Case Reports in Pulmonology The latest articles from Hindawi Publishing Corporation © 2014 , Hindawi Publishing Corporation . All rights reserved. Endobronchial Tuberculosis: Two Case Reports and Review of the Literature Mon, 18 Aug 2014 12:37:55 +0000 Endobronchial tuberculosis commonly affects young patients and presents as acute or insidious onset cough, wheeze, low grade fever, and constitutional symptoms. Although endobronchial lesions usually result in sputum positivity for acid fast bacilli (AFB), a false negative sputum or absence of radiological lesions may result in delayed diagnosis. On the other hand, sputum positivity with presence of signs on chest radiology may lead to consideration of parenchymal TB as the primary diagnosis and the coexistence of endobronchial lesions may be missed until sequelae of the latter ensue. Besides, in elderly patients, consideration of other differentials like malignancy and pneumonia may lead to misdiagnosis. Hence, bronchoscopy is essential for confirmation of endobronchial TB. We hereby report two cases of endobronchial TB which stress the importance of bronchoscopic diagnosis for timely institution of treatment and prevention of permanent sequelae, respectively. Anshum Aneja, Uma Maheswari Krishnaswamy, Vijayashree Thyagaraj, Riyaz P. Moideen, and Mantha Satya Padmaja Copyright © 2014 Anshum Aneja et al. All rights reserved. An Osteolytic Metastasis of Humerus from an Asymptomatic Squamous Cell Carcinoma of Lung: A Rare Clinical Entity Tue, 12 Aug 2014 09:24:05 +0000 Advanced lung cancer is complicated by skeletal metastases either due to direct extension from adjacent primaries or, more commonly, due to haematogenous dissemination of neoplastic cells. Lumber spine is the most common site for bony metastases in bronchogenic carcinoma. Proximal lone bones, especially humerus, are unusual sites for metastases from lung primaries. Small cell and large cell varieties of lung cancer are most commonly associated with skeletal dissemination. It is also unusual that an asymptomatic squamous cell carcinoma of lung presents with painful, soft tissue swelling with osteolytic metastasis of humerus which is reported in our case. Systemic cytotoxic chemotherapy, local palliative radiotherapy, adequate analgesia, and internal fixation of the affected long bone are different modalities of treatment in this advanced stage of disease. But the prognosis is definitely poor in this stage IV disease. Anirban Das, Sudipta Pandit, Sibes k. Das, Sumitra Basuthakur, and Somnath Das Copyright © 2014 Anirban Das et al. All rights reserved. Tracheobronchial Foreign Body Aspiration: Dental Prosthesis Mon, 04 Aug 2014 10:39:29 +0000 It is important to extract foreign bodies for avoiding life-threatening complications. They can lead to death if they are not treated. Different signs and symptoms could occur according to the complete or partial airway obstruction. Foreign body aspiration is a rare incident in adults. The organic foreign materials such as foods are found to be aspirated more commonly and are usually settled in the right bronchial system. However, dental prosthesis and teeth aspirations are rare in literature. In our study, a 52-year-old male patient who had aspirated the front part of his lower dental prosthesis accidentally is presented and the foreign body is extracted by using rigid bronchoscopy. There are many causes of aspiration but dental prosthetic aspirations should be kept in mind during sleep. For this reason, dental apparatus must be taken out while asleep. Ataman Köse, Dilek Kostak, Erol Aramagan, Aslıhan Durak, Nur Sezin Seçkin, Serdar Süha Dönmez, and Hüseyin Melek Copyright © 2014 Ataman Köse et al. All rights reserved. Radiographically Severe but Clinically Mild Reexpansion Pulmonary Edema following Decompression of a Spontaneous Pneumothorax Sun, 03 Aug 2014 11:42:21 +0000 The case is a 48-year-old female who presented with mild dyspnea on exertion and cough with unremarkable vital signs and was found to have a large right sided pneumothorax. She underwent small bore chest tube decompression with immediate reexpansion of the collapsed lung. However, she rapidly developed moderate hypoxemia and radiographic evidence of reexpansion pulmonary edema (REPE) on both the treated and contralateral sides. Within a week, she had a normal chest X-ray and was asymptomatic. This case describes a rare complication of spontaneous