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1.
Medicine (Baltimore) ; 103(39): e39768, 2024 Sep 27.
Artigo em Inglês | MEDLINE | ID: mdl-39331946

RESUMO

RATIONALE: The prevalence of idiopathic retinitis, vasculitis, aneurysms, and neuroretinitis (IRVAN) syndrome is <1/1,000,000, and it tends to occur in young women. Panretinal photocoagulation, focal laser photocoagulation, peripheral cryopexy, vitrectomy, and corticosteroid therapy are the traditional treatments. We reported a case of a senile patient with IRVAN syndrome who presented with severe exudation in both eyes and macular edema in the left eye, successfully treated by serial intravitreal aflibercept injections. PATIENT CONCERNS: A 77-year-old Taiwanese woman complained of progressive blurred vision in the left eye and ocular examinations revealed a visual acuity of 20/125 in the left eye. DIAGNOSIS: Indirect fundoscopy and fluorescein angiography showed bilateral multiple aneurysms, vasculitis, optic nerve staining, and neovascularizations. In addition, optical coherence tomography demonstrated macular edema with subretinal fluid and exudations in the left eye. INTERVENTIONS: Monthly intravitreal injections of antivascular endothelial growth factor with aflibercept 2.0 mg were administered in the left eye. OUTCOMES: The visual acuity in the left eye improved to 20/50 after 18 months of treatment. Macular edema and subretinal fluid regressed. Furthermore, vessel leakage and optic disc staining also improved. LESSONS: This case is the first to demonstrate successful treatment of IRVAN syndrome-related central macular edema and exudations, in the absence of neovascularization, using a series of intravitreal aflibercept injections as monotherapy.


Assuntos
Aneurisma , Injeções Intravítreas , Edema Macular , Receptores de Fatores de Crescimento do Endotélio Vascular , Proteínas Recombinantes de Fusão , Vasculite Retiniana , Retinite , Humanos , Feminino , Receptores de Fatores de Crescimento do Endotélio Vascular/administração & dosagem , Receptores de Fatores de Crescimento do Endotélio Vascular/uso terapêutico , Proteínas Recombinantes de Fusão/administração & dosagem , Proteínas Recombinantes de Fusão/uso terapêutico , Idoso , Edema Macular/tratamento farmacológico , Edema Macular/etiologia , Retinite/tratamento farmacológico , Retinite/diagnóstico , Vasculite Retiniana/tratamento farmacológico , Aneurisma/tratamento farmacológico , Aneurisma/complicações , Síndrome , Inibidores da Angiogênese/administração & dosagem , Inibidores da Angiogênese/uso terapêutico , Acuidade Visual , Tomografia de Coerência Óptica
3.
BMJ Case Rep ; 16(11)2023 Nov 27.
Artigo em Inglês | MEDLINE | ID: mdl-38011962

RESUMO

A woman in her 40s presented with massive haemoptysis and breathlessness for 1 day. She had been diagnosed with pulmonary tuberculosis based on sputum CBNAAT (Cartridge Based Nucleic Acid Amplification Test) and was on antitubercular treatment for previous 2 weeks. Her chest X-ray showed right middle lobe lateral segment dense consolidation with bilateral nodular infiltrates. CT pulmonary angiography (CTPA) revealed a well-defined homogenously enhancing vascular lesion of size 10×11×13 mm in the right hilar region communicating with the descending branch of right pulmonary artery, suggesting a Rasmussen's aneurysm. It was in close proximity to the segmental bronchus that was almost completely occluded, suggesting epituberculosis. Transvenous pulmonary artery glue embolisation successfully achieved complete ablation of the aneurysm with preserved arterial flow. She has later completed 6 months of antitubercular treatment and is cured with no recurrence of haemoptysis. Her lung infiltrates have resolved with some lung scarring.


