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Volume 3 Issue 3, March 2016

Editorial
Chauhan AR

Maternal Autoimmune Disorder diagnosed by Fetal Heart Block
Shilotri M, More V, Satia MN.

Ovarian Hyperstimulation Syndrome In a Spontaneous Twin Pregnancy With Hypothyroidism
Chhatrapati A, Nadkarni T, Mishra I, Jassawalla MJ.

Vulval Leiomyoma: a Case Report
Devalla A, Warke HS, Mayadeo NM.

Ovarian Fibroma: An Unusual Case
Manjrekar VM, Parulekar SV.

Intrapartum Pubic Diastasis
Dwivedi JS, Prasad M, Gupta AS.

Cervical Fibroid Mimicking Uterine Inversion
Mehta V, Prasad M, Gupta AS.

Vaginal Myomectomy For Intrauterine Submucous Leiomyomatous Polyp
Niphadkar M, Parulekar SV.

Torsion Hydrosalpinx – a Diagnostic Puzzle
Dixit K, Setty JP, Jassawalla N, Srikanth.

Spontaneous Conception Through Perforated Transverse Vaginal Septum
Goel A, Shende D, Tiwari N, Chauhan AR.

Editorial

Chauhan AR

Symphysiotomy is the deliberate surgical separation of the pubic symphysis to aid vaginal delivery in cases of obstructed labor. Though the West has condemned this surgery and relegated it to obscurity, symphysiotomy is still practiced and in fact, its revival is recommended in resource poor settings, namely parts of central and south Africa. 
Physiologically, the pubic symphysis widens approximately 3 to 7 mm during pregnancy; when this gap is more than 10 mm, it is abnormal and is referred to as pubic symphysis diastasis. This is a rare condition that may occur either intra or postpartum, and presents with acute pelvic pain. The diagnosis is usually clinical, supported by X-rays, and management is conservative in the form of analgesia, prolonged bed rest in lateral position, pelvic binder, and subsequent physiotherapy. Rarely, if the gap exceed 3 to 4 cm, surgery for fixation and stabilization of the joint may be required. 
Interestingly, Séverin Pineau first described symphysiotomy in 1597 after observing pubic diastasis on a hanged pregnant woman; however it was not until 1777 that Jean-René Sigault performed the first successful symphysiotomy in Paris. This procedure was opposed by the famous French accoucher Baudeloque and subsequently, the fortunes of symphysiotomy waxed and waned in Europe for more than a century, till Gigli of Italy performed pubiotomy using his saw. Debate raged about symphysiotomy versus cesarean delivery, and symphysiotomy again fell into disrepute due to its many complications, namely hemorrhage, difficulty during walking and urinary incontinence and fistulae. "Subcutaneous partial symphysiotomy" described by Zarate of Argentina in the early 1920s is a relatively simple procedure, where the superior and inferior ligaments of the symphysis are not completely divided; this is the technique in use today. 
Literature on symphysiotomy is of two extremes: on the one hand, serious issues about the procedure being carried out on approximately 1500 women in Ireland in the latter half of the last century and its long -term sequelae, and on the other hand, its lifesaving role in resource poor situations. 
The practice of symphysiotomy was prevalent in parts of Ireland in the 1940s up to the 1980s, partly due to the orthodox Catholic aversion to cesarean section (CS), especially repeat CS, as contraception is usually not advocated, and partly due to disregard for women's autonomy. The Journal of Bone and Joint Surgery in 2014 carried an article on the radiographic findings after symphysiotomy in Irish women (mean duration of 41.6 years) and found that late- onset osteoarthritis of the sacroiliac joint and pelvic instability were key findings. Recently these surviving women, many of whom are now in their 70s and 80s, have raised their voice about their horrific experiences in labor, where symphysiotomy was performed without their consent, and quality of life issues including nagging pain, difficulty in walking and urinary incontinence. In 2014, this culminated in the United Nations Human Rights Committee moving the Irish government to offer compensation to these women for their physical and emotional trauma.
On the other hand, symphysiotomy has been advocated as life saving in many resource poor and rural parts of Africa, where maternal and perinatal mortality due to neglected obstructed labor is still high, as also mortality after CS. Advocates of symphysiotomy say that it is a simple underemployed surgical procedure done under local anesthesia or analgesia to widen the pelvic ring, and facilitate vaginal delivery in cases of mild to moderate cephalopelvic disproportion, without the need for sophisticated equipment or an operation theater. it can be performed by a doctor or a midwife. The procedure obviates the need for CS, is socio- culturally acceptable to women who prefer vaginal birth, and leaves the uterus unscarred. A 2012 paper by Monjok in the African Journal of Reproductive Health calls for the "revival and reinstatement of symphysiotomy in the obstetric arsenal of Nigeria and sub- Saharan Africa". Another paper by Ersdal from Zimbabwe in 2014 studied the knowledge, attitudes and practice of doctors and midwives, and found that those working in smaller district hospitals had a more positive attitude toward symphysiotomies than those working in larger referral centers, especially teaching institutes. 
A Cochrane review in 2012 by Hofmeyr and colleagues mentions "failure to progress in labour when cesarean section is unavailable, unsafe or declined by the mother; and obstructed birth of the after-coming head of a breech presenting baby" as special situations where symphysiotomy may be resorted to. Though observational studies from Africa and West Asia have reported low maternal mortality rates and high success with symphysiotomy, a large part of the problem is that there are no randomized trials. The authors suggest that evidence from good quality research is needed to compare efficacy and safety of symphysiotomy versus no symphysiotomy, or various modifications of the technique, or symphysiotomy versus CS in various clinical situations. Hence there are no clear recommendations; rather they state that "because of the possibility that symphysiotomy may be life- saving in certain situations, professional and global bodies should provide guidelines for its use (or non- use) based on best available evidence".
Sadly, symphysiotomy is a reflection on the condition of women globally. Till such time as women continue to be the lowest priority on the health ladder and till basic reproductive rights are not awarded to them, this discrimination and disparity will continue. 
This issue of the journal carries an interesting case report of spontaneous pubic diastasis which prompted this commentary on the renewed interest in symphysiotomy. The question on whether its use in modern obstetrics is justified still remains.

