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Published in: BMC Gastroenterology 1/2019

Open Access 01-12-2019 | Anemia | Case report

Anemia following zinc treatment for Wilson’s disease: a case report and literature review

Authors: Sha Cai, Jing-Yu Gong, Jing Yang, Jian-She Wang

Published in: BMC Gastroenterology | Issue 1/2019

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Abstract

Background

Zinc therapy is considered an effective and safe treatment for Wilson’s disease. Hypocupremia-related anemia is rarely reported after long-term zinc administration or combination therapy with copper-chelating agent.

Case presentation

We herein report a 12-year-old girl with pre-symptomatic Wilson’s disease diagnosed 5 years ago who presented with severe anemia after high-dose oral zinc for 4 years and 4 months. Her hemoglobin was gradually restored to the normal range after the adjustment of zinc dose and diet therapy for 4 months. A review of the literature revealed eight patients with hypocupremia-associated anemia following zinc therapy for Wilson’s disease, including 7 adults and 1 child. The only child patient was a 16-year-old boy, in whom the zinc therapy was succession to penicillamine administration.

Conclusions

This is the first report worldwide that a child developed severe anemia following high-dose single zinc administration for Wilson’s disease. It highlights the importance of regular follow-up during zinc treatment and the involvement of specialists in the long-term management of Wilson’s disease. We hope that this will alert pediatricians the issue of zinc over-treatment.
Literature
1.
go back to reference Walshe JM. History of Wilson's disease:1912 to 2000. Mov Disorder. 2006;21:142–7.CrossRef Walshe JM. History of Wilson's disease:1912 to 2000. Mov Disorder. 2006;21:142–7.CrossRef
2.
go back to reference Bearn AG. A genetical analysis of thirty families with Wilson’s disease (hepatolenticular degeneration). Ann Hum Genet. 1960;24:33–43.CrossRef Bearn AG. A genetical analysis of thirty families with Wilson’s disease (hepatolenticular degeneration). Ann Hum Genet. 1960;24:33–43.CrossRef
3.
go back to reference Roberts EA, Schilsky ML. Diagnosis and treatment of Wilson disease:an update. Hepatology. 2008;47(6):2089–111.CrossRef Roberts EA, Schilsky ML. Diagnosis and treatment of Wilson disease:an update. Hepatology. 2008;47(6):2089–111.CrossRef
4.
go back to reference Ala A, Walker AP, Ashkan K, Dooley JS, Schilsky ML. Wilson’s disease. Lancet. 2007;369(9559):397–408.CrossRef Ala A, Walker AP, Ashkan K, Dooley JS, Schilsky ML. Wilson’s disease. Lancet. 2007;369(9559):397–408.CrossRef
5.
go back to reference Lorincz MT. Neurologic Wilson’s disease. Ann N Y Acad Sci. 2010;1184:173–87.CrossRef Lorincz MT. Neurologic Wilson’s disease. Ann N Y Acad Sci. 2010;1184:173–87.CrossRef
6.
go back to reference Wilson SAK. Progressive lenticular degeneration:a familial nervous disease associated with cirrhosis of the liver. Brain. 1912;34:295–507.CrossRef Wilson SAK. Progressive lenticular degeneration:a familial nervous disease associated with cirrhosis of the liver. Brain. 1912;34:295–507.CrossRef
7.
