Volume 5 Number 11

Editorial

Gupta AS

We are living in an age surrounded with technology and bombarded from all sides with information. In this digital world the public, medical fraternity can all access information from the world wide web. All information does not add to the knowledge. Knowledge should be such that it brings about a change in the attitudes and translates into day today practices so that society benefits.

The need to update the knowledge, skills, attitudes, professional competence and practices of a medical practitioner forms the basis of recommending continuous medical education for medical practitioners. Various evidence based studies have demonstrated decay of knowledge after a medical practitioner leaves medical school. Self motivation rarely is adequate in this hectic life to enable a medical practitioner to keep in touch and educate self with all recent developments in their field. Continuous medical education or CME is a concept which includes active, continuous life long learning by medical fraternity while in practice to gain, update, retain knowledge, acquire and maintain necessary skills in order to give good standard medical care to patients.

Various countries have tried to force the hand of their medical practitioners by insisting on periodic re registrations; renewal of their professional registrations by earning credit hours which then make them eligible for re registrations or renewal of their registrations. However, such compulsory credit hours linked points, legal bindings are not able to reach the goal of true continuous medical education and updating of the skills as these restrict the type of education activities that generate credit hours. The CME programs should be well designed for all types of medical practitioners to enable every practitioner to develop professionally. Proper guidelines for structured learning, both online and offline modules, interactive teaching and learning methodology with active participation, self study, publications, research, attending and learning on grand ward rounds, attending conferences, workshops, symposiums, learning through distance education modes and virtual teaching should be part of the bouquet that allows the medical practitioner to gain continuous medical education.

An alternative terminology to CME would be CPD (Continuous Professional Development) as well designed programs, tailor made for different health professionals will induce behavioral changes, multidisciplinary approach awareness, refinement of their medical, social  and management skills. The challenge to design a well structured CME/CPD program should be well thought, have a wide scope, have clear laid down guidelines and standard operating procedures and a monitoring system that ensures the gainful impact of the program. The program should not get reduced to just obtaining the required credit hours by any means only for the purpose of re-registration. These programs should also be trimmed to curtail waste of time, money, distraction of the mind and must remain focused to its primary goal of providing continuous professional development and medical education or else it will be reduced to a mere formality and a burden to the medical fraternity by the regulating bodies for obtaining points or credit hours for renewing their registrations rather than its ultimate aim of keeping abreast with the recent developments in the medical field and ensuring continuous professional competence. Well designed tests for evaluating competence should be in place as a professional should have the option of maintaining his education, skills and competence through various modalities including self study.

We hope that the November issue of our journal will add to the education of the clinician.

A Rare Case Of Superior Mesenteric Vein Thrombosis In Pregnancy

Author Information
Toshniwal SA*, Chandak PO**, Deshmukh S***.
(*Toshniwal Chest Hospital & Maternity Home, Nanded, **Junior Resident, Department of Obstetrics and Gynecology, Dr Shankarrao Chavan GMC, Nanded, ***Intern, Dr Shankarrao Chavan GMC, Nanded, India.)
Abstract

Superior Mesenteric Venous Thrombosis (SMVT) is a rare entity. Its occurrence in pregnancy is an even rarer possibility. Here a case of a primigravida with term gestation with acute abdomen who underwent laparotomy for suspected ruptured uterus, but actually had ischemic bowel segment due to SMVT is presented. Resection and anastomosis of involved bowel segment, management with anticoagulation and an otherwise uneventful post operative period is described here.
Introduction

Small bowel ischemia is a relatively uncommon disorder and the causes are diverse. Due to vague clinical presentation and the lack of specific diagnostic tests, early diagnosis of intestinal ischemia is difficult, resulting in significant morbidity and mortality. We herein report a case of a woman who presented with signs and symptoms of acute abdomen at 38 weeks gestation. Pregnancy is a hypercoagulable state, and can cause ischemic phenomena. However, intestinal ischemia due to SMVT is rather rare.

Case Report

A 25 year old primigravida presented at 38 weeks gestation with complaints of pain in abdomen and three episodes of vomiting. Pulse was 92/ minute and blood pressure was 110/ 70 mm Hg.  Cardiovascular and respiratory system were unremarkable. Abdominal examination showed a single fetus in cephalic presentation with regular fetal hearts, and vaginal examination was suggestive of latent labor. Urine albumin was nil and preeclampsia was ruled out. Vomiting as a premonitory symptom of eclampsia was considered less likely. Oxytocin drip (2.5 units in 500 ml ringer lactate) was started at 8 drops/ min. Regular vaginal examination showed progress of labor. However, around 4 hours after starting oxytocin, she complained of severe abdominal pain, not related to uterine activity, and examination showed guarding, tenderness and rigidity. There were features of acute abdomen with shoulder tip pain. There was progressive breathlessness, tachycardia, hypotension and distension of abdomen, and there was sudden disappearance of fetal heart sound. Ultrasonography was done on emergency basis and fetal demise was confirmed. It also showed an intact uterus, dilated bowel loops and minimal free fluid in peritoneal cavity. The drop in blood pressure was such that inotropic support was required. Hematological and biochemical profile was largely normal. The only abnormal investigation was a WBC count of 18000/ mm3. The clinical impression was one of uterine rupture and a decision of emergency exploratory laparotomy was taken in view of her deteriorating general condition. Procedure was done under general anesthesia and the abdomen was opened by a Pfannenstiel incision. Intraoperative findings showed presence of 500 cc of serosanguinous ascitic fluid. Uterus was intact and a lower segment cesarean section was performed to deliver a fresh still born male baby of 2.5 kg and there was no evidence of any retro-placental clot. Surprisingly, there was a gangrenous small bowel segment of around 60 cm (figure 1).


Figure 1. Image showing gangrenous small bowel with green arrow pointing to normal segment.

General surgeon was called over.  Volvulus was ruled out and a resection of the gangrenous bowel segment and anastomosis was performed. No stoma was required. One unit whole blood was transfused intraoperatively. Intra-peritoneal drain was kept and abdomen was closed. Close postoperative management was required in surgical ICU, and ionotropic support was tapered and withdrawn on postoperative day 3.  She was kept nil by mouth and was started on oral sips on sixth postoperative day and bowel movement was smooth after that. She was given higher antibiotics and anticoagulant therapy with injection Heparin unfractionated 5000 IU 6 hourly for 5 days with warfarin overlap from day 3 with 2 mg tablet to maintain an International Normalized Ratio (INR) between 2 to 3. Sutures were removed on tenth postoperative day and she was discharged two days later, with advice of continuing anticoagulation till further advice.  Investigations to rule out other causes for thrombosis were advised, but could not be done due to non-affordability. Histopathology report was consistent with bowel ischemia, which showed necrotic changes with denudation of bowel mucosa with inflammatory changes with neutrophilic infiltration with edema all over.
Discussion
Superior Mesenteric Vein drains blood from jejunum and ileum, and amalgamate with the splenic vein to form the hepatic portal vein. Thrombosis of this vein is a rare entity. Around 5 to 15 % cases of mesenteric ischemia are due to SMVT. Lin et al have mentioned that only 17 cases of SMVT have been described in pregnancy.[1]
In general, the causes of SMVT include hypercoagulable states (protein C/S deficiency, Factor V Leiden mutation, antithrombin 3 deficiency),[2] polycythemia vera, neoplasms, abdominal trauma, intra-abdominal infections and autoimmune causes. No specific cause is found in upto 49 % cases. In our patient, causes of hypercoagulation could not be ascertained due to financial non-affordability. Use of oral contraceptive pills can enhance the chances of thrombosis.[3] In our patient, there was no history of using any hormonal contraception. Our patient presented at term. Cases have been reported at early gestation,[4] and at 8th month of gestation.[1]
SMVT can be acute or chronic. In chronic cases, there can be features of portal hypertension. In our patient, there was typical features of the acute presentation (severe pain in abdomen, distension, vomiting, bloating, fever, tachycardia and hypotension). The diagnosis of the condition in this patient was probably delayed due to simultaneous occurrence of labor at term. Initially, the features mimicked normal changes of labor. Only when fetal hearts disappeared, there was a suspicion of something more sinister. Exploratory laparotomy with resection and anastomosis is the optimal management, as was done in our case. Shui et al have described a surgical management, but reversible ischemia due to a volvulus. In that case, resection anastomosis was not required.[5]
Anticoagulant therapy is the most important step in management, immaterial of whether the case has been managed surgically or conservatively. Low molecular weight heparin in the doses of 0.6mg/kg or unfractionated heparin in the dose of 5000 IU 6 hourly followed by overlapping with warfarin to maintain INR between 2 to 3 is the treatment of choice. There are no defined guidelines for the duration of anticoagulation and the decision is to be made as per the case. Our patient is being continued with anticoagulation till date.
The decision on continuation or termination of pregnancy is taken as per the case and available facilities. In our case, it was a concomitant LSCS. Engelhardt had described onset of labor, resulting in vaginal birth soon after the surgical procedure.[6] Lin et al have also described concomitant LSCS. Postpartum cases have also been described.[1]

Conclusion

To conclude, we stress that in a pregnant woman with a clinical presentation of acute abdomen, even with the absence of the other known risk factors, superior mesenteric vein thrombosis should be kept in mind and included in the differential diagnosis to avoid potential complications.

