Journal of Japan Society of Perinatal and Neonatal Medicine
Online ISSN : 2435-4996
Print ISSN : 1348-964X
Volume 57, Issue 2
Journal of Japan Society of Perinatal and Neonatal Medicine
Displaying 1-30 of 30 articles from this issue
Review
  • Yoshiaki Sato
    2021Volume 57Issue 2 Pages 234-242
    Published: 2021
    Released on J-STAGE: September 06, 2021
    JOURNAL FREE ACCESS

     Stem cell therapy has brought hope for patients with various diseases for which no adequate treatments are available. Stem cell therapy has many strengths and advantages. Some stem cells have the potential to replace injured cells and regenerate on their own. In addition, stem cells produce a large number of growth factors that act on a variety of mechanisms to produce therapeutic effects. Stem cells have the potential to home to the location where they are needed, i.e., stem cells can move to or around a lesion, even when they are administered intravenously. Moreover, stem cells act according to their situation and location. Stem cell therapy has not only anti-inflammatory and antiapoptotic effects but also regenerative effects, including the ability to enhance angiogenesis and neurogenesis. Therefore, there is more possibility for stem cell therapy to be effective when administered even at later time point. Offering these strengths and advantages, stem cell therapy may be able to provide therapeutic effects that are not provided by conventional drugs.

     In the development of treatments for hypoxic-ischemic encephalopathy(HIE), several safety and ethical concerns are associated with the use of embryonic stem cells or neural stem cells. However, after the confirmation of their efficacy and safety via animal experiments, treatments using cord blood stem cells and Muse cells have started undergoing clinical trials. If treatments based on cell formulation are developed, which can be administered intravenously, it will become possible to treat many neonates with HIE.

     In the development of treatments for cerebral palsy, clinical trials are ahead of basic research. Several clinical trials have already been reported, and their findings have mainly shown therapeutic effects related to motor skills. Regarding chronic lung diseases, many basic experimental results have been reported and clinical trials are also being conducted. In addition, clinical trials for the treatment of intraventricular hemorrhage are currently underway. Furthermore, animal experiments have been conducted in order to develop treatments for necrotizing enterocolitis, periventricular leukomalacia, and fetal growth restriction. Subsequently, additional future developments are expected in stem cell therapy for various diseases.

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General Remarks
  • Nozomi Kubo, Haruta Mogami, Masaki Mandai, Eiji Kondoh
    2021Volume 57Issue 2 Pages 243-250
    Published: 2021
    Released on J-STAGE: September 06, 2021
    JOURNAL FREE ACCESS

     The microbiome of neonates is influenced by maternal microbiome, prenatal environment and delivery mode. Postnatal factors such as usage of antibiotics, diet, breast feeding, or environmental exposure further modulate the infant’s microbiome. The infant’s microbiome shapes the immune system, and altered microbiome in the early life increases the risk for the diseases such as asthma, inflammatory bowel diseases, and autoimmune diseases. Recently, microbiome-mediated therapeutics such as vaginal seeding have been attempted to improve the microbiota of newborns and prevent the subsequent onset of allergic diseases.

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Originals
  • Itsuka Nagamitsu, Atsuhiko Sakai, Nobutaka Hachisuga, Yuka Sato, Saki ...
    2021Volume 57Issue 2 Pages 251-256
    Published: 2021
    Released on J-STAGE: September 06, 2021
    JOURNAL FREE ACCESS

     Placental abruption is a major disease in obstetrics in terms of the need for rapid and accurate management. Therefore, clinicians need to be familiar with its clinical features. Although placental abruption can occur in extremely early gestational weeks, its clinical features are poorly understood because of its rarity. We performed a retrospective study to reveal the clinical characteristics of placental abruption, occurring between 22 and 28 gestational weeks. In the seven cases included in this study, the mean gestational age at onset was 26 weeks and 1 day. All patients had already known risk factors such as advanced maternal age, hypertensive disorders of pregnancy, and chorioamnionitis. In particular, there were three cases of histological chorioamnionitis. There was no maternal death. There was one intrauterine fetal death, one neonatal death, and two preterm infants who survived without sequelae. In our study, extremely early onset placental abruption tended to be associated with acute intrauterine inflammation. Thus, while treating diseases with an inflammatory background, such as premature rupture of membranes and preterm labor, special caution is required for the possible occurrence of placental abruption. The vital and neurological outcomes of children born after early-onset placental abruption were poor.

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  • Rumiko Yamamoto, Shusaku Hayashi, Nobuaki Mitsuda, Keisuke Ishii
    2021Volume 57Issue 2 Pages 257-262
    Published: 2021
    Released on J-STAGE: September 06, 2021
    JOURNAL FREE ACCESS

     Objective:

     The objectives were to assess the test properties of prolonged deceleration and bradycardia immediate before delivery for predicting neonatal acidemia and to examine changes in test properties by addition of other features on cardiotocogram.

     Method:

     This was a retrospective cohort study of women who labored singleton, cephalic, and non-anomalous fetuses at term between 2016 and 2018 at our hospital. Terminal deceleration was defined as prolonged deceleration or bradycardia within 30 minutes before delivery. The sensitivity, specificity, positive predictive value, and negative predictive value of terminal deceleration for neonatal acidemia(umbilical cord arterial pH <7.2)were calculated. Among cases with terminal deceleration, associated factors among other features on cardiotocogram for neonatal acidemia were explored using logistic regression analysis. Test properties of terminal decelerations combined with associated factors were also calculated.

