Challenges in Diagnosis and Management of Recurrent Uterine Arteriovenous Malformation: A Case Report.

Uterine arteriovenous malformation (AVM) is a potentially life-threatening condition. The vast majority of cases are acquired. Uterine artery embolization (UAE) is currently the treatment of choice for symptomatic women desiring future childbearing. However, there is no consensus on the number of UAE procedures that can be performed on an individual woman. We report a case of recurrent uterine AVM and discuss the challenges in diagnosis and management. A 35-year-old multiparous woman presented with heavy menstrual bleeding (HMB). She had been diagnosed with uterine AVM six years ago and had undergone two previous UAE procedures. Her abdominal examination revealed a healthy Pfannensteil scar. Bimanual examination revealed a normal-sized uterus that was firm, mobile, and fornices were free. Her haemoglobin was 10.2 g/dl. Greyscale two-dimensional ultrasound revealed a normal-sized uterus with multiple hypoechoic lesions in the myometrium. Colour Doppler ultrasound showed intense vascularity with multidirectional flow in the myometrium, suggestive of uterine AVM. In view of previous failed UAE procedures, she opted for a hysterectomy. A total abdominal hysterectomy with a bilateral salpingectomy was performed. Blood loss during the procedure was greater than average, and she was transfused with a unit of packed cells. Her post-operative course was uneventful. Histopathology confirmed the diagnosis of a uterine AVM. To conclude, the UAE is considered the treatment of choice for symptomatic women with uterine AVM desiring future childbearing. In cases of failure of UAE procedures, hysterectomy is therapeutic but may be associated with more than average blood loss.

Successful Management of a Giant Mucinous Cystadenocarcinoma of the Ovary Through Laparotomy: A Case Report.

Giant mucinous cystadenocarcinoma of the ovary is rarely described. Huge ovarian masses are mostly benign, but malignancy should be ruled out by investigations and clinical assessment. Here, we present a case of a large mucinous cystadenocarcinoma of the ovary in a 48-year-old postmenopausal woman. Imaging examinations revealed a large cystic tumor that filled the whole abdominal cavity. Despite the difficulties presented by the size of the tumor and its malignant potential, laparotomy was carried out, which included bilateral salpingo-oophorectomy, total abdominal hysterectomy, exploration of other intra-abdominal organs, and pelvic lymphadenectomy. Histopathology indicated the presence of mucinous cystadenocarcinomas. Adjuvant chemotherapy was given post-operatively, and the patient maintained remission during follow-up. This case emphasizes the need for early detection by simple imaging modalities such as ultrasonography in cases of ovarian masses. Most adnexal masses, if detected early, are amenable to surgical management with a good prognosis. Large masses underline the need for a multidisciplinary approach to improve patient outcomes.

Laparoscopic surgery for endometrial cancer is oncologically safe and improves hospital stay duration: a retrospective single-center study over a 16-year period.

OBJECTIVE: To investigate changes in surgical procedures and patient outcomes of patients diagnosed with endometrial cancer (EC) at a German university hospital between 1998 and 2014. METHODS: A monocentric, retrospective review was conducted to identify patients diagnosed and treated with EC during the aforementioned period at the Department of Gynecology and Obstetrics at the University Hospital Kiel, Germany. RESULTS: 303 patients were identified. Patient demographics, risk factors, histological subtypes and stages of EC remained consistent over time. The most common surgical procedure was total abdominal hysterectomy (TAH) (81.9%). In 2011, the institution carried out its first total laparoscopic hysterectomy (TLH) for EC, resulting in a significant increase in laparoscopic surgical procedures (2011-2014: N = 70; TAH 44.2%; TLH 51.4%). Although the total number of lymph node stagings remained consistent over time, there was a significant increase in the performance of simultaneous pelvic and para-aortic lymphonodectomy (LNE) compared to pelvic LNE alone (2.6 in 2001-2005 vs. 18.0% in 2011-2014, p ≤ 0.001). The duration of hospital stays significantly decreased over time, with a mean of 20.9 days in the first and 8.5 days in the last period. When comparing surgical procedures, TLHs resulted in significantly shorter postoperative stays with an average of 6.58 vs. 13.92 days for TAH. The surgical procedure performed did not affect 5-year overall survival rates in this study (84.9% for TAH and 85.3% for TLH, p = 0.85). CONCLUSIONS: Our retrospective single-center study demonstrates that laparoscopic surgery for endometrial cancer is oncologically safe and shortens hospital stays.

