Volume 4 Issue 2, February 2017

Editorial
Gupta AS

Salmonella Infection Of Ovarian Endometrioma in A Case of Cervico-Vaginal Atresia And Bicornuate Uterus
Mehta D, Parulekar SV.

Torsion Of A Paraovarian Cyst
Mehta N, Parulekar SV.

Atypical Eclampsia With PRES Syndrome
Nasare P, Madhva Prasad S, Gupta AS.

Vulvar Abscess in a Pregnant Woman
Thakare R, Thosar MA, Gupta AS.

Lost and found - Lippes Loop
Shah NH, Doshi D, Kumari K, Paranjpe SH.

Botryomycosis of Anterior Abdominal Wall following Cesarean Section
Chhonkar A, Nayak CS, Tambe S.

Inherited Thrombophilia In Pregnancy: Management And Outcome
Kalappa SB, Shende D, Chauhan AR.

Exaggerated Placental Tissue Site Reaction: A Diagnostic Dilemma
Ostwal P, Shende D, Dhokia T, Chauhan AR.

Successful Management Of a Misdiagnosed Case Of A Transverse Vaginal Septum
Kambhampati L, Mali K, Satia M.

Editorial

Gupta AS

Surgical wounds leave scars. The type of scar that forms depends on wound healing that in turn is affected by variable factors. Patient’s having co morbid conditions like obesity, diabetes, malignancy and vascular insufficiency affect the wound healing and thus the scar formation. Suture materials used, site of the surgery, infections modify the wound healing.  Well healed hair line invisible scars are ideal and provide cosmetic satisfaction for the patients especially women. However, this is not a universal outcome. Surgical wounds that are mainly clean cut incisions heal by primary intention. When a wound is inflicted and sutured various suture materials are used. An inflammatory reaction is triggered, hyperemia at the surgical site brings in the fibroblasts that start laying down collagen for wound healing. However, if the inflammatory reaction is severe, or the suture materials are more reactive or micro organisms have been inoculated at the wound site then this acute inflammatory reaction is so severe that it can lead to hyper granulation tissue formation resulting in poor wound healing, wound breakdown or formation of abnormal scars. Production of excessive collagen can result in the formation of hypertrophic scars or keloids. Hypertrophic scars are formed due to the heaping up of excessive collagen. The scar is elevated above the skin level but it does not extend beyond the scar margins. It does not keep growing not does it result in pruritis. They may regress over time. Biopsy taken from these scars show fibroblasts arranged in a disordered pattern but the fibrous tissue is laid in whorls. Keloid scars are also thickened but they usually go beyond the wound margins and can grow in various directions giving the appearance of projections and claws. They continue to grow and are many a times tender and cause intense itching. Biopsy from these scars show plenty of collagen strands that are infiltrated with eosinophils.

Localized chronic inflammatory reactions mainly to foreign material like sutures, mesh and tape, or to infections result in focal collections and nodules that are known as granulomas. Suture granulomas usually heal well after the foreign body that is the cluster of usually non absorbable sutures are removed. The other rarer type of scar granulomas are due to scar endometriosis, scar sarcoidosis, scar botryomycosis and scar actinomycosis.

Scar endometriosis is commonly encountered in women who have undergone procedures like cesarean births, hysterectomies, myomectomies, hysterotomy or even tubal liagation. These are typical nodular, tender masses formed on skin scars, fascia or muscle. These enlarge and become tender cyclically during menstruation and this typical presentation pinpoints the diagnosis. Wide excisional biopsy treats and conforms the diagnosis. The tissues are seen to be infilterated with the endometrial glands and stroma. Non caseating granulomas that develop in old, even remote surgical, injection or tatoo scars or cause reactivation of the old scars should alert the clinicain to the presence of scar sarcoidosis. Scar actinomycosis is a rare, chronic, suppurative, inflammatory, granulomatous condition that occurs due to infection with microareophilic or anerobic bacteria. It can also cause sinuses, fistulae and abscesses. Scar Botrymycosis is another chronic non inflammatory reactions to the bacterial antigens.

Clinicians when faced with these unusual cases of wound healing and abnorm scars shod keep the above differentials r proper management.

This issue has an interesting article on scar botrymycosis and I hope our dedicated followers and readers enjoy reading all the articles.

Salmonella Infection Of Ovarian Endometrioma in A Case of Cervico-Vaginal Atresia And Bicornuate Uterus

Author Information

Mehta D*, Parulekar SV**.

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

Abstract

Pelvic endometriosis is a known complication of mullerian duct anomalies of obstructive type. Ovarian endometriosis may manifest as an endometrioma. Infection of ovarian endometrioma by Salmonella is very rare. We present a case of genital outflow tract agenesis and ovarian endometrioma which developed asymptomatic Salmonella infection. This is the first case of this type in the world literature.

Introduction

Mullerian duct anomalies of obstructive type like vaginal atresia or transverse septum and cervical atresia in presence of a functioning uterus result in development of hematometra, which progresses to formation of hematosalpinx and retrograde spill leading to pelvic endometriosis. Ovarian endometrioma may get infected as a part of pelvic infections, which can be ascending or acquired by hematogenous route. Salmonella infection of ovarian endometrioma is known to occur, but is rare. We present a case of bicornuate uterus, agenesis of cervix and upper vagina, left sided ovarian endometrioma and asymptomatic Salmonella infection of the endometrioma.

Case Report

A 23 year old unmarried woman presented to the outpatient department with complaints of primary amenorrhoea with cyclical abdominal pain for 5 years. The pain was of a dull aching nature lasting for 3-4 days occurring cyclically every month. It was not associated with vomiting or gastrointestinal complaints. There was no history of delayed menses in her siblings or mother,  delay in development of secondary sexual characteristics, fever, sore throat or urinary complaints. No other medical or surgical history. General and systemic examination revealed no abnormality. Breast development was of Tanners stage V. Axilllary and pubic hair was normal. The abdomen was soft and supple. Her external genitalia and external urinary meatus were normal. The vagina was 1cm in depth, ending as a blind pouch. Rectal examination showed a firm nodule measuring 3 cm in diameter. Cervix was not felt. All routine blood investigations within normal limit. Serum biochemistry within normal limits.

Serum FSH was 4.37 mIU/ml, LH 16.7mIU/ml and prolactin 10.20 ng/ml. Abdominopelvic  ultrasonography showed the uterus measuring  5.3x2.7x3 cm,  normal  right ovary and a cyst measring  5.3x5.2 cm in the left ovary. The kidneys and urinary tract were normal. There was some free fluid in in the pouch of Douglas. Magnetic resonance imaging (MRI) showed a bulky left ovary with a 54x50x60 mm well defined hemorrhagic cyst. Another cyst was noted superior to this cyst. The uterus was displaced to the left by the cyst. The cervix was not seen. The  right ovary was normal. A diagnosis of hypoplastic cervix with left ovarian endometrioma was made.

Figure 1. Ultrasonography of pelvis: The left ovary shows a complex cyst, while the right ovary is normal.

Figure 2. Ultrasonography of pelvis: The uterus (B-B in sagittal view, C-C in transverse view) has some blood in it. The cervix and upper vagina are absent.

The patient and her relatives were counselled about the need for removal of the endometrioma and hysterectomy in view of cervical atresia. However they opted for only removal of the endometrioma. Investigations for fitness for surgery showed results in the normal range. A laparotomy was performed. The findings are shown in figure 1. There was a bicornuate uterus with atresia of the cervix and upper vagina. The two uterine horns were separate, connected by a crescentic fibrous band in the coronal plane. The left horn measured 5x5 cm and was soft, cystic. The right horn measured 3x2 cm and was firm. Both fallopian tubes were normal. Left ovary was replaced by a 5 cm diameter cyst, adherent to the sigmoid colon by flimsy adhesions. The adhesions were separated. The cyst ruptured during enucleation and thick, pultaceous, gray odorless, purulent fluid. It was sent for microbiologic study and for TB-PCR test. The cyst lining was peeled off, and the ovary was reconstructed. Peritoneal wash was given with normal saline. Salmonella typhi was grown from the fluid. The patient was treated with ceftriaxone, gentamicin and metronidazole postoperatively. The same antibiotics were continued after obtaining the culture report because the patient was well and did not have any symptoms and signs of intraperitoneal or any other sepsis. She recovered fully. Histopathology of the cyst confirmed the diagnosis of ovarian endometrioma.

Figure 3. Findings at laparotomy: LH - left horn of uterus, LT – left fallopian tube, LO -  reconstructed left ovary, RH - right uterine horn, RT -  right fallopian tube, RO – right ovary.

