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1.
Medicine (Baltimore) ; 98(31): e16571, 2019 Aug.
Artigo em Inglês | MEDLINE | ID: mdl-31374024

RESUMO

RATIONALE: IgG4-related disease (IgG4-RD) is a systemic autoimmune disease and mixed cryoglobulinemia may be caused by autoimmune diseases. However, so far only 1 case of IgG4-RD complicated with mixed cryoglobulinemia is reported. Our case further confirms the close relationship between these 2 diseases. PATIENT CONCERNS: A 55-year-old female was admitted because of dry mouth and teeth falling off. DIAGNOSES: The patient was diagnosed as IgG4-related sialadenitis (IgG4-RS) complicated with type III mixed cryoglobulinemia. IgG4-RS was confirmed by elevated serum IgG4 levels and diffuse IgG4 plasmocyte infiltration and storiform fibrosis in the interstitium of labial gland. Type III mixed cryoglobulinemia was confirmed by positive serum cryoglobulins and no monoclonal immunoglobulin in serum and urine. INTERVENTIONS AND OUTCOMES: After treatment with prednisone and cyclophosphamide, serum cryoglobulins rapidly turned negative with the remission of IgG4-RS. LESSONS: Type III mixed cryoglobulinemia can be caused by IgG4-RS, and the underlying mechanisms need to be further explored.


Assuntos
Crioglobulinemia/complicações , Doença Relacionada a Imunoglobulina G4/complicações , Sialadenite/complicações , Crioglobulinemia/tratamento farmacológico , Feminino , Humanos , Doença Relacionada a Imunoglobulina G4/tratamento farmacológico , Imunossupressores/uso terapêutico , Pessoa de Meia-Idade , Sialadenite/tratamento farmacológico
2.
Medicine (Baltimore) ; 98(28): e16382, 2019 Jul.
Artigo em Inglês | MEDLINE | ID: mdl-31305439

RESUMO

RATIONALE: Type I monoclonal cryoglobulinemia is usually associated with lymphoproliferative disorders, such as monoclonal gammopathy of undetermined significance (MGUS), myeloma, chronic lymphocytic leukemia (CLL) and lymphoplasmocytic lymphoma (LPL). Clinical symptoms related to Type I cryoglobulin (CG) isotype often include skin, neurological and renal manifestations. PATIENT CONCERNS: A 42-year-old woman who initially presented urticaria, palpable purpura in both her upper extremities and legs, eosinophilia and Raynaud phenomenon. Skin biopsy revealed eosinophil infiltration. Monoclonal immunoglobulin (Ig) G-κprotein was detected and CG was also positive. DIAGNOSES: The patient was finally diagnosed as MGUS related Type I CG. INTERVENTIONS: Cyclophosphamide-dexamethasone-thalidomide (CDT) therapy was initiated. OUTCOMES: The treatment relieved the skin symptoms efficiently. LESSONS: To our knowledge, this is a rare case of Type I cryoglobulinemic vasulitis with eosinophilia complicated by MGUS, and the effective treatment of cyclophosphamide combined with thalidomide and prednisone may provide a new therapeutic option for cryoglobulinemic vasulitis.


Assuntos
Crioglobulinemia/complicações , Eosinofilia/complicações , Gamopatia Monoclonal de Significância Indeterminada/complicações , Vasculite/complicações , Adulto , Crioglobulinemia/diagnóstico , Crioglobulinemia/tratamento farmacológico , Diagnóstico Diferencial , Eosinofilia/diagnóstico , Eosinofilia/tratamento farmacológico , Feminino , Humanos , Gamopatia Monoclonal de Significância Indeterminada/diagnóstico , Gamopatia Monoclonal de Significância Indeterminada/tratamento farmacológico , Vasculite/diagnóstico , Vasculite/tratamento farmacológico
4.
Medicine (Baltimore) ; 98(4): e14201, 2019 Jan.
Artigo em Inglês | MEDLINE | ID: mdl-30681592

