Abstract Background Known causes of superficial punctuate keratopathy (SPK) in children include entropion, viral infection, blepharokeratoconjunctivitis (BKC), and toxicity of eye drops. However, there are some SPK patients whose causes could not be identified well. Herein, we describe the history, diagnosis, treatment, and prognosis of a rare case. Case presentation To report a case of superficial punctate keratopathy (SPK) which coexisted with floppy eyelid syndrome (FES) and presented as intermittent red eye and blurred vision in an 11-year-old boy who slept in the prone position. His condition did not improve despite treatment with topical antibiotics (levofloxacin, tobramycin), steroid eye drops (prednisolone), and artificial tears. The patient was diagnosed with tonsil hypertrophy and nasopharyngeal adenoid hypertrophy and obstructive sleep apnea syndrome (OSAS). He underwent tonsillectomy and adenoidectomy. Then he started sleeping in the supine position postoperatively. The SPK, red eye and blurred vision completely resolved after surgery without additional treatment. The corneal sensation also recovered gradually during the next 7 years. However, the floppy eyelid did not resolve. Conclusion Recurrent SPK of childhood might be related to tonsil hypertrophy, adenoid hypertrophy and OSAS, which can be rehabilitated by a surgical approach.
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