Kagawa Prefectural Central Hospital

“Belly dancer dyskinesia (BDD)” is a descriptive term for involuntary, undulating abdominal movements with diverse reported etiologies, leading to debate about its pathophysiology. Spinal myoclonus is attributed to the disinhibition of spinal central pattern generators (CPGs).1 While some argue for a clear distinction, significant phenomenological overlap exists, suggesting that it may represent a clinical mechanistic spectrum.2, 3 However, this hypothesis lacks definitive histopathological evidence. We present the first autopsy-proven case of BDD-like hyperkinetic movement disorder, providing direct evidence that the generator is localized in a severely inflamed brainstem and spinal cord. A 59-year-old man with no known immunodeficiency was admitted with a three-day history of progressive confusion. His examination was remarkable for continuous, rhythmic, 2–3 Hz wave-like contractions of the entire abdominal wall, phenomenologically consistent with BDD (Video 1). Critically, these movements persisted unabated under deep sedation using anti-seizure medications, benzodiazepines, and propofol. Cerebrospinal fluid (CSF) analysis revealed lymphocytic pleocytosis (125 cells/μL) and elevated protein levels, consistent with meningoencephalitis. His neurological status rapidly declined, requiring mechanical ventilation, and he died of sudden cardiac arrhythmia on day five. A postmortem CSF culture grew Mycobacterium intracellulare; however, as this was a single finding, its pathogenic role was uncertain, and contamination could not be definitively excluded. A comprehensive neuropathological examination was pivotal. Macroscopically, the brain and spinal cord appeared edematous. Microscopic examination revealed extensive lymphohistiocytic inflammatory infiltrates throughout the leptomeninges of the brainstem and the entire spinal cord. Lymphocytic infiltration with edema also involved subpial white matter that is anterior corticospinal, spinothalamic and spinocerebellar tracts, posterior funiculus (Fig. 1B). Lymphocytic infiltration cuffing along vascular externa extended deep into the parenchyma (Fig. 1C). Perivascular lymphocytic cuffing of spinal cord was prominent even in central gray matter and focally surrounded by spongiosis (Fig. 1A) though degeneration of neuronal body wasn't definitive. This distribution involves the anatomical location of the internuncial fibers and propriospinal neurons that constitute the spinal CPGs, rather than the primary somatic motoneurons of the limbs (Fig. 1A,C). The pontomedullary reticular formation was also affected by spongiotic change around perivascular lymphocytic cuffing. In contrast, the cerebral cortex, basal ganglia, and cerebellum were largely unaffected. This case uniquely correlated a clinical phenomenon with precise anatomical pathology. The persistence of movement under deep sedation effectively ruled out a cortical origin. Our pathological findings provide a direct anatomical substrate for this clinical observation, demonstrating severe inflammation and neuronal destruction in the brainstem-spinal cord networks known to contain CPGs. We postulate that the inflammatory process causes fatal disruption of supraspinal inhibitory pathways that normally regulate spinal CPGs. This disinhibition resulted in continuous, pathological, rhythmic motor output observed clinically. Our findings provide the first direct histopathological evidence that the BDD-like movements can be a manifestation of abdominal segmental myoclonus driven by a disordered brainstem-spinal cord CPG. This case supports the concept that these phenomena represent a spectrum of diseases unified by the disinhibition of intrinsic rhythmogenic circuits. This underscores that when a BDD-like phenotype is encountered, particularly with features suggestive of myoclonus, the structural etiology affecting the brainstem or spinal cord must be thoroughly investigated.4, 5 (1) Research project: A. Conception, B. Organization, C. Execution; (2) Manuscript: A. Writing of the first draft, B. Review and Critique. Y.T.: 1A, 1B, 1C, 2A, 2B. Y.N.: 1C, 2B. M.I.: 1C, 2B. K.N.: 2B. Y.S.: 2B. S.T.: 1C, 2B. Y.I.: 1C, 2B. H.I.: 1B, 2B. We thank Editage (www.editage.com) for English language editing. Ethical Compliance Statement: The authors confirm that the approval of an institutional review board was not required for this work. Informed consent for autopsy and for the publication of this case report, including clinical data and video, was obtained from the patient's next of kin. We confirm that we have read the Journal's position on issues involved in ethical publication and affirm that this work is consistent with those guidelines. Funding Sources and Conflict of Interest: No specific funding was received for this work. The authors declare that there are no conflicts of interest relevant to this work. Financial Disclosures for the previous 12 months: The authors declare that there are no additional disclosures to report. The data that support the findings of this study are available on request from the corresponding author. The data are not publicly available due to privacy or ethical restrictions.

