Review Article
Clinical Characteristics of 76 Patients with IgG4-Related Hypophysitis: A Systematic Literature Review
Table 2
Summary of the reported cases of IgG4-related hypophysitis.
| Case no | Age | Sex | IgG4 (mg/dl) | Pituitary function | MRI | Number of other affected organs | Biopsy | Therapy | Response | Authors | AH | DI | Stalk | Pituitary | Pituitary | Others | Symptom | MRI |
| 1 | 66 | F | 485 | + | − | − | + | 4 | − | — | GC | + | + | van der Vliet [3] | 2 | 71 | M | 405 | + | + | − | + | 3 | − | Submandibular gland, retroperitoneum | GC | + | + | Tanabe et al. [8] | 3 | 70 | M | 2220 | + | − | + | − | 2 | − | Submandibular gland | GC | + | + | Yamamoto et al. [9] | 4 | 77 | M | 720 | + | − | − | + | 2 | + | Pancreas, cholecyst | Surgery + GC | nd | nd | Wong et al. [5] | 5 | 55 | M | 1860 | + | + | + | − | 2 | − | Paranasal sinus | GC | + | + | Isaka et al. [10] | 6 | 62 | M | 292 | + | + | + | − | 3 | − | Lung | GC | + | + | Tsuboi et al. [11] | 7 | 74 | F | nd | + | + | + | + | 1 | + | — | Surgery + GC | + | + | Osawa et al. [12] | 8 | 70 | M | 924 | + | + | + | − | 3 | − | Liver, lung, parotid gland | GC | + | + | Hori et al. [14] | 9 | 68 | M | 159 | nd | + | + | + | 2 | − | — | GC | + | + | Haraguchi et al. [13] | 10 | 74 | F | 170 | + | + | + | + | 1 | − | Lymph node | GC | + | + | Haraguchi et al. [13] | 11 | 55 | M | 127 | + | + | + | + | 3 | − | Lacrimal gland, kidney | nd | nd | nd | Patel and Szostek [16] | 12 | 71 | M | 240 | − | + | + | + | 1 | − | Pleural | GC | nd | nd | Nagai et al. [15] | 13 | 75 | M | nd | + | + | + | + | 1 | + | Paranasal sinus | GC | + | + | Leporati et al. [6] | 14 | 47 | M | 94.9 | nd | nd | − | + | 3 | + | Lung, lymph node | GC | + | + | Nishina et al. [17] | 15 | 53 | F | Normal | + | − | − | + | 0 | + | — | Surgery + GC | + | + | Kanoke et al. [20] | 16 | 60 | F | Normal | + | − | + | + | 0 | + | — | Surgery + GC | + | + | Kanoke et al. [20] | 17 | 66 | M | nd | + | − | + | − | 4 | + | Mediastinum | Surgery + GC | + | + | Hsing et al. [19] | 18 | 55 | M | 1010 | − | − | + | + | 0 | + | — | GC | + | + | Hattori et al. [18] | 19 | 53 | M | 24.5 | + | + | + | − | 3 | − | Lung | GC | nd | + | Bando et al. [21] | 20 | 76 | M | 513 | + | + | + | + | 0 | − | — | GC | nd | + | Bando et al. [21] | 21 | 58 | F | 405 | nd | + | + | + | 3 | − | Lung, stomach | GC | nd | + | Bando et al. [21] | 22 | 68 | F | 16.9 | + | − | + | − | 0 | + | — | GC | nd | + | Bando et al. [21] | 23 | 53 | F | 16.9 | + | + | + | + | 1 | + | — | GC | nd | + | Bando et al. [21] | 24 | 67 | F | 82.5 | + | + | + | + | 0 | + | — | GC | nd | + | Bando et al. [21] | 25 | 76 | M | 2100 | + | + | + | + | 2 | + | Pancreas | GC | + | + | Iseda et al. [23] | 26 | 33 | F | nd | + | + | + | − | 0 | + | — | Surgery + GC | + | + | Khong et al. [24] | 27 | 70 | M | 300 | + | + | + | + | 3 | + | Retroperitoneum | GC | + | + | Ohkubo et al. [25] | 28 | 40 | M | 413 | + | + | + | + | 1 | + | Lacrimal gland | GC + azathioprine | + | + | Caputo et al. [22] | 29 | 37 | F | nd | + | + | + | + | 0 | + | — | GC | + | + | Sosa et al. [26] | 30 | 25 | F | nd | + | − | + | + | 0 | + | — | GC | + | + | Sosa et al. [26] | 31 | 71 | M | nd | − | + | + | + | 0 | + | — | nd | nd | nd | Imber et al. [28] | 32 | 87 | M | 285 | + | + | + | + | 0 | − | — | GC | + | + | Nakasone et al. [30] | 33 | 32 | F | nd | + | + | − | + | 0 | + | — | GC | | | Tauziede-Espariat et al. [32] | 34 | 38 | F | Normal | + | + | − | + | 0 | + | — | GC | + | + | Tauziede-Espariat et al. [32] | 35 | 43 | M | 79.2 | + | − | + | + | 1 | + | — | GC | + | − | Ngaosuwan et al. [31] | 36 | 67 | M | 240 | + | + | + | − | 2 | − | Liver | GC + azathioprine | + | + | Joshi et al. [29] | 37 | 62 | M | 307 | − | + | + | + | 2 | − | Kidney | GC | nd | nd | Joshi et al. [29] | 38 | 72 | M | 853 | + | + | + | − | 1 | − | Kidney | GC | + | + | Harano et al. [27] | 39 | 38 | F | 377 | + | + | − | + | 2 | + | Orbita | GC | + | + | Alexandraki et al. [33] | 40 | 55 | M | 800 | + | + | + | + | 0 | + | — | GC | + | + | Bhagwat et al. [34] | 41 | 16 | F | nd | − | + | + | + | 0 | + | — | GC | nd | nd | Decker et al. [35] | 42 | 54 | M | nd | + | + | + | + | 2 | − | Kidney | GC | + | + | Patel et al. [36] | 43 | 76 | M | 1030 | + | − | + | + | 0 | + | — | GC | + | + | Anno et al. [37] | 44 | 57 | M | 83.9 | + | + | + | + | 5 | + | — | GC + azathioprine | + | nd | Gu et al. [38] | 45 | 29 | F | 135 | + | + | − | + | 0 | + | — | Surgery + GC + MMF | + | + | Hadjigeorgiou et al. [39] | 46 | 56 | M | 570.7 | + | + | + | + | 2 | − | — | Surgery + GC | + | + | Huang et al. [40] | 47 | 70 | M | 425 | + | + | + | − | 4 | − | Lip | No | — | — | Kawasaki et al. [41] | 48 | 51 | F | nd | + | + | + | + | 0 | + | — | Surgery + GC | + | + | Lee et al. [42] | 49 | 85 | M | 713 | nd | + | + | − | 1 | − | Kidney | GC | + | + | Matsuda et al. [43] | 50 | 36 | F | nd | + | + | + | + | 0 | + | — | Surgery + GC | + | nd | Rotondo et al. [44] | 51 | 49 | M | nd | + | − | + | + | 0 | + | — | GC | + | + | Rotondo et al. [44] | 52 | 56 | M | 298 | + | − | + | + | 3 | − | — | GC | + | + | Yatabe et al. [45] | 53 | 14 | F | 61 | − | − | + | + | 0 | + | — | GC + rituximab | − | + | Bullock et al. [46] | 54 | 25 | F | 55.7 | + | + | + | + | 0 | + | — | GC | + | + | Koide et al. [48] | 55 | 23 | F | nd | + | + | − | + | 1 | + | Retroperitoneum | Surgery + GC | nd | nd | Guarda et al. [47] | 56 | 50 | M | 426 | + | + | − | + | 7 | + | — | GC | − | − | Liu et al. [49] | 57 | 16 | F | 43.7 | + | + | + | − | 2 | − | Submandibular gland | GC | − | − | Liu et al. [49] | 58 | 57 | F | 2250 | − | + | + | − | 3 | − | Lacrimal gland | GC | + | + | Liu et al. [49] | 59 | 36 | M | 2470 | − | + | + | − | 4 | − | Lacrimal gland | GC | + | + | Liu et al. [49] | 60 | 44 | M | 1910 | − | + | + | − | 3 | − | Pancreas | GC | + | + | Liu et al. [49] | 61 | 46 | M | 327 | + | + | + | − | 1 | − | Lymph node | GC | + | + | Liu et al. [49] | 62 | 51 | M | 1980 | − | + | + | − | 3 | − | Submandibular gland | GC | + | + | Liu et al. [49] | 63 | 58 | M | 5410 | + | + | + | − | 4 | − | — | GC | + | + | Liu et al. [49] | 64 | 64 | M | 199 | + | + | + | + | 0 | − | — | GC | + | + | Liu et al. [49] | 65 | 61 | M | 4680 | + | + | + | + | 2 | − | Paranasal sinus | GC | + | + | Liu et al. [49] | 66 | 58 | F | 58 | + | + | + | + | 0 | + | — | GC | + | + | Murphy et al. [50] | 67 | 36 | M | Normal | + | + | + | + | 0 | + | — | GC | + | + | Sosa et al. [51] | 68 | 67 | F | 865 | nd | + | + | − | 6 | + | Bladder | GC | + | + | Xue et al. [53] | 69 | 50 | M | 13.1 | nd | nd | − | + | 0 | + | — | Surgery | + | + | Tang et al. [52] | 70 | 24 | F | Normal | + | + | + | + | 0 | + | — | Surgery + GC | + | + | Yuen et al. [54] | 71 | 24 | F | Normal | nd | nd | − | + | 0 | + | — | GC | + | nd | Yuen et al. [54] | 72 | 71 | M | Normal | + | + | − | + | 0 | + | — | GC | + | − | Yuen et al. [54] | 73 | 78 | M | 802 | + | nd | − | + | 4 | − | — | Surgery + GC | + | + | Kanie et al. [56] | 74 | 43 | F | 462 | + | + | + | + | 2 | − | Orbita | GC | + | + | Yoshida et al. [58] | 75 | 28 | F | 880 | − | − | − | + | 0 | − | — | Methotrexate + rituximab | + | nd | Goulam-Houssein et al. [55] | 76 | 65 | M | 221 | + | nd | − | + | 0 | + | — | Surgery + GC + rituximab | + | + | Vauchot et al. [57] |
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F, female; M, male; nd, not described; AH, anterior hypophysitis; DI, diabetes insipidus; GC, glucocorticoid; MMF, mycophenolate mofetil.
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