RESUMO

RATIONALE: Concomitant cryoglobulinemic vasculitis and cold agglutinin disease (CAD) is an extremely uncommon clinical scenario. The role of bortezomib in the treatment of cryoglobulinemic vasculitis needs further investigation. PATIENT CONCERNS: A 72-year-old Chinese woman presented with a 25-year history of cyanosis of the extremities after cold exposure, which worsened and was accompanied with purpuric skin lesions and proteinuria in recent years. Laboratory data demonstrated hemolysis. Cold agglutinin and cryoglobulin tests were positive. There was no evidence for malignancies after blood, image, and pathologic tests. DIAGNOSES: Concomitant cryoglobulinemic vasculitis and CAD. INTERVENTIONS: The patient was treated with bortezomib-based regimen, including bortezomib, cyclophosphamide, and dexamethasone. OUTCOMES: The patient responded well to the treatment. Both symptoms and laboratory tests significantly improved. The patient's condition was in a state of sustained remission in the 6-month follow-up. LESSONS: This rare case promotes further understanding of these 2 diseases and suggests that bortezomib is a promising treatment in type I cryoglobulinemic vasculitis.


Assuntos
Anemia Hemolítica Autoimune/tratamento farmacológico , Bortezomib/uso terapêutico , Crioglobulinemia/tratamento farmacológico , Vasculite/tratamento farmacológico , Idoso , Anemia Hemolítica Autoimune/complicações , Crioglobulinemia/complicações , Ciclofosfamida/uso terapêutico , Dexametasona/uso terapêutico , Feminino , Humanos , Resultado do Tratamento , Vasculite/complicações
5.
Clin Rheumatol ; 38(5): 1257-1262, 2019 May.
Artigo em Inglês | MEDLINE | ID: mdl-30628015

RESUMO

Type I cryoglobulinemia is associated with B cell proliferative diseases, whereas essential mixed cryoglobulinemia is classically associated with infections, malignancy, and autoimmune diseases, but may be idiopathic. Prognosis in patients with grave manifestations and renal involvement is often poor. We report a case of a 40-year-old woman, 2 weeks post-partum for pre-eclampsia who was hospitalized with nephritic syndrome and acute renal failure. The patient harbored type I and type II cryoglobulinemia. Renal and cutaneous biopsies confirmed the diagnosis; however, an underlying etiology was not established. A bone marrow biopsy suggested monoclonal gammopathy of undetermined source (MGUS). Despite therapy with intravenous cyclophosphamide, rituximab, plasmapheresis, dialysis, and bortezomib, the patient succumbed after 8 months of hospitalization. We suggest that an overlap entity of types I and II cryoglobulinemia with severe multi-organ involvement not only is rare but also may be resistant to conventional therapy and fatal.


Assuntos
Crioglobulinemia/complicações , Crioglobulinemia/terapia , Gamopatia Monoclonal de Significância Indeterminada/complicações , Gamopatia Monoclonal de Significância Indeterminada/terapia , Adulto , Ciclofosfamida , Diálise , Evolução Fatal , Feminino , Humanos , Cadeias kappa de Imunoglobulina/análise , Rim/patologia , Plasmaferese , Rituximab , Pele/patologia
6.
Medicine (Baltimore) ; 97(39): e12355, 2018 Sep.
Artigo em Inglês | MEDLINE | ID: mdl-30278513

