Repeated recurrence of bilateral gigantomastia after subcutaneous mastectomy caused by tumoral pseudoangiomatous stromal hyperplasia: a case report and review of literature

Abstract

Pseudoangiomatous stromal hyperplasia (PASH) is an uncommon benign proliferative mesenchymal lesion of the breast with a hormonal-sensitive nature. Various manifestations of PASH, ranging from an incidental microscopic finding in a tissue biopsy to a large palpable mass or bilateral gigantomastia, have been described. For tumoral PASH, surgical excision is indicated for a growing and symptomatic mass with a small chance of recurrence. A recurrence of bilateral gigantomastia after surgical excision or reduction mammoplasty is not common but has been occasionally reported, leading to further mastectomy. Repeated recurrence of bilateral gigantomastia is extremely rare. Herein, we report a case of a 13-year-old girl who presented with the third recurrence of bilateral gigantomastia caused by tumoral PASH, after undergoing bilateral reduction mammoplasty, and later subcutaneous mastectomy. Precocious puberty occurred early in this child at the age of 9 years, which may have been a factor unmasking PASH at this young age. The incomplete removal of the PASH could also have been a recurrence risk in our case as extended masses underneath the pectoralis muscle were later identified on the MRI study. This highlights the advantage of preoperative imaging in cases with a very large tumoral PASH in order to maximize the chance of complete tumor removal.

Bilateral gigantomastia due to benign breast tumors: a case series and brief review focusing on bilateral diffuse pseudoangiomatous stromal hyperplasia

“…Gigantomastia is a rare condition characterized by excessive hypertrophy of the connective tissue of the breast. While the exact criteria vary across studies, gigantomastia is typically defined as breast tissue comprising more than 3% of total body weight, excessive breast growth exceeding 1.5 kg per breast, or overly large breasts requiring reduction by 1-2 kg [1]. This condition can cause both physical and emotional distress, making surgical intervention cru-cial for improving patient quality of life.…”

“…mastia, as preoperative imaging examinations sometimes reveal no definitive lesions [1,2]. While substantial breast size can lead to meaningful psychological and physical distress, securing national health insurance coverage for breast reduction surgery in South Korea is challenging.…”