Xanthogranulomatous pleuritis induced by recurrent biliothorax due to a biliopleural fistula: The first case report in the literature.

Xanthogranulomatous pleuritis is an extremely rare pathological entity, characterized by the infiltration of foamy cells and multinucleated giant cells within the pleural space. This condition often mimics infectious and neoplastic processes, presenting significant diagnostic challenges. This report details the first documented case of xanthogranulomatous pleuritis induced by recurrent biliothorax due to a biliopleural fistula, presenting a unique clinical scenario. We describe the clinical presentation, diagnostic hurdles, and both the surgical and medical management of this case. The discovery of biliothorax, evidenced by pleural fluid bilirubin levels that exceed serum bilirubin levels, underscores the importance of considering biliothorax in the differential diagnosis of recurrent pleural effusions, particularly in patients with a history of trauma. This case emphasizes the need for heightened awareness and a multidisciplinary approach in the diagnosis and treatment to effectively manage this complex condition and prevent recurrence.

Lipid-Laden Macrophage Index as a Diagnostic Tool for Pediatric Aspiration: A Systematic Review.

Objective: Lipid-laden macrophage index (LLMI) has been proposed as a marker for aspiration on bronchoalveolar lavage. It has also been studied as a marker for gastroesophageal reflux and other pulmonary diseases. This review aims to determine the clinical correlation between LLMI and pediatric aspiration. Data Sources: PubMed (MeSH search), Scopus, and Cochrane Central Register of Controlled Trials (CENTRAL) portals through December 17th, 2020. Review Methods: Preferred Reporting Items for Systematic Review and Meta-Analysis criteria were followed, and a quality assessment of included studies was performed using the Methodological Index for Non-Randomized Studies. Search criteria included all occurrences in the title or abstract of the terms "pulmonary aspiration" and "alveolar macrophages." Results: Five studies describing 720 patients met inclusion, 3 retrospective case-control studies, and 2 prospective observational studies. Four studies suggested a link between elevated LLMI and aspiration, and 1 found no association. Control groups varied and included healthy nonaspirators to nonaspirators with other pulmonary diseases. Diagnosis of aspiration was not standardized across the studies. Three papers proposed cutoff values for LLMI, all different. Conclusion: The existing literature indicates that LLMI is not a sensitive or specific marker for aspiration. Further study is needed to define the utility of LLMI in pediatric aspiration.

Dysregulation of miR-103a Mediates Cigarette Smoking-induced Lipid-laden Macrophage Formation.

Cigarette smoke (CS) is considered a major risk factor for chronic obstructive pulmonary disease (COPD) that is currently the third leading cause of death in the United States. Studies have indicated that patients with COPD have elevated blood low-density lipoprotein levels, which may contribute to the dysregulation of lipid metabolism. Accumulating data show that microRNAs (miRNAs) are involved in various human diseases. However, the role of microRNAs in the pathogenesis of COPD remains poorly defined. In this study, we found that miR-103a expression was significantly reduced in alveolar macrophages from smokers and patients with COPD versus that in alveolar macrophages from nonsmokers. Our data indicated that reactive oxygen species negatively regulate miR-103a in macrophages. Functionally, miR-103a modulates the expressions of genes involved in lipid metabolism and directly targets low-density lipoprotein receptors in macrophages. Furthermore, overexpression of miR-103a suppressed the accumulation of lipid droplets and reduced the reactive oxygen species, both in vitro and in vivo. Taken together, our findings indicate that downregulation of miR-103a contributes to cigarette smoke-induced lipid-laden macrophage formation and plays a critical role in lipid homeostasis in lung macrophages in the pathogenesis of COPD.

Acute exogenous lipoid pneumonia: Unusual presentation as cavitating lung disease with pneumothorax.

Acute lipoid pneumonia is quite uncommon and is associated with oily or lipid contents within the alveoli. Exogenous lipoid pneumonia due to kerosene poisoning, manifests with a wide clinical spectrum ranging from subtle chemical pneumonitis to marked severe pulmonary and systemic inflammation. We present an interesting case of an adult male with kerosene poisoning. He manifested with severe cavitating lung disease. In addition, he developed spontaneous pneumothorax. Both cavitating lung disease and pneumothorax are unusual manifestations of acute exogenous lipoid pneumonia and perhaps follow severe lung injury following high volume kerosene exposure.

