A rare case of IgG4-related disease involving the uterus

Ohkubo, Hirotsugu; Miyazaki, Mikinori; Oguri, Tetsuya; Arakawa, Atsushi; Kobashi, Yoichiro

doi:10.1093/rheumatology/kev024

Cited by 11 publications

(7 citation statements)

References 6 publications

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“…To clarify the characteristics of IgG4-RD with pleural effusion, we conducted a review of the English-language literature by a MEDLINE search using the following keywords: “IgG4-related disease” AND “pleural effusion” OR “pleuritis.” Cases with unclear clinical data or unmatched clinical symptoms (e.g., without serum IgG4 or pleural effusion) were excluded. We ultimately analyzed 21 cases (including the present case) (Table 3) [[4], [5], [6], [7], [8], [9], [10], [11], [12], [13], [14], [15], [16], [17], [18], [19], [20], [21], [22]].…”

Section: Discussionmentioning

confidence: 99%

Long-term spontaneous remission with active surveillance in IgG4-related pleuritis: A case report and literature review

Makimoto

Ohashi

Taniguchi

et al. 2019

Respiratory Medicine Case Reports

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Pleural effusion is a relatively rare feature of IgG4-related disease (IgG4-RD). Here, we report a case of a 72-year-old woman who presented with pleural effusion. Although the pleural adenosine deaminase level was increased, surgical biopsy of the pleura and left inguinal lymph node indicated that the effusion was due to IgG4-RD. Active surveillance was initiated because serum IgG4 and pleural effusion naturally decreased and then completely disappeared. The patient has shown no recurrence for >4 years. This case suggests that pleural biopsy can be used to distinguish IgG4-RD from tuberculosis; moreover, some cases with pleural effusion could improve without treatment.

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Section: Discussionmentioning

confidence: 99%

Long-term spontaneous remission with active surveillance in IgG4-related pleuritis: A case report and literature review

Makimoto

Ohashi

Taniguchi

et al. 2019

Respiratory Medicine Case Reports

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“…More than 40 different organs have been reported to be involved in IgG4‐RD . As for the gynecological organ, a case of IgG4‐RD involving the uterus was recently published . Nevertheless, involvement of the ovary has never been reported in English literature.…”

Section: Introductionmentioning

confidence: 99%

Immunoglobulin G4‐related disease presenting as bilateral ovarian masses and mimicking advanced ovarian cancer

Maruyama

Sato

Taga

et al. 2015

J of Obstet and Gynaecol

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Immunoglobulin G4-related disease (IgG4-RD) is characterized by extensive infiltration of IgG4(+) plasma cells and fibrosis in various organs. However, the involvement of the ovary in IgG4-RD has never been reported. A 59-year-old woman presented with urinary retention. Magnetic resonance imaging and computed tomography revealed a huge multinodular pelvic mass and common iliac/para-aortic lymph node swelling. A laparotomy was performed under the suspicion of advanced ovarian cancer, and the pelvic mass was identified as ovary in origin. Histopathology of the excised tumor revealed massive lymphoplasmacytic infiltration (>90% were IgG4(+) plasma cells), storiform fibrosis, and obliterative phlebitis; thus leading to a diagnosis of IgG4-RD. We conclude that IgG4-RD can present as a bilateral ovarian mass along with lymphadenopathy, therefore mimicking ovarian cancer.

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“…That study noted that most of the patients (90%) started to show IgG4‐RD after 40 years old 8 . Gynecologists rarely encounter IgG4‐RD, and few cases were reported 4,9 . One case was a 68‐year‐old woman with persistent cough and shortness of breath 4 .…”

Section: Discussionmentioning

confidence: 96%

“…Its clinical features include lesions that present as swellings, nodules and wall thickenings throughout the body, 2 leading to various diseases, such as autoimmune pancreatitis, parotitis and retroperitoneal fibrosis, many of which are common among middle‐aged and elderly men 3 . Uterine mass formation in IgG4‐RD is uncommon 4 . The diagnostic criteria include the following: (i) clinically diffuse or localized enlargements, masses, nodules or hypertrophic lesions in a single or multiple organs; (ii) elevated serum IgG4 (>135 mg/dL) and (iii) the presence of two histological features: (a) massive lymphocyte and plasma cell infiltration with fibrosis; and (b) an increased number of IgG4‐positive plasma cells (IgG4+ : IgG+ ratio must be at least 40%, and the number of IgG4+ cells per high‐power field must be >10) 5,6 .…”

Section: Introductionmentioning

confidence: 99%

A uterine pseudotumor of immunoglobulin G4‐related disease

Senda

Ikeda

Tamauchi

et al. 2020

J of Obstet and Gynaecol

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Immunoglobulin G4‐related disease (IgG4‐RD) is characterized by systemic lesions presenting as swellings, nodules and wall thickenings. The first‐line therapy is glucocorticoids. This disease is common among middle‐aged and elderly men, and its uterine mass formation is uncommon. We encountered a 39‐year‐old nulliparous woman with an IgG4‐related uterine mass and enlarged pelvic lymph nodes. Complete remission was achieved by fertility‐sparing resection of the uterine mass without glucocorticoids. This case showed that IgG4‐RD can present as a uterine mass and we need to be careful of its diagnostic and therapeutic strategies because an IgG4‐related uterine mass is difficult to distinguish from sarcoma or malignant lymphoma. Our case also demonstrated that IgG4‐related lymphadenopathy could regress spontaneously without complete lymphadenectomy or glucocorticoids when IgG4‐RD does not affect vital organs. A pelvic mass with hyperproteinemia may be a hint to remind us of IgG4‐RD and may trigger a preoperative immunoglobulin test.

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A rare case of IgG4-related disease involving the uterus

Cited by 11 publications

References 6 publications

Long-term spontaneous remission with active surveillance in IgG4-related pleuritis: A case report and literature review

Long-term spontaneous remission with active surveillance in IgG4-related pleuritis: A case report and literature review

Immunoglobulin G4‐related disease presenting as bilateral ovarian masses and mimicking advanced ovarian cancer

A uterine pseudotumor of immunoglobulin G4‐related disease

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