Chilaiditi Sign: A Rare Radiographic Encounter and Diagnostic Exploration.

This case report explores the complex diagnostic dilemma between the Chilaiditi sign and the pneumoperitoneum. The patient presented with chronic complaints of vague abdominal pain, abdominal distension, and breathlessness. A chest X-ray indicated an elevated right hemidiaphragm with transverse colon interposition, leading to the diagnosis of Chilaiditi's sign. Subsequent imaging, including abdominal ultrasound and contrast-enhanced computed tomography, revealed a large non-enhancing multilobulated multicystic mass adherent to the anterior wall of the uterus, raising suspicions of malignancy. Managed by gynecologists, the patient underwent a total abdominal hysterectomy, mass excision, and bilateral salpingo-oophorectomy. The discussion delves into Chilaiditi's sign, its historical context, and its complex pathophysiology involving intestinal, hepatic, and/or diaphragmatic components. Various anatomical and functional factors contributing to this condition are explored. This case highlights the importance of considering the Chilaiditi sign in patients with radiologic evidence of subdiaphragmatic air. It emphasizes the need for timely and accurate diagnosis to differentiate it from more severe conditions like pneumoperitoneum. Such consideration aids in optimizing management strategies and preventing unnecessary investigations.

Emergency Hysterectomy Following Placental Abruption in a Patient With a History of Substance Abuse: A Case Report.

Placental abruption is a serious medical condition that can occur during pregnancy, involving the premature separation of the placenta from the inner uterine wall before childbirth. This detachment often leads to severe bleeding, and if conventional methods prove ineffective in managing the bleeding, a hysterectomy may be deemed necessary to ensure the mother's safety. This case report details the management of a 22-year-old female, gravida IV, para III, who experienced placental abruption during her fourth pregnancy. An emergent cesarean section resulted in severe postpartum hemorrhage and disseminated intravascular coagulation (DIC). Positive drug tests for cocaine and methamphetamines added further complexity, leading to an unplanned hysterectomy for life-saving measures. This case underscores the critical importance of early recognition, multidisciplinary collaboration, and timely intervention in managing obstetric emergencies within the context of substance abuse.

Incidental actinomycosis in a 44-year-old female during total abdominal hysterectomy for abnormal uterine bleeding: A case report.

Actinomycosis, a rare chronic bacterial infection caused by Actinomyces species, presents diagnostic challenges due to diverse clinical presentations. This report presents a case of peritoneal actinomycosis incidentally discovered during a total abdominal hysterectomy in a 44-year-old female with refractory abnormal uterine bleeding and a history of long-term intrauterine contraceptive device use. The patient presented with persistent abnormal uterine bleeding despite conservative management. Intraoperative findings during total abdominal hysterectomy revealed peritoneal involvement, prompting histopathological evaluation confirming actinomycosis. This case highlights diagnostic complexities associated with actinomycosis, emphasizing the significance of histopathological confirmation. Postoperative management with antibiotics demonstrated favorable outcomes, supporting their efficacy in treating actinomycosis. The case underscores the importance of considering uncommon infections in pelvic pathology, particularly in patients with prolonged intrauterine contraceptive device usage. It prompts further exploration of actinomycosis in relation to intrauterine contraceptive device use and highlights the need for timely intervention and histopathological confirmation for optimal patient care.

Iatrogenic Bladder Injury During Laparoscopic Hysterectomy: A Case Report and Discussion of Anatomic Variations.

Hysterectomy, one of the most common surgical procedures performed in women worldwide, assumes a very important role in the definitive management of diverse gynecologic conditions. This case report presents a compelling instance of an iatrogenic bladder perforation that occurred during laparoscopically assisted vaginal hysterectomy in a 47-year-old woman with a high body mass index, extensive surgical history, and postural orthostatic tachycardia syndrome. Despite considerable preoperative planning and the use of minimally invasive techniques, the occurrence of physician-induced bladder perforation highlights the significance of understanding anatomical relationships and variations. The patient's previous abdominal surgeries including two cesarean sections, appendectomy, and cholecystectomy likely contributed to scar formation and adhesions, making dissection challenging. The case report and following discussion delve into anatomical variations, as well as the diagnosis and management of iatrogenic bladder injuries. The presented case serves as a valuable addition to the literature, contributing insights into the challenges and considerations surrounding urinary tract injuries during hysterectomy. This paper aims to review current research and guide practicing obstetricians and gynecologists in the management of intraoperative bladder injuries.

