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Sternoclavicular septic arthritis is a rare form of septic arthritis that can lead to fatal complications, such as abscess formation and mediastinitis, in the absence of prompt diagnosis and appropriate treatment. A man in his 40s presented with pain in the right sternoclavicular joint area, and after a joint injection of steroids was administered, he was diagnosed with septic sternoclavicular arthritis caused by Parvimonas micra and Fusobacterium nucleatum. Gram staining of a specimen obtained from the abscess formation area led to early suspicion of anaerobic infection, and appropriate antibiotics were administered.
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Artritis Infecciosa , Firmicutes , Articulación Esternoclavicular , Masculino , Humanos , Fusobacterium nucleatum , Absceso/microbiología , Corticoesteroides , Artritis Infecciosa/tratamiento farmacológico , Artritis Infecciosa/diagnóstico , Articulación Esternoclavicular/diagnóstico por imagen , Articulación Esternoclavicular/microbiologíaRESUMEN
Central nervous system (CNS) tuberculosis (TB) remains a serious disease with high morbidity and mortality but is often difficult to diagnose owing to less sensitive microbiological techniques. Herein, we present a case where the main complaint was staggering gait; however, the patient was diagnosed with CNS TB associated with pulmonary TB. A woman in her 70s was admitted to our hospital with a two-month history of progressive ataxia. Cerebrospinal fluid examination showed an elevated lymphocyte count; however, cranial imaging studies did not show significant findings. However, we performed positron emission tomography-computed tomography imaging owing to suspicions of paraneoplastic syndrome, which showed substantial 18F-fluorodeoxyglucose accumulation in the lungs. A subsequent bronchoscopy exam led to a pulmonary TB diagnosis for which the patient was treated, and the patient's symptoms fully resolved. Finally, we diagnosed ataxia due to CNS TB with pulmonary TB after excluding other causes of ataxia and because of a lymphocyte-predominant increase of cells in the spinal fluid. Thus, TB infection should be considered in cases of cerebellar ataxia of unknown etiology.ââ.
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OBJECTIVE: This study clarifies the involvement of gender and pre-existing diabetes mellitus (DM) in the clinical characteristics of polymyalgia rheumatica (PMR). METHODS: The clinical records of patients diagnosed with PMR in our department between January 2011 and June 2021, especially in terms of gender and DM were retrospectively analysed. RESULTS: We identified 89 patients with the median age of 75.37 cases were men and 52 cases were women. Pre-existing DM was found in 21 patients (23.6%). Male PMR patients exhibited a higher complication rate of pre-existing DM and C-reactive protein (CRP) levels at diagnosis (p = .04 and p < .01, respectively) than female patients, and men were more common in the patient group with pre-existing DM (p = .04). The CRP levels of male PMR patients without pre-existing DM were higher than female PMR patients without pre-existing DM. CONCLUSION: Male PMR patients might have a varying pathophysiology from female patients in terms of high inflammation levels accompanied by a high prevalence rate of pre-existing DM and need a gender-specific approach.
