Case Report: Effective and Safe Treatment With Certolizumab Pegol in Pregnant Patients With Cogan’s Syndrome: A Report of Three Pregnancies in Two Patients

Cogan’s syndrome is a rare autoimmune disease characterized by ocular inflammation and audiovestibular manifestations. Treatment consists of systemic glucocorticoids and other immunosuppressive agents including methotrexate, cyclophosphamide and TNF-α-inhibitors. Due to potential ovarian or fetal toxicity immunosuppressive treatment options are limited during pregnancies. Thus far there is a paucity of reports on pregnancies in Cogan’s syndrome. With minimal transplacental transfer, Certolizumab pegol is considered to be safe for the use in pregnant patients with underlying inflammatory diseases. However, there is no literature on the use of this TNF-α-inhibitor in Cogan’s syndrome in general and especially during gestation. Here we report three pregnancies in two Cogan’s Syndrome-patients treated with Certolizumab pegol. Treatment with Certolizumab pegol was effective and well tolerated in patients with Cogan’s syndrome and seems to be a safe treatment option during pregnancy.

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Atypical Cogan's syndrome: A case report and summary of current treatment options

International Journal of Pediatric Otorhinolaryngology ◽

10.1016/j.ijporl.2014.12.028 ◽

2015 ◽

Vol 79 (3) ◽

pp. 428-431 ◽

Cited By ~ 10

Author(s):

Debora Jančatová ◽

Karol Zeleník ◽

Pavel Komínek ◽

Petr Matoušek

Keyword(s):

Case Report ◽

Treatment Options ◽

Current Treatment ◽

Cogan’S Syndrome ◽

Cogan's Syndrome

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Use of immunosuppressive agents in the treatment of severe ocular and vascular manifestations of Cogan's syndrome

The American Journal of Medicine ◽

10.1016/0002-9343(90)90157-9 ◽

1990 ◽

Vol 88 (3) ◽

pp. 296-301 ◽

Cited By ~ 52

Author(s):

Nancy B. Allen ◽

C. Christine Cox ◽

Mary R. Jacobs ◽

Rex M. McCallum ◽

Barton F. Haynes ◽

...

Keyword(s):

Immunosuppressive Agents ◽

Cogan’S Syndrome ◽

Cogan's Syndrome

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A Rare Case of Atypical Cogan’s Syndrome Presenting as Encephalitis

Modern Rheumatology Case Reports ◽

10.1093/mrcr/rxab055 ◽

2021 ◽

Author(s):

Debashis Maikap ◽

Amrita Pradhan ◽

Prasanta Padhan

Keyword(s):

Immunosuppressive Treatment ◽

Young Patients ◽

Confirmatory Test ◽

High Morbidity ◽

Unknown Aetiology ◽

Cogan’S Syndrome ◽

Cogan's Syndrome ◽

Interstitial Keratitis ◽

Vestibular Symptoms ◽

Systemic Symptoms

Abstract Cogan’s syndrome (CS) is a rare autoimmune vasculitis of unknown aetiology characterised by non-syphilitic interstitial keratitis, audio-vestibular symptoms, sometimes systemic symptoms and multi-organ involvement. Atypical CS has other ocular features such as scleritis, episcelritis, retinitis and optic neuritis. Diagnosis of CS is purely clinical without a confirmatory test. Hereby, we report a case of atypical CS presenting with features of encephalitis who was treated successfully with intravenous pulse methylprednisolone with cyclophosphamide. It is important to consider Cogan’s syndrome in the differential diagnosis of encephalitis with ocular and vestibular symptoms in young patients, as high morbidity and mortality rates are effectively lowered by early immunosuppressive treatment.

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Effective and safe treatment for the inflammatory diseases of the nose and nasopharynx

Russian Medical Inquiry ◽

10.32364/2587-6821-2020-4-4-238-242 ◽

2020 ◽

Vol 4 (4) ◽

pp. 238-242

Author(s):

A.Yu. Ovchinnikov ◽

◽

N.A. Miroshnichenko ◽

Yu.O. Nikolaeva ◽

◽

...