Maternal Autoimmune Disorder Diagnosed by Fetal Complete Heart Block with Postnatally Diagnosed Down Syndrome

Author Information

Shilotri M*, More V**,  Satia MN***
(** Second year resident, **Assistant Professor, *** Professor, Department of Obstetrics and Gynecology, Seth G. S. Medical College and KEM Hospital, Mumbai, India.)

Abstract

Congenital complete atrioventricular block may result from a congenital cardiac anomaly or the presence of anti-Ro and/or anti-La antibodies in women who have systemic lupus erythematosus, Sjogren’s syndrome, undifferentiated autoimmune disorder, or are asymptomatic. The prognosis of the baby depends on the time of diagnosis and presence of a cardiac anomaly. Down syndrome has a high incidence of congenital cardiac anomalies, however, complete atrioventricular block is rarely seen. We present a case of a 23 year old gravida 2, para 1, living 1 who was asymptomatic and was diagnosed to have autoimmune antibodies on evaluation for fetal bradycardia which was later diagnosed as complete heart block on fetal echocardiography. The neonate was later incidentally diagnosed to have Down syndrome with atrial septal defect.

Introduction

Congenital complete atrioventricular block is a rare disorder with an incidence of 1 in 22,000 live births.[1] Congenital cardiac anomalies are seen in 44% of Down syndrome cases.[2] Amongst cases of Down syndrome, varying degrees of heart block are seen with atrioventricular septal defects while atrial septal defect is only associated with PR interval prolongation on ECG.[3] Congenital complete atrioventricular block may be diagnosed antenatally as early as 16 weeks gestation. It has a high perinatal morbidity and mortality. Women with positive anti-Ro and/or anti-La antibodies should be monitored with serial fetal echocardiography to detect early any congenital conduction defects. Although there are no guidelines for antenatal treatment of fetal heart block, there are many promising therapies being studied.

Case Report

A 23 year old gravida 2 para 1, living 1 registered antenatally at our clinic at 23.5 weeks gestation. She had no history of any significant medical or surgical illness. She had a previous full term normal delivery and the child was well. At 28.5 weeks gestation the fetal heart sounds were found to be irregular ranging from 60 beats/ minute to 120 beats/ minute, as heard on a stethoscope and confirmed on a hand-held Doppler device during a routine antenatal examination. She was advised admission for evaluation of possible fetal cardiac anomaly. However she was not willing for the same. An obstetric ultrasonography for fetal anomalies and a fetal echocardiography was advised. At her next antenatal visit at 31.5 weeks, the baseline fetal heart rate was 50 beats/ minute. Ultrasonography for fetal malformations at 29 weeks showed evidence of fetal bradycardia (53 beats per minute) most probably due to conduction defect with a complete heart block, mild pericardial effusion and reverse flow in ductus venosus suggestive of early fetal hydrops. No other anomalies were evident. As she was not willing for admission, she was advised to do blood investigations such as anti-nuclear (ANA) antibodies, anti-double stranded DNA (dsDNA) antibodies, anti-Ro (SSA), anti-La (SSB) antibodies and blood sugars. She was advised to follow up with the reports in the outdoor patient department. Patient later returned to the hospital in active labor at 37.1 weeks and delivered uneventfully. She delivered a female child of 1.672 kg with an Apgar score of 8/10. On examination of the neonate, Mongoloid features were observed (slanting eyes, low set ears and depressed nasal bridge) with clinodactyly and bradycardia. Baby was immediately shifted to the neonatal intensive care unit for further evaluation. ECG of the neonate was suggestive of complete heart block with atrial rate of 160 beats/ minute and ventricular rate of 60 beats/ minute. Echocardiography showed a 3 mm atrial septal defect of ostium secundum type. No other cardiac anomalies were detected. Cardiology opinion was taken who advised monthly follow up of the baby. The neonate’s blood counts, hepatic and renal function tests were within normal limits. Karyotype and thyroid function tests of the baby were sent in view of the Mongoloid features. The baby was discharged on full feeds and advised to follow up with the above reports and was diagnosed to have Down syndrome on karyotyping. On maternal evaluation, the patient tested positive for ANA , anti-Ro and anti-La antibodies and was negative for anti-dsDNA antibodies. Rheumatology opinion was sought and was advised urine routine examination and complement levels (C3, C4) which were normal. CRP levels  were raised and thyroid function tests were suggestive of hypothyroidism. Her complete blood counts, renal and liver function tests and blood sugars were normal. She had no complaints of rash, photosensitivity, arthritis or long standing fever. She was asked to follow up in the rheumatology outpatient department for further evaluation of type of autoimmune disorder. Thus, the patient was diagnosed to have an autoimmune disorder on evaluation for the fetal heart block.

