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Yumi Keida, Misaki Nakashima, Hiroki Yonemoto, Mutsumi Akaishi, Koichi ...
2025Volume 61Issue 2 Pages
244-248
Published: 2025
Released on J-STAGE: November 10, 2025
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Monochorionic monoamniotic twins(MCMA twins)occur in approximately 1-2% of monozygotic twins. Premature birth, low birth weight, congenital anomalies, twin-to-twin transfusion syndrome, and umbilical cord entanglement are complications that can appear in MCMA twins. MCMA twins have the highest perinatal mortality and morbidity. Although the perinatal mortality rate has been reported to be declined in recent years, there are few reports on the clinical characteristics and prognosis of the infants. To investigate the clinical characteristics and prognosis of MCMA twins, we conducted a retrospective chart review of 12 MCMA twins from six pregnancies managed in our hospital. There were four cases(33.3%)of intrauterine fetal death, including three cases occurred before 22 weeks gestation. The median gestational age of the eight live births was 32 weeks and 3 days(29 weeks and 4 days - 35 weeks and 6 days), and the median birth weight was 1,686g(973g-2,368g). Congenital heart disease and intracranial lesions were highly prevalent, and 50% had neurological sequelae, suggesting that long-term follow-up is necessary for all MCMA twins.
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Shinji Hagimoto, Sota Iwatani, Takao Kobayashi, Toshihiko Ikuta, Sachi ...
2025Volume 61Issue 2 Pages
249-255
Published: 2025
Released on J-STAGE: November 10, 2025
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Elevated direct bilirubin(DB)levels, known as cholestasis, are relatively common in very low birth weight (VLBW)infants. To investigate the prevalence and underlying factors, we conducted a gestational age(GA)-matched case-control study at our institution. A total of 161 VLBW infants without congenital disorders born between 2020 and 2023 were included in the analysis. Among them, 9 infants(5.6%, case group)exhibited DB levels of ≥2 mg/dL on multiple occasions between postnatal day 14 and corrected 36 weeks of GA. For comparison, 27 infants with consistently lower DB levels of < 2 mg/dL during the same period and born at the same GA were selected as the control group. Perinatal factors, neonatal factors, neonatal complications, and nutritional management were analyzed and compared between the two groups. The case group had a significantly higher frequency of male sex(89% vs. 44%), abdominal surgery(56% vs. 4%), and fasting for 2 or more days during the week prior to exceeding DB levels of 2 mg/dL(44% vs. 11%)compared to the control group. In conclusion, prolonged direct hyperbilirubinemia was observed in 5.6% of VLBW infants without congenital disorders. This GA-matched case-control study identified male sex, abdominal surgery, and prolonged fasting as potential contributing factors to its development.
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Midori Yokoi, Tsutomu Kondo, Chihiro Takase, Chika Park, Kana Miyahara ...
2025Volume 61Issue 2 Pages
256-261
Published: 2025
Released on J-STAGE: November 10, 2025
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Background: Infants at twenty-two weeks of gestation are considered the lower limit of extrauterine viability, and the mortality rate is higher compared to infants at 23 weeks of gestation.
Methods: We retrospectively investigated the complications and mortality rates in 8 infants at 22 weeks of gestation(22-week group)and 16 infants at 23 weeks(23-week group)born between 2014 and 2021 using medical records.
Results: Five cases(63%)in the 22-week group died during NICU hospitalization, compared to two cases(13%) in the 23-week group. The rates of prenatal factors such as maternal antenatal steroids, and treatments including indomethacin prophylaxis, hydrocortisone, COX inhibitors or ligation for patent ductus arteriosus were similar between the two groups.
Discussion: The overall mortality rate was significantly higher in the 22-week infants than in the 23-week infants even though their treatments were similar. This suggests that immaturity of various organs at the 22-week infants including adrenal function, cardiac function and coagulation function had a significant impact on the outcomes.
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Munetoshi Akazawa, Kazunori Hashimoto
2025Volume 61Issue 2 Pages
262-268
Published: 2025
Released on J-STAGE: November 10, 2025
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Objectives: The prognosis for newborns affected by syphilis is often inadequate due to delays in diagnosis and treatment in the cases of pregnant women with inadequate perinatal care. The number of pregnancies complicated by syphilis, as well as cases of congenital syphilis, has been rapidly increasing in Japan. Therefore, a comprehensive examinations of the clinical characteristics and perinatal prognosis of pregnant women with inadequate perinatal care complicated syphilis infection is essential.
Methods: We conducted a literature review of domestic reports published in the past 10 years and identified 30 cases of syphilis-associated pregnancies with inadequate prenatal care, including 21 cases of congenital syphilis. Clinical characteristics were analyzed.
Results: The mean maternal age was 23.4 years, with 60% of patients engaged in the sex industry. In 53% of cases, delivery occurred within a few days of the initial visit, and the cesarean section rate was 40%. The mean birth weight was 1,977 g, and the preterm birth rate was 64%. A comparison between cases with and without congenital syphilis revealed no significant differences in maternal background.
Conclusion: Pregnant women with inadequate perinatal care and syphilis were predominantly young individuals engaged in the sex industry. The perinatal prognosis was inadequate, with two-thirds of cases involving preterm birth and low birth weight.
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Kohei Kawada, Hidehiko Nakanishi, Rika Sekiya, Yukako Kosaka, Ayano Ya ...
