Image-Assisted Pleural Needle Biopsy or Medical Thoracoscopy: Which Method for Which Patient? - A Randomized Controlled Trial.

BACKGROUND: Image-guided or assisted needle biopsies and the increasing use of medical thoracoscopy (MT) have increased the diagnostic accuracy of pleural diseases significantly. However, no consensus exists regarding which patients with pleural effusion should undergo MT and which patients should undergo image-guided or assisted needle biopsy as the first procedure to ensure greater diagnostic accuracy. RESEARCH QUESTION: Which biopsy method is more appropriate for which patient to provide the highest diagnostic accuracy in the diagnosis of pleural effusion? STUDY DESIGN AND METHODS: This prospective, randomized, parallel study included 228 patients with undiagnosed exudative pleural effusion. Patients were divided into two groups based on CT scan findings. Group 1 included patients with pleural effusion only. Group 2 included patients with pleural thickening or lesion in addition to pleural effusion. Patients in each group were assigned randomly to an image-assisted Abrams needle biopsy (IA-ANPB) or MT arm. The diagnostic sensitivity, reliability, and safety were determined for both groups. RESULTS: The false-negative rate was 30.3% for the IA-ANPB arm and 3.1% for the MT arm in group 1. The same rates were 11.9% for IA-ANPB and 4.7% for MT in group 2. In group 1, the sensitivity for the IA-ANPB arm was 69.7%, and the negative likelihood ratio was 0.30. The same rates for the MT arm were 96.9% and 0.03 (P = .009). In group 2, these values were 88.1% and 0.12 for the IA-ANPB arm and 95.4% and 0.05 for the MT arm (P = .207). The rate of complications between the two biopsy methods was not different (8.5% and 15.8%, respectively; P = .107). INTERPRETATION: MT showed a high diagnostic success in all patients with pleural fluid. However, IA-ANPB showed similar diagnostic success as MT in patients with pleural effusion and associated pleural thickening or lesions. Therefore, in the latter case, IA-ANPB could be preferable to MT. TRIAL REGISTRY: ClinicalTrials.gov; No.: NCT05428891; URL: www. CLINICALTRIALS: gov.

Investigation of MTAP and BAP1 staining loss and P16/CDKN2A deletion in pleural cytology specimens and its role in the diagnosis of mesothelioma.

BACKGROUND: Mesothelioma is a malignant neoplasm with a poor survival rate. We aimed to investigate the importance of BAP1, MTAP (IHC), and p16/CDKN2A homozygous deletion (FISH) in cytologic material obtained from pleural effusion sampling, which is a less invasive procedure in the diagnosis of mesothelioma. METHODS: Our study discussed pleural cytology samples of cases with histopathologically proven mesothelioma diagnoses between 2017 and 2022. As the control group, materials that had pleural effusion sampling for other reasons and reactive mesothelial hyperplasia were included in the study. Cell blocks prepared from these materials were subjected to fluorescent in situ hybridization for p16/CDKN2A homozygous deletion and immunohistochemistry for BAP1 and MTAP. RESULTS: The specificity of the P16/CDKN2A homozygous deletion in diagnosing mesothelioma is 100%. Its sensitivity is 68.75%. The specificity of BAP1 immunohistochemical nuclear expression loss is 95%, while the sensitivity is 60%. Loss of nuclear expression of MTAP alone has the lowest specificity and sensitivity, with a specificity of 86% and a sensitivity of 43%. The highest sensitivity is reached when BAP1 loss and p16/CDKN2A homozygous deletion are evaluated together, increasing to 81%. The specificity is 95%. CONCLUSION: It has been determined that any marker alone cannot be used for a definitive mesothelioma diagnosis in pleural effusion cytological specimens; however, sensitivity increases in some combinations. The combination of BAP1 immunohistochemistry and p16/CDKN2A homozygous deletion detected by FISH, which has a higher specificity and sensitivity, can be routinely used in the diagnosis of mesothelioma under the guidance of clinical and radiologic information.

BAP1, Wilms' tumor 1, and calretinin in predicting survival and response to first-line chemotherapy in patients with pleural mesothelioma.

