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Tetsuro Murakawa, Masahiko Murase, Emi Fukuoka, Mio Igawa, Tokuo Miyaz ...
2022 Volume 58 Issue 1 Pages
18-23
Published: 2022
Released on J-STAGE: May 10, 2022
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Chickenpox, also known as varicella, is an airborne infection. Since almost all Japanese NICUs are shared rooms, there is a high risk of horizontal transmission of disease. However, there are no guidelines on the prevention of horizontal infection in the NICU. One mother who delivered a very low-birth-weight infant in our hospital had contact with her infant without realizing that she had breakthrough chickenpox. A hospitalization limit of 28 days from the date of visit was enforced to prevent infection. Acyclovir or Valaciclovir were administered to all children in our NICU to prevent horizontal transmission of infection. Gamma-globulin was administered to infants who were in contact with the mother, with herpes zoster, and with no history of chickenpox or chickenpox vaccination. The mother had no history of chickenpox or chickenpox vaccination, and her husband received two doses of chickenpox vaccine. Only parents with a known history of chickenpox, those who had received two doses of chickenpox vaccine, and those who have antibodies to chickenpox were allowed to visit the hospital. Parents who did not meet these conditions but wanted to visit the hospital got tested for chickenpox antibody titers. Eleven patients did not meet these conditions, and on testing, they were confirmed as positive for varicella antibodies. No infants or parents in our NICU were infected.
Horizontal transmission can be prevented by individual measures.
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Naomichi Mano, Hirari Ariga, Takehiko Yokoyama, Taihei Tanaka
2022 Volume 58 Issue 1 Pages
24-30
Published: 2022
Released on J-STAGE: May 10, 2022
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There is limited research on vaccine failures for the prevention of mother-to-child transmission of hepatitis B virus(HBV)in Japan. Thus, we conducted a survey aimed at investigating the current status of the prevention of perinatal HBV transmission. The response rates to the primary survey were 42% among 142 delivery facilities and 26% among 1,578 pediatric advocacy facilities. The preliminary response rate was estimated to represent 51% of the total births in the Aichi Prefecture, based on the 162,161 recorded births at the participating facilities. The secondary survey gained a 90% response rate after its administration to 30 facilities with delayed or missed vaccinations, cases of mother-to-child transmission, or vaccine failures. Results showed that 552 children received protection against perinatal HBV transmission, and 14 had missed out on this protection. Antibody titer tests were repeated in only one-third of the facilities, including the secondary survey participants. Primary vaccine failure was observed in five cases, although antibody titers increased in all cases with additional vaccination. This study highlighted the importance of developing follow-up vaccination guidelines and revealed the barriers to additional vaccination and testing in the insurance system.
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Risa Andou, Soromon Kataoka, Fuyuka Igarashi, Midori Imaizumi, Asaka S ...
2022 Volume 58 Issue 1 Pages
31-36
Published: 2022
Released on J-STAGE: May 10, 2022
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Mother-to-child transmission of cytomegalovirus(CMV)is the most frequent TORCH syndrome, with 1% –2% of pregnant women developing primary CMV infection during pregnancy, resulting in a 30% –50% rate of fetal infection. However, a previous report has shown that educating pregnant women on prevention of CMV infection reduces their risk of primary infection. In the current study, the effectiveness of information provision for preventing primary CMV infection in pregnant women was verified. This study included 2,568 women who gave birth during a 4-year period in our hospital. Pregnant women without CMV infection were extracted by testing for the presence of CMV IgG antibodies during their first trimester. The women without CMV infection were made aware of preventative measures against CMV infection. CMV IgG antibodies were measured during the first trimester of 1,283 cases, of which, 380 cases(29.6%)were IgG-negative. Among them, two cases(0.5%) became positive for CMV IgG during the third trimester. The primary infection rate in pregnant women who were educated on infection prevention was lower than the previously reported rate of 1%–2%.
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Yoshinori Fujinaka, Asami Matsunaga, Megumi Takahashi, Megumi Seya, Yo ...
2022 Volume 58 Issue 1 Pages
37-43
Published: 2022
Released on J-STAGE: May 10, 2022
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Of 4,013 term infants born in our hospital during the 3 years from 2014 to 2017, approximately 30%(1,239 infants)were early term. Excluding multiple births, congenital malformations, and small- and large-for-gestationalage infants, 894 early-term and 1, 695 full-term infants were identified. Maternal background, neonatal complications, and hospitalization rates were examined retrospectively. Maternal complications were found to be more common in the early-term group; in terms of delivery method, cesarean section, especially planned cesarean section, was more common in the early-term group. Neonatal asphyxia(5-min Apgar < 7)and hypoglycemia(< 50 mg/dL)were more common in the early-term group in the univariate analysis; however, only hypoglycemia was significantly different in the multivariate analysis. Early-term birth was an independent risk factor for neonatal hospitalization rates and readmission rates(within 1 month after birth). Delivery as close as possible to the full term is desirable; however, considering the risks associated with emergency cesarean section surgery, we consider the planned cesarean section which time is slightly earlier than full term is appropriate.
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Maiko WAGATA, Ryo YAMAMOTO, Jun SASAHARA, Takeshi KANAGAWA, Keisuke IS ...
