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A Patient with Keratoconus, Nanophthalmos, Lipodermoids, and Pigmentary Retinopathy.

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  • المؤلفون: Sammouh FK;Sammouh FK; Baban TA; Baban TA; Warrak EL; Warrak EL
  • المصدر:
    Ophthalmic genetics [Ophthalmic Genet] 2016 Jun; Vol. 37 (2), pp. 228-32. Date of Electronic Publication: 2016 Jan 15.
  • نوع النشر :
    Case Reports; Journal Article
  • اللغة:
    English
  • معلومة اضافية
    • المصدر:
      Publisher: Informa Healthcare Country of Publication: England NLM ID: 9436057 Publication Model: Print-Electronic Cited Medium: Internet ISSN: 1744-5094 (Electronic) Linking ISSN: 13816810 NLM ISO Abbreviation: Ophthalmic Genet Subsets: MEDLINE
    • بيانات النشر:
      Publication: London : Informa Healthcare
      Original Publication: Buren, The Netherlands : Aeolus Press, c1994-
    • الموضوع:
      Keratoconus/*complications ; Lipoma/*complications ; Microphthalmos/*complications ; Orbital Neoplasms/*complications ; Retinitis Pigmentosa/*complications; Adult ; Corneal Topography ; Humans ; Hyperopia/complications ; Keratoconus/diagnosis ; Lipoma/pathology ; Male ; Microphthalmos/diagnosis ; Ophthalmoscopy ; Orbital Neoplasms/pathology ; Retinitis Pigmentosa/diagnosis ; Tomography, Optical Coherence ; Visual Acuity/physiology
    • نبذة مختصرة :
      A 44-year-old male with no pertinent history other than poor vision for more than 25 years was examined. Best corrected visual acuity was 20/80 OD [MR: +14.25 +1.00 × 15°] and 20/200 OS [MR: +15.00 +1.50 × 175°]. Significant limitation in ocular movements and the presence of an orbital lipodermoid in the infero-temporal aspect of each eye were noted. Forced duction test was positive for the same directions of limitation indicating possible extraocular muscle fibrosis. Ophthalmoscopy was remarkable for the presence of peripheral bony spicules. Corneal topography was compatible with keratoconus (Kmax = 55.04D OD and 52.87D OS). A-scan revealed axial lengths of 16.96 mm OD and 16.32 mm OS, compatible with a diagnosis of nanophthalmos. OCT revealed diffuse macular thickening for both eyes with foveal thickness of 350 µm OD and 353 µm OS. Over the next 12 years the patient had stable visual acuity, manifest refractions and anterior segment examination. Ophthalmoscopy revealed only minimal progression of pigmentary changes. We report the first case of these simultaneous multiple findings which may refer to a possible syndromic association of congenital or early childhood onset.
    • Contributed Indexing:
      Keywords: High hyperopia; keratoconus; lipodermoids; muscle fibrosis and retinal dystrophy; nanophthalmos
    • الموضوع:
      Date Created: 20160116 Date Completed: 20171106 Latest Revision: 20220331
    • الموضوع:
      20250114
    • الرقم المعرف:
      10.3109/13816810.2015.1028648
    • الرقم المعرف:
      26771301