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| Item Type: | Article |
|---|---|
| Title: | Butyrylcholinesterase and 24 h-urinary copper excretion as compliance assessment in long-term treated Wilson's disease |
| Creators Name: | Hefter, H., Novak, M., Rosenthal, D., Meuth, S.G., Luedde, T., Albrecht, P., Hartmann, C.J. and Samadzadeh, S. |
| Abstract: | BACKGROUND AND AIM OF THE STUDY: Compliance is the most challenging aspect of long-term therapy in Wilson's disease (WD). Evidence is presented that butyrylcholinesterase (CHE) can be used as a sensitive biomarker to detect compliance problems in long-term treated WD-patients. METHODS: For the present retrospective, monocentric study demographical and treatment related data of 108 WD-patients (who had been treated at the Clinic of Neurology of the university hospital in Düsseldorf (Germany) between 2/2005 and 1/2021) were extracted from the charts. These patients underwent 2003 therapy control visits. The present study focuses on the analysis of three parameters of copper metabolism (serum levels of ceruloplasmin (CER-S), copper (CU-S) and the 24 h-urinary copper excretion (24 h-UCU)) and the serum levels of CHE (CHE-S). A patient was classified to be non-compliant when in his charts at least 8 24 h-UCU-values were found, and all his 24 h-UCU-values were larger than 60 μg/d (N-COM8-group). A patient was classified to be compliant when at least one of at least 8 24 h-UCU-values was lower than or equal to 60 μg/d (COM8-group). RESULTS: CHE-S was significantly (p < 0.05) different between thus defined compliant or non-compliant patients. Neither CU-S nor CER-S nor calculated free (non-ceruloplasmin-bound) serum copper levels (NCC-S) were significantly different between the NCOM8- and COM8-group. Analysis of the area under the curve and sensitivity and specificity by means of ROC-curves underlined the sensitivity of CHE-S in contrast to the insensitivity of CU-S and CER-S to detect patients who had been classified as compliant/non-compliant on the basis of their 24 h-UCU-values. CONCLUSION: When compliance of WD-patients is classified on the basis of their 24 h-urinary copper excretion CHE-S is more sensitive to detect problems of non-compliance than serum levels of copper, of non-ceruloplasmin bound free copper or ceruloplasmin. Therefore, CHE-S may be used as an easy to determine further biomarker for compliance assessment in long-term treatment of WD in addition to 24 h-UCU. |
| Keywords: | Wilson’s Disease, Butyrylcholinesterase, 24 H-Urinary Copper Excretion, Compliance, Long-Term Treatment |
| Source: | Frontiers in Neurology |
| ISSN: | 1664-2295 |
| Publisher: | Frontiers Media SA |
| Volume: | 16 |
| Page Range: | 1553573 |
| Date: | 19 May 2025 |
| Official Publication: | https://doi.org/10.3389/fneur.2025.1553573 |
| PubMed: | View item in PubMed |
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