Abstract

BackgroundWe describe a case of a fever of unknown etiology that was caused by a caseating tubercle granuloma which produced erythropoietin. To our knowledge, this is the first report of an erythropoietin- producing granuloma.Case presentationA 48-year-old Japanese man with a 5-year history of maintenance hemodialysis for diabetic nephropathy presented with an intermittent fever over a few months. During febrile periods he developed erythema nodosum on his legs. Computed tomography showed axillary lymph node enlargement and this was further corroborated by a gallium scan that revealed high gallium uptake in these nodes. A Mantoux test was positive and an interferongamma release assay for tuberculosis diagnosis was also positive. Lymph node tuberculosis was suspected and the patient underwent lymphadenectomy. Histological analysis of the lymph nodes revealed a caseating granuloma that showed positive results on an acid-fast bacteria stain and a Mycobacterium tuberculosis polymerase chain reaction test. After lymphadenectomy, however, the patient’s hemoglobin levels rapidly decreased from 144 to 105 g/L, and this was further compounded by a decrease in serum erythropoietin from 223 mIU/mL to 10.7 mIU/mL by postoperative day 21. We suspected the tubercle to be a source of the erythropoietin and this was further confirmed by in situ hybridization.ConclusionsWe report for the first time ectopic erythropoietin production by a tuberculous lymph node. Our observations are substantiated by a postoperative decline in his erythropoietin level and a clinical requirement for erythropoietin treatment.

Highlights

  • We describe a case of a fever of unknown etiology that was caused by a caseating tubercle granuloma which produced erythropoietin

  • We report for the first time ectopic erythropoietin production by a tuberculous lymph node

  • We present here the first known instance of a tubercle granuloma implicated in ectopic Epo production

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Summary

Conclusions

We present here the first known instance of a tubercle granuloma implicated in ectopic Epo production. Chronic infections like tuberculosis are usually accompanied by anemia because of lowered Epo production caused by cytokines such as tumor necrosis factor [15]. In cases like ours, where the patient was on maintenance dialysis, we speculate that the renal anemia due to chronic dialysis was able to mask the effects of polycythemia caused by the Epo over-production. Other transcription factors like Wilms tumor protein, Wt1 [23] and GATA-4 [24] are involved in Epo gene expression in the liver In this light, the molecular mechanism underlying Epo production in macrophages is not known, it is not an implausible finding. We report, to the best of our knowledge, the first case of Epo-producing tubercle granuloma in a hemodialysis patient.

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