Discussion

Salmonella can cause gastroenteritis, enteric fever, septicemia and localized suppuration. Salmonella is rarely the causative organism in an ovarian abscess. It reaches the ovary either directly from bowel in contact with it or by hematogenous route. In most of the cases there are predisposing factors like ovarian simple cyst,  endometrioma, benign cystic teratoma, or cystadenoma.[1,2,3] Wang et al reported an ovarian abscess secondary to an enteroovarian fistula. [4] Usually Salonella infection is symptomatic. But Kemmann et al reported a case of an ovarian endometrioma which grew Salmonella, when the patient had no features of sepsis.[5]

In the case presented, the patient had cervical and upper vaginal atresia and bicornuate uterus. The left horn was functional and obstruction to drainage of menstrual blood probably resulted in pelvic endometriosis by retrograde spill. The left ovary had developed endometrioma. The left uterine horn was not functional. Pelvic infections are usually ascending. But that was not possible in this case because there was no communication of the pelvis with the exterior. She did not have any gastrointestinal infection or Salmonella septicemia. Hematogenous spread of Salmonella to the ovary from a past forgotten infection might have remained dormant and manifested later in this case. The patient was fortunate in not getting infection of her left hematometra, either preoperatively or postoperatively. It is quite likely that she will develop more lesions of endometriosis. The left uterine horn was placed quite high in the pelvis and pulling it down for creating a new cervix and opening it into a neovagina was not possible. She would have done better by undergoing removal of the left hematometra. But she wanted to retain the uterus despite extensive counseling. It is likely that she may require another surgery for her progressive hematometra.  She has been advised to follow up regularly and have abdomino-pelvic ultrasonography at least every 6 months, so as to detect progression of the hematometra and pelvic endometriosis. If she does so, further surgery can be offered.

Acknowledgment

We than Dr Shreshthaqa Mahanti for taking operative photograph.

References

1. Cohen JI, Barlett JA, Cory GR. Extra-intestinal manifestations of Salmonella infections. Medicine. 1987;66:349-388.
2. Kubota T, Ishi K, Takeuchi H. A study of tubo-ovarian and ovarian abscesses, with a focus on cases with endometrioma. J Obstet Gynaecol Res. 1997;23(5):421-426.
3. Sidahmed H, Hassan A. Salmonella infection of ovarian dermoid cyst. Br Med J. 1975;3(5976):140.
4. Wang CN, Lai CH, Hsueh S, Chou HH. Ovarian endometrioma complicated by a Salmonella abscess caused by an enteroovarian fistula: a case report. J Reprod Med. 2005 Nov;50(11):871-3.
5. Kemmann E, Cummins L, Dietzel H. Salmonella abscess in an ovarian endometrioma. N J Med. 1993 Aug;90(8):596-7.

Citation

Mehta D, Parulekar SV. Salmonella Infection Of Ovarian Endometrioma in A Case of Cervico-Vaginal Atresia And Bicornuate Uterus. JPGO 2017. Volume 4 No. 2. Available from: http://www.jpgo.org/2017/02/salmonella-infection-of-ovarian.html

Torsion Of A Paraovarian Cyst

Author Information

Mehta N*, Parulekar SV**

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

Abstract

Paraovarian cysts are difficult to be differentiated from ovarian cysts clinically, and even on imaging. Their torsion is rare. But if not diagnosed and treated in time, there can be permanent loss of the fallopian tube and ovary of the same side due to ischemic necrosis. We report a case of torsion of a left paraovarian cyst in a 24-year-old girl. 

Introduction

Paraovarian cysts account for approximately 5 % of adnexal cysts.[1] Most paraovarian/ paratubal cysts are non-neoplastic distended remnants of the mesonephric duct, paramesonephric duct or mesothelium of the broad ligament. They are unilocular, thin walled, containing clear fluid. They are usually small, though rarely they may attain a large size and cause abdominal pain.[2] Women with paraovarian cysts are most commonly asymptomatic. Rarely they may present with complications such as hemorrhage, rupture or torsion.  Infrequently, adjacent structures like the ovary, fallopian tube or infundibulopelvic ligament may undergo torsion with it. We present a case of a paraovarian cyst torsion with four turns, in which both the ovary and fallopian tube were conserved successfully.

Case Report

A 24-year-old nullipara, married for 8 months, came with complaints of pain in abdomen and 2-3 episodes of vomiting since one day. These complaints were relieved on giving analgesics and antiemetics. The patient had no other symptoms. She was postmenstrual. Her medical and surgical history was noncontributory. Her vital parameters were within normal limits. The general and systemic examination revealed no abnormality. On abdominal palpation, there was no tenderness, guarding or rigidity and no mass was felt. On per speculum examination, the cervix and vagina were healthy. On per vaginal examination, a mass of 5-6 cm diameter was felt in left anterior fornix, which was smooth, firm and slightly tender, and the uterus was displaced to the right and posteriorly. Ultrasonography (USG) revealed a left paraovarian cyst of 9.4 x 4.6x 7 cm. The left paraovarian vessels showed low resistance flow in the ovarian artery and normal flow in veins, indicating no occlusion of blood flow to the left paraovarian cyst and adnexal sructures. The patient’s investigations for fitness for anesthesia showed normal results. An exploratory laparotomy was performed. A left paraovarian cyst of 6-7 cm diameter was found which had undergone torsion with 4 twists, along with the distal part of the left fallopian tube. The part of the fallopian tube adjacent to the cyst was congested. Both ovaries and the uterus were normal. The torsion of the cyst was reversed by untwisting the turns, the cyst was separated from the cyst wall and sent for histopathology.  The redundant cyst wall was excised and mesosalpinx reconstructed with polyglactin 1-0 in simple interrupted sutures. The left fallopian tube was congested but viable. So it was preserved. The patient made an uneventful recovery. Histopathological examination of the cyst confirmed the diagnosis of torsion of a paraovarian cyst.

Figure 1. Operative findings: large, congested left paraovarian cyst (POC), normal uterus (U), left ovary (LO), right ovary (RO), and right fallopian tube (RFT) are seen.

Discussion

A paraovarian cyst arises from the mesothelium of the broad ligament, remnants of mesonephric duct, or remnants of paramesonephric duct.[3] Torsion of a paraovarian cyst is rare. It is more common in the reproductive age group and three times more common during pregnancy because the cyst gets elevated out of the pelvis due to growth of the uterus.[4] As the cyst lies in broad ligament and has no pedicle of its own, when it undergoes torsion, the ipsilateral fallopian tube and ovary being close to it, may also undergo torsion along with it. It is difficult to differentiate between paraovarian cyst and ovarian cyst clinically. USG and magnetic resonance imaging (MRI) are useful in making the diagnosis, though sometimes the diagnosis may be difficult even with imaging.[5,6,7] A high index of suspicion and prompt surgery is recommended to salvage the fallopian tube and ovary. Other complications of paraovarian cysts include hemorrhage, rupture, infection and neoplastic change. Previously it was believed that untwisting the adnexa could lead to increased risks of thromboembolism. Presently, there is growing evidence that unwinding the involved adnexa to observe for tissue reperfusion and viability is safe.[5] McGovern  et al reported that the occurrence of pulmonary embolism was 0.2 % in cases of adnexal torsion, and these cases were associated with adnexal excision, none to untwisting of the pedicle.[6] The adnexa may be preserved regardless of their appearance following detorsion. A functional integrity and subsequent pregnancy has been reported in almost 95%, with no increased postoperative morbidity.[7] Thus, resection of the associated cyst and salvage of the involved adnexa are the treatment goals.

References

1. Atal R: Torsion of Paraovarian Cyst Resulting in Secondary Torsion of Ovary and Fallopian Tube. International Journal of Obstetrics and Gynaecology Research (IJOGR) 3(8): 432-437, 2016.
2. F. Steinback, Kauppila A. Development and classification of paraovarian cysts: an ultrastructural study. Gynecol Obstet Invest, 12 (1981), pp. 1–10.
3. Honore LH, O’Hara KE. Serous papillary neoplasms arising in paramesonephric paraovarian cysts. A report of 8 cases. Acta Obstet Gynecol Scand, 59 (1980), pp. 8–525.
4. Puri M, Jain K, Negi R. Torsion of para-ovarian cyst: a cause of acute abdomen. Indian journal of medical sciences. 2003 Aug 1;57(8):361.
5. Ghossain MA, Braidy CG, Kanso HN, Farah K, Klein-Tomb L, Trak-Smayra V, Suidan JS, Elhage A, Atallah D, Abboud J. Extraovarian cystadenomas: ultrasound and MR findings in 7 cases. Journal of computer assisted tomography. 2005 Jan 1;29(1):74-9.
6. Gopal K, Lim Y, Dobson M , Keating P, Stringfellow H. A case of torted parafimbrial cyst on MRI: case report and review of literature. Br J Radiol, 79 (2006), pp. e208–e210, 
7. Barloon TJ, Brown BP, Abu-Yusuf MM, Warnock NG. Paraovarian and paratubal cysts: preoperative diagnosis using transabdominal and transvaginal sonography. J Clin Ultrasound. 1996;24:117–22.
8. Sanfilippo JS, Rock JA. Surgery for benign disease of the ovary. In Rock JA, Jones HW III, editors. Te Linde’s operative gynaecology. 11th edition. New Delhi;Wolters Kluwer Pvt Ltd 2015, pp 607.
9. McGovern PG, Noah R, Koenigsberg R, Little AB. Adnexal torsion and pulmonary embolism: case report and review of the literature. Obstetrical & gynecological survey. 1999 Sep 1;54(9):601-8.
10. Cohen SB, Oelsner G, Seidman DS, Admon D, Mashiach S, Goldenberg M. Laparoscopic detorsion allows sparing of the twisted ischemic adnexa. The Journal of the American Association of Gynecologic Laparoscopists. 1999 May 31;6(2):139-43.

