Case Reports in Pulmonology The latest articles from Hindawi Publishing Corporation © 2016 , Hindawi Publishing Corporation . All rights reserved. An Unusual Association in an Uncommon Disease: Two Cases of Spontaneous Pneumomediastinum Associated with Pneumorrhachis Tue, 26 Apr 2016 12:50:07 +0000 Pneumomediastinum, the presence of free air in the mediastinum, is described as spontaneous pneumomediastinum when there is no apparent cause such as trauma, surgery, interventional procedures, or intrathoracic infections. Pneumorrhachis is a rare clinical condition, consisting of intraspinal air. The main causes are iatrogenic, traumatic, and nontraumatic. Spontaneous mediastinum is usually associated with subcutaneous emphysema and, occasionally, with pneumothorax; however, its association with pneumorrhachis is extremely rare. Here, we present two rare cases of spontaneous pneumomediastinum associated with pneumorrhachis caused by vigorous coughing. Luís Martins, Patrícia Dionísio, Susana Moreira, Alda Manique, Isabel Correia, and Cristina Bárbara Copyright © 2016 Luís Martins et al. All rights reserved. Severe Acute Pulmonary Toxicity Associated with Brentuximab in a Patient with Refractory Hodgkin’s Lymphoma Sun, 17 Apr 2016 14:13:58 +0000 Acute pulmonary toxicity associated with brentuximab appears to be a rare but serious adverse effect that can be potentially fatal. We report the case of a twenty-nine-year-old female with Hodgkin’s lymphoma who was treated with brentuximab and later presented with severe acute pulmonary toxicity; she improved after the discontinuation of brentuximab and administration of antibiotics and glucocorticoid therapy. Currently there is very little data in the literature in regard to the clinical manifestations and characteristics of patients taking brentuximab and the potential development of acute severe pulmonary toxicity, as well as the appropriate therapeutic approach, making this particular case of successful treatment and resolution unique. Yasmin Sabet, Saul Ramirez, Elizabeth Rosell Cespedes, Marimer Rensoli Velasquez, Mateo Porres-Muñoz, Sumit Gaur, Juan B. Figueroa-Casas, and Mateo Porres-Aguilar Copyright © 2016 Yasmin Sabet et al. All rights reserved. Mediastinal Mature Teratoma Revealed by Empyema Thu, 07 Apr 2016 06:35:41 +0000 Teratomas are germ cell tumors, manifested with a great variety of clinical features; the most common extragonadal site is the anterior mediastinum. In this case, we report the patient with a large mature mediastinal teratoma with several components of ectodermal and endothermal epithelium. A 24-year-old female patient presented with history of persistent chest pain and progressively aggravating dyspnea for the previous 3 months. A chest X-ray showed a large opacity of the entire left hemithorax. Transcutaneous needle aspiration revealed a purulent fluid. The tube thoracostomy was introduced and the effusion was evacuated. Some weeks later, patient was seen in emergency for persistent cough and lateral chest pain. CT scan revealed a mass of the left hemithorax. The mass showed heterogeneous density, without compressing mediastinum great vessels and left hilar structures. Lipase value was elevated in needle aspiration. The patient underwent a total resection of the mediastinum mass via a left posterolateral thoracotomy. Microscopy revealed a mature teratoma with cystic structures. The patient subsequently made a full recovery. This case provide benign mediastinal teratoma with total atelectasis of left lung and elevated lipase value in needle transcutaneous aspiration; this event is explained by pancreatic component in the cystic tumor. Total removal of the tumor is adequate treatment for this type of teratoma and the prognosis is excellent. Mohammed Raoufi, Laila Herrak, Anas Benali, Leila Achaachi, Mustapha El Ftouh, Salma Bellarbi, Charaf Tilfine, and Firdaous Taouarsa Copyright © 2016 Mohammed Raoufi et al. All rights reserved. Stent-in-Stent Technique for the Treatment of Proximal Bronchial Restenosis after Insertion of Metallic Stents: A Report of Two Cases Thu, 24 Mar 2016 11:40:47 +0000 Endoscopic treatment of a bronchial restenosis previously treated by insertion of a partially covered self-expandable metallic stent (SEMS) can be difficult. Classically, after recanalization of the bronchus, the stent is removed and replaced by a more adapted one. We report on two cases of proximal bronchial restenosis treated by insertion of an additional stent inside the lumen of the previously