Papillary Adenocarcinoma of Minor Salivary Gland Origin in a Child

Abstract

\s=b\A papillary adenocarcinoma with metastasis occurred in a 5-year-old child. The neoplasm arose on the anterolateral sublingual aspect of the tongue and metastasized to a submandibular lymph node. Histologically, the tumor contained broad glandular papillary projections. The tumor cells were cuboidal and had vesicular, "ground glass" nuclei. Colloidlike material was found within the stroma and lumen of the glands. To determine whether the papillary adenocarcinoma was of minor salivary gland or ectopic thyroid in origin, ultrastructural, histochemical, and immunohistochemical techniques were applied to make this clinically important distinction. Ultrastructural and immunohistochemical studies did not indicate the site of origin. Histochemical methods, however, demonstrated the colloidlike material to be sulfated mucopolysaccharide, which was nonreducible by ferric ferrocyanide. These histochemical properties and the anterolateral location identified the tumor as salivary gland in origin.Tumors of minor salivary glands and mucous membranes of the oral cavity and upper respiratory tract have a diversity of histopathologic appearances and a variety of clinical courses. The vast majority of these tumors occurs in older adults.The present case of papillary adeno¬ carcinoma is a rare variety of carcinoma of the minor salivary gland, and its occurrence in a child of 5 years is unique. Because of its rarity, histolog¬ ie features, and location on the tongue, it presented a problem in dif¬ ferentiating between a salivary gland tumor and the more common papil¬ lary carcinoma of ectopie thyroid gland. The diagnostic criteria for Fig 1.-Sublingual tumor was composed of broad papillary projections with sparsely cellular collagenous stroma. Papillary projections formed almost solid tumor mass (hematoxylin-eosin, original magnification X40).

Cribriform adenocarcinoma of the tongue: a hitherto unrecognized type of adenocarcinoma characteristically occurring in the tongue

“…reported a tumour of the tongue in a 59‐year old man which was called ‘tubular adenocarcinoma’, 20 and Crocker et al . described a 5‐year‐old boy with an adenocarcinoma, which they called ‘papillary adenocarcinoma of minor salivary gland’ 21 . Even allowing for the difficulties of proper assessment of figures in reprints, there are many similarities to the microscopic appearance of our cases.…”

“…Even allowing for the difficulties of proper assessment of figures in reprints, there are many similarities to the microscopic appearance of our cases. Particularly reminiscent were the pallor and ground glass quality of the nuclei as seen in both tumours, the solid and cribriform arrangement in the first tumour 20 and the papillary appearance resulting from tumour cell detachment from the stroma as seen in the other report 21 …”

Newly described salivary gland tumors

“…107,108 Yajima et al reported a tumor of the tongue in a 59-year-old man, which was called 'tubular adenocarcinoma', 108 and Crocker et al described a 5-yearold boy with an adenocarcinoma, which the authors designated 'papillary adenocarcinoma of minor salivary gland'. 109 Particularly reminiscent are the pallor and ground glass quality of the nuclei as seen in both tumors, the solid and cribriform arrangement in the first tumor 108 and the papillary appearance resulting from tumor cell detachment from the stroma as seen in the other report. 109 As a consequence, therefore, we strongly suspect that if cases of CAMSG were excluded, then the metastatic rate of true PLGA would be exceptional.…”

“…109 Particularly reminiscent are the pallor and ground glass quality of the nuclei as seen in both tumors, the solid and cribriform arrangement in the first tumor 108 and the papillary appearance resulting from tumor cell detachment from the stroma as seen in the other report. 109 As a consequence, therefore, we strongly suspect that if cases of CAMSG were excluded, then the metastatic rate of true PLGA would be exceptional. In contrast, the patients with CAMSG had lymph node metastases in 19/31 cases already at the time of the presentation of the primary tumor in the published series.…”

Cribriform adenocarcinoma of minor salivary gland: A report of two cases with an emphasis on cytology

“…Moreover, these are not the only published reports where CAMSG is called something else. Likely the first published report that represents CAMSG in the literature came from Crocker et al, who reported on a young child with what they called “papillary adenocarcinoma of minor salivary gland.” In 1989, Yajima et al reported a lesion in the tongue of a middle aged man that was called tubular carcinoma …”