Development of Pulmonary Hypertension in an Infant Treated With Diazoxide for Hyperinsulinism, a Case Report and Literature Review

Albahri Ziad; Lukeš Antonín; Mynářová Kateřina; Krylová Kateřina; Al Mawiri Abdul; Hussain Khaled; Neumann David

doi:doi:10.11648/j.ajp.20150101.11

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Development of Pulmonary Hypertension in an Infant Treated With Diazoxide for Hyperinsulinism, a Case Report and Literature Review

Albahri Ziad, Lukeš Antonín, Mynářová Kateřina, Krylová Kateřina, Al Mawiri Abdul, Hussain Khaled, Neumann David

Published in American Journal of Pediatrics (Volume 1, Issue 1)

Received: 27 June 2015 Accepted: 20 July 2015 Published: 23 July 2015

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Abstract

Congenital hyperinsulinism (CHI) is heterogeneous disorder in the neonatal period characterized by inappropriate insulin secretion in the presence of hypoglycemia. Known etiologies include inactivating mutations of the KATP channel. Its management can be extremely complicated, and may involve medical therapy and surgery. We describe a full term baby male at one month-old without dysmorphic features or congenital anomalies, the baby was presented with asymptomatic hypoglycemia by routine screening at the age of 1.5 hour of life, he was treated with diazoxide for prolonged neonatal hyperinsulinism. Tolerance of diazoxide is usually excellent, but several adverse effects of this drug have been described. During diazoxide therapy he presented with tachycardia and low oxygen saturation, the baby became dyspnoeic with fluid retention and facial edema. Echocardiography showed moderate pulmonary hypertension. Diazoxide was withdrawn, his clinical status and pulmonary hypertension dramatically improved and returned to baseline.

Published in	American Journal of Pediatrics (Volume 1, Issue 1)
DOI	10.11648/j.ajp.20150101.11
Page(s)	1-3
Creative Commons	This is an Open Access article, distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution and reproduction in any medium or format, provided the original work is properly cited.
Copyright	Copyright © The Author(s), 2015. Published by Science Publishing Group

Keywords

Hyperinsulism, Diazoxide, Pulmonary Hypertension

References

[1]	Yorifuji T. Congenital hyperinsulinism: current status and future perspectives. Ann Pediatr Endocrinol Metab. 2014; 19(2):57-68.
[2]	Arnoux JB, Verkarre V, Saint-Martin C, Montravers F, Brassier A, Valayannopoulos V, Brunelle F, Fournet JC, Robert JJ, Aigrain Y, Bellanné-Chantelot C, de Lonlay P. Congenital hyperinsulinism: current trends in diagnosis and therapy. Orphanet J Rare Dis. 2011;6:63.
[3]	Gopal-Kothandapani JS, Hussain K. Congenital hyperinsulinism: Role of fluorine-18L-3, 4 hydroxyphenylalanine positron emission tomography scanning. World J Radiol. 2014 28;6(6):252-260.
[4]	Lovvorn HN 3rd, Nance ML, Ferry RJ Jr, Stolte L, Baker L, O'Neill JA Jr, Schnaufer L, Stanley CA, Adzick NS. Congenital hyperinsulinism and the surgeon: lessons learned over 35 years. J Pediatr Surg. 1999;34(5):786-92; discussion 792-793.
[5]	Parker JJ, Allen DB. Hypertrophic cardiomyopathy after prolonged diazoxide therapy for hyperinsulinemic hypoglycemia. J Pediatr. 1991;118(6):906-909.
[6]	Yildizdas D, Erdem S, Küçükosmanoglu O, Yilmaz M, Yüksel B. Pulmonary hypertension, heart failure and neutropenia due to diazoxide therapy. Adv Ther. 2008;25(5):515-519.
[7]	Price LC, McAuley DF, Marino PS, Finney SJ, Griffiths MJ, Wort SJ. Pathophysiology of pulmonary hypertension in acute lung injury. Am J Physiol Lung Cell Mol Physiol. 2012;302(9):L803-815.
[8]	Silvani P, Camporesi A Drug-induced pulmonary hypertension in newborns: a review. Curr Vasc Pharmacol. 2007;5(2):129-133. Review.
[9]	Demirel F, Unal S, Ç etin II, Esen I, Arasli A. Pulmonary hypertension and reopening of the ductus arteriosus in an infant treated with diazoxide. J Pediatr Endocrinol Metab. 2011;24(7-8):603-605.
[10]	Yildizdas D, Erdem S, Küçükosmanoglu O, Yilmaz M, Yüksel B. Pulmonary hypertension, heart failure and neutropenia due to diazoxide therapy. Adv Ther. 2008;25(5):515-519.
[11]	Nebesio TD, Hoover WC, Caldwell RL, Nitu ME, Eugster EA. Development of pulmonary hypertension in an infant treated with diazoxide. J Pediatr Endocrinol Metab. 2007;20(8):939-944.
[12]	Senguttuvan R, Saleh M, Wheeler M, Patel Ch, Chin C and Patel P. Diazoxide induced pulmonary hypertension in an infant. Endocrine Society's 96th Annual Meeting and Expo, June 21–24, 2014 – Chicago.
[13]	Silvani P, Camporesi A, Mandelli A, Wolfler A, Salvo I. A case of severe diazoxide toxicity. Paediatr Anaesth. 2004;14(7):607-609.

