- Department of Neurology and Neurosurgery, Universidade Federal de São Paulo, Rua Napoleão de Barros, 720, 6th floor, Sao Paulo, Brazil,
- Department of Orthopedics and Traumatology, Universidade Federal de São Paulo, Rua Napoleão de Barros, 720, 1st floor, Sao Paulo, Brazil.
Correspondence Address:
Luis Renato Nakachima
Department of Neurology and Neurosurgery, Universidade Federal de São Paulo, Rua Napoleão de Barros, 720, 6th floor, Sao Paulo, Brazil,
DOI:10.25259/SNI_606_2020
Copyright: © 2020 Surgical Neurology International This is an open-access article distributed under the terms of the Creative Commons Attribution-Non Commercial-Share Alike 4.0 License, which allows others to remix, tweak, and build upon the work non-commercially, as long as the author is credited and the new creations are licensed under the identical terms.How to cite this article: Fabio Veiga de Castro Sparapani1, Marcela Fernandes2, Leonardo Favi Bocca1, Luis Renato Nakachima2, Sergio Cavalheiro1. Acute handlebar syndrome: Two extremes of a challenging diagnosis. 29-Oct-2020;11:366
How to cite this URL: Fabio Veiga de Castro Sparapani1, Marcela Fernandes2, Leonardo Favi Bocca1, Luis Renato Nakachima2, Sergio Cavalheiro1. Acute handlebar syndrome: Two extremes of a challenging diagnosis. 29-Oct-2020;11:366. Available from: https://surgicalneurologyint.com/surgicalint-articles/10356/
Abstract
Background: Ulnar nerve mononeuropathy diagnosis can be challenging depending on where neural lesion is present. Repetitive trauma during cycling is a rare cause of ulnar neuropathy.
Case Description: We describe two patients who developed the handlebar syndrome, an ulnar nerve palsy at Guyon’s canal after cycling. The first patient had the syndrome after a short-distance ride and she was treated surgically, while the second patient developed the classical syndrome after a long ride and received conservative treatment. Surgical treatment of the first patient led to functional recovery.
Conclusion: Handlebar syndrome is a neuropathy caused by extrinsic repetitive compression of ulnar nerve at wrist. Increasing incidence of this disease can be expected after increasing popularity of cycling sports. Avoid of repetitive trauma is the main management goal, with surgical treatment reserved for failure of conservative treatment.
Keywords: Cycling, Guyon’s canal, Nerve compression syndrome, Ulnar neuropathy
INTRODUCTION
The diagnosis of the ulnar nerve lesion at Guyon’s canal could be challenging. The characteristic sensory loss at the ulnar portion of the hand and weakness of all ulnar intrinsic hand muscles occurs when both the superficial and deep branches are affected.[
When only the terminal superficial or deep branches are affected, both due to intrinsic or extrinsic causes, uncommon clinical features could be seen. These unusual clinical presentations can be difficult to be interpreted by nonspecialists. Misdiagnosis can lead to equivocal treatment, delaying or preclude nerve recovery.
CASE DESCRIPTION
Case 1
A 55-year-old female presented to our outpatient facility complaining about 4 weeks history of weakness and clumsiness of the left hand without pain or sensory loss. She spontaneously denied any unusual situation but, after a thorough anamnesis, she remembered some light exercises, including a short bike ride on a weekend trip. Her medical records are unremarkable. Physical examination showed marked weakness and atrophy of all ulnar supplied intrinsic left hand muscles, except for the hypothenar muscles [
Before our evaluation, magnetic resonance imaging (MRI) of the cervical spine was performed showing no cervical myelopathy or compression of cervical roots and an electroneurography (ENMG), done just after initial symptoms, showed an unspecific axonal multineuropathy without motor conduction block. With these examinations, a suspected diagnosis of amyotrophic lateral sclerosis was performed by general physician and patient was sent to our institution. After our evaluation, a forelimb MRI was performed and no nerve compression was observed. The patient was submitted to another ENMG that confirmed ulnar neuropathy.
A surgical exploration was indicated after clinical complementary evaluation confirmed ulnar neuropathy. The procedure revealed a mild compression of the deep ulnar branch under the origin of the flexor digiti quinti brevis and opponens digiti quinti muscles [
Figure 2:
Surgical view showing the pisohamate hiatus. The fibrous arch (white arrow) must be divided to release the deep branch of ulnar nerve. Note the deep branch of ulnar nerve (red arrow) passing posterior to the fibrous arch and the superficial branch of ulnar nerve (black arrow) passing anterior to the arch.
During the outpatient follow-up, the patient had an uneventful recovery with motor and sensory improvement.
Case 2
A 45-year-old female complained about weakness of the left hand with local pain and sensory loss in the IV, V fingers and hypothenar volar region in the 4th day of a 500 km road cycling amateur competition. After the end of the competition, the pain has gone but the rest of the symptoms remained. The patient presented to our outpatient facility 3 weeks after the end of cycling event. Her medical records were unremarkable.
Physical examination revealed mild weakness (Grade IV) and atrophy of all ulnar supplied intrinsic left hand muscles, including the hypothenar muscles, and hypoesthesia in the V finger [
A forearm and wrist MRI were requested and she denied being submitted an electroneuromyography. The MRI did not reveal any anomalies except the amyotrophy related. A conservative treatment was done with a good recovery.
DISCUSSION
The handlebar is one of the three contact areas that support the cyclist, beyond pedals, and the seat. Continuous and hyperextended position of the hand could compress and stretch both ulnar and median nerves at the wrist.[
The Guyon’s canal lesions are usually divided into four places with distinct clinical aspects. Cavallo et al. proposed a clinical classification to distal ulnar neuropathy that can be easily applied to clinical practice.[
The most reported etiologies of these lesions are the compressive in nature: lipoma, cysts, anomalies of ligaments or muscles, hook of the hamate fracture, and ulnar artery aneurysms. Extrinsic causes, such as chronic repetitive trauma or chronic pressure together with vibration, are not commonly reported, except if applied for months or years.[
The prevalence of ulnar and median nerve compression in long-distance cyclists ranges from 10% to 70%[
When history is unclear and the clinical findings are not specific, further diagnostic workups are needed. ENMG and an MRI of the hand should be performed for the exclusion of pathological structures at Guyon’s canal. Important differential diagnosis should be discarded, including radiculopathy of C8 and/or T1 roots. In pure motor syndromes, amyotrophic lateral sclerosis, multifocal motor neuropathy, and hereditary neuropathy with liability to pressure palsy (HNPP) should be ruled out.[
In our first case, the initial diagnosis caused intense stress and great suffering to the patient and her family. Her short history of cycling was an unusual cause of distal ulnar neuropathy, usually related to long-distance and repetitive cycling. Since the first report by Simpson[
Although handlebar syndrome tends to recover with conservative management, it has to be early diagnosed to avoid a worsening of the case, as presented by the second patient. There is a lack of evidence-based recommendations on literature regarding handlebar syndrome treatment.[
CONCLUSION
Handlebar syndrome is a distal ulnar neuropathy cause by extrinsic repetitive compression of ulnar nerve at wrist. Its differential diagnosis can be challenging and must be ruled out before definitive diagnosis.
The incidence of handlebar syndrome should increase more and more with the increasing popularity of cycling sports, including mountain bike or long-distance cycling, so clinicians must be aware of this diagnosis. Surgical treatment is reserved to conservative management failure or anatomical anomaly found after imaging evaluation.
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent.
Financial support and sponsorship
Nil.
Conflicts of interest
There are no conflicts of interest.
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