Citation

Mehta N, Parulekar SV. Torsion Of A Paraovarian Cyst. JPGO 2017. Volume 4 No. 2. Available from: http://www.jpgo.org/2017/02/torsion-of-paraovarian-cyst.html

Atypical Eclampsia With PRES Syndrome

Author Information

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

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

                           

Abstract 

The clinical course of patients having preeclampsia and eclampsia is often unpredictable. A case of eclampsia who developed multiple seizures in the intrapartum and immediate postpartum period, that required multiple anti-epileptics is described. The concept of “delta hypertension” is mentioned in brief and the features of posterior reversible encephalopathy syndrome (PRES) are discussed. 

Introduction

Magnesium sulphate is the recommended drug for prevention and management of seizures in severe preeclampsia/ eclampsia. When a patient convulses despite being on magnesium sulphate, additional anti-epileptics may be considered, and further neurological evaluation is mandated. 

Case Report

A 23 year old, primigravida, in a non-consanguineous marriage, 40 weeks and 3 days of gestation presented to emergency as a diagnosed case of pre-eclampsia. She complained of abdominal pain and vaginal discharge for few hours. She was perceiving good fetal movements. She was on capsule nifedipine 10 mg 6 hourly, that had been started 2 weeks prior to this presentation when pre-eclampsia was detected. She had taken discharge against medical advice during her prior admission. 

On examination, patient was conscious, oriented, and afebrile, with normal pulse rate and a blood pressure of 130/80 mm of Hg measured in the right arm in the supine position. Cardiovascular and respiratory system examination were unremarkable. Abdominal examination showed a full term gravid uterus with live fetus in vertex presentation with a heart rate of 140 beats/ minute (bpm) and minimal uterine activity.  On vaginal examination, internal os was 2 cm dilated, 40% effaced, with vertex at -1 position. Clear liquor was draining. Pelvis was adequate. Urine albumin with dipstick was 2+ and deep tendon reflexes were normal. There were no premonitory signs or symptoms of impending eclampsia. She was then admitted to labor ward, anti-hypertensives were continued, fetal heart sounds, blood pressure (BP), urine output and other parameters were monitored. Investigations (complete blood count, liver and renal function tests, coagulation profile) were sent and were found to be within normal limits. For around 5 hours post admission, highest BP recorded was 140/90 mm of Hg, and uterine activity progressively increased. Then, persistent variable decelerations up to 90 bpm from a baseline of 130 bpm ensued. On vaginal examination internal os was 3 cm dilated, 40% effaced and vertex was at station -1. Decision of emergency cesarean section in view of fetal distress in first stage of labor was taken. Just as she was being shifted to the operation theater, she developed generalized tonic clonic convulsions. The BP recorded immediately post convulsion was 170/110 mm of Hg. Injection magnesium sulphate by Zuspan regimen, (loading dose of 4 grams followed by maintenance dose of 1 gm/hour was started). Tablet labetalol 100 mg was started. Ophthalmology reference taken for fundus examination was suggestive of no papilledema. She and her relatives then denied consent for LSCS for fetal indication. Blood sugar and serum electrolytes were within normal limits at the time of first convulsion. Injection oxytocin was started for augmentation of labor. Within half hour of this, she convulsed again. BP was 140/90 mm of Hg. The interval between the 1st and 2nd convulsion was 1 hour 30 minutes. Injection levetiracetam 1 gram loading dose was given intravenously and the convulsion episode subsided. Injection mannitol 100 ml intravenous 8 hourly was started and tablet labetalol 100 mg orally 8 hourly was continued. Injection magnesium sulphate by Zuspan regime was continued. Oxytocin augmentation was continued and patient delivered vaginally. Apgar score of the neonate was 6/10 at one minute and 9/10 at 5 minutes. Her 3rd and 4th stages of labor were unremarkable. Tablet amlodipine 5 mg was added as her BP recordings remained around 160/100 mm of Hg. She developed a 3rd episode of convulsion 10 hours after her delivery and injection phenytoin 600 mg intravenous bolus was given. CT scan of the brain showed hypodensity mostly involving the white matter of the left frontal lobe. A similar hypodense area involving the left temporal lobe and the left external capsule were also seen. (Figures 1 and 2). She was further managed in the medical intensive care unit (MICU). Within 1 hour of the 3rd convulsion she convulsed again for the 4th time. Her BP was 170/100 mm of Hg. Injection midazolam was given and convulsions subsided. Injection magnesium sulphate, levetiracetam and phenytoin were continued. Oral labetalol and amlodipine were continued and tablet hydralazine 25 mg was added in view of persistent blood pressure of 170/100 mm of Hg. Neurologist advised to continue the same treatment.  Blood pressure stabilized and normalized gradually over 3 weeks. She did not convulse again and she had no residual neurological deficits on discharge. She was discharged on all the three antihypertensive drugs and levetiracetam. 

Figure 1. CT image showing (A arrow) hypodensity involving left frontal lobe mostly involving the white matter and B arrow showing hypodense areas involving left temporal lobe and left external capsule.

Figure 2. CT image with arrow pointing towards temporal lobe hypodense areas.

Discussion

The definitive management of eclampsia is delivery. The drug of choice for control of seizures in eclampsia is magnesium sulphate. Compared to other anticonvulsant agents, magnesium sulphate also reduces the risk ratio of recurrence of seizures and improves maternal and fetal outcomes.[1]

After the initial administration of 4 grams of magnesium sulphate, if a repeat convulsion occurs, the first step is to administer an additional 2 grams of magnesium sulphate. However, this was not administered in our patient. Very few patients require such an additional dose. If convulsions occur despite this, additional anticonvulsants are indicated. In the original study conducted in Parkland hospital which studied the effectiveness of magnesium sulphate, only 5 patients out of 245 patients required additional anticonvulsants to control eclamptic seizures.[2]

Our patient required two additional anticonvulsants; levetiracetam and phenytoin sodium and a benzodizapine.  Benzodizapine midazolam a category ‘D’ drug, is not a usual drug used as an anticonvulsant in pregnancy. Levetiracetam is a newer broad spectrum antiepileptic agent which is being considered to be of good safety in pregnancy. The drug appears to be better than carbamazepine and valproate, with regards to long term neurological outcomes in the fetus also.[3, 4, 5] The drug has also been used safely in the Indian tertiary care setup for recurrent seizures with good outcomes.[6]

Our patient had a sudden rise in blood pressure from 150/90 to 170/110 mm of Hg. While the criterion of only an increase in blood pressure is no longer used in the definition of preeclampsia,[7] the concept of “delta hypertension” is still approved in modern obstetrics. This refers to a sudden increase in blood pressure in otherwise normotensive women during late pregnancy, labor and postpartum period.[8]  Such a delta hypertension has been proven to have higher incidence of readmission in postpartum period with severe preeclampsia/eclampsia.[9] In the study reported by Tank et al, eclamptic patients with high mean arterial pressure had been shown to have adverse neurological outcomes.[10]

Our patient had a neurological complication, namely “posterior reversible leukoencephalopathy syndrome”. A term coined by Hinchey et al, is described as the presence of neurological and radiological signs, with associated confusion, headaches, arousal problems, occasionally coma, visual disturbances and generalized seizures.[11]

In our patient, the highest blood pressure recording was of 170/110 mm of Hg. Though hypertensive emergencies and eclampsia are characteristic causes of PRES, it can occur in seemingly normotensive patients also. While PRES commonly occurs at term, extremely rare cases presenting during the first trimester as a complication of hydatidiform mole [12] and also in late postpartum period [13] have been described.  

Our patient who underwent CT brain, had the classical neuro-radiological findings of PRES that is reversible vasogenic subcortical edema without infarction. MRI brain is superior to  CT brain in identifying the typical abnormalities of PRES. Upon prompt control of the blood pressure or of the offending disease process, the condition appears to resolve and this can be identified by repeat neuroimaging . 

One theory that hypothesizes its pathogenesis states that when severe hypertension exceeds the physiological limits of auto regulation, breakthrough brain edema occurs, precipitating the condition. However, it was earlier believed that hypertension led to cerebral auto regulatory vasoconstriction and in turn led to ischemia, finally resulting in brain edema. The posterior circulation is preferentially affected.  Owing to this, visual disturbances may be a common phenomenon. However, our patient did not report any visual complaints. The radiological imaging in our patient showed more anterior involvement than posterior involvement, which is more classical of PRES due to malignant etiologies rather than obstetric etiology.[14, 15] There exists a beneficial effect of magnesium sulphate in PRES also. Naidu et al studied cerebral artery flow velocity waveforms using ultrasonography by trans-temporal approach. They determined that the wave forms suggested a significant reduction in cerebral vasospasm among those treated with magnesium sulphate than when compared to other anticonvulsant medications.[16]

Though this patient required multiple anticonvulsants, it constitutes a rare scenario. Blood pressure monitoring should continue in the postpartum state also. The above case fits into atypical eclampsia but typical PRES, a condition which is reversible, has classical clinico-radiological features and has good prognosis. 

