Content uploaded by Dina Antina Schott
Author content
All content in this area was uploaded by Dina Antina Schott
Content may be subject to copyright.
Available via license: CC BY-NC 4.0
Content may be subject to copyright.
CASE REPORT
Prepubertal unilateral gynecomastia: a report of two cases
Inge A. Hoevenaren &Dina Antina Schott &
Barto J. Otten &Henriette C. Kroese-Deutman
Received: 18 March 2010 / Accepted: 17 June 2010 / Published online: 27 July 2010
#The Author(s) 2010. This article is published with open access at Springerlink.com
Abstract
Background Gynecomastia is defined as the presence of
excessive breast tissue in males, which can appear
unilateral or bilateral. Bilateral gynecomastia is frequently
found in the neonatal period, early in puberty, and with
increasing age. Prepubertal unilateral gynecomastia in the
absence of endocrine abnormalities is extremely rare, with
only a few cases in literature.
Methods We report the cases of two otherwise healthy boys
of 8 and 11 years old with unilateral breast masses. No
abnormalities were found on ultrasonography and all
endocrine parameters were within normal limits. Treatment
consisted of peripheral liposuction followed by subcutaneous
partial resection of the gland, conducted through an
infra-areolar incision.
Results Microscopy of the subcutaneous mastectomy spec-
imen revealed gynecomastia without signs of malignancy.
Postoperative course of both patients was uncomplicated,
with no signs of recurrence of breast tissue.
Conclusions Atypical presentations of gynecomastia are often
not recognized, with little attention to breast development
in prepubertal non-obese children. Since prepubertal
gynecomastia could be a sign of possible underlying
diseases, a thorough examination and further research is
recommended. If there is no causal treatment, surgical
resection is the therapy of first choice. Peripheral liposuction
and surgical resection of the gland tissue are the mainstay of
treatment. In summary, we describe two cases of prepubertal
unilateral gynecomastia with a normal endocrine workup.
Furtherresearchisneededtoestablishthepathophysiologic
mechanisms of prepubertal gynecomastia, since underlying
etiology in most cases remains unclear.
Keywords Unilateral gynecomastia .Prepub erty .Etiology .
Surgery
Introduction
Gynecomastia is characterized by the presence of unilateral or
bilateral breast tissue in males. This benign condition accounts
for 60% of all disorders of the male breast [1,2]. While
bilateral gynecomastia is common in the neonatal period, early
in puberty, and with increasing age, prepubertal unilateral
gynecomastia is a rare condition, with only a few cases in
literature [3]. We report a unique clinical presentation of two
boys with prepubertal onset of unilateral gynecomastia and
discuss the etiology, workup, and therapeutic intervention.
Case reports
Patient 1
An 8-year-old boy presented with a mass beneath his right
breast, slowly increasing in size over the past 8 months.
I. A. Hoevenaren (*):H. C. Kroese-Deutman
Department of Plastic, Reconstructive and Hand Surgery,
Radboud University Nijmegen Medical Centre,
P.O. Box 9101, 6500 HB Nijmegen, The Netherlands
e-mail: I.Hoevenaren@anat.umcn.nl
D. A. Schott :B. J. Otten
Department of Endocrinology,
Radboud University Nijmegen Medical Centre,
Nijmegen, The Netherlands
Present Address:
I. A. Hoevenaren
Department of Anatomy and Embryology,
Radboud University Nijmegen Medical Centre,
Nijmegen, The Netherlands
Eur J Plast Surg (2011) 34:395–398
DOI 10.1007/s00238-010-0469-6
The patient complained of tenderness of the breast mass
and of being teased extensively for his enlarged breast.
Family history was negative for breast malignancies, but
positive for prepubertal bilateral gynecomastia in the father,
as well as in two of his brothers. They underwent surgical
excision at about age seven without evidence of recurrence
afterwards. Past medical history revealed attention deficit
hyperactivity disorder (ADHD), for which the patient was
treated with Methylphenidate 30 mg/day and Melatonin
5 mg/day. No other drugs or dermal applications were used.