2022 Volume 58 Issue 1 Pages
44-51
Published: 2022
Released on J-STAGE: May 10, 2022
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The objective of the present study was to elucidate the limits of gestational age and birth weight for viability and survival without neurodevelopmental impairment among small for gestational age infants delivered at preterm period. The retrospective observational study including singleton delivered before 34 weeks’ gestation with the birth weight under 3 % tile of local standard between 2010 and 2015 was performed. Outcomes were the prevalence of good short-term prognosis, which was the survival at 28th days after birth, and of good long-term prognosis, which was the survival at three years old without cerebral palsy, mental retardation with the score of Kyoto Scale of Psychological Development under 70, visual disturbance, or acoustic disturbance. Gestational age and birth weight that more than half of infants have good short-term or long-term prognosis were defined as the limit of viability or survival without neurodevelopmental impairment, respectively. Among 61 participants the prevalence of good short-term and long-term prognosis was 92%(56/61)and 71%(39/55), respectively. The limit of viability was 26 gestational weeks and birthweight ≥400g. The limit of survival without neurodevelopmental impairment was 27 gestational weeks and at birthweight ≥400g. Gestational age and birth weight to predict frequent good short-term prognosis were 26 weeks and 550 g and to predict frequent good long-term prognosis were 27 weeks and 650 g, respectively. These results might be useful for the pregnancy management and patient counseling in cases with FGR.
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Takashi Seike, Kosuke Kawakami, Yusuke Kurokawa, Marie Kitagawa, Marin ...
2022 Volume 58 Issue 1 Pages
52-57
Published: 2022
Released on J-STAGE: May 10, 2022
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We retrospectively examined the impact of simulation education of the Japan Council for Implementation of Maternal Emergency Life-Saving System(J-CIMELS)on clinical practice.
The study included 21 cases of critical obstetrical hemorrhage treated in our hospital from January 2016 to December 2019. We classified six cases in the J-MELS group and 15 cases in the non-J-MELS group.
We statistically examined the following parameters: patient background, the severity of illness, initial response, treatment, and time course from the onset.
There were no significant differences in patient background or severity of illness. In terms of the initial response, the J-MELS group responded significantly more appropriately to emergency calls and oxygen administration(P = 0.012 and 0.012, respectively). In terms of treatment, RBC transfusion volume and ABO-incompatible transfusion were significantly higher in the J-MELS group(P = 0.043 and 0.014, respectively). In the time course from the onset, the J-MELS group was significantly faster in each of the following events: declaration of critical obstetrical hemorrhage, the start of RBC transfusion, the start of FFP transfusion, and hemostasis(blood loss of 50 g/h or less)(P = 0.0042, 0.00071, 0.00026, and 0.023, respectively).
This study suggests that the J-CIMELS simulation education may be effective in responding to critical obstetrical hemorrhage. Further dissemination of J-CIMELS is desirable to reduce maternal deaths.
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Kentaro Hirayama, Takeshi Kumagai, Tomoya Tsuchihashi, Takayuki Suzuki ...
2022 Volume 58 Issue 1 Pages
58-62
Published: 2022
Released on J-STAGE: May 10, 2022
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At our NICU, we have newly introduced the Yumizen M100 Banalyst(hereinafter referred to as Yumizen M100)as a point of care testing(POCT)device for CRP measurement. Prior to that, we compared the Latessier M POCT analyzer with the central laboratory analyzer, evaluating and examining the correlation of the CRP value and the error of measurement. From April to August 2021, a total of 76 tests were performed on 27 cases measured by Yumizen M100 and Latessier M, and a total of 57 tests were performed on 23 cases measured by all three analyzers, including the one from the central laboratory. The CRP value measured by Yumizen M100 showed a very strong correlation with the value measured by Lattesier M and the central laboratory respectively(r = 0.977, 0.997). When the central laboratory CRP value was compared with Yumizen M100 and Latessier M, the measurement of error was significantly smaller using the Yumizen M100(0.044 vs. 0.079, p = 0.029). The introduction of POCT analyzers that are quick, simple, highly accurate, and capable of measuring small quantities of sample was considered to be useful in the NICU.
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Shigeru Nishimaki, Katsumi Mizuno
2022 Volume 58 Issue 1 Pages
63-69
Published: 2022
Released on J-STAGE: May 10, 2022
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A questionnaire survey about donor milk and breast milk bank was conducted on baby-friendly hospitals(BFH). Responses were obtained from 61 obstetricians and 77 pediatricians.
Over 70% of the obstetricians and over 80% of the pediatricians knew the existence of breast milk banks; and also that donor milk is given to extremely premature infants and extremely-low-birth-weight infants in the hospitals. Limited to 55 pediatricians working at BFH with NICU, about 85% had intention to administer donor milk from the breast milk bank to babies soon after birth, but on the other hand they concerned about insufficiency of information about milk collection, infection through donor milk, the preservation and transportation of donor milk, and how the baby’s mother would feel. They answered they can cooperate in posting information about breast milk bank and distributing leaflet in the hospitals.
It is necessary to enhance transmission of information about donor milk and breast milk banks to medical staff working at NICU; and measures are awaited to enable administration of donor milk soon after birth without delay.
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Kazutoshi Ueda, Akihito Takahashi, Yuriko Kondo, Eriko Iwasaki, Midori ...
2022 Volume 58 Issue 1 Pages
70-75
Published: 2022
Released on J-STAGE: May 10, 2022
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Background: Patent ductus arteriosus is one of the complications in extremely low birth weight infants and affects their mortality and neurodevelopmental outcomes. We investigated the effects of a change in circulatory management on morbidity such as patent ductus arteriosus and long-term neurological prognosis of extremely low birth weight infants. Methods: We identified extremely low birth weight infants between 2005 and 2017 and classified them into two groups by the treatment timing, namely before and after the introduction of prophylaxis administration of indomethacin. The infants born in other hospitals and those with congenital malformations were excluded. We compared maternal, neonatal, and treatment details, mortality, complications, and the developmental status between the two groups. Results: There were 156 and 220 infants in the early and late groups, respectively. The volume of patent ductus arteriosus surgery significantly increased in the late group. Multivariate analysis showed that indomethacin administration, fluid dose, steroid use in the acute phase, and the use of two sedatives significantly increased in the late group. There were no differences in mortality and developmental delay between the two groups. Conclusions: The change in circulatory management may have caused an increase in patent ductus arteriosus surgery for extremely low birth weight infants.