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Diabetes Mellitus , Arteritis de Células Gigantes , Polimialgia Reumática , Humanos , Masculino , Femenino , Polimialgia Reumática/complicaciones , Polimialgia Reumática/epidemiología , Polimialgia Reumática/diagnóstico , Estudios Retrospectivos , Pueblos del Este de Asia , Arteritis de Células Gigantes/complicaciones , Diabetes Mellitus/epidemiologíaAsunto(s)
Diabetes Mellitus , Inhibidores de la Dipeptidil-Peptidasa IV , Sinovitis , Sedimentación Sanguínea , Inhibidores de la Dipeptidil-Peptidasa IV/uso terapéutico , Dipeptidil-Peptidasas y Tripeptidil-Peptidasas , Edema/complicaciones , Edema/tratamiento farmacológico , Humanos , Hipoglucemiantes , Sinovitis/complicaciones , Sinovitis/tratamiento farmacológicoRESUMEN
The aim of this study was to determine the factors that are associated with prolonged mechanical ventilation in elderly patients.Retrospective cohort studySingle tertiary hospital in JapanWe retrospectively identified 228 patients aged 75 years or older who were admitted to a single tertiary care center in Japan between January 1, 2014 and December 31, 2017 because of endogenous diseases and underwent mechanical ventilation.The primary outcome was extubation difficulty, which was defined as the need for mechanical ventilation for more than 14 days after intubation, reintubation within 72âhours after extubation, tracheotomy or extubation, or death within 14 days after intubation.A multivariate analysis showed that age (odds ratio [OR]â=â0.95; 95% confidence interval [CI]â=â0.66-1.38; Pâ=â.80), gender (ORâ=â0.56; 95%CIâ=â0.27-1.17; Pâ=â.13), body mass index (BMI) (ORâ=â1.05; 95%CIâ=â0.98-1.14; Pâ=â.16), smoking history (ORâ=â0.64; 95%CIâ=â0.29-1.41; Pâ=â.27), Activities of daily living (ADL) (ORâ=â0.95; 95%CIâ=â0.49-1.83; Pâ=â.87), and modified acute physiology and chronic health evaluation (APACHE) II score (ORâ=â1.02; 95%CIâ=â0.95-1.09; Pâ=â.61) were not statistically significantly different. However, there were statistically significant differences in extubation difficulty between patients with diabetes mellitus (ORâ=â2.3; 95%CIâ=â1.01-5.12; Pâ=â.04) and those with cardiovascular disease diagnosis on admission (ORâ=â0.31; 95%CIâ=â0.1-0.97; Pâ=â.04).Diabetes mellitus and cardiovascular disease diagnosis on admission were factors that were associated with prolonged mechanical ventilation in the elderly. The results of this study may help to support shared decision making with patients or surrogate decision makers at the start of intensive care in the elderly.
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APACHE , Evaluación Geriátrica/estadística & datos numéricos , Admisión del Paciente/estadística & datos numéricos , Respiración Artificial/estadística & datos numéricos , Factores de Tiempo , Actividades Cotidianas , Anciano , Anciano de 80 o más Años , Extubación Traqueal/efectos adversos , Índice de Masa Corporal , Enfermedades Cardiovasculares/complicaciones , Enfermedades Cardiovasculares/terapia , Cuidados Críticos , Complicaciones de la Diabetes/complicaciones , Complicaciones de la Diabetes/terapia , Servicio de Urgencia en Hospital , Femenino , Humanos , Japón , Tiempo de Internación , Masculino , Análisis Multivariante , Oportunidad Relativa , Valor Predictivo de las Pruebas , Estudios Retrospectivos , Factores de RiesgoRESUMEN
A man in his 40s with a history of congenitally corrected transposition of the great arteries (CCTGA) and closure of ventricular septal defect was referred to our hospital with purpura and hematuria. Presence of purpura, renal damage, and pathological findings on skin biopsy led to the diagnosis of IgA vasculitis (IgAV). Oral prednisolone (PSL) was initiated. However, Streptococcus pseudoporcinus was isolated from blood cultures, and transthoracic echocardiogram revealed vegetation on the pulmonary valve. From these findings, the diagnosis of infective endocarditis (IE) was made. Although the patient's condition improved after PSL interruption and antibiotic administration, his purpura relapsed. PSL readministration improved symptoms, with no further relapse even after gradual PSL dose reduction. The present case raises awareness of the importance of recognizing the occurrence of IE in IgAV patients, especially in those with congenital heart disease. CCTGA should be acknowledged as a risk factor for IE in the right-sided heart.
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Transposición Congénitamente Corregida de las Grandes Arterias , Endocarditis/complicaciones , Vasculitis/inmunología , Adulto , Humanos , Inmunoglobulina A , Masculino , Arteria PulmonarRESUMEN
WHAT IS KNOWN AND OBJECTIVE: Tumour necrosis factor-α-blocking agents potentially cause vasculitis. However, no study has reported on the association between hypocomplementemic urticarial vasculitis (HUV) and certolizumab pegol (CZP) usage. CASE DESCRIPTION: We present the first case of HUV development during CZP treatment for rheumatoid arthritis. Hypocomplementemic urticarial vasculitis improved after CZP was discontinued and the dose of oral prednisolone was increased. WHAT IS NEW AND CONCLUSION: Clinicians should be aware about the potential development of HUV during CZP treatment, which is presumed to be safe considering its unique structural characteristics that differ from those of other tumour necrosis factor-α-blocking agents.
