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Histiocytosis

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https://www.readbyqxmd.com/read/27908938/a-rare-case-of-extensive-cranial-langerhans-cell-histiocytosis-synchronously-presenting-as-otitis-externa-and-giant-cell-arteritis
#1
Adnan Darr, Zahir Mughal, Thomas Martin
Langerhans cell histiocytosis (LCH) is a rare histiocytic disorder of unpredictable clinical course and varied modes of presentation. The spectrum of presentation is wide, ranging from isolated eosinophilic granulomas to multiple lesions and diffuse systemic involvement. We present the case of a 52-year-old man, who presented with an 8-week history of worsening otalgia and superficial temporal tenderness attributed to otitis externa within the community and subsequently giant cell arteritis. Computed tomography and magnetic resonance imaging were undertaken due to atypical features, which demonstrated bony destruction within the right greater wing of the sphenoid, squamous part of temporal and mastoid bone, with middle cranial fossa communication...
December 1, 2016: Journal of Surgical Case Reports
https://www.readbyqxmd.com/read/27898473/histopathological-and-clinical-findings-in-cutaneous-manifestation-of-erdheim-chester-disease-and-langerhans-cell-histiocytosis-overlap-syndrome-associated-with-the-brafv600e-mutation
#2
Julia Liersch, J Andrew Carlson, Jörg Schaller
The overlap of Erdheim-Chester disease (ECD) and Langerhans cell histiocytosis (LCH) is more common than it was generally accepted. Both diseases seem to be linked by a mutation in oncogenic BRAFV600E, probably an early event which occurs in bone marrow progenitor cells. In this article are described the clinical and histological findings in 2 cases of ECD-LCH overlap syndrome bearing the BRAFV600E mutation in both ECD and LCH lesions in bone and skin. In one case, lesions of ECD and LCH were situated directly site-to-site in the same bone section leading to the assumption of a common myeloid precursor cell for these diseases...
November 23, 2016: American Journal of Dermatopathology
https://www.readbyqxmd.com/read/27894453/usual-and-unusual-manifestations-of-familial-hemophagocytic-lymphohistiocytosis-and-langerhans-cell-histiocytosis
#3
REVIEW
Craig Erker, Paul Harker-Murray, Julie-An Talano
Familial hemophagocytic lymphohistiocytosis (FHL) and Langerhans cell histiocytosis (LCH) are histiocytic diseases that occur most commonly in young children. Improvements in recognition and treatment have been substantial for both diseases in the past decade, although early and late morbidity continue to be major concerns. These two diagnoses behave differently, although the clinical spectra for both diseases are diverse and can lead to confusion and delays in diagnosis and treatment. This article focuses on the clinical and genetic spectrum of FHL as well as the clinical and treatment variations of LCH...
February 2017: Pediatric Clinics of North America
https://www.readbyqxmd.com/read/27891484/-histiocytosis-x-a-rare-case-report
#4
Emmanuel Dhiravia Sargunam Azariah, Deepak Chandrasekaran, Ravindran Chinnaswami, Sivaramakrishnan Balasubramaniam, Eswari Jagdish
Histiocytosis X is an idiopathic disease, characterized by a disorder of the reticulo-endothelial system in the human body. Histopathological studies carried out right from the 1800s have seen a significant similarity in the pathologic process of different stages in particular clinical syndromes showing proliferation of mature histiocytes. It was then modified by Lichenstein in 1953 as "Histiocytosis X". The exact aetiology is unknown; hence, the name "Histiocytosis X". The disease classically presents with three syndromes namely Eosinophilic granuloma, Letterer-Siwe disease and Hand-Schuller-Christian disease...
October 2016: Journal of Clinical and Diagnostic Research: JCDR
https://www.readbyqxmd.com/read/27889325/-langerhans-cell-histiocytosis-and-erdheim-chester-disease-a-continuity
#5
S Parreau, J Haroche, I Pommepuy, J F Emile, J C Bourras, F Archambeaud
INTRODUCTION: Erdheim-Chester disease and langerhans cell histiocytosis are two rare diseases separate on clinical, radiological and histological aspects. However, several cases involving both entities have been described. OBSERVATION: A 70-year-old man had a central diabetes insipidus, xanthelasmas, retroperitoneal fibrosis and osteosclerosis of the legs suggestive of Erdheim-Chester disease. Bone biopsy showed langerhans cell histiocytosis CD1a positive with the presence of BRAF V600E mutation...
