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Cyclosporine A-Induced Conchal Hyperplasia with Nasal Obstruction in a Patient with Membranous Nephropathy

Patient: Female, 66-year-old Final Diagnosis: Cyclosporine induced hyperplasia of the tubinates Symptoms: Dyspnea Medication:— Clinical Procedure: Drug withdrawal Specialty: Dentistry • Nephrology • Otolaryngology OBJECTIVE: Unusual clinical course BACKGROUND: The immunomodulatory and pharmacokineti...

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Autores principales: Tillmann, Frank-Peter, Harth, Ana, Özcan, Fedai, Jörres, Achim
Formato: Online Artículo Texto
Lenguaje:English
Publicado: International Scientific Literature, Inc. 2022
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9131439/
https://www.ncbi.nlm.nih.gov/pubmed/35642551
http://dx.doi.org/10.12659/AJCR.935362
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author Tillmann, Frank-Peter
Harth, Ana
Özcan, Fedai
Jörres, Achim
author_facet Tillmann, Frank-Peter
Harth, Ana
Özcan, Fedai
Jörres, Achim
author_sort Tillmann, Frank-Peter
collection PubMed
description Patient: Female, 66-year-old Final Diagnosis: Cyclosporine induced hyperplasia of the tubinates Symptoms: Dyspnea Medication:— Clinical Procedure: Drug withdrawal Specialty: Dentistry • Nephrology • Otolaryngology OBJECTIVE: Unusual clinical course BACKGROUND: The immunomodulatory and pharmacokinetic effects of cyclosporine A are used to treat diverse disease entities in different medical fields, including organ transplantation and/or autoimmune diseases. It is also applied in patients with nephrotic range proteinuria as an adjunct to steroids and supportive antihypertensive/anti-proteinuric medications. Cyclosporine has a small therapeutic window and is dosed with respect to the underlying disease entity and severity via trough level adaptations. Among its most frequent adverse effects are hypertension, nephrotoxicity, neurotoxicity, and electrolyte disturbances. Hypertrichosis and gingival hyperplasia are obvious and widely recognized adverse effects. CASE REPORT: We report on a 66-year-old woman who was treated with cyclosporine A for primary membranous nephropathy. During treatment with cyclosporine, she developed hirsutism and gingival hyperplasia. Later, she reported having impaired nasal breathing and dyspnea on mild physical exercise. Clinical, rhinoscopic, and radiological evaluations showed marked conchal hyperplasia as a potential cause of her symptoms. An extensive medical work-up did not show evidence of allergic, immunologic, or other drug adverse effects, suggesting cyclosporine-induced hyperplasia of the turbinates as a hypothetical causative factor. Dose reductions did not lead to resolution of symptoms but resulted in increasing proteinuria. Therefore, cyclosporine was stopped, and the patient was treated with rituximab. Thereafter, hirsutism and gingival and conchal hyperplasia gradually regressed over 2–4 months, showing complete resolution of conchal hyperplasia on computed-tomography follow-up after 6 months. CONCLUSIONS: Cyclosporine can not only result in gingival hyperplasia but also in hyperplasia of the turbinates leading to impaired nasal breathing and shortness of breath on exertion. An extensive search for many other known causes of conchal swelling is warranted to finally suggest an adverse effect of cyclosporine. Discontinuation of cyclosporine resulted in complete remission of conchal hyperplasia as well as other adverse effects.
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spelling pubmed-91314392022-06-09 Cyclosporine A-Induced Conchal Hyperplasia with Nasal Obstruction in a Patient with Membranous Nephropathy Tillmann, Frank-Peter Harth, Ana Özcan, Fedai Jörres, Achim Am J Case Rep Articles Patient: Female, 66-year-old Final Diagnosis: Cyclosporine induced hyperplasia of the tubinates Symptoms: Dyspnea Medication:— Clinical Procedure: Drug withdrawal Specialty: Dentistry • Nephrology • Otolaryngology OBJECTIVE: Unusual clinical course BACKGROUND: The immunomodulatory and pharmacokinetic effects of cyclosporine A are used to treat diverse disease entities in different medical fields, including organ transplantation and/or autoimmune diseases. It is also applied in patients with nephrotic range proteinuria as an adjunct to steroids and supportive antihypertensive/anti-proteinuric medications. Cyclosporine has a small therapeutic window and is dosed with respect to the underlying disease entity and severity via trough level adaptations. Among its most frequent adverse effects are hypertension, nephrotoxicity, neurotoxicity, and electrolyte disturbances. Hypertrichosis and gingival hyperplasia are obvious and widely recognized adverse effects. CASE REPORT: We report on a 66-year-old woman who was treated with cyclosporine A for primary membranous nephropathy. During treatment with cyclosporine, she developed hirsutism and gingival hyperplasia. Later, she reported having impaired nasal breathing and dyspnea on mild physical exercise. Clinical, rhinoscopic, and radiological evaluations showed marked conchal hyperplasia as a potential cause of her symptoms. An extensive medical work-up did not show evidence of allergic, immunologic, or other drug adverse effects, suggesting cyclosporine-induced hyperplasia of the turbinates as a hypothetical causative factor. Dose reductions did not lead to resolution of symptoms but resulted in increasing proteinuria. Therefore, cyclosporine was stopped, and the patient was treated with rituximab. Thereafter, hirsutism and gingival and conchal hyperplasia gradually regressed over 2–4 months, showing complete resolution of conchal hyperplasia on computed-tomography follow-up after 6 months. CONCLUSIONS: Cyclosporine can not only result in gingival hyperplasia but also in hyperplasia of the turbinates leading to impaired nasal breathing and shortness of breath on exertion. An extensive search for many other known causes of conchal swelling is warranted to finally suggest an adverse effect of cyclosporine. Discontinuation of cyclosporine resulted in complete remission of conchal hyperplasia as well as other adverse effects. International Scientific Literature, Inc. 2022-05-21 /pmc/articles/PMC9131439/ /pubmed/35642551 http://dx.doi.org/10.12659/AJCR.935362 Text en © Am J Case Rep, 2022 https://creativecommons.org/licenses/by-nc-nd/4.0/This work is licensed under Creative Common Attribution-NonCommercial-NoDerivatives 4.0 International (CC BY-NC-ND 4.0 (https://creativecommons.org/licenses/by-nc-nd/4.0/) )
spellingShingle Articles
Tillmann, Frank-Peter
Harth, Ana
Özcan, Fedai
Jörres, Achim
Cyclosporine A-Induced Conchal Hyperplasia with Nasal Obstruction in a Patient with Membranous Nephropathy
title Cyclosporine A-Induced Conchal Hyperplasia with Nasal Obstruction in a Patient with Membranous Nephropathy
title_full Cyclosporine A-Induced Conchal Hyperplasia with Nasal Obstruction in a Patient with Membranous Nephropathy
title_fullStr Cyclosporine A-Induced Conchal Hyperplasia with Nasal Obstruction in a Patient with Membranous Nephropathy
title_full_unstemmed Cyclosporine A-Induced Conchal Hyperplasia with Nasal Obstruction in a Patient with Membranous Nephropathy
title_short Cyclosporine A-Induced Conchal Hyperplasia with Nasal Obstruction in a Patient with Membranous Nephropathy
title_sort cyclosporine a-induced conchal hyperplasia with nasal obstruction in a patient with membranous nephropathy
topic Articles
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9131439/
https://www.ncbi.nlm.nih.gov/pubmed/35642551
http://dx.doi.org/10.12659/AJCR.935362
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