A rare case of thoracic brucellosis misdiagnosed as malignant tumor and literature review.

Brucellosis remains a major public health problem worldwide. It is commonly found in most developed and developing countries, such as the Mediterranean region, the Middle East, and Latin America. In China, brucellosis is mainly distributed in some of the northern provinces and is relatively rare in Shandong province. Brucellosis has a variety of clinical manifestations, with fever, sweating, fatigue, and migratory joint pain being the most common. Because of the non-specific clinical symptoms, brucellosis is often misdiagnosed as other diseases. Here, we report a rare case of brucellosis of thoracic vertebrae misdiagnosed as thoracic malignant tumor and present a review of related literature.

Improving the diagnostic recognition of thoracic endometriosis: Spotlight on a new histo-morphological indicator.

The diagnosis of thoracic endometriosis (TE) is challenging, hence resulting in under-diagnosis as well as long delays before arriving at a correct definitive diagnosis. Our aim is to review the histopathological findings in TE, summarise the diagnostic features, identify any major histo-morphological indicator(s) hitherto unrecognised as such, suggest diagnostic criteria; all with the aim of improving the diagnostic capacity and reducing observer error even where the clinical suspicion is low. A case-control study in which a search in the pathology archives of a referral hospital over a 10-year period was conducted. Twenty-six cases of TE were identified, reviewed, and compared with a control population of 48 cases taken from common benign thoracic diseases. Nine notable histological features were identified in varying permutations in the test group, namely: endometrioid glands, lymphoid clusters, ceroid macrophages, siderophages, cholesterol crystals, capillary congestion, multinucleated giant cells, smooth muscle bundles and fibrosis. The first 6 features were frequent; each being present in over 13 (13/26; 50%) test cases. The first 8 features showed significant association with TE by the Chi-squared test (P<0.05). In this group, the strength of association is high for the first 4 features (Cramér's V≥0.5). The presence of ceroid macrophages is shown to be a novel key feature, previously unrecognised as such, for the identification of TE. The presence of any three of four features including endometrioid glands, lymphoid clusters, ceroid macrophages and siderophages is a suggested criterion for the definitive diagnosis of TE.

Fetal body MRI and its application to fetal and neonatal treatment: an illustrative review.

This Review depicts the evolving role of MRI in the diagnosis and prognostication of anomalies of the fetal body, here including head and neck, thorax, abdomen and spine. A review of the current literature on the latest developments in antenatal imaging for diagnosis and prognostication of congenital anomalies is coupled with illustrative cases in true radiological planes with viewable three-dimensional video models that show the potential of post-acquisition reconstruction protocols. We discuss the benefits and limitations of fetal MRI, from anomaly detection, to classification and prognostication, and defines the role of imaging in the decision to proceed to fetal intervention, across the breadth of included conditions. We also consider the current capabilities of ultrasound and explore how MRI and ultrasound can complement each other in the future of fetal imaging.

Clinical and molecular practice of European thoracic pathology laboratories during the COVID-19 pandemic. The past and the near future.

BACKGROUND: This study evaluated the consequences in Europe of the COVID-19 outbreak on pathology laboratories orientated toward the diagnosis of thoracic diseases. MATERIALS AND METHODS: A survey was sent to 71 pathology laboratories from 21 European countries. The questionnaire requested information concerning the organization of biosafety, the clinical and molecular pathology, the biobanking, the workload, the associated research into COVID-19, and the organization of education and training during the COVID-19 crisis, from 15 March to 31 May 2020, compared with the same period in 2019. RESULTS: Questionnaires were returned from 53/71 (75%) laboratories from 18 European countries. The biosafety procedures were heterogeneous. The workload in clinical and molecular pathology decreased dramatically by 31% (range, 3%-55%) and 26% (range, 7%-62%), respectively. According to the professional category, between 28% and 41% of the staff members were not present in the laboratories but did teleworking. A total of 70% of the laboratories developed virtual meetings for the training of residents and junior pathologists. During the period of study, none of the staff members with confirmed COVID-19 became infected as a result of handling samples. CONCLUSIONS: The COVID-19 pandemic has had a strong impact on most of the European pathology laboratories included in this study. Urgent implementation of several changes to the organization of most of these laboratories, notably to better harmonize biosafety procedures, was noted at the onset of the pandemic and maintained in the event of a new wave of infection occurring in Europe.