References
  1. Lin H, Lin CC, Huang WT. Idiopathic superior mesenteric vein thrombosis resulting in small bowel ischemia in a pregnant woman. Case Rep Obstet Gynecol. 2011; 2011:687250.
  2. Atakan Al R, Borekci B, Ozturk G, Akcay MN, Kadanali S. Acute mesenteric venous thrombosis due to protein S deficiency in a pregnant woman. Journal of Obstetrics and Gynaecology Research. 2009; 35(4):804–7.
  3. Béliard A, Verreth L, Grandjean P. Oral contraceptive and acute intestinal ischemia with mesenteric venous thrombosis: a case report. Open Access J Contracept. 2017; 8:9-11.
  4. Giannos A, Stavrou S, Goumalatsos N, Fragkoulidis G, Chra E, Argiropoulos D, et al. Mesenteric cysts and mesenteric venous thrombosis leading to intestinal necrosis in pregnancy managed with laparotomy: a case report and review of the literature. J Med Case Rep. 2017; 11(1):184.
  5. Shui LH, Rafi J, Corder A, Mowbray D. Mid-gut volvulus and mesenteric vessel thrombosis in pregnancy: case report and literature review. Archives of Gynecology and Obstetrics. 2011; 283(S1):39–43.
  6. Engelhardt TC, Kerstein MD. Pregnancy and mesenteric venous thrombosis. Southern Medical Journal. 1989; 82(11):1441–3.
Citation

Toshniwal SA, Chandak PO, Deshmukh S. A Rare Case Of Superior Mesenteric Vein Thrombosis In Pregnancy. JPGO 2018. Volume 5 No.11. Available from: https://www.jpgo.org/2018/11/a-rare-case-of-superior-mesenteric-vein.html

Transperitoneal Passage Of Landon's Retractor During Vaginal Hysterectomy For Procidentia

Author Information

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

Abstract

Vaginal hysterectomy has to be performed in limited space and the anatomy is difficult to understand as compared to during an abdominal hysterectomy. The urinary bladder needs to be retracted forwards using a Landon's retractor and the rectum posteriorly using a vaginal speculum. This is possible only after the uterovesical and rectouterine peritoneal pouches have been opened. A curious case is presented in which the Landon's retractor was inadvertently passed in through the uterovesical peritoneal pouch and out through the rectouterine peritoneal pouch.

Introduction

The learning curve is quite steep for vaginal hysterectomy.[1,2] This is because the space available is limited and the anatomy is difficult to understand as compared to while performing an abdominal hysterectomy. Some of the instruments used are quite different than those used in conventional operations performed abdominally. It is necessary to retract the urinary bladder and the rectum away from the uterus so as to be able to operate. That can be done only when the uterovesical and rectouterine peritoneal pouches have been opened.[1,2] A case being operated on by a junior resident is presented in which the Landon's retractor was passed into the pelvic peritoneal cavity after opening the uterovesical peritoneal pouch, and out through the rectouterine peritoneal pouch which has already been opened. The reason for this occurrence is discussed. This is the first case of this type in the world literature.

Case Report

A 51 year old woman, para 4 living 4, presented to us with a complaint of something coming out per vaginum for 2 years. She had no urinary or rectal symptoms. There was no precipitating factor for genital prolapse. She had been menopausal for 5 years. Her past medical and surgical history was not contributory. Her general condition was fair and vital parameters within normal limits. Her general and systemic examination revealed no abnormality. She had a fourth degree uterine prolapse, large cystocele of central transverse type, a large rectocele of central transverse type, and deficient perineum. Her investigations for fitness for anesthesia showed normal results.

A vaginal hysterectomy with anterior colporrhaphy and posterior colpoperineorrhaphy was carried out under spinal anesthesia. The operation was being done by a junior resident, assisted by a junior consultant. After circumferential incision in the vaginal mucosa at the level of junction with the portio vaginalis, the rectouterine peritoneal pouch was opened and Soonawalla's speculum was passed between the uterus in front and the rectum behind. Uterine supports were weak and uterine corpus prolapsed out of the open rectouterine peritoneal pouch. Then the uterovesical peritoneal pouch was opened after dissecting the urinary bladder away from the front of the supravaginal cervix. Then a Landon's retractor was passed into the pelvic peritoneal cavity through the open uterovesical peritoneal pouch (figure 1). Then the cervix was held forward so that the prolapsed uterine corpus could be reposited before performing hysterectomy. That time it was noticed that the Landon's retractor had passed above the uterine fundus and out of the open rectouterine pouch, behind the prolapsed uterine corpus (figure 2). It was withdrawn, the uterine corpus was reposited and then the Landon's retractor was passed into the pelvic peritoneal cavity such that it did not pass out of the rectouterine pouch. The remaining part of the operation was carried out without any difficulty. The patient made an uneventful recovery.


Figure 1. Landon's retractor has been passed through open uterovesical peritoneal pouch. Prolapsed uterine corpus is shown by black arrow.


Figure 2. The blade of the Landon's retractor is seen to have exited through open rectouterine peritoneal pouch, behind the prolapsed uterine corpus.

Discussion

The space available in the lower pelvis for performing a vaginal hysterectomy is quite limited because the vaginal space is small and it is largely occupied by the uterus, rectum and urinary bladder.[3] The instruments used to retract the urinary bladder (Landon's retractor) and the rectum (Soonawalla's speculum) usually pass between the urinary bladder and uterus in case of Landon's retractor and between the uterus and the rectum in case of the Soonawalla's speculum. This is because these instruments are crowded in the limited space by the pelvic organs.[4,5] However in case of a massive uterovaginal prolapse, the urinary bladder, uterus and rectum are outside the pelvic outlet and the restraint of space does not apply. The levator hiatus is also likely to be very large due to separation of the two levatores from each other. The uterus of a postmenopausal woman is often small due to atrophy, which results from loss of estrogen. Such a small size of the uterus, combined with poor supports, tends to prolapse out through open rectouterine peritoneal pouch, as happened in the case presented. That increased the space available between the pelvic organs further. Owing to misdirection of the Landon's retractor during its passage between the urinary bladder and the uterus, the blade of the retractor passed more posteriorly, above the prolapsed uterine corpus and behind it, and then out through the open rectouterine peritoneal pouch.

The occurrence was not serious, because it could be corrected very easily, and no operative mishap could have occurred as further operative steps could not haven been taken without correcting its position first. It is reported only because it was quite curious. Its awareness should help junior surgeons in training, to operate better.

Acknowledment

I thank Dr. Sana Bijapur for taking operative photographs.

References
  1. Zimmerman CW. vaginal Hysterectomy. In: Jones HW, Rock JA editors. TeLinde's Operative Gynecology. 11th ed. Philadelphia: Wolters Kluwer  2014. p.716-737.
  2. Lentz GM. Anatomic defects of the abdominal wall and pelvic floor. In: Katz VL, Lentz GM, Lobo RA, Gershensen DM editors. Comprehensive Gynecology. Philadelphia: Elsevier 2016. p. 500-536.
  3. Brummer TH, Jalkanen J, Fraser J, Heikkinen AM, Kauko M, Mäkinen J, et al. FINHYST, a prospective study of 5279 hysterectomies: complications and their risk factors. Hum Reprod 2011;26:1741.
  4. Fatania K, Vithayathil M, Newbold P, Yoong W. Vaginal versus abdominal hysterectomy for the enlarged non-prolapsed uterus: a retrospective cohort study. Eur J Obstet Gynecol Reprod Biol 2014; 174:111.
  5. Johnson N, Barlow D, Lethaby A, Tavender E, Curr E, Garry R. Surgical approach to hysterectomy for benign gynaecological disease. Cochrane Database Syst Rev 2006;(19):CD003677.
Citation

Parulekar SV. Transperitoneal Passage Of Landon's Retractor During Vaginal Hysterectomy For Procidentia. JPGO 2018. Vol 5 No. 11. Available from: https://www.jpgo.org/2018/11/transperitoneal-passage-of-landons.html

Unilateral Fibrotic Atresia Of Fallopian Tube

Author Information

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

Abstract

A fallopian tube can be absent on one side congenitally, or due to resorption of affected tube after torsion. Partial absence may be due to resorption of an ectopic pregnancy in the tube after its rupture. Absence of a fallopian tube along with the ovary of the same side can be congenital, or due to necrosis after a torsion. An unusual case of congenital atresia of a fallopian tube is presented.

Introduction

A fallopian tube may be absent either alone or with ovary of the same side. Both of these conditions can be congenital or acquired due to a number of causes. Usually such an absence shows nothing in the place of the structure which is missing. An unusual case in which a fallopian tube was replaced in its entirety by a fibrous band congenitally is presented.

Case Report

A 23 year old woman, married for 4 years, presented for management of primary infertility. She had regular menses every 28 to 30 days, with moderate flow for 3-4 days and some pelvic pain. Her sexual history was normal. Her past medical and surgical history were not contributory. She had no episode of acute pelvic pain in the past. Her general and systemic examination revealed no abnormality. Her secondary sex characteristics and external genitals were normal. Abdominal examination showed normal findings. A speculum examination showed normal vagina and cervix. A bimanual pelvic examination showed an anteverted, normal sized, mobile uterus and no abnormality. Her husband's semen report was normal. Follicular study showed presence of spontaneous ovulation. Her investigations for fitness for anesthesia showed normal results.