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Yukiko Taga, Kaoru Kawasaki, Eriko Yasuda, Akeo Kawamura, Ayaka Yamagu ...
2022 Volume 58 Issue 1 Pages
76-82
Published: 2022
Released on J-STAGE: May 10, 2022
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Myomectomy during cesarean section is still controversial because of the potential risk for uncontrolled bleeding. The aim of this study was to clarify the safety of uterine myomectomy during cesarean section. We classified 25 patients who underwent cesarean myomectomy into four groups according to the location of myoma (subserosal + intramural, subserosal, intramural, and cervical). The maximum length and number of enucleated myomas, operation time, blood loss, intraoperative blood transfusion volume, change in hemoglobin concentration (pre-operative level to lowest post-operative level), length of hospital stay after surgery and complications were retrospectively compared among four groups. The cervical group tended to have higher blood loss, hemoglobin changes, autologous blood return, and longer days in postoperative hospital stay than the other groups, but these differences were not significant. There were no serious complications associated with cesarean myomectomy including a case in which myomectomy was also performed at 13 weeks’ gestation. Myomectomy during cesarean section can be safely performed with a proper selection of patients, sufficient preoperative preparations such as autologous and intraoperative strategies to avoid massive blood loss.
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Ryuta Miyake, Katsuhiko Naruse, Junko Hyo, Miho Okamoto, Shoichiro Yam ...
2022 Volume 58 Issue 1 Pages
83-87
Published: 2022
Released on J-STAGE: May 10, 2022
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Postpartum depression has an incidence of 10-15%, and it is important to screen at the 1-month postnatal examination. One of the screening tools is the Edinburgh Postnatal Depression Questionnaire(EPDS). In Nara Prefecture, the first Japanese patient infected with coronavirus infection(COVID-19)was confirmed on January 28, 2020. After the COVID-19 epidemic, our hospital has limitation on visits and attending deliveries, which may be a mental burden on maternity women. In this study, we defined women who delivered at our hospital after the COVID-19 epidemic from January 28 to December 31 2020 as a study group, and women who delivered at the same period in 2019 as a control group. The EPDS values at the time of postnatal medical examination were compared. There were 920 women in the control group and 720 in the study group. The average EPDS values were 3.9 ± 3.9 and 3.9 ± 4.1, respectively, and there was no significant difference between two groups. We analyzed the factors that contributed to the mental load of postpartum women with EPDS of 9 points or more from medical records, but there was no significant difference. The reason for that there was no significant difference in EPDS may be the possibility that the maternity women were able to adapt to the COVID-19 epidemic situation and we and our medical staff provided the appropriate mental support.
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Tatsuya Yoshihara, Yasuhiko Okuda, Satoko Sasazu, Eriko Ogasahara, Shu ...
2022 Volume 58 Issue 1 Pages
88-91
Published: 2022
Released on J-STAGE: May 10, 2022
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Pregnancy with congenital uterine anomaly increase perinatal complications such as miscarriage, premature birth, breach presentation, cesarean section, and fetal growth restriction, but many are under-examined. We retrospectively examined the complications of pregnancy with congenital uterine anomaly at our hospital from 2009 to 2021.39 cases of pregnancy with congenital uterine anomaly were observed, we found that cord insertion site abnormality occurred significantly more frequently. Perinatal complications that are often found in pregnancies with congenital uterine anomaly may be related to cord insertion site abnormality.
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Reiji Nakano, Katsuaki Toyoshima, Hitoshi Yoda
2022 Volume 58 Issue 1 Pages
92-97
Published: 2022
Released on J-STAGE: May 10, 2022
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Critical congenital heart disease(CCHD)is defined as congenital heart disease that requires surgery or other treatment in the neonatal period. Pulse oximetry screening(POS)has been reported to improve the detection of CCHD in European and American countries, although POS algorithm is not established in Japan. The aim of this study is to investigate the implementation challenges of POS to detect CCHD in Japan. In this nationwide survey, 1, 323 of 2, 374 facilities responded to the questionnaire(valid response rate 56%). Ninety one percent of them agree with the need of POS in their facilities. Seventy three percent of them conceive of the implementation of POS in their facilities as a feasible strategy to detect CCHD. This study showed a need for establishment of universal POS protocol in Japan.
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Ayumu Noro, Masato Mizushima, Masaya Uchida, Nobuko Shiono, Tatsuo Sat ...
