Legionella Serositis: A Rare Presentation.

BACKGROUND: Legionella has a higher prevalence in India than in the world. Legionaries' disease most commonly involves the lungs but because of increased awareness, extrapulmonary manifestations are also being diagnosed more frequently. CASE DESCRIPTION: We present a case of a young female with acute onset of fever and chest pain. On initial investigation, an electrocardiogram (ECG) reported widespread pulse rate (PR) depression suggestive of pericarditis which was confirmed by ECG. High-resolution computed tomography (HRCT) thorax suggested mild bilateral pleural effusion with normal lung parenchyma. elevated erythrocyte sedimentation rate (ESR), C-reactive protein (CRP) added to the diagnosis of serositis. Serological study for atypical organisms was remarkable for positive immunoglobulin M (IgM) for Legionella. She was treated with a high dose of steroids and azithromycin successfully. CONCLUSION: Isolated extrapulmonary presentation of legionaries disease is often overlooked and is common. So it should be always included in the diagnostic armamentarium as treatment is highly efficacious if started early.

Serositis and lymphopenia are common features of systemic lupus erythematosus following SARS-CoV-2 infection: A case report and literature review.

The coronavirus disease 2019 (COVID-19) caused by severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2) can affect a number of human systems, including the respiratory, cardiovascular, neurological, gastrointestinal, and musculoskeletal systems. These symptoms persist long after the acute infection has healed and is called "long COVID". Interestingly, there have been a series of reports that SARS-CoV-2 infections trigger the development of various autoimmune diseases such as systemic lupus erythematosus (SLE), inflammatory arthritis, myositis, vasculitis. Here, we report a novel case of SLE characterized by persistent pleural effusion and lymphopenia following SARS-CoV-2 infection. This is the first case in the Western Pacific region to our knowledge. Furthermore, we reviewed 10 similar cases including our case. By looking at the characteristics of each case, we found that serositis and lymphopenia are common features of SLE following SARS-CoV-2 infection. Our finding suggests that patients with prolonged pleural effusion and/or lymphopenia after COVID-19 should be checked for autoantibodies.

Chronic Myelomonocytic Leukemia Presenting With Polyserositis and Seropositivity for Rheumatoid Arthritis.

Chronic myelomonocytic leukemia (CMML) is a rare clonal stem cell disorder associated with clinical and pathologic of myelodysplasia and myeloproliferation. Systemic autoimmune/inflammatory disorders (SAID) and polyserositis have been associated with CMML. These manifestations can be observed concomitantly, shortly before diagnosis or anytime along the course of illness. We report a case of myeloproliferative CMML who presented with polyserositis and positive serology for rheumatoid arthritis. Retrospective studies of myelodysplasia/CMML have reported 15% to 25% incidence of SAID. The most commonly observed disorders include systemic vasculitis, connective tissue diseases, polychondritis, seronegative arthritis, and immune thrombocytopenia. SAID does not confer adverse prognosis in retrospective studies. Polyserositis is less common; this may result from leukemic infiltrate or result from autoimmunity. Treatment of serositis includes steroids and cytoreductive agents. Serositis may confer poor prognosis and hypomethylating therapy may improve the outcome.

A 60-Year-Old Woman With Acute Chest Pain and Bilateral Pleural and Pericardial Effusions.

CASE PRESENTATION: A 60-year-old woman presented with acute-onset, progressively worsening shortness of breath and pleuritic chest pain for 3 days. She also complained of a dry cough, but no fever or chills. There was no history of swelling of the feet; nor was there a history of nausea or diarrhea. She was a lifelong nonsmoker and had no history of recent travel or sick contacts. Her medical history included hypertension and ulcerative colitis. The ulcerative colitis was in remission and she had not been taking medications for this for over 7 years. Her home medications included alendronate, amlodipine, aspirin, atenolol, and vitamin D3 supplements. She had no allergies.

Increased Diagnostic Yield of Tuberculous Serositis by Using Serous Fluid Drainage Flocky Precipitate (SFDFP) as a Testing Sample.

