Lamellar Hole-associated Epiretinal Proliferation in choroideremia: a case report.
CHM
Electron microscopy
LHEP
Lamellar Hole-associated Epiretinal Proliferation
Macular hole
OCT
Peeling
Vitrectomy
Journal
International journal of retina and vitreous
ISSN: 2056-9920
Titre abrégé: Int J Retina Vitreous
Pays: England
ID NLM: 101677897
Informations de publication
Date de publication:
19 Oct 2021
19 Oct 2021
Historique:
received:
02
05
2021
accepted:
02
10
2021
entrez:
20
10
2021
pubmed:
21
10
2021
medline:
21
10
2021
Statut:
epublish
Résumé
To report a clinical case of a patient affected with choroideremia (CHM) who underwent macular surgery for a macular hole (MH) with Lamellar Hole-associated Epiretinal Proliferation (LHEP). We have described a 48-year-old male patient affected with CHM who developed MH with LHEP over a 7-year follow-up. The patient was referred to the Regional Center for Hereditary Retinal Degenerations of the Eye Clinic in Florence (Italy) in April 2012. The patient underwent vitrectomy and Inner Limiting Membrane (ILM) and LHEP peeling with fluid-air exchange. Ultra-structural examination of the excised epiretinal proliferation, carried out using electron microscopy, showed dense amorphous material, mainly composed of abundant clusters of fibrous collagens resembling compact fibrous long spacing collagen (FLSC), embedded in native vitreous collagen (NVC) and type IV collagen. No cells were detected in any of the specimens collected. At the 3rd-week postoperative follow-up the macular hole was closed. Macular hole with LHEP can be detected in CHM patients; in our patient the macular hole showed tractional and degenerative features, with good anatomical results after macular surgery.
Sections du résumé
BACKGROUND
BACKGROUND
To report a clinical case of a patient affected with choroideremia (CHM) who underwent macular surgery for a macular hole (MH) with Lamellar Hole-associated Epiretinal Proliferation (LHEP).
CASE PRESENTATION
METHODS
We have described a 48-year-old male patient affected with CHM who developed MH with LHEP over a 7-year follow-up. The patient was referred to the Regional Center for Hereditary Retinal Degenerations of the Eye Clinic in Florence (Italy) in April 2012. The patient underwent vitrectomy and Inner Limiting Membrane (ILM) and LHEP peeling with fluid-air exchange. Ultra-structural examination of the excised epiretinal proliferation, carried out using electron microscopy, showed dense amorphous material, mainly composed of abundant clusters of fibrous collagens resembling compact fibrous long spacing collagen (FLSC), embedded in native vitreous collagen (NVC) and type IV collagen. No cells were detected in any of the specimens collected. At the 3rd-week postoperative follow-up the macular hole was closed.
CONCLUSION
CONCLUSIONS
Macular hole with LHEP can be detected in CHM patients; in our patient the macular hole showed tractional and degenerative features, with good anatomical results after macular surgery.
Identifiants
pubmed: 34666838
doi: 10.1186/s40942-021-00333-5
pii: 10.1186/s40942-021-00333-5
pmc: PMC8527750
doi:
Types de publication
Journal Article
Langues
eng
Pagination
63Informations de copyright
© 2021. The Author(s).
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