Extensive chronic perianal pyoderma associated with persistent inflammation caused by wearing the same unwashed underwear for more than ten years.

Key Clinical Message: Poor personal hygiene wearing the same unwashed briefs, and prolonged sitting have led to the development of chronic perianal pyoderma. This can be confused with hidradenitis suppurativa and must be differentiated as their treatments are different. Abstract: There are potential risks of persistent inflammation resulting from poor personal hygiene. This comprises wearing the same unwashed briefs and prolonged sitting posture that led to developing chronic perianal pyoderma (CPP) in a smoking man. CPP can be confused with hidradenitis suppurativa, requiring differentiation as their treatment strategies distinctly differ.

Malignancy-related Hypercalcemia Caused by Metameric Cutaneous Metastasis of Parathyroid Hormone-related Protein-producing Bladder Carcinoma with Squamous Cell Differentiation: An Autopsy Case of Cobb Syndrome.

A 74-year-old woman was admitted with hypercalcemia and prolonged disturbance of consciousness. The left buttock to the anterior aspect of the left thigh was swollen and erythematous, with a collection of 1.0-cm large, firm, elastic nodules distributed in a zosteriform pattern in the L1-L4 region. Based on autopsy findings, a very rare case of Cobb syndrome was diagnosed due to a spinal vascular malformation at the Th12-L4 level and L5 vertebral hemangioma. Cobb syndrome-associated cutaneous metastasis extending along the same metamere was complicated by immunohistochemically proven parathyroid hormone-related protein-producing advanced bladder carcinoma in this case.

Tacrolimus Induction Therapy for Nephrotic Syndrome Caused by Minimal Mesangial Lupus Nephritis with Lupus Podocytopathy: A Case-Based Review.

BACKGROUND Nephrotic syndrome caused by minimal mesangial lupus nephritis is considered rare. Nephrotic syndrome can be caused by minimal mesangial lupus nephritis with diffuse epithelial foot-process effacement and lupus podocytopathy. CASE REPORT A 23-year-old Japanese woman diagnosed with mixed connective tissue disease was admitted because of weight gain and generalized edema for 2 weeks prior to admission. She had butterfly-shaped erythema on her cheeks, proteinuria, leukocytopenia with lymphocytopenia, and hypoalbuminemia. She was positive for antinuclear antibodies, and specific autoantibodies were only positive for the ribonucleoprotein (RNP) antigen. She was diagnosed with systemic lupus erythematosus. Renal biopsy showed minor glomerular abnormalities, and immunofluorescence revealed peripheral deposits of IgM and complement C3c. Electron microscopy revealed diffuse podocyte foot-process effacement of >80% of the capillary loop surfaces, with only a few subendothelial deposits. Consequently, we diagnosed minimal mesangial lupus nephritis with lupus podocytopathy. On hospital day 4, we administered 1000 mg/day of methylprednisolone for 3 days, followed by prednisolone 50 mg/day, but proteinuria persisted. On day 12, we administered tacrolimus (3 mg/day). Proteinuria improved and then disappeared on day 17. Prednisolone was gradually tapered and stopped after 3 years, although tacrolimus 3 mg/day was continued. No flare-up was observed 4 years after admission. CONCLUSIONS Tacrolimus showed good efficacy in this case of minimal mesangial lupus nephritis with lupus podocytopathy. Prospective and randomized controlled trials should be conducted to demonstrate the efficacy of tacrolimus for this indication.

Trajectory of SARS-CoV-2 anti-S IgG levels following transfusion and a third dose of BNT162b2 vaccine in a patient with massive postoperative bleeding: A case report.

OBJECTIVE: Vaccination against SARS-CoV-2 has been shown to be effective in preventing infection and severe disease. Massive bleeding and transfusion after vaccination can lead to a decrease in the antibody level. The effect of an additional dose of vaccine after blood transfusion has not been described previously. In this case report, we report the SARS-CoV-2 anti-S IgG trajectory in a male patient who received a third dose of vaccine after a massive postoperative bleed and blood and plasma transfusion. CASE PRESENTATION: A 57-year-old male physician had a SARS-CoV-2 anti-S IgG level of 44 AU/mL, measured using the Lumipulse Presto chemiluminescence assay 3 months after receiving 2 doses of the BNT162b2 vaccine. The patient underwent a hemicolectomy for colon cancer, experienced massive postoperative bleeding, and required a transfusion. The patient's SARS-CoV-2 anti-S IgG level dropped to 9.2 AU/mL. A third dose of BNT162b2 vaccination was administered to reduce the risk of breakthrough infection. Fifteen days after receiving the third vaccine dose, the patient's SARS-CoV-2 anti-S IgG level increased to 421 AU/mL, likely to reflect protection. CONCLUSION: This report suggests that administering an extra dose of vaccine is useful for restoring protective antibody levels in vaccinated patients who experience massive postoperative bleeding.

Multidrug Antibiotic Therapy for a Non-Human Immunodeficiency Virus-Infected Patient With Clarithromycin-Resistant Disseminated Mycobacterium avium Complex Disease.

The management of macrolide-resistant Mycobacterium avium complex (MAC) disease is challenging. It is extremely rare for non-human immunodeficiency virus (HIV)-infected patients to develop disseminated MAC disease. A 73-year-old non-HIV-infected woman was diagnosed with MAC lung disease (MAC-LD) for 20 years and subsequently chronic necrotizing pulmonary aspergillosis for three years. To avoid drug interaction between rifampicin and voriconazole, MAC-LD was treated with clarithromycin (CLR) alone. The results of the bone biopsy and bone marrow culture conducted for back pain were compatible with CLR-resistant MAC vertebral osteomyelitis. The clinical management of CLR-resistant disseminated MAC disease consisting of lung and spinal lesions with no established treatment and a poor prognosis is challenging. In this case, the patient was treated with multidrug antibiotic therapy, including CLR, ethambutol, rifampicin, amikacin, and moxifloxacin. The results show the effectiveness of multidrug antibiotic therapy in treating CLR-resistant disseminated MAC disease.

