Key result
Crizotinib fails to shrink ALK-positive histiocytosis CNS lesions, requiring cytarabine for partial response.
Why the study?
Only a few cases of ALK-positive histiocytosis with central nervous system involvement have been reported, and involvement of the suprasellar region had not been documented.
Case Report (n=1)
No
Additional adjuvant chemotherapy drugs like cytarabine may be needed when ALK-inhibitor treatment is ineffective in ALK-positive histiocytosis with CNS involvement.
Crizotinib may not suffice alone for CNS-involved ALK-positive histiocytosis; hypothesis-generating for cytarabine combinations in this rare pediatric entity.
BACKGROUND: Among the histiocytic disorders, anaplastic lymphoma kinase (ALK)-positive histiocytosis emerged in 2008. As more and more cases of the novel entity are reported, our understanding of it is deepened. However, only a few cases with central nervous system (CNS) involvement have been reported. Furthermore, the lesion in the suprasellar region has not been documented. CASE PRESENTATION: We presented a case of ALK-positive histiocytosis involving the suprasellar region of a one-year-and-four-month-old boy. Through clinical, neuropathological, and genomic analyses, the patient was diagnosed with ALK-positive histiocytosis. After lesions were resected he started treatment with a combination of the three compounds vincristine, prednisolone, and crizotinib, but they did not work. Cytarabine was then added as an additional chemotherapy drug for him, and the lesions in the brain and lungs were shrunk by combining treatment of crizotinib, dexamethasone, vincristine, and cytarabine according to the RECIST (esponse Evaluation Criteria In Solid Tumours). CONCLUSIONS: Additional adjuvant chemotherapy drugs are needed when ALK-inhibitor treatment is ineffective.
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He et al. (2022) conducted a case report in ALK-positive histiocytosis involving the central nervous system (n=1). Crizotinib-based systemic treatment (crizotinib, vincristine, prednisolone) was evaluated on Treatment response (lesion size). In a 16-month-old boy with ALK-positive histiocytosis involving the central nervous system, crizotinib-based systemic treatment failed to shrink lesions, requiring the addition of cytarabine for partial response.
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