A definitive diagnosis of tuberculosis serositis (TS) is still challenging. Our preliminary practice found that Serous Fluid Drainage Flocky Precipitate (SFDFP) was a useful testing sample to diagnose TS. We designed this study to assess the diagnostic performance of SFDPF for TS compared with conventional bacteriology methods on serous fluid (SF). A cohort study was conducted from July 2014 to April 2016. Patients with suspected TS were consecutively screened. SF and SFDFP were collected and tested by Ziehl-Neelsen stain, MTB culture, and Xpert/RIF assay. We compared the diagnostic performance of SF and SFDFP in several test settings. Through this study, 85 patients were enrolled, of whom 70 (82.4%) were confirmed TS or highly probable TS, 13 (15.3%) were none-TS and 2 (2.4%) indeterminate results were ruled out. The overall sensitivity using both SFDFP and SF was significantly higher than each (60% vs. 48% and 41%, p < 0.05). SFDFP and SF samples had similar diagnostic performance (p < 0.05). No false positive was detected in this study. We concluded that SFDFP is a reliable testing sample for diagnosing tuberculous serositis. SFDFP may significantly improve the diagnostic yield as a supplement to conventional tests.

A rare combination: chylous polyserositis and autoimmune myelofibrosis as a presentation of systemic lupus erythematosus.

Chylous polyserositis and autoimmune myelofibrosis occurring concomitantly inn a case of SLE are a rare phenomenon. We here report a case of a 38-year-old woman who was admitted with a history of cough and shortness of breath for 1½ months along with fever and abdominal distension for 1 month. She also had arthralgias, weight loss and pancytopenia. She was diagnosed as a case of SLE with Chylous polyserositis and autoimmune myelofibrosis. She was started on steroids and immunosuppressive therapy, to which she responded. To summarize, this is the first case report where chylous polyserositis and pancytopenia due to autoimmune myelofibrosis occurred which was responsive to steroids and immunosuppressive therapy.

Multifocal osteolysis with chylous polyserositis and intrathoracic vein thrombosis: a diagnostic challenge for rheumatologists.

Vanishing bone disease with multisystemic involvement may mimic systemic autoimmune or autoinflammatory diseases. We present a 19-year-old man who was hospitalized due to chest pain following a progressive osteolysis of the bony thorax. The disease later expanded into the pleura, peritoneum and pericardium in a form of massive chylous polyserositis. The patient also developed thrombosis of multiple central veins, which in turn worsened the chylothorax by increasing the pressure in the thoracic duct. This is the first case of vanishing bone disease complicated by triple chylous effusions and central vein thrombosis.

Anti-SmD1 antibodies are associated with renal disorder, seizures, and pulmonary arterial hypertension in Chinese patients with active SLE.

Detection of autoantibodies in systemic lupus erythematosus (SLE) plays an important role in timely diagnosis and earlier treatment of SLE. In this study, we used a SmD1 polypeptide-based ELISA to determine anti-SmD1 antibody in 269 SLE, including100 naïve (had not been treated with steroids or immunosuppressants at study inception) SLE patients and 169 non-naive SLE patients; 233 controls with other rheumatic diseases (RDC) (70 RA, 40 AS, 73SSc, and 50 SS), and 110 healthy controls (HC) group. The positive rate of anti-SmD1 among all SLE patients was 60.97%, higher than that in the RDC group (13.30%, P = 0.000) or the HC group (9.09%, P = 0.000). The positive rate of anti-SmD1 in non-naive SLE patients was higher than that for anti-dsDNA antibodies (44.97%, P = 0.03). Positivity for anti-SmD1 only was found in 14.00% of naive SLE patients and 16.00% of non-naive SLE patients. In naive SLE patients, the serum concentration of anti-SmD1 was lower after treatment than before treatment (P = 0.039). Active SLE patients positive for anti-SmD1 were more likely to have malar rash, rash, nonscarring alopecia, PAH and hypocomplementemia. High positivity for anti-SmD1 only in patients with SLE indicated the importance and necessity of detection of anti-SmD1 in patients with SLE.

