Publications by authors named "Yoshitsugu Chigusa"

48 Publications

A Case of Torsion in an Otherwise-Normal Ovary with a Giant Hematosalpinx Larger than Enlarged Ovary: Utilization of Diagnostic Laparoscopy for the Accurate Diagnosis.

Case Rep Obstet Gynecol 2021 23;2021:1371611. Epub 2021 Aug 23.

Department of Gynecology and Obstetrics, Graduate School of Medicine, Kyoto University, 54 Shogoinkawahara-cho, Sakyo-ku, Kyoto City, Kyoto, Japan 606-8507.

We report a case of torsion in an otherwise-normal ovary with a giant hematosalpinx. A 23-year-old woman presented with complaints of abdominal pain and nausea. At initial visit, there was few abnormal findings of imaging tests, and we made a diagnosis of ovarian hemorrhage. Three days later, she came back with increased symptoms, and we detected the mass of a complex solid cystic structure with a unilocular cyst much larger than solid component. A diagnostic laparoscopy was performed immediately, and we could make a diagnosis of torsion in an otherwise-normal ovary with a giant hematosalpinx. We performed a salpingectomy and could preserve her ovary. This is the first case of torsion in an otherwise-normal ovary with a giant hematosalpinx which enlarged to a greater extent than the ovary.
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http://dx.doi.org/10.1155/2021/1371611DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC8408003PMC
August 2021

A 36 kg Giant Ovarian Fibroma with Meigs Syndrome: A Case Report and Literature Review of Extremely Giant Ovarian Tumor.

Case Rep Obstet Gynecol 2021 15;2021:1076855. Epub 2021 Aug 15.

Department of Gynecology and Obstetrics, Graduate School of Medicine, Kyoto University, 54 Shogoinkawahara-cho, Sakyo-ku, Kyoto city, Kyoto, Japan 606-8507.

Ovarian tumors can get extremely giant to occupy the whole abdominal cavity. We report a case of 36 kg solid ovarian tumor, which was the largest ovarian solid tumor that have been ever reported. A 54-year-old woman presented to our hospital with a chief complaint of markedly distended abdominal wall. Preoperative imaging examinations revealed that most of the tumor was uniform and its density was like that of subcutaneous fat. Pleural effusion was detected in the right thoracic region. We organized a multidisciplinary team and successfully resected the right adnexa. The patient had an uneventful postoperative course, and she was discharged on the 7 postoperative day and diagnosed with a fibroma of the ovary with Meigs syndrome. A comprehensive literature search revealed 48 cases of extremely giant ovarian tumor in these 20 years. Six out of 48 cases are solid. Twelve out of 48 cases are malignant or borderline malignant, and patients' age and tumor size/weight were not related to the frequency of malignancy/borderline malignancy. As many as 4 out of 48 patients died before their first hospital visit or early after surgery. Clinicians should consider a considerable high mortality and frequency of severe surgical complications when planning the treatment strategy for extremely giant ovarian tumors.
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http://dx.doi.org/10.1155/2021/1076855DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC8382553PMC
August 2021

Diagnostic Accuracy of Magnetic Resonance Imaging for International Federation of Gynecology and Obstetrics 2018 IB to IIB Cervical Cancer Staging: Comparison Among Magnetic Resonance Sequences and Pathologies.

J Comput Assist Tomogr 2021 Aug 16. Epub 2021 Aug 16.

From the Departments of Diagnostic Radiology and Nuclear Medicine Gynecology and Obstetrics, Kyoto University Hospital Department of Radiology, National Hospital Organization Kyoto Medical Center Department of Diagnostic Pathology, Kyoto University Hospital, Kyoto, Japan.

Objective: This study aimed to investigate the most accurate magnetic resonance (MR) sequence for tumor detection, maximal tumor diameter, and parametrial invasion compared with histopathologic diagnoses.

Methods: Fifty-one patients with International Federation of Gynecology and Obstetrics 2018 IB1 to IIB cervical cancer underwent preoperative MR imaging and surgical resection. Two radiologists independently evaluated the tumor detection, parametrial invasion, and tumor size in each of T2-weighted image, diffusion-weighted image, and contrast-enhanced T1-weighted image. Results obtained for squamous cell carcinoma (SCC) and adenocarcinoma were also compared.

Results: Neither the tumor detection rate nor parametrial invasion was found to be significantly different among sequences. Tumor size assessment using MR imaging with pathology showed good correlation: r = 0.63-0.72. The adenocarcinoma size tended to be more underestimated than SCC in comparison with the pathologic specimen.

Conclusions: Cervical cancer staging by MR images showed no significant difference among T2-weighted image, diffusion-weighted image, and contrast-enhanced T1-weighted image. Adenocarcinoma was prone to be measured as smaller than the pathologic specimen compared with SCC.
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http://dx.doi.org/10.1097/RCT.0000000000001210DOI Listing
August 2021

Maternal near-miss attributable to haemorrhagic stroke in patients with hypertensive disorders of pregnancy in Japan: A national cohort study.

Pregnancy Hypertens 2021 Aug 23;25:240-243. Epub 2021 Jul 23.

Department of Gynaecology and Obstetrics, Kyoto University, Kyoto, Japan. Electronic address:

Objectives: To investigate the epidemiologic and clinical characteristics of maternal near-misses attributable to haemorrhagic stroke (HS) occurring in patients with hypertensive disorders of pregnancy (HDP), with a focus on severe neurological morbidity.

Methods: A national retrospective cohort study was conducted using the national database of health insurance claims for the period 2010 to 2017. The subjects were all insured women with a diagnosis of both HDP and HS. Severe neurological morbidity requiring rehabilitation, types of HDP, types of HS, and magnesium sulphate use were tabulated.

Results: The number of women with HDP who were diagnosed with HS was 3.4 per 100,000 deliveries between 2010 and 2017. Forty percent of HDP-related HS cases had neurological morbidities requiring rehabilitation (1.4 per 100,000 deliveries), and 4.4% were in a persistent vegetative state after HS. Of the HDP cases who developed HS, 69.2% were severe HDP, of which 55.6% were without eclampsia. The most common type of HS was intracerebral haemorrhage (2.5 per 100,000 deliveries), followed by subarachnoid haemorrhage due to cerebral aneurysm (1.2 per 100,000 deliveries). The frequency of magnesium sulphate use increased in all patients with HDP-related HS in the second half of the study period (2014-2017) compared with the first half (2010-2013) (p < 0.0001). This was more evident in cases of HDP-related HS with eclampsia (31.9% to 83.8%) compared to those without eclampsia (25.0% to 42.9%).