Keyword(s):

Nasal Cavity ◽

Paranasal Sinuses ◽

Viral Infections ◽

Inflammatory Diseases ◽

Treatment Options ◽

Safe Treatment ◽

Inflammatory Disorders ◽

Acute Rhinosinusitis ◽

Related Quality ◽

Health Related

Inflammatory diseases of the nasal cavity and paranasal sinuses are identified as the leading cause of upper respiratory disorders. Nose breathing is of crucial importance to provide the entry for air to get into the respiratory system. In addition to oxygen saturation, nose breathing provides the exchange of air in the paranasal sinuses. This process is required to maintain normal microflora of the nasal cavity. In contrast, mouth breathing occurring during the inflammatory diseases of the nasal cavity is associated with discomfort and poor health-related quality of life and results in inflammation. Viral inflammatory disorders often exacerbate a patient’s condition and provoke bacterial inflammation. Recent widespread occurrence of antimicrobial resistance forced to change the paradigm in the treatment of inflammatory disorders and to refocus on time-proved topical therapies. Systemic antibiotics are now used only for strict indications. Other treatment options are widely applied. Thus, silver preparations characterized by antiseptic and bactericidal properties allow to avoid complications and to aid the recovery. They are the best choice of topical agents for treating acute rhinosinusitis. KEYWORDS: acute rhinitis, acute rhinosinusitis, acute respiratory viral infections, antibacterial drugs, topical treatment, argentum proteinate. FOR CITATION: Ovchinnikov A.Yu., Miroshnichenko N.A., Nikolaeva Yu.O. Effective and safe treatment for the inflammatory diseases of the nose and nasopharynx. Russian Medical Inquiry. 2020;4(4):238–242. DOI: 10.32364/2587-6821-2020-4-4-238-242.

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Immunogenicity of biologics in inflammatory bowel disease

Therapeutic Advances in Gastroenterology ◽

10.1177/1756283x17750355 ◽

2018 ◽

Vol 11 ◽

pp. 1756283X1775035 ◽

Cited By ~ 62

Author(s):

Séverine Vermeire ◽

Ann Gils ◽

Paola Accossato ◽

Sadiq Lula ◽

Amy Marren

Keyword(s):

Inflammatory Bowel Disease ◽

Bowel Disease ◽

Treatment Options ◽

Certolizumab Pegol ◽

Immunosuppressive Agents ◽

Serum Levels ◽

Clinical Implications ◽

Pharmacokinetic Data ◽

Biologic Therapies ◽

Inflammatory Bowel

Crohn’s disease and ulcerative colitis are chronic inflammatory disorders of the gastrointestinal tract. Treatment options include biologic therapies; however, a proportion of patients lose response to biologics, partly due to the formation of anti-drug antibodies (ADAbs). Concomitant immunosuppressive agents reduce the development of ADAbs. This review article aims to assess the immunogenicity of biologic therapies and their clinical implications. A comprehensive literature search was conducted for articles published January 2009 to August 2015 reporting immunogenicity to adalimumab (ADM), certolizumab pegol (CZP), golimumab, infliximab (IFX), ustekinumab, and vedolizumab in inflammatory bowel disease (IBD). Eligible articles were reviewed and quality assessed by independent reviewers. Overall, 122 publications reporting 114 studies were assessed. ADAbs were reported for all agents, but the percentage of patients developing ADAbs was extremely variable, with the highest (65.3%) being for IFX administration to patients with IBD. ADAb presence was frequently associated with a reduction in primary efficacy and a loss of response, and, for IFX, an increase in adverse events (AEs). Lower serum levels of ADM, CZP and IFX were seen in ADAbs-positive rather than ADAbs-negative patients; pharmacokinetic data were unavailable for other therapies. Little information was available regarding the timing of ADAb development; studies reported their detection from as early as 10–14 days up to months after treatment initiation. Biologic therapies carry an intrinsic risk of immunogenicity, although reported rates of ADAbs vary considerably. The clinical implications of immunogenicity are a concern for effective treatment; further research, particularly into the more recently approved biologics, is required.

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Cogan's syndrome – A rare aortitis, difficult to diagnose but with therapeutic potential

Revista da Associação Médica Brasileira ◽

10.1590/1806-9282.63.12.1028 ◽

2017 ◽

Vol 63 (12) ◽

pp. 1028-1031 ◽

Cited By ~ 3

Author(s):

Raiza Colodetti ◽

Guilherme Spina ◽

Tatiana Leal ◽

Mucio Oliveira Jr ◽

Alexandre Soeiro

Keyword(s):

Disease Control ◽

Rare Case ◽

Aortic Wall ◽

Therapeutic Potential ◽

Immunosuppressive Treatment ◽

Rare Condition ◽

Cogan’S Syndrome ◽

Ocular Manifestations ◽

Cogan's Syndrome

Summary The inflammation of aortic wall, named aortitis, is a rare condition that can be caused by a number of pathologies, mainly inflammatory or infectious in nature. In this context, the occurrence of combined audiovestibular and/or ocular manifestations eventually led to the diagnosis of Cogan's syndrome, making it the rare case, but susceptible to adequate immunosuppressive treatment and satisfactory disease control.

