Aniridic Keratopathy: Conservative Approaches

García, José Santiago López; Lozano, Isabel

doi:10.1007/978-3-319-19779-1_7

Aniridia 2015

DOI: 10.1007/978-3-319-19779-1_7

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Aniridic Keratopathy: Conservative Approaches

José Santiago López García

Isabel Lozano

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2018

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“…16 Management of ARK depends on the severity of the involvement of the ocular surface and can include preservativefree lubricants, autologous serum, and amniotic membrane transplantation, but in severe ARK, a source of limbal stem cells is needed. 17 Limbo-keratoplasty has been shown to be a better treatment option in ARK than penetrating keratoplasty. The latter has a limited prognosis in aniridia patients because of the stem cell deficiency and altered healing responses.…”

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Altered Signaling Pathways in Aniridia-Related Keratopathy

Vicente

Byström

Domellöf

2018

Invest. Ophthalmol. Vis. Sci.

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PURPOSE. To study the Notch1, Wnt/beta-catenin, sonic hedgehog (SHH), and mammalian target of rapamycin (mTOR) cell signaling pathways in naïve and surgically treated corneas of aniridia cases with advanced aniridia-related keratopathy (ARK). METHODS.Two naïve corneal buttons from patients with advanced ARK submitted to penetrating keratoplasty for the first time, one corneal button from an ARK patient that had undergone a keratolimbal allograft (KLAL), two corneal buttons from ARK patients who had previously undergone centered or decentered transplantation, and two adult healthy control corneas were processed for immunohistochemistry in this descriptive study. Antibodies specific against elements of the Notch1 (Notch1; Dlk1; Numb), Wnt/beta-catenin (Wnt5a; Wnt7a; beta-catenin), SHH (glioma-associated oncogene homolog [Gli1]; Hes1), and mTOR (mTOR1; ribosomal protein S6 [rpS6]) signaling pathways were used as well as antibodies against PAX6 and keratin 13 (Krt13). RESULTS.All ARK corneas presented signs of conjunctivalization and analogous signaling pathway changes in the subepithelial pannus and epithelium, with decreased detection of the Notch1 signaling pathway and an increased presence of the Notch1 inhibitors Numb and Dlk1. Increased detections of Wnt/beta-catenin (enhanced presence of Wnt5a, Wnt7a, and beta-catenin), SHH (detection of Gli1 and Hes1), and mTOR (identification of mTOR and rpS6) signaling pathways were found in the subepithelial pannus and epithelium of all ARK corneas, when compared with normal controls. CONCLUSIONS.The similarity in pathway alterations found in all ARK corneas, irrespective of limbal stem cell transplantation, further supports the discussion on the role of host-specific factors and limbal stem cell deficiency in ARK.

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confidence: 99%

Altered Signaling Pathways in Aniridia-Related Keratopathy

Vicente

Byström

Domellöf

2018

Invest. Ophthalmol. Vis. Sci.

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Aniridic Keratopathy: Conservative Approaches

Cited by 1 publication

References 26 publications

Altered Signaling Pathways in Aniridia-Related Keratopathy

Altered Signaling Pathways in Aniridia-Related Keratopathy

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