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Published in: BMC Pediatrics 1/2021

Open Access 01-12-2021 | Portal Vein Thrombosis | Case report

Portal vein thrombosis and food protein-induced allergic proctocolitis in a premature newborn with hypereosinophilia: a case report

Authors: Cheong-Jun Moon, Tae Hee Kwon, Hyun-Seung Lee

Published in: BMC Pediatrics | Issue 1/2021

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Abstract

Background

Peripheral blood eosinophilia is identified in numerous medical conditions associated with allergic, infectious, and inflammatory processes mostly as reactive eosinophilia with or without tissue eosinophilia. In hospitalized neonates, eosinophilia is common with an inverse relationship with gestational age and occurs solely as mild eosinophilia in the majority of cases. In the literature, eosinophilia has been proposed as a possible risk factor for venous thromboembolism. However, few reports are found on thromboembolic events including portal vein thrombosis (PVT) associated with eosinophilia in the newborn period.
Neonates, particularly preterm infants, are vulnerable to thrombosis due to the immature and developing hemostatic system with little reserve capacity, which occurs as catheter-related thrombosis in most cases.

Case presentation

A male newborn at 34+ 5 weeks’ gestation presented with a left portal venous thrombus and hematochezia after initial cow’s milk feeding in the setting of blood hypereosinophilia for a prolonged period of time without central venous catheterization. The infant was diagnosed with PVT and food protein-induced allergic proctocolitis (FPIAP) and showed complete resolution of the conditions with expectant management with food avoidance, including the normalized eosinophil count.

