Case Reports in Obstetrics and Gynecology The latest articles from Hindawi © 2017 , Hindawi Limited . All rights reserved. Cesarean Scar Endometriosis: An Uncommon Surgical Complication on the Rise? Case Report and Literature Review Thu, 23 Feb 2017 07:30:05 +0000 Endometriosis is defined by the presence and growth of ectopic functional endometrial tissue outside the uterus. Scar endometriosis has been described following obstetrical and gynecological surgery. It is a rare condition, though probably on the rise, due to the considerable increase of cesarean sections performed worldwide. Its physiopathology is complex; its symptomatology is rich and diverse but thorough clinical examination along with ultrasound imaging and potentially pretherapeutic cytologic evaluation are usually efficient in diagnosing the condition. Treatment is mostly surgical. We report the case of a cesarean section scar endometriosis, managed at a tertiary level center and emphasize the diagnosis and treatment options. Imane Khachani, Abdelhai Filali Adib, and Rachid Bezad Copyright © 2017 Imane Khachani et al. All rights reserved. Spontaneous Pneumomediastinum in Labor Tue, 21 Feb 2017 00:00:00 +0000 Spontaneous pneumomediastinum and subcutaneous emphysema also known as Hamman’s syndrome is a very rare complication of labor that is often related to the valsalva maneuver during the labor. In most case, Hamman’s syndrome is a self-limiting condition, rarely complicated unless there are underlying respiratory diseases. Chest X-ray can be a useful early diagnostic technique in severe clinical presentation. We report an uneventful pregnancy in a primigravid parturient, which was complicated in the late second stage of labor by the development of subcutaneous emphysema, pneumomediastinum, and mild pneumothorax. Spontaneous recovery occurred after four days of conservative management. This condition shows the major interest of labor analgesia especially locoregional techniques. Mohamed Adnane Berdai, Said Benlamkadem, Smael Labib, and Mustapha Harandou Copyright © 2017 Mohamed Adnane Berdai et al. All rights reserved. Pure Primary Ovarian Squamous Cell Carcinoma Perforating the Rectum Mon, 20 Feb 2017 07:12:34 +0000 Rectal perforation is uncommon in ovarian cancer, even in advanced stages. Pure primary ovarian squamous cell carcinoma is a very rare subtype of ovarian cancer and has not been reported to cause rectal perforation. A 50-year-old woman presented with rectal bleeding. Rectosigmoidoscopy suggested perforation of a pelvic tumor into the rectum. Abdominopelvic magnetic resonance imaging revealed a 9 cm heterogeneous mass in the pouch of Douglas. We performed complete cytoreduction, including an en-bloc resection of the tumor and rectosigmoid colon. Histopathology showed squamous cell carcinoma of the left ovary penetrating the rectal wall. A common symptom of rectal bleeding was caused by a very rare entity of ovarian cancer penetrating the rectal wall, but thorough evaluation led to its accurate diagnosis and appropriate treatment. Kazuya Mimura, Aiko Okada, Naotsugu Haraguchi, Kenjiro Sawada, Takuji Tomimatsu, and Tadashi Kimura Copyright © 2017 Kazuya Mimura et al. All rights reserved. Management of Retained Genital Piercings: A Case Report and Review Sun, 19 Feb 2017 07:00:42 +0000 The prevalence of genital piercing among women is increasing. As the popularity increases, the number of complications from infection, injury, and retained jewelry is likely to rise. Techniques to remove embedded jewelry are not well described in the literature. The purpose of this report was to describe a case of a patient with a retained clitoral glans piercing, discuss a simple technique for outpatient removal, and review current evidence regarding associated risks of clitoral piercings. A 24-year-old female presented to the emergency department with an embedded clitoral glans piercing. Local anesthetic was injected into the periclitoral skin and a small superficial vertical incision was made to remove the ball of the retained barbell safely. In conclusion, among patients with retained genital piercing, outpatient removal of embedded jewelry is feasible. While the practice