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2.
J Cutan Pathol ; 48(4): 567-571, 2021 Apr.
Artigo em Inglês | MEDLINE | ID: mdl-32885480

RESUMO

Patients with primary immunodeficiency are at increased risk for malignancy, especially hematologic neoplasms. This paper reports a unique case of a 47-year-old man with X-linked agammaglobulinemia who presented with progressive asymptomatic violaceous papules and plaques on his face, hands, and trunk for 1 year. Skin biopsies revealed deep, nodular infiltrates of histiocytes and CD8-positive lymphocytes, with a CD4:CD8 ratio of 1:10. Laboratory studies showed cytopenias. Flow cytometry in the skin, blood, and bone marrow (BM) showed a CD3+/CD8+/CD57+ large granular lymphocyte population. BM biopsy showed 30% involvement with these atypical T-cells. T-cell gene rearrangement studies of skin, blood, and BM revealed identical T-cell clones. He was diagnosed with T-large granular lymphocyte leukemia (T-LGLL) with an associated CD8+ cutaneous lymphoproliferation. Skin involvement was suspected to represent infiltration by T-LGLL. However, co-existence of two lymphoproliferative disorders (LPDs), T-LGLL and CD8+ granulomatous LPD, remains a possibility. In general, cutaneous infiltrates associated with LGLL are rare and poorly understood. It has been suggested that they are markers of poor prognosis. Our case report describes skin, blood, and BM findings in an immunosuppressed patient with T-LGLL in detail. These findings have not yet been reported and their significance requires further investigation.


Assuntos
Agamaglobulinemia/genética , Linfócitos T CD8-Positivos/patologia , Doenças Genéticas Ligadas ao Cromossomo X/genética , Leucemia Linfocítica Granular Grande/genética , Leucemia Linfocítica Granular Grande/patologia , Adjuvantes Imunológicos/administração & dosagem , Adjuvantes Imunológicos/uso terapêutico , Agamaglobulinemia/complicações , Agamaglobulinemia/diagnóstico , Agamaglobulinemia/patologia , Biópsia , Medula Óssea/patologia , Linfócitos T CD8-Positivos/imunologia , Fármacos Dermatológicos/administração & dosagem , Fármacos Dermatológicos/uso terapêutico , Diagnóstico Diferencial , Quimioterapia Combinada , Rearranjo Gênico do Linfócito T , Doenças Genéticas Ligadas ao Cromossomo X/complicações , Doenças Genéticas Ligadas ao Cromossomo X/diagnóstico , Doenças Genéticas Ligadas ao Cromossomo X/patologia , Histiócitos/patologia , Humanos , Imiquimode/administração & dosagem , Imiquimode/uso terapêutico , Terapia de Imunossupressão/efeitos adversos , Leucemia Linfocítica Granular Grande/complicações , Leucemia Linfocítica Granular Grande/tratamento farmacológico , Masculino , Metotrexato/administração & dosagem , Metotrexato/uso terapêutico , Pessoa de Meia-Idade , Pele/patologia , Resultado do Tratamento
3.
PLoS One ; 8(10): e78450, 2013.
Artigo em Inglês | MEDLINE | ID: mdl-24205234

RESUMO

BACKGROUND: In June of 2010, an antenatal ultrasound program to perform basic screening for high-risk pregnancies was introduced at a community health care center in rural Uganda. Whether the addition of ultrasound scanning to antenatal visits at the health center would encourage or discourage potential patients was unknown. Our study sought to evaluate trends in the numbers of antenatal visits and deliveries at the clinic, pre- and post-introduction of antenatal ultrasound to determine what effect the presence of ultrasound at the clinic had on these metrics. METHODS AND FINDINGS: Records at Nawanyago clinic were reviewed to obtain the number of antenatal visits and deliveries for the 42 months preceding the introduction of ultrasound and the 23 months following. The monthly mean deliveries and antenatal visits by category (first visit through fourth return visit) were compared pre- and post- ultrasound using a Kruskal-Wallis one-way ANOVA. Following the introduction of ultrasound, significant increases were seen in the number of mean monthly deliveries and antenatal visits. The mean number of monthly deliveries at the clinic increased by 17.0 (13.3-20.6, 95% CI) from a pre-ultrasound average of 28.4 to a post-ultrasound monthly average of 45.4. The number of deliveries at a comparison clinic remained flat over this same time period. The monthly mean number of antenatal visits increased by 97.4 (83.3-111.5, 95% CI) from a baseline monthly average of 133.5 to a post-ultrasound monthly mean of 231.0, with increases seen in all categories of antenatal visits. CONCLUSIONS: The availability of a low-cost antenatal ultrasound program may assist progress towards Millennium Development Goal 5 by encouraging women in a rural environment to come to a health care facility for skilled antenatal care and delivery assistance instead of utilizing more traditional methods.


Assuntos
Assistência Ambulatorial , Atenção à Saúde/métodos , Parto Obstétrico/métodos , Cuidado Pré-Natal/métodos , Instituições de Assistência Ambulatorial , Feminino , Instalações de Saúde , Humanos , Aceitação pelo Paciente de Cuidados de Saúde , Gravidez , População Rural , Uganda , Ultrassonografia/métodos
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