     Result:

     Of the 2, 480 women included in the analysis, 362 experienced terminal deceleration. The sensitivity, specificity, positive predictive value, and negative predictive value of terminal deceleration for neonatal acidemia were 35.6%, 87.0%, 17.7%, and 94.5%, respectively.

     Multivariate logistic regression analysis revealed that baseline >160 bpm before terminal deceleration, cardiotocogram level ≥4 before terminal deceleration, deceleration time ≥6 minutes, and minimal variability at the nadir of terminal deceleration were independent factors associated with neonatal academia among cases of terminal deceleration. Combining with these associated factors, positive predictive value of terminal deceleration for neonatal acidemia was improved.

     Conclusion:

     The positive predictive value of terminal deceleration for neonatal acidemia was below 20%. When accompanied with preexisting fetal tachycardia or fetal distress, duration of ≧ 6 minutes, and minimal variability at the nadir, positive predictive value of terminal deceleration for neonatal acidemia was increased, therefore, it should be considered as sign indicating the need for urgent delivery and neonatal resuscitation.

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  • Maki Toyama, Tetsuhiro Sakihara
    2021Volume 57Issue 2 Pages 263-268
    Published: 2021
    Released on J-STAGE: September 06, 2021
    JOURNAL FREE ACCESS

     Objective: To evaluate factors of neonatal respiratory distress(NRD)using perinatal information in general city hospitals, and develop a prediction tool for NRD.

     Methods: We developed a prediction model from factors related to NRD in the neonates born between January 2018 and December 2019(development dataset)and compared it with a prediction based on the presence of rapid delivery due to nonreassuring fetal status(NRFS). Receiver operating characteristic curve analyses were used to examine the predictive ability for NRD. The prediction ability for NRD was verified in the neonates born between January and December 2020(validation dataset).

     Results: Late term, cesarean section, rapid delivery due to NRFS, and umbilical cord arterial blood pH <7.12 were associated with NRD in development dataset which consists of 1,209 neonates including 73 of NRD. The prediction ability of the prediction model developed from them(area under the curve 0.69; 95% confidence interval 0.63–0.76)was equal to or higher than that of the prediction based on the presence of rapid delivery due to NRFS. The prediction ability of the prediction model was high even in validation dataset which consists of 649 neonates including 44 of NRD(area under the curve 0.66; 95% confidence interval 0.57-0.74).

     Conclusions: The predictive tool for NRD developed from factors below; late term, cesarean section, rapid delivery due to NRFS, and umbilical cord arterial blood pH, might be useful in neonatal care in general city hospitals.

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  • Shuji Ishida, Hidehiko Nakanishi, Ayano Yamaguchi, Mari Ooka, Manabu k ...
    2021Volume 57Issue 2 Pages 269-274
    Published: 2021
    Released on J-STAGE: September 06, 2021
    JOURNAL FREE ACCESS

     There are no previous reports whether a circulatory dysfunction similar to post ligation cardiac syndrome(PLCS), which causes after surgical treatment for patent ductus arteriosus(PDA)in premature newborns, occurs after medical treatment, too. The purpose of this study was to clarify whether circulatory dysfunction similar to PLCS developed after medical treatment. We enrolled 32 premature newborns less than 26 weeks of gestation who were needed surgical or medical treatment for hemodynamic significant PDA between January 2016 and January 2020. The median gestational age was 25 weeks and birth weight was 639 g. Eight newborns developed circulatory dysfunction(five newborns of indomethacin prophylaxis, one newborn of indomethacin treatment, two newborns of surgery)and vasoactive inotropic score increased from 6 points to 10 points after PDA closure. Systolic blood pressure(41→34mmHg vs 44→44mmHg, P<0.01)decreased after PDA closure. CTR on chest X-ray(50→43% vs 52→43%, P=0.88)and LVIDd on echocardiography(10.8→10mm vs 10.4→9.7mm, P=0.96)which are indicators of preload, were no change. On the other hand, LVEF(71→54% vs 75→72%, P<0.01)which is cardiac contractility, decreased. Circulatory dysfunction similar to PLCS was observed after medical treatment. Systolic blood pressure decreased in newborns with circulatory dysfunction, and the cause was decreased cardiac contractility.

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  • Mayuko Takeuchi, Noriko Yoneda, Sayaka Tsuda, Arihiro Shiozaki, Akitos ...
    2021Volume 57Issue 2 Pages 275-281
    Published: 2021
    Released on J-STAGE: September 06, 2021
    JOURNAL FREE ACCESS

     [Background]Pregnancies complicated with heart disease account for 2-3% of total pregnancies. As the number of adult congenital heart disease is increasing because of the progress of medical treatment, clarifying the precautions for the management of pregnancies complicated with heart disease is necessary. The purpose of this study is to clarify the points of caution regarding the management of pregnancies with heart diseases in accordance with case series in our hospital. [Methods]We investigated the maternal background and perinatal outcomes of pregnancies complicated with heart disease in our hospital over the past 10 years. [Results]Out of 31 pregnancies(24 cases) complicated with heart disease, 6 pregnancies had obstetric complications, which include 4 preterm labor cases with hospitalization. One delivery was terminated at the 30 weeks of gestation, because heart failure was elicited after the administration of magnesium sulfate and steroids. [Conclusion]Tocolytic agents and steroids can affect hemodynamics; therefore, strict management in cooperation with cardiologists is required for preterm labor in heart diseases complicated pregnancies.