Hysteroscopy and laparoscopy were performed on her under general anesthesia. Hysteroscopy showed normal cervical canal, uterine cavity, endometrium and tubal ostia. Laparoscopy showed normal uterus, ovaries, left fallopian tube, pelvic peritoneum and bowel. The right fallopian tube was replaced by a fibrous band 2-3 mm in diameter, extending from the right cornua to right lateral pelvic wall (figure 1). The patient made an uneventful recovery.


Figure 1. Laparoscopy findings: RUOL- right uteroovarian ligament, RFT – right fallopian tube (green arrows), RO – right ovary (white arrows).

Discussion

The first case of an absence of a fallopian tube in the world literature was reported by Alexander in 1947.[1] However it was a case of unilateral mesonephric and paramesonephric duct agenesis. There are several reports of unilateral absence of ovary and fallopian tube. Absence of an entire fallopian tube alone is extremely uncommon. Absence of a part of a fallopian tube is more common than total absence.[2, 3] The uterus is the most common site of congenital malformations of the female genital tract. The next common sites is the vagina. The fallopian tube alone is a rare site, though it can be absent in cases of unilateral mullerian agenesis, in which the uterus on the same side is also not developed (unicornuate uterus). Unilateral ovarian and fallopian tube agenesis is believed to occur in 1:11240 cases. [4] When both the ovary and fallopian tube are absent on one side, the most likely cause is torsion of the adnexal structures, the symptoms caused by which may or may not be remembered by the patient. The tube and ovary undergo ischemic necrosis and get resorbed. Less commonly it is due to a congenital malformation. [5, 6, 7] The presence of ovarian and tubal remnants in the peritoneal cavity in such a case supports the theory of torsion being responsible for such cases.[8] Congenital malformations of the fallopian tub include accessory ostia, duplication of the entire tube, diverticulum formation and partial or complete absence of the fallopian tube.[9, 10]

Usually the condition is diagnosed incidentally during a laparoscopy or laparotomy which is being done for an unrelated condition. Pednekar and Parulekar reported unilateral absence of midportion of the left fallopian tube at the time of a cesarean section in a multiparous patient.[2] Partial absence of the tube was reported in 18 cases by Nawroth et al.[11] Total absence of a fallopian tube is even rarer. Paternoster et al reported two such cases. [12] Paterson and Chan reported a curious case with absence of proximal part of the left fallopian tube and separation of the distal end into 3 parts. [13]
The case presented here was extremely unusual in that there was a fibrotic atresia of the entire fallopian tube. Such an occurrence has not been described in the literature. It was asymptomatic and was diagnosed coincidentally. Since the woman had both ovaries and a normal other fallopian tube, her reproductive potential was not affected by the condition. It is reported only for the sake of making the medical community aware of such a condition. It has no treatment.

Acknowledgment

I thank Dr Neha Mathews for taking operative photograph.

References
  1. Alexander HD. True unicornuate uterus and total absence of left broad ligament, round ligament, salpinx, ovary, kidney and ureter. Can Med Assoc J. 1947 May; 56(5):539.
  2. Pednekar R, Parulekar SV. Unilateral Segmental Absence Of Fallopian Tube. JPGO 2015. Volume 3 No. 6. Available from: http://www.jpgo.org/2016/06/unilateral-segmental-absence-of.html
  3. Yazawa H, Yabe M, Endo S, Hayashi S.A Case Of Congenital Unilateral Partial Absence Of Fallopian Tube. Fukushima Journal Of Medical Science. 2010;56:44-49.
  4. Rapisarda G, Pappalardo EM, Arancio A, La Greca M. Unilateral ovarian and fallopian tube agenesis. Arch Gynecol Obstet. 2009 Nov; 280(5):849-50.
  5. Sirisena LA. Unexplained absence of an ovary and uterine tube. Postgrad Med J. 1978;54:423–424.
  6. Eustace DL. Congenital absence of fallopian tube and ovary. Eur J Obstet Gynecol Reprod Biol. 1992;46:157–159.
  7. Vaiarelli A, Luk J, Patrizio P. Ectopic pregnancy after IVF in a patient with unilateral agenesis of the fallopian tube and ovary and with endometriosis: search of the literature for these associations. J Assist Reprod Genet. 2012;29:901–904.
  8. Uckuyu A, Ozcimen EE, Sevinc Ciftci FC. Unilateral congenital ovarian and partial tubal absence: report of four cases with review of literature. Fertil Steril. 2009;91:936 e5–e8.
  9. Beyth  Y,  Kopolovic  J.  Accessory  tubes,  a  possible contributing  factor  in  fertility.  Fertile  Sterile. 1982;38:382-3.
  10. Daw E. Duplication of uterine tube. Obstet Gynecol. 1973;42:137-8.
  11. Nawroth F, Nugent W, Ludwig M. Congenital partial atresia  of  fallopian  tube.  Reprod Biomed  Online. 2006;12:205-8.
  12. Paternoster DM, Costantini W, Uglietti A, Vasile C, Bocconi L. Congenital or torsion-induced absence of Fallopian tubes. Two case reports. Minerva Ginecologica 1998, 50(5):191-194.
  13. Paterson PJ, Chan CLK. Congenital Absence of Fallopian Tube Segments. ANZJPG 1985 Available from: https://obgyn.onlinelibrary.wiley.com/doi/abs/10.1111/j.1479-828X.1985.tb00626.x#
Citation

Parulekar SV. Unilateral Fibrotic Atresia Of Fallopian Tube. JPGO 2018. Vol 5 No. 11. Available from: https://www.jpgo.org/2018/11/unilateral-fibrotic-atresia-of.html

Bicournuate Unicollis Uterus Incidentally Detected At Emergency Cesarean Section

Author Information

Jha N*, Chaudhari HK**, Prasad M***.
(* Junior Resident, ** Associate Professor, *** Assistant Professor. Department of Obstetrics and Gynecology, Seth G S Medical College and K E M Hospital, Mumbai, India.)

Abstract

Cesarean sections are to be performed by a lower uterine segment. Altered anatomy due to the presence of uterine malformations and that too when they are undiagnosed can be tricky. These may result in inadvertant surgical complications One such case of a cesarean section in an undetected case of uterine malformation who luckily did not face undue surgical trauma is being presented here.

Introduction

Cesarean sections are to be done through a lower segment incision. Outcomes of patients with upper segment cesarean section have significantly worse outcomes compared to lower segment cesarean section. A cesarean section wherein an apparent  vertical incision extending from the lower segment of the gravid horn to near the medial wall of the undiagnosed non gravid uterine horn detected only at the time of suturing the uterine incision, is described here.

Case Report

A 20 year old gravida 3 para 2 living 1 IUFD1 came at 35 weeks and 3 days of gestation with complaints of pain in abdomen. Her previous pregnancies had been cesarean sections, details of which were not known precisely. She also reported that in her first pregnancy she was told that there was rupture of the uterus, resulting in fetal demise. The operative notes were not available, and whether the scar was of a lower segment or upper segment could not be ascertained. The delivery occurred at term. Her second pregnancy was a cesarean section which was done in a tertiary care hospital. She had been taken for an elective cesarean section, in view of uncertainty of the details of the first pregnancy. She had not been willing for a vaginal birth after cesarean in any case, because of prior fetal demise. There was no significant medical or surgical illness history.  She had registered with our center in the first trimester, and was followed up regularly. However, no precise decision regarding her obstetric management could be taken because both prior operative notes were not available. It was decided to perform elective cesarean section at 37 weeks of gestation. However, she presented at 35 weeks in preterm labor.
On admission she was having no tachycardia, blood pressure was 110/ 80 mm Hg, cardiovascular and respiratory systems were normal. Uterus was 36 weeks in size deviated to the right side with activity of two contractions in 10 minutes. There was no scar tenderness. There was a well healed Pfannensteil scar.  Cervical os was 1 centimeter dilated, 40% effaced, vertex was at -3 station, membranes were intact and show was present. Pre-operative investigations were done and she was taken up for emergency lower segment cesarean section. No attempt of tocolysis was made as she had given a history of unconfirmed uterine rupture. She had consented for placement of a post placental intrauterine contraceptive device (IUCD).
Intraoperatively, abdomen was opened by midline vertical incision and there were no peritoneal adhesions. Doyens’ retractor was introduced and uterus was easily visualized. Loose uterovesical fold of peritoneum was dissected out and Doyens’ retractor was advanced to retract the urinary bladder. A transverse incision was taken on what was presumed to be the lower uterine segment. Female child of 2.440 kg was delivered and placenta was expelled spontaneously.  Upon attempting to mop the uterine cavity, it was realized that the uterus is bicornuate in nature, with small, inconspicuous left horn that was not seen prior to the delivery (figure 1).

 

Figure 1. Image showing bicornuate uterus with small left uterine horn (blue arrow) and right normal gravid horn (green arrow pointing to right side).

Only after completely exteriorizing the uterus and visualizing the entire uterine contour, the bicornuate status was confirmed after the adnexal structure of the left side came into view (figure 2).


Figure 2. Image showing visualization of left sided adnexal structures (green arrow).