2022 Volume 58 Issue 1 Pages
98-104
Published: 2022
Released on J-STAGE: May 10, 2022
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We conducted logistic regression analysis for symptomatic patent ductus arteriosus(sPDA)in 357 extremely low birth weight infants who were delivered between January 2008 and December 2019, among whom 177 developed sPDA. The infants were admitted to our neonatal intensive care unit and survived to discharge. Symptomatic patent ductus arteriosus, defined as PDA cases in which the condition required indomethacin(or ibuprofen)administration or PDA ligation, with reference to cardiovascular dysfunction scores, was considered the dependent variable. Perinatal factors and acute therapeutic management items were defined as independent variables. In univariate analysis, significant risk factors for sPDA were: short gestational age, low birth weight, first delivery, indomethacin administration to the mother, low Apgar score at 1 min, high pH values in umbilical cord arterial blood, respiratory distress syndrome(RDS), gastrointestinal perforation, high-frequency ventilation, neonatal volume loading, and catecholamine administration. On the other hand, administration of xanthine drugs within two days of birth was detected as a significant inhibiting factor for sPDA. In multivariate analysis (adjusted odds ratio: 95% confidence interval; p-value), administration of xanthine drugs within two days of birth (0.33: 0.16-0.65; p<0.01), gestational age(+1 week) (0.67: 0.53-0.82; p<0.01), RDS(1.94: 1.07-3.55; p= 0.03), and catecholamine administration(2.28: 1.19-4.41; p=0.01)were detected as factors significantly associated with the occurrence of sPDA. The results suggest that early administration of xanthine drugs to extremely low birth weight neonates might suppress the development of sPDA.
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Miyu Akiyama, Takatsugu Maeda, Hiroko Taniguchi, Nami Kirihara, Takash ...
2022 Volume 58 Issue 1 Pages
105-109
Published: 2022
Released on J-STAGE: May 10, 2022
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We conducted a retrospective study to evaluate the association between the severity of polyhydramnios and its causes, neonatal prognosis, and the accuracy of prenatal diagnosis in singleton pregnancies with polyhydramnios.
This study included 84 singleton pregnancies with polyhydramnios, who delivered at our hospital between January 2015 and December 2019. Polyhydramnios was diagnosed if amniotic fluid index(AFI)was ≥25cm. All patients were classified into three groups based on the maximum value of AFI in the third trimester(mild: AFI between 25-30 cm, moderate: AFI between 30-35 cm, and severe: AFI >35 cm).
The incidence of fetal malformations was significantly higher in moderate or severe group(54%)than in mild group(20%). The mortality rate was significantly higher in infants those who required amnioreduction(55.6%)and those with chromosomal abnormalities(72.7%). The accuracy of prenatal diagnosis was 92%, which was slightly higher than that of previous reports.
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Ken-ichi Honda, Shigeo Hashimoto, Ryota Sakurai, Kunihiko Doh, Aki Sug ...
2022 Volume 58 Issue 1 Pages
110-114
Published: 2022
Released on J-STAGE: May 10, 2022
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We report a case of an umbilical cord hematoma in a primi-para woman who was receiving drip infusion of oxytocin for weakened uterine contractions in the second stage of labor. The fetal heart rate suddenly dropped and the variability remained low, and the baby was delivered by vacuum extraction with compression of the fundus. The placenta seemed to have no specific findings, but the pathological examination of the umbilicus near placenta revealed an intra-umbilical cord hematoma and rupture of the umbilical vein within the Warthon jelly wall. The umbilical vein wall was confirmed using immunohistochemistry for smooth muscle actin and elastica van Gieson staining for collagen and elastic fibers.
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Hanamilena Fujimoto, Hiroki Matsubayashi, Masahiro Yasuda, Yasuhiro Wa ...
2022 Volume 58 Issue 1 Pages
115-119
Published: 2022
Released on J-STAGE: May 10, 2022
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The group of disorders with excess X chromosomes and a female phenotype is called poly X syndrome, and 49,XXXXX is the rarest among them. We describe a case of 49,XXXXX with tracheobronchomalacia. In addition to several dysmorphic features and ventricular septal defect, she presented with persistent, severe, labored breathing. We performed bronchoscopy and diagnosed her with tracheobronchomalacia. Although growth retardation is major complication of 49,XXXXX, her body weight gain and height improved after intracardiac repair and nasal continuous positive airway pressure(CPAP)for tracheobronchomalacia.
As far as we know, this is the first case of 49,XXXXX with tracheobronchomalacia. When labored breathing or feeding disorders are observed in 49,XXXXX cases, active evaluation of the airway is desired.
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Kentaro Niwa, Makoto Orisaka, Haruna Sasaki, Chiyo Tamamura, Hiroshi K ...
2022 Volume 58 Issue 1 Pages
120-125
Published: 2022
Released on J-STAGE: May 10, 2022
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Pregnancy and puerperium are at increased risk of venous thromboembolism(VTE), including deep vein thrombosis(DVT)and pulmonary thromboembolism(PTE). In case 1 who developed VTE in the first trimester of pregnancy, an inferior vena cava(IVC)filter was temporarily placed due to coexistence of PTE, but the IVC filter could be removed after anticoagulant therapy, and vaginal delivery was successful. In case 2 who developed VTE in the third trimester of pregnancy, labor has onset before confirming the disappearance of the iliac vein DVT, and the temporary IVC filter was placed immediately before vaginal delivery. Since the mechanism of VTE onset and the time to delivery are different between the first trimester and third trimester, management of DVT, such as IVC filter placement, is also different during each pregnancy period.
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Yusuke Daimon, Minako Kihara, Takeshi Nishida
2022 Volume 58 Issue 1 Pages
126-130
Published: 2022
Released on J-STAGE: May 10, 2022
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Alveolar capillary dysplasia with misalignment of the pulmonary veins(ACD/MPV)is a rare disorder characterized by abnormal development of pulmonary capillaries, and with severe persistent pulmonary hypertension of the newborn(PPHN), and prolonged progressive respiratory impairment. It is most often suspected based on the presence of refractory PPHN and concomitant anomalies of the cardiovascular, gastrointestinal and urogenital systems, and is diagnosed by FOXF1 genetic testing and/or lung histological examination. We experienced a case of ACD/MPV, which was diagnosed by the characteristic findings of pathological autopsy after death, and was difficult to diagnose without extrapulmonary complications. Even in the absence of complications, it is one disease that should be suspected in cases of refractory PPHN of unknown cause, and appropriate genetic counseling and active genetic analysis should be considered while being alive.
