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3.
BMC Neurol ; 24(1): 247, 2024 Jul 17.
Artículo en Inglés | MEDLINE | ID: mdl-39020271

RESUMEN

BACKGROUND: The harlequin syndrome is a rare disorder of the autonomic nervous system characterized by unilateral diminished flushing and sweating of the face following exposure to heat or physical activity. It results from sympathetic dysfunction and most commonly occurs idiopathically. A secondary development due to an underlying pathology (e.g., carotid artery dissection, tumors) must be excluded at first appearance. There is evidence that the cranial autonomic system is involved in the pathophysiology of trigeminal autonomic headaches like hemicrania continua. Therefore, an overlap in the pathophysiology of harlequin syndrome and trigeminal autonomic headache disorders seems plausible. However, the association of a harlequin syndrome with hemicrania continua was never reported. CASE PRESENTATION: This work describes the case of a 42-year-old female patient presenting to our headache unit. The patient reported persisting unilateral headache of the right side of dragging or squeezing character accompanied by trigeminal autonomic symptoms, including lacrimation, nasal congestion, conjunctival injection and Horner's syndrome, and was responsive to treatment with 75mg/d indomethacin. Five months after the initial consultation, the patient noted that the upper right quadrant of her face was pale after jogging. A harlequin syndrome was diagnosed. Further, she developed a short-lasting, bilateral headache of pulsatile character during strenuous exercise consistent with exertional headache. Comprehensive diagnostic evaluations, encompassing cranial and cervical MRI scans, laboratory tests, and biopsies, culminated in the diagnosis of Sjögren's syndrome. This finding suggests that the trigemino-autonomic dysfunction may either be idiopathic or a direct manifestation of Sjögren's syndrome. CONCLUSIONS: This report documents the case of a rare combination of a headache resembling probable hemicrania continua and the harlequin syndrome (and even exertional headache). It illustrates the underlying anatomy of the autonomic nervous system in a clinical context and emphasizes the hypothesis of a pathophysiological link between abnormal sympathetic activity and trigeminal autonomic headaches.


Asunto(s)
Enfermedades del Sistema Nervioso Autónomo , Rubor , Hipohidrosis , Humanos , Femenino , Adulto , Rubor/diagnóstico , Rubor/etiología , Hipohidrosis/diagnóstico , Hipohidrosis/complicaciones , Hipohidrosis/fisiopatología , Enfermedades del Sistema Nervioso Autónomo/diagnóstico , Enfermedades del Sistema Nervioso Autónomo/complicaciones , Enfermedades del Sistema Nervioso Autónomo/fisiopatología , Cefalea/etiología , Cefalea/diagnóstico , Cefalea/fisiopatología
6.
JAMA Dermatol ; 160(6): 612-619, 2024 Jun 01.
Artículo en Inglés | MEDLINE | ID: mdl-38630457