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TNF-α inhibitors in the treatment of hidradenitis suppurativa

Therapeutic Advances in Chronic Disease ◽

10.1177/2040622319851640 ◽

2019 ◽

Vol 10 ◽

pp. 204062231985164 ◽

Cited By ~ 7

Author(s):

Kevin T. Savage ◽

Kelsey S. Flood ◽

Martina L. Porter ◽

Alexa B. Kimball

Keyword(s):

Clinical Trials ◽

Hidradenitis Suppurativa ◽

Complex Disease ◽

Case Reports ◽

Treatment Options ◽

Certolizumab Pegol ◽

Case Series ◽

Phase Iii ◽

Two Phase ◽

Tnf Α

Hidradenitis suppurativa (HS) is a complex disease with a dramatic impact on the quality of life of patients that it afflicts. Despite this, there are few treatment options offering long-term relief. The exact pathophysiology of HS is unclear, although the current theory involves follicular obstruction, rupture, and subsequent inflammation leading to fistula and abscess development in intertriginous skin. Several inflammatory modulators have been implicated in the development of HS, including tumor necrosis factor (TNF)-α as well as interleukin (IL)-1β, IL-10, and IL-17. Initial evidence for the use of TNF-α inhibitors in HS stemmed from recognition that inflammatory bowel disease patients treated with these medications saw a concurrent improvement in their HS symptoms. Early case reports and case series illustrated TNF-α inhibitors’ value in the treatment of HS. Later, two phase III clinical trials, PIONEER I and PIONEER II, demonstrated that adalimumab is an efficacious treatment for HS. Infliximab represents another effective HS treatment option with its main advantage being dosing flexibility. In contrast, clinical trials have failed to show evidence for application of etanercept in HS. There is limited data on other TNF-α inhibitors such as certolizumab-pegol and golimumab. This review outlines the history, dosing, response, and adverse effects of TNF-α inhibitors in the treatment of HS.

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Cogan's syndrome: State of the art of systemic immunosuppressive treatment in adult and pediatric patients

Autoimmunity Reviews ◽

10.1016/j.autrev.2017.02.009 ◽

2017 ◽

Vol 16 (4) ◽

pp. 385-390 ◽

Cited By ~ 8

Author(s):

Paolo Mora ◽

Giacomo Calzetti ◽

S. Ghirardini ◽

P. Rubino ◽

S. Gandolfi ◽

...

Keyword(s):

Pediatric Patients ◽

State Of The Art ◽

Immunosuppressive Treatment ◽

Cogan’S Syndrome ◽

Cogan's Syndrome

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Autoimmune Inner Ear Disease: Immune Biomarkers, Audiovestibular Aspects, and Therapeutic Modalities of Cogan’s Syndrome

Journal of Immunology Research ◽

10.1155/2018/1498640 ◽

2018 ◽

Vol 2018 ◽

pp. 1-8 ◽

Cited By ~ 5

Author(s):

Oded Shamriz ◽

Yuval Tal ◽

Menachem Gross

Keyword(s):

Immunosuppressive Agents ◽

Autoimmune Disorder ◽

Cogan’S Syndrome ◽

Sentence Recognition ◽

Therapeutic Modalities ◽

Immune Biomarkers ◽

Cogan's Syndrome ◽

Include Tumor Necrosis Factor ◽

Inner Ear Disease ◽

Necrosis Factor

Cogan’s syndrome (CS) is a rare autoimmune disorder characterized by audiovestibular dysfunction and ocular inflammation. Currently, there is no specific serum autoantibody used in the diagnostic workup of CS. Treatment is based on immunosuppressive agents, mainly corticosteroids as first-line choice. Recently, novel therapeutic modalities in CS have emerged. These include tumor necrosis factor-α inhibitors and other biologicals. Despite medical treatment, hearing loss may progress to irreversible bilateral profound SNHL in approximately half of CS patients resulting in candidacy for cochlear implantation (CI). Due to the inflammatory nature of the disease that is causing endosteal reaction with partial obliteration or complete neoossification of the intracochlear ducts, early CI is recommended. CI provides excellent and stable hearing rehabilitation with high score of word and sentence recognition. In this review, we will discuss different aspects of CS including clinical presentation, diagnosis, treatment, and future directives.

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