Toxoplasma chorioretinitis in primary myeloperoxidase MPO deficiency: A case report
D.V. Maltsev1, Cand Sc (Med); O.O. Hurzhii2, Ophthalmologist, MD
1 Institute of Experimental and Clinical Medicine at the Bogomolets NMU; Kyiv (Ukraine)
2 Ailas Clinic; Kyiv (Ukraine)
TO CITE THIS ARTICLE: Maltsev DV, Hurzhii OO. Toxoplasma chorioretinitis in primary myeloperoxidase MPO deficiency: A case report. J.ophthalmol.(Ukraine).2019;4:75-81. http://doi.org/10.31288/oftalmolzh201947581
We present a clinical case of primary partial myeloperoxidase (MPO) deficiency in a young female patient with recurrent episodes of opportunistic infections. Initially, she presented to the clinical immunologist (DVM) seeking help with her severe clinical manifestations of chronic fatigue syndrome. Blood polymerase chain reaction (PCR) was positive for Epstein Barr virus. Brain MRI showed signs of external hydrocephalus and bilateral hippocampal sclerosis. After double antiviral therapy with valacyclovir (3 g daily, orally) and recombinant human alpha-2b-interferon (3 mln units every other day intramuscularly) for 3 months, the blood PCR became negative, and clinical symptoms completely resolved. A year later, the patient presented again to the immunologist, but this time due to abruptly decreased vision in her right eye. She was referred to the ophthalmologist. There were ophthalmoscopic and fluorescein angiography (FA) signs of acute chorioretinitis in the right eye. Serological testing revealed increased IgM titers to Toxoplasma gondii. Blood PCR was positive for Toxoplasma gondii. The patient was diagnosed with acute Toxoplasma chorioretinitis based on the clinical-and-laboratory, ophthalmoscopic and FA findings, and was treated for toxoplasmosis with spiramycin (1.5 mln units diluted in 200 ml of 0.9% normal saline, daily for 14 days, intravenously, and thereafter, 1000 mg three times a day after meals, for 14 days, orally). In addition, she received continuous basic immunomodulating therapy with recombinant human interferon gamma (500,000 units every other day at bedtime for 6 months) for compensation of MPO deficiency, as per the national guidelines. This normalized the percentage content and functional activity of MPO in neutrophils, and enabled prevention of further opportunistic infection episodes. This case report demonstrates that treatment of eyes affected by opportunistic infections in immunocompromised patients requires cooperation between ophthalmologists and clinical immunologists.
Keywords: chorioretinitis, toxoplasmosis, myeloperoxidase deficiency
1.Nauseef WM. Diagnostic assays for myeloperoxidase and myeloperoxidase deficiency. Methods Mol Biol. 2014;1124:537-46. doi: 10.1007/978-1-62703-845-4_32.
2.Dinauer MC. Disorders of neutrophil function: an overview. Methods Mol Biol. 2014;1124:501-15. doi: 10.1007/978-1-62703-845-4_30.
3.Xiao X, Saha P, Yeoh BS, et al. Myeloperoxidase deficiency attenuates systemic and dietary iron-induced adverse effects. J Nutr Biochem. 2018 Dec;62:28-34. doi: 10.1016/j.jnutbio.2018.08.003.
4.Pahwa R, Jialal I. Myeloperoxidase Deficiency. In: StatPearls [Internet]. Treasure Island (FL): StatPearls Publishing; 2019-2018 Oct 27.
5.Kazmirchuk VE, Tsaryk VV, Maltsev DV. Myeloperoxidase deficiency – congenital and acquired disorders of neutrophil function. Fundamental and applied sciences today. 2014;3:32–6.
6.Domingues-Ferreira M, Levy A, Barros NC, et al. Case report of myeloperoxidase deficiency associated with disseminated paracoccidioidomycosis and peritoneal tuberculosis. Rev Soc Bras Med Trop. 2017 Jul-Aug;50(4):568-570. doi: 10.1590/0037-8682-0462-2016.
7.Grossl NA, Candel AG, Shrit A, Schumacher HR. Myeloperoxidase deficiency and severe sepsis. South Med J. 1993 Jul;86(7):832-6.
8.Ohno H. Association of primary myeloperoxidase deficiency and myeloproliferative neoplasm. Intern Med. 2010;49(22):2527-8.
9.Kalinski T, Jentsch-Ullrich K, Fill S, et al. Lethal candida sepsis associated with myeloperoxidase deficiency and pre-eclampsia. APMIS. 2007 Jul;115(7):875-80.
10.Milligan KL, Mann D, Rump A, et al. Complete Myeloperoxidase Deficiency: Beware the "False-Positive" Dihydrorhodamine Oxidation. J Pediatr. 2016 Sep;176:204-6. doi: 10.1016/j.jpeds.2016.05.047.
