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1
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DeCicco D, Alshaikhnassir E, Deepak V, Hadique S, Sangani R. Isolated pulmonary amyloidoma: A rare cause of solitary pulmonary nodule. Respir Med Case Rep 2023; 42:101820. [PMID: 36874267 PMCID: PMC9975676 DOI: 10.1016/j.rmcr.2023.101820] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/03/2022] [Revised: 01/23/2023] [Accepted: 02/16/2023] [Indexed: 02/22/2023] Open
Abstract
Pulmonary nodules are a frequent finding on imaging, especially given screening guidelines for lung cancer with low dose computed tomography (CT) scan. Here, we report a case with a single pulmonary nodule in a patient exposed to coal dust and asbestos. The nodule had benign features, but it showed an increase in size on repeated imaging. A CT-guided biopsy followed by mass spectrometry of the sample identified the nodule as the AL subtype of amyloidoma. A bone marrow biopsy was without evidence for malignancy including lymphoma. Nodular pulmonary amyloidosis (NPA) is rare, and a biopsy is required to establish the diagnosis. NPA generally does not affect lung function or impact survival; thus NPA does not require specific therapy. This case is the first documented case associated with coal-dust exposure. High-risk patients need to be followed longitudinally due to association of amyloidosis with lymphoma and other systemic conditions.
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Affiliation(s)
- Danielle DeCicco
- Department of Medicine, West Virginia University, Morgantown, WV, 26505, USA
| | - Esra Alshaikhnassir
- Department of Pathology, West Virginia University, Morgantown, WV, 26505, USA
| | - Vishal Deepak
- Section of Pulmonary, Critical Care and Sleep Medicine, Department of Medicine, West Virginia University Morgantown, WV, 26505, USA
| | - Sarah Hadique
- Section of Pulmonary, Critical Care and Sleep Medicine, Department of Medicine, West Virginia University Morgantown, WV, 26505, USA
| | - Rahul Sangani
- Section of Pulmonary, Critical Care and Sleep Medicine, Department of Medicine, West Virginia University Morgantown, WV, 26505, USA
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2
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Gaultier S, Puscas T, Pastre J, Gibault L, Arlet JB, Cauquil C, Michon A. Transbronchial cryobiopsy proven amyloid diffuse cystic lung disease complicating a transthyretin mutated (ATTRm) amyloidosis: a case report. Ther Adv Respir Dis 2023; 17:17534666231178690. [PMID: 37329289 PMCID: PMC10278398 DOI: 10.1177/17534666231178690] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/14/2022] [Accepted: 05/12/2023] [Indexed: 06/19/2023] Open
Abstract
We present a case report of transbronchial cryobiopsy proven diffuse amyloid cystic lung disease complicating a homozygous Val122Ile (V122I) transthyretin mutated amyloidosis (ATTRm). To the best of our knowledge, this is the first case in the literature reporting such pulmonary lesions in ATTRm amyloidosis, and notably diagnosed through cryobiopsy. A 51-year-old man from Mali with a past medical history of bilateral carpal tunnel syndrome presented erectile dysfunction, asthenia and worsening dyspnoea over the past year. He presented signs of cardiac failure; histological and radiological investigations diagnosed cardiac amyloidosis. He was found homozygote for the V122I mutation in transthyretin. A diffuse cystic lung disease (DCLD) was noted on computed tomography (CT) scan. We performed a transbronchial pulmonary cryobiopsy that revealed histological transthyretin amyloid deposits. This case report illustrates the safety and usefulness of cryobiopsy in the setting of DCLD and extends ATTRm amyloidosis as a possible cause of DCLD.
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Affiliation(s)
- Sébastien Gaultier
- Service de Médecine Interne, Hôpital Européen Georges-Pompidou, Assistance Publique–Hôpitaux de Paris, Université Paris Cité, Paris, France
| | - Tania Puscas
- Service de Cardiologie, Hôpital Européen Georges-Pompidou, Assistance Publique–Hôpitaux de Paris, Université Paris Cité, Paris, France
| | - Jean Pastre
- Service de Pneumologie, Hôpital Européen Georges-Pompidou, Assistance Publique–Hôpitaux de Paris, Université Paris Cité, Paris, France
| | - Laure Gibault
- Service d’Anatomie Pathologie, Hôpital Européen Georges-Pompidou, Assistance Publique–Hôpitaux de Paris, Université Paris Cité, Paris, France
| | - Jean-Benoît Arlet
- Service de Médecine Interne, Hôpital Européen Georges-Pompidou, Assistance Publique–Hôpitaux de Paris, Université Paris Cité, Paris, France
| | - Cécile Cauquil
- Service de Neurologie, Hôpital Bicêtre, Hôpitaux Universitaires Paris Saclay, Le Kremlin Bicêtre, France
| | - Adrien Michon
- Service de Médecine Interne, Hôpital Européen Georges-Pompidou, Assistance Publique–Hôpitaux de Paris, Université Paris Cité, 20 rue Leblanc 750015 Paris, France
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3
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Aslam W, Najafi AG, Lamb CR. Severe tracheobronchial amyloidosis. Am J Med Sci 2022; 364:366-368. [DOI: 10.1016/j.amjms.2022.04.006] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/21/2021] [Revised: 12/13/2021] [Accepted: 04/06/2022] [Indexed: 11/16/2022]
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5
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Glass LN, Sumon M, Goulart H, Ahari J. Disappearing nodules: spontaneously regressing pulmonary amyloidosis. BMJ Case Rep 2019; 12:12/5/e229718. [PMID: 31110070 DOI: 10.1136/bcr-2019-229718] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/04/2022] Open
Abstract
A case report of spontaneous regression of pulmonary amyloidosis, diffuse interstitial pattern, in an elderly patient.