References

1. Duley L, Henderson-Smart DJ, Chou D. Magnesium sulphate versus phenytoin for eclampsia.Cochrane Database Syst Rev. 2010 Oct 6;(10):CD000128.
2. Cunningham FG. Hypertenisve disorders. In Cunningham FG, Leveno KJ, Bloom SL, Spong CY, Dashe JS, Hoffman BS, editors. Williams’ Obstetrics. 24th ed. New York: Mc Graw Hill 2014; pp. 748
3. Iniesta I.Carbamazepine in pregnancy: Levetiracetam and lamotrigine are better options. BMJ. 2011 Jan 25;342:d279. 
4. Mawhinney E, Craig J, Morrow J, Russell A, Smithson WH, Parsons L, et al. Levetiracetam in pregnancy: Results from the UK and Ireland epilepsy and pregnancy registers. Neurology 2013; 80(4):400–5 
5. Shallcross R, Bromley RL, Cheyne CP, García-Fiñana M, Irwin B, Morrow J, et al. In utero exposure to levetiracetam vs valproate: development and language at 3 years of age. Neurology. 2014;82(3):213–21
6. Satia MN, Shilotri MP. A study of the obstetric and perinatal outcomes of eclampsia and the use of levetiracetam in its management. Int J Reprod Contracept Obstet Gynecol. 2016; 5(12): 4266-4270.
7. Dekker G. Hypertension. In James D, Steer PJ, Weiner CP, Gonik B, Crowther CA, Robson SC editors. High risk pregnancy. Management options. 4th ed. Missouri: Elsevier Saunders 2011: pg 800.
8. Cunningham FG. Obstetrical hemorrhage. In Cunningham FG, Leveno KJ, Bloom SL, Spong CY, Dashe JS, Hoffman BS, editors. Williams’ Obstetrics. 24th ed. New York:   Mc Graw Hill 2014: pg 729.  
9. Atterbury JL, Groome LJ, Hoff C. Blood pressure changes in normotensive women readmitted in the postpartum period with severe preeclampsia/eclampsia. J Matern Fetal Med. 1996; 5(4):201–5. 
10. Tank PD, Chauhan AR, Bhattacharya MS, Warke HS, Raut VS. Neurological complications in eclampsia: a case series. Int J Fertil Womens Med. 2004; 49(2):61–9.
11. Ait S, Gilbert T, Cotton F, Bonnefoy M. Cortical blindness and posterior reversible encephalopathy syndrome in an older patient. BMJ Case Rep. 2012 May 26;2012. Available from: http://www.ncbi.nlm.nih.gov/pubmed/22669215
12. Ottanelli S, Simeone S, Serena C, Rambaldi MP, Villanucci A, Tavella K, et al. PP120. Hydatidiform mole as a cause of eclampsia in the first trimester: A case report. Pregnancy Hypertenion An Int J Women’s Cardiovasc Heal. 2012; 2(3):304. 
13. Rijal JP, Giri S, Dawadi S, Dahal K V. Posterior reversible encephalopathy syndrome (PRES) in a patient with late postpartum eclampsia. BMJ Case Rep [Internet]. 2014; 2014. Available from: http://www.ncbi.nlm.nih.gov/pubmed/24577180 
14. Lamy C, Oppenheim C, Mas JL. Posterior reversible encephalopathy syndrome. Handb Clin Neurol. 2014;121:1687-701.
15. Cozzolino M, Bianchi C, Mariani G, Marchi L, Fambrini M, Mecacci F. Therapy and differential diagnosis of posterior reversible encephalopathy syndrome (PRES) during pregnancy and postpartum. Arch Gynecol Obstet . 2015; 292(6):1217–23.
16. Naidu S, Payne AJ, Moodley J, Hoffmann M, Gouws E. Randomised study assessing the effect of phenytoin and magnesium sulphate on maternal cerebral circulation in eclampsia using transcranial Doppler ultrasound. Br J Obstet Gynaecol. 1996; 103(2):111–6.

Citation

Nasare P, Madhva Prasad S, Gupta AS. Atypical Eclampsia With PRES Syndrome. JPGO 2017. Volume 4 No.2. Available from: http://www.jpgo.org/2017/02/atypical-eclampsia-with-pres-syndrome.html

Vulval Abscess In A Pregnant Woman

Author Information

Thakare R*, Thosar MA**, Gupta AS***

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

Abstract

A pregnant woman at 34 weeks of pregnancy was referred to our tertiary care hospital with extensive abscess of the vulva extending to the groin and anterior abdominal wall for management. The abscess was incised and drained under antibiotic cover. Daily dressing with collagen granules and silver colloid gel was done. She had premature rupture of membranes on second day of procedure and delivery was conducted by lower segment cesarean section (LSCS) for fetal distress. The wound healed over a period. The fetal outcome was uneventful.

Introduction

Simple infections of the vulvar skin and subcutaneous tissue can result in the formation of a vulvar abscess which is a common problem seen in gynecological practice. The loose areolar tissue present in the subcutaneous layers of the vulvar area along with the contiguity of  vulvar fascial planes with the groin and anterior abdominal wall facilitates the spread of infection and abscess formation.

Case Report

A 22 year old primigravida married since one year, with 34 weeks of gestation was referred from a peripheral hospital to our tertiary care hospital in view of large left sided vulvar abscess extending to the anterior abdominal wall and inguinal region for incision and drainage. She had a swelling in the vulvar area since seven days. Initially swelling was about 1x1cm and had increased gradually. She also complained of pus discharge from the lesion and fever since one day. She had difficulty in walking and had a wide based gait. She was admitted in a private hospital, where she received parenteral antibiotics for four days. She was referred to a higher center due to non-resolution of the abscess. She had no bowel or bladder complaints. She had no history of tuberculosis, hypertension, diabetes mellitus, asthma, thyroid disorder or epilepsy. There was also no history of any surgical illness, blood transfusion or drug allergies. Ultrasound (USG) was done of the local perineal region. It showed a heterogeneous collection of 3.4×1.8 cm in the left vulvar region extending to the mid-inguinal region as well as to the left medial and anterior aspect of the thigh. Right inguinal region showed a single enlarged lymph node measuring 1.8 cm. On admission her general condition was fair and she was afebrile. Pulse was regular, good volume, she had no tachycardia. Blood pressure was in the normal range. No cardiovascular or respiratory abnormality was detected. Bilateral pedal edema was present up to the knees. On abdominal examination uterus was 34 weeks in size, relaxed, fetus was in vertex presentation, head was 4/5th palpable and fetal heart sounds were present, regular in rhythm with a rate of 140 beats per minute. On local examination left sided lower abdominal wall erythema and induration with warmth on touch, extending to the left labia majora was seen and felt. Excoriation was present over the left labia majora. A spontaneous sealed site of pus drainage was seen over the left labia majora. Swelling and induration was also extending to the medial side of the groin and thigh. No midline extension of the swelling was noted. Right sided labia majora and minora were normal. No enlarged lymph nodes were palpable on either side. (Figure 1)

Figure 1. Abscess of the left labia majora extending to the medial side of the groin and thigh.

The swelling was extremely tender, and she was not co-operative. She could not flex her left leg as the limb muscles were in spasm. Her hematological, serological and biochemical parameters were normal. Her HIV status was also seronegative. General surgeons were consulted and a joint decision to drain the abscess was taken. After preoperative workup and consent incision and drainage of the vulvar abscess was done under spinal anesthesia. Three incisions were taken (Figure 2). The 1st incision was over the left labia majora on the most prominent fluctuant and dependant site. Second and 3rd incisions were taken over the left inguinal region and all pus was drained out. Blunt dissection was extended to the medial side of the thigh. All the pus was drained out; all loculi were broken by blunt dissection. Thick, yellow and foul smelling pus was collected and sent for aerobic, anaerobic and AFB cultures (Figure 3). Broad spectrum parenteral antibiotics were continued. No biopsy was taken as there was no clinical suspicion of malignancy.

Figure 2. Surgical incisions.

Figure 3. Pus drainage.

The abscess cavity was irrigated with hydrogen peroxide, povidone iodine and warm saline. The abscess cavity was packed with povidone iodine soaked gauze pieces and covered with sterile dressing. Both the preliminary and final reports of pus culture were suggestive of no growth. Next day she had premature rupture of membranes. Preinduction cervical ripening was done with Dinoprostone gel. The Bishop’s score failed to improve. Fetal distress was noticed on intrapartum monitoring. A decision of emergency lower segment cesarean section was taken after explaining risk of peritonitis to her and her relatives. The cesarean section was done by vertical midline incision over the abdominal wall after isolating the abscess area to prevent the spread of infection by contiguity. Maternal and fetal outcome were good. Baby’s birth weight was 2.269 kg with APGAR score of 9/10. In the neonatal period, the baby had no signs and symptoms of sepsis.

Post operatively daily dressing of vulvar wound was continued. The vulvar wound healed but the communicating wound at the groin had a wide defect as the she was ambulatory Plastic surgeons reviewed the gaping wound in the left inguinal region. A 3x3 cm fistulous tract was present with signs of inflammation. Daily sitz bath and irrigation of the fistulous tract with vinegar from the upper end to lower end and regular cleaning and dressing with silver colloid gel and collagen granules was advised. Plan of primary closure with debridement followed by split thickness skin grafting was made. However the wound continued to heal. The plastic surgeons reviewed the wound and the decision of grafting was canceled. Daily cleaning and dressing of the wound was continued. The abdominal incision of the cesarean section healed well. On discharge, the vulvar wound was healed, but the wound at the left inguinal region was present for which daily dressing was advised with collagen granules. On follow up with the plastic surgeons, the wound was better and was healing.

