菅原 斉

Aggregatibacter actinomycetemcomitans, an etiological agent associated with periodontitis, endocarditis, and other infections, has rarely been implicated in spondylitis. A 70-year-old man with aortic valve replacement presented with a 4-month history of lower back pain and was diagnosed with spondylitis. Prolonged incubation of blood cultures and a biopsy yielded A. actinomycetemcomitans. Concurrent infective endocarditis (IE) was probable considering the infectious organism and the patients' prosthetic valve. The patient was treated with ceftriaxone and recovered well. Pyogenic spondylitis with possible concurrent IE may be caused by A. actinomycetemcomitans. Extended incubation and repeated cultures should be considered if HACEK infection is suspected.

A 27-year-old woman complaining of persistent fever and sore throat was consulted. Examination of the laryngopharynx revealed multiple aphthous ulcers in the uvula, arytenoids, epiglottis, and laryngeal pyriform fossa. Crohn's disease was diagnosed by colonoscopy.

A 67-year-old man presented with a 15-year history of chronic urticaria. It was distributed symmetrically on the limb trunk (Fig. 1A and B) without pruritus. No fever or joint pain or headache is observed. He showed no signs of angioedema. He was referred to our hospital for treatment of chronic urticaria. An antihistamine was ineffective. He was diagnosed with mild sensorineural hearing loss. He had no family history of autoinflammatory diseases.

We present a case of diffuse aspiration bronchiolitis (DAB) with a false‐positive Gaffky score. “Tree‐in‐bud” opacities detected after aspiration should be considered DAB rather than mycobacterial infection.

It is important to suspect intracranial hypotension based on distinctly frequent orthostatic headaches and diffuse dural hyperplasia. Lumbar puncture is a procedure prone to complications, especially in patients with already existing intracranial hypotension.

An 82‐year‐old man with untreated diabetes mellitus (DM) had anterior chest wall swelling and ulcers 2 years following blunt chest trauma. Contrast‐enhanced computed tomography revealed sternal fracture with osteolytic change and subcutaneous abscess. Blood and sternal cultures were positive for methicillin‐susceptible Staphylococcus aureus (MSSA). Transesophageal echocardiogram showed vegetation on the right coronary cusp and moderate aortic regurgitation. The patient received a diagnosis of infective endocarditis associated with chronic sternal osteomyelitis complicated by subcutaneous abscess because of MSSA. This case report showed that trivial trauma in patients with uncontrolled DM can cause chronic sternal osteomyelitis resulting in infective endocarditis.

Peritonsillar abscess should be considered as a differential in patients presenting with fever, sore throat, and other cold‐like symptoms. Point‐of‐care transcutaneous cervical ultrasound is effective for diagnosing peritonsillar abscess in place of computed tomography (CT) imaging. Tongue movement during ultrasound examination will help confirm the presence of anatomical lesions on tonsils, and comparing the affected and unaffected sides will improve proper diagnosis.

A 49‐year‐old‐Japanese woman visited us with a 1‐year history of verrucous lesions on the oral mucosa and fingers. Her past medical history was unremarkable except for chilblain in winter. She had no family history of collagen disease. Physical examination showed a well‐defined, hyperkeratotic erythematous plaque with ulceration on the palate and verrucous nodules on the fingers (Fig. 1a,b). She had no butterfly rash or discoid lesions on any other sites. Histology from both oral mucosa and finger revealed similar features; marked hyperkeratosis and acanthosis with perivascular lymphocytic infiltrates in the dermis. Liquefaction degeneration with a few necrotic keratinocytes was also found (Fig. 1c,d). We regarded the patient as verrucous lupus erythematosus (LE). Then, leukopenia (2,820/μl) and thrombocytopenia (67,000/μl) were observed as well as positive antinuclear antibody (ANA). However, the diagnosis of systemic lupus erythematosus (SLE) was not confirmed by the negative finding of anti‐DNA antibody, and the lack of requirements in 1997 updated ACR criteria for SLE validated back at that time. She developed skin ulcers on the left lower leg in the next two months (Fig. 2a). Four months later, laboratory examination revealed leukopenia (2,850/μl), thrombocytopenia (89,000/μl), low C3/C4 level (27 mg/dl, 3 mg/dl, respectively), positivity for ANA (1 : 640, speckled type), and anti‐Sm antibody (1 : 2). Histology of the leg ulcer exhibited no vasculitis but the occlusion of the vessels (Fig. 2b). Direct immunofluorescence revealed IgM, C1q, and C3 depositions on vessel walls as well as linear IgM deposition at basement membrane zone (Fig. 2c). Seven months later, she had seizure attacks. Then, SLE was diagnosed based upon ACR (1997) and SLICC criteria. Even under 2019 EULAR/ACR classification criteria,1 she was classified as SLE from total 24 points scored by the findings of fever (38.9 °C), leukopenia, thrombocytopenia, seizure, low C3/C4, and anti‐Sm antibody. Then, the patient was transported to us for dyspnea and high fever. Methylprednisolone pulse therapy was performed. Chest roentgenogram revealed extensive lung infiltrates. Aspergillus fumigatus was isolated from the sputum. Invasive aspergillosis was diagnosed. She died of multiple organ failure 8 months after the first visit.

