Neuromyotonia with Central Nervous System Lesions following Quadrivalent Human Papilloma Virus Vaccination

Abstract

Neuromyotonia is a rare peripheral nerve hyperexcitability syndrome often associated with antibodies directed against contactin-associated protein-like 2 and leucine-rich, glioma inactivated 1. The quadrivalent human papilloma virus vaccine Gardasil®, first approved in 2006, is known to be a highly effective prophylaxis against papillomavirus types 6, 11, 16, and 18. Molecularly, this non-infectious recombinant vaccine is based on purified L1 proteins from the human papilloma virus capsid. Since the approval of this vaccine, several studies have investigated its safety regarding the occurrence of autoimmune conditions following application. Here, we present the first case of neuromyotonia with active Gadolinium enhancing demyelinating central nervous system lesions following vaccination with Gardasil®.

Isaacs syndrome with LGI1 and CASPR2 antibodies after HPV vaccination: A case report

“…Autoimmune CASPR2 and LGI1 diseases usually manifest as Morvan syndrome and/or limbic encephalitis. We report a case of Isaacs with positive CASPR2, LGI1, anti-phospholipid antibody, and anti-Sulfatide antibody after the second dose of qHPV vaccine, and described the classic clinical feature of Isaacs: muscle spasms, which was con rmed by Cerami and Maryam Hatami [4,5]. Although one study reported a case of CASPR2 and LGI1 double antibody positivity, the clinical presentation was GBS-like syndrome that developed into typical respiratory paralysis, and the neurological symptoms in this patient resolved quickly after plasma washing [6].…”

“…Isaacs syndrome is a rare condition, and it is even rarer to appear after HPV vaccination. Studies have found that in ammatory central nervous system diseases are more common in young people [4]. So, there is some overlap between this group and the population that receives HPV vaccination.…”

“…The quadrivalent human papillomavirus (qHPV) vaccine, rst approved in 2006, is a highly effective prophylaxis against papillomavirus types 6, 11, 16 and 18. Since the vaccine was approved, several studies have investigated the possibility of autoimmune disease following application of the vaccine [4]. Here, we report the clinical features of a case of Isaacs syndrome that occurred after the second dose of qHPV vaccination and their response to symptomatic and immunomodulatory treatment, which was positive for CASPR2 antibodies and positive for LGI1 antibodies with positive antiphospholipid antibodies and positive anti-sulfatide antibodies.…”

Isaacs syndrome with LGI1 and CASPR2 antibodies after HPV vaccination: A case report

“…We report a case of Isaacs with positive CASPR2, LGI1, anti-phospholipid antibody, and anti-Sulfatide antibody after the second dose of qHPV vaccine, and described the classic clinical feature of Isaacs: muscle spasms, which was confirmed by Cerami and Maryam Hatami. [ 10 , 11 ] In fact, Isaacs syndrome is an autoimmune disease characterized by neurogenic myotonia. It is distinguished by the presence of a myotonic tremor syndrome resulting from heightened excitability of the peripheral nerves.…”

“…[ 2 , 19 ] Emerging research suggests that vaccines could potentially serve as trigger factors for certain inflammatory autoimmune disorders that affect the nervous system. This phenomenon might be linked to the immune system response to human papilloma virus (HPV) virus-like particles, which can stimulate the immune system via a process known as “bystander activation.” [ 10 , 20 ] Additionally, the secretion of IL-6 and TNF-α by dendritic cells could provide immunomodulatory signals that contribute to inflammatory reactions within the nervous system. [ 10 , 21 ] Overall, research has indicated that inflammatory diseases affecting the central nervous system are more frequently observed in young individuals, Isaacs syndrome, a relatively rare disease, may also have this tendency and may be associated with an immune response following vaccination.…”