RESUMEN

Importance: Treatment of erythema and flushing in rosacea is challenging. Calcitonin gene-related peptide (CGRP) has been associated with the pathogenesis of rosacea, raising the possibility that inhibition of the CGRP pathway might improve certain features of the disease. Objective: To examine the effectiveness, tolerability, and safety of erenumab, an anti-CGRP-receptor monoclonal antibody, for the treatment of rosacea-associated erythema and flushing. Design, Setting, and Participants: This single-center, open-label, single-group, nonrandomized controlled trial was conducted between June 9, 2020, and May 11, 2021. Eligible participants included adults with rosacea with at least 15 days of either moderate to severe erythema and/or moderate to extreme flushing. No concomitant rosacea treatment was allowed throughout the study period. Visits took place at the Danish Headache Center, Copenhagen University Hospital, Rigshospitalet in Copenhagen, Denmark. Participants received 140 mg of erenumab subcutaneously every 4 weeks for 12 weeks. A safety follow-up visit was performed at week 20. Data analysis occurred from January 2023 to January 2024. Intervention: 140 mg of erenumab every 4 weeks for 12 weeks. Main Outcomes and Measures: The primary outcome was mean change in the number of days with moderate to extreme flushing during weeks 9 through 12, compared with the 4-week run-in period (baseline). The mean change in number of days with moderate to severe erythema was a secondary outcome. Adverse events were recorded for participants who received at least 1 dose of erenumab. Differences in means were calculated with a paired t test. Results: A total of 30 participants (mean [SD] age, 38.8 [13.1] years; 23 female [77%]; 7 male [23%]) were included, of whom 27 completed the 12-week study. The mean (SD) number of days with moderate to extreme flushing was reduced by -6.9 days (95% CI, -10.4 to -3.4 days; P < .001) from 23.6 (5.8) days at baseline. The mean (SD) number of days with moderate to severe erythema was reduced by -8.1 days (95% CI, -12.5 to -3.7 days; P < .001) from 15.2 (9.1) days at baseline. Adverse events included transient mild to moderate constipation (10 participants [33%]), transient worsening of flushing (4 participants [13%]), bloating (3 participants [10%]), and upper respiratory tract infections (3 participants [10%]), consistent with previous data. One participant discontinued the study due to a serious adverse event (hospital admission due to gallstones deemed unrelated to the study), and 2 participants withdrew consent due to lack of time. Conclusions and Relevance: These findings suggest that erenumab might be effective in reducing rosacea-associated flushing and chronic erythema (participants generally tolerated the treatment well, which was consistent with previous data), and that CGRP-receptor inhibition holds potential in the treatment of erythema and flushing associated with rosacea. Larger randomized clinical trials are needed to confirm this finding. Trial Registration: ClinicalTrials.gov Identifier: NCT04419259.


Asunto(s)
Anticuerpos Monoclonales Humanizados , Eritema , Rubor , Rosácea , Humanos , Rosácea/tratamiento farmacológico , Rosácea/complicaciones , Femenino , Masculino , Persona de Mediana Edad , Anticuerpos Monoclonales Humanizados/administración & dosificación , Anticuerpos Monoclonales Humanizados/efectos adversos , Eritema/tratamiento farmacológico , Eritema/etiología , Rubor/etiología , Rubor/tratamiento farmacológico , Adulto , Resultado del Tratamiento , Antagonistas del Receptor Peptídico Relacionado con el Gen de la Calcitonina/administración & dosificación , Antagonistas del Receptor Peptídico Relacionado con el Gen de la Calcitonina/efectos adversos , Antagonistas del Receptor Peptídico Relacionado con el Gen de la Calcitonina/farmacología , Anciano , Índice de Severidad de la Enfermedad , Inyecciones Subcutáneas
9.
J Invest Dermatol ; 144(5): 934-935, 2024 May.
Artículo en Inglés | MEDLINE | ID: mdl-38099889
13.
Anaesth Crit Care Pain Med ; 42(1): 101162, 2023 02.
Artículo en Inglés | MEDLINE | ID: mdl-36162787

RESUMEN

BACKGROUND: Mesenteric traction syndrome (MTS) is commonly seen during major abdominal surgery and is characterised by facial flushing, hypotension, and tachycardia 15 min into surgery. MTS also impacts the postoperative course, as severe MTS has been associated with increased postoperative morbidity. However, despite MTS being common and severe MTS causing increased postoperative morbidity, the gaps in the literature are not clearly defined. We aimed to examine the diagnostic criteria, incidence, intraoperative and postoperative impact, and potential preventative measures of MTS while highlighting potential gaps in the literature. METHODS: We followed the Prisma guidelines and performed a systematic literature search. We included only human studies examining MTS. All hits were screened for title and abstract, followed by a full-text review by at least two authors for determining eligibility for inclusion. Data were extracted and risk of bias was assessed by two independent reviewers. RESULTS: A total of 37 studies, comprising 1102 patients were included in the review. The combined incidence of MTS during open abdominal surgery was found to be 76%, with 35% developing severe MTS. It was found that the development of MTS was associated with marked haemodynamic changes. It was also found that several different subjective diagnostic criteria exist and that severe MTS was associated with increased postoperative morbidity. Furthermore, several preventative measures for protecting against MTS have been examined, but only on the incidence of MTS and not on the postoperative course. CONCLUSION: MTS occurs in 76% of patients undergoing major abdominal surgery and is associated with deleterious haemodynamic effects, which are more pronounced in patients developing severe MTS. Severe MTS is also associated with a worse postoperative outcome. However, gaps are still present in the current literature on MTS.