go back to reference Wiernicka A, Jańczyk W, Dądalski M, Avsar Y, Schmidt H, Socha P. Gastrointestinal side effects in children with Wilson's disease treated with zinc sulphate. World J Gastroenterol. 2013;19(27):4356–62.CrossRef Wiernicka A, Jańczyk W, Dądalski M, Avsar Y, Schmidt H, Socha P. Gastrointestinal side effects in children with Wilson's disease treated with zinc sulphate. World J Gastroenterol. 2013;19(27):4356–62.CrossRef
8.
go back to reference Lang CJ, Rabas-Kolominsky P, Engelhardt A, Kobras G, Konig HJ. Fatal deterioration of Wilson’s disease after institution of oral zinc therapy. Arch Neurol. 1993;50:1007–8.CrossRef Lang CJ, Rabas-Kolominsky P, Engelhardt A, Kobras G, Konig HJ. Fatal deterioration of Wilson’s disease after institution of oral zinc therapy. Arch Neurol. 1993;50:1007–8.CrossRef
9.
go back to reference Walshe JM, Munro NA. Zinc-induced deterioration in Wilson's disease aborted by treatment with penicillamine, dimercaprol, and a novel zero copper diet. Arch Neurol. 1995;52:10–1.CrossRef Walshe JM, Munro NA. Zinc-induced deterioration in Wilson's disease aborted by treatment with penicillamine, dimercaprol, and a novel zero copper diet. Arch Neurol. 1995;52:10–1.CrossRef
10.
go back to reference Linn FH, Houwen RH, van Hattum J, van der Kleij S, van Erpecum KJ. Long-term exclusive zinc monotherapy in symptomatic Wilson disease: experience in 17 patients. Hepatology. 2009;50(5):1442–52.CrossRef Linn FH, Houwen RH, van Hattum J, van der Kleij S, van Erpecum KJ. Long-term exclusive zinc monotherapy in symptomatic Wilson disease: experience in 17 patients. Hepatology. 2009;50(5):1442–52.CrossRef
11.
go back to reference Horvath J, Beris P, Giostra E, Martin PY, Burkhard PR. Zinc-induced copper deficiency in Wilson disease. J Neurol Neurosurg Psychiatry. 2010;81(12):1410–1.CrossRef Horvath J, Beris P, Giostra E, Martin PY, Burkhard PR. Zinc-induced copper deficiency in Wilson disease. J Neurol Neurosurg Psychiatry. 2010;81(12):1410–1.CrossRef
12.
go back to reference Dzieżyc K, Litwin T, Sobańska A, Członkowska A. Symptomatic copper deficiency in three Wilson's disease patients treated with zinc sulphate. Neurol Neurochir Pol. 2014;48(3):214–8.CrossRef Dzieżyc K, Litwin T, Sobańska A, Członkowska A. Symptomatic copper deficiency in three Wilson's disease patients treated with zinc sulphate. Neurol Neurochir Pol. 2014;48(3):214–8.CrossRef
13.
go back to reference Rau AR, Usha M, Mallya P, Rau AT. Cytopenia and bone marrow dysplasia in a case of Wilson’s disease. Indian J Hematol Blood Transfus. 2014;30(Suppl1):433–6.CrossRef Rau AR, Usha M, Mallya P, Rau AT. Cytopenia and bone marrow dysplasia in a case of Wilson’s disease. Indian J Hematol Blood Transfus. 2014;30(Suppl1):433–6.CrossRef
14.
go back to reference Cortese A, Zangaglia R, Lozza A, Piccolo G, Pacchetti C. Copper deficiency in Wilson's disease: peripheral neuropathy and myelodysplastic syndrome complicating zinc treatment. Mov Disord. 2011;26(7):1361–2.CrossRef Cortese A, Zangaglia R, Lozza A, Piccolo G, Pacchetti C. Copper deficiency in Wilson's disease: peripheral neuropathy and myelodysplastic syndrome complicating zinc treatment. Mov Disord. 2011;26(7):1361–2.CrossRef
15.
go back to reference Foubert-Samier A, Kazadi A, Rouanet M, Vital A, Lagueny A, Tison F, Meissner W. Axonal sensory motor neuropathy in copper-deficient Wilson's disease. Muscle Nerve. 2009;40(2):294–6.CrossRef Foubert-Samier A, Kazadi A, Rouanet M, Vital A, Lagueny A, Tison F, Meissner W. Axonal sensory motor neuropathy in copper-deficient Wilson's disease. Muscle Nerve. 2009;40(2):294–6.CrossRef