Predicting 72-h mortality in patients with extremely high random plasma glucose levels: A case-controlled cross-sectional study.

ABSTRACT: The risk factors associated with 72-hours mortality in patients with extremely high levels of random plasma glucose (RPG) remain unclear.To explore the risk factors predictive of 72-hours mortality in patients with extremely high RPG under heterogenos pathophysiological conditions.Retrospective, single-center, case-controlled cross-sectional study.University teaching hospital.Adults over age 18 were selected from the medical records of patients at the Saitama Medical Center, Japan, from 2004 to 2013.Extremely high RPG (≥500 mg/dl).Mortality at 72 hours following the RPG test, regardless of hospitalization or in an outpatient setting. Multivariate logistic regression analysis was performed with adjustment for age, sex, body mass index (BMI), and RPG level. The final prediction model was built using the logistic regression model with a higher C-statistic, specificity, and sensitivity.A total of 351 patients with RPG ≥500 mg/dl were identified within the 10-year period. The 72-hours mortality rate was 16/351 (4.6%). The C-statistics of the 72-hours mortality prediction model with serum albumin (ALB) and creatine kinase (CK) was 0.856. The probability of 72-hours mortality was calculated as follows: 1/[1 + exp (-5.142 + 0.901log (CK) -1.087 (ALB) + 0.293 (presence (1) or absence (0) of metastatic solid tumor)]. The sensitivity and specificity of this model was 75.5%.The independent risk factors associated with 72-hours mortality in patients with RPG ≥500 mg/dl are hypoalbuminemia, elevated CK, and presence of a metastatic solid tumour. Further research is needed to understand the mechanisms and possible interventions to prevent mortality associated with extremely high RPG.

Presence of anti-transcriptional intermediary factor-1 gamma antibodies in a dermatomyositis patient with retroperitoneal cancer of unknown primary site.

A 71-year-old woman with dermatomyositis (DM) received glucocorticoid steroid (GCS) and tacrolimus treatment. Relapse of skin symptoms was observed after tapering the GCS dose, and the patient tested positive for anti-transcriptional intermediary factor-1 gamma (TIF1-γ) antibody. Examinations for malignancy were repeatedly performed. However, no obvious findings indicative of a tumour were observed. Two years after, a retroperitoneal tumour was detected and pathologically diagnosed as poorly differentiated adenocarcinoma. The patient developed intestinal and biliary obstruction and eventually died of sepsis. Herein, we report the presence of anti-TIF1-γ antibodies in a DM patient with cancer of unknown primary site.

Spontaneous Pyogenic Spondylitis and Possible Infective Endocarditis Caused by Aggregatibacter actinomycetemcomitans.

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 Haemophilus spp., Aggregatibacter spp, Cardiobacterium spp, Eikenella spp, and Kingella spp. (HACEK) infection is suspected.

Refractory aphthae in the epiglottis and supraglottic area due to Crohn's disease.

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.

Fat ring sign and tumoral pseudocapsule sign in idiopathic mesenteric panniculitis complicated by protein-losing enteropathy.

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.

Giant cell arteritis manifested by chronic dry cough.

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.

Acute splenic infarction presenting as an unusual manifestation of essential thrombocythaemia with normal platelet count.

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 JAK2 V617F mutation was identified. Accordingly, low-dose aspirin was initiated, and no thrombotic episode occurred. Nevertheless, 6 months postdischarge, the platelet count gradually increased to >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.

Unclassified Vasculitis with Episcleritis, Thrombophlebitis, Deep Vein Thrombosis, Pulmonary Vasculitis, and Intracranial Vasculitis: An Autopsy Case Report.

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.

Rapid Progression of Lung Cancer Following Emergency Caesarean Section Led to Postpartum Acute Respiratory Failure.

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.

Aortic Intimal Sarcoma Contributes to Atherosclerotic Renovascular Hypertension: An Autopsy Case Report and Review of the Literature.

An autopsy of a 70-year-old man with multiple bone metastases from a malignancy of unknown origin (MUO) and renovascular hypertension revealed an aortic intimal sarcoma (AIS) in the right renal artery accompanied by atherosclerotic changes. AIS appeared as aggregated mutton fat-like translucent particles arising from the intima of the branching portion of the right renal artery and was composed of undifferentiated, fine spindle cells with thicket-like proliferation. AIS was confirmed by immunohistopathology, showing the loss of the lumen lined by CD31-positive endothelium and the expression of CD31, keratin, and vimentin in the viable part of the tumor. In patients with MUO presenting with both bone metastases and an acute or sub-acute onset of renovascular hypertension, AIS in the renal artery may be responsible.

Candidemia from an upper urinary tract infection complicated by candida endophthalmitis.

A 51-year-old Japanese woman developed candidemia as an outpatient secondary to a Candida albicans upper urinary tract infection complicated by previously undiagnosed type 2 diabetes mellitus with poor glycemic control and ureterolithiasis. The patient did not have any risk factors typically associated with candidemia, such as an indwelling vascular catheter, parenteral nutrition or broad-spectrum antibiotic use. During the clinical course, her condition was complicated by unilateral candida endophthalmitis, which progressed despite the administration of systemic antifungal agents and ultimately required vitreous surgery. The etiology of candidemia in this patient and the reason she developed progressive ocular symptoms after starting antifungal treatment are reviewed.