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Abstract Details

A Tale of 2 Leukodystrophies in DOCK8 Deficiency, a Rare Primary Immunodeficiency
Autoimmune Neurology
P17 - Poster Session 17 (11:45 AM-12:45 PM)
9-002

Describe two cases of neuroinflammation in DOCK8 deficiency.

DOCK8 deficiency is a genetic combined immunodeficiency. It has infrequently been associated with systemic autoimmunity and CNS vasculitis often thought to be infectious.

We report two patients with DOCK8 deficiency and inflammatory brain white matter disease.

Case 1

 9 yo male with DOCK8 deficiency. At 7.5 years of age, he had had an episode of infectious ADEM; CSF non-inflammatory but positive for EBV and CMV PCR. He recovered well with gancyclovir and steroids. Follow-up brain MRI at 9 yo showed some gliosis. The patient was incidentally found on sinus MRI 2 months later to have a contrast enhancing lesion in the right frontal white matter extending across the corpus callosum. He was neurologically asymptomatic. CSF was acellular, positive only for EBV PCR. He was found to be seropositive for MOG-immunoglobulin G (IgG) by cell-based assay. He was treated with ivIG monthly and the MRI improved. He proceeded to HSCT.

Case 2

21 yo male with recurrent sinopulmonary infections and warts whose genetic sequencing revealed DOCK8 deficiency. He developed erythroderma; skin biopsy consistent with cytotoxic T-cell lymphoma (CTCL). PET revealed generalized lymphadenopathy consistent with Sezary syndrome. Neurologically asymptomatic but screening MRI revealed diffuse punctate FLAIR hyperintense lesions, some with contrast enhancement. CSF had 9 WBCs, positive for JC-virus and some atypical T-cells on flow cytometry. Brain biopsy confirmed PML without lymphomatous involvement. In an attempt to prevent further immunosuppression from standard chemotherapy, he was treated with bexarotene with very good partial response. He received JCV-viral specific T-cells engineered from his matched-donor brother to debulk his JC viral load before proceeding to HSCT.

We report two cases of DOCK8 deficiency with neuroinflammatory demyelination: one from PML and the other from MOG antibody recurrent ADEM. CNS autoimmune diseases can present in patients with primary immunodeficiencies.  

Authors/Disclosures
Ariane Soldatos, MD, FAAN
PRESENTER
Dr. Soldatos has nothing to disclose.
Irene Cortese, MD Dr. Cortese has stock in Keires, AG. Dr. Cortese has stock in Nouscom, AG. Dr. Cortese has stock in PDC*line Pharma.
Anita M. Fletcher, MD (AdventHealth Neuroscience Institute) Dr. Fletcher has nothing to disclose.
No disclosure on file
No disclosure on file