Friday, 29 May 2020

Iris Publishers_World Journal of Gynecology & Womens Health (WJGWH)

Struvite “Sand” in the Vagina as Diagnostic Factor for New-onset Vesicovaginal Fistula and Pessary Management When Present

Abstract

Background: In cases of pessary neglect, complications can include vaginal ulcerations, discharge, bleeding, discomfort, voiding and defecatory dysfunction, and fistula formation. In rare occurrences there are case reports involving vaginal stones with the presence of a vesicovaginal fistula (VVF). Case Report: We present the case of a 93-year-old with concern for a possible VVF who required pessary use for her prolapse. Conclusion: This is a unique case since there are no current case reports or management guidelines of sand-like exudate in patients with routine pessary care.
Keywords: Vesicovaginal fistula; Pelvic organ prolapse; Struvite; Pessary management
Abbreviations: VVF: Vesicovaginal Fistula; POP: Pelvic Organ Prolapse

Introduction

Vesicovaginal fistula (VVF) is a known complication of pessary placement, however, most cases are consistent with lack of pessary care. Other complications include vaginal ulcerations, discharge, bleeding, discomfort, voiding and defecatory dysfunction, and fistula formation, however exact complication rates are largely unknown. One oft-cited study quotes a 56% complication rates, however, none were fistulae [1]. Although several case reports of pessary-related fistulae exist, most are in the setting of neglect [2].
Case studies about vaginal stones with the presence of VVF exist, they are rare occurrences, and most case reports describe larger appearing stones [3,4]. The interaction of urinary salt ions, pH, and temperature results in the formation of the microscopic crystals, eventually creating a bladder stone [5]. No reports of copious sand-appearing exudate exist in the literature, and none to guide management of pessary care when present. Furthermore, as described above, most cases of fistula do not occur in a patient with long-term pessary use with regular pessary maintenance.

Case Report

A 93-year-old with vaginal prolapse maintained with ring pessary for the last 6 years, was being evaluated for new incontinence, in retrospect likely the development of a VVF. She had a history of pelvic organ prolapse (POP) and has been managed conservatively with a ring pessary since age 87. She also has history of colon cancer requiring colon resection and anastomosis, and her postoperative course was complicated by an abdominal abscess and wound dehiscence.
She had been receiving regular pessary care with her provider every 3-6 months, and, patient began to complain of copious thin watery discharge. Providers expressed a concern for possible fistula, but none was noted on vaginal exam. Patient noted worsening urinary incontinence, with constant leaking, and pessary was changed from size 1 to size 0. Patient returned with complaints of hematuria. Exam after lavage revealed no heme or erosions, and culture sent from straight catheter and from a vaginal collection were both P. stuarti, further supporting presence of a likely fistula. Estrace was initiated.
The patient began to have copious beige, gritty sand-like debris noted in the posterior vaginal vault (Figure 1). Debris was sent to pathology, which resulted as inflammatory debris, insufficient for diagnosis. Creatinine sent on fluid was 14.0mg/dl, with reference range for first morning void being 28-217mg/dL. A Pyridium test was inconclusive. During consultation with a urogynecologist, a 2mm fistula was noted at the vaginal apex, after clearing the sand-like debris, and the anterior vaginal wall appearing intact. Recommendation was to stop pessary use for a period of time; however, patient declined. She was also unable to apply vaginal estrogen routinely. Given her complex surgical history and medical state she is not a surgical candidate, she was managed conservatively with more frequent vaginal lavage and pessary cleaning (Figure 2).

