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Published in: Virology Journal 1/2022

Open Access 01-12-2022 | Opportunistic Infection | Review

Disseminated cryptococcosis with varicella-zoster virus coinfection of idiopathic CD4 + T lymphocytopenia: a case report and literature review

Authors: Li Fang, Junli Zhang, Fangfang Lv

Published in: Virology Journal | Issue 1/2022

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Abstract

Background

Idiopathic CD4 + T lymphocytopenia (ICL) is a rare immunodeficiency syndrome, unaccompanied by various opportunistic infections. Cryptococcus and varicella-zoster viruse are the most common opportunistic infections.

Method

We described a case of disseminated cryptococcosis with varicella-zoster virus coinfection in a patient with ICL and reviewed all published reports. A total of 26 cases with cryptococcal meningitis in ICL were enrolled.

Discussion

ICL remains poorly understood to clinicians. Patients with cryptococcal meningitis in ICL mostly suffered with headache and fever in a subacute or chronic period, while some patients might have atypical manifestations which makes a difficulty for early diagnosis. Some characteristics of cerebrospinal fluid can help to predict the prognosis of the disease. Cryptococcosis with varicella-zoster virus coinfection is rare but serious.

Conclusion

We recommed CD4 + T cells should be assessed in patients with unusual or recurrent infections. As the underlying pathophysiology is poorly understood, there is no standard therapy for ICL. Increased awareness of the disease and early prevention for CD4 reduction are needed.
Literature
1.
go back to reference Yarmohammadi H, Cunningham-Rundles C. Idiopathic CD4 lymphocytopenia: pathogenesis, etiologies, clinical presentations and treatment strategies. Ann Allergy Asthma Immunol. 2017;119(4):374–8.PubMedPubMedCentral Yarmohammadi H, Cunningham-Rundles C. Idiopathic CD4 lymphocytopenia: pathogenesis, etiologies, clinical presentations and treatment strategies. Ann Allergy Asthma Immunol. 2017;119(4):374–8.PubMedPubMedCentral
2.
go back to reference Centers for Disease Control (CDC). Unexplained CD4+ T-lymphocyte depletion in persons without evident HIV infection—United States. MMWR Morb Mortal Wkly Rep. 1992;41:541. Centers for Disease Control (CDC). Unexplained CD4+ T-lymphocyte depletion in persons without evident HIV infection—United States. MMWR Morb Mortal Wkly Rep. 1992;41:541.
3.
4.
go back to reference Rajasingham R, Smith RM, Park BJ, et al. Global burden of disease of HIV-associated cryptococcal meningitis: an updated analysis. Lancet Infect Dis. 2017;17:873–81.CrossRef Rajasingham R, Smith RM, Park BJ, et al. Global burden of disease of HIV-associated cryptococcal meningitis: an updated analysis. Lancet Infect Dis. 2017;17:873–81.CrossRef
5.
go back to reference Wilson LS, Reyes CM, Stolpman M, et al. The direct cost and incidence of systemic fungal infections. Value Health. 2002;5:26–34.PubMed Wilson LS, Reyes CM, Stolpman M, et al. The direct cost and incidence of systemic fungal infections. Value Health. 2002;5:26–34.PubMed
6.
go back to reference Gholamin M, Bazi A, Abbaszadegan MR. Idiopathic lymphocytopenia. Curr Opin Hematol. 2015;22:46.PubMed Gholamin M, Bazi A, Abbaszadegan MR. Idiopathic lymphocytopenia. Curr Opin Hematol. 2015;22:46.PubMed
7.
go back to reference Zonios DI, Falloon J, Bennett JE, et al. Idiopathic CD4+ lymphocytopenia: natural history and prognostic factors. Blood. 2008;112:287.PubMedPubMedCentral Zonios DI, Falloon J, Bennett JE, et al. Idiopathic CD4+ lymphocytopenia: natural history and prognostic factors. Blood. 2008;112:287.PubMedPubMedCentral
