Cogan syndrome with systemic manifestations

Background. Cogan syndrome is a rare systemic vasculitis with variable arterial involvement. Its true prevalence is unknown. It is characterized by ocular symptoms, hearing loss, vestibular dysfunction. Cogan syndrome can be classified as typical or atypical. Typical syndrome is characterized by a c...

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Main Authors: Nataliya А. Morova, Yulia V. Arbuzova
Format: Article
Language:Russian
Published: Open Systems Publication 2024-03-01
Series:Лечащий Врач
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Online Access:https://journal.lvrach.ru/jour/article/view/1186
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author Nataliya А. Morova
Yulia V. Arbuzova
author_facet Nataliya А. Morova
Yulia V. Arbuzova
author_sort Nataliya А. Morova
collection DOAJ
description Background. Cogan syndrome is a rare systemic vasculitis with variable arterial involvement. Its true prevalence is unknown. It is characterized by ocular symptoms, hearing loss, vestibular dysfunction. Cogan syndrome can be classified as typical or atypical. Typical syndrome is characterized by a combination of bilateral keratitis with hearing loss and vestibular dysfunction. Atypical syndrome includes other types of eye or hearing damage and cases when the interval separating these symptoms onset exceeds 2 years. Systemic manifestations are more common in atypical Cogan syndrome. There are different theories about the development of the disease. Currently, the most popular autoimmune theory is that Cogan syndrome is based on autoimmune vasculitis of the most vascularized layers of the eye, inner ear and other organs. The search for immune markers in Cogan syndrome led to the discovery of antibodies to antigens of the inner ear, cornea, and antiendothelial antibodies, but their diagnostic value is not great. Immune markers that allow a confident diagnosis have not yet been discovered.Objective. Report a case of the Cogan syndrome with systemic manifestations in a female patient.Results. We present the clinical case of Cogan syndrome in a 37-year-old patient. The disease was diagnosed based on the presence of acute conjunctivitis, acute bilateral sensorineural hearing loss, dizziness. These symptoms were preceded by fever, arthralgia, myalgia, diarrhea. Conjunctivitis and audiovestibular symptoms appeared at the same time. Additionally, the patient exhibited systemic manifestations such as polyserositis, pneumonitis, erosive colitis with malabsorption syndrome, abdominal lymphadenopathy, anemia, thrombocytopenia. The notable aspect of the case was a very early diagnosis (a week after the onset of the disease). This factor determined the achievement of a good and rapid response to treatment within the first week. Treatment included glucocorticoids, pulse cyclophosphamide therapy. Within a month, all symptoms were completely resolved.Conclusion. Early diagnosis and timely treatment of Cogan syndrome makes it possible to completely relieve symptoms of the syndrome and prevent deafness.
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spelling doaj-art-b5dba5e6fca048febf4108fcbad532002025-08-20T03:57:51ZrusOpen Systems PublicationЛечащий Врач1560-51752687-11812024-03-010271110.51793/OS.2024.27.2.0011174Cogan syndrome with systemic manifestationsNataliya А. Morova0Yulia V. Arbuzova1Federal State Budgetary Educational Institution of Higher Education Omsk State Medical University of the Ministry of Health of the Russian Federation;Budgetary healthcare institution of the Omsk region Regional Clinical HospitalBackground. Cogan syndrome is a rare systemic vasculitis with variable arterial involvement. Its true prevalence is unknown. It is characterized by ocular symptoms, hearing loss, vestibular dysfunction. Cogan syndrome can be classified as typical or atypical. Typical syndrome is characterized by a combination of bilateral keratitis with hearing loss and vestibular dysfunction. Atypical syndrome includes other types of eye or hearing damage and cases when the interval separating these symptoms onset exceeds 2 years. Systemic manifestations are more common in atypical Cogan syndrome. There are different theories about the development of the disease. Currently, the most popular autoimmune theory is that Cogan syndrome is based on autoimmune vasculitis of the most vascularized layers of the eye, inner ear and other organs. The search for immune markers in Cogan syndrome led to the discovery of antibodies to antigens of the inner ear, cornea, and antiendothelial antibodies, but their diagnostic value is not great. Immune markers that allow a confident diagnosis have not yet been discovered.Objective. Report a case of the Cogan syndrome with systemic manifestations in a female patient.Results. We present the clinical case of Cogan syndrome in a 37-year-old patient. The disease was diagnosed based on the presence of acute conjunctivitis, acute bilateral sensorineural hearing loss, dizziness. These symptoms were preceded by fever, arthralgia, myalgia, diarrhea. Conjunctivitis and audiovestibular symptoms appeared at the same time. Additionally, the patient exhibited systemic manifestations such as polyserositis, pneumonitis, erosive colitis with malabsorption syndrome, abdominal lymphadenopathy, anemia, thrombocytopenia. The notable aspect of the case was a very early diagnosis (a week after the onset of the disease). This factor determined the achievement of a good and rapid response to treatment within the first week. Treatment included glucocorticoids, pulse cyclophosphamide therapy. Within a month, all symptoms were completely resolved.Conclusion. Early diagnosis and timely treatment of Cogan syndrome makes it possible to completely relieve symptoms of the syndrome and prevent deafness.https://journal.lvrach.ru/jour/article/view/1186cogan syndromesystem vasculitisconjunctivitishearing losssystemic manifestationsglucocorticoidscyclophosphamide
spellingShingle Nataliya А. Morova
Yulia V. Arbuzova
Cogan syndrome with systemic manifestations
Лечащий Врач
cogan syndrome
system vasculitis
conjunctivitis
hearing loss
systemic manifestations
glucocorticoids
cyclophosphamide
title Cogan syndrome with systemic manifestations
title_full Cogan syndrome with systemic manifestations
title_fullStr Cogan syndrome with systemic manifestations
title_full_unstemmed Cogan syndrome with systemic manifestations
title_short Cogan syndrome with systemic manifestations
title_sort cogan syndrome with systemic manifestations
topic cogan syndrome
system vasculitis
conjunctivitis
hearing loss
systemic manifestations
glucocorticoids
cyclophosphamide
url https://journal.lvrach.ru/jour/article/view/1186
work_keys_str_mv AT nataliyaamorova cogansyndromewithsystemicmanifestations
AT yuliavarbuzova cogansyndromewithsystemicmanifestations