PURPOSE: There are currently no methods to predict response to chemotherapy in pleural mesothelioma (PM). The aim of this study is to investigate the predictive and prognostic role of BAP1, WT1 and calretinin expression and their combinations in pre-treatment tumor samples by immunohistochemical (IHC) staining. METHODS: The study included consecutive PM patients treated with chemotherapy alone at a University hospital between 2009 and 2020. BAP1 analyses were performed on formalin-fixed, paraffin-embedded tumor tissue samples of the patients, while WT1 and calretinin information were obtained from the histopathological diagnosis records. RESULTS: Of the total 107 patients included, 64% had loss of BAP1 expression, whereas 77% had WT1 and 86% had calretinin expression. Patients with the presence of BAP1 expression, one or both of the other two markers, or loss of expression of all three markers (unfavorable status) were more likely to not respond to chemotherapy than those with the presence of all three markers or loss of BAP1 expression and expression of one or two other markers (favorable status) (p = 0.001). Median survival time of patients with favorable and unfavorable status was 15 ± 1.7 and 8.0 ± 2.4 months, respectively (p = 0.027). After adjustment for histopathology and stage, loss of BAP1 (HR = 0.54, 95%CI 0.35-0.83), WT1 (1.75, 1.06-2.90), calretinin (2.09, 1.14-3.84) expression and favourable panel (0.50, 0.27-0.92) was associated with prognosis. CONCLUSIONS: The IHC biomarkers BAP1, WT1, and calretinin, used in the routine diagnosis of PM and their combinations, are the first biomarkers associated with response to chemotherapy and may be a useful tool to select patients for first-line platinum pemetrexed treatment in PM patients. Validation in a large cohort is ongoing.

Genetic analysis of familial predisposition in the pathogenesis of malignant pleural mesothelioma.

PURPOSE: Mesothelioma is the primary tumor of the mesothelial cell membrane. The most important etiology is asbestos exposure. The development of malignant mesothelioma in very few of the population exposed to asbestos and its frequent occurrence in some families may be significant in terms of genetic predisposition. Again, the presence of relatives with mesothelioma who did not have asbestos contact strengthens this argument. This disease, which has limited treatment options and has a poor prognosis, revealing a genetic predisposition, if any, may prolong survival with early diagnosis and effective treatment. METHODS: Based on the genetic predisposition idea, we diagnosed and followed a total of ten individuals of relatives with mesothelioma. DNA was isolated from peripheral blood and whole genome sequencing analysis was done. Common gene mutations in ten individuals were filtered using bioinformatics. After this filter, from the remaining variants, very rare in the population and damaging mutations are selected. RESULTS: Eight thousand six hundred and twenty-two common variants have been identified in ten individuals with this analysis. In total, 120 variants were found on 37 genes in 15 chromosomes. These genes are PIK3R4, SLC25A5, ITGB6, PLK2, RAD17, HLA-B, HLA-DRB1, HLA-DQB1, GRM, IL20RA, MAP3K7, RIPK2, and MUC16. CONCLUSION: Our finding, PIK3R4 gene, is directly associated with mesothelioma development. Twelve genes, which are associated with cancer, were detected in literature. Additional studies, which scan first-degree relatives of individual, are needed to find the specific gene region.

What has changed in malignant mesothelioma between 1990 and 2019? A time- series analyses in Turkey.

OBJECTIVES: To investigate the changes in epidemiological and survival characteristics of malignant mesothelioma (MM) cases diagnosed in a 30-year period between 1990 and 2019. METHODS: Data were analyzed considering three time periods (1990-1999, 2000-2009, 2010-2019) when treatment practices changed. The Join point Regression Program was used to analyze the change in clinical and epidemiological characteristics of the cases. Kaplan-Meier analysis was used to calculate the overall survival of the patients. Cox regression analysis was used to determine the effect of variables on survival. RESULTS: The study group consisted of 928 MM patients. During the study period, the mean age of the patients and the percentage of epithelioid subtype increased, while the percentage of female and histopathologically unidentified cases decreased. The median survival (95%CI) of patients according to the study periods was 9.0 (7.2-10.9), 9.0 (7.6-10.4) and 12.0 (10.5-13.5) months, respectively. A significant increase in overall survival was observed in the time trend (p = 0.013). There was no significant change in overall survival in patients receiving best supportive care over the 30-year period (p = 0.060), but an improvement of 1.4 (95%CI 0.2 to 2.7) months (p = 0.027) was observed in patient receiving chemotherapy. An improvement in overall survival of 4.8 (1.2 to 8.4) months was also observed in patients receiving multimodality treatment during 2000-2019 (p = 0.014). MM patients who were younger, female, diagnosed after 2000, epithelioid subtype, early stage, and received chemotherapy or multimodal treatment had longer survival. CONCLUSIONS: It was found that histopathological diagnosis and treatment success in MM have improved over the years.