inserted stent using the stent-in-stent (SIS) technique. The indications for the initial stent were malignancy in Patient 1 and posttransplant bronchial stenosis in Patient 2. Restenosis occurred at the proximal end of the stent within months in both cases. Stent removal and insertion of a new stent were considered, but this option was discarded because of an excessive risk of bronchial perforation and preference towards an alternative approach. In both cases, a second customized SEMS was placed using the SIS technique after ablation of the proximal end stenosis of the stent by argon plasma coagulation and/or dilation with a balloon. Recanalization of the bronchus was achieved in both cases without complications. The SIS technique is a valuable alternative to removal of SEMS in case of proximal bronchial restenosis. Benjamin Bondue, Pascal Schlossmacher, Christiane Knoop, Isabelle Etienne, Sylvie Luce, Youri Sokolow, and Dimitri Leduc Copyright © 2016 Benjamin Bondue et al. All rights reserved. TINF2 Gene Mutation in a Patient with Pulmonary Fibrosis Sun, 20 Mar 2016 13:28:54 +0000 Pulmonary fibrosis is a frequent manifestation of telomere syndromes. Telomere gene mutations are found in up to 25% and 3% of patients with familial disease and sporadic disease, respectively. The telomere gene TINF2 encodes an eponymous protein that is part of the shelterin complex, a complex involved in telomere protection and maintenance. A TINF2 gene mutation was recently reported in a family with pulmonary fibrosis. We identified a heterozygous Ser245Tyr mutation in the TINF2 gene of previously healthy female patient that presented with progressive cough due to pulmonary fibrosis as well as panhypogammaglobulinemia at age 52. Retrospective multidisciplinary evaluation classified her as a case of possible idiopathic pulmonary fibrosis. Telomere length-measurement indicated normal telomere length in the peripheral blood compartment. This is the first report of a TINF2 mutation in a patient with sporadic pulmonary fibrosis, which represents another association between TINF2 mutations and this disease. Furthermore, this case underlines the importance of telomere dysfunction and not telomere length alone in telomere syndromes and draws attention to hypogammaglobulinemia as a manifestation of telomere syndromes. T. W. Hoffman, J. J. van der Vis, M. F. M. van Oosterhout, H. W. van Es, D. A. van Kessel, J. C. Grutters, and C. H. M. van Moorsel Copyright © 2016 T. W. Hoffman et al. All rights reserved. Tracheobronchomegaly as a Cause of Bronchiectasis in an Adult Sun, 28 Feb 2016 12:17:37 +0000 Mounier-Kuhn syndrome (MKS) is a rare congenital anomaly characterized by dilated trachea and main bronchi due to atrophy of the elastic fibers and smooth muscle cells of the trachea and major airways. Patients of MKS can have varied presentation. The diagnosis is established radiologically and bronchoscopically. There is no specific treatment for MKS. We present an adult man with MKS who presented with recurrent respiratory tract infection. The diagnosis was confirmed by imaging study and fiberoptic bronchoscopy. Vishnukanth Govindaraj, Madhusmita Mohanty Mohapatra, Balla Nagamalli Kumar, and Suryakala Narayanasami Copyright © 2016 Vishnukanth Govindaraj et al. All rights reserved. Community-Acquired Moraxella catarrhalis Bacteremic Pneumonia: Two Case Reports and Review of the Literature Thu, 18 Feb 2016 11:26:18 +0000 Moraxella (formerly Branhamella) catarrhalis was discovered at the end of the nineteenth century, and for many decades it was considered to be a harmless commensal of the upper respiratory tract. It is a Gram-negative, aerobic diplococcus considered to be the third most common pathogen isolated in childhood sinusitis and otitis media and in adult chronic lower respiratory disease, as well as an etiological agent of pneumonia in immunosuppressed patients or those with chronic obstructive pulmonary disease. Moraxella catarrhalis pneumonia is rarely associated with bacteremia. Here, we present two cases of community-acquired Moraxella catarrhalis bacteremic pneumonia. Miguel Angel Ariza-Prota, Ana Pando-Sandoval, Marta García-Clemente, David Fole-Vázquez, and Pere Casan Copyright © 2016 Miguel Angel Ariza-Prota et al. All rights reserved. Key Features in the Management of Pulmonary Carcinosarcoma Thu, 18 Feb 2016 07:06:35 +0000 Pulmonary carcinosarcoma represents a category of extremely rare tumours accounting for 0.1% of all lung malignancies. It is defined as a poorly differentiated