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  • Nao Taguchi, Tomoko Araki, Ayaka Nakashima, Yuki Kataoka
    2021Volume 57Issue 2 Pages 282-287
    Published: 2021
    Released on J-STAGE: September 06, 2021
    JOURNAL FREE ACCESS

     We investigated the association between intimate partner violence(IPV)in pregnancy and obstetrical complications. This study compared 69 pregnant women who were admitted to a maternal fetal intensive care unit(MFICU)for obstetrical complications such as threatened premature labor, premature rupture of membrane, and fetal growth restriction, with 355 pregnant women who came for outpatient checkups from February to July 2019. The Violence Against Women Screen(VAWS)was used for screening, and the positive rate of IPV was 36.4% in the MFICU group and 23.4% in the outpatient group, showing a significant difference(p = 0.027). However, in the multiple logistic regression analysis adjusted for the number of births, General Anxiety Disorder, history of preterm birth, and smoking, IPV was not significant as an independent factor. As one-third of the MFICU group was found to have possible IPV, it is important to screen for potential IPV for the physical and mental health of pregnant women and their fetuses.

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  • Hiroyuki Yoshio, Kiyofumi Noda, Ippei Miyata, Junko Arimichi, Sumiyo I ...
    2021Volume 57Issue 2 Pages 288-294
    Published: 2021
    Released on J-STAGE: September 06, 2021
    JOURNAL FREE ACCESS

     In order to promote breastfeeding in Japan, we conducted an awareness survey of young students on breastfeeding by using the questionnaire sheet. The questionnaire was anonymously asked to the students, including nursing ones, of 2,971 of 14 departments of schools in 4 prefectures of Okayama, Hiroshima, Yamaguchi and Shimane, and finally tallied the results of 2,742 cases. Their ages surveyed were ranged from 14- to 24-years-old and their mean and median ages were 18.2- and 18-years-old, respectively. The numbers of males and females were 793(29%)and 1949(71%), respectively. The ratios of the answers to a questionnaire of which is better between breastfeeding and formula feeding for nourishing infant were breastfeeding; 59.5%, formula feeding; 1.4%, either available; 28%, and not sure; 11.1%, respectively. The preference of female for breastfeeding was stronger than that of male(64.7 vs 46.9%, p < 0.05), but the ratios of “either available” were both around 30%, which were almost the same as them. The wishes of females themselves for breastfeeding in future also were stronger than those of males(81.8 vs 61.8%, p < 0.05). Comparing 1, 138 of nursing students with 516 of non-nursing ones, whose ages were both adjusted between 18- and 24-years old, 70.3% of nursing students were in favour of breastfeeding for nourishing infant as opposed to 64.6% of non-nursing ones(p < 0.05). But this percentage of nursing students had not increased with age during school. According to this survey, many young students seem to think by intuition that breastfeeding is better than formula feeding for nourishing infants mainly because it is good for infant. However, some students have their anxieties about acting breastfeeding and working simultaneously, which should be improved. It is essential to enlighten evidence-based breastfeeding, and also important to train qualified health care providers for breastfeeding in Japan.

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  • Tomoya Tsuchihashi, Masakazu Miyawaki, Nobuyuki Kakimoto, Takeshi Kuma ...
    2021Volume 57Issue 2 Pages 295-300
    Published: 2021
    Released on J-STAGE: September 06, 2021
    JOURNAL FREE ACCESS

     In Japan, large quantities of iodone-rich food are commonly consumed. Excessive iodine Intake in newborn period is harmful effect to thyroid function. However, there are few reports about an amount of iodine intake of the low-birth-weight infants. The aim of this study was to measure an amount of iodine discharge, and presume an amount of iodine intake of the low-birth-weight infant. We enrolled 37 low-birth-weight infants which could use enteral nutrition sufficiency were measured. Iodine in the urine was measured at the time of thyroid function measurement, and the iodine intake was inferred from urine amount. The median of the urine iodine concentration was 300μg /L, the median of iodine intake was 43.8μg/day. 6 cases exceeded the upper limit of iodine intakes, and all of 3 cases over 1,000μg/day developed thyroid dysfunction. Breast milk is a valuable source of iodine for low-birth-weight infants, but the iodine content of breast milk varies from person to person, and in some cases, it is extremely high. It is important to understand the iodine intakes of low-birth-weight infants from the urinary iodine concentration.

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  • Keisuke Tsumura, Takeshi Ono, Takaaki Shinagawa, Kunio So, Yuko Oshima ...
    2021Volume 57Issue 2 Pages 301-308
    Published: 2021
    Released on J-STAGE: September 06, 2021
    JOURNAL FREE ACCESS

     Objective: This study aims to determine the frequency of detection of ampicillin(ABPC)-resistant bacteria, their species, and time from antibiotics administration to the emergence of the bacteria in the amniotic fluid in preterm premature rupture of membranes(PPROM).

     Methods: We analyzed 29 strains of 23 cases in which amniotic fluid culture was positive at the time of PPROM diagnosis and 11 strains of 8 cases in which amniotic fluid culture became positive after administration of ampicillin(ABPC)+ clarithromycin(CAM)or azithromycin(AZM). Genital mycoplasma was excluded from the study because it is already known to be ABPC-resistant.

