Persistent corneal epithelial defect associated with sleeping position: A case report

Yingsi Li; Shudi Chen; Wenjing Song; Yu Cheng; Yuan Gao; Luoying Xie; Meiting Huang; Xiaoming Yan

doi:10.4103/ijo.IJO_2127_22

CASE REPORT

Year : 2023 | Volume : 3 | Issue : 2 | Page : 278-280

Persistent corneal epithelial defect associated with sleeping position: A case report

Yingsi Li, Shudi Chen, Wenjing Song, Yu Cheng, Yuan Gao, Luoying Xie, Meiting Huang, Xiaoming Yan
Department of Ophthalmology, Peking University First Hospital, Beijing, China

Date of Submission	25-Aug-2022
Date of Acceptance	31-Jan-2023
Date of Web Publication	28-Apr-2023

Correspondence Address:
Xiaoming Yan
Department of Ophthalmology, Peking University First Hospital, No. 8 Xishiku Avenue, Beijing, 100034
China

Source of Support: None, Conflict of Interest: None

DOI: 10.4103/ijo.IJO_2127_22

Abstract

Herein, we report a rare case of persistent corneal epithelial defect (PCED) in a 9-year-old child with floppy eyelid syndrome (FES) and abnormal sleeping position. The boy presented with a two-month history of foreign body sensation in the right eye, a 4 × 5 mm² corneal epithelial defect, irregular astigmatism, a long history of snoring, and intermittent nasal congestion. However, no improvement was observed after basic treatment for one month. The child slept in the prone position and was obese. After changing the sleeping position and undergoing nasal therapy in combination with artificial tear therapy, his corneal epithelial defects disappeared, and corneal astigmatism was reduced.

Keywords: Floppy eyelid syndrome, persistent corneal epithelial defect, sleeping position

How to cite this article:
Li Y, Chen S, Song W, Cheng Y, Gao Y, Xie L, Huang M, Yan X. Persistent corneal epithelial defect associated with sleeping position: A case report. Indian J Ophthalmol Case Rep 2023;3:278-80

How to cite this URL:
Li Y, Chen S, Song W, Cheng Y, Gao Y, Xie L, Huang M, Yan X. Persistent corneal epithelial defect associated with sleeping position: A case report. Indian J Ophthalmol Case Rep [serial online] 2023 [cited 2023 Jun 10];3:278-80. Available from: https://www.ijoreports.in/text.asp?2023/3/2/278/374938

Yingsi Li, Shudi Chen; Contributed equally to this work.

Persistent corneal epithelial defect (PCED) is characterized by a continuous corneal defect over two weeks due to the impaired healing capability of the corneal epithelium even after undergoing corresponding treatments.^[1],[2] Long-lasting corneal defects without reasonable and effective treatments tend to progress to corneal stromal dissolution, ocular infection, and even corneal perforation, which could seriously affect patients' vision.^[2],[3] The concomitance of recurrent keratopathy, floppy eyelid syndrome (FES), and nasopharyngeal abnormalities is rarely seen in children. To our knowledge, only one case has been reported thus far.^[4] In this report, we present such a case in an overweight child.

Case Report

A 9-year-old boy presented with a two-month history of persistent foreign body sensation in the right eye and was referred to our ophthalmology clinic on September 13, 2021. Under slit-lamp examination, corneal epithelial defects with a clear boundary of 4 × 5 mm² above the temporal part were noticed on his right eye [[Figure 1]a, white arrows], and epithelial damage was further confirmed by corneal fluorescein staining [Figure 1]b. An increase in Langerhans cells in the corneal epithelium was observed via confocal microscopy [[Figure 1]c, black arrows]. Irregular astigmatism (7.4 D) on the corneal anterior surface in the right eye was detected by a Pentacam three-dimensional anterior segment analyzer [Figure 1]d. Samples from corneal lesions were taken for microscopy and bacterial culture, with negative isolates for both bacteria and herpes simplex virus (HSV). No other obvious abnormality was found in anterior and posterior segment examinations. The child subsequently received combined treatments with lubricant tear drops, topical steroids, therapeutic contact lenses, and topical antibiotics. However, corneal lesions still existed in the right eye at each visit during the one-month follow-up period.

Figure 1: (a) Slit-lamp photograph showing corneal epithelial defects (white arrows) of the right eye. (b) A 4 × 5 mm² corneal epithelial defect confirmed by fluorescein staining. (c) Confocal microscopy images showing an increase in Langerhans cells in corneal epithelium (black arrows). (d) Pentacam showing 7.4 D astigmatism of the right eye. (e) Photograph of right eye showing eyelash ptosis on the upper eyelid. (f) and (g) Corneal epithelial defects disappeared after the combined treatment. (h) Corneal astigmatism was reduced to 3.1D after treatment

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On October 11, we noticed eyelash ptosis in both eyes of the child [Figure 1]e, slightly easy evagination of the upper eyelid with papillary conjunctivitis, and a width of the upper eyelid over 14 mm. Therefore, the child was diagnosed with PCED and FES. On further questioning, his parents reported that he had a long history of snoring and intermittent nasal congestion, with a habit of sleeping in the prone position tilted to the right side. Given that the child's body mass index (BMI) was 26.3 and that obesity and FES are closely associated with obstructive sleep apnea (OSA), he was referred to the otorhinolaryngology department for further treatment. However, due to insufficient evidence, the child was not diagnosed with OSA. He was given a definitive diagnosis of chronic sinusitis and associated treatment regimens (budesonide nasal spray, clarithromycin, and expectorants twice daily for two weeks). Glucocorticoids and antibiotics were stopped, and he continued treatment with lubricant eye drops (carbomer and deproteinized calf blood extract eye drops).