pneumothorax and highlights the lack of correlation between symptoms, sequelae, and radiographic severity of pneumothorax and reexpansion pulmonary edema. Proposed pathophysiologic mechanisms include increased production of reactive oxygen species with subsequent loss of surfactant and increased vascular permeability, and loss of vasoregulatory tone. William E. Harner and Eric A. Crawley Copyright © 2014 William E. Harner and Eric A. Crawley. All rights reserved. Primary Pulmonary Synovial Sarcoma: A Very Rare Presentation Sun, 03 Aug 2014 08:51:25 +0000 Synovial sarcoma (SS) is a rare tumor originating from mesenchymal tissue and accounting for approximately 5–10% of all soft tissue sarcomas. A rare case of primary pulmonary SS in an asymptomatic 18-year-old man admitted to our hospital for investigation of a 6 × 6.5 cm, oval-shaped, well-delineated pleural based peripheral mass in the left lower lobe in his thorax CT is presented. Left lower lobectomy was done. Immunohistochemically, tumor cells were positive for cytokeratin, epithelial membrane antigen (EMA), and vimentin so that the histopathological diagnosis was compatible with biphasic spindle cell type SS in the lung. Ekrem Cengiz Seyhan, Sinem Nedime Sokucu, Gulsah Gunluoglu, Nurdan Simsek Veske, and Sedat Altin Copyright © 2014 Ekrem Cengiz Seyhan et al. All rights reserved. High-Flow Nasal Cannula Therapy in a Patient with Reperfusion Pulmonary Edema following Percutaneous Transluminal Pulmonary Angioplasty Tue, 15 Jul 2014 00:00:00 +0000 A 62-year-old woman with Wolff-Parkinson-White syndrome was with recent worsening of dyspnea to New York Heart Association functional status Class III. The patient was diagnosed as having central type chronic thromboembolic pulmonary hypertension. By cardiac catheterization, her mean pulmonary artery pressure was 53 mmHg with total pulmonary resistance 2238 dynesseccm−5. After medical therapies with tadalafil, furosemide, ambrisentan, beraprost, and warfarin were initiated, percutaneous transluminal pulmonary angioplasty (PTPA) was performed. Following PTPA, life-threating hypoxemia resulting from postoperative reperfusion pulmonary edema developed. High-flow nasal cannula therapy (HFNC) was applied, and 100% oxygen at 50 L/min of flow was required to keep oxygenation. HFNC was continued for 3 days, and the patient was discharged on 8th postoperative day with SpO2 of 97% on 3 L/min of oxygen inhalation. Because of the simplicity of the technique, the lower cost of equipment, and remarkable patient tolerance to the treatment, we speculate that HFNC can take over the post of noninvasive ventilation as first-line therapy for patients with acute respiratory failure. Kiyoshi Moriyama, Toru Satoh, Akira Motoyasu, Tomoki Kohyama, Mariko Kotani, Riichiro Kanai, Tadao Ando, and Tomoko Yorozu Copyright © 2014 Kiyoshi Moriyama et al. All rights reserved. Sarcoidosis Incidentally Diagnosed: A Case Report Tue, 08 Jul 2014 00:00:00 +0000 Sarcoidosis is a chronic, granulomatous condition with unknown cause. Because most of the patients are free of clinical symptoms, sarcoidosis should be considered in differential diagnosis if noncaseous granuloma is noted in biopsies, performed for other reasons. With no clinical symptoms, our patient was diagnosed with sarcoidosis upon identifying noncaseous granuloma in the lymph node biopsy material collected during the laparoscopic operation, performed for gallbladder polyp. Besir Kesici, Ahmet Burak Toros, Levent Bayraktar, and Adem Dervisoglu Copyright © 2014 Besir Kesici et al. All rights reserved. Endobronchial Lipomatous Polyp: A Rare Benign Tumor of the Lung Sun, 25 May 2014 08:26:57 +0000 Endobronchial lipomatous polyp is a rare nonmalignant tumor of the lung. It comprises 5% of the benign lung tumor, with the majority of benign tumors being hamartoma. Lipomatous polyp often leads to endobronchial lesion, associated with postobstructive pneumonia, hemoptysis, and atelectasis. We hereby present a case and discussion of an elderly man with endobronchial lipomatous polyp, presenting as recurrent pneumonia. Salim Surani, Karen Allen, Saherish Surani, and Joseph Varon Copyright © 2014 Salim Surani et al. All rights reserved. Intestinal Type of Lung Adenocarcinoma in Younger Adults Sun, 23 Mar 2014 11:21:03 +0000 Intestinal type of lung adenocarcinoma (ILADC) was initially described by Tsao and Fraser in 1991. Morphology and immunophenotype of ILADC