     Results: Three Gram-negative bacilli, two fungi, and one MRSA strain were resistant to ABPC at the time of PPROM diagnosis. ABPC-resistant bacteria were detected in 6/25(24%)strains and in 10/11(91%)strains at the time of PPROM diagnosis and after antibiotics administration, respectively. ABPC-resistant bacteria appear as early as day 1 after the start of the administration of antibiotics. All seven strains of Gram-negative bacilli were ABPC-resistant before or after antibiotic administration. Gram-negative bacilli were found in the amniotic fluid of three of the five patients whose neonate developed early-onset neonatal sepsis.

     Conclusion: In PPROM, ABPC appears to be effective against almost all strains of bacteria except Gram-negative rods, fungus, and genital mycoplasma before antibiotic treatment. However, it is unlikely to be effective against new strains of bacteria even at an early period after antibiotic treatment.

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  • Tamina Kino, Yasuyo Maruyama, Sayako Nakagawa, Masafumi Yamamoto, Ayak ...
    2021Volume 57Issue 2 Pages 309-314
    Published: 2021
    Released on J-STAGE: September 06, 2021
    JOURNAL FREE ACCESS

     We conducted a retrospective study to ascertain the prevalence of rubella antibodies during pregnancy and the postpartum rubella vaccination rate. We targeted 3,322 pregnant women who delivered their infants at our hospital from January 2014 to December 2017. We analyzed the rubella antibody prevalence in pregnant women and the rubella vaccination rate during perpetual hospitalization by comparing the number of deliveries, the presence of infertility treatments, and the age at delivery. We determined a rubella antibody titer of HI ≤ 16 times as low antibody titer. The proportion of pregnant women with low antibody titers was 31.5%, of which the rubella vaccination coverage was 43.6%. The antibody-bearing was high in over 35-year-old pregnant and increased with their age, but the vaccination rate was high in Primiparas. Furthermore, there were certain pregnant women with low antibody titers after infertility treatments. The rubella vaccination rate among pregnant women to prevent rubella infection and congenital rubella syndrome is still low. Therefore, to improve vaccination, an integrated policy from government-private collaborations is needed, and it is also important to raise awareness of vaccination among health care workers.

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  • Chika HOMMA, Yoko NISHIMURA, Natsumi FURUYA, Akiko KURASAKI, Haruhiro ...
    2021Volume 57Issue 2 Pages 315-320
    Published: 2021
    Released on J-STAGE: September 06, 2021
    JOURNAL FREE ACCESS

     Purpose: To evaluate the difference of vulnerability in the preterm period in cases of mothers suffered hemorrhagic shock compared to them in the term period.

     Method: Case-control study was attempted. Among the women delivered singleton infant by cesarean section in our perinatal center, cases had massive bleeding more than 2,000 mL during operation between 2011 and 2020 were retrospectively enrolled. The subjects were divided into cases(delivered before 35 weeks of gestation)and controls(delivered after 35 weeks of gestation). Indication of cesarean section, vital signs, amount of blood transfusion were reviewed from medical records. Lowest blood pressure, maximum heart rate, maximum shock index(SI), the minimum hemoglobin concentration, and amount of blood transfusion were compared between two groups.

     Result: Out of 6,194 deliveries during study period, number of the cesarean operation was 2,361. Among them, the subjects were 64 cases(1%). 12 cases(19%)and 52 controls were compared. Distribution of variables in cases and controls(median, range)were 2,466(2,010-4,839 mL vs 2,360(2,013-4,320)mL(ns)in amount of bleeding, 1.1(0.7-1.8)vs 1.0(0.7-2) (ns)in maximum shock index, 100(70-145)bpm vs 100(70-140)bpm in maximum heart rate, 90(70-110) mmHg vs 90(60-120) mmHg in systolic blood pressure, 12.5(0-90) min. vs5(0-75) min.(ns)in duration between placental separation and SI>1, 1,093(210-1,900)mL vs 957(320-3,150)mL(ns) in amount of bleeding until SI>1, and 17.5(0-90)min. vs 7.5(0-75)min(p=0.010)in duration between SI>1 and recovery to SI<1, respectively.

     Conclusion: No differences between cases and controls were found in amount of bleeding, vital signs, and duration until SI>1. However, after mothers suffered shock vitals, time to recover was significantly longer in cases delivered until 35 weeks of gestation compared to cases did after 35 weeks of gestation.

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  • Yurika Hoshi, Manabu Sugie, Masatoshi Imamura, Junichi Shimizu, Tsutom ...
    2021Volume 57Issue 2 Pages 321-325
    Published: 2021
    Released on J-STAGE: September 06, 2021
    JOURNAL FREE ACCESS

     Chronic lung disease(CLD)is an important prognostic factor that affects long-term outcomes in preterm infants. Although there exist different definitions of CLD, widely accepted diagnostic criteria is based on the dependence on non-invasive respiratory support(NRS)including supplemental oxygen at 28days after birth or at 36 weeks of post-conceptional age(PCA).

     However, some infants diagnosed as CLD show improved lung function and become free from any respiratory support before the term gestation.

     This is explained by the fact that the current definition comprises a wide range of respiratory disorder such as functional prematurity of respiratory control system and/or anatomical prematurity of the lung that should resolve with age.

     In this study, we aimed to define the right timing for diagnosing CLD by examining the timing of weaning from NRS in infants below 32 weeks of PMA who entered our NICU.

     Results showed negative correlation between the gestational age at birth and the duration of NRS. That is, the lower the gestational age was, the longer the duration of NRS was. However, more than half infants who required NRS became free from NRS by 42 weeks of PCA.

     In conclusion, the current diagnosis of CLD may include the respiratory disorder other than CLD.