2025Volume 61Issue 2 Pages
269-275
Published: 2025
Released on J-STAGE: November 10, 2025
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Intravitreal injection of anti-VEGF antibody ranibizumab(IVR)was introduced at our institution in 2019 for the treatment of retinopathy of prematurity(ROP). Compared to conventional retinal photocoagulation(PC), IVR has a higher recurrence rate and requires longer follow-up. This study retrospectively analyzed 63 ROP cases admitted to our NICU between January 2018 and March 2022 to assess the impact of IVR introduction on NICU management. Initial treatments included 18 cases(29%)in the PC group and 15 cases(24%)in the IVR group, which was further divided into the IVR-alone group(12 cases)and the IVR + PC group(3 cases). The IVR-alone group had a significantly longer hospital stay than the PC group, whereas the IVR + PC group had a hospital stay comparable to the PC group. While IVR alone requires prolonged hospitalization for retinal vascularization monitoring, the addition of PC at an appropriate time may facilitate earlier discharge. IVR + PC may be a viable treatment option, but further studies are needed to assess long-term outcomes.
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Takeshi Kumagai, Sawako Minami, Takahiro Okutani, Mitsuhiko Riko, Ryuz ...
2025Volume 61Issue 2 Pages
276-282
Published: 2025
Released on J-STAGE: November 10, 2025
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Objective and methods: For improvement of perinatal prognosis, we reviewed perinatal medical survey conducted for 20 years and examined using ICD-PM.
Results: Perinatal mortality decreased from 4.44 to 2.33, especially in the proportion of early neonatal deaths. In the ICD-PM classification, A3: antepartum hypoxia, I3: acute intrapartum event, N1: congenital malformations, and M1: complications of placenta/ cord that was the most frequent cause in each group. The mortality rate of fetal hypoxia with maternal complications of placenta/cord did not improve, and the perinatal mortality rate with maternal gestational hypertension, pre-eclampsia deteriorated.
Conclusions: Fetal management with complications of placenta/ cord, management of maternal medical conditions, and detailed research on the perinatal death cases were considered to be challenges.
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Haruhiro Kuwabara, Masato Tanaka, Jun Nirei, Keisuke Shoji, Akira Koba ...
2025Volume 61Issue 2 Pages
283-289
Published: 2025
Released on J-STAGE: November 10, 2025
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Physical growth during NICU admission in preterm infants has been associated with poor long-term developmental prognosis. Therefore, we investigated the relationship between physical growth during NICU admission and development at 3 years of age. We studied 48 extremely low-birth-weight infants admitted to our NICU, and compared the presence or absence of extrauterine growth restriction(EUGR)with the weight, height, head circumference, and body size at birth SDS. We examined the relationship between the average daily increase in body size and Bayley Scales of Infant Development 3rd Edition(BSID-III)cognitive, language, and motor development scores at the age of 3 years. Motor development scores for weight and head circumference were significantly lower in the presence of EUGR. Additionally, average weight gain during NICU admission and cognitive and motor development scores were positively correlated. Aggressive nutritional management of extremely low-birth-weight infants after birth for good physical growth during NICU admission may lead to good neurological development.
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Eri Tsugeno, Hiroyuki Tsuda, Aki Minoda, Yumi Nakamura, Miho Suzuki, Y ...
2025Volume 61Issue 2 Pages
290-295
Published: 2025
Released on J-STAGE: November 10, 2025
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The purpose of this study is to identify the factors involved in the success of twin vaginal delivery. This study was based on a retrospective review of medical records for twin pregnancies delivered at our hospital from September 2018 to August 2023. The criteria used at our hospital to permit vaginal delivery for twin pregnancies include the following conditions: first twin must be in a cephalic presentation, estimated fetal body weight of both twins is more than 1,800 grams, diamniotic twin, there must be no contraindications for vaginal delivery, and the patient must wish to attempt vaginal delivery.
Among the twin pregnancies that underwent a trial vaginal delivery, those that resulted in vaginal delivery of both children were considered the successful group. To identify the success factors for vaginal delivery in twin pregnancies, we performed statistical analyses on eight variables: maternal age, pre-pregnancy BMI, parity, assisted reproductive technology(ART), chorionicity, induced labor, the fetal presentation of the second twin, and the birth weight discordance between the twins. Of the 338 total twin pregnancies, 92 attempted vaginal delivery, 80(87.0%)in the successful group and 12(13.0%)in the unsuccessful group. The successful group had significantly more multiparous patients and non-ART pregnancies. To increase the rate of vaginal deliveries, multiparous patients and non-ART pregnancies should be aggressively considered for twin vaginal deliveries.
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Takehiko Takeda, Yasuyuki Kishigami, Sho Tano, Mikiya Kato, Rina Shiba ...
2025Volume 61Issue 2 Pages
296-303
Published: 2025
Released on J-STAGE: November 10, 2025
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Background: The COVID-19 pandemic has presented significant challenges in perinatal care, particularly in managing pregnant women who are SARS-CoV-2-positive while ensuring the functionality of regional perinatal medical centers. This study aimed to assess the obstetric management and perinatal outcomes for pregnant women with SARS-CoV-2 infection or close contact with infected persons during the COVID-19 pandemic.