are the same as in colorectal adenocarcinoma. Rectocolonoscopy must be performed to exclude colorectal origin of adenocarcinoma. Colorectal adenocarcinoma claimed to be genetically similar to an ILADC. Patients. We describe 24- and 26-year-old patients of both genders who went under surgery because of a lung tumor mass detected on CT scan. ILADC was diagnosed on resected lung specimens. According to positivity of Cytokeratin20, CDX-2, and Villin, respectively, and negativity of Cytokeratin7, TTF-1, Napsin-A, SurfactantB, MUC-1, and MUC-2, respectively, ILADC was diagnosed. KRAS mutation was detected in tumor tissue of the male patient. Conclusion. Rectocolonoscopy is the only relevant method for distinguishing the intestinal type of lung adenocarcinoma from metastatic colorectal carcinoma because immunohistochemistry and detection of mutation status are frequently the same in both types of adenocarcinoma. More investigations are needed for further understanding of ILADC in purpose of personalized lung carcinoma therapy particularly introducing detection of mutation status, especially in younger patients. Jelena Stojsic, Milica Kontic, Dragan Subotic, Marko Popovic, Dragana Tomasevic, and Jelena Lukic Copyright © 2014 Jelena Stojsic et al. All rights reserved. Improvement with Infliximab of a Disseminated Sarcoidosis in a Patient with Crohn’s Disease Thu, 06 Feb 2014 13:18:09 +0000 Sarcoidosis and Crohn’s disease are systemic granulomatous disorders affecting the lung and the intestine, respectively, with variable involvement of other organs and are seldom associated. While anti-TNFα is a recognized treatment of Crohn’s disease, its usage is discussed in sarcoidosis. A 42-year-old man presented with an 11-year-long history of Crohn’s disease; upon discovery of an abnormal chest CT scan the diagnosis of multivisceral sarcoidosis was made and, later, a treatment with an anti-TNFα agent, infliximab, was started, because of worsening Crohn’s disease recurrences. CT scan demonstrated net regression of pulmonary opacities and hepatosplenic lesions. Pathologies obtained from the intestinal tract and the bronchi of the patient were, respectively, characteristic of Crohn’s disease and sarcoidosis leading to the diagnosis of both diseases. We report a rare case of steroid resistant Crohn’s disease associated with multivisceral sarcoidosis, treated successfully by an anti-TNFα agent, infliximab. Nader Chebib, Fabrice Piégay, Julie Traclet, François Mion, and Jean-François Mornex Copyright © 2014 Nader Chebib et al. All rights reserved. Organizing Pneumonia Preceding Rheumatoid Arthritis Tue, 28 Jan 2014 07:26:11 +0000 Rheumatoid arthritis patients are susceptible to interstitial lung disease, and joint manifestations of rheumatoid arthritis usually precede lung involvements by several years. Organizing pneumonia, as the first manifestation of rheumatoid arthritis, is extremely rare, and its clinical features remain currently unknown. We present a case and a literature review of patients who were pathologically diagnosed with organizing pneumonia first and met the diagnostic criteria of rheumatoid arthritis later. In this review, we observed the following: (1) patients with organizing pneumonia preceding rheumatoid arthritis have a high prevalence of rheumatoid factor or anticyclic citrullinated peptide antibodies; (2) almost all patients developed rheumatoid arthritis within one year after the diagnosis of organizing pneumonia. We suggest that patients with organizing pneumonia and positive for either rheumatoid factor or anticyclic citrullinated peptide antibody should be cautiously followed up regarding the development of rheumatoid arthritis, particularly during the first year after the diagnosis of organizing pneumonia. Yoshiaki Kinoshita, Atsuhiko Sakamoto, and Kouko Hidaka Copyright © 2014 Yoshiaki Kinoshita et al. All rights reserved. Vanishing Lung Syndrome in a Patient with HIV Infection and Heavy Marijuana Use Wed, 08 Jan 2014 07:02:16 +0000 Vanishing lung syndrome (VLS) is a rare and distinct clinical syndrome that usually affects young men. VLS leads to severe progressive dyspnea and is characterized by extensive, asymmetric, peripheral, and predominantly upper lobe giant lung bullae. Case reports have suggested an additive role of marijuana use in the development of this disease in young male tobacco smokers. We herein report a case of a 65-year-old Hispanic male