of female genital piercing is not regulated, piercing of the glans of the clitoris is associated with increased injury to the nerves and blood supply of the clitoris structures leading to future fibrosis and diminished function compared to piercing of the clitoral hood. Laura J. Moulton and Amelia M. Jernigan Copyright © 2017 Laura J. Moulton and Amelia M. Jernigan. All rights reserved. Spontaneous Rupture of Pyometra in a Nonpregnant Young Woman Sun, 19 Feb 2017 00:00:00 +0000 A 40-year-old woman presented with severe vaginal bleeding. Initial workup with an abdominal sonography revealed endometrium for about 3 mm and free fluid in the abdomen. Hemodynamic instability with abdominal pain and free fluid in the abdomen prompted blood transfusion and laparotomy. There were about 1000 cc blood and clots in the abdomen at laparotomy. There was a longitudinal rupture from fundus up to cervix at the left side of the uterus. Tearing was in full thickness from serosa to endometrium. Scar of previous cesarean was transvers and not associated with this tearing. There was not any myomectomy scar. Parvin Mostafa-Gharabaghi, Shima Bordbar, Shabnam Vazifekhah, and Mohammad Naghavi-Behzad Copyright © 2017 Parvin Mostafa-Gharabaghi et al. All rights reserved. Can Surgery Be Avoided? Exclusive Antibiotic Treatment for Pelvic Actinomycosis Thu, 16 Feb 2017 00:00:00 +0000 Pelvic actinomycosis is an uncommon, slowly progressing granulomatous infection that has been associated with the presence of intrauterine devices. Due to its unspecific clinical and radiologic findings, it can mimic pelvic or intra-abdominal malignancy leading to mutilating surgery of high morbidity. Rarely, diagnosis is made preoperatively and in most cases surgical intervention is necessary. The patient in our case is a 42-year-old female with an IUD for 15 years diagnosed with pelvic actinomycosis. Patient was uniquely diagnosed preoperatively through paracentesis and treated conservatively with prolonged antibiotic therapy and without any type of surgical intervention. Follow-up at 1 year showed almost complete radiologic resolution of the inflammatory mass, nutritional recovery, and absence of symptoms. Pelvic actinomycosis can be successfully diagnosed and treated medically without surgical interventions. M. P. Ruiz, E. M. Williams, C. M. Markey, A. M. Johnson, and P. B. Morales-Ramirez Copyright © 2017 M. P. Ruiz et al. All rights reserved. Spontaneous Regression of Uterine Arteriovenous Malformations with Conservative Management Thu, 16 Feb 2017 00:00:00 +0000 Uterine arteriovenous malformation (AVM) can cause massive hemorrhage and is often treated with uterine artery embolization (UAE), which may lead to ovarian insufficiency. Thus, avoiding UAE should be considered, particularly in women undergoing fertility treatments. We present three women diagnosed with postmiscarriage AVM on color Doppler by transvaginal ultrasound imaging. They had no genital bleeding and a small mass, measuring 16–22 mm. If estradiol was >300 pg/mL when AVM was diagnosed, then a gonadotropin-releasing hormone agonist was administered. All three women underwent follow-up observation, revealing spontaneous mass disappearance. To avoid ovarian insufficiency risk with UAE, conservative management and close follow-up observation should be considered in patients with AVM without bleeding, particularly during the fertility treatment. Keiko Mekaru, Sugiko Oishi, Kozue Akamine, Chiaki Heshiki, and Yoichi Aoki Copyright © 2017 Keiko Mekaru et al. All rights reserved. Ovarian Mature and Immature Teratomas in Monozygotic Twins: A Case Report of Simultaneous Presentation Wed, 15 Feb 2017 00:00:00 +0000 Mature cystic teratoma is one of the most common kinds of ovarian tumor, and immature teratoma is a rare tumor, representing less than 1% of all ovarian teratomas. Although there are some reports about familial occurrences of ovarian tumors, literature concerning the clinical cases of monozygotic twins is rare. We report the 5-year clinical courses of a 12-year-old Japanese girl with a recurrent bilateral ovarian mature teratoma and her monozygotic twin with a unilateral ovarian mature teratoma and contralateral