Assuntos
Aneurisma , Hemoptise , Feminino , Humanos , Hemoptise/terapia , Aneurisma/tratamento farmacológico , Artéria Pulmonar/diagnóstico por imagem , Angiografia , Angiografia por Tomografia Computadorizada , Antituberculosos/uso terapêutico
4.
Lancet Child Adolesc Health ; 7(10): 697-707, 2023 10.
Artigo em Inglês | MEDLINE | ID: mdl-37598693

RESUMO

BACKGROUND: Although Kawasaki disease is commonly regarded as a single disease entity, variability in clinical manifestations and disease outcome has been recognised. We aimed to use a data-driven approach to identify clinical subgroups. METHODS: We analysed clinical data from patients with Kawasaki disease diagnosed at Rady Children's Hospital (San Diego, CA, USA) between Jan 1, 2002, and June 30, 2022. Patients were grouped by hierarchical clustering on principal components with k-means parcellation based on 14 variables, including age at onset, ten laboratory test results, day of illness at the first intravenous immunoglobulin infusion, and normalised echocardiographic measures of coronary artery diameters at diagnosis. We also analysed the seasonality and Kawasaki disease incidence from 2002 to 2019 by subgroup. To explore the biological underpinnings of identified subgroups, we did differential abundance analysis on proteomic data of 6481 proteins from 32 patients with Kawasaki disease and 24 healthy children, using linear regression models that controlled for age and sex. FINDINGS: Among 1016 patients with complete data in the final analysis, four subgroups were identified with distinct clinical features: (1) hepatobiliary involvement with elevated alanine transaminase, gamma-glutamyl transferase, and total bilirubin levels, lowest coronary artery aneurysm but highest intravenous immunoglobulin resistance rates (n=157); (2) highest band neutrophil count and Kawasaki disease shock rate (n=231); (3) cervical lymphadenopathy with high markers of inflammation (erythrocyte sedimentation rate, C-reactive protein, white blood cell, and platelet counts) and lowest age-adjusted haemoglobin Z scores (n=315); and (4) young age at onset with highest coronary artery aneurysm but lowest intravenous immunoglobulin resistance rates (n=313). The subgroups had distinct seasonal and incidence trajectories. In addition, the subgroups shared 211 differential abundance proteins while many proteins were unique to a subgroup. INTERPRETATION: Our data-driven analysis provides insight into the heterogeneity of Kawasaki disease, and supports the existence of distinct subgroups with important implications for clinical management and research design and interpretation. FUNDING: US National Institutes of Health and the Irving and Francine Suknow Foundation.


Assuntos
Aneurisma , Síndrome de Linfonodos Mucocutâneos , Estados Unidos , Humanos , Criança , Síndrome de Linfonodos Mucocutâneos/complicações , Síndrome de Linfonodos Mucocutâneos/epidemiologia , Imunoglobulinas Intravenosas/uso terapêutico , Proteômica , Análise por Conglomerados , Aneurisma/tratamento farmacológico
5.
Trials ; 24(1): 60, 2023 Jan 26.
Artigo em Inglês | MEDLINE | ID: mdl-36703139

RESUMO

BACKGROUND: Kawasaki disease (KD) is an acute self-limiting inflammatory vasculitis affecting predominantly medium-sized arteries, particularly the coronary arteries. A number of recent studies conducted in different European countries have demonstrated alarmingly high coronary complications despite treatment with intravenous immunoglobulin (IVIG). These high complication rates now emphasize the need for an urgent reappraisal of IVIG as the sole primary therapeutic agent for KD. The Kawasaki disease CAA prevention (KD-CAAP) trial will test the hypothesis that immediate adjunctive corticosteroid treatment to standard of care IVIG and aspirin will reduce coronary artery aneurysm (CAA) rates in unselected KD patients across Europe. METHODS: KD-CAAP is a multicentre, randomised, controlled, open-label, blinded endpoint assessed trial that will be conducted across Europe supported by the conect4children pan-European clinical trials network. Patients with KD who satisfy the eligibility criteria will be randomised (1:1) to receive either oral prednisolone 2 mg/kg/day plus standard of care therapy IVIG (2 g/kg) and aspirin (40 mg/kg/day); or IVIG and aspirin alone. Further management is dictated by temperature and C-reactive protein (CRP) responses. Co-primary outcomes are as follows: (i) any CAA within the 3 months of trial follow-up; (ii) average estimate of maximum coronary Z-score at weeks 1, 2 and 6 adjusting for rescue treatment. Additional outcomes will be assessed including cost effectiveness, quality of life, corticosteroid toxicity and other safety outcomes. DISCUSSION: Several recent studies have indicated that coronary complications associated with KD across Europe are much higher than early trials of IVIG had initially suggested. KD-CAAP directly addresses this issue by exploring the therapeutic benefit of adjunctive corticosteroids in unselected KD cases. If we find that corticosteroids prevent CAA and are safe, this is a cheap and widely available intervention that could be implemented immediately for the benefit of children. TRIAL REGISTRATION: ISRCTN71987471- March 31, 2020; Eudract 2019-004433-17.


