Key words coronary artery fistura, coronary ishchemia, coronary steal, myocardial ischemia, neonate.Coronary artery fistula (CAF) is a rare heart anomaly defined as abnormal communication of coronary arteries with cardiac chambers or other vessels. Although symptoms such as exercise intolerance with dyspnea, angina, and arrhythmia may occur, more than half of patients are clinically asymptomatic even when they have moderate volume overload. 1 The symptoms have been known to increase with age, and the majority of pediatric CAF are asymptomatic. 2 Rarely, infants with a large CAF have symptoms such as heart murmur or congestive heart failure. 1,2 Myocardial ischemia, however, is extremely rare in infancy, and only three cases have been found in the literature. [3][4][5] In addition, CAF in all three cases originated from the left coronary artery (LCA), and not from the right coronary artery (RCA).Herein, we describe the case of a neonate with a CAF originating from the RCA who was asymptomatic at birth. The Fig. 1 (a) Echocardiography on the first day of age showing a markedly enlarged right coronary artery (RCA). (b) Color Doppler echocardiography showing the coronary artery fistula (CAF; open arrows) draining into the right ventricle (RV) just below the tricuspid valve. (c) Electrocardiography at 7 days of age showing significant ST depression in V1-V4. (d) Coronary angiography of the RCA showing a significantly dilated RCA draining into the RV through the CAF. In addition, the distal RCA is not visible. (e) Collateral arteries (white arrows) from the left coronary artery feed the distal RCA.Neonatal right CAF with coronary ischemia 417 patient rapidly developed heart failure in 1 week because of left-to-right shunt and coronary ischemia. The study was approved by the local ethics committee and informed consent for publication was obtained from the parents.A male neonate born at 39 weeks of gestation was transferred to Asahikawa Kosei General Hospital at 5 h of age because of a heart murmur. On admission, he had a Levine 3/6 to-and-fro murmur at the lower left sternal border. His vitals were stable with normal respiratory status.Chest X-ray showed a normal-sized heart with a cardiothoracic ratio (CTR) of 51% and no pulmonary congestion. Electrocardiogram (ECG) on the first day of age showed no ST-segment or T-wave abnormalities, suggesting no coronary ischemia.Echocardiography indicated an enlarged RCA and a large CAF draining from the RCA into the right ventricle (RV; Fig. 1a,b), with the RV to RCA flow in systole and the reverse flow in diastole. On Doppler echocardiography an apparent diastolic reverse flow was seen in the ascending aorta, suggesting a significant diastolic steal through the CAF to the RV, whereas there was neither chamber enlargement nor valvular regurgitation.His general condition and echocardiography findings were stable at 3 days of age; therefore, he was transferred back to the initial clinic and subsequently discharged 2 days later.Upon his visit to hospital at 7 days of age, he presente...
Introduction Although imatinib is the first-line of therapy for Philadelphia chromosome (Ph)-positive chronic myeloid leukemia (CML), in Japan, it is recommended by the manufacturer that lactating women treated with imatinib mesylate for CML should discontinue breastfeeding their infants. Case A 32-year-old pregnant patient was diagnosed with Ph-positive CML at 13 weeks of gestation. She received imatinib (400 mg/day) after 28 weeks of gestation. A female infant was delivered at a gestational age of 35 weeks and 3/7 days after preterm premature rupture of membranes. It was decided to feed only colostrum to the infant and formula feeding was done subsequently because of the risk of the transfer of imatinib to breast milk. The milk/plasma (M/P) ratio and the relative infant dose (RID) for imatinib were calculated to be 0.35 and 1.4%, respectively at 5 days of life. Moreover, the serum level of imatinib in the child of age 5 days was 27 ng/mL, which was much lower than the target trough value for CML (1000 ng/mL). Conclusion The M/P ratio and RID values for maternally administered imatinib were within the safe range for breastfeeding, as reported in previous studies. In addition, it was found that the serum concentration of imatinib in the child was relatively low during short-term breastfeeding.
Background Iron deficiency during infancy is associated with poor neurological development, but iron overload causes severe complications. Appropriate iron supplementation is therefore vital. Reticulocyte hemoglobin content (RET‐He) provides a real‐time assessment of iron status and chracterezes hemoglobin synthesis in preterm infants. However, the existing literature lacks detailed reports assessing chronological changes in RET‐He. The aim of this study was to assess the chronological changes in RET‐He during oral iron dietary supplementation, and concomitant therapy with recombinant human erythropoietin (rHuEPO) in preterm very low birthweight infants. Methods Very low birthweight infants, admitted to our neonatal intensive care unit were analyzed retrospectively. Hemoglobin (Hb), reticulocyte percentage (Ret), mean corpuscular volume, RET‐He, serum iron (Fe), and serum ferritin were recorded. Data at birth (T0), the initial day of rHuEPO therapy (T1), the initial day of oral iron supplementation (T2), 1–2 weeks (T3), 3–4 weeks (T4), 5–6 weeks (T5), and 7–8 weeks (T6) from the initial day of oral iron supplementation were extracted, and their changes over time were examined. Results Reticulocyte hemoglobin content was highest at birth and declined rapidly thereafter, especially after starting rHuEPO therapy. There was no upward trend in RET‐He after the initiation of oral iron supplementation, with a slower increase during 5–6 weeks after the initiation of iron therapy. Conclusions During the treatment of anemia of prematurity, low RET‐He levels may be prolonged. Anemia of prematurity should therefore be assessed and treated on a case‐by‐case basis, while considering the iron metabolic capacity of preterm infants.
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