Zaimi Yosra, Ayari Myriam, Mensi Asma, Bel Hadj Kacem Linda, Achouri Leila, Bouzrara Meriem, Said Yosra, Mouelhi Leila, Debbeche Radhouane
Department of Gastroenterology, Charles Nicolle Hospital, Tunis, Tunisia.
Department of Pathology, Charles Nicolle Hospital, Tunis, Tunisia.
Appl Clin Genet. 2021 Apr 15;14:235-239. doi: 10.2147/TACG.S306298. eCollection 2021.
H syndrome is an extremely rare autosomal recessive affection caused by biallelic mutations in the SLC29A3 gene encoding the human equilibrative nucleoside transporter hENT3. The hallmark signs are cutaneous consisting of hyperpigmentation and hypertrichosis patches. Besides, associated systemic manifestations are highly various reflecting phenotypic pleiotropism. Herein, we report a first case of pseudo-Meigs' syndrome occurring in a young Tunisian H syndrome diagnosed patient with a novel homozygous frameshift mutation in exon 2 of the SLC29A3 gene: p.S15Pfs*86 inducing a premature stop codon. The patient developed ascites associated with left ovarian mass and she underwent surgery. After tumor resection, ascites disappeared rapidly. Histological examination showed serous cystadenoma of the ovary orienting the diagnosis towards pseudo-Meigs' syndrome.
H综合征是一种极其罕见的常染色体隐性疾病,由编码人类平衡核苷转运蛋白hENT3的SLC29A3基因的双等位基因突变引起。其标志性体征是皮肤出现色素沉着和多毛斑。此外,相关的全身表现多种多样,反映了表型多效性。在此,我们报告首例发生在一名年轻突尼斯H综合征诊断患者中的假性梅格斯综合征,该患者在SLC29A3基因第2外显子存在一种新的纯合移码突变:p.S15Pfs*86,导致提前出现终止密码子。该患者出现腹水并伴有左侧卵巢肿块,随后接受了手术。肿瘤切除后,腹水迅速消失。组织学检查显示为卵巢浆液性囊腺瘤,据此诊断为假性梅格斯综合征。