cystadenocarcinoma.
The diagnostic standard for distinguishing mucinous carcinoma from borderline tumours lies in the presence of stromal invasion. Two invasive patterns, ‘destructive’ and ‘expansile’, have been well recognized in primary ovarian carcinoma. An ‘expansile’ pattern is the most common pattern in primary ovarian mucinous carcinoma whereas a ‘destructive’ pattern is less common and should raise the concern for metastatic carcinoma. [10,13] However, it can often be challenging to report a carcinoma in a minor age group without definitive infiltration into the stroma, rendering an equivocal report as in this case. For stage I epithelial cancers in adults, surgery alone is adequate. Here surgery is used for both staging and therapeutic purposes. Surgery includes total abdominal hysterectomy, bilateral salpingo-oophorectomy and omentectomy along with tumor debulking or cytoreduction. Fertility sparing surgery may be preferred in young patients inspite of possible metastasis since fertility is an important issue.[1] Furthermore, some studies have compared fertility sparing surgery with radical surgery in borderline ovarian tumors.[14] In these studies, though the recurrence rate was slightly higher in the fertility sparing group as compared to radical group, these recurrences could be managed by subsequent surgeries. A study done by Aggarwal et al. pointed out that low malignant recurrences have been reported more than ten years after initial surgery even in an adult patient population.[15] Therefore, like in adults young patients should also be kept under close follow up to monitor recurrence which should be treated with another salvage surgery.
Several adjuvant regimens ranging from single agent to multi agent regime have been tried for the treatment of malignant ovarian neoplasms. Our patient was referred to an oncology institute for further follow up and management. In conclusion, prognosis of ovarian cancers presenting at younger age remains variable and depends on the stage of presentation. Most of the cases of metastatic variant reported in the literature have had bad prognosis with almost all the patients dying within five years of detection of the lesion. In the mucinous variety, primary mucinous carcinomas have a favourable prognosis though it is less common than the metastatic variant. Although there are a no clear indications for ovarian cancer screening, testing is recommended for women at high risk such as those with a significant family history, but consultation with a genetic counsellor is recommended to discuss limitations and alternatives to genetic testing. Potential screening tests include for CA-125, other tumor markers and USG. However, these tests are neither sensitive nor specific for ovarian cancer. Since signs and symptoms are vague, patient often presents late and at an advanced stage, management of these cases differ from their adult counterparts. Instead of radical surgery, fertility sparing conservative approach should be preferred in these cases.

References
  1. Brown J, Frumovitz M. Mucinous Tumors of the Ovary: Current Thoughts on Diagnosis and Management. Curr Oncol Rep. 2014 Jun;16(6):389.
  2. Seidman JD, Kurman RJ, Ronnett BM. Primary and metastatic mucinous adenocarcinoma in the ovaries: incidence in routine practice with a new approach to improve intraoperative diagnosis. Am J Surg Pathol. 2003;27:985–93.
  3. Morowitz M, Huff D, von Allmen D. Epithelial ovarian tumors in children: a retrospective analysis. J Pediatr Surg. 2003;38:331–35.
  4. Shimada M, Kigawa J, Ohishi Y, et al. Clinicopathological characteristics of mucinous adenocarcinoma of the ovary. Gynecol Oncol. 2009;113:331–4.
  5. Tian Q, Lu B, Ye J, Lu W, Xie X, Wang X. Early stage primary ovarian mucinous carcinoma: Outcome-based clinicopathological study in comparison with serous carcinoma. Journal of International Medical Research. 2016;44:357-366.
  6. Horiuchi A, Kameoka K, Sato K, Yamamoto Y, Watanabe Y. Huge mucinous borderline ovarian cystadenoma in a premenarchal girl. Open J Pediatrics. 2012;2: 82–86.
  7. Shankar KR, Wakhlu A, Kokai GK, McDowell H, Jones MO. Ovarian adenocarcinoma in premenarchal girls. J Pediatr Surg. 2001;36:511–15.
  8. Iwasaki M, Taira K, Kobayashi H, Saiga T. Ovarian mucinous cystadenoma of borderline malignancy in a premenarchal girl. J Pediatr Adolesc Gynecol. 2010;23(3):e119–e123.
  9. Leen, SL, Singh, N. Pathology of primary and metastatic mucinous ovarian neoplasms. J Clin Pathol 2012;65:591–95.
  10. Lee, KR, Young, RH. The distinction between primary and metastatic mucinous carcinomas of the ovary: gross and histologic findings in 50 cases. Am J Surg Pathol 2003;27:281–92.
  11. Nustad K, Bast RC Jr, Brien TJ, Nilsson O, Seguin P, Suresh MR et al. Specificity and affinity of 26 monoclonal antibodies against the CA 125 antigen: first report from the ISOBM TD-1 workshop. International Society for Oncodevelopmental Biology and Medicine. Tumour Biol. 1996;17(4):196–219.
  12. Bast RC Jr, Klug TL, St John E, Jenison E, Niloff JM, Lazarus H et al. A radioimmunoassay using a monoclonal antibody to monitor the course of epithelial ovarian cancer. N Engl J Med. 1983 Oct 13;309(15):883-87.
  13. Tabrizi, AD, Kalloger SE, Köbel M, Cipollone J, Roskelley CD, Mehl E et al. Primary ovarian mucinous carcinoma of intestinal type: significance of pattern of invasion and immunohistochemical expression profile in a series of 31 cases. Int J Gynecol Pathol 2010 Mar; 29(2):99–107.
  14. Park JY, Kim DY, Kim JH, Kim YM, Kim YT, Nam JH. Surgical management of borderline ovarian tumors: the role of fertility-sparing surgery. Gynecol Oncol. 2009;113:75–82.
  15. Aggarwal A, Lucco KL, Lacy J, Kives S, Gerstle JT, Allen L. Ovarian epithelial tumors of low malignant potential: a case series of 5 adolescent patients. J Pediatr Surg. 2009;44:2023–27.
Citation

