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

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

A, Clinical appearance of the red-purple fluid-filled nodule that appeared adjacent to the operative scar tissue resulting from the removal of the previous tumor. B, The tumor consisted of discrete nodules that were well circumscribed and, as was the case with the previous specimen, the tumor contained hemorrhages, although there were fewer tubular structures than in the previous specimen (hematoxylin and eosin ×20). C, Neoplastic cells exhibit nuclear atypicality and an increased number of mitoses compared with the previous specimen (hematoxylin and eosin ×400). D, Distinctive squamous foci were present (hematoxylin and eosin ×400). E, p63 staining showing that tumor cells, with the exception of those lining the tubular structures, were positive for p63 expression (×100).
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Figure 2: A, Clinical appearance of the red-purple fluid-filled nodule that appeared adjacent to the operative scar tissue resulting from the removal of the previous tumor. B, The tumor consisted of discrete nodules that were well circumscribed and, as was the case with the previous specimen, the tumor contained hemorrhages, although there were fewer tubular structures than in the previous specimen (hematoxylin and eosin ×20). C, Neoplastic cells exhibit nuclear atypicality and an increased number of mitoses compared with the previous specimen (hematoxylin and eosin ×400). D, Distinctive squamous foci were present (hematoxylin and eosin ×400). E, p63 staining showing that tumor cells, with the exception of those lining the tubular structures, were positive for p63 expression (×100).

Mentions: Two years later, the small nodule had regrown and physical examination revealed a 4.5 mm red-purple fluid-filled nodule located adjacent to the previous postoperative scar (Fig. 2A). There was no lymphadenopathy or organomegaly. Histological evaluation with H&E staining revealed a multilobular, solid tumor that contained fewer tubular structures than in the previous specimen (Fig. 2B). Tubular structures were mainly found at margins of the tumor and the central region consisted of sheets of neoplastic cell proliferation. Within the overlying epidermis was a blister containing red blood cells. As was the case with the previous specimen, the tumor contained hemorrhages. Higher magnification revealed that neoplastic cells presented with nuclear atypicality and increased mitoses (62 mitoses per 10 high power fields) (Fig. 2C). Necrosis and stromal hyalinization were presented focally. In addition, the tumor showed foci of squamous differentiation (Fig. 2D). There was no microscopic finding of invasion into the surrounding tissue. Although we considered the tumor to be a malignant sweat gland neoplasm based on the above findings, we took a wait-and-see approach because of the free margin in the histological preparation and the lack of perineural or vascular invasion.


Aggressive Digital Papillary Adenocarcinoma With Multiple Organ Metastases: A Case Report and Review of the Literature
A, Clinical appearance of the red-purple fluid-filled nodule that appeared adjacent to the operative scar tissue resulting from the removal of the previous tumor. B, The tumor consisted of discrete nodules that were well circumscribed and, as was the case with the previous specimen, the tumor contained hemorrhages, although there were fewer tubular structures than in the previous specimen (hematoxylin and eosin ×20). C, Neoplastic cells exhibit nuclear atypicality and an increased number of mitoses compared with the previous specimen (hematoxylin and eosin ×400). D, Distinctive squamous foci were present (hematoxylin and eosin ×400). E, p63 staining showing that tumor cells, with the exception of those lining the tubular structures, were positive for p63 expression (×100).
© Copyright Policy - open-access
Related In: Results  -  Collection

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

Figure 2: A, Clinical appearance of the red-purple fluid-filled nodule that appeared adjacent to the operative scar tissue resulting from the removal of the previous tumor. B, The tumor consisted of discrete nodules that were well circumscribed and, as was the case with the previous specimen, the tumor contained hemorrhages, although there were fewer tubular structures than in the previous specimen (hematoxylin and eosin ×20). C, Neoplastic cells exhibit nuclear atypicality and an increased number of mitoses compared with the previous specimen (hematoxylin and eosin ×400). D, Distinctive squamous foci were present (hematoxylin and eosin ×400). E, p63 staining showing that tumor cells, with the exception of those lining the tubular structures, were positive for p63 expression (×100).
Mentions: Two years later, the small nodule had regrown and physical examination revealed a 4.5 mm red-purple fluid-filled nodule located adjacent to the previous postoperative scar (Fig. 2A). There was no lymphadenopathy or organomegaly. Histological evaluation with H&E staining revealed a multilobular, solid tumor that contained fewer tubular structures than in the previous specimen (Fig. 2B). Tubular structures were mainly found at margins of the tumor and the central region consisted of sheets of neoplastic cell proliferation. Within the overlying epidermis was a blister containing red blood cells. As was the case with the previous specimen, the tumor contained hemorrhages. Higher magnification revealed that neoplastic cells presented with nuclear atypicality and increased mitoses (62 mitoses per 10 high power fields) (Fig. 2C). Necrosis and stromal hyalinization were presented focally. In addition, the tumor showed foci of squamous differentiation (Fig. 2D). There was no microscopic finding of invasion into the surrounding tissue. Although we considered the tumor to be a malignant sweat gland neoplasm based on the above findings, we took a wait-and-see approach because of the free margin in the histological preparation and the lack of perineural or vascular invasion.

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