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Aggressive Digital Papillary Adenocarcinoma With Multiple Organ Metastases: A Case Report and Review of the Literature

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ABSTRACT

Aggressive digital papillary adenocarcinoma (ADPA) is a rare sweat gland neoplasm with a high recurrence rate and metastatic potential. In this study, the authors describe a case that originally appeared to benign spiradenoma, but took an ominous course eventually resulting in the diagnosis of ADPA. A 73-year-old woman developed a gradually growing nodule on the second toe of her left foot, which she had first noticed 4 years previously. An excisional biopsy was performed followed by histological examination. The authors initially considered the tumor to be a benign spiradenoma and did not perform reexcision. However, she experienced local recurrence 24 months later, and multiple pulmonary metastasis 31 months later. On histological examination, both the primary and locally recurrent tumors were found to be composed of discrete and well-circumscribed solid nodules, lacking cystic space. All tumors (the primary tumor, locally recurrent tumor, and lung metastases) presented with a pattern of fused back-to-back tubular structures and myoepithelial differentiation confirmed by immunohistochemical examination. On the basis of these findings, the authors finally diagnosed ADPA with multiple pulmonary metastases. The patient underwent chemotherapy, but died of disease 49 months later. This case highlights the importance of high clinical suspicion of ADPA when digital lesions present.

No MeSH data available.


Related in: MedlinePlus

A, Clinical appearance of the red-brown nodule on the second toe of the patient's left foot. B, Well-circumscribed tumor nodule in the dermis containing tubular structures and hemorrhage (hematoxylin and eosin ×20). C, Closely aggregated back-to-back tubular structures lined by cuboidal or columnar epithelium (hematoxylin and eosin ×100). D, Some tubules displayed evidence of decapitation secretion. Neoplastic cells presented with atypical nuclei and mitoses (hematoxylin and eosin ×400). E, Papillary structures formed by heaped-up epithelium with fibrovascular cores and necrosis surrounding hemorrhages (hematoxylin and eosin ×100). F, Tumor cells, with the exception of those lining the tubular structures, were positive for p63 expression (×100).
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Figure 1: A, Clinical appearance of the red-brown nodule on the second toe of the patient's left foot. B, Well-circumscribed tumor nodule in the dermis containing tubular structures and hemorrhage (hematoxylin and eosin ×20). C, Closely aggregated back-to-back tubular structures lined by cuboidal or columnar epithelium (hematoxylin and eosin ×100). D, Some tubules displayed evidence of decapitation secretion. Neoplastic cells presented with atypical nuclei and mitoses (hematoxylin and eosin ×400). E, Papillary structures formed by heaped-up epithelium with fibrovascular cores and necrosis surrounding hemorrhages (hematoxylin and eosin ×100). F, Tumor cells, with the exception of those lining the tubular structures, were positive for p63 expression (×100).

Mentions: A 73-year-old woman developed a gradually growing nodule on the second toe of her left foot, which she had first noticed 4 years previously. She denied any preexisting lesions at the site and her medical and family histories were unremarkable. On physical examination, the tumor was found to be a 3 mm red-brown nodule with tenderness, which presented on the second toe of the patient's left foot (Fig. 1A). An excisional biopsy was performed followed by histological examination. Hematoxylin and eosin (H&E) staining revealed a well-circumscribed solid tumor in the dermis containing tubular structures and hemorrhages (Fig. 1B). Higher magnification revealed that the tumor presented with closely aggregated back-to-back tubular structures lined by cuboidal or columnar epithelium (Fig. 1C). Some tubules showed evidence of decapitation secretion, and the neoplastic cells presented with nuclear atypicality and numerous mitoses (45 mitoses per 10 high power fields) (Fig. 1D). The tumor also had papillary structures formed by heaped-up epithelium with fibrovascular cores, and necrosis surrounding hemorrhages (Fig. 1E). There was no microscopic finding of invasion into the surrounding tissue. We initially considered the tumor to be a benign spiradenoma as the overall features resembled spiradenoma with apparent tubules and hemorrhages. Moreover, because histological examination did not reveal an apparent positive margin, we did not perform reexcision.