Assuntos
Corticosteroides , Aneurisma , Aspirina , Imunoglobulinas Intravenosas , Síndrome de Linfonodos Mucocutâneos , Criança , Humanos , Lactente , Corticosteroides/efeitos adversos , Corticosteroides/uso terapêutico , Aneurisma/complicações , Aneurisma/tratamento farmacológico , Aspirina/efeitos adversos , Aspirina/uso terapêutico , Vasos Coronários , Imunoglobulinas Intravenosas/efeitos adversos , Imunoglobulinas Intravenosas/uso terapêutico , Síndrome de Linfonodos Mucocutâneos/complicações , Síndrome de Linfonodos Mucocutâneos/diagnóstico , Síndrome de Linfonodos Mucocutâneos/tratamento farmacológico , Estudos Multicêntricos como Assunto , Qualidade de Vida , Ensaios Clínicos Controlados Aleatórios como Assunto , Pré-Escolar , Adolescente
6.
Ocul Immunol Inflamm ; 31(2): 437-444, 2023 Feb.
Artigo em Inglês | MEDLINE | ID: mdl-35081005

RESUMO

PURPOSE: Idiopathic retinal vasculitis, aneurysms and neuroretinitis is a rare vision-threatening condition. If untreated vision loss occurs due to complications of progressive retinal ischaemia including retinal neovascularisation, neovascular glaucoma and retinal exudation. Despite the proposed underlying inflammatory aetiology this condition demonstrates poor response to corticosteroid treatment. The aim was to describe two paediatric cases of idiopathic retinal vasculitis, aneurysms and neuroretinitis treated with infliximab. METHODS: Two case reports. RESULTS: Infliximab treatment led to resolution of aneurysmal dilatations and retinal vasculitis, and reversal of some retinal capillary non-perfusion. CONCLUSION: Early infliximab treatment should be considered in cases of idiopathic retinal vasculitis, aneurysms and neuroretinitis.


Assuntos
Aneurisma , Coriorretinite , Vasculite Retiniana , Retinite , Humanos , Criança , Vasculite Retiniana/diagnóstico , Vasculite Retiniana/tratamento farmacológico , Vasculite Retiniana/complicações , Infliximab/uso terapêutico , Retinite/diagnóstico , Retinite/tratamento farmacológico , Coriorretinite/complicações , Aneurisma/diagnóstico , Aneurisma/tratamento farmacológico , Aneurisma/complicações , Angiofluoresceinografia
7.
Ocul Immunol Inflamm ; 31(3): 627-630, 2023 Apr.
Artigo em Inglês | MEDLINE | ID: mdl-35316150

RESUMO

PURPOSE: To report a case of IRVAN in a 13-year-old girl responding well to Adalimumab and Azathioprine. RESULTS: A 13-year-old girl presented to us with central scotoma for a duration of 10 months. She was treated earlier with oral steroids with poor response. Fundus examination revealed features of IRVAN. She was treated with intravitreal dexamethasone implant in both eyes with oral Mycophenolate Mofetil (MMF) with transient response to it. So she was switched over to subcutaneous Adalimumab 40 mg once in 2 weeks and oral Azathioprine 50 mg BD. The disease activity was well controlled with the current regime. CONCLUSION: Though various treatment modalities have been described in literature for the treatment of IRVAN. This is the first case of IRVAN to be treated with Adalimumab along with Azathioprine to be reported.