8.
go back to reference Ahmad DS, Esmadi M, Steinmann WC. Idiopathic CD4 lymphocytopenia: spectrum of opportunistic infections, malignancies, and autoimmune diseases. Avicenna J Med. 2013;3(2):37–47.PubMedPubMedCentral Ahmad DS, Esmadi M, Steinmann WC. Idiopathic CD4 lymphocytopenia: spectrum of opportunistic infections, malignancies, and autoimmune diseases. Avicenna J Med. 2013;3(2):37–47.PubMedPubMedCentral
9.
go back to reference Browne SK. Anticytokine autoantibodies in infectious diseases: pathogenesis and mechanisms-ScienceDirect. Lancet Infect Dis. 2010;10(12):875–85.PubMed Browne SK. Anticytokine autoantibodies in infectious diseases: pathogenesis and mechanisms-ScienceDirect. Lancet Infect Dis. 2010;10(12):875–85.PubMed
11.
go back to reference Kuo CY, Wang SY, Shih HP, et al. Disseminated cryptococcosis due to anti-granulocyte-macrophage colony-stimulating factor autoantibodies in the absence of pulmonary alveolar proteinosis. J Clin Immunol. 2017;37:143–52.PubMed Kuo CY, Wang SY, Shih HP, et al. Disseminated cryptococcosis due to anti-granulocyte-macrophage colony-stimulating factor autoantibodies in the absence of pulmonary alveolar proteinosis. J Clin Immunol. 2017;37:143–52.PubMed
12.
go back to reference Saijo T, Chen J, Chen SC, Rosen LB, Yi J, Sorrell TC, et al. Anti- granulocyte-macrophage colony-stimulating factor autoantibodies are a risk factor for central nervous system infection by Cryptococcus gattii in otherwise immunocompetent patients. MBio. 2014;5(2):e00912-e914.PubMedPubMedCentral Saijo T, Chen J, Chen SC, Rosen LB, Yi J, Sorrell TC, et al. Anti- granulocyte-macrophage colony-stimulating factor autoantibodies are a risk factor for central nervous system infection by Cryptococcus gattii in otherwise immunocompetent patients. MBio. 2014;5(2):e00912-e914.PubMedPubMedCentral
13.
go back to reference Sim BNH, Hui LY, Krishnan D, et al. Idiopathic CD4 lymphopenia in a case of disseminated cryptococcosis with brain, vertebral spine and reproductive organ involvement. Clin Med (Lond). 2019;19(2):133–4. Sim BNH, Hui LY, Krishnan D, et al. Idiopathic CD4 lymphopenia in a case of disseminated cryptococcosis with brain, vertebral spine and reproductive organ involvement. Clin Med (Lond). 2019;19(2):133–4.
14.
go back to reference Eshwara VK, Garg R, Chandrashekhar GS, et al. Fatal Cryptococcus gattii meningitis with negative cryptococcal antigen test in a HIV-non-infected patient. Indian J Med Microbiol. 2018;36(3):439–40.PubMed Eshwara VK, Garg R, Chandrashekhar GS, et al. Fatal Cryptococcus gattii meningitis with negative cryptococcal antigen test in a HIV-non-infected patient. Indian J Med Microbiol. 2018;36(3):439–40.PubMed
15.
go back to reference Malone C, Gupta ND, Kothari A, Palacios E, Neitzschman H. Radiology case of the month: idiopathic CD4 lymphocytopenia. J La State Med Soc. 2017;169(3):85–7.PubMed Malone C, Gupta ND, Kothari A, Palacios E, Neitzschman H. Radiology case of the month: idiopathic CD4 lymphocytopenia. J La State Med Soc. 2017;169(3):85–7.PubMed
17.
go back to reference Shribman S, Noyce A, Gnanapavan S, et al. Cryptococcal meningitis in apparently immunocompetent patients: association with idiopathic CD4+ lymphopenia. Pract Neurol. 2018;18:166–9.PubMed Shribman S, Noyce A, Gnanapavan S, et al. Cryptococcal meningitis in apparently immunocompetent patients: association with idiopathic CD4+ lymphopenia. Pract Neurol. 2018;18:166–9.PubMed
18.
go back to reference Pavić I, Cekinović D, Begovac J, et al. Cryptococcus neoformans meningoencephalitis in a patient with idiopathic CD4+ T lymphocytopenia. Coll Antropol. 2013;37(2):619–23.PubMed Pavić I, Cekinović D, Begovac J, et al. Cryptococcus neoformans meningoencephalitis in a patient with idiopathic CD4+ T lymphocytopenia. Coll Antropol. 2013;37(2):619–23.PubMed
