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Langerhans histiocytosis

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https://www.readbyqxmd.com/read/28630846/chronic-recurrent-multifocal-osteomyelitis-a-case-report-with-atypical-presentation
#1
Miguel Pádua Figueiredo, Marco Pato, Fernando Amaral
INTRODUCTION: Chronic recurrent multifocal osteomyelitis (CRMO) is a rare autoinflammatory condition. The clinical picture consists of sterile osteomyelitis, typically with multiple-site lesions in the metaphysis of long bones and not uncommonly, symmetrical bone involvement. It is a poorly understood entity, whose prognosis, etiology and ideal treatment are still controversial. The authors report a case of unifocal presentation with an atypical location. CASE REPORT: A previously healthy 12-year-old Caucasian girl came to our institution due to progressive pain on her left thigh for the previous 3 months...
January 2017: Journal of Orthopaedic Case Reports
https://www.readbyqxmd.com/read/28626593/langerhans-cell-sarcoma-a-case-report-demonstrating-morphological-and-immunophenotypical-variability-within-a-single-lesion
#2
Rasika Singh, Charles Edward Keen, Christopher Stone, Patrick Sarsfield
Langerhans cells are antigen presenting dendritic cells and tumours arising from these are rare. The tumours arising from these dendritic cells are divided into two categories according to a WHO classification: Langerhans cell histiocytosis and Langerhans cell sarcoma. It is the degree of atypia and clinical aggressiveness that distinguishes the two subtypes. Langerhans cell sarcoma (LCS) is a neoplastic proliferation of Langerhans cells which can occur in skin, bone marrow, lymph nodes, spleen, liver, and lung...
2017: Case Reports in Pathology
https://www.readbyqxmd.com/read/28623120/association-between-ischemic-stroke-and-erdheim-chester-disease-a-case-report-and-review-of-literature
#3
Guillaume Fargeot, Sabrina Stefanizzi, Sebastien Depuydt, Frederic Klapczynski, Alain Ameri
Erdheim-Chester disease (ECD) is a rare form of non-Langerhans cell histiocytosis characterized by a xanthogranulomatous infiltration of tissues by spumous histiocytes. Neurological involvement is frequent, but ischemic strokes have been exceptionally described. We report the case of a 68-year-old woman who presented with an acute ischemic stroke associated with a multisystemic disorder including insipidus diabetes, infiltration of the aorta and the carotid arteries, perirenal infiltration, aortitis, and lytic bone lesions...
June 13, 2017: Journal of Stroke and Cerebrovascular Diseases: the Official Journal of National Stroke Association
https://www.readbyqxmd.com/read/28622183/cyclin-d1-is-expressed-in-neoplastic-cells-of-langerhans-cell-histiocytosis-but-not-reactive-langerhans-cell-proliferations
#4
Vignesh Shanmugam, Jeffrey W Craig, Jason L Hornick, Elizabeth A Morgan, Geraldine S Pinkus, Olga Pozdnyakova
Langerhans cell histiocytosis (LCH) is characterized by frequent activating mutations involving the mitogen-activated protein kinase (MAPK) pathway. Therefore, downstream markers of MAPK pathway activation such as cyclin D1 may be useful as novel diagnostic markers of neoplasia in LCH. The goal of this study was to investigate cyclin D1 expression in LCH and reactive Langerhans cell accumulations using immunohistochemistry on archival tissue. All LCH cases tested (39/39) showed cyclin D1 expression in CD1a/Langerin cells...
June 15, 2017: American Journal of Surgical Pathology
https://www.readbyqxmd.com/read/28617889/erdheim-chester-disease-presenting-with-histiocytic-colitis-and-cytokine-storm
#5
George P Christophi, Yeshika Sharma, Quader Farhan, Umang Jain, Ted Walker, Gregory S Sayuc, Deborah C Rubin
BACKGROUND: Non-Langerhans histiocytosis is a group of inflammatory lymphoproliferative disorders originating from non-clonal expansion of hematopoietic stem cells into cytokine-secreting dendritic cells or macrophages. Erdheim-Chester Disease (ECD) is a rare type of non-Langerhans cell histiocytosis characterized by tissue inflammation and injury caused by macrophage infiltration and histologic findings of foamy histiocytes. Often ECD involves the skeleton, retroperitoneum and the orbits...
