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https://www.readbyqxmd.com/read/29157165/successful-cochlear-implantation-in-langerhans-cell-histiocytosis-a-rare-case
#1
Gaurav Gupta, Avani Jain, Mohnish Grover
An 8-year-old girl presented with a 1-year history of bilateral progressive hearing loss and vertigo for 6 months. High-resolution computed tomography of the temporal bones demonstrated multiple lytic lesions. Histopathology examination confirmed a diagnosis of Langerhans cell histiocytosis (LCH). She underwent chemotherapy for 12 months. Following treatment, she was in remission. However, the bilateral profound sensorineural hearing loss persisted. She underwent right cochlear implantation with very good functional audiological outcomes...
November 20, 2017: Cochlear Implants International
https://www.readbyqxmd.com/read/29125707/vascularized-humeral-periosteal-flap-to-treat-lateral-humeral-condyle-nonunion-an-anatomical-study-and-report-of-two-successfully-treated-pediatric-cases
#2
S Sergi Barrera-Ochoa, Francisco Soldado, Alfonso Rodriguez-Baeza, Paula Diaz-Gallardo, Felipe Hodgson, Pablo S Corona, Jorge Knörr
PURPOSE: Nonunion is a common complication of lateral condyle humeral (LCH) fractures in children. In situ fixation with a screw and bone grafting is the classically-recommended method of treatment. The purpose of this study is to analyze the feasibility of obtaining a vascularized periosteal flap obtained from the lateral humerus and based on the posterior collateral radial vessels (PCRV). Second, to report the results after the application in two pediatric cases. METHODS: Periosteal branches of PCRV were studied in ten upper limbs from fresh human cadavers...
November 10, 2017: Microsurgery
https://www.readbyqxmd.com/read/29118704/spontaneous-remission-of-severe-systemic-langerhans-cell-histiocytosis-with-bladder-involvement-a-case-study
#3
Isotta M Magaton, Alexandar Tzankov, Fatime Krasniqi, Christof Rottenburger, Rosanna Zanetti-Daellenbach, Peter Grendelmeier, Viola Heinzelmann-Schwarz, Michael Mayr, Fabienne D Schwab
Background: The clinical presentation of Langerhans cell histiocytosis (LCH) is heterogeneous ranging from single-organ involvement to systemic disease causing substantial morbidity and mortality. We describe an unusual course of severe multisystem LCH with spontaneous remission. Case Presentation: We report on a 45-year-old Caucasian woman with cervical cancer, FIGO stage IVB. Five months after the end of combined radiochemotherapy and brachytherapy, the patient was readmitted because of severe dysuria...
September 2017: Case Reports in Oncology
https://www.readbyqxmd.com/read/29107666/jak2-mutated-langerhans-cell-histiocytosis-associated-with-primary-myelofibrosis-treated-with-ruxolitinib
#4
Arturo Bonometti, Filippo Bagnoli, Daniele Fanoni, Luigia Venegoni, Laura Corti, Paola Bianchi, Elena Maria Elli, Giuseppe Isimbaldi, Vincenzo L'Imperio, Gianluca Nazzaro, Emanuela Passoni, Emilio Berti
The pathogenesis and cellular origin of Langerhans cell histiocytosis (LCH) are debated. Recently, mutations on MAPK and PI3K pathways have been linked to disrupted cell proliferation in LCH. Janus Kinase 2 (JAK2) mutations play the same role in Philadelphia-negative chronic myeloproliferative neoplasms. We describe the case of a patient affected by JAK2-positive Primary Myelofibrosis (PMF) who developed a clonally related LCH while in treatment with Ruxolitinib. JAK-inhibitors are well known to affect function and differentiation of different hematological lineages, including mononuclear phagocytes precursors...
