Phosphoglyceride Crystal Deposition Disease of the Scapula: A Case Report and Literature Review.

CASE: A 76-year-old woman presented with a 2-month history of right shoulder pain with no apparent cause. Radiography revealed an ill-defined osteolytic lesion in the right scapular spine with a pathological fracture. Malignant bone tumor was suspected, and a biopsy was performed. Pathological examination with gold hydroxamic acid staining revealed phosphoglyceride crystal deposition. Lesion curettage was performed, and her symptoms improved. No recurrence was observed at the 3-year postoperative follow-up. CONCLUSION: Phosphoglyceride crystal deposition in the bone is an extremely rare disease. The gold hydroxamic acid staining method might be useful for the diagnosis of this condition.

Spontaneous Regression of Multiple Pulmonary Metastasis of Sacral Chordoma: A Case Report.

CASE: A 53-year-old man was referred to our institution for the treatment of sacral chordoma and underwent a wide resection. Multiple lung metastases were observed in both the lungs, a year after the surgery, and the diagnosis was confirmed by biopsy. The patient refused treatment, and the lesions continued to increase in size gradually. However, 3 years and 6 months after the surgery, computed tomography of both the lungs showed spontaneous regression of the lesions without any obvious causes. The metastatic lung lesions had disappeared at the final follow-up, 7 years and 5 months after the multiple pulmonary metastases were diagnosed. CONCLUSION: We report the first case of spontaneous regression of pathologically proven pulmonary metastases of a sacral chordoma.

The 'other' bone sarcomas in Japan: a retrospective study of primary bone sarcomas other than osteosarcoma, Ewing sarcoma and chondrosarcoma, using data from the Bone Tumuor Registry in Japan.

BACKGROUND: To date, no standard treatments for primary bone sarcomas other than those for osteosarcoma, Ewing sarcoma and chondrosarcoma have been developed. METHODS: The clinical characteristics and prognostic factors of 330 patients with primary bone sarcomas other than osteosarcoma, Ewing sarcoma and chondrosarcoma, listed in a nationwide tumour registry (Bone and Soft Tissue Tumor Registry in Japan) were investigated. The effects of adjuvant chemotherapy were determined by comparing the outcomes of patients with non-metastatic bone tumours who received surgery plus chemotherapy with those of patients who underwent surgery alone. RESULTS: The most common diagnosis was undifferentiated high-grade pleomorphic sarcoma. Axial site tumours (47.8%) and distant metastases at presentation (24.5%) were frequent. The 5-year overall and progression-free survival rates were 44.9 and 39.9%, respectively. Prognostic factor analysis identified surgery as an independent predictor of overall survival, and distant metastases at presentation was significant and independent predictor of both overall and progression-free survival. No significant difference in outcome was observed between patients treated with surgery alone and those treated with surgery plus chemotherapy (P = 0.71). CONCLUSIONS: Patients with bone sarcomas other than osteosarcoma, Ewing sarcoma and chondrosarcoma without metastasis at presentation have a relatively good prognosis with definitive surgery; however, the benefit of adjuvant chemotherapy is unclear.

Life history and nesting ecology of a Japanese tube-nesting spider wasp Dipogon sperconsus (Hymenoptera: Pompilidae).

To clarify the life history of the Japanese spider wasp Dipogon sperconsus, bionomical studies using bamboo-cane trap nests were carried out in Japan. Based on weekly and consecutive daily surveys of trap nests and rearing of broods from collected nests, we evaluated the production of cells and eggs per day, prey spiders, and seasonal patterns of nesting activities. We found a relatively short critical period of switching from the summer generation into the overwintering generation. We also found that the voltinism is affected by the timing of egg production of the second generation in relation to this critical period. The developmental period for each generation and sex, voltinism and cell production per day were determined based on data for a large number of individuals for the first time.

The evaluation of lymph node cell proliferation response by liposomes loaded with major histocompatibility complex class II binding aquaporin 4 antigen peptide.