The incision was found to be extending between the 2 horns almost vertically into the loose fold of the peritoneum that was part of the vesico-rectal fold. The non gravid horn’s medial wall was not injured or opened up and its cavity was also not seen. As the cavity was not opened it was not possible to see where the two cavities communicated. The lower end of the non gravid horn did not open in the incised lower segment of the gravid horn. The cervix was traced from the right horn down and the uterine incision was approximated, closed and hemostasis was achieved. There were no problems in suturing or achieving hemostasis. The almost vertical nature of the incision (but not classical section), though inadvertent, was clearly evident after the closure of the uterine incision (figure 3). The non gravid left horn was normal in size, soft to touch and did not have an obvious hematometra. Her cervix was single as felt during the digital examination that was done on admission  There was no sign of endometriosis on the left tube or the ovary or on the uterosacrals which were inspected after closing the uterus. Both horns were inspected to see for any evidence of previous scar of cesarean section and uterine rupture but no apparent scars of either could be identified. Postoperative recovery was uneventful and she was discharged on day 5 of her surgery.


Figure 3. Intraoperative image after closure of the uterus. Arrow shows the extension of the incision in the anterior part of the vesico-rectal fold of the peritoneum. 

Even though a consent for intrauterine postplacental IUCD insertion had been taken, but IUCD could not be inserted as only the cavity of the pregnant horn was accessible and that of the non pregnant horn was not accessible as that cavity was not opened. She was informed about this and the husband decided to undergo a vasectomy subsequently. She is scheduled for a hysterosalpingography 3 months after the cesarean section. 

Discussion

When compared to a normal population, women with uterine malformations have a higher risk of preterm births, cesarean sections and poor neonatal outcomes.[1] Our patient needed a preterm cesarean section but had good neonatal outcome. While considering this patient’s first pregnancy outcome, it appeared to be a spontaneous uterine rupture in an unscarred bicornuate uterus. Such cases have been reported by Nitzsche et al and Ashelby et al.[2,3]
Case reports wherein such patients underwent a vaginal delivery successfully, but presented with a ruptured rudimentary horn postdelivery have also been described.[4] Our patient was not given a vaginal delivery due to previous two scars on the uterus. Due to the lack of availability of papers, we could not ascertain any particular detail about the first pregnancy outcome.
In her second pregnancy, she underwent a planned cesarean section. Though no specific guidelines exist on the matter, most case series which describe pregnancy outcome after a uterine rupture, uneventful cesarean sections appear to be the most common mode of delivery.[5-7]
Regarding the pregnancy outcome of the index pregnancy, which has been described in this report, it is noted that operative details and difficulties in specific cases of uterine malformations have not been described.
In this case, the surgeon uterine malformation was undiagnosed despite her having undergone a previous surgery for rupture uterus. Though this patient was registered in the first trimester, and ultrasonography had been done then, it was not able to pick up the uterine abnormality. Chan et al, in their large systematic review had concluded that two-dimensional ultrasonography is a poor modality to identify uterine abnormalities. Three dimensional ultrasonography and MRI are better.[8] These were not utilized in our case, and could be considered in future similar cases.
As it has been shown in the figures, the bicornuate uterus came to light only after the delivery of the fetus, and just prior to uterine incision closure.
The bladder appeared to be pulled up and it was attributed to her previous 2 surgeries, and since dissection of the uterovesical fold of peritoneum was done, it appeared as though the lower uterine segment was reached. Whereas, in actuality, only on the right side the lower segment of the uterus was reached. On the left side, the incision extended into the junction of the two horns without actually entering the cavity of the other horn.
There was no problem in approximating this oblique incision. Despite a comprehensive literature search, the authors have not been able to find any reported cases of similar description.
Luckily for this patient the incision did not injure the non gravid horn. However, such a surgical error can occur in rare cases of undiagnosed malformed uteri. One method of prevention of such an injury would be to inspect the uterine contour and the round ligaments prior to the incision on the uterus. However, the operating surgeons in this case, did not do it. They did not entertain the possibility of a pregnancy in a single horn of a bicornuate uterus prior to delivery of the fetus as she had a 1st trimester ultrasound scan and she had a prior cesarean birth. This appears to be the main learning point for readers here.
Even if such inspection is done, there can be a possibility that the non-pregnant horn lies posteriorly and comes to vision only after delivery of the fetus. 
Whether the non-confirmation of uterine malformation prior to incision on the uterus constitutes a complication or a medical error is debatable. Pettker, in a systematic review, has described that human and systemic errors are an innate part of medical and surgical procedures. It has also been opined that many obstetric errors, specifically those that are not reportable to the governmental authorities, tend to be under reported and are assumed not to be happening at all, which is erroneous. Further, it has been opined that such events should be considered for disclosure to not only the patients, but also to medical professionals.[9,10]

Conclusions

Whenever there is abnormal perinatal outcome (uterine rupture in this case), women must be clearly counseled about preserving medical records and providing it to attending doctors of subsequent pregnancies. In patients with prior cesarean sections with poor outcomes, three dimensional ultrasonography or MRI could be considered in the early pregnancy, considering the possibility of a malformed uterus. Intra operatively, uterus should be inspected in-toto, both round ligaments should be identified to centralize the uterus before an incision is taken. Failure to identify both round ligaments should alert the obstetrician to the possibility of an underlying uterine malformation.

References

  1. Żyła MM, Wilczyński J, Nowakowska-Głąb A, Maniecka-Bryła I, Nowakowska D. Pregnancy and Delivery in Women with Uterine Malformations. Adv Clin Exp Med 2015; 24(5):873–9.
  2. Nitzsche B, Dwiggins M, Catt S. Uterine rupture in a primigravid patient with an unscarred bicornuate uterus at term. Case reports women’s Heal. 2017; 15:1–2.
  3. Ashelby L, Toll G, Patel RR, Abdel-Fattah S, Hunter A. Live birth after rupture of a non-communicating horn of a bicornuate uterus. BJOG. 2005; 112(11):1576–7.
  4. Mishra N, Yadav N, Koshiya D, Jhanwar V. Ruptured rudimentary horn pregnancy with a history of an uneventful vaginal delivery. J Med Ultrason. 2015 Jan; 42(1):117–20.
  5. Al Sakka M, Dauleh W, Al Hassani S. Case series of uterine rupture and subsequent pregnancy outcome. Int J Fertil Womens Med. 2005; 44(6):297–300
  6. Chibber R, El-Saleh E, Al Fadhli R, Al Jassar W, Al Harmi J. Uterine rupture and subsequent pregnancy outcome – how safe is it? A 25-year study. J Matern Fetal Neonatal Med. 2010; 23(5):421–4.
  7. Lim AC, Kwee A, Bruinse HW. Pregnancy after uterine rupture: a report of 5 cases and a review of the literature. Obstet Gynecol Surv. 2005; 60(9):613–7.
  8. Chan YY, Jayaprakasan K, Tan A, Thornton JG, Coomarasamy A, Raine-Fenning NJ. Reproductive outcomes in women with congenital uterine anomalies: a systematic review. Ultrasound Obstet Gynecol. 2011;38(4):371-82. 
  9. Pettker CM. Systematic approaches to adverse events in obstetrics, Part I: Event identification and classification. Semin Perinatol. 2017;41(3):151-155.
  10. Pettker CM. Systematic approaches to adverse events in obstetrics, Part II: Event analysis and response. Semin Perinatol. 2017;41(3):156-160.
Citation

Jha N, Chaudhari HK, Prasad M. Bicournuate Unicollis Uterus Incidentally Detected At Emergency Cesarean Section. JPGO 2018. Volume 5 No.11. Available from: https://www.jpgo.org/2018/11/bicournuate-unicollis-uterus.html

A Case Of Severe Restrictive Lung Disease In Pregnancy

Author Information

Shetty RS*, Hatkar PA**, Desai GS***.
(* Senior Resident, ** Associate Professor, *** Assistant Professor, Department of Obstetrics & Gynaecology, Seth G S Medical College and K E M Hospital, Parel, Mumbai, India.)

Abstract

Severe restrictive lung disease as a result of post tuberculosis sequelae occurring in pregnancy is a rare phenomenon. We present one such case which was managed upto term, with a severe restrictive lung disease, needing cesarean section. The importance of multidisciplinary management is highlighted here.

Introduction

Dyspnea in a pregnant woman may be physiological or pathological. During pregnancy, pulmonary functional reserve, including functional residual capacity and total lung capacity, are reduced; while oxygen consumption by the placenta, fetus, and maternal organs is raised.[1] Any restriction in ventilation during pregnancy has serious consequences for both the mother and fetus. Here, we present a case of right lung collapse due to tuberculous sequelae in a pregnant woman and its management.

Case Report

A 33 year old primigravida, presented at 32 weeks of gestation with complaints of breathlessness and orthopnea. She had tuberculous pleural effusion at 11 years of age, and had completed 6 months of treatment. Thereafter, she had been asymptomatic. She conceived spontaneously two years after marriage and registered at a private hospital. She had an uneventful pregnancy when she started to experience breathlessness, prior to presentation in our hospital.  On examination, there was tachypnea (30 per minute), and respiratory system examination showed asymmetry between the chest walls, with reduced air entry on the right side. Abdominal examination showed uterus of 32 weeks gestation with live fetus in cephalic presentation and vaginal examination showed her not to be in labor. Chest X ray (prior to presentation) showed right middle zone/ lower zone homogenous opacity with tracheal shift to right and left lower zone haziness (figure 1).
Figure 1. Image of chest x ray showing restriction with a well ventilated left lung but almost completely fibrosed right lung.

