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Chronic spontaneous urticaria presents with wheals and/or angioedema for >6 weeks without any specific triggers. The incidence of chronic spontaneous urticaria is increased in patients with comorbid autoimmune conditions. Here, we present a case of chronic spontaneous urticaria in a 9-year-old with type 1 diabetes and autoimmune thyroid disease who first presented with insulin pump site reactions concerning an insulin-related allergy. The patient was successfully treated with antihistamines and later immunosuppression with resumption of insulin pump therapy and remission of chronic spontaneous urticaria symptoms 18 months after onset.

This case report examines a rare occurrence of chronic spontaneous urticaria (CSU) following the administration of the CoronaVac vaccine for COVID-19. The patient developed persistent urticarial lesions that appeared and disappeared over an extended period after receiving the vaccine. The diagnosis of CSU was supported by histopathological examination and the close temporal correlation between symptom onset and vaccination. The discussion focuses on the immune mechanisms involved in CSU, the potential triggers of allergic reactions to COVID-19 vaccines, and the importance of further research to identify specific allergenic components. This case underscores the need for vigilance in monitoring and reporting adverse events related to COVID-19 vaccination to ensure vaccine safety and optimize public health strategies.

UNASSIGNED: Chronic spontaneous urticaria (CSU) is defined as the spontaneous occurrence of hives, angioedema, or both for more than 6 weeks; several inciting triggers including vaccines have been implicated. Coronavirus disease 2019 (COVID-19) vaccinations have been well tolerated by patients with CSU. However, reports have emerged of CSU triggered by COVID-19 vaccination and this study describes a South African case series.
UNASSIGNED: To provide details of the first case series of new-onset CSU post-COVID-19 vaccination in Africa and summarize the global literature of reported cases to date.
UNASSIGNED: All patients referred to our Urticaria Center of Excellence in Cape Town from the initiation of the COVID-19 vaccine rollout in South Africa (from February 2021 to August 2022) were reviewed to identify patients who developed new-onset CSU within 12 weeks of receiving a COVID-19 vaccine. Medical history, physical examinations, and laboratory investigations were reviewed.
UNASSIGNED: More than 20 million adults received COVID-19 vaccinations in South Africa during the study period. Eight patients had new-onset chronic urticaria post-COVID-19 vaccination; 6 of the 8 patients were female, the median age was 41 years (interquartile range [IQR], 38-44), and all had a history of atopy. Only 1 reported COVID-19 infection post vaccination. Chronic urticaria occurred following Pfizer-BioNTech, AstraZeneca, and Janssen Ad26.COV2.S vaccination in 6, 1, and 1 patient, respectively, with a median of 12 days (IQR, 3-38) from vaccination to symptoms onset. The baseline median score for Urticarial Activity Score 7 was 34 (IQR, 29-40), and 5 of the 8 patients (63%) had a total IgE level of more than 43 IU/L. All patients received high-dose antihistamines, with only 3 patients controlled.
UNASSIGNED: New-onset CSU can rarely be triggered by COVID-19 vaccinations, most commonly mRNA vaccines. COVID-19 vaccine-triggered CSU appears to have a phenotype similar to that triggered by other inciting agents and across populations.