Life-threatening Medical Complications Due to Ovarian Hyperstimulation Syndrome: A Hidden Etiology.

Ovarian hyperstimulation syndrome is usually an iatrogenic complication in women taking ovulation induction medications during assisted reproduction. We hereby report the case of a 25 years old female who presented with hypertension, polyserositis with tense ascites and large cystic ovaries. She developed sigmoid and transverse sinus thrombosis. She had undergone a clandestine ovulation induction therapy as a commercial ovum donor. She fitted in severe category of ovarian hyperstimulation syndrome.

Chronic GvHD-associated serositis and pericarditis.

Serositis is a rare manifestation of chronic GvHD (cGvHD). No risk factors or laboratory changes associated with this syndrome have been recognized to date, and outcomes have not been described in a large series. We searched our institutional database for patients undergoing allogeneic hematopoietic cell transplant identified as having serositis or pericarditis. Laboratory studies from prior to diagnosis, at diagnosis and post diagnosis of serositis, as well as outcomes from invasive procedures were included. Twenty patients met criteria for cGvHD-associated serositis, and all but three patients had a prior diagnosis of cGvHD. Fifteen were male, and the complication occurred in the setting of immunosuppressant taper in 12 cases. Ten patients required invasive interventions, including pericardial window or stripping. A significant increase in blood monocytes and decrease in serum albumin were identified at diagnosis compared with pre-diagnosis. Out of 20 patients, 17 were treated with steroids, with 12 demonstrating a complete response. These data suggest that cGvHD-associated serositis occurs mainly in the setting of treated as opposed to de novo cGvHD and biomarkers associated with the syndrome include a decrease in albumin and an increase in absolute monocyte count. Outcome data from larger series are required to better understand the optimal management of this rare complication.

Xanthogranulomatous inflammation of the perimetrium with infiltration into the uterine myometrium in a postmenopausal woman: a case report.

BACKGROUND: Xanthogranulomatous inflammation is an uncommon form of chronic inflammation that is destructive to the normal tissue of affected organs. Although xanthogranulomatous endometritis and xanthogranulomatous salpingitis of the female genital tract has been described previously, to the best of our knowledge, this is the first report of xanthogranulomatous inflammation with infiltration into the uterine myometrium from the perimetrium without endometritis. CASE PRESENTATION: A 68-year-old Japanese woman with intermittent lower abdominal pain and low-grade fever who was initially treated with antibiotics underwent hysterectomy due to abscess formation in the posterior wall of the myometrium and perimetrium (the outer serosal layer of the uterus). Histopathological findings revealed that the abscess was caused by xanthogranulomatous inflammation with the granulation tissue and chronic inflammatory cells that consisted of focal and sheets of foam cells. The inflammation destroyed the perimetrial elastic lamina, and the myometrium was deeply infiltrated by the xanthoma cells. Neither endometritis nor salpingitis was coexistent with the xanthogranulomatous inflammation. CONCLUSION: The patient was diagnosed as xanthogranulomatous inflammation, most likely arising from the perimetrium. Our findings suggest that the perimetrium, as well as the endometrium and adnexae, is one of the origins of xanthogranulomatous inflammation in female genital tract.

Utility of T-cell interferon-γ release assays for diagnosing tuberculous serositis: a prospective study in Beijing, China.

