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Cogan syndrome
No full textTo lead ophthalmologists to consider Cogan syndrome
when managing a patient presenting with keratitis or other ocular
inflammation accompanied by sensorineural hearing loss. Methods.
Seven patients affected by Cogan syndrome were studied: two
males and five females, ranging from 27 to 65 years of age (mean
age: 41 years). Subjects were evaluated for a period ranging from
22 to 46 months (mean follow up time: 29.2 months). All patients
were treated with immunosuppressive drug combination therapy
(IDCT). Results. Three patients were affected by classic Cogan
syndrome (i.e., vestibuloauditory symptoms and later sensorineural
hearing loss and interstitial keratitis). Four patients presented
atypical Cogan syndrome (i.e., sensorineural hearing loss and
chronic ocular inflammation such as uveitis, scleritis, conjunctivitis,
retinal vasculitis, etc.). Four of these patients had a late diagnosis.
Two of them were diagnosed when they already had a
cochlear implant, one with bilateral deafness underwent cochlear
implantation 1 year after the beginning of IDCT, one had severe
bilateral hearing loss that improved during the first year of IDCT,
and then rapidly worsened to total deafness in 1 month following
an episode of severe systemic hypotension. Three patients who had
an early diagnosis of Cogan syndrome had no worsening of vestibuloauditory
dysfunction during the follow up period. Conclusion.
Diagnosis of Cogan syndrome should not be overlooked by
ophthalmologists in all patients with recurrent ocular inflammatory
disease associated with vestibuloauditory symptoms. Early diagnosis
is essential to commence the appropriate immunosuppressive
therapy that may prevent permanent hearing loss and ocular
dysfunctionPurpose. To lead ophthalmologists to consider Cogan syndrome when managing a patient presenting with keratitis or other ocular inflammation accompanied by sensorineural hearing loss. Methods. Seven patients affected by Cogan syndrome were studied: two males and live females, ranging from 27 to 65 years of age (mean age: 41 years). Subjects were evaluated for a period ranging from 22 to 46 months (mean follow up time: 29.2 months). All patients were treated with immunosuppressive drug combination therapy (IDCT). Results. Three patients Were affected by classic Cogan syndrome (i.e., vestibuloauditory symptoms and later sensorineural hearing loss and interstitial keratitis). Four patients presented atypical Cogan syndrome (i.e., sensorineural hearing loss and chronic ocular inflammation such as uveitis, scleritis, conjunctivitis, retinal vasculitis, etc.). Four of these patients had a late diagnosis. Two of them were diagnosed when they already had a cochlear implant, one with bilateral deafness underwent cochlear implantation I year after the beginning of IDCT, one had severe bilateral hearing loss that improved during the first year of IDCT, and then rapidly worsened to total deafness in 1 month following an episode of severe systemic hypotension. Three patients who had an early diagnosis of Cogan syndrome had no worsening of vestibuloauditory dysfunction during the follow up period. Conclusion. Diagnosis of Cogan syndrome should riot be overlooked by ophthalmologists in all patients with recurrent ocular inflammatory disease associated with vestibuloauditory symptoms. Early diagnosis is essential to commence the appropriate immunosuppressive therapy that may prevent permanent hearing loss and ocular dysfunction
Discontinuous drug combination therapy in autoimmune ocular disorders
No full textAbstract
PURPOSE: This study aimed to assess the effectiveness of a steroid-sparing immunosuppressive treatment (IST) protocol in the control of severe or steroid-resistant autoimmune ocular inflammatory diseases.
METHODS: We carried out a prospective, non-randomized clinical study. Patients presenting with ocular inflammations that failed to respond adequately to steroids alone after monotherapy for a mean period of 9 +/- 2 months (internal control) were offered the option to switch to a combined IST. The protocol consisted of different immunosuppressive drugs added in a stepladder sequence, where each drug (including the steroids) was administered discontinuously. Main outcome measures were control of inflammation, visual acuity and safety of treatment.
RESULTS: A total of 76 subjects (121 affected eyes) enrolled in the IST protocol. Mean length of follow-up was 43 +/- 15 months. Complete control of inflammation was achieved in 86% of patients. During the first year of IST, the rate of inflammatory recurrences/patient was 0.78 +/- 1.13. This ratio diminished further during succeeding follow-up. Mean best corrected visual acuity improved from 0.31 logMAR to 0.24 logMAR (p < 0.001). Blood pressure and uric acid blood levels significantly altered for the worse in the study group.
CONCLUSIONS: Immunosuppressive treatment was effective in achieving inflammatory quiescence in a large majority of patients. The study also demonstrated the longterm safety of the protocol and its steroid-sparing effect
I pazienti che sviluppano oftalmopatia tiroidea (OT) progressiva presentano valori di esoftalmometria (Hertel) più elevati all'esordio.
No full textCapacità predittiva di un modello di rete neurale per la progressione dell'oftalmopatia tiroidea: confronto con la valutazione oftalmologica.
No full textIncidence of ocular Zoonoses referred to the Inflammatory and autoimmune ocular diseases service of the University of Parma - Italy.
No full textPrediction of the progression of thyroid-associated ophthalmopathy at the first patient examination: comparison between a neural network model and clinical ophthalmological assessment.
No full textThe use of a neural network model for the prediction of progression of thyroid-associated ophthalmopathy (TAO) at the first patient examination
No full textCogan syndrome: confocal microscopy assessment of corneal damage
No full textPURPOSE: To perform in vivo assessment of corneal alterations in patients with Cogan syndrome (CS) and to correlate these findings with prior histopathologic descriptions.
METHODS: Four consecutive patients (8 eyes) presenting with typical CS underwent confocal microscopy examination. At the moment of evaluation, ocular inflammation was quiescent in all the patients. The images were studied singularly, and then compared to those obtained from 5 healthy controls. Statistical analysis was performed with Student t test.
RESULTS: All the cases showed multiple brightly reflective deposits in the stroma with moderate reflectivity of the surrounding stromal tissue. The outer corneal layers and the endothelium were spared. In 3/4 of the patients, the nerve fibers of the subepithelial plexus were thin and poorly reflective, with interruptions and lack of the typical branching pattern. No cells different from keratocytes could be detected in either case or control eyes. Corneal vascularization was present in one case. In CS-affected eyes, corneal thickness was 552+/-62 microm on average, and endothelial cell count was 2466+/-288 cells/mm(2). Statistical significance was not reached compared to the control group.
CONCLUSIONS: Confocal microscopy was able to provide detailed images of corneal alterations in CS. This confirmed the interstitial nature of the keratitis and showed an excellent accordance with prior histopathologic findings. The absence of inflammatory cell infiltration may be explained by the prolonged control of ocular inflammation in our patients, which may also have limited stimuli for corneal neovascularization
Retrospective analysis of clinical ophthalmological signs in patient with thyroid-associated ophthalmopathy: implementation of a neural network model for the prediction of disease progression.
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