16.
go back to reference van den Hamer CJ, Hoogenraad TU. Copper deficiency in Wilson's disease. Lancet. 1989;2(8660):442.CrossRef van den Hamer CJ, Hoogenraad TU. Copper deficiency in Wilson's disease. Lancet. 1989;2(8660):442.CrossRef
17.
go back to reference Mohamed M, Johnston A, Maclaine Cross A, Sharma A. Reversible pancytopenia caused by severe copper deficiency in a patient with Wilson disease. Med J Aust. 2018;209:1112.CrossRef Mohamed M, Johnston A, Maclaine Cross A, Sharma A. Reversible pancytopenia caused by severe copper deficiency in a patient with Wilson disease. Med J Aust. 2018;209:1112.CrossRef
18.
go back to reference Nicastro E, Ranucci G, Vajro P, Vegnente A, Iorio R. Re-evaluation of the diagnostic criteria for Wilson disease in children with mild liver disease. Hepatology. 2010;52:1948–56.CrossRef Nicastro E, Ranucci G, Vajro P, Vegnente A, Iorio R. Re-evaluation of the diagnostic criteria for Wilson disease in children with mild liver disease. Hepatology. 2010;52:1948–56.CrossRef
19.
go back to reference Socha P, Janczyk W, Dhawan A, Baumann U, D'Antiga L, Tanner S, et al. Wilson's disease in children: a position paper by the hepatology committee of the European Society for Paediatric gastroenterology, hepatology and nutrition. J Pediatr Gastroenterol Nutr. 2018;66:334–44.CrossRef Socha P, Janczyk W, Dhawan A, Baumann U, D'Antiga L, Tanner S, et al. Wilson's disease in children: a position paper by the hepatology committee of the European Society for Paediatric gastroenterology, hepatology and nutrition. J Pediatr Gastroenterol Nutr. 2018;66:334–44.CrossRef
20.
go back to reference Chang H, Xu A, Chen Z, Zhang Y, Tian F, Li T. Long-term effects of a combination of D-penicillamine and zinc salts in the treatment of Wilson's disease in children. Exp Ther Med. 2013;5(4):1129–32.CrossRef Chang H, Xu A, Chen Z, Zhang Y, Tian F, Li T. Long-term effects of a combination of D-penicillamine and zinc salts in the treatment of Wilson's disease in children. Exp Ther Med. 2013;5(4):1129–32.CrossRef
21.
go back to reference Brewer GJ, Hill GM, Prasad AS, Cossack ZT, Rabbani P. Oral zinc therapy for Wilson’s disease. Ann Intern Med. 1983;99:314–9.CrossRef Brewer GJ, Hill GM, Prasad AS, Cossack ZT, Rabbani P. Oral zinc therapy for Wilson’s disease. Ann Intern Med. 1983;99:314–9.CrossRef
22.
go back to reference European Association for Study of Liver. EASL clinical practice guidelines:Wilson's disease. J Hepatol. 2012;56(3):671–85.CrossRef European Association for Study of Liver. EASL clinical practice guidelines:Wilson's disease. J Hepatol. 2012;56(3):671–85.CrossRef
23.
go back to reference Abuduxikuer K, Wang J-S. Zinc mono-therapy in pre-symptomatic Chinese children with Wilson disease: a single center, retrospective study. PLoS One. 2014;9(1):e86168.CrossRef Abuduxikuer K, Wang J-S. Zinc mono-therapy in pre-symptomatic Chinese children with Wilson disease: a single center, retrospective study. PLoS One. 2014;9(1):e86168.CrossRef
24.