non-small-cell carcinoma that contains a component of sarcoma or sarcoma-like elements. These biphasic tumours typically have a poor prognosis due to late diagnosis and early metastases. Preoperative tissue diagnosis is usually difficult due to the heterogeneity of the tumour, with biopsies often just reflecting one element of the tumour. By means of a case illustration and review of the literature, we discuss the optimal management of patients with pulmonary carcinosarcoma. Nikolaos Panagiotopoulos, Davide Patrini, Benjamin Adams, Jonathan Pararajasingham, Rajeev Shukla, Elaine Borg, Martin Hayward, and David Lawrence Copyright © 2016 Nikolaos Panagiotopoulos et al. All rights reserved. Acute Respiratory Failure due to Neuromyelitis Optica Treated Successfully with Plasmapheresis Wed, 17 Feb 2016 11:35:12 +0000 Neuromyelitis Optica (NMO) is a demyelinating autoimmune disease involving the central nervous system. Acute respiratory failure from cervical myelitis due to NMO is known to occur but is uncommon in monophasic disease and is treated with high dose steroids. We report a case of a patient with NMO who developed acute respiratory failure related to cervical spinal cord involvement, refractory to pulse dose steroid therapy, which resolved with plasmapheresis. Massa Zantah, Timothy B. Coyle, and Debapriya Datta Copyright © 2016 Massa Zantah et al. All rights reserved. Mucous Membrane Pemphigoid with Tracheal Involvement Wed, 03 Feb 2016 12:03:34 +0000 34-year-old African American female with history of pemphigoid presented with hemoptysis. Patient was found to have mucous membrane pemphigoid involving the oropharynx and extending to trachea, till just above main stem carina. Four other cases described mucosal pemphigoid involving the trachea. We hereby present a brief review of current consensus on management of mucous membrane pemphigoid with airway involvement. Arash Minaie and Salim R. Surani Copyright © 2016 Arash Minaie and Salim R. Surani. All rights reserved. Humoral Immunity in Bronchiectasis: Finding Good’s Syndrome Tue, 29 Dec 2015 14:07:51 +0000 We present a case of a 37-year-old man with a past history of a surgically removed thymoma, who presented with recurrent pulmonary infections and bronchiectasis. On further testing, he was found to have low total immunoglobulin levels, a constellation of findings known as Good’s syndrome. He responded well to immunoglobulin replacement, in addition to the usual treatments for bronchiectasis. We present this case to emphasize the association of bronchiectasis, low immunoglobulins, and thymomas and the role of immunoglobulin replacement as a treatment option. C. Pu, S. Sukhal, and S. Fakhran Copyright © 2015 C. Pu et al. All rights reserved. Endobronchial Enigma: A Clinically Rare Presentation of Nocardia beijingensis in an Immunocompetent Patient Thu, 24 Dec 2015 08:19:42 +0000 Nocardiosis is an opportunistic infection caused by the Gram-positive weakly acid-fast, filamentous aerobic Actinomycetes. The lungs are the primary site of infection mainly affecting immunocompromised patients. In rare circumstances even immunocompetent hosts may also develop infection. Diagnosis of pulmonary nocardiosis is usually delayed due to nonspecific clinical and radiological presentations which mimic fungal, tuberculous, or neoplastic processes. The present report describes a rare bronchoscopic presentation of an endobronchial nocardial mass in a 55-year-old immunocompetent woman without underlying lung disease. The patient exhibited signs and symptoms of unresolving community-acquired pneumonia with a computed tomography (CT) scan that showed a space-occupying lesion and enlarged paratracheal lymph node. This patient represents the unusual presentation of pulmonary Nocardia beijingensis as an endobronchial mass. Pathology obtained during bronchoscopy demonstrated polymerase chain reaction (PCR) confirmation of nocardiosis. Symptoms and clinical findings improved with antibiotic treatment. This patient emphasizes the challenge in making the diagnosis of pulmonary nocardiosis, especially in a low risk host. A literature review presents the difficulties and pitfalls in the clinical assessment of such an individual. Nader Abdel-Rahman, Shimon Izhakain, Walter G. Wasser, Oren Fruchter, and Mordechai R. Kramer Copyright © 2015 Nader Abdel-Rahman et al. All rights reserved. Interstitial Lung Disease in Werner Syndrome: A Case Report of a 55-Year-Old Male Patient Wed, 16 Dec 2015 12:41:54 +0000 Werner syndrome (WS) is