Failure of crizotinib based systemic treatment in ALK positive histiocytosis involving the central nervous system: a case report and literature review

Abstract

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.

Letter to the Editor Regarding “Local ALK-Positive Histiocytosis with Unusual Morphology and Novel TRIM33::ALK Gene Fusion” by Tran et al.

“…Treatment options are evaluated based on disease severity and site of involvement; these options include surgical excision, radiation therapy, medical treatment, and supportive care. 2,6–8 In the current case, positron emission tomography/computed tomography did not show any abnormalities in other systems. The patient is being observed without treatment and is symptom-free at 6 months following the procedure.…”

Case report: Systemic presentation of ALK-positive Histiocytosis

“…Prognosis is generally better for multiple lesions, while systemic presentations vary depending on the affected sites. Reviewing literature cases with follow-ups ranging from 2 to 14 years, one case resulted in death, one survived the disease, and the rest showed no abnormalities or disease recurrence (2,3,(5)(6)(7). Recently, some scholars have successively reported isolated cases, raising whether they are related to systemic ALK-HSs, which requires further investigation (10).…”

“…The prognosis for this condition is primarily favorable, but in a few cases, it can be poor. Searching in the international medical literature, fewer than 50 cases have been reported to date, making systemic ALK-HSs extremely rare (2,3). This article presents one case of systemic ALK-HSs, detailing the patient's treatment, follow-up, and prognosis.…”

The causal relationship between human brain morphometry and knee osteoarthritis: a two-sample Mendelian randomization study

“…Brain-wide volumes, or the comprehensive volumetric measures of different regions within the human brain, play pivotal roles in various physiological processes and are crucial for understanding the complex interplay between brain structure and function ( Hasan et al, 2011 ; Immonen et al, 2020 ). These volumetric attributes of the brain are not only indicative of individual neuroanatomical variability but also provide invaluable insights into the underlying mechanisms of numerous diseases and disorders, particularly those with neuropsychological components ( Varghese et al, 2017 ; He et al, 2022 ; Statsenko et al, 2022 ; Díez-Cirarda et al, 2023 ). The significance of brain-wide volumes extends across multiple dimensions of human health and disease.…”