previously diagnosed with severe emphysema and acquired immune deficiency syndrome (AIDS), with a history of intravenous heroin use and active marijuana smoking who presents to the emergency department with severe progressive shortness of breath he was found to have multiple large subpleural bullae occupying more than one-third of the hemithorax on chest computerized tomography (CT), characteristic of vanishing lung syndrome. The patient was mechanically ventilated and later developed a pneumothorax requiring chest tube placement and referral for surgical bullectomy. Surgical bullectomy has shown high success rates in alleviating the debilitating symptoms and preventing the life threatening complications of this rare syndrome. This case further emphasizes the importance of recognizing VLS in patients with severe emphysema and heavy marijuana smoking. Basheer Tashtoush, Fernando Gonzalez-Ibarra, Roya Memarpour, Anas Hadeh, and Laurence Smolley Copyright © 2014 Basheer Tashtoush et al. All rights reserved. Pulmonary Embolism with Vertebral Augmentation Procedures Mon, 09 Dec 2013 15:12:40 +0000 With the prevalence of an aging American population on the rise, osteoporotic vertebral fractures are becoming a common occurrence, resulting in an increase in vertebral augmentation procedures and associated complications such as cement leakage, vertebral compressions, and pulmonary embolism. We describe a patient who presented with respiratory distress three years following kyphoplasty of the lumbar vertebra. Computed tomography (CT) angiogram of the chest confirmed the presence of polymethylmethacrylate (PMMA) cement in the lung fields and pulmonary vessels. We conducted a systematic review of the published literature identifying effective management strategies for the treatment of vertebroplasty-associated pulmonary embolism. Swetha Bopparaju, Joseph Varon, and Salim Surani Copyright © 2013 Swetha Bopparaju et al. All rights reserved. Diffuse Alveolar Hemorrhage due to Acute Mitral Valve Regurgitation Sun, 08 Dec 2013 14:37:05 +0000 Diffuse alveolar hemorrhage (DAH) can be caused by several etiologies including vasculitis, drug exposure, anticoagulants, infections, mitral valve stenosis, and regurgitation. Chronic mitral valve regurgitation (MR) has been well documented as an etiological factor for DAH, but there have been only a few cases which have reported acute mitral valve regurgitation as an etiology of DAH. Acute mitral valve regurgitation can be a life-threatening condition and often requires urgent intervention. In rare cases, acute mitral regurgitation may result in a regurgitant jet which is directed towards the right upper pulmonary vein and may specifically cause right-sided pulmonary edema and right-sided DAH. Surgical repair of the mitral valve results in rapid resolution of DAH. Acute MR should be considered as a possible etiology in patients presenting with unilateral pulmonary edema, hemoptysis, and DAH. Creticus P. Marak, Parijat S. Joy, Pragya Gupta, Yana Bukovskaya, and Achuta K. Guddati Copyright © 2013 Creticus P. Marak et al. All rights reserved. Transition from Hepatopulmonary Syndrome to Portopulmonary Hypertension: A Case Series of 3 Patients Sun, 10 Nov 2013 15:54:48 +0000 Hepatopulmonary syndrome (HPS) and portopulmonary hypertension (PPHTN) are the two major pulmonary vascular complications of liver disease. While HPS is characterized by low pulmonary vascular resistance, PPHTN is defined by the presence of elevated pulmonary vascular resistance. Given these seemingly opposing pathophysiologic mechanisms, these conditions were traditionally felt to be mutually exclusive. In this series, we present three patients with severe hepatopulmonary syndrome who had spontaneous resolution of their HPS with the subsequent development of PPHTN. To our knowledge, this is the largest case series presented of this phenomenon in nontransplanted patients. One proposed mechanism for the occurrence of this phenomenon involves dysregulation of the same vascular signaling pathway, which may lead to both pulmonary vascular dilatations and pulmonary arterial remodeling in the same patient. Another theory involves the possible differential binding of endothelin-1, a vasoactive signaling peptide that induces vasoconstriction when bound to receptor A and vasodilation when bound to receptor B. Although the mechanisms for this phenomenon remain unclear, it is important to