In view of respiratory failure, she was admitted in the respiratory critical care unit, and was started on oxygen, bronchodilators & injectable steroids. Her vital parameters were monitored. Subsequently she improved and oxygen requirement reduced. Steroids were tapered. Sputum showed no mycobacterial growth. Gene Xpert was negative. She was maintained on inhalational broncholidator (Formeterol 6 mg and budesonide 400 mcg twice a day via rotahaler) and salbutamol 200 mcg when needed. Throat swab was sent and respiratory infection (H1N1, bacterial growth) was ruled out. Throughout this period of admission, she maintained good fetal kick count and there were no obstetric complaints.  One week after the above management, chest X ray was repeated which showed a similar picture. In view of her stable condition, she was discharged in 6 days, and advised to follow up in antenatal outpatient department. She was compliant and then was admitted close to term for observation and a labor plan. It was imperative to avoid the stress of labor on the already decompensated lung which could add to further complications. Though pulmonary function tests are not normally done, in view of severe nature of the disease, the decision to perform the same was taken by the chest physician. This revealed a severe restriction, and based on this report, the chest physicians advised that vaginal birth should be avoided. Anesthesiologists were consulted for the same, and they opined that for cesarean birth, regional anesthesia will be preferred. Apart from mild mitral regurgitation and mild tricuspid regurgitation, cardiac echocardiography was largely normal and cardiologist opined there is a moderate risk for cardiac failure in the event of an operative procedure like LSCS. In the event of failure of regional anesthesia, and presence of significant lung collapse, and possible difficult intubation, a prophylactic tracheostomy was considered as one of the options. However, ENT surgeons opined that this be done only on an emergency basis, and not electively. Hence, a combined multidisciplinary opinion of cesarean section was confirmed. For optimization of respiratory status, she was advised spirometry.
She went into labor at 37 weeks of gestation, and a high dependency unit was made available for her.  Epidural analgesia with spinal anaesthesia was given, and LSCS was performed delivering a live term baby with a good Apgar score. Usual antibiotics (ceftriaxone, gentamycin, metronidazole) used. Initial postoperative period in ICU was uneventful, orals were started and she was ambulated. Around 36 hours post-procedure, she developed breathlessness and cough with one episode of haemoptysis. Chest physicians reviewed and apart from continuing inhalational bronchodilators, injectable steroids (injection hydrocortisone 100 mg) was added. Injection tranexamic acid 500 mg four times a day and ethamsylate 500 mg intravenous three times a day were added. Investigations (arterial blood gases, electrocardiography, platelet count, coagulation profile, sputum AFB) were within normal limits. ENT specialists ruled out an upper airway cause of haemoptysis. Sputum culture grew Pseudomonas species sensitive to amikacin/ cefepime/ ceftazidine/ ceftriaxone/ cefepime/ ciprofloxacin/ levofloxacin/ piperacillin tazobactam. High resolution CT scan was done which showed post- infective sequelae, with ground glass consolidation in superior segment of left lower lobe and right lung collapse (figure 2).

Figure 2.  Image of CT scan showing reduced air entry in the right lung, with red arrow pointing towards well aerated area and yellow arrow pointing towards collapsed lung area.

She was transferred to medical ward and was started on injection piperacillin tazobactam 4.5 gm intravenous twice a day, tablet azithromycin 500 mg once a day and injection furosemide 20 mg twice along with the rest of the prior medications.
With these medications, she improved symptomatically, and never required any mechanical ventilation. In view of coexisting non-productive cough also, tablet oseltamivir 75 mg twice a day was started, and throat swab was sent for H1N1 (which turned out to be negative). A week after the procedure, cardiologists reviewed and advised reassessment after 6 weeks. She was shifted back to obstetric postnatal ward on day 9 of procedure. She was monitored for one more week and due to constant improvement, she was discharged at 16th postoperative day, with advice for regular follow up. She was counseled about various options of contraception, and she chose Injection depot medroxyprogesterone acetate.

Discussion

Tuberculosis continues to be a major health problem in India. More than 210 cases of tuberculosis per 1,00,000 people was the estimate for 2016, according to the WHO predictions for India. Successful treatment is based on clearance of tuberculosis bacilli from the involved site or completion of the drug dose but does not assess structural and functional effects on the involved organ.[2] Our patient had a long standing structural effect, and problems arose during pregnancy.  TB can give rise to long term pulmonary complications such as fibrosis, bronchiectasis, chronic pulmonary aspergillosis, airway stenosis and chronic obstructive pulmonary disease. It is believed to associated with cancer also.[3]

Singla et al reported on 51 MDR-TB patients who were successfully treated. Of them, 78% had persistent respiratory symptoms. Almost all had residual radiological sequelae and ventilatory defects. Two-thirds showed a mixed type of ventilatory abnormality and 19 % had pure restriction and 11 % had pure obstruction.[4] Hndizo et al and Amaral et al have also reported similar outcomes.[5,6] Like the majority, our patient had radiological sequelae, ventilatory defects and a mixed ventilatory abnormality, confirmed on pulmonary function tests also.
Ventilator management in woman carrying a pregnancy, with pre-existing respiratory difficulty poses a dilemma. Endotracheal intubation carries more than normal risk, and failed intubation is many times more common. The altered respiratory dynamics (reduced functional residual capacity and increased oxygen consumption) cause rapid oxygen desaturation during apnea or hypoventilation. Non-invasive ventilation is ideal for short-term ventilatory support, and avoids the potential complications of intubation.[7] In our patient also, maximum attempts were made to avoid intubation. Stress of labor would have precipitated episodes of respiratory difficulty and hence was avoided. Regional anesthesia was used and high flow oxygen was used, thereby successfully avoiding intubation at any time.

Interventions such as inhaled nitric oxide and prone positioning in pregnancy have also been used successfully. Nair et al have reported the successful use of extracorporeal life-support for ARDS in pregnant patients with good outcomes, especially during the H1N1 epidemic in 2009.[8]
Delivery of the fetus in a patient with respiratory failure will not cause a significant benefit to the mother, except relief from mechanical discomfort. However, Tomlinson et al in their case series have opined that induction of labor in a respiratory-status-compromised mother is more likely to cause harm than benefits.[9] In the study by Jenkins et al also, a higher proportion of patients underwent a cesarean section.[10] This is similar to our decision to prefer a cesarean section, based on the opinion of the pulmonologists, and supported by a multi-discipinary discussion of specialists.  Although cesarean section may allow more rapid delivery, the increased physiological stress, and slightly higher risk of higher mortality should be taken into account. ICU arrangements must be made available in the event of sudden maternal or fetal deterioration.[10]  As expected, our patient also needed ICU stay for six days. However, the recovery and overall outcome was good, as described.

Conclusion

Sequelae of pulmonary tuberculosis can become symptomatic during pregnancy, after a long time of quiescence. Severe restriction of pulmonary function may necessitate pulmonary function tests in pregnancy. The risks of performing a cesarean section should be balanced against the benefits obtained by avoidance of exertion during labor. Successful management of  cases of severe restrictive lung disease with good feto-maternal outcome is possible by following a multi-disciplinary approach.

References
  1. Wong MK, Leung WC, Wang JK, Lao TT, Ip MS, Lam WK, et al. Recurrent pneumothorax in pregnancy: What should we do after placing an intercostals drain. Hong Kong Med J. 2006; 12: 375-80.
  2.  Shah M, Reed C. Complications of tuberculosis. Curr Opin Infect Dis 2014; 27(5):403-10.
  3. Liang HY, Li XL, Yu XS, Peng G, Zhi-Hua Y, He QC, et al. Facts and Fiction of the relationship between pre-existing tuberculosis and lung cancer risk: a systematic review. Int J Cancer 2009; 125: 2936-44.
  4. Singla N, Singla R, Fernandes S, Behera D. Post treatment sequelae of multidrug resistant tuberculosis patients. Indian J Tuberc 2009;56(4):206–12.
  5. Hnzido E, Singh T, Churchyard G. Chronic Pulmonary function impairment by initial and recurrent pulmonary tuberculosis following treatment. Thorax 2000; 55:32–8.
  6. Amaral AF, Coton S, Kato B, Tan WC, Studnicka M, Janson C, et al. Tuberculosis associates with both airflow obstruction and low lung function: BOLD results. Eur Respir J. 2015 Oct;46(4):1104-12
  7. Al-Ansari MA, Hameed AA, Al-Jawder SE, Saeed HM. Use of non invasive positive pressure ventilation in pregnancy: Case series. Ann Thoracic Medicine 2007; 2: 23–25.
  8. Nair P, Davies AR, Beca J, Bellomo R, Ellwood D, Forrest P, et al. Extracorporeal membrane oxygenation for severe ARDS in pregnant and postpartum women during the 2009 H1N1 pandemic. Intensive Care Med 2011; 37: 648–654.
  9. Tomlinson MW, Caruthers TJ, Whitty JE, Gonik B. Does delivery improve maternal condition in the respiratory-compromised gravida? Obstet Gynecol 1998; 91: 108–111.
  10. Jenkins TM, Troiano NH, Graves CR, Baird SM, Boehm FH. Mechanical ventilation in an obstetric population: Characteristics and delivery rates. Am J Obstet Gynecol 2003; 188: 549–52.
Citation

Shetty RS, Hatkar P, Desai GS. A Case Of Severe Restrictive Lung Disease In Pregnancy. JPGO 2018. Volume 5 No.11. Available from:https://www.jpgo.org/2018/11/a-case-of-severe-restrictive-lung.html

Idiopathic Bell’s Palsy In Pregnancy


Author Information

Koshewara P*, Prasad M**, Gupta A.S***.
(* Senior Resident, ** Assistant Professor, *** Professor. Department of Obstetrics and Gynecology, Seth GS Medical college and KEM Hospital, Mumbai, India.)