a progeroid or premature aging syndrome characterized by early onset of age-related pathologies and cancer. The average life expectancy of affected people is 52.8 years and tends to increase. The major causes of death are malignancy and myocardial infarction. Increased telomere attrition and decay are thought to play a causative role in the clinical and pathological manifestations of the disease. Although telomere length, with or without germline mutation, is known to be associated with interstitial lung disease, the latter is not associated with WS. To the best of our knowledge, we report the first case describing a WS patient with fatal ILD. This case suggests that older patients with WS could develop ILD. Clinical outcome of WS patients may thus be improved by counselling them regarding smoking cessation or other exposure and by proposing antifibrotic therapy. Tiphaine Goletto, Flora Crockett, Selim Aractingi, Cecile Toper, Patricia Senet, Jacques Cadranel, and Jean-Marc Naccache Copyright © 2015 Tiphaine Goletto et al. All rights reserved. Pulmonary Vein Stenosis Mimicking Nonspecific Interstitial Pneumonia Tue, 08 Dec 2015 08:31:13 +0000 Pulmonary vein stenosis (PVS) is a known complication after catheter ablation of arrhythmias. Surprisingly, little information is available on its manifestations in the lung. We describe the case of a 39-year-old woman who presented from an outside hospital with worsening shortness of breath after catheter ablation of pulmonary veins for atrial fibrillation. After an initial diagnosis of pneumonia and its nonimprovement with antibiotics, a surgical lung biopsy was done and interpreted as nonspecific interstitial pneumonia (NSIP) with vascular changes consistent with pulmonary arterial hypertension. Later, she was admitted to our institution where a transthoracic echocardiogram (TTE) and subsequent computed tomography (CT) angiogram of the heart showed severe stenosis of all four pulmonary veins. The previous lung biopsy was rereviewed and reinterpreted as severe parenchymal congestion mimicking NSIP. Our case demonstrates that PVS is an underrecognized complication of catheter ablation, and increased awareness among both clinicians and pathologists is necessary to avoid misdiagnosis. Karthika R. Linga, Andras Khoor, Jonathan A. Phelan, and Isabel Mira-Avendano Copyright © 2015 Karthika R. Linga et al. All rights reserved. Silicone Breast Implants: A Rare Cause of Pleural Effusion Thu, 26 Nov 2015 13:45:55 +0000 Pleural effusions are one of the rarest complications reported in patients with silicone gel filled breast implants. The silicone implants have potential to provoke chronic inflammation of pleura and subsequent pulmonary complications such as pleural effusion. Herein, we report a 44-year-old female who presented with left sided pleural effusion, six weeks after a silicone breast implantation surgery. The most common infectious, inflammatory, and malignant causes of pleural effusion were excluded with pleural fluid cytology and cultures. With recurrent effusion in the setting of recent surgery, the chemical reaction to silicone breast implants was sought and exploration was performed which revealed foreign body reaction (FBR) to silicone material. The symptoms dramatically improved after the explantation. Imam H. Shaik, Bindu Gandrapu, Fernando Gonzalez-Ibarra, David Flores, Jyoti Matta, and Amer K. Syed Copyright © 2015 Imam H. Shaik et al. All rights reserved. Untreated Active Tuberculosis in Pregnancy with Intraocular Dissemination: A Case Report and Review of the Literature Thu, 26 Nov 2015 12:09:39 +0000 Background. Tuberculosis (TB) is a disease that affects hundreds of millions of people across the world. However, the incidence in developed countries has decreased over the past decades causing physicians to become unfamiliar with its unspecific symptoms. Pregnant individuals are especially difficult because many symptoms of active TB can mimic normal physiological changes of pregnancy. We present a case report of a 26-year-old multiparous woman, G4P3003, at 38-week gestation with a history of positive PPD who emigrated from Ghana 6 years ago. She came to the hospital with an initial complaint of suprapubic pain, pressure, and possible leakage of amniotic fluid for the past week. Patient also complained of a productive cough for the past 3 to 4 months with a decrease in vision occurring with the start of pregnancy. Visual acuity was worse than 20/200 in both eyes. Definitive diagnosis of active TB was