Discussion

Hair follicles, sweat and sebaceous glands of the vulvar skin are common sites of infection and abscess formation. The contiguity of vulvar fascial spaces with other anatomic compartments permits spread of infection from the vulva to the inner thigh, abdominal wall, or ischiorectal fossa.[1] The differential diagnosis of a vulvar abscess includes infectious and non-infectious vulvar lesions. Among the infectious causes, vulvovaginitis presents mostly as erythema and less likely as a suppurative mass. Necrotizing fasciitis though a rare condition may present as an abscess and should be suspected when the lesion is extensive with persistent pain, erythema, and induration.[2] Malignant lesions of the vulva may present as a firm lesion with surrounding erythema. A biopsy should be taken whenever there is a suspicion of malignancy.

In our case the abscess had spread over a wide area. The only history which was elicited was of itching and scratching over the left side of vulva few days prior to onset of symptoms. A close watch was kept for further extension of the abscess and development of necrotising fasciitis. But fortunately even though a large area was involved, necrosis and gangrene did not occur. Probably the parenteral antibiotics received in the private hospital prevented the causative organisms from growing and multiplying (as seen by negative pus culture report). The decision of conventional incision and open drainage by the surgeons was made in view of the large extension of the abscess. In some cases closure by primary suturing may be done under antibiotic cover.[3] She had preterm premature rupture of membranes probably due to some ascending infection that was not detected on cervical swab due to antibiotic use. Fortunately her abdominal wound healed well and she had no spread of sepsis as special care was taken to isolate her abscess site during the cesarean section. A midline vertical incision for cesarean section that was taken avoided the inevitable lateral extension into the infected left lower abdominal tissue planes.

This interesting case highlights the need of a multidisciplinary approach involving the general surgeons and reconstructive plastic surgeons. Timely drainage and breaking of all loculi and regular dressing can bring out a favorable outcome even in a large vulvar abscess.

References

1. Lazenby GB, Thurman AR, Soper DE. Vulvar abscess. UpToDate. 2016;1-20. Available from:http://www.uptodate.com/contents/vulvar-abscess
2. Kdous M, Hachicha R, Iraqui Y, Jacob D, Piquet PM, Truc JB. Necrotizing fasciitis of the perineum secondary to a surgical treatment of Bartholin's gland abscess. Gynecol Obstet Fertil 2005; 33(11):887-90.
3. Larsen T, Larsen PN, Christophersen S, Moesgaard F, Nielsen ML. Treatment of abscesses in the vulva. Conventional open treatment versus primary suture under antibiotic cover. Acta Obstet Gynecol Scand. 1986;65(5):459-61.

Citation

Thakare R, Thosar MA, Gupta AS. Vulvar Abscess In A Pregnant Woman. JPGO 2017. Volume 4 No.2. Available from: http://www.jpgo.org/2017/02/vulval-abscess-in-pregnant-woman.html

Lost and found - Lippes Loop

Author Information

Shah NH*, Doshi D**, Kumari K**, Paranjpe SH***

(* Consulting Gynecologist/Obstetrician, ** Gynecologist Consultant, *** Director Gynecologist Consultant, Dr.Paranjpe Maternity Home, Chembur, Mumbai, India. )

Abstract

Intra uterine contraceptive devices (IUD’s) like Lippes loop were intended for long term use, and are retained for years inside the uterine cavity. Post menopausal women have often presented with Lippes loop in situ but having completely forgotten about its insertion. We report a case of Lippes loop removal in a 70 year old, postmenopausal, woman who was unaware of its insertion. She was complaining of pain in abdomen and vaginal discharge. Considering the complication of long term use of this device, it was removed hysteroscopically.

Introduction

Intrauterine contraceptive devices have been used from the 19th century.[1] In 1960, Dr. Jack Lippes made his first model of the ‘Double-S’ intrauterine contraceptive device named as “Lippes Loop” (LL). It was a flexible polyethylene plastic loop of appropriate size for the uterine cavity which subsequently went on to become the gold standard for other IUD’s to be compared with.[2] These early IUD’s like Lippes loop were intended for long term use until even after menopause.[2] Literature has presented a large and varying number of side effects & complications after long term use of any IUD. Here, we report a case of diagnosis and hysteroscopic removal of a Lippes loop in a 70 year old woman who had no idea about its insertion and presence.

Case Report

A 70 year old menopausal, parous woman, came to our hospital out patient department with the chief complaints of pain in abdomen and a lot of vaginal white discharge since last 5 months. She did not give any history of use of any kind of contraceptive. On per speculum examination cervix was atrophic and a lot of curdy white discharge was present. No thread was seen during the speculum examination. A PAP smear was taken and an ultrasonography (USG) scan of the pelvis was advised. On USG, an echogenic substance was identified suggestive of Lippes loop. A radiograph of the pelvis was obtained to confirm the diagnosis. It showed the loop clearly. As the thread was not visible outside the cervix, a decision for removal under anesthesia was taken.

Figure 1. Radiograph of pelvis, showing the Lippes loop.

Figure 2. Hysteroscopic view of the Lippes loop.

Retrieval was not achieved even after trying removal with a simple artery forceps. After failure to remove with simple curettage also, hysteroscopy was done, wherein the loop was clearly identified and removed with a hysteroscopic grasper.

Figure 3. Removing the loop with a hysteroscopic grasper.

Figure 4. Lippes loop after removal.

Endometrial curettage was also sent for histopathological examination and both endometrium and PAP smear turned out to be negative for malignancy.

Discussion

Lippes loop was an inert IUD intended for long term use till menopause, but today, it is no longer in use having been replaced by the now popular copper bearing devices introduced in the 1970’s. Pregnancy rates decreased with increase in length of use. The probability of ectopic pregnancy was observed to be constant and it had a 1 in 10 chance, if the device remained in situ for more than three years.[3] Though infections were common within the first year of inserting the device, they could occur at anytime.[4] Actinomycosis, as such is a rare cause of pelvic inflammatory disease (PID). However, it is associated to IUD insertion as few cases of PID due to Actinomycosis have been reported in IUD users in literature.[5] It has been documented that removal of a long standing IUD becomes very difficult because of accumulation of small calcium deposits thus causing corrosion in the plastic of the IUD. This in turn weakens the strength of the device and its tail making it friable to break. The longer the loop is kept inside, it has a tendency to bury itself in the endometrium thus causing difficulty in its removal. Removal in such circumstances is associated with pain and bleeding. Removal can become difficult after menopause as there is atrophy of the uterus and the cervical canal.[6]  Pukale et al  reported a similar case who underwent hysterectomy for PID ( pain in abdomen, fever with chills, foul smelling vaginal discharge, and feeling of right sided abdopelvic mass ) due to a forgotten Lippes loop. Cut section of the uterus following hysterectomy showed a deeply embedded loop.[7] A forgotten IUD can lead to various problems including infertility, postmenopausal bleeding,  abdominal pain, pelvic inflammatory disease, and lastly fever which may be due to actinomycosis. Since long, literature has mentioned a lot of side effects and various complications of using various IUD’s. These complications consists of ectopic pregnancy, bowel obstruction, uterine perforation, infections and also death. WHO recommends removal of a misplaced IUD as soon as a diagnosis has been made.[8]

Conclusion 

Case reports are the only helpful source of documentation when there is lack of  sufficient literature and evidence. Reporting of rare cases, especially of a rare complication, or events associated with prolonged IUD use should be promptly reported and also widely encouraged.

After reviewing the literature and various case reports, our opinion is to remove the IUD even if it is inert and not causing any symptoms as complications cannot be predicted. The complications occurring due to this, although rare, are very severe and unusual causing a lot of morbidity to the patient.

References

1. Harrison RF. Adenocarcinoma of the uterine body following use of intrauterine contraceptive device. Ir J Med Sci. 1971; 140(9):407-9.
2. Thomsen RJ, Rayl DL. Dr. Lippes and his loop. Four decades in perspective. J Reprod Med. 1999; 44(10):833-6.
3. Vessey MP, Yeates D, Flavel R. Risk of ectopic pregnancy and duration of use of an intrauterine device. Lancet. 1979; 2(8141):501-2.
4. Vessey MP, Yeates D, Flavel R, McPherson K. Pelvic inflammatory disease and the intrauterine device: findings in a large cohort study. Br Med J (Clin Res Ed). 1981; 282(6267):855-7.
5. Purdie DW, Carty MJ, McLeod TI. Tubo-ovarian actinomycosis and the IUCD. Br Med J. 1977; 2(6099):1392.
6. Bromham DR. IUD removal problems: a study of associated factors. British Journal of Family Planning. 1982;8(2):43–50.
7. Ravindra P, Okram S, Vijayalakshmi S. Forgotten Lippes loop.Jr of Dental and Medical Sciences. 2013;8(5):19–20
8. Ozgun MT, Batukan C, Serin IS, Ozcelik B, Basbug M, Dolanbay M. Surgical management of intra-abdominal mislocated intrauterine devices. Contraception. 2007;75(2):96-100.