Conclusions

Our experience suggests that in the setting of hypereosinophilia with a prolonged duration in premature neonates, FPIAP should be suspected in case of hematochezia in otherwise healthy infants, and considering the increased thrombotic risk by the hypereosinophilia and premature newborn status, evaluation for neonatal thrombosis may be needed, including PVT with the potential risk for the more serious, but uncommon, late complications encompassing portal hypertension.
Literature
1.
go back to reference Sullivan SE, Calhoun DA. Eosinophilia in the neonatal intensive care unit. Clin Perinatol. 2000;27:603–22.CrossRef Sullivan SE, Calhoun DA. Eosinophilia in the neonatal intensive care unit. Clin Perinatol. 2000;27:603–22.CrossRef
2.
go back to reference Juul SE, Haynes JW, McPherson RJ. Evaluation of eosinophilia in hospitalized preterm infants. J Perinatol. 2005;25:182–8.CrossRef Juul SE, Haynes JW, McPherson RJ. Evaluation of eosinophilia in hospitalized preterm infants. J Perinatol. 2005;25:182–8.CrossRef
3.
go back to reference Yen JM, Lin CH, Yang MM, Hou ST, Lin AH, Lin YJ. Eosinophilia in very low birth weight infants. Pediatr Neonatol. 2010;51:116–23.CrossRef Yen JM, Lin CH, Yang MM, Hou ST, Lin AH, Lin YJ. Eosinophilia in very low birth weight infants. Pediatr Neonatol. 2010;51:116–23.CrossRef
5.
go back to reference Valent P. Pathogenesis, classification, and therapy of eosinophilia and eosinophil disorders. Blood Rev. 2009;23:157–65.CrossRef Valent P. Pathogenesis, classification, and therapy of eosinophilia and eosinophil disorders. Blood Rev. 2009;23:157–65.CrossRef
6.
go back to reference Kuang FL. Approach to patients with eosinophilia. Med Clin North Am. 2020;104:1–14.CrossRef Kuang FL. Approach to patients with eosinophilia. Med Clin North Am. 2020;104:1–14.CrossRef
7.
go back to reference Larsen RL, Savage NM. How I investigate eosinophilia. Int J Lab Hematol. 2019;41:153–61.CrossRef Larsen RL, Savage NM. How I investigate eosinophilia. Int J Lab Hematol. 2019;41:153–61.CrossRef
8.
go back to reference Wahidi LS, Sherman J, Miller MM, Zaghouani H, Sherman MP. Early persistent blood eosinophilia in necrotizing enterocolitis is a predictor of late complications. Neonatology. 2015;108:137–42.CrossRef Wahidi LS, Sherman J, Miller MM, Zaghouani H, Sherman MP. Early persistent blood eosinophilia in necrotizing enterocolitis is a predictor of late complications. Neonatology. 2015;108:137–42.CrossRef
9.
go back to reference Calbi M, Giacchetti L. Low breast milk IgA and high blood eosinophil count in breast-fed newborns determine higher risk for developing atopic eczema after an 18-month follow-up. J Investig Allergol Clin Immunol. 1998;8:161–4.PubMed Calbi M, Giacchetti L. Low breast milk IgA and high blood eosinophil count in breast-fed newborns determine higher risk for developing atopic eczema after an 18-month follow-up. J Investig Allergol Clin Immunol. 1998;8:161–4.PubMed
10.
go back to reference Liu Y, Meng X, Feng J, Zhou X, Zhu H. Hypereosinophilia with concurrent venous thromboembolism: clinical features, potential risk factors, and short-term outcomes in a Chinese cohort. Sci Rep. 2020;10:8359.CrossRef Liu Y, Meng X, Feng J, Zhou X, Zhu H. Hypereosinophilia with concurrent venous thromboembolism: clinical features, potential risk factors, and short-term outcomes in a Chinese cohort. Sci Rep. 2020;10:8359.CrossRef
11.
go back to reference Maino A, Rossio R, Cugno M, Marzano AV, Tedeschi A. Hypereosinophilic syndrome, Churg-Strauss syndrome and parasitic diseases: possible links between eosinophilia and thrombosis. Curr Vasc Pharmacol. 2012;10:670–5.CrossRef Maino A, Rossio R, Cugno M, Marzano AV, Tedeschi A. Hypereosinophilic syndrome, Churg-Strauss syndrome and parasitic diseases: possible links between eosinophilia and thrombosis. Curr Vasc Pharmacol. 2012;10:670–5.CrossRef
12.
go back to reference Ames PR, Aloj G, Gentile F. Eosinophilia and thrombosis in parasitic diseases: an overview. Clin Appl Thromb Hemost. 2011;17:33–8.CrossRef Ames PR, Aloj G, Gentile F. Eosinophilia and thrombosis in parasitic diseases: an overview. Clin Appl Thromb Hemost. 2011;17:33–8.CrossRef
13.
go back to reference Ames PR. Recurrent abdominal thrombosis despite heparin thromboprophylaxis in a patient with transient eosinophilia. Clin Appl Thromb Hemost. 2011;17:229–31.CrossRef Ames PR. Recurrent abdominal thrombosis despite heparin thromboprophylaxis in a patient with transient eosinophilia. Clin Appl Thromb Hemost. 2011;17:229–31.CrossRef
14.
go back to reference Sano H, Fukuoka T, Maruyama H, Hayashi T, Tanahashi N. Cerebral sinovenous thrombosis in a patient with transient eosinophilia. Intern Med. 2014;53:2139–42.CrossRef Sano H, Fukuoka T, Maruyama H, Hayashi T, Tanahashi N. Cerebral sinovenous thrombosis in a patient with transient eosinophilia. Intern Med. 2014;53:2139–42.CrossRef
