Literature DB >> 3565516

Keratitis associated with the multiple endocrine deficiency, autoimmune disease, and candidiasis syndrome.

R D Wagman, J J Kazdan, S W Kooh, D Fraser.   

Abstract

Optimal management of the ocular and systemic components of the multiple endocrine deficiency, autoimmune disease, candidiasis syndrome requires early identification of affected individuals. This autosomal recessive syndrome is characterized by hypoparathyroidism, Addison's disease, and chronic mucocutaneous candidiasis among other endocrinopathies and immune disorders. We retrospectively reviewed 16 patients, 14 with the full syndrome and two with the syndrome partially manifested. Four of these patients manifested a self-limited, bilateral keratitis in which the age of onset ranged from 2 to 9 years. Keratitis preceded the onset of any endocrinopathy in two of four patients and was among the first signs of the syndrome. The keratitis was not caused by hypoparathyroidism or candidiasis. The anterior stromal vascularization and scarring resulted in a visual acuity of 20/50 or worse in four of eight affected eyes. We recommend medical management of the corneal disease without surgical intervention.

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Year:  1987        PMID: 3565516     DOI: 10.1016/s0002-9394(14)74281-3

Source DB:  PubMed          Journal:  Am J Ophthalmol        ISSN: 0002-9394            Impact factor:   5.258


  4 in total

1.  Resolution of autoimmune polyglandular syndrome-associated keratopathy with keratolimbal stem cell transplantation: case report and historical literature review.

Authors:  Manan Shah; Edward Holland; Chi-Chao Chan
Journal:  Cornea       Date:  2007-06       Impact factor: 2.651

Review 2.  Ocular involvement in primary immunodeficiency diseases.

Authors:  Sima Hosseinverdi; Hassan Hashemi; Asghar Aghamohammadi; Hans D Ochs; Nima Rezaei
Journal:  J Clin Immunol       Date:  2013-11-30       Impact factor: 8.542

3.  Long term keratits treatment with topical cyclosporin a in autoimmune polyglandular syndrome type 1.

Authors:  Omar AlAbbasi; Moustafa S Magliyah; Muhammad Ahad
Journal:  Am J Ophthalmol Case Rep       Date:  2021-01-06

Review 4.  Autoimmune Addison's Disease as Part of the Autoimmune Polyglandular Syndrome Type 1: Historical Overview and Current Evidence.

Authors:  Roberto Perniola; Alessandra Fierabracci; Alberto Falorni
Journal:  Front Immunol       Date:  2021-02-26       Impact factor: 7.561

  4 in total

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