Osteopathy in Complex Lymphatic Anomalies

Solorzano, Ernesto; Alejo, Andrew; Ball, Hope C.; Magoline, Joseph; Khalil, Yusuf; Kelly, Michael; Safadi, Fayez F.

doi:10.3390/ijms23158258

Cited by 6 publications

(7 citation statements)

References 124 publications

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“…Lymphatic vessels are not found in normal healthy bone; however, bone involvement in KLA is observed on imaging and in tissue specimens 33,34 . Osseous lesions in KLA localize to trabecular bone and marrow, and they may significantly affect structural integrity leading to fractures, pain, and disability 35 . Treatment with steroids, used in the initial treatment of KLA, can weaken bone due to glucocorticoid‐induced osteoporosis 36 .…”

Section: Bone and Bone Marrow Involvementmentioning

confidence: 99%

“…33,34 Osseous lesions in KLA localize to trabecular bone and marrow, and they may significantly affect structural integrity leading to fractures, pain, and disability. 35 Treatment with steroids, used in the initial treatment of KLA, can weaken bone due to glucocorticoidinduced osteoporosis. 36 Bone resorption markers, such as carboxyterminal cross-linking telopeptide (CTX-1), may be elevated.…”

Section: Bone and Bone Marrow Involvementmentioning

confidence: 99%

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Kaposiform lymphangiomatosis: Diagnosis, pathogenesis, and treatment

McDaniel

Adams

Steele³

et al. 2023

Pediatric Blood & Cancer

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Kaposiform lymphangiomatosis (KLA) is a life‐threatening rare disease that can cause substantial morbidity, mortality, and social burdens for patients and their families. Diagnosis often occurs long after initial symptoms, and there are few centers in the world with the expertise to diagnose and care for patients with the disease. KLA is a lymphatic anomaly and significant advancements have been made in understanding its pathogenesis and etiology since its first description in 2014. This review provides multidisciplinary, comprehensive, and state‐of‐the‐art information on KLA patient presentation, diagnostic imaging, pathology, organ involvement, genetics, and pathogenesis. Finally, we describe current therapeutic approaches, important areas for research, and challenges faced by patients and their families. Further insights into the pathogenesis of KLA may advance our understanding of other vascular anomalies given that similar signaling pathways may be involved.

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Section: Bone and Bone Marrow Involvementmentioning

confidence: 99%

Section: Bone and Bone Marrow Involvementmentioning

confidence: 99%

Kaposiform lymphangiomatosis: Diagnosis, pathogenesis, and treatment

McDaniel

Adams

Steele³

et al. 2023

Pediatric Blood & Cancer

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“…2 Chronic inflammation inhibits osteoblasts (OBs) differentiation while activating OCs, and the equilibrium between bone repair and resorption becomes unstable. 3 Despite the fact that immune cells, mesenchymal cells, and the environment are all involved in inflammatory osteolysis, OCs are essential actors in the process. 4 OCs are the major bone-resorbing cells generated from mononuclear macrophages.…”

Section: Introductionmentioning

confidence: 99%

“…Acute inflammation is mostly caused by endogenous or exogenous unfavourable stimuli and can progress to chronic inflammation if not treated in time to preserve tissue homeostasis 2 . Chronic inflammation inhibits osteoblasts (OBs) differentiation while activating OCs, and the equilibrium between bone repair and resorption becomes unstable 3 . Despite the fact that immune cells, mesenchymal cells, and the environment are all involved in inflammatory osteolysis, OCs are essential actors in the process 4 …”

Section: Introductionmentioning

confidence: 99%

IKK/NF‐κB and ROS signal axes are involved in Tenacissoside H mediated inhibitory effects on LPS‐induced inflammatory osteolysis

Xie

Chen

et al. 2023

Cell Proliferation

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Periodontal disease and arthroplasty prosthesis loosening and destabilization are both associated with osteolysis, which is predominantly caused by abnormal bone resorption triggered by pro‐inflammatory cytokines. Osteoclasts (OCs) are critical players in the process. Concerns regarding the long‐term efficacy and side effects of current frontline therapies, however, remain. Alternative therapies are still required. The aim of this work was to investigate the involvement of Tenacissoside H (TDH) in RANKL‐mediated OC differentiation, as well as inflammatory osteolysis and associated processes. In vitro, bone marrow‐derived macrophages (BMMs) cultured with RANKL and M‐CSF were used to detect TDH in the differentiation and function of OCs. Real‐time quantitative PCR was used to measure the expression of specific genes and inflammatory factors in OCs. Western blot was used to identify NFATc1, IKK, NF‐κB, MAPK pathway, and oxidative stress‐related components. Finally, an LPS‐mediated calvarial osteolysis mouse model was employed to explore TDH's role in inflammatory osteolysis. The results showed that in vivo TDH inhibited the differentiation and resorption functions of OCs and down‐regulated the transcription of osteoclast‐specific genes, as well as Il‐1β, Il‐6 and Tnf‐α. In addition, TDH inhibited the IKK and NF‐κB signalling pathways and down‐regulated the level of ROS. In vivo studies revealed that TDH improves the bone loss caused by LPS. TDH may be a new candidate or treatment for osteoclast‐associated inflammatory osteolytic disease.

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“…Complex lymphatic anomalies (CLAs) are categorized by idiopathic boney lesions because of abnormal lymphatic invasion [1]. CLAs include Gorham-Stout disease (GSD), generalized lymphatic anomaly (GLA), kaposiform lymphangiomatosis (KLA), and central conducting lymphatic anomaly (CCLA).…”

Section: Introductionmentioning

confidence: 99%

The Lymphatic Endothelial Cell Secretome Inhibits Osteoblast Differentiation and Bone Formation

Solorzano,

Alejo,

Ball

et al. 2023

Cells

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Complex lymphatic anomalies (CLAs) are a set of rare diseases with unique osteopathic profiles. Recent efforts have identified how lymphatic-specific somatic activating mutations can induce abnormal lymphatic formations that are capable of invading bone and inducing bone resorption. The abnormal bone resorption in CLA patients has been linked to overactive osteoclasts in areas with lymphatic invasions. Despite these findings, the mechanism associated with progressive bone loss in CLAs remains to be elucidated. In order to determine the role of osteoblasts in CLAs, we sought to assess osteoblast differentiation and bone formation when exposed to the lymphatic endothelial cell secretome. When treated with lymphatic endothelial cell conditioned medium (L-CM), osteoblasts exhibited a significant decrease in proliferation, differentiation, and function. Additionally, L-CM treatment also inhibited bone formation through a neonatal calvaria explant culture. These findings are the first to reveal how osteoblasts may be actively suppressed during bone lymphatic invasion in CLAs.

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Osteopathy in Complex Lymphatic Anomalies

Cited by 6 publications

References 124 publications

Kaposiform lymphangiomatosis: Diagnosis, pathogenesis, and treatment

Kaposiform lymphangiomatosis: Diagnosis, pathogenesis, and treatment

IKK/NF‐κB and ROS signal axes are involved in Tenacissoside H mediated inhibitory effects on LPS‐induced inflammatory osteolysis

The Lymphatic Endothelial Cell Secretome Inhibits Osteoblast Differentiation and Bone Formation

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