Reducing Unnecessary Chest X-Ray Films After Thoracic Surgery: A Quality Improvement Initiative.

BACKGROUND: Previous work has identified that inpatient post-thoracic surgery chest x-ray films (CXR) are overutilized. METHODS: A three-phase rapid cycle quality improvement initiative was performed to reduce empiric post-thoracic surgery CXR use by 25% over 1 year. We adapted evidence-based guidelines and implemented "plan-do-study-act" (PDSA) cycle methodology. The PDSA cycles included (1) education with literature and preintervention statistics; (2) electronic medical record order-set modification; and (3) audit and feedback with monthly status reports. Each cycle lasted 3 months. Use of CXR was tracked in the post-anesthesia care unit and as a daily rate of non-post-anesthesia care unit CXRs. Cost data were estimated from Centers for Medicare & Medicaid Services fees. RESULTS: During the initiative, 292 thoracic surgery inpatients were monitored. Before intervention, 99% of patients (69 of 70) received a post-anesthesia care unit CXR, and the daily rate of other CXRs was 1.6. Overall, there was a significant reduction in CXR utilization (P < .001). Post-anesthesia care unit CXRs decreased by 42%, lowering to 89% (68 of 76) to 68% (50 of 74) to 57% (41 of 72) in PDSA cycles 1 through 3, respectively. The daily rate of other CXRs decreased by 38%, lowering to 1.4 to 1.3 to 1.0. Patient perioperative characteristics and health care quality measures were not different between cycles. After quality improvement implementation, cost savings were estimated to be at least $73,292 per year. CONCLUSIONS: Implementation of our quality improvement initiative safely and systematically reduced empiric CXR use after inpatient thoracic surgery. Results will be used in future quality improvement initiatives to reduce unnecessary postoperative testing.

Atypical localisation of pleomorphic adenoma issue from heterotopic salivary tissue in the supraclavicular region: diagnosis and management.

Heterotopic salivary tissue (HST) is a normal salivary tissue located outsides the major and minor salivary glands. Multiple sites of localisation of heterotopias have been described, the occurrence of HST in the neck is rare and it may have several clinical manifestation. We report the case of 72-year-old Caucasian man presenting a suspect mass on the left-sided supraclavicular region. He is known for melanoma of the right elbow, surgically treated 11 years ago. The final diagnosis of pleomorphic adenoma of HST was retained. Methods of diagnosis and management are documented. Neoplasms arising from HST are uncommon with approximately 80% of benign tumours. Warthin's tumour is the most frequent. The localisation of these neoplasms in the lower neck is very rare and often causes a diagnostic pitfall. Histological analysis provides certainty of diagnosis, defines management and follow-up.

Refractory thoracic endometriosis.

Thoracic endometriosis syndrome (TES) is a rare entity caused by thoracic implantation of endometrial tissue, manifesting as catamenial pneumothorax, pleural effusion and haemoptysis in young female individuals. Its management and long-term prevention of recurrences, can be challenging. We present the case of a young woman who presented with recurrent pneumothorax, haemopneumothorax and pleural effusion. The diagnosis of TES was confirmed based on cytological findings of pleural fluid. She underwent treatment with mechanical pleurodesis twice but continued to have recurrences. Hormonal treatment failed to produce a satisfactory resolution. She underwent chemical pleurodesis, which successfully induced remission of her TES. A review of the literature suggests that chemical pleurodesis produces better results compared with mechanical pleurodesis and that hormonal treatment with gonadotropin-releasing hormone agonists is effective at preventing recurrences.

Thoracic splenosis: Case report of a symptomatic case.