Xanthogranulomatus inflammatory lesion mimicker of malignancy: A clinicopathological study from rural India.

OBJECTIVE: Xanthogranulomatus inflammation is an uncommon variant of chronic inflammation and a well-established pathological entity involving various organs and systems. It may be associated with infection and obstruction, defective lipid transport, immunological disturbances, and often confused as a malignant neoplasm. The confirmative diagnosis is made on histopathology. METHODS: This is a retrospective study conducted from January 2008 to April 2020 on histopathologically diagnosed xanthogranulomatus lesions. All the relevant available data regarding age, sex, and organ involvement were collected from histopathology lab records. The macroscopic and microscopic evaluation of cases was also done. This study was aimed to determine the significance of histopathology in the diagnosis of xanthogranulomatus lesions, revealing pathological changes, and clinicopathological correlation. RESULTS: In the current study, there were 93 cases of xanthogranulomatus inflammatory lesion. Gall bladder was frequently involved 70 (75.27%), followed by 5 (5.37%) kidney, gastrointestinal Tract 6 (3 [3.23%] cases in colon and 3 [3.23%] in appendix, respectively), and others. The maximum number of cases was in the age group of 31-40 years with 24 (25.80%) cases. The female to male sex ratio was 2.3:1. CONCLUSION: Awareness and knowledge of xanthogranulomatus inflammatory lesion is significant to the pathologist and surgeon to prevent extensive surgery. This lesion often mimics as malignancy and confirmatory diagnosis is made on histopathology. Thus, every excised specimen must be examined histopathologically to diagnose and rule out differential.

Shine like gold and sparkle like glitter: Three cases of lipoid pneumonia.

Lipoid pneumonia (LP) is an unwonted, mostly asymptomatic entity which has no classical radiological appearance. It can be endogenous or exogenous depending upon the type of exposure or underlying milieu. It simulates a number of infective and malignant respiratory conditions and can go undiagnosed or delayed leading to morbidity and mortality. We put forward three cases that initially presented as classical pneumonia, but on further assessment and investigations were diagnosed to be LP. All the three cases manifested with symptoms of fever, productive cough and breathlessness. Chest Xray and CT scan were indicative of consolidation. Bronchoalveolar lavage (BAL) evinced lipid laden macrophages that stained positive with fat stains (Sudan IV and Oil Red O). Two cases were endogenous and one was exogenous type. LP, owing to its nonspecific clinical presentation and radiographic signs, needs a high index of suspicion, and a detailed clinical history for accurate diagnosis. Corroboration of lipid laden alveolar macrophages in BAL is the crux to the diagnosis. Hence, clinicians should be cognizant of this condition and rule out LP in cases of non-resolving pneumonia in an appropriate clinical context.

Flexible Bronchoscopy in Evaluation of Persistent Wheezing in Children-Experiences from National Pediatric Center.

Background and objectives: Persistent wheezing (PW) is defined as prolonged or recurrent episodes of wheezing despite regular treatment. Flexible bronchoscopy (FB) is recommended to determine the etiology of PW in children. This study aimed to determine the etiology of PW based on FB findings in a national pediatric center. Materials and Methods: Children presenting with PW that underwent flexible bronchoscopy from April 2016 to August 2019 at the Mother and Child Health Institute of Serbia were included in this observational study. After endoscopic evaluation, bronchoalveolar lavage fluid (BALF) samples were taken and further analyzed. Quantitative microbiology, cytological analysis and oil-red staining of specimens were performed to determine cellular constituents and presence of lipid laden macrophages (LLM). Upper gastrointestinal series were performed to exclude gastroesophageal reflux disease, swallowing dysfunction and vascular ring. Results: Pathological findings were revealed in 151 of 172 study participants, with bacterial lower airway infection (BLAI) (48.3%) and primary bronchomalacia (20.4%) as the most common. Younger participants were hospitalized for significantly longer periods (ρ = -0.366, p < 0.001). Study participants with BLAI and associated mucus plugging were notably younger (p < 0.001). Presence of LLM in BALF was not associated with findings of upper gastrointestinal series. All patients with confirmed BLAI were treated with oral antibiotics. Although FB is considered to be invasive, there were no complications associated with the procedure. Conclusions: Flexible bronchoscopy has an exceptional diagnostic value in evaluation of PW. In younger patients with BLAI, presence of mucus plugs may complicate the clinical course, so significant benefits can be achieved with therapeutic lavage during bronchoscopy.