ovarian immature teratoma. This is the first clinical report on mature and immature teratomas of the ovary in monozygotic twins. Our cases support future clarification of the molecular mechanism and pathogenesis of teratoma. Daisuke Shigemi, Naoki Kawai, and Toshiyuki Takeshita Copyright © 2017 Daisuke Shigemi et al. All rights reserved. Prenatal and Peripartum Management of Patients with Hypofibrinogenemia Resulted in Two Successful Deliveries Tue, 14 Feb 2017 00:00:00 +0000 Fibrinogen is an essential agent involved in maintaining pregnancy and coagulation. Since inherited fibrinogen disorders introduce greater risks for conditions such as placental abruption and postpartum hemorrhage, careful prenatal and perinatal management is essential for this patient population. We report two cases of successful deliveries in patients with hypofibrinogenemia. Case 1 is of a 26-year-old (gravida 1, para 1) woman. The patient’s fibrinogen level increased spontaneously to higher than 300 mg/dL during pregnancy, without treatment. She delivered at week 38 of gestation, with no complications. Case 2 is of a 30-year-old (gravida 3, para 1) woman. We performed repeated infusions of fibrinogen to maintain the level higher than 100 mg/dL during pregnancy and at least 200 mg/dL in the perioperative period; the patient delivered a healthy infant. We identified a new mutation, Hiroshima I (γ278Tyr→His). It is important to maintain appropriate fibrinogen levels in cases of inherited fibrinogen disorders for successful prenatal and peripartum management. Yuko Teraoka, Hiroshi Miyoshi, Kumi Oshima, Satoshi Urabe, Norifumi Tanaka, and Yoshiki Kudo Copyright © 2017 Yuko Teraoka et al. All rights reserved. Low-Grade Appendiceal Mucinous Neoplasm Presenting as an Adnexal Mass Mon, 13 Feb 2017 06:41:10 +0000 Appendiceal tumors are rare, late diagnosed neoplasms that may not be differentiated from adnexal masses even by advanced imaging methods and other diagnostic procedures. They may be asymptomatic and remain undiagnosed until surgery. We report a case of an 80-year-old postmenopausal woman presenting with a pelvic mass and a history of weight loss. The patient underwent laparotomy which revealed an appendiceal mucocele, for which she received a full oncological procedure. The histology report showed a low-grade appendiceal mucinous neoplasm, and the patient underwent six cycles of chemotherapy. Appendiceal tumors should be kept in mind in patients with adnexal mass. P. Panagopoulos, T. Tsokaki, E. Misiakos, V. Domi, C. Christodoulaki, D. Sioutis, and N. Papantoniou Copyright © 2017 P. Panagopoulos et al. All rights reserved. Severe Preeclampsia in the Setting of Myasthenia Gravis Thu, 09 Feb 2017 00:00:00 +0000 Myasthenia gravis (MG) is a rare autoimmune disease that leads to progressive muscle weakness and is common during female reproductive years. The myasthenic mother and her newborn must be observed carefully, as complications during all stages of pregnancy and the puerperium may arise suddenly. Preeclampsia is a common obstetrical condition for which magnesium sulfate is used for seizure prophylaxis. However, magnesium sulfate is strongly contraindicated in MG as it impairs already slowed nerve-muscle connections. Similarly, many first-line antihypertensive medications, including calcium channels blockers and β-blockers, may lead to MG exacerbation. This case describes the effective obstetrical management of a patient with MG who developed severe preeclampsia. The effective use of levetiracetam and various antihypertensive medications including intravenous labetalol is described. A review of the ten reported cases of MG complicated by preeclampsia is examined to aggregate observations of clinical care, with focus on delivery methods, anticonvulsants, and antihypertensive medications. Adam J. Lake, Antoun Al Khabbaz, and Renée Keeney Copyright © 2017 Adam J. Lake et al. All rights reserved. Takayasu’s Arteritis in Pregnancy: A Rare Case Report from a Tertiary Care Infirmary in India Tue, 07 Feb 2017 12:05:07 +0000 Background. Takayasu’s arteritis (TA) is a rare, chronic, inflammatory, progressive, idiopathic arteriopathy, afflicting young women of reproductive