Discussion

Congenital complete atrioventricular heart block may manifest any time from 16 weeks gestation to young adulthood. In the antenatal period it may first be detected as bradycardia on auscultation during an obstetric examination, as in our case, or on a routine obstetric ultrasonography. Fetal congenital heart block may be a result of an underlying cardiac structural anomaly as seen in 30 – 53% of cases.[1] The most common cardiac anomaly associated with heart block is left atrial isomerism with or without atrioventricular septal defect and levo-transposition of great arteries.[4] In a structurally normal heart, congenital heart block in the form of neonatal lupus is a result of maternal autoantibodies to intracellular ribonucleoprotiens {anti-Ro (SS-A) and/ or anti-La (SS-B)}, detected on enzyme linked immunosorbent assay. Women that test positive for anti-Ro antibodies have a 3% risk of bearing a child with neonatal lupus.[2] At the time of diagnosis of the fetal heart block the mother may be a known case of systemic lupus erythematosus (26%), Sjogren’s disease (14%), an undifferentiated autoimmune disorder (19%) or may be completely asymptomatic (40%).[3] It is postulated that these autoantibodies are passively transferred through placental circulation which in turn injure a formerly normal fetal heart. The heart block varies from first to third degree and the antibody mediated injury may result in a late cardiomyopathy as seen in 5-11% of cases.[4] Neonatal lupus has other manifestations such as an annular, photosensitive rash on the face, low counts of red blood cells, white blood cells, platelets and deranged liver function tests. These manifestations usually resolve spontaneously, however, the cardiac changes are permanent.
Down syndrome is associated with atrioventricular septal defects (30%), atrial septal defect (25%), ventricular septal defect (22%), patent ductus arteriosus (5%), coarctation of aorta (5%) and tetralogy of Fallot (3%).[2] The type of conduction disorder seen in Down syndrome depends upon the cardiac anomaly. Varying degrees of heart block are seen with atrioventricular septal defects while atrial septal defect is only associated with PR interval prolongation on ECG. Isolated complete congenital heart block in a case of Down syndrome is rare.[7] Thus, in our case, the fetal complete heart block was most likely caused by the maternal autoimmune antibodies.
On diagnosis of congenital heart block, the fetus is monitored by serial echocardiograms. Several antenatal therapies for neonatal lupus have been attempted such as dexamethasone therapy, plasmapheresis, beta stimulation by salbutamol, hydroxychloroquine, intravenous immunoglobulins etc.[8] However, no therapy has been approved as standard protocol for antenatal management of neonatal lupus. In neonates and children cardiac pacing may be required if they are symptomatic, have congestive cardiac failure or in asymptomatic neonates with awake baseline heart rate of less than 55/min.[1] As the neonate in our case had a baseline heart rate of 60/minute and was asymptomatic, pacing was not done.
Complete heart block predisposes the baby to cardiac failure, hydrops fetalis, fetal or neonatal death. Prognosis depends on the presence of an underlying cardiac structural abnormality with a survival beyond the neonatal period of only 14% as compared to 85% in autoimmune congenital heart block.[1] Prognosis is also determined by the time of diagnosis of the disease, with those diagnosed in the newborn period having a better fate than those that are detected in-utero as it is thought that those with severe disease perish in-utero and those with a milder disease survive for a longer time. Although women who test positive for anti-Ro and/or anti-La antibodies may be asymptomatic at the time of diagnosis, there are reports of them becoming symptomatic a few years after the affected delivery, the median time being 1.5 years.[3] Thus it is worthwhile to advise these patients to follow up with a rheumatologist.

References
  1. Kertesz NJ, Fenrich AL, Friedman RA. Congenital complete atrioventricular block. Tex Heart Inst J. 1997;24(4):301-307.
  2. Stoll C, Dott B, Alembik Y, Roth MP. Associated congenital anomalies among cases with Down syndrome. Eur J Med Genet. 2015 Dec;58(12):674-80.
  3. Clark EB, Kugler JD. Preoperative secundum atrial septal defect with coexisting sinus node and atrioventricular node dysfunction. Circulation. 1982;65(5):976-80. PubMed PMID: 7074763
  4. Friedman DM, Duncanson LJ, Glickstein J, Buyon JP. A review of congenital heart block. Images Paediatr Cardiol. 2003;5(3):36-48.
  5. Waltuck J, Buyon JP. Autoantibody-associated congenital heart block: outcome in mothers and children. Ann Intern Med 1994;120:544-51.
  6. Moak JP, Barron KS, Hougen TJ, Wiles HB, Balaji S, Sreeram N, et al. Congenital heart block: development of late-onset cardiomyopathy, a previously underappreciated sequela. J Am Coll Cardiol. 2001;37(1): 238–242. [PubMed: 11153745]
  7. Machakanur V. Congenital Complete Heart Block in Down Syndrome: A Rare Case Report. Journal of Evolution of Medical and Dental Sciences. 2015; 47(4): 8254-8257, DOI: 10.14260/jemds/2015/1197
  8. Hunter LE, Simpson JM. Atrioventricular block during fetal life. J Saudi Heart Assoc. 2015; 27(3): 164-178. doi:10.1016/j.jsha.2014.07.001
Citation

Shilotri M, More V, Satia MN. Maternal Autoimmune Disorder diagnosed by Fetal Heart Block. JPGO 2016. Volume 3 No. 3. Available from: http://www.jpgo.org/2016/03/maternal-autoimmune-disorder-diagnosed.html

Ovarian Hyperstimulation Syndrome In a Spontaneous Twin Pregnancy With Hypothyroidism

Author Information
Chhatrapati A*, Nadkarni T**, Mishra I*, Jassawalla MJ.***.
(*Assistant Professor, ** Additional Professor, ***Honorary Professor and unit head Department of Obstetrics and Gynecology, Nowrosjee Wadia Maternity Hospital, Mumbai, India.)

Abstract

Most of the ovarian hyperstimulation (OHSS) cases occur due to ovulation induction followed by HCG trigger. But OHSS may rarely occur in cases of multifetal gestation, hypothyroidism or polycystic ovarian syndrome. We report a case of OHSS in a spontaneous pregnancy in a primigravida with twin pregnancy with severe hypothyroidism. The clinical presentation included severe abdominal discomfort. After ultrasound examination the diagnosis was confirmed. Patient was managed conservatively.

Introduction

Ovulation induction therapy sometimes leads to ovarian hyperstimulation syndrome which occurs in the luteal phase especially after HCG trigger for ovulation. The syndrome was first described in 1941. In very rare spontaneous pregnancies OHSS can occur due to follicle stimulating hormone receptor gene mutations. The likelihood is more if it is associated with maternal hypothyroidism or multifetal pregnancy. The symptoms vary; nausea, vomiting and abdominal discomfort in mild OHSS and ascites, hydrothorax, thromboembolism and renal failure in severe form. Treatment is mainly supportive.