Testing for lipid-laden macrophages in bronchoalveolar lavage fluid to diagnose vaping-associated pulmonary injury. Are we there yet?

Vaping-associated pulmonary injury (VAPI) is a severe respiratory disorder associated with the inhalation of nicotine, tetrahydrocannabinol, cannabidiol, or other active substances through a personal vaporizer ("vaping"). Within 2 months after its description, the disease has reached epidemic proportion, affecting over 2000 people and resulting in a number of fatalities. The substance(s) responsible for the lung injury are still unknown, and the pathophysiology of the disease is still incompletely understood. The manifestations of the disease are protean, and the insidiously starting respiratory, gastrointestinal, and constitutional symptoms may initially resemble a viral flu-like illness. The disease may increase in severity, requiring hospitalization, and in more severe cases, mechanical ventilation. The diagnosis of VAPI currently relies on the identification of pulmonary infiltrates on imaging studies in patients who have used vaping products, after excluding infections and other plausible alternative diagnoses. Because VAPI is currently a diagnosis of exclusion, some authors have suggested the use of lipid-laden macrophages (LLM) as a diagnostic test to confirm the disease. We review the current state of the knowledge about the pathologic basis of VAPI, and the literature on the analytic performance of the LLM test to better understand the potential utility of this test in the diagnosis of VAPI. Our review finds little evidence to suggest the use of LLM in the diagnosis of VAPI, since its underlying pathology is acute lung injury, which is unrelated to LLM, and the frequency of their detection varies greatly in different reported series.

Airway inflammation and injury in children with prevalent weakly acidic gastroesophageal refluxes.

BACKGROUND: In children with gastroesophageal reflux (GER) both acid refluxes (AR) and weakly acidic refluxes (WAR) can induce respiratory symptoms (RS). METHODS: To characterize the airway inflammation in children with more prevalent WAR or AR (defined according a ROC curve analysis), we performed a 3 year-retrospective review of the medical records of patients who underwent fiberoptic bronchoscopy for difficult-to-treat chronic/recurrent respiratory symptoms and who had a positive multiple intraluminal esophageal impedance (pH/MII) monitoring. RESULTS: In the 13 WAR and 11 AR children, the number of cells recovered by bronchoalveolar lavage (BAL) was similar [0.78 (0.29-1.28) x 106 cells, and 1.05 (0.68-1.64) x 106 cells, respectively] (P = 0.22). A neutrophilic alveolitis and an elevated lipid-laden-macrophage (LLM) index were detected in both groups: no differences were found in neutrophils and lymphocyte percentages or in LLM index between WAR and AR children. In contrast, higher BAL epithelial cell proportions were seen in WAR [10.4 (4.85-23.45) %], as compared to AR [2.5 (1.25-7.25) %] children (P = 0.0045), suggesting greater airway damage in the formers. In the whole patient population a significant correlation was found between the proportions of BAL epithelial cells and the number of WAR events (r = 0.43; P = 0.037). Finally, elevated BAL concentrations of substance P and of pepsin were observed, not statistically different in the WAR and AR groups. CONCLUSIONS: In this patient population, WAR events can be associated with a significant airway inflammation and injury that, because of the biochemical mechanisms involved, are likely not completely preventable and/or counteracted by anti-acid treatments.

Adult exogenous lipoid pneumonia: A rare and underrecognized entity in cytology - A case series.