Bathija S, Hatkar P. Malignant Ovarian Tumor In A Teenage Girl. JPGO 2019. Volume 6 No.5. Available from: https://www.jpgo.org/2019/05/malignant-ovarian-tumor-in-teenage-girl.html

Surgical Adhesive Dressing induced Allergic Contact Dermatitis

Image
Author Information

Srinivasan N*,  Desai G**,  Hatkar P***.
(* Junior resident, ** Assistant Professor, *** Associate Professor, Department of Obstetrics & Gynecology, Seth G. S. Medical College and K. E. M. Hospital, Mumbai, India).

Abstract

Allergic contact dermatitis is a variety of dermatitis or eczematous reaction caused by an allergen when it comes in contact with skin. The allergen otherwise does not cause any harm to people who are not allergic to it. We present a case of a young post operative woman developing allergic contact dermatitis to adhesive surgical dressing.

Introduction

Allergic contact dermatitis is common skin condition amongst general population. Women are more likely to suffer from allergic contact dermatitis. Most common ones are due to nickel and acrylate allergy. Allergic contact dermatitis is a common occupational disease especially seen in metal workers, hair dressers, beauticians, health care workers, painters and flower decorators.[1] Patients with improper first line defence of immunity like leg ulcers, perianal dermatitis, chronic irritant contact dermatitis, systemic illnesses like diabetes, autoimmune diseases are more prone to develop allergic contact dermatitis.
Allergic contact dermatitis is also seen in people suffering from atopic dermatitis as they lack filaggrin, a structural protein in stratum corneum.

Case Report

A 22 year old primipara on day 3 of emergency cesarean delivery presented with multiple, well demarcated, reddish maculo-papular vesicles filled with clear transparent fluid and plaques with skin excoriations and fine scaling on the dorsum of her left hand and similar lesions in the lower abdomen, which were the sites of application of adhesive surgical dressings of intravenous access and cesarean wound respectively. The shape of areas of distribution of such lesions corresponded exactly to the shape of the adhesive dressing used. The lesions were intensely pruritic. Multiple such skin excoriations were found on the face and elbows. She gives past history of generalized reddish skin eruptions that cropped up suddenly 2 years back, but there was no history of any treatment or medications taken for the same.
Dermatology opinion was sort. A diagnosis of allergic contact dermatitis to adhesive surgical dressing was made. Aseptic puncturing and aspiration of fluid from the bigger vesicles was advised. She was started on topical fluocinolone acetonide and mupirocin ointments, tablet levocetrizine OD, and augmented betamethasone dipropionate scalp solution. Avoidance of adhesive surgical dressing was advised. She was encouraged to wear loose fitting clothes of 100% natural fabrics, and to wash her clothes multiple times prior to use.

Figure 1. Contact dermatitis on the dorsum of the left hand where adhesive dressing for securing iv access was present; before start of treatment.

Figure 2. Contact dermatitis on the dorsum of the left hand in phases of healing, where adhesive dressing for securing iv access was present; on treatment.