Physical examination showed a healthy-appearing boy,
136 cm tall (SDS 0), without accelerated growth velocity,
weighing 30 kg (SDS 0) with a body mass index (BMI) of
16.2 (SDS 1). Palpation of the right breast revealed a firm
mobile mass measuring approximately 8 cm in diameter,
with tenderness on deep palpation, Tanner stage III [4]. The
right nipple−areola complex was also enlarged, measuring
1.5 cm compared to 1 cm on the left. There was no history
or sign of galactorrhea and there were no physical findings
indicating onset of puberty. A summary of all endocrine
parameters tested with corresponding results is given in
Table 1. All parameters were found to be within normal
limits. Ultrasound examination of the right breast showed
glandular tissue without cysts or signs of malignancy. Surgery
(conducted by HKD and IAH) under general anesthesia
consisted of peripheral liposuction (with the 2 mm power-
assisted lipoplasty (PAL) liposuction system) and subcutane-
ous partial resection of the gland, conducted through an infra-
areolar incision. The dissected gland specimen measured
2.5×1.6×0.5 cm and weighed 15 g. The resulting
depression was reconstructed by leaving part of the
glandular tissue in situ and repositioning it, in order to
attain a symmetrical result. Pathologic examination
revealed normal glandular breast tissue, without evidence
of malignancy. Microscopy of the subcutaneous mastecto-
my specimen showed fibrotic breast tissue with mammary
ducts with epithelial hyperplasia, periductal cellular stroma,
and stroma hyperplasia. The concluding diagnosis was
florid gynecomastia. Recovery from the surgery was
uneventful. At a postoperative clinical evaluation 8 months
later, no signs of recent breast development were seen. The
patient is currently planned for a minor correction under
local anesthesia of a minimal residual areolar swelling.
Patient 2
An 11-year-old boy presented with a 12-month history of
left breast enlargement. The patient also complained of
tenderness of the breast mass and of great inconvenience in
everyday life. In this case, there was no family history of
breast malignancies or gynecomastia. No drugs or dermal
applications were used. Physical examination showed a
healthy-appearing lively boy. The patient was 138 cm tall
(SDS −1) without accelerated growth velocity, weighing
32 kg (SDS 0) with a BMI of 16.8 (SDS 0). Palpation of the
left breast revealed a firm mass measuring approximately
5 cm in diameter, with tenderness on deep palpation,
Tanner stage III. There was a pronounced asymmetry of the
breast, with a normal-appearing right breast (Fig. 1a). No
history or sign of galactorrhea existed, and there were no
physical findings indicating onset of puberty. A summary
of all endocrine parameters tested with corresponding
results is given in Table 1. All parameters were found to
be within normal limits. Ultrasound examination of the
right breast showed retro-aureolar glandular tissue with
normal aspects and swelling containing subcutaneous fat
tissue. An abdominal computed tomography scan excluded
any estrogen-producing tumor. Surgery (conducted by HKD)
Endocrine study Patient 1 Patient 2 Normal reference range
TSH 1.0 1.2 0.4–4.0 mU/L
FT4 13.5 12.3 8–22 pmol/L
LH <0.15 <0.15 <0.15–1.3 U/L
FSH 0.76 0.94 <0.15–3.7 U/L
Estradiol (E2) <10 30 <30–70 pmol/L
Estrone 110 210 65–220 pmol/L
Testosterone <0.02 0.27 0.03–0.65 nmol/L
hCG <1.0 <1.0 <1.0 ng/ml
Prolactine 98 128 80–660 mU/L
17OHP 0.3 1.0 0.2–7.4 nmol/L
SHBG 123 117 30–210 nmol/l
Androstenedione <0.05 0.65 0.2–2.4 nmol/L
DHEAS <0.4 1.18 0.29–1.90 μmol/l
Estrogen receptor 19 <10 <20 fmol/mg
Progesterone receptor 13 10 <20 fmol/mg
Table 1 Endocrinologic
findings
TSH thyroid-stimulating
hormone, FT4 free thyroxine,
LH luteinizing hormone,
FSH follicle-stimulating
hormone, hCG human chorionic
genodotropin, 17OHP
17-hydroxy-progesterone,
SHBG sex hormone-binding
globulin, DHEAS dehydroe-
piandrosterone
396 Eur J Plast Surg (2011) 34:395–398
under general anesthesia consisted of peripheral liposuction
(with the 2 mm PAL liposuction system) and subcutaneous
partial resection of the gland, conducted through an infra-
areolar incision. The specimen dissected measured 5.0× 3.0×
2.5 cm. Pathologic examination revealed normal glandular
breast tissue, without evidence of malignancy. Microscopy of
the subcutaneous mastectomy specimen showed mammary
ducts with hypertrophic ductuli and mild lymphocytic
infiltrates lining the ducts. The concluding diagnosis was
normal gynecomastia tissue. Recovery was uneventful.
At a postoperative clinical evaluation 11 months later,
there were no signs of breast development (Fig. 1b).
