Massive idiopathic spontaneous hemothorax complicating anti-N-methyl-d-aspartate receptor encephalitis

Abstract

Rationale:Spontaneous hemothorax is a subcategory of hemothorax which can be life threatening. The etiology of spontaneous hemothorax can be various, and in some rare cases the causes remained unknown. Hence, it is quite difficult to establish the diagnosis. Here, we report a case of spontaneous hemothorax in a young female patient who was recently diagnosed with anti-N-methyl-d-aspartate receptor encephalitis (anti-NMDAR encephalitis).Patient concerns:A 20-year-old female was transferred to emergency department of our hospital from local hospital presented with insomnia, mood lability, tonic-clonic seizure, and decreased level of consciousness.Diagnoses:The diagnosis of anti-NMDAR encephalitis was established by detection of cerebrospinal fluid (CSF) and serum antibodies to the NMDA receptor. During the hospital stay, the patient developed massive spontaneous hemothorax and was confirmed by closed-tube thoracostomy.Interventions and outcomes:Video-assisted thoracotomy was performed to evacuate the blood clots and also to obtain pleural biopsy specimen for diagnostic evaluation. However, the reason of hemothorax remained idiopathic. The postoperative status of this patient was uneventful, and she was discharged on postoperative day 45 as her mental status improved markedly.Lessons:In this case, the patient had both anti- NMDAR encephalitis and autoimmune thyroid disease. Based on it, we suspected that the patient subjected to severe autoimmune response and inflammatory reaction, which might explain the pathologic changes of parietal pleura and visceral pleura. We recommend the suspicion of spontaneous hemothorax should be considered when the patients with autoimmune diseases present with hemorrhage-related signs or symptoms.

Anti-N-methyl-D-aspartate receptor encephalitis

“…Due to the frequent acute onset of anti-NMDAR encephalitis, and lacking specific features in imaging examinations, anti-NMDAR encephalitis is often confused with viral encephalitis in the early stages. [12] Viral encephalitis also manifests itself as an acute-onset, rapidly progressing disease, and has symptoms such as fever, headache, vomiting, psychiatric symptoms, epilepsy, and disturbance of consciousness during the course of the disease. For patients with involuntary mouth-face-tongue movements, central hypoventilation syndrome, and a poor response to antiviral therapy during disease diagnosis and treatment, anti-NMDAR encephalitis should be considered.…”

Anti-N-Methyl-D-Aspartate Receptor Encephalitis with Serum Anti-Thyroid Antibodies: A Case Report and Literature Review

“…This finding supported the hypothesis, previously suggested by Tuzun et al [ 26 ], that patients with Thy Abs are inclined to develop anti-neuronal-immune response and acute idiopathic encephalitis. A literature search of patients with the co-occurrence of anti-NMDArE and increased level of serum anti-Thy Abs is presented in Table 1 [ 26 – 31 ]. In particular, De Leu et al [ 27 ] and Lu J et al [ 28 ] described 2 patients with Graves’ disease; the remaining 5 patients had variable and different titers for Anti-Thy Abs, ranging from 58 IU/mL to 3000 IU/mL for TPO and from 92 IU/mL to 920 IU/mL for TG.…”