RESUMO

RATIONALE: Type I cryoglobulinemia is a rare and life-threatening condition. It occurs mainly in B-cell lymphoproliferative disorder. In almost half of the patients, type I cryoglobulinemia is characterized by severe cutaneous involvement. PATIENT CONCERNS: A 71-year-old man comes to our observation because of the onset of persistence of paresthesia and painful acrocyanosis in the fingers and toes. IgG-k multiple myeloma treated with chemotherapy and radiation therapy on the left iliac wing characterized his clinical history. At the evaluation of response after the first-line therapy, the patient achieved a very good partial response. At the time of progression, the same myelomatous disease has taken a typical behavior of cryoglobulinemia. DIAGNOSES: Type I multiple myeloma-associated cryoglobulinemia was diagnosed. INTERVENTIONS: The patient underwent to an immediate composite therapeutic approach based on prostanoid infusion, plasmapheresis along with second-line chemotherapy. OUTCOMES: Despite the rapid biochemical response, the ischemia of the feet worsened. Moreover, a bacterial infection overlapped. The surgical amputation of both feet was necessary. Allowing MM cytoreduction continuation the patient's clinical condition became stabilized. LESSONS: Though rare, type I cryoglobulinemia can be associated with plasma cell dyscrasias. Any delay in diagnosis and the start of therapy can cause worsening of organ damage and endanger the patient's life. Therapeutic strategies in these cases should be directed to the underlying diseases.


Assuntos
Crioglobulinemia/complicações , Mieloma Múltiplo/complicações , Úlcera Cutânea/etiologia , Idoso , Amputação , Crioglobulinemia/diagnóstico , Crioglobulinemia/terapia , Pé/patologia , Doenças do Pé/etiologia , Humanos , Masculino , Mieloma Múltiplo/diagnóstico , Mieloma Múltiplo/terapia
8.
Clin Exp Rheumatol ; 36 Suppl 111(2): 12-32, 2018 Mar-Apr.
Artigo em Inglês | MEDLINE | ID: mdl-29799395
9.
Intern Med ; 57(18): 2739-2745, 2018 Sep 15.
Artigo em Inglês | MEDLINE | ID: mdl-29709950

RESUMO

Anti-neutrophil cytoplasmic antibody (ANCA)-associated vasculitis (AAV) and cryoglobulinemic vasculitis (CV) rarely coexist. An 83-year-old woman was admitted with rapidly progressive renal failure, gastrointestinal hemorrhage and purpura with myeloperoxidase (MPO)-ANCA positivity and cryoglobulinemia. Despite intensive immunosuppressive treatment, she died of aspergillus pneumonia. Autopsy revealed necrotizing crescentic glomerulitis in the majority of the glomeruli, accompanied by partially membranoproliferative-like glomerular changes. Immunofluorescence staining revealed the presence of neutrophil extracellular trap (NET) formation in the glomeruli and cutaneous arteries. These pathological findings suggested that MPO-AAV and/or CV caused NET formation, leading to lethal systemic vasculitis.


Assuntos
Vasculite Associada a Anticorpo Anticitoplasma de Neutrófilos/complicações , Crioglobulinemia/complicações , Crioglobulinemia/patologia , Vasculite Sistêmica/complicações , Vasculite Sistêmica/patologia , Idoso de 80 Anos ou mais , Vasculite Associada a Anticorpo Anticitoplasma de Neutrófilos/imunologia , Autopsia , Armadilhas Extracelulares/imunologia , Evolução Fatal , Feminino , Trato Gastrointestinal/patologia , Glomerulonefrite/imunologia , Humanos , Rim/patologia , Glomérulos Renais/imunologia , Glomérulos Renais/patologia , Peroxidase/imunologia , Pele/imunologia , Pele/patologia
10.
Intern Med ; 57(17): 2511-2515, 2018 Sep 01.
Artigo em Inglês | MEDLINE | ID: mdl-29607961

RESUMO

We report the case of a 75-year-old woman who was found to have hepatitis C virus (HCV) infection in 1987. Before treatment in 2016, she was found to have mixed cryoglobulinemia (MC). Direct-acting antiviral (DAA) treatment produced a sustained virological response 12 (SVR12). She noticed gradual muscle weakness in 2015 and the gradual development of dysarthria and dysphagia in 2017. We performed a muscle biopsy that showed inclusion body myositis (IBM). To the best of our knowledge, this is first case of a patient with HCV infection, MC, and IBM, in which MC and IBM did not improve after an SVR12 was obtained by DAA treatment.


