Unexplained late‐onset hemolysis and methemoglobinemia in a preterm infant

Jinnou, Hideo; Sugiura, Hiroshi; Kikuchi, Shin; Shirai, Kohji; Hirose, Etsuko; Ohki, Shinya

doi:10.1111/j.1442-200x.2011.03389.x

Cited by 4 publications

(6 citation statements)

References 11 publications

(25 reference statements)

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“…The symptoms of the disease were similar to those previously reported in Japan 2–5 . The severity of the disease was variable but severe anemia with Hb <7 g/dL and jaundice with TSB concentration ˃15 mg/dL were observed in 33% and 54% of all cases, respectively.…”

Section: Discussionsupporting

confidence: 83%

“…We previously reported four cases of acute hemolytic episodes of unknown cause in premature infants whose birthweights were <1,000 g at 2–4 weeks after birth 2 . Since then, there have been some case reports with similar characteristics in Japan 3–5 . These patients had some features in common, which are as follows: (i) all were very low birthweight (VLBW) infants who weighed <1,500 g at birth; (ii) episodes occurred suddenly at 10–30 days after birth; (iii) they had no family history of hemolytic diseases or blood type incompatibility between mother and baby; (iv) hemolysis was accompanied by an increase in methemoglobin (MetHb) concentration or the presence of Heinz bodies in erythrocytes, and (v) the hemolytic episodes were transient and not recurrent.…”

mentioning

confidence: 97%

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Nationwide survey of late‐onset hemolysis in very low birthweight infants

Miyazono

Arai

Kanai

et al. 2021

Pediatrics International

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Background In Japan, some cases of late‐onset acute hemolysis in very low birthweight (VLBW) infants have been reported. These cases had common features but the cause of hemolysis was unknown. The incidence and prognosis of this disease are also unknown. However, there are only few reports of such hemolytic episodes in countries other than Japan. Thus, this study aimed to examine the incidence and clinical course of late‐onset acute hemolysis and to establish it as a new disease concept. Methods A nationwide prospective survey was conducted from 2011 to 2015 as a rare disease surveillance project of the Japan Society for Neonatal Health and Development. Results Twenty‐four cases were confirmed. The median (range) gestational age, birthweight, and onset of hemolytic episodes were 26 weeks and 2 days (23 weeks and 4 days–31 weeks and 2 days), 898 g (627–1,416 g), and 19 days after birth (9–33 days), respectively. Phototherapy, blood transfusion, and exchange transfusion were required in 22 (96%), 24 (100%), and 7 (29%) cases, respectively. During the observation period, no recurrence of the hemolytic episode occurred. All patients survived; however, one case developed kernicterus and suffered severe neurological sequelae. Conclusions In this study, at least 1 out of 1,259 VLBW infants developed hemolysis at 9–33 days after birth in Japan. Owing to the risk of kernicterus, this disease should be recognized as among the important pathological conditions of VLBW infants, suggesting the need to manage jaundice and anemia until 5 weeks after birth.

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Section: Discussionsupporting

confidence: 83%

mentioning

confidence: 97%

Nationwide survey of late‐onset hemolysis in very low birthweight infants

Miyazono

Arai

Kanai

et al. 2021

Pediatrics International

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“…Reportedly, however, none of the Japanese kernicterus cohort was G6PD deficient, a finding consistent with the low prevalence of G6PD deficiency in Japan [4]. Analogous late-onset hemolytic events have been reported in extremely preterm neonates in Japan [5, 6]. These are often characterized by Heinz body formation and mild methemoglobinemia [5, 6] possibly resulting from oxidative damage in the context of immature antioxidant defenses [7].…”

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confidence: 73%

The Enigma of Preterm Late Hyperbilirubinemia Kernicterus in Japan

Watchko¹

2021

Neonatology

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“…To our knowledge, this is the first report describing the prediction of unexplained late-onset hemolysis using TcB monitoring in a VLBW infant. The following characteristics are seen in unexplained late-onset hemolysis: (1) the infant is born preterm, especially with a VLBW; (2) despite a stable general condition, hemolytic hyperbilirubinemia occurs suddenly 2–3 weeks after birth; (3) SpO 2 decreases as a result of methemoglobinemia, and this is followed by elevation of blood COHb; (4) the peripheral blood picture shows Heinz bodies; (5) the hemolytic attack is transient and does not relapse; and (6) the condition may develop into severe hyperbilirubinemia, which requires exchange transfusion in addition to phototherapy [ 3 , 4 ]. The present case showed all these characteristics, allowing a diagnosis of unexplained late-onset hemolysis.…”

Section: Discussionmentioning

confidence: 99%

“…The etiology of the condition remains uncertain. One possible causative factor in this disease is oxidative stress, perhaps caused by a combination of the immature antioxidant capacity of the erythrocyte and exposure to highly concentrated oxygen and drugs or toxic agents [ 3 , 8 ]. Although neonatal Heinz body hemolytic anemia can be induced by exposures of vitamin C [ 9 ], phenolic disinfectant [ 10 ], toluidine blue [ 11 ], or methylene blue [ 12 ], the present case did not expose to these agents.…”

Section: Discussionmentioning

confidence: 99%

Transcutaneous bilirubin monitoring predicts unexplained late-onset hemolysis in a very low birthweight infant

et al. 2016

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Background:In term infants, transcutaneous bilirubin (TcB) monitoring can be used to predict hemolytic hyperbilirubinemia. However, it is not clear whether the technique can also be used to predict unexplained late-onset hemolysis in very low birthweight (VLBW) infants.Case presentation: The case was an infant with a birthweight of 1154 g who developed unexplained late-onset hemolysis at 8 days of age. The hyperbilirubinemia rapidly worsened, and therefore both phototherapy and exchange transfusion were performed. TcB levels were measured using the JM-105 jaundice meter and found to have increased by >3 mg/dL since before the onset, demonstrating for the first time that the device clearly detects changes in hemolytic rate. Conclusions:Although TcB levels did not correspond directly with total serum bilirubin levels in VLBW infants, the two values exhibited parallel changes in this case. Therefore, serial TcB monitoring may be useful in the early prediction of unexplained late-onset hemolysis in VLBW infants.

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Unexplained late‐onset hemolysis and methemoglobinemia in a preterm infant

Cited by 4 publications

References 11 publications

Nationwide survey of late‐onset hemolysis in very low birthweight infants

Nationwide survey of late‐onset hemolysis in very low birthweight infants

The Enigma of Preterm Late Hyperbilirubinemia Kernicterus in Japan

Transcutaneous bilirubin monitoring predicts unexplained late-onset hemolysis in a very low birthweight infant

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