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TECHNIQUES IN UROLOGY
Cite as: Can Urol Assoc J 2017;11(5):E244-7. http://dx.doi.org/10.5489/cuaj.4169
Published online May 9, 2017
Abstract
Perineal nodular induration (PNI), or biker’s nodule, is a rare, both-
ersome, pseudotumour. Herein, we describe the surgical technique
used to treat a healthy cyclist who developed an enlarging PNI
for five years that grew into a perineal mass. The mass prevented
him from cycling due to worsening discomfort and heaviness. The
PNI-associated mass was successfully removed by wide surgical
excision and a local advancement flap. Subsequently, the patient
resumed cycling. Histopathology report demonstrated a benign
lesion with abundant ropy collagen with native smooth muscle,
vessels, and rare fibroblast-like spindle cells. With the increasing
popularity of cycling, PNI may become more common, and health
providers should be aware of this rare entity and how it can be
safely removed.
Introduction
Bicycle riding is one of the most popular methods for trans-
portation, exercise, and leisure. According to U.S. Bicycling
Participation Benchmarking 2015 report, 34% or 103.7
million Americans (ages three and older) rode a bicycle at
least once in the past year, with 14% of this population
reporting frequent riding more than 104 times/year.1 The
health benefits of bicycling as an aerobic exercise are well-
known.2-4 Endurance cycling may cause injuries affecting the
genitourinary system.5 Perineal nodular induration (PNI) also
known as biker’s nodule, cyclist’s nodule, ischiatic hygroma,
third testicle, or an accessory testicle is a benign pseudo-
tumour that presents most commonly in avid male cyclists
with repetitive perineal microtrauma.6 It usually develops
posterior to the scrotum in the soft tissue of the perineum,
as two masses on both sides of the median raphe, or as a
single mass located midline, or lateralized over the ischial
tuberosity.6 Herein, we report our surgical technique for PNI
treatment and the histopathological characteristics of PNI.
History
A 48-year-old healthy male who is an avid cyclist presented
with a progressive inferior perineal and scrotal swelling for
five years. He had been actively cycling for 20 years, riding an
average of 100 miles/week. When he presented to our clinic,
he complained of redundant scrotal and perineal skin. He
reported a history of saddle sores that resolved in the past. On
physical examination, there was a large, mobile, non-tender,
and soft mound of perineal tissue that was posterior to the
scrotum with redundant tissue, covered by normal skin (Fig.
1). Ultrasound of the scrotum demonstrated redundant soft tis-
sue in the perineum inferior to the raphe. The bulge restricted
the patient’s daily activity and he opted for surgical excision.
Surgical technique
The patient was placed in a lithotomy position. An elliptical
incision was made from the apex, near the scrotum, down
to 2 cm above the anus. The dissection was superior to
the bulbospongiosus muscle. After complete mobilization,
the mass was transected and sent to pathology. It measured
approximately 14 x 5 cm (Fig. 2). In order to close the inci-
sion in a tension-free manner, lateral flaps were raised that
were approximately 15 x 3 cm. The subcutaneous layers
were closed using interrupted 2-0 vicryl for the deep and
superificial layers. In total, there were four layers of closure.
The patient tolerated the procedure well and was discharged
in the same day in a good condition.