Assuntos
Aneurisma , Coriorretinite , Vasculite Retiniana , Retinite , Feminino , Humanos , Adolescente , Vasculite Retiniana/diagnóstico , Vasculite Retiniana/tratamento farmacológico , Adalimumab/uso terapêutico , Azatioprina/uso terapêutico , Angiofluoresceinografia , Retinite/diagnóstico , Retinite/tratamento farmacológico , Aneurisma/diagnóstico , Aneurisma/tratamento farmacológico
9.
Genes (Basel) ; 13(5)2022 04 22.
Artigo em Inglês | MEDLINE | ID: mdl-35627118

RESUMO

Objectives: Protein C (PC) deficiency is an inherited thrombophilia with a prevalence of 0.5% in the general population and 3% in subjects with a first-time deep vein thrombosis (DVT). Here we report a series of 14 PC-deficient Polish patients with comprehensive clinical and molecular characteristics, including long-term follow-up data and a deep mutational analysis of the PROC gene. Patients and Methods: Fourteen unrelated probands (mean ± SD age 43.8 ± 13.0 years) with suspicion of PC deficiency, who experienced thromboembolic events and a majority of whom received anticoagulants (92.8%), were screened for PROC mutations by sequencing the nine PROC exons and their flanking intron regions. Results: Ten probands (71.4%) had missense mutations, two patients (14.3%) carried nonsense variants, and the other two subjects (14.3%) had splice-site mutations, the latter including the c.401-1G>A variant, reported here for the very first time. The proband carrying the c.401-1A allele had a hepatic artery aneurysm with a highly positive family history of aneurysms and the absence of any mutations known to predispose to this vascular anomaly. Conclusion: A novel detrimental PROC mutation was identified in a family with aneurysms, which might suggest yet unclear links of thrombophilia to vascular anomalies, including aneurysms at atypical locations in women. The present case series also supports data indicating that novel oral anticoagulants (NOACs) are effective in PC deficient patients.


Assuntos
Aneurisma , Deficiência de Proteína C , Trombofilia , Trombose , Administração Oral , Adulto , Aneurisma/tratamento farmacológico , Anticoagulantes/uso terapêutico , Feminino , Humanos , Pessoa de Meia-Idade , Mutação , Polônia , Proteína C/genética , Proteína C/metabolismo , Proteína C/uso terapêutico , Deficiência de Proteína C/tratamento farmacológico , Deficiência de Proteína C/genética , Trombose/genética
10.
ACS Appl Mater Interfaces ; 13(43): 50705-50715, 2021 Nov 03.
Artigo em Inglês | MEDLINE | ID: mdl-34689548

RESUMO

Thrombosis is closely related to the instability of intracranial aneurysm (IA), whose rupture is associated with high morbidity and mortality. It is difficult to detect an IA-related thrombus because traditional magnetic resonance imaging (MRI) and even contrast-enhanced MRI cannot clearly distinguish a thrombus from the surrounding tissues. Herein, a nanoplatform [(MFe2O4-ZnDPA nanoparticles (NPs)], consisting of Zn0.4Co0.6Fe2O4@Zn0.4Mn0.6Fe2O4 NPs for imaging and Zn(II)-bis(dipicolylamine) (ZnDPA) for thrombus targeting, is constructed to target an experimental aneurysm-related thrombus in rabbits via MRI. In vitro experiments including platelet safety evaluation primarily prove that MFe2O4-ZnDPA NPs with a high MRI transverse relaxation time (T2) have good biocompatibility. MFe2O4-ZnDPA NPs could target a thrombus via the special interaction between ZnDPA and phosphatidylserine of activated platelets in the thrombus through MRI and Fe quantification assays. Moreover, after MFe2O4-ZnDPA NPs are injected into the ear vein of common carotid artery aneurysm model rabbits, MRI shows that MFe2O4-ZnDPA NPs could accumulate in the aneurysm-related thrombus from 0 to 15 min after injection and decrease in the next 45 min. Meanwhile, MFe2O4-ZnDPA NPs could decrease the MRI T2 signal of the aneurysm-related thrombus to enhance the outline of the aneurysm. This study demonstrates that a nanoplatform can enhance the detection of an aneurysm-related thrombus as well as aneurysm itself to assist further treatment of IA.


