Association between recent pregnancy or hormonal contraceptive exposure and outcome of desmoid-type fibromatosis

Debaudringhien, M.; Blay, J.-Y.; Bimbai, André-Michel; Bonvalot, Sylvie; Italiano, Antoîne; Rousset-Jablonski, C.; Corradini, Nadège; Piperno‐Neumann, Sophie; Chevreau, Christine; Kurtz, Jean‐Emmanuel; Guillemet, Cécile; Bompas, Emmanuelle; Collard, Olivier; Salas, Sébastien; Cesne, Axel Le; Orbach, Daniel; Thery, Julien; Deley, M.-C. Le; Mir, Olivier; Penel, Nicolas

doi:10.1016/j.esmoop.2022.100578

Cited by 10 publications

(11 citation statements)

References 16 publications

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“…Until now, no validated and reliable biomarker exists that can predict the outcome of DF. Data about the prognostic value of the nature of somatic CTNNB1 pathogenic variant are questionable; in prior publication from the ALTITUDES study, we did not find an association between the CTTNB1 mutational status and outcome 8 . Excluding CTNNB1 , there is no other putative biomarker that could predict the outcome.…”

Section: Discussionmentioning

confidence: 59%

“…This heterogeneity in patient management reflects how DF is a hard-to-treat tumor, but is a study limitation. As previously published, 8,9 we have used a complex composite endpoint (EFS) that could be debated, we strongly think that this EFS definition catch the different scenarios of DF worsening. However, the results of our current study were very stable when focusing on patients with active surveillance as first approach, with no significant heterogeneity in terms of the prognostic value associated with pain according to first-line management (interaction test, P = .27).…”

Section: Discussionmentioning

confidence: 95%

“…As previously reported, the events considered were local relapse after complete (R0/R1) resection, disease progression according to the local investigator after R2 resection, during active surveillance and systemic treatments or death from any cause. 8,9 EFS was censored at the date of the last contact when no event was reported in the follow-up and at the date of second-line treatment if treatment was started because of worsening symptoms but without documented progression or relapse. The association between pain and EFS was assessed by Log-rank test in the univariate analysis and by multivariate Cox models to control for possible confounders.…”

Section: Discussionmentioning

confidence: 99%

“…Data about the prognostic value of the nature of somatic CTNNB1 pathogenic variant are questionable; in prior publication from the ALTITUDES study, we did not find an association between the CTTNB1 mutational status and outcome. 8 Excluding CTNNB1, there is no other putative biomarker that could predict the outcome. Pain is possibly an easily obtainable bedside biomarker that could identify DF associated with high risk of relapse of progression.…”

Section: Discussionmentioning

confidence: 99%

“…The association between pain (as well as quality of life, anxiety and depression) and outcome was analyzed using the event‐free survival (EFS) as the endpoint. As previously reported, the events considered were local relapse after complete (R0/R1) resection, disease progression according to the local investigator after R2 resection, during active surveillance and systemic treatments or death from any cause 8,9 . EFS was censored at the date of the last contact when no event was reported in the follow‐up and at the date of second‐line treatment if treatment was started because of worsening symptoms but without documented progression or relapse.…”

Section: Methodsmentioning

confidence: 99%

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Pain in desmoid‐type fibromatosis: Prevalence, determinants and prognosis value

Penel

Bonvalot

Deley

et al. 2023

Intl Journal of Cancer

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The aim of this study is to evaluate the prevalence, determinants and prognostic value of pain at diagnosis in patients with desmoid‐type fibromatosis (DF). We selected patients from the ALTITUDES cohort (NCT02867033), managed by surgery, active surveillance or systemic treatments, with pain assessment at diagnosis. Patients were invited to fill QLQ‐C30 questionnaire and Hospital Anxiety Depression Scale. Determinants were identified using logistic models. Prognostic value on event‐free survival (EFS) was evaluated using the Cox model. Overall, 382 patients were included in the current study (median age: 40.2 years; 117 men). The prevalence of pain was 36%, without significant difference according to first‐line treatment (P = .18). In the multivariate analysis, pain was significantly associated with tumor size >50 mm (P = .013) and tumor site (P < .001); pain was more frequent in the neck and shoulder locations (odds ratio: 3.05 [1.27‐7.29]). Pain at baseline was significantly associated with poor quality of life (P < .001), depression (P = .02), lower performance status (P = .03) and functional impairment (P = .001); we also observed a nonsignificant association with anxiety (P = .10). In the univariate analysis, baseline pain was associated with poor EFS; the 3‐year EFS was 54% in patients with pain compared to 72% in those without pain. After adjustment for sex, age, size and line of treatment, pain was still associated with poor EFS (hazard ratio: 1.82 [1.23‐2.68], P = .003). One third of recently diagnosed patients with DF experienced pain, especially those with larger tumors and neck/shoulder locations. Pain was associated with unfavorable EFS after adjustment for the confounders.

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Section: Discussionmentioning

confidence: 59%

Section: Discussionmentioning

confidence: 95%

Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Section: Methodsmentioning

confidence: 99%

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Pain in desmoid‐type fibromatosis: Prevalence, determinants and prognosis value

Penel

Bonvalot

Deley

et al. 2023

Intl Journal of Cancer

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Desmoid Tumors: A Comprehensive Review

Bektas

Bell²,

Khan

et al. 2023

Adv Ther

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Desmoid tumors (DT) are rare, locally aggressive, fibroblastic soft-tissue tumors that are characterized by infiltrative growth and can affect organs and adjacent structures, resulting in substantial clinical burden impacting patients’ health-related quality of life. Searches of PubMed, Embase, Cochrane, and key conferences were conducted in November 2021 and updated periodically through March 2023 to identify articles describing the burden of DT. Of 651 publications identified, 96 relevant ones were retained. Diagnosis of DT is challenging because of its morphologic heterogeneity and variable clinical presentation. Patients visit multiple healthcare providers, often facing delays in correct diagnosis. The low incidence of DT (estimated 3–5 cases per million person-years) limits disease awareness. Patients with DT experience a high symptom burden: up to 63% of patients experience chronic pain, which leads to sleep disturbance (73% of cases), irritability (46% of cases), and anxiety/depression (15% of cases). Frequently mentioned symptoms are pain, limited function and mobility, fatigue, muscle weakness, and swelling around the tumor. Overall, quality of life in patients with DT is lower than in healthy controls. There is no treatment approved by the US Food and Drug Administration for DT; however, treatment guidelines reference available options, such as active surveillance, surgery, systemic therapy, and locoregional therapy. Choice of active treatment may depend on tumor location, symptoms, and risk of morbidity. The substantial burden of illness of DT is related to difficulties in timely and accurate diagnosis, high symptom burden (pain and functional limitations), and decreased quality of life. There is a high unmet need for treatments that specifically target DT and improve quality of life. Supplementary Information The online version contains supplementary material available at 10.1007/s12325-023-02592-0.

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