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2.
An Bras Dermatol ; 96(2): 240-242, 2021.
Artigo em Inglês | MEDLINE | ID: mdl-33579583

RESUMO

Small vessel vasculitis with anti-proteinase antibodies 3 is an atypical clinical presentation of tuberculosis. The authors present the case of a 47-year-old male patient, with palpable purpura and palmoplantar hemorrhagic blisters, with subsequent dissemination. He presented severe pulmonary symptoms with cavitation, fever, hemoptysis, and high levels of anti-proteinase 3. Histopathological assessment of the skin revealed small vessel vasculitis; pulmonary histopathology showed granulomas with caseation. Bronchoalveolar lavage was positive for alcohol-acid-fast bacilli. In countries with a high prevalence of tuberculosis, the presence of autoantibodies in a patient with vasculitis, fever, and pulmonary cavitation requires investigation of infectious causes.


Assuntos
Dermatopatias Vasculares , Vasculite , Adulto , Anticorpos Anticitoplasma de Neutrófilos , Hemoptise/diagnóstico , Hemoptise/etiologia , Humanos , Pulmão/diagnóstico por imagem , Masculino , Pessoa de Meia-Idade , Mieloblastina , Vasculite/diagnóstico
3.
Medicine (Baltimore) ; 99(39): e22341, 2020 Sep 25.
Artigo em Inglês | MEDLINE | ID: mdl-32991448

RESUMO

RATIONALE: The Goodpasture syndrome is an extremely rare disease, with renal and pulmonary manifestations, and is mediated by anti-glomerular basement membrane (anti-GBM) antibodies. Renal pathological changes are mainly characterized by glomerular crescent formation and linear immunofluorescent staining for immunoglobulin G on the GBM. There are few reports on the atypical course of the syndrome involving serum-negative anti-GBM antibodies. Therefore, we present a case of Goodpasture syndrome that presented with nephrotic-range proteinuria and was seronegative for anti-GBM antibodies. PATIENT CONCERNS: A 38-year-old Chinese man presented with a lung lesion that was discovered by physical examination a month prior to presentation. The chief concern was occasional hemoptysis without fever, cough, chest pain, and edema. DIAGNOSES: Laboratory testing revealed that the urinary protein level and urine erythrocyte count were 7.4 g/24 hours and 144/high-power field (HPF), respectively. Serological testing for anti-GBM antibodies was negative. Chest computed tomography revealed multiple exudative lesions in both lungs, indicating alveolar infiltration and hemorrhage. Electronic bronchoscopy and pathological examination of the alveolar lavage fluid indicated no abnormalities. However, kidney biopsy suggested cellular crescent formation and segmental necrosis of the globuli, with linear IgG and complement C3 deposition on the GBM. These findings were consistent with the diagnosis of anti-GBM antibody nephritis. INTERVENTIONS: The patient underwent 7 sessions of double filtration plasmapheresis. He was also administered with intravenous methylprednisolone and cyclophosphamide. After renal function stabilization, he was discharged under an immunosuppressive regimen comprising of glucocorticoids and cyclophosphamides. OUTCOMES: Three months later, follow-up examination revealed that the 24-hour urine protein had increased to 13 g. Furthermore, the urine erythrocyte count was 243/HPF. After a 6-month follow-up, the patient achieved partial remission, with a proteinuria level of 3.9 g/24 hours and a urine erythrocyte count of 187/HPF. LESSONS: This extremely rare case of Goodpasture syndrome manifested with seronegativity for anti-GBM antibodies and nephrotic-range proteinuria. Our findings emphasize the importance of renal biopsy for the clinical diagnosis of atypical cases. Furthermore, because renal involvement achieved only partial remission despite therapy, early detection and active treatment of the Goodpasture syndrome is necessary to improve the prognosis of patients.