Aggressive Digital Papillary Adenocarcinoma With Multiple Organ Metastases: A Case Report and Review of the Literature
A, Clinical appearance of the red-brown nodule on the second toe of the patient's left foot. B, Well-circumscribed tumor nodule in the dermis containing tubular structures and hemorrhage (hematoxylin and eosin ×20). C, Closely aggregated back-to-back tubular structures lined by cuboidal or columnar epithelium (hematoxylin and eosin ×100). D, Some tubules displayed evidence of decapitation secretion. Neoplastic cells presented with atypical nuclei and mitoses (hematoxylin and eosin ×400). E, Papillary structures formed by heaped-up epithelium with fibrovascular cores and necrosis surrounding hemorrhages (hematoxylin and eosin ×100). F, Tumor cells, with the exception of those lining the tubular structures, were positive for p63 expression (×100).
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Related In: Results  -  Collection

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Show All Figures
getmorefigures.php?uid=PMC5120768&req=5

Figure 1: A, Clinical appearance of the red-brown nodule on the second toe of the patient's left foot. B, Well-circumscribed tumor nodule in the dermis containing tubular structures and hemorrhage (hematoxylin and eosin ×20). C, Closely aggregated back-to-back tubular structures lined by cuboidal or columnar epithelium (hematoxylin and eosin ×100). D, Some tubules displayed evidence of decapitation secretion. Neoplastic cells presented with atypical nuclei and mitoses (hematoxylin and eosin ×400). E, Papillary structures formed by heaped-up epithelium with fibrovascular cores and necrosis surrounding hemorrhages (hematoxylin and eosin ×100). F, Tumor cells, with the exception of those lining the tubular structures, were positive for p63 expression (×100).
Mentions: A 73-year-old woman developed a gradually growing nodule on the second toe of her left foot, which she had first noticed 4 years previously. She denied any preexisting lesions at the site and her medical and family histories were unremarkable. On physical examination, the tumor was found to be a 3 mm red-brown nodule with tenderness, which presented on the second toe of the patient's left foot (Fig. 1A). An excisional biopsy was performed followed by histological examination. Hematoxylin and eosin (H&E) staining revealed a well-circumscribed solid tumor in the dermis containing tubular structures and hemorrhages (Fig. 1B). Higher magnification revealed that the tumor presented with closely aggregated back-to-back tubular structures lined by cuboidal or columnar epithelium (Fig. 1C). Some tubules showed evidence of decapitation secretion, and the neoplastic cells presented with nuclear atypicality and numerous mitoses (45 mitoses per 10 high power fields) (Fig. 1D). The tumor also had papillary structures formed by heaped-up epithelium with fibrovascular cores, and necrosis surrounding hemorrhages (Fig. 1E). There was no microscopic finding of invasion into the surrounding tissue. We initially considered the tumor to be a benign spiradenoma as the overall features resembled spiradenoma with apparent tubules and hemorrhages. Moreover, because histological examination did not reveal an apparent positive margin, we did not perform reexcision.

View Article: PubMed Central - PubMed

ABSTRACT

Aggressive digital papillary adenocarcinoma (ADPA) is a rare sweat gland neoplasm with a high recurrence rate and metastatic potential. In this study, the authors describe a case that originally appeared to benign spiradenoma, but took an ominous course eventually resulting in the diagnosis of ADPA. A 73-year-old woman developed a gradually growing nodule on the second toe of her left foot, which she had first noticed 4 years previously. An excisional biopsy was performed followed by histological examination. The authors initially considered the tumor to be a benign spiradenoma and did not perform reexcision. However, she experienced local recurrence 24 months later, and multiple pulmonary metastasis 31 months later. On histological examination, both the primary and locally recurrent tumors were found to be composed of discrete and well-circumscribed solid nodules, lacking cystic space. All tumors (the primary tumor, locally recurrent tumor, and lung metastases) presented with a pattern of fused back-to-back tubular structures and myoepithelial differentiation confirmed by immunohistochemical examination. On the basis of these findings, the authors finally diagnosed ADPA with multiple pulmonary metastases. The patient underwent chemotherapy, but died of disease 49 months later. This case highlights the importance of high clinical suspicion of ADPA when digital lesions present.

No MeSH data available.


Related in: MedlinePlus