19.
go back to reference Sancesario G, Palmieri G, Viola G, et al. Difficulty diagnosing chronic cryptococcal meningitis in idiopathic CD4+ lymphocytopenia. Neurol Sci. 2011;32(3):519–24.PubMed Sancesario G, Palmieri G, Viola G, et al. Difficulty diagnosing chronic cryptococcal meningitis in idiopathic CD4+ lymphocytopenia. Neurol Sci. 2011;32(3):519–24.PubMed
20.
go back to reference Tsalik EL, Jaggers LB. Life-threatening asymptomatic incidentaloma: a case report of idiopathic CD4 lymphocytopenia and opportunistic infections. Am J Med Sci. 2010;340(2):158–9.PubMed Tsalik EL, Jaggers LB. Life-threatening asymptomatic incidentaloma: a case report of idiopathic CD4 lymphocytopenia and opportunistic infections. Am J Med Sci. 2010;340(2):158–9.PubMed
21.
go back to reference Sharma A, Lal V, Modi M, et al. Idiopathic CD4 lymphocytopenia presenting as refractory cryptococcal meningitis. Ann Indian Acad Neurol. 2010;13(2):136–8.PubMedPubMedCentral Sharma A, Lal V, Modi M, et al. Idiopathic CD4 lymphocytopenia presenting as refractory cryptococcal meningitis. Ann Indian Acad Neurol. 2010;13(2):136–8.PubMedPubMedCentral
22.
go back to reference Augustine R, Khalid M, Misri ZK, et al. Idiopathic CD4+ T-lymphocytopenia-a diagnostic dilemma. J Assoc Physicians India. 2010;58:45–7.PubMed Augustine R, Khalid M, Misri ZK, et al. Idiopathic CD4+ T-lymphocytopenia-a diagnostic dilemma. J Assoc Physicians India. 2010;58:45–7.PubMed
23.
go back to reference Augusto E, Raguenaud ME, Kim C, et al. Idiopathic CD4+ T-lymphocytopenia with cryptococcal meningitis: first case report from Cambodia. Trop Doct. 2009;39(3):176–7.PubMed Augusto E, Raguenaud ME, Kim C, et al. Idiopathic CD4+ T-lymphocytopenia with cryptococcal meningitis: first case report from Cambodia. Trop Doct. 2009;39(3):176–7.PubMed
24.
go back to reference Yilmaz-Demirdag Y, Wilson B, Lowery-Nordberg M, et al. Interleukin-2 treatment for persistent cryptococcal meningitis in a child with idiopathic CD4(+) T lymphocytopenia. Allergy Asthma Proc. 2008;29(4):421–4.PubMed Yilmaz-Demirdag Y, Wilson B, Lowery-Nordberg M, et al. Interleukin-2 treatment for persistent cryptococcal meningitis in a child with idiopathic CD4(+) T lymphocytopenia. Allergy Asthma Proc. 2008;29(4):421–4.PubMed
25.
go back to reference Juhi T, BibhaBati M, Aradhana B, et al. Cryptococcal meningitis in a tertiary care hospital. Nippon Ishinkin Gakkai Zasshi. 2009;50(2):95–9. Juhi T, BibhaBati M, Aradhana B, et al. Cryptococcal meningitis in a tertiary care hospital. Nippon Ishinkin Gakkai Zasshi. 2009;50(2):95–9.
26.
go back to reference Jha S, Ghosh P, Agarwal V. Cryptococcal meningitis unmasking idiopathic CD4 lymphocytopenia. Neurol India. 2007;55(3):312–4.PubMed Jha S, Ghosh P, Agarwal V. Cryptococcal meningitis unmasking idiopathic CD4 lymphocytopenia. Neurol India. 2007;55(3):312–4.PubMed
27.
go back to reference Salit RB, Hankey KG, Yi R, et al. Detection of CD4(+) T-cell antibodies in a patient with idiopathic CD4 T lymphocytopenia and cryptococcal meningitis. Br J Haematol. 2010;139(1):133–7. Salit RB, Hankey KG, Yi R, et al. Detection of CD4(+) T-cell antibodies in a patient with idiopathic CD4 T lymphocytopenia and cryptococcal meningitis. Br J Haematol. 2010;139(1):133–7.
28.
go back to reference Diamantis PK, Zacharenia S, Ireni K, et al. Idiopathic CD4+ T lymphocytopenia disclosed by recurrent cryptococcal meningitis. First case report from Greece. Int J Infect Dis. 2005;9:347–8. Diamantis PK, Zacharenia S, Ireni K, et al. Idiopathic CD4+ T lymphocytopenia disclosed by recurrent cryptococcal meningitis. First case report from Greece. Int J Infect Dis. 2005;9:347–8.
