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Idiopathic sclerosing inflammation presenting as sinusitis

Idiopathic sclerosing orbital inflammation is a rare finding that is poorly delineated, immune mediated, and causes severe symptoms and disability. It has been described affecting the orbit in addition to other sites within the head and neck, but has rarely been described presenting as sinusitis. A...

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Detalles Bibliográficos
Autores principales: Barham, Henry P., Dishop, Megan K., Prager, Jeremy D.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: OceanSide Publications, Inc. 2012
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC3548607/
https://www.ncbi.nlm.nih.gov/pubmed/23342288
http://dx.doi.org/10.2500/ar.2012.3.0031
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author Barham, Henry P.
Dishop, Megan K.
Prager, Jeremy D.
author_facet Barham, Henry P.
Dishop, Megan K.
Prager, Jeremy D.
author_sort Barham, Henry P.
collection PubMed
description Idiopathic sclerosing orbital inflammation is a rare finding that is poorly delineated, immune mediated, and causes severe symptoms and disability. It has been described affecting the orbit in addition to other sites within the head and neck, but has rarely been described presenting as sinusitis. A case report and literature review were performed. A 14-year-old girl with right-sided face and eye pain and pressure for >1 month presented 3 days after endoscopic sinus surgery for presumed acute sinusitis. She subsequently developed ipsilateral vision loss and hypesthesia of the infraorbital nerve. MRI revealed a mildly enhancing soft tissue intensity lesion extending from the right maxillary sinus into the pterygopalatine fossa and orbital apex through the inferior orbital fissure. Biopsy specimens of the lesion were consistent with a sclerosing inflammatory lesion. High-dose steroids led to rapid improvement in vision and pain; however, the patient was unable to tolerate steroid weaning because of recurrence of eye pain and headache. Repeat imaging showed progression of the lesion. Rheumatology was consulted and the patient's steroid therapy was altered and her medications were expanded to include azathioprine. The patient's symptoms improved and subsequent imaging showed a reduction in the size and extent of the lesion. Idiopathic sclerosing inflammation is characterized by primary, chronic, and immunologically mediated fibrosis. Patients typically have a poor response to corticosteroid treatment or radiotherapy. Immunosuppressive therapy in addition to corticosteroids is the recommended treatment.
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spelling pubmed-35486072013-01-22 Idiopathic sclerosing inflammation presenting as sinusitis Barham, Henry P. Dishop, Megan K. Prager, Jeremy D. Allergy Rhinol (Providence) Articles Idiopathic sclerosing orbital inflammation is a rare finding that is poorly delineated, immune mediated, and causes severe symptoms and disability. It has been described affecting the orbit in addition to other sites within the head and neck, but has rarely been described presenting as sinusitis. A case report and literature review were performed. A 14-year-old girl with right-sided face and eye pain and pressure for >1 month presented 3 days after endoscopic sinus surgery for presumed acute sinusitis. She subsequently developed ipsilateral vision loss and hypesthesia of the infraorbital nerve. MRI revealed a mildly enhancing soft tissue intensity lesion extending from the right maxillary sinus into the pterygopalatine fossa and orbital apex through the inferior orbital fissure. Biopsy specimens of the lesion were consistent with a sclerosing inflammatory lesion. High-dose steroids led to rapid improvement in vision and pain; however, the patient was unable to tolerate steroid weaning because of recurrence of eye pain and headache. Repeat imaging showed progression of the lesion. Rheumatology was consulted and the patient's steroid therapy was altered and her medications were expanded to include azathioprine. The patient's symptoms improved and subsequent imaging showed a reduction in the size and extent of the lesion. Idiopathic sclerosing inflammation is characterized by primary, chronic, and immunologically mediated fibrosis. Patients typically have a poor response to corticosteroid treatment or radiotherapy. Immunosuppressive therapy in addition to corticosteroids is the recommended treatment. OceanSide Publications, Inc. 2012 2012-09-18 /pmc/articles/PMC3548607/ /pubmed/23342288 http://dx.doi.org/10.2500/ar.2012.3.0031 Text en Copyright © 2012, OceanSide Publications, Inc., U.S.A. This publication is provided under the terms of the Creative Commons Public License ("CCPL" or "License"), in attribution 3.0 unported (Attribution Non-Commercial No Derivatives (CC BY-NC-ND)), further described at: http://creativecommons.org/licenses/by-nc-nd/3.0/legalcode. The work is protected by copyright and/or other applicable law. Any use of the work other then as authorized under this license or copyright law is prohibited.
spellingShingle Articles
Barham, Henry P.
Dishop, Megan K.
Prager, Jeremy D.
Idiopathic sclerosing inflammation presenting as sinusitis
title Idiopathic sclerosing inflammation presenting as sinusitis
title_full Idiopathic sclerosing inflammation presenting as sinusitis
title_fullStr Idiopathic sclerosing inflammation presenting as sinusitis
title_full_unstemmed Idiopathic sclerosing inflammation presenting as sinusitis
title_short Idiopathic sclerosing inflammation presenting as sinusitis
title_sort idiopathic sclerosing inflammation presenting as sinusitis
topic Articles
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC3548607/
https://www.ncbi.nlm.nih.gov/pubmed/23342288
http://dx.doi.org/10.2500/ar.2012.3.0031
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