Although mandibular odontogenic deep neck infections are occasionally fatal, the transmission pathway has not been elucidated. This multicenter retrospective study was comprised of the patients of both sexes who were over 18 years of age and who had mandibular odontogenic deep neck abscesses. The patients' characteristics, laboratory tests, and radiographic findings were analyzed. One hundred eighteen patients with mandibular odontogenic deep neck abscesses were included. Bone resorption superior to the mylohyoid line and the related abscess formation in submandibular space or submental space were both significantly associated with the presence of sublingual space abscess. In addition, the type of causative tooth was not a risk factor for abscess formation in both the sublingual space and "submandibular or submental" space. When an odontogenic lesion is located superior to the mylohyoid line, the abscess tends to initially form in the sublingual space and subsequently spread to the submandibular or submental space. Since any mandibular tooth can lead to abscess formation in these regions, oral and maxillofacial surgeons should carefully assess the anatomical position of the lesion and accurately identify the causative tooth.

Medication-related osteonecrosis of the jaw is currently classified only by clinical staging, with no imaging-based qualitative classification available. To establish foundational data for a new classification method that could help guide treatment decisions and predict prognosis, this study investigated the computed tomography findings commonly observed in patients with medication-related osteonecrosis of the jaw and their frequencies. Computed tomography scans from 784 patients across 19 medical institutions were analyzed for the presence of osteolysis, sequestrum separation, periosteal reactions, mixed-type osteosclerosis, and the bone-within-bone appearance. Clinical factors associated with each finding were also examined. Osteolysis appeared as localized in 55.1% of cases, extended in 27.7%, and advanced in 13.8%. In contrast, 3.4% of patients showed no osteolysis. This non-osteolytic pattern was more common among patients treated with denosumab. Sequestrum separation was seen in 34.7% of patients, most frequently among those with osteoporosis. Periosteal reactions were observed in 5.9% of cases as the attached type, 12.4% as the gap type, and 5.6% as the irregular type. Mixed-type osteosclerosis occurred in 31.9% of cases, most often in the mandibles of patients with malignant tumors. The bone-within-bone appearance was observed in 3.1% of cases. Patients with sequestrum separation had more favorable treatment outcomes. In contrast, those without osteolysis and those with periosteal reactions, mixed-type osteosclerosis, or bone-in-bone appearance were more likely to have poorer outcomes. This multicenter study clarified the prevalence of several key computed tomography features in medication-related osteonecrosis of the jaw, highlighting their potential relevance to clinical outcomes. These findings lay the groundwork for future research into their prognostic and therapeutic implications to support the development of a new imaging-based classification system.

Introduction: Early detection of duodenal neoplasms is crucial because of the complications associated with their treatment and their increasing recognition. However, standardised protocols for duodenal observation during screening esophagogastroduodenoscopy (EGD) have not been established. This study was performed to survey the current practical status of duodenal observation and to evaluate the clinical relevance of our systematic photo-documentation protocol, known as the Seven Pictures Rule (7 PR). Methods: A cross-sectional survey using a web-based self-administered questionnaire was conducted among endoscopists in a regional area of Japan. The questionnaire included items on respondents’ professional background, duodenal observation practices during screening EGD (observed locations, observation time, ampulla visualisation, and patient discomfort), as well as awareness, application, and perceived effects of the 7 PR, and preferences for future protocols. Data were anonymised and analysed descriptively. Results: Of the 99 endoscopists included, 96.7% reported observing the duodenum based on their own individual methods. The intentional observation rates by location were highest in the following order: bulb (99%), ampulla in descending (94%), contralateral ampulla in descending (84%), superior duodenal angle (77%), contralateral superior duodenal angle (79%), and transverse duodenum (33%). However, only 14% of respondents reported successfully observing the ampulla in more than 75% of cases. While 62% were aware of the 7 PR, only one-fourth had implemented it in practice. Notably, 43% of respondents familiar with the 7 PR felt that it had led to an increased detection of duodenal neoplasms. Conclusions: Current duodenal observation practices vary among endoscopists. The establishment of a systematic photo-documentation protocol such as the 7 PR may serve as a useful strategy for improving the detection of duodenal neoplasms. Further optimisation of the protocol is warranted to support broader adoption in clinical settings.

