Ocular involvement (Conjunctivitis/Scleritis) as a form of presentation in ER-IgG4 in pediatric age.
Keywords:
Ocular involvement in ER-IgG4 – pediatrics – eye – conjunctivitis – IgG4
Abstract
Abstract Introduction: The disease related to IgG4 (ER-IgG4) in pediatric age has different manifestations than in adults and there are only case reports and some systematic reviews in the literature. Furthermore, ocular involvement in this population (EOR-IgG4), although common in children, continues to be poorly defined due to the rarity of the disease and the lack of suspicion of it, which leads to a late diagnosis. Clinical case: 12-year-old girl with EOR-IgG4 with a diagnostic delay of 4 years. She presented conjunctivitis, scleritis, ptosis, edema and ocular pain, unilaterally, without extraophthalmic involvement. Increased IgG4 and serum IgG4/IgG ratio, involvement of the central belly of the left internal rectus muscle and involvement of the eyelid on images. Lymphoplasmacytic infiltrate, with immunohistochemistry compatible with ER-IgG4. Conclusion: It is important that pediatric doctors take this disease into account and know its different forms of presentation in this population.References
BIBLIOGRAFÍA
1- Hui Lu, Fei Teng, Panpan Zhang y col. Differences in clinical characteristics of IgG4-related disease across age groups: a prospective study of 737 patients. Rheumatology 2021;60:2635–2646. doi:10.1093/rheumatology/keaa651.
2- Roubini G. Smerla, Dimitra Rontogianni, George E. Fragoulis. Ocular manifestations of IgG4-related disease in children. More common than anticipated? Review of the literature and case report. Clinical Rheumatology. 2017. https://doi.org/10.1007/s10067-017-3934-9.
3- Umehara H, Okazaki K, Kawa S, et al.; Research Program for Intractable Disease by the Ministry of Health, Labor and Welfare (MHLW) Japan. The 2020 revised comprehensive diagnostic (RCD) criteria for IgG4-RD. Mod Rheumatol 2021. May;31(3):529-533.
4- Wallace ZS, Naden RP, Chari S, Choi H, Della-Torre E, Dicaire JF, et al. The 2019 American College of Rheumatology/European League Against Rheumatism Classification Criteria for IgG4-Related Disease. Ann Rheum. Dis 2020 Jan;79(1):77-87.
5- Satoshi Haraa, Misaki Yoshidab, Hajime Sanada y col. Pediatric IgG4-related disease: a descriptive review. EXPERT REVIEW OF CLINICAL IMMUNOLOGY. 2023. https://doi.org/10.1080/1744666X.2023.2274358.
6- Griepentrog GJ, Vickers RW, Karesh JW, et al. A clinicopathologic case study of two patients with pediatric orbital IgG4-related disease. Orbit.2013;32:389–391.
7- Kalapesi FB, Garrott HM, Moldovan C, et al. IgG4 orbital inflammation in a 5-year-old child presenting as an orbital mass. Orbit. 2013;32:137–140.
8- Sane M, Chelnis J, Kozielski R, et al. Immunoglobulin G4-related sclerosing disease with orbital inflammation in a 12-year-old girl. JAAPOS. 2013;17:548–550.
9- Susan Ruyu Qi, Mélanie Hébert, Eunice You y col. Conjunctival Infiltration in a Child as a Rare Manifestation of IgG4-Related Disease. Cornea 2022;41:496–498.
10- Ang Li, Thomas P. Plesec, Linda Mileti y col. Isolated Conjunctival Inflammation as a Manifestation of IgG4-Related Disease. Cornea 2018;0:1–3.
11- Jing Li, Rui Liu, Tingting Ren y col. A case of IgG4-positive ligneous conjunctivitis mistaken for a conjunctival mass. Diagnostic Pathology (2023) 18:77. https://doi.org/10.1186/s13000-023-01366-0.
12- Wei-Yu Chiang, Ting-Ting Liu, Wan-Ting Huang y col. Co-existing ligneous conjunctivitis and IgG4-related disease. Indian J Ophthalmol. 2016 Jul; 64(7): 532–534. doi: 10.4103/0301-4738.190154.
13- Faiz Karim, Jan Loeffen, Wichor Bramer y col. IgG4-related disease: a systematic review of this unrecognized disease in pediatrics. Pediatr Rheumatol Online J. 2016; 14: 18. doi: 10.1186/s12969-016-0079-3.
14- B. de Sainte Marie, M. Ebbo, A. Grados y col. A descriptive study of IgG4-related disease in children and young adults. Autoimmunity Reviews. Volume 21, Issue 4, April 2022, 103035. https://doi.org/10.1016/j.autrev.2022.103035.
