Acute nonbacterial pleuritis caused by spontaneous rupture of metastatic pulmonary adenocarcinoma

Nonbacterial pleuritis caused by rupture of a metastatic adenocarcinoma is extremely rare and has not yet been reported. A 59-year-old man with a history of rectal cancer surgery 6 years earlier presented with a solitary lung mass in the periphery of the right lower lobe on computed tomography. Transbronchial biopsy disclosed a suspected metastatic adenocarcinoma, and he was admitted for pulmonary metastasectomy. He had been asymptomatic, but 4 weeks after the diagnostic bronchoscopy, the patient suddenly complained of a right back pain and dyspnea at rest; shortly after that, he developed a fever of 39 degrees C. A chest X-ray showed right pleural effusion, collapse of the right lower lobe, and elevation of the right diaphragmatic dome, but without pneumothorax. Emergent video-assisted thoracoscopy revealed a perforated tumor in the collapsed lower lobe, and a right lower lobectomy was carried out. The postoperative course was uneventful, and he was well without recurrent disease 2 years after pulmonary resection.     

Analyses of the transglutaminase 1 gene mutation and ultrastructural characteristics in a Japanese patient with lamellar ichthyosis

We described a Japanese female with lamellar ichthyosis whose transglutaminase 1 gene (TGM1 gene) was mutated. DNA sequence analysis revealed that the patient had a homozygous mutation, i.e. a point mutation from G to A at nucleotide 1494 resulting in the substitution of glycine for arginine at codon 143. Her mother was heterozygous for this mutation. In situ transglutaminase assay in the patient's skin showed loss of enzyme activity. Ultrastructural examination revealed incomplete formation of cornified cell envelopes and electron-dense materials adjacent to plasma membranes. These results suggest that defective transglutaminase activity caused by homozygous TGM1 gene mutation (G143R) results in disruption of cornified envelope assembly and the clinical phenotype of lamellar ichthyosis.     

Atypical cervical astrocytoma manifesting as occipitalgia

A 23-year-old female presented with chronic occipitalgia without signs of increased intracranial pressure followed by worsening headache and slight gait unsteadiness. Cerebral magnetic resonance (MR) imaging showed no corresponding lesions. Cervical MR imaging revealed a cervical intramedullary tumor. Intraoperatively the subpial tumor was found to stretch the 3rd-5th dorsal nerve roots posteriorly, which was thought to cause the intolerable headache. Total tumor resection was achieved without requiring myelotomy using electrophysiological monitoring with somatosensory and motor evoked potentials. Histological examination identified diffuse astrocytoma. Postoperatively the headache had completely resolved. Cervical astrocytoma of subpial location is a very rare cause of headache in adults. The subpial location enables surgical extirpation because minimal or no myelotomy is needed.     

Orbital pseudotumor associated with retrobulbar hematoma: case report

A 75-year-old male suffered sudden onset of retroorbital pain, visual loss, periorbital ecchymosis, and double vision without preceding trauma, paranasal sinus surgery, or infectious signs. Neuroophthalmological inspection also revealed marked restriction of the extraocular movements, visual defect, and exophthalmos on the affected side. Neuroimaging showed an irregular-shaped retrobulbar mass centered in the inferolateral aspect and partially protruding between the inferior and lateral rectus muscles without enhancement by contrast medium. High dose steroid therapy provided little improvement in the visual symptoms. Needle aspiration biopsy revealed only fluid hematoma. Surgical exploration via the lateral wall of the orbit resulted in escape of chocolate-colored, liquefied hematoma during dissection between the inferior and lateral rectus muscles. No obvious vascular lesion was recognized. A small purplish elastic soft mass with irregular contours was recognized adherent to the inferior rectus muscle within the capsule of the hematoma. The mass was subtotally resected. The histological diagnosis was inflammatory pseudotumor with hemorrhagic change. No component of vascular malformation was found. Visual function and extraocular movement improved postoperatively and neuroimaging showed no abnormal structures in the affected orbit. Pseudotumor may bleed and form a retrobulbar hematoma which carries the risk of visual loss.     

