Combined Treatment of Progressive Encephalitis in an X-linked Agammaglobulinemia Patient
-
Marco Antonio Yamazaki-Nakashimada
,
Patricia Herrera-Mora
,
Alfonso Mahrx-Bracho
,
Gabriela López-Herrera
,
Juan Carlos Bustamante-Ogando
,
Selma Cecilia Scheffler-Mendoza
Abstract
Most patients with X-linked agammaglobulinemia are susceptible to infections, while some cases also suffer from inflammatory or autoimmune complications. We describe a patient with progressive encephalitis who improved after the use of immunomodulatory treatment with corticosteroids, fluoxetine, and nitazoxanide. In most of the cases the evolution of the progressive encephalitis is complicated and catastrophic. Based on our experience and the review of the literature, we propose the use of this combined treatment to control this devastating complication.
1. Hernandez-Trujillo VP, Scalchunes C, Cunningham-Rundles C, Ochs HD, Bonilla FA, Paris K, et al. Autoimmunity and inflammation in X-linked agammaglobulinemia. J Clin Immunol. 2014;34(3):627-32.
2. Kasahara Y, Imamura M, Shin C, Shimizu H, Utsumi J, Hosokai R, et al. Fatal Progressive Meningoencephalitis Diagnosed in Two Members of a Family With X-Linked Agammaglobulinemia. Front. Pediatr. 2020;8:579.
3. Teramoto T, Kaneko H, Funato M, Sawa H, Nagashima K, Hirose Y, et al. Progressive multifocal leukoencephalopathy in a patient with X-linked agammaglobulinemia. Scand J Infect Dis. 2003;35(11-12):909-10.
4. Gall T, Hawley J, May Y. Progressive neurodegeneration in X-linked agammaglobulinemia (meeting abstract). Neurology 2019;95(15 Suppl):P1.7-005.
5. Tuzankina I, Kobeleva Y, Kiseleva N, Bolkov M, Reuter G, Maródi L. Cytotoxic T Lymphocytes Mediate Neuronal Injury in Patients With X-linked Agammaglobulinemia and Progressive Neurodegenerative Disease. Allergy. 2011;66:1617-8.
6. Domingo A, Schmidt TG, Barcelon E, Lukban M, Westenberger A, Klein C. X-linked agammaglobulinemia with hearing impairment, dystonia-parkinsonism, and progressive neurodegeneration. J Neurol. 2014;261:2225-7.
7. Sag AT, Saka E, Ozgur TT, Sanal O, Ayvaz DC, Elibol B, et al. Progressive Neurodegenerative Syndrome in a Patient With X-linked Agammaglobulinemia Receiving Intravenous Immunoglobulin Therapy. Cogn Behav Neurol. 2014;27:155-9.
8. Mohammadzadeh I, Yeganeh M, Khaledi M, Salehiomran MR, Aghamohammadi A, Rezaei N. Debilitating Progressive Encephalitis in a Patient With BTK Deficiency. Acta Microbiol Immunol Hung. 2012;59:335-42.
9. Saini AG, Radotra BD, Bhattarai D, Rawat A, Bhatia V. X-Linked Agammaglobulinemia With Chronic Meningoencephalitis: A Diagnostic Challenge. Indian Pediatr. 2021.15;58(2):169-173.
10. Frémond ML, Pérot P, Muth E, Cros G, Dumarest M, Mahlaoui N, et al. Next-Generation Sequencing for Diagnosis and Tailored Therapy: A Case Report of Astrovirus-Associated Progressive Encephalitis. J Pediatric Infect Dis Soc. 2015;4:e53-7.
11. Wilson MR, Suan D, Duggins A, Schubert RD, Khan LM, Sample HA, et al. A Novel Cause of Chronic Viral Meningoencephalitis: Cache Valley Virus. Ann Neurol. 2017;82(6):105-114.
12. Nakano A, Yamasaki R, Miyazaki S, Horiuchi N, Kunishige M, Mitsui T. Beneficial effect of steroid pulse therapy on acute viral encephalitis. Eur Neurol. 2003;50(4):225-9.
13. Gofshteyn J, Cárdenas AM, Bearden D. Treatment of Chronic Enterovirus Encephalitis With Fluoxetine in a Patient With X-Linked Agammaglobulinemia. Pediatr Neurol. 2016;64:94-98.
14. Chetty K, Cheng I, Kaliakatsos M, Gonzalez-Granado LI, Klapsa D, Martin J, et al. Case report: Novel treatment regimen for enterovirus encephalitis in SCID. Front Immunol. 2022;13:930031.
15. Sham L, Bitnun A, Branson H, Hazrati LN, Dell SD, Yeung RSM, et al. Treatment of rituximab-associated chronic CNS enterovirus using IVIg and fluoxetine. Neurology. 2019;92(19):916-18.
