Extended treatment response to imatinib in Rosai‐Dorfman disease

Singh, Arvind; Simons, Yael; Biren-Fetz, John; Mohapatra, Gayatry; Ni, Hongyu; Gaitonde, Sujata; Khan, Irum

doi:10.1111/ejh.13605

Cited by 5 publications

(2 citation statements)

References 5 publications

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“…This provides an opportunity to use the immunotherapeutic approach for the treatment of this disease. Some authors reported beneficial effects of the use imatinib (a tyrosine kinase inhibitor) and rituximab (CD20 monoclonal antibody) in therapy of systemic RDD [91,92] . However, their efficacy has not been evaluated in the case of RDD with intracranial involvement.…”

Section: Immunotherapymentioning

confidence: 99%

Intracranial Rosai-Dorfman Disease: pathophysiology, diagnosis and treatment

Szymoniuk

Dryla

Pytka

et al. 2022

J Educ Health Sport

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Introduction and purpose: Rosai-Dorfman Disease (RDD), known also as sinus histiocytosis with massive lymphadenopathy(SHML) is a benign histiocytic proliferative syndrome. The etiology and pathogenesis of RDD remains unclear. Central nervous system involvement is a rare event and concerns approximately 7.8% of RDD cases, whereas intracranial lesions constitute almost 90% of CNS-RDD cases. The aim of this literature review was to summarize current knowledge about the diagnosis and treatment of intracranial manifestation of RDD. We also described possible hypotheses regarding the pathophysiology of this disorder. State of knowledge: Even though Rosai-Dorfman disease was thought to be a reactive process, recent evidencedemonstrate the presence of clonality, which means that in this histiocytosis the process that underlies the pathology is neoplastic. Intracranial lesions caused by RDD can be easily misdiagnosed with many diseases such as meningiomas, malignant gliomas or metastatic tumors. The final diagnosis of Rosai-Dorfman disease should be made based on histologic and immunohistochemical examinations. Current therapeutic options for this condition include surgery, radiotherapy, chemotherapy, corticosteroids and immunotherapy. Surgical treatment often constitutes the first-line treatment for intracranial RDD and is the most beneficial treatment option. However, the implementation of adjuvant therapies is very important to avoid the recurrence of lesions, which appear in approximately 14% of subjects after about 10 years from surgery. Conclusion: This literature review presents current data about pathophysiology, diagnosis and treatment of intracranial involvement of Rosai-Dorfman disease. Further studies on this topic should focus on exploring etiologic mechanisms underlying on this pathology and comparing available treatment methods.

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Section: Immunotherapymentioning

confidence: 99%

Intracranial Rosai-Dorfman Disease: pathophysiology, diagnosis and treatment

Szymoniuk

Dryla

Pytka

et al. 2022

J Educ Health Sport

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“…Imatinib has also been used successfully in clinical cases of other histiocytic disorders. Although no histiocytic disorder has known mutations in KIT, ABL or PDGFR, some histiocytic lesions appear to have a great expression of PDGFR 16,17 . Results in seven patients with Erdheim‐Chester disease (a type of histiocytosis) treated with imatinib were variable, although it could be considered as a therapeutic strategy when first‐line treatments fail.…”

Section: What Is New and Conclusionmentioning

confidence: 99%

Thalidomide as treatment of refractory thoracic Rosai‐Dorfman disease

Hoyo-Muñoz

Collado-Borrell

Escudero‐Vilaplana

et al. 2021

Clinical Pharmacy Therapeu

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What is Known and Objective Rosai‐Dorfman disease (RDD) is an infrequent entity of unknown aetiology. Currently, there is no clear consensus on the treatment, and nothing has shown definitive safety and efficacy. We describe the case of a woman diagnosed with pulmonary RDD, who responded to thalidomide treatment after failure of four previous lines of systemic chemotherapy. Case Description We present the case of a 74‐year‐old woman diagnosed with pulmonary RDD and autoimmune complications. We decided to use thalidomide as a rescue treatment after the failure of corticosteroids and several chemotherapies. Our patient achieved remission of the disease and remained stable for years. What is New and Conclusion To the authors’ knowledge, this is the first reported case in which thalidomide treatment induced remission in refractory pulmonary RDD. Thalidomide showed a rapid onset of action, with lasting responses, which could make it an exciting option for treating this life‐threatening.

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Surgical management and outcome of primary intracranial Rosai-Dorfman disease: a single-institute experience and pooled analysis of individual patient data

Zhang

et al. 2023

Neurosurg Rev

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Extended treatment response to imatinib in Rosai‐Dorfman disease

Cited by 5 publications

References 5 publications

Intracranial Rosai-Dorfman Disease: pathophysiology, diagnosis and treatment

Intracranial Rosai-Dorfman Disease: pathophysiology, diagnosis and treatment

Thalidomide as treatment of refractory thoracic Rosai‐Dorfman disease

Surgical management and outcome of primary intracranial Rosai-Dorfman disease: a single-institute experience and pooled analysis of individual patient data

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