A rare case of congenital pupillary abnormality: a case report.
Case report
Microcoria
Pupillary abnormality
Pupilloplasty
Journal
BMC ophthalmology
ISSN: 1471-2415
Titre abrégé: BMC Ophthalmol
Pays: England
ID NLM: 100967802
Informations de publication
Date de publication:
02 May 2022
02 May 2022
Historique:
received:
13
02
2022
accepted:
25
04
2022
entrez:
3
5
2022
pubmed:
4
5
2022
medline:
6
5
2022
Statut:
epublish
Résumé
Congenital anomalies of the pupil are quite varied, including abnormal size, shape, color, response to stimulus, and function. We are here reporting an unusual case presented with the absence of pupillary opening with folds of iris tissue at the center. Only an extremely small pupil (diameter < 0.5 mm) could be observed during the operation. A 15-year-old male patient visited our outpatient clinic due to vision difficulty in his right eye for more than ten years. The best-corrected visual acuity was 2.0 logMAR and 0 logMAR for the right and left eye, respectively. There were amblyopia, astigmatism and constant exotropia in his right eye. Ophthalmic examination of the right eye showed flat iris root, minimal iris pigmentation, and the pupil area was entirely covered by iris tissue. Lens status and fundus evaluation could not be commented. The left eye was found to be within normal limit. Based on ophthalmic examination, the admission diagnosis was given as acorea. Pupilloplasty was performed on the right eye due to the situation that the iris tissue blocked the visual axis, which led to visual impairment and stimulus deprivation amblyopia. However, an extremely small pupil at the center of his pupillary area was observed during the operation. The postoperative course was favorable, and a normal pupil was secured. Hospital discharge diagnosis was given as microcoria, and amblyopia treatment was followed. We report a rare case of congenital pupillary abnormality. The further diagnosis was given as microcoria, which should be differentiated from acorea. For this kind of pupil disorder which blocks the visual axis, early diagnosis and treatment can help prevent the development of stimulus deprivation amblyopia.
Sections du résumé
BACKGROUND
BACKGROUND
Congenital anomalies of the pupil are quite varied, including abnormal size, shape, color, response to stimulus, and function. We are here reporting an unusual case presented with the absence of pupillary opening with folds of iris tissue at the center. Only an extremely small pupil (diameter < 0.5 mm) could be observed during the operation.
CASE PRESENTATION
METHODS
A 15-year-old male patient visited our outpatient clinic due to vision difficulty in his right eye for more than ten years. The best-corrected visual acuity was 2.0 logMAR and 0 logMAR for the right and left eye, respectively. There were amblyopia, astigmatism and constant exotropia in his right eye. Ophthalmic examination of the right eye showed flat iris root, minimal iris pigmentation, and the pupil area was entirely covered by iris tissue. Lens status and fundus evaluation could not be commented. The left eye was found to be within normal limit. Based on ophthalmic examination, the admission diagnosis was given as acorea. Pupilloplasty was performed on the right eye due to the situation that the iris tissue blocked the visual axis, which led to visual impairment and stimulus deprivation amblyopia. However, an extremely small pupil at the center of his pupillary area was observed during the operation. The postoperative course was favorable, and a normal pupil was secured. Hospital discharge diagnosis was given as microcoria, and amblyopia treatment was followed.
CONCLUSIONS
CONCLUSIONS
We report a rare case of congenital pupillary abnormality. The further diagnosis was given as microcoria, which should be differentiated from acorea. For this kind of pupil disorder which blocks the visual axis, early diagnosis and treatment can help prevent the development of stimulus deprivation amblyopia.
Identifiants
pubmed: 35501768
doi: 10.1186/s12886-022-02422-x
pii: 10.1186/s12886-022-02422-x
pmc: PMC9063307
doi:
Types de publication
Case Reports
Journal Article
Langues
eng
Sous-ensembles de citation
IM
Pagination
201Informations de copyright
© 2022. The Author(s).
Références
Am J Hum Genet. 2015 Apr 2;96(4):631-9
pubmed: 25772937
Br J Ophthalmol. 1923 Sep;7(9):401-19
pubmed: 18168325
Br J Ophthalmol. 2013 Sep;97(9):1155-60
pubmed: 23832966
Arq Bras Oftalmol. 2019 Aug 29;82(5):425-428
pubmed: 31482961
Indian J Ophthalmol. 2018 Mar;66(3):450
pubmed: 29480264
Genes (Basel). 2021 Apr 22;12(5):
pubmed: 33922078
Neurol Clin. 2010 Aug;28(3):657-77
pubmed: 20637995
Ophthalmol Glaucoma. 2018 Sep - Oct;1(2):145-147
pubmed: 32672565
Eur J Med Genet. 2020 May;63(5):103918
pubmed: 32200002