Thoracic splenosis is the autotransplantation of splenic tissue in the left thoracic cavity as a result of a splenic injury. This rare pathology is usually asymptomatic and may be discovered on incidental imaging, but the diagnosis often requires invasive procedures such as surgery in order to eliminate a neoplasic origin. We report a rare symptomatic case of a 39-year-old man presenting with chest pain and multiple nodules revealed on a computed tomography scan. The patient underwent a surgical exploration and the pathological studies concluded to a thoracic splenosis. Indeed, the previous medical history of the patient revealed a left thoraco-abdominal traumatism during childhood. The aim of this paper is to emphasize that the diagnosis can now be performed using only imaging techniques such as technetium-99 sulfur colloid or labelled heat-denatured red blood cell scintigraphy to avoid unnecessary invasive procedures including thoracotomy.

Video-assisted thoracoscopic surgery is a safe and effective method to treat intrathoracic unicentric Castleman's disease.

BACKGROUND: Castleman's disease (CD) is a rare non-clonal lymphadenopathy. Application of video-assisted thoracoscopic surgery (VATs) in intrathoracic unicentric Castleman's disease (UCD) is rarely reported. This study is aimed to clarify the role of VATs for diagnosis and treatment in intrathoracic UCD. METHODS: The authors reviewed and identified patients who had received a histologic diagnosis of CD through VATs at our hospital from January2010 to June 2018. Clinical and radiologic variables, histopathology, type of approach, complications, and long-term effect were analyzed to evaluate the safety and efficacy of VATs. RESULTS: A total of 10 patients were included in this study, with 8 hyaline vascular type and 2 plasma cell type. The mean maximum diameter of the lesions was 4.66 cm. Nine cases underwent complete surgical excision by VATs, and 1 case was converted to thoracotomy. All patients had no postoperative complications. With a median follow-up of 5 years (range: 1-9 years), no tumor recurrence was found in 9 patients receiving complete tumor resection, and 1 patient with incomplete tumor resection remained symptom free without clinical or radiographic progression. CONCLUSIONS: VATs is an alternative, minimally invasive technique for the diagnosis and treatment in patients with intrathoracic UCD.

Chondromesenchymal hamartomas in a 24-year-old male mimicking a posterior mediastinal tumor and a 5-month-old boy with postoperative disseminated intravascular coagulation: two case reports.

BACKGROUND: Chondromesenchymal hamartoma of the chest wall is a rare, benign disease that usually presents at birth or in early infancy. It typically involves one or more ribs, forming a unilateral or bilateral extrapleural mass. Patients may be asymptomatic or complain of mild respiratory distress depending on tumor size and location. To the best of our knowledge, only two of the approximately 100 cases reported so far are adults. CASE PRESENTATION: We present two cases of chondromesenchymal hamartoma. The first case involved the left fifth rib in a 24-year-old male, in close proximity to the fifth vertebral body in the left posterior mediastinum, mimicking a posterior mediastinal tumor on imaging. The tumor was excised via thoracoscopy and the patient had an uneventful postoperative course. The second case was that of a 5-month-old boy, who had a tumor involving the left fifth and sixth ribs which caused thoracic cage collapse. Following en bloc resection of the tumor and the involved rib segments, the patient was transferred to the intensive care unit for treatment of pulmonary infection and disseminated intravascular coagulation (DIC). He was discharged from the hospital in stable condition 11 days later. On histopathology, the tumor was found to be a chondromesenchymal hamartoma with immature spindle-shaped mesenchymal cells, plate-like hyaline cartilage, areas of woven bone formation, endochondral ossification and calcification, osteoclastic giant cells, and secondary aneurysmal bone cysts. CONCLUSIONS: Although the presently reported cases have morphological characteristics similar to previously reported ones, they had distinct radiological and clinical characteristics. Patient 1 is only the third report of an adult with chondromesenchymal hamartoma. His case was characterized by its radiological appearance mimicking a posterior mediastinal tumor. Patient 2 represents the first documentation of DIC as a postoperative complication following excision of a chondromesenchymal hamartoma. We present these two cases to provide clinicopathological insights regarding this extremely rare tumor that are relevant to both pathologists and clinicians.

Understanding Respiratory Restrictions as a Function of the Scoliotic Spinal Curve in Thoracic Insufficiency Syndrome: A 4D Dynamic MR Imaging Study.