Evaluation of the contribution of fine-needle non-aspiration cytology to diagnosis in cases with pulmonary malignant lesions.

BACKGROUND: Fine-needle non-aspiration cytology (FNNAC) is an easy-to-apply, minimally invasive diagnostic method that contributes to the diagnosis and staging of lung cancer. FNNAC can be performed from peripheral lymph nodes as well as in peripheral lung lesions. This study aimed to evaluate the contribution of FNNAC performed from peripheral lesions or lymph nodes to diagnosis in patients with pulmonary malignant lesions. METHODS: FNNAC was applied from a peripherally located mass in the lung, chest wall lesion, or peripheral lymph node using a needle without an injector or active suction. The collected material was evaluated using the cytoblock method. The FNNAC accuracy was obtained by dividing the true positivity value by a number of needle biopsies performed. The 95% confidence interval of the obtained rate was also calculated. RESULTS: The mean age of 56 patients, two female (3.6%) and 54 male (96.4%), was 63.9 ± 9.1 (38-80) years. FNNAC was performed from the peripheral lymph node in 48 patients, the peripheral pulmonary lesion in four, and the accompanying chest wall lesion in four. While true positivity was present in 42 patients, two patients had true negativity, and 12 had false negativity. In five of the 12 cases reported as false negative, the collected material was evaluated as insufficient, while the malignant diagnoses of the remaining seven cases were confirmed by other diagnostic methods. The diagnostic success of FNNAC was determined as 78.57% (95% CI: 65.56-88.41). FNNAC was more successful in diagnosis when performed from the peripheral lymph node compared to the peripheral pulmonary lesion (p=0.033).

Primary Extraosseous Ewing's Sarcoma of the Lung: Radiologic and Pathologic correlation.

Ewing's sarcoma (ES) is a rare and highly aggressive tumor belonging to a family of neoplasms of neuroectodermal origin, which primarily affects the bones or soft tissues. ES originating from lung parenchyma without chest wall involvement is extremely rare with less than 40 cases reported in the English literature. A 41-year-old man admitted to the thoracic surgery department presenting with intermittent non-productive cough, dyspnea, left-sided chest pain for two months for further evaluation and treatment with a preliminary diagnosis of pulmonary mass. Contrast-enhanced thorax CT and MRI revealed a large heterogeneous soft-tissue mass in the left lower lobe with no distant metastases or occult primary tumor. Following the percutaneous transthoracic biopsy, histopathological and immunohistochemical results were consistent with primary pulmonary ES. Though rare, primary pulmonary ES should be considered in the differential diagnosis of young patients presenting with a large heterogeneous soft tissue mass in the lung. This case report highlights the diagnosis, radiologic and pathologic findings, and management of primary pulmonary ES.

Detection of Lung Cancer on Computed Tomography Using Artificial Intelligence Applications Developed by Deep Learning Methods and the Contribution of Deep Learning to the Classification of Lung Carcinoma.

BACKGROUND: Every year, lung cancer contributes to a high percentage deaths in the world. Early detection of lung cancer is important for its effective treatment, and non-invasive rapid methods are usually used for diagnosis. INTRODUCTION: In this study, we aimed to detect lung cancer using deep learning methods and determine the contribution of deep learning to the classification of lung carcinoma using a convolutional neural network (CNN). METHODS: A total of 301 patients diagnosed with lung carcinoma pathologies in our hospital were included in the study. In the thorax, Computed Tomography (CT) was performed for diagnostic purposes prior to the treatment. After tagging the section images, tumor detection, small and non-small cell lung carcinoma differentiation, adenocarcinoma-squamous cell lung carcinoma differentiation, and adenocarcinoma-squamous cell-small cell lung carcinoma differentiation were sequentially performed using deep CNN methods. RESULTS: In total, 301 lung carcinoma images were used to detect tumors, and the model obtained with the deep CNN system exhibited 0.93 sensitivity, 0.82 precision, and 0.87 F1 score in detecting lung carcinoma. In the differentiation of small cell-non-small cell lung carcinoma, the sensitivity, precision and F1 score of the CNN model at the test stage were 0.92, 0.65, and 0.76, respectively. In the adenocarcinoma-squamous cancer differentiation, the sensitivity, precision, and F1 score were 0.95, 0.80, and 0.86, respectively. The patients were finally grouped as small cell lung carcinoma, adenocarcinoma, and squamous cell lung carcinoma, and the CNN model was used to determine whether it could differentiate these groups. The sensitivity, specificity, and F1 score of this model were 0.90, 0.44, and 0.59, respectively, in this differentiation. CONCLUSION: In this study, we successfully detected tumors and differentiated between adenocarcinoma- squamous cell carcinoma groups with the deep learning method using the CNN model. Due to their non-invasive nature and the success of the deep learning methods, they should be integrated into radiology to diagnose lung carcinoma.