October 28, 2017: Human Pathology
https://www.readbyqxmd.com/read/29107340/langerhans-cell-histiocytosis-a-neoplastic-disorder-driven-by-ras-erk-pathway-mutations
#5
REVIEW
Gary Tran, Thy N Huynh, Amy S Paller
Langerhans cell histiocytosis (LCH) is a disorder of myeloid neoplasia of dendritic cells that affects 1 in 200,000 children <15 years of age and even fewer adults. LCH presents with a spectrum of clinical manifestations. High-risk stratification is reserved for infiltration of blood, spleen, liver, and lungs. After decades of debate on the disease pathogenesis, a neoplastic mechanism is now favored on the basis of LCH cell clonality, rare cases of familial clustering, and recent evidence of mutations involving the Ras/Raf/MEK (mitogen-activated protein kinase kinase)/ERK (extracellular signal-regulated kinase) pathway in lesional biopsy specimens...
October 26, 2017: Journal of the American Academy of Dermatology
https://www.readbyqxmd.com/read/29096034/cns-erdheim-chester-disease-a-challenge-to-diagnose
#6
Zenggang Pan, Bette K Kleinschmidt-DeMasters
Erdheim-Chester disease (ECD) is a rare nonLangerhans cell histiocytosis. Although approximately 50% of cases eventually involve the central nervous system (CNS), the CNS has seldom been reported as the initial biopsy site. The diagnosis of CNS ECD can be challenging due to morphologic overlap with reactive histiocytic proliferation, Langerhans cell histiocytosis (LCH), and extranodal Rosai-Dorfman disease (RDD). We present 3 cases from our files that illustrate the protean manifestations of ECD. Case 1 was a 47-year-old man with ataxia, dysarthria, and intermittent ophthalmoplegia whose cerebellar biopsy had shown only profuse, nonspecific Rosenthal fiber-rich piloid gliosis; ECD was diagnosed only at autopsy...
December 1, 2017: Journal of Neuropathology and Experimental Neurology
https://www.readbyqxmd.com/read/29084059/insl3-expression-in-leydig-cell-hyperplasia-and-leydig-cell-tumors
#7
Nelli S Lakis, Kara A Lombardo, Shamlal Mangray, George J Netto, Daniela Salles, Andres Matoso
Insulin-like 3 (INSL3) is a hormone produced by Leydig cells (LCs) and leads to physiological testicular descent during embryonic development. We investigated the expression of INSL3 by immunohistochemistry in normal LCs, in Leydig cell tumor (LCT) (n=17 including 15 testes and 2 ovaries) and in Leydig cell hyperplasia (LCH) (n=10). Normally distributed LCs showed strong immunostaining in the cytoplasm in all cases. All 10 cases (100%) of LCH were strongly and diffusely positive in the intertubular areas. Six cases of LCH had nodules raging in size from 0...
October 27, 2017: Applied Immunohistochemistry & Molecular Morphology: AIMM
https://www.readbyqxmd.com/read/29074592/finding-active-lch-cells-in-the-blood
#8
Oussama Abla
No abstract text is available yet for this article.
October 26, 2017: Blood
https://www.readbyqxmd.com/read/29044633/extensive-nail-changes-in-a-toddler-with-multisystemic-langerhans-cell-histiocytosis
#9
Vimal Kumar, Dhanalakshmi Angappan, Julius Scott, Deenadayalan Munirathnam, Mukul Vij, Naresh Shanmugam
Langerhans cell histiocytosis (LCH) is a multisystem disorder involving various organs. Nail changes in LCH are extremely rare. We present this case report of extensive nail changes in an 18-month-old child with multisystem LCH.
October 17, 2017: Pediatric Dermatology
https://www.readbyqxmd.com/read/29043209/perianal-langerhans-cell-histiocytosis-a-rare-presentation-in-an-adult-male
#10
Asmaa Gaber Abdou, Doha MaherTaie
Langerhans cell histiocytosis (LCH) is a rare disease characterized by a proliferation of cells that show immunophenotypic and ultrastructural similarities with antigen-presenting Langerhans cells of mucosal sites and skin. LCH in adults is rare, and there are still many undiagnosed/misdiagnosed patients. We describe LCH involvement of the perianal region of a 33-year-old male with a previous history of diabetes insipidus. The differential diagnosis and all the reported cases of LCH of the perianal skin involvement since its description in 1984 till 2016 are discussed...