Case Report

A 21 year old primigravida married since 1 year 6 months was referred to our hospital with 2 and a half months amenorrhea and severe abdominal discomfort. Urine pregnancy test was done which was positive. Patient was stable clinically. Ultrasound (USG) was done. Her USG detected, a monochorionic monoamniotic twin pregnancy with multiple ovarian cysts bilaterally and mild free fluid in the pelvis. Right ovary measured 9.4x7.2 cm with multiple cysts, largest being 3x3 cm. Left ovary measured 11x8 cm with multiple cysts, largest measuring 5x5 cm. Patient did not give history of receiving any ovulation induction agents. Patient was admitted for evaluation and further management.
On physical examination, patients’ vital parameters were stable. Abdomen was soft non tender. On per vaginal examination uterine size was 12 weeks. Bilateral ovaries were cystic. Laboratory investigations revealed severe anemia ( Hb 6.4 gm% ), hematocrit ( 20.1% ), normal blood electrolytes, blood urea nitrogen ( 10 mg/ dL ), creatinine ( 0.8 mg/ dL ) and coagulation profile ( PT/INR 1.0 ). SGOT/SGPT was slightly raised ( 43/35 units/l ). Serum proteins were normal ( Total 6.9 g/ dL; A/G 4.2/2.7 g/ dL). Serum β HCG was above 300,000 mIU/ ml which was corresponding to the weeks of gestation. Serum TSH was very high (> 100 uIU/ ml) suggestive of severe hypothyroidism. Serum estradiol (E2) was 4720 pg/ ml and serum progesterone was 353.41 ng/ ml which was high. Patient was managed conservatively. Hemoglobin electrophoresis was normal. Correction was done with 2 units of packed red cell transfusion. Post transfusion Hb was 9.3 gm% and PCV was 31.7%. For hypothyroidism 100 ug of levo thyroxine was started after consultation with the endocrinologist. At 11 weeks nuchal translucency/ nasal bone (NT/ NB) scan was performed which was normal. There was no change in the size of the ovarian cysts. As patient was symptomatically better she was discharged after 15 days observational period. She followed up in OPD regularly; but she aborted spontaneously; complete abortion at 17 weeks of gestation. USG pelvis done after 10 months of abortion was completely normal. Patient was advised to try for natural conception with early ANC registration.


Figure 1. Hyperstimulated Right Ovary


Figure 2. Hyperstimulated Left Ovary


Figure 3. Monoamniotic Twin

Discussion

Ovarian hyperstimulation syndrome very rarely occurs in a spontaneous pregnancy. Iatrogenic OHSS occurs following HCG trigger for ovulation, hence is seen at about 3-5 weeks of amenorrhea. But spontaneous occurs between 8-14 weeks. Recently the Follicle Stimulating Hormone (FSH) receptor gene mutations are identified which leads to increased sensitivity to circulating HCG in spontaneous OHSS.[1]  De Leener has proposed a classification of spontaneous OHSS syndrome into three types according to clinical presentation and FSH receptor mutation.
Type I : Due to mutation in FSH receptor gene. The recurrence rate of spontaneous OHSS is very high in this type.
Type II : Due to high circulating HCG which is seen in multifetal pregnancies or in molar pregnancies, this is most common type.
Type III : Associated with maternal hypothyroidism.[2]
In pregnancies where ovulation induction is done by FSH, the follicular recruitment and enlargement is found during exogenous FSH administration and can lead to iatrogenic OHSS. In spontaneous pregnancies, the follicular recruitment and growth occur later due to abnormal stimulation of the mutated FSH receptor that is abnormally sensitive to normal levels of circulating HCG, produced by syncytiotrophoblast of pregnancy. Hence the spontaneous cases of OHSS generally develops at 8 weeks amenorrhea and resolves spontaneously at the end of the first trimester of pregnancy.[3]
In literature many cases were described as spontaneous pregnancy with OHSS and maternal hypothyroidism. In these cases it is assumed that high levels of thyroid stimulating hormone stimulate ovaries directly due to structural resemblance to FSH and can cause ovarian hyperstimulation.[4,5] In our case hypothyroidism was present. The pathophysiology of spontaneous OHSS associated with hypothyroidism is not well understood. The various theories are (a) in hypothyroid patients, there are high levels of circulating estriol produced through 16 hydroxylation  (less potent estriol) as against 2 hydroxylation pathways (more potent estriol) seen in normal patients. This causes decreased negative feedback regulation and excessive release of gonadotropins by ovary.  (b) High levels of thyroid stimulating hormone can directly stimulate ovaries in women with hypothyroidism and can cause ovarian hyperstimulation.[6]
OHSS can be classified according to the degree of severity of affection as mild, moderate severe and critical. Royal College of Obstetricians and Gynecologists in 2006 [7,8] classifies OHSS as mild OHSS, in which patients have symptoms of abdominal bloating and mild abdominal pain; moderate OHSS, when nausea, vomiting, moderate abdominal pain, is present with ultrasound evidence of ascites. and severe OHSS, identifiable by clinical ascites, renal affection  oliguria, hemoconcentration, PCV 45%, and hypoproteinemia; and critical OHSS, with tense ascites, oliguria or anuria, hematocrit > 55%, and leukocytosis (WBC > 25,000).
The management of OHSS depends on the degree of severity. Early recognition and prompt appropriate treatment will avoid serious consequences. Medical treatment, undertaken in first line, may be insufficient. Therapy is supportive, syndrome being self-limiting. Mild cases can be managed on OPD basis with daily measurement of weight and monitoring urinary output. Serial measurement of hematocrit, electrolyte and creatinine are done. Severe OHSS requires hospital admission and prompt management to replace lost intravascular volume and prevent its potentially fatal complications namely renal failure and thromboembolic events.[9] Sometimes drainage of fluid or debulking of ovarian cysts may be needed.[10]

Conclusion

OHSS is very rare but potentially serious complication. The exact etiology is not known conclusively but further research is going on. Certain factors like high levels of HCG hormone found in early pregnancy in cases of multifetal gestation and molar pregnancy and mutations in the follicle stimulating hormone (FSH) receptor gene, which leads to an increased sensitivity to HCG, may be some of the possible etiologies. Twin pregnancies and hypothyroid patients are more prone to OHSS. If we diagnose and manage spontaneous OHSS at the appropriate time we can prevent any severe complication.

Acknowledgment

Dr. Umesh Athawale, Athawale Diagnostic center for the ultrasound examinations.