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Wataru Hattori, Noriko Kato, Yoshitaka Shiraishi, Mai Ogawa, Haruna Ta ...
2022 Volume 58 Issue 1 Pages
131-136
Published: 2022
Released on J-STAGE: May 10, 2022
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COVID-19 is an infectious disease caused by SARS-CoV-2. It originated from "unknown pneumonia" in Wuhan, Hubei Province, announced by the China Health Organization in December 2019, and was identified as a new type of coronavirus. In February 2020, the World Health Organization(WHO)named the disease caused by this virus “COVID-19”, and the International Virus Naming Commission named the virus “Severe Acute Respiratory Syndrome Coronavirus-2(SARS-Cov-2)” The virus spread to China and other countries, and on March 11, 2020, the WHO declared a pandemic. Most patients are asymptomatic and clinical cases are mild. Although it naturally resolves, different pathological conditions, such as severe cases requiring artificial respiratory management and death, have been reported. At present, there are few reports on the effects of COVID-19 on pregnant women and fetuses. We report a severe case of COVID-19 in a pregnant woman. COVID-19 developed during pregnancy and cesarean section was performed to terminate the pregnancy, but the respiratory condition did not improve and the patient was admitted to the ICU to undergo ECMO.
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Seiya Yoshida, Masako Araki, Manabu Adachi
2022 Volume 58 Issue 1 Pages
137-141
Published: 2022
Released on J-STAGE: May 10, 2022
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Diabetes insipidus occurs very rarely during pregnancy. The prevalence of this disease is estimated to be about 4 per 100,000 pregnancies. This is caused from the transient increase of placental production of vasopressin. Only one case is reported fetal polyuria and polyhydramnios induced by maternal diabetes insipidus in the past. We are now reporting the second case of diabetes insipidus during pregnancy causing fetal polyuria and polyhydramnios. Furthermore, in our report, immunohistochemically staining of the placenta was performed using an anti-vasopressin antibody, confirming the strong expression of vasopressin’s in chorionic villi. There are several reports of diabetes insipidus during pregnancy, but it is unclear why fetal polyuria and polyhydramnios occurred only in our previous reports and in our case. Diabetes insipidus during pregnancy can occur consecutive, and in our case, if the disease recurs in the next pregnancy, more detailed studies show the exact physiological role of placental vasopressin’s and pathology of this disease may be revealed.
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Masato Yoneyama, Junya Sakuma, Hikari Kotaki, Mayumi Takano, Sumito Na ...
2022 Volume 58 Issue 1 Pages
142-146
Published: 2022
Released on J-STAGE: May 10, 2022
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Fetal intracranial hemorrhage is a rare disease, occurring in 0.5-0.9 cases per 1,000 cases. We report a case of fetal intracranial hemorrhage due to maternal vitamin K deficiency. At 28 weeks and 6 days of gestation, a 37 years old woman, 3 gravida 0 para, visited our hospital due to an eating disorder accompanied by worsening of psychiatric symptoms. She had not taken sufficient diet for several weeks. She was hospitalized and peripheral parenteral nutrition was administered. Ultrasonography at 30 weeks 3 days of gestation showed a left intracranial occupying lesion, and fetal MRI revealed intracranial hemorrhage. Fetal demise was confirmed at 30 weeks 4 days of gestation. Considering the worsening of the mother’s mental state, the stillborn baby was delivered by cesarean section under general anesthesia. Although maternal blood tests showed no abnormal coagulation, vitamin K1 and K2 were both ≦ 0.05 ng/mL, indicating vitamin K deficiency, which was presumed to be the cause of fetal intracranial hemorrhage. Even in cases such as this case, where maternal blood tests showed no abnormalities in coagulation capacity, management with fetal bleeding in mind should be considered. In addition, fetal MRI can provide detailed evaluation of fetal intracranial hemorrhage, and can be useful in explaining the condition to the family and in deciding on a policy.
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Yui AOSHIMA, Tomomi SHIGA, Ken-ichirou MORISHIGE
2022 Volume 58 Issue 1 Pages
147-151
Published: 2022
Released on J-STAGE: May 10, 2022
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A 35-year-old primipara at her 30th week of gestation presented with polydipsia and polyuria. When she was 17 years old, she had a medical history of traumatic central diabetes insipidus, cured in a month with desmopressin(dDAVP). She was diagnosed with gestational diabetes mellitus at 26 weeks of gestation. At 35 weeks of gestation, she was referred to our hospital for a medical examination. Laboratory data showed low arginine vasopressin and urine osmolality. At 36 weeks of gestation, after she was administered nasal dDAVP, her urine volume decreased and urine osmolality elevated rapidly. She was diagnosed to have central diabetes insipidus. Although fetal growth was normal, amniotic fluid deficiency was observed. A healthy baby was delivered via spontaneous vaginal delivery at 39 weeks and four days. The patient’s symptoms did not recur despite the tapering and termination of dDAVP administration after birth.
In this case, the subclinical deficiency of AVP or insufficiency of additional AVP due to past trauma was thought to have caused the manifestation of central diabetes insipidus.
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Hidehiro Takahashi, Tomoaki Nomura, Daisuke Hatanaka, Michiko Kusakari ...
