Legros L, Revencu N, Nassogne M-C, Wese F-X, Feyaerts A
Service d'urologie, cliniques universitaires Saint-Luc, Bruxelles, Belgique.
Centre de génétique humaine, cliniques universitaires Saint-Luc, Bruxelles, Belgique.
Arch Pediatr. 2015 Nov;22(11):1147-50. doi: 10.1016/j.arcped.2015.07.017. Epub 2015 Sep 16.
We report on the case of a child who presented with recurrent, multiple, and voluminous bladder diverticula. Bladder diverticula are defined as a herniation of the mucosa through the bladder muscle or the detrusor. Causes are numerous and diverticula can be classified into primary congenital diverticula (para-ureteral - or Hutch diverticula - and posterolateral diverticula); secondary diverticula (resulting from chronic mechanical obstruction or from neurological disease; and diverticula secondary to connective tissue or muscle fragility. The latter is seen in disease entities such as prune belly syndrome, Ehlers-Danlos syndrome, cutis laxa syndrome, OHS (occipital horn syndrome), Menkes disease, and Williams-Beuren syndrome. In this patient, the cause of these diverticula was OHS, a genetic, recessive X-chromosome-linked syndrome, responsible for abnormal tissue caused by a disorder in copper metabolism. This case reminds us of the importance of pushing the diagnostic workup when presented with multiple and/or large bladder diverticula, and in particular to search for rare malformation syndromes after exclusion of an obstacle.
我们报告了一例患有复发性、多发性和大量膀胱憩室的儿童病例。膀胱憩室被定义为黏膜通过膀胱肌肉或逼尿肌的疝出。病因众多,憩室可分为原发性先天性憩室(输尿管旁憩室或哈钦森憩室以及后外侧憩室);继发性憩室(由慢性机械性梗阻或神经疾病引起;以及继发于结缔组织或肌肉脆弱性的憩室。后者见于如梅干腹综合征、埃勒斯-丹洛斯综合征、皮肤松弛综合征、枕角综合征、门克斯病和威廉姆斯-贝伦综合征等疾病实体。在该患者中,这些憩室的病因是枕角综合征,这是一种遗传性、隐性X染色体连锁综合征,由铜代谢紊乱导致组织异常。该病例提醒我们,当出现多发性和/或大的膀胱憩室时,推进诊断检查的重要性,尤其是在排除梗阻后寻找罕见的畸形综合征。