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Antirreumáticos/efectos adversos , Certolizumab Pegol/efectos adversos , Urticaria/inducido químicamente , Vasculitis/inducido químicamente , Antirreumáticos/administración & dosificación , Artritis Reumatoide/tratamiento farmacológico , Certolizumab Pegol/administración & dosificación , Femenino , Enfermedades por Deficiencia de Complemento Hereditario/inducido químicamente , Enfermedades por Deficiencia de Complemento Hereditario/diagnóstico , Enfermedades por Deficiencia de Complemento Hereditario/tratamiento farmacológico , Humanos , Persona de Mediana Edad , Prednisolona/administración & dosificación , Urticaria/diagnóstico , Urticaria/tratamiento farmacológico , Vasculitis/diagnóstico , Vasculitis/tratamiento farmacológicoRESUMEN
Objectives: To analyze the association among remitting seronegative symmetrical synovitis with pitting edema (RS3PE) syndrome, diabetes mellitus (DM), and antidiabetic drugs.Methods: We retrospectively analyzed the clinical and serologic manifestations of patients who had RS3PE syndrome with and without pre-existing DM. Then we performed a subanalysis in which patients with pre-existing DM were classified into two groups according to whether they were or were not taking a dipeptidyl peptidase 4 inhibitor (DPP4i), an antidiabetic drug that was suggested to have an association with the pathogenesis of RS3PE syndrome.Results: Pre-existing DM was found in 13 (35.1%) of 37 patients with RS3PE syndrome. No significant differences in age, gender, physical manifestations, and laboratory findings were observed between the patients with DM and those without DM. DPP4i had been administered to 6 of 13 patients with RS3PE and pre-existing DM. We observed no significant differences in manifestations of RS3PE syndrome before treatment; however, one relapse occurred in a patient with poorly controlled DM who had been continuing DPP4i therapy.Conclusion: This study revealed no evidence suggesting an association among RS3PE syndrome, DM, and antidiabetic drugs. DPP4i would be safe for use by most of patients with RS3PE syndrome. However, elderly patients and patients with poorly controlled DM might require careful observation.
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Complicaciones de la Diabetes/epidemiología , Diabetes Mellitus/tratamiento farmacológico , Inhibidores de la Dipeptidil-Peptidasa IV/efectos adversos , Edema/epidemiología , Hipoglucemiantes/efectos adversos , Sinovitis/epidemiología , Anciano , Inhibidores de la Dipeptidil-Peptidasa IV/uso terapéutico , Femenino , Humanos , Hipoglucemiantes/uso terapéutico , Masculino , Persona de Mediana EdadRESUMEN
We report the case of an 80-year-old man with generalized granuloma annulare (GGA) who subsequently developed giant cell arteritis (GCA). Steroid treatment was effective for both diseases in this case. Although cases of concomitant GGA and GCA have rarely been reported, previous studies suggest that common histological characteristics underlie the two diseases. It is therefore necessary to recognize that GGA can be complicated by GCA, particularly when typical symptoms, such as headache and visual disturbance, are present.