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Affiliation(s)
- Lisa N Glass
- Division of Pulmonary, Critical Care and Sleep Disorders Medicine, George Washington University School of Medicine and Health Sciences, Washington, DC, USA
| | - Mahbubur Sumon
- Internal Medicine, Howard University College of Medicine, Washington, DC, USA
| | - Hannah Goulart
- School of Medicine and Health Sciences, The George Washington University, Washington, DC, USA
| | - Jalil Ahari
- Pulmonary and Critical Care, The George Washington University, Washington, DC, USA
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6
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Baumgart JV, Stuhlmann-Laeisz C, Hegenbart U, Nattenmüller J, Schönland S, Krüger S, Behrens HM, Röcken C. Local vs. systemic pulmonary amyloidosis—impact on diagnostics and clinical management. Virchows Arch 2018; 473:627-637. [DOI: 10.1007/s00428-018-2442-x] [Citation(s) in RCA: 25] [Impact Index Per Article: 3.6] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/19/2018] [Revised: 07/16/2018] [Accepted: 08/14/2018] [Indexed: 12/11/2022]
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Nakao M, Muramatsu H, Yamamoto E, Suzuki Y, Arakawa S, Tomooka K, Sakai Y, Fujita K, Sato H. A case of wild-type transthyretin amyloidosis associated with organizing pneumonia. J Rural Med 2017; 12:130-134. [PMID: 29255531 PMCID: PMC5721298 DOI: 10.2185/jrm.2935] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/20/2017] [Accepted: 06/08/2017] [Indexed: 12/04/2022] Open
Abstract
An 81-year-old man was referred to our hospital with bilateral multiple patchy opacities
on chest radiography. His chief complaints were a few months’ history of intermittent mild
cough and slightly yellow sputum. Chest computed tomography (CT) showed non-segmental
air-space consolidations with ground-glass opacities. Amyloid deposition with organizing
pneumonia (OP) was seen in transbronchial lung biopsy (TBLB) specimens from the left S8.
Three months later, the infiltration originally seen in the left lower lobe was remarkably
diminished, and new infiltrations in the lingual and right lower lobes were detected on
chest CT. Amyloid deposition with OP was seen in TBLB specimens from the left S4.
Transthyretin was detected following immunohistochemical examination. The presence of
wild-type transthyretin (ATTRwt) was proven using genetic analysis. The present report
describes a rare case of ATTRwt amyloidosis associated with OP.
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Affiliation(s)
- Makoto Nakao
- Department of Respiratory Medicine, Kainan Hospital Aichi Prefectural Welfare Federation of Agricultural Cooperatives, Japan
| | - Hideki Muramatsu
- Department of Respiratory Medicine, Kainan Hospital Aichi Prefectural Welfare Federation of Agricultural Cooperatives, Japan
| | - Eriko Yamamoto
- Department of Respiratory Medicine, Kainan Hospital Aichi Prefectural Welfare Federation of Agricultural Cooperatives, Japan
| | - Yuto Suzuki
- Department of Respiratory Medicine, Kainan Hospital Aichi Prefectural Welfare Federation of Agricultural Cooperatives, Japan
| | - Sousuke Arakawa
- Department of Respiratory Medicine, Kainan Hospital Aichi Prefectural Welfare Federation of Agricultural Cooperatives, Japan
| | - Ken Tomooka
- Department of Respiratory Medicine, Kainan Hospital Aichi Prefectural Welfare Federation of Agricultural Cooperatives, Japan
| | - Yusuke Sakai
- Department of Respiratory Medicine, Kainan Hospital Aichi Prefectural Welfare Federation of Agricultural Cooperatives, Japan
| | - Kouhei Fujita
- Department of Respiratory Medicine, Kainan Hospital Aichi Prefectural Welfare Federation of Agricultural Cooperatives, Japan
| | - Hidefumi Sato
- Department of Respiratory Medicine, Kainan Hospital Aichi Prefectural Welfare Federation of Agricultural Cooperatives, Japan
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9
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Abstract
Amyloidosis is a disorder of protein folding in which normally soluble plasma proteins aggregate in an abnormal fibrillar form causing progressive disruption to tissue structure and organ function. This review covers systemic AA and AL amyloidosis which may arise as a consequence of chronic respiratory conditions; the manifestations of both systemic and of localised amyloid deposition within the respiratory tract and provides a summary of current approaches to diagnosis and management.
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Affiliation(s)
- H J Lachmann
- National Amyloidosis Centre and Centre for Amyloidosis and Acute Phase Proteins, Department of Medicine, Royal Free and University College Medical School, Royal Free Campus, London, UK.