Methods: We conducted a retrospective analysis of 132 pregnant women treated at our regional perinatal medical center from July 2020 to January 2023. Their delivery outcomes were compared with those of all deliveries during the same timeframe and with data from 2019, before the pandemic. Among these women, 34(25.8%) delivered in isolation, with 31(91.2%)of them undergoing emergency transfers from primary or secondary care facilities to our perinatal medical center.
Results: No cesarean sections were performed solely due to SARS-CoV-2 infection; all cesarean deliveries were based on obstetric indications. The cesarean section rate among SARS-CoV-2-positive women or those with close contact with infected persons was 17.6%, which was lower than the overall institutional rate of 27.5% during the same period. The neonatal intensive care unit admission rate was 23.5%, with no reported cases of neonatal SARS-CoV-2 infection or in-hospital transmission.
Conclusion: Our findings indicate that safe obstetric management during the COVID-19 pandemic is possible, even in nondesignated infectious disease hospitals, while achieving a lower cesarean section rate when appropriate infection control measures are implemented. These results offer valuable insights for enhancing pandemic preparedness in perinatal care.
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Yuya Yamada, Hajime Yasuhara, Shioka Watanabe, Yoshinori Takeda, Ayako ...
2025Volume 61Issue 2 Pages
304-310
Published: 2025
Released on J-STAGE: November 10, 2025
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Several drug package inserts have recommendations regarding lactation cessation; however, their descriptions differ from those in the technical literature. Under these circumstances, the package inserts were revised between April 2019 and March 2024. In this study, we investigated the changes in recommendations for breastfeeding mothers before and after the revision of package inserts for 414 oral medications used in our hospital. Additionally, the differences in the recommendations between the revised package inserts and those in the technical literature were examined. The results showed that for approximately two-thirds of the drugs, the references to cessation of lactation were changed while considering the therapeutic benefits of breastfeeding. However, some recommendations in the revised package inserts did not correspond to those in the technical literature. Moreover, we found that the recommendations varied in the technical literature. Thus, when administering medication to a breastfeeding mother, it is essential to fully consider not only the package insert, but also the contents of the technical literature.
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Masanaga Matsumoto, Tetsuro Kamimura, Yukihiro Toriigahara, Kyosuke Ta ...
2025Volume 61Issue 2 Pages
311-317
Published: 2025
Released on J-STAGE: November 10, 2025
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All the seven cases of neonatal necrotizing enterocolitis(NEC)in full-term infants treated at our hospital had congenital heart disease(CHD), which we defined as CHD-NEC. We retrospectively examined the clinical backgrounds, pathogenesis, patterns of onset, and outcomes of CHD-NEC.
Single ventricle circulation was detected in five cases(71%). In all seven patients, mesenteric hypoperfusion prior to onset was suspected; high pulmonary blood flow was implicated in five; and in two, onset followed cardiac arrest and multiorgan failure. Surgical interventions for NEC were performed in five cases(71%). Necrotizing intestinal lesions were beyond the distal end of the ileum in all cases, colonic lesions were detected in six cases(86%). The mortality rate was 57%, but all deaths occurred in the long-term postoperative period. They had few risk factors typically associated with preterm NEC(PT-NEC).
In contrast to PT-NEC associated primarily with intestinal immaturity, CHD-NEC was caused by mainly decreasing intestinal blood flow. Predominance of colon in necrotizing lesion was particularly characteristic in CHD-NEC. We suggest that CHD-NEC should be regarded as a distinct pathophysiological mechanism to PT-NEC, and it is essential to optimize targeted prevention and management strategies for CHD-NEC.
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Megumi Tsunakake, Maiko Sagawa, Naofumi Yamane, Yumiko Kan, Hiroko Nak ...
2025Volume 61Issue 2 Pages
318-323
Published: 2025
Released on J-STAGE: November 10, 2025
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We report two cases of ring chromosomes with fetal ultrasound abronalities that were prenatally diagnosed by amniocentesis. Case1 is 28-year-old woman, gravida 2, para 1, conceived spontaneously. She presented to our hospital at 18 weeks gestation with fetal edema and fetal growth restriction. The fetus was diagnosed with ring chromosome 13 on amniocentesis. After genetic counseling, the couple opted for an abortion and delivered a 422g male baby at 21 weeks 4 days gestation. Case2 is 38-year-old woman, gravida 1, para 0, conceived by in vitro fertization. She presented to our hospital at 15 weeks gestation with cystic hygroma and fetal growth restriction. The fetus was diagnosed with ring chromosome 15 on amniocentesis. After genetic counseling, the couple opted for an abortion and delivered a 174g male baby at 20 weeks 2 days gestation.
Ring chromosome 13 and 15 are rare genetic disorder, the phenotypes vary from mild to severe because of the site of deletion. Therefore, it is important to provide appropriate information based on the deletion site and ultrasonography findings to encourage parents to make decisions.
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Yoshimasa Uematsu, Ken-ichiro Konishi, Kiyoshi Tanaka, Hajime Takayasu ...
2025Volume 61Issue 2 Pages
324-328
Published: 2025
Released on J-STAGE: November 10, 2025
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【Introduction】Advancements in fetal ultrasonography have increased the detection of pyriform sinus fistula(PSF)as a fetal cervical cyst. Since PSF often presents with respiratory distress or infection, early diagnosis is crucial. However, right-sided cases are rare and frequently lead to delayed diagnosis.