AimHeadache is one of the most frequent somatic complaints in psychiatric practice and is often attributed to underlying mental disorders. However, primary headache disorders—particularly migraine and tension‐type headache (TTH)—commonly coexist with psychiatric conditions. Evidence from psychiatric outpatient settings remains limited.MethodsWe conducted a retrospective chart review of all psychiatric outpatients who visited our 600‐bed regional general hospital between April 1, 2023, and March 31, 2024. Among 2525 patients, we identified 360 individuals with headache‐related insurance diagnoses and extracted data on headache labels, treating departments, and prescribed medications. For calcitonin gene–related peptide (CGRP)‐targeted monoclonal antibodies, we extended the observation period to March 31, 2025, to describe an exploratory case series including additional prescriptions.ResultsOf 2525 psychiatric outpatients, 360 (14.3%) carried a headache‐related insurance diagnosis. The most frequent labels were “headache” (203/360, 56.4%), migraine (92/360, 25.6%), and TTH (46/360, 12.8%); cluster headache and medication‐overuse headache (MOH) were each recorded in 1/360 (0.3%). Headache care was most often delivered within psychiatry (153/360, 42.5%), followed by neurology (42/360, 11.7%), neurosurgery (40/360, 11.1%), general internal medicine (28/360, 7.8%), and rheumatology/collagen‐vascular disease (15/360, 4.2%). Commonly documented agents included nonsteroidal anti‐inflammatory drug (NSAIDs) (40/360, 11.1%), acetaminophen (38/360, 10.6%), triptans (23/360, 6.4%), Japanese Kampo formulas (16/360, 4.4%), and CGRP monoclonal antibodies (6/360, 1.7%). At the agent level, acetaminophen (n = 38), loxoprofen (n = 33), zolmitriptan (n = 14), goreisan (n = 8), sumatriptan (n = 6), kakkonto (n = 6), diclofenac (n = 4), valproic acid (n = 4), and naratriptan (n = 3) were among the most frequently listed. In the exploratory CGRP analysis (total seven patients through March 31, 2025), six were women; the mean age was 48.4 ± 9.2 years. Psychiatric comorbidities were heterogeneous, including eating disorder, bipolar disorder, post‐traumatic stress disorder, dysthymia with social anxiety disorder, schizophrenia, autism spectrum disorder, and neurotic depression. All cases experienced headache improvement; two required switching to another CGRP agent for recurrent attacks yet maintained benefit. One patient temporarily discontinued due to a rash before resuming a different CGRP agent. In contrast, medium‐term changes in mood/anxiety were limited.ConclusionIn a psychiatric outpatient cohort, primary headaches were common and frequently managed within psychiatry. CGRP‐targeted therapy yielded headache relief even under psychiatric comorbidity, while psychiatric symptoms did not uniformly improve, underscoring the need for parallel mental‐health interventions alongside headache‐specific care. Strengthening cross‐specialty pathways and early headache evaluation within psychiatry are warranted.

