January 2020 Case Reports 221 Transorbital orbitocranial penetrating injury due to bicycle brake civilians: Risk factors on admission head computed tomography. handle in a child. Pediatr Neurosurg 2007;43:498‑500. J Neurosurg; 2015;122:219‑26. 4. Kasamo S, Asakura T, Kusumoto K, Nakayama M, Kadota K, 7. Grafman J, Schwab K, Warden D, Pridgen A, Brown HR, Atsuchi M, et al. Transorbital penetrating brain injury [in Japanese]. Salazar AM. Frontal lobe injuries, violence, and aggression. No Shinkei Geka 1992;20:433‑8. Neurology 199646:1231. 5. Kasper EM, Luedi MM, Zinn PO, Rubin PA, Chen C. Retained 8. Kazim SF, Shamim MS, Tahir MZ, Enam SA, Waheed S. Management transorbital foreign body with intracranial extension after pipe of penetrating brain injury. J Emerg Trauma Shock 2011;4:395‑402. bomb explosion. Surg Neurol Int 2010;1:94. 9. Le C, Strong EB, Luu Q. Management of anterior skull 6. Bodanapally UK, Saksobhavivat N, Shanmuganathan K, Aarabi B, base cerebrospinal fluid leaks. J Neurol Surg. B Skull Base Roy AK. Arterial injuries after penetrating brain injury in 2016;77:404‑11. Oral mucosa for reconstructive surgery and redness. On examination, we can find distorted or occluded episcleral blood vessels, scleral necrosis, and sometimes anterior in a case of severe inflammatory uveitis (sclero‑uveitis).[5] The prevalence of sclero‑uveitis is necrotizing sclero‑uveitis low, unless associated with a systemic disease.[5] Among the most common causes of sclero‑uveitis, we can include herpes virus infection, rheumatoid arthritis, tuberculosis, sarcoidosis, José Lamarca‑Mateu1,2, Borja Salvador‑Culla2, or other rheumatologic diseases.[5] Treatment of necrotizing Alba Gómez‑Benlloch2, Rafael I Barraquer1,2 sclero‑uveitis (NSU) consists of systemic corticosteroids, combined with immunosuppressants and/or immunomodulators.[6,7] Surgery is reserved only to treat complications, mainly ocular The purpose of this case is to show the efficacy of buccal mucosa perforation. In these cases, a conjunctival flap or other autologous as an alternative to treat a case of severe necrotizing sclero‑uveitis tissue grafts may be an option.[8] (NSU) associated with ocular perforation. We show a severe inflammatory NSU case that did not improve with topical Case Report treatment and scleral patch. We performed a buccal mucosa graft taken from the lower lip with excellent functional and anatomical An 86‑year‑old male presented to our hospital for a second result, with no signs of relapse of the NSU after 2 years of opinion with a history of NSU in the right eye (RE) refractory to follow‑up. Buccal mucosa can be a safe, useful, and effective treatment with ofloxacin drops three times a day, brinzolamide alternative for the reconstruction of the scleral wall. twice a day, and latanoprost every night. His past ocular history included evisceration of the left eye 1 year before, secondary Key words: Amniotic membrane transplantation, buccal mucosa, to a corneal perforation. Uncorrected VA was 0.3 decimal necrotizing scleritis, necrotizing sclero‑uveitis, oral mucosa (20/60) and best‑corrected VA was 0.6 (20/32). On examination, RE presented conjunctival hyperemia, very engorged scleral Scleritis is an inflammatory disease that presents with edema and vessels, and an area of de‑epithelialization with scleral thinning infiltrates that affect the entire thickness of the sclera. It is frequently with perforation [Fig. 1]. Anterior chamber was deep, with associated with systemic inflammatory diseases (80%).[1,2] moderate Tyndall flare, abundant cells, and endothelial Necrotizing scleritis constitutes the most aggressive form of keratic precipitates. Intraocular pressure (IOP) was 8 mmHg. scleritis. It typically appears in the elderly ages, bilaterally (50%), Fundus examination confirmed that the vitreous cavity was and can severely affect visual acuity (VA) (>50%).[3] The risks of uninflamed. Thus, a diagnosis of NSU was suspected. associated systemic disease and visual loss in necrotizing scleritis is high (80% and 50%, respectively).