This Research Topic highlights high-quality Case Reports in Neonatology. Our Case Reports aim to highlight unique cases of patients that present with an unexpected/unusual diagnosis, treatment outcome, or clinical course. Case Reports provide insight into the differential diagnosis, decision making, and clinical management of unusual cases and are a valuable educational tool. This Research Topic will only accept Case Report submissions, reports must be original with the aim to significantly advance the field to be considered. All Case Reports should contain “Case Report" in the title to be considered for this Research Topic. We welcome all case reports covering aspects of Neonatology.

Medical University of Vienna

Saarland University Hospital

University of Health Sciences (Turkey)

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Frontiers in Pediatrics

Neonatology

Case Reports in Neonatology 2022

Editorial on the Research Topic <a href="https://www.frontiersin.org/research-topics/32262/case-reports-in-neonatology-2022">Case reports in neonatology 2022</a>This Research Topic publishes high-quality case reports and case series in the field of neonatology, ranging from lethal poisoning, adverse drug reactions, congenital pathologies, neonatal metabolic disease, survival and care practices of periviable births to malignancies in newborns (<a href="https://doi.org/10.3389/fped.2023.877550">Baptiste et al.</a> <a href="https://doi.org/10.3389/fped.2022.913050">Cheng et al.</a> <a href="https://doi.org/10.3389/fped.2022.979763">Gaertner et al.</a> <a href="https://doi.org/10.3389/fped.2022.991488">Boddu et al.</a> <a href="https://doi.org/10.3389/fped.2022.993922">Chen et al.</a> <a href="https://doi.org/10.3389/fped.2022.905089">Yan et al.</a>). Case reports provide important insights into the differential diagnosis, decision making, and clinical management of unusual cases. They provide a valuable tool for (a) recognizing new or rare diseases, (b) evaluating therapeutic effects, adverse events, and costs of interventions; as well as (c) improving problem-based medical education in a real-world clinical setting, Conversely, randomized clinical trials generate evidence for the efficacy of interventions in a controlled setting. Undoubtedly, both types of reports are necessary and complementary in modern medicine. Of note, case reports today make up a substantial proportion of articles published in medical journals, and they continue to further our understanding of a great variety of diseases. Consequently, case reports are an important contributor to and driver of medical progress. The CARE guidelines help increase the completeness, accuracy, and transparency of published case reports (<a href="#B1">1</a>).The importance of having access to case reports in our continuously changing modern world is key in the provision of best clinical care. In the immortal words of the famous Canadian physician, first Professor of Medicine and founder of the Medical Service at Johns Hopkins Hospital, Sir William Osler (July 12, 1849 to December 29, 1919): “Always note and record the unusual … Publish it. Save it on a permanent record as a short, concise note. Such communications are always of value” (<a href="#B2">2</a>).No doubt, the value of case reports may be underestimated compared to other publications that are more detailed and supported by evidence (eg, randomized controlled clinical trials). Nevertheless, this experience-based informative method has nowadays been transformed into an accepted academic form of communication and publication, thus contributing to the rapid dissemination of medical knowledge to a wide medical professional audience, including neonatologists.Therefore, we hope that this Special Collection is both informative and stresses the importance and role of case reports in the field of neonatology, and will also translate into improved neonatal care practices around the world.This editorial was jointly conceptualized and written together. All authors contributed to the article and approved the submitted version.The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.All claims expressed in this article are solely those of the authors and do not necessarily represent those of their affiliated organizations, or those of the publisher, the editors and the reviewers. Any product that may be evaluated in this article, or claim that may be made by its manufacturer, is not guaranteed or endorsed by the publisher.

Editorial: Case reports in neonatology 2022

During a 2-year period, eight cases of a distinct illness were seen among 1,424 neonates admitted to a newly established neonatal care unit in southern Haiti. The newborns presented with a picture of sepsis with shock, vomiting, hypotonia, lethargy, and abdominal distention. Five cases proved fatal and another case left the hospital against advice in extremis with little chance of survival. In each case, the illness was associated with a history of ingestion of teas that included castor oil, known as lok in Haitian Creole. The presumptive cause of the illness was established by the presence of a dark, oily substance in drainage from the nares and nasogastric tubes and by subsequent admission on direct questioning of the caregivers, who said that the infants had been given large amounts of lok. The castor oil tea had been given to three infants in the immediate neonatal period where its use is attributed to encouraging the passage of meconium. The five remaining infants were between 15 and 30 days of life when they were given lok shortly before admission to the neonatal unit for treatment of an undefined illness. All of them were term infants with no identified risks at birth. As nasogastric tubes are not routinely placed in sick neonates, and the parents did not volunteer information about lok administration, the practice may be more widespread than that recorded here. Although our data are confined to observations in Haiti, the use of traditional medicines is a globally widespread phenomenon. Attention must be drawn to the potential toxicity of such preparations and means found to ban their use in neonates.