Conclusion: Of the maternal near-miss cases due to HDP-related HS, 40.0% were rehabilitated and 69.2% were HDP without either eclampsia or severe hypertension.
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http://dx.doi.org/10.1016/j.preghy.2021.07.244DOI Listing
August 2021

Correction to: Diagnostic performance of preoperative MR imaging findings for differentiation of uterine leiomyoma with intraligamentous growth from subserosal leiomyoma.

Abdom Radiol (NY) 2021 Jul 16. Epub 2021 Jul 16.

Department of Diagnostic Imaging and Nuclear Medicine, Kyoto University Graduate School of Medicine, 54 Kawaharacho, Shogoin, Sakyo-ku, Kyoto, 606-8507, Japan.

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http://dx.doi.org/10.1007/s00261-021-03208-3DOI Listing
July 2021

Three-dimensional human placenta-like bud synthesized from induced pluripotent stem cells.

Sci Rep 2021 Jul 8;11(1):14167. Epub 2021 Jul 8.

Department of Gynecology and Obstetrics, Kyoto University Graduate School of Medicine, 54 Shogoin Kawahara-cho, Sakyo, Kyoto, 606-8507, Japan.

Placental dysfunction is related to the pathogenesis of preeclampsia and fetal growth restriction, but there is no effective treatment for it. Recently, various functional three-dimensional organs have been generated from human induced-pluripotent cells (iPSCs), and the transplantation of these iPSCs-derived organs has alleviated liver failure or diabetes mellitus in mouse models. Here we successfully generated a three-dimensional placental organ bud from human iPSCs. The iPSCs differentiated into various lineages of trophoblasts such as cytotrophoblast-like, syncytiotrophoblast-like, and extravillous trophoblast-like cells, forming organized layers in the bud. Placental buds were transplanted to the murine uterus, where 22% of the buds were successfully engrafted. These iPSC-derived placental organ buds could serve as a new model for the study of placental function and pathology.
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http://dx.doi.org/10.1038/s41598-021-93766-9DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC8266876PMC
July 2021

Sebaceous carcinoma of the vulva treated with sentinel lymph node biopsy: a case report and literature review.

Int Cancer Conf J 2021 Jul 5;10(3):239-243. Epub 2021 May 5.

Department of Gynecology and Obstetrics, Kyoto University, 54 Shogoin Kawahara-cho, Sakyo-ku, Kyoto, 606-8507 Japan.

Sebaceous carcinoma (SC) is a rare and aggressive cutaneous malignancy. It often occurs on the eyelid, where it is called periocular SC, while extraocular SC mainly occurs on the head and heck. Extraocular vulvar SC is extremely rare; only nine cases have been described in the literature, and the optimal treatment strategy is unknown. We herein report a case of vulvar SC that was successfully treated with local excision in combination with sentinel lymph node biopsy (SNB). A 66-year-old female presented with vulvar discomfort. An 8 mm ulcerated mass was palpable in her left labia minora. Skin biopsy suggested SC. Imaging showed no swelling of the pelvic and inguinal lymph nodes and no metastasis. Sentinel lymph node scintigraphy using technetium-99 m showed three sentinel lymph nodes. The patient underwent local excision with SNB; intraoperative frozen-section examination revealed no nodal metastasis, and no further inguinal lymphadenectomy was performed. The final diagnosis was SC of the vulva, FIGO stage IB (pT1bN0M0). At the 14-month follow-up, she remained asymptomatic and had no signs of recurrence. The scientific rationale for SNB in extraocular SC has not yet been established, although SNB can be considered for periocular SC. However, considering the insufficient data on the management of vulvar SC and the aggressive nature of both periocular and extraocular SCs, SNB can be a reasonable and useful method for avoiding inadequate treatment and reducing the complications caused by unnecessary inguinal lymphadenectomy.
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http://dx.doi.org/10.1007/s13691-021-00488-wDOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC8206379PMC
July 2021

Chronic abruption-oligohydramnios sequence (CAOS) revisited: possible implication of premature rupture of membranes.

J Matern Fetal Neonatal Med 2021 May 20:1-7. Epub 2021 May 20.

Department of Gynecology and Obstetrics, Kyoto University, Kyoto, Japan.

Aim: The pathogenic mechanism of chronic abruption-oligohydramnios sequence (CAOS) remains unknown, and there are no objective standards for diagnosis on imaging or using pathological evidence. We aimed to reconsider and clarify the true pathology of CAOS by integrating clinical, magnetic resonance imaging (MRI) and histopathological findings of the placenta.

Material And Methods: This is a case series of patients with CAOS managed at our hospital between 2010 and 2020. The clinical data of the patients, including MRI findings and placental pathology, were reviewed retrospectively.

Results: A total of 18 patients were eligible. Preterm birth occurred in 17 (94%) cases; the median gestational age at delivery was 25. Three neonates (17%) died within two years, and 10 neonates (56%) developed chronic lung disease. MRI was performed in 13 cases and clearly showed intrauterine hematoma and hemorrhagic amniotic fluid. Pathologically, in all cases, retroplacental hematoma was not detected, and fetal membranes were extremely fragile and ragged. Shedding and necrosis of the amniotic epithelium was a characteristic finding, which was confirmed in 17 cases (94%). Diffuse chorionic hemosiderosis (DCH) was detected in all cases.

Conclusions: The fundamental cause of CAOS is repeated intrauterine hemorrhage and subsequent subchorionic hematoma, which induces hemorrhagic amniotic fluid and DCH. Consequently, these factors result in the necrosis and weakening of the amnion. Therefore, the true pathology of CAOS is believed to be premature rupture of membranes rather than chronic abruption.
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http://dx.doi.org/10.1080/14767058.2021.1929159DOI Listing
May 2021

Diagnostic performance of preoperative MR imaging findings for differentiation of uterine leiomyoma with intraligamentous growth from subserosal leiomyoma.

Abdom Radiol (NY) 2021 08 2;46(8):4036-4045. Epub 2021 Apr 2.