Effect of two different modalities of hysterectomy on wound infection and wound dehiscence in obese patients.

This research intended to investigate the influence of the operation of both kinds of hysterectomies in the risk of wound infection and the degree of wound dehiscence. Both of them were open field and laparoscope. In this research, we looked into four databases: PubMed, Web of Science, Embase and Cochrane Library. Research was conducted on various operative methods for hysterectomy in obese patients between 2000 and October 2023. Two independent investigators performed an independent review of the data, established the inclusion and exclusion criteria, and managed the results with Endnote software. It also evaluated the quality of the included literature. Finally, the data were analysed with RevMan 5.3. This study involved 874 cases, 387 cases received laparoscopy and 487 cases received open access operation. Our findings indicate that there is a significant reduction in the rate of post-operative wound infection among those who have received laparoscopy compared with who have received open surgical procedures (odds ratio [OR], 0.04; 95% confidence interval [CI], 0.01-0.15; p < 0.001); There was no statistical difference between the rate of post-operative wound dehiscence and those who received laparotomy compared with those who received open surgical procedures (OR, 0.33; 95% CI, 0.10-1.11; p = 0.07); The estimated amount of blood lost during the operation was less in the laparoscopy group compared with the open procedure (mean difference, -123.72; 95% CI, -215.16 to -32.28; p = 0.008). Generally speaking, the application of laparoscopy to overweight women who have had a hysterectomy results in a reduction in the expected amount of bleeding during surgery and a reduction in the risk of post-operative wound infections.

An Unforeseen Complication: Intestinal Prolapse After Total Abdominal Hysterectomy.

After total abdominal hysterectomy (TAH), intestinal prolapse is uncommon. We report an instance of a 48-year-old woman who had TAH and then intestinal prolapse. Two weeks after the operation, symptoms started to show up, and the vaginal vault developed a bulging bulge. The problem was satisfactorily treated with an urgent laparotomy. The significance of being vigilant for unusual complications following TAH is shown by this example.

A Comparison between Total Abdominal Hysterectomy versus Total Laparoscopic Hysterectomy.

Objectives: To compare the operative and postoperative outcomes of total laparoscopic hysterectomy (TLH) and total abdominal hysterectomy (TAH). Materials and Methods: In this retrospective comparative study, we reviewed all hysterectomies performed in the Al-Karak Governmental Hospital in Al-Karak, Jordan, from September 2018 to July 2022. We enrolled 129 patients who underwent hysterectomy. The patients were divided into the TLH (n = 39) and TAH (n = 90) groups. Patient data were accessed through hospital records and analyzed using SPSS 25.0. Results: The most common indication for TLH was uterine fibroid, and that for TAH was abnormal uterine bleeding, although the specimen weights were comparable. There was no significant between-group difference in the patient's demographics. Although the TLH group had longer operative time, the hospital stay was shorter and there were no reported cases of wound infection. The estimated blood loss was significantly lower in the TLH group than in the TAH group, but there was no difference between the two groups in terms of blood transfusion requirement and postoperative hemoglobin level. Conclusion: TLH and TAH had comparable overall outcomes in the Al-Karak Governmental Hospital. However, TLH was superior to TAH in terms of blood loss, and patients with TLH recovered faster without postoperative wound infection.

A Case Report of Uterine Müllerian Adenosarcoma With Sarcomatous Overgrowth.

Uterine adenosarcoma remains a highly aggressive tumor and is less described in the literature, with an unfavorable prognosis and an increased risk of local and distant recurrence. However, surgery, chemotherapy, and radiotherapy offer local control of the disease, and overall survival remains reduced. We report the case of a 79-year-old patient with stage IIIB uterine adenosarcoma, confirmed by immunohistochemistry and initially diagnosed with postmenopausal metrorrhagia. The patient was managed through a multimodal treatment by conducting a multidisciplinary consultation.

The uterine adenosarcoma with postoperative residual in a woman treated by total abdominal hysterectomy/bilateral salpingo-oophorectomy: A case report and review of literature.

