30 November 2022
: Case report 
An Infant with Bilateral Keratitis: From Infectious to Genetic Diagnosis
Challenging differential diagnosis
Louis-Philippe Thibault1ABCDEF*, Grant A. Mitchell2BCDEF, Brigitte Parisien1BCDE, Patrick Hamel
3BCDE, Ana C. Blanchard4ABCDEF
DOI: 10.12659/AJCR.937967
Am J Case Rep 2022; 23:e937967
Table 1. Comparison of main characteristics of previously published TYRII case reports, with emphasis on ocular lesions at initial clinical presentation (presented in author alphabetical order).
| References | Sex | Age at onset of symptoms | Age at presentation | Symptoms and/or clinical findings at presentation | Initial diagnosis and treatment | Clinical evolution following initial treatment |
|---|---|---|---|---|---|---|
| Charlton []10 | Case 1: MCase 2: F | Case 1: 6 wCase 2: 6½ mo | Case 1: 3 moCase 2: 11 mo | Case 1: Bilateral photophobia and conjunctival injection, with central corneal dendritic-like lesions.Case 2: Conjunctival injection and photophobia in her left eye. Dendritic lesions without conjunctival injection in her right eye | Case 1: Initial diagnosis not defined, treated with topical antibiotics for 6 weeks, then refered for HSV keratitis.Case 2: Bilateral recalcitrant HSV keratitis, treated with topical antibiotics, cycloplegics, topical glucocorticoids, arabinoside, trifluorothymidine, and debridement | Case 1: No improvement.Case 2: Sporadic improvement and exacerbations apparently not related to treatment |
| Colditz []12 | Case 1: MCase 2: FCase 3 and 4: N/A (palmo-plantar lesions at presentation) | Case 1: 3 moCase 2: 5 w | Case 1: 4 yoCase 2: 2 yo | Case 1: Unilateral epiphora, photophobia, and blepharospasm. Dendritic corneal ulcers at 5 mo.Case 2: Conjonctival injection and photophobia. Central corneal dendritic ulcers at 5 mo | Case 1: HSV, treated with topical idoxuridine for 2 months.Case 2: N/A | Case 1: Increased photophobia.Case 2: N/A |
| Goddé-Jolly []13 | F | 10 days | 7 mo | Conjonctivitis of the left eye, with mild corneal haze | Purulent conjonctivitis, treated with antibiotics eyedrops for 3 weeks | Increased corneal haze and extension of conjunctivitis symptoms to the right eye |
| Gokhale []14 | N/A | 2 mo | 5 mo | Photophobia, corneal haze, followed by bilateral dendritic keratitis | Conjunctivitis, treated with antibiotics eyedrops for 2 months, followed by suspicion of HSV keratitis, treated by acyclovir ointment for 2 weeks | No improvement |
| Hervé []15 | Case 1: FCase 2: F | Case 1: First days of life.Case 2: First days of life. | Case 1: 16 yoCase 2: 8 mo | Case 1: Conjunctivitis, photophobia, epiphora.Case 2: Conjunctivitis, photophobia, pain | Case 1: Chronic conjunctivitis and HSV keratitis, treated with antibiotics, allergy medications, homeopathy.Case 2: HSV keratitis, treated with eyedrops medication (N/S) | Case 1: No improvement.Case 2: No improvement, dendritic keratitis noticed at 1 yo |
| Kymionis []18 | M, twins | 9 mo | 15 mo | Eye rubbing, photophobia, and epiphora | Bilateral HSV keratitis, treated with Topical trifluridine for 2 months prior to presentation | No improvement |
| Macsai []8 | Report of 9 cases.Details given for Case 1 (F) and Case 9 (M) | Birth to 11 moCase 1: 1 moCase 9: 11 mo | 3 mo to 57 yoCase 1: 5 mo 1/2Case 9: 11 mo | Photophobia, redness, tearing, blepharospasm, pain, eye rubbing.Case 1: PhotophobiaCase 9: Eye rubbing, photophobia | 7/9 (78%) diagnosed and treated for HSV keratitis with topical trifluridine.Case 1: HSV keratitis, treated with eyedrops trifluridine.Case 9: Diagnosis N/S, treated with eyedrops trifluridine | 4/7 (57%) showed transient resolution.Case 1: Resolution of symptoms, clinical signs still present (superficial right corneal haze, and dendritiform pattern in left cornea).Case 9: Less symptomatic, large dendritiform epithelial lesion on right cornea |
| Podglajen-Wecxsteen []16 | M | 9 mo | 20 yo | Tearing, conjunctival injection | Allergic conjunctivitis, treated accordingly (N/S) | No improvement |
| Tsai []17 | F | Birth | 2 yo | Photophobia, epiphora | HSV keratitis, treated accordingly (N/S) | No improvement |
| Soares []11 | M | 2 mo | 2 yo | Photophobia | HSV keratitis, treated with acyclovir eye ointment, followed by oral acyclovir | No improvement |
| N/A – not available; N/S – not specified. | ||||||