Department of Diagnostic Imaging and Nuclear Medicine, Kyoto University Graduate School of Medicine, 54 Kawaharacho, Shogoin, Sakyo-ku, Kyoto, 606-8507, Japan.

Purpose: To evaluate the diagnostic performance of MRI findings for differentiating uterine leiomyoma with intraligamentous growth, or broad ligament fibroid, from subserosal leiomyoma.

Methods: This study included 37 patients with surgically confirmed uterine smooth muscle tumors (36 leiomyomas and one smooth muscle tumor of uncertain malignant potential) with intraligamentous growth (IL) and size-matched control of 37 patients with subserosal leiomyoma (SS). Two radiologists independently evaluated eight preoperative MRI findings: tumor shape, degeneration, attachment to uterus, ovary elevation, ureter displacement, bladder deformation, rectal displacement, and separation of round ligament (RL) and uterine artery (UA). The diagnostic values of these findings and interobserver agreement were assessed. Receiver-operating characteristic (ROC) analysis of the number of positive MRI findings for diagnosing IL was performed. Clinical outcomes including surgical method, operation time, intraoperative blood loss, perioperative complications, and postoperative hospital stay of the two groups were compared.

Results: Significant differences in tumor shape, attachment to uterus, ovary elevation, ureter displacement, and separation of RL and UA were found between IL and SS. Four of these findings, excluding ureter displacement, showed moderate to substantial interobserver agreement. When two or more of these four findings were positive, sensitivity, specificity, and area under the ROC curve were 91%, 77%, 0.90 in reader 1 and 82%, 89%, 0.91 in reader 2. The operation time was significantly longer for IL than for SS.

Conclusion: Tumor shape, attachment to uterus, ovary elevation, and separation of RL and UA are useful MRI findings for differentiating intraligamentous leiomyoma from subserosal leiomyoma.
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http://dx.doi.org/10.1007/s00261-021-03042-7DOI Listing
August 2021

Histological distribution pattern of hemosiderin deposition on the chorionic plate and fetal membrane of diffuse chorioamniotic hemosiderosis related to chronic abruption oligohydramnios sequence.

Placenta 2021 02 20;105:1-6. Epub 2021 Jan 20.

Department of Diagnostic Pathology, Kyoto University Hospital, Japan.

Introduction: Chronic abruption oligohydramnios sequence (CAOS) is histologically characterized by diffuse chorioamniotic hemosiderosis (DCH). However, the criteria for the histological evaluation of the extent of CAOS-related hemosiderin deposition (HD) of the membranes and the difference in HD between the chorionic plate (CP) and fetal membrane (FM) are not well studied. This case control study compared the degree and distribution pattern of HD on CP and FM to present the histological features of DCH and the criteria for histological evaluation.

Methods: From the medical records of Kyoto University Hospital (2010-2019), we selected 20 CAOS cases that were clinically diagnosed by Elliot's criteria. Twenty non-CAOS cases matched to the CAOS group by gestational age were selected as controls. We compared the clinical data and pathological features in the two groups. We performed iron staining in all the cases and analyzed HD in CP and FM according to the histological score (H-Score: 0-12), which was determined as the density (0-3) multiplied by the extent of staining (0-4).

Results: HD was found in 100% (20/20) of CAOS and 15% (3/20) of control cases. In both the FM and CP, CAOS cases showed a significantly higher HS than control cases (CAOS, HS = 4-12; Control, HS = 0-1, p < 0.0001). Three CAOS patients presented HD alone in the CP. The HS of the CP was significantly higher than that of the FM (p = 0.0003).

Discussion: CAOS presented DCH with HS ≥ 4. This study showed that the CP might be more suitable for evaluating DCH than the FM.
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http://dx.doi.org/10.1016/j.placenta.2021.01.013DOI Listing
February 2021

Three cases of seromucinous carcinoma of the ovary arising in endometriotic cysts.

Int Cancer Conf J 2021 Jan 21;10(1):46-53. Epub 2020 Sep 21.

Department of Gynecology and Obstetrics, Kyoto University, 54 Shogoin Kawahara-cho, Sakyo-ku, Kyoto, 606-8507 Japan.

Ovarian seromucinous carcinoma (SMC) is an uncommon neoplasia and is composed predominantly of serous and endocervical-type mucinous epithelium. Due to its low frequency and difficult diagnosis, the natural history, characteristic imaging findings, and pathological features of SMC have not been adequately described in the literature thus far. We herein report three cases of ovarian SMC along with magnetic resonance imaging (MRI) findings. The diagnosis of SMC was made after staging laparotomy in all cases, and systemic chemotherapy was performed in two cases. No recurrence was observed in any of the cases. The MRI findings in SMC were so varied that characteristic imaging features useful for diagnosis were not found. In two cases, MRI suggested endometriotic cysts, and endometriosis and seromucinous borderline tumors (SMBTs) were detected concurrently in all cases by histological examination. Thus, it was suggested that SMC develops in multiple stages via endometriosis and SMBT. The cooccurrence of endometriosis and SMBT could also make the diagnosis of SMC more convincing.
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http://dx.doi.org/10.1007/s13691-020-00447-xDOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7797404PMC
January 2021

Predictive factors for flares of established stable systemic lupus erythematosus without anti-phospholipid antibodies during pregnancy.

J Matern Fetal Neonatal Med 2020 Nov 3:1-6. Epub 2020 Nov 3.

Department of Gynecology and Obstetrics, Kyoto University, Kyoto, Japan.

Purpose: To identify predictors of systemic lupus erythematosus (SLE) flares during pregnancy in patients previously considered to be at low risk.

Materials And Methods: The retrospective cohort study included 54 singleton pregnancies, managed between 2005 and 2019, involving maternal diagnosed SLE at a low disease activity (SLE Disease Activity Index ≤4) for ≥12 months before conception and without anti-phospholipid antibodies. Pregnancy outcomes were compared between patients who had SLE exacerbations during pregnancy (flare group,  = 21) and patients that did not have a flare (non-flare group,  = 33).