References
  1. Delbaere A, Smits G, Olatunbosun O, Pierson R, Vassart G, Costagliola S. New insights into the pathophysiology of ovarian hyperstimulation syndrome- What makes the difference between spontaneous and iatrogenic syndrome? Hum Reprod. 2004 Mar;19(3):486–9.
  2. De Leener A, Montanelli L, Van Durme J, Chae H, Smits G, Vassart G, et al. Presence and absence of follicle-stimulating hormone receptor mutations provide some insights into spontaneous ovarian hyperstimulation syndrome physiopathology. J Clin Endocrinol Metab. 2006;91(2):555–62.
  3. Ozer Oztekin, Ferit Soylu, Orkan Tatli. Spontaneous ovarian hyperstimulation syndrome in a normal singleton pregnancy. Taiwan J Obstet Gynecol. 2006 Sep;45(3):272–5.
  4. Ayhan A, Tincer ZS, Aksu AT. Ovarian hyperstimulation syndrome associated with spontaneous pregnancy. Human Reproduction. 1996;11(8):1600–1601.
  5. Rothmensch S. Spontaneous ovarian hyperstimulation syndrome associated with hypothyroidism. Am J Obstet Gynecol. 1989;160:1220–1222.
  6. Taher BM, Ghariabeh RA, Jarrah NS, Hadidy AM, Radaideh AM, Ajlouni KM. Spontaneous ovarian hyperstimulation syndrome caused by hypothyroidism in an adult. Eur J Obstet Gynecol Reprod Biol. 2004;112:107–9.
  7. Jenkins JM, Drakeley AJ, Mathur RS. The Management of Ovarian Hyperstimulation Syndrome. Green-top Guideline. 2006 Sep;(5):1–11.
  8. Vloeberghs V, Peeraer K, Pexsters A, D'Hooghe T. Ovarian hyperstimulation syndrome and complications of ART. Best Pract Res Clin Obstet Gynaecol. 2009 Oct;23(5):691–709. [PubMed]
  9. Davis M, Kennedy R. Ovarian hyperstimulation syndrome: Aetiology, prevention and management. Reviews in Gynaecological and Perinatal Practice. 2006;6:26–32.
  10. Aboulghar MA, Mansour RT, Serour GI, Amin Y. Ultrasonically guided vaginal aspiration of ascites in the treatment of severe ovarian hyperstimulation syndrome. Fertil Steril. 1990 May;53(5):933–5.
Citation

Chhatrapati A, Nadkarni T, Mishra I, Jassawalla MJ. Ovarian Hyperstimulation Syndrome In a Spontaneous Twin Pregnancy With Hypothyroidism. JPGO 2016. Volume 3 No. 3. Available from: 

Vulval Leiomyoma: a Case Report

Author Information

Devalla A*, Warke HS**, Mayadeo NM***.
(* Second Year Resident, **Associate Professor; *** Professor; Department of Obstetrics and Gynaecology, Seth G S Medical College & KEM Hospital, Mumbai, India.)

Abstract

Vulval leiomyomas are rare in occurrence with as few as 300 cases reported in the literature. Smooth muscle tumors may arise from the erector pili muscle, the erectile tissue or at the insertions of the round ligaments and are usually well- circumscribed. Purely epithelioid lesions are identified only in the vulva and their hyalinisation is more common. We present a case of a 46 year old woman with bilateral multiple vulval masses with symptoms of discomfort while sitting and walking. Clinically it was diagnosed as a labial lipoma due to its soft consistency and surgical excision was done. Histopathology report was suggestive of leiomyoma with hyaline changes on microscopic examination.

Introduction

Extrauterine leiomyomas commonly affect the urinary bladder, labia majora, labia minora, ovaries and urethra; labia majora being the most common. Presence of concurrent uterine leiomyomas or a history of hysterectomy done for the same may be suggestive of the diagnosis.[1] Among those affected by these tumors, the mean age at presentation varies from 13 to 71 years. The average tumor size varies from 0.5 to 15 cm. With regard to pathologic origin, the tumors are thought to arise from the smooth muscle cells within erectile tissue or blood vessel walls or the remnants of the round ligament. These may also originate from the dartos muscle of the genitalia.[2] Vulval leiomyomas may be clinically misdiagnosed as Bartholin’s cysts, lipomas or fibromas.[3] Although considered benign tumors, they can be locally aggressive and can also metastasize. Most tumors are solitary and well circumscribed masses. Symptoms include pain and difficulty in walking, discomfort while sitting or difficulty in micturition. Owing to the size and appearance of the tumor, the patient may suffer from stress, anxiety and cosmetic issues. Treatment options include surgical excision and removal of the entire tumor, when symptomatic. Follow-up care with regular screening and examination are very important as the chances of recurrence are not uncommon. Excision of the labial masses was performed in our patient admitted with complaints of increase in the size of the swelling causing her discomfort while walking. The patient was discharged on day 7 and followed up after 2 weeks and 6 weeks.

Case Report

A 46 year old, married since 26 years, Para 3 was admitted with the complaints of multiple vulval masses since 1 year which gradually increased, largest mass being 5x4 cm  and smallest being 3x2 cm, causing discomfort while sitting and walking with occasional bleeding from the mass. There was no history of fever, vaginal discharge and bladder or bowel disturbances. The patient gave history of undergoing total abdominal hysterectomy 5 years ago for fibroid uterus with abnormal uterine bleeding. On general examination, vital parameters of the patient were normal. On local examination, multiple masses (grossly four masses) were present on both sides (two on either side) of the labia majora and smaller masses on the lateral walls of vagina near labia majora, the smallest being 3x2 cm and largest 5x5 cm. Clinically, the masses appeared to be lipomas or degenerated fibromyomas due to its soft consistency. All the routine investigations were normal. Ultrasonography of the pelvis was done which indicated hyperechoic lesions in the labia majora and essentially a normal pelvic study. On clinical examination, the innermost extent of one of the vulval mass on the left side could not be appreciated. Hence, a multiplanar-multiecho magnetic resonance imaging (MRI) with post-contrast imaging was done which was suggestive of a fairly large space occupying lesion involving the lower pelvis and perineal region measuring 10x7x5 cm in the vagina extending into the peri-urethral region and distinct from the rectum. The fat planes from the rectum were well-preserved. Antero-inferiorly, it was seen extending up to the vulva and superiorly till the vault. The patient was planned for excision of the vulval mass with an abdomino-perineal approach with surgical stand by. Procedure was done under spinal and epidural anesthesia. A 3 cm incision was made at the muco-cutaneous junction and a firm to cystic encapsulated mass was enucleated after dissection along its capsular plane. The size of the cavity formed after removal of the mass was reduced with interrupted sutures followed by closure of the overlying skin. Similar procedure was done for the other vulval masses. There was no extension of the vulval masses till the vault and into the pelvis. The vulval mass on the upper left side, clinically and on MRI thought to be extending into the pelvis was also enucleated completely through the vaginal approach. On cut section of the largest mass a whorl-like appearance with central degenerative changes were noted and our intra-operative diagnosis was that of a vulval leiomyoma. She had an uneventful postoperative recovery and was discharged on the seventh postoperative day. At 6 weeks follow up she was completely asymptomatic.
Histopathology report showed tissue bits greyish white in color with whorled pattern in gross appearance that was confirmed on microscopic examination that revealed features of leiomyoma with hyaline changes with no evidence of malignancy.


