{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,3,26]],"date-time":"2026-03-26T22:01:21Z","timestamp":1774562481039,"version":"3.50.1"},"reference-count":56,"publisher":"Georg Thieme Verlag KG","issue":"10","content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Klin Monbl Augenheilkd"],"published-print":{"date-parts":[[2023,10]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:p>\n          Background Achromatopsia (ACHM) as a hereditary cone disease might manifest in a stationary and progressive manner. The proper clinical and genetic diagnosis may allow an individual\n          prognosis, accurate genetic counselling, and the optimal choice of low vision aids. The primary aim of the study was to determine the spectrum of clinical and genetic diagnostics required to\n          characterize the ACHM.<\/jats:p><jats:p>\n          Methods A retrospective analysis was performed in 8 patients from non-related families (5 \u2640,3 \u2642); age at diagnosis: 3\u200a\u2013\u200a56\u2009y, mean 18.13 (SD\u2009\u00b1\u200918.22). Clinical phenotyping, supported\n          by colour vision test, fundus photography-, autofluorescence- (FAF), infra-red- (IR), OCT imaging and electroretinography provided information on the current status and the course of the\n          disease over the years. In addition, genetic examinations were performed with ACHM relevant testing (CNGA3, CNGB3, GNAT2, PDE6C, PDE6H and the transcription factor ATF6).<\/jats:p><jats:p>\n          Results All patients suffered photophobia and reduced visual acuity (mean: 0.16 [SD \u00b1\u20090.08]). Nystagmus was identified in 7 from 8 subjects and in one patient a head-turn right\n          helped to reduce the nystagmus amplitude. Colour vision testing confirmed complete achromatopsia in 7 out of 8 patients. Electrophysiology found severely reduced photopic- but also scotopic\n          responses. Thinning and interruption of the inner segment ellipsoid (ISe) line within the macula but also FAF- and IR abnormalities in the fovea and\/or parafovea were characteristic in all\n          ACHM patients. Identification of pathogenic mutations in 7 patients helped to confirm the diagnosis of ACHM (3 adults, 4 children; 3 \u2640 and 4 \u2642). Achromatopsia was linked to CNGA3 (2\n          \u2640, 1 \u2642) and CNGB3 variants (2 \u2640, 3 \u2642). The youngest patient (\u2640, 10\u2009y) had 3 different CNGB3 variants on different alleles. In a patient (\u2642, 29\u2009y) carrying 2 pathogenic\n          digenic-triallelic CNGA3- and CNGB3-mutations, a severe progression of ISe discontinuity to coloboma-like macular atrophy was observed during the 12-year follow-up. The oldest\n          female (67\u2009y) showed a compound homozygous CNGA3- and heterozygous CNGB3-, as well as a heterozygous GUCY2D variants. The destruction of her ISe line was significantly\n          enlarged and represented a progressive cone-rod phenotype in comparison to other ACHM patients. In a patient (\u2642, 45\u2009y) carrying a pathogenic CNGB3 and USH2 mutation, a severe\n          macular oedema and a rod-cone phenotype was observed. In addition, two variants in C2ORF71 considered as VOS were found. One patient showed the rare ATF6 mutation, where a\n          severe coloboma-like macular atrophy was observed on the left eye as early as at the age of three years.<\/jats:p><jats:p>\n          Conclusion Combining multimodal ophthalmological diagnostics and molecular genetics when evaluating patients with ACHM helps in characterizing the disease and associated modifiers,\n          and is therefore strongly recommended for such patients.<\/jats:p>","DOI":"10.1055\/a-2176-4233","type":"journal-article","created":{"date-parts":[[2023,9,15]],"date-time":"2023-09-15T22:51:33Z","timestamp":1694818293000},"page":"1158-1173","source":"Crossref","is-referenced-by-count":3,"title":["Analysis of Suspected Achromatopsia by Multimodal Diagnostic Testing"],"prefix":"10.1055","volume":"240","author":[{"given":"Sylvia A.","family":"Kugler","sequence":"additional","affiliation":[{"name":"Department of Ophthalmology, Cantonal Hospital St.\u00a0Gallen, Switzerland"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Christophe","family":"Valmaggia","sequence":"additional","affiliation":[{"name":"Department of Ophthalmology, Cantonal Hospital St.\u00a0Gallen, Switzerland"},{"name":"Department of Ophthalmology, University of Z\u00fcrich, Switzerland"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Veit","family":"Sturm","sequence":"additional","affiliation":[{"name":"Department of Ophthalmology, University of Z\u00fcrich, Switzerland"},{"name":"Ophthalmology, Eye Center Rosengarten, Arbon, Switzerland"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Daniel F.","family":"Schorderet","sequence":"additional","affiliation":[{"name":"Faculty of Biology and Medicine, University of Lausanne and Faculty of Life Sciences, \u00c9cole polytechnique f\u00e9d\u00e9rale de Lausanne, Switzerland"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Margarita G.","family":"Todorova","sequence":"additional","affiliation":[{"name":"Department of Ophthalmology, Cantonal Hospital St.\u00a0Gallen, Switzerland"},{"name":"Department of Ophthalmology, University of Z\u00fcrich, Switzerland"},{"name":"Department of Ophthalmology, University Hospital Basel, Switzerland"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"194","published-online":{"date-parts":[[2023,10,23]]},"reference":[{"key":"ref1","doi-asserted-by":"publisher","first-page":"437","DOI":"10.1001\/jamaophthalmol.2013.7987","article-title":"Spectral-domain optical coherence tomography staging and autofluorescence imaging in achromatopsia","volume":"132","author":"J P Greenberg","year":"2014","journal-title":"JAMA Ophthalmol"},{"key":"ref2","doi-asserted-by":"publisher","first-page":"291","DOI":"10.1136\/bjo.2003.027102","article-title":"The cone dysfunction syndromes","volume":"88","author":"M Michaelides","year":"2004","journal-title":"Br J 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