Results: The flare group had shorter gestational durations ( = .01), lower birth weights ( = .02), and a higher risk of emergent cesarean section ( = .002) compared with the non-flare group. The flare group demonstrated higher doses of prednisone ( = .04) at the time of conception as well as an increased rate of low 50% hemolytic complement (CH50) activity ( = .03) in the first trimester compared to the non-flare group. A decision tree drawn using a prednisone dose ≥10.5 mg/day and a low CH50 predicted SLE flares with a net accuracy of 78%.

Conclusions: A prednisone dose ≥10.5 mg daily and CH50 hypocomplementemia in early pregnancy are useful in the early detection of patients at a high risk of SLE exacerbation.
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http://dx.doi.org/10.1080/14767058.2020.1843626DOI Listing
November 2020

Severe fetal anemia as a consequence of extra-abdominal umbilical vein varix: A case report and review of the literature.

Congenit Anom (Kyoto) 2021 Jan 5;61(1):4-8. Epub 2020 Nov 5.

Department of Gynecology and Obstetrics, Kyoto University Graduate School of Medicine, Kyoto, Japan.

Umbilical vein varix is associated with a high incidence of fetal anomalies and perinatal complications. There are two types of umbilical vein varix: fetal intra-abdominal and extra-abdominal. Herein, a case is reported of severe fetal anemia with extra-abdominal umbilical vein varix. A 33-year-old primigravida was referred to our hospital for fetal growth restriction, fetal cardiomegaly, and decreased fetal movements at 26 weeks' gestation. A Doppler assessment showed an elevated middle cerebral artery peak systolic velocity at 2.2 MoM, suggesting fetal anemia. Umbilical vein varix had caused intermittent turbulent flow, provoking hemolytic anemia. Intrauterine transfusion improved fetal circulatory failure and anemia and prolonged gestational period. At 33 weeks' gestation, the patient underwent cesarean delivery due to nonreassuring fetal status. Pathological analysis revealed focal loss of vascular smooth muscle of the umbilical vein. Extra-abdominal umbilical vein varix has been reported in 14 cases including this case. The antenatal diagnosis rate is reported to be 79%; fetal heartbeat abnormalities and fetal deaths were reported as 50% and 14%, respectively. Eighty-six percent of patients had intra-umbilical cord thrombosis, but currently this is the only case of hemolytic anemia. Furthermore, extra-abdominal umbilical vein varix may present as fetal hydrops with anemia. During ultrasound examination of fetal anemia, umbilical cord screening should be performed with caution.
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http://dx.doi.org/10.1111/cga.12397DOI Listing
January 2021

osteomyelitis of the distal radius in a healthy pregnant woman.

J Bone Jt Infect 2020 13;6(1):1-5. Epub 2020 Jul 13.

Department of Orthopaedic Surgery, Graduate School of Medicine, Kyoto University, Kyoto, 606-8507, Japan.

Although characteristic, is a rare cause of osteomyelitis, especially in healthy individuals. A 25-year old primigravida at 29 weeks' gestation noticed pain and swelling in her right wrist. Her leukocyte count was normal, but her C-reactive protein level was slightly elevated, at 1.1 mg dL (normal range,  mg dL ). Plain radiography showed an osteolytic lesion in the distal radius, and magnetic resonance imaging (MRI) showed an extraosseous fluid collection with bone edema in addition to the osseous lesion. After a needle biopsy was performed, the skin overlying the lesion became ulcerated at the site of the needle tract. We drained whitish pus from the site; both this pus and the original biopsy specimen grew on culture. We diagnosed osteomyelitis and began intravenous antibiotic therapy, avoiding oral quinolones to prevent fetotoxicity. Her symptoms resolved, as did the bone edema and fluid collection. Ossification occurred at the site of osteolysis, with localized abnormal signal intensity persisting on MRI. This rare case of osteomyelitis was treated without surgery; the patient's pregnancy influenced the treatment course.
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http://dx.doi.org/10.5194/jbji-6-1-2020DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7517663PMC
July 2020

Conservative management for retained products of conception after less than 22 weeks of gestation.

J Obstet Gynaecol Res 2020 Oct 5;46(10):1982-1987. Epub 2020 Aug 5.

Department of Gynecology and Obstetrics, Kyoto University, Kyoto, Japan.

Aim: The aim of the study was to investigate the efficacy of conservative treatment in cases of retained products of conception (RPOC) with a preceding pregnancy of less than 22 weeks and to assess whether serum beta-human chorionic gonadotropin (hCG) levels could be a useful index to monitor the progress of treatment.

Methods: This is a case series of patients with RPOC developed after less than 22 weeks of gestation and managed expectantly with serial serum hCG measurement between 2011 and 2017. The clinical data of subjects were reviewed retrospectively. Cases that did not require invasive treatment such as surgery were designated as conservative management success.

Results: A total of 19 cases were eligible: 14 miscarriages and 5 induced abortions. Eleven patients underwent dilatation and curettage. The diagnosis of RPOC was made 35 (8-80) days after abortion. All patients were successfully treated with conservative management. Serum hCG levels at diagnosis were 29.6 (3.2-1585) mIU/mL. Serial measurement of serum hCG was continued until the levels became lower than the cutoff value, and the mean duration to hCG disappearance was 67.5 (6-183) days. In all cases, RPOC vanished spontaneously 77 (27-184) days after diagnosis. The disappearance of RPOC in the uterine cavity was subsequent to a significant decrease in serum hCG. Once serum hCG levels reached the cutoff value, no bleeding episodes were observed.

Conclusion: Conservative management for RPOC might be acceptable and effective. Furthermore, serial serum hCG levels reflect the activity of RPOC, and hCG may be a reliable index to monitor the progress of treatment.
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http://dx.doi.org/10.1111/jog.14405DOI Listing
October 2020

New era of trophoblast research: integrating morphological and molecular approaches.

Hum Reprod Update 2020 09;26(5):611-633

Human Health Sciences, Kyoto University Graduate School of Medicine, Kyoto, Japan.