Figure 1. Vulval leiomyomas (arrows).


Figure 2. Vulval leiomyomas after removal. One leiomyoma is bisected (arrow) to show its archtecture.

Discussion

Leiomyomas are an important health concern as they are the most frequent indication for the performance of hysterectomy, accounting for nearly 2,40,000 such procedures in the United States.[4] About 40% of the patients with fibroids have chromosomal abnormalities that include deletions of chromosome 7 and trisomy of chromosome 12. The genes known to play an important role in their occurrence are CLAD1 and PLAG1 gene.[5] Vaginal leiomyomas were first described by Denys de Leyden in 1733.[6] Vulval leiomyomas are most commonly seen in the anterior vaginal wall. The size of the tumor may vary from 2 to 10 cm, the most common site being the labia majora. Myxoid changes and hyalinization are common over labia majora. Majority of the patients are asymptomatic though some may present with vaginal bleeding, dyspareunia, discomfort while sitting and urinary disturbances. Most tumors occur as solitary, well circumscribed and slow-growing masses.[7] Diagnosis may involve complete clinical examination with thorough evaluation of medical history. Ultrasound and MRI are most commonly used for diagnosis though histopathology of the surgical specimen is the gold standard for diagnosis. Treatment of choice would be surgical excision of the tumor with urethral catheterisation (to avoid damaging the urethra) if limited to the vulva. In cases of large tumors, abdomino-perineal approach is preferred. The patient needs to be explained about the chances for recurrence and hence the need for regular follow up.[8]

Conclusion

Vulvar leiomyomas are rare, seen more commonly in middle aged women. Concurrent uterine leiomyomas or a past history of abdominal hysterectomy for the same helps in its diagnosis. These tumors rarely metastasize but chances of their recurrence are known. Clinical examination, USG and MRI help in the diagnosis. However, histopathology of the surgical specimen is the gold standard. Surgical excision of the tumors is the treatment of choice.

References
  1. Szklarukj, Tamm EP, Choi H, Varavithya V. MR imaging of common and uncommon large pelvic masses. Radiographics 2003; 23(2): 403-24.
  2. Fasih N, Prasad SAK, Macdonald DB, Fraser-Hill MA, Papadatos D, Kielar AZ et al. Leiomyomas beyond the uterus: unusual locations, rare manifestations. Radiographics 2008, Nov-Dec;28(7): 1931-48.
  3. Kaufman RH, Gardner HL. Tumors of the vulva and vagina. Benign mesodermal tumors. Clin Obstet Gynecol 1965 Dec;8(4):953-81.
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  7. Cobanoğlu O, Gürkan Zorlu C, Ergun Y, Kutluay L. Leiomyosarcoma of the vagina.Eur J Obstet Gynecol Reprod Biol. 1996 Dec 27; 70(2): 205-7.
  8. Bae J H, Choi SK, Kim JW. Vaginal leiomyoma: A case report and review of the literature. J Women's Med. 2008;1:92–4.
Citation

Devalla A, Warke HS, Mayadeo NM. Vulval Leiomyoma: a Case Report. JPGO 2016. Volume 3 No. 3. Available from: http://www.jpgo.org/2016/03/vulval-leiomyoma-case-report.html

Ovarian Fibroma: An Unusual Case

Author Information

Manjrekar VM*, Parulekar SV**
(* Second year Postgraduate student, ** Professor and Head, Department of Obstetrics and Gynaecology, Seth G.S. Medical College & KEM Hospital, Mumbai, India.)

Abstract

The ovarian fibroma is a rare benign tumor which grows from the connective tissue of the ovarian cortex. Among the sex cord-stromal tumors, fibromas are the most commonly encountered subtype accounting for almost two-thirds of neoplasms in this group. We present an unusual case of Fibroma of the ovary presenting later in life.

Introduction

Fibromas, thecomas and fibro-thecomas are rare benign tumors which grow from the connective tissue of the ovarian cortex. The mean age of occurrence is 45-55 years.[1,2] They are classified under Sex cord-stromal tumors which also include tumors composed of granulosa cells, theca cells, Sertoli cells, Leydig cells, and fibroblasts of stromal origin, singly or in various combinations.[3,4] According to the WHO, thecoma-fibroma tumors are classified into the following categories:
Thecoma-fibroma group
  • Thecoma, not otherwise specified
-Typical
-Luteinized
  • Fibroma
-Cellular fibroma
-Fibrosarcoma
  • Stromal tumor with minor sex cord elements
  • Sclerosing stromal tumor
  • Signet ring cell stromal tumor
  • Unclassified (fibrothecoma)
Fibromas are benign tumors developing in the postmenopausal women (over 40 years of age). They usually present as unilateral, solid, firm to hard masses with a bosselated external surface, off white to pearly white in color. They can be bilateral, and usually measure more than 6 cm in diameter. The consistency may be soft and cyst formation is common if edema is associated with the tumor.[5,6] The cut surface shows a whorled pattern with a grayish white stroma and occasional areas of calcification. We present an unusual case of fibroma of the ovary presenting later in life and its surgical management.

Case Report

A 65 year old postmenopausal woman was referred from a peripheral hospital in view of a large left ovarian cyst (6.2x7.8 cm) diagnosed on ultrasonography (plates not available). She had pain in the left side of the lower abdomen for 7 months, dull aching in nature with no aggravating and relieving factors. She also perceived a mass in her lower abdomen which had gradually progressed in size over 3 months, non tender and of firm consistency. She was a known case of hypertension diagnosed 12 years ago, receiving T. Amlodipine 5mg OD and T. Aspirin 150 mg OD. She had attained menopause 16 years ago, her previous cycles being regular. She had 3 full term normal deliveries followed by tubal ligation done 24 years ago. On examination her vital parameters were within normal limits, blood pressure was 130/80 mm Hg, and general and systemic examination revealed no abnormality. Per abdomen examination revealed a solitary, intraperitoneal, firm to hard mass of 14 to 16 weeks' size arising from the pelvis. It had free side to side mobility. The cervix and vagina appeared healthy on per speculum examination. On vaginal examination the uterus appeared to be of normal size, pushed to the left and posteriorly by an anterolateral firm to hard mass of about 7x8 cm. A provisional diagnosis of left ovarian tumor was made.
Pap smear was atrophic inflammatory. Her hemogram, plasma sugara levels, liver and renal function test results were within normal limits. Contrast enhanced computerized tomography (CECT) of the abdomen and pelvis showed a well defined left ovarian cyst measuring 10 cm in diameter, causing smooth extrinsic impression on the fundus of the urinary bladder. The fat planes were preserved and minimal fluid was noted in pouch of Douglas. Tumor marker CA-125 was 14.49 U/ml.


