BiliNorm is based on the Indonesian National Guideline on Hyperbilirubinemia [6]. It can be accessed at www.bilinorm.babyhealthsby.org with two language options: Indonesian and English [7]. When opening the BiliNorm app, users have to fill in the following patient data: gestational age (weeks), date and time of birth, date and time of record, birth weight (g), TSB level (mg/dL or µmol/L) and risk factors (Figure 1A). The risk factors incorporated in this tool were adapted from the American Academy of Pediatrics (AAP) guidelines [8] and include ABO/Rhesus incompatibility, haemolysis (G6PD deficiency or spherocytosis), other illnesses (asphyxia, infection) and hypoalbuminemia (< 30 mg/L). When “no risk factors” is selected, the case is regarded as uncomplicated hyperbilirubinemia. When “unknown risk factors” is selected, the patient is categorized as having risk factors other than those listed or as not being able to check the risk factors. After these data are provided, the actual TSB level of the infant is shown in the TSB nomogram, which displays treatment thresholds for PT and exchange transfusion over time. Advice for the caretaker regarding how to treat the infant is provided as follows: no treatment, start PT or immediately start intensive PT and consider exchange transfusion. Figure 1 shows an example of the data entered into the BiliNorm app for a preterm infant born at 33 weeks of gestation. She was admitted to a neonatal unit with jaundice at day two. Her birth weight was 2100 grams. Blood tests showed ABO incompatibility and a TSB of 17.2 mg/dL. After all data were entered, the results section showed the treatment thresholds for PT and exchange transfusion for this patient. The recommendation for the paediatrician for this patient is to immediately start intensive PT and consider exchange transfusion.
Different nomograms are included in the BiliNorm app: one for infants with a gestational age of more than 35 weeks and four for preterm infants. Infants born before 35 weeks have different thresholds than term babies according to the new Indonesian National Guideline on Hyperbilirubinemia. In Indonesia, it is often difficult to determine exact gestational age; therefore, guidelines for preterm babies are categorized by birth weight categories: ≤1000 g, 1001-1499 g, 1500-1999 g, and >1999 g.
In addition to providing advice about potential treatments for hyperbilirubinemia, information is provided about the risks of complications due to acute bilirubin encephalopathy (ABE). This information is based on the modified Bilirubin Induced Neurological Dysfunction-Modified (BIND-M) scoring, adapted from Radmacher et al. [9]. Mental status, muscle tone, altered cry, and altered gaze need to be examined. The results are given in four categories: 0: no ABE, 1-4: mild ABE, 5-6: moderate ABE, and >6: severe ABE. In the example case, the patient had mild hypotonia and a high-pitched cry, so the BIND-M score is 2, which is classified as mild ABE with a likely low risk of neurological complications (if appropriate treatment is provided in a timely manner).
Another feature of the BiliNorm is the advice to complete a follow-up examination at the outpatient clinic. This is based on the possible diagnosis of kernicterus spectrum disorder (KSD) and consists of a scoring system that includes the highest TSB level, the presence of risk factors, the findings of neurological examinations performed at first presentation and at follow-up, the presence of enamel dysplasia, the results of the auditory brainstem response (ABR) test, and MRI findings [10]. The result is given as one of four categories: definite kernicterus (10-14), probable kernicterus (6-9), possible kernicterus (3-5) and no kernicterus (0-2). This feature might help professionals to prepare for the possible long-term complications of severe hyperbilirubinemia.
Communication with the patients’ family is often neglected and can be difficult for health care workers in low- and middle-income countries, especially in Indonesia. Therefore, the BiliNorm also provides an educational checklist on what should be told to the patients’ family. The checklist was adopted and adapted from the NICE guidelines on neonatal jaundice [11].
We introduced the BiliNorm app to health care workers, including midwives, paediatric residents and paediatricians, from two general district hospitals in East Java (Dr Soetomo General Hospital, Surabaya, Indonesia, and Dr. Saiful Anwar General Hospital, Malang, Indonesia) in March 2019. After the introduction of the app, we asked the participants to use BiliNorm.
To evaluate how BiliNorm was perceived in practice, we sent a questionnaire via Google form to health care workers who had used the application. This questionnaire used for this study was adapted and developed from Davis’ Technology Acceptance Model (TAM) [12]. The questionnaire had four main parts: 1. Perceived usefulness; 2. Perceived ease of use; 3. Subjective norms, and 4. Intention to use BiliNorm in the future. There were 22 questions in total, and for each question, seven possible answers ranging from 1 (strongly disagree) to 7 (strongly agree) could be given. The English version of our questionnaire is shown in the Supplementary File 1.
To investigate whether the introduction of the BiliNorm app had an effect on the treatment of infants with hyperbilirubinemia, we collected data from the medical records of all patients with neonatal hyperbilirubinemia admitted to the neonatal units of both hospitals during the six-month period before BiliNorm was introduced (September 2018 to March 2019) and the seven-month period after its introduction (April to September 2019).
Data on gestational age, birth weight, birth date, risk factors, and TSB were collected to determine which treatment should have been given to the patients based on the Indonesian Hyperbilirubinemia Guideline. Next, the actual treatment that was given was compared with the treatment that was indicated by the guidelines. All cases were divided into four groups: under-treatment, correct treatment, over-treatment, and inappropriate treatment. Under-treatment meant that the infant did not receive any treatment despite having a TSB level above the PT threshold. Over-treatment meant that the baby received PT despite having a TSB level below the PT threshold. Correct treatment was defined as treatment consistent with the Indonesian Hyperbilirubinemia Guideline. Inappropriate treatment indicated that treatment was given without any TSB measurement.
Ethical approval was granted by the Ethical Committee in Health Research of Dr Soetomo General Hospital Surabaya (Number 1060/KEPK/III/2019). Written informed consent was obtained from all participating health care workers and from the parents of all infants included in this study.
Data and Statistical Analysis
The data collected from the medical records were analysed using SPSS for Windows, Version 21 (IBM., Corp. Armonk, N.Y., USA). Pearson's chi-square test was used to calculate the p value of the proportion of infants within a gestational age category, birth weight category, risk factor category, and treatment classifications for the pre-introduction period versus the post-introduction period of the BiliNorm. We also calculated the p values of correct treatment, under-treatment, over-treatment and inappropriate treatment pre- versus post-introduction of the BiliNorm using Pearson's chi-square test. Probability values < 0.05 were considered statistically significant.