A 66-year-old woman complaining of painless bulging in the right flank abdomen exhibited redness with belt-like scab formation in her right lower quadrant (Th11-12 level).

A 77-year-old man visited the hospital with a chronic cough persisting for 2.5 months accompanied with night sweats, weight loss (3.5 kg) and elevated C-reactive protein level. Chest CT of the lung field was normal, but aortic wall thickening accompanied by a contrast effect was noted. Positron emission tomography–CT (PET–CT) showed that the aorta and subclavian artery were inflamed, suggesting large-vessel vasculitis. Ultrasonography showed thickening of the superficial temporal artery wall (macaroni sign). Biopsy revealed lymphocytic infiltration in the tunica media and foreign-body giant cell reaction with the elastic lamina, resulting in a diagnosis of giant cell arteritis (GCA). The cough was considered a symptom of GCA as it resolved following prednisolone administration. Cough may rarely be an initial GCA symptom. However, for chronic cough accompanied with elevated inflammatory findings but with a normal lung field, imaging studies such as PET–CT are useful for the differential diagnosis.

A 66-year-old man complained of lower limb edema was diagnosed with mesenteric panniculitis because of fat ring sign and tumoral pseudocapsule sign on contrast-enhanced CT. Patient had edema due to protein-losing enteropathy associated with mesenteric panniculitis.

Background: Brucellosis, an important zoonotic disease, is endemic in various parts of the world. Patients diagnosed with brucellosis in developed countries are often travelers and immigrants from endemic areas. This pathogen is listed as biosafety level 3, which means that it is highly contagious and therefore a risk to clinical laboratory technicians. Case presentation: A 43-year-old Chinese man, who could not understand Japanese, visited our hospital because of an intermittent fever that had persisted for 5 months. Associated symptoms included muscle pain whenever he had a fever. He reported currently working as a welder in Japan. However, his previous employment working in animal husbandry in Heilongjiang, mainland China was not determined at the initial visit owing to language barriers. Two sets of blood culture showed nonfermenting gram-negative bacilli, initially misidentified as Ochrobactrum anthropi and subsequently identified as Brucella abortus. Six-week doxycycline and rifampicin were administered, with intravenous gentamicin for the initial 1 week. The patient recovered without relapse, confirmed by the negative result of a Brucella agglutination test. The patient’s wife and three laboratory technicians were required to undergo blood examinations, which revealed no evidence of infection; however, they received prophylaxis with 3 weeks’ doxycycline and rifampicin. Conclusion: In nonendemic countries, immigrants with imported brucellosis can be treated, to prevent secondary brucellosis infection, an occupational hazard among laboratory technicians. Greater attention is needed for positive findings of blood cultures, which may initially be misidentified as O. anthropi. When providing medical care for immigrants with fever of unknown origin, it is especially important for primary care physicians to overcome language barriers so as to assess pertinent information regarding their home country, such as previous employment, to prevent the spread the imported zoonoses in the era of a dramatically increasing number of immigrants and foreign travelers.

Essential thrombocythaemia (ET) is characterised by elevated platelet count by a clonal stem cell disorder of megakaryocytes. Although thrombosis is a common complication of ET, splenic infarction (SI) is extremely rare. Here, we present the case of a 31-year-old Japanese man who presented with sudden-onset severe pain at the left hypochondrium on the day before admission. Enhanced abdominal CT revealed SI. The laboratory test results revealed a normal platelet count (439×10⁹/L). Subsequently, the patient was diagnosed with ET because the platelet count gradually increased to 50.0×10⁴/μL, and <italic>JAK2 V617F</italic> mutation was identified. Accordingly, low-dose aspirin was initiated, and no thrombotic episode occurred. Nevertheless, 6 months postdischarge, the platelet count gradually increased to &gt;650 × 10⁹/L, and anagrelide was initiated. This case demonstrates an unusual complication of acute SI due to ET under the rare situation of the normal platelet count.