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Takahashi N, Glockner J, Howe BM, Hartman RP, Kawashima A. Taxonomy and Imaging Manifestations of Systemic Amyloidosis. Radiol Clin North Am 2016; 54:597-612. [DOI: 10.1016/j.rcl.2015.12.012] [Citation(s) in RCA: 7] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/17/2022]
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11
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Baulier G, Victor J, Mercié P, Lazaro E, Duffau P. Amylose pulmonaire AL révélée par une toux : à propos d’une observation. Rev Med Interne 2016; 37:363-6. [DOI: 10.1016/j.revmed.2015.08.008] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/22/2015] [Revised: 08/04/2015] [Accepted: 08/17/2015] [Indexed: 10/23/2022]
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12
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Saraya T, Nunokawa H, Fujiwara M, Ohkuma K, Tsujimoto N, Tsukahara Y, Ishii H, Goto H, Takizawa H. Tracheobronchial Amyloidosis in a Patient with Sjögren's Syndrome. Intern Med 2016; 55:981-4. [PMID: 27086816 DOI: 10.2169/internalmedicine.55.5108] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 11/06/2022] Open
Abstract
A 65-year-old woman was referred to our respiratory department because of incidentally detected endobronchial deposits. She had been diagnosed with Sjögren's syndrome 12 years earlier. Bronchoscopy showed protrusion of the reddened, shiny or edematous mucosa at the orifice of the lower lobe bronchus, suggesting a submucosal tumor. Based on the pathological findings of the transbronchial biopsied specimens, the patient was diagnosed with non-classified type tracheobronchial amyloidosis associated with Sjögren's syndrome, which was negative for both λ and κ chains, transthyretin and amyloid A. She has remained in good health without a relapse of the tumor.
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Affiliation(s)
- Takeshi Saraya
- Department of Respiratory Medicine, Kyorin University School of Medicine, Japan
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13
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[Tracheobronchial amyloidosis: Diagnosis, treatment and evolution in 5 patients]. Med Clin (Barc) 2015; 145:460-2. [PMID: 25865612 DOI: 10.1016/j.medcli.2015.01.024] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 12/25/2014] [Revised: 01/23/2015] [Accepted: 01/29/2015] [Indexed: 10/23/2022]
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Abstract
OBJECTIVE Recent technical advances, including the routine use of CT thin sections and techniques such as 2D minimum-intensity-projection and 3D volume images, have increased our ability to detect large airways diseases. Furthermore, dedicated CT protocols allow the evaluation of dynamic airway dysfunction. CONCLUSION With diseases of the large airways more commonly seen in daily practice, it is important that radiologists be familiar with the appearances, differential diagnosis, and clinical implications of these entities.
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Abstract
Primary tracheobronchial amyloidosis (TBA) is a rare disease characterized by extracellular focal or diffuse submucosal deposits of amyloid proteins. Various types of endobronchial lesions have been described in TBA when bronchoscopy is performed using white light. Narrow-band imaging bronchoscopy has been mainly employed for detecting preneoplastic and neoplastic endobronchial lesions as it provides more detailed images of the microvasculature reflective of an altered angiogenesis process. Here, we describe bronchoscopic findings with white light and narrow-band imaging in 2 patients presenting with central airway obstructive disease later confirmed as having primary TBA.
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Abstract
A 50-year-old man presented to clinic with a two-year history of progressive exertional dyspnea and voice hoarseness. This history suggested upper airways obstruction, which was confirmed on computed tomography imaging that revealed extensive thickening of the proximal tracheal wall causing severe luminal narrowing. Bronchoscopic debulking was then performed and the samples obtained confirmed tracheal amyloidoisis. Extensive investigation confirmed that disease was localized solely to the trachea. Ultimately, after multiple discussions, the chosen treatment modality was radiotherapy, which proceeded relatively uneventfully and achieved excellent radiological and clinical response. Although tracheal amyloidosis is rare, it is most commonly observed as part of a multisystem presentation. The present report describes the even more uncommon diagnosis of isolated tracheal amyloidosis and highlights the role of radiotherapy in its management.
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Abstract
Pleural effusion is a common problem dealt by most of the practicing clinicians. Some causes for pleural effusion are less often considered as a differential diagnosis owing to its rarity. Here we report a case of renal amyloidosis on alternate day haemodialysis for about two months time presenting with left sided pleural effusion. On evaluation this turned out to be a case of amyloidosis on thoracoscopic pleural biopsy suggesting the possibility of Primary systemic amyloidosis.
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18
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Higo H, Fujiwara K, Watanabe H, Makimoto G, Kameyama N, Matsushita M, Rai K, Sato K, Inomata T, Sunami K, Shibayama T. Diffuse parenchymal pulmonary amyloidosis showing an objective response to bortezomib-based chemotherapy. Intern Med 2014; 53:1809-12. [PMID: 25130116 DOI: 10.2169/internalmedicine.53.1728] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 11/06/2022] Open
Abstract
A 77-year-old woman was admitted because of bilateral hand numbness and dyspnea on exertion. Her serum IgG was increased, and a bone marrow aspiration analysis supported a diagnosis of multiple myeloma. Additionally, computed tomography scans of the chest showed bilateral ground glass attenuations, linear opacities, and consolidations. Transbronchial lung biopsy revealed Congo Red-positive amorphous eosinophilic deposits. She was therefore diagnosed with diffuse parenchymal pulmonary amyloidosis accompanied by multiple myeloma. Following combination chemotherapy including bortezomib, her serum monoclonal protein levels were normalized, and pulmonary function and oxygenation improved.
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Affiliation(s)
- Hisao Higo
- Department of Respiratory Medicine, National Hospital Organization Okayama Medical Center, Japan
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19
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Abstract
There are multiple etiologies for tracheal stenosis, postintubation stenosis being the most common. In this study, we briefly report a unique form of tracheal stenosis with circumferential calcification at the site of the lesion. The patient underwent multiple therapeutic bronchoscopies for palliation without much relief.
