Abu-Rumeileh Samir, Barschke Peggy, Oeckl Patrick, Baiardi Simone, Mammana Angela, Mastrangelo Andrea, Al Shweiki Mhd Rami, Steinacker Petra, Ladogana Anna, Capellari Sabina, Otto Markus, Parchi Piero
Department of Neurology, Martin-Luther-University Halle-Wittenberg, 06120 Halle (Saale), Germany.
Department of Neurology, Ulm University Hospital, 89081 Ulm, Germany.
Int J Mol Sci. 2022 Feb 12;23(4):2051. doi: 10.3390/ijms23042051.
Proenkephalin (PENK) and prodynorphin (PDYN) are endogenous opioid peptides mainly produced in the striatum and, to a lesser extent, in the cerebral cortex. Dysregulated metabolism and altered cerebrospinal fluid (CSF) levels of PENK and PDYN have been described in several neurodegenerative diseases. However, no study to date investigated these peptides in the CSF of sporadic Creutzfeldt-Jakob disease (sCJD). Using liquid chromatography-multiple reaction monitoring mass spectrometry, we evaluated the CSF PDYN- and PENK-derived peptide levels in 25 controls and 63 patients with sCJD belonging to the most prevalent molecular subtypes (MM(V)1, VV2 and MV2K). One of the PENK-derived peptides was significantly decreased in each sCJD subtype compared to the controls without a difference among subtypes. Conversely, PDYN-derived peptides were selectively decreased in the CSF of sCJD MV2K, a subtype with a more widespread overall pathology compared to the sCJD MM(V)1 and the VV2 subtypes, which we confirmed by semiquantitative analysis of cortical and striatal neuronal loss and astrocytosis. In sCJD CSF PENK and PDYN were associated with CSF biomarkers of neurodegeneration but not with clinical variables and showed a poor diagnostic performance. CSF PDYN and PENK-derived peptides had no significant diagnostic and prognostic values in sCJD; however, the distinct marker levels between molecular subtypes might help to better understand the basis of phenotypic heterogeneity determined by divergent neuronal targeting.
前脑啡肽原(PENK)和前强啡肽原(PDYN)是主要在纹状体产生的内源性阿片肽,在大脑皮层产生的较少。在几种神经退行性疾病中,已描述了PENK和PDYN代谢失调及脑脊液(CSF)水平改变的情况。然而,迄今为止尚无研究在散发性克雅氏病(sCJD)患者的脑脊液中对这些肽进行研究。我们使用液相色谱-多反应监测质谱法,评估了25名对照者和63例属于最常见分子亚型(MM(V)1、VV2和MV2K)的sCJD患者脑脊液中源自PDYN和PENK的肽水平。与对照组相比,每种sCJD亚型中一种源自PENK的肽均显著降低,各亚型之间无差异。相反,在sCJD MV2K患者的脑脊液中,源自PDYN的肽选择性降低,与sCJD MM(V)1和VV2亚型相比,该亚型具有更广泛的整体病理改变,我们通过对皮质和纹状体神经元丢失及星形细胞增多的半定量分析证实了这一点。在sCJD患者的脑脊液中,PENK和PDYN与神经退行性变的脑脊液生物标志物相关,但与临床变量无关,且诊断性能较差。脑脊液中源自PDYN和PENK的肽在sCJD中无显著诊断和预后价值;然而,分子亚型之间不同的标志物水平可能有助于更好地理解由不同神经元靶向决定的表型异质性的基础。