Assuntos
Antivirais/uso terapêutico , Crioglobulinemia/complicações , Hepatite C Crônica/complicações , Hepatite C Crônica/tratamento farmacológico , Miosite de Corpos de Inclusão/complicações , Idoso , Anilidas/uso terapêutico , Carbamatos/uso terapêutico , Crioglobulinemia/diagnóstico , Crioglobulinemia/tratamento farmacológico , Feminino , Hepatite C Crônica/diagnóstico , Humanos , Compostos Macrocíclicos/uso terapêutico , Miosite de Corpos de Inclusão/diagnóstico , Miosite de Corpos de Inclusão/tratamento farmacológico , Ritonavir/uso terapêutico , Resposta Viral Sustentada
11.
Pediatr Rheumatol Online J ; 16(1): 31, 2018 Apr 24.
Artigo em Inglês | MEDLINE | ID: mdl-29690915

RESUMO

BACKGROUND: This study aimed to evaluate the demographic, clinical features, laboratory data, pathology and other survey in pediatric patients with cryofibrinogenemia. METHODS: A 12-year retrospective chart review identified eight pediatric patients at Mackay Memorial Hospital, Taipei, Taiwan. RESULTS: The female-to-male ratio was 3:1. The mean age at symptom onset and of diagnosis was 10.3 ± 4.6 years and 12.3 ± 4 years, respectively. One child (12.5%) had primary cryofibrinogenemia. The common symptoms were purpura, arthralgia, and muscle weakness (100%). On laboratory examination, cryofibrinogen was positive in all patients. All patients had increased anti-thrombin III while 87.5% and 62.5% had abnormal protein S and protein C, respectively. All eight also complained of neurologic symptoms. One had vertebral artery narrowing, two showed increased T2-weighted signal intensity on the thalamus or white matter, and one had acute hemorrhagic encephalomyelitis on brain magnetic resonance imaging. CONCLUSIONS: This study reports on the presentations of cryofibrinogenemia, which is rare in children. Most cases are associated with autoimmune disease and have severe and complex presentations. Central nervous system involvement is common.


Assuntos
Crioglobulinemia/diagnóstico , Adolescente , Criança , Pré-Escolar , Crioglobulinemia/complicações , Feminino , Humanos , Imagem por Ressonância Magnética , Masculino , Estudos Retrospectivos , Taiwan
12.
Clin Exp Rheumatol ; 36(3): 490-493, 2018 May-Jun.
Artigo em Inglês | MEDLINE | ID: mdl-29533748

RESUMO

OBJECTIVES: Guidelines for preventing Pneumocystis pneumonia (PCP) in HIV patients are based on CD4 below 200/mm3. Such cut-off value is suggested to guide prophylaxis in non-HIV conditions (NHIV) especially in autoimmune and inflammatory diseases (AD). We aimed to determine if CD4 could be used to guide PCP prophylaxis in AD. METHODS: CD4 and lymphocyte-count were retrospectively studied in patients diagnosed with PCP between January 2013 and February 2016. RESULTS: 129 patients were included. The median CD4-count was 302/mm3 in AD, which was significantly higher than in HIV patients (19/mm3; p<0.0001). Fifty percent (n=10) of AD patients had CD4 counts greater than 300/mm3. CONCLUSIONS: Prophylaxis for PCP cannot rely solely on CD4-count in NHIV patients especially in AD.


