Hyperammonemia in a case of herpes simplex and anti-N-methyl-D-aspartate receptor encephalitis

Jung Min Ko, Woo Joong Kim, Soo Yeon Kim, Jun Hwa Lee, Jong Hee Chae, Kijoong Kim, Byung Chan Lim

Research output: Contribution to journalArticleResearchpeer-review

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.

Original languageEnglish
Pages (from-to)634-637
Number of pages4
JournalBrain and Development
Volume41
Issue number7
DOIs
StatePublished - 1 Aug 2019

Fingerprint

Anti-N-Methyl-D-Aspartate Receptor Encephalitis
Herpes Simplex Encephalitis
Hyperammonemia
Herpes Simplex
Metabolic Diseases
Brain Diseases
Autoimmune Diseases
Pediatrics
Lysinuric Protein Intolerance

Keywords

  • Anti-N-methyl-D-aspartate receptor encephalitis
  • Herpes simplex encephalitis
  • Lysinuric protein intolerance

Cite this

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title = "Hyperammonemia in a case of herpes simplex and anti-N-methyl-D-aspartate receptor encephalitis",
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.",
keywords = "Anti-N-methyl-D-aspartate receptor encephalitis, Herpes simplex encephalitis, Lysinuric protein intolerance",
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Hyperammonemia in a case of herpes simplex and anti-N-methyl-D-aspartate receptor encephalitis. / Ko, Jung Min; Kim, Woo Joong; Kim, Soo Yeon; Lee, Jun Hwa; Chae, Jong Hee; Kim, Kijoong; Lim, Byung Chan.

In: Brain and Development, Vol. 41, No. 7, 01.08.2019, p. 634-637.

Research output: Contribution to journalArticleResearchpeer-review

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T1 - Hyperammonemia in a case of herpes simplex and anti-N-methyl-D-aspartate receptor encephalitis

AU - Ko, Jung Min

AU - Kim, Woo Joong

AU - Kim, Soo Yeon

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AU - Chae, Jong Hee

AU - Kim, Kijoong

AU - Lim, Byung Chan

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AB - 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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