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Corticoesteroides/uso terapéutico , Arteritis de Células Gigantes/etiología , Arteritis de Células Gigantes/fisiopatología , Granuloma Anular/complicaciones , Granuloma Anular/fisiopatología , Anciano de 80 o más Años , Arteritis de Células Gigantes/diagnóstico , Granuloma Anular/diagnóstico , Humanos , Masculino , Resultado del TratamientoRESUMEN
Aim: To analyze the clinical course and prognosis in patients diagnosed with polymyalgia rheumatica (PMR) complicated by the presence of malignancies.Methods: We retrospectively screened the case files of 216 patients hospitalized in our department between 2011 and December 2016 for the results of a thorough physical examination and data on treatment for rheumatic diseases. We identified 53 patients with PMR according to Bird's criteria and 43 patients with 2012 European League Against Rheumatism (EULAR)/American College of Rheumatology (ACR) classification criteria for PMR, then analyzed the clinical and serologic manifestations of PMR in patients with (n = 6) and without (n = 47 in Bird' criteria, n = 37 in 2012 EULAR/ACR criteria) malignancy.Result: No significant differences in age, gender, smoking, and serum markers were observed between PMR patients with and without malignancy, but there were statistically significant differences in tender joint counts and the presence of swollen joints and peripheral arthritis in both Bird's criteria and 2012 EULAR/ACR criteria.Conclusion: The presence of swollen joints and peripheral arthritis may be useful signs that indicate the coexistence of malignancies in patients with PMR.
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Artritis/diagnóstico , Articulaciones/patología , Neoplasias/diagnóstico , Polimialgia Reumática/diagnóstico , Anciano , Artritis/complicaciones , Femenino , Humanos , Masculino , Persona de Mediana Edad , Neoplasias/complicaciones , Polimialgia Reumática/complicaciones , PronósticoRESUMEN
BACKGROUND: The aim of this study was to identify fibrosis-related serological surrogate outcome measures in patients with immunoglobulin G4-related disease (IgG4-RD). METHODS: This was a clinical observational study of 72 patients with untreated IgG4-RD from four institutions in Japan. The serum concentrations of growth differentiation factor 15 (GDF-15), CCL2, hyaluronic acid (HA), amino-terminal propeptide of type III procollagen (PIIINP), and tissue inhibitor of metalloproteinases 1 (TIMP-1) were measured by enzyme-linked immunosorbent assays. The enhanced liver fibrosis (ELF) score was calculated from the TIMP-1, PIIINP, and HA values. We evaluated associations between the values of these biomarkers and laboratory data, the IgG4-RD responder index (IgG4-RD RI) score, and organ involvements. RESULTS: Compared with the 44 healthy controls, the patients with IgG4-RD showed significantly elevated serum concentrations of GDF-15, MCP-1, HA, PIIINP, and TIMP-1 and ELF scores. The patients' serum concentrations of GDF-15, CCL2, HA, and TIMP-1 (but not PIIINP) were positively correlated with each other. Among them, serum GDF-15 most efficiently distinguished patients with IgG4-RD from healthy controls. Serum GDF-15 was not associated with the IgG4-RD RI score or the number of organ involvements but was independently associated with the presence of retroperitoneal fibrosis and with parotid gland involvement. CONCLUSIONS: We observed increased serological surrogate outcome measures of fibrosis in IgG4-RD. GDF-15 may precisely reflect the fibrotic degree in patients with IgG4-RD.
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Biomarcadores/sangre , Enfermedad Relacionada con Inmunoglobulina G4/sangre , Cirrosis Hepática/sangre , Anciano , Quimiocina CCL2/sangre , Ensayo de Inmunoadsorción Enzimática , Femenino , Factor 15 de Diferenciación de Crecimiento/sangre , Humanos , Ácido Hialurónico/sangre , Enfermedad Relacionada con Inmunoglobulina G4/patología , Cirrosis Hepática/patología , Masculino , Persona de Mediana Edad , Fragmentos de Péptidos/sangre , Procolágeno/sangre , Estudios Retrospectivos , Inhibidor Tisular de Metaloproteinasa-1/sangreRESUMEN
We report a female in her twenties who developed generalized edema. She was diagnosed as systemic lupus erythematous (SLE) and Sjögren's syndrome (SS) based on her physical manifestations and positive findings for antinuclear antibody and anti-SS-A/SS-B-antibody. Although she manifested hypoproteinemia, a possibility of lupus nephritis was denied due to a lack of significant abnormality in kidney function tests and urinalysis. The nature of hypoproteinemia and related symptoms was identified as protein losing gastroenteropathy (PLGE) based on α 1-antitrypsin clearance and histopathology findings. Physicians should be aware that PLGE may develop as an underlying cause of edema in SLE and SS.
