RESUMO
OBJECTIVES: Juvenile onset recurrent respiratory papillomatosis (JORRP) can cause severe or disseminated disease. Surgical treatment may be inadequate. Systemic bevacizumab has shown initial success for severe JORRP. The objective of this systematic review was to assess usage, effectiveness, and safety of this treatment. METHODS: We searched PubMed, Embase, and Web of Science for studies of humans with JORRP treated with systemic bevacizumab. Two researchers independently reviewed the studies to determine inclusion and aggregate data on patient characteristics, dosing protocols, treatment response, adverse events, and level of evidence. RESULTS: Of 80 identified articles, 12 studies were included detailing 20 distinct cases. At a mean age of 12.8 years (range = 1-43 years) patients received initial dosing of 5 to 10 mg/kg of bevacizumab followed by ongoing doses at a mean 3-week intervals (range = 2-5 weeks). All patients had clinically significant disease reduction with reduced need for surgery. Six patients (30%) had complete response in at least one involved anatomic site. Eleven (55%) required no surgery after initiating treatment. There was recurrence in all four patients whose treatment was stopped, but had rapid improvement with treatment resumption. Six (30%) experienced mild or moderate adverse events. CONCLUSIONS: Marked improvement in severe JORRP has been reported from systemic bevacizumab. Treatment protocols vary, and treatment discontinuation was not feasible in any reported patient. Based on currently available data, systemic bevacizumab can be considered for severe JORRP as it appears to be well tolerated and effective. A clinical trial could enhance the understanding of its safety and efficacy for this indication. Laryngoscope, 131:1138-1146, 2021.
Assuntos
Inibidores da Angiogênese/administração & dosagem , Bevacizumab/administração & dosagem , Infecções por Papillomavirus/tratamento farmacológico , Infecções Respiratórias/tratamento farmacológico , Prevenção Secundária/métodos , Inibidores da Angiogênese/efeitos adversos , Bevacizumab/efeitos adversos , Esquema de Medicação , Humanos , Infusões Intravenosas , Infecções por Papillomavirus/diagnóstico , Recidiva , Infecções Respiratórias/diagnóstico , Índice de Gravidade de Doença , Fatores de Tempo , Resultado do TratamentoRESUMO
Vocal fold paralysis (VFP) is an important cause of respiratory and feeding compromise in infants. The causes of neonatal VFP are varied and include central nervous system disorders, birth-related trauma, mediastinal masses, iatrogenic injuries, and idiopathic cases. Bilateral VFP often presents with stridor or respiratory distress and can require rapid intervention to stabilize an adequate airway. Unilateral VFP presents more subtly with a weak cry, swallowing dysfunction, and less frequently respiratory distress. The etiology and type of VFP is important for management. Evaluation involves direct visualization of the vocal folds, with additional imaging and testing in select cases. Swallowing dysfunction, also known as dysphagia, is very common in infants with VFP. A clinical assessment of swallowing function is necessary in all cases of VFP, with some patients also requiring an instrumental swallow assessment. Modification of feeding techniques and enteral access for feedings may be necessary. Airway management can vary from close monitoring to noninvasive ventilation, tracheostomy, and laryngeal surgery. Long-term follow-up with otolaryngology and speech-language pathology service is necessary for all children with VFP to ensure adequate breathing, swallowing, and phonation. The short- and long-term health and quality-of-life consequences of VFP can be substantial, especially if not managed early.
Assuntos
Manuseio das Vias Aéreas , Transtornos de Deglutição , Doenças do Recém-Nascido , Transtornos Respiratórios , Sons Respiratórios , Paralisia das Pregas Vocais , Transtornos de Deglutição/diagnóstico , Transtornos de Deglutição/etiologia , Transtornos de Deglutição/terapia , Humanos , Recém-Nascido , Doenças do Recém-Nascido/diagnóstico , Doenças do Recém-Nascido/etiologia , Doenças do Recém-Nascido/terapia , Transtornos Respiratórios/diagnóstico , Transtornos Respiratórios/etiologia , Transtornos Respiratórios/terapia , Sons Respiratórios/diagnóstico , Sons Respiratórios/etiologia , Paralisia das Pregas Vocais/complicações , Paralisia das Pregas Vocais/diagnóstico , Paralisia das Pregas Vocais/etiologia , Paralisia das Pregas Vocais/terapiaRESUMO
OBJECTIVE: Thyroglossal duct cysts (TGDCs) are relatively common congenital midline neck masses that are treated with surgical excision. Traditionally these are removed along with any associated tract and the central portion of the hyoid bone through an anterior neck incision. Some patients with TGDCs want to avoid an external neck scar. METHODS: We describe the details of a transoral endoscopic vestibular excision of a TGDC and the associated hyoid bone in an adolescent patient. RESULTS: This novel approach was successful and there were no complications. CONCLUSION: We propose that cervical TGDCs can be safely and completely removed with this approach in appropriately selected patients while avoiding a neck scar.