Citation

Shah NH, Doshi D, Kumari K, Paranjpe SH. Lost and found - Lippes Loop. JPGO 2017. Volume 4 No.2. Available from: http://www.jpgo.org/2017/02/lost-and-found-lippes-loop.html

Botryomycosis of Anterior Abdominal Wall following Cesarean Section

Author Information

Chhonkar A*, Nayak CS**, Tambe S***

(*Third Year Resident, **Professor and Head of Department, *** Assistant Professor, Department of Skin and V.D., T.N.M.C. & B.Y.L. Ch. Nair hospital, Dr. A.L.Nair Road, Mumbai central, Mumbai, Maharashtra-400008) 

Abstract 

Botryomycosis is a chronic granulomatous inflammatory reaction to bacterial antigens. It may present with cutaneous or, less commonly, visceral involvement.  It is a relatively rare infection that is more prevalent among immunocompromised patients. In cutaneous botryomycosis, the lesions may be pleomorphic including cysts, abscesses, fistulas, nodules, plaques or ulcers. Here we report an immunocompetent patient who developed botryomycosis after lower segment cesarean section. High frequency ultrasound imaging is very helpful in evaluating completion of healing.

Introduction

Botryomycosis is a chronic granulomatous inflammatory reaction to bacterial antigens.[1] It was initially mistaken for a fungal infection, thus the term botryomycosis (from Greek word botrys which means a bunch of grapes and mycosis which means of fungal origin) was used. Other less commonly used terms are bacterial pseudomycosis, staphylococcal actinophytosis and granular bacteriosis.[2,3] In cutaneous botryomycosis, the lesions may be pleomorphic including cysts, abscesses, fistulas, nodules, plaques or ulcers.The general swollen and suppurative aspect of the lesion suggests local inoculation of a foreign body as the initial cause of infection and perpetuation of the disease.[4] It is more prevalent among immunocompromised patients.[5-7] However, it has also been reported in immunocompetent individuals as seen in our patient. 

Case Report

A 24 year old housewife presented with painful reddish swelling in groin of seven months duration with pus discharge following a lower segment cesarean section nine months earlier. A week after the surgery, she developed wound site infection which spread despite taking oral antibiotics and the wound site was excised and re-sutured. One week later, a tender boggy swelling developed over the excision site with multiple discharging sinuses. On examination, two oval, tender sinuses of 0.5x1cm with hyperpigmentation of the surrounding skin in the right inguinal region and a solitary, oval, tender, boggy swelling, with few pustules over it in the left inguinal region were seen and felt. (Figure 1). Routine hemogram, liver and renal function tests and other biochemical tests were normal. A differential diagnosis of actinomycosis, deep mycotic infection, botryomycosis and lupus vulgaris were considered. On investigation erythrocyte sedimentation rate (ESR) was 80mm/ hour while chest x-ray and mantoux test were negative. Cultures for AFB, fungal and bacterial infections were negative. Smear from discharge showed gram positive cocci. Histopathology revealed chronic granulomatous inflammation. She had received oral cefixime and amoxicillin-clavulinic acid combination in the past which led to partial resolution of the lesions but the lesions recurred on stopping the treatment. Ultrasonography (USG) of the local part revealed hypoechoic mass lesions involving the skin and subcutaneous tissue in both the inguinal regions suggestive of abscesses with bilateral lymphadenopathy. (Figure-2,3). She was given tablet cotrimoxazole (trimethoprin 160 mg + sulphamethoxazole 800 mg) twice a day for four months with complete resolution of the lesions (Figure-4) which was confirmed on USG before discontinuing the treatment.(Figure-5).

Figure 1. Two oval, tender sinuses with hyperpigmentation of surrounding skin in the right inguinal region and a solitary, oval, tender, boggy swelling, with few pustules over it in the left inguinal region. 

Figure 2. Hypoechoic masses in skin and subcutaneous tissue in left inguinal region suggestive of abscesses with lymphadenopathy.

Figure 3. Hypoechoic masses in skin and subcutaneous tissue in right inguinal region suggestive of abscesses with lymphadenopathy.

Figure 4. Post treatment photograph showing resolution of lesion with scarring.

Figure 5. Post treatment USG showing decrease in size of left inguinal lymph node and resolution of abscess. Left image is pre treatment and right image is post treatment.

Discussion

Botryomycosis was first described in 1870 by Bollinger, by observing granulomatous lesions as a complication after horse castration. In 1884, Rivolta coined the name Botryomycosis.[1] The pathogenesis of the disease is not clear but it may be related to low virulence of infectious agents, large bacterial inoculum or change in specific cellular immunity or in humoral immune response.[2]

Associated risk factors include alcoholism, diabetes mellitus, trauma and surgery, HIV infection, cystic fibrosis, chronic granulomatous disease.[3-5]  A history of injury is common in cutaneous form of botryomycosis, which shows the role of foreign body as well as infection in the causation of the disease.[6]

Cellular response in botryomycosis is similar to that seen in actinomycetoma and eumycetoma. The grain consisting of bacterial colonies is surrounded by acute suppurative response. The inflammatory infiltrate consists of numerous neutrophils surrounding the grain, lymphocytes, histiocytes, eosinophils, plasma cells, few foreign body giant cells and fibroblasts surrounding the central suppuration. Surrounding this infiltrate is the fibrosis and granulation response, with new vessel formation.[7]

The most common causative agent is Staphylococcus aureus (40%), followed by Pseudomonas species (20%).[2] Other microorganisms reported are Escherichia coli, Proteus vulgaris, Klebsiella, Neisseria, Streptococcus, Staphylococcus epidermidis, N Bacillus species and Actinobacillus lignieresii.[1,2]

There are two forms of botryomycosis- cutaneous and visceral. In cutaneous form, hands, pinna, feet and head are commonly affected. Skin folds in overweight individuals are also vulnerable areas.[7] The lesions may be pleomorphic including cysts, abscesses, fistulas, nodules, plaques or ulcers. In visceral involvement, pulmonary botryomycosis is the most common entity.[7,8,9] There have also been reports of intraoral granulomatous pyogenic botryomycosis.[10]

Diagnosis is confirmed on histopathological examination which reveals ‘Splendore Hoeppli’ phenomenon; grains of bacteria surrounded by eosinophilic material and inflammatory cells consisting of epitheloid cells, histiocytes, eosinophils and giant cells.[11] Since multiple agents may be responsible in the formation of granules, cultures are essential to pin-point the causative organism.[1] Ultrasonography with frequency higher than 7 MHz allows the visualization of superficial as well as deeper structures. Frequencies higher than 15 MHz are increasingly used in dermatology as they allow differentiation of skin layers.[12] High frequency ultrasound along with color Doppler helps in the measurement of skin thickness as well as in investigation of tumors and inflammatory diseases and also helps in the evaluation of effectiveness of proposed treatments. [13]

Treatment requires antibiotic therapy and, in most cases surgical debridement. The selection of antibiotics should be in keeping with the results of bacterial cultures.[14]

This case highlights the importance of prolonged anti-bacterial therapy, preferably based on antibiotic sensitivity reports, till complete clinical cure. This case also highlights the efficacy of age old but less used anti-bacterials like co-trimoxazole in some cases. Ultrasonography of skin helped us to locate the pus pockets and the repeat scan helped us confirm that all pus collections had cleared before we decided to stop the treatment. Therefore, USG of skin is useful in confirming complete resolution of inflammatory lesions.

References

1. Mehregan DA, Su WP, Anhalt JP. Cutaneous botryomycosis. J Am Acad Dermatol. 1991; 24(3):393-6       
2. Bonifaz A, Carrasco E. Botryomycosis. Int J Dermatol. 1996;35(6):381-8       
3. Brunken RC, Lichon-Chao N, van der Broek H. Immunologic abnormalities in botryomycosis. A case report with review of the literature. J Am Acad Dermatol 1983; 9(3): 428- 34.
4. Patterson JW, Kitces EN, Neafie RC. Cutaneous botryomycosis in a patient with acquired immunodeficiency syndrome. J Am Acad Dermatol 1987;16 (1 Pt 2):238- 42.
5. Olmstead PM, Finn M. Botryomycosis in pierced ears. Arch Dermatol 1982;118(11): 925- 7.
6. Hay RJ and Adriaans BM.  Bacterial Infections. In Burns T, Breathnach S, Cox N, Griffiths C, editors.  Rook's Textbook of Dermatology, 8th ed. Oxford, UK: Wiley-Blackwell, 2010;1-82.
7. Rippon JW. Medical mycology: the pathogenic fungi and the pathogenic actinomycetes. 3rd ed. U.S.A: W.B. Saunders Company; 1988. p. 116.
8. Waisman M. Staphylococcus actinophytosis (Botryomycosis):Granular bacteriosis of skin. Arch Dermatol 1962;86:525-9.
9. Bishop GF, Greer KE, Horwitz DA. Pseudomonas botryomycosis. Arch Dermatol 1976;112:1568-70.
10. Mackinnon JE, Conti-Diaz IA. Experimental botryomycosis produced by Pseudomonas aeruginosa. J Med Microbiol 1963;3:369-73.
11. Mechow N, Göppner D, Franke I, Kolesnik M, Bonnekoh B, Gollnick HPM, et al. Cutaneous botryomycosis diagnosed long after an arm injury. Journal of the American Academy of Dermatology.2014;71(4):e155-6.
12. Wortsman X. Common Applications of Dermatologic Sonography. J Ultrasound Med. 2012;31:97–111.
13. Lucas VS, Burk RS, Creehan S, Grap MJ. Utility of high-frequency ultrasound: moving beyond the surface to detect changes in skin integrity. Plast Surg Nurs. 2014;34(1):34–8.
14. Neafie RC, Marty AM: Unusual infections in humans. Clin Microbiol Rev. 1993; 6(1): 34–56.