Assuntos
Aneurisma/tratamento farmacológico , Imageamento por Ressonância Magnética , Estruturas Metalorgânicas/farmacologia , Nanopartículas/química , Trombose/tratamento farmacológico , Aneurisma/diagnóstico por imagem , Animais , Sobrevivência Celular/efeitos dos fármacos , Estruturas Metalorgânicas/síntese química , Estruturas Metalorgânicas/química , Estrutura Molecular , Ativação Plaquetária/efeitos dos fármacos , Coelhos , Trombose/diagnóstico por imagem
11.
Sci Rep ; 11(1): 13856, 2021 07 05.
Artigo em Inglês | MEDLINE | ID: mdl-34226641

RESUMO

Coronary artery (CA) stenosis is a detrimental and often life-threatening sequela in Kawasaki disease (KD) patients with coronary artery aneurysm (CAA). Therapeutic strategies for these patients have not yet been established. All-trans-retinoic acid (atRA) is a modulator of smooth muscle cell functions. The purpose of this study was to investigate the effect of atRA on CA stenosis in a mouse model of KD. Lactobacillus casei cell wall extract (LCWE) was intraperitoneally injected into 5-week-old male C57BL/6 J mice to induce CA stenosis. Two weeks later, the mice were orally administered atRA (30 mg/kg) 5 days per week for 14 weeks (LCWE + atRA group, n = 7). Mice in the untreated group (LCWE group, n = 6) received corn oil alone. Control mice were injected with phosphate-buffered saline (PBS, n = 5). Treatment with atRA significantly suppressed CA inflammation (19.3 ± 2.8 vs 4.4 ± 2.8, p < 0.0001) and reduced the incidence of CA stenosis (100% vs 18.5%, p < 0.05). In addition, atRA suppressed the migration of human coronary artery smooth muscle cells (HCASMCs) induced by platelet-derived growth factor subunit B homodimer (PDGF-BB). In conclusion, atRA dramatically alleviated CA stenosis by suppressing SMC migration. Therefore, it is expected to have clinical applications preventing CA stenosis in KD patients with CAA.


Assuntos
Aneurisma/tratamento farmacológico , Estenose Coronária/tratamento farmacológico , Síndrome de Linfonodos Mucocutâneos/tratamento farmacológico , Tretinoína/farmacologia , Aneurisma/induzido quimicamente , Aneurisma/patologia , Animais , Movimento Celular/efeitos dos fármacos , Parede Celular/química , Estenose Coronária/induzido quimicamente , Estenose Coronária/patologia , Vasos Coronários/efeitos dos fármacos , Vasos Coronários/patologia , Modelos Animais de Doenças , Humanos , Lacticaseibacillus casei/química , Lipopolissacarídeos/química , Lipopolissacarídeos/toxicidade , Camundongos , Síndrome de Linfonodos Mucocutâneos/induzido quimicamente , Síndrome de Linfonodos Mucocutâneos/patologia , Miócitos de Músculo Liso/efeitos dos fármacos
13.
BMJ Case Rep ; 13(12)2020 Dec 17.
Artigo em Inglês | MEDLINE | ID: mdl-33334747

RESUMO

A 25-year-old Indian man presented with low-grade fever followed by gradually increasing swelling of neck and face. Physical examination showed bilateral neck swelling, facial swelling and dilated veins in the upper chest. Superior vena cava (SVC) obstruction due to an underlying malignancy was suspected. CT thorax showed large saccular aneurysm with thrombosis of bilateral subclavian arteries of which the right one caused external compression of right innominate vein draining into the SVC. A history of recurrent oral and scrotal ulcers was obtained following which skin pathergy test was done, which was suggestive of a diagnosis of Behcet's disease (BD). He responded to treatment with steroids and azathioprine. This report illustrates that rare nonmalignant cause such as BD could also present with SVC obstruction.