Assuntos
Doença Antimembrana Basal Glomerular/complicações , Doença Antimembrana Basal Glomerular/imunologia , Autoanticorpos/sangue , Proteinúria/etiologia , Administração Intravenosa , Adulto , Assistência ao Convalescente , Doença Antimembrana Basal Glomerular/diagnóstico , Doença Antimembrana Basal Glomerular/terapia , Grupo com Ancestrais do Continente Asiático/etnologia , Complemento C3/metabolismo , Ciclofosfamida/administração & dosagem , Ciclofosfamida/uso terapêutico , Quimioterapia Combinada , Glucocorticoides/administração & dosagem , Glucocorticoides/uso terapêutico , Hemoptise/diagnóstico , Hemorragia/etiologia , Hemorragia/patologia , Humanos , Imunoglobulina G/metabolismo , Imunossupressores/administração & dosagem , Imunossupressores/uso terapêutico , Rim/patologia , Glomérulos Renais/metabolismo , Glomérulos Renais/patologia , Pulmão/diagnóstico por imagem , Pulmão/patologia , Masculino , Metilprednisolona/administração & dosagem , Metilprednisolona/uso terapêutico , Nefrite/diagnóstico , Nefrite/imunologia , Plasmaferese/métodos , Tomografia Computadorizada por Raios X/métodos , Resultado do Tratamento
5.
Medicine (Baltimore) ; 99(21): e20281, 2020 May 22.
Artigo em Inglês | MEDLINE | ID: mdl-32481307

RESUMO

RATIONALE: Acute idiopathic pulmonary hemorrhage (AIPH) in infants is a rare condition, and a clear treatment protocol has not yet been established. PATIENT CONCERNS: We report 2 infant cases of AIPH in a 3-month-old male and a 1-month-old female, who presented at an emergency room with epistaxis and respiratory distress. Both were immediately intubated, which revealed a bloody intratracheal aspirate. DIAGNOSIS: Pulmonary hemorrhage was confirmed by X-ray and computed tomography imaging in both cases. The extensive evaluation revealed no specific etiology for the acute pulmonary hemorrhage, and AIPH was therefore diagnosed in both cases. INTERVENTIONS: Intravenous methylprednisolone resulted in a rapid improvement in oxygenation and a reduction in high airway pressure during mechanical ventilation. Methylprednisolone was subsequently tapered off within 13 and 3 days in cases 1 and 2, respectively. In case 1, intratracheal administration of a surfactant also resulted in an immediate improvement in respiratory condition and the patient was extubated after 2 days; no effect was seen in case 2, and the patient was extubated after 10 days. OUTCOME: Both infants recovered well without sequelae or further relapse after 23 and 71 months of follow-up, respectively. LESSONS: Early administration of corticosteroid therapy and intratracheal administration of diluted surfactant should be considered for severe acute pulmonary hemorrhage in infants.


Assuntos
Hemoptise/tratamento farmacológico , Metilprednisolona/administração & dosagem , Surfactantes Pulmonares/uso terapêutico , Doença Aguda , Quimioterapia Combinada , Feminino , Seguimentos , Glucocorticoides/administração & dosagem , Hemoptise/diagnóstico , Humanos , Lactente , Recém-Nascido , Injeções Intravenosas , Masculino , Fatores de Tempo , Tomografia Computadorizada por Raios X
6.
Rev Mal Respir ; 37(3): 275-279, 2020 Mar.
Artigo em Francês | MEDLINE | ID: mdl-32171443

RESUMO

INTRODUCTION: Haemangiomas are vascular malformations, frequently cutaneous, hepatic and splenic. Respiratory involvement and multiple localisations are uncommon. CASE REPORT: We present a rare case of multiple cavernous haemangiomas in a 35 year old woman presenting with repeated haemoptysis. Thoracic CT scanning showed a mass in the left lower lobe associated with lymph node enlargement above and below the diaphragm, heterogeneous splenomegaly and a single spinal lesion without hypermetabolism on PET scanning. Enbronchial ultrasound-guided trans-bronchial needle aspiration was not contributory. Histopathological diagnostic was made firstly by splenectomy with lumbar-aortic curettage and then by lobectomy for haemostasis. A final diagnosis of multiple cavernous haemangiomas involving lung, lymph nodes, spleen and bone was made. CONCLUSIONS: Bronchopulmonary cavernous haemangiomas associated with extra-thoracic lesions are exceptionally rare and their presentation, suggesting, a malignant cause, often leads to surgical resection for diagnostic and, eventually, therapeutic management. We report an original case of cavernous haemangiomas involving lung, lymph nodes, spleen and bone.