15- Ümmüşen Kaya Akca, Erdal Atalay, Müşerref Kasap Cüceoğlu y col. IgG4‑related disease in pediatric patients: a single‑center experience. Rheumatology International. 2021. https://doi.org/10.1007/s00296-021-04885-5.
1- Hui Lu, Fei Teng, Panpan Zhang y col. Differences in clinical characteristics of IgG4-related disease across age groups: a prospective study of 737 patients. Rheumatology 2021;60:2635–2646. doi:10.1093/rheumatology/keaa651.
2- Roubini G. Smerla, Dimitra Rontogianni, George E. Fragoulis. Ocular manifestations of IgG4-related disease in children. More common than anticipated? Review of the literature and case report. Clinical Rheumatology. 2017. https://doi.org/10.1007/s10067-017-3934-9.
3- Umehara H, Okazaki K, Kawa S, et al.; Research Program for Intractable Disease by the Ministry of Health, Labor and Welfare (MHLW) Japan. The 2020 revised comprehensive diagnostic (RCD) criteria for IgG4-RD. Mod Rheumatol 2021. May;31(3):529-533.
4- Wallace ZS, Naden RP, Chari S, Choi H, Della-Torre E, Dicaire JF, et al. The 2019 American College of Rheumatology/European League Against Rheumatism Classification Criteria for IgG4-Related Disease. Ann Rheum. Dis 2020 Jan;79(1):77-87.
5- Satoshi Haraa, Misaki Yoshidab, Hajime Sanada y col. Pediatric IgG4-related disease: a descriptive review. EXPERT REVIEW OF CLINICAL IMMUNOLOGY. 2023. https://doi.org/10.1080/1744666X.2023.2274358.
6- Griepentrog GJ, Vickers RW, Karesh JW, et al. A clinicopathologic case study of two patients with pediatric orbital IgG4-related disease. Orbit.2013;32:389–391.
7- Kalapesi FB, Garrott HM, Moldovan C, et al. IgG4 orbital inflammation in a 5-year-old child presenting as an orbital mass. Orbit. 2013;32:137–140.
8- Sane M, Chelnis J, Kozielski R, et al. Immunoglobulin G4-related sclerosing disease with orbital inflammation in a 12-year-old girl. JAAPOS. 2013;17:548–550.
9- Susan Ruyu Qi, Mélanie Hébert, Eunice You y col. Conjunctival Infiltration in a Child as a Rare Manifestation of IgG4-Related Disease. Cornea 2022;41:496–498.
10- Ang Li, Thomas P. Plesec, Linda Mileti y col. Isolated Conjunctival Inflammation as a Manifestation of IgG4-Related Disease. Cornea 2018;0:1–3.
11- Jing Li, Rui Liu, Tingting Ren y col. A case of IgG4-positive ligneous conjunctivitis mistaken for a conjunctival mass. Diagnostic Pathology (2023) 18:77. https://doi.org/10.1186/s13000-023-01366-0.
12- Wei-Yu Chiang, Ting-Ting Liu, Wan-Ting Huang y col. Co-existing ligneous conjunctivitis and IgG4-related disease. Indian J Ophthalmol. 2016 Jul; 64(7): 532–534. doi: 10.4103/0301-4738.190154.
13- Faiz Karim, Jan Loeffen, Wichor Bramer y col. IgG4-related disease: a systematic review of this unrecognized disease in pediatrics. Pediatr Rheumatol Online J. 2016; 14: 18. doi: 10.1186/s12969-016-0079-3.
14- B. de Sainte Marie, M. Ebbo, A. Grados y col. A descriptive study of IgG4-related disease in children and young adults. Autoimmunity Reviews. Volume 21, Issue 4, April 2022, 103035. https://doi.org/10.1016/j.autrev.2022.103035.
15- Ümmüşen Kaya Akca, Erdal Atalay, Müşerref Kasap Cüceoğlu y col. IgG4‑related disease in pediatric patients: a single‑center experience. Rheumatology International. 2021. https://doi.org/10.1007/s00296-021-04885-5.
Published
2024-08-07
How to Cite
1.
Zunino LV. Ocular involvement (Conjunctivitis/Scleritis) as a form of presentation in ER-IgG4 in pediatric age. Rev. Argent. Reumatol. [Internet]. 2024Aug.7 [cited 2024Sep.16];35(2). Available from: https://ojs.reumatologia.org.ar/index.php/revistaSAR/article/view/799
Section
Clinical Case