Autoimmune responses to aquaporin 4 (AQP4) cause neuromyelitis optica (NMO); thus, specific immunotolerance to this self-antigen could represent a new NMO treatment. We generated the liposome-encapsulated AQP4 peptide 201-220 (p201-220) to induce immunotolerance. Liposomes were generated using phosphatidylserine and the polyglycidol species PG8MG. The in vivo tissue distribution of the liposomes was tested using an ex vivo imaging system. To confirm the antigen presentation capacity of PG8MG liposomes, dendritic cells were treated with PG8MG liposome-encapsulated AQP4 p201-220 (AQP4-PG8MG liposomes). Immunotolerance induction by AQP4-PG8MG liposomes was evaluated using the ex vivo cell proliferation of lymph node cells isolated from AQP4 p201-220-immunized AQP4-deficient mice. Fluorescent dye-labeled PG8MG liposomes were distributed to the lymph nodes. AQP4 p201-220 was presented on dendritic cells. AQP4-PG8MG liposomes were tended to suppress immune responses to AQP4 p201-220. Thus, the encapsulation of AQP4 peptides in PG8MG liposomes represents a new strategy for suppressing autoimmune responses to AQP4.

Association of Hydrophobic Carboxyl-Terminal Dendrimers with Lymph Node-Resident Lymphocytes.

Delivery systems to lymph node-resident T cells around tumor tissues are essential for cancer immunotherapy, in order to boost the immune responses. We previously reported that anionic dendrimers, such as carboxyl-, sulfonyl-, and phosphate-terminal dendrimers, were efficiently accumulated in lymph nodes via the intradermal injection. Depending on the terminal structure, their cell association properties were different, and the carboxyl-terminal dendrimers did not associate with any immune cells majorly. In this study, we investigated the delivery of carboxyl-terminal dendrimers with different hydrophobicity to lymph node-resident lymphocytes. Four types of carboxyl-terminal dendrimers-succinylated (C) and 2-carboxy-cyclohexanoylated (Chex) dendrimers with and without phenylalanine (Phe)-were synthesized and named C-den, C-Phe-den, Chex-den, and Chex-Phe-den, respectively. Chex-Phe-den was well associated with lymphocytes, but others were not. Chex-Phe-den, intradermally injected at the footpads of mice, was accumulated in the lymph node, and was highly associated with the lymphocytes, including T cells. Our results suggest that Chex-Phe-den has the potential for delivery to the lymph node-resident T cells, without any specific T cell-targeted ligands.

Carboxyl-, sulfonyl-, and phosphate-terminal dendrimers as a nanoplatform with lymph node targeting.

The development of drug delivery vehicles to cancer and/or immune cells in lymph nodes is important for cancer diagnosis, therapy, and immunotherapy. We previously reported that anionic carboxyl-terminal dendrimers were accumulated in lymph nodes. In this study, three anionic dendrimers with carboxyl-, sulfonyl-, and phosphate-terminal groups were prepared to examine the lymph node targeting and the association with immune cells in the lymph nodes. These anionic dendrimers were accumulated in the lymph node by intradermal injection. Although the carboxyl- and sulfonyl-terminal dendrimers were diffused from the injection site, the phosphate-terminal dendrimers were mostly retained. The phosphate-terminal dendrimer was recognized by the macrophages, dendritic cells, and B cells in the lymph node, whereas the carboxyl- and sulfonyl-terminal dendrimers were not. Our results show that these anionic dendrimers were accumulated in the lymph node where the association with immune cells could be controlled by the terminal structure of the dendrimer. The phosphate-terminal dendrimer can be used as a nanoplatform for the delivery of some bioactive molecules to some immune cells, including B cells, in the lymph node.

Revision of the subgenus Stigmatodipogon Ishikawa of the genus Dipogon Fox (Hymenoptera: Pompilidae: Pepsinae).

The systematics of the subgenus Stigmatodipogon Ishikawa of the genus Dipogon Fox is revised. Six species are listed. Two new species, Dipogon (Stigmatodipogon) kurokawai Shimizu, sp. nov. (Honshu, Japan) and D. (S.) lao Loktionov Lelej, sp. nov. (Laos) are described and illustrated and other four species from Japan, the Russian Far East and Eastern Siberia are redescribed. A new combination is proposed for D. (S.) budrisi (Loktionov Lelej, 2014), comb. nov. Nesting records of D. (S.) kurokawai and prey records of D. (S.) macrostigmatus Ishikawa, 1959 and D. (S.) petiolatus Lelej, 1986 are presented. These are the first records of biology for this subgenus. A key to species is provided.