【Case 1】A fetal cervical cyst was detected on the right side during the prenatal period. Postnatal ultrasonography revealed an irregularly shaped, simple cyst in the right neck. On the first day of life, air was observed within the cyst following the initiation of oral feeding. By day three, debris appeared, and by day five, signs of cyst infection were noted. Surgical excision of the cyst was performed on day eight.
【Case 2】A fetal cervical cyst was detected on the right side during the prenatal period. Postnatal ultrasonography revealed an irregularly shaped, simple cyst containing air on the right side of the neck. The air content increased following oral feeding. By day two, debris appeared, and by day five, signs of cyst infection were noted. Surgical excision of the cyst was performed on day seven.
【Discussion】PSF exhibits characteristic ultrasonographic findings, with the sequential appearance of air and debris within a previously simple cyst. Frequent ultrasonographic evaluation is essential for early diagnosis, and PSF should be considered in the differential diagnosis, even for right-sided cervical cysts.
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Masaaki Kuda, Tsukasa Nakama, Satoshi Ieiri, Mitsuhisa Takatsuki
2025Volume 61Issue 2 Pages
329-334
Published: 2025
Released on J-STAGE: November 10, 2025
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Neonatal cervical cysts are rare but significant clinical findings. This case report discusses a 15-day-old girl with a left cervical cyst, highlighting the diagnostic challenges and surgical management of neonatal pyriform sinus cyst. On day 5 after birth, the patient presented with left cervical swelling and wheezing. Initial imaging revealed a left cervical cyst displacing the trachea with significant inflammation. Incision and drainage were performed, and the patient was referred with suspected cervical lymphangioma. Subsequent CT scans indicated air in the cyst, suggesting a pharyngeal cleft remnant. Despite inconclusive ultrasonography, CT angiography, barium swallow test, and cyst puncture contrast study, a pyriform sinus fistula was highly suspected. Following inflammation subsidence, surgery was performed on day 29. Preoperative endoscopy confirmed a left pyriform sinus fistula. The cyst was excised, and a guidewire was used to ensure complete fistula removal. This case underscores the increasing incidence of congenital pyriform sinus fistula in neonates. Accurate diagnosis and complete surgical excision are crucial for effective management. This report, along with a review of 48 cases in Japan, emphasizes the importance of precise fistula localization and appropriate surgical techniques.
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Keisuke Fukuda, Naoki Yamada, Natsumi Ito, Momoko Seki, Kazue Miyamoto ...
2025Volume 61Issue 2 Pages
335-339
Published: 2025
Released on J-STAGE: November 10, 2025
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Noninvasive prenatal genetic testing(NIPT)is a nonconfirmatory test. We report a case of a false-positive NIPT result due to a supernumerary marker chromosome derived from chromosome 18.
Case: A 44-year-old G4P3 woman. She requested prenatal diagnosis and was referred to our hospital at 10 weeks and 5 days’ gestation. After genetic counseling, NIPT was performed. The result was positive for trisomy 18. Amniocentesis was performed. The chromosome analysis revealed a 47,XX,+mar.ish der(18)(D18Z1+)dn karyotype. The chromosome karyotypes of both parents were normal, and the marker chromosome was found to be de novo. The marker chromosome was derived from the centromere region of chromosome 18, and as it is extremely small, it was thought that the risks of clinically significant symptoms were fewer than the general risks. At 38 weeks and 3 days’ gestation, a cesarean section was performed to prevent the rupture of a maternal aneurysm. The baby was a 2,568-g girl. She had a normal phenotype at birth, and no developmental disorder was observed at the age of 3 years.
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Yudai Kubo, Yukako Kawasaki, Akiko Hiraiwa, Kentaro Nakamura, Mitsuhid ...
2025Volume 61Issue 2 Pages
340-345
Published: 2025
Released on J-STAGE: November 10, 2025
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Pyruvate dehydrogenase complex(PDHC)deficiency is a rare inborn error of metabolism characterized by developmental delay and hyperlactatemia. The neonatal form presents with brain structural abnormalities and frequently progresses to West syndrome with refractory seizures. We report the case of a female neonate with ventriculomegaly, born at 37 weeks’ gestation, weighing 2,154g(-1.8SD), and with a head circumference of 29.5cm(-2.4 SD). Blood gas analysis at birth revealed lactic acidosis, with elevated serum lactate and pyruvate levels(66.9/4.2mg/dL). Genetic testing confirmed a mutation in the PDHA1 gene. Intractable apneic seizures appeared starting at day 13, epileptic waveforms consistent with apneic seizures were observed, leading to a diagnosis of apneic seizures due to epilepsy. Treatment included a ketogenic diet and phenobarbital for metabolic stabilization and seizure control. Despite initial stabilization and a transition to home care, the patient experienced a relapse of focal epilepsy with apneic seizures at 4 months, triggered by a rhinovirus infection. This case underscores the importance of timely diagnosis and early therapeutic intervention, particularly through electroencephalographic monitoring during seizure episodes, for the effective management of neonatal PDHC deficiency.
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Masaho Suetsugu, Koki Shimura, Yukiko Tanaka, Miyoko Waratani, Taisuke ...
2025Volume 61Issue 2 Pages
346-350
Published: 2025
Released on J-STAGE: November 10, 2025
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In pregnancies complicated by Basedow’s disease, maternal thyroid-stimulating hormone(TSH)receptor antibodies(TRAb)and antithyroid drugs can traverse the placenta, potentially inducing fetal thyroid dysfunction and fetal goiter. Fetal goiter can lead to significant complications, including polyhydramnios and postnatal respiratory distress due to tracheal compression, underscoring the importance of precise prenatal diagnosis and timely therapeutic intervention.