age group, causing narrowing, occlusion, and aneurysms of systemic and pulmonary arteries, especially the aorta and its branches. During pregnancy, such patients warrant special attention. An interdisciplinary collaboration of obstetricians, cardiologists, and neurologists is necessary to improve maternal and fetal prognosis. Here a case is reported where a patient with diagnosis of TA, complicated by neurological sequelae, successfully fought the vagaries of the condition twice to deliver uneventfully. Case. 25-year-old G2P1L1 presented at 34 weeks of gestation, with chronic hypertension, with TA, with epilepsy, and with late-onset severe IUGR. Following a multidisciplinary approach, she delivered an alive born low birth weight baby (following induction). Her postpartum course remained uneventful. Conclusion. Pregnancy with TA poses a stringent challenge to an obstetrician. Despite advancements in cardiovascular management and advent of new-fangled drugs, the optimal management for pregnant patients with this disease still remains elusive. Sheeba Marwah, Monika Rajput, Ritin Mohindra, Harsha S. Gaikwad, Manjula Sharma, and Sonam R. Topden Copyright © 2017 Sheeba Marwah et al. All rights reserved. Double Balloon Cervical Ripening Catheter for Control of Massive Hemorrhage in a Cervical Ectopic Pregnancy Thu, 02 Feb 2017 07:59:27 +0000 Cervical pregnancy can be complicated by perfuse vaginal bleeding. Mechanical compression directed at tamponing the cervical vessels can control hemostasis. There are several types of balloon catheters that have been described for cervical compression. However use of a double balloon catheter is a novel approach for cervical tamponade, as one balloon is positioned below the external cervical os and the second balloon is situated above in the internal cervical os. This compresses the cervix from internal os to external os between the two balloons, forming a “cervical sandwich.” We describe this method of cervical tamponade using a silicone double balloon cervical ripening catheter that rapidly controlled hemorrhage in a patient that failed conservative management with methotrexate. Nabila Zambrano, James Reilly, Michael Moretti, and Nisha Lakhi Copyright © 2017 Nabila Zambrano et al. All rights reserved. Peripartum Cardiomyopathy Presenting as Bradycardia Wed, 01 Feb 2017 08:58:59 +0000 Peripartum cardiomyopathy (PPCM) is a disease that typically affects young otherwise healthy women. As PPCM is associated with significant mortality, timely diagnosis is necessary to ensure appropriate care. To our knowledge, this represents the first reported case of PPCM presenting as symptomatic bradycardia. We describe the patient’s clinical presentation and relevant findings and review the potential etiology and ramifications of bradycardia in patients with PPCM. Elisabeth Codsi, Carl H. Rose, Marysia S. Tweet, Sharonne N. Hayes, Patricia J. M. Best, and Lori A. Blauwet Copyright © 2017 Elisabeth Codsi et al. All rights reserved. Sigmoid Volvulus Complicating Postpartum Period Tue, 31 Jan 2017 14:02:51 +0000 Background. Sigmoid volvulus is a rare complication of pregnancy and the puerperium. Case. A 19-year-old patient, gravida 1 para 0 at 41 0/7 weeks of gestation, admitted for late-term induction of labor underwent an uncomplicated primary low transverse cesarean delivery for arrest of descent. Her postoperative period was complicated by sudden onset of abdominal pain and the ultimate diagnosis of sigmoid volvulus. Conclusion. Prompt surgical evaluation of an acute abdomen in the postpartum period is essential; delayed diagnosis and treatment can lead to significant maternal morbidity and mortality. Kelsey E. Ward, Erin Blake, Eduardo Gonzalez, Frederic Pieracci, and Meredith Alston Copyright © 2017 Kelsey E. Ward et al. All rights reserved. Pregnancy Outcome in Cartilage-Hair Hypoplasia, a Rare Form of Dwarfism Tue, 31 Jan 2017 10:44:30 +0000 Background. This case report discusses the pregnancy outcome of a patient with cartilage-hair hypoplasia, a rare form of dwarfism, and multiple previous orthopedic surgeries. Literature on pregnancy outcomes in patients with cartilage-hair hypoplasia is limited. Case. A 