Many pregnancy complications are the result of dysfunction in the placenta. The pathogenic mechanisms of placenta-mediated pregnancy complications, however, are unclear. Abnormal placental development in these conditions begins in the first trimester, but no symptoms are observed during this period. To elucidate effective preventative treatments, understanding the differentiation and development of human placenta is crucial. This review elucidates the uniqueness of the human placenta in early development from the aspect of structural characteristics and molecular markers. We summarise the morphogenesis of human placenta based on human specimens and then compile molecular markers that have been clarified by immunostaining and RNA-sequencing data across species. Relevant studies were identified using the PubMed database and Google Scholar search engines up to March 2020. All articles were independently screened for eligibility by the authors based on titles and abstracts. In particular, the authors carefully examined literature on human placentation. This review integrates the development of human placentation from morphological approaches in comparison with other species and provides new insights into trophoblast molecular markers. The morphological features of human early placentation are described in Carnegie stages (CS), from CS3 (floating blastocyst) to CS9 (emerging point of tertiary villi). Molecular markers are described for each type of trophoblast involved in human placental development. We summarise the character of human trophoblast cell lines and explain how long-term culture system of human cytotrophoblast, both monolayer and spheroid, established in recent studies allows for the generation of human trophoblast cell lines. Due to differences in developmental features among species, it is desirable to understand early placentation in humans. In addition, reliable molecular markers that reflect normal human trophoblast are needed to advance trophoblast research. In the clinical setting, these markers can be valuable means for morphologically and functionally assessing placenta-mediated pregnancy complications and provide early prediction and management of these diseases.
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http://dx.doi.org/10.1093/humupd/dmaa020DOI Listing
September 2020

Two Cases of Ectopic Pregnancy Mimicking Gestational Trophoblastic Disease.

Case Rep Obstet Gynecol 2020 16;2020:2417428. Epub 2020 Jun 16.

Department of Gynecology and Obstetrics, Kyoto University, 54 Shogoin Kawahara-cho, Sakyo, Kyoto 606-8507, Japan.

A well-known typical feature of ectopic pregnancy is an evident gestational sac structure outside of the uterus. However, some cases show atypical appearance that is described as a heterogeneous hypervascular mass. We report two cases of ectopic pregnancy that presented heterogeneous findings mimicking gestational trophoblastic diseases but were correctly diagnosed as ectopic pregnancies on MRI. The first case was an interstitial pregnancy in which the patient underwent surgical treatment. The second case was a cesarean scar pregnancy that was treated conservatively but showed spurious enlargement of pregnancy-related lesions after the treatment. Both cases lacked myometrial invasion on MRI, and the patients were diagnosed with ectopic pregnancies. Invasive findings on MRI may discriminate ectopic pregnancy from trophoblastic tumors and avoid unnecessary hysterectomy.
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http://dx.doi.org/10.1155/2020/2417428DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7315310PMC
June 2020

Mechanisms of thrombin-Induced myometrial contractions: Potential targets of progesterone.

PLoS One 2020 4;15(5):e0231944. Epub 2020 May 4.

Department of Gynecology and Obstetrics, Kyoto University, Graduate School of Medicine, Kyoto, Japan.

Intrauterine bleeding during pregnancy is a major risk factor for preterm birth. Thrombin, the most abundant coagulation factor in blood, is associated with uterine myometrial contraction. Here, we investigated the molecular mechanism and signaling of thrombin-induced myometrial contraction. First, histologic studies of placental abruption, as a representative intrauterine bleeding, revealed that thrombin was expressed within the infiltrating hemorrhage and that thrombin receptor (protease-activated receptor 1, PAR1) was highly expressed in myometrial cells surrounding the hemorrhage. Treatment of human myometrial cells with thrombin resulted in augmented contraction via PAR1. Thrombin-induced signaling to myosin was then mediated by activation of myosin light chain kinase- and Rho-induced phosphorylation of myosin light chain-2. In addition, thrombin increased prostaglandin-endoperoxidase synthase-2 (PTGS2 or COX2) mRNA and prostaglandin E2 and F2α synthesis in human myometrial cells. Thrombin significantly increased the mRNA level of interleukine-1β, whereas it decreased the expressions of prostaglandin EP3 and F2α receptors. Progesterone partially blocked thrombin-induced myometrial contractions, which was accompanied by suppression of the thrombin-induced increase of PTGS2 and IL1B mRNA expressions as well as suppression of PAR1 expression. Collectively, thrombin induces myometrial contractions by two mechanisms, including direct activation of myosin and indirect increases in prostaglandin synthesis. The results suggest a therapeutic potential of progesterone for preterm labor complicated by intrauterine bleeding.
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http://journals.plos.org/plosone/article?id=10.1371/journal.pone.0231944PLOS
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7197857PMC
July 2020

HER2-amplified cervical gastric-type mucinous carcinoma with a primitive enterocyte phenotype.

Histopathology 2020 09 29;77(3):511-513. Epub 2020 Jun 29.

Department of Diagnostic Pathology, Kyoto University, Kyoto, Japan.

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http://dx.doi.org/10.1111/his.14119DOI Listing
September 2020

Placenta Accreta in a Woman with Childhood Uterine Irradiation: A Case Report and Literature Review.

Case Rep Obstet Gynecol 2019 5;2019:2452975. Epub 2019 Nov 5.

Department of Obstetrics and Gynecology, Kyoto University Hospital, Kyoto, Japan.

The pregnancies of childhood cancer survivors who have received uterine irradiation are associated with a high risk of several obstetrical complications, including placenta accreta. The present case was a 26-year-old pregnant woman with a history of myelodysplastic syndrome treated with umbilical cord blood transplantation following chemotherapy and total body irradiation at the age of 10. Despite every possible measure to prevent preterm labor, uterine contractions became uncontrollable and a female infant weighing 892 g was vaginally delivered at 27 weeks of gestation. Under the postpartum ultrasonographic diagnosis of placenta accreta, we selected to leave the placenta . Although emergency bilateral uterine artery embolization was required, complete resorption of the residual placenta was accomplished on the 115 day postpartum. Our experience highlighted the following points. (1) The expectant management of placenta accreta arising in an irradiated uterus may not only fulfill fertility preservation, but may also reduce possible risks associated with cesarean hysterectomy. (2) Due to extreme thinning of and a poor blood supply to the myometrium, reaching an antepartum diagnosis of placenta accreta in an irradiated uterus is difficult. (3) The recurrence of placenta accreta in subsequent pregnancies needs to be considered after successful preservation of the uterus.
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http://dx.doi.org/10.1155/2019/2452975DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6875035PMC
November 2019

Novel intrauterine balloon tamponade systems for postpartum hemorrhage.

Acta Obstet Gynecol Scand 2019 12 12;98(12):1612-1617. Epub 2019 Sep 12.

