Małgorzata Dębska-Rutkowska, Piotr Kwast, Monika Jabłońska-Jesionowska, *Lidia Zawadzka-Głos
Diagnostic difficulties in salivary gland tumors in children
Trudności diagnostyczne guzów dużych gruczołów ślinowych u dzieci
Department of Pediatric Otolaryngology, Medical University of Warsaw, Poland
Head of Department: Associate Professor Lidia Zawadzka-Głos, MD, PhD
Wstęp. Guzy dużych gruczołów ślinowych, ze względu na rzadkość występowania oraz szerokie spektrum możliwych rozpoznań, stanowią wyzwanie dla lekarza. Badania obrazowe są kluczowym elementem procesu diagnostycznego, choć ich wyniki często nie dają możliwości postawienia ostatecznego rozpoznania.
Cel pracy. Celem pracy była ocena przydatności badań dodatkowych w diagnostyce guzów dużych gruczołów ślinowych u dzieci.
Materiał i metody. Dokonano analizy retrospektywnej danych medycznych dzieci ze zmianami guzowatymi ślinianki przyusznej lub podżuchwowej hospitalizowanych w Klinice Otolaryngologii Dziecięcej Warszawskiego Uniwersytetu Medycznego w latach 2012-2017.
Wyniki. U 8 dziewczynek i 7 chłopców w wieku od 4 miesięcy do 17 lat rozpoznano 6 guzów mieszanych, 3 naczyniaki krwionośne oraz po jednym przypadku naczyniaka limfatycznego, mięsaka prążkowanokomórkowego i raka śluzowo-naskórkowego. U wszystkich pacjentów wykonano badanie ultrasonograficzne (USG) ślinianek oraz tomografię komputerową (TK) i/lub rezonans magnetyczny (MRI) twarzoczaszki. Na podstawie USG rozpoznano większość (66%) guzów mieszanych. USG oraz TK lub MRI z kontrastem pozwoliły postawić rozpoznanie u wszystkich 4 pacjentów z naczyniakami. U 2 pacjentów na podstawie badania histopatologicznego rozpoznano nowotwór złośliwy ślinianki przyusznej.
Wnioski. Podstawowym badaniem diagnostycznym guzów dużych gruczołów ślinowych pozostaje USG, uzupełniane w razie konieczności o TK lub MRI. Badania obrazowe w niektórych przypadkach nie pozwalają na postawienie ostatecznej diagnozy. Rozpoznanie ostateczne guzów ślinianek (oprócz naczyniaków) ustala się na podstawie badania histopatologicznego. Najczęstszym guzem ślinianek u dzieci starszych jest guz mieszany, a u niemowląt – naczyniak. Należy pamiętać o możliwości wystąpienia u dzieci bardzo rzadkich guzów ślinianek, takich jak mięsak lub rak.
Introduction. Tumors of major salivary glands, due to their low incidence and the wide spectrum of possible diagnoses, are a challenge for physicians. Imaging studies are the key elements of the diagnostic process, although their results often do not provide a definitive diagnosis.
Aim. The aim of the study was to assess the usefulness of additional tests in the diagnosis of tumors of major salivary glands in children.
Material and Methods. A retrospective analysis of medical data of children with tumors of parotid or submandibular salivary gland hospitalized in the Department of Pediatric Otolaryngology of Medical University of Warsaw from the years 2012-2017 was performed.
Results. In 8 girls and 7 boys aged between 4 months and 17 years, the following diagnoses were established: 6 mixed tumors, 3 hemangiomas, 1 lymphangioma, 1 rhabdomyosarcoma, and 1 mucoepidermoid carcinoma. In all of the patients, ultrasound (US) of salivary gland, computed tomography (CT) and/or magnetic resonance imaging (MRI) of the head were performed. US examination enabled to diagnose the majority (66%) of mixed tumors. US and CT or MRI with contrast enabled to recognize angiomas in all 4 patients with this lesion. In 2 patients, malignant neoplasm was diagnosed based on the histological examination.
Conclusions. US remains the primary diagnostic test in patients with tumors of major salivary gland. When necessary, it can be supplemented with CT or MRI. In some cases, imaging studies do not enable to establish a definite diagnosis. The definite diagnosis of salivary gland tumors is established based on the results of the pathological examination (except for angiomas). The most common salivary gland tumor in older children is mixed tumor, and in infants – angioma. It must be remembered that very rare salivary gland tumors, such as sarcomas and carcinomas, may also occur in children.
Salivary gland tumors are not common in pediatric patients (1-6). Only 3-5% of all salivary gland tumors occur at the developmental age (1, 7). Salivary gland tumors constitute 8% of pediatric head and neck tumors (8). All the histological types of salivary gland tumors that are reported in adult patients also occur in children, but their incidence is different (1, 5, 7). According to the World Health Organization (WHO) classification from 2005, almost 50 histological types of salivary gland tumors can be distinguished (9). Malignant salivary gland tumors occur relatively more often in children than in adults and are more common in younger children (4, 5). Malignant tumors occur more frequently in minor salivary glands, and tumors of major salivary glands are more likely to be benign (6).
