Cases reported "Lymphopenia"

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1/19. Treatment of idiopathic CD4 T lymphocytopenia with IL-2.

    Idiopathic CD4 T lymphocytopenia (ICL) is an unusual immune defect in which there is an unexplained deficit of CD4 T cells, leading to fungal, parasitic or other serious opportunistic infections. Current treatment efforts are directed at eliminating infections. Here we describe the use of a novel treatment, subcutaneous polyethylene glycol (PEG)-IL-2 injections, in a woman with this disorder, who had chronic severe mycobacterial disease which led to repeated hospitalizations, and advancing respiratory insufficiency. For this patient, PEG-IL-2, 50 000 U/m2, has been given by weekly subcutaneous injections for 5.5 years. This treatment has resulted in marked (and still continuing) long-term immunological improvement with normalized T cell functions and increased CD4 cell numbers. She has had substantial clinical improvement with clearing of mycobacterial disease, reducing hospitalizations and improved lung functions. The improvement seen in this patient suggests that low-dose IL-2 is a safe and practical therapy, which might be useful in other subjects with this potentially serious immune defect.
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ranking = 1
keywords = idiopathic
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2/19. Isolated human papillomavirus 18-positive extragenital bowenoid papulosis and idiopathic CD4 lymphocytopenia.

    We report a case of isolated extragenital bowenoid papulosis (BP) in a young man with an idiopathic low CD4 count. The lesions occurred on the dorsal aspect of his left middle finger and were not associated with genital involvement. polymerase chain reaction studies of a biopsy demonstrated human papillomavirus 18. As far as we are aware, this is the first documented case of BP (genital or extragenital) associated with idiopathic CD4 lymphocytopenia.
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ranking = 1.5
keywords = idiopathic
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3/19. Muscular-skeletal cryptococcosis in a patient with idiopathic CD4 lymphopenia.

    A healthy 27-year-old woman presented, four months after childbirth, ingravescent pain and claudication of the left lower limb. magnetic resonance imaging of the lumbosacral and iliac regions showed widespread muscular-skeletal lesions. The patient underwent surgery; cryptococcus neoformans was isolated from surgical samples. Liposomal amphotericin b, fluconazole and itraconazole were administered. Laboratory findings showed lymphocytopenia, with reduction of CD4 lymphocytes (23 cells per cubic millimeter) in the absence of hiv infection and any other defined immunodeficiency. This is a rare case of muscular-skeletal cryptococcal infection isolated in a subject affected with idiopathic CD4 lymphocytopenia.
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ranking = 1.25
keywords = idiopathic
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4/19. Diffuse large cell lymphoma and t(8;22) (q24;q11) in a patient with idiopathic CD4 T-lymphopenia.

    We describe a unique case of a patient with a three-year history of idiopathic CD4( ) T cell lymphopenia (hiv negative) who presented with stage IV diffuse large cell non Hodgkin's lymphoma with t(8;22). Despite the severe lymphopenia, the patient tolerated intensive chemotherapy well and at 18 months, remains in complete remission.
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ranking = 1.25
keywords = idiopathic
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5/19. Idiopathic CD4 T cell lymphocytopenia with the absence of B cells and CD8 28 cells in peripheral blood.

    The absence of B cells and a severe decrease in CD8 28 cells were observed in two female children with CD4 T cell lymphocytopenia. Idiopathic (primary) CD4 lymphocytopenia is a rare entity and its pathogenesis and genetics are not yet known. The literature was reviewed, in particular for severe alterations in B and CD8 28 cells and for the role of nf-kappa b and p56 (lck) in the immunopathogenesis. Whether the underlying mechanism in idiopathic CD4 lymphocytopenia is found or not, these patients who present with severe symptoms of a combined immunodeficiency must be treated with intravenous immunoglobin regularly until they have a compatible donor for bone marrow transplantation.
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ranking = 0.25
keywords = idiopathic
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6/19. Recurrent 'sterile' verrucous cyst abscesses and epidermodysplasia verruciformis-like eruption associated with idiopathic CD4 lymphopenia.