Assuntos
Doenças Autoimunes/tratamento farmacológico , Linfócitos T CD4-Positivos/imunologia , Infecções por HIV/terapia , Imunossupressores/efeitos adversos , Linfopenia/tratamento farmacológico , Pneumonia por Pneumocystis/prevenção & controle , Adulto , Idoso , Idoso de 80 Anos ou mais , Vasculite Associada a Anticorpo Anticitoplasma de Neutrófilos/complicações , Vasculite Associada a Anticorpo Anticitoplasma de Neutrófilos/tratamento farmacológico , Vasculite Associada a Anticorpo Anticitoplasma de Neutrófilos/imunologia , Artrite Reumatoide/complicações , Artrite Reumatoide/tratamento farmacológico , Artrite Reumatoide/imunologia , Doenças Autoimunes/complicações , Doenças Autoimunes/imunologia , Contagem de Linfócito CD4 , Crioglobulinemia/complicações , Crioglobulinemia/tratamento farmacológico , Crioglobulinemia/imunologia , Dermatomiosite/complicações , Dermatomiosite/tratamento farmacológico , Dermatomiosite/imunologia , Gerenciamento Clínico , Feminino , Arterite de Células Gigantes/complicações , Arterite de Células Gigantes/tratamento farmacológico , Arterite de Células Gigantes/imunologia , Infecções por HIV/complicações , Hepatite Autoimune/complicações , Hepatite Autoimune/tratamento farmacológico , Hepatite Autoimune/imunologia , Humanos , Hospedeiro Imunocomprometido , Linfopenia/etiologia , Linfopenia/imunologia , Masculino , Pessoa de Meia-Idade , Neoplasias/complicações , Neoplasias/imunologia , Neoplasias/terapia , Transplante de Órgãos , Pneumonia por Pneumocystis/etiologia , Pneumonia por Pneumocystis/imunologia , Guias de Prática Clínica como Assunto , Estudos Retrospectivos
13.
Nephrol Ther ; 14(2): 118-126, 2018 Apr.
Artigo em Francês | MEDLINE | ID: mdl-29545130

RESUMO

Cryoglobulins are immunoglobulins that undergo reversible precipitation at low temperatures. They can induce systemic vasculitis, characterized by purpuric cutaneous lesions, arthritis, peripheral neuropathy, hypocomplementemia and glomerular disease. Renal pathology reveals membranoproliferative glomerulonephritis, with particularly intense mesangial cell proliferation and infiltration by macrophages, associated with intracapillary thrombi. This renal disease presents as a nephritic syndrome, with heavy proteinuria, haematuria severe hypertension and rapidly progressive kidney failure that can lead to end-stage renal disease. Hepatitis C is the main cause of mixed (type 2 or 3) cryoglobulinemia and requires the initiation of a specific antiviral therapy, together with immunosuppressive drugs. Rituximab is now considered as the best immunosuppressive therapy in this situation, inducing B-cell depletion, clearance of circulating cryoglobulin and resolution of renal symptoms. Monoclonal (type 1) cryoglobulinemia, is a rare condition, but it usually reveals an B-cell or a plasma cell proliferation, that require a specific hematological treatment to obtain remission of the renal disease.


Assuntos
Crioglobulinemia/complicações , Nefropatias/etiologia , Hepatite C/complicações , Humanos , Imunossupressores/uso terapêutico , Rim/patologia , Nefropatias/diagnóstico , Nefropatias/tratamento farmacológico
14.
Wiad Lek ; 71(1 pt 1): 59-63, 2018.
Artigo em Polonês | MEDLINE | ID: mdl-29558353

RESUMO

Cryoglobulinemia is defined as the presence of cryoglobulins in the blood. Cryoglobulinemia is often observed in the course of many diseases (infection, hematological disorders, autoimmune disorders) or has an idiopathic character. The classification of cryoglobulinemia is based on the immunological analysis of cryoglobulins and the activity of the rheumatoid factor (RF). The presence of cryoglobulins may induce cryoglobulinemic vasculitis (CV) which manifests with skin changes, arthritis and the dysfunction of internal organs. Diagnosis of cryoglobulinemia refers to the detection of cryoprecipitate in the blood serum sample. In the treatment of cryoglobulinemia various groups of medications may be used: corticosteroids, cyclophosphamide, azathioprine and rituximab. The purpose of the treatment is to reduce the production of cryoglobulins and the inflammation caused by their presence. Additionally, the treatment of organs complication is also crucial. If the viral infection is the causative agent of vasculitis, combination therapy will be recommended: antiviral and immunosuppressive therapy.