Assuntos
Aneurisma/diagnóstico , Síndrome de Behçet/diagnóstico , Artéria Subclávia/imunologia , Síndrome da Veia Cava Superior/diagnóstico , Adulto , Aneurisma/tratamento farmacológico , Aneurisma/imunologia , Anticoagulantes/administração & dosagem , Azatioprina/administração & dosagem , Síndrome de Behçet/sangue , Síndrome de Behçet/complicações , Síndrome de Behçet/imunologia , Proteína C-Reativa/análise , Glucocorticoides/administração & dosagem , Humanos , Imageamento Tridimensional , Masculino , Testes Cutâneos , Artéria Subclávia/diagnóstico por imagem , Síndrome da Veia Cava Superior/sangue , Síndrome da Veia Cava Superior/tratamento farmacológico , Síndrome da Veia Cava Superior/etiologia , Tomografia Computadorizada por Raios X , Resultado do Tratamento , Veia Cava Superior/diagnóstico por imagem
14.
Biomolecules ; 10(3)2020 02 26.
Artigo em Inglês | MEDLINE | ID: mdl-32111073

RESUMO

BACKGROUND: Matrix metalloproteinases (MMPs) are involved in vascular wall degradation, and drugs able to modulate MMP activity can be used to prevent or treat aneurysmal disease. In this study, we evaluated the effects of statins on MMP-2, MMP-9, and neutrophil gelatinase-associated lipocalin (NGAL) in both plasma and tissue in patients with aneurysmal disease. METHODS: We performed a prospective, single-blind, multicenter, control group clinical drug trial on 184 patients of both sexes >18 years old with a diagnosis of arterial aneurysmal disease. Enrolled patients were divided into two groups: Group I under statin treatment and Group II not taking statins. In addition, 122 patients without aneurysmal disease and under statin treatment were enrolled as a control group (Group III). The expression of MMPs and NGAL in plasma was evaluated using ELISA, while their expression in endothelial tissues was evaluated using Western blot. RESULTS: The ELISA test revealed greater plasma levels (p < 0.01) of MMPs and NGAL in Groups I and II vs. Group III. Western blot analysis showed higher expression (p < 0.01) of MMPs and NGAL in Group II vs. Group I, and this increase was significantly higher (p < 0.01) in patients treated with low potency statins compared to high potency ones. CONCLUSIONS: MMPs and NGAL seem to play a major role in the development of aneurysms, and their modulation by statins suggests that these drugs could be used to prevent arterial aneurysmal disease.


Assuntos
Aneurisma/tratamento farmacológico , Aneurisma/patologia , Inibidores de Hidroximetilglutaril-CoA Redutases/uso terapêutico , Lipocalina-2/análise , Metaloproteinase 2 da Matriz/análise , Metaloproteinase 9 da Matriz/análise , Idoso , Aneurisma/sangue , Feminino , Humanos , Lipocalina-2/sangue , Masculino , Metaloproteinase 2 da Matriz/sangue , Metaloproteinase 9 da Matriz/sangue , Pessoa de Meia-Idade , Projetos Piloto , Estudos Prospectivos
15.
Intern Med ; 59(3): 377-381, 2020 Feb 01.
Artigo em Inglês | MEDLINE | ID: mdl-31645531

RESUMO

A 44-year-old man was admitted to our hospital due to heart failure. Transthoracic echocardiography demonstrated global hypokinesis with an ejection fraction of 25%, prominent trabeculation and deep intertrabecular recesses, and apical aneurysm with multiple thrombi (10×13 mm in the inferior wall, 15×8 mm in the anterior wall). Cardiac magnetic resonance imaging showed an increased ratio of noncompacted (NC) to compacted (C) myocardium (NC/C ratio >2.3) and apical aneurysm. Coronary angiography revealed no significant stenosis. He was therefore diagnosed with left ventricular noncompaction complicated by apical aneurysm. Four weeks after starting anticoagulation, the multiple apical thrombi disappeared without clinical signs of embolism.


