Treatment of relapsing Mycobacterium avium infection with interferon-gamma and interleukin-2 in an HIV-negative patient with low CD4 syndrome

Sternfeld, Thomas; Nigg, Axel; Belohradsky, Bernd H.; Bogner, Johannes R.

doi:10.1016/j.ijid.2009.08.004

Cited by 24 publications

(20 citation statements)

References 9 publications

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“…All of the above cases are summarized in a report of a 39-year-old man who had a Mycobacterium avium intracellulare infection that was treated with IL-2 for 35 months and who was apparently cured while his CD4 count increased from 365 to 470/mm 3 at the end of therapy [41]. A 64-year-old man with recurrent disseminated Mycobacterium avium intracellulare infection was also treated with IL-2 in three cycles of 5 consecutive days each every other month and had a favorable clinical outcome and CD4 number increases after each IL-2 cycle [42]. Finally, a recent report described a series of four ICL patients who received several courses of recombinant IL-2 for up to 3 years [18].…”

Section: Treatmentmentioning

confidence: 99%

“…Three patients with ICL were included in a study of IFN-γ in refractory disseminated non-tuberculous mycobacteria infection, and two of them had clinical improvement and CD4 count increases [43]. Two more patients with a potential benefit from IFN-γ treatment have been documented in the literature [16,42], whereas others had no clear benefit [37,38]. …”

Section: Treatmentmentioning

confidence: 99%

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Idiopathic CD4 lymphocytopenia: a case of missing, wandering or ineffective T cells

Zonios¹,

Sheikh

Sereti

2012

Arthritis Res Ther

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Idiopathic CD4 lymphocytopenia (ICL) is a presumed heterogenous syndrome with key element low CD4 T-cell counts (below 300/mm3) without evidence of HIV infection or other known immunodeficiency. The etiology, pathogenesis, and management of ICL remain poorly understood and inadequately defined. The clinical presentation can range from serious opportunistic infections to incidentally diagnosed asymptomatic individuals. Cryptococcal and non-tuberculous mycobacterial infections and progressive multifocal leukoencephalopathy are the most significant presenting infections, although the spectrum of opportunistic diseases can be similar to that in patients with lymphopenia and HIV infection. Malignancy is common and related to opportunistic pathogens with an oncogenic potential. Autoimmune diseases are also seen in ICL with an increased incidence. The etiology of ICL is unknown. Mechanisms implicated in CD4 reduction may include decreased production, increased destruction, and tissue sequestration. New distinct genetic defects have been identified in certain patients with ICL, supporting the hypothesis of the lack of a common etiology in this syndrome. The management of ICL is focused on the treatment of opportunistic infections, appropriate prophylactic antibiotics, and close monitoring. In selected patients with life-threatening infections or profound immunodeficiency, strategies to increase T-cell counts or enhance immune function could be considered and have included interleukin-2, interferon-gamma, interleukin-7, and hematopoietic stem cell transplantation. The prognosis is influenced by the accompanying opportunistic infections and may be affected by publication bias of severe cases with unfavorable outcomes. As newer laboratory investigation techniques are being developed and targeted experimental treatments become available, our comprehension and prognosis of this rare syndrome could be significantly improved.

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Section: Treatmentmentioning

confidence: 99%

Section: Treatmentmentioning

confidence: 99%

Idiopathic CD4 lymphocytopenia: a case of missing, wandering or ineffective T cells

Zonios¹,

Sheikh

Sereti

2012

Arthritis Res Ther

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“…31 Other studies have reported success with recombinant IL-2 with or without interferon-γ. 22,30,32,33 IL-7 also has attracted attention, because impaired CD4 T-cell responses to IL-7 in ICL might contribute to cell depletion 34 ; this cytokine was used in an open-label phase I and IIa clinical trial and 1 case of progressive multifocal leukoencephalopathy in ICL, with some benefit. 35,36 The choice of subjects for cytokine therapy and the parameters to be used for follow-up are unclear and further studies would be required to evaluate efficacy.…”

Section: Discussionmentioning

confidence: 99%

Idiopathic CD4 lymphocytopenia

Yarmohammadi

Cunningham‐Rundles

2017

Annals of Allergy, Asthma & Immunology

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Background Idiopathic CD4 lymphocytopenia (ICL) is a rare condition characterized by an unexplained deficit of circulating CD4 T cells leading to increased risk of serious opportunistic infections. The pathogenesis, etiology, clinical presentation, and best treatment options remain unclear. Objective To describe the clinical presentation, treatment strategies, and outcome of patients with ICL seen in a single referral center. Methods In a retrospective study, from January 1993 to January 2014, the demographic characteristics, clinical presentation, and treatments of patients diagnosed with ICL were reviewed. Results Twenty-four patients (14 female [58%] and 10 male [42%]) were evaluated. The mean age was 45 ± 17.6 years (range 7–76 years). Mean CD4 and CD8 T-cell counts at the time of diagnosis were 119 ± 84/mm3 (range 4–294/mm3) and 219 ± 258/mm3 (range 7–630/mm3), respectively. Seventeen patients (71%) had opportunistic infections, 4 (17%) had malignancies, and 3 (13%) had unexplained demyelinating disease and neurologic problems. Most patients had normal levels of immunoglobulins. Thirteen patients had abnormally low to absent response to phytohemagglutinin, concanavalin A, and antigens (candida and tetanus). Three patients had resolution of warts and 1 had mycobacterial lung infection on interleukin-2 with increases in CD4 count. The 11 patients on trimethoprim and sulfamethoxazole had no further hospital admissions for infections. Conclusion The pathogenesis of ICL remains unclear. Although only some patients are healthy, most patients present with opportunistic infections. There is no known standard treatment aside from prophylactic antibiotics.

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“…41 Pegylated IL-2 was given with successful control of MAC disease, improved pulmonary function, and slow improvement of lymphocyte number and function. 41 Pegylated IL-2 was given with successful control of MAC disease, improved pulmonary function, and slow improvement of lymphocyte number and function.…”

Section: Management Of Icl and Secondary Complicationsmentioning

confidence: 99%