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Idiopathic Interstitial Keratitis in a Child 35 Summer 2021 • Vol 16 | No 1 Case report SA Ophthalmology Journal Idiopathic interstitial keratitis in a child 35 Idiopathic interstitial keratitis in a child DA Erasmus MBBCh, MMed, Dip Ophth (SA); Registrar, Department of Neurosciences, Division of Ophthalmology, University of the Witwatersrand, Johannesburg, South Africa ORCID: https://orcid.org/0000-0002-8395-7080 R Höllhumer MBChB, MMed, FC Ophth(SA); Consultant ophthalmologist, Department of Neurosciences, Division of Ophthalmology, University of the Witwatersrand, Johannesburg, South Africa ORCID: https://orcid.org/0000-0002-4375-2224 Corresponding author: Dr DA Erasmus, 40 2nd Avenue, Parktown North, 2193; tel.: +27 76 462 3355; email: [email protected] Abstract Interstitial keratitis represents inflammation and new vessel investigations can help to establish the underlying cause of formation in the middle layers of the cornea without tissue interstitial keratitis. loss. The most important infective aetiologies include herpes viruses, tuberculosis and syphilis. Keywords: interstitial, keratitis, inflammation, cornea, stroma Non-infectious causes such as Cogan’s syndrome should be considered in those cases with associated neurosensory Funding: No external funding was received. hearing loss. The pattern and laterality of inflammation together with the Conflict of interest: The authors have no conflicts of interest presence or absence of systemic features as well as relevant to declare. Severe Mild Moderate Moderate/Mild A * Dry Eye range like NEVER BEFORE Introduction systemic features provides a clue to the a tuberculosis (TB) endemic area such as Interstitial keratitis is a non-ulcerative underlying cause of interstitial keratitis. South Africa. Parasitic infections causing inflammation and subsequent Interstitial keratitis can be broadly interstitial keratitis are very uncommonly vascularisation of the corneal stroma divided into infectious and non-infectious encountered in South Africa but imported which does not primarily involve the aetiologies. There is a paucity of cases may be due to entities such as epithelium or endothelium. The underlying information regarding interstitial keratitis leishmaniasis, onchocerciasis (river pathophysiology is believed to be a in Sub-Saharan Africa and in the paediatric blindness) and trypanosomiasis. cellular and humoral immune-mediated population more generally worldwide. The Non-infectious cases include Cogan’s type IV hypersensitivity response directed majority of cases that have an infectious syndrome, sarcoidosis, Hodgkin’s and Contains towards corneal stromal antigens, aetiology in adults are viral (with herpes T-cell lymphoma, Kaposi’s sarcoma, gold HA** infective antigens or a combination of simplex virus comprising 35.1%) or and arsenic toxicity, and intraocular both.1,2 Clinically the infiltration may be treponemal (comprising approximately sclerosing inflammatory pseudotumour.1-3 diffuse, focal or multifocal and may be 18.6% of cases of both congenital and We present a case of idiopathic interstitial Taking care of all types of Dry Eyes located superficially or, more commonly, acquired infection); no identifiable cause keratitis in a South African child. deep within the stroma. It may be is found in 32% of cases. Other important unilateral or bilateral and be further viral infections in adults include Epstein- Case presentation *Pertains to the unique actuator of Artelac Splash Eye Drops and the unique formulation of Artelac Intense Rebalance Eye Drops. **Hyaluronic Acid. divided into active and inactive forms Barr virus (1%), human T-lymphocytic virus A seven-year-old black African female was Proprietary name: Artelac® Advanced Lipids Eye Drops. Contains 2 mg carbomer, medium chain triglycerides. Preservative: Cetrimide (10 g multi-dose only). Proprietary name: Artelac® Intense with the latter defined as a period of type I, mumps and varicella zoster virus bought in by her mother with a one-week Rebalance Eye Drops. Contains 0,15 % hyaluronic acid (as sodium hyaluronate), 0,5 % polyethylene glycol 8000, vitamin B12. Preservative: Oxyd® (a gentle preservative that converts to water, oxygen and salt at the surface of the eye in the 10 ml multi-dose unit only). Proprietary name: Artelac® Splash Eye Drops. Contains 0,24 % hyaluronic acid (as sodium hyaluronate) in the 10 ml multi-dose unit and no characteristic signs and symptoms (3.1%). Another important mycobacterial history of decreased vision (left eye worse 0,2 % hyaluronic acid (as sodium hyaluronate) in the single dose units. Both the 10 ml multi-dose and single dose units are preservative free. Proprietary name: Artelac® Moisture Eye Drops. Contains 0,32 % hypromellose. Preservative: Cetrimide (10 ml multi-dose only). in the preceding year. The topographical cause is tuberculosis (3.1%), although this than the right) with associated pain and For full prescribing information, refer to the instructions for use. Further information is available on request from Bausch + Lomb. © 2017 Bausch & Lomb Incorporated. ®/™ denote trademarks of distribution together with the presence of is expected to have greater relevance in redness. Furthermore, both of her wrists Bausch & Lomb Incorporated. Distributed by: Sofl ens (Pty) Ltd. Reg. No.: 1968/011787/07. 