delayed due to patient refusal of chest X-ray. Fortunately, delay in diagnosis was minimized since patient delivered within 24 hours of admission. Active TB was confirmed with intraocular dissemination. Patient had optic atrophy OS (left eye) and papillitis, choroiditis, and uveitis OD (right eye) due to TB infiltration. Fetus was asymptomatic and anti-TB therapy was started for both patients. Shadi Rezai, Stephen LoBue, Daniel Adams, Yewande Oladipo, Ramses Posso, Tiffany Mapp, Crystal Santiago, Manisha Jain, William D. Marino, and Cassandra E. Henderson Copyright © 2015 Shadi Rezai et al. All rights reserved. A Large Pleural Effusion following Abdominal Aortic Surgery Mon, 09 Nov 2015 13:02:13 +0000 Chylous ascites and coexistent chylothorax is a rare but important complication following retroperitoneal abdominal surgery. We report a 70-year-old male who developed gradual abdominal distension, chest tightness, and dyspnea five months after having an uncomplicated aortobifemoral bypass performed. Physical examination was consistent with a large right sided effusion and ascites which were confirmed by computed tomography. Thoracentesis yielded an opaque milky fluid with analysis consistent with a chylothorax with a paracentesis revealing fluid that was similar in both appearance and biochemistry. The patient failed initial conservative management so a chest tube was placed followed by chemical pleurodesis. We review the literature of the pathophysiology and treatment approach to such a pleural effusion. Vinoo K. Ramsaran, Vandana K. Seeram, James Cury, and Adil Shujaat Copyright © 2015 Vinoo K. Ramsaran et al. All rights reserved. Severe Respiratory Distress in a Child with Pulmonary Idiopathic Hemosiderosis Initially Presenting with Iron-Deficiency Anemia Sun, 08 Nov 2015 11:57:54 +0000 Idiopathic pulmonary hemosiderosis (IPH) is a rare cause of alveolar hemorrhage in children but should be considered in children with anemia of unknown origin who develop respiratory complications. It is commonly characterized by the triad of recurrent hemoptysis, diffuse parenchymal infiltrates, and iron-deficiency anemia. Pathogenesis is unclear and diagnosis may be difficult along with a variable clinical course. A 6-year-old boy was admitted to the hospital with a severe iron-deficiency anemia, but he later developed severe acute respiratory failure and hemoptysis requiring intubation and mechanical ventilation. The suspicion of IPH led to the use of immunosuppressive therapy with high dose of corticosteroids with rapid improvement in clinical condition and discharge from hospital. A. Potalivo, L. Finessi, F. Facondini, A. Lupo, C. Andreoni, G. Giuliani, and C. Cavicchi Copyright © 2015 A. Potalivo et al. All rights reserved. Community-Acquired Pneumonia and Empyema Caused by Citrobacter koseri in an Immunocompetent Patient Sun, 08 Nov 2015 09:48:34 +0000 Citrobacter species, belonging to the family Enterobacteriaceae, are environmental organisms commonly found in soil, water, and the intestinal tracts of animals and humans. Citrobacter koseri is known to be an uncommon but serious cause of both sporadic and epidemic septicemia and meningitis in neonates and young infants. Most cases reported have occurred in immunocompromised hosts. The infections caused by Citrobacter are difficult to treat with usual broad spectrum antibiotics owing to rapid generation of mutants and have been associated with high death rates in the past. We believe this is the first case described in the literature of a community-acquired pneumonia and empyema caused by Citrobacter koseri in an immunocompetent adult patient. Miguel Angel Ariza-Prota, Ana Pando-Sandoval, Marta García-Clemente, Ramón Fernández, and Pere Casan Copyright © 2015 Miguel Angel Ariza-Prota et al. All rights reserved. Pulmonary Peripheral Carcinoids after Diffuse Idiopathic Pulmonary Neuroendocrine Cell Hyperplasia and Tumorlets: Report of 3 Cases Tue, 03 Nov 2015 09:29:07 +0000 Diffuse idiopathic pulmonary neuroendocrine cell hyperplasia (DIPNECH) and tumorlets are neuroendocrine cells proliferations smaller than 5 mm. The former confines to bronchial/bronchiolar wall, while the latter broke through epithelial basement membrane. The authors present 3 cases of DIPNECH and tumorlets associated with a typical peripheral carcinoid tumor without underlying lung disease. The patients presented with nonspecific pulmonary symptoms: 3 females, 60, 72, and 84 years old, whose CT-scans showed well-defined pulmonary nodules, 2.2, 1.6, and 