Figure 1. CECT showing ovarian mass.


Figure 2. Ovarian fibroma is seen during laparotomy.

An exploratory laparotomy was performed under epidural anaesthesia through a vertical midline infraumbilical incision. Peritoneal fluid was collected for cytology. In situ findings were of a normal sized uterus with a normal right ovary and fallopian tube. A 6x8x9 cm solid, firm to hard, off white to pearly white colored mass was noted on the left side arising from the left ovary, free from other pelvic structures. Left salpingo-oophorectomy was done and specimen sent for frozen section. Frozen section was suggestive of sex cord stromal tumour favoring thecoma-fibroma. Total abdominal hysterectomy with right salpingo-ophorectomy was done. Infracolic omentectomy was also performed to complete the procedure. Post operative course was uneventful and patient was discharged on the 5th post operative day.
Final histopathology report revealed a mildly cellular spindle cell tumor replacing the entire left ovary. The cells were arrange in multiple fascicles, storiform pattern and in sheets suggestive of left ovarian sex cord stromal tumour – fibroma.

Discussion

Ovarian fibromas are predominantly benign tumors occurring in the postmenopausal group. Most of these tumors are asymptomatic with the clinical presentation being of vague abdominal pain and discomfort. Sometimes the patient may appreciate a mass in her abdomen which is firm to hard in consistency. They are stromal tumors composed of spindle, oval or round cells producing collagen[7]. Fibromas are usually solid, unilateral, spherical or oval, with a bosselated surface and a glistening white or pearly white appearance.[8] Preoperative diagnosis is difficult. Diagnosis can be aided by imaging and tumor markers. CA 125 is not specific but can be used to suspect malignant change. Elevated CA-125 is usually associated with the presence of ascites[10,11] CECT is helpful in delineating the tumor, its origin and preservation of fat planes. Fibroma should be differentiated from stromal hyperplasia and fibrothecoma. Diagnosis is confirmed only on histopathology. The treatment consists of surgical resection of the tumor which is associated with very low recurrence rates and laparoscopic surgery can be an effective and safe alternative approach. Laparoscopic assisted vaginal hysterectomy can also be done , the tumour being removed in an endobag.[12]
The case presented here was unusual in some respects. The solid tumor was perceived to be a cystic tumor on ultrasonography as well as on CECT. There appears to be an error of both of these investigations. The tumor was much larger than the average size of a fibroma at the time of presentation. Its frozen section report did not diagnose a fibroma and hence, especially considering her age, a total abdominal hysterectomy with bilateral salpingo-oophorectomy with an infracolic omentectomy was performed, though the peritoneal fluid did not show any malignant cells.

Conclusion

Ovarian fibromas are benign tumours of the the ovary, difficult to diagnose pre-operatively and can clinically and biochemically mimic ovarian cysts, tuboovarian mass, uterine myoma or ovarian malignancy. CA 125 levels are not specific for diagnosis but with imaging are helpful to decide the course of treatment which usually consists of surgical resection either by laparotomy or laparoscopic approach.

References
  1. Sivanesaratnam V, Dutta R, Jayalakshmi P. Ovarian fibroma--clinical and histopathological characteristics.Int J Gynaecol Obstet. 1990 Nov. 33(3):243-7.
  2. Târcoveanu E, Niculescu D, Bradea C, Dimofte G, Vasilescu A, Ferariu D, Crumpei F. Incidence and management of the ovarian fibroma and thecoma. Experience of The First Surgical Clinic Iaşi]. Chirurgia (Bucur). 2006;101(3):325-30.
  3. Tavassoli FA, Mooney E, Gersell DJ, McCluggage WG, and Konishi. Sex cord-stromal tumors. Pathology and Genetics of Tumors of the Breast and Female Genital Organs. Lyon, France: IARC Press; 2003.
  4. Mircea R, Anton E, Anton C, Tarniceriu C, Nedelcu AH, Pricop FZ. Specific features of the stromal ovarian tumors. Rev Med Chir Soc Med Nat Iasi 2012 Oct-Dec. 116(4):1123-30.
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Citation

Manjrekar VM, Parulekar SV. Ovarian Fibroma: An Unusual Case. JPGO 2016. Volume 3 No. 2. Available from: http://www.jpgo.org/2016/03/ovarian-fibroma-unusual-case.html

Intrapartum Pubic Diastasis

Author Information

Dwivedi JS*, Prasad M**, Gupta AS***.
(* Third Year resident, ** Assistant Professor, *** Professor, Department of Obstetrics and Gynaecology, Seth G.S. Medical College and K.E.M. Hospital, Mumbai, India.)

Abstract

There have been reported cases of pubic diastasis. It is a recognized complication of pregnancy. In the modern era, pubic diastasis is rare. It may present in antepartum, intrapartum or postpartum period and might cause serious distress to the patient.

Introduction

Pubic diastasis is defined as the separation or gap between the pubic bones at the symphysis without concomitant fracture. A width of more than 10mm is considered diagnostic. We report a case of postpartum diagnosed pubic diastasis managed conservatively.