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A 64-year-old woman with leg edema was diagnosed with protein-losing gastroenteropathy and Sjögren's syndrome. Central venous nutrition led to infection of her catheter, ascites, and deep vein thrombosis. Following successful treatment of these conditions with antibiotics and anticoagulants, she was treated unsuccessfully with prednisolone and steroid pulse therapy. Mizoribine add-on markedly reduced edema and normalized serum albumin. This is the first report of a steroid-resistant protein-losing gastroenteropathy patient with Sjögren's syndrome successfully treated with mizoribine.
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Glucocorticoides/uso terapéutico , Inmunosupresores/uso terapéutico , Prednisolona/uso terapéutico , Enteropatías Perdedoras de Proteínas/tratamiento farmacológico , Ribonucleósidos/uso terapéutico , Síndrome de Sjögren/tratamiento farmacológico , Femenino , Glucocorticoides/administración & dosificación , Humanos , Inmunosupresores/administración & dosificación , Persona de Mediana Edad , Prednisolona/administración & dosificación , Enteropatías Perdedoras de Proteínas/complicaciones , Ribonucleósidos/administración & dosificación , Síndrome de Sjögren/complicacionesRESUMEN
BACKGROUND: HLA-DRB1 alleles are major determinants of genetic predisposition to rheumatic diseases. We assessed whether DRB1 alleles are associated with susceptibility to particular clinical features of adult onset Still's disease (AOSD) in a Japanese population by determining the DRB1 allele distributions. METHODS: DRB1 genotyping of 96 patients with AOSD and 1,026 healthy controls was performed. Genomic DNA samples from the AOSD patients were also genotyped for MEFV exons 1, 2, 3, and 10 by direct sequencing. RESULTS: In Japanese patients with AOSD, we observed a predisposing association of DRB1*15:01 (p = 8.60 × 10-6, corrected p (Pc) = 0.0002, odds ratio (OR) = 3.04, 95% confidence interval (95% CI) = 1.91-4.84) and DR5 serological group (p = 0.0006, OR = 2.39, 95% CI = 1.49-3.83) and a protective association of DRB1*09:01 (p = 0.0004, Pc = 0.0110, OR = 0.34, 95% CI = 0.18-0.66) with AOSD, and amino acid residues 86 and 98 of the DRß chain were protectively associated with AOSD. MEFV variants were identified in 49 patients with AOSD (56.3%). The predisposing effect of DR5 was confirmed only in patients with AOSD who had MEFV variants and not in those without MEFV variants. Additionally, DR5 in patients with AOSD are associated with macrophage activation syndrome (MAS) and steroid pulse therapy. CONCLUSION: The DRB1*15:01 and DR5 are both associated with AOSD susceptibility in Japanese subjects. A protective association between the DRB1*09:01 allele and AOSD was also observed in these patients. Our data also highlight the effects of DRB1 alleles in susceptibility to AOSD.
