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May 2020 DOI 10.14302/issn.2641-5518.jcci-20-3317
This case report describes facial nerve palsy due to Ramsay Hunt syndrome, highlighting otic vesicles, audiovestibular symptoms, and imaging considerations. Management with antivirals, corticosteroids, and supportive care is summarized. Early recognition can improve outcomes and limit sequelae.
May 2019 DOI 10.14302/issn.2329-9487.jhc-19-2787
Introduction Cardiac non-specific inflammatory pseudotumors (NSIPT) are exceptionally associated to Behçet's disease (BD) and represent a real diagnostic and therapeutic challenge. The meaning and the mechanisms of this association are not yet well understood. The purpose of this paper is to study the epidemiologic, therapeutic, and evolutionary characteristics of cardiac NSIPT during BD Methods Systemic review of all reported cases of cardiac NSIPT associated with BD. Results We found only 6 cases of NSIPT associated with BD. Of these six patients, 4 were men (66.66%) and 2 were women (33.33%): Sex ratio =2. The average age was 26.66 years (9-35 years). The pseudotumor was unique in all cases. The chronology of occurrence of these NSIPT compared to the underlying angiitis was variable: inaugural of the disease in 4 cases, and complicating a previously known BD in 2 cases. The surgery was performed in all cases. It was carried out for diagnostic purpose in 4 cases, and therapeutic in the other 2. Additional medical treatment based on systemic corticosteroids with or without immunosuppressants was indicated in 4 patients. The evolution was favorable in 5 cases and a single case was quickly fatal. Recurrence of NSIPT was reported in one patient (20%). Conclusion The results of this review suggest a very likely association between BD and cardiac NSIPT; especially because of the scarcity of these two conditions in the general population, and the epidemiological characteristics clearly different from those of cardiac NSIPTs in the general population. The pathogenic mechanisms common to these two conditions (immune, inflammatory, reactive, and vascular) reinforce this causal link. The main differential diagnoses of these pseudotumors during BD remain cancer and intracardiac thrombosis.
Jan 2018 DOI 10.14302/issn.2470-5020.jnrt-17-1926
Since the first description in 2013, 39 cases of anti-DPPX-encephalitis have been described. Main features of this autoimmune encephalitis characterized by antibodies against the potassium-channel-associated regulatory protein DPPX are gastrointestinal symptoms, cognitive dysfunction and signs of CNS hyperexcitability. While the majority of patients responds to immunotherapy relapses are frequent and often successfully treated with rituximab. Here we report another case of anti-DPPX-encephalitis presenting with the above mentioned triad. However, this is the first case of anti-DPPX-encephalitis in the context of a connective tissue disease combined with cerebral arteriopathy along with brain parenchymal lesions that we interpreted as a secondary, CTD-associated cerebral vasculitis. While the latter resolved under immunosuppressive treatment, comprising glucocorticosteroids, cyclophosphamide, rituximab and plasmapheresis, deterioration of the CTD and multiple infectious complications finally led to the patient's death. As histological evidence for cerebral vasculitis is lacking, other differential diagnoses for the observed cerebral arteriopathy, especially reversible cerebral vasoconstriction syndrome, have to be considered.
Sep 2017 DOI 10.14302/issn.2471-2175.jdrt-17-1774
Topical corticosteroids are main therapeutic agents for the treatment of a wide variety of dermatological disorders. Three cases of chronic cutaneous ulceration affecting the skin of the breasts that were caused presumably by previous and continued use of potent topical corticosteroids for the treatment of psoriasis and/or intertrigo lesions were reported here. These three cases were presented to emphasize the potential serious local side effects of topical steroids even progressing to ulceration in breast' skin. These cases highlight the importance of appropriate use of topical corticosteroids, necessity of a strict follow up for adverse effects and the need of warning every patient about possible side effects of topical corticosteroids
Aug 2017 DOI 10.14302/issn.2470-0436.jos-17-1666
Purpose: To evaluate early versus late immunomodulatory therapy (IMT) for patients following initial diagnosis of Vogt-Koyanagi-Harada (VKH) disease. Methods: Retrospective review including all VKH patients seen 5/1/2014 to 4/1/2016 at LAC+USC. Early IMT was defined as starting an immunomodulatory agent within 3 months of corticosteroid initiation. Results: Twenty-seven patients were included, of whom 15 received early IMT and 8 received late IMT. Early IMT patients trended toward greater improvement in vision compared to late IMT (logMAR 0.59 vs. 0.11; p=0.14) with no differences in ocular complications including ocular hypertension (p=0.53) and cataract (p=1.0). Patients receiving early IMT averaged 0.93 recurrences versus 2.13 recurrences for late IMT (p=0.092). Of patients successfully taper off oral corticosteroids, the early IMT group was tapered in an average of 8.3 months versus 19.8 months for late IMT (p=0.0019). Conclusions: Early IMT in VKH may allow for shorter duration of corticosteroids with similar visual outcomes, ocular complications, and disease recurrences.