Case Report

A 26 yr old, registered, primigravida married since one year, with gestational hypothyroidism presented in spontaneous active labor at 38.2 weeks of gestation. She had regular uneventful antenatal follow up. In the 7th month of gestation, she had undergone treatment with oral metronidazole for Blastocystis Hominis cysts in stools. For gestational hypothyroidism, she was on tablet levo thyroxine 50 micrograms. Pelvis was clinically adequate. Fetus was in the left occipito-anterior position. First stage of labor lasted around 8 hours. She had inco-ordinate uterine activity at the end of 1st stage of labor. Dilute oxytocin was started. Titration was not needed. In view of poor maternal bearing down efforts, after approximately 2 1/2 hours of second stage of labor, outlet forceps application was done, delivering a male child of 3.608 kg with Apgar score of 9/10. Application of the outlet forceps was very easy. There were no cervical tears and episiotomy was uneventfully sutured. There was no postpartum hemorrhage.
Few hours post-delivery, the patient complained of difficulty in walking, squatting, lifting of legs and had a waddling gait. She had suprapubic pain and tenderness. Patient was given adequate oral analgesia. The clinical impression of postpartum pubic diastasis was confirmed by radiological findings of separation of pubic bones. A gap of around 3.5cm on anteroposterior (AP) view of the pelvis was seen at the pubic symphysis. Ultrasonography ruled out retro pubic hematoma. Orthopaedic reference was taken for the same and a pelvic binder was tied. History was reviewed and patient reported using veil constantly, in accordance with her religious tradition. In light of this, bone mineral density (BMD) was assessed using DEXA (dual energy x-ray absorptiometry) scan. Knee, hips and lower limbs were assessed, which revealed normal BMD. Patient was discharged on analgesics and pelvic binder and advised to follow up after 6 weeks. Post 6 weeks of delivery, patient stopped using the binder. X-ray pelvis AP view was repeated in the 7th week. It showed decrease in the gap between the two bones as compared to previous one. Patient was symptomatically better, had a normal gait and had resumed all her daily routine activities.


Figure 1. Radiograph of the pelvis AP view at diagnosis. The yellow line shows the pubic symphysis separation .


Figure 2. Radiograph of the pelvis AP view after treatment. The red line shows the decrease in the distance between the two pubic bones.

Discussion

An incomplete or a total rupture of the symphysis pubis is known as pubic diastasis. Pubic diastasis is defined as the separation of the pubic bones by more than 10 mm.[1] The first case of pubic symphysis separation during labor was reported in 1932.[2] Pubic diastasis continues to be reported from the developing countries even till recently, following uncomplicated vaginal delivery.[3] While a large number of sporadic case reports continue to be reported, few prospective studies are available which have studied the incidence of this condition. One such study estimates the incidence to be 1 in 385 deliveries.[4] The incidence can range from 1:300 to 1:30000. Various associative factors are fetal macrosomia, cepahalopelvic disproportion and epidural anesthesia. Knowledge of anatomy helps better understanding of this condition. Pubic symphysis is a synovial joint separated by a fibrocartilaginous disc and supported by superior and inferior or arcuate ligaments, which are mainly responsible for the strength of the joint.[5] Experiments have suggested that a force between 400-2600 pounds are required to produce symphyseal separation.[6] The physiological process of joint relaxation starts at around 10 weeks and reaches maximum at or near term, and returns to normal by 4 to 12 weeks' postpartum. Relaxin plays a vital role in the maternal accommodation of pregnancy, which includes pelvic joint relaxation.[7] It is of interest to note that relaxin is recently reported to be related to pre-eclampsia and preterm labor.[8]
Pubic diastasis characteristically presents as suprapubic pain, tenderness, swelling, and edema and radiating pain. Pseudoparalysis may occur due to voluntary splinting of lower extremities. Waddling or duck like gait have also been described.[9] Simultaneous disruption of pubic symphysis and sacroiliac joint during vaginal birth has also been reported. Hence a survey of the other bones should also be done.[10] While pelvic radiographs and CT are the initial imaging modalities, it has been recently reported that pelvic radiographs magnifies the pubic diastasis. CT screening is more accurate. However, CT scan was not required in our case.[11] Complications such as retropubic hematoma formation, pubic osteomyelitis, urinary and fecal incontinence have been reported. Conservative management using analgesics and physiotherapy is usually associated with complete recovery. Operative interventions might be required in cases with symphyseal separation of more than 3 cm.[4,9]
In case of next pregnancy, the mode of delivery will have to be discussed with the patient due to traumatic past and patient’s fear of recurrence of symptoms. Vaginal delivery can be advised, leaving a choice upon the patient with discussion about prevention and therapeutic possibilities.[12] The obstetrician conducting the delivery should be alert to the recurrence or a gap developing in the sacroiliac joints or a resultant fracture in case the gap widens significantly to prevent instability to the pelvic girdle.
In our patient, DEXA scan showed no osteoporotic changes. A report which studied bone mineralization patterns reported an increase in remodeling with bone demineralization in pregnant mothers.[13] Though not reported as yet, we postulate that DEXA scan may help in counseling the patient regarding decision making in the future pregnancies. 

References
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  7. Vrachnis N, Grigoriadis C, Siristatidis C, Vlachadis N, Balakitsas N, Mastorakos G, et al.The Janus face of maternal serum relaxin: a facilitator of birth, might it also induce preterm birth? J Matern Fetal Neonatal Med. 2015;28(18):2187-91. doi: 10.3109/14767058.2014.981804. Epub 2014 Nov 18.
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  9. Mhaske N, Madhva Prasad S, Kharat D, Fonseca MN. A Case Of Postpartum Pubic Diastasis. JPGO 2015. Volume 2 No. 3. Available from:  http://www.jpgo.org/2015/03/a-case-of-postpartum-pubic-diastasis.html
  10. Çıçek H, Keskın HL, Tuhanıoğlu Ü, Kiliçarslan K,Oğur HU. Simultaneous Disruption of the Pubic Symphysis and Sacroiliac Joint during Vaginal Birth. Case Reports in Orthopedics. Volume 2015 (2015), Article ID 812132, 5 pages. Available from: http://dx.doi.org/10.1155/2015/812132.
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  13. Martina DM, Biasioli A, Vascotto L, Rinuncini D, Menegato AA, Liva S. Bone ultrasonometry measurements during pregnancy. Arch Gynecol Obstet. 2010; 281(3): 401-7.
Citation

Dwivedi JS, Prasad M, Gupta AS. Intrapartum Pubic Diastasis. JPGO 2016. Volume 3 No. 3. Available from: http://www.jpgo.org/2016/03/intrapartum-pubic-diastasis.html