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Spontaneous regression of immunoglobulin G4-related dacryoadenitis and multiple organ involvement: A case report

Immunoglobulin G4 (IgG4)-related dacryoadenitis is rarely resolved spontaneously without steroids. Here, we report a case of IgG4-related dacryoadenitis and extra-ophthalmic lesions with spontaneous regression. METHODS: This is a clinical case report. A 56-years-old man had a 1-year and 7-month-old...

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Autores principales: Ito, Mayari, Vaidya, Aric, Kakizaki, Hirohiko, Takahashi, Yasuhiro
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Lippincott Williams & Wilkins 2023
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9829289/
https://www.ncbi.nlm.nih.gov/pubmed/36607859
http://dx.doi.org/10.1097/MD.0000000000032618
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author Ito, Mayari
Vaidya, Aric
Kakizaki, Hirohiko
Takahashi, Yasuhiro
author_facet Ito, Mayari
Vaidya, Aric
Kakizaki, Hirohiko
Takahashi, Yasuhiro
author_sort Ito, Mayari
collection PubMed
description Immunoglobulin G4 (IgG4)-related dacryoadenitis is rarely resolved spontaneously without steroids. Here, we report a case of IgG4-related dacryoadenitis and extra-ophthalmic lesions with spontaneous regression. METHODS: This is a clinical case report. A 56-years-old man had a 1-year and 7-month-old histories of neck and eyelid swelling, respectively. On the first examination, the lacrimal and submandibular glands were palpable bilaterally. Computed tomographic images showed enlargement of the lacrimal gland on both sides, right pulmonary hilar lymph node, and pancreas, and thickening of the abdominal aortic wall. Blood tests demonstrated elevated serum IgG4 level and positive hepatitis B surface antibody. Pathological examination of the biopsied lacrimal gland specimens revealed marked IgG4-positive plasma cell infiltration. RESULTS: The patient was monitored carefully without steroid administration. Serum IgG4 level had gradually decreased during follow-up period and reached the normal range 3 years after the biopsy. At 4-year follow-up, the lacrimal and submandibular glands were not palpable on either side. Computed tomographic images demonstrated no enlargement of the lacrimal gland, submandibular gland, or lymph nodes, and improvement of the enlarged pancreas and thickened abdominal aortic wall. CONCLUSION: Our case indicates that careful observation can be an option in selected cases with risks of steroid treatment or silent clinical course.
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spelling pubmed-98292892023-01-24 Spontaneous regression of immunoglobulin G4-related dacryoadenitis and multiple organ involvement: A case report Ito, Mayari Vaidya, Aric Kakizaki, Hirohiko Takahashi, Yasuhiro Medicine (Baltimore) 5800 Immunoglobulin G4 (IgG4)-related dacryoadenitis is rarely resolved spontaneously without steroids. Here, we report a case of IgG4-related dacryoadenitis and extra-ophthalmic lesions with spontaneous regression. METHODS: This is a clinical case report. A 56-years-old man had a 1-year and 7-month-old histories of neck and eyelid swelling, respectively. On the first examination, the lacrimal and submandibular glands were palpable bilaterally. Computed tomographic images showed enlargement of the lacrimal gland on both sides, right pulmonary hilar lymph node, and pancreas, and thickening of the abdominal aortic wall. Blood tests demonstrated elevated serum IgG4 level and positive hepatitis B surface antibody. Pathological examination of the biopsied lacrimal gland specimens revealed marked IgG4-positive plasma cell infiltration. RESULTS: The patient was monitored carefully without steroid administration. Serum IgG4 level had gradually decreased during follow-up period and reached the normal range 3 years after the biopsy. At 4-year follow-up, the lacrimal and submandibular glands were not palpable on either side. Computed tomographic images demonstrated no enlargement of the lacrimal gland, submandibular gland, or lymph nodes, and improvement of the enlarged pancreas and thickened abdominal aortic wall. CONCLUSION: Our case indicates that careful observation can be an option in selected cases with risks of steroid treatment or silent clinical course. Lippincott Williams & Wilkins 2023-01-06 /pmc/articles/PMC9829289/ /pubmed/36607859 http://dx.doi.org/10.1097/MD.0000000000032618 Text en Copyright © 2022 the Author(s). Published by Wolters Kluwer Health, Inc. https://creativecommons.org/licenses/by/4.0/This is an open access article distributed under the Creative Commons Attribution License 4.0 (CCBY) (https://creativecommons.org/licenses/by/4.0/) , which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.
spellingShingle 5800
Ito, Mayari
Vaidya, Aric
Kakizaki, Hirohiko
Takahashi, Yasuhiro
Spontaneous regression of immunoglobulin G4-related dacryoadenitis and multiple organ involvement: A case report
title Spontaneous regression of immunoglobulin G4-related dacryoadenitis and multiple organ involvement: A case report
title_full Spontaneous regression of immunoglobulin G4-related dacryoadenitis and multiple organ involvement: A case report
title_fullStr Spontaneous regression of immunoglobulin G4-related dacryoadenitis and multiple organ involvement: A case report
title_full_unstemmed Spontaneous regression of immunoglobulin G4-related dacryoadenitis and multiple organ involvement: A case report
title_short Spontaneous regression of immunoglobulin G4-related dacryoadenitis and multiple organ involvement: A case report
title_sort spontaneous regression of immunoglobulin g4-related dacryoadenitis and multiple organ involvement: a case report
topic 5800
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9829289/
https://www.ncbi.nlm.nih.gov/pubmed/36607859
http://dx.doi.org/10.1097/MD.0000000000032618
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