Adamantinomatous craniopharyngioma in the molecular age and the potential of targeted therapies: a review

Whelan, Ros; Hengartner, Astrid; Folzenlogen, Zach; Prince, Eric; Hankinson, Todd C.

doi:10.1007/s00381-020-04677-5

Cited by 16 publications

(14 citation statements)

References 46 publications

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“…Immune checkpoint blockade (ICB) has been proposed as a promising treatment for ACP, but its efficacy is interfered by the complex immunosuppressive TME in ACP (47,48). Previous studies in our laboratory and others have suggested complex expression patterns of B7 family ligands/receptors in craniopharyngiomas (7,31,49,50). To further elucidate the functional characteristics of tumor-infiltrating T cells, we performed differential expression analysis of T reg and cytotoxic T cells derived from neoplastic and nonneoplastic tissues (Fig.…”

Section: T Cell Clustering and Subtype Analysis In Acpmentioning

confidence: 99%

Single-cell RNA sequencing highlights intratumor heterogeneity and intercellular network featured in adamantinomatous craniopharyngioma

et al. 2023

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Despite improvements in microscopically neurosurgical techniques made in recent years, the prognosis of adamantinomatous craniopharyngioma (ACP) is still unsatisfactory. Little is known about cellular atlas and biological features of ACP. Here, we carried out integrative analysis of 44,038 single-cell transcriptome profiles to characterize the landscape of intratumoral heterogeneity and tumor microenvironment (TME) in ACP. Four major neoplastic cell states with distinctive expression signatures were defined, which further revealed the histopathological features and elucidated unknown cellular atlas of ACP. Pseudotime analyses suggested potential evolutionary trajectories between specific neoplastic cell states. Notably, a distinct oligodendrocyte lineage was identified in ACP, which was associated with immunological infiltration and neural damage. In addition, we described a tumor-centric regulatory network based on intercellular communication in TME. Together, our findings represent a unique resource for deciphering tumor heterogeneity of ACP, which will improve clinical diagnosis and treatment strategies.

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Section: T Cell Clustering and Subtype Analysis In Acpmentioning

confidence: 99%

Single-cell RNA sequencing highlights intratumor heterogeneity and intercellular network featured in adamantinomatous craniopharyngioma

et al. 2023

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“…This type harbors CTNNB1 or APC mutations encoding for beta-catenin, which are being investigated for potential targeted therapy. 40,41…”

Section: Discussionmentioning

confidence: 99%

Is Postoperative Radiotherapy Needed in the Management of Adult Craniopharyngiomas?

Khriguian

Tolba

Khosrow‐Khavar

et al. 2022

Can. J. Neurol. Sci.

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Background: The optimal treatment of adult craniopharyngioma (CP) remains controversial. Although benign, these tumors tend to recur locally. The choice between gross total resection (GTR) versus subtotal resection (STR) with adjuvant or delayed radiotherapy (RT) is debated. The objective of this study is to review our experience with adult CPs over a 20-year period and identify an optimal management strategy. Methods: From 1999 to 2020, we reviewed all patients diagnosed with CP at our institution. We collected data regarding tumor characteristics, treatments, and toxicity. Disease progression was defined as growth on imaging. Descriptive statistics were used to assess patient characteristics. The Kaplan Meier method was used to assess progression-free survival (PFS) and corresponding 95% confidence intervals (CI) from the time since treatment initiation. Results: Twenty-four patients with a median age of 50 were included in this study. The median follow-up was 85 months. Seven patients had initial GTR, 10 STR, and 7 STR + RT. The overall 5-year PFS was 56% (95% CI: 38–83%): 100% in the STR + RT group, 69% in the GTR group, and 18% in the STR group (p = 0.01). Of the 17 patients initially treated with surgery alone, 3 with GTR and 6 with STR required salvage RT at a median of 46 months, with no further progression after salvage RT. Conclusions: Our study underscores the importance of RT for local control and suggests that STR + RT should be considered a viable option in the management of these tumors as it may be associated with improved PFS compared to surgery alone.

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“…To the best of our knowledge, amylase is being reported for the first time in craniopharyngioma cystic fluid in this study. Studies on the biochemical composition of the cystic fluid are scanty, and there have been reports of lipids, proteins, glucose, urea, creatinine, sodium, calcium, magnesium, phosphorous, chlorine, ALT, AST, and GGT, but none on amylase ( 7 , 8 , 14 , 34 , 35 ). The significance of this important finding remains to be elucidated.…”

Section: Discussionmentioning

confidence: 99%

“…Preferred management has shifted from radical to conservative resection, with an acceptance that this is a chronic disease, and the goal of therapy is to provide a good quality of life rather than a cure. The majority of options favour partial debulking of the tumour, intra-cystic therapies, and radiotherapy (8)(9)(10)(11)(12)(13). Raised awareness of the cystic component for Ommaya reservoirs and intra-cystic drug therapies has resulted in several studies that explore the nature and contents of this cyst.…”

Section: Introductionmentioning

confidence: 99%

Biochemical profile of paediatric adamantinomatous craniopharyngiomas: A prospective cohort study

Jokonya

Mduluza-Jokonya²,

Mwale³

et al. 2022

Front. Surg.

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IntroductionCraniopharyngioma is a rare brain tumour. Despite being histologically benign, it behaves aggressively and is often difficult to manage. Descriptive epidemiological data on the tumour is lacking in sub-Saharan Africa, and there is none for Zimbabwe. The tumour usually has a cystic component that has been raising interest in the past decade. Few studies have looked at the biochemical composition thereof. This study aims to give a landscape view of craniopharyngiomas (CPs) in Zimbabwe and then profile the biochemical properties of the cystic component of paediatric adamantinomatous craniopharyngioma.MethodologyA prospective cohort study was done in Zimbabwe over a 2-year period to study the epidemiological distribution of craniopharyngioma and examine the biochemical composition of adamantinomatous craniopharyngioma cystic fluid in the paediatric population. Fifteen patients were recruited who had craniopharyngiomas, and of those, nine paediatric adamantinomatous craniopharyngiomas had fluid analysed for biochemical components. SPSS statistical package was used to analyse the data. Descriptive statistics were used for epidemiological data.ResultsThe incidence of CP was calculated to be 0.53 per million person-years. Incidence among the paediatric population 0–14 years was 1.2 per 100,000 person-years. Several biological components were found to be elevated significantly compared to serum and cerebral spinal fluid (CSF). These are sodium, potassium, urea, alkaline phosphatase, phosphate, magnesium, albumin, gamma-glutamyl transferase, calcium, low-density lipids, and glucose.ConclusionThe incidence of CP in Zimbabwe is similar to the rest of the world. Some biochemical components have been noted to be markedly elevated in the cystic fluid and were mirroring serum rather than CSF in concentration.

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Adamantinomatous craniopharyngioma in the molecular age and the potential of targeted therapies: a review

Cited by 16 publications

References 46 publications

Single-cell RNA sequencing highlights intratumor heterogeneity and intercellular network featured in adamantinomatous craniopharyngioma

Single-cell RNA sequencing highlights intratumor heterogeneity and intercellular network featured in adamantinomatous craniopharyngioma

Is Postoperative Radiotherapy Needed in the Management of Adult Craniopharyngiomas?

Biochemical profile of paediatric adamantinomatous craniopharyngiomas: A prospective cohort study

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