IT-DEX and B cell depletion in a child with anti-GAD 65 autoimmune encephalitis presenting as NORSE: A case report

被引：0

作者：

Yarimi, Jonathan M. ^{[1
,2
]}

Sandweiss, Alexander J. ^{[1
,2
]}

Salazar, Karla P. ^{[1
,2
]}

Massrey, Chrissie ^{[1
,2
]}

Ankar, Alexander ^{[1
,2
]}

Muscal, Eyal ^{[3
,4
]}

Lai, Yi-Chen ^{[5
]}

Cokley, Jon A. ^{[6
]}

Davila-Williams, Daniel ^{[1
,2
]}

Shukla, Nikita M. ^{[1
,2
]}

Fisher, Kristen S. ^{[1
,2
]}

机构：

[1] Baylor Coll Med, Dept Pediat, Div Pediat Neurol & Dev Neurosci, 6701 Fannin St,Ste 1250, Houston, TX 70030 USA

[2] Texas Childrens Hosp, 6701 Fannin St,Ste 1250, Houston, TX 70030 USA

[3] Baylor Coll Med, Dept Pediat, Sect Rheumatol, 6701 Fannin St, Houston, TX 70030 USA

[4] Texas Childrens Hosp, 6701 Fannin St, Houston, TX 70030 USA

[5] Texas Childrens Hosp, Dept Pediat, Div Pediat Crit Care Med, Baylor Coll Med, 6701 Fannin St, Houston, TX 70030 USA

[6] Texas Childrens Hosp, Dept Pharm, 6701 Fannin St, Houston, TX 70030 USA

来源：

JOURNAL OF NEUROIMMUNOLOGY | 2024年 / 395卷

关键词：

New-onset refractory status epilepticus; Glutamic acid decarboxylase antibody; Autoimmune encephalitis; Intrathecal dexamethasone; Cytokines;

D O I：

10.1016/j.jneuroim.2024.578430

中图分类号：

R392 [医学免疫学]; Q939.91 [免疫学];

学科分类号：

100102 ;

摘要：

New-onset refractory status epilepticus (NORSE) is a devastating clinical condition that often leads to severe disability. Intrathecal dexamethasone (IT-DEX) has been reported to improve refractory status epilepticus. We present an 11-year-old female with anti-GAD 65 encephalitis presenting as NORSE who had minimal response to standard anti-seizure medications and first-line immunotherapies. The patient received 6 doses of IT-DEX in conjunction with rituximab which correlated with subsequent decreased neuroinflammation, reduced seizure burden and aided in weaning anesthetic infusions. Our case with literature review suggests IT-DEX may be utilized as an early intervention in those with refractory status epilepticus from various etiologies.

引用

页数：4

共 16 条

[1] Antibody-Mediated Encephalitis [J].

Dalmau, Josep ;

Graus, Francesc .

NEW ENGLAND JOURNAL OF MEDICINE, 2018, 378 (09) :840-851

[2] Intrathecal dexamethasone therapy for febrile infection-related epilepsy syndrome [J].

Horino, Asako ;

Kuki, Ichiro ;

Inoue, Takeshi ;

Nukui, Megumi ;

Okazaki, Shin ;

Kawawaki, Hisashi ;

Togawa, Masao ;

Amo, Kiyoko ;

Ishikawa, Junichi ;

Ujiro, Atsushi ;

Shiomi, Masashi ;

Sakuma, Hiroshi .

ANNALS OF CLINICAL AND TRANSLATIONAL NEUROLOGY, 2021, 8 (03) :645-655

[3] Anti-inflammatory and immune-mediated therapy for a case of febrile infection-related epilepsy syndrome with rapid recurrence [J].

Kurimoto, Tomonori ;

Matsuoka, Tsuyoshi ;

Ami, Yuki ;

Kanno, Koji ;

Fujii, Takashi ;

Fujiwara, Naoki ;

Matsuoka, Takashi .

CLINICAL CASE REPORTS, 2022, 10 (06)

[4] Intrathecal Dexamethasone in Febrile Infection-Related Epilepsy Syndrome [J].

Mehta, Niyati P. ;

Sawdy, Rachel ;

Maloney, Kathleen ;

Overlee, Brittany ;

Johnson, Renee K. ;

Howe, Charles L. ;

Farias-Moeller, Raquel .

