A Case of Functional Testicular Torsion Caused by Incarcerated Inguinal Hernia in a Newborn

Testicular torsion in neonates is a urologic emergency with an incidence of 6.1 per 100,000 live births. Incarcerated inguinal hernia is also an emergency with an incidence of approximately 6% to 31% in children with inguinal hernia. Functional testicular torsion from an incarcerated inguinal hernia is a rare event in children, often not considered in the initial evaluation. A 19-day-old newborn boy was admitted to our neonatal intensive care unit after visiting a primary clinic. He presented with inconsolable crying and right scrotal swelling. Upon initial physical examination, a hard palpable mass and swelling was found in the inguinoscrotal region, accompanied with redness and warmth of the scrotum. Scrotal ultrasound with color Doppler showed inguinal hernia with strangulation and scanty blood flow to the testis. The patient underwent a right orchiectomy and partial resection of the affected small bowel. Surgical findings confirmed a testicular torsion and incarcerated inguinal hernia with testis with small bowel necrosis. Here, we report a rare case of a newborn with functional testicular torsion as a result of prolonged spermatic cord compression due to incarcerated inguinal hernia.

Case of a Male Newborn with Incontinentia Pigmenti Initially Misdiagnosed as a Recurrent Skin Infection

Incontinentia pigmenti (IP), also known as Bloch-Sulzberger syndrome, is a rare X-linked dominant disorder that is generally lethal to males and almost always leads to death in utero. This disorder is a genodermatosis with characteristic cutaneous lesions and manifestations affecting the eyes, teeth, hair, and central nervous system. Genodermatosis is a hereditary disease caused by mutations in the nuclear factor-kappa B essential modulator gene mapped to chromosome Xq28. This gene encodes a variety of cytokines and chemokine regulators and is indispensable for protecting cells from tumor necrosis factor-induced apoptosis. Here we describe a case of male newborn with vesiculobullous cutaneous lesions over the left thigh and leg. We first considered the cutaneous lesions a skin infection, as they improved with intravenous antibiotics. However, recurrence and the need for repeated hospitali zations made us consider the differential diagnosis of IP, for which we performed a skin biopsy and chromosome analysis. The histology results were compatible with IP, that is, eosinophilic infiltration in the dermis and epidermis, and individual cell dyskeratinization. The chromosome analysis result was a normal 46, XY karyotype.Here we report the case of a male newborn with IP that manifested as multiple vesiculobullous skin lesions and was initially misdiagnosed as a recurrent skin infection.

Comparison of Safety of Sedatives Versus General Anesthesia in Laser Therapy for Retinopathy of Prematurity

PURPOSE: Laser therapy for retinopathy of prematurity (ROP) is commonly performed under general anesthesia (GA). However, the use of GA for laser therapy in neonates who have already undergone invasive ventilation may lead to postoperative complications such as severe apnea or the development of ventilator dependency. This study aimed to examine the safety of administering only sedatives instead of GA in extremely low birth weight (ELBW) infants, who are the usual recipients of laser therapy for ROP. METHODS: Among ELBW infants who were admitted to the neonatal intensive care unit (NICU) at Samsung Medical Center between January and December 2012, we studied 30 patients treated with laser therapy for ROP. RESULTS: The mean gestational age of the patients was 24.6±1.9 weeks, with a mean birth weight of 646±140 g. The mean age and weight of patients at the time of laser therapy for ROP was 36.3±2.3 weeks and 1,470±423 g. In terms of sedatives, 14 patients (46.7%) were administered chloral hydrate alone, 14 (46.7%) were administered a combination of chloral hydrate and midazolam, one was administered midazolam alone, and one received fentanyl. Prior to laser therapy, 16 patients (53.5%) had established self-respiration, 13 (43.3%) relied on non-invasive ventilation and one patient relied on invasive mechanical ventilation. Following laser therapy, two patients who initially had exhibited self-respiration required respiratory assistance via non-invasive positive pressure ventilation and no patient required intratracheal intubation. CONCLUSIONS: We conclude that the use of sedatives may be safe for ELBW infants who undergo laser therapy for ROP.

