Nummular keratopathy in a patient with Hyper-IgD Syndrome

Courtney L. Kraus; Susan M. Culican

doi:10.1186/1546-0096-7-14

Nummular keratopathy in a patient with Hyper-IgD Syndrome

Courtney L. Kraus, Susan M. Culican

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14 Scopus citations

Abstract

Purpose: To report a case of recurrent nummular keratitis in a pediatric patient with Hyperimmunoglobulinemia D syndrome. Methods: A retrospective chart review. Results: A 14-year-old boy with Hyperimmunoglobulinemia D syndrome (HIDS) presented with photophobia and ocular irritation concomitant with disease exacerbation. He was found on exam to have significant nummular keratitis, which responded to a short course of topical steroids. Despite acute response to local immunosuppression, the patient had several recurrent attacks and eventually developed a large corneal scar and decreased vision. After initiation of infliximab therapy his ocular sequelae improved dramatically and his vision returned to 20/20. Conclusion: One possible form of end-organ damage associated with HIDS is vision threatening nummular keratopathy.

Original language	English (US)
Article number	14
Journal	Pediatric Rheumatology
Volume	7
DOIs	https://doi.org/10.1186/1546-0096-7-14
State	Published - Aug 5 2009
Externally published	Yes

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title = "Nummular keratopathy in a patient with Hyper-IgD Syndrome",

abstract = "Purpose: To report a case of recurrent nummular keratitis in a pediatric patient with Hyperimmunoglobulinemia D syndrome. Methods: A retrospective chart review. Results: A 14-year-old boy with Hyperimmunoglobulinemia D syndrome (HIDS) presented with photophobia and ocular irritation concomitant with disease exacerbation. He was found on exam to have significant nummular keratitis, which responded to a short course of topical steroids. Despite acute response to local immunosuppression, the patient had several recurrent attacks and eventually developed a large corneal scar and decreased vision. After initiation of infliximab therapy his ocular sequelae improved dramatically and his vision returned to 20/20. Conclusion: One possible form of end-organ damage associated with HIDS is vision threatening nummular keratopathy.",

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AB - Purpose: To report a case of recurrent nummular keratitis in a pediatric patient with Hyperimmunoglobulinemia D syndrome. Methods: A retrospective chart review. Results: A 14-year-old boy with Hyperimmunoglobulinemia D syndrome (HIDS) presented with photophobia and ocular irritation concomitant with disease exacerbation. He was found on exam to have significant nummular keratitis, which responded to a short course of topical steroids. Despite acute response to local immunosuppression, the patient had several recurrent attacks and eventually developed a large corneal scar and decreased vision. After initiation of infliximab therapy his ocular sequelae improved dramatically and his vision returned to 20/20. Conclusion: One possible form of end-organ damage associated with HIDS is vision threatening nummular keratopathy.

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Nummular keratopathy in a patient with Hyper-IgD Syndrome

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