Discussion

There are case reports in the literature regarding vaginal and vesical stones caused by VVF. One review of the literature analyzed 1190 cases of pessary use and reported rare complication. In this analysis the majority of rare VVF reported were with the use of a Gellhorn or Shelf pessary [11]. There were no vaginal stones caused by VVF. In our case, it was not a single stone that was created but copious fine sand- like crystals. Although there are known risks of fistula formation in patients using pessaries, most are associated with neglect and lack of pessary maintenance. In one case report an 8cm calcified vaginal stone was found in a 56-year-old patient who had neglected care of her pessary over the course of a year [4].
For elderly women, the risk of VVF is mainly associated with surgery and/or radiation, with fistula rates ranging from 1.4- 9% depending on order of surgery and radiation, with greater complication rates if surgery is followed by radiation therapy [5]. The fistulas associated with surgery are mainly due to injury to the bladder at the time of surgery, which accounts for 90% of VVF in the industrialized world, and most present with leakage of urine 5-10 days postoperatively [6]. Patients with radiation have been known to develop VVF at a distant interval, up to 20 years later, after pelvic radiation for malignancy, however this patient did not receive radiation, and her surgery was remote, having occurred 4 years prior to the fistula formation.
The literature notes that prevention of fistulae is key in pessary use, as well as regular follow-up and maintenance. If a patient is unable to remove a pessary on her own, she has to have regular follow-up; although there are no clear guidelines. Manufacturers recommend follow-up every 4-6 weeks on package inserts, but other studies have noted safe management with visits every 3 months for the first year after fitting and every 6 months in subsequent years with no serious complications [7,8]. Erosions and lesions should not be ignored and should be treated with pessary removal and vaginal estrogen cream until ulcers heal. The only consideration our case is that she was not on maintenance estrogen cream; however, given her complex surgical history, it is unclear if estrogen would have prevented formation of a VVF.
No guidelines exist on medical or expectant management of VVF in the extremely elderly. One case in the literature reported a delayed VVF in an 82-year-old managed with a urinary diversion via ileal conduit [9]. Another case report, of a VVF resulting from a well-cared-for pessary after 12 years of use performed a Latzko fistula repair and LeFort colpocleisis with subsequent complete resolution of POP and VVF [10]. We postulate, however, that with frequent pessary cleanings and lavages of the vagina to remove struvite sand, a surgical procedure can be avoided.

Conclusion

To conclude, pessaries are safe and play an important role in managing pelvic organ prolapse. Severe complications such as rectovaginal fistula and VVF are rare, with most related to neglect; however, in patients with prior pelvic surgery, especially if they also received radiation therapy for malignancy, fistulae are also possible.



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Thursday, 28 May 2020

Iris Publishers_World Journal of Gynecology & Womens Health (WJGWH)

Rate and Risk Profile of Deep Venous Thrombosis in Pregnancy and Postpartum Period Among Sudanese Women

Abstract

Background: Deep venous thrombosis (DVT) during pregnancy is associated with high mortality, morbidity, and costs. DVT can also result in long-term complications that include post thrombotic syndrome (PTS) adding to its morbidity.
Objective: To determine rate, timing and risk profile of deep venous thrombosis during pregnancy and puerperium among Sudanese women.
Methodology: It was prospective case control and hospital-based study carried out at Shendi Teaching and Elmec-Nimir University Hospitals -Sudan from March 2017 to March 2018. Seventy-eight pregnant women or in puerperium with Doppler confirmed deep venous thrombosis were enrolled in the study, representing the main study group, while another 156 pregnant women without DVT were selected as the control group.
Results: The current study showed the frequency of DVT was (0.622%), 78 out of 12,542 deliveries during the whole study period, with (0.176%) and (0.446%) occurring antenatal and postnatal respectively. The rate was 622 DVT per 100,000 births. This study revealed that women who are primigravida, had positive family history of VTE, had past history of DVT, had Anti phospholipids, anemia and delivered by C/S showed statistically significant association with DVT.
Conclusions: The prevalence of DVT in our study was 622 per 100 000 births per year in pregnant and postpartum women. There is an urgent need for prophylaxis measures against DVT for pregnant women who found at higher risk for DVT.
Keywords: Deep Venous thrombosis; Pregnancy; Postpartum; Rate; Risk profile; Sudanese women

Introduction

Venous thromboembolism is a leading cause of maternal death in developed and developing world. Venous thromboembolism can occur throughout pregnancy with an estimated antenatal and postnatal incidence of 6-12 and 3-7 per10, 000 maternities respectively. Over 40% of antenatal venous thromboembolism occurs in the first trimester of pregnancy [1].
The daily risk of venous thromboembolism is four-fold higher in post-natal period compared with antenatal period [2]. The factors which increase the risk of venous thromboembolism in pregnant and postnatal women are age more than 35, prime party, pre-eclampsia, varicose veins, obesity, cesarean section, previous VTE, family history of VTE , patients with inherited thrombophilias, excessive blood loss, long haul travel, prolonged labor, instrumental delivery and immobility after delivery [3].
Clinical diagnosis of deep venous thrombosis by, leg pain, swelling, tenderness, high temperature, oedema, lower abdominal pain and high total white blood cell count. It may also present with features of pulmonary embolism; dyspnea, chest pain, collapse, haemoptesis, fainting and increased jugular venous pressure [4].
Compression duplex ultrasound of the entire proximal venous system is considered the optimal first-line diagnostic test for DVT in pregnancy [5,6]. An apparently normal ultrasound examination in a patient with significant symptoms and signs or risk factors for VTE does not exclude a calf DVT, so serial ultrasound examinations should be repeated [7,8]. When iliac vein thrombosis is suspected because the woman reports back pain and swelling of the entire limb, pulsed Doppler, magnetic resonance venography, or conventional contrast venography should be considered [9,10]. This study attempts to determine rate, timing and risk profile of deep venous thrombosis during pregnancy and puerperium. among Sudanese women.