32-year-old patient with cartilage-hair hypoplasia presented at 12 weeks’ gestation to the high-risk obstetrics clinic for care. Preterm labor resulted in cesarean delivery at 34 weeks’ gestation with general anesthetic. Breastfeeding was stopped at 6 weeks due to neonatal complications. Conclusion. Pregnancy and delivery were uncomplicated. A multidisciplinary approach allowed for effective management during pregnancy and postnatal care. This is the first known documented case of prenatal care, delivery, and breastfeeding in a woman with this rare disorder. Harshithaa Thavarajah and Anne Berndl Copyright © 2017 Harshithaa Thavarajah and Anne Berndl. All rights reserved. A Diagnostic Dilemma of Recurrent Disseminated Peritoneal Leiomyomatosis with Hypertrophied Omental Vessels: Imaging and Embolization of Omental Branches with Positive Outcome Sun, 29 Jan 2017 06:25:54 +0000 We present a case report of recurrent disseminated peritoneal leiomyomatosis in a 30-year-old female with a past history of laparoscopic myomectomy by a technique of morcellation for a large fibroid in 2014. After one year she presented in 2015 with a well-defined oval shaped fibroid along the anterior abdominal wall, which was supplied by the 10th intercostal artery and a branch of right internal mammary artery. She was again presented after 1 year in 2016 with a large pelvic-abdominal fibroid with blood supply from the omental artery, a branch from the right gastroepiploic artery, and sigmoid branches of inferior mesenteric artery. Jitendra Parmar, Chander Mohan, Deepak Hans, and Maulik Vora Copyright © 2017 Jitendra Parmar et al. All rights reserved. Cystic Endometrioma with Coexisting Fibroma Originating in a Supernumerary Ovary in the Rectovaginal Pouch Sun, 22 Jan 2017 00:00:00 +0000 A supernumerary ovary is an exceedingly rare disorder, in which the structure containing ovarian tissue is located at some distance from the normally placed ovary. 16 cases of endometriosis or tumors originating in a supernumerary ovary have been published in the English literature, but no case of coexisting endometriosis and a tumor has been published. We present the case of a 40-year-old female with cystic endometrioma with coexisting fibroma originating in a supernumerary ovary in the rectovaginal pouch. The present case is the first to be reported with coexisting endometriosis and a tumor originating in a supernumerary ovary. Our experience with this case and the results of our previous studies of rectovaginal endometriosis indicated that the possibility of originating in a supernumerary ovary shall be examined in cases of cystic endometrioma in the rectovaginal pouch. Daiki Ogishima, Asumi Sakaguchi, Hiroko Kodama, Kanako Ogura, Ayako Miwa, Yayoi Sugimori, Shozo Matuoka, and Toshiharu Matsumoto Copyright © 2017 Daiki Ogishima et al. All rights reserved. Salmonella enterica Serotype Typhi Bacteremia Complicating Pregnancy in the Third Trimester Thu, 19 Jan 2017 08:01:41 +0000 Background. Salmonella enterica serotype Typhi (S. Typhi) is an anaerobic gram-negative enteric rod that causes infection when contaminated food or water is ingested and may cause illness in pregnancy. Case. This is a patient who presented at 31 weeks’ gestation with abdominal pain and fever and was diagnosed with S. Typhi bacteremia. Conclusion. S. Typhi should be considered in febrile patients with recent travel presenting with abdominal discomfort with or without elevated liver enzymes. George F. Guirguis, Krunal Patel, Lisa Gittens-Williams, Joseph J. Apuzzio, Kristina Martimucci, and Shauna F. Williams Copyright © 2017 George F. Guirguis et al. All rights reserved. A Case of Malignant Melanoma of the Uterine Cervix with Disseminated Metastases throughout the Vaginal Wall Wed, 18 Jan 2017 07:50:47 +0000 Malignant melanoma (MM) in the female genital tract accounts for less than 2% of all melanomas, and the vast majority associated occur in the vulva and vagina. Primary MM of the uterine cervix is extremely rare and its prognosis is very poor. We report a case of primary MM of the cervix with dissemination throughout the vaginal wall. A 66-year-old woman presented with postmenopausal bleeding. Gynecologic