Prophylactic versus Early Rescue Surfactant Treatment in Preterm Infants Born at Less than 30 Weeks Gestation or with Birth Weight Less than or Equal 1,250 Grams

Prophylactic surfactant is known to be effective to reduce chronic lung disease in preterm infants compared with rescue surfactant treatment. In Korea, early prophylactic surfactant therapy was introduced in 2011. However, recently, the increased utilization of antenatal steroids and early stabilization through continuous positive airway pressure (CPAP) in the delivery room may have changed the risks and benefits of prophylactic surfactant therapy of infants at high risk of respiratory distress syndrome (RDS). We compared the effects and safety of prophylactic surfactant therapy (within 30 minutes after birth) and early selective surfactant therapy (within 3 hours after birth) in preterm infants born at < 30 weeks gestation or with birth weight ≤ 1,250 g. The clinical data of 193 infants in period 1 (from 2008 to 2010, early selective surfactant therapy group) were collected retrospectively; those of 191 infants in period 2 (from 2012 to 2014, prophylactic surfactant therapy group) were collected prospectively. Compared to period 1, the rate of intubation and surfactant use were significantly increased in period 2. The use of multiple doses of surfactant in period 2 was significantly increased compared with period 1. Despite more invasive and aggressive management in period 2, there was no difference in the duration of mechanical ventilation, the incidence of bronchopulmonary dysplasia (BPD) or death, and the risk of other adverse neonatal outcomes between the 2 groups. In conclusion, the benefit of prophylactic surfactant therapy in infants treated under current practices is no longer clear compared to early selective surfactant therapy.

Delayed Cord Clamping Compared with Cord Milking in Preterm Neonates

PURPOSE: To evaluate the safety and feasibility of delayed cord clamping compared with umbilical cord milking in premature infants less than 32 weeks of gestation. METHODS: This study was performed by 1:2 case-control match. Infants received delayed cord clamping (DCC) for one minute (DCC group, n=10, May 2014-October 2015) were compared with perinatal factors-matching controls, who received umbilical cord milking (CM, CM group, n=20, May 2014-October 2015) or who received immediate cord clamping (ICC, ICC group, n=20, January 2008-December 2008). The primary outcome was hematocrit during the first 28 days. Secondary outcomes included delivery room management, selected neonatal morbidities and mortality. RESULTS: Baseline characteristics were comparable in all the three groups. The median hematocrit level at 1st day and 3rd day was significantly higher in the DCC group (54.3±6.2%, 53.6±5.6%) as compared with the CM group (48.0±7.7%, 43.2±7.8%) or ICC group (47.2±7.5%, 45.8±6.3%). The DCC group had reductions in red blood cell transfusion within the first two weeks of life compared to the CM group (10% vs. 50%, P=0.03). The DCC group compared to the CM group had no increment in respiratory intervention in the delivery room and hypothermia on admission. There was no difference between DCC and CM in mortality, intraventricular hemorrhage, bronchopulmonary dysplasia, necrotizing enterocolitis, severe retinopathy of prematurity and sepsis. CONCLUSION: Delayed cord clamping for 1 minute in preterm infants may be a safe and feasible method to increase initial hematocrit and reduce transfusion compared with umbilical cord milking.

Clinical Characteristics of Early Onset Sepsis in Micropreemie Born at 25 or Less than 25 Weeks of Gestational Age / 대한주산의학회잡지

PURPOSE: The aim of this study is to determine the clinical characteristics of early onset sepsis (EOS) in micropreemie. METHODS: We retrospectively reviewed medical records of 107 extremely preterm infants born at 25 or less than 25 weeks of gestation and admitted to the neonatal intensive care unit of Samsung Medical Center from January 2013 to August 2015. Infants were divided into two groups based on the presence of culture-proven EOS in the first 7 days of life. Retrospective analysis of perinatal factors and laboratory findings within the first week of life was done between two groups. We compared the neonatal outcomes among two groups. RESULTS: Culture-proven EOS was diagnosed in 11 of 107 infants (10.3%). Main pathogen of EOS was Staphylococcus epidermidis (45.5%). There were no significant differences between control group and EOS group in gestational age, birth weight, Apgar score, delivery type and pathologic chorioamnionitis. Among 11 infants with EOS, 9 showed fetal tachycardia (P=0.001). And EOS group presented lower platelet count at 3rd day and 7th day of life than that of control group (P=0.033, P=0.045). Neonatal outcomes in EOS group were compatible with control group. Main cause of death was sepsis in EOS group. CONCLUSION: In micropreemie, EOS is important factor of mortality. Our data suggest that fetal tachycardia and low platelet count during the first 7 days of life were associated with EOS.