Abstract

Bell’s palsy is rare in pregnancy. Successful pregnancy outcome in a patient who developed acute onset Bell’s palsy in the later part of pregnancy, and continued to have the same during delivery, and marginal improvement post delivery is presented here.

Introduction

Neurological disorders in pregnancy are rare. A patient of Bell’s palsy who had sudden onset of this condition during late third trimester is described below. She underwent successful vaginal delivery and her treatment was similar to that of a non-pregnant patient. 

Case Report

A 30 year old gravida 4 para 1 abortion 2 presented to our tertiary care center at 39 weeks of gestation. She was apparently well till 6 days prior when she developed tingling sensation at left side of face, difficulty on closure of left eye, loss of taste sensation, diplopia, vomiting and headache. She presented to the place of her registration, and evaluation by physician was performed. However, no specific treatment was initiated. Local symptomatic measures such as eye drops had been advised. She was referred to our hospital with labor pains. 
History was reviewed and she had uneventful antenatal period with regular follow ups. She received three doses of iron sucrose intravenous therapy in view of iron deficiency anemia. Her first delivery was uneventful followed by two spontaneous abortions. Antenatal profile was otherwise normal. Blood sugar level (random blood sugar value of 90 mg/ dl) was normal. There was no other significant medical or surgical history. She was averagely built with a normal BMI. On examination, she was afebrile, with blood pressure of 130/80 mm of Hg, pulse of 86/ min, respiratory rate of 18/ min. Cardio-pulmonary examination was normal. Examination showed chemosis of the left eye, swollen lips, tongue deviated to right side, angle of mouth deviated to right side, and loss of wrinkling of forehead and face on the left side. All of these findings were suggestive of lower motor neuron palsy. (She did not consent for her photograph).

Abdominal examination showed a 36 weeks gestation with fetus in breech presentation with regular fetal heart rate of 130/ minute and good uterine activity of 3 to 4 contractions lasting 40 seconds in 10 minutes duration. Vaginal examination showed 4 cm dilated internal os suggestive of established labor. In view of breech presentation in active labor, preparation for cesarean section was initiated. However, in view of Bell’s palsy,  neurologists and oto-laryngologists were consulted. In the mean while the labor progressed spontaneously and assisted breech vaginal delivery was performed, delivering a 2.3 kg male child with no complications.

Neurologists and oto-laryngologists re-examined, and confirmed the case to be Bell’s palsy. She was advised low dose steroid (tablet prednisolone 60 mg once a day for one week, and then gradual weekly tapering of the drug), multivitamins, ciprofloxacin eye drops and chloromphenicol eye ointment to prevent dry eye. Tablet acyclovir 800 mg three times a day was also started.  Physiotherapists taught the patient facial massage and physiotherapy exercises. Neurologists and otolaryngologists advised continuation of steroid injection in the peripartum period.  Post delivery course was uneventful. The signs and symptoms of Bell’s palsy improved and on day 5 of her admission she was discharged with advise to follow up with the physician, neurologist and us.

Discussion

The condition of Bell’s palsy in pregnancy is an intriguing one. It is widely speculated that the famous Italian painting “Mona Lisa” by DaVinci was modeled on a lady who was suffering from this condition. Loosely, this condition is also referred to as “Mona Lisa syndrome”.[1]
The incidence of Bell’s palsy is reported to be around 15- 40 per 100000 people.[2] The incidence is approximately the same in pregnancy also (17 in 100000), as reported by Katz et al.[3]The occurrence of Bell’s palsy in pregnancy is approximately 3 times more during the pregnant state when compared to non-pregnant state.[4]
In this review, it was noted that occurrence of Bell’s palsy during pregnancy was associated with obesity, chronic hypertension and preeclampsia. However, our patient did not have any of the above conditions.  Juan et al and Fawale et al have also reported the similar association of hypertension and Bell’s palsy.[5,6]  Bilateral facial paralysis in pregnancy have been reported by Kovo et al and Vogell et al.[7,8] In the case by the latter, there was early onset bilateral Bell’s palsy with severe preeclampsia developing later after the onset of palsy. 
It is reported to be more common in diabetics. However, our patient did not have this risk factor.[2]Our patient came with spontaneous established labor. Induction of labor has also been reported, mainly for accompanying obstetric indication.[9]
Our patient had good perinatal outcome. As reviewed by Katz et al, there is no association between Bell’s palsy and worsening of perinatal outcome.[3]  Presentation in the puerperium has also been described.[10]  Our patient was started on corticosteroids. Steroids for treatment of Bell’s palsy are evidence based.[11] Our patient was started on antiviral treatment also. Evidence suggests that antiviral treatment for Bell’s palsy should be used only in combination with corticosteroids.[12] Gillman et al followed up patients with Bell’s palsy upto one year after pregnancy completion. The recovery of this group of patients was worse when compared to Bell’s palsy occurring in non-pregnant patients. Our patient will be followed up regularly, to monitor the course of recovery.[13]
While the management of this condition in pregnancy is almost similar to that of non-pregnant state, subtle differences have been highlighted in a review by Hussain et al. [14] In obstetric patients, the main differences were need for multidisciplinary involvement, a search for atypical causes of Bells palsy and earlier institution of corticosteroids. No specific anesthetic precautions are recommended if operative delivery is indicated.[15] and this is also supported by  Overall, Bell’s Palsy is a condition with good recovery rate. However, when it occurs during pregnancy it appears to have a longer time for recovery and probably worse long term outcomes, as reviewed by Philips et al.[16]

References
  1. Hellebrand MC, Friebe-Hoffmann U, Bender HG, Kojda G, Hoffmann TK. [Mona Lisa syndrome: idiopathic facial paralysis during pregnancy]. [Article in German] Z Geburtshilfe Neonatol. 2006; 210(4):126-34.
  2. Somasundara D, Sullivan F. Management of Bell's palsy. Aust Prescr. 2017; 40(3):94-7.
  3. Katz A, Sergienko R, Dior U, Wiznitzer A, Kaplan DM, Sheiner E. Bell's palsy during pregnancy: is it associated with adverse perinatal outcome? Laryngoscope. 2011; 121(7):1395-8
  4. Cohen Y., Lavie O., Granovsky-Grisaru S., Aboulafia Y., Diamant Y.Z. Bell palsy complicating pregnancy: a review; Obstet Gynecol Surv.2000; 55(3): 184-8.
  5. Juan YC, Au HK, Hsu JJ, Ma PC, Liu WM, Jeng CJ. Bell palsy and preeclampsia superimposed on chronic hypertension. Taiwan J Obstet Gynecol. 2010; 49(2):223-4.
  6. Fawale MB, Owolabi MO, Ogunbode O. Bell's palsy in pregnancy and the puerperium: a report of five cases. Afr J Med Med Sci. 2010; 39(2):147-51.
  7. Kovo M, Sagi Y, Lampl Y, Golan A. Simultaneous bilateral Bell's palsy during pregnancy. J Matern Fetal Neonatal Med. 2009; 22(12):1211-3.
  8. Vogell A, Boelig RC, Skora J, Baxter JK. Bilateral Bell palsy as a presenting sign of preeclampsia. Obstet Gynecol. 2014; 124(2 Pt 2 Suppl 1):459-61.
  9. Aditya V. LMN Facial Palsy in Pregnancy: An Opportunity to Predict Preeclampsia—Report and Review. Case Reports in Obstetrics and Gynecology. 2014; 626871:5
  10. Mylonas I, Kästner R, Sattler C, Kainer F, Friese K. Idiopathic facial paralysis (Bell's palsy) in the immediate puerperium in a patient with mild preeclampsia: a case report. Arch Gynecol Obstet. 2005; 272(3):241-3.
  11. Madhok VB, Gagyor I, Daly F, Somasundara D, Sullivan M, Gammie F, et al. Corticosteroids for Bell's palsy (idiopathic facial paralysis). Cochrane Database Syst Rev. 2016 Jul 18;7: CD001942.
  12. Gagyor I, Madhok VB, Daly F, Somasundara D, Sullivan M, Gammie F, et al. Antiviral treatment for Bell's palsy (idiopathic facial paralysis). Cochrane Database Syst Rev. 2015;(11):CD001869.
  13. Gillman GS, Schaitkin BM, May M, Klein SR. Bell's palsy in pregnancy: a study of recovery outcomes. Otolaryngol Head Neck Surg. 2002 Jan; 126(1):26-30.
  14. Hussain A, Nduka C, Moth P, Malhotra R. Bell's facial nerve palsy in pregnancy: a clinical review. J Obstet Gynaecol. 2017;37(4):409-15
  15. Dorsey DL, Camann WR. Obstetric anesthesia in patients with idiopathic facial paralysis (Bell's palsy): a 10-year survey. Anesth Analg. 1993 Jul;77(1):81-3
  16. Phillips KM, Heiser A, Gaudin R, Hadlock TA, Jowett N. Onset of Bell's palsy in late pregnancy and early puerperium is associated with worse long-term outcomes. Laryngoscope. 2017; 127(12):2854-9.
Citation