29.
go back to reference Lepur D, Vranjican Z, Barsić B, et al. Idiopathic CD4+ T-lymphocytopenia-two unusual patients with cryptococcal meningitis. J Infect. 2005;51(2):E15–8.PubMed Lepur D, Vranjican Z, Barsić B, et al. Idiopathic CD4+ T-lymphocytopenia-two unusual patients with cryptococcal meningitis. J Infect. 2005;51(2):E15–8.PubMed
30.
go back to reference Netea MG, Brouwer AE, Hoogendoorn EH, et al. Two patients with cryptococcal meningitis and idiopathic CD4 lymphopenia: defective cytokine production and reversal by recombinant interferon-gamma therapy. Clin Infect Dis. 2004;39(9):e83–7.PubMed Netea MG, Brouwer AE, Hoogendoorn EH, et al. Two patients with cryptococcal meningitis and idiopathic CD4 lymphopenia: defective cytokine production and reversal by recombinant interferon-gamma therapy. Clin Infect Dis. 2004;39(9):e83–7.PubMed
32.
go back to reference Ho CL, Chang BC, Hsu GC, et al. Pulmonary cryptococcoma with CD4 lymphocytopenia and meningitis in an HIV-negative patient. Respir Med. 1998;92:120–2.PubMed Ho CL, Chang BC, Hsu GC, et al. Pulmonary cryptococcoma with CD4 lymphocytopenia and meningitis in an HIV-negative patient. Respir Med. 1998;92:120–2.PubMed
33.
go back to reference Yinnon AM, Bernard R, Ephaim S, et al. Invasive cryptococcosis in a family with epidermodysplasia verruciformis and idiopathic CD4 cell depletion. Clin Infect Dis Off Publ Infect Dis Soc Am. 1997;5:1252–3. Yinnon AM, Bernard R, Ephaim S, et al. Invasive cryptococcosis in a family with epidermodysplasia verruciformis and idiopathic CD4 cell depletion. Clin Infect Dis Off Publ Infect Dis Soc Am. 1997;5:1252–3.
34.
go back to reference Ramirez JA, Srinath L, Ahkee S, et al. HIV-negative “AIDS” in Kentucky: a case of idiopathic CD4+ lymphopenia and cryptococcal meningitis. South Med J. 1994;87(7):751–2.PubMed Ramirez JA, Srinath L, Ahkee S, et al. HIV-negative “AIDS” in Kentucky: a case of idiopathic CD4+ lymphopenia and cryptococcal meningitis. South Med J. 1994;87(7):751–2.PubMed
35.
36.
go back to reference Duncan RA, von Reyn CF, Alliegro GM, et al. Idiopathic CD4+ T-lymphocytopenia-four patients with opportunistic infections and no evidence of HIV infection. N Engl J Med. 1993;328(6):393–8.PubMed Duncan RA, von Reyn CF, Alliegro GM, et al. Idiopathic CD4+ T-lymphocytopenia-four patients with opportunistic infections and no evidence of HIV infection. N Engl J Med. 1993;328(6):393–8.PubMed
37.
go back to reference Zhu LP, Wu JQ, Xu B, et al. Cryptococcal meningitis in non-HIV-infected patients in a Chinese tertiary care hospital, 1997–2007. Med Mycol. 2010;48(4):570–9.PubMed Zhu LP, Wu JQ, Xu B, et al. Cryptococcal meningitis in non-HIV-infected patients in a Chinese tertiary care hospital, 1997–2007. Med Mycol. 2010;48(4):570–9.PubMed
38.
go back to reference Chen CH, Sy HN, Lin LJ, et al. Epidemiological characterization and prognostic factors in patients with confirmed cerebral cryptococcosis in central Taiwan. J Venom Anim Toxins Incl Trop Dis. 2015;21:12.PubMedPubMedCentral Chen CH, Sy HN, Lin LJ, et al. Epidemiological characterization and prognostic factors in patients with confirmed cerebral cryptococcosis in central Taiwan. J Venom Anim Toxins Incl Trop Dis. 2015;21:12.PubMedPubMedCentral
39.
go back to reference Pappas PG, Perfect JR, Cloud GA, et al. Cryptococcosis in human immunodeficiency virus-negative patients in the era of effective azole therapy. Clin Infect Dis. 2001;33(5):690–9.PubMed Pappas PG, Perfect JR, Cloud GA, et al. Cryptococcosis in human immunodeficiency virus-negative patients in the era of effective azole therapy. Clin Infect Dis. 2001;33(5):690–9.PubMed