Asunto(s)
Hipotensión , Tracción , Humanos , Incidencia , Tracción/efectos adversos , Hipotensión/epidemiología , Hipotensión/terapia , Hemodinámica , Rubor/etiología
17.
J Endocrinol Invest ; 45(3): 687-689, 2022 Mar.
Artículo en Inglés | MEDLINE | ID: mdl-34241830

RESUMEN

PURPOSE: The Primavera is considered amongst the greatest and controversial artistic masterpieces worldwide painted by renaissance artist Sandro Botticelli. The aim was to identify any underlying medical foundations for the painting. METHODS: Observational study. RESULTS: The painting reveals, a 'butterfly' malar rash, bilateral ptosis and a clear neck swelling consistent with a goitre in the figure of Flora. This could be explained by concomitant Graves' disease and systemic lupus erythematosus, or other presentations of multiple autoimmune syndrome. CONCLUSION: These findings highlight the likely presentation of the earliest pictorial depictions of thyroid disease with systemic lupus erythematosus and emphasize the exactitude of depiction demonstrated by Botticelli in renaissance era.


Asunto(s)
Enfermedad de Hashimoto , Lupus Eritematoso Sistémico , Medicina en las Artes/historia , Pinturas , Neoplasias de la Tiroides , Autoinmunidad , Blefaroptosis/diagnóstico , Diagnóstico Diferencial , Exantema/diagnóstico , Exantema/etiología , Rubor/diagnóstico , Rubor/etiología , Enfermedad de Hashimoto/diagnóstico , Enfermedad de Hashimoto/inmunología , Historia del Siglo XV , Humanos , Italia , Lupus Eritematoso Sistémico/diagnóstico , Lupus Eritematoso Sistémico/inmunología , Neoplasias de la Tiroides/diagnóstico , Neoplasias de la Tiroides/fisiopatología
18.
Artículo en Inglés | MEDLINE | ID: mdl-34769908

RESUMEN

Association between drinking and smoking has remained controversial since the association between two studies were influenced by various confounding. Thus, our study aimed to explore the causal effect of alcohol consumption and cigarette smoking using alcohol flushing as an instrument variable, which is free from confounders. We analyzed cross-sectional survey data from 2500 Korean young adults (1600 men and 900 women). Alcohol flushing was strongly associated with log transformed alcohol consumption (F = 272). In men, alcohol non-flushers were 1.41 times (95% CI 1.28-1.55) more likely to smoke 100 cigarettes in their lifetime in logistic regression analysis. Alcohol non-flushers were also 1.3 times (95% CI 1.21-1.40) more likely to become daily smokers and 1.39 times (95% CI 1.27-1.51) more likely to be current smokers than alcohol flushers. However, in an IV analysis, no causal relationships between alcohol consumption and smoking status were found. Alcohol consumption, on the other hand, was causally associated with lowering nicotine dependence and former smoking in men. Alcohol consumption determined by alcohol flushing status does not appear to be causally linked to the smoking behavior of young adults. The relationship between alcohol consumption and nicotine dependence and smoking cessation needs further study.


Asunto(s)
Fumar Cigarrillos , Consumo de Bebidas Alcohólicas/epidemiología , Estudios Transversales , Femenino , Rubor/epidemiología , Rubor/etiología , Humanos , Masculino , Humo , Adulto Joven
20.
Pediatr Dermatol ; 38(4): 984-985, 2021 Jul.
Artículo en Inglés | MEDLINE | ID: mdl-34227168

RESUMEN

Harlequin syndrome is a rare disorder of the autonomic nervous system, presenting as unilateral reduced flushing and sweating of the face induced by exercise, stress, or heat. It is caused by a cervical sympathetic deficit located at the preganglionic or postganglionic level on the non-flushing side. We present a case of an 8 year old with harlequin syndrome and review the other dermatological conditions for which the term "harlequin" is part of the nomenclature.


Asunto(s)
Enfermedades del Sistema Nervioso Autónomo , Hipohidrosis , Enfermedades del Sistema Nervioso Autónomo/diagnóstico , Niño , Femenino , Rubor/diagnóstico , Rubor/etiología , Humanos , Hipohidrosis/diagnóstico , Sudoración
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