Koshewara P, Prasad M, Gupta A.S. Idiopathic Bell’s palsy in pregnancy. JPGO 2018. Volume 5 No.11. Available from: https://www.jpgo.org/2018/11/idiopathic-bells-palsy-in-pregnancy.html

Intraplacental Hematoma As A Cause of Fetal Insufficiency

Author Information
Naykodi P *, Bharti S**, Samant PY***.
(* Senior Resident, ** Junior Resident, *** Additional Professor, Department of Gynecology and Obstetrics, Seth GS Medical College and KEM Hospital, Mumbai, India.)
Abstract
Intraplacental hematoma is a rare entity. A pregnancy with such hematomas can result in serious hemodynamic changes in the placental and fetal circulation resulting in adverse perinatal outcomes. We present a rare case of intraplacental hematoma with intrauterine growth restriction (IUGR) with feto-placental insufficiency, but with good neonatal outcome.
Introduction
Intraplacental hematoma is associated with pre-eclampsia, second trimester miscarriage, preterm delivery, intrauterine growth restriction and stillbirth.[1-3] Also known as the Kline’s hemorrhage or placental cavernae, intraplacental hematoma is a hemorrhagic area in the parenchyma of the placenta, quite commonly associated with microangiopathy.[4]  On ultrasonography, these hematomas are identified in the placental intervillous cavity. We present a case of intrauterine growth restriction and deterioration in the placental and fetal Doppler parameters in a case of intraplacental hematoma.
Case Report

A twenty three year old G2P1L1 with 32 weeks gestation was referred to our hospital with ultrasonography suggestive of intaplacental bleeding. She was registered and had regular followup elsewhere. Her previous pregnancy was uncomplicated and she had a full term delivery. Ultrasonography of 28 weeks was suggestive of intraplacental hematoma of 7.4 x 2.1 x 2.9 cm, and the placenta was located anterior and fundal. One week after this, there was an increase in size of the intraplacental bleed to 5.7 x 6 x 2.5 cm, after which she was referred to our hospital. She continued to perceive good fetal movements. Meanwhile she was given progesterone support and steroid dose was completed. At arrival in our hospital, her pulse rate was 88/ minute and blood pressure was 120/80 mm Hg. Uterine height was 32 weeks with longitudinal lie with cephalic presentation and fetal hearts were regular, at 140 beats per minute.
Hemoglobin was 10.5 gm %, platelet count was 2,80,000/cu mm, INR was 0.89 and fibrinogen was 293 mg %. Liver and renal function test reports were normal. Urine albumin was nil. Ultrasonography at our center was suggestive of single live intrauterine gestation of 30 weeks with estimated fetal weight of 1379 grams with adequate liquor, with asymmetric IUGR with intraplacental hematoma of 9 x 8 x 3 cm. Fetal doppler was suggestive of feto-placental insufficiency with raised umbilical artery S/D ratio of 4, fetal middle cerebral artery and pulsatility index of 1.5 (normal).

Figure 1. Ultrasonography showing hematoma.
A decision of induction of labor with prostaglandin E2 gel was taken in view of increasing size of intraplacental hematoma with IUGR and feto-placental insufficiency. She required cesarean section due to failure of induction, and delivered a female child of 1566 gram with Apgar score of 8/10. Intraoperatively, liqor was clear. Neonate was transferred to NICU and was later discharged after a two week stay.  Placenta showed a defect of 6 x 8 cm well organized blood clot with calcifications (figure 2). The postoperative course was uneventful and she was discharged on day 6.




Figure 2. Yellow arrow pointing to intraplacental hematoma
Discussion
Intraplacental hematomas are rare entities. While retroplacental hematomas are more common in pregnancies less than 20 weeks gestation, intraplacental hematomas occur at higher gestation.[5] The source of bleeding in the intraplacental hematomas is usually maternal and linked to the trophoblastic activity.[6] Kline’s hemorrhage is intervillous thrombosis. It is also referred to as “Placenta Hohlraum”, (cavity in German) or placental cavernae due to intraregional hemorrhagic areas in the middle of the placental cotyledon. The possible etiologies include thrombophilias, antiphospholipid antibody syndrome, pre-eclampsia and disseminated intravascular coagulopathy.[4]  Our patient did not have any of these conditions. Most of the bleeding is of maternal origin. Despite this, these hematomas cause a leak in the fetal circulation resulting in under perfusion and fetal anemia.
The risk of placental insufficiency, intrauterine growth restriction and preterm delivery is more in intraplacental hematoma. However, the risk of intrauterine fetal death is more in cases of retroplacental hematoma.[1] The rapidity of progression of the condition is much lesser in intraplacental hematoma, than in retroplacental hematomas. 
Intraplacental bleeding may sometimes cause non immune hydrops fetalis due to fetomaternal hemorrhage, as reported by Parekh et al.[7] Apart from correct timing of delivery, at present there are no treatment options available for the such hematomas. It is suggested that low molecular weight heparin and aspirin may be beneficial in cases of placental infarcts.[8] The intraplacental hematomas are linked to pre-eclampsia, IUGR, intrauterine fetal deaths and abnormal neurodevelopment in the surviving fetuses.[9] The neonate will be followed up for neurodevelopmental delay, but was neurologically normal at discharge.
Conclusion
Fetomaternal risk from intraplacental hematomas is different from that of retroplacental hematoma. Timely diagnosis and investigations can result in good maternal and perinatal outcome.

References
  1. Ott J, Pecnik P, Promberger R, Pils S, Binder J, Chalubinski KM. Intra- versus retroplacental hematomas: a retrospective case-control study on pregnancy outcomes. BMC Pregnancy and Childbirth. 2017;17: 366.
  2. Borlum KG, Thompsen A, Clausen I, Ericksen G. “Long –term prognosis of pregnancies in women with intrauterine hematomas. Obstet  Gynecol . 1989; 74:231-3.
  3. Alma AG, Edgar HA, Roberto R, Qureshi F, Ahn H, Jacques SM, et al. Prenatal Diagnosis of a Placental Infarction Hematoma Associated with Fetal Growth Restriction, Preeclampsia and Fetal Death: Clinicopathological Correlation. Fetal Diagn Ther. 2014; 36:154-61.
  4. Habek D. Multiple intraplacental hematomas – kline’s haemorrhage. Acta Clin Croa. 2015; 50:423-5.
  5. Xiang L, Wei Z, Cao Y. Symptoms of an intrauterine hematoma associated with pregnancy complications: a systematic review. PLoS One. 2014; 9:11.
  6. Jaffe R, Jauniaux E, Hustin J. Maternal circulation in the first trimester human placenta – myth or reality? AJOG. 1997; 176:695-705.
  7. Parekh N, Agrawal A, Badade A, Satoskar P. Intraplacental Hematoma: A Rare Cause of Non-immune Hydrops. JPGO. 2015; 2: 3.  Available from: http://www.jpgo.org/2015/03/intraplacental-hematoma-rare-cause-of.html
  8. Alkazaleh F, Viero S, Smichen M, Walker M, Smith G, Laskin C,et al. Ultrasound diagnosis of severe thrombotic placental damage in second trimester: an observational study. Ultrasound Obstet Gynecol. 2004; 23:472-6.
  9. Redline RW. Severe fetal placental vascular lesion in term infants with neurologic impairment. AJOG. 2005; 192:452-7.
Citation

Naykodi P, Bharti S, Samant PY. Intraplacental Hematoma As A Cause of Fetal Insufficiency. JPGO 2018. Volume 5 No.11. Available from:https://www.jpgo.org/2018/11/intraplacental-hematoma-as-cause-of.html

A Rare Case Of Fetus With Limb Agenesis

Author Information
Jain P*, Shah R**, Warke HS***.
(* Junior resident, ** Assistant Professor, *** Associate Professor, Department of Obstetrics and Gynecology, Seth GS Medical College and KEM Hospital, Mumbai, India.)
Abstract

A grvavida 3 para 2 living 2 with an ultrasonography report suggestive of live fetus with absent upper limb, and extremely short femur, absent tibia and fibula presented at 19 weeks. After explaining poor prognosis, medical termination of pregnancy was done. Skeletal agenesis and dysgenesis is discussed.
Introduction

Early identification of fetuses with congenital anomalies, chromosomal defects, metabolic diseases or other genetic syndromes is of paramount importance in modern obstetrics, since the option of termination of pregnancy is available. Limb anomalies and skeletal dysgenesis in offspring can cause a great harm to society, in view of consumption of resources as well as social and mental injury to the parents. Often an incidental ultrasonography finding, it can occur even in the absence of history of any drug intake or any familial history of skeletal dysgenesis.
Case Report

A 35 years old, gravida 3 para 2 with 2 healthy living children, came to our hospital with an ultrasonography report suggestive of a single live intrauterine gestation of 19 weeks. Skeletal survey showed absent left upper limb, extremely short left femur, absent tibia and fibula on left side and Congenital Talipus Equino Varus (CTEV) on the right side. There was no contributory family history and no history of drug intake or any radiation exposure. The mother did not suffer from any medical disorders. Amniotic band was less likely as more than one limb was involved. Specific genetic testing of parents or the abortus was not considered as the anomaly has a multifactorial cause unlike specific skeletal dysplasia syndromes. General and systemic examination revealed no abnormality. Uterus was 20 weeks size.
Pediatric surgery reference was taken and poor prognosis and need for postnatal NICU and possibly multiple reconstructive surgeries was explained. The patient opted for termination of pregnancy, and due consent was taken considering the substantial risk to the child. Anesthesia fitness was taken and the patient was posted for hysterotomy in view of prior two cesarean section.
Postoperatively the X ray of the abortus was done (figure 1) On gross examination (figure 2) the abortus had absent upper limb, extremely short femur, absent tibia and fibula on left side. CTEV on the right side could not be appreciated. Postoperative course of the patient was uneventful.