16. Hargest V, Sharp B, Livingston B, Cortez V, Schultz-Cherry S. Astrovirus Replication Is Inhibited by Nitazoxanide In Vitro and In Vivo. J Virol. 2020;94(5):e01706-19
17. Shakya A, Bhat HR, Ghosh SK. Update on Nitazoxanide: A Multifunctional Chemotherapeutic Agent. Curr Drug Discov Technol. 2018;15(3):201-13
18. Sukumaran S, Marzan K, Shaham B, Church JA. A Child With X-Linked Agammaglobulinemia and Enthesitis-Related Arthritis. Int J Rheumatol. 2011;2011:175973.
19. Han SP, Lin YF, Weng HY, Tsai SF, Fu LS. A Novel BTK Gene Mutation in a Child with Atypical X-Linked Agammaglobulinemia and Recurrent Hemophagocytosis: A Case Report. Front Immunol. 2019;10(2):1953.
20. Rivas-Larrauri F, Aguilar-Zanela L, Castro-Oteo P, Rosales-Hernandez LA, Otero-Mendoza F, López-Herrera G, et al. Kawasaki disease and immunodeficiencies in children: case reports and literature review. Rheumatol Int. 2019;39:1829-1838.
21. Barmettler S, Otani IM, Minhas J, Abraham RS, Chang Y, Dorsey MJ, Ballas ZK, et al. Gastrointestinal Manifestations in X-linked Agammaglobulinemia. J Clin Immunol. 2017;37(3):287-94.
22. Lavrador V, Correia F, Sampaio R, Candido C, Sameiro-Faria M, Marques L, Mota C. Membranoproliferative Glomerulonephritis and X-Linked Agammaglobulinemia: An Uncommon Association. Case Rep Pediatr. 2014;2014:480947.
23. Tan Q, Ren FL, Wang H. Pyoderma Gangrenosum in a Patient with X-Linked Agammaglobulinemia. Ann Dermatol. 2017;29(4):476-8.
24. Mao L, Kitani A, Hiejima E, Montgomery-Recht K, Zhou W, Fuss I, Wiestner A, Strober W. Bruton tyrosine kinase deficiency augments NLRP3 inflammasome activation and causes IL-1β-mediated colitis. J Clin Invest. 2020;130(21):1793-1807.
25. González-Serrano ME, Estrada-García I, Mogica-Martínez D, González-Garay A, López-Herrera G, Berrón-Ruiz L, et al. Increased proinflammatory cytokine production after lipopolysaccharide stimulation in patients with X-linked agammaglobulinemia. J Clin Immunol. 2012;32(15):967-74.
26. Ray S, Kathuria H, Chakravarty K, Rawat A, Takkar A, Mehta S, et al. Seronegative panencephalitis complicated by viral encephalomyelitis in a case of Good's syndrome - a neuropathological report. Int J Neurosci. 2020:1-6.
2. Kasahara Y, Imamura M, Shin C, Shimizu H, Utsumi J, Hosokai R, et al. Fatal Progressive Meningoencephalitis Diagnosed in Two Members of a Family With X-Linked Agammaglobulinemia. Front. Pediatr. 2020;8:579.
3. Teramoto T, Kaneko H, Funato M, Sawa H, Nagashima K, Hirose Y, et al. Progressive multifocal leukoencephalopathy in a patient with X-linked agammaglobulinemia. Scand J Infect Dis. 2003;35(11-12):909-10.
4. Gall T, Hawley J, May Y. Progressive neurodegeneration in X-linked agammaglobulinemia (meeting abstract). Neurology 2019;95(15 Suppl):P1.7-005.
5. Tuzankina I, Kobeleva Y, Kiseleva N, Bolkov M, Reuter G, Maródi L. Cytotoxic T Lymphocytes Mediate Neuronal Injury in Patients With X-linked Agammaglobulinemia and Progressive Neurodegenerative Disease. Allergy. 2011;66:1617-8.
6. Domingo A, Schmidt TG, Barcelon E, Lukban M, Westenberger A, Klein C. X-linked agammaglobulinemia with hearing impairment, dystonia-parkinsonism, and progressive neurodegeneration. J Neurol. 2014;261:2225-7.
7. Sag AT, Saka E, Ozgur TT, Sanal O, Ayvaz DC, Elibol B, et al. Progressive Neurodegenerative Syndrome in a Patient With X-linked Agammaglobulinemia Receiving Intravenous Immunoglobulin Therapy. Cogn Behav Neurol. 2014;27:155-9.
8. Mohammadzadeh I, Yeganeh M, Khaledi M, Salehiomran MR, Aghamohammadi A, Rezaei N. Debilitating Progressive Encephalitis in a Patient With BTK Deficiency. Acta Microbiol Immunol Hung. 2012;59:335-42.
9. Saini AG, Radotra BD, Bhattarai D, Rawat A, Bhatia V. X-Linked Agammaglobulinemia With Chronic Meningoencephalitis: A Diagnostic Challenge. Indian Pediatr. 2021.15;58(2):169-173.
10. Frémond ML, Pérot P, Muth E, Cros G, Dumarest M, Mahlaoui N, et al. Next-Generation Sequencing for Diagnosis and Tailored Therapy: A Case Report of Astrovirus-Associated Progressive Encephalitis. J Pediatric Infect Dis Soc. 2015;4:e53-7.