BACKGROUND: In patients with uterine adenosarcoma, a total abdominal hysterectomy (TAH) with bilateral salpingo-oophorectomy (BSO) is typically recommended as an initial treatment. There is no consensus on adjuvant therapies. CASE: We report the case of a patient with uterine adenosarcoma with postoperative residual disease. We performed four courses of adjuvant chemotherapy, including Ifosfamide, Mesna, and Adriamycin, and whole pelvic radiation with a dose of 50.4 Gy/28 Fr. CONCLUSION: A combination of chemotherapy and radiotherapy may be a promising treatment option for uterine adenosarcoma with postoperative residual disease.

Improvement in Patient Satisfaction and Anxiety With Perioperative Music Therapy in Patients Undergoing Total Abdominal Hysterectomy: A Single-Blind Prospective Study.

Introduction Listening to music is a safe and low-cost way to reduce preoperative anxiety among patients, but more research is needed to evaluate its effectiveness fully. Aims The aim of the study is to identify the effect of intraoperative music therapy on the visual analogue scale for anxiety (VASA) scores (VASA 1 and VASA 2) and patient satisfaction score (PSS) perioperatively. Methods In a study of 188 patients aged 40-70, those in group A (94 patients) listened to pre-approved music during their surgery for abdominal hysterectomy, while group B (94 patients) did not. Both groups wore noise-cancelling earphones. VASA was recorded before (VASA 1) and after (VASA 2) the surgery. PSS was recorded in the postoperative ward. Music preferences were kept confidential from the investigator recording the scores. Result The two groups of patients had similar demographic profiles and baseline characteristics. The VASA 1 of both groups was similar, with a mean value of 4.36 ± 1.13 for group A and 4.23 ± 1.05 for group B (p = 0.606). However, group A had lower VASA 2 (1.79 ± 0.83) than group B (3.77 ± 0.98). The difference was statistically significant (p < 0.001). The patient satisfaction score in group A was notably higher than those in group B. A total of 52 patients were highly satisfied in group A as compared to none in group B (p < 0.001), and a total of 42 patients were moderately satisfied as compared to eight patients in group B (p < 0.001). Eighty-six patients in group B were unsatisfied. Conclusion According to our research, playing specific music at the right volume can significantly lower anxiety levels and increase patients' satisfaction scores for those who have had abdominal hysterectomy surgeries.

Efficacy of Magnesium Sulfate as an Adjuvant to Bupivacaine in Transversus Abdominis Plane Block for Abdominal Hysterectomy Surgeries.

Background Major abdominal surgeries are often accompanied by excruciating pain, which, if not adequately managed, can reduce patient comfort and satisfaction, delay mobilization, compromise respiratory and cardiac functioning, and increase healthcare costs. The transversus abdominis plane (TAP) block is an efficient and safe complement to multimodal postoperative analgesia for abdominal surgery. This study evaluates the efficacy of combining magnesium sulfate (MgSO4) with bupivacaine for TAP block in patients posted for total abdominal hysterectomy (TAH). Methodology Seventy female patients between the ages of 35 and 60 who were scheduled to have a TAH under spinal anesthesia were divided randomly into two groups of 35 each: group Bupivacaine (B) and group Bupivacaine-Magnesium sulfate (BM). Group B received 18 milliliters (mL) of bupivacaine 0.25 percentage (%) 45 milligrams (mg) with 2 mL normal saline (NS) whereas group BM received 18 mL of bupivacaine 0.25% (45 mg) with 1.5 mL of 10% weight/volume (w/v) MgSO4 (150 mg) and 0.5 mL NS in the ultrasonography-guided (USG) bilateral TAP block performed after the end of surgery. Groups were compared for the postoperative visual analog scale (VAS) scores, the time required for first rescue analgesia, the number of analgesic rescues at various time intervals, patient satisfaction score, and any side effects. Results Postoperative VAS scores at 4, 6, 12 and 24th hour (hr) (p < 0.05) in group BM were lower compared to group B. Time required for rescue analgesia was significantly prolonged in group BM (882.94 ± 70.22 minutes) compared to group B (459 ± 100.53 minutes) with minimal usage of rescue analgesia (p < 0.05) up to 12 hr. In group BM, the patient satisfaction score was higher (p = 0.001). Conclusion In addition to a considerable reduction in post operative VAS scores and overall use of rescue analgesia, the addition of magnesium to bupivacaine significantly prolongs the TAP block and increases the initial postoperative period of bearable pain.