BACKGROUND: Systemic vasculitides constitute heterogenous conditions affecting many organs and systems through blood vessel inflammation. Although there are some classifications for vasculitis, several vasculitides are “unclassified” because they cannot be clearly assigned to one of the known entities. CASE REPORT: We report an autopsy case of a 67-year-old Japanese man who presented with fever, ocular pain, erythema, chest pain, and headache. The disease caused episcleritis, thrombophlebitis, extensive deep vein thrombosis, multiple pulmonary nodules and masses, hypertrophic pachymeningitis, and hyper-intensity areas in brain parenchyma on magnetic resonance images. Histopathology of the pulmonary nodule confirmed vasculitis affecting medium-to-small veins and arteries without necrotizing vasculitis or granulomatous inflammation. We diagnosed the patient with unclassified vasculitis based on the clinicopathological characteristics. Steroids in combination with immunosuppressants were used, but the disease was refractory and relapsing. The disease activity was eventually controlled with rituximab, but the patient died of bronchopneumonia. On autopsy, lung and brain findings indicated healed vascular inflammation. CONCLUSIONS: This is the first case report of unclassified vasculitis, which is characterized as medium-to-small-sized arteritis and phlebitis, causing episcleritis, thrombophlebitis, deep vein thrombosis, pulmonary vasculitis, and intracranial vasculitis. The clinical conditions share some similarities with granulomatosis with polyangiitis and Behçet’s disease; however, they meet no classification criteria of any specific vasculitis. More cases need to be analyzed to confirm our findings.

Our case patient was a 38-year-old pregnant Japanese woman who underwent emergency Caesarean section because of massive vaginal bleeding due to a low-lying placenta. Immediately after delivery, she presented with rapidly progressive dyspnea. Contrast-enhanced computed tomography revealed bilateral pleural effusion, lung nodules, multiple liver tumors, and multiple osteolytic lesions. Accordingly, epidermal growth factor receptor-mutant advanced lung adenocarcinoma was diagnosed. This report highlights the occurrence of rapid progression of lung cancer following delivery that led to postpartum acute respiratory failure, rather than due to pulmonary thromboembolism associated with the existing deep venous thrombosis of the inferior vena cava.

Objectives: Symptomatic cerebral infarction (CI) can occur in patients without main cerebral artery stenosis or occlusion. This study investigated the unique features of carotid artery plaque and white matter disease (WMD) in patients with symptomatic CI and transient ischemic attack (TIA) but without stenosis or occlusion of a main cerebral artery. Patients and methods: We studied 647 patients who underwent both carotid ultrasound examination and brain magnetic resonance images. Plaque score (PS), plaque number, maximal plaque intima-media thickness and grades of WMD were examined. Subjects were divided into four groups, the CI group, TIA group, myocardial infarction (MI) group and risk factor (RF) group. Plaque and WMD were analyzed in cerebral ischemia group (CI and TIA), compared to non-cerebral ischemia groups and to a high PS group and a high WMD grade group from the RF group. Results: Both of each value of plaque and grades of WMD in the cerebral ischemia group were significantly higher than those in other groups. Grades of WMD in the cerebral ischemia group were significantly higher than those in the high PS group, although there was no significant difference of the each value of plaque between the two groups. The each value of plaque in the cerebral ischemia group was also significantly higher than those in the high WMD grade group, although there was no significant difference of grade of WMD between the two groups. Conclusion: Simultaneous increases in carotid artery plaque and WMD are associated with symptomatic CI, which is not caused by stenosis or occlusion of a main cerebral artery.

The causes of chronic unilateral leg swelling include common manifestations such as venous insufficiency, varicosis, deep vein thrombosis, and persistent cellulitis, less commonly are secondary lymphedema (tumor, radiation, and surgery), pelvic tumors or lymphoma causing pressure on the veins, and reflex sympathetic dystrophy, and rarely congenital venous malformation, pregnancy, and iliac vein compression syndrome. Here, we describe a 78‐year‐old nonsmoking man with history of type 2 diabetes mellitus, dyslipidemia, and hypertension presented with left leg swelling that had been getting worse over several months.

A D-shaped left ventricle (D-LV), predominantly during systole, on a short axis view of two-dimensional echocardiography indicates pulmonary hypertension(PH). Here we describe a rare case involving a 35-year-oldmale smoker who developed typical D-LV secondary to fatal pulmonary Langerhans cell histiocytosis(PLCH)-related PH (PLCH-PH). To the best of our knowledge, there is no article to exhibit the image of D-LV caused by PLCH -PH.