- Email: [email protected]
Isolated Peripheral Nerve Palsies in Thoracic Outlet Syndrome
Accepted Manuscript Isolated Peripheral Nerve Palsies in Thoracic Outlet Syndrome Kathryn A. Lee, Jeremy E. Newman, Andrew W. Garnham, Michael L. Wall PII:
S0890-5096(17)30696-9
DOI:
10.1016/j.avsg.2017.05.020
Reference:
AVSG 3404
To appear in:
Annals of Vascular Surgery
Received Date: 11 April 2017 Accepted Date: 7 May 2017
Please cite this article as: Lee KA, Newman JE, Garnham AW, Wall ML, Isolated Peripheral Nerve Palsies in Thoracic Outlet Syndrome, Annals of Vascular Surgery (2017), doi: 10.1016/ j.avsg.2017.05.020. This is a PDF file of an unedited manuscript that has been accepted for publication. As a service to our customers we are providing this early version of the manuscript. The manuscript will undergo copyediting, typesetting, and review of the resulting proof before it is published in its final form. Please note that during the production process errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain.
ACCEPTED MANUSCRIPT
Isolated Peripheral Nerve Palsies in Thoracic Outlet Syndrome
2
Kathryn A. Lee, Jeremy E. Newman, Andrew W. Garnham, Michael L. Wall.
3
Department of Vascular Surgery, Black Country Vascular Network, Dudley. DY1 2HQ, United Kingdom.
4 5
Corresponding Author:
Mr. Michael Wall Black Country Vascular Network
7
Russell’s Hall Hospital
8
Dudley
9
DY1 2HQ
United Kingdom
11
Email:
12
Telephone:
15 16 17 18 19 20 21
TE D
+44 (0) 1384 456 111
EP
14
[email protected]
AC C
13
M AN U
10
SC
6
RI PT
1
ACCEPTED MANUSCRIPT Abstract
23
Thoracic outlet syndrome poses a difficult diagnostic entity with varied aetiology and clinical
24
presentation. We present a 35-year-old gentleman with unilateral long thoracic nerve palsy and
25
contralateral subscapular paralysis caused by aberrant scalenus medius anatomy. Thoracic outlet
26
syndrome ought to be considered in patients presenting with isolated nerve palsies.
RI PT
22
27 28
SC
29
M AN U
30 31 32 33
37 38 39 40 41 42 43 44
EP
36
AC C
35
TE D
34
ACCEPTED MANUSCRIPT Introduction
46
Thoracic outlet syndrome (TOS) covers a large spectrum of symptomatology and pathology.
47
Differentiating venous, arterial and neurological symptoms can be complicated and tests directed at
48
certain pathologies are not always useful. We highlight an unusual case of a man with bilateral
49
isolated nerve palsies presenting with other classical thoracic outlet syndrome symptomatology
50
corrected by thoracic outlet decompression.
51
RI PT
45
Case Report
53
A 35-year-old Mechanic who enjoyed weight-lifting and boxing was referred to the Vascular
54
Outpatient Clinic from the Orthopaedic Surgeons with a seven-year history of right-sided neck and
55
scapular pain associated with intermittent ipsilateral digital pallor disturbance most prominent in
56
winter and on raising his arm. He had a long-standing winging of his right scapula and tenderness in
57
the posterior triangle of the neck on examination. There was also tingling in the ulna distribution of the
58
hand. Roos test was positive for loss of pulse on elevation and tiredness on active use of the limb.
59
Electromyography and nerve conduction study tests were unremarkable, and he had no cervical ribs
60
on chest X-ray. Magnetic resonance angiography (MRA) of his upper limbs revealed significant
61
impingement of both subclavian arteries upon elevation and external rotation.
62
EP
TE D
M AN U
SC
52
Figure 1: Patient MRA showing right subclavian artery (SCA) occlusion and left subclavian artery
64
stenosis.
65
AC C
63
66
He underwent right-sided anterior scalenectomy with division of his scalenus medius and first-rib
67
resection. The fibres of his scalenus medius muscle were found to be interdigitating with his brachial
68
plexus throughout all nerve roots. Post-operatively, he had normal sensation in his right upper limb
69
and shoulder girdle bar some numbness over his pectoralis major muscle, which was thought to be
70
neuropraxia. The winging of his scapula had resolved and he was able to return to work and back to
71
the gym.
