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Immunoglobulin G4-related disease with features of Mikulicz's disease and autoimmune pancreatitis which firstly presented as asymptomatic lymphadenopathy: a case report.

Wu Y, Xu ZR, Zhou WJ, Yang YM - Chin. Med. J. (2015)

View Article: PubMed Central - PubMed

Affiliation: Department of Geriatrics, First Affiliated Hospital, College of Medicine, Zhejiang University, Hangzhou, Zhejiang 310013, China.

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To the Editor: Mikulicz's disease (MD), also known as benign lymphoepithelial lesion, refers to idiopathic, bilateral, painless, and symmetrical swelling of the lacrimal, parotid, and submandibular glands... The complications of MD include autoimmune pancreatitis, retroperitoneal fibrosis, tubulointerstitial nephritis, autoimmune hypophysitis, and Riedel's thyroiditis, all of which show elevated serum immunoglobulin G4 (IgG4) levels and prominent infiltration of IgG4-positive plasmacytes... An 82-year-old man was admitted to our hospital on April 16, 2012 because of anorexia, anergy, emaciation, and painless lumps in postaurem... Five years ago, he found a painless lump (about 2.3 cm × 1.2 cm) both in his left and right postaurem and the left one was surgically excised after 1 year... Subsequent positron emission tomography and computed tomography (PET-CT) scan images showed abnormal 18-fluorodeoxyglucose (FDG) uptake in the bilateral submandibular glands and parotid glands... The maximum standardized uptake value (SUV) of the submandibular glands was 11.16 and of the parotid glands was 10.86... These clinical, laboratory, and imaging findings indicated the possibility of IgG4-RD involving the lymph nodes, submandibular glands, and pancreas, according to the comprehensive diagnostic criteria for IgG4-RD... Treatment guidelines for IgG4-RD have not been developed... Corticosteroid therapy has been regarded as an effective treatment... Himi et al., initiated treatment with prednisolone at 30–40 mg/day against MD without encountering organ failure, and suggested that it is necessary to continue administering at 5–10 mg/day or to combine it with an immunosuppressant... Taking into account the advanced age of our patient, we decreased the dose of corticosteroid, and he was successfully treated... However, the optimal initial doses of steroids, tapering procedures, and maintenance doses are still controversial... Further investigation is necessary to establish the most effective therapy... Here, we have described a typical case of IgG4-RD, previously mis-diagnosed... This time he was successfully treated.

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Positron emission tomography and computed tomography (PET-CT) images. (a) Abnormal 18-fluorodeoxyglucose uptake in bilateral submandibular glands and parotid glands. The maximum standardized uptake value (SUV) of the submandibular glands was 11.16, and of the parotid glands was 10.86; (b) The tail of the pancreas was mildly swollen. The maximum SUV was about 4.48, and about 5.06 after a delayed scan.
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Figure 1: Positron emission tomography and computed tomography (PET-CT) images. (a) Abnormal 18-fluorodeoxyglucose uptake in bilateral submandibular glands and parotid glands. The maximum standardized uptake value (SUV) of the submandibular glands was 11.16, and of the parotid glands was 10.86; (b) The tail of the pancreas was mildly swollen. The maximum SUV was about 4.48, and about 5.06 after a delayed scan.

Mentions: Ultrasonography showed multiple nodes in the thyroid gland and lymph node swelling in both parotid regions. Abdominal magnetic resonance imaging (MRI) scans showed changes in appearance and signals in the tail of the pancreas, consisted with manifestations of autoimmune pancreatitis. Gastroscopy revealed chronic superficial atrophic gastritis. Nasendoscopy showed nasal mucosal swelling. Subsequent positron emission tomography and computed tomography (PET-CT) scan images showed abnormal 18-fluorodeoxyglucose (FDG) uptake in the bilateral submandibular glands and parotid glands. The maximum standardized uptake value (SUV) of the submandibular glands was 11.16 and of the parotid glands was 10.86. Swelling of the mediastinal lymph node also could be seen, with a maximum diameter of the flow path of about 1.66 cm and a maximum SUV of about 5.21. The tail of the pancreas was mildly swollen. Diffuse radioactive uptake of the tail of the pancreas was increased, and the maximum SUV was about 4.48. Delayed scan radioactive uptake still showed an increase, with a maximum SUV of about 5.06 [Figure 1].


Immunoglobulin G4-related disease with features of Mikulicz's disease and autoimmune pancreatitis which firstly presented as asymptomatic lymphadenopathy: a case report.