November 23, 2016: La Revue de Médecine Interne
https://www.readbyqxmd.com/read/27886798/multicentric-reticulohistiocytosis-rheumatology-perspective
#6
REVIEW
Bahtiyar Toz, Nesimi Büyükbabani, Murat İnanç
Multicentric reticulohistiocytosis (MRH) is a rare, multisystemic non-Langerhans cell histiocytosis characterized by skin and articular involvement, and rarely involves various other organs. There are no specific laboratory findings for MRH. Diagnosis is based on clinical findings and skin or synovial biopsy results. There is currently no consensus for the treatment of MRH. Here, we review the differential diagnosis and treatment options of MRH from the rheumatologist's perspective. We also report an index case of MRH associated with Sjögren's syndrome and pulmonary embolism...
April 2016: Best Practice & Research. Clinical Rheumatology
https://www.readbyqxmd.com/read/27867869/thyroid-langerhans-cell-histiocytosis-and-papillary-thyroid-carcinoma
#7
Rajab AlZahrani, Mohammed Algarni, Hadi Alhakami, Haia AlSubayea, Naif Alfattani, Mohammet Guler, Mohamed Satti
A 27-year-old female, married with two children, presented to our clinic with a 1-year history of thyroid swelling and pressure symptoms on lying backward and bilateral cervical lymphadenopathy. The patient was a known case of panhypopituitarism for 5 years. Comprehensive patient evaluation including FNAC with papillary thyroid cancer result then she underwent total thyroidectomy and bilateral neck dissection and final histologic examination confirmed papillary thyroid carcinoma in the background of lymphocytic thyroiditis, associated with Langerhans cell histiocytosis (LCH)...
October 2016: Gland Surgery
https://www.readbyqxmd.com/read/27853924/retrospective-analysis-of-nonendodontic-periapical-lesions-misdiagnosed-as-endodontic-apical-periodontitis-lesions-in-a-population-of-taiwanese-patients
#8
Hsun-Yu Huang, Yuk-Kwan Chen, Edward Cheng-Chuan Ko, Fu-Hsiung Chuang, Ping-Ho Chen, Ching-Yi Chen, Wen-Chen Wang
OBJECTIVE: We aimed to evaluate nonendodontic periapical lesions clinically misdiagnosed as endodontic periapical pathoses in a population of Taiwanese patients. MATERIALS AND METHODS: Cases (2000-2014) of histopathological diagnoses of nonendodontic periapical lesions were retrieved from all cases with a clinical diagnosis of radicular cyst, apical granuloma, or apical periodontitis in the institution. These cases were regarded as misdiagnosed nonendodontic periapical lesions, of which the types and frequencies, in addition to the demographic data, were determined...
November 16, 2016: Clinical Oral Investigations
https://www.readbyqxmd.com/read/27847666/lipid-histiocytosis-of-the-gallbladder-neck-lymph-node
#9
Adriana Handra-Luca, Mohamed Habib Ben Romdhane, Beate Katharina Straub
Lipid histiocytosis of the gallbladder neck lymph node is rarely reported nowadays. Two obese patients presented with gallbladder lithiasis detected on CT scan. The treatment consisted in coelioscopic cholecystectomy. Microscopy revealed subacute/chronic lithiasic cholecystitis and foci of vacuolated cells in the gallbladder neck lymph node. These cells were positive for CD68, CD31, S100 protein, and adipophilin and negative for cytokeratin and Alcian blue. In conclusion, we report lymph node lipid histiocytosis diagnosed microscopically after cholecystectomy...
2016: Case Reports in Pathology
https://www.readbyqxmd.com/read/27846659/the-non-langerhans-cell-histiocytoses-rare-histiocytoses-clinical-aspects-and-therapeutic-approaches
#10
C F Classen, M Minkov, T Lehrnbecher
Rare histiocytoses, also called non-Langerhans cell histiocytoses, include all proliferative disorders of histiocytes, macrophages and dendritic cells that are not classified as Langerhans cell histiocytosis (LCH) and do not belong to the hemophagocytic lymphohistiocytosis (HLH) group of diseases. Thus, the term includes numerous benign or malignant, localized or systemic, adult or pediatric diseases. The classification of the histiocytic disorders has been revised several times. Here, we follow the classification recently published by Jean Francois Emile and an international expert panel, defining subgroups of histiocytoses described as L-Group, C-Group, M-Group, R-Group, and H-Group, which stands for LCH-like, cutaneous or mucocutaneous, malignant, Rosai-Dorfman-Disease like and HLH like...
