Korean Journal of Radiology

The Korean Society of Radiology (대한영상의학회)
권호 표지
DOI QR코드

DOI QR Code

Clinical and Radiological Features of Diffuse Lacrimal Gland Enlargement: Comparisons among Various Etiologies in 91 Biopsy-Confirmed Patients

  • Sae Rom Chung (Department of Radiology and Research Institute of Radiology, Asan Medical Center, University of Ulsan College of Medicine) ;
  • Gye Jung Kim (Department of Ophthalmology, Asan Medical Center, University of Ulsan College of Medicine) ;
  • Young Jun Choi (Department of Radiology and Research Institute of Radiology, Asan Medical Center, University of Ulsan College of Medicine) ;
  • Kyung-Ja Cho (Department of Pathology, Asan Medical Center, University of Ulsan College of Medicine) ;
  • Chong Hyun Suh (Department of Radiology and Research Institute of Radiology, Asan Medical Center, University of Ulsan College of Medicine) ;
  • Soo Chin Kim (Department of Radiology and Research Institute of Radiology, Asan Medical Center, University of Ulsan College of Medicine) ;
  • Jung Hwan Baek (Department of Radiology and Research Institute of Radiology, Asan Medical Center, University of Ulsan College of Medicine) ;
  • Jeong Hyun Lee (Department of Radiology and Research Institute of Radiology, Asan Medical Center, University of Ulsan College of Medicine) ;
  • Min Kyu Yang (Department of Ophthalmology, Asan Medical Center, University of Ulsan College of Medicine) ;
  • Ho-Seok Sa (Department of Ophthalmology, Asan Medical Center, University of Ulsan College of Medicine)
  • Received : 2022.04.05
  • Accepted : 2022.07.25
  • Published : 2022.10.01
Download PDF
⟨ Previous Next ⟩

Abstract

Objective: To compare the clinical and radiological features of various etiologies of chronic diffuse lacrimal gland enlargement. Materials and Methods: We retrospectively reviewed 91 consecutive patients who underwent surgical biopsy for chronic diffuse lacrimal gland enlargement and were diagnosed with non-specific dacryoadenitis (DA) (n = 42), immunoglobulin G4-related dacryoadenitis (IgG4-RD) (n = 33), and lymphoma (n = 16). Data on patient demographics, clinical presentation, and CT imaging findings (n = 73) and MRI (n = 43) were collected. The following radiologic features of lacrimal gland enlargement were evaluated: size, unilaterality, wedge sign, angle with the orbital wall, heterogeneity, signal intensity, degree of enhancement, patterns of dynamic contrast-enhanced, and apparent diffusion coefficient value. Radiological features outside the lacrimal glands, such as extra-lacrimal orbital involvement and extra-orbital head and neck involvement, were also evaluated. The clinical and radiological findings were compared among the three diseases. Results: Compared to the DA and IgG4-RD groups, the lymphoma group was significantly older (mean 59.9 vs. 46.0 and 49.4 years, respectively; p = 0.001) and had a higher frequency of unilateral involvement (62.5% vs. 31.0% and 15.2%, respectively; p = 0.004). Compared to the IgG4-RD and lymphoma groups, the DA group had significantly smaller lacrimal glands (2.3 vs. 2.8 and 3.3 cm, respectively; p < 0.001) and a lower proportion of cases with a wedge sign (54.8% vs. 84.8% and 87.5%, respectively; p = 0.005). The IgG4-RD group showed more frequent involvement of the extra-orbital head and neck structures, including the infraorbital nerve (36.4%), paranasal sinus (72.7%), and salivary gland (58.6%) compared to the DA and lymphoma groups (4.8%-28.6%) (all p < 0.005). Conclusion: Patient age, unilaterality, lacrimal gland size, wedge sign, and extra-orbital head and neck involvement differed significantly different between lymphoma, DA, and IgG4-RD. Our results will be useful for the differential diagnosis and proper management of chronic lacrimal gland enlargement.