BACKGROUND: Over the past 100 years, many procedures have been developed for correcting restrictive thoracic deformities which cause thoracic insufficiency syndrome. However, none of them have been assessed by a robust metric incorporating thoracic dynamics. In this paper, we investigate the relationship between radiographic spinal curve and lung volumes derived from thoracic dynamic magnetic resonance imaging (dMRI). Our central hypothesis is that different anteroposterior major spinal curve types induce different restrictions on the left and right lungs and their dynamics. METHODS: Retrospectively, we included 25 consecutive patients with thoracic insufficiency syndrome (14 neuromuscular, 7 congenital, 4 other) who underwent vertical expandable prosthetic titanium rib surgery and received preimplantation and postimplantation thoracic dMRI for clinical care. We measured thoracic and lumbar major curves by the Cobb measurement method from anteroposterior radiographs and classified the curves as per Scoliosis Research Society (SRS)-defined curve types. From 4D dMRI images, we derived static volumes and tidal volumes of left and right lung, along with left and right chest wall and left and right diaphragm tidal volumes (excursions), and analyzed their association with curve type and major curve angles. RESULTS: Thoracic and lumbar major curve angles ranged from 0 to 136 and 0 to 116 degrees, respectively. A dramatic postoperative increase in chest wall and diaphragmatic excursion was seen qualitatively. All components of volume increased postoperatively by up to 533%, with a mean of 70%. As the major curve, main thoracic curve (MTC) was associated with higher tidal volumes (effect size range: 0.7 to 1.0) than thoracolumbar curve (TLC) in preoperative and postoperative situation. Neither MTC nor TLC showed any meaningful correlation between volumes and major curve angles preoperatively or postoperatively. Moderate correlations (0.65) were observed for specific conditions like volumes at end-inspiration or end-expiration. CONCLUSIONS: The relationships between component tidal volumes and the spinal curve type are complex and are beyond intuitive reasoning and guessing. TLC has a much greater influence on restricting chest wall and diaphragm tidal volumes than MTC. Major curve angles are not indicative of passive resting volumes or tidal volumes. LEVEL OF EVIDENCE: Level II-diagnostic.

Unmasking catamenial hemoptysis in the era of CFTR modulator therapy.

BACKGROUND: Thoracic endometriosis syndrome (TES) is a rare condition that occurs in women when endometriosis implants into the thoracic cavity. Catamenial hemoptysis, the occurrence of hemoptysis with menstruation, is a recognized clinical manifestation of TES commonly treated with hormonal therapy. CASE SUMMARY: We present the first documented case describing the recrudescence of catamenial hemoptysis in the setting of Lumacaftor/Ivacaftor administration in a 25-year-old woman with cystic fibrosis (CF). DISCUSSION: We review the literature on TES, pharmacologic management, and reported cystic fibrosis transmembrane conductance regulator (CFTR) modulator drug interactions. We propose that our patient's recrudescence of catamenial hemoptysis was secondary to a drug-drug interaction between Lumacaftor/Ivacaftor and oral contraceptive therapy. CONCLUSION: Our case suggests that women with CF who have catamenial hemoptysis and a genetic mutation approved for Tezacaftor/Ivacaftor or Elexacaftor/Tezacaftor/Ivacaftor can be managed effectively with either CFTR modulator and hormonal contraceptive therapy.

Thoracic endometriosis-related non-catamenial pneumothorax with peculiar histological findings.

Thoracic endometriosis-related non-catamenial pneumothorax is a rare entity whose pathogenesis is still less unclear than catamenial pneumothorax one. Hormonal therapy and/or talc pleurodesis are not sufficient for successful management. Surgical videothoracoscopic resection has a central role in the treatment. We displace a case of thoracic endometriosis-related non-catamenial pneumothorax presenting with recurrent right pneumothorax, surgically treated three times and misdiagnosed at first two interventions. At third operation, unusual histological findings on diaphragmatic and pulmonary specimens were disclosed. These results could partially clarify the presentation of some complicated misdiagnosed cases. More has to be investigated about pathogenesis of the disease and influence of the hormonal balance on it.

Extrapelvic Endometriosis: A Systematic Review.