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Khan AM, Manzoor K, Jain V, Mahadevia P, Berman A. Detection of nodular pulmonary amyloid by PET positive scan--deception for lung cancer. REVISTA PORTUGUESA DE PNEUMOLOGIA 2012; 18:299-303. [PMID: 22622031 DOI: 10.1016/j.rppneu.2012.01.007] [Citation(s) in RCA: 8] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/16/2011] [Accepted: 01/31/2012] [Indexed: 10/28/2022] Open
Abstract
Amyloidosis results from proteins being deposited as insoluble β-pleated sheets and disrupting organ function. Each precursor protein induces a separate spectrum of organ involvement, and different disease manifestations within the lung. Although autopsy findings often demonstrate amyloid deposits in various compartments of the lung, few are manifested pathologically. Amyloid lung nodules with positron emission tomography (PET) uptake are rare. We describe a rare case where PET uptake was detected in a pulmonary amyloid nodule. To our knowledge there are six previously reported cases in the English literature. This review also focuses on amyloid derived from immunoglobulin light-chain protein (AL disease), which most frequently involves the lung in both systemic and localized forms of the disease. Manifestations of AL-related lung disease range from nodules identified on incidental chest films to diffuse alveolar and septal deposition mimicking malignancy and or diffuse alveolar damage.
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Affiliation(s)
- A M Khan
- Department of Pulmonary, Albert Einstein College of Medicine, Montefiore Medical Center, Bronx, NY 10467, USA.
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22
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Bhavsar T, Huang Y, Gaughan C, Inniss S, Thomas R. Bilateral pulmonary nodular amyloidosis: A case report and review of the literature. World J Respirol 2012; 2:6-8. [DOI: 10.5320/wjr.v2.i2.6] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 02/06/2023] Open
Abstract
Pulmonary amyloidosis is uncommon and is usually associated with systemic amyloidosis. Localized pulmonary involvement in amyloidosis without systemic amyloidosis is even rarer; it is generally tracheobronchial or parenchymal in location. Parenchymal pulmonary amyloidosis is generally asymptomatic and an incidental finding, presenting as nodules varying in size and number, unilateral or bilateral. We present an unusual case of primary bilateral pulmonary nodular amyloidosis in an elderly female.
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Serraj M, Kamaoui I, Znati K, Kouara S, Sahnoune F, Amara B, Biaze ME, Tizniti S, Amarti A, Benjelloun MC. Pseudotumoral tracheobronchial amyloidosis mimicking asthma: a case report. J Med Case Rep 2012; 6:40. [PMID: 22289269 PMCID: PMC3285053 DOI: 10.1186/1752-1947-6-40] [Citation(s) in RCA: 13] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/15/2011] [Accepted: 01/30/2012] [Indexed: 11/29/2022] Open
Abstract
Introduction Tracheobronchial amyloidosis is an uncommon localized form of amyloidosis that can simulate a tracheal tumor. Clinical signs are not specific and the diagnosis is rarely given before performing a bronchoscopy with multiples biopsies. Case presentation We report the case of a 60-year-old Moroccan woman, complaining of dyspnea and wheezing for three years, who was treated at our institution for management of severe asthma. A bronchoscopy revealed a tumor formation of her trachea; multiples biopsies were performed and a diagnosis made of amyloid light-chain amyloidosis. She successfully received an endoscopic resection. Conclusion This case highlights the importance of routinely carrying out an endoscopy in any patient complaining of atypical bronchial symptoms or with uncontrolled asthma. Tracheal amyloidosis is a rare disease, confirmed by histological examination of bronchial biopsies, and the treatment of choice is based on the bronchoscopic resection.
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Affiliation(s)
- Mounia Serraj
- Department of Lung Disease, Hassan II University Hospital of Fez, Fez, Morocco.
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Zendah I, Khelifa MB, Ayadi A, Khattab A, Hantous S, Ghedira H. Systemic Al amyloidosis of the tracheobronchial tract and lungs: a rare finding. TANAFFOS 2012; 11:49-53. [PMID: 25191415 PMCID: PMC4153201] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Download PDF] [Figures] [Subscribe] [Scholar Register] [Received: 02/05/2012] [Accepted: 03/30/2012] [Indexed: 12/03/2022]
Abstract
A 67- year old man presented with cough, weight loss and night sweats. Fiberoptic bronchoscopy did not show any abnormality. Chest computed tomography scan revealed peribronchovascular thickening, sheathing and narrowing of some bronchi. There were also mediastinal and interbronchial Lymphadenopathies. The patient became lost to follow-up. He presented 5 years later with pneumonia. Flexible bronchoscopy showed diffuse infiltration of the bronchi suggesting lung cancer. Histopathological study with histochemical staining revealed tracheobronchial tract AL amyloidosis. Chest CT-scan revealed extension of the broncho-vascular thickening and superimposed pulmonary calcified nodules and lymphadenopathies. Labial biopsy revealed AL amyloidosis. No specific treatment of amyloidosis was thought to be necessary for the patient. At 6 years follow-up the disease had not progressed. This case report highlights the fact that even very rarely, systemic AL amyloidosis can involve the tracheobronchial tract. Moreover, the lungs and the tracheobronchial tract can, although rarely, be affected in the same patient.