June 2017: Journal of Gastrointestinal and Liver Diseases: JGLD
https://www.readbyqxmd.com/read/28616240/metastatic-pancreatic-carcinoma-masquerading-as-cystic-lung-disease-a-rare-presentation
#6
Emily Stern, Taha Huseini, YiJin Kuok, Fiona Lake
This 52-year-old male ex-smoker presented with a six-month history of progressive breathlessness and weight loss. He deteriorated acutely, and was admitted with severe type 1 respiratory failure. Apart from diffuse coarse crackles on chest auscultation, physical examination was unremarkable. High-resolution computed tomography (HRCT) showed diffuse cystic changes throughout the lungs. A diagnosis of pulmonary Langerhans cell histiocytosis (PLCH) was considered. Further workup identified a coincidental pancreatic lesion of uncertain significance, which remained indeterminate on magnetic resonance imaging (MRI) and on positron emission tomography (PET)...
September 2017: Respirology Case Reports
https://www.readbyqxmd.com/read/28614815/a-case-of-pulmonary-langerhans-cell-sarcoma-simultaneously-diagnosed-with-cutaneous-langerhans-cell-histiocytosis-studied-by-whole-exome-sequencing
#7
Si-Wook Kim, Moon Ki Choi, Hye Sook Han, Hyojin Song, Youngil Koh, Seung-Myoung Son, Ok-Jun Lee, Ji Yeoun Lee, Ki Man Lee, Ki Hyeong Lee, Jihyun Kwon
Langerhans cell histiocytosis (LCH) and Langerhans cell sarcoma (LCS) are clonal proliferations of Langerhans-type cells. Unlike in LCH, the pathophysiology and clinical course of LCS are unclear due to its rarity. Here, we report the case of a 73-year-old male patient who was diagnosed with cutaneous LCH and pulmonary LCS at the same time. Pathological review of these 2 tumors revealed similar immunohistochemical findings. However, the tumor cells in LCS had more aggressive cytological features than those in LCH...
June 15, 2017: Acta Haematologica
https://www.readbyqxmd.com/read/28614210/congenital-syphilis-of-bone-a-potential-mimicker-of-childhood-histiocytoses
#8
Youran Zou, Matthew A Marcus, C Guy Castles, Scott E Kilpatrick
Involvement of the skeletal system by congenital syphilis is well documented in the literature, chiefly in the form of radiologic studies, including periostitis, osteitis, and osteochondritis. Because congenital syphilis is generally recognized clinically, tissue biopsy is virtually never performed. Therefore, the histopathologic findings are less well documented and mostly exist in the older literature. We report herein the clinicoradiologic and pathologic features of a 2-month-old infant who initially presented with absence of left arm movement...
June 13, 2017: American Journal of Surgical Pathology
https://www.readbyqxmd.com/read/28612007/erdheim-chester-disease-presenting-with-secondary-hypertension-as-a-result-of-bilateral-proximal-renal-artery-stenosis-a-case-report
#9
Farid Arman, Hania Shakeri, Niloofar Nobakht, Anjay Rastogi, Mohammad Kamgar
Erdheim-Chester disease (ECD) is a rare, non-Langerhans cell histiocytosis presenting most commonly with bone and central nervous system symptoms, including but not limited to bone pain and diabetes insipidus. We present a known case of ECD, which was referred for secondary hypertension workup and diagnosed with severe, proximal, bilateral renal artery stenosis.
May 2017: Case Reports in Nephrology and Dialysis
https://www.readbyqxmd.com/read/28607780/homonymous-superior-quadrantanopia-due-to-erdheim-chester-disease-with-asymptomatic-pituitary-involvement
#10
Roaa Ridha Amer, Sara Mohammed Qubaiban, Eman Abdulkarim Bakhsh
Polyostotic sclerosing histiocytosis, also known as Erdheim-Chester disease (ECD), is a rare form of non-Langerhans histiocytosis. ECD has wide clinical spectrums which mainly affect skeletal, neurological, dermatological, retroperitoneal, cardiac, and pulmonary manifestations. Here we describe a case of ECD in a 45-year-old female who presented initially with bilateral knee pain and homonymous superior quadrantanopia progressed to ophthalmoplegia and complete visual loss of the left eye over a period of one year...
2017: Case Reports in Neurological Medicine
https://www.readbyqxmd.com/read/28606227/-clinical-features-and-prognosis-of-langerhans-cell-histiocytosis-in-children-an-analysis-of-34-cases
#11
Dan Li, Hui Li, Hong Shi
OBJECTIVE: To investigate the clinical features and prognosis of children with Langerhans cell histiocytosis (LCH). METHODS: A retrospective analysis was performed for the clinical data of 34 children with newly diagnosed LCH. RESULTS: The 34 children had a median age of 14.5 months (range: 22 d to 60 months). Of all 34 children, 23 were aged 0-2 years and 11 were aged >2 years. There were 17 children in the high-risk group and 17 in the low-risk group...