Pathology
Grossly, the specimen consisted of a 14 x 5.5 cm x 2 cm
thick fragment of hair-bearing skin and subcutaneous tissue
Mohannad A. Awad, MD1,2; Gregory P. Murphy, MD1; Thomas W. Gaither, MD1; E. Charles Osterberg, MD3;
Thomas A. Sanford, MD1; Andrew E. Horvai, MD4; Benjamin N. Breyer1,5
1Department of Urology, University of California–San Francisco, San Francisco, CA, United States; 2Department of Surgery, King Abdulaziz University, Rabigh, Saudi Arabia; 3Department of Surgery, University
of Texas, Dell Medical School, Austin, TX, United States; 4Department of Pathology, University of California–San Francisco, San Francisco, CA, United States; 5Department of Biostatistics and Epidemiology,
University of California–San Francisco, San Francisco, CA, United States
Surgical excision of perineal nodular induration:
A cyclist’s third testicle
CUAJ • May 2017 • Volume 11, Issue 5
© 2017 Canadian Urological Association
E244
CUAJ • May 2017 • Volume 11, Issue 5 E245
with a homogeneous, fibrotic cut surface. The epidermis and
dermis were unremarkable microscopically (not shown). The
deeper soft tissues showed abundant ropy collagen bundles
mixed with native smooth muscle, vessels, and rare fibro-
blast-like spindle cells. There was no atypia, necrosis, or
mitotic activity (Fig. 3).
Followup
The patient began cycling roughly two months after the
operation with mild discomfort that improved over time.
He denied any sexual or urinary side effects. Fig. 4 shows
the patient’s perineum and scrotum in followup, with no
signs of residual mass at two months’ followup.
Discussion
Although the pathogenesis of PNI is unknown, it may be
related to the repetitive compression and friction of the
perineal fascia between the bike’s saddle and ischial tuber-
osities.7 Beyond cyclists, this complication has been docu-
mented in a patient who was an equestrian and another who
tested lawn mowers.8,9 It is more commonly seen in males,
but recently, five female cases have been reported.9,10 Its
latency period varies from a few weeks to one year. Although
the nodule in our case was mobile, most reports state that it
is fixed to the soft tissue or ischial tuberosity, has a fibrous
to elastic consistency, and is covered by normal skin.6,7,11
Perineal nodular induration
Fig. 1. Large, perineal fat posterior to the scrotum with redundant tissue
resembling an accessory testicle (arrow).
Fig. 2. Excised mass measuring approximately 14 x 5 cm.
Fig. 3. (A) Microscopically, the excision specimen demonstrated abundant ropy collagen with rare, bland fibroblast-like spindle cells mixed with native smooth
muscle and small vessels; (B) at higher magnification, some of the collagen bundles showed a hyaline, keloidal quality.
The histopathological appearance of PNI varies. It is often
characterized by a central pseudocystic space surrounded by
fibrous tissue containing fibroblast spindle cells and clusters
of small-sized vessels.7,11,12 The central pseudocystic space
somewhat resembles that seen in ischemic fasciitis, which
occur in elderly patients over the bony prominences who are
often, though not always, immobilized.13-15 Other possible
differential diagnoses are listed in Table 1.
In most cases, history and physical examination can
diagnose PNI. Imaging modalities, such as ultrasound and
magnetic resonance imaging, may be needed in some cases.
Final diagnosis is made by histopathological examination.
Primary treatment options include adjustment of cyclist
position to change the distribution of weight on the saddle,
as well as limiting the source of friction, which includes
using adequate saddle/perineal lubricant and proper fitting
cycling clothing. Patients can be counselled that there are no
reports of spontaneous regression, but it is possible to limit
progression and discomfort. Second, some authors reported
that intralesional corticosteroids or hyalurodinase injection
may offer some relief. These injections are only preferable
for small nodules and may cause subcutaneous atrophy.22,23
Both of the previous treatment options were not suitable for
our patient, as the mass was quite large and bothersome.
Finally, surgical excision of the mass is the most often used
treatment with superior results.6 The technique to remove this
mass could vary depending on the exact size and location.
With smaller masses, the need to raise local skin flaps as in
our case, may not be necessary. For larger masses, if skin
flaps cannot be mobilized well enough, a split-thickness
skin graft could also be applied to any areas not able to be
closed in a tension-free manner.
Conclusion
PNI is a poorly recognized, rare, bothersome benign pseu-
dotumour that mostly appears in professional and avid
cyclists. Diagnosis is made by clinical history and exami-
nation. Surgical excision is the treatment of choice. With
the increasing popularity of cycling, PNI may become more
common, and healthcare providers should be aware of this
entity and how it can be safely removed.