    rupture of follicular (epidermoid) cysts is believed to be the consequence of bacterial infection. We report a 24-year-old man with idiopathic CD4 lymphopenia and chronic mycobacterium avium intracellulare infection who developed multiple, recurring painful abscesses over the distal extremities that increased in number and severity when systemic steroid and interferon-gamma treatment was instituted for interstitial lung disease. Cultures were consistently negative for microorganisms, but pathological examination revealed ruptured epidermoid cyst walls with human papillomavirus (HPV) viropathic changes (keratinocytes with perinuclear halos and abundant basophilic keratohyaline granules). Cutaneous examination showed numerous, widespread flat-topped papules and achromic macules over the extremities, head and neck. Nested polymerase chain reaction analysis for HPV dna revealed that the abscess-related cyst walls harboured epidermodysplasia verruciformis (EV)-associated HPV types 20, 24, alb-7 (AY013872) and 80. His cutaneous lesions harboured HPV types 3, 8 and 80. Similar to past reports, our patient developed an EV-like eruption in the setting of immunodeficiency. In this instance, EV-associated HPV infection of the follicular infundibular epithelium or pre-existing cysts in the setting of immunodeficiency may have led to cystic growth, rupture and subsequent painful inflammation.
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ranking = 1.25
keywords = idiopathic
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7/19. A new syndrome of long-term idiopathic, severe CD4 lymphocytopenia: isolated paraparesis and conjunctival ischemic microangiopathy.

    An extraordinary case report of an adult patient followed-up for a decade with an extremely severe idiopathic CD4 T-lymphocytopenia (as expressed by an absolute CD4 count of 8-25 cells/microL), associated with an isolated paraparesis and a conjunctival ischemic microangiopathy is described, and discussed on the grounds of the available literature. Despite such a severe and prolonged immunodeficiency, no opportunistic disease occurred, in a observation period longer than ever reported to date. The neurological disorder was diagnosed concurrently with idiopathic CD4 lymphocyte depletion, while the ocular complication occurred two years later, but remained stable thereafter. Both disorders remained stable during the subsequent eight years. Despite extensive and repeated instrumental and laboratory workout, only very limited immunological abnormalities were detected (besides the extremely low CD4 lymphocyte count), and no apparent explaination was found for the disabling paraparesis syndrome. Idiopathic CD4 lymphocytopenia, whose pathogenesis deserves careful investigation, has been associated with a very broad spectrum of signs and symptoms, ranging from negligible or no disturbances, to severe lymphoproliferative disorders, different opportunistic infections, and other focal diseases, including neurological pathologies. However, the association of a long-lasting profound peripheral CD4 lymphocyte depletion in absence of any opportunistic infection or neoplasm, and isolated paraparesis and conjunctival microangiopathy, represents an absolutely unique finding, especially due to the apparently stable course of the above-mentioned syndrome.
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ranking = 1.5
keywords = idiopathic
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8/19. A case of pulmonary cryptococcosis with non-small cell lung cancer in idiopathic CD4 T-lymphocytopenia.

    cryptococcus neoformans commonly causes opportunistic infections in immunocompromised patients, especially in patients with AIDS. CD4 T-lymphocytopenia in AIDS indicates an increased risk of opportunistic infection and a decline in immunological function. Idiopathic CD4 T-lymphocytopenia (ICL) is characterized by depletions in the CD4 T-cell subsets, without evidence of hiv infection. Immunodeficiency can exist in the absence of laboratory evidence of hiv infection, and T-cell subsets should be evaluated in patients who present with unusual opportunistic infections. We report a case of pulmonary cryptococcosis and lung cancer in a patient with persistently low CD4 cell counts, without evidence of hiv infection.
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ranking = 1
keywords = idiopathic
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9/19. Localized cutaneous cryptococcosis in an immunosuppressed man.

    Isolated cutaneous cryptococcosis developed in a 62-year-old white man with an idiopathic deficiency of t-lymphocytes. The patient was healthy, except for the cutaneous infection, and displayed no other evidence of an immunodeficiency disorder. An immunologic workup should be conducted in all patients with cryptococcal infection, regardless of their apparent health status.
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ranking = 0.25
keywords = idiopathic
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10/19. Sinus histiocytosis with massive lymphadenopathy: a case with unusual skin involvement and a therapeutic response to vinblastine-loaded platelets.

    A patient with sinus histiocytosis with massive lymphadenopathy had unusual skin lesions and progressive internal involvement. Both aspects responded dramatically to the administration of vinblastine-loaded platelets. vinblastine-loaded, idiopathic thrombocytopenic purpura antibody-coated platelets are a rationale therapy for a disease characterized by the presence of actively phagocytosing histiocytes.
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ranking = 0.25
keywords = idiopathic
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