Comparative Study of Vaginal Hysterectomy and Total Abdominal Hysterectomy in Non-descent Uterus in a Rural Tertiary Care Center.

Aim To study the role of vaginal hysterectomy in non-descent uterus and to compare it with abdominal hysterectomy with respect to operative time, intraoperative blood loss and complications, ambulation, and postoperative complications. Materials and methods A prospective non-randomized study was carried out on 200 cases at a rural tertiary care center in B.G. Nagara, Karnataka, India for a period of 18 months after obtaining institutional ethical committee approval. One hundred patients underwent a vaginal hysterectomy, and there other 100 underwent an abdominal hysterectomy for similar indications. Results Mean age, parity, mode of delivery, BMI, uterine size, and anesthesia were similar between the groups. The most common indication was fibroid uterus (50%). It was found that the vaginal hysterectomy group was associated with significantly reduced mean operative duration and a decline in postoperative Hemoglobin when compared to the abdominal hysterectomy group. Patients who underwent vaginal hysterectomy had less postoperative pain and were ambulated earlier and discharged earlier. Also, postoperative complications were more common in those who underwent abdominal hysterectomy. Conclusion Vaginal hysterectomy is a safe and the least invasive route and is associated with lesser complications and should be chosen as the preferred method of hysterectomy, whenever feasible.

Therapeutic plasma exchange in hyperthyroidism prior to surgery.

PURPOSE: Therapeutic plasma exchange (TPE) is a treatment option to reduce thyroid hormones in the event of contraindication or unresponsiveness to antithyroid drugs (ATDs). METHODS: We analyzed 11 patients with hyperthyroidism who received TPE prior to surgery between January 2008 and December 2016 at our center. RESULTS: In total, 41 processes were applied to 11 patients with hyperthyroidism. The median age was 40 years, and 90.9% of the patients were female. Seven patients had Graves' disease, while four had a toxic multinodular goiter. The distribution of TPE indications comprised contraindication to ATDs (64%) and insufficient response to ATDs (36%). An adequate response was not obtained with TPE in two patients, and cholestyramine plus methimazole and Lugol solution were applied. The median number of TPE sessions was 3. During the TPE period, a ß-blocker was applied concurrently except in one patient who was contraindicated for the drug. The reduction in FT3 and FT4 hormones and the increase in TSH levels were statistically significant after TPE application (p values of 0.003, 0.033 and 0.008, respectively). Regarding adverse events of TPE application, an allergic reaction was seen in one patient, while prolongation of prothrombin time without any clinical findings was seen in another patient. Ten patients underwent total thyroidectomy, and one patient underwent a gynecological surgery procedure without any major complications. CONCLUSION: The American Society for Apheresis guideline, which is the most referenced guideline, mentions the utilization of TPE before thyroid surgery, only in patients with thyrotoxicosis despite the wider necessity of this treatment choice under the condition of uncontrolled hyperthyroidism prior to any kind of surgery. We concluded that TPE is a reliable and effective application in patients with hyperthyroidism before any surgical procedure, according to our study results.

Comparison of the Efficacy of Single-Injection Regional Analgesia Techniques for Total Abdominal Hysterectomy: A Systematic Review and Network Meta-Analysis.

BACKGROUND: Single-injection regional analgesia techniques can provide effective analgesia for abdominal hysterectomy. However, few randomized controlled trials (RCTs) have directly compared these techniques for total abdominal hysterectomy (TAH), and the best strategy remains unknown. OBJECTIVES: In this network meta-analysis, we compared the analgesic efficacy of single-injection regional analgesia techniques in patients who underwent TAH. STUDY DESIGN: A systematic review and network meta-analysis. METHODS: We searched the PubMed, Embase, Cochrane, and CINAHL databases for relevant trials from inception until April 2022. RCTs that examined single-injection regional analgesia techniques for TAH were included. Random-effects network meta-analyses were performed using the frequentist approach. The primary outcome was 24-hour cumulative morphine equivalent consumption. The secondary outcomes were pain scores, time to first request for rescue analgesia, and rates of postoperative nausea and vomiting (PONV). RESULTS: In total, 36 RCTs were included. Network meta-analyses indicated that the erector spinae plane block provided superior analgesia in terms of reduced morphine consumption, low PONV incidence, and longer time to first analgesia request. Moreover, compared with control (i.e., sham or placebo), the quadratus lumborum block provided superior analgesia in terms of time to first analgesia request and resting pain scores. LIMITATIONS: (1) Few studies have examined single-injection regional analgesia techniques other than the transversus abdominis plane block (TAPB) and wound infiltration, leading to a few indirect effect estimates. (2) Heterogeneity existed due to analgesic type/dose, plane block timing, and injection site. (3) Objective outcomes, such as length of hospital stay, were lacking; most studies only included the patient-reported subjective pain score. CONCLUSION: Single-injection blocks are effective analgesic techniques for TAH. Among them, the erector spinae plane block and quadratus lumborum block seem to have superior effects. Further studies should evaluate techniques other than TAPB and wound infiltration to draw definitive conclusions.