15.
go back to reference Kenet G, Cohen O, Bajorat T, Nowak-Göttl U. Insights into neonatal thrombosis. Thromb Res. 2019;181:S33–6.CrossRef Kenet G, Cohen O, Bajorat T, Nowak-Göttl U. Insights into neonatal thrombosis. Thromb Res. 2019;181:S33–6.CrossRef
16.
go back to reference Dubbink-Verheij GH, Visser R, Roest AA, van Ommen CH, Te Pas AB, Lopriore E. Thrombosis after umbilical venous catheterisation: prospective study with serial ultrasound. Arch Dis Child Fetal Neonatal Ed. 2020;105:299–303.CrossRef Dubbink-Verheij GH, Visser R, Roest AA, van Ommen CH, Te Pas AB, Lopriore E. Thrombosis after umbilical venous catheterisation: prospective study with serial ultrasound. Arch Dis Child Fetal Neonatal Ed. 2020;105:299–303.CrossRef
17.
go back to reference Morag I, Epelman M, Daneman A, Moineddin R, Parvez B, Shechter T, et al. Portal vein thrombosis in the neonate: risk factors, course, and outcome. J Pediatr. 2006;148:735–9.CrossRef Morag I, Epelman M, Daneman A, Moineddin R, Parvez B, Shechter T, et al. Portal vein thrombosis in the neonate: risk factors, course, and outcome. J Pediatr. 2006;148:735–9.CrossRef
18.
go back to reference Cetinkaya PG, Kahveci M, Karaatmaca B, Esenboga S, Sahiner UM, Sekerel BE, et al. Predictors for late tolerance development in food protein-induced allergic proctocolitis. Allergy Asthma Proc. 2020;41:e11–8.CrossRef Cetinkaya PG, Kahveci M, Karaatmaca B, Esenboga S, Sahiner UM, Sekerel BE, et al. Predictors for late tolerance development in food protein-induced allergic proctocolitis. Allergy Asthma Proc. 2020;41:e11–8.CrossRef
19.
go back to reference Martin VM, Virkud YV, Seay H, Hickey A, Ndahayo R, Rosow R, et al. Prospective assessment of pediatrician-diagnosed food protein-induced allergic proctocolitis by gross or occult blood. J Allergy Clin Immunol Pract. 2020;8:1692–9.e1.CrossRef Martin VM, Virkud YV, Seay H, Hickey A, Ndahayo R, Rosow R, et al. Prospective assessment of pediatrician-diagnosed food protein-induced allergic proctocolitis by gross or occult blood. J Allergy Clin Immunol Pract. 2020;8:1692–9.e1.CrossRef
20.
go back to reference Williams S, Chan AK. Neonatal portal vein thrombosis: diagnosis and management. Semin Fetal Neonatal Med. 2011;16:329–39.CrossRef Williams S, Chan AK. Neonatal portal vein thrombosis: diagnosis and management. Semin Fetal Neonatal Med. 2011;16:329–39.CrossRef
21.
go back to reference Cabannes M, Bouissou A, Favrais G, Sembély-Taveau C, Morales L, Favreau A, et al. Systematic ultrasound examinations in neonates admitted to NICU: evolution of portal vein thrombosis. J Perinatol. 2018;38:1359–64.CrossRef Cabannes M, Bouissou A, Favrais G, Sembély-Taveau C, Morales L, Favreau A, et al. Systematic ultrasound examinations in neonates admitted to NICU: evolution of portal vein thrombosis. J Perinatol. 2018;38:1359–64.CrossRef
22.
go back to reference Bhatt MD, Patel V, Butt ML, Chan AKC, Paes B. Thrombosis and hemostasis in newborns (THiN) group. Outcomes following neonatal portal vein thrombosis: a descriptive, single-center study and review of anticoagulant therapy. Pediatr Blood Cancer. 2019;66:e27572.CrossRef Bhatt MD, Patel V, Butt ML, Chan AKC, Paes B. Thrombosis and hemostasis in newborns (THiN) group. Outcomes following neonatal portal vein thrombosis: a descriptive, single-center study and review of anticoagulant therapy. Pediatr Blood Cancer. 2019;66:e27572.CrossRef
23.
go back to reference Jo J, Garssen J, Knippels L, Sandalova E. Role of cellular immunity in cow's milk allergy: pathogenesis, tolerance induction, and beyond. Mediat Inflamm. 2014;2014:249784.CrossRef Jo J, Garssen J, Knippels L, Sandalova E. Role of cellular immunity in cow's milk allergy: pathogenesis, tolerance induction, and beyond. Mediat Inflamm. 2014;2014:249784.CrossRef
24.
go back to reference Fredens K, Dahl R, Venge P. In vitro studies of the interaction between heparin and eosinophil cationic protein. Allergy. 1991;46:27–9.CrossRef Fredens K, Dahl R, Venge P. In vitro studies of the interaction between heparin and eosinophil cationic protein. Allergy. 1991;46:27–9.CrossRef
25.
go back to reference Rucinski B, Niewiarowski S, Strzyzewski M, Holt JC, Mayo KH. Human platelet factor 4 and its C-terminal peptides: heparin binding and clearance from the circulation. Thromb Haemost. 1990;63:493–8.CrossRef Rucinski B, Niewiarowski S, Strzyzewski M, Holt JC, Mayo KH. Human platelet factor 4 and its C-terminal peptides: heparin binding and clearance from the circulation. Thromb Haemost. 1990;63:493–8.CrossRef
26.
