TY - JOUR
T1 - Management of acute hydrops with perforation in a patient with keratoconus and cone dystrophy
T2 - Case report and literature review
AU - Yeh, Steven
AU - Smith, Janine A.
PY - 2008/10
Y1 - 2008/10
N2 - Purpose: To describe a patient with cone dystrophy who presented with acute hydrops and perforation, leading to the diagnosis of keratoconus. Methods: Case report and literature review. Results: A 21 -year-old male patient with a history of cone dystrophy presented with a flat anterior chamber, diffuse corneal stromal edema with an intrastromal cleft, and ruptured Descemet membrane, findings consistent with acute hydrops with corneal perforation. After bandage contact lens placement and instillation of a cycloplegic agent, the anterior chamber reformed within 24 hours. Over the next week, conservative management with a bandage lens, pressure patching, topical fluoroquinolone antibiotic, and topical cycloplegic led to the reformation and maintenance of anterior chamber stability. Corneal topography of the unaffected eye showed global corneal thinning and steep sim K readings suspicious for early keratoconus. Conclusions: Although the association between keratoconus and cone dystrophy is extremely rare, our patient's vision-threatening complication of acute hydrops with corneal perforation highlights the importance of corneal evaluation including topography in cone dystrophy. Conservative management was successful in the restoration of anatomic integrity in this situation.
AB - Purpose: To describe a patient with cone dystrophy who presented with acute hydrops and perforation, leading to the diagnosis of keratoconus. Methods: Case report and literature review. Results: A 21 -year-old male patient with a history of cone dystrophy presented with a flat anterior chamber, diffuse corneal stromal edema with an intrastromal cleft, and ruptured Descemet membrane, findings consistent with acute hydrops with corneal perforation. After bandage contact lens placement and instillation of a cycloplegic agent, the anterior chamber reformed within 24 hours. Over the next week, conservative management with a bandage lens, pressure patching, topical fluoroquinolone antibiotic, and topical cycloplegic led to the reformation and maintenance of anterior chamber stability. Corneal topography of the unaffected eye showed global corneal thinning and steep sim K readings suspicious for early keratoconus. Conclusions: Although the association between keratoconus and cone dystrophy is extremely rare, our patient's vision-threatening complication of acute hydrops with corneal perforation highlights the importance of corneal evaluation including topography in cone dystrophy. Conservative management was successful in the restoration of anatomic integrity in this situation.
KW - Cone dystrophy
KW - Corneal perforation
KW - Hydrops
KW - Keratoconus
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U2 - 10.1097/ICO.0b013e31817618c2
DO - 10.1097/ICO.0b013e31817618c2
M3 - Article
C2 - 18812772
AN - SCOPUS:58149172121
SN - 0277-3740
VL - 27
SP - 1062
EP - 1065
JO - Cornea
JF - Cornea
IS - 9
ER -