Materials and Methods

It was prospective case control and hospital-based study carried out at Shendi Teaching and Elmec-Nimir University Hospitals -Sudan from March 2017 to March 2018. Seventy eight pregnant women or in puerperium who presented with symptoms suggestive of DVT and Doppler confirmed deep venous thrombosis were enrolled in the study, representing the main study group, while another 156 pregnant women from the population of patients presenting to the same hospitals without symptoms of VTE were selected as the control group. Participants completed a questionnaire on personal data and clinical history. Questions regarding known risk factors for DVT such as age, parity, mode of delivery, family history or past history of VTE, medical disease history, personal history of antiphospholipid, sickle disease and thrombophilia. BMI was calculated which defined as the weight in kilograms divided by the square of the height in meters (kg/m2). The BMI was determined by using World Health Organization (WHO) classification for obesity.
Statistical analysis was performed via SPSS software (SPSS, Chicago, IL, USA). Continuous variables were compared using student’s t test (for paired data) or Mann–Whitney U test for nonparametric data. For categorical data, comparison was done using Chi-square test (χ²) or Fisher’s Exact test when appropriate. A P value of <0.05 was considered statistically significant.
Ethical clearance and approval for conducting this research was obtained from the general manager of the hospitals and informed written consent was obtained from every respondent who agreed to participate in the study. The respondents informed that the study is not associated with experimental or therapeutic intervention while information was collected from them.
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Tuesday, 26 May 2020

Iris Publishers_World Journal of Gynecology & Womens Health (WJGWH)

Haemoglobin D Iran With Beta Thalassemia in A Primigravida With Anaemia


Abstract

Anaemia is quite common in female population of developing countries including Pakistan. Beside haemoglobinopathies are often seen in India, Pakistan and Iran due to traditional practices of consanguineous marriages. Haemoglobin D-Punjab is one of the most common subvariants (55%) of Haemoglobin D, which can be inherited as a homozygous or a heterozygous trait with other haemoglobinopathies. Although, Haemoglobin D-Punjab is commonly seen but another variant of HB D like Hb D Iran with heterozygous trait of β thalassemia is rarely reported. We present a rare case of co-inheritance of Haemoglobin D- Iran and β thalassemia in a young primigravida of Kashmiri origin in her twenties. She was six months pregnant and was diagnosed to have anaemia. She had history of diarrhea. On examination, she was pale looking and mildly icteric. Systemic examination was insignificant except for mild splenomegaly. The fundal height was normal for age to date. On investigation, she had mild anaemia with microcytic hypochromic blood picture. Hemoglobin electrophoresis showed a band of Hb A2+Hb F+S/D. Molecular studies of combined Haemoglobin D –Iran and beta thalassemia. She was counseled about his disease and advised follow-up up at 6 months after delivery after delivery.
Keywords: Anaemia; Haemoglobinopathy; Primigravida; Thalassemia

Introduction

Hemoglobin D (Hb D), is a variant of hemoglobin that occurs mainly in north-west India, Pakistan and Iran [1]. The replacement of glutamic acid with glutamine in normal Hemoglobin A at 121 position on its beta chain leads to a new structurally different variant of haemoglobin called Hb D [2]. Hb D is clinically silent in its heterozygous state, but coinheritance of Hb D with either Hb S or thalassemia produces clinically significant conditions like sickle cell anemia and chronic hemolytic anemia of moderate severity [3].
The Hb D Iran trait and homozygous cases have been already reported in the literature [4,5]. However, few studies have reported compound heterozygotes of Hb D Iran with other Hb variants [6]. We report a rare case of compound heterozygous condition of Hb-D β thalassemia trait in a Kashmiri family living in Azad Kashmir, Pakistan.