go back to reference Slungaard A, Vercellotti GM, Tran T, Gleich GJ, Key NS. Eosinophil cationic granule proteins impair thrombomodulin function. A potential mechanism for thromboembolism in hypereosinophilic heart disease. J Clin Invest. 1993;91:1721–30.CrossRef Slungaard A, Vercellotti GM, Tran T, Gleich GJ, Key NS. Eosinophil cationic granule proteins impair thrombomodulin function. A potential mechanism for thromboembolism in hypereosinophilic heart disease. J Clin Invest. 1993;91:1721–30.CrossRef
27.
go back to reference Mukai HY, Ninomiya H, Ohtani K, Nagasawa T, Abe T. Major basic protein binding to thrombomodulin potentially contributes to the thrombosis in patients with eosinophilia. Br J Haematol. 1995;90:892–9.CrossRef Mukai HY, Ninomiya H, Ohtani K, Nagasawa T, Abe T. Major basic protein binding to thrombomodulin potentially contributes to the thrombosis in patients with eosinophilia. Br J Haematol. 1995;90:892–9.CrossRef
28.
go back to reference Levi M, Hack CE, de Boer JP, Brandjes DP, Büller HR, ten Cate JW. Reduction of contact activation related fibrinolytic activity in factor XII deficient patients. Further evidence for the role of the contact system in fibrinolysis in vivo. J Clin Invest. 1991;88:1155–60.CrossRef Levi M, Hack CE, de Boer JP, Brandjes DP, Büller HR, ten Cate JW. Reduction of contact activation related fibrinolytic activity in factor XII deficient patients. Further evidence for the role of the contact system in fibrinolysis in vivo. J Clin Invest. 1991;88:1155–60.CrossRef
29.
go back to reference Ojima-Uchiyama A, Masuzawa Y, Sugiura T, Waku K, Fukuda T, Makino S. Production of platelet-activating factor by human normodense and hypodense eosinophils. Lipids. 1991;26:1200–3.CrossRef Ojima-Uchiyama A, Masuzawa Y, Sugiura T, Waku K, Fukuda T, Makino S. Production of platelet-activating factor by human normodense and hypodense eosinophils. Lipids. 1991;26:1200–3.CrossRef
30.
go back to reference Rohrbach MS, Wheatley CL, Slifman NR, Gleich GJ. Activation of platelets by eosinophil granule proteins. J Exp Med. 1990;172:1271–4.CrossRef Rohrbach MS, Wheatley CL, Slifman NR, Gleich GJ. Activation of platelets by eosinophil granule proteins. J Exp Med. 1990;172:1271–4.CrossRef
31.
go back to reference Moosbauer C, Morgenstern E, Cuvelier SL, Manukyan D, Bidzhekov K, Albrecht S, et al. Eosinophils are a major intravascular location for tissue factor storage and exposure. Blood. 2007;109:995–1002.CrossRef Moosbauer C, Morgenstern E, Cuvelier SL, Manukyan D, Bidzhekov K, Albrecht S, et al. Eosinophils are a major intravascular location for tissue factor storage and exposure. Blood. 2007;109:995–1002.CrossRef
32.
go back to reference Santilli F, Basili S, Ferroni P, Davì G. CD40/CD40L system and vascular disease. Intern Emerg Med. 2007;2:256–68.CrossRef Santilli F, Basili S, Ferroni P, Davì G. CD40/CD40L system and vascular disease. Intern Emerg Med. 2007;2:256–68.CrossRef
33.
go back to reference Monterrubio Villar J, Córdoba López A, Macayo Sánchez AJ. Idiopathic eosinophilia associated with portal vein and massive thrombosis: successful thrombolysis with streptokinase. Med Sci Monit. 2006;12:CS53–6.PubMed Monterrubio Villar J, Córdoba López A, Macayo Sánchez AJ. Idiopathic eosinophilia associated with portal vein and massive thrombosis: successful thrombolysis with streptokinase. Med Sci Monit. 2006;12:CS53–6.PubMed
34.
go back to reference Mates M, Nesher G, Roth B, Rosenberg R, Heyd J, Hershko C. Transient severe eosinophilia precipitating massive venous thrombosis in a patient with hereditary thrombophilia. Acta Haematol. 2004;112:209–11.CrossRef Mates M, Nesher G, Roth B, Rosenberg R, Heyd J, Hershko C. Transient severe eosinophilia precipitating massive venous thrombosis in a patient with hereditary thrombophilia. Acta Haematol. 2004;112:209–11.CrossRef
35.
go back to reference An HS, Baek JS, Kwon BS, Kim GB, Bae EJ, Noh CI, et al. A case of pulmonary thromboembolism associated with hypereosinophilia in a child. Korean Circ J. 2009;39:382–5.CrossRef An HS, Baek JS, Kwon BS, Kim GB, Bae EJ, Noh CI, et al. A case of pulmonary thromboembolism associated with hypereosinophilia in a child. Korean Circ J. 2009;39:382–5.CrossRef
36.
37.
go back to reference Makatsariya A, Bitsadze V, Khizroeva J, Vorobev A, Makatsariya N, Egorova E, et al. Neonatal thrombosis. J Matern Fetal Neonatal Med. 2020:1–9. Makatsariya A, Bitsadze V, Khizroeva J, Vorobev A, Makatsariya N, Egorova E, et al. Neonatal thrombosis. J Matern Fetal Neonatal Med. 2020:1–9.
Metadata
Title
Portal vein thrombosis and food protein-induced allergic proctocolitis in a premature newborn with hypereosinophilia: a case report
Authors
Cheong-Jun Moon
Tae Hee Kwon
Hyun-Seung Lee
Publication date
01-12-2021
Publisher
BioMed Central
Published in
BMC Pediatrics / Issue 1/2021
Electronic ISSN: 1471-2431
DOI
https://doi.org/10.1186/s12887-021-02510-9

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