The case pertains to a 40-year-old primiparous woman, diagnosed with Basedow’s disease in the first trimester. She commenced treatment with propylthiouracil(PTU)and potassium iodide(KI). However, at 26 weeks of gestation, polyhydramnios and fetal goiter was detected, prompting referral to our institution. Ultrasonographic findings confirmed the diagnosis of fetal hypothyroidism-associated goiter. Discontinuation of KI and PTU at 34 weeks resulted in the normalization of amniotic fluid volume and a marked reduction in the size of the fetal goiter. Considering the potential risk of neonatal airway obstruction, an elective cesarean section was performed at 38 weeks of gestation. The female neonate, weighing 2,716 g, had Apgar scores of 9 and 10 at 1 and 5 minutes, and no respiratory compromise was observed.
In cases of Basedow’s disease complicated by fetal goiter, the assessment of fetal thyroid function and modulation of maternal antithyroid drugs are crucial for favorable perinatal outcomes.
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Ayano Tsutsumi, Hiroki Kitaoka, Makiko Kirino, Chiharu Irisa, Jun Kako ...
2025Volume 61Issue 2 Pages
351-357
Published: 2025
Released on J-STAGE: November 10, 2025
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Congenital syphilis has been increasingly reported in Japan, mirroring the rise in syphilis cases nationwide. Early diagnosis and treatment of syphilis in pregnant women are critical to preventing congenital syphilis. However, a lack of prenatal care in some cases results in unrecognized infections until the later stages of pregnancy, delaying appropriate treatment.
This report presents two preterm cases of early congenital syphilis in neonates born to mothers who did not receive prenatal care. Despite challenges in diagnostic testing due to the critical condition of the infants, PCR successfully identified the pathogen. Antibacterial therapy, combined with supportive care, led to favorable short-term outcomes.
These cases highlight the pressing need to raise public awareness of sexually transmitted infections and to strengthen support systems for women.
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Hiroha Tokui, Ai Anami
2025Volume 61Issue 2 Pages
358-361
Published: 2025
Released on J-STAGE: November 10, 2025
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This is a report of an unique urethral duplication with urethrorectal fistula in which fetal intestinal dilation and multiple foci of calcified intraluminal meconium were observed, accompanied by oligohydramnios, making fetal diagnosis difficult. The patient was a 28-year-old primigravida. During a routine antenatal check-up at 25 weeks of gestation, fetal ventricular septal defect and intrauterine growth restriction(-1.5SD)were noted. At 27 weeks, the amniotic fluid volume began to decrease, and we diagnosed oligohydramnios at 29 weeks. In addition, a dilated hyperechogenic bowel with ball-like echogenic structures were observed at the dorsal side of the fetal bladder. These findings suggested a fetal condition involving anorectal malformations, urethrorectal fistula, and lower urinary tract obstruction. Due to the presence of oligohydramnios, the patient was hospitalized from 35 weeks. At 36 weeks and 1 day, spontaneous rupture of membranes occurred, and an emergency cesarean section was performed due to breech presentation. During delivery, a large amount of intraluminal meconium calculi was expelled from the infant’s anus. No anorectal malformations ware observed, and urethral closure was confirmed. The infant was transferred to a specialized medical facility for surgical treatment at the second day of life. Further examination revealed a diagnosis of urethral duplication and urethrorectal fistula. A surgical procedure to separate the urethra and rectum is planned.
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Yuya Akari, Go Ebihara, Takeshi Kusuda, Azusa Kajiyama, Mao Sasaki, Ko ...
2025Volume 61Issue 2 Pages
362-366
Published: 2025
Released on J-STAGE: November 10, 2025
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Apert syndrome is one of craniosynostosis with syndactyly and various complications. But gastrointestinal complications are rare. Here we present a case of Apert syndrome with esophageal and multiple jejunal atresia. In this case, Apert syndrome was suspected because of fetal brachycephaly, long biparietal diameter and exophthalmos and syndactyly of hands and feet. There was lack of stomach bubble accompanied by intestinal dilatation and maternal polyhydramnios, so we considered the possibility of esophageal and intestinal atresia. The baby was born by caesarean section at 37 weeks 5 days gestation, bile-like bronchial secretions were observed in trying to intubate. After we diagnosed esophageal atresia and small intestinal atresia by the radiography, esophageal banding, gastrostomy and jejunostomy were performed on the day of birth. We finally diagnosed multiple jejunal atresia. Only one case of small intestinal atresia in Apert syndrome has been reported, and multiple small intestinal atresia has not been reported. When diagnosing Apert syndrome, gastrointestinal tract disorder should be considered. In this report, we present this case focusing on the pathogenesis of gastrointestinal tract disorder.