BACKGROUND: Exogenous lipoid pneumonia (ELP) is a rare benign entity without specific clinical or imaging presentation. Although cytological studies - either bronchoalveolar lavage (BAL) or fine-needle aspiration (FNA) - may be pursued in patients with ELP, a definitive diagnosis is frequently rendered only on histology. The aim of this study is to highlight the cytological features of ELP. METHODS: A search of cytopathology (CP) and surgical pathology (SP) diagnoses of ELP was conducted. The corresponding clinical and imaging features were obtained, and the morphology, particularly the presence and size of the intracytoplasmic vacuoles and background, was assessed. RESULTS: Nine cases of ELP were identified, including eight with corresponding CP and SP. A neoplasm was suspected in three based on imaging, but ELP was not in the differential clinically or radiographically in any. Among the cases, six patients had BALs and three FNAs. All of the samples showed multiple large vacuoles within macrophages with at least some equal to or larger than the size of the cell nucleus. Similar vacuoles were noted extracellularly on smears. CONCLUSIONS: ELP is typically described in case reports in the clinical or radiological literature. To the best of our knowledge, this represents the largest series of adult ELP in CP. When large vacuoles are present in macrophages in cytology specimens, at least a suspicion of ELP can be suggested to initiate appropriate therapy, identify/remove the inciting agent, and preclude a more invasive procedure.

Lung injury associated with electronic cigarettes inhalation diagnosed by transbronchial lung biopsy.

A 46-year-old healthy man developed respiratory distress, night sweats, fever, and weight loss after using electronic cigarettes (e-cigs) for approximately 1 month. He presented to the hospital when the symptoms worsened 2 months after onset. The findings of bronchoalveolar lavage (BAL) fluid examination and the following transbronchial lung biopsy examination led to the diagnosis of acute alveolitis: intra-alveolar fibrosis accompanied with exudate containing abundant lipid-laden macrophages, eosinophils, and neutrophils. Eventually, e-cig-induced acute lung injury was diagnosed. The symptoms were rapidly alleviated upon e-cig use termination and methylprednisolone pulse therapy, and no subsequent recurrence was observed. There have been only a few reported cases of e-cig-induced lung injury. In e-cig users presenting with atypical pneumonia, close examination by BAL and biopsy should be performed to verify the presence or absence of lipid-laden macrophages.

Case Of Xanthogranulomatous Oophoritis.

Xanthogranulomatous inflammation is characterized by destruction of the tissues of the organ involved and replacement by chronic inflammatory cells such as lymphocytes, plasma cells, occasional neutrophils with or without multinucleated or Touton giant cells. Exact aetiology is not known but the theory of infection with organisms like Proteus, E coli, and Bacteroides fragilis is most popular. Xanthogranulomatous inflammation of the female genital tract is not common and usually involves the endometrium; however, xanthogranulomatous inflammation of the ovaries is a rare entity.

Lipid-laden bronchoalveolar macrophages in asthma and chronic cough.

BACKGROUND: The presence of lipids in alveolar macrophages (AMs) may impair their phagocytic response, and determine airway inflammation and obstruction. OBJECTIVE: To determine the factors such as severity of asthma, chronic cough, airway inflammation and obesity that may influence the presence of lipids in lung macrophages. METHODS: Bronchoalveolar lavage fluid (BALF) was obtained from 38 asthmatics (21 severe and 17 mild/moderate), 16 subjects with chronic cough and 11 healthy control subjects. The presence of lipids in macrophages was detected using an Oil-red-O stain and an index of lipid-laden macrophages (LLMI) was obtained. RESULTS: LLMI scores were higher in healthy subjects (median 48 [IQR 10-61]) and the severe asthma group (37 [11.5-61]) compared to mild/moderate asthmatics (7 [0.5-37]; p < 0.05 each). Subjects reporting a history of gastro-oesophageal reflux disease (GORD) had higher LLMI values (41.5 [11.3-138] versus 13 [0-39.3], p = 0.02). There was no significant correlation between LLMI and chronic cough, BAL cell differential counts, FEV1, FEV1/FVC or body mass index (BMI). CONCLUSIONS: The reduced LLMI in mild/moderate asthma may be related to lower incidence of GORD. However, this was not related to the degree of airflow obstruction, obesity or airway inflammation.

Lipoid Pneumonia Following Aspiration of Ghee (animal fat) in an Omani Infant.

We are reporting, what we believe, is the first case of biopsy proven exogenous lipoid pneumonia in an Omani infant. Exogenous lipoid pneumonia is caused by aspiration of oily products .One reason is the traditional practice of giving infants oily products (Ghee) as in our patient.