be vigilant of this phenomenon as it may change the patient's overall treatment plan, especially in regard to appropriateness and timing of liver transplant. Radhika Zopey, Irawan Susanto, Igor Barjaktarevic, and Tisha Wang Copyright © 2013 Radhika Zopey et al. All rights reserved. Lymphoepithelioma-Like Carcinoma of the Lung: An Unusual Case and Literature Review Wed, 30 Oct 2013 14:09:55 +0000 We described a case of lymphoepithelioma-like carcinoma (LELC) of the lung of a 65-year-old man with initial symptoms of intermittent chest pain and mild shortness of breath for 2 weeks. A right-lung mass was noted on chest computed tomography (CT) scan and was proved histopathologically as LELC of lung after video-assisted thorascopic lobectomy. He was successfully treated with lobectomy with postoperative adjuvant chemotherapy and is alive without signs of recurrence for 36 months after the diagnosis. It is important for clinicians, pathologists, and radiologists to understand the clinical, pathological, and radiological presentations of this neoplasm to avoid improper clinical decision making and misdiagnosis. Yuan-Chun Huang, Ching Hsueh, Shang-Yun Ho, and Chiung-Ying Liao Copyright © 2013 Yuan-Chun Huang et al. All rights reserved. Malignant Mesothelioma Presenting as a Gradually Enlarging Pneumothorax Wed, 02 Oct 2013 16:24:08 +0000 Malignant mesothelioma is an extremely aggressive tumor arising from the pleura with median survival of approximately 9–12 months. It can rarely present as a spontaneous pneumothorax. Less than 35 cases of malignant mesothelioma presenting as spontaneous pneumothorax have been reported in the literature. Pathology may show florid mesothelial hyperplasia. We herein report a case of mesothelioma presenting as a pneumothorax that gradually enlarged over a one-year period and also review the relevant literature. Ashish Prasad, Diana Olsen, and P. S. Sriram Copyright © 2013 Ashish Prasad et al. All rights reserved. Mycobacterium interjectum Lung Infection Mon, 30 Sep 2013 08:13:46 +0000 A 62-year-old male presented with productive cough, weight loss, and night sweats. CXR revealed a right upper lobe cavitary lesion. Evaluation was negative for Mycobacterium tuberculosis, and sputum revealed Mycobacterium avium intracellulare (MAI). Since his clinical course was atypical for MAI, further investigations were pursued which identified Mycobacterium interjectum in lung specimens, a very rarely described etiology of pulmonary disease. Appropriate therapy with rifampin, intravenous amikacin, trimethoprim/sulfamethoxazole (TMP/SMX), and ethambutol resulted in clinical and radiographic improvement. This is the third case described over a period of 20 years of destructive lung disease in an immunocompetent adult due to M. interjectum. M. C. Mirant-Borde, S. Alvarez, and M. M. Johnson Copyright © 2013 M. C. Mirant-Borde et al. All rights reserved. One Lung Wheezing due to Endobronchial Solitary Papilloma Thu, 19 Sep 2013 08:20:52 +0000 A 55-year-old man was referred to our hospital due to intolerant severe short of breathing and persistent cough for two hours. He had similar attacks in the past two years which were treated with bronchodilators. Computed tomography revealed endobronchial mass, which was endoscopically resected by laser. Histology revealed benign squamous papilloma. In this paper we will deal with the various nonspecific clinical presentations, etiopathology, predisposing factors, and diagnosing tests of these benign tumors, especially the important role of computed tomography. Suheil Artul and Faozi Artoul Copyright © 2013 Suheil Artul and Faozi Artoul. All rights reserved. Multifocal Extranodal Involvement of Diffuse Large B-Cell Lymphoma Sun, 15 Sep 2013 13:34:42 +0000 Endobronchial involvement of extrapulmonary malignant tumors is uncommon and mostly associated with breast, kidney, colon, and rectum carcinomas. A 68-year-old male with a prior diagnosis of colon non-Hodgkin lymphoma (NHL) was admitted to the hospital with a complaint of cough, sputum, and dyspnea. The chest radiograph showed right hilar enlargement and opacity at the right middle zone suggestive of a mass lesion. Computed tomography of thorax revealed a right-sided mass lesion extending to thoracic wall with the destruction of the third and the fourth ribs and a right hilar mass lesion. Fiberoptic bronchoscopy was performed in order to evaluate endobronchial involvement