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Steroid responsive encephalopathy associated with autoimmune thyroiditis following ipilimumab therapy: a case report.

ABSTRACT: Ipilimumab is a cytotoxic T-lymphocyte-associated protein 4 receptor antibody used for immunotherapy in cancer. Several immune-related adverse events are known. Steroid responsive encephalopathy associated with autoimmune thyroiditis is an autoimmune encephalopathy associated with Hashimoto's Disease and elevated serum levels of the related antibodies (anti-thyroid-peroxidase antibody or anti-thyroglobulin antibody). Our case implies that steroid responsive encephalopathy associated with autoimmune thyroiditis may be another previously unreported side effect of ipilimumab therapy.We report the case of a 64 years old caucasian patient with prostatic cancer who received ipilimumab therapy in a clinical trial. He presented with aphasia, tremor and ataxia, myocloni, hallucinations, anxiety and agitation in turns with somnolence. Cranial nerves, deep tendon reflexes, motor and sensory functions were normal. Electroencephalography showed background slowing but no epileptic discharges. Brain magnetic resonance imaging was normal and showed no signs of hypophysitis. Cerebrospinal fluid findings ruled out infection and neoplastic meningitis. Anti-thyroid antibodies (anti-thyroid-peroxidase antibody and anti-thyroglobulin antibody) were heavily increased. Assuming steroid responsive encephalopathy associated with autoimmune thyroiditis the patient was treated with 1,000 mg methylprednisolone i.v. for 3 days and continued with 1 mg/kg orally. On the 3rd day of treatment the patient's condition started to improve. Within the next few days he gradually returned to his previous state, and electroencephalography eventually showed only slight slowing. Seven months later the patient's condition was stable, and anti-thyroid antibodies were no more detectable.Steroid responsive encephalopathy associated with autoimmune thyroiditis may be a hitherto unrecognized complication of ipililumab treatment and should be taken into consideration in patients developing central nervous symptoms undergoing this treatment.

SUBMITTER: Carl D

PROVIDER: S-EPMC4514969 | biostudies-literature | 2015 Jul

REPOSITORIES: biostudies-literature

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Steroid responsive encephalopathy associated with autoimmune thyroiditis following ipilimumab therapy: a case report.

Carl David D Grüllich Carsten C Hering Steffen S Schabet Martin M

BMC research notes 20150726

<h4>Background</h4>Ipilimumab is a cytotoxic T-lymphocyte-associated protein 4 receptor antibody used for immunotherapy in cancer. Several immune-related adverse events are known. Steroid responsive encephalopathy associated with autoimmune thyroiditis is an autoimmune encephalopathy associated with Hashimoto's Disease and elevated serum levels of the related antibodies (anti-thyroid-peroxidase antibody or anti-thyroglobulin antibody). Our case implies that steroid responsive encephalopathy asso ...[more]

PMID: 26209970

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Project description:A few patients with Hashimoto's thyroiditis or Graves' disease develop a multiform syndrome of the central nervous system (CNS) termed Hashimoto's encephalopathy or steroid-responsive encephalopathy associated with autoimmune thyroid disease (HE/SREAT). They have high levels of thyroid autoantibodies (TgAb, TPOAb and/or TSH-R-Ab) in blood and cerebrospinal fluid. Autoantibodies against alpha-enolase, aldehyde reductase-I (AKRIA) and/or dimethylargininase-I (DDAHI), proteins expressed in the CNS among other tissues, were detected in the blood and, when searched, in the cerebrospinal fluid of HE/SREAT patients. Recently, we reported that alpha-enolase, AKRIA and DDAHI share local sequence homology with each of the three autoantigens (TgAb, TPOAb, TSH-R-Ab), often in epitope-containing segments of the thyroid autoantigens. We hypothesized that there might be additional CNS-expressed proteins homologous to thyroid autoantigens, possibly overlapping known epitopes of the thyroid autoantigens. We used bioinformatic methods to address this hypothesis. Six, 27 and 47 of 46,809 CNS-expressed proteins share homology with TSH-R, Tg and TPO, respectively. The homologous regions often contain epitopes, and some match regions of thyroid autoantigens which have homology with alpha-enolase, AKRIA and/or DDAHI. Several of the aforementioned proteins are present in CNS areas that show abnormalities at neuroimaging in HE/SREAT patients. Furthermore, autoantibodies against some of the said six, 27 and 47 proteins were reported to be associated with a number of autoimmune diseases. Not only we validated our hypothesis, but we think that such a variety of potential CNS targets for thyroid Ab against epitopes contained in regions that have local homology with CNS proteins may explain the polymorphic phenotypes of HE/SREAT. Only when elevated amounts of these Ab are synthesized and trespass the blood-brain barrier, HE/SREAT appears. This might explain why HE/SREAT is so relatively rare.

Project description:We report a case of overlooked Subacute Thyroiditis (SAT) potentially induced by the administration of a COVID-19 vaccine. This case prompted a thorough review of the existing literature to elucidate possible mechanisms by which immune responses to the COVID-19 vaccine might precipitate thyroid damage. The primary objective is to enhance the clinical understanding and awareness of SAT among healthcare professionals. Subacute thyroiditis is a prevalent form of self-limiting thyroid disorder characterized by fever, neck pain or tenderness, and palpitations subsequent to viral infection. The development of numerous SARS-CoV-2 vaccines during the COVID-19 pandemic was intended to mitigate the spread of the virus. Nevertheless, there have been documented instances of adverse reactions arising from SARS-CoV-2 vaccines, such as the infrequent occurrence of subacute thyroiditis. While the majority of medical practitioners can discern classic subacute thyroiditis, not all cases exhibit typical characteristics, and not all systematic treatments yield positive responses. In this study, we present a rare case of subacute thyroiditis linked to the administration of the SARS-CoV-2 vaccine. A previously healthy middle-aged female developed fever and sore throat 72 h post-inoculation with the inactivated SARS-CoV-2 vaccine. Initially attributing these symptoms to a common cold, she self-administered ibuprofen, which normalized her body temperature but failed to alleviate persistent sore throat. Suspecting a laryngopharyngeal disorder, she sought treatment from an otolaryngologist. However, the pain persisted, accompanied by intermittent fever over several days. After an endocrinology consultation, despite the absence of typical neck pain, her examination revealed abnormal thyroid function, normal thyroid antibodies, heterogeneous echogenicity on thyroid ultrasonography, and elevated levels of Erythrocyte Sedimentation Rate (ESR) and C-Reactive Protein (CRP). These findings led to a consideration of the diagnosis of SAT. Initially, she was treated with non-steroidal anti-inflammatory drugs (NSAIDs) for her fever, which proved effective, but her neck pain remained uncontrolled. This suggested a poor response to NSAIDs. Consequently, steroid therapy was initiated, after which her symptoms of fever and neck pain rapidly resolved.

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Steroid responsive encephalopathy associated with autoimmune thyroiditis following ipilimumab therapy: a case report.

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Steroid responsive encephalopathy associated with autoimmune thyroiditis following ipilimumab therapy: a case report.

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