Idiopathic Cd4+T-lymphocytopenia—two unusual patients with cryptococcal meningitis

Lepur, Dragan; Vranjican, Zoran; Baršić, Bruno; Himbele, J; Klinar, Igor

doi:10.1016/j.jinf.2004.07.002

Cited by 13 publications

(3 citation statements)

References 19 publications

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“…Some examples are: a decreased numbers of naive T cells (CD54RA+), a relative expansion of memory T cells (CD45 RO+) and a higher percentage of T regulatory cells, increased CD4 T-cell activation and turnover, increased apoptosis of CD4 lymphocytes, reduced lymphocytes proliferation response and restricted oligoclonal T cells antigen repertoire, disturbance in the signaling of T cells antigen receptor and deficient IL2 production 1,13 . These similarities between the ICL and immune CMV effects and the occurrence of CMV and EBV encephalitis, CMV digestive compromise and retinitis in some reported ICL cases 1,14,15 lead us to propose that some of the ICL cases reported before, could be really secondary to CMV or EBV or other ganciclovir sensible chronic human herpes virus. In summary, the rapid response of lymphocytopenia and CD4+ T lymphocytopenia in all 6 patients chronically infected by human herpes virus CMV plus EBV, with specific treatment of these viruses, is good evidence of a new associated clinical manifestation to chronic herpes virus infection by CMV with active replication or reinfection, EBV or herpes 6.…”

Section: Discussionmentioning

confidence: 86%

Profound CD4+ T lymphocytopenia in human immunodeficiency virus negative individuals, improved with anti-human herpes virus treatment

2012

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Lymphocytopenia and CD4+ T lymphocytopenia can be associated with many bacterial, fungal, parasite and viral infections. They can also be found in autoimmune and neoplastic diseases, common variable immunodeficiency syndrome, physical, psychological and traumatic stress, malnutrition and immunosuppressive therapy. Besides, they can also be brought into relation, without a known cause, with idiopathic CD4+ T lymphocytopenia. Among viral infections, the Retrovirus, specially the human immunodeficiency virus, is the most frequently cause. However, many acute viral infections, including cytomegalovirus and Epstein Barr virus can be associated with transient lymphocytopenia and CD4+ T lymphocytopenia. As is well known, transient lymphocytopenia and CD4+ T lymphocytopenia are temporary and overcome when the disease improves. Nonetheless, severe CD4+ T Lymphocytopenia associated with chronic infections by human herpes virus has not been reported. We describe 6 cases of human immunodeficiency virus negative patients, with chronic cytomegalovirus and Epstein Barr virus infections and profound lymphocytopenia with clinical symptoms of cellular immunodeficiency. These patients improved rapidly with ganciclovir or valganciclovir treatment. We claim here that it is important to consider the chronic human herpes virus infection in the differential diagnosis of profoundly CD4+ T lymphocytopenia etiology, when human immunodeficiency virus is absent, in order to start effective treatment and to determine, in future studies, the impact of chronic human herpes virus infection in human beings’ health.

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Section: Discussionmentioning

confidence: 86%

Profound CD4+ T lymphocytopenia in human immunodeficiency virus negative individuals, improved with anti-human herpes virus treatment

2012

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“…Only English language papers were reviewed. We found 258 cases diagnosed with ICL in 143 published papers[134567891011121314151617181920212223242526272829303132333435363738394041424344454647484950515253545556575859606162636465666768697071727374757677787980818283848586878889909192939495969798…”

Section: Methodsmentioning

confidence: 99%

Idiopathic CD4 Lymphocytopenia: Spectrum of opportunistic infections, malignancies, and autoimmune diseases

2013

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Idiopathic CD4 lymphocytopenia (ICL) was first defined in 1992 by the US Centers for Disease Control and Prevention (CDC) as the repeated presence of a CD4+ T lymphocyte count of fewer than 300 cells per cubic millimeter or of less than 20% of total T cells with no evidence of human immunodeficiency virus (HIV) infection and no condition that might cause depressed CD4 counts. Most of our knowledge about ICL comes from scattered case reports. The aim of this study was to collect comprehensive data from the previously published cases to understand the characteristics of this rare condition. We searched the PubMed database and Science Direct for case reports since 1989 for Idiopathic CD4 lymphocytopenia cases. We found 258 cases diagnosed with ICL in 143 published papers. We collected data about age, sex, pathogens, site of infections, CD4 count, CD8 count, CD4:CD8 ratio, presence of HIV risk factors, malignancies, autoimmune diseases and whether the patients survived or died. The mean age at diagnosis of first opportunistic infection (or ICL if no opportunistic infection reported) was 40.7 ± 19.2 years (standard deviation), with a range of 1 to 85. One-sixty (62%) patients were males, 91 (35.2%) were females, and 7 (2.7%) patients were not identified whether males or females. Risk factors for HIV were documented in 36 (13.9%) patients. The mean initial CD4 count was 142.6 ± 103.9/mm3 (standard deviation). The mean initial CD8 count was 295 ± 273.6/mm3 (standard deviation). The mean initial CD4:CD8 ratio was 0.6 ± 0.7 (standard deviation). The mean lowest CD4 count was 115.4 ± 87.1/mm3 (standard deviation). The majority of patients 226 (87.6%) had at least one infection. Cryptococcal infections were the most prevalent infections in ICL patients (26.6%), followed by mycobacterial infections (17%), candidal infections (16.2%), and VZV infections (13.1%). Malignancies were reported in 47 (18.1%) patients. Autoimmune diseases were reported in 37 (14.2%) patients.

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“…Cerebrospinal fluid were analysed in 22 cases with adequate information in Table 3 [ 13 – 19 , 21 – 24 , 26 , 27 , 29 , 31 – 36 ]. It showed a median leukocyte of 61 cells/μL (ranging 0–700), mainly constituted by lymphocytic, glucose of 39.1 mg/dL (ranging 1.98–87 mg/dL), below 40 mg/dL (1 mM/L = 18 mg/dL) in 50% of the patients, and protein of 116 mg/dL (ranging 20–266 mg/dL).…”

Section: Literature Review and Discussionmentioning

confidence: 99%

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

Zhang

2022

Virol J

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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.

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Idiopathic Cd4+T-lymphocytopenia—two unusual patients with cryptococcal meningitis

Cited by 13 publications

References 19 publications

Profound CD4+ T lymphocytopenia in human immunodeficiency virus negative individuals, improved with anti-human herpes virus treatment

Profound CD4+ T lymphocytopenia in human immunodeficiency virus negative individuals, improved with anti-human herpes virus treatment

Idiopathic CD4 Lymphocytopenia: Spectrum of opportunistic infections, malignancies, and autoimmune diseases

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

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