Assuntos
Neoplasias Ósseas/diagnóstico , Hemangioma Cavernoso/diagnóstico , Neoplasias Pulmonares/diagnóstico , Neoplasias Primárias Múltiplas/diagnóstico , Neoplasias Esplênicas/diagnóstico , Adulto , Neoplasias Ósseas/patologia , Neoplasias Ósseas/cirurgia , Feminino , Hemangioma Cavernoso/patologia , Hemangioma Cavernoso/cirurgia , Hemoptise/diagnóstico , Hemoptise/etiologia , Hemoptise/cirurgia , Humanos , Neoplasias Pulmonares/patologia , Neoplasias Pulmonares/cirurgia , Imagem por Ressonância Magnética , Neoplasias Primárias Múltiplas/patologia , Neoplasias Primárias Múltiplas/cirurgia , Esplenectomia/efeitos adversos , Neoplasias Esplênicas/patologia , Neoplasias Esplênicas/cirurgia , Esplenomegalia/etiologia , Esplenomegalia/cirurgia , Tomografia Computadorizada por Raios X
9.
Chest ; 157(1): 77-88, 2020 01.
Artigo em Inglês | MEDLINE | ID: mdl-31374211

RESUMO

Massive hemoptysis is a medical emergency with high mortality presenting several difficult diagnostic and therapeutic challenges. The origin of bleeding and underlying etiology often is not immediately apparent, and techniques for management of this dangerous condition necessitate an expedient response. Unlike hemorrhage in other circumstances, a small amount of blood can rapidly flood the airways, thereby impairing oxygenation and ventilation, leading to asphyxia and consequent cardiovascular collapse. Of paramount importance is early control of the patient's airway and immediate isolation of hemorrhage in an attempt to localize and control bleeding. A coordinated team response is essential to guarantee the best chances of patient survival. Prompt control of the airway and steps to limit the spread of hemorrhage take precedence. Bronchial artery embolization, rigid and flexible bronchoscopy, and surgery all serve as potential treatment options to provide definitive control of hemorrhage. Several adjunctive therapies described in recent years may also assist in the control of bleeding; however, their role is less defined in life-threatening hemoptysis and warrants additional studies. In this concise review, we emphasize the steps necessary for a systematic approach in the management of life-threatening hemoptysis.


Assuntos
Hemoptise/diagnóstico , Hemoptise/terapia , Broncoscopia , Lista de Checagem , Diagnóstico Diferencial , Diagnóstico por Imagem , Eletrocoagulação , Embolização Terapêutica , Fibrinolíticos , Hemoptise/etiologia , Hemoptise/fisiopatologia , Humanos , Doença Iatrogênica , Intubação Intratraqueal , Prognóstico
12.
BMC Pulm Med ; 19(1): 185, 2019 Oct 24.
Artigo em Inglês | MEDLINE | ID: mdl-31651292