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Affiliation(s)
- Inès Zendah
- Abderrahmane Mami Hospital of Thoracic Diseases. Department I of Pneumology. 2080. Ariana, Tunisia
| | - Mouna Ben Khelifa
- Abderrahmane Mami Hospital of Thoracic Diseases. Department I of Pneumology. 2080. Ariana, Tunisia
| | - Aïda Ayadi
- Abderrahmane Mami Hospital of Thoracic Diseases. Department of Pathology. 2080. Ariana, zunisia
| | - Amel Khattab
- Abderrahmane Mami Hospital of Thoracic Diseases. Department I of Pneumology. 2080. Ariana, Tunisia
| | - Saoussan Hantous
- Abderrahmane Mami Hospital of Thoracic Diseases. Department of Radiology. 2080. Ariana, Tunisia
| | - Habib Ghedira
- Abderrahmane Mami Hospital of Thoracic Diseases. Department I of Pneumology. 2080. Ariana, Tunisia
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Eguchi T, Yoshida K, Kobayashi N, Saito G, Hamanaka K, Shiina T, Kurai M, Endo M. Localized nodular amyloidosis of the lung. Gen Thorac Cardiovasc Surg 2011; 59:715-7. [DOI: 10.1007/s11748-010-0748-y] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 09/27/2010] [Accepted: 11/16/2010] [Indexed: 10/17/2022]
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Ozyigit LP, Kiyan E, Okumus G, Yilmazbayhan D. Isolated laryngo-tracheal amyloidosis presenting as a refractory asthma and longstanding hoarseness. J Asthma 2009; 46:314-7. [PMID: 19373642 DOI: 10.1080/02770900802660956] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 10/20/2022]
Abstract
Respiratory tract amyloidosis is characterized by deposition of amyloid fibril protein at any site along the respiratory tract ranging from the larynx to the pulmonary parenchyma. Usually, it is not associated with primary systemic amyloidosis. We report a case with isolated laryngotracheal amyloidosis, which is relatively a rare condition, together with a history of refractory asthma accompanied with longstanding hoarseness.
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Affiliation(s)
- Leyla Pur Ozyigit
- Department of Chest Medicine, Istanbul University, Istanbul Medical Faculty, Istanbul, Turkey
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28
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Kumaran R, Saleh A, Amin B, Raoof S. A 73-year-old woman with mild shortness of breath and multiple central calcified pulmonary nodules. Chest 2008; 134:460-464. [PMID: 18682468 DOI: 10.1378/chest.07-1955] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/01/2022] Open
Affiliation(s)
- Rani Kumaran
- Department of Pulmonary and Critical Care Medicine, New York Methodist Hospital, Brooklyn, NY
| | - Anthony Saleh
- Department of Pulmonary and Critical Care Medicine, New York Methodist Hospital, Brooklyn, NY
| | - Bijal Amin
- Department of Pathology, New York Weill Cornell Medical Center, New York, NY
| | - Suhail Raoof
- Department of Pulmonary and Critical Care Medicine, New York Methodist Hospital, Brooklyn, NY.
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Abstract
The term "amyloidoma" has been used to describe localized pulmonary nodular amyloidosis when it is a solitary lesion. Amyloidoma is an uncommon and infrequently reported cause of benign pulmonary lesions. We report the case of a 45-year-old man with hemoptysis, eosinophilia, and a large mass involving both lobes of the left lung, the chest wall, and, via extension through the diaphragm, the liver. Clinical suspicion of echinococcal cyst led to treatment via en bloc excision rather than attempting tissue biopsy for diagnosis. Complete resection of the isolated pulmonary amyloidoma was achieved with no evidence of recurrence.
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30
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Gaurav K, Panda M. An uncommon cause of bilateral pulmonary nodules in a long-term smoker. J Gen Intern Med 2007; 22:1617-20. [PMID: 17846845 PMCID: PMC2219804 DOI: 10.1007/s11606-007-0324-z] [Citation(s) in RCA: 7] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 08/25/2006] [Revised: 01/17/2007] [Accepted: 07/19/2007] [Indexed: 10/22/2022]
Abstract
BACKGROUND Dyspnea caused by pulmonary disease is a common symptom encountered by internists. The most likely diagnosis of pulmonary nodules in a long-term smoker is lung cancer. PATIENT/PARTICIPANT: We report a case of an elderly male with a 70-pack-year smoking history, presenting with exertional dyspnea for 6 months. INTERVENTIONS Detailed review of history was negative. Examination was normal except for diminished breath sounds in all lung fields. Chest x-ray showed bilateral nodular opacities. Computed tomography of thorax revealed multiple bilateral lung masses. A whole-body positron emission tomography revealed enhancement only of the pulmonary masses. Bronchoalveolar lavage was negative for acid fast bacilli, nocardia, and fungi. MAIN RESULTS Lung biopsy showed findings consistent with amyloidosis. Bone marrow biopsy done to investigate primary amyloidosis showed no clonal plasma cells or amyloid staining, thus suggesting a diagnosis of localized pulmonary amyloidosis. Patient is being managed conservatively with close follow-up for signs of progression.
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Affiliation(s)
- Kumar Gaurav
- Department of Medicine, University of Tennessee, College of Medicine-Chattanooga Unit, 975 East Third St., P.O. Box 94, Chattanooga, Tennessee 37403, USA.
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Gilad R, Milillo P, Som PM. Severe diffuse systemic amyloidosis with involvement of the pharynx, larynx, and trachea: CT and MR findings. AJNR Am J Neuroradiol 2007; 28:1557-8. [PMID: 17846211 PMCID: PMC8134395 DOI: 10.3174/ajnr.a0604] [Citation(s) in RCA: 31] [Impact Index Per Article: 1.7] [Reference Citation Analysis] [Abstract] [MESH Headings] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/07/2022]
Abstract
Amyloidosis is a term applied to a diverse group of disorders that share the deposition of amyloid protein in various extracellular tissues. Systemic amyloidosis may involve almost any organ system in the body including regions in the head and neck; however, pharyngeal involvement is rare, with only 12 cases having been previously reported. Ten of these cases were localized disease, and only 2 cases were systemic amyloidosis. We present the case of a patient with severe diffuse systemic amyloidosis with extensive involvement of the pharynx, larynx, trachea, lungs, eyelids, and breasts. We also review the imaging characteristics and pertinent literature.
