Shan L, Nakamura Y, Murakami M, Nakamura M, Naito A, Kawahara K, Utsunomiya H, Mori I, Kakudo K
Department of Pathology, Wakayama Medical College.
Jpn J Cancer Res. 1999 Sep;90(9):965-9. doi: 10.1111/j.1349-7006.1999.tb00842.x.
It is difficult to differentiate between parathyroid neoplasia and hyperplasia. In an attempt to elucidate the clonality of uremic parathyroid hyperplasia and the molecular genetic abnormalities accounting for clonal emergence, we analyzed 20 cases of uremic parathyroid hyperplasia. Clonalities were determined using the X-chromosome-linked human androgen receptor (HUMARA) gene and the phosphoglycerate kinase (PGK) gene, and multiple endocrine neoplasia type 1 (MEN1) gene abnormality was analyzed by studying loss of heterozygosity (LOH) in 11q13 and somatic mutations in the MEN1 gene. As a positive control, a case of MEN1 with Zollinger-Ellison syndrome was analyzed simultaneously. Our analysis revealed that a majority (75%) of the uremic parathyroid hyperplasia tissues, including an autograft with recurrent hyperparathyroidism, was of monoclonal origin. Clonality did not correlate with serum carboxyl-terminal parathyroid hormone (C-PTH) level, calcium level, hemodialytic duration, gland weight or pathological features. Neither LOH in 11q13 nor somatic mutation in the MEN1 gene was detected. For the MEN1 case, a germline mutation (W198X) was detected in exon 3. We concluded that a majority of the uremic parathyroid hyperplasia cases was in fact monoclonal neoplasia. MEN1 gene abnormality played a minor role, if any, in the clonal emergence in uremic parathyroid hyperplasia.
甲状旁腺肿瘤和增生难以区分。为了阐明尿毒症性甲状旁腺增生的克隆性以及导致克隆出现的分子遗传异常,我们分析了20例尿毒症性甲状旁腺增生病例。使用X染色体连锁的人类雄激素受体(HUMARA)基因和磷酸甘油酸激酶(PGK)基因确定克隆性,并通过研究11q13的杂合性缺失(LOH)和MEN1基因的体细胞突变来分析多发性内分泌肿瘤1型(MEN1)基因异常。作为阳性对照,同时分析了1例患有卓艾综合征的MEN1病例。我们的分析显示,大多数(75%)尿毒症性甲状旁腺增生组织,包括伴有复发性甲状旁腺功能亢进的自体移植组织,起源于单克隆。克隆性与血清羧基末端甲状旁腺激素(C-PTH)水平、钙水平、血液透析持续时间、腺体重量或病理特征无关。未检测到11q13的LOH或MEN1基因的体细胞突变。对于MEN1病例,在外显子3中检测到一个种系突变(W198X)。我们得出结论,大多数尿毒症性甲状旁腺增生病例实际上是单克隆肿瘤。MEN1基因异常在尿毒症性甲状旁腺增生的克隆出现中即使有作用也很小。