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  • Yasunori Toyoshi, Fuyo Kameda
    2021Volume 57Issue 2 Pages 326-333
    Published: 2021
    Released on J-STAGE: September 06, 2021
    JOURNAL FREE ACCESS

     Background: There are few reported cases of NCPR training sessions being conducted for nursing students.

     Objective: To report on the practical experience of the NCPR primary course conducted for nursing students and to discuss the future of life-saving education in basic nursing education based on a questionnaire.

     Methods: Third- and fourth-year nursing students of a nursing school were invited to attend the workshop, and questionnaires were collected before and after the workshop.

     Results: Data on motivations for taking the primary NCPR course, the timing of the course, and the level of understanding of the lecture content were obtained through questionnaires.

     Discussion: It is thought that the inclusion of NCPR and other training sessions in basic nursing education as a part of lifesaving education is effective in motivating students to learn and fostering their desire to improve their learning.

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  • Bunmei Yoneda, Masashi Zuiki, Hiroshi Komatsu
    2021Volume 57Issue 2 Pages 334-338
    Published: 2021
    Released on J-STAGE: September 06, 2021
    JOURNAL FREE ACCESS

     The pulse wave transit time(PWTT), calculated with electrocardiograph and saturation of percutaneous oxygen monitor, is known to be useful for non-invasive blood pressure(BP)monitoring. However, little is known about the efficacy of PWTT in newborns. The purpose of this study was to investigate the relationship between PWTT and BP in neonates. The infants, in which invasive BP and PWTT were measured stably, were enrolled 〔n:17, median gestational age:35(29 - 39)weeks, median birth weight: 2,776(1,288 – 3,284)g〕. With calculating the change PWTT and BP every 5 minutes(⊿PWTT and ⊿BP), ⊿PWTT were negatively correlated with change in ⊿BP. Furthermore, receiver operating characteristic curve analysis revealed that ⊿PWTT cut-off values for predicting systolic-BP fluctuation were -7ms at systolic-BP: +10mmHg[area under the curve(AUC)= 0.939, 95% CI=0.873-1.0, sensitivity =94%, specificity =91%], and +9ms at systolic-BP: -10mmHg(AUC = 0.862, 95% CI=0.647-1.0, sensitivity =97%, specificity = 75%). This study suggested that PWTT has the ability to detect non-invasively and continuously the change of blood pressure in neonates.

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  • Fumio Asanao, Shinji Tanigaki, Aya Kitamura, Miho Matsushima, Yoichi K ...
    2021Volume 57Issue 2 Pages 339-342
    Published: 2021
    Released on J-STAGE: September 06, 2021
    JOURNAL FREE ACCESS

     Uterine fibroids are often found in pregnant women with the estimated prevalence of 1.4-3.9%. The most common complaint of uterine fibroids during pregnancy is acute abdominal pain. Acute pain due to uterine fibroids degenerating is severe to require hospitalization for pain relief. In the current study, we aimed to elucidate the risk factors for fibroid-related abdominal pain during pregnancy. We conducted a retrospective cohort study on 109 pregnant women with uterine fibroids delivered at Kyorin University Hospital between April 2015 and June 2019. We reviewed the maternal backgrounds(age, body mass index, parity, history of myomectomy, gestational age, preterm birth rate, birth weight), the fibroids characteristics(size, number, type) and the treatment(medication, hospitalization or not). Among group 1(no symptoms, N=70), group 2(oral medication only, N=24)and group 3(hospitalization, N=15), there was significant difference in the frequency size and type of uterine fibroids. Increasing size of uterine fibroids and subserosal uterine fibroids were associated with higher rates of admissions for fibroid-related pain. In addition, we detected subserosal uterine fibroids were an independent risk factor of fibroid-related pain during pregnancy by the multivariate analysis.

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Case reports
  • Serina Yamada, Kentaro Tamura, Mitsuhide Nagaoka, Satomi Inomata, Yuka ...
    2021Volume 57Issue 2 Pages 343-347
    Published: 2021
    Released on J-STAGE: September 06, 2021
    JOURNAL FREE ACCESS

     Congenital cutaneous candidiasis(CCC)is an unusual disorder caused by an in utero acquired infection of Candida species. Extremely preterm infants are at risk for bloodstream dissemination and death. However, there is no clear indication for treatment since late preterm and term infants may follow a benign course. A female baby born by vaginal delivery at 34 weeks and six days, with a birth weight of 2,358 g, was admitted with dyspnea. At birth, erythema and papules appeared all over her body, but slowly it faded away. Respiratory status gradually worsened from the age of three days, and chest X-ray revealed infiltration in the right upper lung field. Since Candida albicans was detected in the gastric juice and vernix caseosa at birth, we suspected a fungal infection. Candida albicans was also found in the maternal cervical mucus. The umbilical cord had yellow-white plaques. A diagnosis of CCC was made after microscopic findings of spores and pseudohyphae in skin scrapings. Cultures of blood and urine came back negative. We also suspected Candida pneumonia upon finding direct pulmonary spread of the fungus. Systemic antifungal treatment relieved respiratory distress. Therefore, it is important to test for fungal infection in cases where skin lesions are present at birth, and to consider treatment, even in late preterm or term infants, if CCC is diagnosed.