Department of Gynecology and Obstetrics, Kyoto University, Kyoto, Japan.

Introduction: Postpartum hemorrhage is the most common cause of maternal death worldwide. Although intrauterine balloon tamponade has been widely used as an effective procedure to control atonic postpartum hemorrhage, intrauterine balloon tamponade fails to control postpartum hemorrhage in approximately one-fifth of cases. The aim of this study was to evaluate the efficacy of novel intrauterine balloon tamponade systems for postpartum hemorrhage.

Material And Methods: We have developed two novel intrauterine balloon tamponade systems to maintain proper balloon placement. One was a shaft cover with its fixture system and the other was "the Kyoto balloon system" designed to provide direct pressure onto the upper uterine cavity. The efficacy of the intrauterine balloon tamponade systems was evaluated using a silicone three-dimensionally printed postpartum uterine cavity model.

Results: Measurements of balloon displacement during inflation showed that the shaft cover significantly prevented the Bakri balloon from being displaced. The residual fluid volume in the upper uterine cavity was significantly less with the Kyoto balloon system than with the Bakri balloon system, indicating the effectiveness of the Kyoto balloon for upper uterine cavity tamponade.

Conclusions: These innovative intrauterine balloon tamponade systems were effective for prevention of balloon displacement and for balloon tamponade of the upper uterine cavity in a 3D-printed postpartum-specific uterine cavity model.
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http://dx.doi.org/10.1111/aogs.13692DOI Listing
December 2019

Intracervical elastomeric sealant in an model.

J Matern Fetal Neonatal Med 2021 Apr 19;34(7):1109-1111. Epub 2019 Jun 19.

Department of Gynecology and Obstetrics, Kyoto University, Kyoto, Japan.

Background: Premature rupture of membranes occurring in previable midtrimester patients is associated with perinatal mortality, and optimal therapeutic methods are yet to be established.

Objective: This study's objective was to investigate whether an elastomeric sealant, which has been used as a hemostatic agent for arterial anastomosis, could plug the uterine endocervical canal to prevent leakage of intrauterine fluid in an model.

Methods: The elastomeric sealant or fibrin glue was applied to the cervix of uteri removed for benign gynecological disease ( = 4). Normal saline was administered into the uterine cavity through a catheter using a pressure infusion bag. Intrauterine pressure was measured using a digital pressure gauge, and the pressure at which normal saline started leaking out of the uterine cervix was compared between both the sealants.

Results: No fluid leakage was observed with the elastomeric sealant until the pressure exceeded 20 kPa (150 mmHg), while the leakage onset pressure with fibrin sealant was 6.6 ± 1.8 kPa (50 ± 14 mmHg). The threshold leak pressure where the onset of liquid flow was initiated was significantly different between both the sealants ( < .0001).

Conclusions: Intracervical elastomeric sealant exhibited powerful fluid leakage prevention in an model. The sealant would have potential to prevent the leakage of amniotic fluid in pregnancies with previable premature rupture of membranes.
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http://dx.doi.org/10.1080/14767058.2019.1626367DOI Listing
April 2021

Placental sonic hedgehog pathway regulates foetal growth via insulin-like growth factor axis in preeclampsia.

J Clin Endocrinol Metab 2019 May 23. Epub 2019 May 23.

Department of Obstetrics and Gynecology, Kyoto University, Kyoto, Japan.

Background: Placental dysfunction is the underlying cause of common major disorders of pregnancy, such as foetal growth restriction (FGR) and preeclampsia. However, the mechanisms of placental dysfunction are not entirely elucidated. We previously reported 10 reliable preeclampsia pathways based on multiple microarray data sets, among which was the sonic hedgehog (SHH) pathway. Here we describe the significant role of SHH signalling involved in placental development and foetal growth.

Methods: The placental expression levels of surrogate markers of the SHH pathway, patched homolog 1 (PTCH1) and glioma-associated oncogene homolog 2 (GLI2), were evaluated using quantitative real-time polymerase chain reaction (qPCR), western blot analysis and immunohistochemistry. We investigated the underlying mechanisms of the SHH pathway in trophoblast syncytialization, a critical process for placental development and maturation, using primary cytotrophoblasts. Moreover, the potential roles of placental SHH signalling in the regulation of the insulin-like growth factor (IGF) axis were explored by pathway analysis of microarray data. Finally, the influence of SHH signalling on foetal growth was examined by placental administration of cyclopamine, an SHH pathway inhibitor, to pregnant mice.

Results: The SHH pathway was downregulated in preeclampsia placentas and its activation was highly correlated with birth weight. Trophoblast syncytialization was modulated by non-canonical SHH-adenylate cyclase (ADCY) signalling rather than canonical SHH-GLI signalling. The IGF1R pathway was regulated by both non-canonical SHH-ADCY signalling and canonical SHH-GLI signalling. Inhibition of placental SHH signalling significantly reduced foetal weight in mice.

Conclusion: Placental development and foetal growth were regulated through the SHH pathway via the IGF axis.
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http://dx.doi.org/10.1210/jc.2019-00335DOI Listing
May 2019

Maternal Glucocorticoids Make the Fetal Membrane Thinner: Involvement of Amniotic Macrophages.

Endocrinology 2019 04;160(4):925-937

Department of Gynecology and Obstetrics, Kyoto University Graduate School of Medicine, Kyoto, Japan.

Glucocorticoid use during pregnancy is known to increase the risk of preterm birth and preterm premature rupture of membranes (pPROM). Here, we investigated the mechanism of how glucocorticoids weaken the fetal membranes. The amnion mesenchymal layer was significantly thinner in pregnant women treated with prednisolone and in corticosterone-injected mice than in control groups. Matrix metalloproteinase (MMP)-9 mRNA and its activity, COX2 mRNA levels, and prostaglandin E2 synthesis were increased, whereas type 1 collagen (COL1A1) mRNA levels were decreased in the fetal membranes of corticosterone-injected mice. Unexpectedly, the proliferation and migration of macrophages were observed around the corticosterone-injected amnion, and IL-1β was released from these macrophages. In human amnion mesenchymal cells, cortisol did not change MMP mRNA expression, whereas IL-1β treatment robustly increased MMP and COX2 mRNA expression. COL1A1 mRNA level was decreased by both cortisol and IL-1β. These data suggest that the recruitment of amniotic macrophages by glucocorticoids plays a pivotal role in weakening of the fetal membranes, leading to the pathogenesis of pPROM.
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http://dx.doi.org/10.1210/en.2018-01039DOI Listing
April 2019

Human chorionic gonadotropin value and its change prior to methotrexate treatment can predict the prognosis in ectopic tubal pregnancies.

