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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:INTRODUCTION:Diuretics are a commonly used for the treatment of acute pulmonary edema. However, inappropriate administration of diuretic drugs can result in clinical treatment failure and cause acute pulmonary edema. This is due to rapid decreases in intravascular volume as a result of diuretic treatment. To date, the clinical phenomenon of inappropriate use of diuretics leading to acute pulmonary edema remains unexplored and unrecognized. Here, we report the first case of this problem-pulmonary edema following diuretic therapy. PATIENT CONCERNS:A 71-year-old male patient who was intubated and transferred to the intensive care unit (ICU) due to respiratory failure was initially diagnosed with pneumonia as a complication of acute respiratory distress syndrome (ARDS). After treatments including antibiotics, lung protective ventilation strategies, and restrictive fluid management, his respiratory symptoms improved. However, the patient's dyspnea became more severe after experimental diuretic therapy. DIAGNOSIS:A point-of-care ultrasound (POCUS) examination showed increased extravascular lung water retention during a hypovolemic state. After full examinations and analysis, the diagnosis of acute pulmonary edema was determined. INTERVENTIONS:The most likely cause of acute pulmonary edema was left ventricular (LV) hyperdynamic status due to a hypovolemic status caused by excessive diuretic therapy. Consequently, we administrated intravenous fluids and a ?-receptor blocker to the patient. OUTCOMES:Following these treatment, the patient's respiratory distress improved remarkably. CONCLUSION:We report the first case of pulmonary edema following diuretic therapy to stress the need of physicians to follow guidelines of clinical practice. Maintaining an appropriate volume status and treatment of ?-receptor blockers is the key to reversing the progress of this adverse effect. In this process, POCUS is a reliable diagnostic tool to identify the cause of acute pulmonary edema and can increase the accuracy of clinical evaluations. It is likely that a wider use of POCUS will help physicians to obtain a faster, and more accurate, diagnosis of the etiology of acute pulmonary edema, thus allowing a more appropriate therapy.

Dataset Information

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

Publications

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

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