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Published in: Graefe's Archive for Clinical and Experimental Ophthalmology 3/2014

01-03-2014 | Inflammatory Disorders

Clinical features of IgG4-related dacryoadenitis

Authors: Satomi Koizumi, Terumi Kamisawa, Sawako Kuruma, Taku Tabata, Susumu Iwasaki, Kazuro Chiba, Keigo Setoguchi, Shinichiro Horiguchi, Noriko Ozaki

Published in: Graefe's Archive for Clinical and Experimental Ophthalmology | Issue 3/2014

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Abstract

Background

To elucidate the clinical characteristics of IgG4-related dacryoadenitis.

Methods

Clinical features, laboratory findings, radiological findings, associated diseases, treatment, and prognosis were prospectively examined in 12 patients (seven men, five women; mean age, 60.9 ± 15.1 years) with IgG4-related dacryoadenitis.

Results

In addition to eyelid swelling, other ophthalmologic symptoms were observed in seven patients, including diplopia (n = 4), ptosis (n = 2), visual field disturbance (n = 2), eye pain (n = 2), decrease of visual acuity (n = 2), eye-movement disturbance (n = 1), dry eye (n = 1), corneal ulcer (n = 1), and epiphora (n = 1). Swelling of the lacrimal glands was bilateral in half of the patients. Other IgG4-related diseases were present in nine patients, including sialadenitis (n = 5), autoimmune pancreatitis (n = 4), retroperitoneal fibrosis (n = 2), and lymphadenopathy (n = 8). Serum IgG4 levels were significantly higher in patients with other IgG4-related disease (1070 ± 813 mg/dl) than in those without (197 ± 59 mg/dl, p = 0.017). Allergic histories and elevated serum IgE levels were each detected in six patients. Eight patients showed inflammatory extension beyond the lacrimal gland, such as thickened rectus muscle (n = 6), inflammation of the optic nerve (n = 2), and retrobulbar inflammation (n = 3). Steroid therapy was effective in seven patients, but dacryoadenitis relapsed in two patients with markedly higher serum IgG4 levels and autoimmune pancreatitis.

