Experience with Cidofovir as an adjunctive therapy in a patient of adenovirus-induced macrophage activation syndrome in
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CASE BASED REVIEW
Experience with Cidofovir as an adjunctive therapy in a patient of adenovirus-induced macrophage activation syndrome in systemic arthritis Priyankar Pal 1
&
Niladri Bose 2 & Avishek Poddar 3 & Kaustabh Chowdhury 3 & Agnisekhar Saha 3
Received: 13 March 2020 / Revised: 17 April 2020 / Accepted: 23 April 2020 # International League of Associations for Rheumatology (ILAR) 2020
Abstract A 5-year-old female child, with known systemic juvenile idiopathic arthritis diagnosed at 18 months of age (on low dose Prednisolone + Methotrexate + Leflunomide + Tocilizumab), presented with fever for 1 day, vomiting, drowsiness followed by seizures. On admission to PICU, she was drowsy, tachycardic, tachypneic, with rashes, and hepatosplenomegaly. Lab findings showed thrombocytopenia, leucopenia, low ESR, normal CRP, elevated liver enzymes, high ferritin, LDH, and triglycerides suggestive of macrophage activation syndrome (MAS). Chest X-ray showed left basal pneumonia and DNA PCR of throat swab revealed adenovirus. She was diagnosed as adenovirus-triggered MAS and was initiated on pulse methylprednisolone (6 mg/kg). Because of suboptimal response after 2 doses, manifested by increasing drowsiness, further fall in platelets and rising ferritin, methylprednisolone dosage was increased to 30 mg/kg/day with the addition of oral cyclosporine (4 mg/kg/day). In view of worsening of the chest X-ray and increasing oxygen requirement, Cidofovir infusion (1 mg/kg thrice weekly) was also started simultaneously considering increased activity of the adenoviral infection concurrent to immunosuppression. Within 48 h, the child showed signs of recovery with improved consciousness, lower oxygen requirements, and improving lab parameters. She was discharged after 3 weeks of IV Cidofovir, on oral prednisolone and cyclosporine. To the best of our knowledge, this is the first reported use of Cidofovir in adenovirus-induced MAS. Keywords Adenovirus . Cidofovir . Macrophage activation syndrome . Systemic arthritis
Introduction Macrophage activation syndrome (MAS) is a potentially lethal complication of systemic inflammatory diseases of childhood, commonest association being with systemic juvenile idiopathic arthritis (sJIA). MAS may occur spontaneously, Electronic supplementary material The online version of this article (https://doi.org/10.1007/s10067-020-05133-0) contains supplementary material, which is available to authorized users. * Priyankar Pal [email protected] 1
Institute of Child Health, 11, Dr Biresh Guha St., Kolkata 700017, India
2
Department of Pediatrics, Apollo Gleneagles Hospitals, Kolkata, India
3
Pediatric Intensive Care Unit, Apollo Gleneagles Hospitals, Kolkata, India
as a complication of active underlying disease, or may be triggered by an infection, a change in drug therapy, or toxic effect of a medication. The acute and dramatic onset of this entity often mimics a flare of the underlying disease or a severe sepsis [1]. Adenoviruses are emerging as an important cause of respiratory tract infecti
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