Zhao Xi-Chen, Zhao Li, Sun Xiao-Yun, Xu Zeng-Shan, Ju Bo, Meng Fan-Jun, Zhao Hong-Guo
Department of Hematology, The Central Hospital of Qingdao West Coast New Area, Qingdao 266555, Shandong Province, China.
Department of Hematology, The Affiliated Hospital of Qingdao University, Qingdao 266000, Shandong Province, China.
World J Clin Cases. 2020 Jan 26;8(2):425-435. doi: 10.12998/wjcc.v8.i2.425.
Cumulative evidence suggests that the aberrant immune responses in acquired aplastic anemia (AA) are sustained by active chronic infections in genetically susceptible individuals. Recently, the constant source to trigger and sustain the pathophysiology has been proposed to come from the altered gut microbiota and chronic intestinal inflammation. In this case, our serendipitous finding provides convincing evidence that the persistently dysregulated autoimmunity may be generated, at least in a significant proposition of AA patients, by the altered gut microbiota and compromised intestinal epithelium.
A 30-year-old Chinese male patient with refractory severe AA experienced a 3-month-long febrile episode, and his fever was refractory to many kinds of injected broad-spectrum antibiotics. When presenting with abdominal cramps, he was prescribed oral mannitol and gentamycin to get rid of the gut infection. This treatment resulted in a quick resolution of the fever. Unanticipatedly, it also produced an excellent hematological response. He had undergone three episodes of recurrence within the one-year treatment, with each recurrence occurring 7-8 wk from the gastrointestinal inflammation eliminating preparations. However, subsequent treatments were able to produce subsequent remissions and consecutive treatments were successful in achieving durative hematological improvements, strongly indicating an etiological association between chronic gut inflammation and the development of AA. Interestingly, comorbid diseases superimposed on this patient (namely, psychiatric disorders, hypertension, insulin resistance, and renal dysfunction) were ameliorated together with the hematological improvements.
Chronic gut inflammation may be responsible for AA pathogenesis. The comorbidities and AA may share a common etiological association.
累积证据表明，在获得性再生障碍性贫血（AA）中，异常免疫反应在遗传易感个体中由活跃的慢性感染维持。最近，有人提出触发和维持病理生理过程的持续来源来自肠道微生物群的改变和慢性肠道炎症。在这种情况下，我们的意外发现提供了令人信服的证据，即至少在相当一部分AA患者中，持续失调的自身免疫可能是由肠道微生物群的改变和受损的肠上皮产生的。
一名30岁的中国男性难治性重型AA患者经历了长达3个月的发热期，多种注射用广谱抗生素对其发热均无效。出现腹部绞痛时，他被开了口服甘露醇和庆大霉素以消除肠道感染。这种治疗使发热迅速消退。出乎意料的是，它还产生了出色的血液学反应。在一年的治疗中，他经历了三次复发，每次复发都发生在消除胃肠道炎症的治疗后7 - 8周。然而，后续治疗能够产生后续缓解，连续治疗成功实现了持续性血液学改善，强烈表明慢性肠道炎症与AA的发生之间存在病因学关联。有趣的是，叠加在该患者身上的合并症（即精神障碍、高血压、胰岛素抵抗和肾功能不全）随着血液学改善而得到改善。
慢性肠道炎症可能是AA发病机制的原因。合并症和AA可能有共同的病因学关联。