Gene Signature-Based Approach Identified MEK1/2 as a Potential Target Associated With Relapse After Anti-TNFα Treatment for Crohn's Disease.

Background: Anti-tumor necrosis factor alpha (anti-TNFα) therapy has become the mainstay of therapy for Crohn's disease (CD). However, post-therapy, the recurrence rate is still high. The aim of this study was to dissect the molecular mechanism for recurrence of CD treated with anti-TNFα therapy and investigate novel therapeutic options that could induce complete remission. Methods: We re-analyzed publicly available mucosal gene expression data from CD patients pre- and post-infliximab therapy to extract the transcriptional differences between responders and healthy controls. We used a systematic computational approach based on identified differences to discover novel therapies and validated this prediction through in vitro and in vivo experimentation. Results: We identified a set of 3545 anti-TNFα therapy-untreatable genes (TUGs) that are significantly regulated in intestinal epithelial cells, which remain altered during remission. Pathway enrichment analysis of these genes clearly showed excessive growth state and suppressed terminal differentiation, whereas immune components were clearly resolved. Through in silico screening strategy, we observed that MEK inhibitors were predicted to revert expression of genes dysregulated in infliximab responders. In vitro transcriptome analysis demonstrated that selective MEK1/2 inhibitor significantly normalized reference genes from TUGs. In addition, in vitro functional study proved that MEK1/2 inhibitor facilitated intestinal epithelial differentiation. Finally, using murine colitis model, administration of MEK1/2 inhibitor significantly improved diarrhea and histological score. Conclusions: Our data revealed the abnormalities in anti-TNFα responders' CD colons that would be cause of recurrence of CD. Also, we provided evidence regarding MEK1/2 inhibitor as a potential treatment against CD to achieve sustainable remission.

Fluorescence-labeled liposome accumulation in injured colon of a mouse model of T-cell transfer-mediated inflammatory bowel disease.

Drug delivery systems maximize the efficacy of drugs by improving their pharmacokinetic profiles, pharmacodynamic effects, or both and reducing their adverse effects. One of the most advanced, clinically available formulations are liposome-encapsulated drugs. In this study, we aimed to determine if liposomes can selectively deliver compounds in gastrointestinal diseases. Initially, we evaluated the correlation between the diarrhea score and accumulation of fluorescence (FL)-labeled liposome using in vivo imaging systems in various disease states of an inflammatory bowel disease mouse model. The result showed that FL-labeled liposome accumulation and colon tissue weight, which reflect the disease state were highly and positively correlated. Then, to confirm the accumulation of liposomes at injured sites of the colon, we administered both FL-labeled liposomes and luminescence probes for detecting reactive oxygen species (ROS) to the mouse model. The imaging data showed that liposome accumulation tended to coincide with ROS detected sites and the correlation coefficient indicated a significantly positive correlation between liposome accumulation and ROS detection levels. Finally, we evaluated the involvement of macrophages in the uptake mechanism of the liposomes by analyzing the relationship between FL-labeled liposome accumulation and macrophage marker gene expression levels. The result showed that the expression of each macrophage marker gene and liposome accumulation showed a significant positive correlation. Therefore, the macrophages considerably contributed to the uptake mechanism of the liposomes. These data suggest that liposomes could be an attractive delivery tool for enhancing the accumulation of drug candidates through macrophages in injured colonic tissues. This approach is expected to provide new treatment options for patients with colitis.

Suprascapular Nerve Entrapment Caused by Protrusion of an Intraosseous Ganglion of the Glenoid into the Spinoglenoid Notch: A Rare Cause of Posterior Shoulder Pain.