November 2016: Klinische Pädiatrie
https://www.readbyqxmd.com/read/27845884/central-diabetes-insipidus-clinical-profile-that-suggests-organicity-in-peruvian-children-lima-peru-2001-2013
#11
Miguel Angel De Los Santos, Carlos Manuel Del Águila, Maria Isabel Rojas, Juan Manuel Falen, Oswaldo Nuñez, Eliana Manuela Chávez, Oscar Antonio Espinoza, Paola Marianella Pinto, Martha Rosario Calagua
BACKGROUND: Central diabetes insipidus (CDI) is a heterogeneous disease caused by arginine vasopressin deficiency; its management implies a profound understanding of the pathophysiology and the clinical spectrum. The aim of the study was to describe the clinical characteristics that indicate organicity in children and adolescents with central diabetes insipidus treated at the Department of Endocrinology from The Child Health's Institute during 2001 to 2013. METHODS: Cross-sectional, retrospective study...
December 1, 2016: Journal of Pediatric Endocrinology & Metabolism: JPEM
https://www.readbyqxmd.com/read/27833773/a-fatal-case-of-congenital-langerhans-cell-histiocytosis-with-disseminated-cutaneous-lesions-in-a-premature-neonate
#12
Michio Inoue, Yoko Tomita, Tsuyoshi Egawa, Tomoaki Ioroi, Masaaki Kugo, Shinsaku Imashuku
Background. The outcome of neonates with congenital cutaneous Langerhans cell histiocytosis (LCH) is variable. Observations. We report a case of LCH in a female premature neonate born at 33-week gestation. She had disseminated cutaneous lesions, which consisted of hemorrhagic papules and vesicles, with sparse healthy skin areas, and the hands and feet were contracted with scarring and blackened. She was in respiratory failure although no apparent pulmonary or bone lesions on X-rays were noted. Skin biopsy confirmed a diagnosis of LCH due to observation of CD1a(+) Langerhans cells, which lacked expression of E-cadherin and CD56...
2016: Case Reports in Pediatrics
https://www.readbyqxmd.com/read/27824326/99mtc-sestamibi-avid-soft-tissue-lesions-in-rosai-dorfman-disease
#13
Philip Kin-Wai Wong, Daniel J Lee, Saul Harari, Bruce J Barron
Langerhan's histiocytosis is a not uncommon disease, but a related disorder, Rosai-Dorfman (RDD) with extensive or exclusive extranodal distribution, is relatively rare. Ga and PET/CT imaging have typically been used for diagnosis and tracking response to treatment. The authors present a very unusual case wherein lesions of RDD actively accumulated Tc-sestamibi, which was injected as part of a scan to localize a parathyroid adenoma.
December 2016: Clinical Nuclear Medicine
https://www.readbyqxmd.com/read/27823979/high-frequency-of-clonal-ig-and-t-cell-receptor-gene-rearrangements-in-histiocytic-and-dendritic-cell-neoplasms
#14
Wenting Huang, Tian Qiu, Linshu Zeng, Bo Zheng, Jianming Ying, Xiaoli Feng
The 2008 World Health Organization (WHO) diagnostic criteria of histiocytic and dendritic cell neoplasms from hematopoietic and lymphoid tissues no longer required the absence of clonal B-cell/T-cell receptor gene rearrangements. It is true that the clonal B-cell/T-cell receptor gene rearrangements have been identified in rare cases of histiocytic and dendritic cell neoplasms, such as those with or following lymphoma/leukemia or in some sporadic histiocytic/dendritic cell sarcomas, but the clonal features of such group of tumor are still not clear...
November 3, 2016: Oncotarget
https://www.readbyqxmd.com/read/27816731/the-ccl2-ccr2-axis-in-lymph-node-metastasis-from-oral-squamous-cell-carcinoma-an-immunohistochemical-study
#15
Shuichi Fujita, Tohru Ikeda
PURPOSE: Cytokine or chemokine networks involve lymphatic and distant metastasis of various malignancies, including oral squamous cell carcinoma (OSCC). Immunohistochemical analysis was used to investigate the contribution of the axis of the CC chemokine receptor-2 (CCR2) and the CC chemokine receptor-2 ligand (CCL2) to lymphatic metastasis, particularly the relation between primary OSCC and marginal sinus histiocytosis in regional lymph nodes. MATERIALS AND METHODS: Thirteen metastasis-free cases, 15 metastatic cases at resection of primary tumor resection, and 13 postresection metastasis cases were examined...