examination demonstrated a 2 cm polypoid blackish-pigmented tumor on the cervix with multiple small blackish-pigmented lesions throughout the vaginal wall. Cervical Pap smear cytology showed malignant melanoma. MRI and PET/CT did not detect any distant or lymph node metastases. She underwent radical hysterectomy, pelvic lymphadenectomy, and total vaginectomy. The pathological diagnosis was FIGO stage IIIA primary cervical MM. She received adjuvant chemotherapy with 6 courses of dacarbazine, but 6 months later, multiple lung metastases were detected. Despite 4 courses of anti-PD-1 antibody (nivolumab) treatment, she died of the disease 13 months after surgery. Tomoko Noguchi, Nami Ota, Yasushi Mabuchi, Shigetaka Yagi, Sawako Minami, Hisako Okuhira, Yuki Yamamoto, Yasushi Nakamura, and Kazuhiko Ino Copyright © 2017 Tomoko Noguchi et al. All rights reserved. Vulvovaginal Infralevator Haematoma Mimicking the Second Stage of Labour Wed, 18 Jan 2017 07:27:15 +0000 Even though they are quite uncommon, puerperal genital haematomas can be associated with serious maternal morbidity. Key findings are significant perineal pain and, depending on the location, visible swelling. However, attention can be drawn to its progression by the rare occurrence of persistent painful “bearing down” efforts, even after the successful delivery of the baby. The final size of this haematoma and the rare presentation make it truly uncommon. The primary goals of treatment include the prevention of further blood loss, minimizing tissue damage, relieving pain, and reducing the risk of infection. Management is generally conservative for small collections, but surgery is indispensable when they acutely expand in size or are large with worsening symptoms. J. O. Awoleke and O. M. Ipinnimo Copyright © 2017 J. O. Awoleke and O. M. Ipinnimo. All rights reserved. Case Report of Successful Childbearing after Conservative Surgery for Cervical Mullerian Adenosarcoma Thu, 05 Jan 2017 00:00:00 +0000 Mullerian adenosarcoma (MA) is a rare tumor variant with low malignancy potential and is reported to account for 8% of all uterine sarcomas. Cervical MAs are reported to occur in relatively younger patients with the mean age of 27 years, while those in the uterine corpus generally present in postmenopausal women. Due to the rarity of cervical MAs, optimal management for these patients (especially younger women) is still under exploration. Here, we describe a case of cervical MA in a woman of reproductive age who was treated by fertility-preserving surgery and successfully delivered a child 18 months later. Seiji Kanayama, Masako Nakamura, Hidekazu Oi, Sumire Sugimoto, Yoshikazu Sasaki, Tomoko Uchiyama, Chiho Ohbayashi, and Hiroshi Kobayashi Copyright © 2017 Seiji Kanayama et al. All rights reserved. Spontaneous Rupture of a Leiomyoma Causing Life-Threatening Intra-Abdominal Hemorrhage Tue, 03 Jan 2017 00:00:00 +0000 Background. Uterine fibroids are common benign tumors in women. Clinical manifestations are well known. Acute complications necessitating emergent surgical intervention are rare. Case. We report a case of a 53-year-old woman with a history of uterine fibroids presenting with acute-onset severe abdominal pain. Imaging indicated massive free fluid and a large partially solid uterine mass. Vitals were consistent with hypovolemic shock. Examination revealed a surgical abdomen. She underwent an emergent laparotomy and total hysterectomy. Surgery revealed 4.5 L of hemoperitoneum and a 15 cm degenerated uterine fibroid with active bleeding. Pathology was consistent with intraoperative findings. She required transfusion of numerous blood products perioperatively. Her postoperative course was uncomplicated. Conclusion. It is rare for a uterine fibroid to spontaneously rupture. However, prompt recognition of this severe complication is critical for expeditious, life-saving surgical management. Melissa Schwartz and Kristin Powell Copyright © 2017 Melissa Schwartz and Kristin Powell. All rights reserved. A Report of Two Cases of Age-Related Changes in Cervical Morphology in Postmenopausal Women with Vaginal Adenosis Mon, 02 Jan 2017 14:39:17 +0000 This paper presents two cases