RESUMO

BACKGROUND: Diffuse alveolar haemorrhage (DAH) is characterized by the diffuse accumulation of red blood cells within the alveoli, presence of ground glass opacities and/or consolidation on computed tomography (CT). Aside from identifiable non-immune causes, DAH is classically subdivided into idiopathic (idiopathic pulmonary haemosiderosis, IPH) and autoimmune DAH. Here we describe three cases presenting with recurrent pulmonary haemorrhage, initially classified as IPH, who, several years after first presentation, develop anti myeloperoxidase antibodies (MPO) positivity, emphysema on CT and, in one case, renal involvement. CASE PRESENTATION: Patient 1 was diagnosed with IPH aged 14. Her disease remained poorly controlled despite immunosuppression, although ANCA remained negative over the years. Nineteen years from initial presentation, she developed MPO-ANCA positive antibodies and mild renal impairment. She was treated with Rituximab with good response. From first presentation, the chest CT was consistently characterized by diffuse ground-glass opacities and interlobular septal thickening. Ten years later, cystic opacities consistent with emphysema, with a striking peribronchovascular distribution, developed. Patient 2 was diagnosed with IPH aged 32. He was treated with corticosteroids and methotrexate, with fluctuating response. At 11 years from initial presentation, MPO-ANCA positivity was identified, and emphysema with a peribronchovascular distribution was observed on CT, with subsequent significant increase in extent. Patient 3 was diagnosed with IPH at the age of seven, and had recurrent episodes of haemoptysis of varying degree of severity, treated with intermittent courses of corticosteroids until age 11, when he was intubated due to severe DAH. Eight years after the diagnosis emphysematous changes were noted on CT and MPO-ANCA positivity developed for the first time 11 years after initial diagnosis. CONCLUSIONS: We believe these three cases highlight: 1) the possibility of development of ANCA positivity several years down the line from first DAH presentation 2) the possibility that DAH may lead to cystic/emphysematous changes with peribronchovascular distribution on CT. Moreover, the need for ongoing immunosuppressive treatment and the development of emphysema, emphasize a possible role played by autoimmune phenomena, even when DAH is initially diagnosed as "idiopathic". Further studies are required to better understand the relationship between DAH, ANCA positivity and development of emphysema.


Assuntos
Anticorpos Anticitoplasma de Neutrófilos/sangue , Glucocorticoides/administração & dosagem , Hemoptise , Metotrexato/administração & dosagem , Peroxidase/imunologia , Enfisema Pulmonar , Rituximab/administração & dosagem , Adolescente , Adulto , Criança , Diagnóstico Diferencial , Feminino , Hemoptise/diagnóstico , Hemoptise/etiologia , Hemoptise/imunologia , Hemossiderose/diagnóstico , Humanos , Imunossupressores/administração & dosagem , Pulmão/diagnóstico por imagem , Pneumopatias/diagnóstico , Masculino , Administração dos Cuidados ao Paciente , Enfisema Pulmonar/complicações , Enfisema Pulmonar/diagnóstico , Enfisema Pulmonar/imunologia , Enfisema Pulmonar/fisiopatologia , Insuficiência Renal/diagnóstico , Insuficiência Renal/imunologia , Tomografia Computadorizada por Raios X/métodos
13.
Clin Lab ; 65(9)2019 Sep 01.
Artigo em Inglês | MEDLINE | ID: mdl-31532086

RESUMO

BACKGROUND: G-lipopolysaccharide, a component of the cell wall of Gram-negative bacteria, is called lipopolysaccharide. The detection of G-lipopolysaccharide can be used for the early diagnosis of infectious diseases, but some-times G-lipopolysaccharide provides limited help. We report a case of a patient with hemoptysis and high-density shadow of both lungs combined with elevated serum G-lipopolysaccharide which mimicked bronchiectasis with Gram-negative bacterium infection. It was ultimately confirmed as Mycobacterium iranicum infection by CT-guided percutaneous lung biopsy and next generation sequencing. METHODS: The chest computed tomography (CT) scan, CT-guided percutaneous lung biopsy, and NGS were performed for diagnosis and blood tests explored for the latent etiology. RESULTS: The chest CT scan showed a high-density shadow of both lungs, atelectasis of right middle lobe, multiple enlarged lymph nodes in mediastinum and right hilum. Pathology of CT-guided percutaneous lung biopsy indicated fibrous tissue proliferation and granulation tissue formation and some alveolar epithelial cells slightly proliferated with focal carbon powder deposition in alveolar sacs and spaces. The lung tissue NGS confirmed Mycobacterium iranicum infection. CONCLUSIONS: Elevated serum G-lipopolysaccharide is not a specific index for infectious diseases. CT-guided percutaneous lung biopsy and lung tissue NGS has high specificity in pathogen detection of infectious diseases.


