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This report presents two cases of patients with long-standing, treatment-resistant Hailey-Hailey disease (HHD) who experienced significant symptom relief through a combination therapy of oral naltrexone and dupilumab injections. The therapeutic potential of targeting the Th2 pathway and Ca2+ signaling with dupilumab in managing HHD manifestations is highlighted. The findings suggest that Th2 blockade with dupilumab, in conjunction with naltrexone, effectively controls recalcitrant HHD, indicating a role of cytokine response in altering disease pathogenesis. This case contributes to the growing body of literature on biologic treatments for HHD and suggests avenues for further research in HHD management.
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Carcinoma de Células Escamosas , Linfonodo Sentinela , Neoplasias Cutâneas , Humanos , Biópsia de Linfonodo Sentinela , Carcinoma de Células Escamosas/cirurgia , Carcinoma de Células Escamosas/patologia , Neoplasias Cutâneas/cirurgia , Neoplasias Cutâneas/patologia , Metástase Linfática/patologia , Linfonodo Sentinela/patologia , Linfonodos/patologiaRESUMO
Acne fulminans (AF) is a rare disorder marked by severe eruptions of inflamed nodules, hemorrhagic crusts, and ulcers accompanied by systemic symptoms and often laboratory abnormalities. Commonly affecting adolescent males with pre-existing acne, AF has been associated with isotretinoin therapy and elevated testosterone levels. With unknown pathogenesis, lesions frequently involve the trunk and face and are managed standardly with corticosteroids and isotretinoin. Uncontrolled or recurrent cases pose challenges due to prolonged high-dose corticosteroid use with increased scarring. In this study, we present a case of AF in a 17-year-old male unresponsive to corticosteroid and isotretinoin therapy, successfully treated with ustekinumab, an interleukin (IL)-12/23 inhibitor. The introduction of ustekinumab facilitated a controlled corticosteroid taper and isotretinoin dose escalation, resulting in significant clinical improvement of skin lesions and systemic symptoms. This case report underscores the potential of ustekinumab as a viable therapeutic option in the treatment of AF, particularly in cases where corticosteroid and isotretinoin combination therapy have proven ineffective.
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BACKGROUND: Oral benzodiazepines (BZDs) are useful tools for periprocedural anxiolysis. The United States Food and Drug Administration (FDA) recently issued a black-box warning of their risks of abuse and dependence. We performed a systematic review evaluating the safety and efficacy of oral BZDs for periprocedural anxiolysis in outpatient dermatologic, plastic surgery, dental, and ophthalmologic procedures performed under local anesthesia. METHODS: A systematic review of 5 databases was performed in accordance with the Preferred Reporting Items for Systematic Reviews and Meta-Analyses guidelines. Oral benzodiazepine safety and efficacy data were extracted from eligible articles. RESULTS: A total of 43 articles and 4,060 subjects were included. Oral BZDs consistently imparted a positive anxiolytic effect and demonstrated superior or equivalent scores in patient satisfaction, surgeon satisfaction, pain, and anxiety to comparator groups in most studies. Thirty-five subjects experienced transient hypoxia, and 2 experienced transient hypertension. A total of 195 mild, self-limited adverse effects were reported. None of the studies addressed the risks of abuse and dependence in this clinical setting. CONCLUSION: Short-term use of oral BZDs for periprocedural anxiolysis seems to be safe and effective. The 2020 FDA black-box warning should not deter their appropriate use in dermatologic surgery and other low-risk outpatient procedures.
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Anestesia , Ansiolíticos , Efeitos Colaterais e Reações Adversas Relacionados a Medicamentos , Ansiolíticos/efeitos adversos , Ansiedade , Benzodiazepinas/efeitos adversos , Humanos , Estados UnidosRESUMO
INTRODUCTION: Colchicine, because of its anti-inflammatory and possible anti-viral properties, has been proposed as potential therapeutic option for COVID-19. The role of colchicine to mitigate "cytokine storm" and to decrease the severity and mortality associated with COVID-19 has been evaluated in many studies. OBJECTIVE: To evaluate the role of colchicine on morbidity and mortality in COVID-19 patients. METHODS: This systematic review was conducted in accordance with the PRISMA recommendations. The literature search was conducted in 6 medical databases from inception to February 17, 2021 to identify studies evaluating colchicine as a therapeutic agent in COVID-19. All included studies were evaluated for risk of bias (ROB) using the Revised Cochrane ROB tool for randomised controlled trials (RCTs) and Newcastle-Ottawa Scale (NOS) for case-control and cohort studies. RESULTS: Four RCTs and four observational studies were included in the final analysis. One study evaluated colchicine in outpatients, while all others evaluated inpatient use of colchicine. There was significant variability in treatment protocols for colchicine and standard of care in all studies. A statistically significant decrease in all-cause mortality was observed in three observational studies. The risk of mechanical ventilation was significantly reduced only in one observational study. Length of hospitalisation was significantly reduced in two RCTs. Risk for hospitalisation was not significantly decreased in the study evaluating colchicine in outpatients. Very few studies had low risk of bias. CONCLUSION: Based on the available data, colchicine shall not be recommended to treat COVID-19. Further high-quality and multi-center RCTs are required to assess the meaningful impact of this drug in COVID-19.KEY MESSAGESColchicine, an anti-inflammatory agent has demonstrated anti-viral properties in in-vitro studies by degrading the microtubules, as well as by inhibiting the production of pro-inflammatory cytokines.Colchicine has been studied as a potential therapeutic option for COVID-19, with variable results.Until further research can establish the efficacy of colchicine in COVID-19, the use of colchicine in COVID-19 shall be restricted to clinical trials.
