Rapidly Progressing Fatal Adult Multi-Organ Langerhans Cell Histiocytosis Complicated with Fatty Liver Disease

Yuasa, Mitsuhiro; Fujiwara, Shinichirou; Oh, I.Y.; Yamaguchi, Takehiko; Fukushima, Noriyoshi; Morimoto, Akira; Ozawa, Keiya

doi:10.3960/jslrt.52.121

Cited by 9 publications

(9 citation statements)

References 23 publications

(28 reference statements)

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“…Langerhans cell histiocytosis is characterized by cells similar to bone marrow-derived Langerhans cells infiltrated in various tissues and organs [38]. Nonalcoholic fatty liver disease originates from hyperlipidemia, leading to liver failure and Langerhans cell histiocytosis infiltration [39]. In the present study, hepatic Langerhans cell histiocytosis was also observed in liver sections of untreated HFD-fed rats.…”

Section: Hfd-fed Group Treated With Chitosan (Hfd+cos)supporting

confidence: 61%

Evaluating the Anti-Obesity Effects of Chitosan in High-Fat Diet-Induced Obesity in Rat Model

Ghoneim,

Abdou,

Elsehly

et al. 2024

Journal of Medical and Life Science

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Background: Chitosan oligosaccharide (COS) exhibits hypolipidemic and anti-obesity properties. Nevertheless, it remains uncertain whether its efficiency exhibits sex-specific variations. Therefore, this study seeks to fill this gap, systematically evaluating the impact of chitosan oligosaccharides across genders under obesogenic conditions. Material and methods: 36 rats: 18 male, 18 female; split into 6 groups, each with equal male/female. G1 had a standard diet; G2 had a high-fat diet for 4 weeks, then standard for another 4 weeks; G3 had a high-fat diet for 4 weeks, then 500 mg/kg chitosan + standard diet for another 4 weeks. At the study's end, rats were euthanized, blood was collected for biochemical analysis, and liver tissues were excised for histological assessment. Results: Male rats on a high-fat diet displayed significantly elevated body weight, triglycerides, cholesterol, lactate dehydrogenase, ALT, AST, and total protein levels, coupled with noticeable liver damage compared to female rats. However, when treated with COS, male and female rats exhibited reduced weight, lowered triglycerides, cholesterol, lactate dehydrogenase, ALT, and AST levels, and improved liver function and structure. Interestingly, these therapeutic effects were more evident in female and male rats. Conclusion:The study found that COS administration to rats, especially females, reduced weight, and improved liver health, suggesting its potential gender-specific effects on a high-fat diet.

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Section: Hfd-fed Group Treated With Chitosan (Hfd+cos)supporting

confidence: 61%

Evaluating the Anti-Obesity Effects of Chitosan in High-Fat Diet-Induced Obesity in Rat Model

Ghoneim,

Abdou,

Elsehly

et al. 2024

Journal of Medical and Life Science

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“…85 It should be noted that although most generally have indolent and chronic clinical courses, some adult LCH patients exhibit rapid progression and fatal courses. 86 However, the optimal systemic therapy for adult LCH has not yet been developed. [87][88][89][90] Multifocal adult LCH is an orphan disease.…”

Section: Adult Lchmentioning

confidence: 99%

“…Systemic chemotherapy is required for multifocal SS or MS disease also in adults . It should be noted that although most generally have indolent and chronic clinical courses, some adult LCH patients exhibit rapid progression and fatal courses . However, the optimal systemic therapy for adult LCH has not yet been developed .…”

Section: Treatment and Outcomementioning

confidence: 99%

Recent advances in Langerhans cell histiocytosis

Morimoto

Shibata

et al. 2014

Pediatrics International

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165

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The purpose of this review is to provide an updated overview of the pathogenesis and treatment of Langerhans cell histiocytosis (LCH). The pathogenesis of LCH remains obscure and the optimal treatment for LCH has not been established, although incremental progress has been made. Proinflammatory cytokines and chemokines are known to play a role in LCH, which suggests that LCH is an immune disorder. However, the oncogenic BRAF mutation is also detected in more than half of LCH patients, which suggests that LCH is a neoplastic disorder. Remaining major issues in the treatment of LCH are how to rescue patients who have risk-organ involvement but do not respond to first-line therapy, the optimal treatment for the orphan disease of multifocal adult LCH, and how to reduce and treat central nervous system-related consequences, such as central diabetes insipidus and neurodegeneration. More research is needed to better understand the pathogenesis of this disease and to resolve the treatment issues.

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“…LettererSiwe disease is seen in infants (usually within the first year of life), but occurrence in adult has been reported. 24,25 These patients present with fever, weight loss, pancytopenia, lymphadenopathy, hepatosplenomegaly, cuta neous lesions, and bone lesions. HandSchüllerChristian disease is seen in younger children who present with osteo lytic skull lesions, diabetes insipidus (due to hypopituita rism), and exophthalmos.…”

Section: Clinical Featuresmentioning

confidence: 99%

Langerhans Cell Histiocytosis and Langerhans Cell Sarcoma: Current Understanding and Differential Diagnosis

Nakamine

Yamakawa

Yoshino

et al. 2016

JCEH

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Langerhans cell (LC) histiocytosis (LCH) and LC sarcoma (LCS) are proliferative processes consisting of cells having morphologic and phenotypic features of Langerhans cells (LCs), although the latter may have lost some of these features. Because neoplastic nature of LCH as well as LCS is more likely by recent studies, a category of LC hyperplasia can be better characterized. LCH and LCS are rarely seen in daily pathology practice, but it is important to accurately characterize these lesions. For this purpose, an outline covering proliferations of LC and related cells was constructed. The scheme of this outline is based especially on evaluating borderline lesions, neoplastic trans-differentiation, and degree of similarity with the normal counter-parts. In addition, the organization and update of the current classification scheme for histiocytic and dendritic-cell proliferations is presented.

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Rapidly Progressing Fatal Adult Multi-Organ Langerhans Cell Histiocytosis Complicated with Fatty Liver Disease

Cited by 9 publications

References 23 publications

Evaluating the Anti-Obesity Effects of Chitosan in High-Fat Diet-Induced Obesity in Rat Model

Evaluating the Anti-Obesity Effects of Chitosan in High-Fat Diet-Induced Obesity in Rat Model

Recent advances in Langerhans cell histiocytosis

Langerhans Cell Histiocytosis and Langerhans Cell Sarcoma: Current Understanding and Differential Diagnosis

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