A rare cause of unilateral breast swelling in a male infant caused by fibrous hamartoma of infancy combined with pseudoangiomatous stromal hyperplasia

Abstract

We report a case of a male infant who underwent resection of a unilateral breast mass with a histopathological diagnosis of a fibrous hamartoma of infancy (FHI) combined with a pseudoangiomatous stromal hyperplasia (PASH). Breast lumps are uncommon in infants and children, especially in boys. FHI and PASH are very rare causes of breast lumps, especially in infants. To our knowledge, this is the first report of a combination of both pathologies in 1 lesion in the breast of an infant.

Pseudoangiomatous stromal hyperplasia in a healthy young adult male

“…This is further supported by the finding of progesterone, estrogen, and androgen receptor positivity on immunohistochemical testing. Although PASH occurs predominantly in females, 7 cases have also been reported in the male population [4] , [5] , [6] , [7] , [8] , [9] , [10] . Generally, males who develop this condition have some extent of underlying gynecomastia, indicating that increased breast tissue and hormonal imbalance are important risk factors for disease progression [8] .…”

“…Similar to the clinical and imaging features of fibroadenoma, a PASH tumor is described as a single, discrete, well-demarcated mass that ranges from solid to elastic in consistency. The rate of growth and the size of PASH vary widely from one case to another, ranging from microscopic foci to lesions that measure up to 18 cm in diameter [7] . Histologically, a PASH tumor may resemble the histological presentation of a low-grade angiosarcoma or a phyllodes tumor, largely due to the slit-like spaces lined by outstretched spindle cells through the acellular mammary stroma [ 12 , 13 ].…”

“…However, the development of a tumorous form is much more rare [3] . The development of this tumor type in men is even more rare, with only 7 cases previously reported in the English literature [4] , [5] , [6] , [7] , [8] , [9] , [10] . Herein, we describe the rare presentation of a young adult male patient with a large, rapidly growing unilateral PASH tumor.…”

Pseudoangiomatous stromal hyperplasia of thebreast: a rare condition – from diagnosis to treatment

“…PASH predominantly affects women in the pre or perimenopause, and those in the post-menopause, especially the ones under hormone replacement therapy 3,4,10 . It can also more rarely affect people of the male sex and individuals in childhood [14][15][16] . When comparing the clinical presentation among women in the post-menopausal phase with those who have not yet undergone this physiological event, the lesions are usually larger in women in pre and perimenopause 5 .…”

“…In general, on imaging studies, the lesions present characteristics of benignity. Mammography, ultrasonography (especially in cases of inconclusive mammography and in people of an earlier age) and magnetic resonance imaging (which is not routinely used, but can help with lesion assessment and surgical planning) can be used as diagnostic tools 16 .…”