핸들바 마비(Handlebar Palsy)로 오인된 척골관증후군

Case Report
The Korean Journal of Sports Medicine 2015;33(2):139-142
pISSN 1226-3729 eISSN 2288-6028
http://dx.doi.org/10.5763/kjsm.2015.33.2.139
핸들바 마비(Handlebar Palsy)로 오인된 척골관증후군
경희대학교 의과대학 정형외과학교실
김영준ㆍ정덕환ㆍ백종훈
Misdiagnosed Handlebar Palsy: Giant Cell Tumor
of the Tendon Sheath in Guyon’s Canal
Young Jun Kim, Duke Whan Chung, Jong Hun Baek
Department of Orthopedic Surgery, Kyung Hee University School of Medicine, Seoul, Korea
Ulnar tunnel syndrome (UTS) is a compressive neuropathy of the upper extremity that shows various clinical
symptoms according to the anatomic region of the compression site. Numerous factors may cause UTS, and most
publications are case reports describing various etiologies; thus, obtaining a correct diagnosis is often challenging.
Giant cell tumor of the tendon sheath (GCTTS) is well described to be a common benign soft tissue tumor of the
hand; however, it is rarely reported to cause UTS. We report a case of GCTTS in Guyon’s canal causing UTS
that was misdiagnosed as handlebar palsy.
Keywords: Ulnar neuropathies, Ulnar nerve compression syndromes, Giant cell tumors
Introduction
surrounding structures and may even erode the bony structure
in a confined space. Numerous factors may cause ulnar tunnel
Giant cell tumor of the tendon sheath (GCTTS) is the second
syndrome (UTS). Any mass growing within Guyon’s canal is
most common benign soft tissue tumor of the hand. GCTTS occurs
a classical cause of UTS, and chronic repetitive trauma on the
particularly on the volar surface of the finger or hand, and tends
hypothenar eminence has been implicated as a common cause
to involve the radial three digits and distal interphalangeal joint
of UTS in cyclists .
region1). This tumor type has been known to impinge upon
Received: October 7, 2015 Revised: November 9, 2015
Accepted: November 18, 2015
Correspondence: Duke Whan Chung
Department of Orthopedic Surgery, Kyung Hee University
School of Medicine, 23 Kyungheedae-ro, Dongdaemun-gu,
Seoul 02447, Korea
Tel: +82-2-958-8346, Fax: +82-2-964-3865
E-mail: dukech@khmc.or.kr
Copyright ©2015 The Korean Society of Sports Medicine
CC This is an Open Access article distributed under the terms of the Creative
Commons Attribution Non-Commercial License (http://creativecommons.org/
licenses/by-nc/4.0) which permits unrestricted non-commercial use, distribution,
and reproduction in any medium, provided the original work is properly cited.
2,3)
Here, we report a case of UTS caused by GCTTS in Guyon’s
canal that was misdiagnosed as a sports-related neuropathy,
handlebar palsy.
Case Report
A 53-year-old male patient presented with right-hand weakness
and pain over the hypothenar eminence. He was a habitual cyclist
who had experienced pain in the prior 7 months. He had been
take a bike twice in a day, usually 1 – 1.5 hours of duration for
one time, since 2 years ago. Also he was experienced aggravated
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YJ Kim, et al. Misdiagnosed Handlebar Palsy: Giant Cell Tumor of the Tendon Sheath in Guyon’s Canal
numbness and pain when grip on mountain bicycle type handlebar.
canal. The mass was identified and was found to originate at
Weakness was noticeable by the patient 6 months before admission.
the distal and deep portions of Guyon’s canal (Fig. 1A). The
There was marked atrophy of the first web space with a relatively
deep branch of the ulnar nerve was obviously compressed, and
spared hypothenar eminence, and he showed decreased grip
tenting was shown by the mass. The mass was located directly
strength compared with the contralateral side. Tinnel’s sign was
under the deep branch of the ulnar nerve, distal to the bifurcation
positive with no palpable mass over the ulnar side of the wrist.
point of the nerve over the pisohamate ligament (Fig. 1B). After
Egawa’s sign was also positive. There was no sensory loss in
careful dissection, the mass was loosely associated with the tendon
the affected hand, and the patient complained of intermittent
sheath of the flexor carpi ulnaris tendon (Fig. 1C). The canal
numbness over the ulnar two digits. Simple radiography showed
was decompressed after mass excision. The mass was well
no definitive bony lesion or alteration of the soft tissue density
capsulized to about in 1×1.5 cm in size and appeared yellowish
around the ulnar side of the wrist.
and solid (Fig. 1D). The surgical finding was correlated with
A nerve conduction study revealed that the first dorsal interossei
the electrodiagnosis of ulnar neuropathy which representing motor
muscles showed a small amplitude and delayed latency; addi-
nerve pathology. Photomicrography revealed uniform distribution
tionally, sensory nerve conduction was normal. Electromyography
of mononuclear stromal cells with multinucleated giant cells
revealed abnormal spontaneous activities and a decreased inter-
characteristic of GCTTS (Fig. 2).
ference pattern of the first dorsal interossei muscles, suggesting
incomplete ulnar neuropathy around the wrist level.
The patient had fully restored sensory function with improved
motor function of the hand 6 months after the operation.