Keywords

References

  1. Teo L, Seah LL, Choo CT, Chee SP, Chee E, Looi A. A survey of the histopathology of lacrimal gland lesions in a tertiary referral centre. Orbit 2013;32:1-7 https://doi.org/10.3109/01676830.2012.736595
  2. Ahn C, Kang S, Sa HS. Clinicopathologic features of biopsied lacrimal gland masses in 95 Korean patients. Graefes Arch Clin Exp Ophthalmol 2019;257:1527-1533 https://doi.org/10.1007/s00417-019-04327-w
  3. Andrew NH, McNab AA, Selva D. Review of 268 lacrimal gland biopsies in an Australian cohort. Clin Exp Ophthalmol 2015;43:5-11 https://doi.org/10.1111/ceo.12371
  4. Priel A, Vishnevskia-Dai V, Hochma L, Gildener-Leapman J, Ben Simon GJ, Rosner M, et al. The epidemiological, clinical, and histopathological characteristics of lacrimal gland biopsies in a tertiary care center in Israel. Isr Med Assoc J 2018;20:104-108
  5. McNab AA, McKelvie P. IgG4-related ophthalmic disease. Part II: clinical aspects. Ophthalmic Plast Reconstr Surg 2015;31:167-178 https://doi.org/10.1097/IOP.0000000000000364
  6. Sa HS, Lee JH, Woo KI, Kim YD. IgG4-related disease in idiopathic sclerosing orbital inflammation. Br J Ophthalmol 2015;99:1493-1497 https://doi.org/10.1136/bjophthalmol-2014-305528
  7. Wallace ZS, Perugino C, Matza M, Deshpande V, Sharma A, Stone JH. Immunoglobulin G4-related disease. Clin Chest Med 2019;40:583-597 https://doi.org/10.1016/j.ccm.2019.05.005
  8. Lemaitre S, Esquerda GM, Guardiola AC, Agustin JT, Sanda N, Gonzalez-Candial M. Colon cancer and IgG4-related disease with orbital inflammation and bilateral optic perineuritis: a case report. Medicine (Baltimore) 2018;97:e12197
  9. Yamamoto M, Takahashi H, Tabeya T, Suzuki C, Naishiro Y, Ishigami K, et al. Risk of malignancies in IgG4-related disease. Mod Rheumatol 2012;22:414-418 https://doi.org/10.3109/s10165-011-0520-x
  10. Yu T, Wu Y, Liu J, Zhuang Y, Jin X, Wang L. The risk of malignancy in patients with IgG4-related disease: a systematic review and meta-analysis. Arthritis Res Ther 2022;24:14
  11. Taniguchi T, Kobayashi H, Nishikawa K, Iida E, Michigami Y, Morimoto E, et al. Diffusion-weighted magnetic resonance imaging in autoimmune pancreatitis. Jpn J Radiol 2009;27:138-142 https://doi.org/10.1007/s11604-008-0311-2
  12. Hiwatashi A, Yoshiura T, Togao O, Yamashita K, Kikuchi K, Fujita Y, et al. Diffusivity of intraorbital lymphoma vs. IgG4- related DISEASE: 3D turbo field echo with diffusion-sensitised driven-equilibrium preparation technique. Eur Radiol 2013;24:581-586 https://doi.org/10.1007/s00330-013-3058-9
  13. Yuan Y, Chu G, Gong T, Du L, Xie L, Yuan Q, et al. To explore MR imaging radiomics for the differentiation of orbital lymphoma and IgG4-related ophthalmic disease. Biomed Res Int 2021;2021:6668510
  14. Ro SR, Asbach P, Siebert E, Bertelmann E, Hamm B, Erb-Eigner K. Characterization of orbital masses by multiparametric MRI. Eur J Radiol 2016;85:324-336 https://doi.org/10.1016/j.ejrad.2015.11.041
  15. Haradome K, Haradome H, Usui Y, Ueda S, Kwee TC, Saito K, et al. Orbital lymphoproliferative disorders (OLPDs): value of MR imaging for differentiating orbital lymphoma from benign OPLDs. AJNR Am J Neuroradiol 2014;35:1976-1982 https://doi.org/10.3174/ajnr.A3986
  16. Xu XQ, Hu H, Su GY, Zhang L, Liu H, Hong XN, et al. Orbital indeterminate lesions in adults: combined magnetic resonance morphometry and histogram analysis of apparent diffusion coefficient maps for predicting malignancy. Acad Radiol 2016;23:200-208 https://doi.org/10.1016/j.acra.2015.10.015
  17. Klingenstein A, Garip-Kuebler A, Priglinger S, Hintschich C, Mueller-Lisse UG. Morphologic cross-sectional imaging features of IgG4-related orbitopathy in comparison to ocular adnexal lymphoma. Clin Ophthalmol 2021;15:1119-1127 https://doi.org/10.2147/OPTH.S299655