October 8, 2016: Journal of Oral and Maxillofacial Surgery
https://www.readbyqxmd.com/read/27816346/-neuro-langerhans-cell-histiocytosis
#16
Loïc Le Guennec, Nadine Martin-Duverneuil, Karima Mokhtari, Maria Santiago-Ribeiro, Eléonore Bayen, Antoine Del Cul, Daniel Delgadillo, Aurélie Kas, Carine Courtillot, Julien Haroche, Fleur Cohen, Jean Donadieu, Khê Hoang-Xuan, Ahmed Idbaih
Langerhans cell histiocytosis (LCH) is a rare multisystemic disease. LCH is characterized by proliferation of myeloid progenitors with altered differentiation program and similar phenotypic features to epidermal dendritic cells termed Langerhans cell. LCH cells express CD1a+ and langerin and exhibit BRAF V600E mutation in ∼50% of cases. Neurological involvement or neuro-LCH is observed in 5 to 10% of cases. Three subtypes of neuro-LCH are individualized. The tumor type, accounting for 45% of neuro-LCH, affect mainly young adults...
November 2, 2016: La Presse Médicale
https://www.readbyqxmd.com/read/27816342/-adult-pulmonary-langerhans-cell-histiocytosis
#17
Gwenaël Lorillon, Véronique Meignin, Abdellatif Tazi
There is a strong relationship between tobacco smoking and pulmonary Langerhans cell histiocytosis (LCH) in adults. Definitive diagnosis relies on lung histology. In certain cases, the diagnosis can be retained on a typical nodulo-cystic pattern on lung high-resolution computed tomography (HRCT) and appropriate clinical setting. The main differential diagnoses to consider vary according to nodulo-cystic or only cystic pattern on lung HRCT and the clinical context. The natural history of the disease is better known and regular evaluation of respiratory function is essential during the follow-up of the patients...
November 2, 2016: La Presse Médicale
https://www.readbyqxmd.com/read/27816338/-adult-langerhans-cell-histiocytosis
#18
Mathilde de Menthon, Véronique Meignin, Alfred Mahr, Abdellatif Tazi
Langerhans cell histiocytosis (LCH) is a rare disease affecting both genders and can occur at any age. It often evolves through successive flares, and its severity varies from benign forms that don't require treatment to life threatening disease. Some patients have important functional impairment with psychological and social consequences and prolonged disability. LCH may affect only one organ, with uni- or multifocal involvement or be multisystem disease involving multiple organs. The organs most frequently involved are bones, lung, skin and the endocrinal system...
November 2, 2016: La Presse Médicale
https://www.readbyqxmd.com/read/27816337/-histiocytosis-better-understanding-for-better-care
#19
EDITORIAL
Jean Donadieu, Abdellatif Tazi
No abstract text is available yet for this article.
November 2, 2016: La Presse Médicale
https://www.readbyqxmd.com/read/27804235/a-phase-iia-study-of-afuresertib-an-oral-pan-akt-inhibitor-in-patients-with-langerhans-cell-histiocytosis
#20
Robert J Arceci, Carl E Allen, Ira J Dunkel, Eric Jacobsen, James Whitlock, Robert Vassallo, Shannon R Morris, Alison Portnoy, Beth Ann Reedy, Deborah A Smith, Robert Noble, Amy Murnane, Mark Cornfeld, Carlos Rodriguez-Galindo, Mark L Heaney, Kenneth McClain, Sarah Vaiselbuh
BACKGROUND: Langerhans cell histiocytosis (LCH) is a clonal neoplasm characterized by widely varied clinical presentations, including multisystem involvement and systemic inflammatory symptoms. The AKT pathway is relevant to survival and proliferation of dendritic cells, and is also often upregulated in hematopoietic malignancies. A clinical response in an adult patient with LCH participating in the first-in-human trial of afuresertib prompted this prospective trial. PROCEDURE: The population in the current study included treatment-naïve (n = 7) and recurrent/refractory patients with LCH (n = 10), who received oral afuresertib (125 mg)...
November 2, 2016: Pediatric Blood & Cancer
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