2022 Volume 58 Issue 1 Pages
152-157
Published: 2022
Released on J-STAGE: May 10, 2022
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Infantile hemangiomas are common in preterm and low birth weight infants. Infantile hemangiomas proliferate most rapidly during the proliferating phase, especially between 5.5 and 7.5 weeks of age. However, the safety of propranolol in low birth weight infants and infants younger than 5 weeks has not been established. We treated two premature and very low birth weight infants with propranolol for infantile hemangiomas from early infancy. In a case we had treated successfully, but in another case we had to discontinue propranolol therapy after 6 weeks treatment because of liver dysfunction. It was effective, but safety needed attention. Even preterm and low birth weight infants may be given propranolol early to prevent the sequelae of infantile hemangiomas with careful monitoring. But if side effects are observed, we should discontinue administration.
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Megumi Fujino, Kumiko Nakata, Arisa Kitano, Nami Ota, Shigetaka Yagi, ...
2022 Volume 58 Issue 1 Pages
158-162
Published: 2022
Released on J-STAGE: May 10, 2022
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Placental surface cysts are cystic lesions that develop on the fetal surface of the placenta and have little effect on the course of pregnancy; however, an association with fetal growth restriction and maternal collagen diseases has been reported. We report a case of fetal growth restriction due to placental cysts during pregnancy management for dermatomyositis. Ultrasonography at 28 weeks of gestation revealed many cystic lesions on the fetal surface of the placenta, with the largest being 6.0 cm in diameter. Fetal growth restriction of -2.4 SD was observed at 30 weeks of gestation and emergency cesarean section was performed under a diagnosis of fetal dysfunction. A female baby weighing 1,043 g was delivered(Apgar score 6/9), but there was no abnormality in the postnatal course. Fibrin deposition and X-cells were suggested to be related to the development of placental cysts, and placental pathological findings in this case demonstrated fibrin deposition around the cysts and X-cell proliferation.
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Yuri Kato, Hiroshi Kawamura, Akiko Shinagawa, Makoto Orisaka, Tetsuji ...
2022 Volume 58 Issue 1 Pages
163-168
Published: 2022
Released on J-STAGE: May 10, 2022
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We present a case of 37-year-old Japanese pregnant woman referred to our hospital with rapid increase of systemic edema. Transabdominal ultrasonography revealed complete hydatidiform mole with coexistent fetus(CHMCF). Evacuation and curettage were performed at 11+0 weeks’ gestation because of developing hypertension and proteinuria, and thereafter, pulmonary congestion and bilateral pleural effusion with continuous oxygen desaturation was emerged. She responded well to treatments. After that, persistent gestational trophoblastic neoplasia was developed and she needed multiple regimens of chemotherapy. Even if termination of pregnancy was selected in CHMCF due to clinical sign as PE, close monitoring vital sign and physical findings of pregnant women for hemodynamic change before and after abortion is mandatory.
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Ayumi Nagai, Saki Kido, Miyuko Suenaga, Shunsuke Kamura, Atsuhiko Saka ...
2022 Volume 58 Issue 1 Pages
169-175
Published: 2022
Released on J-STAGE: May 10, 2022
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Spontaneous septostomy of the dividing membrane is a rare complication of monochorionic diamniotic twin gestation. Although antenatal diagnosis is preferable, the cause of spontaneous septostomy as well as its diagnostic criteria and management have not been clarified. We experienced two cases of spontaneous septostomy in monochorionic diamniotic twins with proximate umbilical cord insertion. Case 1 was diagnosed as spontaneous septostomy at 25 weeks’ gestation, and umbilical cord entanglement could not be confirmed. Case 2 was diagnosed at 20 weeks’ gestation, and cord entanglement was suspected. Selective cesarean section was performed at 34 weeks’ gestation, and all babies were born healthy. The umbilical cord insertion sites were close in both cases, but cord entanglement was observed only in case 2. Proximate umbilical cord insertion was observed in 90% of previous cases, which is characteristic of spontaneous septostomy in monochorionic diamniotic twins and seems useful for diagnosis and risk recognition. Since it is difficult to estimate fetal risk due to the timing of diagnosis or presence of umbilical cord entanglement, antenatal diagnosis of septostomy and careful management are important.
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Yukako Mizuno, Shota Kanbayashi, Miki Yasuda, Miho Masuda, Yukiko Ando ...
2022 Volume 58 Issue 1 Pages
176-180
Published: 2022
Released on J-STAGE: May 10, 2022
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PMCD may improve the circulatory dynamics of pregnant women in cardiopulmonary arrest and assist in resuscitation, and clinicians believe that immediately initiating PMCD after the onset of cardiac arrest reduces mortality for pregnant women. We report a case in which we performed PMCD to save the lives of a pregnant woman and her baby after the occurrence of cardiac arrest during a planned induced labor with epidural analgesia.
A 34-year-old primipara with an uneventful pregnancy and no relevant medical history or allergies developed CPA during planned painless delivery under epidural anesthesia at 39 weeks and 2 days of gestation. During transport to our hospital, she alternated between regaining spontaneous circulation and going into cardiopulmonary arrest. PMCD was started under general anesthesia in the catheter room, and the newborn was delivered 8 min after arrival at the hospital. Bleeding from the uterine incision continued after suture-based closure of the uterine muscle, so we packed gauze into her pelvis and used the vacuum-packing technique for secure wound closure. After the operation, she was admitted to the intensive care unit. Uterine bleeding persisted, so a supracervical hysterectomy was performed. Twenty-two months after the operation, the mother had short-term memory impairments, but she had neither mental nor motor disability. The baby was born with severe asphyxia, so targeted temperature management was implemented. However, 22 months after birth, the baby had severe motor and mental disabilities due hypoxic ischemic encephalopathy. The baby is still on a ventilator. In the future, we aim to introduce a protocol for PMCD based on thorough discussions with multidisciplinary medical teams.