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Norie Tojo, Koki Shimura, Yukiko Tanaka, Miyoko Waratani, Taisuke Mori
2025Volume 61Issue 2 Pages
367-372
Published: 2025
Released on J-STAGE: November 10, 2025
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Aorto-left ventricular tunnel(ALVT)is a rare congenital heart disease characterized by an abnormal communication between the ascending aorta and the left ventricle, bypassing the aortic valve. It is extremely rare, accounting for less than 0.1% of congenital heart diseases, with very few reported cases of prenatal diagnosis; however, prenatal diagnosis is crucial due to the risk of fetal death or sudden death in some cases. A 28-year-old primiparous woman was referred at 36 weeks’ gestation due to fetal cardiomegaly. Fetal ultrasound revealed no signs of hydrops or anemia. The four-chamber view demonstrated significant cardiac enlargement, with a total cardiac dimension of 43.4 mm and a cardiothoracic area ratio of 0.44. Left ventricular systolic function was reduced, with a left ventricular fractional shortening(LVFS)of 0.23. The three-vessel view revealed a dilated aortic diameter, and the three-vessel trachea view showed diastolic aortic regurgitation. Color Doppler imaging of the left ventricular long-axis showed diastolic aortic regurgitation bypassing the aortic valve and flowing into the left ventricle, leading to a prenatal diagnosis of ALVT. A cesarean section was performed at 37 weeks due to the potential need for postnatal emergency surgery. The male neonate, weighing 2,931 grams with Apgar scores of 8 at 1 and 5 minutes, developed hypoxemia requiring endotracheal intubation. Postnatal echocardiography confirmed ALVT, and surgical closure was performed on the same day. Prenatal diagnosis of ALVT enabled timely postnatal intervention. In the fetal diagnosis of ALVT, assessment of the left ventricular outflow tract with color Doppler is crucial.
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Hina Kameoka, Riki Nishimura, Masashi Sagara, Hironori Shimozawa, Yuka ...
2025Volume 61Issue 2 Pages
373-378
Published: 2025
Released on J-STAGE: November 10, 2025
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Anti-Diegoa(Dia)antibodies can cause hemolytic disease of the newborn(HDN). This is a case report of severe HDN due to an anti-Dia immune response. A female neonate was born at 39 weeks and 6 days of gestation, weighing 3,958g. Her transcutaneous bilirubin level was 7.4mg/dL at 14 hours of life, but rapidly increased to 17.1mg/dL by 38 hours. Laboratory testing revealed a total bilirubin of 23.6mg/dL, exceeding the threshold for exchange transfusion, and she was transferred to our hospital for treatment. Upon admission, her total bilirubin level was 24.8mg/dL, and her unbound bilirubin was 1.69μg/dL, both above the exchange transfusion criteria. However, no symptoms of acute bilirubin encephalopathy were observed. Intensive phototherapy and intravenous fluids were immediately initiated, and intravenous immunoglobulin was added after diagnosing HDN caused by anti- Dia antibodies, followed by exchange transfusion. After these treatments, total bilirubin levels decreased rapidly, with no rebound after two additional days of phototherapy. At her six-months check-up, she had successfully reached all developmental milestones, and showed no symptoms suggestive of athetosis. T2-weighted MRI images of the head revealed no signs of bilirubin encephalopathy. Since anti-Dia antibodies carry a risk of severe HDN, appropriate management during pregnancy and after delivery in a tertiary hospital is crucial for both mother and infant.
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Ryo Takano, Haruna Abe, Natsumi Itoh, Fukuyoshi Higashi, Keiko Nishida ...
2025Volume 61Issue 2 Pages
379-383
Published: 2025
Released on J-STAGE: November 10, 2025
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Augmentation cystoplasty is a surgical procedure that is sometimes performed for refractory bladder storage dysfunction owing to conditions such as spina bifida. With treatment advances, women who have undergone augmentation cystoplasty have subsequently been able to become pregnant and deliver their babies; however, few studies have investigated the perinatal management of these pregnancies. We describe the management of pregnancy in a 36-year-old woman with underlying spina bifida, who had undergone augmentation cystoplasty during early childhood and achieved pregnancy for the first time using in vitro fertilization-embryo transfer. The patient had recurrent urinary tract infections and hydronephrosis during the second trimester of pregnancy. Owing to difficulties with ureteral stent placement, she underwent bilateral nephrostomy(at 17 weeks and at 22 weeks of gestation). The patient was generally well thereafter; however, at 37 weeks of gestation she developed ileus, and a decision was made to terminate the pregnancy. Labor was induced, but delivery was stopped, and an emergency cesarean section was performed. Despite the ureter being highly deviated, delivery was completed without any organ damage, and both the mother and child progressed well. The risk of urinary tract infections is high in pregnancies after augmentation cystoplasty, and appropriate therapeutic interventions in collaboration with other departments are required.
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Yuki Temma, Fumihito Miyake, Kentaro Watanabe, Mariko Yui, Kouji Motok ...
2025Volume 61Issue 2 Pages
384-388
Published: 2025
Released on J-STAGE: November 10, 2025
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The patient was a newborn boy. Fetal ascites was detected at 18 weeks of gestation, then it resolved spontaneously. At 35 weeks of gestation, the mother was admitted to our hospital due to a fetal transient bradycardia. Subsequently, an emergency cesarean section was performed at 37 weeks and 3 days of gestation because of non-reassuring fetal status. At birth, the infant exhibited gasping and bag-mask ventilation was initiated without success. The vocal cord was not observed, and the intubation was unsuccessful. Effective ventilation was finally achieved 30 minutes after birth with an emergency tracheostomy performed by pediatric surgeons. On day 5, the subglottic lumen could not be identified with a laryngeal fiberscope and the patient was diagnosed with congenital laryngeal obstruction. Further evaluations, including swallow esophagography and cervicothoracic contrast CT scan, revealed no evidence of tracheoesophageal fistulas. After stable spontaneous breathing was confirmed, the patient was discharged on day 56. At 10 months of age, a diagnosis of congenital laryngeal web was made based on laryngeal fiberscope, which revealed a slight opening in the glottis and contraction of the false vocal cords. Despite the lack of prenatal diagnosis, rapid collaboration with pediatric surgery teams led to the patient’s survival and discharge without major comorbidity. The characteristic imaging findings of congenital high airway obstruction syndrome(CHAOS)can sometimes resolve spontaneously. In cases of transient fetal ascites, the possibility of CHAOS should be considered.
