Case Report
Non-Operative Treatment of Intracystic Papilloma in the Male Breast
Chung Heeseok1,2*, Park Jaewoo3 and Park Sunhee4
1Department of Surgery, Gwangju Veterans Hospital, South Korea
2Department of Surgery, Chosun University, South Korea
3Department of Pathology, Gwangju Veterans Hospital, South Korea
4Department of Clinical Pharmacy, Chosun University, South Korea
*Corresponding author: Chung Heeseok, Department of Surgery, Gwangju Veterans Hospital, Gwangju, South Korea
Published: 18 Jul, 2018
Cite this article as: Heeseok C, Jaewoo P, Sunhee P.
Non-Operative Treatment of Intracystic
Papilloma in the Male Breast. Clin Surg.
2018; 3: 2037.
Abstract
Intracystic Papilloma (ICP) in male breast is rare and only 13 cases of ICP in men have been reported worldwide so far. In all cases, local excision or mastectomy is performed immediately upon diagnosis for treatment of ICP. This case report describes a patient who was diagnosed with a benign cyst by core-needle biopsy and had received only non-operative management for 10 years. Then, this patient underwent local excision and diagnosed with ICP. The lump did not become malignant after 10 years from initial diagnosis. We suggest that immediate surgical excision may not always be necessary for ICP of the male breast after diagnosis by core-needle biopsy.
Introduction
Intracystic Papilloma (ICP) of the breast is one of the benign breast tumour in which a papillary
lesion grows inside a cyst. ICP is rare in male breasts, and only 13 cases of ICP in men have been
reported worldwide so far [1,2].
In a review of the literature on men with ICP, there was no predilection for age or the right or
left breast [1]. All patients had palpable cystic masses. The size of these lesions ranged from 2 cm to
10 cm. The duration of the disease before diagnosis was more than 1 year in 40% of the patients [2].
The clinical features of ICP in men are similar, including a palpable cystic mass, bloody or serous
nipple discharge, and pain. Clinically, there are no medical conditions associated with ICP in the
male breast. However, 2 cases raised the possibility of a relationship between ICP in men and longterm
therapy with phenothiazine, which has been known to increase the level of prolactin [2,3].
However, an explanation for this possible association is not clear yet.
Mammography or sonography is required to diagnose ICP and a definitive diagnosis can be
made by a sonographically-guided core needle biopsy [1]. Pathologically, ICP is a hyperplastic
polypoid lesion within the duct. The cyst of ICP is actually a cystically dilated duct containing the
papilloma. Therefore, there is no histopathologic difference between intraductal papilloma and ICP,
and the origin of its cyst is likely different from that of cysts that arise in the lobules [1].
In most cases, local excision or mastectomy is performed immediately upon diagnosis for
treatment of ICP [1,2,4,5]. Here, however, we report a case of ICP in an 83-year-old man who was
treated with aspiration, not surgery, for 10 years after diagnosis.
Case Presentation
An 83-year-old man visited our hospital with a lump in his left breast and nipple. He first
discovered the lump 10 years ago. Ultrasonography and mammography were performed when the
lump was originally found, and a definitive diagnosis of a benign cyst was made by core-needle
biopsy. The lump size increased gradually after the diagnosis and was reduced by aspirating the
cystic fluid. The aspiration treatment was repeated every time the cyst reappeared.
During his visit at our hospital, physical examination showed a 6 cm hard, movable lump below
the nipple in the left breast. There were no enlarged axillary lymph nodes or nipple retraction.
The patient had no record of medications that cause male breast enlargement, no known medical
conditions, and no pleural injury.
Lumpectomy was performed under local anaesthesia without additional diagnostic examinations
in our hospital because the patient had already been diagnosed with a
benign cyst. The excised specimen was a cystoma with a volume of 6
× 6 × 3 cm3 and the intracystic papillary lesion was 1.5 cm.
The cyst was identified as ICP by histopathology after surgery. In
our case, the epithelium lining the papillary fibrovascular fronds in
the lesion was mostly apocrine, containing large nuclei, nucleoli, and
abundant cytoplasm. The patient has shown no recurrence for 1 year
after the surgery.
Discussion
ICP in the male breast is very rare because of the rudimentary state
of the mammary gland [1]. Since Simpson and Barson first reported
ICP of the male breast in 1969, it has been reported in only13 other
patients [2,6].