[4] Symptoms include pain Although clinically the lesion did not look infectious, corneal scrapes were taken to rule out infective etiology, Access this article online which came back negative. In addition, blood samples for Quick Response Code: Website: immunological (p‑ANCA, c‑ANCA, ANAs, rheumatoid factor, www.ijo.in ECA, B27) and infectious (hepatitis, HIV, syphilis) markers, Mantoux/quantiferon‑TB Gold test, and radiodiagnosis DOI: tests were performed to rule out systemic diseases. General 10.4103/ijo.IJO_671_19 examination did not show any abnormalities or signs of systemic PMID: disease, such as rheumatoid arthritis. Given the seriousness ***** This is an open access journal, and articles are distributed under the terms of the Creative Commons Attribution‑NonCommercial‑ShareAlike 4.0 License, 1 Universitat Internacional de Catalunya, Facultad de Medicina, Campus which allows others to remix, tweak, and build upon the work non‑commercially, as long as appropriate credit is given and the new creations are licensed under Sant Cugat, 2Centro de Oftalmología Barraquer, Instituto Universitario the identical terms. Barraquer, Universitat Autònoma de Barcelona, Barcelona, Spain Correspondence to: Dr. José Lamarca-Mateu, Centro de For reprints contact: reprints@medknow.com Oftalmología Barraquer, c/Muntaner 314, Barcelona ‑ 08021, Spain. E‑mail: jlamarca@barraquer.com Cite this article as: Lamarca-Mateu J, Salvador-Culla B, Gómez-Benlloch A, Received: 08-Apr-2019 Revision: 29-May-2019 Barraquer RI. Oral mucosa for reconstructive surgery in a case of severe inflammatory necrotizing sclero-uveitis. Indian J Ophthalmol 2020;68:221-3. Accepted: 09-Aug-2019 Published: 19-Dec-2019 222 Indian Journal of Ophthalmology Volume 68 Issue 1 Figure 1: Image of the scleral ulcer in the right eye showing moderate conjunctival hyperemia, engorged superficial vessels, and marked scleral thinning with calcification a b Figure 2: (a) Immediate postoperative image of the semilunar scleral patch (8 × 8 mm), with amniotic membrane covering the entire surface of the graft. (b) Image of the eye 2 months later showing signs of advanced necrosis on the scleral graft a b Figure 3: (a) Image of the buccal mucosa graft (18 × 12 mm) the day after surgery. (b) At last follow‑up, 2 years later, the buccal mucosa graft looks healthy, with no signs of recurrence of the sclero‑uveitis of the case, and being an “only eye”, oral corticosteroids AG, Vienna, Austria) and 10/0 nylon sutures (Ethilon® nylon (1 mg/Kg/day) and dexamethasone drops three times a day were suture, ©Ethicon US, LLC), with an amniotic membrane over, given. Ten days later, and based on the minimal response to the to cover the area of perforation [Fig. 2a]. Blood tests, Mantoux/ treatment, we performed an 8 × 8 mm scleral patch that was QuantiFERON‑TB Gold test, chest x‑ray, thoracic CT scan, and fixed with the help of a tissue adhesive (Tissucol Duo®, Baxter cerebral MRI were unremarkable. January 2020 Case Reports 223 Two months later, the graft presented signs of advanced the context of NSU refractory to medical treatment, preferably necrosis [Fig. 2b], and a resection of the necrotic scleral patch after a scleral patch has been attempted. was performed, combined with a buccal mucosa graft from the lower lip (18 × 12 mm) [Fig. 3a]. Postoperative treatment Declaration of patient consent consisted of growth‑factor‑enriched plasma four times a The authors certify that they have obtained all appropriate day, gentamycin/retinol/methionine ointment (Epithelizing patient consent forms. In the form the patient(s) has/have ointment; 3 mg gentamycin, 5.5 mg retinol, and 5 mg given his/her/their consent for his/her/their images and other methionine; Laboratories Thea, Clermont‑Ferrand, France) clinical information to be reported in the journal. The patients three times a day, prednisolone acetate 10 mg/mL (Pred‑forte; understand that their names and initials will not be published Allergan S.A, Madrid, Spain) five times a day, artificial and due efforts will be made to conceal their identity, but tears (Thealoz duo; Laboratories Thea, Clermont‑Ferrand, anonymity cannot be guaranteed. France) every 1–2 h, Combigan two times a day, and oral Financial support and sponsorship prednisolone 60 mg/day, tapered down 10 mg/7 days with a final maintenance dose of 10 mg/day. Nil. At the last follow‑up, 2 years later, and after cataract Conflicts of interest extraction and intraocular lens implantation, VA with There are no conflicts of interest. correction was 0.1 decimal (20/200). The buccal mucosa graft looked healthy, and the cornea showed swirling epitheliopathy References [Fig. 3b]. IOP was 14 mmHg. The patient is currently on 1. Sainz de la Maza M, Molina N, Gonzalez‑Gonzalez LA, Combigan two times a day, Pred‑forte two times a day, Doctor PP, Tauber J, Foster CS. Clinical characteristics of a large and Thealoz duo as required, with no signs of recurrence of cohort of patients with scleritis and episcleritis. Ophthalmology the sclero‑uveitis. 