Conclusions

IgG4-related dacryoadenitis showed various ophthalmologic symptoms due to extensive inflammation beyond the lacrimal gland, frequent association with other IgG4-related disease or allergic phenomena, and steroid responsiveness.
Literature
1.
go back to reference Khosroshahi A, Stone JH (2011) A clinical overview of IgG4-related systemic disease. Curr Opin Rheumatol 23:57–66PubMedCrossRef Khosroshahi A, Stone JH (2011) A clinical overview of IgG4-related systemic disease. Curr Opin Rheumatol 23:57–66PubMedCrossRef
2.
go back to reference Stones JH, Khosroshahi A, Deshpande V, Chan JK, Heathcote JG, Aalberse R, Azumi A, Bloch DB, Brugge WR, Carruthers MN, Cheuk W, Cornell L, Castillo CF, Ferry JA, Forcione D, Klöppel G, Hamilos DL, Kamisawa T, Kasashima S, Kawa S, Kawano M, Masaki Y, Notohara K, Okazaki K, Ryu JK, Saeki T, Sahani D, Sato Y, Smyrk T, Stone JR, Takahira M, Umehara H, Webster G, Yamamoto M, Yi E, Yoshino T, Zamboni G, Zen Y, Chari S (2012) Recommendations for the nomenclature of IgG4-related disease and its individual organ system manifestations. Arthritis Rheum 64:3061–3067CrossRef Stones JH, Khosroshahi A, Deshpande V, Chan JK, Heathcote JG, Aalberse R, Azumi A, Bloch DB, Brugge WR, Carruthers MN, Cheuk W, Cornell L, Castillo CF, Ferry JA, Forcione D, Klöppel G, Hamilos DL, Kamisawa T, Kasashima S, Kawa S, Kawano M, Masaki Y, Notohara K, Okazaki K, Ryu JK, Saeki T, Sahani D, Sato Y, Smyrk T, Stone JR, Takahira M, Umehara H, Webster G, Yamamoto M, Yi E, Yoshino T, Zamboni G, Zen Y, Chari S (2012) Recommendations for the nomenclature of IgG4-related disease and its individual organ system manifestations. Arthritis Rheum 64:3061–3067CrossRef
3.
go back to reference 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), 2011. Mod Rheumatol 22:21–30PubMedCrossRef 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), 2011. Mod Rheumatol 22:21–30PubMedCrossRef
4.
go back to reference Kamisawa T, Funata N, Hayashi Y, Eishi Y, Koike M, Tsuruta K, Okamoto A, Egawa N, Nakajima H (2003) A new clinicopathological entity of IgG4-related autoimmune disease. J Gastroenterol 38:982–984PubMedCrossRef Kamisawa T, Funata N, Hayashi Y, Eishi Y, Koike M, Tsuruta K, Okamoto A, Egawa N, Nakajima H (2003) A new clinicopathological entity of IgG4-related autoimmune disease. J Gastroenterol 38:982–984PubMedCrossRef
5.
go back to reference Takuma K, Kamisawa T, Anjiki H, Egawa N, Igarashi Y (2010) Metachronous extrapancreatic lesions in autoimmune pancreatitis. Intern Med 49:529–533PubMedCrossRef Takuma K, Kamisawa T, Anjiki H, Egawa N, Igarashi Y (2010) Metachronous extrapancreatic lesions in autoimmune pancreatitis. Intern Med 49:529–533PubMedCrossRef
6.
go back to reference Hamano H, Arakura N, Muraki T, Ozaki Y, Kiyosawa K, Kawa S (2006) Prevalence and distribution of extrapancreatic lesions complicating autoimmune pancreatitis. J Gastroenterol 41:1197–1205PubMedCrossRef Hamano H, Arakura N, Muraki T, Ozaki Y, Kiyosawa K, Kawa S (2006) Prevalence and distribution of extrapancreatic lesions complicating autoimmune pancreatitis. J Gastroenterol 41:1197–1205PubMedCrossRef
7.
go back to reference Mikulicz JH (1892) Uber eine eigenartige symmetrische Erkrankung der Tranen- und Mundspeicheldrusen. Beitr Chir Fortsch Gewidmet Theodor Billroth:610–630 Mikulicz JH (1892) Uber eine eigenartige symmetrische Erkrankung der Tranen- und Mundspeicheldrusen. Beitr Chir Fortsch Gewidmet Theodor Billroth:610–630
8.