During a 2-year period, eight cases of a distinct illness were seen among 1,424 neonates admitted to a newly established neonatal care unit in southern Haiti. The newborns presented with a picture of sepsis with shock, vomiting, hypotonia, lethargy, and abdominal distention. Five cases proved fatal and another case left the hospital against advice in extremis with little chance of survival. In each case, the illness was associated with a history of ingestion of teas that included castor oil, known as lok in Haitian Creole. The presumptive cause of the illness was established by the presence of a dark, oily substance in drainage from the nares and nasogastric tubes and by subsequent admission on direct questioning of the caregivers, who said that the infants had been given large amounts of lok. The castor oil tea had been given to three infants in the immediate neonatal period where its use is attributed to encouraging the passage of meconium. The five remaining infants were between 15 and 30 days of life when they were given lok shortly before admission to the neonatal unit for treatment of an undefined illness. All of them were term infants with no identified risks at birth. As nasogastric tubes are not routinely placed in sick neonates, and the parents did not volunteer information about lok administration, the practice may be more widespread than that recorded here. Although our data are confined to observations in Haiti, the use of traditional medicines is a globally widespread phenomenon. Attention must be drawn to the potential toxicity of such preparations and means found to ban their use in neonates.

The dangers of castor oil tea in neonates in rural Haiti: A case report

BackgroundData from the China Neonatal Network currently shows that the survival rate of very preterm infants in China has improved in recent years. However, due to the unequal economic and medical development of each city in China, the national data do not completely represent the level of neonatal care in the metropolitan areas. Though many studies have published their short- or long-term survival, very few have described the care practices and the course of stay of these neonates in detail. Our objective was to examine the survival and clinical practices among preterm infants born at <24 weeks’ gestational age (GA) in a high-income city in China, from 2015 to 2021.</sec>MethodsRetrospective study of preterm infants <24 weeks GA in a level 3 neonatal intensive care unit in China, over a period of 7 years (2015–2021). Care practices in neonatal intensive care units (NICU) and short- and long-term survival were measured.</sec>ResultsA total of 32 periviable infants were included, with a median GA of 23.0 weeks and mean (SD) birth weight of 497 (94) g; 17 infants (53.1%) were female. While none of the infants born at 21 weeks of gestation survived until discharge, the survival rates were 25.0% (3 of 12) for infants born at 22 weeks and 58.8% (10 of 17) at 23 weeks. Antenatal corticosteroids were used in 56.3%, and 100% were vaginal birth. In the delivery room, surfactant was prescribed for 46.9% of the infants, and postnatal dexamethasone (≥2 courses) was prescribed to 61.5% of the infants. Logistic regression analysis showed that Apgar score at 5 minutes (OR = 2.007, 95% CI, 1.031 to 3.906, P < 0.05) increased the risk of death, while the increase in gestational age (OR = 0.238, 95% CI, 0.060–0.936, P < 0.05), antenatal use of steroids (OR = 0.287, 95% CI, 0.106–0.778, P < 0.01), and premature rupture of membranes (OR = 0.141, 95% CI, 0.024 −0.847, P = 0.032) could decrease the risk. No or mild neurodevelopmental impairment in surviving infants was 76.9% (10 of 13).</sec>ConclusionsAlthough the survival rate of periviable infants was shown to be improved in our study, there is still much room for improvement, and active follow-up information should be conducted.</sec>