Reprod Med Biol 2019 Jan 11;18(1):51-56. Epub 2018 Oct 11.

Department of Gynecology and Obstetrics Kyoto University Kyoto Japan.

Purpose: To investigate the role of beta-human chorionic gonadotropin (HCG) level and its change prior to methotrexate (MTX) treatment as predictors of treatment success and to access the posttreatment observation period for ectopic tubal pregnancy.

Methods: Clinical data of 41 females treated with MTX for tubal pregnancies were reviewed and analyzed retrospectively.

Results: Among 41 patients, 34 achieved complete resolution without surgery. No statistically significant difference was observed in the presence of hemorrhagic ascites, serum progesterone levels, or diameters of adnexal mass between the MTX success and failure groups. Serum HCG levels on the day of MTX administration (day 1) were significantly lower in the MTX success group. Moreover, % HCG change per day, which represents the increment ratio of HCG prior to MTX treatment, was significantly lower in the MTX success group. Receiver operating characteristic (ROC) curves demonstrated that the treatment success was predicted by % HCG change per day less than +12.6% per day with a sensitivity of 87% and a specificity of 71%. The duration from treatment to complete recovery was strongly correlated with day 1 HCG levels.

Conclusions: Pretreatment HCG change is a significant predictor of therapeutic success of MTX treatment, and the treatment period may be predicted from initial HCG levels.
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http://dx.doi.org/10.1002/rmb2.12247DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6332740PMC
January 2019

Novel subtype of atonic postpartum hemorrhage: dynamic computed tomography evaluation of bleeding characteristics and the uterine cavity.

J Matern Fetal Neonatal Med 2020 Oct 31;33(19):3286-3292. Epub 2019 Jan 31.

Department of Gynecology and Obstetrics, Kyoto University, Kyoto, Japan.

Uterine atony is the leading cause of severe postpartum hemorrhage (PPH); however, the underlying cause of intractable atonic PPH unresponsive to conventional treatments (such as uterotonics and intrauterine balloon tamponade) remains unclear. The aim of this study was to investigate whether intractable atonic PPH is associated with the type of bleeding (arterial or nonarterial) and its location, along with variations in the size and shape of the uterine cavity after delivery. This retrospective study included women who had undergone a dynamic computed tomography (CT) scan for the management of severe PPH at Kyoto University Hospital between April 2011 and March 2017. Patients' electronic medical records were reviewed, and relevant clinical information was collected. The presence of contrast extravasation (CE) on CT images in the early phase (40 s) was regarded as active arterial bleeding. Bleeding sites and size of the uterine cavity were evaluated using an coordinate system. The size of the uterine cavity was compared between groups with CE into the upper and lower parts of the uterine body. Of the 60 women assessed for eligibility, 30 were included in the current analysis. Contrast extravasation was detected in 19 women, with 14 showing CE in the early phase. The presence of CE in the early phase was significantly associated with the need for transarterial embolization (Fisher's exact test,  = .0017). The upper and lower parts of the uterine cavity were 97.4 ± 2.7 mm (mean ± standard error of the mean) and 87.2 ± 3.5 mm in length, respectively. The maximum anteroposterior diameters of the upper and lower parts of the uterine cavity were 23.1 ± 2.6 and 76.0 ± 3.0 mm, respectively, and the largest transverse diameters were 67.3 ± 1.9 and 81.1 ± 2.3 mm, respectively. The group that showed CE into the upper uterine cavity had significantly larger qualitative parameters of the upper uterine cavity compared to the group with CE into the lower uterine cavity. The gate from the lower uterine cavity toward the upper uterine cavity was narrow (anteroposterior diameter of 22.6 ± 2.0 mm, transverse diameter of 40.7 ± 3.3 mm), and the intrauterine balloon was always found in the lower uterine cavity on the CT scan. The upper uterine body was characterized by a flat oval-shaped cavity ( plane), thick uterine wall, and lack of uniformity among bleeding sites (  =  62.4 ± 14.8 mm). In contrast, the lower uterine cavity was a circular shape ( plane) with thin walls, and bleeding sites were located at lateral sides around the level of the internal os ( =  -18.8 ± 4.9 mm). Atonic PPH has a significant subtype, named "PRACE," which is characterized by PPH, resistance to treatment, and arterial CE. The need for embolization can be predicted by the presence of arterial bleeding and its location, along with the shape of the uterine cavity.
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http://dx.doi.org/10.1080/14767058.2019.1571033DOI Listing
October 2020

A Case of Recurrent Esophageal Cancer Treated with Concurrent Chemoradiation Therapy in Pregnancy.

Case Rep Obstet Gynecol 2018 3;2018:1280582. Epub 2018 Dec 3.

Department of Gynecology and Obstetrics, Kyoto University, 54 Shogoin Kawahara-cho, Sakyo-ku, Kyoto 606-8507, Japan.

Esophageal cancer rarely coincides with pregnancy, and only five cases have been reported thus far. The management of esophageal cancer during pregnancy is extremely challenging due to its aggressive nature. We herein report a case of recurrent esophageal cancer in pregnancy. A 41-year-old multigravida with a history of esophageal squamous cell cancer treated with esophagectomy and perioperative chemotherapy was diagnosed with local recurrent carcinoma of the residual esophagus at 16 weeks of gestation. The patient strongly desired to continue the pregnancy, and concurrent chemoradiation therapy (CRT) consisting of 50.4 Gy of radiation, cisplatin, and 5-fluorouracil was carried out from 19 weeks of gestation. CRT was dramatically effective, and the recurrent lesion disappeared. At 38 weeks of gestation, she underwent cesarean section and delivered a healthy female baby. Both maternal and fetal courses were satisfactory, and the patient has been free of disease for 12 months. This is the first case of recurrent esophageal cancer in pregnancy in which CRT was completed without reducing treatment intensity and led to a complete response. Nevertheless, little is known regarding the safety and possible adverse effects of CRT on the fetus. Therefore, deliberate selection of patients and long-term follow-up of the child are necessary.
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http://dx.doi.org/10.1155/2018/1280582DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6304601PMC
December 2018

Metabolomic Profiles of Placenta in Preeclampsia.