Wu Y, Xu ZR, Zhou WJ, Yang YM - Chin. Med. J. (2015)

Positron emission tomography and computed tomography (PET-CT) images. (a) Abnormal 18-fluorodeoxyglucose uptake in bilateral submandibular glands and parotid glands. The maximum standardized uptake value (SUV) of the submandibular glands was 11.16, and of the parotid glands was 10.86; (b) The tail of the pancreas was mildly swollen. The maximum SUV was about 4.48, and about 5.06 after a delayed scan.
© Copyright Policy - open-access
Related In: Results  -  Collection

License
Show All Figures
getmorefigures.php?uid=PMC4834790&req=5

Figure 1: Positron emission tomography and computed tomography (PET-CT) images. (a) Abnormal 18-fluorodeoxyglucose uptake in bilateral submandibular glands and parotid glands. The maximum standardized uptake value (SUV) of the submandibular glands was 11.16, and of the parotid glands was 10.86; (b) The tail of the pancreas was mildly swollen. The maximum SUV was about 4.48, and about 5.06 after a delayed scan.
Mentions: Ultrasonography showed multiple nodes in the thyroid gland and lymph node swelling in both parotid regions. Abdominal magnetic resonance imaging (MRI) scans showed changes in appearance and signals in the tail of the pancreas, consisted with manifestations of autoimmune pancreatitis. Gastroscopy revealed chronic superficial atrophic gastritis. Nasendoscopy showed nasal mucosal swelling. Subsequent positron emission tomography and computed tomography (PET-CT) scan images showed abnormal 18-fluorodeoxyglucose (FDG) uptake in the bilateral submandibular glands and parotid glands. The maximum standardized uptake value (SUV) of the submandibular glands was 11.16 and of the parotid glands was 10.86. Swelling of the mediastinal lymph node also could be seen, with a maximum diameter of the flow path of about 1.66 cm and a maximum SUV of about 5.21. The tail of the pancreas was mildly swollen. Diffuse radioactive uptake of the tail of the pancreas was increased, and the maximum SUV was about 4.48. Delayed scan radioactive uptake still showed an increase, with a maximum SUV of about 5.06 [Figure 1].

View Article: PubMed Central - PubMed

Affiliation: Department of Geriatrics, First Affiliated Hospital, College of Medicine, Zhejiang University, Hangzhou, Zhejiang 310013, China.

AUTOMATICALLY GENERATED EXCERPT
Please rate it.

To the Editor: Mikulicz's disease (MD), also known as benign lymphoepithelial lesion, refers to idiopathic, bilateral, painless, and symmetrical swelling of the lacrimal, parotid, and submandibular glands... The complications of MD include autoimmune pancreatitis, retroperitoneal fibrosis, tubulointerstitial nephritis, autoimmune hypophysitis, and Riedel's thyroiditis, all of which show elevated serum immunoglobulin G4 (IgG4) levels and prominent infiltration of IgG4-positive plasmacytes... An 82-year-old man was admitted to our hospital on April 16, 2012 because of anorexia, anergy, emaciation, and painless lumps in postaurem... Five years ago, he found a painless lump (about 2.3 cm × 1.2 cm) both in his left and right postaurem and the left one was surgically excised after 1 year... Subsequent positron emission tomography and computed tomography (PET-CT) scan images showed abnormal 18-fluorodeoxyglucose (FDG) uptake in the bilateral submandibular glands and parotid glands... The maximum standardized uptake value (SUV) of the submandibular glands was 11.16 and of the parotid glands was 10.86... These clinical, laboratory, and imaging findings indicated the possibility of IgG4-RD involving the lymph nodes, submandibular glands, and pancreas, according to the comprehensive diagnostic criteria for IgG4-RD... Treatment guidelines for IgG4-RD have not been developed... Corticosteroid therapy has been regarded as an effective treatment... Himi et al., initiated treatment with prednisolone at 30–40 mg/day against MD without encountering organ failure, and suggested that it is necessary to continue administering at 5–10 mg/day or to combine it with an immunosuppressant... Taking into account the advanced age of our patient, we decreased the dose of corticosteroid, and he was successfully treated... However, the optimal initial doses of steroids, tapering procedures, and maintenance doses are still controversial... Further investigation is necessary to establish the most effective therapy... Here, we have described a typical case of IgG4-RD, previously mis-diagnosed... This time he was successfully treated.

Show MeSH