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Alelos , Pueblo Asiatico/genética , Predisposición Genética a la Enfermedad/genética , Cadenas HLA-DRB1/genética , Enfermedad de Still del Adulto/diagnóstico , Enfermedad de Still del Adulto/genética , Adulto , Anciano , Femenino , Predisposición Genética a la Enfermedad/epidemiología , Humanos , Masculino , Persona de Mediana Edad , Receptores del Ligando Inductor de Apoptosis Relacionado con TNF/genética , Enfermedad de Still del Adulto/epidemiologíaRESUMEN
BACKGROUND: MicroRNAs (miRNAs) are important regulators of a variety of inflammatory mediators. The present study was undertaken to elucidate the role of miRNAs in the rheumatoid cytokine network. METHODS: We analyzed miRNA expression in rheumatoid synovial fibroblasts (RASFs). miRNA array-based screening was used to identify miRNAs differentially expressed between tumor necrosis factor-α (TNF-α)-activated RASFs and untreated RASFs. Transfection of RASFs with miR-155 was used to analyze the function of miR-155. Real-time polymerase chain reaction (PCR) was used to measure the levels of miR-155 in RASFs. RESULTS: miRNA microarray analysis revealed that miR-155-5p was the most highly induced miRNA in TNF-α-stimulated RASFs. TNF-α-induced miR-155 expression in RASFs was time-dependent and TNFα dose-dependent, whereas, IL-6 stimulation did not affect miR-155 expression in RASFs. Transfection of miR-155 mimics into RASFs resulted in the decrease JAK2/STAT3 phosphorylation in IL-6-treated RASFs. CONCLUSIONS: The current results demonstrate that TNF-α modulated miRNA expressions in RASFs. Our data showed that miR-155, which is highly induced by TNF-α stimulation, inhibits IL-6-mediated JAK2/STAT3 activation in RASFs. These findings suggest that miR-155 contributes to the cross-regulation between TNF-α and IL-6-mediated inflammatory pathways in RA.
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Fibroblastos/efectos de los fármacos , Interleucina-6/farmacología , MicroARNs/genética , Factor de Necrosis Tumoral alfa/farmacología , Artritis Reumatoide/genética , Artritis Reumatoide/patología , Células Cultivadas , Relación Dosis-Respuesta a Droga , Fibroblastos/metabolismo , Perfilación de la Expresión Génica/métodos , Regulación de la Expresión Génica/efectos de los fármacos , Humanos , Janus Quinasa 2/metabolismo , Fosforilación/efectos de los fármacos , Factor de Transcripción STAT3/metabolismo , Transducción de Señal/efectos de los fármacos , Transducción de Señal/genética , Membrana Sinovial/metabolismo , Membrana Sinovial/patologíaRESUMEN
We report a case of reactive arthritis (ReA) triggered by Yersinia enterocolitica enteritis. A 24-year-old Japanese man developed polyarthritis in the lower limbs. Two weeks prior to these symptoms, he noted diarrhea, right lower abdominal pain and a fever. Y. enterocolitica was not isolated from a stool culture; however, he was diagnosed with ReA based on the colonoscopic findings of a high anti-Y. enterocolitica antibody titer and HLA-B27 antigen positivity. Following treatment with methotrexate and steroids, his arthritis improved. This is the first reported Japanese case of ReA in the English literature after a gastrointestinal infection caused by Y. enterocolitica.
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Antirreumáticos/uso terapéutico , Artritis Reactiva/tratamiento farmacológico , Artritis Reactiva/etiología , Metotrexato/uso terapéutico , Esteroides/uso terapéutico , Yersiniosis/complicaciones , Adulto , Pueblo Asiatico , Enfermedades Gastrointestinales/complicaciones , Enfermedades Gastrointestinales/tratamiento farmacológico , Humanos , Masculino , Prohibitinas , Resultado del Tratamiento , Yersinia enterocolitica/efectos de los fármacos , Adulto JovenRESUMEN
BACKGROUND: Pneumococcal pneumonia is the most frequent form of pneumonia. We herein assessed the effectiveness of the 23-valent pneumococcal polysaccharide vaccine (PPSV23) in the prevention of pneumonia overall in rheumatoid arthritis (RA) patients at risk for infections. We hypothesized that PPSV23 vaccination is superior in preventing pneumococcal pneumonia compared with placebo in RA patients. METHODS: A prospective, multicenter, double-blinded, randomized, placebo-controlled (1:1) trial was conducted across departments of rheumatology in Japanese National Hospital Organization hospitals. RA patients (n = 900) who had been treated with biological or immunosuppressive agents were randomly assigned PPSV23 or placebo (sodium chloride). The primary endpoints were the incidences of all-cause pneumonia and pneumococcal pneumonia. The secondary endpoint was death from pneumococcal pneumonia, all-cause pneumonia, or other causes. Cox regression models were used to estimate the risk of pneumonia overall for the placebo group compared with the vaccine group. RESULTS: Seventeen (3.7%) of 464 patients in the vaccine group and 15 (3.4%) of 436 patients in the placebo group developed pneumonia. There was no difference in the rates of pneumonia between the two study groups. The overall rate of pneumonia was 21.8 per 1000 person-years for patients with RA. The presence of interstitial pneumonia (hazard ratio: 3.601, 95% confidence interval: 1.547-8.380) was associated with an increased risk of pneumonia in RA patients. CONCLUSION: PPSV23 does not prevent against pneumonia overall in RA patients at relative risk for infections. Our results also confirm that the presence of interstitial lung disease is associated with pneumonia in Japanese patients with RA. TRIAL REGISTRATION: UMIN-CTR UMIN000009566 . Registered 17 December 2012.