Intracystic tumours can now be detected easily using new imaging
modalities, including ultrasonography, computed tomography and
magnetic resonance imaging. However, even with these imaging
modalities, diagnosis of these lesions is difficult except in cases of
intracystic carcinoma with invasive features on imaging [2].
Complex cystic breast masses have a possibility of being
malignant and therefore confirmative diagnosis is usually indicated
[7]. In order to make appropriate treatment decisions, ICP should
be distinguished from Intracystic Papillary Carcinoma (ICPC).
However, it is difficult to distinguish between benign and malignant
lesion preoperatively by imaging or cytologic examinations. In
particular, cytologic examination of specimens obtained by puncture
aspiration are frequently misleading because cellular atypia is very
low in the majority of ICPCs, and the floating cells in the cyst fluid
are easily denatured [8]. Therefore, histological diagnosis is required,
which means an excisional procedure. Alternatively, sonographically
guided core needle biopsy can be performed as a less invasive method
for differential diagnosis of ICP and ICPC when a solid component is
present in the cyst [1].
The natural history or treatment of ICP in male breasts is not
clearly defined. Therefore, the patients are being treated according
to the protocol for intraductal papilloma in women. In cases
of intraductal papilloma in women, surgery is recommended
immediately upon diagnosis based on a report saying that the risk
for breast cancer is elevated two-fold [4]. All previously reported
cases of men diagnosed with ICP have also received local excision or
mastectomy immediately once intraductal papilloma was diagnosed
[1,2,4,5]. However, it cannot be concluded that the incidence of
breast cancer in men with ICP is equal to that in women, because
there have been no studies about the relationship between breast
cancer incidence and ICP in men.
Navas, et al. reported the case of a patient who was diagnosed with
ICP based on the results from laboratory tests after local excision of
a lump that had existed for 6 years [5]. The current case had received
only conservative management without surgery for 10 years. Then,
our patient underwent local excisional surgery and was diagnosed
with ICP based on laboratory test results. In both cases, the lump did
not become malignant after 6 and 10 years, respectively, from the
initial appearance or diagnosis.
In conclusion, appropriate treatment of ICP is controversial. We
suggest that immediate surgical excision may not always be necessary
for ICP of the male breast after diagnosis by core-needle biopsy. Case
studies so far have only discussed the clinical features or pathogenesis
of ICP in men. However, we have focused on the natural progression
or treatment of ICP in men. We currently cannot come to any
definitive conclusions about its natural progression or treatment due
to the small number of cases of ICP in men that have been reported
to date. However, it is expected that accumulation of studies like this
will help to reveal the clinical features and treatment principles of ICP
in men in the future, and make it possible to analyse the differences in
ICP between men and women.
References
- Shim JH, Son EJ, Kim EK, Kwak JY, Jeong J, Hong SW. Benign intracystic papilloma of the male breast. J Ultrasound Med. 2008;27(9):1397-400.
- Yamamoto H, Okada Y, Taniguchi H, Handa R, Naoi Y, Oshima S, et al. Intracystic papilloma in the breast of a male given long-term phenothiazine therapy: A case report. Breast Cancer 2006;13(1):84-8.
- Sara AS, Gottfried MR. Benign papilloma of the male breast following chronic phenothiazine therapy. Am J Clin Pathol 1987; 87(5):649-50.
- Ra YM, Sohn JS, Kim KW, Lee JU, Park HD, Choi IS, et al. Clinicopathologic review of the intraductal papilloma of breast. J Breast Cancer 2010;13(1):31-6.
- Martorano Navas MD, Raya Povedano JL, Añorbe Mendivil E, Muñoz Hernandez A, Ramos Gonzalez A, Vilarrasa Andres A, et al. Intracystic papilloma in male breast: Ultrasonography and pneumocystography diagnosis. J Clin Ultrasound 1993;21(1):38-40.
- Simpson JS, Barson AJ. Breast tumours in infants and children: A 40-year review of cases at a children’s hospital. Can Med Assoc J. 1969;101(2):100-2.
- Berg WA, Campassi CI, Ioffe OB. Cystic lesions of the breast: Sonographic-pathologic correlation. Radiology 2003;227(1):183-91.
- Ahmet Dağ, Erdem Yüce, Tuba Kara. Intracyst papilloma in male breast. J Breast Health. 2012;8:40-2.