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Affiliation(s)
- R Gilad
- Department of Neurosurgery, Mount Sinai School of Medicine of New York University, New York, USA
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32
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Chew HC, Low SY, Eng P, Agasthian T, Cheah FK. Cough and persistent wheeze in a patient with long-standing asthma. Chest 2007; 132:727-31. [PMID: 17699149 DOI: 10.1378/chest.06-2810] [Citation(s) in RCA: 8] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [MESH Headings] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/01/2022] Open
Affiliation(s)
- Huck Chin Chew
- Department of Respiratory and Critical Care Medicine, Singapore General Hospital, Outram Rd, Singapore.
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33
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NIEPOLSKI L, GRZEGORZEWSKA AE, SZYMAŚ J. Nodular pulmonary amyloidosis and Sjögren's syndrome in a patient treated with intermittent hemodialysis. Hemodial Int 2007; 11:406-10. [DOI: 10.1111/j.1542-4758.2007.00209.x] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/30/2022]
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34
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Eder L, Zisman D, Wolf R, Bitterman H. Pulmonary hypertension and amyloidosis--an uncommon association: a case report and review of the literature. J Gen Intern Med 2007; 22:416-9. [PMID: 17356978 PMCID: PMC1824747 DOI: 10.1007/s11606-006-0052-9] [Citation(s) in RCA: 21] [Impact Index Per Article: 1.2] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 11/25/2022]
Abstract
Only a limited number of patients with amyloidosis and pulmonary hypertension have been reported in the literature. We report a 73-year-old female with AL type amyloidosis who developed respiratory insufficiency and right heart failure because of severe pulmonary hypertension. There were no signs of cardiac involvement with amyloid or findings consistent with interstitial lung disease. Previous reports of pulmonary hypertension without an apparent parenchymal lung or myocardial involvement with amyloidosis are summarized. Pulmonary hypertension due to deposition of amyloid in the pulmonary vasculature is an uncommon finding; however, it should be considered in cases of unexplained pulmonary hypertension in patients with amyloidosis.
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Affiliation(s)
- Lihi Eder
- Department of Internal Medicine, Carmel Medical Center, Faculty of Medicine, Technion-Israel Institute of Technology, Haifa, Israel.
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Abstract
Familial Mediterranean Fever (FMF) is a hereditary periodic fever syndrome expressed by acute episodes of fever and painful manifestations. The gravest consequence of FMF is kidney involvement by secondary amyloidosis of AA type, which gradually leads to nephrotic syndrome and uremia. Nephropathic amyloidosis of the AA type, which complicates FMF in most untreated patients, may progress to effect other organs, including the lungs. This kind of organ involvement rarely produces noticeable symptoms and is associated with symptomatic involvement of other organs while remaining subclinical in itself. In this report, one case who had nephropathic and pulmonary amyloidosis of the secondary amyloidosis of AA type, wich complicates the FMF was presented and the pulmonary manifestations of FMF were reviewed.
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Affiliation(s)
- C Sahan
- Ondokuz Mayis University, School of Medicine, Department of Nephrology, Samsun, Turkey
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37
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Pitz MW, Gibson IW, Johnston JB. Isolated pulmonary amyloidosis: case report and review of the literature. Am J Hematol 2006; 81:212-3. [PMID: 16493622 DOI: 10.1002/ajh.20518] [Citation(s) in RCA: 26] [Impact Index Per Article: 1.4] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/10/2022]
Abstract
The respiratory tract is rarely the site of localized tracheobronchial or nodular amyloid deposits. We present an unusual case of isolated nodular amyloidosis of the lung and review the current literature related to this disorder.
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Affiliation(s)
- Marshall W Pitz
- Department of Internal Medicine, University of Manitoba, Winnipeg, Manitoba, Canada.
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38
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Chung MJ, Lee KS, Franquet T, Müller NL, Han J, Kwon OJ. Metabolic lung disease: imaging and histopathologic findings. Eur J Radiol 2005; 54:233-45. [PMID: 15837404 DOI: 10.1016/j.ejrad.2004.07.003] [Citation(s) in RCA: 53] [Impact Index Per Article: 2.7] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/24/2004] [Revised: 07/02/2004] [Accepted: 07/05/2004] [Indexed: 01/15/2023]
Abstract
Metabolic lung disease includes pulmonary alveolar proteinosis (PAP), pulmonary amyloidosis, metastatic pulmonary calcification, dendritic pulmonary ossification, pulmonary alveolar microlithiasis, and storage diseases. In pulmonary alveolar proteinosis, CT demonstrates air-space consolidation with thickened interlobular septa, producing the so-called "crazy paving" appearance. Pulmonary amyloidosis can appear as parenchymal nodules (nodular parenchymal form), diffuse interstitial deposit (diffuse interstitial form), or submucosal deposits in the airways (tracheobronchial form). Metastatic pulmonary calcification may appear on high-resolution CT as numerous 3- to 10-mm diameter calcified nodules or, more commonly as fluffy and poorly defined nodular opacities. In pulmonary microlithiasis, high-resolution CT demonstrates diffuse punctuate micronodules showing slight perilobular predominance resulting in apparent calcification of interlobular septa. Niemann-Pick disease appears as ground-glass attenuation in the upper lung zone and thickening of the interlobular septa in the lower lung zone. Radiologic study including high-resolution CT will be helpful for the diagnosis and follow-up of these diseases.