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Albahri Ziad, Lukeš Antonín, Mynářová Kateřina, Krylová Kateřina, Al Mawiri Abdul, et al. (2015). Development of Pulmonary Hypertension in an Infant Treated With Diazoxide for Hyperinsulinism, a Case Report and Literature Review. American Journal of Pediatrics, 1(1), 1-3. https://doi.org/10.11648/j.ajp.20150101.11

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Albahri Ziad; Lukeš Antonín; Mynářová Kateřina; Krylová Kateřina; Al Mawiri Abdul, et al. Development of Pulmonary Hypertension in an Infant Treated With Diazoxide for Hyperinsulinism, a Case Report and Literature Review. Am. J. Pediatr. 2015, 1(1), 1-3. doi: 10.11648/j.ajp.20150101.11

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AMA Style

Albahri Ziad, Lukeš Antonín, Mynářová Kateřina, Krylová Kateřina, Al Mawiri Abdul, et al. Development of Pulmonary Hypertension in an Infant Treated With Diazoxide for Hyperinsulinism, a Case Report and Literature Review. Am J Pediatr. 2015;1(1):1-3. doi: 10.11648/j.ajp.20150101.11

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@article{10.11648/j.ajp.20150101.11,
  author = {Albahri Ziad and Lukeš Antonín and Mynářová Kateřina and Krylová Kateřina and Al Mawiri Abdul and Hussain Khaled and Neumann David},
  title = {Development of Pulmonary Hypertension in an Infant Treated With Diazoxide for Hyperinsulinism, a Case Report and Literature Review},
  journal = {American Journal of Pediatrics},
  volume = {1},
  number = {1},
  pages = {1-3},
  doi = {10.11648/j.ajp.20150101.11},
  url = {https://doi.org/10.11648/j.ajp.20150101.11},
  eprint = {https://article.sciencepublishinggroup.com/pdf/10.11648.j.ajp.20150101.11},
  abstract = {Congenital hyperinsulinism (CHI) is heterogeneous disorder in the neonatal period characterized by inappropriate insulin secretion in the presence of hypoglycemia. Known etiologies include inactivating mutations of the KATP channel. Its management can be extremely complicated, and may involve medical therapy and surgery. We describe a full term baby male at one month-old without dysmorphic features or congenital anomalies, the baby was presented with asymptomatic hypoglycemia by routine screening at the age of 1.5 hour of life, he was treated with diazoxide for prolonged neonatal hyperinsulinism. Tolerance of diazoxide is usually excellent, but several adverse effects of this drug have been described. During diazoxide therapy he presented with tachycardia and low oxygen saturation, the baby became dyspnoeic with fluid retention and facial edema. Echocardiography showed moderate pulmonary hypertension. Diazoxide was withdrawn, his clinical status and pulmonary hypertension dramatically improved and returned to baseline.},
 year = {2015}
}

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TY  - JOUR
T1  - Development of Pulmonary Hypertension in an Infant Treated With Diazoxide for Hyperinsulinism, a Case Report and Literature Review
AU  - Albahri Ziad
AU  - Lukeš Antonín
AU  - Mynářová Kateřina
AU  - Krylová Kateřina
AU  - Al Mawiri Abdul
AU  - Hussain Khaled
AU  - Neumann David
Y1  - 2015/07/23
PY  - 2015
N1  - https://doi.org/10.11648/j.ajp.20150101.11
DO  - 10.11648/j.ajp.20150101.11
T2  - American Journal of Pediatrics
JF  - American Journal of Pediatrics
JO  - American Journal of Pediatrics
SP  - 1
EP  - 3
PB  - Science Publishing Group
SN  - 2472-0909
UR  - https://doi.org/10.11648/j.ajp.20150101.11
AB  - Congenital hyperinsulinism (CHI) is heterogeneous disorder in the neonatal period characterized by inappropriate insulin secretion in the presence of hypoglycemia. Known etiologies include inactivating mutations of the KATP channel. Its management can be extremely complicated, and may involve medical therapy and surgery. We describe a full term baby male at one month-old without dysmorphic features or congenital anomalies, the baby was presented with asymptomatic hypoglycemia by routine screening at the age of 1.5 hour of life, he was treated with diazoxide for prolonged neonatal hyperinsulinism. Tolerance of diazoxide is usually excellent, but several adverse effects of this drug have been described. During diazoxide therapy he presented with tachycardia and low oxygen saturation, the baby became dyspnoeic with fluid retention and facial edema. Echocardiography showed moderate pulmonary hypertension. Diazoxide was withdrawn, his clinical status and pulmonary hypertension dramatically improved and returned to baseline.
VL  - 1
IS  - 1
ER  -

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Author Information

Albahri Ziad

Department of Pediatrics, Faculty Hospital, Charles University, Hradec Kralove, Czech Republic
Lukeš Antonín
Mynářová Kateřina
Krylová Kateřina
Al Mawiri Abdul