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Masahiro Miwa, Risa Asui, Hiroshi Mizumoto
2025Volume 61Issue 2 Pages
389-393
Published: 2025
Released on J-STAGE: November 10, 2025
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This case involves a preterm infant born at 34 weeks of gestation, who remained stable in terms of respiratory and circulatory status after birth. On day 21 of life, direct bilirubin increased to 1.5 mg/dL, prompting further investigation. The infant was found to have hyperthyroidism, with TSH < 0.01 μU/mL and free T4 at 3.24 ng/dL. Additionally, thyroid-stimulating antibody(TSAb)was positive. Treatment with methimazole was initiated on day 21 of life, and by day 48, direct bilirubin had normalized. The mother was asymptomatic; however, on postpartum day 22, she was found to be TSAb-positive and was diagnosed with Graves’ disease. Retrospective serum analysis at birth showed that both the mother and infant had undetectable TSH levels, while free T4 remained within the normal range. The mother tested positive for both TSAb and TSH stimulation-blocking antibody(TSBAb), suggesting that both antibodies had been transferred to the infant. By day 37 of life, the infant remained TSAb-positive, while TSBAb had become negative. During late pregnancy, TSBAb influenced thyroid function, keeping it normal in both the mother and infant. However, after birth, TSBAb disappeared first, leading to the delayed onset of neonatal hyperthyroidism.
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Eriko Omi, Eisaku Endo, Ryo Takano, Ayaka Tsuihiji, Masayuki Kumazaki, ...
2025Volume 61Issue 2 Pages
394-398
Published: 2025
Released on J-STAGE: November 10, 2025
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Echovirus 11(E-11), a member of the Enterovirus genus, causes a broad spectrum of illnesses ranging from asymptomatic to life-threatening manifestations. In recent years, E-11 has been increasingly recognized as a cause of severe neonatal infections, often presenting with sepsis, multi-organ failure, and high mortality rates. In May 2023, an initial report from France described nine cases of severe E-11 infection. In Japan, severe neonatal fatalities due to E-11 have been reported since 2024. We present a case of neonatal E-11 infection. The mother, at 35 weeks and 2 days of gestation, was admitted to the obstetrics unit for persistent fever and abdominal pain. Laboratory findings revealed an elevated C-reactive protein(2.26mg/dL)with a normal white blood cell count. Suspecting chorioamnionitis, an emergency cesarean section was performed. The neonate had a birth weight of 2,561g and Apgar scores of 9 at both 1 and 5 minutes. Postpartum, the mother’s symptoms resolved, and the mother-newborn contact was permitted. On day three, the neonate developed apnea requiring respiratory support. On day four CSF sample was taken and E-11 PCR was positive. By the day eight, the infant developed liver failure, which rapidly progressed to multi-organ failure, and severe coagulopathy, leading to death. The clinical presentation suggested maternal E-11 infection with vertical transmission to the newborn. The mild CRP elevation with a normal white blood cell count further supported a viral rather than a bacterial infection. When maternal fever with abdominal pain occurs during pregnancy, given the increasing reports of severe neonatal E-11 infections, clinicians should maintain a suspicion of the possibility of E-11 infection. The timing and mode of delivery, as well as postnatal mother-infant contact, should be carefully considered.
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Sumika Kambara, Maki Kondo, Yuichiro Sugiyama, Akiko Saito, Atsushi Na ...
2025Volume 61Issue 2 Pages
399-403
Published: 2025
Released on J-STAGE: November 10, 2025
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A low Apgar score and the need for cardiopulmonary resuscitation in preterm infants are associated with high mortality and neurodevelopmental impairment. We experienced a case of an extremely preterm infant who was born in an ambulance and underwent cardiopulmonary arrest upon arrival at the hospital but recovered following resuscitation and survived without complications. The infant was a male born at 24 weeks and 4 days of gestation, weighing 620g. The mother had experienced abdominal pain and called for emergency services; she was then delivered in the ambulance. Emergency medical technicians initiated chest compression and positive pressure ventilation, and the neonate was transported without umbilical cord clamping. Upon the patient’s arrival at the hospital, the neonatologist examined the patient and resumed resuscitation due to cardiopulmonary arrest. He subsequently recovered, and chest compression was stopped at 26 minutes of age. The patient was then admitted to the neonatal intensive care unit. No intracranial hemorrhage was observed, and his condition gradually stabilized. The hospitalization course was uneventful, and the patient was discharged at the corrected age of 46 weeks without complications or the need for home medical care. Magnetic resonance imaging of the head revealed no abnormalities, and at a chronological age of 3 years, his developmental quotient on the Kyoto Scale of Psychological Development was 79, without neurodevelopmental impairment. Delayed umbilical cord clamping, although incidental, might have contributed to the avoidance of neurodevelopmental impairment by increasing blood volume and stabilizing circulation through continued blood flow between the mother and infant.