July 2017: Autopsy & case reports
https://www.readbyqxmd.com/read/29021671/atlantoaxial-langerhans-cell-histiocytosis-radiographic-characteristics-and-corresponding-prognosis-analysis
#11
Lihua Zhang, Liang Jiang, Huishu Yuan, Zhongjun Liu, Xiaoguang Liu
BACKGROUND: Langerhans cell histiocytosis (LCH) may affect atlas and axis, and there were very few published cases describing a characteristic of LCH of atlantoaxial. OBJECTIVE: The objective of the study is to investigate the image manifestations of atlantoaxial LCH to improve the in-depth comprehension on it. MATERIALS AND METHODS: A retrospective study was done of computed tomography (CT) and magnetic resonance imaging in atlas and axis and prognosis was analyzed...
July 2017: Journal of Craniovertebral Junction and Spine
https://www.readbyqxmd.com/read/29017309/langerhans-cell-histiocytosis-of-the-thoracic-spine-in-an-adult
#12
Myeong Cheol Kim, Sun Hee Sung, Yongjae Cho
We report a case of a 45-year-old man with a complaint of both leg weakness and hypoesthesia. Radiological evaluation revealed an osteolytic lesion of the ninth thoracic vertebra. The patient underwent posterior corpectomy with total excision of the tumor, mesh cage insertion with posterior screw fixation and subsequent radiotherapy. Histology confirmed the diagnosis of Langerhans cell histiocytosis (LCH). This case report presents the diagnostic work-up, histopathological evaluation, and the treatment procedures of rare LCH in the thoracic spine...
September 2017: Korean Journal of Spine
https://www.readbyqxmd.com/read/28993226/vulvar-lesions-in-an-8-year-old-girl-cutaneous-manifestations-of-multisystem-langerhans-cell-histiocytosis
#13
Elisa M Jorgensen, Peter P Chen, Sarah Rutter, Julia A Cron
BACKGROUND: Langerhans cell histiocytosis (LCH) is a rare localized or systemic disease characterized by proliferation of myeloid-derived dendritic cells. Vulvar lesions may be the herald symptom of LCH and may mimic other cutaneous lesions. Prognosis varies widely based on the extent and spread of disease. CASE: An 8-year-old girl with a 4-month history of vulvar lesions resistant to topical steroids was referred by her pediatrician. Vulvar biopsy was diagnostic for LCH...
October 6, 2017: Journal of Pediatric and Adolescent Gynecology
https://www.readbyqxmd.com/read/28969735/langerhans-cell-histiocytosis-involving-both-jaws-in-an-adult
#14
Hira Salam, Ruqaiya Shahid, Talat Mirza
Langerhans cell histiocytosis (LCH) is the latest terminology for a disorder of reticulo-endothelial system, previously known as histiocytosis X, and marked by aberrant proliferation of bone marrow derived Langerhans cells with variable inflammatory infiltrate including neutrophils, lymphocytes, plasma cells, eosinophils, and multinucleated giant cells. Although rare, the disorder frequently inflicts children with peak incidence recorded in 2-4 years age group. LCH is rare in adults. A22-year adult male presented with the complaint of teeth mobility...
September 2017: Journal of the College of Physicians and Surgeons—Pakistan: JCPSP
https://www.readbyqxmd.com/read/28954111/congenital-self-healing-reticulohistiocytosis-with-spontaneous-regression
#15
Leela Rani Parimi, Jiabao You, Liu Hong, Furen Zhang
Congenital self-healing reticulohistiocytosis is a rare, benign, self-limiting variant of Langerhans cell histiocytosis (LCH). LCH encompasses a group of idiopathic disorders characterized by the clonal proliferation of Langerhans cells. Congenital self-healing reticulohistiocytosis typically appears at birth or in the neonatal period as isolated cutaneous lesions, often appearing as multiple crusted papules with no systemic findings. Although clinical features seem aggressive, the lesions tend to involute spontaneously within weeks to a few months leaving residual hypo or hyperpigmented macules...
