IgG4-related disease of the paratestis in a patient with Wells syndrome: a case report

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• 作者：Takashi Karashima (1)
  Yoshinori Taniguchi (2)
  Tsutomu Shimamoto (1)
  Tomoya Nao (1)
  Hiroshi Nishikawa (1)
  Satoshi Fukata (1)
  Masayuki Kamada (1)
  Keiji Inoue (1)
  Kentaro Oko (3)
  Hideki Nakajima (3)
  Shigetoshi Sano (3)
  Manabu Matsumoto (4)
  Naoto Kuroda (5)
  Yoshihiro Kamei (6)
  Taro Shuin (1)
  
  1. Department of Urology ; Kochi Medical School ; Kohasu ; Oko ; Nankoku ; Kochi ; 783-8505 ; Japan
  2. Department of Endocrinology ; Metabolism and Nephrology ; Kochi Medical School ; Nankoku ; 783-8505 ; Japan
  3. Department of Dermatology ; Kochi Medical School ; Nankoku ; 783-8505 ; Japan
  4. Laboratory of Diagnostic Pathology ; Kochi Medical School Hospital ; Nankoku ; 783-8505 ; Japan
  5. Department of Diagnostic Pathology ; Kochi Red Cross Hospital ; Kochi ; 780-0062 ; Japan
  6. Kamei Private Clinic ; Kochi ; 780-0085 ; Japan
  
• 关键词：IgG4 ; related disease ; Paratestis ; Pseudotumor ; Wells syndrome
• 刊名：Diagnostic Pathology
• 出版年：2014
• 出版时间：December 2014
• 年：2014
• 卷：9
• 期：1
• 全文大小：1,075 KB
• 参考文献：1. Umehara, H, Okazaki, K, Masaki, Y, Kawano, M, Yamamoto, M, Saeki, T, Matsui, S, Sumida, T, Mimori, T, Tanaka, Y, Tsubota, K, Yoshino, T, Kawa, S, Suzuki, R, Takegami, T, Tomosugi, N, Kurose, N, Ishigaki, Y, Azumi, A, Kojima, M, Nakamura, S, Inoue, D (2012) A novel clinical entity, IgG4-related disease (IgG4RD): general concept and details. Mod Rheumatol 22: pp. 1-14 CrossRef
  2. B枚sm眉ller, H, Weyhern, CH, Adam, P, Alibegovic, V, Mikuz, G, Fend, F (2011) Paratesticular fibrous pseudotumor鈥揳n IgG4-related disorder?. Virchows Arch 458: pp. 109-113 CrossRef
  3. Hart, PA, Moyer, AM, Yi, ES, Hogan, MC, Pearson, RK, Chari, ST (2012) IgG4-related paratesticular pseudotumor in a patient with autoimmune pancreatitis and retroperitoneal fibrosis: an extrapancreatic manifestation of IgG4-related disease. Hum Pathol 43: pp. 2084-2087 CrossRef
  4. Migita, K, Miyashita, T, Mizuno, A, Jiuchi, Y, Ito, M, Matsuo, M, Izumi, Y, Takeoka, A, Nishino, A, Hayashi, M (2014) IgG4-related epididymo-orchitis associated with bladder cancer: possible involvement of BAFF/BAFF-R interaction in IgG4-related urogenital disease. Mod Rheumato 24: pp. 188-194 CrossRef
  5. Dieckmann, KP, Struss, WJ, Frey, U, Nahler-Wildenhain, M (2013) Paratesticular fibrous pseudotumor in young males presenting with histological features of IgG4-related disease: two case reports. J Med Case Rep 7: pp. 225 CrossRef
  6. Buy Wenniger, LM, Scheltema, JM, Verheij, J, Beuers, U (2013) Testicular inflammation as a new manifestation of IgG4-associated disease. Urology 82: pp. e15-e16 CrossRef
  7. Haddad, F, Helm, TN (2014) Wells syndrome. Cutis 93: pp. 38-39
  8. Umehara, H, Okazaki, K, Masaki, Y, Kawano, M, Yamamoto, M, Saeki, T, Matsui, S, Yoshino, T, Nakamura, S, Kawa, S, Hamano, H, Kamisawa, T, Shimosegawa, T, Shimatsu, A, Nakamura, S, Ito, T, Notohara, K, Sumida, T, Tanaka, Y, Mimori, T, Chiba, T, Mishima, M, Hibi, T, Tsubouchi, H, Inui, K, Ohara, H (2012) Comprehensive diagnostic criteria for IgG4-related disease (IgG4-RD). Mod Rheumatol 22: pp. 21-30 CrossRef
  9. Dangle, PP, Wang, WP, Pohar, KS (2008) Inflammatory myofibroblastic tumor of epididymis: a case report and review of literature. World J Surg Oncol 6: pp. 119 CrossRef
  10. Nagao, Y, Yamanaka, H, Harada, H (2012) A patient with hypereosinophilic syndrome that manifested with acquired hemophilia and elevated IgG4: a case report. J Med Case Rep 6: pp. 63 CrossRef
  11. Sato, Y, Notohara, K, Kojima, M, Takata, K, Masaki, Y, Yoshino, T (2010) IgG4-related disease: historical overview and pathology of hematological disorders. Pathol Int 60: pp. 247-258 CrossRef
  12. Espa帽a, A, Sanz, ML, Sola, J, Gil, P (1999) Wells鈥?syndrome (eosinophilic cellulitis): correlation between clinical activity, eosinophil levels, eosinophil cation protein and interleukin-5. Br J Dermatol 140: pp. 127-130 CrossRef
  
• 刊物主题：Pathology;
• 出版者：BioMed Central
• ISSN：1746-1596

文摘

Background We report a case of a 33-year-old man who presented with immunoglobulin (Ig)G4-related disease (IgG4-RD) forming a pseudotumor in the left paratesticular region during oral administration of corticosteroid for Wells syndrome, which involves cellulitis with eosinophilia. Case presentation The patient was introduced to our institution from a private hospital with a 3-month history of asymptomatic left scrotal mass. A 5-cm diameter nodule was palpable in the left scrotum. Tumor lesion in the left paratestis involving the epididymis and spermatic cord was observed on computed tomography and magnetic resonance imaging. Blood testing showed no abnormalities other than a minimal increase in C-reactive protein levels. Urine examination likewise revealed no significant findings. Left radical orchidectomy was performed under a diagnosis of left paratesticular neoplasm suspected as malignant tumor. The tumor was pathologically identified as IgG4-RD of the left paratestis involving the epididymis and spermatic cord. Conclusions We present a first description of IgG4-RD in a patient with Wells syndrome and the ninth case of IgG4-RD in a scrotal organ, and discuss this very rare entity with reference to the literature. Virtual Slides The virtual slide(s) for this article can be found here: http://www.diagnosticpathology.diagnomx.eu/vs/13000_2014_225