Published online Jan 26, 2020. doi: 10.12998/wjcc.v8.i2.425
Peer-review started: November 8, 2019
First decision: November 19, 2019
Revised: December 2, 2019
Accepted: December 21, 2019
Article in press: December 21, 2019
Published online: January 26, 2020
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.
Core tip: The aberrant immune responses in aplastic anemia had been proposed to be triggered by altered gut microbiota, and our serendipitous finding that a 30-year-old man with refractory severe aplastic anemia had gained an inadvertently excellent hematological response following oral administration of mannitol and gentamycin provides convincing evidence to support the hypothetical but plausible pathogenic association. Several comorbid diseases on this patient were ameliorated together with the hematological improvements, indicating commonly shared etiological associations between the comorbidities and aplastic anemia in a background consisting of altered gut microbiota, chronic intestinal inflammation, increased epithelial permeability, and an autoimmune nature, instead of the adverse events of cyclosporine and iron overload.