go back to reference Scheinberg IH, Jaffe ME, Sternlieb I. The use of trientine in preventing the effects of interrupting penicillamine therapy in Wilson's disease. N Engl J Med. 1987;317:209–13.CrossRef Scheinberg IH, Jaffe ME, Sternlieb I. The use of trientine in preventing the effects of interrupting penicillamine therapy in Wilson's disease. N Engl J Med. 1987;317:209–13.CrossRef
25.
go back to reference Gollan JL, Hussein S, Hoffbrand AV, Sherlock S. Red cell aplasia following prolonged D-penicillamine therapy. J Clin Pathol. 1976;29:135–9.CrossRef Gollan JL, Hussein S, Hoffbrand AV, Sherlock S. Red cell aplasia following prolonged D-penicillamine therapy. J Clin Pathol. 1976;29:135–9.CrossRef
26.
go back to reference Harada M, Miyagawa K, Honma Y, Hiura M, Shibata M, Matsuhashi T, et al. Excess copper chelating therapy for Wilson disease induces anemia and liver dysfunction. Intern Med. 2011;50:1461–4.CrossRef Harada M, Miyagawa K, Honma Y, Hiura M, Shibata M, Matsuhashi T, et al. Excess copper chelating therapy for Wilson disease induces anemia and liver dysfunction. Intern Med. 2011;50:1461–4.CrossRef
27.
go back to reference Halfdanarson TR, Kumar N, Li CY, Phyliky RL, Hogan WJ. Hematological manifestations of copper deficiency:a retrospective review. Eur J Haematol. 2008;80:523–31.CrossRef Halfdanarson TR, Kumar N, Li CY, Phyliky RL, Hogan WJ. Hematological manifestations of copper deficiency:a retrospective review. Eur J Haematol. 2008;80:523–31.CrossRef
28.
go back to reference Gabreyes AA, Abbasi HN, Forbes KP, McQuaker G, Duncan A, Morrison I. Hypocupremia associated cytopenia and myelopathy:a national retrospective review. Eur J Haematol. 2013;90(1):1–9.CrossRef Gabreyes AA, Abbasi HN, Forbes KP, McQuaker G, Duncan A, Morrison I. Hypocupremia associated cytopenia and myelopathy:a national retrospective review. Eur J Haematol. 2013;90(1):1–9.CrossRef
29.
go back to reference Myint ZW, Oo TH, Thein KZ, Tun AM, Saeed H. Copper deficiency anemia: review article. Ann Hematol. 2018;97(9):1527–34.CrossRef Myint ZW, Oo TH, Thein KZ, Tun AM, Saeed H. Copper deficiency anemia: review article. Ann Hematol. 2018;97(9):1527–34.CrossRef
30.
go back to reference Klevay LM. “Myelodysplasia,” myeloneuropathy, and copper deficiency. Mayo Clin Proc. 2006;81(1):132 author reply 132.CrossRef Klevay LM. “Myelodysplasia,” myeloneuropathy, and copper deficiency. Mayo Clin Proc. 2006;81(1):132 author reply 132.CrossRef
31.
go back to reference Allred JW, Aulino JM. Hypocupremia-associated myelopathy. J Comput Assist Tomogr. 2007;31:157–9.CrossRef Allred JW, Aulino JM. Hypocupremia-associated myelopathy. J Comput Assist Tomogr. 2007;31:157–9.CrossRef
32.
go back to reference Merle U, Schaefer M, Ferenci P, Stremmel W. Clinical presentation, diagnosis and long-term outcome of Wilson’s disease:a cohort study. Gut. 2007;56(1):115–20.CrossRef Merle U, Schaefer M, Ferenci P, Stremmel W. Clinical presentation, diagnosis and long-term outcome of Wilson’s disease:a cohort study. Gut. 2007;56(1):115–20.CrossRef
Metadata
Title
Anemia following zinc treatment for Wilson’s disease: a case report and literature review
Authors
Sha Cai
Jing-Yu Gong
Jing Yang
Jian-She Wang
Publication date
01-12-2019
Publisher
BioMed Central
Published in
BMC Gastroenterology / Issue 1/2019
Electronic ISSN: 1471-230X
DOI
https://doi.org/10.1186/s12876-019-1038-5

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