and showed stenosis with mucosal tumor infiltration in right upper lobe bronchus. The pathological examination of bronchoscopic biopsy specimen reported diffuse large B-cell lymphoma and the patient was accepted as the endobronchial recurrence of sigmoid colon NHL. The patient is still under treatment of R-ICE (rituximab-ifosfamide-carboplatin-etoposide) chemotherapy and partial regression of pulmonary lesions was noted after 3 courses of treatment. Devrim Cabuk, Y. Taha Gullu, Ilknur Basyigit, Ozgur Acikgoz, Kazım Uygun, Kursat Yildiz, and Fusun Yildiz Copyright © 2013 Devrim Cabuk et al. All rights reserved. Snake Envenomation Causing Distant Tracheal Myonecrosis Thu, 05 Sep 2013 16:22:12 +0000 Snakebites are often believed to be poisonous. However, this is not always the case. In fact, each bite differs from snake to snake, depending on if the snake is poisonous and if there is envenomation. Venom in pit viper snakebites is often associated with local necrosis. The abundant literature selections and research articles justify local myonecrosis due to envenomation, but there is not much in the literature regarding myonecrosis at a site distant from the snakebite. We hereby present a case of a 42-year-old man who was transferred to our emergency department after a rattlesnake bit him twice. The patient, besides developing local myonecrosis at the site of the snakebite, developed necrosis of the scrotum as well as tracheal pressure myonecrosis at the site of the endotracheal tube balloon. In this review, we will attempt to discuss the myonecrosis pathophysiology and management related to the rattle snakebite. Amina Khimani, Afton Mcnierney, Sara Surani, and Salim Surani Copyright © 2013 Amina Khimani et al. All rights reserved. Sleeptalking! Sleepwalking! Side Effects of Montelukast Thu, 05 Sep 2013 09:22:51 +0000 A 16-year-old Caucasian female presented to the pulmonary clinic for a followup on her asthma. Due to the worsening of allergy-related symptoms, therapy with montelukast 10 mg daily was started and resulted in good relief of the patient’s symptoms. In the nights following initiating therapy with montelukast, the patient’s mother reported daily parasomnias in the form of sleeptalking and sleepwalking. Montelukast was discontinued, and that resulted in absence of the parasomnias. In a second attempt montelukast was reinstituted to control the patient’s symptoms. Parasomnias were immediately reported after resuming therapy. Montelukast was then discontinued indefinitely. Our patient has never had any history of parasomnias, and since the discontinuation of montelukast, parasomnias were never reported again. Parasomnias in the form of sleeptalking or sleepwalking were not previously reported as adverse effects of montelukast. Alternative modalities to treat allergy-related symptoms in patients, who develop parasomnias while receiving montelukast, should be explored. Samer Alkhuja, Natalya Gazizov, and Mary Ellen Alexander Copyright © 2013 Samer Alkhuja et al. All rights reserved. A Case of Pulmonary Tumor Thrombotic Microangiopathy Diagnosed by Transbronchial Lung Biopsy and Treated with Chemotherapy and Long-Term Oxygen and Anticoagulation Therapies Thu, 29 Aug 2013 10:22:21 +0000 A 41-year-old woman, who underwent breast resection for cancer of the right breast and adjuvant chemotherapy 2 years ago, was admitted to our hospital due to shortness of breath upon exertion. High-resolution computed tomography of the chest showed small nodular opacities in the peribronchiolar area in both lungs, as well as mediastinal and hilar lymphadenopathy. A transbronchial lung biopsy revealed breast cancer metastasis and pulmonary tumor thrombotic microangiopathy (PTTM). Treatment of PTTM is rarely reported due to the difficulty of antemortem diagnosis; however, the patient was effectively treated with chemotherapy and oxygen and anticoagulation therapies for 3 months. Atsushi Kitamura, Naoki Nishimura, Torahiko Jinta, Rika Suda, Yasuhiko Yamano, Genta Ishikawa, Yutaka Tomishima, Tsuyoshi Hamaoka, Koyu Suzuki, and Naohiko Chohnabayashi Copyright © 2013 Atsushi Kitamura et al. All rights reserved. Undiagnosed Chronic Granulomatous Disease, Burkholderia cepacia complex Pneumonia, and Acquired Hemophagocytic Lymphohistiocytosis: A Deadly Association Sat, 24 Aug 2013 09:11:55 +0000 Background. Chronic granulomatous disease is a rare inherited disorder of the phagocyte nicotinamide