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All Journal Paediatrica Indonesiana
Sri Lilijanti Widjaja
Department of Child Health, Universitas Sebelas Maret Medical School/Dr. Moewardi Hospital, Surakarta, Central Java
Health Professions Medicine & Pharmacology

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Baby girl with pentalogy of Cantrell: a case report on an extremely rare condition Widiastuti Soewondo; Suyono Suyono; Johannes Berchmans Prasodjo; Sri Lilijanti Widjaja
Paediatrica Indonesiana Vol 59 No 1 (2019): January 2019
Publisher : Indonesian Pediatric Society

Show Abstract | Download Original | Original Source | Check in Google Scholar | Full PDF (346.475 KB) | DOI: 10.14238/pi59.1.2019.51-4

Abstract

In 1958, Cantrell et al. described an extremely rare syndrome of congenital defects involving the abdominal wall, sternum, diaphragm, pericardium, and heart malformation.1 The incidence of pentalogy of Cantrell (POC) is one in 65,000 live births.1,2,3 Only one case was reported in Dr. Moewardi Public Hospital between 1999 and 2016. The exact etiology of this condition is unknown, but developmental failure of mesoderm at 4 weeks of gestational age may contribute to the condition.4 The prognosis depends on the degree of heart failure and the malformations that occur. Heart failure, arrhythmia, cardiac rupture, cardiac tamponade, endocarditis, and peripheral emboli are described as the main complications and causes of death.5,6 The aim of this report was to add to reference data about complete POC and the prognostic outcome.
TNF-? as a predictive factor of pulmonary hypertension in children with Down syndrome with and without congenital heart disease Latifah Rahmi Hariyanti; Sri Lilijanti Widjaja; Dwi Hidayah
Paediatrica Indonesiana Vol 62 No 1 (2022): January 2022
Publisher : Indonesian Pediatric Society

Show Abstract | Download Original | Original Source | Check in Google Scholar | DOI: 10.14238/pi62.1.2022.61-5

Abstract

Background Down syndrome (DS) is a chromosomal disorder due to trisomy 21 that may involve congenital heart disease (CHD). Pulmonary hypertension (PH) may be present in DS with and without CHD. TNF-α is a cytokine involved in the pathogenesis of inflammation in PH. Objective To determine the association between TNF-α and the risk of PH in children with DS with and without congenital heart disease. Methods This observational study was conducted in DS children aged two months to five years who visited the outpatient clinic of a regional referral hospital in Indonesia. Subjects underwent echocardiography and were classified into four groups (CHD-PH, CHD-no PH, no CHD-PH, no CHD-no PH). Serum TNF-α was measured in all subjects. We used the ANOVA test to compare mean TNF-α between the groups and to determine the optimal TNF-α cut-off point. We compared the risk of PH in subjects with TNF-α above and below the cut-off point. Results We included 36 DS children in this study. Mean TNF-α in the CHD-PH, CHD-no PH, no CHD-PH, and no CHD-no PH groups was 2,564.44 (SD 177.00) pg/mL, 2,112.89 (SD 382.00) pg/mL, 2,211.56 (SD 330.70) pg/mL, and 1,118.89 (SD 1056.65) pg/mL, respectively (p<0.001). The optimal TNF-α cut-off point was 2,318 pg/mL. DS children with TNF-α ≥2,318 pg/mL had a higher risk of CHD (RR=2.6; 95%CI 1.17 to 5.78; p=0.008) and PH (RR=3.5; 95%CI 1.43 to 8.60; p=0.001). Conclusions DS children with CHD accompanied by PH have significantly higher TNF-α levels than those without PH and those without CHD. In children with DS, an elevated TNF-α level (≥2,318 pg/mL) is associated with a higher risk of CHD and PH.
Co-Authors Dwi Hidayah Johannes Berchmans Prasodjo Latifah Rahmi Hariyanti Suyono Suyono Widiastuti Soewondo