We describe a case of suprascapular nerve entrapment caused by protrusion of an intraosseous ganglion of the glenoid into the spinoglenoid notch. A 47-year-old man with left shoulder pain developed an intraosseous cyst in the left glenoid, which came into contact with the suprascapular nerve. The area at which the patient experienced spontaneous shoulder pain was innervated by the suprascapular nerve, and 1% xylocaine injection into the spinoglenoid notch under ultrasonographic guidance relieved the pain. Therefore, we concluded that the protrusion of an intraosseous cyst of the glenoid into the spinoglenoid notch was a cause of the pain, and performed curettage. Consequently, the shoulder pain was resolved promptly without suprascapular nerve complications, and the cyst was histologically diagnosed as an intraosseous ganglion. This case demonstrated that the intraosseous ganglion of the glenoid was a benign lesion but could be a cause of suprascapular nerve entrapment syndrome. Curettage is a useful treatment option for a ganglion inside bone and very close to the suprascapular nerve.

Primary bone sarcomas in patients over 40 years of age: A retrospective study using data from the Bone Tumor Registry of Japan.

BACKGROUND: Optimal treatments of patients aged >40 years with osteosarcoma and Ewing sarcoma and of patients with other types of bone sarcoma have not yet been established. METHODS: The clinical characteristics of 746 patients with primary bone sarcomas aged >40 years listed in the nationwide tumor registry (Bone and Soft Tissue Tumor Registry of Japan) were investigated. Patients with Ewing sarcoma, conventional chondrosarcoma, multiple myeloma, and malignant lymphoma were excluded. The effects of adjuvant chemotherapy were determined by comparing outcomes in patients aged 40-70 years with non-metastatic high grade bone tumors who received surgery plus chemotherapy with those who underwent surgery alone. RESULTS: Axial site tumors (38.6%) and distant metastasis at presentation (21.4%) were frequent in patients aged >40 years with primary bone sarcomas. Chemotherapy was administered to 407 (54.6%) patients. Their 5-year overall survival (OS) and progression free survival (PFS) rates were 47.6% and 39.8%, respectively. Multivariate analysis showed that distant metastases at presentation, histological high grade, and radiation therapy were significantly correlated with both OS and PFS. Age and tumor size were also significantly correlated with OS. Patients aged 40-70 years who underwent surgery plus chemotherapy and those who underwent surgery alone had 5-year OS rates of 69.3% and 63.2%, respectively (p = 0.092), and 5-year PFS rates of 55.4% and 50.7%, respectively (p = 0.63). CONCLUSIONS: Bone sarcoma patients aged >40 years had distinct clinical features and a poor prognosis. Chemotherapy did not improve OS or PFS in patients aged 40-70 years.

Metastatic Prostate Cancer of Hand.

Soft tissue metastases of prostate cancer to other sites are extremely rare, and, to our best knowledge, there have been no reports of metastasis to soft tissue of the hand. A 63-year-old man was diagnosed with prostatic cancer. During treatment, bone and soft tissue metastases to the right hand, appearing in the first web space, were observed. The tumor was resected, along with both the first and second metacarpal bones. The thumb was reconstructed by pollicization of the remaining index finger, enabling the patient to use the pollicized thumb for activities of daily living. This is the first case report of prostate cancer metastasizing to the soft tissue in hand. After wide resection, pollicization was able to reconstruct a functional hand and thumb.

Co-occurrence of ecologically equivalent cryptic species of spider wasps.

Many cryptic species have been discovered in various taxonomic groups based on molecular phylogenetic analyses and mating experiments. Some sympatric cryptic species share equivalent resources, which contradicts the competitive exclusion principle. Two major theories have been proposed to explain the apparent lack of competitive exclusion, i.e. niche-based coexistence and neutral model, but a conclusive explanation is lacking. Here, we report the co-occurrence of cryptic spider wasp species appearing to be ecologically equivalent. Molecular phylogenetic analyses and mating experiments revealed that three phylogenetically closely related species are found sympatrically in Japan. These species share the same resources for larval food, and two of the species have the same niche for nesting sites, indicating a lack of competitive exclusion. This evidence may suggest that ecologically equivalent species can co-occur stably if their shared resources are sufficiently abundant that they cannot be over-exploited.

Malignant solitary fibrous tumor of the lumbar spinal root mimicking schwannoma: a case report.

