IgG4-related Lung Pseudotumor and Pleural Inflammation with Autoimmune Hepatitis

Nagashima, Kazunori; Sano, Itsuki; Kobayashi, Tomoe; Eto, Kazunori; Nagai, Ken; Ninomiya, Ryusuke; Suzuki, Akira; Oohata, Yoshihiro; Konishi, Kouhei; Nakano, Tsuyoshi; Yamamoto, Fumiyasu

doi:10.2169/internalmedicine.9026-17

Cited by 10 publications

(5 citation statements)

References 26 publications

(38 reference statements)

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“…11 There are multiple reports on patients with underlying autoimmune disease such as autoimmune pancreatitis and Sjogren syndrome with hepatic IPT that responded to steroid therapy. [14][15][16] In our patient, it is possible that the decrease in the size of her liver lesion occurred in the setting of her higher dose of steroid therapy for her polymyalgia rheumatica.…”

Section: Discussionmentioning

confidence: 85%

Enlarging Liver Mass: Inflammatory Pseudotumor in a Patient With Polymyalgia Rheumatica

Kanagalingam

Dulymamode

Jafroodifar

et al. 2022

Journal of Investigative Medicine High Impact Case Reports

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Inflammatory pseudotumors of the liver are rare, non-neoplastic liver tumors. Due to the nonspecific clinical presentation, imaging features, and histopathological findings, they can mimic malignant tumors requiring invasive diagnostics. We present a case of a 61-year-old female patient with a history of type 2 diabetes mellitus, hypothyroidism, hyperlipidemia, and polymyalgia rheumatica who had initially presented with abdominal pain for 3 weeks. Further workup showed normal liver chemistries and tumor markers: AFP and CA 19-9. Magnetic resonance imaging (MRI) of the abdomen showed a segment 6 lesion measuring 4.1 × 4.0 × 3.7 cm. A liver biopsy then confirmed the diagnosis of an inflammatory pseudotumor of the liver with negative IgG4. On follow-up imaging, a rapid growth of this liver lesion was noted. Laparoscopy was done but did not show any distinct liver lesion. Follow-up imaging confirmed a decrease in the size of the mass. Interestingly, the patient had been on a higher dose of steroids for her polymyalgia rheumatic leading up to the follow-up imaging. This is the first case of an inflammatory pseudotumor of the liver in a patient with polymyalgia rheumatica. With this case, we would like to increase the awareness for inflammatory pseudotumors of the liver as a differential diagnosis of liver lesions in patients with underlying autoimmune disorders.

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Section: Discussionmentioning

confidence: 85%

Enlarging Liver Mass: Inflammatory Pseudotumor in a Patient With Polymyalgia Rheumatica

Kanagalingam

Dulymamode

Jafroodifar

et al. 2022

Journal of Investigative Medicine High Impact Case Reports

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“…The most common manifestation of IgG4-RD is type 1 autoimmune pancreatitis affecting 13% of patients [12] . In addition to salivary gland and pancreatic involvement, other associations of IgG4-RLD that have been reported, include autoimmune hepatitis, constrictive pericarditis, asthma, Sjogren syndrome, psoriasis, and lymphomas [13] , [14] , [15] .…”

Section: Discussionmentioning

confidence: 99%

IgG4-related lung disease mimicking lung cancer

Shafiq

Ghorab

Abuzakouk

et al. 2022

Radiology Case Reports

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“…Chung et al proposed that the degree of accumulation of IgG4-positive liver cells is associated with the serum IgG4 response in patients with IgG4-AIH [ 11 ]. Furthermore, synchronous or metachronous development of other IgG4-related disease is observed in most cases of IgG4-AIH [ 9 , 12 – 15 ]. This evidence suggests that IgG4-AIH is a hepatic manifestation of IgG4-related disease and, while sharing pathological findings with AIH, is not a subtype of classic AIH [ 9 ].…”

Section: Discussionmentioning

confidence: 99%

Immunoglobulin G4-associated autoimmune hepatitis with peripheral blood eosinophilia: a case report

et al. 2020

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Background Immunoglobulin G4 (IgG4) associated autoimmune hepatitis (AIH) has been recognized as a type of autoimmune disease that responds to corticosteroid. The diagnosis is based on elevation of the serum IgG4 level, abundance of IgG4 enhanced plasma cell infiltration in the portal region of the liver, and satisfaction of the criteria for “definite AIH” under the revised International Autoimmune Hepatitis Group (IAIHG) scoring system. However, the clinical course of the disease is unclear. Case presentation A 65-year-old man with jaundice and peripheral blood eosinophilia. His IAIHG and simplified score was compatible with definite AIH and his IgG4 level was elevated. Magnetic resonance imaging did not reveal abnormalities in the hepatobiliary system or pancreas. A liver biopsy revealed interface hepatitis with IgG4 positive plasma cell infiltration in the portal region, without evidence of bile duct injury. He responded to 4-week period of induction prednisolone therapy and had no recurring symptoms under maintenance therapy of 5 mg prednisolone during the 3-year follow up. Conclusions This was a rare case that demonstrated an association between IgG4 associated AIH and the presence of peripheral blood eosinophilia.

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IgG4-related Lung Pseudotumor and Pleural Inflammation with Autoimmune Hepatitis

Cited by 10 publications

References 26 publications

Enlarging Liver Mass: Inflammatory Pseudotumor in a Patient With Polymyalgia Rheumatica

Enlarging Liver Mass: Inflammatory Pseudotumor in a Patient With Polymyalgia Rheumatica

IgG4-related lung disease mimicking lung cancer

Immunoglobulin G4-associated autoimmune hepatitis with peripheral blood eosinophilia: a case report

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