ACCEPTED MANUSCRIPT A year later, he re-presented to clinic with a three-week history of severe pain at the medial border of
73
his left scapula that kept him awake at night, and subjective ipsilateral upper limb heaviness and
74
weakness. On examination, his scapula was not winged, but he had a positive Gerber’s Lift-Off Test
75
suggesting subscapularis paralysis. Again, Roos test was positive.
76
He underwent left-sided anterior and medius scalenectomy with first-rib resection. This time, the
77
fibres of his scalenus medius muscle passed posterior to the C5-6 trunks of the brachial plexus before
78
passing anterior to the trunks of C7, C8 and T1, causing compression at this point (Figures 2 & 3).
RI PT
72
SC
79
Figure 2: Intra-operative images of his left-sided thoracic outlet decompression, using the supraclavicular
81
approach.
M AN U
80
82
2
The Disabilities of the Arm, Shoulder and Hand (DASH) Questionnaire was used to assess his pre-
84
and post-operative upper limb function. Prior to his left thoracic outlet decompression, his DASH
85
score was 35. At his six-week follow-up appointment, his score was 13.4 and his subscapularis
86
weakness had resolved.
87
TE D
83
Conclusion
89
Roos described seven different types of congenital anomalies in his seminal paper on TOS in 1976 .
90
The discovery of fibres of scalenus medius are described best in this classification as Type 4.
91
However, this may not fully reflect our findings of scalenus medius interdigitating the trunks of the
92
brachial plexus, but highlights the different anatomies that present in this challenging operative area.
93
Isolated nerve injury is a rare clinical finding in TOS. Compression of the long thoracic nerve is not
94
unusual in shoulder pathology
95
cases of long thoracic nerve injury in TOS in the literature and these were treated with orthosis as
96
opposed to surgery . This was successful due to deformity of posture in the subjects. The anatomical
97
course of the long thoracic nerve having originated off the C5-7 nerve roots takes it anterior to
98
scalenus posterior muscle before coursing distal and laterally deep to the clavicle and superficial to
6
AC C
EP
88
3,5
but is limited in TOS. The authors were only able to identify two
4
ACCEPTED MANUSCRIPT 99
the first and second ribs. This classically is not involved in TOS but entrapment in these cases must have been present as decompression was successful.
101
The authors were unable to identify any cases of subscapularis paralysis due to TOS in the literature.
102
This may reflect a very low incidence or possibility that isolated nerve injuries are under reported. The
103
upper and lower subscapular nerves originate from the posterior cord of the brachial plexus and this
104
may have been the area of compression in this case.
105
Physicians must be aware of the possible anatomical abnormalities when investigating and treating
106
these patients. This case highlights congenital aberration in scalenus medius muscle anatomy as a
107
cause for two nerve palsies; in the long thoracic, and subscapular nerves. Clinicians presented with
108
patients with isolated nerve dysfunction should consider thoracic outlet syndrome in their differential
109
diagnoses.
M AN U
SC
RI PT
100
110 111
References
112
1. Gerber, C. and Krushell, RJ. Isolated rupture of the tendon of the subscapularis muscle. Clinical
114
features in 16 cases. J Bone Joint Surg Br. 1991 May; 73(3): 389-94.
TE D
113
2. Hudak, PL., Amadio, PC., and Bombardier, C. Development of an upper extremity outcome measure: the DASH (disabilities of the arm, shoulder and hand) [corrected]. The Upper Extremity
116
Collaborative Group (UECG). Am J Ind Med. 1996 Jun; 29(6):602-8. Erratum in: Am J Ind Med
117
1996 Sep; 30(3):372. The Institute for Work & Health are the copyright owners of the DASH and
118
QuickDASH.
AC C
EP
115
119
3. Scapular Winging: A Great Masquerader of Shoulder Disorders: AAOS Exhibit Selection.
120
Srikumaran, U., Wells, J., Freehill, M. et al., J Bone Joint Surg Am. 2014; 96: e122(1-13)
121 122 123 124
4. Nakatsuchi, Y., Saitoh, S., Hosaka, M., Uchiyama, S. Long thoracic nerve paralysis associated with thoracic outlet syndrome. J Shoulder Elbow Surg. 1994 Jan; 3(1): 28-33. 5. Freedman, J., Shankwiler, J. Nerve compression syndromes about the shoulder girdle. Current Orthopaedic Practice. 2008; 19(5): p524–529.
ACCEPTED MANUSCRIPT 125 126
6. Roos DB. Congenital anomalies associated with thoracic outlet syndrome. Anatomy, symptoms, diagnosis, and treatment. Am J Surg. 1976 Dec;132(6): 771-8.