OBJECTIVE: To conduct a systematic review of the literature on patients with extrapelvic deep endometriosis. DATA SOURCES: A thorough search of the PubMed/Medline, Embase, and Cochrane databases was performed. METHODS OF STUDY SELECTION: Studies in the last 20 years that reported on primary extrapelvic endometriosis were included (PROSPERO registration number CRD42019125370). TABULATION, INTEGRATION, AND RESULTS: The initial search identified 5465 articles, and 179 articles, mostly case reports and series, were included. A total of 230 parietal (PE), 43 visceral (VE), 628 thoracic (TE), 6 central nerve system, 12 extrapelvic muscle or nerve, and 1 nasal endometriosis articles were identified. Abdominal endometriosis was divided into PE and VE. PE lesions involved primary lesions of the abdominal wall, groin, and perineum. When present, symptoms included a palpable mass (99%), cyclic pain (71%) and cyclic bleeding (48%). Preoperative clinical suspicion was low, the use of tissue diagnosis was indeterminate (25%), and a few (8%) malignancies were suspected. Surgical treatment for PE included wide local excision (97%), with 5% recurrence and no complications. Patients with VE involving abdominal organs - kidneys, liver, pancreas, and biliary tract - were treated surgically (86%) with both conservative (51%) and radical resection (49%), with 15% recurrence and 2 major complications reported. In patients with TE involving the diaphragm, pleura, and lung, isolated and concomitant lesions occurred and favored the right side (80%). Patients with TE presented with the triad of catamenial pain, pneumothorax, and hemoptysis. Thoracoscopy with resection followed by pleurodesis was the most common procedure performed, with 29% recurrence. Adjuvant medical therapy with gonadotropin-releasing hormone was administered in 15% of cases. Preoperative magnetic resonance imaging was performed in all cases of nonthoracic and nonabdominal endometriosis. Common symptoms were paresthesia and cyclic pain with radiation. Surgical resection was reported in 84%, with improvement of symptoms. CONCLUSION: Extrapelvic endometriosis, traditionally thought to be rare, has been reported in a considerable number of cases. Heightened awareness and clinical suspicion of the disease and a multidisciplinary approach are recommended to achieve a prompt diagnosis and optimize patient outcomes. Currently, there are no comparative studies to provide recommendations regarding optimal diagnostic methods, treatment options, and outcomes for endometriosis involving extrapelvic sites.

Thoracic Endometriosis Syndrome: A Review of Diagnosis and Management.

BACKGROUND: Endometriosis is characterized by the presence of endometrial-like glands and stroma outside the uterine cavity and is believed to affect 6%-10% of reproductive-age women. Endometriosis within the lung parenchyma or on the diaphragm and pleural surfaces produces a range of clinical and radiological manifestations. This includes catamenial pneumothorax, hemothorax, hemoptysis, and pulmonary nodules, resulting in an entity known as thoracic endometriosis syndrome (TES). DATABASE: Computerized searches of MEDLINE and PubMed were conducted using the key words "thoracic endometriosis," "catamenial pneumothorax," "catamenial hemothorax," and "catamenial hemoptysis." References from identified sources were manually searched to allow for a thorough review. CONCLUSION: TES can produce incapacitating symptoms for some patients. Symptoms of TES are nonspecific, so a high degree of clinical suspicion is warranted. Medical management represents the first-line treatment approach. When this fails or is contraindicated, definitive surgical treatment for cases of suspected TES uses a combined video laparoscopy performed by a gynecologic surgeon and video-assisted thoracoscopic surgery performed by a thoracic surgeon. Postoperative hormonal suppression may further reduce disease recurrence.

Intrathoracic Splenosis Without Clinical Evidence of Diaphragmatic Rupture.

Intrathoracic splenosis is a rare diagnosis that is usually made after an invasive procedure. Most cases report concomitant rupture of the spleen and left hemidiaphragm with autotransplantation of splenic tissue into the left hemithorax. We report a case of intrathoracic splenosis with no evidence of diaphragmatic rupture. The mechanism may be explained by hematogenous spread. The patient underwent video-assisted thoracoscopic surgery for diagnosis, which could have been avoided if splenosis was suspected.