Assuntos
Crioglobulinemia/complicações , Vasculite/etiologia , Doenças Autoimunes/complicações , Crioglobulinemia/diagnóstico , Crioglobulinemia/tratamento farmacológico , Crioglobulinemia/etiologia , Crioglobulinas , Humanos , Infecção/complicações , Vasculite/diagnóstico , Vasculite/tratamento farmacológico
15.
Can J Cardiol ; 34(3): 343.e1-343.e3, 2018 03.
Artigo em Inglês | MEDLINE | ID: mdl-29395708

RESUMO

Cryoglobulinemic vasculitis is a rare and frequently fatal type of myocarditis. Cardiac manifestations in type 1 cryoglobulinemic vasculitis have never been reported to our knowledge. We report a rare case of type 1 cryoglobulinemic vasculitis with cardiac involvement in a patient who experienced progressive heart failure during the diagnosis. The diagnosis was made by the presence of cryoglobulins and endomyocardial biopsy results. After bortezomib-containing treatments, plasma cryoglobulin levels returned to normal, and the patient's clinical condition gradually improved.


Assuntos
Bortezomib/uso terapêutico , Crioglobulinemia/complicações , Insuficiência Cardíaca/fisiopatologia , Vasculite/tratamento farmacológico , Vasculite/etiologia , Adulto , Biópsia por Agulha , Crioglobulinemia/diagnóstico , Crioglobulinemia/tratamento farmacológico , Progressão da Doença , Seguimentos , Insuficiência Cardíaca/etiologia , Humanos , Imuno-Histoquímica , Masculino , Doenças Raras , Medição de Risco , Índice de Gravidade de Doença , Resultado do Tratamento , Vasculite/patologia
16.
Dermatol Online J ; 24(1)2018 Jan 15.
Artigo em Inglês | MEDLINE | ID: mdl-29469770

RESUMO

A 55-year-old man presented with a history of asymptomatic, bilateral, figurate dermatosis consisting of erythematous, annular, linear, andarciform, indurated nodules and plaques on the lateral walls of thorax, flanks, and hypogastrium. His medical history was positive for chronic hepatitis C. Skin biopsy was compatible with the diagnosisof interstitial granulomatous dermatitis. Additionalinvestigation revealed positive rheumatoid factor, antinuclear antibodies, and cryoglobulins. Thepatient started treatment with high-potency topical corticosteroid. After 12 weeks of treatment there was a partial clinical improvement of thedermatosis. Interstitial granulomatous dermatitis exhibits a distinct histopathological pattern with a diverse clinical appearance, which can be associated with autoimmune systemic diseases, neoplasms, several drugs, and infections. Optimal therapy for this condition is yet to be established, but topical corticosteroids have been a mainstay of treatment.


Assuntos
Crioglobulinemia/complicações , Dermatite/etiologia , Granuloma/etiologia , Hepatite C Crônica/complicações , Anticorpos Antinucleares/sangue , Biópsia , Dermatite/patologia , Granuloma/patologia , Humanos , Masculino , Pessoa de Meia-Idade , Fator Reumatoide/sangue , Pele/patologia
17.
BMC Gastroenterol ; 18(1): 19, 2018 Jan 25.
Artigo em Inglês | MEDLINE | ID: mdl-29370761