Background: Data from the China Neonatal Network currently shows that the survival rate of very preterm infants in China has improved in recent years. However, due to the unequal economic and medical development of each city in China, the national data do not completely represent the level of neonatal care in the metropolitan areas. Though many studies have published their short- or long-term survival, very few have described the care practices and the course of stay of these neonates in detail. Our objective was to examine the survival and clinical practices among preterm infants born at &lt;24 weeks’ gestational age (GA) in a high-income city in China, from 2015 to 2021.Methods: Retrospective study of preterm infants &lt;24 weeks GA in a level 3 neonatal intensive care unit in China, over a period of 7 years (2015–2021). Care practices in neonatal intensive care units (NICU) and short- and long-term survival were measured.Results: A total of 32 periviable infants were included, with a median GA of 23.0 weeks and mean (SD) birth weight of 497 (94) g; 17 infants (53.1%) were female. While none of the infants born at 21 weeks of gestation survived until discharge, the survival rates were 25.0% (3 of 12) for infants born at 22 weeks and 58.8% (10 of 17) at 23 weeks. Antenatal corticosteroids were used in 56.3%, and 100% were vaginal birth. In the delivery room, surfactant was prescribed for 46.9% of the infants, and postnatal dexamethasone (≥2 courses) was prescribed to 61.5% of the infants. Logistic regression analysis showed that Apgar score at 5 minutes (OR = 2.007, 95% CI, 1.031 to 3.906, P &lt; 0.05) increased the risk of death, while the increase in gestational age (OR = 0.238, 95% CI, 0.060–0.936, P &lt; 0.05), antenatal use of steroids (OR = 0.287, 95% CI, 0.106–0.778, P &lt; 0.01), and premature rupture of membranes (OR = 0.141, 95% CI, 0.024 −0.847, P = 0.032) could decrease the risk. No or mild neurodevelopmental impairment in surviving infants was 76.9% (10 of 13).Conclusions: Although the survival rate of periviable infants was shown to be improved in our study, there is still much room for improvement, and active follow-up information should be conducted.

Survival and care practices of periviable births of <24 weeks’ gestation—a single center retrospective study in China, 2015–2021

BackgroundBoth calcium (Ca) and phosphorus (P) are needed to prevent and treat metabolic bone disease (MBDP). However, the predominant focus of many treating neonatologists lies in supplementing P and vitamin D. In this report, we describe a VLBW infant with severe MBDP due to inadequately treated calcium deficiency and discuss the need to recognize this entity.</sec>Case details and managementA 25-week, 700 gm baby boy had chronic lung disease and necrotizing enterocolitis. He received total parenteral nutrition, budesonide, furosemide, and caffeine. With high serum alkaline phosphatase (ALP: 1,700 IU/L) and low P (2.8 mg/dl), MBDP was diagnosed at 12 weeks, started on oral phosphate, human milk fortifier, and 1,400 IU/d of vitamin D before discharge. He was readmitted 2 weeks later with decreased lower limb mobility and respiratory distress. X-rays revealed severe osteopenia and fractures of both femurs. Serum P was 4.6 mg/dl but ALP was high (1,700 IU/L), and Ca was low (6.4 mg/dl). Parathyroid hormone (PTH: 605 pg/ml) and 25-hydroxy Vitamin D (25 OHD > 200 ng/ml) were very high. We discontinued his P and vitamin D, hypocalcemia treated with IV Ca gluconate, later oral Ca citrate, and calcitriol. Phosphate was added after normalization of Ca. Over the next many weeks, X-rays and biochemistry improved.</sec>DiscussionMBDP results from both Ca and P deficiencies, especially in VLBW infants with comorbidities. P supplementation without treating underlying calcipenia can precipitate hypocalcemia and worsen osteopenia with disastrous consequences. In severe calcipenia, active vitamin D might have a role in addition to an appropriate dose of elemental calcium.</sec>

Background: Both calcium (Ca) and phosphorus (P) are needed to prevent and treat metabolic bone disease (MBDP). However, the predominant focus of many treating neonatologists lies in supplementing P and vitamin D. In this report, we describe a VLBW infant with severe MBDP due to inadequately treated calcium deficiency and discuss the need to recognize this entity.Case details and management: A 25-week, 700 gm baby boy had chronic lung disease and necrotizing enterocolitis. He received total parenteral nutrition, budesonide, furosemide, and caffeine. With high serum alkaline phosphatase (ALP: 1,700 IU/L) and low P (2.8 mg/dl), MBDP was diagnosed at 12 weeks, started on oral phosphate, human milk fortifier, and 1,400 IU/d of vitamin D before discharge. He was readmitted 2 weeks later with decreased lower limb mobility and respiratory distress. X-rays revealed severe osteopenia and fractures of both femurs. Serum P was 4.6 mg/dl but ALP was high (1,700 IU/L), and Ca was low (6.4 mg/dl). Parathyroid hormone (PTH: 605 pg/ml) and 25-hydroxy Vitamin D (25 OHD &gt; 200 ng/ml) were very high. We discontinued his P and vitamin D, hypocalcemia treated with IV Ca gluconate, later oral Ca citrate, and calcitriol. Phosphate was added after normalization of Ca. Over the next many weeks, X-rays and biochemistry improved.Discussion: MBDP results from both Ca and P deficiencies, especially in VLBW infants with comorbidities. P supplementation without treating underlying calcipenia can precipitate hypocalcemia and worsen osteopenia with disastrous consequences. In severe calcipenia, active vitamin D might have a role in addition to an appropriate dose of elemental calcium.