Surprisingly, the symptoms of foreign body sensation and corneal lesions disappeared in the right eye [Figure 1]f and [Figure 1]g after one month of nasal therapy synchronized with artificial tear drops and corrected sleep position. Moreover, corneal astigmatism was significantly reduced to 3.1 D [Figure 1]h.

Discussion

In this case study, we examined an overweight child who slept in the prone position and had PCED in the right eye, mild FES, and chronic sinusitis. After a change in sleep posture, his corneal epithelial defect healed, his corneal astigmatism decreased, discomfort in the right eye disappeared, and ocular surface inflammation was improved. Although the degree of eyelid laxity and BMI did not change significantly during the period of treatment, we showed that lax eyelids and poor sleeping posture could be the pathogenic and precipitating factors for PCED.

The concomitance of recurrent keratopathy, FES, and nasopharyngeal abnormalities is rarely seen in children. To our knowledge, only one case has been reported thus far.^[4] In this report, an 11-year-old child with FES and OSA experienced rapid healing of recurrent keratopathy after a combined operation of tonsillectomy and adenoidectomy.^[4] The child in this case showed snoring symptoms and OSA risk indicators, including obesity and a prone sleeping position, even though he had not been diagnosed with OSA. Since there are regional variations in the diagnostic standards for OSA in children, this patient may be diagnosed with mild OSA in various nations. Therefore, in addition to sinusitis-related treatment, we advise children to try management strategies for mild OSA, such as an improved sleeping posture and weight loss.^[5] Long-term ocular discomfort existed due to persistent corneal epithelial defects, and we focused on aggressive eye lubrication and nighttime ocular protection. During the follow-up period, there was no recurrence and no tendency of keratoconus. Although beneficial results have been achieved after nasal treatment for chronic sinusitis in combination with artificial tear therapy and sleeping posture intervention, the relationship between sleep breathing status and keratopathy could not be confirmed due to insufficient evidence.

Obesity is an independent risk factor for FES. Serum leptin levels are elevated in obese patients due to leptin resistance. Leptin may be associated with the upregulation of matrix metalloproteinase 9 (MMP-9) in the eyelids, which may contribute to the development of FES.^[6] FES makes the upper eyelid prone to ectropion and exposure and dryness on the ocular surface, coupled with ocular compression and increased temperature, resulting in the increased activity of inflammatory mediators in the long-term right-sided prone position.^[4],[7] The combination of these factors eventually leads to persistent keratopathy.

Conclusion

In summary, our case showed that PCED might be related to FES, sleeping posture, and obesity. At present, the management of PCED is quite difficult and challenging. Therefore, ophthalmologists should pay due attention to the eyelid laxity, sleep position, and body shape of patients. In cases where it is difficult to change BMI and eyelid laxity in a short period of time, improving sleeping posture may be the key to the early treatment of PECD.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient (s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initial s will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

References

1.	Tsubota K, Goto E, Shimmura S, Shimazaki J. Treatment of persistent corneal epithelial defect by autologous serum application. Ophthalmology 1999;106:1984-9.
2.	Wirostko B, Rafii M, Sullivan DA, Morelli J, Ding J. Novel therapy to treat corneal epithelial defects: A hypothesis with growth hormone. Ocul Surf 2015;13:204-12.
3.	Berman M. The pathogenesis of corneal epithelial defects. Acta Ophthalmol Suppl 1989;192:55-64.
4.	Gao YY, Wang HJ, Wu Y. Superficial punctate keratopathy in a pediatric patient was related to adenoid hypertrophy and obstructive sleep apnea syndrome: A case report. BMC Ophthalmol 2018;18:55.
5.	Marcus CL, Brooks LJ, Draper KA, Gozal D, Halbower AC, Jones J, et al. Diagnosis and management of childhood obstructive sleep apnea syndrome. Pediatrics 2012;130:576-84.
6.	Idowu OO, Ashraf DC, Vagefi MR, Kersten RC, Winn BJ. Floppy eyelid syndrome: Ocular and systemic associations. Curr Opin Ophthalmol 2019;30:513-24.
7.	Mazharian A, Panthier C, Courtin R, Jung C, Rampat R, Saad A, et al. Incorrect sleeping position and eye rubbing in patients with unilateral or highly asymmetric keratoconus: a case-control study. Graefes Arch Clin Exp Ophthalmol 2020;258:2431-9.