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Affiliation(s)
- Myung Jin Chung
- Department of Radiology and Imaging Science, Samsung Medical Center, Sungkyunkwan University School of Medicine, 50 Ilwon-Dong, Kangnam-Ku, Seoul 135-710, South Korea
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Affiliation(s)
- Om P Sharma
- Department of Medicine, Keck School of Medicine, Los Angeles, California 90033, USA.
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40
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Abstract
Amyloidosis is a disease caused by the deposition of abnormal proteins within extracellular soft tissues, and affects the respiratory tract in 50% of cases. Pulmonary involvement may be secondary to systemic disease or due to the accumulation of locally produced amyloid, and this differentiation has important prognostic and therapeutic implications. This pictorial essay, using cases of biopsy-proven respiratory tract amyloidosis, aims to depict the spectrum of imaging findings, and those of CT in particular, that can be of use in distinguishing between the 2 typical patterns of the disease.
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41
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Besancon MF, Stacy BA, Kyles AE, Moore PF, Vernau W, Smarick SD, Rasor LA. Nodular immunocyte-derived (AL) amyloidosis in the trachea of a dog. J Am Vet Med Assoc 2004; 224:1302-6, 1280-1. [PMID: 15112779 DOI: 10.2460/javma.2004.224.1302] [Citation(s) in RCA: 7] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/20/2022]
Abstract
A 7-year-old castrated male Miniature Schnauzer was examined because of labored breathing and episodes of respiratory distress that progressed to collapse. On cervical radiographs, a focal soft tissue mass in the caudal cervical portion of the trachea was observed, and during tracheoscopy, a 1 x 1 cm, pedunculated, multinodular, pink, intraluminal mass extending from the dorsal tracheal membrane and obstructing approximately 80% of the tracheal lumen was seen. Tracheal resection and anastomosis was performed to remove the mass, and the dog recovered without complications. On histologic examination, the mass consisted of a large accumulation of homogeneous, faintly fibrillar eosinophilic material admixed with a predominantly plasma cell infiltrate; examination of sections stained with thioflavin T and Congo red stain confirmed that the eosinophilic material was amyloid. A diagnosis of nodular, immunocyte-derived (AL) amyloidosis was made. Seventeen months after surgery, the dog had a relapse of respiratory distress because of an extramedullary plasmacytoma involving the trachea.
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Affiliation(s)
- M Faulkner Besancon
- Veterinary Medical Teaching Hospital, School of Veterinary Medicine, University of California, Davis, CA 95616, USA
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43
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Monroe AT, Walia R, Zlotecki RA, Jantz MA. Tracheobronchial amyloidosis: a case report of successful treatment with external beam radiation therapy. Chest 2004; 125:784-9. [PMID: 14769766 DOI: 10.1378/chest.125.2.784] [Citation(s) in RCA: 52] [Impact Index Per Article: 2.5] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/01/2022] Open
Abstract
Tracheobronchial amyloidosis (TBA) refers to the deposition of localized amyloid deposits within the upper airways. Treatments have historically focused on bronchoscopic techniques including debridement, laser ablation, balloon dilation, and stent placement. Local excisions often prove temporarily effective, with multiple local recurrences and progressive compromise pulmonary function occurring frequently. We present a case of TBA managed with definitive external beam radiation therapy. Eighteen months after moderate-dose radiation, the patient demonstrated improvements in functional status, pulmonary function, bronchoscopic visualization, and CT-based luminal diameters. The literature involving the role of radiation therapy in the treatment of TBA is reviewed.
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Affiliation(s)
- Alan T Monroe
- Department of Radiation Oncology, University of Florida College of Medicine, Health Science Center, Gainesville, FL 32610-0385, USA
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44
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45
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Ozer C, Nass Duce M, Yildiz A, Apaydin FD, Eğilmez H, Arpaci T. Primary diffuse tracheobrochial amyloidosis: case report. Eur J Radiol 2002; 44:37-9. [PMID: 12350409 DOI: 10.1016/s0720-048x(01)00437-5] [Citation(s) in RCA: 15] [Impact Index Per Article: 0.7] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/30/2022]
Abstract
Respiratory amyloidosis is a rare disease that occurs in three forms: tracheobronchial, nodular parenchymal and diffuse parenchymal involment. Tracheobronchial amyloidosis is characterized by focal or diffuse deposition of amyloid in the submucosa of the trachea and proximal bronchi. Herein, we report a case of diffuse tracheobronchial amyloidosis with plain radiography and thorax computed tomography findings.
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Affiliation(s)
- Caner Ozer
- Department of Radiology, Faculty of Medicine, Mersin University, Zeytinlibahçe Cad. Eski Otogar Yani 33070 Mersin, Turkey.
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46
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Murthy SC, Rice TW. The solitary pulmonary nodule: a primer on differential diagnosis. Semin Thorac Cardiovasc Surg 2002; 14:239-49. [PMID: 12232865 DOI: 10.1053/stcs.2002.34450] [Citation(s) in RCA: 18] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/11/2022]
Abstract
Despite significant advances in noninvasive imaging techniques, management of the solitary pulmonary nodule (SPN) remains a challenge for chest physicians. Patients with SPNs are frequently asymptomatic, and the physical examination is seldom revealing. Accurate diagnosis is essential, because >50% of patients will require prompt disease-specific therapy. The complexity of the problem is best appreciated by reviewing the differential list, which includes nearly 80 distinct clinical entities. Consequently, a thorough understanding of the more common etiologies is necessary to adequately treat patients with SPNs.