Clinical Characteristics of Early Onset Sepsis in Micropreemie Born at 25 or Less than 25 Weeks of Gestational Age / 대한주산의학회잡지

PURPOSE: The aim of this study is to determine the clinical characteristics of early onset sepsis (EOS) in micropreemie. METHODS: We retrospectively reviewed medical records of 107 extremely preterm infants born at 25 or less than 25 weeks of gestation and admitted to the neonatal intensive care unit of Samsung Medical Center from January 2013 to August 2015. Infants were divided into two groups based on the presence of culture-proven EOS in the first 7 days of life. Retrospective analysis of perinatal factors and laboratory findings within the first week of life was done between two groups. We compared the neonatal outcomes among two groups. RESULTS: Culture-proven EOS was diagnosed in 11 of 107 infants (10.3%). Main pathogen of EOS was Staphylococcus epidermidis (45.5%). There were no significant differences between control group and EOS group in gestational age, birth weight, Apgar score, delivery type and pathologic chorioamnionitis. Among 11 infants with EOS, 9 showed fetal tachycardia (P=0.001). And EOS group presented lower platelet count at 3rd day and 7th day of life than that of control group (P=0.033, P=0.045). Neonatal outcomes in EOS group were compatible with control group. Main cause of death was sepsis in EOS group. CONCLUSION: In micropreemie, EOS is important factor of mortality. Our data suggest that fetal tachycardia and low platelet count during the first 7 days of life were associated with EOS.

Comparison of Clinical Manifestation and Prognosis of Preterm and Full-Term Infants with Down Syndrome

PURPOSE: To investigate and compare the clinical manifestation and prognosis of preterm and full-term infants with Down syndrome (DS). METHODS: We retrospectively reviewed 80 patients diagnosed with DS and confirmed by chromosomal study at the Samsung Medical Center between January 1994 and July 2014. Data on demographic characteristics, associated anomalies, treatment, prognosis and cause of death were compared between preterm and full-term DS infants. RESULTS: Of the 80 confirmed DS patients, there were 49 (61%) full-term and 31 (38%) preterm DS infants. The mean gestational age of full-term DS infants was 38(+1)+/-0(+2) weeks (range, 37(+0)-40(+0) weeks) and the mean birth weight was 3,007+/-418 g (range, 1,930-4,100 g). The mean gestational age of preterm infants was 34(+1)+/-2(+1) weeks (range, 29(+1)-36(+6) weeks) and the mean birth weight was 2,181+/-598 g (range, 890-3,500 g). There were no differences in demographics, associated anomalies, mortality or related factors, or the rate of active treatment between full-term and preterm DS infants. CONCLUSION: In this single center study, the mortality rate of preterm DS infants was comparable to that of full-term DS infants. Larger national cohort studies might be needed to further investigate the prognosis of preterm DS infants.

A 3-year-old girl with Graves' disease with literature review

Graves' disease, the main cause of hyperthyroidism in the pediatric age group, is very rare in children younger than 4 years old but can seriously interfere with growth and development if not recognized and treated. Here we report a case of a 3-year-old girl with Graves' disease who presented with goiter, exophthalmos, heat intolerance, and hyperactivity. At her first visit, her serum concentrations of triiodothyronine (T3) and free thyroxine (free T4) were normal, whereas that of thyroid-stimulating hormone (TSH) was decreased. Antimicrosomal antibody was 7,053.94 IU/mL, and TSH-binding inhibitory immunoglobulin was 31.62%. A thyroid scan showed diffuse enlargement with markedly increased uptake of both thyroid glands. Although T3 and free T4 levels were initially normal, she developed hyperthyroidism 3 months later. She was finally diagnosed with Graves' disease and treated with methimazole for 6 months. This is the first report of Graves' disease in children younger than 4 years old in Korea.

A Comparison of Clinical Manifestations of Patients with Tsutsugamushi Disease between Children and Adults / 소아감염

PURPOSE: We compared the clinical manifestations of patients with tsutsugamushi disease between children and adults. METHODS: From January 2003 to December 2012, 768 patients diagnosed with tsutsugamushi disease were retrospectively reviewed, and the clinical characteristics, laboratory findings, and complications were compared between children and adults. RESULTS: No patterns of annual increases in the number of patients were noted in both children and adults. The higher incidences occurred in October and November respectively. By gender, male outnumbered female in children, but the opposite trend was seen in adults. By residential area, the urban distribution of children was higher than that of adults. Rashes (P=0.001) and eschar (P=0.004) were more common in children, while myalgia was more common in adults. Children had a high prevalence of anemia (P=0.041), and low incidence rates of thrombocytopenia, abnormal liver and renal function. Children yielded better results in the duration of their hospital stay and the incidence of complications (P<0.001). A comparison of the therapeutic effects of doxycycline and macrolide antibiotics, which was performed only on the children, did not reveal any significant differences. CONCLUSION: Compared to adults, children had higher incidence rates of male patients and more often suffered from rashes and eschar. Children yielded better results in the laboratory findings and duration of the hospital stay and complications. Therefore, when children are suspected to have tsutsugamushi disease, especially during its peak occurrence period, detailed physical examination and serological test should be performed to ensure a prompt diagnosis, and the use of macrolide antibiotics, which have fewer side effects, is expected to yield the same therapeutic effects.