In most of the studies, ca. 60-70% of salivary gland tumors are benign (1), although some studies report a 1:1 ratio of malignant to benign lesions (3, 10). The most common benign tumors include: pleomorphic adenoma, also known as mixed tumor (łac. adenoma pleomorphum, tumor mixtus), hemangioma, and lymphangioma. Mucous retention cysts, usually localized in minor salivary glands, are usually not included in the statistical analyses of salivary gland tumors, although they represent 2-5% of the findings in the parotid gland (11).
The aim of the study was to assess the usefulness of additional tests in the diagnosis of tumors of major salivary glands in children.
Material and Methods
Retrospective analysis of medical data of 15 children with tumors of parotid and submandibular salivary glands hospitalized in the Department of Pediatric Otolaryngology of Medical University of Warsaw in the years 2012-2017 was performed. Medical history of the patients, along with additional tests, including imaging studies, were analyzed.
During 5 years between October 2012 and July 2017, 15 children with tumors of parotid and submandibular glands were hospitalized in the Department of Pediatric Otolaryngology, including 8 girls and 7 boys aged between 4 months and 17 years.
In most cases, salivary gland tumor was accidentally identified by the child’s physician or their parents. The time between the first identification of the tumor and the definite diagnosis varied from ca. 1 month (in case of hemangiomas) to a few years (in case of mixed tumors). The tumor was asymptomatic in 13 children, with no pain or facial nerve palsy. Two patients developed inflammation of the hemangioma. These children complained of pain, fever was also observed. Antibiotic treatment was required.
Tumor size varied from 15x18x11 mm to 37x34x24 mm. Palpatively, solid tumors were round, cohesive, hard. Hemangiomas that were currently not inflamed were soft, whereas inflamed hemangiomas were hard, with altered and warmed skin above the tumor.
All patients underwent basic blood tests (with no significant abnormal findings), ultrasound (US) of salivary gland and computed tomography (CT) and/or magnetic resonance imaging (MRI) of the head in case of diagnostic difficulties. The tumors were localized in parotid gland in 14 cases (7 on the right and 7 on the left) and in the right submandibular gland in 1 case.
Definite diagnoses included 6 cases of mixed tumors, 3 hemangiomas, 1 lymphangioma, 1 rhabdomyosarcoma, and 1 mucoepidermoid carcinoma. In 3 cases, pathological examination did not confirm the neoplastic nature of the lesion. These cases included 1 case of enlarged intraglandular lymph nodes, 1 case of fibrous tissue with necrosis and hemorrhages, and 1 case of non-specific inflammatory lesions.
In case of mixed tumor (6 patients: 4 girls and 2 boys aged between 11 and 17 years of age, mean age: 14.3 years), findings in ultrasound were described as solid, well demarcated, polycystic, although in 2 cases liquid content was found and cysts were suspected. In these patients, computed tomography was also performed, and it revealed a limited heterogenous tumor with low contrast gain. One patient underwent MRI. US examination enabled to diagnose the majority (66%) of mixed tumors. The patients were qualified for surgical treatment.
In patients with hemangiomas of salivary glands (3 girls aged 4 months, 7 months, and 7 years), ultrasound examination revealed a well-vascularized lesion or a lesion with visible blood flow, and in a patient with lymphangioma (12-year-old boy) – with no visible flow. The diagnostics was supplemented with CT or MRI with contrast, which demonstrated contrast enhancement in case of hemangiomas. These imaging studies enabled to establish diagnosis in all 4 patients. Biopsy of the lesions was not performed. Children with hemangiomas were treated conservatively – propranolol was used with good effect. The child with lymphangioma remains in the outpatient care, where the lesion is observed.
Two patients were diagnosed with malignant tumor of the parotid gland. In the case of a 6-year-old boy with rhabdomyosarcoma (lat. rhabdomyosarcoma embryonale), ultrasound examination revealed a well-vascularized solid polycyclic lesion measuring 20x18x14 mm. After 2 weeks, MRI examination revealed a mass in the superficial and deep lobe of parotid gland measuring 53x45x41 mm, reaching parapharyngeal space, modelling the lumen of nasopharynx, and compressing internal jugular vein. The invasion of neighboring structures was not observed. In the case of an 8-year-old boy with mucoepidermoid carcinoma (lat. carcinoma mucoepidermale), ultrasound examination revealed an oval, heterogenous, hypoechogenic tumor measuring 17x13x14 mm. Ultrasound-guided fine needle aspiration (FNA) was performed, and the results were identical with subsequent histopathological examination. No further imaging studies were performed. In both cases, the patients were qualified for surgical treatment, and then referred for further oncological treatment.
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