Competing interests: This research was funded by the Bicycle React Grant. The authors report no
competing personal or financial interests.
This paper has been peer-reviewed.
References
1. PeopleforBikes. U.S. Bicycling Participation Benchmarking Study Report 2015. July 20, 2016]; Available
at http://b.3cdn.net/bikes/7b69b6010056525bce_ijm6vs5q1.pdf. Accessed March 24, 2017.
CUAJ • May 2017 • Volume 11, Issue 5
E246
Awad et al.
Fig. 4. Patient perineum and scrotum at two months’ followup showing no signs
of residual mass.
Table 1. Differential diagnoses for perineal nodular induration
Differential diagnosis Histolopathological features
Ischemic fasciitis13-15 Outer fringe of mitotically active fibroblasts and occasional ganglion-like cells and small vessels
surrounding a central zone of necrobiosis
Cellular angofibrom*16 Uniformly moderately cellular with plump, bland spindle cells, hyalinized round, thick and walled
vessels may contain adipose tissue
Angiomyofibroblastoma-like tumour
of the male genital tract*17
Alternating hypercellular and hypocellular areas with clusters of rounded, epithelioid cells around
numerous small vessels
Mammary type fibroblastoma*18 Well-circumscribed, more cellular, and lacks a central zone pattern. Bundles of thick, ropy collagen
with inconspicuous vessels may contain adipose tissue
Massive localized lymphedema19 Edematous stroma, dilated vascular channels, and mature adipose tissue separated by fibrous
septa
Prepuberal vulvar formation20 Poorly marginated, hypocellular tumours, formed by bland of spindle-shaped cells in a
collagenous to edematous or myxoid stroma
Childhood asymmetric labium majus
enlargement21
Rare to moderate cellular interconnected fibrous bands, encircled lobules of fat, vessels, and
nerves
*These three entities are closely related and may represent variants of a single entity.
CUAJ • May 2017 • Volume 11, Issue 5 E247
2. Powell KE, Thompson PD, Caspersen CJ, et al., Physical activity and the incidence of coronary heart disease.
Annu Rev Public Health 1987;8:253-87. https://doi.org/10.1146/annurev.pu.08.050187.001345
3. Williams PT, Reduction in incident stroke risk with vigorous physical activity. Evidence from 7.7-year
followup of the National Runners’ Health Study. Stroke 2009;40:1921-3. https://doi.org/10.1161/
STROKEAHA.108.535427
4. Williams PT. Vigorous exercise, fitness, and incident hypertension, high cholesterol, and diabetes. Med Sci
Sports Exerc 2008;4:998. https://doi.org/10.1249/MSS.0b013e31816722a9
5. Leibovitch I, Mor Y. The vicious cycling: Bicycling related urogenital disorders. Eur Urol 2005; 47:277-87.
https://doi.org/10.1016/j.eururo.2004.10.024
6. Creff A, Melki F, Ceccaldi M, et al. L’hygroma ischiatique ou “troisième testicule du stayer”. Réflexion à
propos de l’étiopathogénie, du traitement et de la prévention. Med Sport 1985;59:296-300.
7. de Saint Aubain Somerhausen N, Geurde B, Couvreur Y. Perineal nodular induration: The ‘third
testicle of the cyclist’, an under‐recognized pseudotumour. Histopathology 2003;42:615-6.
https://doi.org/10.1046/j.1365-2559.2003.01592.x
8. Pedio G, Zoebeli L, Rust B. A rare case of” occupational disease.” Acta Cytol 1986;30:453-4.
9. Devers KG, Heckman SR, Muller C, et al. Perineal nodular induration: a trauma-induced mass in a female
equestrian. Int J Gynecol Pathol 2010;29:398-401. https://doi.org/10.1097/PGP.0b013e3181ce1341
10. McCluggage WG, Smith JH. Reactive fibroblastic and myofibroblastic proliferation of the vulva (cyclist’s
nodule): A hitherto poorly described vulval lesion occurring in cyclists. Am J Surg Pathol 2011;35:110-4.