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  • Junko Nakanishi, Seiichi Tomotaki, Koji Motokura, Ryosuke Araki, Yutar ...
    2021Volume 57Issue 2 Pages 348-352
    Published: 2021
    Released on J-STAGE: September 06, 2021
    JOURNAL FREE ACCESS

     Neonatal pneumopericardium(PPC)is a very rare neonatal air-leak syndrome. PPC with cardiac tamponade has a high mortality rate. We report the first case of an extremely low-birth-weight infant with PPC and cardiac tamponade, saved by emergent pericardial drainage. The patient was born at 24 weeks and 6 days of gestational age, at 667 g. She developed left pneumothorax and PPC immediately after birth, and underwent left thoracic drainage. Because her PPC gradually improved spontaneously, we observed without invasive treatments. However, on Day 2, her PPC rapidly worsened, progressed to cardiac tamponade and then to sudden cardiac arrest. We immediately began cardiopulmonary resuscitation. However, she did not recover by chest compression and adrenaline administration. After emergent pericardial drainage, she recovered immediately. Physicians must consider that PPC can rapidly worsen and progress to cardiac tamponade.

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  • Chihiro Watanabe, Yasuko Togawa, Mari Sugimoto, Takao Togawa, Takahiro ...
    2021Volume 57Issue 2 Pages 353-359
    Published: 2021
    Released on J-STAGE: September 06, 2021
    JOURNAL FREE ACCESS

     Here, we report a case of a male infant born at 35 weeks and 5 days of gestation as the first of monochorionic diamniotic twins. There was no abnormal finding such as heart failure or amniotic fluid volume except weight difference between twins during the fetal period. Circulatory disorders were evident from the early postnatal period, so he was transported to our institute with suspected twin-to-twin transfusion syndrome. Echocardiogram revealed persistent right chamber volume overload and diastolic regurgitation in the descending aorta, even after some improvement of circulatory insufficiency due to heart failure and pulmonary hypertension. Subsequent ultrasonography revealed abnormal blood flow in the left common carotid artery and other arteries at the left skull base. Finally, dural arteriovenous fistula was diagnosed by head computed tomography and magnetic resonance imaging. Surgical ligation of the left external carotid artery, the main collateral vessel, was performed under cerebral angiography at 15 days of age. Additional catheter embolization was performed at 4 months of age, and the collateral vessel was completely blocked. No recurrence or neurological deficits were observed at 8 years of age. His development was comparable to that of the healthy twin. These results suggest that careful physical and imaging examinations are useful for diagnosis and treatment decisions regarding high-flow arteriovenous shunts in cases of prolonged heart failure.

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  • Yui Takahashi, Kyohei Miyazaki, Kenichi Sato, Nozomi Kashiwabara, Mina ...
    2021Volume 57Issue 2 Pages 360-365
    Published: 2021
    Released on J-STAGE: September 06, 2021
    JOURNAL FREE ACCESS

     Neurocutaneous melanosis(NCM)is a rare congenital disease caused by proliferation of melanocytes in the skin and central nervous system. The prognosis is poor if neurological symptoms develop. In this paper, we report a four-day-old boy who had a giant pigmented nevus at birth. High-intensity lesions on magnetic resonance imaging(MRI)T1-weighted images of brain tissue including the amygdala suggested melanocytic proliferation in the central nervous system and we diagnosed this case as NCM from a remarkably elevated level of serum 5-S-cycteinyldopa(5-S-CD). Although pathological findings in the central nervus system or neurological symptoms are necessary for the definitive diagnosis of NCM, recent reports indicated that NCM is often diagnosed by the combination of giant pigmented nevi and brain MRI findings. In addition to the giant pigmented nevi and MRI finding including T1-high intensity lesions or leptomeningeal gadolinium-enhancement, melanocytes in cerebrospinal fluid or remarkably high levels of 5-S-CD in cerebrospinal fluid or serum suggestive of melanocyte proliferation in the central nervus system would improve the accuracy of the diagnosis. Early diagnosis of NCM during the neonatal period makes it possible to support the family psychologically and provide early intervention for central nervous system disorders.

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  • Naoki Otsuka, Toshiya Teshigawara, Keisuke Ichida, Yukie Ezaka, Toshim ...
    2021Volume 57Issue 2 Pages 366-370
    Published: 2021
    Released on J-STAGE: September 06, 2021
    JOURNAL FREE ACCESS

     Fulminant group A streptococcal infection is sepsis that develops suddenly and rapidly progresses to multiple organ dysfunction, including shock and necrotizing soft tissue inflammation. Group A streptococcal infection during pregnancy causes maternal death. It has been reported many cases of maternal death in Japan. In this case, mother was in shock and obstetric DIC at the time of examination. Delivered baby has died and showed remarkable exfoliation findings throughout the body. Group A streptococci causes tonsillitis, pharyngitis, soft tissue inflammation, and myositis, and it is known that the production of exotoxin causes a rash throughout the body and causes epidermal exfoliation. Because the presence of the infectious disease could be suspected from the epidermal exfoliation of the baby, injection of ampicillin and clindamycin was started from an early stage. Although there are few cases of maternal lifesaving cases, it was achieved by early administration of antibiotics in addition to multidisciplinary treatment. We report a case of fulminant group A streptococcal infection that succeeded in saving the maternal life, with a review of the literature.