of women who had extensive vaginal adenosis from prenatal DES exposure, extending almost halfway down the vaginal canal. Both women were followed for decades with annual exams and Pap smears until after menopause. Clinical examination in both cases initially showed an absent pars vaginalis of the cervix, vaginal adenosis, and shallowness of the fornices. Several decades of annual exams showed these stigmata of DES exposure gradually disappear as the upper vagina progressively contracted. After menopause the upper vagina in both cases transformed into what appeared to be a normal cervix with all adenosis involuted into a normal endocervical canal. A timeline was created to show the morphological changes that were observed over time. This timeline illustrates how severe vaginal stenosis above the level of the squamocolumnar junction developed in middle age and was followed in the postmenopause by fusion of the upper vaginal walls in the midline resulting in the appearance of a normal, but prolapsed, cervix. Marguerite B. Vigliani Copyright © 2017 Marguerite B. Vigliani. All rights reserved. A Third Surgically Managed Ectopic Pregnancy after Two Salpingectomies Involving the Opposite Tube Mon, 02 Jan 2017 08:18:35 +0000 Recurrent ectopic pregnancy in a remnant fallopian tube after ipsilateral salpingectomy is clinically rare. We report the extremely rare case of a third recurrent ectopic pregnancy after two previous salpingectomy procedures involving the opposite tube. A 26-year-old woman, gravida 3 para 0, experienced three ectopic pregnancies brought about by natural conception, all of which were treated surgically (right partial salpingectomy, right remnant tube resection, and left total salpingectomy). During the two salpingectomy procedures involving the right tube, the patency of the intact left tube was intraoperatively confirmed with indigo carmine. The most appropriate surgical intervention should be discussed when managing recurrent ectopic pregnancies. It might be necessary to perform total salpingectomy to reduce the risk of future recurrence on the remaining tube. Naoyuki Iwahashi, Yoko Deguchi, Yuko Horiuchi, Kazuhiko Ino, and Kenichi Furukawa Copyright © 2017 Naoyuki Iwahashi et al. All rights reserved. Endocervical Carcinogenesis and HPV Vaccination: An Occasional Circumstance or a Gap in the Chain? Mon, 02 Jan 2017 07:21:57 +0000 As a result of the Human Papillomavirus (HPV) vaccination program, the prevalence of precancerous dysplasia and invasive cervical cancer has substantially decreased. In this brief report, we present a case of a young patient who was diagnosed with in situ adenocarcinoma of the cervix. This 30-year-old female had completed the HPV vaccination after she became sexually active and has been undergoing annual gynecological assessments, including clinical examination and Pap test, all of which had been negative. This year, her Pap test revealed a low grade squamous intraepithelial lesion (LGSIL) and additionally a colposcopy was performed. Given the extent of the lesion and since the colposcopy was inadequate, the patient underwent a type 3 large loop excision of the transformation zone and a curettage of the endocervix under local anesthesia. The pathological diagnosis from cervical biopsy revealed an in situ adenocarcinoma of the endocervix with negative limits. The HPV subtypes 16 and 83 were detected with PCR. After proper consultation she decided to preserve her fertility and to undergo a regular follow-up, postponing hysterectomy after the completion of her family planning. In conclusion, this case report highlights the need for diagnostic surveillance regarding HPV-related cervical cancer even after vaccination. Georgios-Marios Makris, Petros Karakitsos, Eugenia Kotsifa, Niki Margari, Nikiforita Poulakaki, Theodoros N. Sergentanis, Marco-Johannes Battista, Charalampos Chrelias, and Nicolaos Papantoniou Copyright © 2017 Georgios-Marios Makris et al. All rights reserved. Trichoepithelioma Arising in an Ovarian Mature Cystic Teratoma Thu, 29 Dec 2016 15:07:31 +0000 Here, we report an extremely rare case of trichoepithelioma (TE)—a benign epithelial tumor originating from the outer root sheath of