Background and AimsOzanimod is a once-daily, oral, selective sphingosine 1-phosphate receptor 1 and 5 modulator. The objective of the randomized, phase 2/3 J-True North study (NCT03915769) was to assess the efficacy and safety of ozanimod in Japanese patients with moderately to severely active ulcerative colitis.MethodsIn the 12-week induction period (IP), patients were randomized 1:1:1 to receive placebo, ozanimod 0.46 mg, or ozanimod 0.92 mg. Patients who completed the IP with a clinical response at week (w) 12 were eligible to enter a 40-week maintenance period where they received the same treatment as they did in the IP. The primary endpoint was clinical response (complete Mayo score) at w12; clinical and mucosal secondary endpoints were assessed at w12 and w52.ResultsOf 198 patients randomized, 176 completed the IP. Of these patients, 97 entered and 77 completed the maintenance period. A significantly higher proportion of patients receiving ozanimod achieved clinical response at w12 versus placebo (ozanimod 0.46 mg: 52.9%, P = .0158; ozanimod 0.92 mg: 61.5%, P = .0006; vs placebo: 32.3%). Similar results were observed in the secondary endpoints where patients receiving ozanimod achieved higher rates of clinical remission, endoscopic improvement, and mucosal healing at w12 than those receiving placebo. Efficacy was maintained at w52 for all endpoints. Both doses of ozanimod were well tolerated, with no unexpected safety signals.ConclusionThis large-scale clinical trial demonstrated the efficacy and safety of once-daily oral ozanimod in Japanese patients with moderately to severely active ulcerative colitis. This is the first time that the efficacy and safety of ozanimod were verified in a large number of patients in Asia.

Cytokine storm syndrome (CSS), commonly associated with hemophagocytic lymphohistiocytosis (HLH), is a fatal hyperinflammatory syndrome. Differentiating the underlying diseases responsible for CSS is essential for timely therapeutic decisions. This study explored the clinical usefulness of serum cytokine profiling in distinguishing underlying diseases in patients with CSS. Serum samples were collected from 143 adult and pediatric patients with CSS and 22 healthy controls. The cohort included patients with various diagnoses of primary and secondary HLH and Kawasaki disease (KD)-like hyperinflammatory syndromes. Serum levels of 48 cytokines were analyzed in 97 patients using a bead-based multiplex immunoassay (Luminex assay). Serum levels of interferon alpha (IFN-α), interleukin-18 (IL-18), IL-6, CXCL9, and soluble tumor necrosis factor receptor II (sTNF-RII) were measured in 165 participants using enzyme-linked immunosorbent assay (ELISA). Luminex assay categorized patients with CSS into five clusters based on serum cytokine patterns. ELISA revealed distinct cytokine patterns, wherein patients with histiocytic necrotizing lymphadenitis-associated HLH and systemic lupus erythematosus-associated macrophage activation syndrome (MAS) showed elevated IFN-α; systemic juvenile idiopathic arthritis-associated and adult-onset Still's disease-associated MAS, X-linked inhibitor of apoptosis protein deficiency with HLH, and NLRC4-associated autoinflammatory disorder exhibited higher IL-18 levels. Additionally, KD shock syndrome had higher IL-6 levels than the other groups. CXCL9 was significantly elevated in patients with virus-associated HLH, familial HLH, malignant lymphoma-associated HLH, and KD-MAS. Multisystem inflammatory syndrome in children and toxic shock syndrome also showed moderate elevations of CXCL9 and IL-6 levels. Serum cytokine profiling effectively differentiates CSS subtypes, facilitating better diagnosis and personalized treatment strategies based on specific disease backgrounds.

Intertrochanteric femoral fractures are mainly managed by internal fixation. However, failures such as over-telescoping, cut-out, nonunion, or implant failure can occur, especially in osteoporotic elderly patients. We report the case of a patient in whom we performed artificial hip replacement surgery after fixation failure following internal fixation of an intertrochanteric femoral fracture. We report the case of an 85-year-old woman who sustained a left intertrochanteric femoral fracture treated with a dynamic hip screw (DHS). One week postoperatively, radiographs revealed over-telescoping of the lag screw. The patient did not complain of pain, but she underwent conversion to cemented bipolar hemiarthroplasty under general anesthesia. One possible cause of over-telescoping of the lag screw after surgery was that the longitudinal fracture line in the calcar of the proximal bone fragment, as seen in the initial CT image, may have extended horizontally at the neck level. During surgery, a fracture at the same site caused the anterior medial fragment to fail, resulting in a coronal shear fracture and fixation failure. When a longitudinal fracture line is observed in the calcar of the proximal fragment, it is necessary to keep in mind that it may extend horizontally at the neck level.