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Artritis Reumatoide/complicaciones , Vacunas Neumococicas/uso terapéutico , Neumonía Neumocócica/prevención & control , Streptococcus pneumoniae/efectos de los fármacos , Anciano , Método Doble Ciego , Femenino , Interacciones Huésped-Patógeno/efectos de los fármacos , Interacciones Huésped-Patógeno/inmunología , Humanos , Estimación de Kaplan-Meier , Masculino , Persona de Mediana Edad , Evaluación de Resultado en la Atención de Salud/métodos , Evaluación de Resultado en la Atención de Salud/estadística & datos numéricos , Vacunas Neumococicas/inmunología , Neumonía Neumocócica/complicaciones , Neumonía Neumocócica/microbiología , Modelos de Riesgos Proporcionales , Estudios Prospectivos , Streptococcus pneumoniae/inmunología , Streptococcus pneumoniae/fisiologíaRESUMEN
A 78-year-old man was admitted to our hospital for multiple lung and liver tumors. Initial clinical diagnosis was hepatocellular carcinoma (HCC) with lung metastases because of a high value of serum protein induced by vitamin K absence or antagonist II (PIVKA-II) (6,705 mAU/mL). However, a review of a prior CT showed the lung tumor had existed 6 months before liver tumors were detected. The tumors progressed rapidly and the patient died 37 days after admission. Autopsy revealed that both lung and liver tumors exhibited the histology of large cell neuroendocrine carcinoma (LCNEC). Immunohistochemistry revealed that the tumor cells expressed not only neuroendocrine markers but also PIVKA-II diffusely. Hepatoid differentiation was not detected. Background liver did not show any chronic liver disease. The final diagnosis was PIVKA-II producing LCNEC of the lung with multiple liver metastases. PIVKA-II producing tumors other than HCC are extremely rare. To our best knowledge, this is the first case report of PIVKA-II producing neuroendocrine tumors of the lung.
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Carcinoma de Células Grandes/secundario , Carcinoma Neuroendocrino/secundario , Neoplasias Hepáticas/secundario , Neoplasias Pulmonares/patología , Precursores de Proteínas/biosíntesis , Protrombina/biosíntesis , Anciano , Biomarcadores , Biomarcadores de Tumor/análisis , Carcinoma de Células Grandes/metabolismo , Carcinoma Neuroendocrino/metabolismo , Resultado Fatal , Humanos , Inmunohistoquímica , Neoplasias Pulmonares/metabolismo , MasculinoRESUMEN
The most common arthritic involvement in familial Mediterranean fever (FMF) is acute self- limiting monoarthritis which typically lasts for 72 h. Hip joint involvement is uncommon in FMF and can result either from a process specific to this disease or from a coexisting inflammatory joint disease. We describe a 37-year-old woman with FMF and right osteoarthritis secondary to congenital hip dislocation. Periodic fever with right coxalgia lasting for 6 months was treated using colchicine. Genetic analysis revealed homozygous mutation in the MEFV gene (L110P-E148Q/L110P-E148Q), confirming the FMF diagnosis. Although the clinical presentation and course of FMF arthritis are diverse, delineating these clinical patterns may help with early recognition and treatment to prevent destructive arthritis in FMF. Clinicians should consider the possibility of FMF development in unusual monoarthritis patients with recurrent febrile attacks.