11.El Messaoudi K, Verheyden AM, Thiry L, et al. Human recombinant myeloperoxidase antiviral activity on cytomegalovirus. J Med Virol. 2002 Feb;66(2):218-23.
12.Chochola J, Yamaguchi Y, Moguilevsky N, et al. Virucidal effect of myeloperoxidase on human immunodeficiency virus type 1-infected T cells. Antimicrob Agents Chemother. 1994 May;38(5):969-72.
13.Patıroğlu T, Güngör HE, Belohradsky JS, et al. Myeloperoxidase deficiency: the secret under the flag of unstained cell. Turk J Haematol. 2013 Jun; 30(2): 232–3.
14.Bell AL, Markey GM, Alexander HD, et al. Myeloperoxidase deficiency in a patient with rheumatoid arthritis: oxygenation and radical activity by phagocytic cells. Br J Rheumatol. Br J Rheumatol. 1993 Feb;32(2):162-5.
15.Rudolph TK, Wipper S, Reiter B. et al. Myeloperoxidase deficiency preserves vasomotor function in humans. Eur Heart J. 2012 Jul;33(13):1625-34. doi: 10.1093/eurheartj/ehr193.
16.Kazimirchuk VE, Maltsev DV, Slobodin TN, Golovchenko IuI. [Parkinson's syndrome in young women with myeloperoxidase deficiency of phagocytes]. Mezhdunarodnyi Nevrologicheskii Zhurnal. 2011; 1(39):15-24. Russian.
17.d'Onofrio G, Mancini R, Vallone R, et al. Acquired neutrophil myeloperoxidase deficiency: an indicator of subclinical activation of blood coagulation? Blood Cells. 1983;9(3):455-66.
18.Russo AJ, Krigsman A, Jepson B, Wakefield A. Low serum myeloperoxidase in autistic children with gastrointestinal disease. Clin Exp Gastroenterol. 2009;2:85-94.
19.Kettle AJ, Winterbourn CC. Superoxide-dependent hydroxylation by myeloperoxidase. J Biol Chem. 1994 Jun 24;269(25):17146-51.
20.Albrett AM, Ashby LV, Dickerhof N, et al. Heterogeneity of hypochlorous acid production in individual neutrophil phagosomes revealed by a rhodamine-based probe. J Biol Chem. 2018 Oct 5;293(40):15715-15724. doi: 10.1074/jbc.RA118.004789.
21.Azarova LA, Vasilenko LP, Oblamskaia GV. [Hereditary myeloperoxidase deficiency: its diagnosis on the Technicon H.1 hematological analyzer]. Gematol. Transfuziol.1993 Feb;38(2):41-2. Russian.
22.Becker R, Pflüger KH. Myeloperoxidase deficiency: an epidemiological study and flow-cytometric detection of other granular enzymes in myeloperoxidase-deficient subjects. Ann Hematol. 1994 Oct;69(4):199-203.
23.Rośniak-Bąk K, Jeleń A, Bąk M. Analysis of Separation of White Blood Cells in Peripheral Blood as an Indicator of MPO Deficiency. Ann Clin Lab Sci. 2017 Aug;47(4):422-431.
24.Marchetti C, Patriarca P, Solero GP, et al. Genetic studies on myeloperoxidase deficiency in Italy. Jpn J Infect Dis. 2004 Oct;57(5):S10-2.
25.[Locally Adapted National Evidence-based Guidelines for Managing Myeloperoxidase Deficiency. State Expert Center of the Ministry of Health of Ukraine and Ukrainian Society of Specialists for Immunology, Allergology and Immunorehabilitation]. Ukrainian. Available at http://mtd.dec.gov.ua/images/dodatki/2016_609_DefMiel_Fago/2016_609_AKN_...
26.[Unified Clinical Pathway for Providing Care to Patients with Myeloperoxidase Deficiency]. 2016. Ukrainian. Available at http://mtd.dec.gov.ua/images/dodatki/2016_609_DefMiel_Fago/2016_609_YKPM...
27.Maltsev DV. [Efficacy of long-term continuous immunomodulating therapy with recombinant human interferon gamma for patients with clinically manifested forms of myeloperoxidase deficiency]. Immunologiia I allergologiia. 2015; 1:44-53. Russian.
28.Maltsev DV. 2015. The effectiveness of combined antiviral therapy in chronic mononucleosis caused by Epstein-Barr virus. European EBV meeting. Karolinska Institutet. Stockholm. p. 33.
29.Ramsaransing G, Teelken A, Prokopenko VM. Low leucocyte myeloperoxidase activity in patients with multiple sclerosis. J Neurol. Neurosurg Psychiatry. 2003;74(7):953-5.
The authors certify that they have no conflicts of interest in the subject matter or materials discussed in this manuscript.