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Ikuko Hama, Shin-ichiro Mizutomi, Hidenori Iwasaki, Yusuke Mitani, Tak ...
2025Volume 61Issue 2 Pages
404-408
Published: 2025
Released on J-STAGE: November 10, 2025
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Hirschsprung’s disease(HD)is characterized by functional intestinal obstruction due to the absence of ganglion cells in the intestine. The prevalence of preterm infants with HD has increased in recent years. While Hirschsprung-associated enterocolitis, bowel perforation and ileus are well-documented complication, small intestinal volvulus without malrotation has not been previously reported. We report a case of an extremely low birth weight infant with HD, complicated by intestinal volvulus without malrotation. The patient was a girl born at 25 weeks of gestation with a birth weight of 711 g and had a family history of HD. HD was suspected due to abdominal distension. She tolerated enteral feeding with frequent enemas and bougie insertion. At 70 days of age, abdominal distension worsened following sedation and ventilatory management for photocoagulation for retinopathy of prematurity. Although bowel decompression was initiated, abdominal distention did not fully resolve. At 83 days of age, the distention further exacerbated, accompanied by respiratory deterioration and elevated inflammatory markers. Given the suspicion of HD-associated ileus or enteritis, bowel decompression and antibiotic therapy were administered. However, her condition did not improve, and laparotomy was performed. Intestinal volvulus without malrotation was identified, and a portion of the ileum subsequently progressed to gangrene, necessitating resection. Biopsy of the sigmoid colon during laparotomy and rectal biopsy at 145 days of age confirmed the diagnosis of HD. This case suggests that preterm infants with HD may develop small intestinal volvulus, a complication that should not delay the decision for laparotomy.
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Mayumi Kamiyama, Atsushi Watanabe, Ken Takada, Tomohiro Saito, Motohir ...
2025Volume 61Issue 2 Pages
409-413
Published: 2025
Released on J-STAGE: November 10, 2025
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Neonatal suppurative submandibular adenitis is a rare disease that often occurs in preterm infants. In this report, we describe the clinical course of a full-term baby with this disease. The patient was born at 39 weeks of gestational age with a birth weight of 3,012 g. At day age 15, she was admitted to the hospital with fever. She had a 15-mm mass in the right submandibular region and erythema around the mass. Pus drained from the Wharton’s canal orifice by compression of the mass, and a pus smear revealed gram-positive cocci, leading to a diagnosis of suppurative submandibular adenitis. After initial infusion and administration of ABPC/SBT, the patient recovered from peripheral circulatory failure and her fever resolved the next day. Pus, pharyngeal, and mother milk cultures detected Staphylococcus aureus, suggesting an ascending infection from the oral cavity. The mass disappeared on the 6th day of treatment, and the patient was discharged from the hospital after completion of antibiotic therapy on the 14th day. Two months after discharge, an outpatient examination revealed no recurrence. It is important to consider submandibular adenitis when a submandibular grand is swelled, for immediate diagnose and treatment even in a full-term baby.
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Tomomasa Terada, Tokuo Miyazawa, Kousuke Oikawa, Gakuto Ujiie, Akio Eb ...
2025Volume 61Issue 2 Pages
414-418
Published: 2025
Released on J-STAGE: November 10, 2025
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Peripherally Inserted Central Venous Catheters(PICC)are widely used in various pediatric settings, including neonatal care. However, catheter replacement is necessary when occlusion occurs. Catheter occlusion can be caused by mechanical factors, such as catheter kinking or damage, as well as intraluminal obstruction due to blood components or drug-derived deposits.
It is generally difficult to determine the cause of catheter occlusion, and in clinical practice, the exact cause is rarely identified. In this study, we analyzed the tip of an occluded PICC using scanning electron microscopy and energy-dispersive X-ray spectroscopy, which revealed that the occlusion was caused by sodium and chloride deposits. We report this case because the cause of occlusion was not the commonly reported blood cell components or calcium phosphate, and because the composition of the occlusive material was identified relatively easily using this analytical method.
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Ranna Kai, Masahiro Kinoshita, Mitsuaki Umino, Eimei Harada, Yasuki Ma ...
2025Volume 61Issue 2 Pages
419-423
Published: 2025
Released on J-STAGE: November 10, 2025
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Since 2015, our hospital has provided neonatal medical support via video calls in collaboration with local obstetric clinics. We report a case of neonatal asphyxia in which remote support proved effective. A female neonate was delivered at 38 weeks and 6 days of gestation by emergency cesarean section due to umbilical cord prolapse and fetal distress. Our hospital was contacted before delivery, and a video call was initiated to conduct a pre-delivery briefing and provide real-time resuscitation support. Remote guidance enabled appropriate clinical decision-making and instructions throughout the resuscitation process. Following birth, the neonate developed respiratory distress and was transferred to our hospital. She recovered well and was discharged without any neurological sequelae. Prior to this case, our hospital and the obstetric clinic had conducted regular video conference meetings, which contributed to familiarization with the equipment and strengthened interprofessional collaboration. These factors likely facilitated smooth coordination during the emergency. This case demonstrates the effectiveness of remote neonatal resuscitation support. It also highlights the importance of ongoing inter-facility communication and training, and suggests potential areas for future development in remote neonatal care.
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