Internal medicine- Faculty Hospital, Charles University, Hradec Kralove, Czech Republic
Hussain Khaled

Department of Paediatric Endocrinology, Institute of Child Health, University College, London, United Kingdom
Neumann David

Department of Pediatrics, Faculty Hospital, Charles University, Hradec Kralove, Czech Republic

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APA Style

Albahri Ziad, Lukeš Antonín, Mynářová Kateřina, Krylová Kateřina, Al Mawiri Abdul, et al. (2015). Development of Pulmonary Hypertension in an Infant Treated With Diazoxide for Hyperinsulinism, a Case Report and Literature Review. American Journal of Pediatrics, 1(1), 1-3. https://doi.org/10.11648/j.ajp.20150101.11

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ACS Style

Albahri Ziad; Lukeš Antonín; Mynářová Kateřina; Krylová Kateřina; Al Mawiri Abdul, et al. Development of Pulmonary Hypertension in an Infant Treated With Diazoxide for Hyperinsulinism, a Case Report and Literature Review. Am. J. Pediatr. 2015, 1(1), 1-3. doi: 10.11648/j.ajp.20150101.11

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AMA Style

Albahri Ziad, Lukeš Antonín, Mynářová Kateřina, Krylová Kateřina, Al Mawiri Abdul, et al. Development of Pulmonary Hypertension in an Infant Treated With Diazoxide for Hyperinsulinism, a Case Report and Literature Review. Am J Pediatr. 2015;1(1):1-3. doi: 10.11648/j.ajp.20150101.11

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@article{10.11648/j.ajp.20150101.11,
  author = {Albahri Ziad and Lukeš Antonín and Mynářová Kateřina and Krylová Kateřina and Al Mawiri Abdul and Hussain Khaled and Neumann David},
  title = {Development of Pulmonary Hypertension in an Infant Treated With Diazoxide for Hyperinsulinism, a Case Report and Literature Review},
  journal = {American Journal of Pediatrics},
  volume = {1},
  number = {1},
  pages = {1-3},
  doi = {10.11648/j.ajp.20150101.11},
  url = {https://doi.org/10.11648/j.ajp.20150101.11},
  eprint = {https://article.sciencepublishinggroup.com/pdf/10.11648.j.ajp.20150101.11},
  abstract = {Congenital hyperinsulinism (CHI) is heterogeneous disorder in the neonatal period characterized by inappropriate insulin secretion in the presence of hypoglycemia. Known etiologies include inactivating mutations of the KATP channel. Its management can be extremely complicated, and may involve medical therapy and surgery. We describe a full term baby male at one month-old without dysmorphic features or congenital anomalies, the baby was presented with asymptomatic hypoglycemia by routine screening at the age of 1.5 hour of life, he was treated with diazoxide for prolonged neonatal hyperinsulinism. Tolerance of diazoxide is usually excellent, but several adverse effects of this drug have been described. During diazoxide therapy he presented with tachycardia and low oxygen saturation, the baby became dyspnoeic with fluid retention and facial edema. Echocardiography showed moderate pulmonary hypertension. Diazoxide was withdrawn, his clinical status and pulmonary hypertension dramatically improved and returned to baseline.},
 year = {2015}
}

Copy | Download

TY  - JOUR
T1  - Development of Pulmonary Hypertension in an Infant Treated With Diazoxide for Hyperinsulinism, a Case Report and Literature Review
AU  - Albahri Ziad
AU  - Lukeš Antonín
AU  - Mynářová Kateřina
AU  - Krylová Kateřina
AU  - Al Mawiri Abdul
AU  - Hussain Khaled
AU  - Neumann David
Y1  - 2015/07/23
PY  - 2015
N1  - https://doi.org/10.11648/j.ajp.20150101.11
DO  - 10.11648/j.ajp.20150101.11
T2  - American Journal of Pediatrics
JF  - American Journal of Pediatrics
JO  - American Journal of Pediatrics
SP  - 1
EP  - 3
PB  - Science Publishing Group
SN  - 2472-0909
UR  - https://doi.org/10.11648/j.ajp.20150101.11
AB  - Congenital hyperinsulinism (CHI) is heterogeneous disorder in the neonatal period characterized by inappropriate insulin secretion in the presence of hypoglycemia. Known etiologies include inactivating mutations of the KATP channel. Its management can be extremely complicated, and may involve medical therapy and surgery. We describe a full term baby male at one month-old without dysmorphic features or congenital anomalies, the baby was presented with asymptomatic hypoglycemia by routine screening at the age of 1.5 hour of life, he was treated with diazoxide for prolonged neonatal hyperinsulinism. Tolerance of diazoxide is usually excellent, but several adverse effects of this drug have been described. During diazoxide therapy he presented with tachycardia and low oxygen saturation, the baby became dyspnoeic with fluid retention and facial edema. Echocardiography showed moderate pulmonary hypertension. Diazoxide was withdrawn, his clinical status and pulmonary hypertension dramatically improved and returned to baseline.
VL  - 1
IS  - 1
ER  -

Copy | Download