June 2017: Zhongguo Dang Dai Er Ke za Zhi, Chinese Journal of Contemporary Pediatrics
https://www.readbyqxmd.com/read/28604476/pet-mr-in-the-assessment-of-pediatric-histiocytoses-a-comparison-to-pet-ct
#12
Andrew C Sher, Robert Orth, Kenneth McClain, Carl Allen, Shireen Hayatghaibi, Victor Seghers
PURPOSE: The aim of this study was to analyze the feasibility and diagnostic performance of F-FDG PET/MR compared with F-FDG PET/CT in a cohort of pediatric histiocytosis patients with regard to image quality, lesion detection, and FDG quantification. METHODS: Children with a diagnosis of Langerhans cell histiocytosis or Rosai-Dorfman disease were prospectively recruited. Seventeen PET/CT and PET/MR examinations were performed on 9 patients (mean age, 6.2 years) following a single-injection dual-imaging protocol...
June 9, 2017: Clinical Nuclear Medicine
https://www.readbyqxmd.com/read/28596665/langerhans-cell-histiocytosis-lch-of-the-tonsil-in-adult-patient-an-uncommon-disease-at-an-uncommon-site
#13
Rachna Khera, Faiq Ahmed, Sudha Murthy, Krishna Mohan Mallavarapu
Langerhans cell histiocytosis (LCH) is a rare disease characterized by clonal neoplastic proliferation of normal antigen presenting cell (APC), the Langerhans cell. Most cases occur in childhood and the disease is rare in adults. LCH can involve solitary organ or can present as a multi-system disease in children. In adults, isolated pulmonary LCH is the commonest presentation. Tonsillar infiltration as a sole manifestation is extremely rare. We herewith report a case with isolated tonsillar involvement by LCH in an adult patient...
June 2017: Indian Journal of Hematology & Blood Transfusion
https://www.readbyqxmd.com/read/28590337/spontaneous-pneumothorax-in-diffuse-cystic-lung-diseases
#14
Joseph Cooley, Yun Chor Gary Lee, Nishant Gupta
PURPOSE OF REVIEW: Diffuse cystic lung diseases (DCLDs) are a heterogeneous group of disorders with varying pathophysiologic mechanisms that are characterized by the presence of air-filled lung cysts. These cysts are prone to rupture, leading to the development of recurrent spontaneous pneumothoraces. In this article, we review the epidemiology, clinical features, and management DCLD-associated spontaneous pneumothorax, with a focus on lymphangioleiomyomatosis, Birt-Hogg-Dubé syndrome, and pulmonary Langerhans cell histiocytosis...
July 2017: Current Opinion in Pulmonary Medicine
https://www.readbyqxmd.com/read/28587427/mri-features-of-growth-hormone-deficiency-in-children-with-short-stature-caused-by-pituitary-lesions
#15
Chao Xu, Xinxian Zhang, Lina Dong, Bin Zhu, Tao Xin
We verified the advantages of using magnetic resonance imaging (MRI) for improving the diagnostic quality of growth hormone deficiency (GHD) in children with short stature caused by pituitary lesions. Clinical data obtained from 577 GHD patients with short stature caused by pituitary lesions were retrospectively analyzed. There were 354 cases (61.3%) with anterior pituitary dysplasia; 45 cases (7.8%) of pituitary stalk interruption syndrome (PSIS); 15 cases (2.6%) of pituitary hyperplasia due to primary hypothyroidism; 38 cases (6...
June 2017: Experimental and Therapeutic Medicine
https://www.readbyqxmd.com/read/28586276/dendritic-cell-trafficking-and-function-in-rare-lung-diseases
#16
Huan Liu, Claudia Jakubzick, Andrew R Osterburg, Rebecca L Nelson, Nishant Gupta, Francis X McCormack, Michael T Borchers
Dendritic cells (DCs) are highly specialized immune cells that capture antigens and then migrate to lymphoid tissue and present antigen to T cells. This critical function of DCs is well defined and recent studies further demonstrate that DCs are also key regulators of several innate immune responses. Studies focused on the roles of DCs in the pathogenesis of common lung diseases such as asthma, infection and cancer have traditionally driven our mechanistic understanding of pulmonary DC biology. The emerging development of novel DC reagents, techniques and genetically modified animal models has provided abundant data revealing distinct populations of DCs in the lung and allow us to examine mechanisms of DC development, migration and function in pulmonary disease with unprecedented detail...
