Hyperammonemia in a case of herpes simplex and anti-N-methyl-d-aspartate receptor encephalitis |
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| Authors: | Jung Min Ko Woo Joong Kim Soo Yeon Kim Jun Hwa Lee Jong Hee Chae Ki Joong Kim Byung Chan Lim |
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| Institution: | 1. Department of Pediatrics, Seoul National University College of Medicine, Seoul National University Children’s Hospital, Seoul, Republic of Korea;2. Department of Pediatrics, Samsung Changwon Hospital, Sungkyunkwan University School of Medicine, Changwon, Republic of Korea |
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| Abstract: | Herpes simplex encephalitis (HSE) is a widely accepted risk factor for anti N-methyl-d-aspartate receptor (NMDAR) encephalitis. Association of inherited metabolic disease has never been reported in a patient with HSE and anti-NMDAR encephalitis. Herein, we report a case of pediatric HSE complicated by development of anti-NMDAR encephalitis; this patient showed subsequent recurrent, unexplained episodes of encephalopathy associated with hyperammonemia. The patient was diagnosed with lysinuric protein intolerance (LPI), a rare inborn metabolic disorder. Although it would be difficult to make conclusions regarding the casual link of HSE and anti-NMDAR encephalitis with LPI from a single case, there have been many reports that autoimmune diseases and immunologic abnormalities are frequently associated with LPI. Thus, we speculate that LPI may contribute to the development of anti-NMDAR encephalitis following HSE. |
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| Keywords: | Corresponding author at: Department of Pediatrics Seoul National University Children’s Hospital Daehakro 101 Jongnogu Seoul 03080 Republic of Korea Herpes simplex encephalitis Lysinuric protein intolerance |
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