July 2017: Anais Brasileiros de Dermatologia
https://www.readbyqxmd.com/read/28950648/unraveling-intermittent-features-in-single-particle-trajectories-by-a-local-convex-hull-method
#16
Yann Lanoiselée, Denis S Grebenkov
We propose a model-free method to detect change points between distinct phases in a single random trajectory of an intermittent stochastic process. The local convex hull (LCH) is constructed for each trajectory point, while its geometric properties (e.g., the diameter or the volume) are used as discriminators between phases. The efficiency of the LCH method is validated for six models of intermittent motion, including Brownian motion with different diffusivities or drifts, fractional Brownian motion with different Hurst exponents, and surface-mediated diffusion...
August 2017: Physical Review. E
https://www.readbyqxmd.com/read/28944988/evaluation-and-treatment-of-langerhans-cell-histiocytosis-patients-with-central-nervous-system-abnormalities-current-views-and-new-vistas
#17
REVIEW
E Ann Yeh, Jay Greenberg, Oussama Abla, Giulia Longoni, Eli Diamond, Michelle Hermiston, Brandon Tran, Carlos Rodriguez-Galindo, Carl E Allen, Kenneth L McClain
Central nervous system (CNS) involvement in Langerhans cell histiocytosis (LCH) can include mass lesions of the hypothalamic pituitary axis, choroid plexus, cerebrum, and cerebellum or magnetic resonance imaging (MRI) signal abnormalities of the cerebellum, pons, and basal ganglia. The term neurodegenerative (ND) CNS-LCH has been given to the MRI signal abnormalities and neurologic dysfunction, although initially patients may have no clinical symptoms. Standardized evaluations to better understand the natural history and response to therapy are needed...
September 25, 2017: Pediatric Blood & Cancer
https://www.readbyqxmd.com/read/28937439/langerhans-cell-histiocytosis-with-clinical-and-histologic-features-of-hidradenitis-suppurativa-brief-report-and-review
#18
Jessica E Kalen, Divya Shokeen, Mona Mislankar, Michael Wangia, Kiran Motaparthi
Langerhans cell histiocytosis (LCH) is an uncommon histiocytic disorder in adults. Clinically, this rare entity can mimic other dermatologic conditions, including hidradenitis suppurativa. A case of LCH is reported with clinical and histologic features of hidradenitis suppurativa, along with a review of these unusual findings. Clinical dermatologists and dermatopathologists benefit from awareness of this unique presentation, which may prompt earlier identification and diagnosis of adult patients with LCH.
September 11, 2017: American Journal of Dermatopathology
https://www.readbyqxmd.com/read/28935696/a-genome-wide-association-study-of-lch-identifies-a-variant-in-smad6-associated-with-susceptibility
#19
LETTER
Erin C Peckham-Gregory, Rikhia Chakraborty, Michael E Scheurer, John W Belmont, Harshal Abhyankar, Amel G Sengal, Brooks P Scull, Olive Eckstein, Daniel J Zinn, Louisa Mayer, Albert Shih, Miriam Merad, D Williams Parsons, Kenneth L McClain, Philip J Lupo, Carl E Allen
No abstract text is available yet for this article.
November 16, 2017: Blood
https://www.readbyqxmd.com/read/28932806/langerhans-cell-histiocytosis-limited-to-the-female-genital-tract-a-review-of-literature-with-three-additional-cases
#20
Rebekah Wieland, Jenna Flanagan, Elise Everett, Sharon Mount
•LCH of the female reproductive tract has four patterns of involvement.•A comprehensive literature review revealed 35 cases of pure genital LCH.•We report two new cases of pure LCH lesions of the vulva and one of the cervix.•Treatment of LCH varies and there is no standard for pure genital involvement.•Prognosis of LCH confined to the gynecologic tract appears to be favorable.
November 2017: Gynecologic Oncology Reports
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