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. 2022 Aug 19;14(16):4007.
doi: 10.3390/cancers14164007.

Suprarenal Masses in Very Young Infants: Is It Safe to Watch and Wait? Report of a SIOPEN Observational Study Results

Vassilios Papadakis  1 , Vanessa Segura  2 , Massimo Conte  3 , Dominique Plantaz  4 , Andrea Di Cataldo  5 , Gudrun Schleiermacher  6 , Kate Wheeler  7 , Jose D Bermúdez  8 , Shifra Ash  9 , Bénédicte Brichard  10 , Ruth Ladenstein  11 , Valérie Combaret  12 , Sabine Sarnacki  13 , Anna Maria Fagnani  14 , Claudio Granata  15 , Adela Cañete  16   17
Affiliations

Suprarenal Masses in Very Young Infants: Is It Safe to Watch and Wait? Report of a SIOPEN Observational Study Results

Vassilios Papadakis et al. Cancers (Basel). .

Abstract

Background: To assess whether expectant observation of infants ≤ 90 days old with small suprarenal masses (sSRMs) could avoid unnecessary surgery without impacting outcome. Methods: Infants ≤ 90 days with a ≤ 5 cm mass, without midline extension or lymph node or distant spread were registered (ClinicalTrials.org:NCT01728155). Once staging was completed, they were followed with ultrasound, MRI and urinary catecholamines. Surgical resection was only planned if there was a ≥40% mass volume increase or for a mass persisting after 48 weeks of the planned observation. Results: Over a 5-year period, 128 infants were registered. No infant had detectable MYCN amplification in the peripheral blood. Surgery was performed in 39 (30.5%) patients, in 18 during and in 21 after the planned 48-week observation, and 74% were confirmed to be neuroblastomas. Non-life-threatening surgical complications occurred in two cases. The 3-year overall survival and event-free survival were 100% and 87.1%, respectively. The 16 events observed were volume increase (N = 11) and progression to neuroblastoma stage MS (N = 5). Patients with solid masses or MIBG-positive masses had lower EFS. Conclusions: Expectant observation for infants with sSRMs with clinical follow-up and timely imaging (including MRI scan) is safe and effective, allowing surgery to be avoided in the majority of them.

Keywords: infant; neonate; neuroblastoma; observation; suprarenal masses; surgery.

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Conflict of interest statement

V.S., M.C., D.P., A.D.C., G.S., K.W., J.D.B., S.A., B.B., V.C., S.S., A.M.F. and C.G. declare no conflict of interest. A.C. and R.L. report a grant from FP7/2007–2013 ENCCA during the conduct of the study. V.P. has received honoraria for lecturing (Sobi, EUSA Pharma) and travel support for scientific congress attendance (Sanofi, EUSA Pharma, Novartis).

Figures

Figure 1
Figure 1
Therapeutic management of suprarenal masses with follow-up investigations at prescribed intervals during the observational course of the study.
Figure 2
Figure 2
(A) Left: Event-free survival for the whole cohort (n = 128) patients enrolled in the study. Right: EFS for three cohorts of patients based on MIBG procedure: positive (Group 1, n= 42), negative (Group 2, n = 33) and patients with MIBG not done (Group 3, n = 53). (B) Left: Event-free survival by mass structure for the whole cohort (n = 128) patients enrolled in the study. Right: EFS by mass structure for the cohort of patients with MIBG-positive masses (Group 1 n = 42). (C) Left: Resection-free survival for the whole cohort (n = 128) patients enrolled in the study. Right: RFS for three cohorts of patients based on MIBG procedure: positive (Group 1, n= 42), negative (Group 2, n = 33) and patients with MIBG not done (Group 3, n = 53). (D) Left: Resection-free survival by mass structure for the whole cohort (n = 128) patients enrolled in the study. Right: RFS by mass structure for the cohort of patients with MIBG-positive masses (Group 1, n = 42).
Figure 3
Figure 3
Event-free survival effect of volume and nature of mass for the whole cohort (n = 96 available patients with 3D tumor measures from first postnatal US). The predictive evolution of 9 hypothetical patients is shown, combining the three types of mass structure ((A) cystic, (B) mixed and (C) solid) with three volumes: 1 cm3, in red; 6 cm3, in green, and 12 cm3 blue.
Figure 4
Figure 4
Swimmer/Bubble combined plot graph of patients with a SRM who underwent surgery and had (A) or did not have (B) an event. (C) Patients who achieved complete remission during the 48-week observation period. The plot displays evolution of the mass over time until surgery. Each patient is represented as a single horizontal line. Bubbles depict tumor volume (3D measurements in ml) for each US imaging monitoring point. Type of mass is color coded: orange (cystic), green (mixed) and blue (solid).
Figure 4
Figure 4
Swimmer/Bubble combined plot graph of patients with a SRM who underwent surgery and had (A) or did not have (B) an event. (C) Patients who achieved complete remission during the 48-week observation period. The plot displays evolution of the mass over time until surgery. Each patient is represented as a single horizontal line. Bubbles depict tumor volume (3D measurements in ml) for each US imaging monitoring point. Type of mass is color coded: orange (cystic), green (mixed) and blue (solid).
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References

    1. Holgersen L.O., Subramanian S., Kirpekar M., Mootabar H., Marcus J.R. Spontaneous resolution of antenatally diagnosed adrenal masses. J. Pediatr. Surg. 1996;31:153–155. doi: 10.1016/S0022-3468(96)90339-5. - DOI - PubMed
    1. Kerbl R., Urban C.E., Lackner H., Höfler G., Ambros I.M., Ratschek M., Ambros P.F. Connatal localized neuroblastoma: The case to delay treatment. Cancer. 1996;77:1395–1401. doi: 10.1002/(SICI)1097-0142(19960401)77:7<1395::AID-CNCR26>3.0.CO;2-W. - DOI - PubMed
    1. Kerbl R., Urban C.E., Ambros I.M., Dornbusch H.J., Schwinger W., Lackner H., Ladenstein R., Strenger V., Gadner H., Ambros P.F. Neuroblastoma mass screening in late infancy: Insights into the biology of neuro-blastic tumors. J. Clin. Oncol. 2003;21:4228–4234. doi: 10.1200/JCO.2003.10.168. - DOI - PubMed
    1. Nadler E.P., Barksdale E.M. Adrenal Masses in the Newborn. Semin. Pediatr. Surg. 2000;9:156–164. doi: 10.1053/spsu.2000.7560. - DOI - PubMed
    1. Sauvat F., Sarnacki S., Brisse H., Medioni J., Rubie H., Aigrain Y., Gauthier F., Audry G., Helardot P., Landais P., et al. Outcome of suprarenal localized masses diagnosed during the perinatal period: A retrospective multicenter study. Cancer. 2002;94:2474–2480. doi: 10.1002/cncr.10502. - DOI - PubMed

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