Prospective Study of the Utility of Computed Tomography Triage of Pleural Biopsy Strategies in Patients With Pleural Diseases.

BACKGROUND: This study aimed to prospectively evaluate the efficacy and reliability of a diagnostic workup, triaging pleural biopsy method according to baseline computerized tomography (CT) findings in the diagnosis of pleural diseases. METHODS: Patients with pleural pathology were divided into 3 arms according to findings on CT scan images. Arm A: patients with pleural thickening/lesion in addition to pleural effusion. These patients underwent CT scan-guided Abrams' needle pleural biopsy. Arm B: patients with pleural effusion alone or suspected benign asbestos pleurisy. This group underwent medical thoracoscopy (MT). Arm C: patients with only pleural thickening. This group underwent ultrasonography-guided cutting needle pleural biopsy. MT was planned in patients who did not have a specific diagnosis in the CT scan-guided Abrams' needle pleural biopsy group. When patients with a histopathologic diagnosis of fibrinous pleuritis after MT were assessed in terms of the risk factors for malignant pleural diseases, we offered a further invasive procedure. RESULTS: A total of 164 patients were enrolled in the study. Diagnostic sensitivity after the initial procedure was 90.2% in Arm A, 93.3% in Arm B, 95.2% in Arm C, and 92.4% in the entire workup. The negative predictive value of the entire workup was 90.4% for malignant pleural mesothelioma, 97.1% for metastatic malignant pleural diseases, and 100% for tuberculous pleurisy. Five cases who had a diagnosis of fibrinous pleuritis after MT were detected to have risk factors, 4 of which (80%) indicated malignant disease. Complication rates were low and acceptable. CONCLUSION: Use of CT scans to triage an appropriate pleural biopsy method is associated with high diagnostic success. We recommend that the proposed diagnostic workup in this study may be used as a diagnostic algorithm for pleural diseases that require a histopathologic analysis. Determination of risk factors predicting malignant disease in patients where fibrinous pleuritis is reported after MT would be useful for clinical practice.

Water-Clear Cell Adenoma of the Mediastinal Parathyroid Gland.

Water-clear cell adenoma of the parathyroid gland is a rare neoplasm that consists of cells with abundant clear-pink cytoplasm. There have only been 19 cases reported in the English literature. Here we report a case of water-clear cell adenoma of the mediastinal parathyroid gland. A 70-year-old male patient presented to the hospital with back pain and a mediastinal mass 6 cm in size was detected. After excision and microscopic evaluation, uniform, large clear cells with fine cytoplasmic vacuolization, without nuclear atypia, and arranged in solid and acinar patterns were revealed. The cells formed nests that were separated by fine fibrovascular septae and stained positively with anti-parathyroid hormone. To the best of our knowledge, this has not been previously reported in this location. In the differential diagnosis of clear cell lesions of the mediastinum, water-clear cell parathyroid adenoma should be considered.

Prognostic Significance of CD24 in Clear Cell Renal Cell Carcinoma.