Hypertension 2019 03;73(3):671-679

Department of Obstetrics and Gynaecology, National Hospital Organization Kyoto Medical Center, Japan (K.K., I.K.).

Preeclampsia is one of the leading causes of maternal and neonatal mortality and morbidity worldwide. We have previously reported that magnesium sulfate therapy is effective for early-onset (EO) preeclampsia. To investigate the molecular mechanisms underlying this favorable effect, metabolomics analysis of magnesium sulfate-treated preeclamptic placentas was performed using capillary electrophoresis time of flight mass spectrometry. There were significant metabolic differences between EO-preeclamptic placentas (n=7) and other placentas (late-onset preeclampsia [n=3], normal pregnancies [n=10]). In EO-preeclamptic placentas, the glutathione metabolism pathway was markedly upregulated, whereas single-sample gene-set enrichment analysis using a publicly available microarray dataset (GSE75010) showed that the glutathione metabolism pathway was significantly downregulated in EO-preeclamptic placentas compared with nonpreeclamptic controls. Metabolomic profiles showed that magnesium sulfate significantly promoted glutathione production in an immortalized trophoblast cell line under oxidative stress conditions but not under normal conditions. Magnesium sulfate suppressed hydrogen peroxide-induced production of reactive oxygen species. Exploratory analysis revealed that urinary 8-isoprostane was decreased in all 5 women treated with magnesium sulfate for preeclampsia with severe features. These findings suggest that magnesium sulfate is effective for treating EO-preeclampsia partly because of its antioxidant effects on trophoblasts.
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http://dx.doi.org/10.1161/HYPERTENSIONAHA.118.12389DOI Listing
March 2019

Enoxaparin administration within 24 hours of caesarean section: a 6-year single-centre experience and patient outcomes.

J Obstet Gynaecol 2019 May 22;39(4):451-454. Epub 2018 Dec 22.

a Department of Gynaecology and Obstetrics , Kyoto University , Kyoto , Japan.

A caesarean section (CS) is a major risk factor for a venous thromboembolism, and enoxaparin, a low-molecular-weight heparin, has been widely used for thromboprophylaxis. However, it remains unclear whether an enoxaparin thromboprophylaxis has an acceptable safety profile when given early after CS compared to delayed administration, especially in the presence of an epidural catheter. This study aimed to survey cases in which enoxaparin administration was performed within 24 hours of CS and to evaluate patient outcomes with or without epidural anaesthesia. The number of eligible cases were 578: 328 patients received an epidural anaesthesia (epidural group), and 250 did not (non-epidural group). In both groups, no patient developed a spinal epidural haematoma. A wound or a subcutaneous bleeding occurred in 22 (6.7%) and 20 (8.0%) cases in the epidural and non-epidural groups, respectively. One patient developed a mild pulmonary embolism, and one case of asymptomatic deep vein thrombosis was detected. An enoxaparin administration within 24 hours of CS appears to be reasonable, regardless of an epidural anaesthesia. Impact statement What is already known on this subject? A venous thromboembolism (VTE) after a caesarean section (CS) remains a significant cause of maternal morbidity and mortality. Therefore, a thromboprophylaxis using enoxaparin, a low-molecular-weight heparin, has been widely recommended and accepted. However, there is no consensus regarding the optimal timing to initiate an enoxaparin administration after CS in the presence of an epidural catheter. What do the results of this study add? This is the largest study that has collected cases receiving enoxaparin within 24 hours of a CS. Irrespective of the presence of an epidural catheter, no patient developed a spinal epidural haematoma after an early administration of enoxaparin. Furthermore, the incidence of haemorrhagic complications did not increase. What are the implications of these findings for clinical practice and/or further research? Given the significant incidence of VTE after CS and the extremely low frequency of spinal epidural haematomas, it can be justified to initiate thromboprophylaxis with enoxaparin soon after CS. However, appropriately designed, large clinical trials are necessary to examine the safety and efficacy of an early enoxaparin administration after CS. Based on such studies, the starting time of thromboprophylaxis after a CS should be decided.
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http://dx.doi.org/10.1080/01443615.2018.1527300DOI Listing
May 2019

Moyamoya Disease in Pregnancy: A 20-Year Single-Center Experience and Literature Review.

World Neurosurg 2019 Feb 19;122:684-691.e2. Epub 2018 Oct 19.

Department of Gynecology and Obstetrics, Kyoto University Graduate School of Medicine, Kyoto, Japan.

Background: Pregnancy is a potential risk factor for stroke in women with Moyamoya disease. However, the rarity of the disease has limited clinical expertise to ensure a healthy pregnancy. The aim of the present study was to explore the possible risk factors for hemorrhagic and ischemic stroke in pregnant women with Moyamoya disease.

Methods: A retrospective review of cases in our hospital during a 20-year period and a review of the reported data were conducted to investigate pregnancy-related cerebrovascular events in women with Moyamoya disease.

Results: Thirty pregnancies in 20 women with Moyamoya disease were identified in the case review of our hospital. All were previously diagnosed cases, and no stroke had occurred during the study period. In the reported data review, pregnancy-related stroke in women with Moyamoya disease was identified in 54 (44 intracranial hemorrhage and 10 cerebral infarction). Intracranial hemorrhage occurred most commonly during the antepartum period (n = 39; 88.6%), with most events occurring at ≥24 weeks. Of the intracranial hemorrhage cases, 7 (15.9%) were complicated by hypertensive disorders of pregnancy, and 8 patients (18.2%) died of stroke. The onset of cerebral infarction was either in the antepartum (n = 4; 40.0%) or postpartum (n = 6; 60.0%) period. All postpartum cases occurred within 3-7 days after delivery.

Conclusion: Pregnancy-related stroke in patients with Moyamoya disease might be susceptible to gestational age. Intracranial hemorrhage is prone to occur during the antepartum period, especially at ≥24 weeks, and cerebral infarction tends to occur postpartum.
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http://dx.doi.org/10.1016/j.wneu.2018.10.071DOI Listing
February 2019
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