BACKGROUND: Diagnosis of tuberculous serositis remains a challenge. The aim of this study was to evaluate the diagnostic efficiency of T-SPOT.TB on serous effusion mononuclear cells (SEMC) for diagnosing tuberculous serositis in a high TB burden area. METHODS: The present prospective study enrolled patients with suspected tuberculous serositis in a tertiary referral hospital in Beijing, China, to investigate the diagnostic sensitivity, specificity, predictive value (PV), and likelihood ratio(LR) of these tests. Clinical assessment, T-SPOT.TB on SEMC, and T-SPOT.TB on PBMC were performed. Test results were compared with the final confirmed diagnosis. RESULTS: Of the 187 participants, 74 (39.6%) were microbiologically or clinically diagnosed as tuberculous serositis and 93(49.7%) were ruled out. The remaining 20 (10.7%) patients were clinically indeterminate and excluded from the final analysis. Compared to that on PBMC, T-SPOT.TB on SEMC showed higher sensitivity (91.9%vs73.0%, P = 0.002), specificity (87.1%vs.73.1%, P = 0.017), PPV (85.0%vs.68.4%, P = 0.013), NPV (93.1%vs.77.3%, P = 0.003), LR+ (7.12vs.2.72) and LR- (0.09vs.0.37), respectively. The frequencies of spot forming cells (SFCs) for T-SPOT.TB on SEMC were 636 per million SEMC (IQR, 143-3443) in patients with tuberculous serositis, which were 4.6-fold (IQR, 1.3-14.3) higher than those of PBMC. By ROC curve analysis, a cut-off value of 56 SFCs per million SEMC for T-SPOT.TB on SEMC showed a sensitivity of 90.5% and specificity of 89.2% for the diagnosis of tuberculous serositis. CONCLUSIONS: T-SPOT.TB on SEMC could be an accurate diagnostic method for tuberculous serositis in TB endemic settings. And 56 SFCs per million SEMC might be the optimal cut-off value to diagnose tuberculous serositis.

Refractory ascites as the first presentation of systemic lupus erythematosus.

Serositis (peritonitis, pleuritis, and pericarditis) can be observed in approximately 13% of patients with systemic lupus erythematosus (SLE). However, peritoneal serositis presenting as painless massive ascites is extremely rare with only few cases been reported. Indeed, ascites in SLE has been reported to occur only when complicated by nephrotic syndrome, congestive cardiac failure, or portal hypertension. We describe herein a very unique case of SLE related serositis presenting with a massive refractory ascites, normal albumin, and absence of a clear autoimmune disorder or protein-losing enteropathy (PLE) at the time of her presentation, which only responded to a pulse course of corticosteroid. SLE was confirmed 2 years later.

Hashimoto's thyroiditis concomitant with sequential autoimmune hepatitis, chorea and polyserositis: a new entity of autoimmune polyendocrine syndrome?

We herein report a case of Hashimoto's thyroiditis (HT) with sequential autoimmune hepatitis (AIH), chorea and polyserositis. The patient was a 24-year-old man who underwent subtotal thyroidectomy due to compression symptoms caused by goiter and was diagnosed with HT postoperatively based on pathological examinations two years previously. He had exhibited liver dysfunction and intermittent chorea since 2008. His liver function and polyserositis improved remarkably following the administration of ursodeoxycholic acid (UDCA) and methylprednisolone. This is a very rare case that can be classified as autoimmune polyglandular syndrome (APS) type 3. Early and adequate UDCA and glucocorticoid treatment may lead to a favorable prognosis.

Tuberculosis mesenteric adenopathy and polyserositis.

A 41-year-old female patient was admitted into Surgery Clinic accusing abdominal pain, diarrhea, fever and chills. Based on clinical, biological and imaging data, it was established a diagnosis of pelviperitonitis and it was initiated an antibiotic and anti-inflammatory treatment. As fever and abdominal pain continued, it was decided to go on with surgery that revealed suppurated and perforated mesenteric adenopathy. Pus was sampled for bacteriological exam and also biopsy was performed for pathological exam. The result of pathological exam was suggestive for a specific granulomatous lesion (TB lesion). It was established diagnosis of TB mesenteric adenopathy and it was initiated specific anti-TB treatment according to WHO guidelines. After three, respectively five months of treatment, patient developed a right laterocervical adenopathy that fistulized in both cases, despite the correct treatment administered. No resistant TB strain and no atypical mycobacteria was discovered.