Submandibular Sialoblastoma in a Newborn: A Case Report and Surgical Approach

Submandibular Sialoblastoma in a Newborn: A Case Report and Surgical Approach | Authorea try { document.documentElement.classList.add('js'); } catch (e) { } var _gaq = _gaq || []; _gaq.push(['_setAccount', 'G-8VDV14Y67G']); _gaq.push(['_trackPageview']); (function() { var ga = document.createElement('script'); ga.type = 'text/javascript'; ga.async = true; ga.src = ('https:' == document.location.protocol ? 'https://ssl' : 'http://www') + '.google-analytics.com/ga.js'; var s = document.getElementsByTagName('script')[0]; s.parentNode.insertBefore(ga, s); })(); Skip to main content Preprints Collections Wiley Open Research IET Open Research Ecological Society of Japan All Collections About About Authorea FAQs Contact Us Quick Search anywhere Search for preprint articles, keywords, etc. Search Search ADVANCED SEARCH SCROLL This is a preprint and has not been peer reviewed. Data may be preliminary. 21 July 2025 V2 Latest version Share on Submandibular Sialoblastoma in a Newborn: A Case Report and Surgical Approach Authors : Gabriela Martínez 0009-0008-3973-3144 [email protected] , Selene Venegas , Carolina Sued , and Rafael Estévez Authors Info & Affiliations https://doi.org/10.22541/au.175260127.76573557/v2 258 views 79 downloads Contents Abstract Information & Authors Metrics & Citations View Options References Figures Tables Media Share Abstract Sialoblastoma is a rare tumor of the salivary glands, rarely diagnosed in infancy, usually presenting in the first months of life. This report describes a case of sialoblastoma in an 18-day-old newborn who presented with a progressively growing mass in the right facial region. A cervical ultrasound identified a right submandibular adenopathy, leading to the decision to perform a right submandibular sialodenectomy under general anesthesia. The surgery was successful, and the pathology report confirmed the presence of a tumor composed mostly of basaloid cell nests. Although sialoblastomas may present local recurrences or metastases, the prognosis for cases diagnosed and treated early is generally favorable. This case highlights the importance of early diagnosis and prompt surgical intervention to enhance outcomes in pediatric patients. KEYWORDS: Sialoblastoma, submandibular gland, pediatric surgery, sialadenectomy, pediatric pathology. Increased volume of the right submaxillary region, predominantly preauricular and right malar. Soft tissue ultrasound at the right submandibular joint. It shows a heterogeneous image with a vascularized center measuring 2.7 x 1.7 cm. Diagnostic impression of right submandibular adenopathy. After the incision in the skin of the neck, just below the jaw, the right submandibular gland is located. Macroscopically, the excised tumor. Three irregular fragments with a whitish, multinodular surface. Layered suturing of the incision to ensure proper healing. A submandibular gland is seen with a tumor composed predominantly of basaloid cell nests. These cells surround preserved ducts of the gland in areas. The basaloid cells show few mitoses and granular chromatin. The stroma shows abundant fibrosis (stained with hematoxylin and eosin). Immunohistochemistry for smooth muscle actin shows positivity in the peripheral myoepithelial cells of the tumor nests, demonstrating a periductal distribution. This expression suggests myoepithelial differentiation, a characteristic finding in neoplasms with a basaloid component such as sialoblastoma. The staining pattern is cytoplasmic and focal, reinforcing the salivary histogenesis of the tumor. Immunohistochemical staining for CK7 reveals intense cytoplasmic positivity in the ductal cells of the tumor, with preservation of the ductal architecture in multiple foci. This expression confirms the ductal differentiation of the epithelial component of sialoblastoma, supporting its origin from embryonic salivary structures. Three irregular fragments, the largest measuring 4.5 x 3 x 1.4 cm, have a whitish, multinodular surface. Sectioned, it is homogeneously whitish. The smallest measures 1.8 x 0.7 cm with a yellowish surface. Representative sections are included in four blocks. x4. DISCUSSION Primary salivary gland tumors in childhood are rare, with an even lower incidence in children under five years of age (6). Although salivary gland tumors represent less than 0.25% of cases in the first decade of life, the most common are of non-epithelial origin, such as hemangioma (6). However, among epithelial neoplasms, sialoblastoma stands out as a rare tumor, observed mainly in the neonatal or childhood period (3). Most cases are diagnosed in the first months of life, and their prevalence is higher in males (3). This tumor, which commonly originates in the parotid and submandibular glands, presents as a painless, slow-growing subcutaneous mass, ranging in size from 1.5 to 15 cm (3). Histologically, sialoblastoma is characterized by the presence of basaloid cells arranged in clusters and a lack of acinar differentiation, reflecting its embryonic origin in the salivary glands (7). The differential diagnosis of sialoblastomas includes other salivary gland tumors, such as adenoid cystic carcinoma and basal cell adenoma, whose histological patterns are distinct (8). Immunohistochemical studies have identified positivity for cytokeratin in ductal cells and for markers such as vimentin and smooth muscle actin, which corroborate the epithelial nature of these tumors (8). Despite their rarity, it has been suggested that sialoblastomas may be associated with liver markers, such as alpha-fetoprotein, suggesting a possible relationship with liver tumors, although further research is required to confirm this association (8). Regarding prognosis, sialoblastomas exhibit unpredictable behavior, with a tendency to recur locally or metastasize to lymph nodes and lungs (3). Despite