June 6, 2017: American Journal of Respiratory Cell and Molecular Biology
https://www.readbyqxmd.com/read/28583775/-a-lumbar-vertebral-eosinophilic-granuloma-with-spinal-instability-in-an-adolescent
#17
K Bin, S Jacopin, E Poinsot, C Stoven, J-C Ruzic, S Maurin, E Enaud
This is a case report of a rare isolated eosinophilic granuloma of vertebra L3, in a 15-year-old adolescent. Vertebral instability, due to aggravation of the osteolysis, occurred without neurological symptoms despite orthopedic treatment. Surgery was necessary to correct and stabilize the spinal deformation. Postoperative chemotherapy (vinblastine and corticoids) was given as recommended for Langerhans cell histiocytosis (HL2010). No recurrence was observed after 5 years of follow-up.
June 2, 2017: Archives de Pédiatrie: Organe Officiel de la Sociéte Française de Pédiatrie
https://www.readbyqxmd.com/read/28583274/benign-bone-conditions-that-may-be-fdg-avid-and-mimic-malignancy
#18
REVIEW
Thomas C Kwee, John M H de Klerk, Maarten Nix, Ben G F Heggelman, Stefan V Dubois, Hugo J A Adams
Positron emission tomography with the radiotracer (18)F-fluoro-2-deoxy-d-glucose (FDG) plays an important role in the evaluation of bone pathology. However, FDG is not a cancer-specific agent, and knowledge of the differential diagnosis of benign FDG-avid bone alterations that may resemble malignancy is important for correct patient management, including the avoidance of unnecessary additional invasive tests such as bone biopsy. This review summarizes and illustrates the spectrum of benign bone conditions that may be FDG-avid and mimic malignancy, including osteomyelitis, bone lesions due to benign systemic diseases (Brown tumor, Erdheim-Chester disease, Gaucher disease, gout and other types of arthritis, Langerhans cell histiocytosis, and sarcoidosis), benign primary bone lesions (bone cysts, chondroblastoma, chondromyxoid fibroma, desmoplastic fibroma, enchondroma, giant cell tumor and granuloma, hemangioma, nonossifying fibroma, and osteoid osteoma and osteoblastoma), and a group of miscellaneous benign bone conditions (post bone marrow biopsy or harvest status, bone marrow hyperplasia, fibrous dysplasia, fractures, osteonecrosis, Paget disease of bone, particle disease, and Schmorl nodes)...
July 2017: Seminars in Nuclear Medicine
https://www.readbyqxmd.com/read/28583250/oral-juvenile-xanthogranuloma-report-of-two-cases
#19
Mônica Simões Israel, Roman Carlos, Fábio Ramôa Pires
Juvenile xanthogranuloma is a rare non-Langerhans cell histiocytosis that usually occurs in the skin of children. Extracutaneous involvement is rare, and few cases affecting the oral cavity have been reported. The purpose of the present study was to report two cases of oral juvenile xanthogranuloma affecting the lower lip of a 14-year-old girl and the soft palate of a second 14-month-old girl, both present as yellowish papules. The lesions were surgically excised, and histological and immunohistochemical analysis showed a proliferation of non-Langerhans cells histiocytes and foamy cells, fulfilling the morphologic features of juvenile xanthogranuloma...
May 15, 2017: Pediatric Dentistry
https://www.readbyqxmd.com/read/28582647/inflammatory-serum-cytokines-and-chemokines-increase-associated-with-the-disease-extent-in-pediatric-langerhans-cell-histiocytosis
#20
Akira Morimoto, Yukiko Oh, Sachie Nakamura, Yoko Shioda, Tomomi Hayase, Toshihiko Imamura, Kazuko Kudo, Shinsaku Imashuku
OBJECTIVE: Langerhans cell histiocytosis (LCH) is characterized by immature dendritic cell proliferation, infiltration of LCH lesions by various inflammatory cells, and a lesional cytokine storm. It is classified into three groups on the basis of disease extent, namely, multisystem with risk-organ involvement (MS+), multisystem without risk-organ involvement (MS-), and single-system (SS) disease. We comprehensively analyzed whether serum levels of cytokines/chemokines reflect the disease extent...
June 2, 2017: Cytokine
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