1.4 cm, respectively; first patient was submitted to lobectomy and the others underwent surgical biopsy. Whitish/brownish lobulated tumors corresponded to typical carcinoids (less than 2 mitoses/2 mm2 and without necrosis); polygonal/elongated cells under lobular pattern expressed CD56, chromogranin A, synaptophysin, and CK7; Ki-67 positivity was between 1 and 3%. Bronchial/bronchiolar wall neuroendocrine cell hyperplasia and several neuroendocrine nodules under 5 mm, with identical morphologic and immunoexpression, were observed, without lung disease. Typical carcinoid associated with DIPNECH and tumorlets without other pulmonary diseases is rare. Sporadic cases may recall embryonal neuroendocrine differentiation potentiality to develop peripheral hyperplasia, most commonly in underoxygenated parenchyma. The described cases are elucidative of peripheral spectrum of neuroendocrine cell tumour evolution, reinforcing higher female incidence as in central carcinoids, still without a clear preneoplastic lesion. Carlos Abrantes, Rui Caetano Oliveira, Joana Saraiva, João Bernardo, and Lina Carvalho Copyright © 2015 Carlos Abrantes et al. All rights reserved. Giant Right Intrathoracic Myxoid Fusocellular Lipoma Mon, 05 Oct 2015 11:53:28 +0000 Intrathoracic lipomas are rare benign tumors; their behavior is not completely clear and their surgical removal may be challenging. We report a case of a giant right intrathoracic myxoid fusocellular lipoma compressing the lung, tracheobronchial tree, and esophagus which was removed through a posterolateral thoracotomy. Complete removal resulted in resolution of the chest pain and improvement of the dyspnea, with no recurrence at 4-year follow-up. Petre V. H. Botianu, Anda Mihaela Cerghizan, and Alexandru M. Botianu Copyright © 2015 Petre V. H. Botianu et al. All rights reserved. Pulmonary Nocardiosis in the Immunocompetent Host: Case Series Wed, 30 Sep 2015 13:49:32 +0000 Pulmonary nocardiosis is commonly recognized as an opportunistic infection in patients with predisposing immunosuppressive conditions. However, reports of pulmonary nocardiosis in the immunocompetent host are rare. Here, we report a case series of four patients with pulmonary nocardiosis without a predisposing condition. Inderjit Singh, Frances Mae West, Abraham Sanders, Barry Hartman, and Dana Zappetti Copyright © 2015 Inderjit Singh et al. All rights reserved. The Rapid Initiation, Titration, and Transition from Intravenous to Oral Treprostinil in a Patient with Severe Pulmonary Arterial Hypertension Thu, 17 Sep 2015 11:57:37 +0000 In patients who require urgent initiation of pulmonary arterial hypertension medications due to disease progression, it is customary to start intravenous prostacyclin therapy, typically during a hospital admission. If there are complicating factors or relative contraindications to intravenous and subcutaneous prostanoids, oral treprostinil provides another pathway to prostanoid therapy, but this usually requires a prolonged titration. We describe the case of a thirty-six-year-old male with severe pulmonary arterial hypertension and contraindication to intravenous and subcutaneous prostanoid therapy due to congenital deafness and the risk of not hearing the intravenous pump alarms. Intravenous treprostinil was initiated, titrated to high dose, and then rapidly transitioned to oral treprostinil. A rapid initiation, titration, and transition from intravenous to oral treprostinil can be safely performed under watchful supervision in order to achieve higher and more efficacious doses of oral treprostinil in a timely manner. James Benjamin Gleason, Justin Dolan, Pirouz Piran, and Franck Farzad Rahaghi Copyright © 2015 James Benjamin Gleason et al. All rights reserved. A Cause of Bilateral Chylothorax: A Case of Mesothelioma without Pleural Involvement during Initial Diagnosis Wed, 16 Sep 2015 14:21:32 +0000 Chylothorax is characterized by fluid accumulation in the pleural cavity containing chylomicrons due to disruption of lymphatic drainage in the thoracic ductus and development of chylothorax. A 60-year-old male patient presented to our clinic with shortness of breath and displayed bilateral pleural effusion and diffuse mediastinal lymph nodes in his computed chest tomography images. There were no thickening and nodular formation on the pleural surfaces. PET-CT showed no pathological FDG uptake. Thoracentesis showed a chylous effusion. Drainage reduced during monitoring could not be stopped; therefore, surgical intervention