BACKGROUND: Anaphylaxis is an acute, potentially life-threatening allergic reaction that typically occurs after exposure to a trigger, while idiopathic anaphylaxis (IA) occurs in the absence of a trigger. Acute management of both triggered anaphylaxis and IA relies on the use of epinephrine. In some patients with recurrent IA, glucocorticoid prophylaxis with prednisone can be effective. While there is currently no high quality evidence for the use of other prophylactic options to prevent recurrent IA, evolving data exists to support the consideration of biologics that target IgE or the Th2 pathway.
METHODS: We present the case of a 28 year old female with no atopic or autoimmune history with recurrent episodes of IA since childhood occurring up to twice weekly. There was improvement in acute symptoms with administration of first or second generation antihistamines and/or intramuscular epinephrine. Without an identifiable trigger, she was diagnosed with IA and frequent idiopathic urticaria and omalizumab was added to her treatment regimen with improvement in symptom frequency. After being lost to follow up, she had recurrence of symptom frequency and severity without omalizumab therapy and subsequently presented to our institution. Her workup at this point was negative for food allergy, alpha gal syndrome, systemic mastocytosis, hereditary alpha tryptasemia, carcinoid syndrome, and pheochromocytoma, and she was trialed on dupilumab with near resolution of her symptom frequency over a six month time period.
CONCLUSIONS: Recurrent IA is a diagnosis of exclusion that is associated with high morbidity. Prophylaxis remains an area of uncertainty, although prednisone has been effective in some cases. When prednisone is contraindicated or ineffective for the prevention of IA, biologic therapies that target IgE or the Th2 pathway may present a reasonable consideration. This case adds support to the suggestion that dupilumab may be a logical off-label consideration for prophylaxis of recurrent IA. The data for dupilumab in this clinical scenario is still very limited, and further research is required before any recommendation can be made.

Wheat-dependent exercise-induced anaphylaxis is a rare but severe form of anaphylaxis that occurs after consuming wheat products and engaging in physical activity. A case study of a 30-year-old woman suffering from chronic urticaria for the last 5 years highlights the difficulty in diagnosing this condition, as specific triggers were not identified. A diagnostic study called MADx revealed a positive analysis for omega-5-gliadin, leading to a diagnosis of wheat-dependent exercise-induced anaphylaxis. Delayed diagnosis is a common issue, and it can be challenging to distinguish wheat-dependent exercise-induced anaphylaxis from other conditions with similar symptoms. The treatment involves avoiding wheat products and always carrying an epinephrine auto-injector. When evaluating patients with similar symptoms, healthcare providers should include wheat-dependent exercise-induced anaphylaxis in their differential diagnosis. Patients should be educated about the symptoms, triggers, and management to seek immediate medical attention in an emergency.

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Urticaria is a disease triggered by mast cells and characterized by recurrent symptoms such as wheals and/or angioedema. Most patients benefit from treatment with antihistamines (AH) or, if not effective enough, from additional therapy with omalizumab, which leads to significant symptom relief. Here we present a patient with therapy-resistant chronic spontaneous urticaria treated with AH, omalizumab, and glucocorticosteroids. The subsequent recommended therapy according to the current guideline, cyclosporine A, was contraindicated in this patient. Therefore, therapy with dupilumab was initiated, resulting in a complete control of symptoms. In this case report, we present a case of successful dual therapy with omalizumab and dupilumab.

UNASSIGNED: Chronic spontaneous urticaria (CSU) can be extremely debilitating to the patient and challenging for the treating clinician. The National Institute of Health and Clinical Excellence (NICE) in the United Kingdom (UK) recommendation of omalizumab for patients who fail to respond to high-dose anti-histamines has improved treatment options and quality of life. However, there is still lack of clear guidelines for treatment of patients resistant to standard and anti-IgE therapies.
UNASSIGNED: We discuss the therapeutic strategies employed among nine extremely resistant CSU cases and the heterogeneity between guidelines from different societies.
UNASSIGNED: Patients with anti-histamine-resistant urticaria either remained on omalizumab or started on immunosuppressive drugs (dapsone or ciclosporin) when they stopped responding to omalizumab. We used clinical assessment, skin biopsies (when available) and previous published reports to consider dapsone (for predominantly neutrophilic infiltration), or ciclosporin at doses between 2 and 4 mg/kg/day. One patient with ciclosporin-resistant urticaria responded to mycophenolate mofetil. Two patients remain on long-term omalizumab due to its relative safety and efficacy including 1 patient with underlying antibody deficiency where omalizumab was preferred over risks of using immunosuppressive medications.
UNASSIGNED: These case studies bring to light the real-world difficulties in managing patients with resistant CSU and the need for generating the evidence base on alternative therapeutic options such as synergistic use of biologics and immunosuppressive drugs.

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