NEUROLOGY-CLINICAL PRACTICE, 2023, 13 (03)

[5] Neurologic syndromes related to anti-GAD65 Clinical and serologic response to treatment [J].

Munoz-Lopetegi, Amaia ;

de Bruijn, Marienke A. A. M. ;

Boukhrissi, Sanae ;

Bastiaansen, Anna E. M. ;

Nagtzaam, Mariska M. P. ;

Hulsenboom, Esther S. P. ;

Boon, Agnita J. W. ;

Neuteboom, Rinze F. ;

de Vries, Juna M. ;

Sillevis Smitt, Peter A. E. ;

Schreurs, Marco W. J. ;

Titulaer, Maarten J. .

NEUROLOGY-NEUROIMMUNOLOGY & NEUROINFLAMMATION, 2020, 7 (03)

[6] The influence of intrathecal injection of methotrexate and dexamethasone on neuropsychiatric systemic lupus erythematosus (NPSLE): a retrospective cohort study of 386 patients with NPSLE [J].

Nie, Yuxue ;

Sun, Boyuan ;

He, Xin ;

Zheng, Minmin ;

Wu, Di ;

Yang, Yunjiao ;

Zhang, Li ;

Bai, Wei ;

Jiang, Nan ;

Qiao, Lin ;

Huang, Can ;

Zhou, Shuang ;

Zhou, Jiaxin ;

Peng, Linyi ;

Niu, Jingwen ;

Li, Mengtao ;

Zhao, Yan ;

Zeng, Xiaofeng ;

Wang, Li ;

Zhang, Wen .

ARTHRITIS RESEARCH & THERAPY, 2023, 25 (01)

[7] Use and Safety of Immunotherapeutic Management of N-Methyl-D-Aspartate Receptor Antibody Encephalitis A Meta-analysis [J].

Nosadini, Margherita ;

Eyre, Michael ;

Molteni, Erika ;

Thomas, Terrence ;

Irani, Sarosh R. ;

Dalmau, Josep ;

Dale, Russell C. ;

Lim, Ming .

JAMA NEUROLOGY, 2021, 78 (11) :1333-1344

[8] Clinical presentation of new onset refractory status epilepticus in children (the pSERG cohort) [J].

Sculier, Claudine ;

Barcia Aguilar, Cristina ;

Gaspard, Nicolas ;

Gainza-Lein, Marina ;

Sanchez Fernandez, Ivan ;

Amengual-Gual, Marta ;

Anderson, Anne ;

Arya, Ravindra ;

Burrows, Brian T. ;

Brenton, James N. ;

Carpenter, Jessica L. ;

Chapman, Kevin E. ;

Clark, Justice ;

Gaillard, William D. ;

Glauser, Tracy A. ;

Goldstein, Joshua L. ;

Goodkin, Howard P. ;

Gorman, Mark ;

Lai, Yi-Chen ;

McDonough, Tiffani L. ;

Mikati, Mohamad A. ;

Nayak, Anuranjita ;

Peariso, Katrina ;

Riviello, James ;

Rusie, Allison ;

Sperberg, Katherine ;

Stredny, Coral M. ;

Tasker, Robert C. ;

Tchapyjnikov, Dmitry ;

Vasquez, Alejandra ;

Wainwright, Mark S. ;

Wilfong, Angus A. ;

Williams, Korwyn ;

Loddenkemper, Tobias .

EPILEPSIA, 2021, 62 (07) :1629-1642

[9] Favorable response to classic ketogenic diet in a child with anti-GAD 65 antibody mediated super refractory status epilepticus [J].

Sivathanu, Deepika ;

Kewalramani, Deepti ;

Manokaran, Ranjith Kumar .

EPILEPSY & BEHAVIOR REPORTS, 2022, 19

[10] Seizure burden and neuropsychological outcomes of new-onset refractory status epilepticus: Systematic review [J].

Taraschenko, Olga ;

Pavuluri, Spriha ;

Schmidt, Cynthia M. M. ;

Pulluru, Yashwanth Reddy ;

Gupta, Navnika .

FRONTIERS IN NEUROLOGY, 2023, 14

← 1 2 →