a hair follicle—arising in an ovarian mature cystic teratoma (MCT) with fluorodeoxyglucose-positron emission tomography (FDG-PET) findings. A 48-year-old Japanese woman presented to our hospital for her annual follow-up of adenomyosis. Ultrasonography and magnetic resonance imaging revealed a left ovarian tumor with irregular-shaped septum, which was suspicious of malignancy. However, tumor marker levels were within normal range. On FDG-PET, the maximum standardized uptake value (SUVmax) of the tumor was 2.9. Laparotomy with left salpingooophorectomy was performed. Pathologic examination revealed the probability of TE, rather than basal cell carcinoma (BCC), arising in an ovarian MCT. After five years of follow-up, the patient had no sign of recurrence. The FDG-PET SUVmax was low in TE, as with other benign tumor. However, future investigation is needed to evaluate the findings of FDG-PET imaging in TE cases. Takashi Suzuki, Satoru Nakayama, Haruka Muto, Shinichi Shimizu, Yoshiro Otsuki, Hiroshi Adachi, and Takeshi Murakoshi Copyright © 2016 Takashi Suzuki et al. All rights reserved. Vulvar Lobular Capillary Hemangioma: A Rare Location for a Frequent Entity Thu, 29 Dec 2016 08:03:39 +0000 Lobular capillary hemangioma, or pyogenic granuloma, is an acquired hemorrhagic benign vascular lesion of the skin and mucous membranes. The pyogenic granuloma of the vulva is a rare finding and a limited number of case reports are available in the literature. To the best of our knowledge this is the first case described as a single pyogenic granuloma on the vulva. F. Abreu-dos-Santos, S. Câmara, F. Reis, T. Freitas, H. Gaspar, and M. Cordeiro Copyright © 2016 F. Abreu-dos-Santos et al. All rights reserved. Uterine Rupture with Cesarean Scar Heterotopic Pregnancy with Survival of the Intrauterine Twin Mon, 26 Dec 2016 09:54:02 +0000 Background. Heterotopic pregnancy is a multiple gestation with both intrauterine and ectopic fetuses. A cesarean scar ectopic pregnancy is when the fetus has implanted over the previous hysterotomy site. A known complication of cesarean scar ectopic pregnancy is uterine rupture, which can cause great morbidity and mortality. Case. 28-year-old G5P3105 at 10 weeks with a dichorionic diamniotic gestation was found to have a ruptured uterus with expulsion of a cesarean scar ectopic pregnancy and retention of the intrauterine fetus. After uterine repair, the singleton gestation reached viability was delivered by emergent cesarean section for placental abruption. Conclusion. Safe management of cesarean ectopic pregnancy requires early diagnosis by ultrasonography. With early detection, management can focus on preventing maternal morbidity of uterine rupture and life-threatening hemorrhage. Kimberly R. Lincenberg, Eric R. Behrman, James S. Bembry, and Christine M. Kovac Copyright © 2016 Kimberly R. Lincenberg et al. All rights reserved. Prenatal Evidence of Persistent Notochord and Absent Sacrum Caused by a Mutation in the T (Brachyury) Gene Mon, 26 Dec 2016 06:01:05 +0000 Caudal regression syndrome (CRS) is a rare congenital disorder characterized by developmental abnormalities of caudal spinal segments. To date, the etiology of CRS is unclear; sporadic cases are strongly associated with maternal diabetes, while familiar recurrence is infrequent. We describe in detail the prenatal clinical and sonographic findings of a recently described hereditary caudal regression syndrome, in four fetuses reported to be homozygous for a mutation in the T (brachyury) gene. The syndrome occurred in three consanguineous, but unrelated families, originating from the same geographical area. All affected fetuses had persistence of the notochord in association with abnormal vertebral ossification, sacral agenesis, and bilateral clubfoot. These findings suggest that, in case of prenatal diagnosis of sacral agenesis, an advanced ultrasound examination should assess the vertebral ossification and the rare persistence of the notochord, in order to rule the involvement of the T gene. F. Fontanella, M. C. van Maarle, P. Robles de Medina, R. J. Oostra, R. R. van Rijn, E. Pajkrt, and C. M. Bilardo Copyright © 2016 F. Fontanella et al. All rights reserved.