CASE REPORT
 
KEYWORDS
TOPICS
ABSTRACT
Introduction:
The report discusses the management of ocular surface complications in the late phase of toxic epidermal necrolysis, with an emphasis on strategies to mitigate these debilitating consequences.

Case Report:
A 75-year-old male presented with severe ocular complications, including visual acuity loss, pain, and tearing in the right eye (RE), stemming from chronic-phase Toxic Epidermal Necrolysis (TEN) triggered by allopurinol two years previously. Initial examination revealed cicatricial entropion in both eyes (BE), with the right eye (RE) exhibiting conjunctival cicatrization, symblepharon, corneal neovascularization, and a 3 mm corneal perforation. The patient underwent a combination of surgical and medical interventions, including fornix reconstruction, customized mini-keratoplasty, amniotic membrane transplantation, and partial tarsorrhaphy. Post-operative treatment included topical dexamethasone, tobramycin, cyclosporine, artificial tears, and oral prednisone. Despite ongoing treatment, including the addition of platelet-rich plasma, the right eye’s best-corrected visual acuity (BCVA) remained at light perception, with persistent corneal haziness, neovascularization, progressive conjunctival cicatrization, and keratinization.

Conclusions:
This case highlights the significant challenges of managing late-phase ocular complications in SJS/TEN, emphasizing the need for a multidisciplinary approach and aggressive intervention to preserve ocular structure and function.
REFERENCES (15)
1.
Gregory DG. New Grading System and Treatment Guidelines for the Acute Ocular Manifestations of Stevens-Johnson Syndrome. Ophthalmol. 2016 Aug;123(8):1653–1658. doi:10.1016/j.ophtha.2016.04.041. Epub 2016 Jun 11. PMID:27297404.
 
2.
Jain R, Sharma N, Basu S, Iyer G, Ueta M, Sotozono C, et al. Stevens-Johnson syndrome: The role of an ophthalmologist. Surv Ophthalmol. 2016 Jul-Aug;61(4):369–99. doi:10.1016/j.survophthal.2016.01.004. Epub 2016 Jan 30. PMID:26829569.
 
3.
Kojima M, Mieno H, Ueta M, Nakata M, Teramukai S, Sunaga Y, et al. Improvement of the Ocular Prognosis of Stevens-Johnson Syndrome and Toxic Epidermal Necrolysis: A National Survey in Japan. Am J Ophthalmol. 2024 Nov;267:50–60. doi:10.1016/j.ajo.2024.05.011. Epub 2024 May 24. PMID:38795750.
 
4.
Bennett TO, Sugar J, Sahgal S. Ocular manifestations of toxic epidermal necrolysis associated with allopurinol use. Arch Ophthalmol. 1977 Aug;95(8):1362–4. doi:10.1001/archopht.1977.04450080072005. PMID:889509.
 
5.
Lee HS, Ueta M, Kim MK, Seo KY, Sotozono C, Kinoshita S, Yoon KC. Analysis of Ocular Manifestation and Genetic Association of Allopurinol-Induced Stevens-Johnson Syndrome and Toxic Epidermal Necrolysis in South Korea. Cornea. 2016 Feb;35(2):199–204. doi:10.1097/ICO.0000000000000708. PMID:26655481.
 
6.
Hoyer D, Atti C, Nuding S, Vogt A, Sedding DG, Schott A. Toxic Epidermal Necrolysis Caused by Allopurinol: A Serious but Still Underestimated Adverse Reaction. Am J Case Rep. 2021 Oct 11;22:e932921. doi:10.12659/AJCR.932921. PMID:34634004; PMCID:PMC8522529.
 
7.
Mieno H, Ueta M, Kinoshita F, Teramukai S, Kinoshita S, Sotozono C. Corticosteroid Pulse Therapy for Stevens-Johnson Syndrome and Toxic Epidermal Necrolysis Patients With Acute Ocular Involvement. Am J Ophthalmol. 2021 Nov;231:194–199. doi:10.1016/j.ajo.2021.06.015. Epub 2021 Jun 29. PMID:34214456.
 
8.
Rashad R, Kwan JT, Shanbhag SS, Ngowyutagon P, Saeed M, Tahboub MA, et al. Long-term outcomes of glued (sutureless) amniotic membrane transplantation in acute Stevens-Johnson syndrome/toxic epidermal necrolysis: a comparative study. Br J Ophthalmol. 2024 Oct 22;108(11):1508–1513. doi:10.1136/bjo-2023-324076. PMID:38490716.
 
9.
Sharma S, Singh S, Basu S, Shanbhag SS. Chronic Ocular Sequelae and Subsequent Surgical Interventions in Stevens-Johnson Syndrome After Amniotic Membrane Transplantation. Cornea. 2022 May 1;41(5):632–634. doi:10.1097/ICO.0000000000002816. PMID:34294633.
 
10.
Mahmood AH, Alharbi AS, Almanea BA, Alsaati AF. Sutureless Amniotic Membrane (ProKera®) and Intravenous Immunoglobulin in the Management of Ocular Complications of Stevens-Johnson Syndrome-Toxic Epidermal Necrolysis Overlap. Cureus. 2021 Aug 8;13(8):e16989. doi:10.7759/cureus.16989. PMID:34540392; PMCID:PMC8422256.
 
11.
Bonneau S, Tong CM, Yang Y, Harissi-Dagher M. The treatment of end-stage corneal disease: penetrating keratoplasty compared with Boston type 1 keratoprosthesis. Graefes Arch Clin Exp Ophthalmol. 2022 Sep;260(9):2781–2790. doi:10.1007/s00417-022-05646-1. Epub 2022 Apr 6. PMID:35384455.
 
12.
Tan A, Tan DT, Tan XW, Mehta JS. Osteo-odonto keratoprosthesis: systematic review of surgical outcomes and complication rates. Ocul Surf. 2012 Jan;10(1):15–25. doi:10.1016/j.jtos.2012.01.003. Epub 2012 Jan 8. PMID:22330056.
 
13.
Sayegh RR, Ang LP, Foster CS, et al. The Boston keratoprosthesis in Stevens–Johnson syndrome. Am J Ophthalmol. 2008;145:438–444.
 
14.
Solomon A, Ellies P, Anderson DF, Touhami A, Grueterich M, Espana EM, et al. Long-term outcome of keratolimbal allograft with or without penetrating keratoplasty for total limbal stem cell deficiency. Ophthalmology. 2002 Jun;109(6):1159–66. doi:10.1016/s0161-6420(02)00960-0. PMID:12045060.
 
15.
Tóth G, Lukács A, Schirra F, Sándor GL, Killik P, Maneschg OA, et al. Ophthalmic Aspects of Stevens-Johnson Syndrome and Toxic Epidermal Necrolysis: A Narrative Review. Ophthalmol Ther. 2023 Aug;12(4):1795–1811. doi:10.1007/s40123-023-00725-w. Epub 2023 May 4. PMID:37140876; PMCID:PMC10157599.
 
eISSN:1898-2263
ISSN:1232-1966
Journals System - logo
Scroll to top