Assuntos
Aneurisma/complicações , Aneurisma/tratamento farmacológico , Anticoagulantes/uso terapêutico , Insuficiência Cardíaca/complicações , Insuficiência Cardíaca/tratamento farmacológico , Ventrículos do Coração/fisiopatologia , Trombose/fisiopatologia , Adulto , Aneurisma/diagnóstico por imagem , Aneurisma/fisiopatologia , Angiografia Coronária , Ecocardiografia , Insuficiência Cardíaca/diagnóstico por imagem , Insuficiência Cardíaca/fisiopatologia , Ventrículos do Coração/diagnóstico por imagem , Humanos , Imageamento por Ressonância Magnética , Masculino , Trombose/etiologia , Resultado do Tratamento
17.
Am J Case Rep ; 20: 1805-1811, 2019 Dec 04.
Artigo em Inglês | MEDLINE | ID: mdl-31796724

RESUMO

BACKGROUND Pulmonary artery aneurysm (PAA) is a rare disease in cardiovascular system. This disease is difficult to diagnose and less often considered due to its non-specific clinical manifestations. Until now there are no clear guidelines about its optimal management because of the small number of reported cases. CASE REPORT We report a 56-year-old male with chief complain of atypical bilateral chest pain and shortness of breath. Initial electrocardiogram (ECG) and laboratory evaluation showed no sign of ischemic heart disease. After the patient was stabilized, he was evaluated using chest x-ray, transthoracic echocardiography (TTE), and multi slice computed tomography (MSCT). The patient was then diagnosed with PAA secondary to suspicion of pulmonary hypertension (PH) with chronic obstructive pulmonary disease and heart failure. Conservative treatment was chosen because of the limited resources for surgery and patient's refusal to be referred. The treatment aims to lower the pulmonary artery pressure while monitoring the aneurysm. His 6-month follow-up evaluation showed an improvement in pulmonary artery pressure and persistent of the PAA without any increasement of the diameter. CONCLUSIONS PAA is a rare disease that is difficult to diagnose because of its non-specific nature. Persistent atypical chest pain can be an early symptom of PAA, thus clinicans should be aware in a high-risk patient suffered persistent chest pain, despite normal ECG and laboratory findings. TTE and MSCT evaluation are reliable for diagnosing PH and PAA. With conservative treatment and routine follow-up, patient with PAA secondary to PH could be managed well.


Assuntos
Aneurisma/diagnóstico por imagem , Aneurisma/etiologia , Hipertensão Pulmonar/complicações , Hipertensão Pulmonar/diagnóstico por imagem , Artéria Pulmonar/diagnóstico por imagem , Artéria Pulmonar/patologia , Aneurisma/tratamento farmacológico , Dor no Peito , Quimioterapia Combinada , Dispneia , Ecocardiografia , Insuficiência Cardíaca/diagnóstico por imagem , Insuficiência Cardíaca/tratamento farmacológico , Insuficiência Cardíaca/etiologia , Humanos , Hipertensão Pulmonar/tratamento farmacológico , Masculino , Pessoa de Meia-Idade , Artéria Pulmonar/efeitos dos fármacos , Tomografia Computadorizada por Raios X
19.
Pediatr Pulmonol ; 54(6): E13-E15, 2019 06.
Artigo em Inglês | MEDLINE | ID: mdl-30938918

RESUMO

We report the first case of Hughes Stovin Syndrome successfully treated with long-lasting tumor necrosis factor α (TNF-α) blocker (infliximab) treatment. Because of the failure of the standard therapeutic regimen with steroids and cyclophosphamide, infliximab was started achieving a stable disease remission and a complete resolution of pulmonary aneurysms. Hughes Stovin Syndrome, although rare, is a life-threatening condition that needs to be timely identified and treated aggressively. Our report underlines the importance of TNF-α blocker treatment in Hughes Stovin Syndrome, suggesting its use as long-term safe and useful.


