AbstractWe report findings of monocular and binocular assessments of colour vision and thresholds for rod‐ and cone‐mediated sensitivity in two young patients (P1 and P2) diagnosed with monocular dyschromatopsia. Altered, monocular, colour perception with no clear diagnosis was reported in each patient based on results from a number of clinical visual assessment tests carried out over several years. Electrodiagnostic tests also revealed reduced Yellow / Blue (YB) chromatic sensitivity in P1. Both patients exhibited unexpected binocular interactions of colour signals. This abstract focuses mostly on the results obtained in P1, but similar results using the same visual assessment tests will also be presented for P2.P1 experienced visual discomfort, often manifested as differences between monocular and binocular viewing, noise‐like appearance of uniform fields and colours over the field, when viewed monocularly with either eye. As a result of these reports, we re‐examined P1's visual deficits using the Colour Assessment and Diagnosis (CAD) and the Flicker‐Plus (FP) tests (http://www.city-occupational.co.uk/). The FP test measures rod‐ and cone‐mediated rapid flicker sensitivity centrally (along the direction of gaze) and at 6 deg eccentricity in each of the four quadrants.Colour Vision. P1 shows significant loss of YB colour vision when tested monocularly in the right eye, but normal red‐green (RG) and YB colour vision in the left eye. When tested binocularly, P1 continues to show the same loss of YB colour vision observed when using only the right eye. These results were unexpected since P1 shows ‘normal’ binocular summation for both colour and flicker sensitivity, even when the thresholds are above the normal range.Rod‐ / Cone‐sensitivity. The patient's cone‐mediated, flicker sensitivity at all five locations investigated (under binocular viewing) was normal (with flicker detection thresholds, often under 2%), but only for the cone‐enhanced condition. When tested with rod‐enhanced stimuli, P1 shows much reduced sensitivity with larger thresholds in the periphery, but only in binocular viewing. Such responses have not been observed previously.In conclusion, P1 shows loss of YB colour vision in the right eye and also significant loss of rod‐mediated sensitivity at each of the five locations investigated. The patient's cone mediated thresholds, at every location tested, were consistently below the mean expected for normal trichromats, both binocularly and with each eye. The YB colour vision measured binocularly remained severely abnormal with thresholds similar to the right eye, in spite of normal colour vision in the left eye. Preliminary observations also suggest that the patient's colour and rapid flicker sensitivity can be affected by fatigue and other factors.Although patient, P2, varied significantly to P1 in monocular thresholds, a similar binocular, inhibitory interaction between the two eyes was observed when processing colour signals. These observations and the unusual interactions between signals derived from the two eyes point towards abnormal central processing of colour signals.