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左甲状腺素治疗的先天性甲状腺功能减退症伴疲劳患者肉碱 - 酰基肉碱谱的改变:一项来自孟加拉国的基于液相色谱 - 串联质谱的研究

Altered carnitine-acylcarnitine profiles in levothyroxine-treated congenital hypothyroid patients with fatigue: An LC-MS/MS-based study from Bangladesh.

作者信息

Begum Mst Noorjahan, Sarker Suprovath Kumar, Islam Md Tarikul, Bhuyan Golam Sarower, Mahtarin Rumana, Patwary Mohammad Hridoy, Konika Tasnia Kawsar, Qadri Syeda Kashfi, Ahmed Tasnuva, Banu Hurjahan, Sultana Nusrat, Rahat Asifuzzaman, Shyamaly Kohinoor Jahan, Bhuiyan Taufiqur Rahman, Hasan Mizanul, Hasanat Mohammad A, Sajib Abu A, Islam Abul B M M K, Mannoor Kaiissar, Akhteruzzaman Sharif, Qadri Firdausi

机构信息

Institute for Developing Science and Health Initiatives (ideSHi), ECB Chattar, Mirpur, Dhaka, Bangladesh.

Department of Genetic Engineering & Biotechnology, University of Dhaka, Dhaka, Bangladesh.

出版信息

PLoS One. 2025 Sep 25;20(9):e0331474. doi: 10.1371/journal.pone.0331474. eCollection 2025.

Abstract

Congenital hypothyroidism (CH), characterized by insufficient thyroid hormone production at birth, is frequently associated with fatigue, particularly in cases with delayed diagnosis. This study employed liquid chromatography-tandem mass spectrometry (LC-MS/MS) to profile carnitine and acylcarnitines in late-diagnosed congenital hypothyroid patients receiving levothyroxine (LT4) therapy, with the aim of identifying metabolic alterations that may be associated with fatigue symptoms. A total of 56 late-diagnosed congenital hypothyroid patients and 107 age-, sex-, and BMI-matched healthy controls were enrolled. Blood samples were collected in EDTA tubes and as dried blood spots (DBS) on Whatman® 903 filter paper. LC-MS/MS was used to quantify free carnitine and 28 acylcarnitines, and plasma triglyceride (TG) levels were measured using a biochemical analyzer. Compared to healthy controls, congenital hypothyroid patients showed higher mean (±SD) concentrations of free carnitine (45.38 ± 12.61 vs. 41.54 ± 9.85 µmol/L; P = 0.049), total carnitines (67.33 ± 18.27 vs. 62.51 ± 14.13 µmol/L), and total acylcarnitines (21.95 ± 7.66 vs. 20.96 ± 5.61 µmol/L), although only free carnitine levels were statistically significant. Long-chain acylcarnitines were significantly lower in congenital hypothyroid patients (2.67 ± 0.87 µmol/L) compared to controls (3.15 ± 0.93 µmol/L; P = 0.0014). The β-oxidation ratio C0/(C16 + C18), a proxy for Carnitine Palmitoyltransferase I (CPT-I) activity, was significantly elevated in patients compared to healthy controls (34.55 ± 14.88 vs. 25.73 ± 6.87; P < 0.0001). Plasma TG levels were also significantly higher in patients (88.92 ± 59.54 mg/dL) than in controls (58.33 ± 15.79 mg/dL; P = 0.02). Metabolic profiling in congenital hypothyroid patients revealed impaired long-chain fatty acid oxidation and elevated triglyceride levels. These metabolic changes may contribute to fatigue symptoms and are potentially associated with reduced CPT-I activity, which is essential for mitochondrial β-oxidation. Additionally, mutations in the TPO and TSHR genes identified within this cohort may be linked to the observed metabolic alterations. Collectively, these findings suggest a possible interplay between genetic variants, disrupted lipid metabolism, and clinical features of congenital hypothyroidism.

摘要

先天性甲状腺功能减退症(CH)的特征是出生时甲状腺激素分泌不足,常伴有疲劳,尤其是在诊断延迟的病例中。本研究采用液相色谱 - 串联质谱法(LC-MS/MS)对接受左甲状腺素(LT4)治疗的诊断延迟的先天性甲状腺功能减退患者的肉碱和酰基肉碱进行分析,目的是确定可能与疲劳症状相关的代谢改变。共纳入56例诊断延迟的先天性甲状腺功能减退患者和107例年龄、性别和BMI匹配的健康对照。血液样本采集于EDTA管中,并作为干血斑(DBS)采集在Whatman® 903滤纸上。LC-MS/MS用于定量游离肉碱和28种酰基肉碱,血浆甘油三酯(TG)水平使用生化分析仪测量。与健康对照相比,先天性甲状腺功能减退患者的游离肉碱平均(±标准差)浓度更高(45.38±12.61 vs. 41.54±9.85 μmol/L;P = 0.049)、总肉碱(67.33±18.27 vs. 62.51±14.13 μmol/L)和总酰基肉碱(21.95±7.66 vs. 20.96±5.61 μmol/L),尽管只有游离肉碱水平具有统计学意义。先天性甲状腺功能减退患者的长链酰基肉碱显著低于对照组(2.67±0.87 μmol/L)(3.15±0.93 μmol/L;P = 0.0014)。β-氧化比率C0/(C16 + C18),作为肉碱棕榈酰转移酶I(CPT-I)活性的指标,与健康对照相比在患者中显著升高(34.55±14.88 vs. 25.73±6.87;P < 0.0001)。患者的血浆TG水平也显著高于对照组(88.92±59.54 mg/dL)(58.33±15.79 mg/dL;P = 0.02)。先天性甲状腺功能减退患者的代谢谱分析显示长链脂肪酸氧化受损和甘油三酯水平升高。这些代谢变化可能导致疲劳症状,并且可能与CPT-I活性降低有关,CPT-I活性对于线粒体β-氧化至关重要。此外,在该队列中鉴定出的TPO和TSHR基因的突变可能与观察到的代谢改变有关。总体而言,这些发现表明遗传变异、脂质代谢紊乱和先天性甲状腺功能减退症的临床特征之间可能存在相互作用。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/fc5a/12463203/9f8eb4d3cba4/pone.0331474.g001.jpg

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