adenine dinucleotide phosphate (NADPH) oxidase. The clinical course of the disease is marked by recurrent infections, including Burkholderia cepacia complex infection. Case Report. Here we report the case of a 21-year-old male hospitalized for a Burkholderia cepacia complex pneumonia. Despite the broad spectrum antibiotic treatment, fever continued and patient’s condition worsened. Anemia and thrombocytopenia developed together with hypofibrinogenemia. The patient died of multiple organ dysfunction 17 days after his admission. Autopsy revealed hemophagocytosis, suggesting the diagnosis of acquired hemophagocytic lymphohistiocytosis. DNA analysis showed a deletion in the p47phox gene, confirming the diagnosis of autosomal recessive chronic granulomatous disease. Discussion. In addition to chronic granulomatous disease, recent findings have demonstrated that Burkholderia cepacia complex can decrease activity of the NADPH oxidase. Interestingly, hemophagocytic lymphohistiocytosis is characterized by an impaired function of the T-cell mediated inflammation which is partly regulated by the NADPH oxidase. Physicians should therefore pay particular attention to this deadly association. Maxime Maignan, Colin Verdant, Guillaume F. Bouvet, Michael Van Spall, and Yves Berthiaume Copyright © 2013 Maxime Maignan et al. All rights reserved. Compression of the Superior Vena Cava by an Interatrial Septal Lipoma: A Case Report Thu, 01 Aug 2013 10:07:24 +0000 Primary cardiac tumours are rare; their prevalence ranges from 0.0017% to 0.28% in various autopsy series. Cardiac lipomas are well-encapsulated benign tumours typically composed of mature fat cells, and their reported size ranges from 1 to 15 cm. They are usually seen in the left ventricle and the right atrium. Lipomas are true neoplasms, as opposed to lipomatous hypertrophy of the interatrial septum, which is a nonencapsulated hyperplastic accumulation of mature and foetal adipose tissue. Cardiac lipomas occur in patients of all ages, and the frequency of occurrence has been found to be equal in both sexes. Patients are usually asymptomatic, although the manifestation of symptoms depends upon both size and location of the tumour. We present the case of a patient with an interatrial septal lipoma, causing obstruction of the superior vena cava. R. Grech, A. Mizzi, and S. Grech Copyright © 2013 R. Grech et al. All rights reserved. Empiric Treatment of Focal Organizing Pneumonia in a Patient with a Low-Risk Lung Mass Tue, 30 Jul 2013 13:17:55 +0000 The authors present a case of a 57-year-old man presenting with a solitary lung mass. Transbronchial biopsy showed an organizing pneumonia pattern. A therapeutic trial of corticosteroids resulted in complete resolution avoiding surgery. The authors discuss the diagnosis of focal organizing pneumonia without surgical resection. Mir Alikhan and Srihari Veeraraghavan Copyright © 2013 Mir Alikhan and Srihari Veeraraghavan. All rights reserved. Delayed Presentation of Traumatic Diaphragmatic Rupture with Herniation of the Left Kidney and Bowel Loops Mon, 15 Jul 2013 14:25:39 +0000 Rupture of the diaphragm mostly occurs following major trauma. We report a case of delayed presentation of traumatic diaphragmatic hernia on the left side in a 44-year-old male who presented two weeks after a minor blunt trauma. Left kidney and intestinals coils were found to herniate through the diaphragmatic tear. This case demonstrates the importance of considering the diagnosis in all cases of blunt trauma of the trunk. It also illustrates the rare possibility of herniation of kidney through the diaphragmatic tear. Amiya Kumar Dwari, Abhijit Mandal, Sibes Kumar Das, and Sudhansu Sarkar Copyright © 2013 Amiya Kumar Dwari et al. All rights reserved. A Behcet’s Disease Patient with Right Ventricular Thrombus, Pulmonary Artery Aneurysms, and Deep Vein Thrombosis Complicating Recurrent Pulmonary Thromboembolism Tue, 18 Jun 2013 19:02:08 +0000 Intracardiac thrombus, pulmonary artery aneurysms, deep vein thrombosis, and pulmonary thromboembolism are rarely seen symptoms of Behcet’s disease. A 20-year-old female patient was admitted for complaints of cough, fever, palpitations, and chest pain. On the dynamic thorax computed tomograms (CT) obtained because of significantly enlarged hilar structures seen on chest radiograms, aneurysmal dilatation of the pulmonary artery segments bilaterally, chronic thrombus with collapse, and