  18. Vandenbroucke JP, von Elm E, Altman DG, Gotzsche PC, Mulrow CD, Pocock SJ, et al. Strengthening the Reporting of Observational Studies in Epidemiology (STROBE): explanation and elaboration. Ann Intern Med 2007;147:W163-W194 https://doi.org/10.7326/0003-4819-147-8-200710160-00010-w1
  19. von Elm E, Altman DG, Egger M, Pocock SJ, Gotzsche PC, Vandenbroucke JP; STROBE Initiative. The strengthening the reporting of observational studies in epidemiology (STROBE) statement: guidelines for reporting observational studies. J Clin Epidemiol 2008;61:344-349 https://doi.org/10.1016/j.jclinepi.2007.11.008
  20. Goto H, Takahira M, Azumi A; Japanese Study Group for IgG4-Related Ophthalmic Disease. Diagnostic criteria for IgG4-related ophthalmic disease. Jpn J Ophthalmol 2015;59:1-7 https://doi.org/10.1007/s10384-014-0352-2
  21. Tamboli DA, Harris MA, Hogg JP, Realini T, Sivak-Callcott JA. Computed tomography dimensions of the lacrimal gland in normal Caucasian orbits. Ophthalmic Plast Reconstr Surg 2011;27:453-456 https://doi.org/10.1097/IOP.0b013e31821e9f5d
  22. Lorenzano D, Rose GE. The "wedge sign": an imaging sign for aggressive lacrimal gland disease. Ophthalmology 2017;124:1081-1083 https://doi.org/10.1016/j.ophtha.2017.03.004
  23. Choi YJ, Lee JH, Sung YS, Yoon RG, Park JE, Nam SY, et al. Value of dynamic contrast-enhanced MRI to detect local tumor recurrence in primary head and neck cancer patients. Medicine (Baltimore) 2016;95:e3698
  24. Go H, Kim JE, Kim YA, Chung HK, Khwarg SI, Kim CW, et al. Ocular adnexal IgG4-related disease: comparative analysis with mucosa-associated lymphoid tissue lymphoma and other chronic inflammatory conditions. Histopathology 2012;60:296-312 https://doi.org/10.1111/j.1365-2559.2011.04089.x
  25. Luemsamran P, Rootman J, White VA, Nassiri N, Heran MKS. The role of biopsy in lacrimal gland inflammation: a clinicopathologic study. Orbit 2017;36:411-418 https://doi.org/10.1080/01676830.2017.1352608
  26. Dagi Glass LR, Freitag SK. Orbital inflammation: corticosteroids first. Surv Ophthalmol 2016;61:670-673 https://doi.org/10.1016/j.survophthal.2016.01.005
  27. Borenstein SH, Gerstle T, Malkin D, Thorner P, Filler RM. The effects of prebiopsy corticosteroid treatment on the diagnosis of mediastinal lymphoma. J Pediatr Surg 2000;35:973-976 https://doi.org/10.1053/jpsu.2000.6945
  28. Kubota T, Moritani S. Orbital IgG4-related disease: clinical features and diagnosis. ISRN Rheumatol 2012;2012:412896
  29. Behzadi F, Suh CH, Jo VY, Shanmugam V, Morgan EA, Guenette JP. Imaging of IgG4-related disease in the head and neck: a systematic review, case series, and pathophysiology update. J Neuroradiol 2021;48:369-378 https://doi.org/10.1016/j.neurad.2021.01.006
  30. Tiegs-Heiden CA, Eckel LJ, Hunt CH, Diehn FE, Schwartz KM, Kallmes DF, et al. Immunoglobulin G4-related disease of the orbit: imaging features in 27 patients. AJNR Am J Neuroradiol 2014;35:1393-1397 https://doi.org/10.3174/ajnr.A3865
  31. Ohshima K, Sogabe Y, Sato Y. The usefulness of infraorbital nerve enlargement on MRI imaging in clinical diagnosis of IgG4-related orbital disease. Jpn J Ophthalmol 2012;56:380-382 https://doi.org/10.1007/s10384-012-0151-6
  32. Soussan JB, Deschamps R, Sadik JC, Savatovsky J, Deschamps L, Puttermann M, et al. Infraorbital nerve involvement on magnetic resonance imaging in European patients with IgG4-related ophthalmic disease: a specific sign. Eur Radiol 2017;27:1335-1343 https://doi.org/10.1007/s00330-016-4481-5
  33. Choi JW, Kim SY, Moon KC, Cho JY, Kim SH. Immunoglobulin G4-related sclerosing disease involving the urethra: case report. Korean J Radiol 2012;13:803-807 https://doi.org/10.3348/kjr.2012.13.6.803
  34. Lee JY, Cheng KL, Lee JH, Choi YJ, Kim HW, Sung YS, et al. Detection of local recurrence in patients with head and neck squamous cell carcinoma using voxel-based color maps of initial and final area under the curve values derived from DCEMRI. AJNR Am J Neuroradiol 2019;40:1392-1401 https://doi.org/10.3174/ajnr.A6130