Assuntos
Osso Hioide/cirurgia , Cirurgia Endoscópica por Orifício Natural/métodos , Procedimentos Cirúrgicos Otorrinolaringológicos/métodos , Cisto Tireoglosso/cirurgia , Adolescente , Endoscopia/métodos , Humanos , Osso Hioide/diagnóstico por imagem , Imageamento por Ressonância Magnética , Masculino , Boca , Cisto Tireoglosso/diagnóstico por imagemRESUMO
Exercise provides protection against myocardial ischemia-reperfusion (IR) injury. Understanding the mechanisms of this protection may lead to new interventions for the prevention and/or treatment of heart disease. Although presently these mechanisms are not well understood, reports suggest that manganese superoxide dismutase (MnSOD) and calpain may be critical mediators of this protection. We hypothesized that an exercise-induced increase in MnSOD would provide cardioprotection by attenuating IR-induced oxidative modification to critical Ca(2+)-handling proteins, thereby decreasing calpain-mediated cleavage of these and other proteins attenuating cardiomyocyte death. After IR, myocardial apoptosis and infarct size were significantly reduced in hearts of exercised animals compared with sedentary controls. In addition, exercise prevented IR-induced calpain activation as well as the oxidative modification and calpain-mediated degradation of myocardial Ca(2+)-handling proteins (L-type Ca(2+) channels, phospholamban, and sarcoplasmic/endoplasmic reticulum calcium ATPase). Further, IR-induced activation of proapoptotic proteins was attenuated in exercised animals. Importantly, prevention of the exercise-induced increase in MnSOD activity via antisense oligonucleotides greatly attenuated the cardioprotection conferred by exercise. These results suggest that MnSOD provides cardioprotection by attenuating IR-induced oxidation and calpain-mediated degradation of myocardial Ca(2+)-handling proteins, thereby preventing myocardial apoptosis and necrosis.
Assuntos
Proteínas de Ligação ao Cálcio/metabolismo , Calpaína/metabolismo , Traumatismo por Reperfusão Miocárdica/prevenção & controle , Esforço Físico/fisiologia , Superóxido Dismutase/metabolismo , Animais , Apoptose , Proteína Agonista de Morte Celular de Domínio Interatuante com BH3/metabolismo , Caspase 12/metabolismo , Masculino , Modelos Cardiovasculares , Traumatismo por Reperfusão Miocárdica/metabolismo , Traumatismo por Reperfusão Miocárdica/patologia , Miócitos Cardíacos/metabolismo , Miócitos Cardíacos/patologia , Necrose , Oligonucleotídeos Antissenso/genética , Oligonucleotídeos Antissenso/farmacologia , Ratos , Ratos Sprague-Dawley , Superóxido Dismutase/antagonistas & inibidores , Superóxido Dismutase/genéticaRESUMO
Macrophage migration inhibitory factor acts via its intrinsic thiol-protein oxidoreductase activity to negatively regulate the neuronal chronotropic actions of angiotensin II in normotensive rat neurons. Because the chronotropic action of angiotensin II is potentiated in spontaneously hypertensive rat neurons, we investigated whether this negative regulatory mechanism is absent in these rats. Angiotensin II (100 nM) elicited an approximately 89% increase in neuronal firing in Wistar-Kyoto rat hypothalamus and brain stem cultured neurons and an increase in intracellular macrophage migration inhibitory factor levels in the same cells. The chronotropic action of angiotensin II was significantly greater (approximately 212% increase) in spontaneously hypertensive rat neurons, but angiotensin II failed to alter macrophage migration inhibitory factor expression in these cells. Intracellular application of recombinant macrophage migration inhibitory factor (0.8 nM) or its specific neuronal overexpression via Ad5-SYN-MIF (1x10(7) infectious units) significantly attenuated the chronotropic action of angiotensin II in spontaneously hypertensive rat neurons, similar to results from Wistar-Kyoto rat neurons. In contrast, C60S-macrophage migration inhibitory factor (0.8 nM), which lacks thiol-protein oxidoreductase activity, failed to alter the chronotropic action of angiotensin II in neurons from either rat strain. Thus, whereas macrophage migration inhibitory factor has the potential to depress the chronotropic action of angiotensin II in spontaneously hypertensive rat neurons, it is unlikely that this regulatory mechanism occurs, because angiotensin II does not increase the expression of this protein. The lack of this regulatory mechanism may contribute to the increased chronotropic action of angiotensin II in spontaneously hypertensive rat neurons.