11. Wilson MR, Suan D, Duggins A, Schubert RD, Khan LM, Sample HA, et al. A Novel Cause of Chronic Viral Meningoencephalitis: Cache Valley Virus. Ann Neurol. 2017;82(6):105-114.
12. Nakano A, Yamasaki R, Miyazaki S, Horiuchi N, Kunishige M, Mitsui T. Beneficial effect of steroid pulse therapy on acute viral encephalitis. Eur Neurol. 2003;50(4):225-9.
13. Gofshteyn J, Cárdenas AM, Bearden D. Treatment of Chronic Enterovirus Encephalitis With Fluoxetine in a Patient With X-Linked Agammaglobulinemia. Pediatr Neurol. 2016;64:94-98.
14. Chetty K, Cheng I, Kaliakatsos M, Gonzalez-Granado LI, Klapsa D, Martin J, et al. Case report: Novel treatment regimen for enterovirus encephalitis in SCID. Front Immunol. 2022;13:930031.
15. Sham L, Bitnun A, Branson H, Hazrati LN, Dell SD, Yeung RSM, et al. Treatment of rituximab-associated chronic CNS enterovirus using IVIg and fluoxetine. Neurology. 2019;92(19):916-18.
16. Hargest V, Sharp B, Livingston B, Cortez V, Schultz-Cherry S. Astrovirus Replication Is Inhibited by Nitazoxanide In Vitro and In Vivo. J Virol. 2020;94(5):e01706-19
17. Shakya A, Bhat HR, Ghosh SK. Update on Nitazoxanide: A Multifunctional Chemotherapeutic Agent. Curr Drug Discov Technol. 2018;15(3):201-13
18. Sukumaran S, Marzan K, Shaham B, Church JA. A Child With X-Linked Agammaglobulinemia and Enthesitis-Related Arthritis. Int J Rheumatol. 2011;2011:175973.
19. Han SP, Lin YF, Weng HY, Tsai SF, Fu LS. A Novel BTK Gene Mutation in a Child with Atypical X-Linked Agammaglobulinemia and Recurrent Hemophagocytosis: A Case Report. Front Immunol. 2019;10(2):1953.
20. Rivas-Larrauri F, Aguilar-Zanela L, Castro-Oteo P, Rosales-Hernandez LA, Otero-Mendoza F, López-Herrera G, et al. Kawasaki disease and immunodeficiencies in children: case reports and literature review. Rheumatol Int. 2019;39:1829-1838.
21. Barmettler S, Otani IM, Minhas J, Abraham RS, Chang Y, Dorsey MJ, Ballas ZK, et al. Gastrointestinal Manifestations in X-linked Agammaglobulinemia. J Clin Immunol. 2017;37(3):287-94.
22. Lavrador V, Correia F, Sampaio R, Candido C, Sameiro-Faria M, Marques L, Mota C. Membranoproliferative Glomerulonephritis and X-Linked Agammaglobulinemia: An Uncommon Association. Case Rep Pediatr. 2014;2014:480947.
23. Tan Q, Ren FL, Wang H. Pyoderma Gangrenosum in a Patient with X-Linked Agammaglobulinemia. Ann Dermatol. 2017;29(4):476-8.
24. Mao L, Kitani A, Hiejima E, Montgomery-Recht K, Zhou W, Fuss I, Wiestner A, Strober W. Bruton tyrosine kinase deficiency augments NLRP3 inflammasome activation and causes IL-1β-mediated colitis. J Clin Invest. 2020;130(21):1793-1807.
25. González-Serrano ME, Estrada-García I, Mogica-Martínez D, González-Garay A, López-Herrera G, Berrón-Ruiz L, et al. Increased proinflammatory cytokine production after lipopolysaccharide stimulation in patients with X-linked agammaglobulinemia. J Clin Immunol. 2012;32(15):967-74.
26. Ray S, Kathuria H, Chakravarty K, Rawat A, Takkar A, Mehta S, et al. Seronegative panencephalitis complicated by viral encephalomyelitis in a case of Good's syndrome - a neuropathological report. Int J Neurosci. 2020:1-6.
| Files | ||
| Issue | Vol 22 No 5 (2023) | |
| Section | Brief Communication | |
| DOI | https://doi.org/10.18502/ijaai.v22i5.13999 | |
| Keywords | ||
| Agammaglobulinemia Immunomodulators Primary immunodeficiency diseases Neuroinflammatory diseases Therapy | ||
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This work is licensed under a Creative Commons Attribution-NonCommercial 4.0 International License. |
How to Cite
1.
Yamazaki-Nakashimada M, Herrera-Mora P, Mahrx-Bracho A, López-Herrera G, Bustamante-Ogando J, Scheffler-Mendoza S. Combined Treatment of Progressive Encephalitis in an X-linked Agammaglobulinemia Patient. Iran J Allergy Asthma Immunol. 2023;22(5):504-509.