Assuntos
Bronquiectasia/patologia , Infecções por Bactérias Gram-Negativas/diagnóstico , Hemoptise/diagnóstico , Lipopolissacarídeos/sangue , Pulmão/patologia , Infecções por Mycobacterium/diagnóstico , Idoso , Biópsia/métodos , Diagnóstico Diferencial , Erros de Diagnóstico , Sequenciamento de Nucleotídeos em Larga Escala/métodos , Humanos , Pulmão/microbiologia , Masculino , Tomografia Computadorizada por Raios X
14.
J Pediatr ; 214: 66-70, 2019 11.
Artigo em Inglês | MEDLINE | ID: mdl-31540762

RESUMO

OBJECTIVES: To describe the etiologies of hemoptysis in patients without pre-existing bronchiectasis or cardiac disease; to assess odds of recurrent hemoptysis by diagnostic category; and to assess odds of mortality by diagnostic category. STUDY DESIGN: This retrospective case series included all patients with hemoptysis documented during an admission to Boston Children's Hospital from January 1, 2007 to June 1, 2017. Patients with bronchiectasis, congenital heart disease, primary pulmonary hypertension, bleeding above the glottis, hemoptysis before 38 weeks of corrected gestational age, hematemesis, foreign body, and trauma were excluded. Patients were also characterized by coagulation status. Primary outcomes were recurrent hemoptysis and death. Univariate analysis was performed to determine ORs for recurrence and death per diagnostic category with infection as the reference category. RESULTS: In total, 257 patients met study criteria and were analyzed. The most common causes of hemoptysis were infection (n = 122), neoplasm (n = 58), and other diagnoses (n = 49). Of the patients with infection, recurrence was 28% and all-cause mortality was 12%. Neoplasm had lower odds of recurrence (OR 0.3, P = .012) but higher odds of mortality (OR 15.8, P < .001). Thrombocytopenia had lower odds of recurrence (OR 0.2, P = .005) but higher odds of mortality (OR 5.9, P < .001). Patients with a tracheostomy had higher odds of recurrence (OR 6.3, P < .001), but lower odds of death (OR 0.4, P = .042). CONCLUSIONS: This study confirms that infection is the most common cause of hemoptysis in patients without severe underlying pulmonary or cardiac disease. Hemoptysis associated with neoplasm and/or thrombocytopenia confers mortality risk. Tracheostomy confers risk of recurrence. Future prospective research on diagnoses associated with hemoptysis is warranted.


Assuntos
Hemoptise/etiologia , Hemoptise/mortalidade , Adolescente , Adulto , Boston/epidemiologia , Bronquiectasia/complicações , Criança , Pré-Escolar , Feminino , Seguimentos , Cardiopatias/complicações , Hemoptise/diagnóstico , Humanos , Lactente , Recém-Nascido , Modelos Logísticos , Masculino , Razão de Chances , Recidiva , Estudos Retrospectivos , Medição de Risco , Fatores de Risco , Adulto Jovem
16.
J Card Surg ; 34(10): 1110-1113, 2019 Oct.
Artigo em Inglês | MEDLINE | ID: mdl-31269305

RESUMO

Mechanical circulatory support is increasingly used and may bring about unique challenges. Most support systems require systemic anticoagulation and the need for anticoagulation must be balanced against the increased risk for bleeding. We report the case of a young man awaiting heart retransplantation, who was supported with a temporary extracorporeal ventricular assist device with the addition of an oxygenator. He developed hemoptysis that forced the cessation of anticoagulation exposing to increased thromboembolic risk. We discuss this distinct clinical scenario with no clearly defined solution and explore the risks and benefits of the different treatment options.


Assuntos
Remoção de Dispositivo/métodos , Oxigenação por Membrana Extracorpórea/efeitos adversos , Coração Auxiliar/efeitos adversos , Hemoptise/etiologia , Hemorragia Pós-Operatória/etiologia , Alvéolos Pulmonares/patologia , Adulto , Biópsia , Hemoptise/diagnóstico , Hemoptise/cirurgia , Humanos , Masculino , Hemorragia Pós-Operatória/diagnóstico , Hemorragia Pós-Operatória/cirurgia , Alvéolos Pulmonares/irrigação sanguínea , Tomografia Computadorizada por Raios X
18.
BMC Pulm Med ; 19(1): 104, 2019 Jun 06.
Artigo em Inglês | MEDLINE | ID: mdl-31170962