Citation

Chhonkar A, Nayak CS, Tambe S. Botryomycosis of Anterior Abdominal Wall following Cesarean Section. JPGO 2017. Volume 4 No.2. Available from: http://www.jpgo.org/2017/02/botryomycosis-of-anterior-abdominal.html

Inherited Thrombophilia In Pregnancy: Management And Outcome

Author Information

Kalappa SB*, Shende D**, Chauhan AR***.

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

Abstract

Thrombophilias refer to inherited or acquired deficiencies of one or more inhibitory proteins of the coagulation cascade which can lead to hypercoagulabilty and recurrent venous thromboembolism. Normal pregnancy itself increases the thrombogenic potential of inherited disorders as it is associated with increased stasis and alterations in the levels of various coagulation factors that promote thrombosis. These combined thrombophilias complicating pregnancy frequently pose a challenge to obstetricians. Here we present a case of inherited antithrombin III and protein S deficiency in a pregnant patient, associated with various maternal and fetal complications, managed successfully through a multi-disciplinary approach.

Introduction

Antithrombin III is an important inhibitor of thrombin in clot formation. Protein S is a natural circulating anticoagulant, which is activated by protein C and inactivates factors Va and VIIIa of the coagulation cascade. The prevalence of antithrombin III deficiency is estimated to be 1 in 2000 to 5000 individuals and the prevalence of protein S deficiency is 2 per 1000.[1] More than 100 autosomal dominant mutations can lead to the deficiency of either of these factors. Inherited thrombophilias have been linked with various pregnancy complications such as recurrent pregnancy loss, preeclampsia, placental abruption and fetal growth restriction.[2]

Case Report

A 30 year old gravida 4, para 3, with no living issue, 26 weeks of gestation, a known case of antithrombin III and protein S deficiency, was referred from hematologist in view of bad obstetric history and recently diagnosed preeclampsia for obstetric management. Her first pregnancy had resulted in an intrauterine fetal death at seven months of gestation with abruptio placentae. This was followed by a second intrauterine fetal death at 6 months of gestation, with intrauterine growth restriction and oligohydramnios. Her third pregnancy resulted in a term still birth with abruptio placentae. Inherited thrombophilia was diagnosed four years ago during the course of her evaluation for bad obstetric history; she was not on any medication for the same. 

She was admitted in the antenatal ward. On examination, her general condition was fair, her pulse was 86 beats /minute, and blood pressure was in the range of 150/ 90 to 160/ 100 mm Hg. No abnormalities were detected in the respiratory and cardiovascular systems. Grade 3 pitting edema was present in both lower limbs. Abdominal wall edema and facial puffiness was also present. On per abdominal examination, uterus size corresponded to 26 weeks of gestation and fetal heart sounds were regular. On per vaginal examination the cervical os was closed and uneffaced. 

She was thoroughly investigated: her routine biochemical, hematological and serological investigations including hemoglobin, platelet count, lipid profile and serum electrolytes were normal. Prothrombin time was 10.3 (control 9.9), international normalized ratio (INR) was 1.04 and partial thromboplastin time was 33.1 (control 28.0); all in the normal range. Her serum TSH had previously not been done and was elevated. It was 6.27 µIU/ ml. Alkaline phosphatase was also raised to 255.30 IU/L (normal range 15 - 112 IU/L) while rest of the liver function tests were normal. Renal function tests were deranged with serum uric acid 7.5 mg % (normal range 2.4 – 4.9 mg %); blood urea nitrogen 20 mg % (normal range 3 – 13 mg %) and serum creatinine was 1.4 mg % (normal range 0.4 – 0.8 mg %). Her 24 hours urine protein was also high at 2860 mg/ day (normal range 47 – 186 mg/ day). Renal ultrasound was normal. Her urine culture showed insignificant bacteruria. 

Anti nuclear antibody, double stranded DNA antibody, anti cardiolipin antibody and anti phospholipid antibody tests were negative. Lupus anticoagulant was absent. Her antithrombin III levels were 10 % (normal range 70 – 130 %), protein S was 94 % (normal range 70- 140 %) and protein S activity was 4 % (normal range 65 – 140 %). 

She was started on low molecular weight heparin (enoxaparin) 0.6 ml subcutaneously twice a day along with tablet aspirin 150 mg daily when the pregnancy was diagnosed. Preeclampsia was detected at 26 weeks of gestation and she was started on antihypertensives; tablet labetalol 200 mg twice daily, tablet alpha methyldopa 500 mg four times a day and capsule nifedipine 10 mg four times a day were added to control the progressively increasing blood pressure. Routine examination of the fundus revealed no evidence of papilledema. Hypothyroidism detected during routine evaluation was treated with tablet thyroxine 25 µg daily. 2D echocardiography was done as patient complained of breathlessness; it revealed moderate mitral regurgitation secondary to minor chordal rupture. Hence, patient was started on tablet digoxin 0.25 mg daily and tablet furesemide 40 mg twice daily as per cardiologist’s advice.

Hematology, nephrology, cardiology and ophthalmology consultants were involved periodically for comprehensive management of all pregnancy related complications. Fetal well being was monitored by bi-weekly non stress test and weekly obstetric Doppler studies. Two doses of injection betamethasone 12 mg were given intramuscularly 24 hours apart at 28 weeks of gestation. Obstetric Doppler revealed severe utero-placental and feto-placental insufficiency which progressed to absent diastolic flow in the umbilical artery followed by reversal of flow in the umbilical artery at 29 weeks of gestation. In consultation with neonatologist, decision to terminate the pregnancy was taken and emergency lower segment cesarean section was done. Outcome was a female baby with birth weight of 720 grams and an Apgar score of 4/10 and 9/10 at 1 and 5 minutes respectively. Baby was intubated and shifted to the neonatal intensive care unit where it was intensively managed for three months and discharged. The patient was monitored in the intensive care unit in the immediate post operative period. Injection low molecular weight heparin was continued for the entire puerperal period. All other medications were tapered or changed to meet post partum requirements and patient was discharged on day 10 post operatively.

Discussion

Numerous pregnancy related complications have been linked with various thrombophilias, both acquired and inherited. Pregnancy associated changes in various coagulation factors that increase the risk of thrombogenic events are: a) increased resistance to activated protein C from the second trimester b) decreased protein S activity c) increase in fibrinogen and factors II, VII, VIII, X  d) levels and activity of some fibrinolytic inhibitors like thrombin activatable fibrinolysis inhibitor, plasminogen activator inhibitor 1 and plasminogen activator inhibitor 2 increase. In particular, antithrombin III deficiency has been associated with increased risk of early pregnancy loss, preeclampsia and placental abruption, while protein S deficiency is more likely to cause late pregnancy loss.[2]

Pathological changes in the placental vasculature have been suspected to be one of the main causes of pregnancy related complications in patients with thrombophilias.[3] These changes are characterized by superficial invasion of cytotrophoblast in the myometrial spiral arteries, acute atherosis and thrombi in the spiral arteries and the inter-villous space. The thrombotic nature of these lesions and the increased thrombotic risk associated with thrombophilias suggests a direct cause-and-effect relationship between inherited thrombophilias and the above mentioned severe obstetric complications.

A recently conducted meta-analysis has suggested that severe preeclampsia is associated with thrombophilias, mainly Factor V Leiden mutation, deficiencies of antithrombin III, protein C and protein S and hyperhomocysteinemia.[3]

There is circumstantial eveidence that treatment with low molecular weight heparin may improve the pregnancy outcome in patients with inherited thrombophilias.[4] But, the use of anticoagulant therapy in pregnancy is challenging in view of the potential fetal and maternal complications. In asymptomatic women who have never experienced an episode of venous thromboembolism, the recommendation is either clinical surveillance or prophylactic therapy during the third trimester of pregnancy and for two to six weeks postpartum. Either unfractionated heparin 5000 IU twice daily or a low molecular weight heparin 40 mg once daily is used. In women with antithrombin III deficiency, this therapy is probably indicated throughout pregnancy in view of the substantially greater risk associated with the condition.[5]

Conclusion

There is insufficient evidence to draw any conclusions regarding the strength of association between inherited thrombophilia and adverse pregnancy outcomes. The guidelines for the prophylaxis and treatment of these conditions with anticoagulants are also in need of large scale randomized controlled trials to confirm the advantage and risk reduction for pregnancy complications. The best strategy at present is to conduct individual based risk assessment and provide treatment with a multi-disciplinary approach for successful pregnancy outcomes.