Comparison of transversus abdominis plane blocks with liposomal bupivacaine versus ropivacaine in open total abdominal hysterectomy.

Regional anesthesia is frequently employed in efforts to improve postoperative analgesia and reduce opioid requirements following abdominal surgery. The purpose of the current analysis was to determine if there was a difference in postoperative pain and opioid consumption between patients who underwent open total abdominal hysterectomy (TAH) and received ultrasound-guided bilateral transversus abdominis plane (TAP) blocks using either liposomal bupivacaine or ropivacaine. A single-center retrospective analysis was conducted of 215 patients from November 2018 through March 2020 who underwent an open TAH and received bilateral TAP blocks with either liposomal bupivacaine or ropivacaine. The primary outcome measure was opioid consumption at regular intervals until discharge, and the secondary outcome measures included pain scores, incidence of nausea/vomiting, and use of antiemetics at the same time intervals. Intraoperative opioid consumption and postanesthesia recovery unit opioid requirements were similar between the two groups. Opioid requirements at 24 hours (P < 0.04) and 48 hours (P < 0.01), as well as total morphine equivalent requirements (P < 0.05), were significantly lower in the liposomal bupivacaine group compared to the ropivacaine group. Patients undergoing open TAH who received liposomal bupivacaine TAP blocks required fewer postoperative opioids to achieve similar pain scores when compared to patients who received ropivacaine TAP blocks.

A Rare Case of Recurrent Hematometra of Unknown Etiology.

An abnormal blood collection in the uterus is referred to as hematometra. Obstruction of the genitourinary outflow system caused by earlier surgeries or congenital defects is most frequently related to this rare disorder. The symptoms of hematometra include acute pelvic pain and a history of absent menarche. Here is a case of a 42-year-old female who presented with complaints of severe lower abdominal pain, and pain during urination that was accompanied by vulval itching in June 2021. She had undergone two Caesarean sections and a myomectomy in the past. She was given three monthly injections of gonadotropin-releasing hormone (GnRH) analogue after receiving USG-guided drainage because of a diagnosis of hematometra in January 2021. However, in June 2021, she experienced a recurrence of the same symptoms, necessitating a total abdominal hysterectomy and bilateral salpingo-oophorectomy, which completely resolved the patient's complaints. For a deeper understanding of this issue, further case reporting is necessary.

Concealed Uterine Rupture in the Broad Ligament in an Unscarred Uterus With Postpartum Hemorrhage.

Uterine rupture is an obstetric emergency that traditionally occurs during delivery in a mother with previously known risk factors, especially a uterine scar. However, a rupture that occurs in an unscarred uterus is uncommon. We report a case of a low-risk mother who presented for induction of labor for late-term pregnancy, without a history of uterine surgery, required vacuum-assisted vaginal delivery for maternal exhaustion, and one hour later was noted to have postpartum hemorrhage. The postpartum hemorrhage was managed medically but was taken for curettage due to continued hemorrhage and hemodynamic instability, followed by laparotomy which identified an 11 cm vertical uterine rupture parallel to the ascending uterine artery concealed in the broad ligament and extending inferiorly to the lateral cervix, necessitating a total abdominal hysterectomy. The patient tolerated the procedures well and was discharged home on postoperative day 2. Highlighting the importance of a high index of suspicion for uterine rupture, even without risk factors, this report describes an atypical presentation and describes an effective stepwise approach to management.