2012;119:43‑50. 2. Bielefeld P, Saadoun D, Héron E, Abad S, Devilliers H, Deschasse C, Discussion et al. Scleritis and systemic diseases: What should know the internist? Rev Med Interne 2018;39:711‑20. The high risk of perforation in NSU can have fatal consequences.[3,9] Thus, it is essential to carry out a rapid 3. Bowling B. Epiesclera y esclera. In: Kanski. Oftalmología clínica: Un enfoque sistemático. 8th ed. 2016. Elsevier España S.L.U. p. diagnosis of suspicion in order to identify the etiology and 254‑67 apply the necessary treatment.[10] Sometimes, despite a correct 4. Héron E, Gutzwiller‑Fontaine M, Bourcier T. Scleritis and episcleritis: diagnosis and treatment, complications may occur, given Diagnosis and treatment. Rev Med Interne 2014;35:577‑85. the great aggressiveness of the disease. In our case, there is previous history of evisceration in the contralateral eye due 5. Sainz de la Maza M, Foster CS, Jabbur NS. Scleritis‑associated uveitis. Ophthalmology 1997;104:58‑63. to a similar episode 1 year before. Thus, the management of NSU must be aggressive, focused on controlling the underlying 6. Sands DS, Chan SCY, Gottlieb CC. Methotrexate for the treatment disease.[7,11] In cases, refractory to treatment with imminent risk of noninfectious scleritis. Can J Ophthalmol 2018;53:349‑53. of perforation, an urgent surgical approach is necessary. Several 7. Oray M, Meese H, Foster CS. Diagnosis and management of options have been suggested in the past.[8] Amniotic membrane non‑infectious immune‑mediated scleritis: Current status and is usually used as a complement thanks to its anti‑inflammatory future prospects. Expert Rev Clin Immunol 2016;12:827‑37. and epithelium proliferation stimulating effect. [12] It is 8. Nguyen QD, Foster CS. Scleral patch graft in the management of important to note that these options do not solve the underlying necrotizing scleritis. Int Ophthalmol Clin 1999;39:109‑31. pathology and may not be a definitive solution, as shown in our 9. Sainz de la Maza M, Jabbur NS, Foster CS. Severity of scleritis and patient who presented scleral graft necrosis, which required episcleritis. Ophthalmology 1994;101:389‑96. replacement within a few days. In these cases, the oral mucosa 10. Sainz de la Maza M, Foster CS. Necrotizing scleritis after ocular of autologous origin can be an effective and lasting alternative, surgery. A clinicopathologic study. Ophthalmology 1991;98:1720‑6. mainly due to its immunological privileges.[13‑15] They have been 11. Sainz de la Maza M, Molina N, Gonzalez‑Gonzalez LA, Doctor PP, used previously for cases of scleral melt in chemical burns.[16] Tauber J, Foster CS. Scleritis therapy. Ophthalmology 2012;119:51‑8. This situation makes the adaptation to the surrounding tissue 12. Jirsova K, Jones G. Amniotic membrane in ophthalmology: more favorable in the buccal mucosa grafts than in scleral ones. Properties, preparation, storage and indications for grafting ‑ A review. Cell Tissue Bank 2017;18:193‑204. Another positive aspect of the oral mucosa is that it is easily 13. Grixti A, Malhotra R. Oral mucosa grafting in periorbital adaptable to practically any surface since it has elastic fibers reconstruction. Orbit 2018;6:1‑18. that provide distensibility, unlike the sclera. In addition, it allows more extensive and easier to manipulate autografts 14. Henderson H, Collin J. Mucous membrane grafting. Dev Ophthalmol 2008;41:230‑42. with respect to the conjunctival tissue. In fact, it is the tissue used in the osteo‑odonto‑ and osteo‑keratoprosthesis.[17] As a 15. Hodge C, Sutton G, Devasahayam R, Georges P, Treloggen J, Cooper S, et al. The use of donor scleral patch in ophthalmic disadvantage, it is worth noting that in cases of evident uveal surgery. Cell Tissue Bank 2017;18:119‑28. exposure that require a significant physical resistance, buccal mucosa does not have enough strength with respect to the graft 16. Wang S, Tian Y, Zhu H, Cheng Y, Zheng X, Wu J. Tenonplasty combined with free oral buccal mucosa autografts for repair of scleral origin or other synthetic patches.[8] of sclerocorneal melt caused by chemical burns. Cornea 2015;34:1240‑4. Conclusion 17. Charoenrook V, Michael R, de la Paz MF, Temprano J, In conclusion, the application of buccal mucosa can be safe, Barraquer RI. Comparison of long‑term results between useful, and effective for the reconstruction of the scleral wall osteo‑odonto‑keratoprosthesis and tibial bone keratoprosthesis. in cases of marked thinning, with minimal uveal exposure, in Ocul Surf 2018;16:259‑64.