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Yuika Yamaoka, Yuki Abe, Yuto Suzuki, Reiko Nagata, Tomomi Hashimoto, ...
2022 Volume 58 Issue 1 Pages
181-184
Published: 2022
Released on J-STAGE: May 10, 2022
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Maternal infection with cytomegalovirus(CMV)during pregnancy results in a CMV infection rate of 0.3% in the delivered child, which can cause mental retardation, developmental delay, and hearing loss. This paper reports a case in which the mother was diagnosed with CMV infection due to ulcerative colitis(UC)during pregnancy, and the child was diagnosed with asymptomatic congenital CMV infection. The patient was a 31-year-old gravida 2 para 1 woman with concomitant UC. UC symptoms re-emerged around the 28th week of pregnancy, and she started taking oral mesalazine from the 31st week of pregnancy. She was diagnosed with CMV infection via a blood test conducted to exclude CMV enteritis. The patient underwent vaginal delivery at 38 weeks of pregnancy, and her son was diagnosed with asymptomatic congenital CMV infection. Valganciclovir was administered for 6 weeks from age 6 days, and the urine CMV was negative. Mental development was satisfactory in the first year of life. In this case, we were able to diagnose and treat the child promptly for congenital CMV infection after birth.
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Yasuhiro Yanagishita, Yuichiro Matsuoka, Chie Kobayashi, Noriko Hasega ...
2022 Volume 58 Issue 1 Pages
185-189
Published: 2022
Released on J-STAGE: May 10, 2022
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A female infant weighing 2,928 grams was delivered vaginally at 39 weeks of gestation. She began direct breastfeeding from day 1 after birth. On day 16, blisters appeared on the mother’s nipples and around the areola, and papules appeared on the infant’s lips. On day 23, the papules developed into blisters, causing poor feeding and weight loss. After 2 weeks of intravenous acyclovir administration, the infant’s symptoms resolved slowly without neurological sequelae. Since the herpes simplex virus(HSV)-DNA PCR test from the infant’s lip lesion and the HSV antigen test from the mother’s breast lesion were positive, a diagnosis of neonatal HSV disease limited to the skin, eyes, and mouth due to maternal HSV transmission was made. The infant received suppressive oral acyclovir therapy for 6 months and showed normal development at the 17-month evaluation. In this case, recognition of the mother’s breast lesions led to an early diagnosis of neonatal HSV disease. Thus, it is vital for perinatal health care providers to be aware of such conditions and check for herpetic lesions on the breast, extraoral area, and genitalia.
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Mari Yokokawa, Muneichiro Sumi
2022 Volume 58 Issue 1 Pages
190-194
Published: 2022
Released on J-STAGE: May 10, 2022
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Noonan syndrome(NS)is a distinctive facial appearance, lymphoid dysplasia, and a congenital anomaly syndrome characterized by heart anomaly. We herein report a female infant in whom right pleural effusion and hepatomegaly were noted at 29 weeks of gestation with bilateral pleural effusion pooling progressively at 30 weeks. She was delivered via Caesarean section at 32 weeks and 2 days of gestation due to a non-reassuring fetal status. She had symptoms specific to NS, such as significant anasarca, distinctive facial features, myeloproliferative disorder(MPD), and atrial septal defect. In addition, she had also non-specific symptoms, such as hepatomegaly with malformation of the portal vein and elevated liver fibrosis markers. Genetic screening showed a mutation of the PTPN11 gene, which confirmed the diagnosis of NS. Thrombocytopenia, monocytosis and blast cells in the peripheral blood since birth remitted without any treatment and eventually was diagnosed as Noonan syndrome-related myeloproliferative disorder. In cases of fetal pleural effusion and hepatomegaly, chromosomal or genetic abnormalities should be considered as the differential diagnosis. Furthermore, hematologic and liver function including fibrosis markers need to be evaluated to identify any complications such as MPD and liver malformation.
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Tomoki Morikawa, Ryo Itoshima, Ryo Ogawa, Arata Oda, Takehiko Hiroma, ...
2022 Volume 58 Issue 1 Pages
195-199
Published: 2022
Released on J-STAGE: May 10, 2022
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We report a male newborn with congenital syphilis born to a mother whose serological test for syphilis was negative at early gestation. The mother was a 22-year-old, whose serological test for syphilis at 13 weeks of gestation was negative. She had a undiagnosed vulvar ulcer during this period, and it disappeared spontaneously. She delivered a male newborn who weighed 1,536g by emergency cesarean section at 33 weeks and 5 days of gestation. He was transferred to our hospital due to severe birth asphyxia. We suspected congenital infection and commenced treatment with ampicillin(ABPC)and cefotaxime(CTX)on admission. As he had saddle nose, epidermolysis and hepatosplenomegaly, we checked his fluorescent treponemal antibody-absorption(FTA-ABS) IgM, which proved to be positive. We made a diagnosis of congenital syphilis, and we changed antibiotic to benzylpenicillin(PCG)and treated him for 10 days.
The number of congenital syphilis and syphilis among young women has been increasing in Japan recently. Maternal serological syphilis test is recommended only at early gestation, but it is necessary to pay attention to the possibility of syphilis infection in the second trimester.
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Naofumi Hanyu, Shonosuke Nara, Tomoko Maeda, Midori Nishibata, Maria N ...