was considered. The patient underwent right thoracotomy. There were no pathological findings in the parietal and visceral pleura during the surgery. Initially lymphoma was considered. Perioperative samples were collected from the mediastinal lymph node. The pathology analysis reported metastasis of malignant mesothelioma. Evaluation of a repeated chest computed tomography showed nodular formations on the pleural surfaces. Mediastinal lymph nodes compressed the ductus thoracicus, resulting with chylothorax. The present case, with malignant mesothelioma, bilateral chylothorax, and mediastinal lymph node without any pleural involvement during initial diagnosis, is rare and will hence contribute to the literature. Ercan Kurtipek, Meryem İlkay Eren Karanis, Nuri Düzgün, Hıdır Esme, and Mustafa Çaycı Copyright © 2015 Ercan Kurtipek et al. All rights reserved. Legionella micdadei: A Forgotten Etiology of Growing Cavitary Nodules: A Case Report and Literature Review Tue, 15 Sep 2015 10:45:41 +0000 Background. Legionella micdadei is a Gram negative bacterium that can stain weakly acid fast. It was first described in 1979 after immunosuppressed patients developed pneumonia at a Pittsburgh VA, initially given the name Pittsburgh Pneumonia Agent. It is the second most common Legionella species causing infection after pneumophila, and typically infects immunocompromised hosts. It is not easy to be cultured which makes diagnosing difficult. Case Presentation. A 31-year-old female with ulcerative colitis, primary sclerosing cholangitis, and cirrhosis presented with fever, chills, shortness of breath, dry cough, and chest pain for five days after being started on immunosuppression for autoimmune hepatitis two months earlier. The first chest CT showed small bilateral cavitary nodules. The nodules continued to grow on subsequent imaging despite what was thought to be appropriate therapy. A transthoracic biopsy was performed which grew Legionella micdadei and the patient improved after being treated with levofloxacin. Conclusion. Legionella micdadei is an atypical pathogen known to cause pneumonia in immunosuppressed patients. This case highlights a typical presentation of an atypical infection not commonly thought about and should be considered when nodules are growing despite being on broad antimicrobial therapy. Daniel Lachant and Paritosh Prasad Copyright © 2015 Daniel Lachant and Paritosh Prasad. All rights reserved. Recurrent Pleural Effusions Occurring in Association with Primary Pulmonary Amyloidosis Wed, 02 Sep 2015 12:03:09 +0000 Recurrent pleural effusions occurring in association with immunoglobulin light chain amyloidosis and not associated with amyloid cardiomyopathy are rare. These portend an overall poor prognosis with mean survival time of approximately 1.8 months. We hereby report a case of a 59-year-old Caucasian female with recurrent pleural effusions and an ultimate diagnosis of pulmonary amyloidosis in association with plasma cell myeloma. The optimal treatment for recurrent pleural effusions in amyloidosis has not been determined; however, our patient responded to therapy with Cyclophosphamide-Bortezomib- (Velcade-) Dexamethasone (CyBorD) and had no repeat hospitalizations or recurrence of pleural effusion at four-month follow-up after initiation of therapy. Lauren Tada, Humayun Anjum, W. Kenneth Linville, and Salim Surani Copyright © 2015 Lauren Tada et al. All rights reserved. Right Heart Transvalvular Embolus with High Risk Pulmonary Embolism in a Recently Hospitalized Patient: A Case Report of a Therapeutic Challenge Mon, 31 Aug 2015 12:23:37 +0000 Thrombus-in-transit is not uncommon in pulmonary embolism but Right Heart Transvalvular Embolus (RHTVE) complicating this is rare. A 54-year-old obese male with recent hospitalization presented with severe dyspnea and collapse. Initial investigations revealed elevated d-dimer and troponin. CTA showed saddle pulmonary embolus and bedside echocardiogram revealed right ventricular (RV) pressure overload and dilatation (RV > 41 mm), McConnell’s sign, and mobile echodensity attached to tricuspid valve. Patient was immediately resuscitated and promptly transferred for surgical embolectomy under cardiopulmonary bypass. A long segment of embolus traversing through the tricuspid valve and extensive bilateral pulmonary artery embolus were removed. IVC filter was placed for a persistent right lower extremity DVT. Hypercoagulable work-up was negative. Patient continued to do well after discharge on Coumadin. Open