Assuntos
Aneurisma/tratamento farmacológico , Infliximab/uso terapêutico , Fator de Necrose Tumoral alfa/antagonistas & inibidores , Adolescente , Aneurisma/diagnóstico , Síndrome de Behçet , Ciclofosfamida/uso terapêutico , Humanos , Pulmão/patologia , Masculino , Prednisona/uso terapêutico , Síndrome
20.
World Neurosurg ; 126: 146-150, 2019 06.
Artigo em Inglês | MEDLINE | ID: mdl-30858000

RESUMO

BACKGROUND: Cranial artery dolichoectasia is characterized by dilated, elongated, and tortuous cranial arteries. Dolichoectasia and dolichoectatic aneurysm-particularly of the internal carotid arteries (ICAs)-are rare but can lead to stroke. We report a case of suspected recurrent acute cerebral infarction and dolichoectatic aneurysm of the left ICA combined with right dolichoectatic aneurysm, vertebrobasilar dolichoectasia, and atrial fibrillation. CASE DESCRIPTION: An 80-year-old man experienced 3 days of recurrent aphasia episodes. He had a history of atrial fibrillation and hypertension. Neurologic findings indicated Wernicke aphasia. Magnetic resonance imaging showed acute cerebral infarction in the left temporal and parietal lobes. Magnetic resonance angiography and cerebral angiography showed dolichoectatic aneurysms in the C1-2 portion of the bilateral ICAs and vertebrobasilar dolichoectasia. Possible diagnoses included convulsion, cardioembolic infarction, atherothrombotic cerebral infarction, and acute ischemic stroke associated with the dolichoectatic aneurysm of the left ICA. Oral antiplatelet drugs, direct oral anticoagulants, and anticonvulsants were administered. Nine months after initial hospitalization, the patient had a recurrence of acute ischemic stroke and was rehospitalized. He was treated with a new direct oral anticoagulant, clopidogrel, statins, and ethyl icosapentate. At 2 years after initial hospitalization, he has had no recurrence of stroke, has a modified Rankin Scale score of 2, and is currently being followed as an outpatient. CONCLUSIONS: A direct oral anticoagulant (edoxaban) and clopidogrel were effective for suspected recurrent acute ischemic stroke associated with dolichoectatic aneurysm of the ICA combined with contralateral dolichoectatic aneurysm, vertebrobasilar dolichoectasia, and atrial fibrillation.


Assuntos
Aneurisma/complicações , Isquemia Encefálica/complicações , Doenças das Artérias Carótidas/complicações , Acidente Vascular Cerebral/complicações , Insuficiência Vertebrobasilar/complicações , Idoso de 80 Anos ou mais , Aneurisma/diagnóstico por imagem , Aneurisma/tratamento farmacológico , Anticoagulantes/uso terapêutico , Isquemia Encefálica/diagnóstico por imagem , Isquemia Encefálica/tratamento farmacológico , Doenças das Artérias Carótidas/diagnóstico por imagem , Doenças das Artérias Carótidas/tratamento farmacológico , Artéria Carótida Interna/diagnóstico por imagem , Clopidogrel/uso terapêutico , Humanos , Masculino , Inibidores da Agregação Plaquetária/uso terapêutico , Piridinas/uso terapêutico , Recidiva , Acidente Vascular Cerebral/diagnóstico por imagem , Acidente Vascular Cerebral/tratamento farmacológico , Tiazóis/uso terapêutico , Insuficiência Vertebrobasilar/diagnóstico por imagem , Insuficiência Vertebrobasilar/tratamento farmacológico
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