RESUMO

BACKGROUND: Peripheral neuropathy is one most common, limiting and invalidating neurological symptom in subjects with hepatitis C virus and mixed cryoglobulinemia. Notably, the medical therapy proposed to eradicate HCV, can frequently exacerbate the painful neuropathy. Therefore, neuropathy therapies are insufficient and inadequate, and comprise immunosuppressive drugs, such as steroid or cyclosporine, intravenous immunoglobulin or plasma exchange. These have shown variable success in case reports, with a presumably temporary effect, but with major side effects. METHODS: We assessed the effects of oxcarbazepine treatment in 67 cases of cryoglobulinemia related neuropathy, who did not respond to either steroid or Gabapentin, or Pregabalin. Oxcarbazepine was chosen based on the promising preliminary results. RESULTS: Patients treated with Oxcarbazepine showed a rapid, discrete and persistent relief of polyneuropathic signs, without consistent side effects, and with a limited interaction with concomitant drugs. CONCLUSIONS: These data favor the use of oxcarbazepine as a useful tool in the management of neuropathic pain associated with Hepatitis-C cryoglobulin neuropathy.


Assuntos
Analgésicos/uso terapêutico , Carbamazepina/análogos & derivados , Crioglobulinemia/complicações , Hepatite C/complicações , Neuralgia/tratamento farmacológico , Analgésicos/efeitos adversos , Carbamazepina/efeitos adversos , Carbamazepina/uso terapêutico , Feminino , Humanos , Masculino , Pessoa de Meia-Idade , Neuralgia/etiologia , Oxcarbazepina
18.
BMJ Case Rep ; 20182018 Jan 04.
Artigo em Inglês | MEDLINE | ID: mdl-29301807

RESUMO

We describe a patient with untreated hepatitis C virus (HCV) infection presenting with pulmonary renal syndrome. He rapidly developed bilateral lung infiltrates and respiratory failure, and bronchoscopy confirmed acute alveolar haemorrhage secondary to cryoglobulinaemic vasculitis. Early bronchoscopy to confirm the diagnosis and consequent institution of immunosuppressive therapy led to excellent outcomes, which otherwise is reported in the literature to carry significant mortality. Therefore, in patients with HCV presenting with bilateral lung infiltrates, physicians must maintain a high degree of clinical suspicion for alveolar haemorrhage secondary to cryoglobulinaemic vasculitis.


Assuntos
Crioglobulinemia/complicações , Hemorragia/virologia , Hepatite C/complicações , Pneumopatias/virologia , Vasculite/complicações , Crioglobulinemia/virologia , Hepacivirus , Hepatite C/virologia , Humanos , Masculino , Pessoa de Meia-Idade , Alvéolos Pulmonares/virologia , Vasculite/virologia
19.
Arch Virol ; 163(4): 961-967, 2018 Apr.
Artigo em Inglês | MEDLINE | ID: mdl-29318374

RESUMO

Mixed cryoglobulinemia (MC) is the most common extrahepatic manifestation of chronic hepatitis C (CHC), with the presence of symptoms in 10-15% of cases. There have been encouraging data regarding immunological and clinical responses in patients treated with the novel combinations of direct-acting antivirals (DAAs), but the role of ribavirin (RBV) in the treatment of MC has not yet been demonstrated. We prospectively enrolled 132 patients affected by MC and CHC, and virological, immunological and clinical responses were evaluated at 12 weeks after completion of treatment. All subjects were treated with interferon (IFN)-free regimens according to clinical guidelines, with or without RBV. All patients achieved a virological response. A complete immunological response (CR) was observed in 71 subjects (53.8%), a partial response in 44 (33.3%), and no response in 17 (12.8%). Ten patients showed a complete resolution of symptoms (7.6%), and 31 showed a significant improvement (23.5%). CR was significantly higher in patients taking RBV (71.1 vs. 44.8%, p < 0.001) and in treatment-naïve patients (62.5 vs. 43.3%, p < 0.001). In logistic regression analysis, duration of HCV infection of less than 20 years (OR 2.448; 95% IC 1.335-6.202; p = 0.019), treatment-naïve status (OR 2.885; 95% IC 1.404-9.660; p = 0.025) and the use of RBV (OR 6.961; 95% IC 3.912-26.885; p < 0.001) were predictors of CR. In MC patients, IFN-free regimens are effective and well tolerated, and RBV seems to significantly increase the immunological response and promote a decline in cryocrit.