Figure 3. Contact dermatitis on the anterior abdominal wall where adhesive dressing for surgical site was present; before start of treatment.

Figure 4. Contact dermatitis on the anterior abdominal wall in phases of healing, where adhesive dressing for surgical site was present; on treatment.

Discussion

Allergic contact dermatitis is a type 4 hypersensitivity reaction or delayed hypersensitivity that is caused by an allergen when it comes in contact with skin. It  manifests 48-72 hours after exposure to the allergen. It is mediated by CD4+ T lymphocytes which recognize the antigen on skin surface and respond by releasing cytokines that stimulate the immune system and cause allergic reaction.[2] Allergic contact dermatitis on histopathology shows psoriasiform epidermal hyperplasia with spongiosis and parakeratosis with perivasular lymphocytic infiltration. Microscopic evaluation of the lesions in our case was not necessary as a confirmatory diagnosis was made on its gross appearance itself.

Conclusion

Allergic contact dermatitis is treated usually by glucocorticoids, avoidance of offending allergen sources.[3] Prognosis is dependent on patient education, compliance in avoiding allergens and appropriate skin care.

References
  1. Hatch KL, Maibach HI. Textile dye dermatitis. J Am Acad Dermatol 1995;32(4):631-639.
  2. Lazarov A, Trattner A, David M, Ingber A. Symptoms and signs reported during patch testing. Am J Contact Dermat 2000;11(1):26-9.
  3. Belsito DV. The diagnostic evaluation, treatment, and prevention of allergic contact dermatitis in the new millennium. J Allergy Clin Immunol. 2000;105(3):409–20.
Citation

Srinivasan N,  Desai G,  Hatkar P.  Surgical Adhesive Dressing induced Allergic Contact Dermatitis. JPGO 2019. Vol 6 No. 5. Available from: https://www.jpgo.org/2019/05/remembering-past-greats-ingemar.html

Remembering Past Greats: Ingemar Ingemarsson

Author Information

Prasad M
(Assistant Professor, Department of Obstetrics and Gynecology, Vydehi Institute of Medical Sciences and Research Centre, Whitefield, Bangalore, India.)

Ingemar Ingemarsson was a Swedish obstetrician and gynecologist who passed away few years back. Born and brought up in Southern Sweden and educated in the University of Lund, Ingemarsson became faculty in the department of obstetrics and gynecology in the same institute.[1] In the 1970s, he is credited to have revolutionized the way Sweden’s hospitals performed with respect to maternity care. It is noted that the first cardiotocograph was introduced under his supervision, by the encouragement of his teacher, Prof. Lars Bengston. His main areas of interest included preterm birth and fetal surveillance during labor. A simple PubMed search of the name Ingemarsson reveals that he had published extensively from early 1980s to 2005, over varying topics in obstetrics. The impact of his research has been significant. Though now losing prominence, terbutaline was a well established tocolytic agent for over three decades. However, it can also be used in birth asphyxia.[2] One of the main research projects which established terbutaline for this use was Ingemarsson’s thesis project done in coordination with a pharmacologist of his institute. Most standard books carrying chapters on tocolytic therapy regularly quoted many of Ingemarsson’s publications. In 1993, his book titled ‘Fetal Heart Rate Monitoring: A practical guide’ was rather famous and guided obstetricians in the correct usage and interpretation of the cardiotocograph.
He was a guide and mentor to scores of students and he was respected and revered by all. In the late 1990s and 2000s, he was a regularly invited speaker in perinatal meets all over the world. He particularly coordinated with institutions in Singapore as a visiting consultant. He visited India in 2004 and lectured at many institutions. Honoring the yeoman service rendered by Ingemarsson, the Nordic society of obstetrics and gynecology conducted a symposium on preterm birth in the FIGO world congress held in 2015.[3] Ingemarsson’s life and work shall remain an inspiration for obstetricians world over.

References

Montan S, Arulkumaran S. Tribute to a great obstetrician: in memory of Ingemar Ingemarsson. Acta Obstet Gynecol Scand. 2015 Mar;94(3):333-5.
Ingemarsson I, Arulkumaran S, Ratnam SS. Single injection of terbutaline in term labor. I. Effect on fetal pH in cases with prolonged bradycardia. Am J Obstet Gynecol. 1985 Dec 15;153(8):859-65.
Jørgensen JS, Jacobsson B, Vinter CA, Lamont RF, Maršál K. Ingemar Ingemarsson Memorial Symposium on preterm delivery at the XXI FIGO World Congress. Acta Obstet Gynecol Scand. 2016 May;95(5):495-500.

