Perry Ariane, Douillard Claire, Jonca Frederic, Glowacki Francois, Leroy Xavier, Caveriviere Paul, Hubert Aurélie, Labrune Philippe
APHP, Hôpitaux Universitaires Paris Sud, Hôpital Antoine Béclère, Centre de référence des maladies héréditaires du métabolisme hépatique Clamart France.
Lille University Hospital, Hôpital Jeanne de Flandres, Centre de référence des maladies héréditaires du métabolisme Lille France.
JIMD Rep. 2020 Jan 29;52(1):17-22. doi: 10.1002/jmd2.12096. eCollection 2020 Mar.
Glycogen storage disease type Ia (GSD Ia) is a rare metabolic disease due to glucose-6-phosphatase deficiency. Chronic kidney disease is a frequent complication that may manifest itself by glomerular lesions and tubular dysfunction from the second decade of life. We report two young GSDIa patients with malignant renal tumor. The first patient was a 25-year-old man. He had chronic metabolic imbalance without kidney involvement. The tumor, a type 2 papillary renal carcinoma, was accidentally discovered during follow-up. The second patient was a 27-year-old woman with chronic metabolic imbalance and chronic kidney involvement. The tumor, a grade 2 papillary carcinoma, was accidentally discovered during follow-up. These two observations are, to date, the first to be reported. We suggest that annual monitoring of kidney imaging in GSDI patients should be systematic to detect renal cancer, from the second decade of life.
糖原贮积病Ia型(GSD Ia)是一种由于葡萄糖-6-磷酸酶缺乏引起的罕见代谢性疾病。慢性肾脏病是一种常见并发症,可能在生命的第二个十年开始表现为肾小球病变和肾小管功能障碍。我们报告了两名患有恶性肾肿瘤的年轻GSDIa患者。第一名患者是一名25岁男性。他有慢性代谢失衡但无肾脏受累。该肿瘤为2型乳头状肾细胞癌,在随访期间意外发现。第二名患者是一名27岁女性,有慢性代谢失衡和慢性肾脏受累。该肿瘤为2级乳头状癌,在随访期间意外发现。这两例病例是迄今为止首次报道。我们建议,从生命的第二个十年开始,对GSDI患者应进行系统性的肾脏影像学年度监测,以检测肾癌。