Assuntos
Antivirais/uso terapêutico , Crioglobulinemia/tratamento farmacológico , Hepacivirus/efeitos dos fármacos , Hepatite C Crônica/tratamento farmacológico , Ribavirina/uso terapêutico , Adulto , Anilidas/uso terapêutico , Carbamatos/uso terapêutico , Crioglobulinemia/complicações , Crioglobulinemia/imunologia , Crioglobulinemia/virologia , Quimioterapia Combinada , Feminino , Genótipo , Hepacivirus/genética , Hepacivirus/crescimento & desenvolvimento , Hepacivirus/imunologia , Hepatite C Crônica/complicações , Hepatite C Crônica/imunologia , Hepatite C Crônica/virologia , Humanos , Imidazóis/uso terapêutico , Interferons , Compostos Macrocíclicos/uso terapêutico , Masculino , Pessoa de Meia-Idade , Estudos Prospectivos , Ritonavir/uso terapêutico , Sofosbuvir/uso terapêutico , Sulfonamidas/uso terapêutico , Resultado do Tratamento , Uracila/análogos & derivados , Uracila/uso terapêutico
20.
Retin Cases Brief Rep ; 12(3): 159-165, 2018 Summer.
Artigo em Inglês | MEDLINE | ID: mdl-27902540

RESUMO

PURPOSE: Macular perivenous retinal whitening results from hypoperfusion-induced ischemia of the middle retina that can occur in central retinal vein occlusion (CRVO). We describe an unusual case of recurrent CRVO with macular perivenous retinal whitening and retino-ciliary venous sparing in the setting of 2 prothrombotic diseases, antiphospholipid syndrome and Type II cryoglobulinemia. METHODS: A 50-year-old man presented with intermittent loss of vision in his right eye related to a recurrent CRVO. Color photography, optical coherence tomography, and fluorescein angiography were performed and compared with those obtained during a previous CRVO that occurred 6 years earlier in the same eye. RESULTS: On presentation, visual acuity was hand motion in the right eye, 20/30 in the left eye. Funduscopic examination of the right eye showed vascular tortuosity, scattered retinal hemorrhages, and retinal whitening in the macula. Optical coherence tomography showed hyperreflectivity of the middle layers of the retina that correlated with the areas of retinal whitening. A discrete area of retinal sparing was noted in the superonasal macula that, on fluorescein angiography, corresponded to the distribution of a single retino-ciliary vein. A review of retinal imaging obtained during the patient's previous CRVO showed similar but more subtle findings of retino-ciliary sparing. Laboratory testing revealed antiphospholipid syndrome and Type II cryoglobulinemia. As the patient's CRVO progressed and subsequently stabilized after treatment in the following months, this area of venous sparing remained the only functional, nonischemic retinal tissue in his macula. Presumably, this vein possessed privileged and uncompromised blood flow by circumventing the occluded venous circulation. CONCLUSION: Macular perivenous retinal whitening should be considered in the differential diagnosis of retinal whitening and occurs in CRVO secondary to hypoperfusion-induced middle retinal ischemia. To our knowledge, this case represents the first description of retino-ciliary venous sparing of the retina in CRVO.


Assuntos
Síndrome Antifosfolipídica/complicações , Crioglobulinemia/complicações , Macula Lutea/efeitos da radiação , Doenças Retinianas/etiologia , Oclusão da Veia Retiniana/complicações , Vasos Retinianos/patologia , Humanos , Masculino , Pessoa de Meia-Idade , Oclusão da Veia Retiniana/etiologia , Transtornos da Visão/etiologia
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