Are we undertreating calcium deficiency in metabolic bone disease of prematurity? A case report and review

BackgroundChest wall rigidity is a known side effect of fentanyl use, which is why fentanyl is usually combined with a muscle relaxant such as mivacurium. Verifying endotracheal intubation is difficult in case of a rigid chest wall.</sec>Case presentationWe present the case of a preterm infant (29 completed weeks gestation, birth weight 1,150 g) with a prolonged chest wall rigidity after fentanyl administration for intubation despite adequate doses of mivacurium. This resulted in a pronounced desaturation without any effect on heart rate. Clinically, the infant showed no chest wall movement despite intubation and common tools to verify intubation (including end-tidal carbon dioxide measurement and auscultation) were inconclusive. However, using electrical impedance tomography (EIT), we were able to demonstrate minimal tidal volumes at lung level and thereby, EIT was able to accurately show correct placement of the endotracheal tube.</sec>ConclusionsThis case may increase vigilance for fentanyl-induced chest wall rigidity in the neonatal population even when simultaneously administering mivacurium. Higher airway pressures exceeding 30 mmHg and the use of μ-receptor antagonists such as naloxone should be considered to reverse opioid-induced chest wall rigidity. Most importantly, our data may imply a relevant clinical benefit of using EIT during neonatal intubation as it may accurately show correct endotracheal tube placement.</sec>

Background: Chest wall rigidity is a known side effect of fentanyl use, which is why fentanyl is usually combined with a muscle relaxant such as mivacurium. Verifying endotracheal intubation is difficult in case of a rigid chest wall.Case presentation: We present the case of a preterm infant (29 completed weeks gestation, birth weight 1,150 g) with a prolonged chest wall rigidity after fentanyl administration for intubation despite adequate doses of mivacurium. This resulted in a pronounced desaturation without any effect on heart rate. Clinically, the infant showed no chest wall movement despite intubation and common tools to verify intubation (including end-tidal carbon dioxide measurement and auscultation) were inconclusive. However, using electrical impedance tomography (EIT), we were able to demonstrate minimal tidal volumes at lung level and thereby, EIT was able to accurately show correct placement of the endotracheal tube.Conclusions: This case may increase vigilance for fentanyl-induced chest wall rigidity in the neonatal population even when simultaneously administering mivacurium. Higher airway pressures exceeding 30 mmHg and the use of μ-receptor antagonists such as naloxone should be considered to reverse opioid-induced chest wall rigidity. Most importantly, our data may imply a relevant clinical benefit of using EIT during neonatal intubation as it may accurately show correct endotracheal tube placement.

Case report: Intrapulmonary tidal volumes in a preterm infant with chest wall rigidity

This study reports a case of hepatoblastoma with onset at 30-weeks' gestation and rapid growth rate. The postnatal enhanced CT confirmed an intrahepatic mass with a size of 8.5 cm × 6.6 cm and a clear boundary accompanied by uneven enhancement, displacement, and narrow lumen of the hepatic vein due to compression. The alpha-fetoprotein (AFP) at birth was 1,002,632 ng/ml (normal level 48,406 [±34,718] ng/ml). A diagnosis of congenital hepatoblastoma was established based on the imaging and laboratory outcomes. The infant received chemotherapy of Cisplatin-5 fluorouracil-Vincristine (C5V) on the fourth day after birth. After four courses of C5V, a complete tumor resection was performed, and the postoperative pathology was consistent with mixed epithelial and mesenchymal hepatoblastoma. Four more courses of C5V and one course of C5VD (C5V plus doxorubicin) followed the surgery. Infectious diarrhea and acute kidney injury (stage I) occurred during chemotherapy, which recovered after anti-infection and symptomatic treatment. The patient is currently 2 years old and still in complete remission. In this case, the onset of hepatoblastoma was early, and the tumor grew rapidly, resulting in an obvious compression effect. Chemotherapy was started early after birth, and the curative effect was satisfactory, suggesting that the hepatoblastoma based on clinical diagnosis with rapid tumor progression and severe dysfunction of surrounding organs caused by compression should undergo chemotherapy as soon as possible if a pathological diagnosis cannot be obtained temporarily, which also plays an important role in improving the complete resection rate of intraoperative tumor and reducing the recurrence rate of postoperative tumor.