Discussion
Gynecomastia is a condition in which the glandular
components of the male breast proliferate, resulting in an
enlargement of one or both breasts. In the age distribution,
three distinct peaks are identified. The first is found in the
neonatal period, in which palpable breast tissue develops in
60% to 90% of all newborns due to transfer of estrogens
across the placenta. The second peak occurs during puberty, as
a result of an imbalance between estrogens and androgens
within the breast tissue. The last peak is found in the adult
population, with the highest prevalence among 50- to 80-year-
old males [1,3]. Common known causes for gynecomastia in
adults include liver disease, as well as the use of drugs such
as digitalis and tricyclic antidepressants [5]. In contrast to
gynecomastia in adolescent boys and adult men, prepubertal
gynecomastia is rare. A specific cause is hardly ever
identified, and in 90% of patients, prepubertal gynecomastia
is classified as idiopathic [2,6]. Known causes of breast
enlargement in children are diverse [3,6–8]. Therefore,
further exploration of the etiology is advised, particularly to
rule out any endocrine or malignant abnormalities. A variety
of endocrinopathies, mostly as a result of an increased ratio
of circulating estrogens to androgens, induce stimulation of
breast tissue leading to gynecomastia. Calzada et al. showed
that the presence of hormone receptors in gynecomastia may
provide a setting favorable for mammary glands to develop
gynecomastia [9]. In patient 1, there were low levels of both
receptors and, in patient 2, only the progesterone receptor
was detected. We conclude that the gynecomastia in our
patients was not mediated by an abnormal increase in the
numbers of these receptors. The family history of our first
patient revealed gynecomastia on the father'sside.Stratakis
et al. described increased extraglandular aromatization of
androgens to cause the unusual entity of familial gyneco-
mastia, named aromatase excess syndrome [10]. This
syndrome has been correlated to serum estradiol and estrone
excess, which were within normal limits in both of our
patients. Therefore, a mild aromatase excess syndrome could
be excluded as a cause of the prepubertal gynecomastia and
familial occurrence. None of our patients were exposed to
exogenous estrogens. However, our first patient was using
Melatonin, of which in literature only one case is reported.
The case described a possible relationship of the usage of
this drug and a painful bilateral gynecomastia in an adult
male [11], making the relationship between this drug and
gynecomastia in our first patient very unlikely. Furthermore,
obesity is documented in 31% of the boys, with excessive fat
tissue as a possible cause of prepubertal gynecomastia [3].
Increased adipose tissue causes an increased aromatization of
mostly testosterone in adipose tissue, leading to an imbalance
between estradiol and testosterone levels. Our patients had no
signs of being overweight; therefore, obesity was excluded as
the etiology for the gynecomastia.
Fig. 1 a An 11-year-old boy with unilateral gynecomastia of his left breast. bThe same boy at an 11-month postoperative evaluation, without any
signs of breast development
Eur J Plast Surg (2011) 34:395–398 397
In summary, atypical presentations of gynecomastia are
frequently underappreciated, with little attention to breast
enlargement in the nonobese prepubertal boy. Since
prepubertal gynecomastia could be a sign of possible
underlying diseases, a thorough examination and further
research is recommended. If a reversible cause can be
excluded, peripheral liposuction in combination with
surgical resection of the gland tissue by an infra-areolar
approach is the first therapy of choice. We report two rare
cases of prepubertal unilateral gynecomastia, in whom the
cause of the gynecomastia remains unclear, while none of
the above discussed causes could be identified.
Open Access This article is distributed under the terms of the
Creative Commons Attribution Noncommercial License which per-
mits any noncommercial use, distribution, and reproduction in any
medium, provided the original author(s) and source are credited.
References
1. Braunstein GD (1993) Gynecomastia. N Engl J Med 328(7):490–
495
2. Cho YR, Jones S, Gosain AK (2008) Neurofibromatosis: a
cause of prepubertal gynecomastia. Plast Reconstr Surg 121
(3):34e–40e
3. Einav-Bachar R, Phillip M, Aurbach-Klipper Y, Lazar L (2004)
Prepubertal gynaecomastia: aetiology, course and outcome. Clin
Endocrinol 61(1):55–60
4. Marshall WA, Tanner JM (1970) Variations in the pattern of
pubertal changes in boys. Arch Dis Child 45(239):13–23
5. Harigopal M, Murray MP, Rosen PP, Shin SJ (2005) Prepubertal
gynecomastia with lobular differentiation. Breast J 11(1):48–51
6. Henley DV, Lipson N, Korach KS, Bloch CA (2007) Prepubertal
gynecomastia linked to lavender and tea tree oils. N Engl J Med
356(5):479–485
7. Ersoy B, Yoleri L, Riza Kandiloğglu A (2002) Unilateral
galactocele in a male infant. Plast Reconstr Surg 109(1):401–402
8. Kauf E (1998) Gynecomastia in childhood: pathologic causes
unusual but serious. Fortschr Med 116(35–36):23–26
9. Calzada L, Torres-Calleja J, Martinez JM, Pedrón N (2001)
Measurement of androgen and estrogen receptors in breast tissue
from subjects with anabolic steroid-dependent gynecomastia. Life
Sci 69(13):1465–1469
10. Stratakis CA, Vottero A, Brodie A et al (1998) The aromatase
excess syndrome is associated with feminization of both sexes and
autosomal dominant transmission of aberrant P450 aromatase
gene transcription. J Clin Endocrinol Metab 83(4):1348–1357
11. De Bleecker JL, Lamont BH, Verstraete AG, Schelfhout VJ
(1999) Melatonin and painful gynecomastia. Neurology 53
(2):435–436
398 Eur J Plast Surg (2011) 34:395–398