their potentially malignant nature, most cases have a favorable prognosis when detected early and treated appropriately (3). Surgical excision is the standard treatment, although radiotherapy and chemotherapy are used in cases with incomplete resection or metastasis (8). Neoadjuvant treatments, such as chemotherapy, are also considered for locally invasive tumors to avoid mutilating surgeries (8). Since the disease primarily occurs in children, the risks of radiotherapy side effects, especially in terms of bone growth and facial development, must be carefully considered (8). Finally, the pathogenesis of sialoblastoma is not fully understood, but it is thought to arise from blastema cells retained in the salivary gland (9). Chromosomal findings have revealed specific aberrations, such as deletion of the long arm of chromosome 3, which differentiates sialoblastoma from other salivary neoplasms (9). Despite the rarity of these tumors, it is crucial to continue research to improve the understanding of their biology, diagnosis, and treatment, thus ensuring adequate follow-up of patients, especially due to possible recurrences up to years after the intervention (9, 10). CONCLUSION Sialoblastoma is a rare tumor of the salivary glands, especially in pediatric patients, as evidenced in this case report of a newborn with a lesion in the submandibular gland. This tumor, although it generally presents in the first months of life and is usually benign, can have unpredictable biological behavior, with risks of local recurrence or distant metastasis. Early diagnosis and complete surgical resection are essential for a favorable prognosis. However, due to the possibility of recurrence, long-term follow-up is recommended, as some cases may present complications years after surgery. This report underscores the importance of an appropriate diagnostic approach and the need for early surgical intervention for the effective management of sialoblastoma in children, with special attention to prognosis and the potential effects of additional treatments in the event of recurrence. BIBLIOGRAPHICAL REFERENCES 1. Vawter GF, Tefft M. Congenital tumors of the parotid gland. ArchPathol. 1966; 82:242-24. 2. Taylor GP. Congenital epithelial tumor of the parotidsialoblastoma. Pathol Pediatr. 1988;8:447 – 452 3. Ellis GL. What’s New in the AFIP Fascicle on Salivary Gland Tumors: A Few Highlights from the 4th Series Atlas. Head and Neck Pathology [Internet]. 2009 Jul 20 [cited 2025 Mar 3];3(3):225–30. Available from: https://link.springer.com/article/10.1007/s12105-009-0128-z 4. Fonseca I, Martins AG, Soares J. Epithelial salivary gland tumors of children and adolescents in southern Portugal: a clinicopathologic study of twenty-four cases. Oral Surg Oral Med Oral Pathol. 1991;72:696 -701. 5. Kwak J, Kim E. Thyroid Imaging Reporting and Data System Ultrasound Lexicon. Ultrasound Med Biol [Internet]. 2019 [cited 2025 Mar 3];25(5):75–82. Available from: https://socpr.org.pe/wpcontent/uploads/2024/06/rev-vol21-caso-1.pdf 6. Peerayut Sitthichaiyakul, Julintorn Somran, Nongluk Oilmungmool, Worasakwuttipong S, Noppadol Larbcharoensub. Sialoblastoma of the cheek: A case report and review of the literature. Molecular and Clinical Oncology [Internet]. 2016 Mar 30 [cited 2025 Mar 3];4(6):925–8. Available from: https://www.spandidos-publications.com/10.3892/mco.2016.840 7. Ersoz S, Turgutalp H, Cobanoglu U, Bektas D, Yaris N. Sialoblastoma in the parotid gland: A case report. Pediatrics International [Internet]. 2010 Aug [cited 2025 Mar 4];52(4):670–2. Available from: https://onlinelibrary.wiley.com/doi/10.1111/j.1442-200X.2010.03108.x 8. Dardick I, Daley TD, McComb RJ. Sialoblastoma in adults: distinction from adenoid cystic carcinoma. Oral Surgery Oral Medicine Oral Pathology Oral Radiology and Endodontology [Internet]. 2009 Nov 1 [cited 2025 Mar 4];109(1):109–16. Available from: https://www.oooojournal.net/article/S10792104(09)00533-2/abstract 9. M. Prigent, N. Teissier, M. Peuchmaur, M. El Maleh-Berges, P. Philippe-Chomette, P Cardin, et al. Sialoblastoma of salivary glands in children: Chemotherapy should be discussed as an alternative to mutilating surgery. International Journal of Pediatric Otorhinolaryngology [Internet]. 2010 Jun 8 [cited 2025 Mar 4];74(8):942–5. Available from: https://www.sciencedirect.com/science/article/abs/pii/S0165587610000480?via%3Dihub 10. Karaman E, Duman C, Cansız H, Yildiz I, Dervişoglu S, Ozdemir G. Sialoblastoma in cheek region: Report of a case. International Journal of Pediatric Otorhinolaryngology Extra [Internet]. 2010 Mar [cited 2025 Apr 14];5(2):47–9. Available from: https://www.sciencedirect.com/science/article/abs/pii/S1871404809000100 Information & Authors Information Version history V1 Version 1 15 July 2025 V2 Version 2 21 July 2025 Copyright This work is licensed under a Non Exclusive No Reuse License. Keywords pediatric pathology pediatric surgery sialadenectomy sialoblastoma submandibular gland Authors Affiliations Gabriela Martínez 0009-0008-3973-3144 [email protected] Department of Medical Research, PUCMM View all articles by this author Selene Venegas Pediatric Surgeon, Corominas Clinic View all articles by this author Carolina Sued Surgeon Cervical-oral-maxillofacial Surgery, Corominas Clinic View all articles by this author Rafael Estévez Pathologist, Corominas Clinic View all articles by this author Metrics & Citations Metrics Article Usage 258 views 79 downloads .FvxKWukQNSOunydq8rnd { width: 100px; } Citations Download citation Gabriela Martínez, Selene Venegas, Carolina Sued, et al. Submandibular Sialoblastoma in a Newborn: A Case Report and Surgical Approach. Authorea . 21 July 2025. DOI: https://doi.org/10.22541/au.175260127.76573557/v2 If you have the appropriate software installed, you can download article citation data to the citation manager of your choice. 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