Purpose: Vascular anomalies (VAs) are a heterogeneous group of congenital disorders characterized by variable clinical presentation, biological behaviour, and therapeutic response. The classification system developed by the International Society for the Study of Vascular Anomalies (ISSVA) is currently the global standard, categorizing VAs based on histopathological features, hemodynamic, and clinical progression. Lymphatic malformations (LMs), including Lymphangioma Circumscriptum (LC)& horbar;a localized and often superficial variant & horbar;are stratified into macrocystic, microcystic, or mixed subtypes. Despite the system's widespread adoption, the absence of standardized dimensional criteria and consistent diagnostic protocols limits its applicability. The rarity of oral LC, especially in paediatric patients alongw with its clinical overlap with other vascular or neoplastic lesions, further complicates diagnosis and management. This study introduces an integrated High-Tech protocol combining High-Definition Intraoral Ultrasonography (IHDUS) and Diode Laser for the management of LC occuring in the tongue of paediatric patients. A comprehensive literature review further contextualizes the clinic-pathological significance of the herein reported approach within contemporary frameworks for vascular anomaly management. Methods: All patients underwent a structured three-step protocol: IHDUS for the precise assessment of lesion depth, vascularity, and extension; Diode Laser transmucosal photocoagulation to promote volume and vascular supply reduction with subsequent Diode Laser-assisted surgical excision; histological examination withimmunohistochemical analysis too. Results: 7 paediatric cases with histologically confirmed LC of the tongue were treated, all demonstrating comparable clinical and radiological characteristics; after surgery, complete healing was observed in all cases without complications. Conclusion: Data from literature mainly describe invasive procedures for LC treatment, lacking standardization and often unsuitable for paediatric populations. The proposed protocol demonstrated substantial clinical value as a minimally invasive, safe, and reproducible approach for the diagnosis and management of LC of the tongue in paediatric patients. (c) 2025 The Author(s). Published by Elsevier Inc. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).
Lymphangioma circumscriptum of the tongue in paediatric patients: a clinic-pathological and immunohistochemical study with high-tech protocol and comprehensive literature analysis
Limongelli L.;Forte M.
;Cascardi E.;Cervinara F.;Cianciotta G.;Dell'Olio F.;Scaringella G.;Favia G.;Capodiferro S.
2025-01-01
Abstract
Purpose: Vascular anomalies (VAs) are a heterogeneous group of congenital disorders characterized by variable clinical presentation, biological behaviour, and therapeutic response. The classification system developed by the International Society for the Study of Vascular Anomalies (ISSVA) is currently the global standard, categorizing VAs based on histopathological features, hemodynamic, and clinical progression. Lymphatic malformations (LMs), including Lymphangioma Circumscriptum (LC)& horbar;a localized and often superficial variant & horbar;are stratified into macrocystic, microcystic, or mixed subtypes. Despite the system's widespread adoption, the absence of standardized dimensional criteria and consistent diagnostic protocols limits its applicability. The rarity of oral LC, especially in paediatric patients alongw with its clinical overlap with other vascular or neoplastic lesions, further complicates diagnosis and management. This study introduces an integrated High-Tech protocol combining High-Definition Intraoral Ultrasonography (IHDUS) and Diode Laser for the management of LC occuring in the tongue of paediatric patients. A comprehensive literature review further contextualizes the clinic-pathological significance of the herein reported approach within contemporary frameworks for vascular anomaly management. Methods: All patients underwent a structured three-step protocol: IHDUS for the precise assessment of lesion depth, vascularity, and extension; Diode Laser transmucosal photocoagulation to promote volume and vascular supply reduction with subsequent Diode Laser-assisted surgical excision; histological examination withimmunohistochemical analysis too. Results: 7 paediatric cases with histologically confirmed LC of the tongue were treated, all demonstrating comparable clinical and radiological characteristics; after surgery, complete healing was observed in all cases without complications. Conclusion: Data from literature mainly describe invasive procedures for LC treatment, lacking standardization and often unsuitable for paediatric populations. The proposed protocol demonstrated substantial clinical value as a minimally invasive, safe, and reproducible approach for the diagnosis and management of LC of the tongue in paediatric patients. (c) 2025 The Author(s). Published by Elsevier Inc. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).I documenti in IRIS sono protetti da copyright e tutti i diritti sono riservati, salvo diversa indicazione.