The role of cancer stem cells in the initiation and progression of cancer has become a well-studied area of emerging research, and stem cells with different surface markers have been identified in various types of cancer. CD24 is a membrane protein that acts as the ligand for P-selectin and has been defined as a stem cell marker of colonic cancer. The immunohistochemical expression of CD24 is associated with worse patient outcomes in small cell lung cancer, hepatocellular carcinoma, breast cancer, and colon cancer. In this study, we used immunohistochemistry to determine CD24 expression in clear cell, papillary and chromophobe renal cell carcinoma and investigated its relationship with other clinicopathological parameters and prognosis. A total of 108 cases of clear cell, 12 papillary and 13 choromophobe renal cell carcinoma were examined. Clinicopathological features including age, gender, vascular invasion, tumor necrosis, and T stage were recorded. Clinical stage and overall survival and disease-free survival times were recorded. The immunohistochemical expression of CD24 was classified as low or high based on the percentage and intensity of positive staining. CD24 expression was associated with both tumor grade and recurrence rates. The survival analysis revealed that patients with high CD24 expression exhibited significantly lower overall and disease-free survival. Increased expression of CD24 is related to the prognosis of clear cell renal cell carcinoma. This is the first study identifying a strong association between CD24 expression levels and survival. Thus, CD24 expression may aid in predicting prognosis in clear cell renal cell carcinoma.

Adrenocortical Oncocytic Carcinoma: A Case Report and Review of the Histopathologic Diagnostic Criteria.

Oncocytic tumors are rare in the adrenal gland. The histopathological diagnosis of adrenocortical carcinoma is difficult due to the lack of precise diagnostic criteria for malignancy. A 44-year-old man was admitted to our hospital with left flank pain. Radiologically an adrenal mass was detected. After the excision and histopathologic evaluation of the mass, a diagnosis of adrenocortical oncocytic carcinoma was made. At least one of the features of more than 5 mitoses in 50 high power fields, atypical mitotic figures or venous invasion is required for the diagnosis of malignancy in adrenocortical tumors. It has been suggested that tumors that have more than one of the minor criteria of large size ( > 10 cm or > 200 gr), necrosis, capsular or sinusoidal invasion, should be evaluated as having uncertain malignant potential.

An Unusual Presentation of Gout: Tophi in the Middle Ear.

Gout is a common metabolic disease and is characterized by episodic acute and chronic arthritis caused by the deposition of monosodium urate (MSU) crystals in joints and connective tissues. The most commonly involved joint is the first metatarsophalangeal joint. It may also present with nonspecific complaints caused by gouty tophi in unusual sites of the body such as the spine, bone, and middle ear. Such involvements can be disabling and impair the quality of life. In this report, we describe the case of a patient who had complaints of hearing loss and otorrhea. A 34 year-old male patient presented with complaints of recurrent otorrhea and hearing loss for several months. He had no history of joint pain or constitutional symptoms at the time of admission. An otoscopic examination of the left ear revealed subtotal tympanic membrane perforation, and white middle ear plaques were detected. The patient underwent intact canal mastoidectomy under general anesthesia, and middle ear sclerosis was determined as Wieling Kerr classification type 2. Postoperative histopathologic examination of the specimen revealed amorphous eosinophilic deposits within fibrous stroma and tophi containing negatively birefringent MSU crystals by polarized light. After the histopathologic diagnosis of gout, the patient was assessed for peripheral joint involvement. He had no radiographic abnormalities. The patient was administered a serum urate-lowering therapy, given a low purine diet, and followed with the diagnosis of tophaceous gout. Unusual localizations of gouty tophi have been reported in the literature. Gouty tophi should also be considered in the differential diagnosis of middle ear masses alongside common pathologies.

The role of growth factors on hepatic damage in rats with obstructive jaundice.

In this study, we investigated the affect and the role of growth factors on liver damage. 110 Sprague-Dawley rats were divided into 11 groups: a sham group, a control group, HGF, EGF, IGF, TGF groups of irreversible jaundiced rats and a control group and HGF, EGF, IGF, and TGF groups of reversible jaundiced rats (n = 10). In the irreversible jaundiced groups, the common bile duct was explorated, double ligated, and cut. 150 µg/kg/day HGF, 5 µg/kg/day EGF, 5 µg/kg/day IGF, and 5 µg/kg/day TGF ß-1 were injected intraperitoneally after the seventh post-operative day. In the reversible jaundiced group, the common bile duct was ligated and the ligation was resolved on the seventh post-operative day. For 5 days, growth factors were injected at the same dose. Ductal proliferation scores significantly decreased after growth factor administration in the EGF-A and TGF-A groups. Furthermore, ductal proliferation was decreased in the TGF-B group. As a result of this study, HGF was effective in the irreversible jaundiced groups and ineffective in the reversible jaundice groups. EGF was effective in the reversible jaundiced groups and ineffective in the irreversible jaundiced groups. In both the irreversible jaundiced and reversible jaundiced groups, IGF was ineffective, although TGF ß-1 was effective. We believe that these results arise from the positive effects of effective doses of growth factor on liver damage.