consolidation substances compatible with pulmonary embolism involving both lower lobes have been observed. It is learned that, four years ago, the patient had been diagnosed with Behcet’s disease and received colchicine treatment but not regularly. The patient was hospitalized. On the transthoracic echocardiogram, a thrombosis with a dimension of 4.2 × 1.6 cm was recognized in the right ventricle. On abdomen CT, aneurysmal iliac veins and deep vein thrombus on Doppler ultrasonograms were diagnosed. At the controls after three months of immunosuppressive and anticoagulant therapies, some clinical and radiological improvements were recognized. The patient suspended the treatment for a month and the thrombus recurred. We present our case in order to show the effectiveness of immunosuppressive and anticoagulant therapies and rarely seen pulmonary thromboembolism in recurrent Behcet’s disease. Selvi Aşker, Müntecep Aşker, Özgür Gürsu, Rıdvan Mercan, and Özgür Bülent Timuçin Copyright © 2013 Selvi Aşker et al. All rights reserved. Primary Pleural Angiosarcoma in a 63-Year-Old Gentleman Thu, 13 Jun 2013 11:03:13 +0000 Primary pleural angiosarcomas are extremely rare. As of 2010, only around 50 case reports have been documented in the literature. Herein, we report the case of a 63-year-old gentleman who presented with a 3-month history of right-sided chest pain, dyspnea, and hemoptysis. Chest X-ray showed bilateral pleural effusion with partial bibasilar atelectasis. Ultrasound-guided thoracocentesis showed bloody and exudative pleural fluid. Cytologic examination was negative for malignant cells. An abdominal contrast-enhanced computed tomography (CT) scan showed two right diaphragmatic pleural masses. Whole-body positron emission tomography/computed tomography (PET/CT) scan showed two hypermetabolic fluorodeoxyglucose- (FDG-) avid lesions involving the right diaphragmatic pleura. CT-guided needle-core biopsy was performed and histopathological examination showed neoplastic cells growing mainly in sheets with focal areas suggestive of vascular spaces lined by cytologically malignant epithelioid cells. Immunohistochemical analysis showed strong positivity for vimentin, CD31, CD68, and Fli-1 markers. The overall pathological and immunohistochemical features supported the diagnosis of epithelioid angiosarcoma. The patient was scheduled for surgery in three weeks. Unfortunately, the patient died after one week after discharge secondary to pulseless ventricular tachycardia arrest followed by asystole. Moreover, we also present a brief literature review on pleural angiosarcoma. Ahmed Abu-Zaid and Shamayel Mohammed Copyright © 2013 Ahmed Abu-Zaid and Shamayel Mohammed. All rights reserved. Prenatal Diagnosis and Postnatal Findings of Bronchogenic Cyst Thu, 23 May 2013 18:41:37 +0000 Bronchogenic cysts arise from abnormal buds from the primitive esophagus and tracheobronchial tree, which do not extend to the site where alveolar differentiation occurs. Bronchogenic cysts are typically unilocular mucus field lesions arising from posterior membranous wall of the air way. The prenatal diagnosis usually is realized by two-dimensional ultrasound showing the large unilocular cystic image in the chest fetus. The prenatal percutaneous aspiration can reduce the risk of heart compression and permit better respiratory conditions to newborn. We present a case of a primiparous pregnant 23 year-old-woman prenatal ultrasound showed a large unilocular cyst in the left hemithorax with compression of the normal left lung tissue and contralateral mediastinal shift. This cyst was percutaneously aspirated without subsequent reaccumulation of fluid. The newborn did not have respiratory distress and the computed tomography scan confirmed the finding of a fluid-filled cyst in the left chest. The chest X-ray showed the displacement of the heart and the mediastinum from the left to the right. The prenatal diagnosis of bronchogenic cyst is very important to assess the degree of the compression of the normal lung and the mediastinum shift. Furthermore, the prenatal diagnosis permits planning delivery in the tertiary hospital with multidisciplinary team because of the risk of respiratory distress. Livia Teresa Moreira Rios, Edward Araujo Júnior, Luciano Marcondes Machado Nardozza, Antonio Fernandes Moron, and Marília da Glória Martins Copyright © 2013 Livia Teresa Moreira Rios et al. All rights reserved.