go back to reference Yamamoto M, Takahashi H, Ohara M, Suzuki C, Naishiro Y, Yamamoto H, Shinomura Y, Imai K (2006) A new conceptualization for Mikulicz’s disease as an IgG4-related plasmacytic disease. Mod Rheumatol 16:335–340PubMedCentralPubMedCrossRef Yamamoto M, Takahashi H, Ohara M, Suzuki C, Naishiro Y, Yamamoto H, Shinomura Y, Imai K (2006) A new conceptualization for Mikulicz’s disease as an IgG4-related plasmacytic disease. Mod Rheumatol 16:335–340PubMedCentralPubMedCrossRef
9.
go back to reference Sato Y, Ohshima K, Ichimura K, Sato M, Yamadori I, Tanaka T, Takata K, Morito T, Kondo E, Yoshino T (2008) Ocular adnexal IgG4-related disease has uniform clinicopathology. Pathol Int 58:465–470PubMedCrossRef Sato Y, Ohshima K, Ichimura K, Sato M, Yamadori I, Tanaka T, Takata K, Morito T, Kondo E, Yoshino T (2008) Ocular adnexal IgG4-related disease has uniform clinicopathology. Pathol Int 58:465–470PubMedCrossRef
10.
go back to reference Kubota T, Moritani S, Katayama M, Terasaki H (2010) Ocular adnexal IgG4-related lymphoplasmacytic infiltrative disorder. Arch Ophthalmol 128:577–584PubMedCrossRef Kubota T, Moritani S, Katayama M, Terasaki H (2010) Ocular adnexal IgG4-related lymphoplasmacytic infiltrative disorder. Arch Ophthalmol 128:577–584PubMedCrossRef
11.
go back to reference Takahira M, Ozawa Y, Kawano M, Zen Y, Hamaoka S, Yamada K, Sugiyama K (2012) Clinical aspects of IgG4-related orbital inflammation in a case series of ocular adnexal lymphoproliferative disorders. Int J Rheumatol 2012:635473PubMedCentralPubMedCrossRef Takahira M, Ozawa Y, Kawano M, Zen Y, Hamaoka S, Yamada K, Sugiyama K (2012) Clinical aspects of IgG4-related orbital inflammation in a case series of ocular adnexal lymphoproliferative disorders. Int J Rheumatol 2012:635473PubMedCentralPubMedCrossRef
12.
go back to reference Plaza JA, Garrity JA, Dogan A, Ananthamurthy A, Witzig TE, Salomao DR (2011) Orbital inflammation with IgG4-positive plasma cells. Manifestation of IgG4 systemic disease. Arch Ophthalmol 129:421–428PubMedCrossRef Plaza JA, Garrity JA, Dogan A, Ananthamurthy A, Witzig TE, Salomao DR (2011) Orbital inflammation with IgG4-positive plasma cells. Manifestation of IgG4 systemic disease. Arch Ophthalmol 129:421–428PubMedCrossRef
13.
go back to reference Cheuk W, Yuen HKL, Chan JKC (2007) Chronic sclerosing dacryoadenitis: part of the spectrum of IgG4-related sclerosing disease? Am J Surg Pathol 31:643–645PubMedCrossRef Cheuk W, Yuen HKL, Chan JKC (2007) Chronic sclerosing dacryoadenitis: part of the spectrum of IgG4-related sclerosing disease? Am J Surg Pathol 31:643–645PubMedCrossRef
14.
go back to reference Lee LY, Chen TC, Kuo TT (2011) Simultaneous occurrence of IgG4-related chronic sclerosing dacryoadenitis and chronic sialadenitis in a patient also with involvement of lymph node and Warthin’s tumor. Int J Surg Pathol 19:369–372PubMedCrossRef Lee LY, Chen TC, Kuo TT (2011) Simultaneous occurrence of IgG4-related chronic sclerosing dacryoadenitis and chronic sialadenitis in a patient also with involvement of lymph node and Warthin’s tumor. Int J Surg Pathol 19:369–372PubMedCrossRef
15.
go back to reference Jakobiec FA, Stacy RC, Mehta M, Fay A (2010) IgG4-positive dacryoadenitis and Kuttner submandibular sclerosing inflammatory tumor. Arch Ophthalmol 128:942–944PubMedCrossRef Jakobiec FA, Stacy RC, Mehta M, Fay A (2010) IgG4-positive dacryoadenitis and Kuttner submandibular sclerosing inflammatory tumor. Arch Ophthalmol 128:942–944PubMedCrossRef
16.