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  • Natsuho Adachi, Chiharu Irisa, Kohei Kashima, Satsuki Kakiuchi, Riki N ...
    2021Volume 57Issue 2 Pages 371-375
    Published: 2021
    Released on J-STAGE: September 06, 2021
    JOURNAL FREE ACCESS

     A 2-month-old female with Beckwith-Wiedemann syndrome, delivered at a gestational age of 25 weeks and birth weight 1, 083g, developed multiple liver masses. It was difficult to differentiate hepatoblastoma from hepatic hemangioma because she was too immature to perform a liver biopsy. We used, therefore, propranolol hydrochloride as a therapeutic and diagnostic treatment although it is not recommended to use for preterm infants in general. The patient showed a decrease tumor size significantly, which led to diagnosis as infantile hepatic hemangiomas.

     We suggest that propranolol hydrochloride may be useful to manage preterm infants with liver tumors suspected as infantile hepatic hemangioma.

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  • Masayuki Hane, Tomoaki Yokokura, Kazuki Miyabayashi, Mayumi Ifuku, Nat ...
    2021Volume 57Issue 2 Pages 376-380
    Published: 2021
    Released on J-STAGE: September 06, 2021
    JOURNAL FREE ACCESS

     The expansion of the treatment indications of everolimus for neonatal tuberous sclerosis complex(TSC)has been approved, with its effectiveness toward the cardiac rhabdomyoma being continuously reported. However, many of the issues related to its usage have not been fully resolved. We encountered a case of TSC with cardiac rhabdomyoma in a 2,488 g low-birth-weight preterm infant born through emergency cesarean section at 35 weeks and 5 days of gestation owing to a cardiac tumor discovered during the fetal period. This tumor located in the interventricular septum and the tumor was enlarged postnatally. The administration of everolimus was commenced, considering the risk of right ventricular outflow tract stenosis. Shrinkage was confirmed at 50 days old with no adverse reaction. In December 2019, expert opinion consensus for everolimus treatment was given for emergencies caused by neonatal cardiac rhabdomyoma in TSC. However, there are only few reports of therapeutic intervention for preterm infants. Based on the course of this case, we consider the appropriate treatment for the cardiac rhabdomyoma in the preterm infant and report it here.

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  • Keisuke Ichida, Toshimitsu Furui, Yuri Nakao, Naoki Otsuka, Yukie Ezak ...
    2021Volume 57Issue 2 Pages 381-384
    Published: 2021
    Released on J-STAGE: September 06, 2021
    JOURNAL FREE ACCESS

     The incidence of intracranial acute subdural hematoma after spinal anesthesia is very rare(1/500,000-1/1,000,000), but it is a complication that can cause severe neurological symptoms and sequelae. The mechanism of occurrence of intracranial acute subdural hematoma is thought to be caused by the leakage of spinal fluid from puncture as well as post-dural puncture headaches, and the similarities of symptoms often make it difficult to distinguish between them. In this study, we show a case in which the patient who had a headache on the second day after surgery after selective caesarean section with spinal anesthesia for twin pregnancy and we were diagnosed with intracranial acute subdural hematoma by a simple CT examination. It is a rare but serious complication after spinal anesthesia. So it is necessary to treat the patient with intracranial acute subdural hematoma in mind.

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  • Kaoru Kitamura, Masashi Sagara, Kei Wakabayashi, Sakiko Komori, Hirono ...
    2021Volume 57Issue 2 Pages 385-389
    Published: 2021
    Released on J-STAGE: September 06, 2021
    JOURNAL FREE ACCESS

     Spondylocostal dysostosis(SCDO)is a rare disorder characterized by multiple vertebral malformations and rib deformities. We present two infants with SCDO and spinal abnormalities. One had defects and deformities of vertebrae and ribs, low-lying conus, spinal lipoma, and cleft foot. The other had defects and deformations of vertebrae and ribs, low-lying conus, lipomyelomeningocele, and unilateral renal aplasia. Both cases showed minor external findings, such as a deviated gluteal cleft or shallow sacral dimple in the lower back, but had spinal cord abnormalities that were found by ultrasound examination in the neonatal period. Although spinal cord abnormality in SCDO is reported to be rare, screening by ultrasound in the neonatal period is useful for diagnosis and management.

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  • Chihiro Hirama, Yoko Nishimura, Yuriko Iwahata, Natsumi Furuya, Chika ...
    2021Volume 57Issue 2 Pages 390-394
    Published: 2021
    Released on J-STAGE: September 06, 2021
    JOURNAL FREE ACCESS

     【Case】

     A 37-year-old multiparous pregnant woman was taken regular pregnancy checkups at maternity clinic. Pregnancy course was uneventful, but ritodrine chloride tablet was administered after 30 weeks of gestation due to small uterine contraction. She was followed up for her thirst, malaise, polyuria, nocturia and polydipsia at this clinic form 32 weeks of gestation. However, she was transferred to our perinatal center at 35 weeks of gestation due to develop hepatic and renal dysfunction. Physical findings were as follows: clear consciousness, afebrile, pulse 98/min, blood pressure 123/97mmHg, soft abdomen, no tenderness, estimated fetal body weight 2,380g(-0.4SD), amniotic fluid pocket 2.1cm. Laboratory data were as follows: Platelets 160,000/μL, AST 347U/L, ALT 397U/L, ALP 654U/L, LDH 595U/L, γGTP 34U/L, Creatinine 1.03mg/dL, UA 9.0mg/dL, plasma osmolality 301 mOsm/kg, urine osmolality 126 mOsm/kg. Abdominal ultrasound did not reveal fatty liver. Head MRI was taken due to suspicious of diabetes insipidus and high signal loss in the posterior pituitary was observed in T1-weighted image. Therefore, diagnosis of transient diabetes insipidus was made and desmopressin was administered. Despite desmopressin administration, amniotic fluid decreased to none and hepatic dysfunction worsened. We decided termination of pregnancy. After delivery, symptoms, and hepatic and renal dysfunction was improved and head MRI revealed improved high signal in posterior pituitary. She no longer needed desmopressin and discharged 6th day after delivery.