CT Scan-Guided Abrams' Needle Pleural Biopsy versus Ultrasound-Assisted Cutting Needle Pleural Biopsy for Diagnosis in Patients with Pleural Effusion: A Randomized, Controlled Trial.

BACKGROUND: Image-guided pleural biopsies, both using ultrasound (US) or computed tomography (CT), are important in the diagnosis of pleural disease. However, no consensus exists regarding which biopsy needles are appropriate for specific procedures. OBJECTIVES: In this randomized, prospective study, we aimed to compare CT scan-guided pleural biopsy using an Abrams' needle (CT-ANPB) with US-assisted pleural biopsy using a cutting needle (US-CNPB) with respect to both diagnostic yield and safety. METHODS: Between February 2009 and April 2013, 150 patients with exudative pleural effusion who could not be diagnosed by cytological analysis were included in the study. The patients were randomized into either the US-CNPB group or the CT-ANPB group. The two groups were compared in terms of diagnostic sensitivity and complications. RESULTS: Of the 150 patients enrolled in this study, 45 were diagnosed with malignant mesothelioma, 46 were diagnosed with metastatic pleural disease, 18 were diagnosed with pleural tuberculosis, 34 were diagnosed with benign pleural disease, and 7 were lost to follow-up. In the US-CNPB group, the diagnostic sensitivity was 66.7%, compared with 82.4% in the CT-ANPB group; the difference between the two groups was statistically significant (p = 0.029). The sensitivity of CT-ANPB increased to 93.7% for patients with a pleural thickness ≥1 cm. The complication rates were low and acceptable. CONCLUSIONS: The first diagnostic intervention that should be preferred in patients with pleural effusion and associated pleural thickening on a CT scan is CT-ANPB. US-CNPB should be used primarily in cases for which only pleural thickening but no pleural effusion is noted.

MicroRNA and mRNA features of malignant pleural mesothelioma and benign asbestos-related pleural effusion.

INTRODUCTION: We investigated the expression of microRNAs and mRNAs in pleural tissues from patients with either malignant pleural mesothelioma or benign asbestos-related pleural effusion. METHODS: Fresh frozen tissues from a total of 18 malignant pleural mesothelioma and 6 benign asbestos-related pleural effusion patients were studied. Expression profiling of mRNA and microRNA was performed using standard protocols. RESULTS: We discovered significant upregulation of multiple microRNAs in malignant pleural mesothelioma compared to benign asbestos-related pleural effusion. Hsa-miR-484, hsa-miR-320, hsa-let-7a, and hsa-miR-125a-5p were able to discriminate malignant from benign disease. Dynamically regulated mRNAs were also identified. MET was the most highly overexpressed gene in malignant pleural mesothelioma compared to benign asbestos-related pleural effusion. Integrated analyses examining microRNA-mRNA interactions suggested multiple altered targets within the Notch signaling pathway. CONCLUSIONS: Specific microRNAs and mRNAs may have diagnostic utility in differentiating patients with malignant pleural mesothelioma from benign asbestos-related pleural effusion. These studies may be particularly helpful in patients who reside in a region with a high incidence of mesothelioma.

A late and extreme complication of lumboperitoneal shunt.

BACKGROUND CONTEXT: Lumboperitoneal (LP) shunting is an effective treatment modality for commmunicating hydrocephalus, pseudotumor cerebri, postoperative pseudomeningoceles, and cerebrospinal fluid fistulas. However, LP shunts are associated with some complications. Here, we report an extreme complication resulting in the formation of granulation tissue. PURPOSE: To report a late extreme complication of LP shunt. STUDY DESIGN: A case report. METHODS: A late and unusual complication of an LP shunt with unique radiologic findings is presented. RESULTS: Spinal magnetic resonance imaging studies depicted a mass on the right anterior section of the dural sac encircling the catheter throughout its course from the L3-L4 interspinous level to the catheter tip. CONCLUSIONS: The etiopathologic mechanism, the prolonged duration, and the unusual radiologic findings in this case are unique and remarkable.