RESUMO

BACKGROUND: Bronchial Dieulafoy's disease (BDD) is a rare disease that is known to be a cause of hemorrhage. The characteristics of this disease are still unknown. The present study describes the disorder based on a review of the world's literature, emphasizing the diagnostic and therapeutic views. METHODS: A comprehensive research of BDD of the PubMed, Google Scholar, and Web of Science databases was performed. The following data were collected: patient characteristics; chest imaging, bronchoscopy, vascular angiography, and histopathologic examination findings; and treatment rendered. RESULTS: 73 cases of BDD have been reported from 1995 to 2019. Most of the cases occurred in Asia (52.1%), followed by Europe (31.5%). Chest imaging findings were non-specific. The main bronchoscopy finding was a nodular or protruding lesion (60.9%). 19 patients underwent bronchoscopic biopsies, 17 had bleeding, and 6 died. Four patients were successfully shown to have vascular malformations under mucosal protrusion by endobronchial ultrasound scan (EBUS). Vascular angiography mainly showed tortuous, dilated bronchial arteries. Vascular angiography mainly showed tortuous, dilated bronchial arteries. The arterial supply was mainly provided by bronchial arteries (48 cases) and the pulmonary circulation (4 cases). The lesions were mainly located in the right bronchus (53 cases). Selective bronchial artery embolization (BAE) was attempted in 38 patients and 20 patients underwent lobectomies. Emergency resection was performed in 15 patients, all of whom survived and had no recurrent hemoptysis. CONCLUSIONS: Massive hemoptysis was the common manifestation of BDD. Vascular angiography and EBUS is a very useful examination before biopsy. BAE may be used in stable patients, or patients who cannot tolerate surgery, while surgical resection should be considered in patients who are unstable, patients with uncontrolled hemoptysis, or following BAE failure.


Assuntos
Brônquios/patologia , Broncopatias/diagnóstico , Hemoptise/diagnóstico , Adulto , Idoso , Angiografia , Broncopatias/cirurgia , Broncoscopia , Embolização Terapêutica , Feminino , Hemoptise/etiologia , Hemoptise/cirurgia , Humanos , Masculino , Pessoa de Meia-Idade , Estudos Retrospectivos
19.
Ann Thorac Surg ; 108(6): e357-e359, 2019 12.
Artigo em Inglês | MEDLINE | ID: mdl-31158352

RESUMO

Congenital pulmonary venous stenosis is an uncommon congenital heart defect and is a consequence of failed incorporation of the common right or left pulmonary vein into the left atrium during embryologic development. Clinical manifestations include progressive exertional dyspnea, cough, chest pain, and hemoptysis. In this report, we describe a 37-year-old woman with known congenital unilateral pulmonary venous stenosis and a unilateral hypoplastic right pulmonary artery who presented with recurrent episodes of hemoptysis, requiring multiple catheter-based interventions, and symptomatic pleural effusions, requiring multiple large-volume thoracenteses. The patient underwent successful video-assisted thoracoscopic pneumonectomy with resolution of symptoms.


Assuntos
Angiografia por Tomografia Computadorizada/métodos , Artéria Pulmonar/anormalidades , Estenose de Veia Pulmonar/congênito , Cirurgia Torácica Vídeoassistida/métodos , Malformações Vasculares/cirurgia , Anormalidades Múltiplas/diagnóstico por imagem , Anormalidades Múltiplas/cirurgia , Adulto , Dispneia/diagnóstico , Dispneia/etiologia , Feminino , Seguimentos , Hemoptise/diagnóstico , Hemoptise/etiologia , Humanos , Pneumonectomia/métodos , Artéria Pulmonar/cirurgia , Recuperação de Função Fisiológica , Medição de Risco , Estenose de Veia Pulmonar/diagnóstico por imagem , Estenose de Veia Pulmonar/cirurgia , Resultado do Tratamento , Malformações Vasculares/diagnóstico por imagem
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