embolectomy offers great promises where there is no consensus in optimal management approach in such patients. Bedside echocardiogram is vital in risk stratification and deciding choice of advanced PE treatment. Gyanendra Kumar Acharya, Ajibola Monsur Adedayo, Hejmadi Prabhu, Derek R. Brinster, and Parvez Mir Copyright © 2015 Gyanendra Kumar Acharya et al. All rights reserved. Pleuroparenchymal Fibroelastosis: A New Entity within the Spectrum of Rare Idiopathic Interstitial Pneumonias Wed, 26 Aug 2015 09:00:53 +0000 Pleuroparenchymal fibroelastosis (PPFE) is a rare entity that has been recently included in the official American Thoracic Society/European Respiratory Society (ATS/ERS) statement in 2013 as a group of rare idiopathic interstitial pneumonias (IIPs). PPFE is characterized by pleural and subpleural parenchymal thickening due to elastic fiber proliferation, mainly in the upper lobes. The etiology of the disease is unclear, although some cases have been associated as a complication after bone marrow transplantation, lung transplantation (LT), chemotherapy, and recurrent respiratory infections. The patients usually report progressive dyspnea and dry cough and are predisposed to develop spontaneous or iatrogenic pneumothoraces after surgical lung biopsy (SLB) for its diagnosis. That is why better awareness with the clinical and radiologic features can help optimal management by the multidisciplinary team. Novel invasive techniques such as cryobiopsy may become useful tools in these patients as it could spare SLB. We present the first reported cases in Spain. Karina Portillo, Ignasi Guasch, Caroline Becker, Felipe Andreo, Maria Teresa Fernández-Figueras, José Ramirez Ruz, Carlos Martinez-Barenys, Samuel García-Reina, Pedro Lopez de Castro, Irene Sansano, Ana Villar, and Juan Ruiz-Manzano Copyright © 2015 Karina Portillo et al. All rights reserved. Metastatic Squamous Cell Carcinoma Component from an Adenosquamous Carcinoma of the Lung with Identical Epidermal Growth Factor Receptor Mutations Sun, 23 Aug 2015 08:12:08 +0000 The case reported is a young “light” ex-smoker who initially had a localized adenosquamous carcinoma bearing an epidermal growth factor receptor (EGFR) sensitizing mutation. He first recurred six months after initial treatment within the brain with a pure squamous histology and the same EGFR mutation. Surgical resection and radiation rendered him disease-free. Subsequent isolated recurrence within the lung eighteen months later was a pure adenocarcinoma, again with the same identified EGFR mutation. These histologic changes (from adenosquamous to pure squamous to pure adenocarcinoma) have been described but not before in the absence of any selection pressure with EGFR tyrosine kinase inhibitors. This case points out the histologic “flexibility” of EGFR mutant lung cancers and the importance for appropriate molecular testing in nonsmokers with lung cancer of any histologic type. Jarred Burkart, Konstantin Shilo, Weiqiang Zhao, Efe Ozkan, Amna Ajam, and Gregory A. Otterson Copyright © 2015 Jarred Burkart et al. All rights reserved. Successful Treatment of Bulla with Endobronchial Valves Wed, 12 Aug 2015 06:43:45 +0000 Emphysematous bullae are a complication of end-stage COPD. Patients with large bullae and poor respiratory function have limited treatment options. Surgical resection is a recognized treatment, but functional improvement after bullectomy is not satisfactory in patients with forced expiratory volume in 1 s (FEV1) < 35% predicted. When this 59-year-old male end-stage COPD patient was assessed, he was cachectic and lung function tests showed a FEV1 of 0.56 L (19% predicted) and a RV of 7 L (314% predicted), while 6MWT was 315 m and MRC dyspnea score was 4. Chest X-ray revealed a massive bulla of 10 cm in diameter in the right middle lobe. A fibrobronchoscopy was performed under local anesthesia and 2 Zephyr 4.0 valves were placed in the right middle lobe. Chest X-ray and CT scan performed 36 days later showed the complete resolution of the bulla. Seven months later, the patient demonstrated an improvement in FEV1 (+30%) and a decrease in RV from 314 to 262% predicted. This case report shows that the Zephyr valves may be successfully used to treat a large bulla in the right middle lobe in a patient with diffuse emphysema and severely impaired lung function. Erdoğan Çetinkaya, Mehmet Akif Özgül, Şule Gül, Hilal Boyacı, Ertan Cam, Emine Kamiloglu, and Mustafa Çörtük Copyright © 2015 Erdoğan Çetinkaya et al. All rights reserved.