Figure 1. X ray of abortus. Yellow arrow points to normal right upper limb. Orange arrows point to normal right lower limb. Green arrows point to normal spine. Red arrow points to location of left limb (absence). Blue arrows point to short femur and absent tibia/fibula.


Figure 2. Image of abortus. Absent upper limb is well appreciated.
Discussion

Skeletal dysplasia can occur in a number of syndromes like achondroplasia which is caused by mutations in FGFR3 gene.[1] Larsen syndrome is also such a syndrome which affects the bone development throughout the body. They generally have small extra bones in their wrists seen on X- rays.[2] It was considered as a differential diagnosis in this case.
National birth defect surveillance is carried out by the Birth Defects Registry of India. There are multiple individual hospital studies as well. The estimated predelivery rate of congenital anomalies diagnosed is around 11 per 1000 births and the rate of abortions due to congenital anomalies is 4.39 per 1000 births.[3] The large gap is obvious and the need for prenatal diagnosis of major anomalies and possibility for timely intervention or abortion if needed.
The option of termination of pregnancy is offered in all these cases. Induction of labour with prostaglandin E1 analogue misoprostol, is now a common practice and several studies have reported excellent results, including in second trimester of pregnancy.[4,5] Surgical second trimester abortion is preferred over medical methods in view of better outcome and success rates  in a patient with previous uterine surgeries.[6] Our patient underwent a surgical method of termination of pregnancy.
Skeletal defects are rare in uncomplicated pregnancies without family history or history of drug intake, most common being talipes equinovarus. The rate is 49/10000 live births as per the study conducted by Bhide et al.[3]  Majority of anomalies are cardiovascular or neural tube defects. The skeletal dysplasias or osteochondrodysplasias are a heritable group of more than 450 well delineated disorders that affect primarily bone and cartilage, but can also have significant effects on muscle, tendons and ligaments. Skeletal dysplasias are inherited in mainly a dominant mode, but can also be recessive. Knowledge of these is important for genetic counseling. Our patient was referred for genetic counselling with available data regarding this fetus and abortus.
Our patient was diagnosed with the fetal abnormality by an ultrasonography. In a large study by Dicke et al, the sensitivity of ultrasonography in picking up limb reduction defects involving long bones was 76%. The authors analyzed other parameters of fetal limb abnormalities also, and concluded that ultrasonography is an excellent modality for detection of fetal limb anomalies.[7]  Andrikopoulou et al analyzed the factors which improved ultrasonographic detection of isolated fetal limb abnormalities, and found that even a minimal increment in time spent at each ultrasonography gives rise to a significant improvement in detection rates of fetal limb abnormalities.[8]
In most cases of skeletal dysplasia, lethality usually results from small chest, pulmonary hypoplasia and respiratory compromise. Diagnosis is made based on clinical and radiographic findings. Nonlethal skeletal dysplasias are associated with short stature but overall affected individuals are cognitively normal and have a good quality of life.
Conclusion
In this era, ultrasonography can pick up fetal limb abnormalities. Continuation of a pregnancy with a fetus carrying an absent limb merits termination. Surgical termination may be required in scarred uteri. We present this case report and suggest that all efforts must be taken to identify every fetal limb abnormality.

References
  1. Horton WA, Hall JG, Hecht JT. Achondroplasia. Lancet. 2007; 370(9582):162-72.
  2. Bicknell LS, Farrington-Rock C, Shafeghati Y, Rump P, Alanay Y, Alembik Y et al. A molecular and clinical study of Larsen syndrome caused by mutations in FLNB. J Med Genet. 2007 Feb;44(2):89-98.
  3. Bhide P, Gund P, Kar A. Prevalence of Congenital Anomalies in an Indian Maternal Cohort: Healthcare, prevention, and Surveillance Implications. Public Library of Science.  2016;11(11): e0166408.
  4. Bhattacharjee N, Ganguly RP, Saha SP. Misoprostol for termination of mid-trimester post-Caesarean pregnancy. Aust N Z J Obstet Gynaecol. 2007 Feb;47(1):23-5.
  5. Ho PC, Ngai SW, Liu KL, Wong GC, Lee SW. Vaginal misoprostol compared with oral misoprostol in termination of second‐trimester pregnancy. Obstet Gynecol 1997;90: 735–738
  6. Lama S, Chhetri S. Outcome of Medical versus Surgical Methods in Second Trimester Abortion. Nepal Journal of Obstetrics and Gynaecology 2015; 20(2):50-3.
  7. Dicke JM, Piper SL, Goldfarb CA.  The utility of ultrasound for the detection of fetal limb abnormalities--a 20-year single-center experience. Prenat Diagn. 2015 Apr;35(4):348-53.
  8. Andrikopoulou M, Vahanian SA, Chavez MR, Murphy J, Hanna N, Vintzileos AM. Improving the ultrasound detection of isolated fetal limb abnormalities. J Matern Fetal Neonatal Med. 2017 Jan;30(1):46-9.
Citation
Jain P, Shah R, Warke HS. A Rare Case Of Fetus With Limb Agenesis. JPGO 2018. Volume 5 No.11. Available from: https://www.jpgo.org/2018/11/a-rare-case-of-fetus-with-limb-agenesis.html

Remembering Past Greats: Raoul Palmer

Author Information

Prasad M
(Assistant Professor, Department of Obstetrics and Gynecology, Seth GS Medical College and KEM Hopsital, Mumbai)

The surgical revolution which consisted of introduction of laparoscopy for pelvic procedures was pioneered by Raoul Palmer (1906-86).[1] His exceptional stature has been revered by many and well documented.[2] He was a Frenchman with an international influence. He was a surgical gynecologist, and did most of his work along with his wife, Elisabeth Palmer. He is believed to be one of the first to use the colposcope routinely for all his patients, and also started using the Papnicolaou smear regularly. However, the highlight of his pioneering career was the advancement in laparoscopy. Initially, it was called as “celioscopy”. 

Pneumoperitoneum creation was investigated extensively by Palmer. The use of carbon-dioxide, the quantity of gas to be insufflated and the speed of insufflation were looked into in detail, and published by him in the late 1940s. Though his initial experience was with local anesthesia, he had come to the conclusion that general anesthesia should be preferred for laparoscopy procedures.[3]
He designed many instruments for use in laparoscopic surgeries. Most of his work was from Hospital Broca, Paris. The resilience with which he and his supportive wife continued their pioneering medical work, despite the difficulties of German occupation of Paris during the second world war, is described well by Litynski.[4] He is credited with making the first motion picture of laparoscopy, soon after the introduction of an efficient lighting system in 1952. His visionary ideas included the belief of usage of the newly introduced techniques in reproductive enhancement.[5]

He authored many texts and mentored many pupils. Notable gynecologists who were tutored by him include Patrick Steptoe and Melvin Cohen. The modern operative laparoscopy doyen Semm, has also acknowledged Palmer as a founding father in gynecological laparoscopy. He had famously stated that the Paris clinic of Palmer was considered to be a “temple of laparoscopy.

Operative endoscopic gynecology, which is now standard of care, owes a lot to this great individual.

References
  1. Bruhat MA. [Operative laparoscopy: genuine surgical advance or simple temptation by the feasible?].[Article in French] Bull Acad Natl Med. 1994 Feb;178(2):199-208.
  2. Cohen J. Raoul Palmer: a maker of fate. Reprod Biomed Online. 2007 Jan;14(1):125-6.
  3. Schlogel G. [Raoul Palmer and the coelio-surgical adventure from 1940 to 1995]. [Article in French]. Hist Sci Med. 1996;30(2):281-7.
  4. Litynski GS. Raoul Palmer, World War II, and transabdominal coelioscopy. Laparoscopy extends into gynecology. JSLS. 1997 Jul-Sep;1(3):289-92.
  5. Biographies. In O’Dowd MJ, Philipp EE, editors. The History of Obstetrics and Gynecology. 1st ed. Lancs: Parthenon Publishing Group 2000; pp. 640.
Citation

Prasad M. Remembering Past Greats: Raoul Palmer. JPGO 2018. Volume 5 No.11. Available from:https://www.jpgo.org/2018/11/remembering-past-greats-raoul-palmer.html