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伴有盆腔尤因肉瘤的前蛋白转化酶1/3缺乏症

PROPROTEIN CONVERTASE 1/3 DEFICIENCY WITH PELVIC EWING SARCOMA.

作者信息

Oral H, Guven D C, Özdemir D Ateş, Usubütün A, Gonc N, Arik Z

机构信息

Department of Internal Medicine, Hacettepe University Faculty of Medicine, Ankara, Turkey.

Department of Internal Medicine, Division of Oncology, Hacettepe University Faculty of Medicine, Ankara, Turkey.

出版信息

Acta Endocrinol (Buchar). 2022 Oct-Dec;18(4):508-511. doi: 10.4183/aeb.2022.508.

Abstract

Proprotein convertase 1/3 (PC 1/3) deficiency is a rare, autosomal recessive disorder caused by mutations in the PCSK1 gene. The disease is characterized by early-onset chronic diarrhea/malabsorption, followed by severe obesity and hormonal deficiencies such as hypocortisolism, hypothyroidism, diabetes insipidus, hypogonadism, growth deficiency, and diabetes mellitus. Ewing's sarcoma is a rare tumor, usually of small dimensions of neuroectodermal origin that is difficult to distinguish pathologically from a primitive neuroectodermal tumor. A 22-year-old female patient with PC 1/3 deficiency was admitted to our clinic with recurrent urinary tract infections. Magnetic resonance imaging (MRI) revealed an 11x12 cm pelvic mass displacing the uterus. A core-needle biopsy was performed on the pelvic mass. As a result of the pathological evaluation, ıt was diagnosed with pelvic Ewing's sarcoma. The patient was started on the VAC-IE chemotherapy protocol. We report a case of pelvic Ewing's sarcoma in a patient with PC 1/3 deficiency. Further research is needed to assess malignancy risk in metabolic disorders including very rare disorders like PC 1/3 deficiency.

摘要

前蛋白转化酶1/3(PC 1/3)缺乏症是一种罕见的常染色体隐性疾病,由PCSK1基因突变引起。该疾病的特征是早发性慢性腹泻/吸收不良,随后出现严重肥胖和激素缺乏,如皮质醇减少症、甲状腺功能减退、尿崩症、性腺功能减退、生长发育迟缓以及糖尿病。尤因肉瘤是一种罕见肿瘤,通常体积较小,起源于神经外胚层,在病理上难以与原始神经外胚层肿瘤区分开来。一名患有PC 1/3缺乏症的22岁女性患者因反复尿路感染入住我院。磁共振成像(MRI)显示盆腔有一个11×12厘米的肿块,使子宫移位。对盆腔肿块进行了粗针活检。病理评估结果显示,该患者被诊断为盆腔尤因肉瘤。患者开始接受VAC-IE化疗方案治疗。我们报告了一例患有PC 1/3缺乏症的患者发生盆腔尤因肉瘤的病例。需要进一步研究来评估包括PC 1/3缺乏症等非常罕见疾病在内的代谢紊乱中的恶性肿瘤风险。

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