دورية أكاديمية
A rare manifestation of IgG4-related disease and secondary hypereosinophilic syndrome: A case report.
| العنوان: | A rare manifestation of IgG4-related disease and secondary hypereosinophilic syndrome: A case report. |
|---|---|
| المؤلفون: | Takeuchi M; Kidney Unit, National Hospital Organization Fukuokahigashi Medical Center, Fukuoka, Japan., Shojima M; Kidney Unit, National Hospital Organization Fukuokahigashi Medical Center, Fukuoka, Japan., Matsueda S; Kidney Unit, National Hospital Organization Fukuokahigashi Medical Center, Fukuoka, Japan., Nagae H; Kidney Unit, National Hospital Organization Fukuokahigashi Medical Center, Fukuoka, Japan., Kuroiwa M; Division of Hematology, National Hospital Organization Fukuokahigashi Medical Center, Fukuoka, Japan., Fujita A; Division of Pathology, National Hospital Organization Fukuokahigashi Medical Center, Fukuoka, Japan., Kawano M; Innovative Clinical Research Center, Department of Rheumatology, Kanazawa University Hospital, Ishikawa, Japan., Inoue D; Department of Radiology, Kanazawa University Hospital, Ishikawa, Japan., Komori T; Department of Radiology, Kanazawa University Hospital, Ishikawa, Japan., Takeuchi M; Department of Pathology, Kurume University, Fukuoka, Japan., Ooshima K; Department of Pathology, Kurume University, Fukuoka, Japan., Kuroki Y; Kidney Unit, National Hospital Organization Fukuokahigashi Medical Center, Fukuoka, Japan., Katafuchi R; Kidney Unit, National Hospital Organization Fukuokahigashi Medical Center, Fukuoka, Japan.; Kidney Unit, Medical Corporation Houshikai Kano Hospital, Fukuoka, Japan. |
| المصدر: | Modern rheumatology case reports [Mod Rheumatol Case Rep] 2024 Jul 08; Vol. 8 (2), pp. 329-338. |
| نوع المنشور: | Case Reports; Journal Article |
| اللغة: | English |
| بيانات الدورية: | Publisher: Oxford University Press Country of Publication: England NLM ID: 101761026 Publication Model: Print Cited Medium: Internet ISSN: 2472-5625 (Electronic) Linking ISSN: 24725625 NLM ISO Abbreviation: Mod Rheumatol Case Rep Subsets: MEDLINE |
| أسماء مطبوعة: | Publication: 2021- : Oxford : Oxford University Press Original Publication: Abingdon, Oxfordshire : Taylor & Francis, [2017]- |
| مواضيع طبية MeSH: | Immunoglobulin G4-Related Disease*/diagnosis , Immunoglobulin G4-Related Disease*/complications , Hypereosinophilic Syndrome*/diagnosis , Hypereosinophilic Syndrome*/complications , Hypereosinophilic Syndrome*/etiology , Hypereosinophilic Syndrome*/drug therapy , Prednisolone*/therapeutic use , Prednisolone*/administration & dosage, Humans ; Female ; Aged ; Immunoglobulin G/blood ; Immunoglobulin G/immunology ; Nephritis, Interstitial/diagnosis ; Nephritis, Interstitial/etiology ; Nephritis, Interstitial/drug therapy ; Nephritis, Interstitial/immunology ; Treatment Outcome ; Biopsy |
| مستخلص: | We report a case of IgG4-related disease (IgG4-RD) with marked eosinophilia. A 79-year-old woman was admitted due to diarrhoea and weight loss. Cervical lymphadenopathy, bilateral submandibular glands swelling, anaemia (Hb8.5 g/dl), hypereosinophilia (9750/μl), elevated serum creatinine (1.57 mg/dl), pancreatic amylase (191 IU/l), and IgG4 (3380 mg/dl) were found. Diffusion-weighted image on magnetic resonance imaging showed high-intensity signals inside both the pancreas and the kidneys. The echogram of submandibular glands revealed cobblestone pattern. Kidney biopsy revealed acute tubulointerstitial nephritis. Biopsies of lip, gastrointestinal tract, and bone marrow showed infiltration of lymphoplasmacytic cells and IgG4-positive plasma cells (30-67/HPF). Gastrointestinal and bone marrow biopsies also showed eosinophilic infiltration. Adrenal insufficiency, rheumatic disease, tuberculosis, parasite infection, drug-induced eosinophilia, and eosinophilic leukaemia were all ruled out. We started treatment with 40 mg of prednisolone (PSL) and her general condition rapidly improved. The eosinophil count, serum IgG4, and serum creatinine decreased. We gradually tapered PSL and maintained 5 mg/day. During the 5 years of treatment, she had no recurrence of the symptom. According to the 2019 American College of Rheumatology/European League Against Rheumatism classification criteria for IgG4-RD, eosinophils >3000/μl is one of the exclusion criteria. If we comply with this criterion, the diagnosis of IgG4-RD should be avoided. However, our case fit the diagnostic criteria of type I autoimmune pancreatitis, IgG4-related sialadenitis, and global diagnosis of IgG4-RD. We finally diagnosed our case as IgG4-RD with secondary hypereosinophilic syndrome. This case suggests that IgG4-RD with eosinophils >3000/μl does exist in the real world. (© Japan College of Rheumatology 2024. Published by Oxford University Press.) |
| فهرسة مساهمة: | Keywords: IgG4; IgG4-related disease; eosinophilia; hypereosinophilic syndrome |
| المشرفين على المادة: | 9PHQ9Y1OLM (Prednisolone) 0 (Immunoglobulin G) |
| SCR Disease Name: | Acute Tubulointerstitial Nephritis |
| تواريخ الأحداث: | Date Created: 20240226 Date Completed: 20240708 Latest Revision: 20240708 |
| رمز التحديث: | 20240709 |
| DOI: | 10.1093/mrcr/rxae009 |
| PMID: | 38407323 |
| قاعدة البيانات: | MEDLINE |
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