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Tratamento Farmacológico da COVID-19 , Colchicina/uso terapêutico , Humanos , Morbidade , Estudos Observacionais como Assunto , Respiração Artificial , SARS-CoV-2RESUMO
INTRODUCTION: In the unprecedented era of COVID-19, ongoing research and evolution of evidence has led to ever-changing guidelines for clinical monitoring and therapeutic options. Formulating treatment protocols requires the understanding and application of the evolving research. OBJECTIVE: The primary objective of this study is to present a systematic evidence-based approach to synthesize the necessary data in order to optimize the management of COVID-19. METHODS: At Mayo Clinic Florida, we developed a multidisciplinary centralized COVID Treatment Review Panel (TRP) of expert pulmonologists, intensivists, infectious disease specialists, anesthesiologists, hematologists, rheumatologists, and hospitalists that in real-time reviews the latest evidence in peer-reviewed journals, the available clinical trials, and help guide the rapid application of therapeutics or interventions to the patient and the bedside provider. RESULTS/CONCLUSIONS: The multi-disciplinary team approach of synthesizing clinical data and coordinating care is effective in responding to rapidly evolving and changing evidence. Systematic data collection and evidence-based treatment algorithms enable physicians to rapidly translate the current literature to clinical practice, and improve care and outcomes of patients.
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Brooke-Spiegler Syndrome (BSS) is a rare autosomal dominant familial disorder resulting in dermatologic neoplasms of copious nodular appendages. Here, we report a case of Familial Cylindromatosis (FC), a subtype of BSS, in a patient with the largest cylindroma of 7.4 × 5.6 × 3.8 cm on the scalp. The patient had undiagnosed cylindromas growing for 36 years at presentation; however, he did not seek out healthcare evaluation. Excision and pathologic investigation of three large masses from different body sites determined a shared phenotype of cylindromas. Subsequent evaluation of the patient's son separately, after primary patient excision, confirmed cylindroma development as well. The pathologic evidence of cylindromas in the patient with a new history of family incidence confirmed the diagnosis of the FC variant of BSS.
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Hydroxychloroquine, initially used as an antimalarial, is used as an immunomodulatory and anti-inflammatory agent for the management of autoimmune and rheumatic diseases such as systemic lupus erythematosus. Lately, there has been interest in its potential efficacy against severe acute respiratory syndrome coronavirus 2, with several speculated mechanisms. The purpose of this review is to elaborate on the mechanisms surrounding hydroxychloroquine. The review is an in-depth analysis of the antimalarial, immunomodulatory, and antiviral mechanisms of hydroxychloroquine, with detailed and novel pictorial explanations. The mechanisms of hydroxychloroquine are related to potential cardiotoxic manifestations and demonstrate potential adverse effects when used for coronavirus disease 2019 (COVID-19). Finally, current literature associated with hydroxychloroquine and COVID-19 has been analyzed to interrelate the mechanisms, adverse effects, and use of hydroxychloroquine in the current pandemic. Currently, there is insufficient evidence about the efficacy and safety of hydroxychloroquine in COVID-19. KEY MESSAGES HCQ, initially an antimalarial agent, is used as an immunomodulatory agent for managing several autoimmune diseases, for which its efficacy is linked to inhibiting lysosomal antigen processing, MHC-II antigen presentation, and TLR functions. HCQ is generally well-tolerated although severe life-threatening adverse effects including cardiomyopathy and conduction defects have been reported. HCQ use in COVID-19 should be discouraged outside clinical trials under strict medical supervision.
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Antimaláricos/uso terapêutico , Cardiotoxicidade/etiologia , Infecções por Coronavirus/tratamento farmacológico , Hidroxicloroquina/uso terapêutico , Pneumonia Viral/tratamento farmacológico , Antimaláricos/farmacologia , COVID-19 , Ensaios Clínicos como Assunto , Humanos , Hidroxicloroquina/farmacologia , Pandemias , Tratamento Farmacológico da COVID-19RESUMO
Francisella tularensis is disseminated in nature by biting arthropods such as mosquitoes. The relationship between mosquitoes and F. tularensis in nature is highly ambiguous, due in part to the fact that mosquitoes have caused significant tularemia outbreaks despite being classified as a mechanical vector of F. tularensis. One possible explanation for mosquitoes being a prominent, yet mechanical vector is that these insects feed on flower nectar between blood meals, allowing for transmission of F. tularensis between mosquitoes. Here, we aimed to assess whether F. tularensis could survive in flower nectar. Moreover, we examined if mosquitoes could interact with or ingest and transmit F. tularensis from one source of nectar to another. F. tularensis exhibited robust survivability in flower nectar with concentrations of viable bacteria remaining consistent with the rich growth medium. Furthermore, F. tularensis was able to survive (albeit to a lesser extent) in 30% sucrose (a nectar surrogate) over a period of time consistent with that of a typical flower bloom. Although we observed diminished bacterial survival in the nectar surrogate, mosquitoes that fed on this material became colonized with F. tularensis. Finally, colonized mosquitoes were capable of transferring F. tularensis to a sterile nectar surrogate. These data suggest that flower nectar may be capable of serving as a temporary source of F. tularensis that could contribute to the amplification of outbreaks. Mosquitoes that feed on an infected mammalian host and subsequently feed on flower nectar could deposit some F. tularensis bacteria into the nectar in the process. Mosquitoes subsequently feeding on this nectar source could potentially become colonized by F. tularensis. Thus, the possibility exists that flower nectar may allow for vector-vector transmission of F. tularensis.