Citation

Prasad M. Remembering Past Greats: Ingemar Ingemarsson. JPGO 2019. Volume 6 No.5. Available from: https://www.jpgo.org/2019/05/remembering-past-greats-ingemar.html

Volume 6 Number 4

Editorial
Parulekar SV

A Rare Case Of A Large Asymptomatic Cervical Leiomyoma
Mathews N, Parulekar SV.

An Unusual Case Of A Large Leiomyoma Complicating Pregnancy
Joshi S, Parulekar SV.

Absence Of Ipsilateral Adnexa And Its Implications
Rane P, Samant P.

Torsion Of Hematosalpinx In A Case Of Unicornuate Uterus
Jha N, Chaudhari HK, More V.

Subserosal Fibroid Misdiagnosed As Ovarian Mass In Pregnancy: Even MRI Is Not 100% Diagnostic
Verma ML, Sachan R, Singh A, Kumari M, Singh U, Sankhwar PL.

Vaginal Pull Through Procedure To Treat Isolated Lower One Third Vaginal Agenesis
Saxena A, Pardeshi S, Gupta AS.

Alloimmunisation With Rare Blood Group In Twin Pregnancy
Singhania N, Vaidya A, Gupta AS.

Successful Management Of Essential Thrombocytosis In Pregnancy
Tagad M, Tiwari N, Chaudhari HK.

Remembering Past Greats: Grigoris Lambrakis
Prasad M, Venkatesh S.

Editorial

Parulekar SV

Obesity during pregnancy is on the rise, and so are the conditions that go with obesity. Obstructive sleep apnea (OSA)  is one such condition. It is characterized by repetitive episodes of obstruction of the upper airway during sleep, leading to absence or severe reduction in airflow despite respiratory effort. The gravida experiences hypoxemia and recurrent arousal from sleep. Its prevalence is directly proportional to maternal age and weight independent of each other. Physiological changes of pregnancy like reduced diameter of the oropharynx, nasal obstruction due to mucosal edema and increase in Mallampati grade lead to increased resistance of the upper airway and greater negative intra-pharyngeal pressure, which in turn lead to snoring and obstructed breathing in sleep. The increase in the circulating blood volume causes adverse effect on the upper airway function in recumbency (sleep) and risk of OSA. The true incidence of OSA in pregnancy is not known, but is probably much more than what is believed, because women tend to report it less often. Questionnaires are not found to be very useful to detect which gravidas suffer from it. An objective test is more likely to detect OSA. A full-night, attended, in-laboratory polysomnogram is the best method of diagnosing OSA. An unattended, home sleep apnea testing is more convenient, but is not cleared for use in pregnancy by American Academy of Sleep Medicine. When apnea hypopnea index (average number of apneas plus hypopneas per hour slept) is 5 to 15, 15-30, and more than 30 events per hour, OSA is said to be mild, moderate and severe respectively. It must be kept in mind that OSA is not the only cause of disturbance of sleep in pregnancy, and there are causes related to pregnancy, e.g. leg cramps, fetal movements, urge for urination and dyspepsia. If OSA is not treated, risk of complications like fatigue, excessive daytime sleepiness, lack of attention, systemic hypertension, cerebrovascular disease, cardiac arrhythmias, ischemic heart disease, cardiomyopathy, pulmonary embolism,  insulin resistance, gestational diabetes, preeclampsia, gestational hypertension, fetal growth restricition, effect on neurological growth, preterm labor and fetal death. Pregnancy may exacerbate OSA. Thus treating OSA during pregnancy is important. All gravidas with moderate or severe disease are treated. The treatment includes use of oral mandibular repositioning appliances, continuous positive airway pressure and behavior modification. Modafinil, which is used to treat OSA, is a category C drug. It is also found in the breast milk. Hence its use is not recommended in pregnancy and lactation.  Upper airway surgery like uvulopalatopharyngoplasty is not recommended in pregnancy. Complications listed above need to be watched for and managed aggressively. Continuous pulse oximetry is used in labor and after childbirth. Any hypoxemia is evaluated and treated energetically, maintaining oxygen saturation at least 96%. Regional anesthesia for labor pain management is preferred to opioids. As the awareness of this condition during pregnancy increases, more and more cases will come to light. We hope to create this awareness in our readers through this editorial.