This study reports a case of hepatoblastoma with onset at 30-weeks&apos; gestation and rapid growth rate. The postnatal enhanced CT confirmed an intrahepatic mass with a size of 8.5 cm &#xd7; 6.6 cm and a clear boundary accompanied by uneven enhancement, displacement, and narrow lumen of the hepatic vein due to compression. The alpha-fetoprotein (AFP) at birth was 1,002,632 ng/ml (normal level 48,406 [&#xb1;34,718] ng/ml). A diagnosis of congenital hepatoblastoma was established based on the imaging and laboratory outcomes. The infant received chemotherapy of Cisplatin-5 fluorouracil-Vincristine (C5V) on the fourth day after birth. After four courses of C5V, a complete tumor resection was performed, and the postoperative pathology was consistent with mixed epithelial and mesenchymal hepatoblastoma. Four more courses of C5V and one course of C5VD (C5V plus doxorubicin) followed the surgery. Infectious diarrhea and acute kidney injury (stage I) occurred during chemotherapy, which recovered after anti-infection and symptomatic treatment. The patient is currently 2 years old and still in complete remission. In this case, the onset of hepatoblastoma was early, and the tumor grew rapidly, resulting in an obvious compression effect. Chemotherapy was started early after birth, and the curative effect was satisfactory, suggesting that the hepatoblastoma based on clinical diagnosis with rapid tumor progression and severe dysfunction of surrounding organs caused by compression should undergo chemotherapy as soon as possible if a pathological diagnosis cannot be obtained temporarily, which also plays an important role in improving the complete resection rate of intraoperative tumor and reducing the recurrence rate of postoperative tumor.

Case Report of Congenital Hepatoblastoma With the Onset at 30-Weeks' Gestation

We summarized two cases of congenital factor V deficiency (FVD) associated with a novel F5 mutation, and analyzed the relationship of the clinical features and genetic characteristics in congenital FVD. Case 1 was a female newborn infant with remarkable bleeding who died of severe intracranial hemorrhage on day 42 after birth. She had significant prolongation of prothrombin time (PT) and activated partial thromboplastin time (APTT). The percentage activity of FV (PFV) was lower than 3% in case 1. The mother of case 1 showed no tendency to bleed. She had mild prolongation of PT and APTT. The PFV was only 43%. Both cases were found to have the same novel mutation in F5, which was c.5419G>A (p.Ala1807Thr) in exon 16. The variant in case 1 was inherited from the mother of case 1. Whole-exome sequencing (WES) also found a splice site mutation: a 103 Mb maternal uniparental disomy (UPD) of 1q21.1-qter in case 1, in which the F5 gene is located in this segment. So case 1 was homozygote and the mother of case 1 was heterozygote. The novel mutation of F5 was predicted to be harmful by bioinformatics software including Sorting Intolerant From Tolerant (SIFT), Polyphen2, LRT, and Mutation Taster. In summary, c.5419G>A (p.Ala1807Thr) in exon 16 of F5 is a pathogenic mutation, which causes severe congenital FVD in homozygote patients.

We summarized two cases of congenital factor V deficiency (FVD) associated with a novel F5 mutation, and analyzed the relationship of the clinical features and genetic characteristics in congenital FVD. Case 1 was a female newborn infant with remarkable bleeding who died of severe intracranial hemorrhage on day 42 after birth. She had significant prolongation of prothrombin time (PT) and activated partial thromboplastin time (APTT). The percentage activity of FV (PFV) was lower than 3% in case 1. The mother of case 1 showed no tendency to bleed. She had mild prolongation of PT and APTT. The PFV was only 43%. Both cases were found to have the same novel mutation in F5, which was c.5419G&gt;A (p.Ala1807Thr) in exon 16. The variant in case 1 was inherited from the mother of case 1. Whole-exome sequencing (WES) also found a splice site mutation: a 103 Mb maternal uniparental disomy (UPD) of 1q21.1-qter in case 1, in which the F5 gene is located in this segment. So case 1 was homozygote and the mother of case 1 was heterozygote. The novel mutation of F5 was predicted to be harmful by bioinformatics software including Sorting Intolerant From Tolerant (SIFT), Polyphen2, LRT, and Mutation Taster. In summary, c.5419G&gt;A (p.Ala1807Thr) in exon 16 of F5 is a pathogenic mutation, which causes severe congenital FVD in homozygote patients.