Hyperacute reactivation of cytomegalovirus-induced gastroduodenitis during remission induction in a young male patient with granulomatosis with polyangiitis: a case report and review of literature

一例年轻男性肉芽肿性多血管炎患者在缓解诱导期发生巨细胞病毒性胃十二指肠炎超急性复发:病例报告及文献复习

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Abstract

BACKGROUND: Cytomegalovirus is a pathogen known to aggravate the inflammatory response in autoimmune diseases via molecular mimicry. Although it is recognized that cytomegalovirus activation can happen during extended but variable periods of immunosuppression (14-90 days), it is rarely reported in conjunction with an acute flare-up of an autoimmune disease. Currently, there is no consensus on cytomegalovirus prophylaxis for patients initiating remission induction. CASE PRESENTATION: Here, we present the case of a 31-year-old male patient of South Indian ethnicity, presenting with a 2-month history of fever, conductive hearing loss, and ear discharge. This was associated with symmetrical inflammatory polyarthritis for 1 month, unilateral painful conjunctivitis, and skin erythema for 5 days. Blood analyses showed elevated inflammatory markers; strongly positive anti-proteinase 3 and cytoplasmic antineutrophil cytoplasmic antibody levels; normal procalcitonin and complement levels; and negative anti-myeloperoxidase and perinuclear antineutrophil cytoplasmic antibody levels. A nasal endoscopy revealed a midline granuloma with vasculitis features on biopsy. Imaging revealed pulmonary nodules and otomastoiditis. Now diagnosed with granulomatosis with polyangiitis, the patient developed signs of gastroduodenitis within a day of initiation of immunosuppression with high-dose "pulse" intravenous methylprednisolone. We evaluated him for mesenteric ischemia/gastrointestinal vasculitis. However, the duodenal biopsies from the bleeding ulcers revealed a probable cytomegalovirus infection, confirmed with high serum viral loads. We treated him with a ganciclovir regimen and transitioned him to steroid-sparing immunosuppressant therapy with mycophenolate mofetil, which was selected over cyclophosphamide for its noninferior effectiveness and better safety profile in non-life-threatening granulomatosis with polyangiitis disease. The patient recovered uneventfully and is currently in remission. CONCLUSION: Cytomegalovirus reactivation is possible during short-term steroid pulse therapy. Further research is needed to evaluate whether routine cytomegalovirus screening is warranted before starting immunosuppressive treatment with high-dose steroids in autoimmune conditions.

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