     【Conclusion】

     The mechanism of the onset of transient diabetes insipidus is considered that the metabolism of vasopressinase (ADHase)in the liver is lowered due to some preceding hepatic dysfunction, the blood concentration of ADHase is increased, and vasopressin is decomposed, or that ADHase is increased by increased placental weight due to multiple pregnancies. In this case, there was little improvement with desmopressin and worsening of liver dysfunction due to dehydration, so we chose to terminate the pregnancy to reduce maternal distress.

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  • Masahiro Kawasaki, Masakatsu Sase, Keiko Hasegawa, Yoshiko Nawata, Tsu ...
    2021Volume 57Issue 2 Pages 395-398
    Published: 2021
    Released on J-STAGE: September 06, 2021
    JOURNAL FREE ACCESS

     We report a case of a male baby with Megacystis microcolon intestinal hypoperistalsis syndrome(MMIHS)that was not diagnosed prenatally despite presenting with a megacystis.

     A male fetus was referred to our hospital at 34 weeks of gestation because routine fetal ultrasound scan revealed a cyst in his abdominal cavity at 30 weeks of gestation, which was subsequently diagnosed as a megacystis with bilateral hydronephrosis at 32 weeks of gestation. Although lower urethral obstruction(LUTO)was suspected, the amniotic fluid volume was normal and MRI examination showed no abnormality in his urethra, though mild gastric dilatation was observed. He was born at 38 weeks and 6 days of gestation with birth weight 3,032 g. A large amount of bilious gastric juice was aspirated at birth. He was diagnosed with MMIHS, as microcolon was seen on enema imaging and normal ganglion cells were confirmed by rectal biopsy. At 11 months of age he has been hospitalized for chronic intestinal and urinary dysfunction, and managed with a gastrostomy, a cyststomy, and parenteral nutrition.

     MMIHS should also be considered as one of the differential diagnosis of suspected LUTO although it is an extremely rare disease. Since polyhydramnios and gastrointestinal dilatation of MMIHS may appear gradually during late gestation, it is useful to examine the gastrointestinal tract in detail with MRI in addition to ultrasonography.

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  • Saki Kurotaki, Asami Ito, Juria Maeda, Minako Yokoyama, Daisuke Matsuk ...
    2021Volume 57Issue 2 Pages 399-403
    Published: 2021
    Released on J-STAGE: September 06, 2021
    JOURNAL FREE ACCESS

     The patient was a 32-year-old woman with gravida 4 para 2 and had a medical history of childhood asthma. After spontaneous pregnancy, she was managed for pregnancy and underwent preventive cervical cerclage at 15 weeks of gestation at her prior hospital. She was aware of shortness of breath, respiratory distress, and edema, and she consulted her previous internal medicine facility at 19 weeks of gestation. The results of the examination showed right heart failure, pulmonary hypertension, and thrombocytopenia. Contrast-enhanced computed tomography showed no pulmonary thromboembolism, and the patient was transferred to our hospital for examination and treatment. The echocardiography showed marked right heart load, and her platelet number decreased to 29,000/μL, suggesting thrombotic microangiopathy. She was scheduled to be treated after a platelet transfusion on the next day, but her condition suddenly changed and she died. A pathological autopsy was performed, and confirmed a diagnosis of idiopathic pulmonary arterial hypertension(IPAH). IPAH is more prevalent in young women, with a high mortality rate of 30-50% in cases of perinatal complications, and pregnancy is generally contraindicated. Secondary pulmonary hypertension was ruled out in this case because there was no history of collagen disease, cardiac disease, or pulmonary or liver disease. If a patient complains of shortness of breath or respiratory distress during pregnancy, we must keep IPAH in mind and provide prompt examination and treatment.

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  • Shotaro Matsudera, Shun Watanabe, Yukiko Tani, Takeshi Yamaguchi, Kei ...
    2021Volume 57Issue 2 Pages 404-408
    Published: 2021
    Released on J-STAGE: September 06, 2021
    JOURNAL FREE ACCESS

     The patient was a girl, at 38 week’s gestation, born in the breech presentation with a prolapsed umbilical cord. Immediately after birth, cardiopulmonary resuscitation was performed. On admission to our NICU, because severe metabolic acidosis and convulsions were observed, She was diagnosed with hypoxic encephalopathy. Therefore, hypothermia therapy was initiated. At day 1, hypotension and anemia were observed, and abdominal ultrasonography revealed a hematoma of approximately 3 cm in the right lobe of the liver, the patient was then referred to our department. Her vitals were not stable, and the hypothermia therapy was discontinued on day 2, judging that it would be difficult to continue with conservative treatment, emergency surgery was performed. When the abdomen was opened, a ruptured area was found in the posterior region of the right lobe of the liver, which was judged to be the source of the bleeding. An absorbable hemostatic agent was applied to the injured area, and the bleeding was controlled by manual compression hemostasis. Oral intake was started on the 11th postoperative day, and the patient was discharged on the 36th day. Therefore, we believe it is important to perform surgical treatment of liver injury in neonates while working in close collaboration with other departments such as pediatrics and radiology for optimal treatment.

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