Takotsubo syndrome after a minimally invasive tooth extraction in a patient without dental phobia: A case report

Takotsubo syndrome (TTS) is a rare, stress-induced acute cardiac disorder. Its precipitating factors include emotionally or physically stressful events and exogenous and endogenous adrenaline. In this report, we describe a case of atypical TTS in a 73-year-old woman who reported no dental fear and required acute cardiac care in an outpatient setting. She underwent routine extraction of an upper left premolar under local anesthesia. She reported heart palpitations after the injection, and the procedure was completed in 15 min. After presenting symptoms of sweating, pale skin, vomiting, low blood pressure, and ST-segment elevation, cardiologists ordered echocardiography, coronary angiography, and ventriculography. Upon receiving a TTS diagnosis, the patient was hospitalized and administered an intra-aortic balloon pump and beta-blocker. Her symptoms resolved, and she was discharged with no sequelae. We found no precipitating factors in the progression of TTS in this case, which suggests that TTS can develop in the absence of precipitating factors. All general dentists and oral surgeons should recognize the possible risk of TTS, even during minimally invasive dental procedures, such as routine extractions in patients without dental phobia.     

CTLA4-Ig Directly Inhibits Osteoclastogenesis by Interfering With Intracellular Calcium Oscillations in Bone Marrow Macrophages

CTLA4-Ig (cytotoxic T-lymphocyte antigen 4-immunoglobulin; Abatacept) is a biologic drug for rheumatoid arthritis. CTLA4 binds to the CD80/86 complex of antigen-presenting cells and blocks the activation of T cells. Although previous reports showed that CTLA4-Ig directly inhibited osteoclast differentiation, the whole inhibitory mechanism of CTLA4-Ig for osteoclast differentiation is unclear. Bone marrow macrophages (BMMs) from WT mice were cultured with M-CSF and RANKL with or without the recombinant mouse chimera CTLA4-Ig. Intracellular calcium oscillations of BMMs with RANKL were detected by staining with calcium indicator fura-2 immediately after administration of CTLA4-Ig or after one day of treatment. Calcium oscillations were analyzed using Fc receptor gamma- (FcRγ-) deficient BMMs. CTLA4-Ig inhibited osteoclast differentiation and reduced the expression of the nuclear factor of activated T cells NFATc1 in BMMs in vitro. Calcium oscillations in BMMs were suppressed by CTLA4-Ig both immediately after administration and after one day of treatment. CTLA4-Ig did not affect osteoclastogenesis and did not cause remarkable changes in calcium oscillations in FcRγ-deficient BMMs. Finally, to analyze the effect of CTLA4-Ig in vivo, we used an LPS-induced osteolysis model. CTLA4-Ig suppressed LPS-induced bone resorption in WT mice, not in FcRγ-deficient mice. In conclusion, CTLA4-Ig inhibits intracellular calcium oscillations depending on FcRγ and downregulates NFATc1 expression in BMMs. © 2019 American Society for Bone and Mineral Research.     

Serum interleukin 8 levels correlate with synovial fluid levels in patients with aseptic loosening of hip prosthesis

Levels of inflammatory cytokines (tumor necrosis factor alpha, interleukin [IL] 6, and IL-8) in serum from patients with osteolysis on radiographs after hip arthroplasty [osteolysis(+), n = 28], patients without osteolysis after hip arthroplasty [osteolysis(-), n = 24], and nonoperated healthy subjects [controls, n = 20] were determined. In addition, cytokine levels in synovial fluid from patients undergoing revision total hip arthroplasty (n = 14) for loosening were measured and compared with each other and with the area of osteolysis on radiographs. Serum IL-6 and IL-8 levels were significantly higher in the osteolysis(+) group than in the osteolysis(-) or the control groups. Furthermore, a significant correlation was found between the serum and synovial fluid IL-8 levels and between synovial fluid IL-8 levels and the area of osteolysis in patients undergoing revision total hip arthroplasty. Therefore, serum IL-8 levels could be a useful periprosthetic osteolysis marker.