254 Hall Street, Centurion. Tel: +27 10 025 2100. www.bausch.co.za BL386/19 Case report Vol 16 | No 1 • Summer 2021 36 Idiopathic interstitial keratitis in a child SA Ophthalmology Journal Figure 1a. No abnormal facies, no saddle nose deformity or low- Figure 1b. Normal dentition set ears had begun to swell and become stiff General examination revealed no pallor, swollen, tender and warm to the touch approximately two weeks prior. Although jaundice, lymphadenopathy or pitting with clinical effusions as seen in Figure 1d. the patient denied this was painful, her oedema. Importantly there were no Both ankles were mildly tender without mother noted that it improved throughout stigmata of congenital syphilis infection any gross deformity. the day and was exacerbated by cold including Hutchinson’s teeth, mulberry Vision was recorded as 6/18 on the weather. There were no episodes of molars or sabre tibias (Figures 1a and right and counting fingers on the left with headaches, nausea, vomiting or tinnitus. b). The patient was able to walk unaided no improvement on pinhole. Both eyes Although the patient was HIV-exposed, without difficulty. Her left hand revealed had ciliary injection with multifocal areas her birth PCR was confirmed negative. a ‘Z’-shaped deformity with bilaterally of stromal inflammation and marginal The remaining maternal booking swollen and mildly tender proximal infiltrates that were more prominent on the bloods (including RPR) were negative. interphalangeal joints with associated Immunisation history was inconsistent, Boutonniere deformities as seen in with a period of six years elapsing without Figure 1c. Similarly, both wrists were coverage. Developmentally the patient’s mother reported that she had achieved normal milestones although she gave the history that her hearing was poor from a ‘young age’. Despite this she was coping well academically in Grade 2. At the age of 2 years the patient was diagnosed with juvenile idiopathic arthritis (JIA) following an episode of joint swelling. She was commenced on methotrexate, chloroquine and prednisone for the period 2012 to 2017 during which time she was reportedly well controlled. She was subsequently lost to follow-up and no further symptoms were reported until the most recent Figure 1c. Dorsal view of the hands presentation. She was otherwise well with showing ‘Z’-shaped deformity of the no other admissions. There are no known thumbs with swollen interphalangeal Figure 1d. Lateral view of the hands and contacts with TB. joints and wrists wrists showing wrist swelling Summer 2021 • Vol 16 | No 1 Case report SA Ophthalmology Journal Idiopathic interstitial keratitis in a child 37 Figure 2a. Left eye showing multifocal areas of inflammation and Figure 2b. Right eye showing multifocal areas of stromal deep vascularisation. Note the ‘salmon-patch’ lesion inferiorly inflammation and vascularisation left (Figures 2a and b). Three hundred-and- film was normal (Figure 3) and a tuberculin patient was started on dexamethasone sixty degrees of new vessels were noted skin test was negative (<5 mm) at 48 hours. 0.1% QID in both eyes, atropine 1% BD and deep within the stroma. There was a large, Auditory testing suggested moderately lanolin ointment at night. The patient was deep 3×3.2 mm ‘salmon-coloured’ nodule on severe conductive hearing loss in both the re-referred to paediatric rheumatology the inferior cornea on the left. Decreased right (Figure 4a) and left ear (Figure 4b). and restarted on methotrexate 12.5 mg corneal sensation and generalised punctate Thus, a working diagnosis of idiopathic once weekly, prednisone 20 mg daily epithelial erosions without larger areas interstitial keratitis was made and the and chloroquine 200 mg five times a of staining were present bilaterally. The Table I: Serological investigations week for serologically negative JIA. The anterior chambers were active with 2+ Blood Value patient showed significant improvement cells and 1+ flare although iris nodules or in symptoms and visual acuity – at her WCC 5.79 transillumination defects were absent. iCare two weeks follow-up her best corrected tonometry was 12 and 10 mmHg for the Hb 12.4 visual acuity had improved to 6/9 in both right and left eye respectively. The vitreous Platelets 387
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