BACKGROUND CONTEXT: Malignant solitary fibrous tumors (SFTs) arising from the spinal cord are extremely rare and poorly understood mesenchymal neoplasms. To date, only one malignant SFT located in the spinal canal of the sacrum has been described, but none arising from the lumbar nerve root have been reported. Although most SFTs with benign histological features can be treated by complete surgical excision alone, malignant SFTs may require adjuvant therapy. However, systemic chemotherapy and radiotherapy have not been shown effective in patients with malignant SFTs. PURPOSE: To describe a patient with a malignant SFT arising from the lumbar nerve root. STUDY DESIGN: A case report and review of literature. METHODS: We describe the clinical course of the patient and the radiological and pathological findings of the tumor. The effect of systemic chemotherapy was evaluated and the relevant literature was reviewed. This work has no disclosure of funding and was approved by the Institutional Review Board of Gifu University. RESULTS: The tumor had been resected previously at another hospital, but it recurred and showed multiple metastatic lesions on both lungs within 3 months. Although the patient received systemic chemotherapy, both primary and metastatic lesions were found to be stable disease according to Response Evaluation Criteria in Solid Tumors. The patient died due to cachexia 6 months after her first visit. CONCLUSION: This patient presented with a highly unusual tumor. Even if a tumor is a dumbbell-shaped mass, similar to a neural tumor, SFT should be considered in the differential diagnosis.

A wheelchair operation assistance control for a wearable robot using the user's residual function.

Cervical Cord Injury (CCI) is a dysfunction of the upper limb. In an individual with C5-level CCI, which is the most frequent of all eight levels, force can be applied in the direction of flexion by the biceps brachii, but extension force cannot be applied because of the triceps brachii paralysis. Persons with C5-level CCI therefore cannot operate a wheelchair up an incline and over carpet. In this study, we estimated the wheelchair velocity during elbow flexion depending on the angular velocity of the elbow. A wearable assistive robot can assist with the elbow extension movement using this estimated velocity while the wheelchair is being operated.

Control of wearable motion assist robot for upper limb based on the equilibrium position estimation.

In this paper, we propose a robotic system for assisting patients who have upper limb dysfunction in performing reaching movements through flexion. Since upper limb motion is more strongly needed than lower limb mobility for near work, a patient's level of recovery of upper limb function influences daily life. Recently, with the widespread application of robotic technology in rehabilitation medicine, active movement has often been noted to be more important than passive movement for rapid recovery. A novel control method for assisting upper limb movement by using a control system with two degrees of freedom is proposed. In the process of estimating the trajectory, the minimum jerk criterion is used to compute the velocity trajectory and to determine the reach position. The aim is to eventually develop a movement assistance system for the upper limb which will enable wearers to perform flexion and extension covering ranges of motion which are otherwise impossible to achieve autonomously. The effectiveness of the developed system is demonstrated experimentally.

Is "black geode" sign a characteristic MRI finding for extracranial schwannomas?

PURPOSE: To evaluate whether the "black geode" sign is a characteristic magnetic resonance imaging (MRI) finding for extracranial schwannomas. MATERIALS AND METHODS: Forty-three patients with pathologically confirmed extracranial schwannomas underwent preoperative gadolinium-enhanced MRI. The black geode sign was defined as the appearance of enhanced outer and inner rings. MR images were retrospectively reviewed for size, configuration, and signal intensity of the lesions in addition to the presence of the black geode sign. RESULTS: Gadolinium-enhanced T1-weighted images revealed the black geode sign in seven of 43 patients (16%). The thickness of inner rings (mean 0.6 cm, range 0.3-0.8 cm) was significantly greater than that of outer rings (mean 0.2 cm, range 0.1-0.3 cm) (P < 0.01). While outer rings were circular or elliptical in shape with smooth contours, inner rings had a lobular configuration with irregular thickness and contours. The degrees of enhancement were significantly stronger with inner rings than with outer rings (P < 0.01). In histopathological correlation of five patients who underwent total excision, inner and outer rings corresponded to peridegenerative areas and fibrous capsules, respectively. CONCLUSION: The black geode sign may be fairly specific to extracranial schwannomas on gadolinium-enhanced MR images.