2022 Volume 58 Issue 1 Pages
200-204
Published: 2022
Released on J-STAGE: May 10, 2022
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The patient was a preterm infant born at 31 weeks and 6 days, weighing 1,816 g, admitted to the NICU for general management.
Phototherapy for unconjugated hyperbilirubinemia was initiated on day 2 and discontinued on day 3, however, the bilirubin rose again repeatedly, requiring intermittent phototherapy for about 2 months.
Based on the clinical course, bilirubin UDP-glucuronosyltransferase(UGT1A1)gene mutation was suspected, and phenobarbital(PB)2 mg/kg/d was started from day 48.
Afterwards, the bilirubin level decreased and the patient was discharged on day 64(the infant was 41 weeks and 0 days old).
UGT1A1 gene analysis revealed that the affected child was homozygous for a missense variant of G71R(Glu71Arg). Genetic test should be considered when breast milk jaundice due to UGT1A1 gene mutation is suspected. Free bilirubin levels(unbound bilirubin: UB)can be a useful marker whether to treat the case of prolonged jaundice.
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Momoko Sawamura, Chieko Matsushima, Ritsuko Kitamura, Jun Iio, Yoshino ...
2022 Volume 58 Issue 1 Pages
205-210
Published: 2022
Released on J-STAGE: May 10, 2022
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For the patient, Giant congenital melanocytic nevus(GCMN)was found in the whole body, and hemangioma and nevus sebaceous were found in head and neck region at birth. Magnetic resonance imaging(MRI)of the head was performed after hospitalization and the high signal intensity area(HSIA)which thought to be melanin deposition, was observed in the cerebellum and brainstem at T1-weighted images. As a result, she was diagnosed with neurocutaneous melanosis. Since anemia and thrombocytopenia were progressed, daily blood transfusions were required. The contrast-enhanced computed tomography(CT)scan, the contrast-enhanced MRI, and the abdominal ultrasonography(AUS)were undertaken to find the cause of pancytopenia. The results showed hepatosplenomegaly without hemangioma and the suspicion of lymphoma in multiple regions including bilateral neck region, mediastinum, retroperitoneum and mesentery. It was difficult to select the effective treatment due to the difficulty of identifying the cause, so it did not save lives. Considering autoimmune diseases, gamma globulin was given to the patient. While it did not work, steroid pulse therapy was effective temporarily. The clinical course of it will be described.
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Eisuke Fukama, Tomoaki Nomura, Daisuke Hatanaka, Michiko Kusakari, Hid ...
2022 Volume 58 Issue 1 Pages
211-215
Published: 2022
Released on J-STAGE: May 10, 2022
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A female infant was born at 38 week’s gestation with a birth weight of 3, 188 g. She was treated with nasal CPAP after birth for transient tachypnea of the newborn and pharyngeal collapse associated with glossoptosis from the 11th day of life. At age 20 days, her right parotid region became swollen and red and appeared sore to the touch. She was diagnosed as having acute parotitis and immediately started on intravenous administration of cefazolin. Staphylococcus aureus was isolated from her blood and near the Stensen duct. Although acute suppurative parotitis in neonates is rare, this is the first report, to our knowledge, of the condition in a neonate receiving CPAP treatment.
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Chihiro Tano, Seiji Hayashi, Maiko Matsusawa, Kaname Matsusawa
2022 Volume 58 Issue 1 Pages
216-220
Published: 2022
Released on J-STAGE: May 10, 2022
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A male infant weighing 2,679g was delivered vaginally at 39 weeks and 4 days of gestation. He was the surviving co-twin of a monochorionic diamniotic(MD)twin pregnancy achieved by natural conception, while death of the other fetus occurred at 14 weeks of gestation. The extensive skin defect on his abdomen was noticed at birth in the NICU was diagnosed as aplasia cutis congenita and was initially covered using Vaseline-impregnated gauze as a conservative treatment option. On the fourth day of life, treatment involved AQUACEL Ag® due to the persistent yellow exudate around the lesion area since the third day of life. Subsequently, the exudate ceased, as re-epithelialization of the lesion occurred; therefore, the patient was discharged on the 19th day of life. In the following month, the lesion eventually cicatrized. This is a rare but noteworthy condition which may present with the surviving co-twin of a monochorionic diamniotic(MD)twin pregnancy with vanishing twin syndrome. In this case, maintaining a moist environment and applying AQUACEL Ag® were both effective.
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Michiko Ito, Tatenobu Goto, Takanori Ota, Megumi Suyama, Tomoaki Maji, ...
2022 Volume 58 Issue 1 Pages
221-225
Published: 2022
Released on J-STAGE: May 10, 2022
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This case report is a neonate with hypophosphatasia(HPP). She has respiratory problems immediately after birth, and developed vitamin B6-dependent seizures from day 13. She was diagnosed with severe perinatal hypophosphatasia because she had significant low serum ALP level, thinning of ribs and rickets-like changes in the limbs.
Seizures and abnormal brain waves rapidly disappeared after intravenous administration of pyridoxine. She received ALP enzyme replacement therapy(ERT)from day 22. Respiratory failure improved and she could be weaned off ventilator at 2 months of age. In addition, bone mineralization improved and rickets-like changes mostly disappeared at 3 months of age. She left the hospital at 5 months of age. Most of the perinatal severe HPP have reported homozygous mutations in the responsible gene. But she had a heterozygous mutation in c.1559delT and should be noted. Although the clinical course of HPP can be predicted to some extent from the ALPL variants, the severity of symptoms may differ even in patients with the same variants.
Additional tests may identify other genetic modifiers. Future physical growth and development, and dental follow-up are important for evaluating the relationship between genotype and clinical course.
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