References

1. ACOG Practice Bulletin No 138: Inherited thrombophilias in pregnancy. Obstet Gynecol. 2013; 122(3): 706-17.
2. Simcox LE, Ormesher L, Tower C, Greer IA. Thrombophilia and Pregnancy Complications. International Journal of Molecular Sciences. 2015; 16(12): 28418-28.      
3. Kupferminc MJ. Thrombophilia and pregnancy. Reproductive Biology and Endocrinology. 2003; 1:111.
4. Skeith L, Carrier M, Kaaja R, Martinelli I, Petroff D, Schleubner E, et al. A meta-analysis of low-molecular-weight heparin to prevent pregnancy loss in women with inherited   thrombophilia. Blood. 2016; 127(13): 1650-55.
5. Bates SM, Greer IA, Middeldorp S, Veenstra DL, Prabulos AM, Vandvik PO. VTE, thrombophilia, antithrombotic therapy, and pregnancy: Antithrombotic Therapy and Prevention of Thrombosis, 9th ed: American College of Chest Physicians Evidence-Based Clinical Practice Guidelines. Chest. 2012;141(2 Suppl), e691S–e736S.

Citation

Kalappa SB, Shende D, Chauhan AR. Inherited Thrombophilia In Pregnancy: Management And Outcome. JPGO 2017. Volume 4 No. 2. Available from: http://www.jpgo.org/2017/02/inherited-thrombophilia-in-pregnancy.html

Exaggerated Placental Tissue Site Reaction: A Diagnostic Dilemma

Author Information

Ostwal P*, Shende D**, Dhokia T ***, Chauhan AR****.

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

Abstract

Exaggerated placental site (EPS) reaction is a non neoplastic lesion characterized by invasion of extravillous intermediate trophoblast into the myometrium and walls of blood vessels of the normal placental bed. It is very commonly seen following spontaneous or elective abortions and may also present as an etiology of 3rd stage post partum hemorrhage. We present a case of exaggerated placental tissue site reaction that caused a diagnostic dilemma as it simulated uterine sarcoma.

Introduction

Exaggerated placental tissue site reaction (EPS) is an exuberant pathologic process in which intermediate trophoblasts infiltrate the underlying endometrium and myometrium at the normal placental implantation site. These lesions may occur following normal pregnancy, ectopic pregnancy specifically scar and cervical ectopic pregnancies, elective or spontaneous abortion, and molar pregnancy.[1,2] They are seen as a sequelae in 1.6% of first trimester abortions. These lesions are neither inflammatory, nor are they limited to the endometrial zone. Therefore, a different class has been created for such lesions by WHO by the name “Exaggerated Placental Tissue Site Reaction”.[1] EPS is a difficult and confusing differential diagnosis for clinicians, and extensive literature search reveals that it has not been studied in much detail.[3] We discuss a case of EPS which had a confusing presentation.

Case Report

A 45 yr old woman, married since 25 years, para 4, with 4 previous normal deliveries, presented to the emergency room of our tertiary center with a history of heavy vaginal bleeding since 2 - 3 months. She gave history of amenorrhea of 4 months,after which she had consumption of MTP

(Medical Termination of Pregnancy) pills which was 3 months prior to her presentation, following which she started bleeding heavily with passage of fleshy clots. The symptoms persisted till date of presentation, with usage of 5 - 6 pads per day. On examination, her general condition was moderate; she was poorly built, anorexic and pale. Systemic and abdominal examinations were unremarkable. On vaginal examination, uterus was 12 weeks’ size and soft; heavy bleeding with passage of clots was noted.

She was admitted and investigated. Her UPT (urinary pregnancy test) was negative; β HCG level was 15.68 mIU/ml. Her hemoglobin was 8 gm%. Urgent transabdominal sonography was suggestive of bulky uterus with heteroechoic vascular lesion in fundus and endometrium with likely invasion of the anterior myometrium (features favoring malignant etiology). CT scan confirmed increased vascularity in anterior myometrium and endometrium, suggestive of carcinoma uterus, as seen in Figure 1. Tumor markers, serum LDH and TSH were done which were all within normal limits. 

Figure 1. Heteroechoic area showing tissue with blood vessels encroaching on the anterior wall

of uterus suggestive of malignancy.

She was stabilized and treatment in the form of intravenous tranexamic acid 500 mg 8 hourly was instituted along with one unit of blood. As she had an episode of uncontrolled bleeding not responding to medical management, uterine curettage was performed and tissue histopathology was suggestive of exaggerated placental tissue site reaction. She was advised endometrial ablation therapy; however she opted for total abdominal hysterectomy. Hysterectomy specimen showed a bulky, adenomyotic uterus. Histopathology examination revealed fragments of myometrium and hemorrhagic material with few degenerated villi and decidual cells. Multinucleated trophoblastic cells were seen scattered in between myometrial fibres.

Discussion

Intermediate trophoblast is called so because it intermediate in differentiation between cytotrophoblast and syncytiotrophoblast. In early gestation, implantation site intermediate trophoblast is significant for the continuation of pregnancy, and to establish feto-maternal circulation, it infiltrates the decidua and invades the myometrium and spiral arteries. Normally, only the upper half of the myometrium and decidua is invaded by these cells during early gestation. This layer regresses progressively over time during later pregnancy. The condition is labeled as EPS when the intermediate trophoblast infiltrates exaggeratedly into the myometrium and fails to regress or involute over time.

This was a case of menorrhagia presenting as uterine sarcoma or vascular tumor, which on further evaluation turned out to be incomplete abortion. It is a rare diagnosis of intrauterine pregnancy made only on the basis of presence of extravillous trophoblastic cells in myometrium along with the chorionic villi. These cells are highly degenerative and are also called as wandering cells, migratory cells, megalokaryocytes, diplokaryocytes. In hypoxic conditions, there is increased incidence of syncytial knotting leading to abnormal villous shapes.[4]

It is always a dilemma to label a case of EPS in curettage samples based on the histopathological criteria alone, as there are no clear cut guidelines about the degree of myometrial invasion by intermediate trophoblasts in the implantation site at various gestational ages required for it to be labeled as EPS. The standard of care for EPS is endometrial ablation;[5] however our patient opted for hysterectomy as she had completed her family and was unable to ensure follow up. Another important aspect here is distinguishing the lesion from other differentials like uterine sarcoma, other gestational lesions like placental site nodule (PSN), and placental site trophoblastic tumors (PSST). Immuno-histochemical markers used for diagnosis of EPS are cytokeratin (CKAE1/AE3, endomysial antibodies, β-HCG and human placental lactogen, which indicate intermediate trophoblastic origin.[5,6] Recently, CD-146 has been used as a marker for intermediate trophoblasts in the implantation site. It is a cell adhesion molecule which is known to function in the processes of tumor progression, metastasis, implantation, and placentation.[7]

However, recent studies have shown Ki 67 specific (MIB -1) antibody is most useful antibody as is it increased in all other differentials of this lesion but is present in less than 1 % of EPS. A cytogenetic non fluorescent in situ hybridization (NISH) study in cases of EPS indicated that often bizarre nuclei are found in these lesions namely polyploidy or aneuploidy, perhaps arising by cell fusion or endo-reduplication of chromosomes. The intermediate trophoblast in our lesion stained negatively, which favored the diagnosis of EPS.[4,8]

Conclusion

EPS is a rare benign trophoblastic lesion, without any risk of persistent gestational trophoblastic disease. However, awareness of this entity is essential as it must be distinguished from other borderline and neoplastic lesions which might require aggressive treatment. More literature and documentation of cases is required to establish the criteria of diagnosing the lesion confidently on the basis of morphology and immuno-histochemistry. Treating obstetricians should be cognizant of this entity and it should be considered in cases which have unconventional presentation.

References

1. Shih IM, Kurman RJ. The pathology of intermediate trophoblastic tumors and tumor-like lesions. Int J Gynecol Pathol. 2001; 20(1):31-47.
2. Choi JJ, Emmadi R. Incidental placental site nodule in a fallopian tube. Int J Surg Pathol. 2014; 22(1):90-2.
3. Shetty A, Narasimha A, Jyalakshmi VJ. Exaggerated placental site reaction: case report of a rare benign trophoblastic lesion. Int J Reprod Contracept Obstet Gynecol. 2015; 4(5): 1647-1649.
4. Benischke K, Kaufmann P. Trophoblastic neoplasms. Pathology of the human placenta. 4th ed. Germany: Springer 2000; pp.754-767.
5. O'Neill CJ, Cook I, McCluggage WG. Postcesarean delivery uterine diffuse intermediate trophoblastic lesion resembling placental site plaque. Hum Pathol. 2009; 40(9):1358-60.
6. Chen B, Cheng C, Chen W. Transformation of a post-cesarean section placental site nodule into a coexisting epithelioid trophoblastic tumor and placental site trophoblastic tumor: a case report. Diagn Pathol. 2013; 8:85
7. Shih IM. The role of CD146 (Mel-CAM) in biology and pathology. J Pathol 1999; 189:4–11.
8. Luiza JW, Taylor SE, Gao FF,  Edwards RP. Placental site trophoblastic tumor: Immunohistochemistry algorithm key to diagnosis and review of literature. Gynecol Oncol Case Rep. 2013;7:13-5.

Citation

Ostwal P, Shende D, Dhokia T, Chauhan AR. Exaggerated Placental Tissue Site Reaction: A Diagnostic Dilemma. JPGO 2017. Volume 4 No. 2. Available from: http://www.jpgo.org/2017/02/exaggerated-placental-tissue-site.html