https://doi.org/10.1097/PAS.0b013e3181ffd8ab
11. Vuong P, Camuzard P, Schoonaert M. Perineal nodular indurations (“accessory testicles”) in cyclists. Fine-
needle aspiration cytologic and pathologic findings in two cases. Acta Cytol 1987;32:86-90.
12. Khedaoui R, Martín-Fragueiro LM, Tardío JC. Perineal nodular induration (“biker’s nodule”): Report of two
cases with fine-needle aspiration cytology and immunohistochemical study. Int J Surg Pathol 2014;22:71-
5. https://doi.org/10.1177/1066896912465008
13. Montgomery EA, Meis JM, Mitchell MS, et al. Atypical decubital fibroplasia: A distinctive fibro-
blastic pseudotumour occurring in debilitated patients. Am J Surg Pathol 1992;16:708-15.
https://doi.org/10.1097/00000478-199207000-00009
14. Perosio P, Weiss S. Ischemic fasciitis: A juxta-skeletal fibroblastic proliferation with a predilection for elderly
patients. Mod Pathol1993;6:69-72.
15. Liegl B, Fletcher CD. Ischemic fasciitis: Analysis of 44 cases indicating an inconsistent association
with immobility or debilitation. Am J of Surg Pathol 2008;32:1546-52. https://doi.org/10.1097/
PAS.0b013e31816be8db
16. Nucci MR, Granter SR, Fletcher CD. Cellular angiofibroma: A benign neoplasm distinct
from angiomyofibroblastoma and spindle cell lipoma. Am J Surg Pathol 1997;21:636-44.
https://doi.org/10.1097/00000478-199706000-00002
17. Laskin WB, Fetsch JF, Mostofi FK. Angiomyofibroblastomalike tumour of the male genital tract: Analysis
of 11 cases with comparison to female angiomyofibroblastoma and spindle cell lipoma. Am J of Surg
Pathol 1998;22:6-16. https://doi.org/10.1097/00000478-199801000-00002
18. McMenamin ME, Fletcher CD. Mammary-type myofibroblastoma of soft tissue: A tumour closely related
to spindle cell lipoma. Am J Surg Pathol 2001;25:1022-9. https://doi.org/10.1097/00000478-
200108000-00006
19. Kurt H, Arnold CA, Payne JE, et al. Massive localized lymphedema: A clinicopathological study of 46
patients with an enrichment for multiplicity. Mod Pathol 2016;29:75-82. https://doi.org/10.1038/
modpathol.2015.135
20. Iwasa Y, Fletcher CD. Distinctive prepubertal vulval fibroma: A hitherto unrecognized mesen-
chymal tumour of prepubertal girls: Analysis of 11 cases. Am J Surg Pathol 2004;28:1601-8.
https://doi.org/10.1097/00000478-200412000-00008
21. Vargas SO, Kozakewich HP, Boyd TK, et al. Childhood asymmetric labium majus enlargement: Mimicking
a neoplasm. Am J Surg Pathol 2005;29:1007-16.
22. Köhler P, Utermann S, Kahle B, et al. [Biker’s nodule—perineal nodular induration of the cyclist]. Hautarzt
2000;51:763-5. https://doi.org/10.1007/s001050051211
23. Bauer P, Etienney I. Affections périnéales liées au sport. 2008. Available at: http://www.em-consulte.
com/en/article/99380. Accessed April 13, 2017.
Correspondence: Dr. Mohannad A. Awad, Department of Urology, University of California–San
Francisco, San Francisco, CA, United States; mohannad.awad@ucsf.edu
Perineal nodular induration