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Affiliation(s)
- S C Murthy
- Department of Thoracic and Cardiovascular Surgery, Section of General Thoracic Surgery, Cleveland Clinic Foundation, Cleveland, OH 44195, USA
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47
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Giménez A, Franquet T, Prats R, Estrada P, Villalba J, Bagué S. Unusual primary lung tumors: a radiologic-pathologic overview. Radiographics 2002; 22:601-19. [PMID: 12006690 DOI: 10.1148/radiographics.22.3.g02ma25601] [Citation(s) in RCA: 63] [Impact Index Per Article: 2.7] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/11/2022]
Abstract
Although the great majority of lung carcinomas are histologically characterized as adenocarcinoma, squamous cell carcinoma, large cell undifferentiated carcinoma, or small cell carcinoma, a variety of rare benign and malignant lung tumors may sporadically affect the lung. Several nonneoplastic tumorlike lesions are seen infrequently but are also part of the differential diagnosis for lung masses. Conventional radiographic findings, although of limited value in the diagnosis of these entities, should be examined carefully when lung tumors are suspected. Computed tomography (CT) is well suited for making a definitive diagnosis of some disease processes. CT helps determine the location and features of the lesions and depicts associated findings to help document the extent of disease. The differential diagnosis can be narrowed when there are typical CT features (eg, the presence of fat in lipoid pneumonia). Although unusual primary lung tumors are difficult to diagnose on the basis of imaging findings alone because such findings are nonspecific in the majority of cases, cross-sectional imaging can play an important role in the diagnostic work-up of these unusual tumors by delineating their extent and directing the radiologist or bronchoscopist to the appropriate biopsy site.
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Affiliation(s)
- Ana Giménez
- Department of Radiology, Hospital de la Santa Creu i Sant Pau, Universitat Autònoma de Barcelona, Sant Antoni M. Claret 167, 08025 Barcelona, Spain.
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48
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Cazalets C, Belleguic C, Sost G, Caulet-Maugendre S, Kernec J, Droz D, Grosbois B. [Tracheobronchial amyloidosis: apropos of 2 cases]. Rev Med Interne 2002; 23:317-21. [PMID: 11928380 DOI: 10.1016/s0248-8663(01)00557-4] [Citation(s) in RCA: 13] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 10/17/2022]
Abstract
INTRODUCTION Tracheo-bronchial amyloidosis is an uncommon localized form of amyloidosis. We report two new cases. EXEGESIS Two patients had developed expiratory dyspnea for several months. CT-scan and flexible bronchoscopy confirmed tracheal narrowing and a diagnosis of tracheo-bronchial amyloidosis was made by tissue biopsies. The immunohistochemical type was AL in one case, undetermined in the other case. There was no argument for systemic involvement. The two patients benefited from bronchoscopic dilatation. This treatment improved clinical symptoms and pulmonary function tests with a follow up of 12 and 18 months respectively. CONCLUSION Tracheo-bronchial amyloidosis is a localised form of amyloidosis with various respiratory symptoms. Diagnosis is made by CT-scan and flexible bronchoscopy that allows biopsies. Immunohistochemical type is more often AL. Recurrence, respiratory insufficiency and tracheo-bronchial metaplasia are the most important complications. Treatment consists of bronchoscopic dilatation or excision, and bronchoscopic laser-YAG. Pulmonary function testing allows precise follow-up.
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Affiliation(s)
- C Cazalets
- Service de médecine interne, hôpital Sud, 16, boulevard de Bulgarie, BP 5612, 35056 Rennes, France
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49
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Clark JM, Weissler MC. Localized laryngotracheobronchial amyloidosis: Case report and review of the literature. EAR, NOSE & THROAT JOURNAL 2001. [DOI: 10.1177/014556130108000908] [Citation(s) in RCA: 11] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/15/2022] Open
Abstract
Although localized laryngotracheobronchial amyloidosis is rare, the otolaryngologist–head and neck surgeon should be familiar with this condition. Its characteristic appearance can suggest its presence in a patient who has the typical initial symptoms. Biopsies during direct laryngoscopy and bronchoscopy can play both a diagnostic and therapeutic role. After an appropriate examination to rule out systemic involvement, the patient should be managed with conservative surgery, although the use of a CO2 laser might be more efficacious than conventional surgery. With appropriate diagnosis and treatment, patients should expect a favorable prognosis. In this article, we describe a new case of localized laryngotracheobronchial amyloidosis in a 67-year-old woman, and we review the literature on this subject.
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Affiliation(s)
- J. Madison Clark
- From the Department of Otolaryngology-Head and Neck Surgery, Medical University of South Carolina, Charleston
| | - Mark C. Weissler
- Department of Otolaryngology-Head and Neck Surgery, University of North Carolina School of Medicine, Chapel Hill
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50
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Yamamoto T, Maeda M, Mizuno S, Saga T, Itoh H. Primary diffuse tracheobronchial amyloidosis: radiologic findings. J Thorac Imaging 2001; 16:177-80. [PMID: 11428418 DOI: 10.1097/00005382-200107000-00007] [Citation(s) in RCA: 7] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/26/2022]
Abstract
Reports detailing the radiologic features of tracheobronchial amyloidosis are scant and typically describe only the computed tomography findings of this disease. No reports have described the magnetic resonance (MR) findings of tracheobronchial amyloidosis. With this case, the authors describe the radiologic findings in a patient with diffuse tracheobronchial amyloidosis, focusing particularly on the MR and bone scintigraphy findings in this disease.
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Affiliation(s)
- T Yamamoto
- Department of Diagnostic Radiology, Maizuru Kyosai Hospital, Maizuru City, Kyoto, Japan
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