Attention upturned: Bias toward and away from the affected side of the body and near space in a case of complex regional pain syndrome

Abstract

People with Complex Regional Pain Syndrome (CRPS) following limb injury can show neuropsychological symptoms in the absence of observable brain pathologies. These can include sensory changes, distorted body representation, and inattention to their affected limb and its surrounding space, resembling post-stroke hemispatial neglect. The precise nature and mechanisms of these neuropsychological symptoms are unclear, however insights could be gained by testing for dissociations and associations that have been observed in stroke patients. Drawing from clinical and experimental methods for investigating spatial attention bias and related symptoms in stroke patients, we conducted a detailed investigation of neuropsychological symptoms in a woman with CRPS of her left arm who initially presented to us with pronounced inattention to her affected side. The patient showed visual and tactile neglect and extinction on her affected side on confrontation tests, but no attention deficits on “bedside” tests of neglect. On sensitive computer-based measures, attention biases were found in the patient's body and near space (in Temporal Order Judgements), but not far or imagined space (on the Greyscales task and Mental Number Line Bisection). Unique to the current literature, the patient showed a reversal in her Temporal Order Judgement bias across time, from inattention (first and second session) to hyperattention (third session) to her affected side. In contrast, pain and self-reported body representation distortion were similar across the three sessions. The patient had reduced central and peripheral visual acuity, however these deficits were near symmetrical and therefore could not explain her performance on the visual attention tasks. Given that spatial attention bias has been linked to imbalance in relative activation of the two cerebral hemispheres, we administered a Global-Local processing task to test for hemispheric asymmetry. This revealed no difference in global compared to local interference refuting any hemispheric imbalance. Instead, the patient showed impaired performance (compared to controls) on incongruent trials regardless of trial type, consistent with executive impairment. We conclude that spatial attention bias in CRPS can generalize across different sensory modalities and extend beyond the affected limb to the external space around it, independent of any low-level sensory disturbances. This bias is not necessarily directed away from the affected side or stable over time. People with CRPS can also demonstrate more generalized neuropsychological changes in sensory and executive functions. Our observations refute several existing theories about the mechanisms of attention bias in CRPS, and their relationship to pain, and have potential implications for treatment.