We considered that the cause of the ulnar neuropathy was
repetitive stress injury related to sports activity, also known as
Discussion
handlebar palsy. Surgical exploration was performed without
further imaging study, such as sonography or magnetic resonance
imaging.
Although the anatomic course of the ulnar nerve in the wrist
has been well documented in standard texts, it is important to
A skin incision was made directly over Guyon’s canal. We
emphasize specific distinctions concerning the anatomy with
performed ulnar nerve exploration from the proximal side of the
respect to compression of the ulnar nerve. The ulnar tunnel
Fig. 1. Intraoperative view of the
ulnar tunnel. (A) The mass was
identified in Guyon’s canal, immediately beneath the ulnar nerve.
(B) After further surgical exploration, it was located distal to the
ulnar nerve bifurcation point; the
deep branch of the ulnar nerve
was obviously compressed and
tented. (C) The mass was loosely
attached to the flexor carpi ulnaris
tendon. (D) The removed mass.
br.: branch.
140
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김영준 외. 핸들바 마비(Handlebar Palsy)로 오인된 척골관증후군
occurs.
UTS is caused by various etiologic factors. Common causes
are acute or chronic trauma most likely related to an occupation,
5)
and ganglia . Other causes include carpal bone fracture, ulnar
artery or hypothenar muscle pathology, and concurrent case of
idiopathic carpal tunnel syndrome. Chronic repetitive trauma over
the hypothenar eminence has been implicated as a cause of UTS
and is not uncommon among long-distance cyclists, classically
described as handlebar palsy2,6). Typical findings are an isolated
Fig. 2. A photomicrographic picture showing uniform distribution of mononuclear stromal cells with multinucleated
giant cells which was characteristics of giant cell tumor of
the tendon sheath. H&E, ×100.
lesion of the deep terminal branch, and an additional lesion of
2)
the superficial sensory branch may be present . Handlerbar palsy
is occurred mostly in long distance cycling or professional cyclist.
6)
Patterson et al. investigated the incidence of ulnar neuropathy
in 25 cyclists. The author investigated cyclists, who underwent
originates at the proximal edge of the palmar carpal ligament
a 600 km tour over 4 days. 70% of the participants experienced
and extends distally to the fibrous arch of the hypothenar muscles
some form of sensory or motor neurological symptoms by the
4)
at the level of the hook of hamate . During its course in the
end of the tour. There is a gap in the literature regarding evidence
fibro-osseous tunnel, the ulnar nerve bifurcates into a superficial
based information for the management of handlebar palsy. The
sensory and deep motor branch distal to the distal pole of the
most commonly recommended treatment is to prevent its
3)
pisiform . The motor branch to the hypothenar muscles separates
occurrence, including avoid local pressure using some forms of
from the main ulnar nerve just distal to the pisiform at the wrist.
gloves and/or modifications of handlebar grip. However, There
Thus, the hypothenar muscle was relatively unaffected by
are some treatment guidelines of UTS in European HANDGUIDE
compression of the deep branch. The deep branch of the ulnar
study in 2013 . They concluded that main factor for treatment
nerve courses around the hook of the hamate to innervate the
choice were severity of symptoms, duration of symptoms and
other intrinsic muscles of the hand. This deep branch is susceptible
previous treatment received. In the presented case, patient’s first
to nerve compression from overlying tendinous bands in the
web space atrophy was severe, which suggesting chronic UTS.
hypothenar muscles and from its excursion as it courses around
Surgery is an option to explore ulnar nerve compression in the
3)
the hook of the hamate . Therefore, it is important to inspect
7)
canal.
the course of the deep branch because it may require decompression
GCTTS causing UTS located in Guyon’s canal is rare. There
and because satellite lesions may be present. The relationship
have been few reports concerning GCTTS in the English-language
between the three anatomical zones of ulnar nerve compression
literature, but it should be included in the possible pathologic
within Guyon’s canal and symptoms can be classified into three
structure of Guyon’s canal and the differential diagnosis
4)
8-10)
.
categories . Zone 1 refers to the region proximal or within Guyon’s
This case demonstrates the possibility of a misdiagnosis of
canal, before bifurcation of the ulnar nerve that manifests as
UTS. Clinical history and examination may be nonspecific or
motor weakness and sensory deficits, or pure sensory or pure
related to coexisting pathologies, making an accurate diagnosis
motor deficits. Zone 2 surrounds the deep branch, and compression
confusing3). When external causes like handlebar palsy was
in this region results in paralysis of the intrinsic muscle and
suspected to cause compression of the ulnar nerve in Guyon’s
may involve or spare the hypothenar muscle. Zone 3 surrounds
canal, appropriate imaging studies are advised in addition to
the superficial branch, and a lesion in this zone produces sensory
electrodiagnostic studies. Careful exploration of the nerve down
symptoms. Clinical findings can appear as various aspects in the
to the rest of the ulnar tunnel should be performed during surgical
mass lesion of Guyon’s canal depending on where the compression
exploration because of the propensity for space-occupying lesions.
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YJ Kim, et al. Misdiagnosed Handlebar Palsy: Giant Cell Tumor of the Tendon Sheath in Guyon’s Canal
Conflict of Interest
No potential conflict of interest relevant to this article was
6.
reported.
7.
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