40.
go back to reference Hung CW, Chang WN, Kung CT, et al. Predictors and long-term outcome of seizures in human immuno-deficiency virus (HIV)-negative cryptococcal meningitis. BMC Neurol. 2014;14:208.PubMedPubMedCentral Hung CW, Chang WN, Kung CT, et al. Predictors and long-term outcome of seizures in human immuno-deficiency virus (HIV)-negative cryptococcal meningitis. BMC Neurol. 2014;14:208.PubMedPubMedCentral
41.
go back to reference Wu JQ, Xu B, Ou XT, et al. Factors associated with mortality in cryptococcal meningitis. Zhonghua Yi Xue Za Zhi. 2010;90(1):33–7.PubMed Wu JQ, Xu B, Ou XT, et al. Factors associated with mortality in cryptococcal meningitis. Zhonghua Yi Xue Za Zhi. 2010;90(1):33–7.PubMed
42.
go back to reference Diamond RD, Bennett JE. Prognostic factors in cryptococcal meningitis. A study in 111 cases. Ann Intern Med. 1974;80(2):176–81.PubMed Diamond RD, Bennett JE. Prognostic factors in cryptococcal meningitis. A study in 111 cases. Ann Intern Med. 1974;80(2):176–81.PubMed
43.
go back to reference Lee YC, Wang JT, Sun HY, et al. Comparisons of clinical features and mortality of cryptococcal meningitis between patients with and without human immunodeficiency virus infection. J Microbiol Immunol Infect. 2011;44(5):338–45.PubMed Lee YC, Wang JT, Sun HY, et al. Comparisons of clinical features and mortality of cryptococcal meningitis between patients with and without human immunodeficiency virus infection. J Microbiol Immunol Infect. 2011;44(5):338–45.PubMed
44.
go back to reference Anekthananon T, Manosuthi W, Chetchotisakd P, et al. Predictors of poor clinical outcome of cryptococcal meningitis in HIV-infected patients. Int J STD AIDS. 2011;22(11):665–70.PubMed Anekthananon T, Manosuthi W, Chetchotisakd P, et al. Predictors of poor clinical outcome of cryptococcal meningitis in HIV-infected patients. Int J STD AIDS. 2011;22(11):665–70.PubMed
45.
go back to reference Cabello Úbeda A, Fortes Alen J, Gadea I, et al. Cryptococcal meningoencephalitis. Epidemiology and mortality risk factors in pre- and post-HAART era. Med Clin (Barc). 2016;146(9):397–401. Cabello Úbeda A, Fortes Alen J, Gadea I, et al. Cryptococcal meningoencephalitis. Epidemiology and mortality risk factors in pre- and post-HAART era. Med Clin (Barc). 2016;146(9):397–401.
46.
go back to reference Hakyemez IN, Erdem H, Beraud G, et al. Prediction of unfavorable outcomes in cryptococcal meningitis: results of the multicenter Infectious Diseases International Research Initiative (ID-IRI) cryptococcal meningitis study. Eur J Clin Microbiol Infect Dis. 2018;37(7):1241–2.PubMed Hakyemez IN, Erdem H, Beraud G, et al. Prediction of unfavorable outcomes in cryptococcal meningitis: results of the multicenter Infectious Diseases International Research Initiative (ID-IRI) cryptococcal meningitis study. Eur J Clin Microbiol Infect Dis. 2018;37(7):1241–2.PubMed
47.
go back to reference Zonios DI, Falloon J, Huang CY, et al. Cryptococcosis and idiopathic CD4 lymphocytopenia. Medicine. 2007;86(2):78–92.PubMed Zonios DI, Falloon J, Huang CY, et al. Cryptococcosis and idiopathic CD4 lymphocytopenia. Medicine. 2007;86(2):78–92.PubMed
Metadata
Title
Disseminated cryptococcosis with varicella-zoster virus coinfection of idiopathic CD4 + T lymphocytopenia: a case report and literature review
Authors
Li Fang
Junli Zhang
Fangfang Lv
Publication date
01-12-2022
Publisher
BioMed Central
Published in
Virology Journal / Issue 1/2022
Electronic ISSN: 1743-422X
DOI
https://doi.org/10.1186/s12985-022-01765-7

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