go back to reference Okazaki K, Kawa S, Kamisawa T, Ito T, Inui K, Irie H, Irisawa A, Kubo K, Notohara K, Hasebe O, Fujinaga Y, Ohara H, Tanaka S, Nishino T, Nishimori I, Nishiyama T, Suda K, Shiratori K, Shimosegawa T, Tanaka M (2009) Japanese clinical guidelines for autoimmune pancreatitis. Pancreas 38:849–866PubMedCrossRef Okazaki K, Kawa S, Kamisawa T, Ito T, Inui K, Irie H, Irisawa A, Kubo K, Notohara K, Hasebe O, Fujinaga Y, Ohara H, Tanaka S, Nishino T, Nishimori I, Nishiyama T, Suda K, Shiratori K, Shimosegawa T, Tanaka M (2009) Japanese clinical guidelines for autoimmune pancreatitis. Pancreas 38:849–866PubMedCrossRef
17.
go back to reference Kamisawa T, Takuma K, Egawa N, Tsuruta K, Sasaki T (2010) Autoimmune pancreatitis and IgG4-related sclerosing disease. Nat Rev Gastroenterol Hepatol 7:401–409PubMedCrossRef Kamisawa T, Takuma K, Egawa N, Tsuruta K, Sasaki T (2010) Autoimmune pancreatitis and IgG4-related sclerosing disease. Nat Rev Gastroenterol Hepatol 7:401–409PubMedCrossRef
18.
go back to reference Kamisawa T, Imai M, Egawa N, Okamoto T (2008) Serum IgG4 levels and extrapancreatic lesions in autoimmune pancreatitis. Eur J Gastroenterol Hepatol 20:1167–1170PubMedCrossRef Kamisawa T, Imai M, Egawa N, Okamoto T (2008) Serum IgG4 levels and extrapancreatic lesions in autoimmune pancreatitis. Eur J Gastroenterol Hepatol 20:1167–1170PubMedCrossRef
19.
go back to reference Kuruma S, Kamisawa T, Tabata T, Hara S, Fujiwara T, Kuwata G, Egarashira H, Koizumi K, Setoguchi K, Fujiwara J, Arakawa T, Momma K, Mitsuhashi T, Sasaki T (2013) Clinical characteristics of autoimmune pancreatitis with or without Mikulicz’s disease and Mikulicz’s disease alone. Gut Liver 7(1):96–99PubMedCentralPubMedCrossRef Kuruma S, Kamisawa T, Tabata T, Hara S, Fujiwara T, Kuwata G, Egarashira H, Koizumi K, Setoguchi K, Fujiwara J, Arakawa T, Momma K, Mitsuhashi T, Sasaki T (2013) Clinical characteristics of autoimmune pancreatitis with or without Mikulicz’s disease and Mikulicz’s disease alone. Gut Liver 7(1):96–99PubMedCentralPubMedCrossRef
20.
go back to reference Kamisawa T, Anjiki H, Egawa N, Kubota N (2009) Allergic manifestations in autoimmune pancreatitis. Eur J Gastroenterol Hepatol 21:1136–1139PubMedCrossRef Kamisawa T, Anjiki H, Egawa N, Kubota N (2009) Allergic manifestations in autoimmune pancreatitis. Eur J Gastroenterol Hepatol 21:1136–1139PubMedCrossRef
21.
go back to reference Kamisawa T, ShimosegawaT OK, Nishino T, Watanabe H, Kanno A, Okumura F, Nishikawa T, Kobayashi K, Ichiya T, Takatori H, Yamakita K, Kubota K, Hamano H, Okamura K, Hirano K, Ito T, Ko SB, Omata M (2009) Standard steroid therapy for autoimmune pancreatitis. Gut 58:1504–1507PubMedCrossRef Kamisawa T, ShimosegawaT OK, Nishino T, Watanabe H, Kanno A, Okumura F, Nishikawa T, Kobayashi K, Ichiya T, Takatori H, Yamakita K, Kubota K, Hamano H, Okamura K, Hirano K, Ito T, Ko SB, Omata M (2009) Standard steroid therapy for autoimmune pancreatitis. Gut 58:1504–1507PubMedCrossRef
Metadata
Title
Clinical features of IgG4-related dacryoadenitis
Authors
Satomi Koizumi
Terumi Kamisawa
Sawako Kuruma
Taku Tabata
Susumu Iwasaki
Kazuro Chiba
Keigo Setoguchi
Shinichiro Horiguchi
Noriko Ozaki
Publication date
01-03-2014
Publisher
Springer Berlin Heidelberg
Published in
Graefe's Archive for Clinical and Experimental Ophthalmology / Issue 3/2014
Print ISSN: 0721-832X
Electronic ISSN: 1435-702X
DOI
https://doi.org/10.1007/s00417-013-2541-y

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