Case Report

A young six-month prime gravida of Kashmiri origin from Muzaffarabad, Azad Kashmir Pakistan in her twenties presented with chronic diarrhea with history of chronic anemia. She was nonsmoker, nonalcoholic. There was no past history of parasitic infestation, blood loss, weight loss, tuberculosis, any other disease. She was treated by various GPs with oral hematinics without much improvement of her symptoms & correction of anaemia. She was believed to have diarrhea due to iron therapy and was lately administered parenteral iron therapy. However, her Hb remained 9gm/dl and did not improve. On examination, she was conscious and oriented in time place and person. Her BP was100/70mm Hg and pulse rate of 80 beats per minute. She was fair colored with mild jaundice. There was no cyanosis, clubbing, koilonychia, lymphadenopathy or edema. Systemic examination was normal except for mild splenomegaly. Ultrasound abdomen showed 24 weeks fetus, normal sized liver and an enlarged spleen of 5cms in size. Her stool examination revealed cysts of Entamoeba histolytica. She was treated with tablet metronidazole 800mg tid for 10 days. Her diarrheal symptoms disappeared and follow up stool examination was normal. Retrospectively, her family history revealed consanguine marriage of her parents. Complete blood picture showed Hb of 8.8g/dl, WBC count of 13.5 X 103/ul with 68% polymorphs. RBC count was 5.16x 106/ul. MCV was 60.8 fL, MCH 17.1pg and HCT 31.4%. Platelets were 232x103/ul. Peripheral blood film showed microcytic hypochromic blood picture with moderate anisopiokilocytosis and a few target cells. Reticulocyte count was 5.5%. Hemoglobin electrophoresis was advised to rule out haemoglobinopathy. It showed a band of Hb A2 plus Hb F plus Hb S/ D. Hb F was 0.8% and H A2 3.1%. Sickling test was negative. PCR for thalassemia showed a mutation in Fr 8-9(+G) and another Hb D (Iran). A diagnosis of combined haemoglobin D –Iran and β thalassemia were made. Urine examination was normal. The diagnosis was conveyed to the patient. No intervention was needed, however, she was counselled regarding the condition and advised to follow up at 6 months after delivery for blood screening for haemoglobinopathy.

Discussion

Hemoglobinopathies are one of the common blood disorders seen in Pakistani population. Mutations in the globin genes can cause either a quantitative reduction in output from that gene or alter the amino acid sequence of the protein produced. Quantitative defects cause thalassemia, whereas qualitative changes are referred to as Hb variants. These hereditary disorders of Hb pose a massive health problem in many third world countries including India, Pakistan, and Iran [7].
In a retrospective study conducted at National Institute of Blood Disease & Bone Marrow Transplantation, Karachi, Pakistan, a total of 2739 patients blood samples were analyzed from 2010-2014. The overall frequency of hemoglobinopathies was 34.2%, among which beta Thalassemia minor was 51.8% and Hb D trait was 6.7% and Beta Thalassemia major was 24.1%.7 Nearly similar findings were observed in other studies conducted by in Pakistan [8,9].
Thalassemias are inheritable common genetic disorder worldwide due to alteration in haemoglobin (Hb) production. About 4.83% of world’s populations carries of globin chain variants including 1.67% of the population which is heterozygous for α-thalassemia and β-thalassemia [7].

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Monday, 25 May 2020

Iris Publishers_World Journal of Gynecology & Womens Health (WJGWH)

Enormous Ovarian Borderline Serous Cystic Fibroma in Postmenopausal Patient. A Case

Ovarian serous cystadenomas are benign tumors that arise from ovarian epithelium. They represent the commonest type accounting for approximately 60%.
They affect all ages with a peak incidence between the 4th and 5th decade of life. Their definite differentiation from other ovarian masses is based on histological findings. Even though serous cystadenomas are quite commonly diagnosed ovarian masses, the diagnosis of unusually enormous masses is still quite rare.
This case report refers to the case of a post-menopausal 52-year-old woman, admitted at our Department, complaining of abdominal swelling over the past 3 years. An abdominal CT scan performed, revealing an enormous mass, originating from the right ovary, extending throughout the whole abdominal cavity towards the diaphragm up to the xiphoid process. Patient underwent exploratory laparotomy revealing an ovarian mass with median diameter 30x25x15cm.
She underwent a total abdominal hysterectomy with bilateral salpigoopherectomy and dissection of the momentum, as part of surgical staging of the lesion. Final histopathological report confirmed the details of the frozen biopsy, announcing the presence of serous cystic fibroma of borderline malignancy.
Multidisciplinary approach proposed follow up of the patient with assiduous imaging findings.

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