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Progressive keratolysis with pseudopterygium associated with erythema elevatum diutinum.
Ophthalmology. 2011 May; 118(5):927-33.O

Abstract

OBJECTIVE

To report a patient with a rare case of progressive keratolysis with pseudopterygium associated with erythema elevatum diutinum (EED).

DESIGN

Interventional case report and literature review.

PARTICIPANT

One patient with ocular presentation of EED was identified.

METHODS

A 64-year-old woman presented with recurrent pterygium and painful corneal thinning with impending perforation of the left eye. Examination revealed intensely inflamed pseudopterygium, paracentral and peripheral corneal ulcerations, and descemetocele with a pinpoint leakage.

MAIN OUTCOME MEASURES

Clinical course and laboratory and histopathologic findings.

RESULTS

Disseminated skin lesions developed during hospital admission. Examination revealed multiple, well-demarcated, discrete, red to violaceous, firm nodules and plaques on the knuckles and dorsum of the hands, and the extensor surface of the forearms, elbows, knees, right ankle, and buttocks. A skin biopsy of the recent lesions showed a dense and diffuse inflammatory cell infiltration, predominantly neutrophilic infiltrates and nuclear dust (leukocytoclasis), located around small blood vessels in the dermis, consistent with the diagnosis of EED. The diagnosis of progressive keratolysis with pseudopterygium associated with EED was made. Spontaneous, rapid resolution of both cutaneous and ocular lesions occurred after dapsone therapy.

CONCLUSIONS

Erythema elevatum diutinum is a rare, chronic, and recurrent disease that has both dermatologic and ocular manifestations. Peripheral ulcerative keratitis seems to be the most common ocular finding and may be the initial presenting feature of EED. Ophthalmologists should be aware of this rare entity in the differential diagnosis of ulcerative or nonulcerative peripheral keratitis.

Authors+Show Affiliations

Department of Ophthalmology, Ramathibodi Hospital, Mahidol University, Bangkok, Thailand. lekhanont@yahoo.comNo affiliation info availableNo affiliation info availableNo affiliation info availableNo affiliation info available

Pub Type(s)

Case Reports
Journal Article
Review

Language

eng

PubMed ID

21211842

Citation

Lekhanont, Kaevalin, et al. "Progressive Keratolysis With Pseudopterygium Associated With Erythema Elevatum Diutinum." Ophthalmology, vol. 118, no. 5, 2011, pp. 927-33.
Lekhanont K, Patarakittam T, Mantachote K, et al. Progressive keratolysis with pseudopterygium associated with erythema elevatum diutinum. Ophthalmology. 2011;118(5):927-33.
Lekhanont, K., Patarakittam, T., Mantachote, K., Waiyawatjamai, P., & Vongthongsri, A. (2011). Progressive keratolysis with pseudopterygium associated with erythema elevatum diutinum. Ophthalmology, 118(5), 927-33. https://doi.org/10.1016/j.ophtha.2010.09.013
Lekhanont K, et al. Progressive Keratolysis With Pseudopterygium Associated With Erythema Elevatum Diutinum. Ophthalmology. 2011;118(5):927-33. PubMed PMID: 21211842.
* Article titles in AMA citation format should be in sentence-case
TY - JOUR T1 - Progressive keratolysis with pseudopterygium associated with erythema elevatum diutinum. AU - Lekhanont,Kaevalin, AU - Patarakittam,Thanikan, AU - Mantachote,Kanoktip, AU - Waiyawatjamai,Premjit, AU - Vongthongsri,Anun, Y1 - 2011/01/06/ PY - 2010/01/07/received PY - 2010/08/11/revised PY - 2010/09/14/accepted PY - 2011/1/8/entrez PY - 2011/1/8/pubmed PY - 2011/7/9/medline SP - 927 EP - 33 JF - Ophthalmology JO - Ophthalmology VL - 118 IS - 5 N2 - OBJECTIVE: To report a patient with a rare case of progressive keratolysis with pseudopterygium associated with erythema elevatum diutinum (EED). DESIGN: Interventional case report and literature review. PARTICIPANT: One patient with ocular presentation of EED was identified. METHODS: A 64-year-old woman presented with recurrent pterygium and painful corneal thinning with impending perforation of the left eye. Examination revealed intensely inflamed pseudopterygium, paracentral and peripheral corneal ulcerations, and descemetocele with a pinpoint leakage. MAIN OUTCOME MEASURES: Clinical course and laboratory and histopathologic findings. RESULTS: Disseminated skin lesions developed during hospital admission. Examination revealed multiple, well-demarcated, discrete, red to violaceous, firm nodules and plaques on the knuckles and dorsum of the hands, and the extensor surface of the forearms, elbows, knees, right ankle, and buttocks. A skin biopsy of the recent lesions showed a dense and diffuse inflammatory cell infiltration, predominantly neutrophilic infiltrates and nuclear dust (leukocytoclasis), located around small blood vessels in the dermis, consistent with the diagnosis of EED. The diagnosis of progressive keratolysis with pseudopterygium associated with EED was made. Spontaneous, rapid resolution of both cutaneous and ocular lesions occurred after dapsone therapy. CONCLUSIONS: Erythema elevatum diutinum is a rare, chronic, and recurrent disease that has both dermatologic and ocular manifestations. Peripheral ulcerative keratitis seems to be the most common ocular finding and may be the initial presenting feature of EED. Ophthalmologists should be aware of this rare entity in the differential diagnosis of ulcerative or nonulcerative peripheral keratitis. SN - 1549-4713 UR - https://www.unboundmedicine.com/medline/citation/21211842/Progressive_keratolysis_with_pseudopterygium_associated_with_erythema_elevatum_diutinum L2 - https://linkinghub.elsevier.com/retrieve/pii/S0161-6420(10)00981-4 DB - PRIME DP - Unbound Medicine ER -