127
RI PT
128 129 130
SC
131 132
M AN U
133 134 135
139 140 141 142 143 144 145 146 147
EP
138
AC C
137
TE D
136
ACCEPTED MANUSCRIPT Figure Legends
149
Figure 1: Patient MRA showing right subclavian artery (SCA) occlusion and left subclavian artery
150
stenosis. Intravenous contrast-enhanced T1-weighted MRA imaging performed as part of his
151
diagnostic work-up. The right-sided occlusion and left-sided stenosis suggests arterial-type thoracic
152
outlet syndrome, which did not completely explain the clinical symptoms and signs.
RI PT
148
153
Figure 2: Intra-operative images of his left-sided thoracic outlet decompression, using the supraclavicular
155
approach. Superficial dissection (A) shows the scalenus anterior and its relations to the phrenic nerve and
156
the nerve roots of the brachial plexus. Deep dissection (B) shows the scalenus medius fibres emerging
157
posterior to nerve roots C5 and C6 before merging with scalenus anterior.
AC C
EP
TE D
M AN U
SC
154
AC C
EP
TE D
M AN U
SC
RI PT
ACCEPTED MANUSCRIPT
AC C
EP
TE D
M AN U
SC
RI PT
ACCEPTED MANUSCRIPT
AC C
EP
TE D
M AN U
SC
RI PT
ACCEPTED MANUSCRIPT
S0890-5096(17)30696-9
DOI:
10.1016/j.avsg.2017.05.020
Reference:
AVSG 3404
To appear in:
Annals of Vascular Surgery
Received Date: 11 April 2017 Accepted Date: 7 May 2017
Please cite this article as: Lee KA, Newman JE, Garnham AW, Wall ML, Isolated Peripheral Nerve Palsies in Thoracic Outlet Syndrome, Annals of Vascular Surgery (2017), doi: 10.1016/ j.avsg.2017.05.020. This is a PDF file of an unedited manuscript that has been accepted for publication. As a service to our customers we are providing this early version of the manuscript. The manuscript will undergo copyediting, typesetting, and review of the resulting proof before it is published in its final form. Please note that during the production process errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain.
ACCEPTED MANUSCRIPT
Isolated Peripheral Nerve Palsies in Thoracic Outlet Syndrome
2
Kathryn A. Lee, Jeremy E. Newman, Andrew W. Garnham, Michael L. Wall.
3
Department of Vascular Surgery, Black Country Vascular Network, Dudley. DY1 2HQ, United Kingdom.
4 5
Corresponding Author:
Mr. Michael Wall Black Country Vascular Network
7
Russell’s Hall Hospital
8
Dudley
9
DY1 2HQ
United Kingdom
11
Email:
12
Telephone:
15 16 17 18 19 20 21
TE D
+44 (0) 1384 456 111
EP
14
[email protected]
AC C
13
M AN U
10
SC
6
RI PT
1
ACCEPTED MANUSCRIPT Abstract
23
Thoracic outlet syndrome poses a difficult diagnostic entity with varied aetiology and clinical
24
presentation. We present a 35-year-old gentleman with unilateral long thoracic nerve palsy and
25
contralateral subscapular paralysis caused by aberrant scalenus medius anatomy. Thoracic outlet
26
syndrome ought to be considered in patients presenting with isolated nerve palsies.
RI PT
22
27 28
SC
29
M AN U
30 31 32 33
37 38 39 40 41 42 43 44
EP
36
AC C
35
TE D
34
ACCEPTED MANUSCRIPT Introduction
46
Thoracic outlet syndrome (TOS) covers a large spectrum of symptomatology and pathology.
47
Differentiating venous, arterial and neurological symptoms can be complicated and tests directed at
48
certain pathologies are not always useful. We highlight an unusual case of a man with bilateral
49
isolated nerve palsies presenting with other classical thoracic outlet syndrome symptomatology
50
corrected by thoracic outlet decompression.
51
RI PT
45
Case Report
53
A 35-year-old Mechanic who enjoyed weight-lifting and boxing was referred to the Vascular
54
Outpatient Clinic from the Orthopaedic Surgeons with a seven-year history of right-sided neck and
55
scapular pain associated with intermittent ipsilateral digital pallor disturbance most prominent in
56
winter and on raising his arm. He had a long-standing winging of his right scapula and tenderness in
57
the posterior triangle of the neck on examination. There was also tingling in the ulna distribution of the
58
hand. Roos test was positive for loss of pulse on elevation and tiredness on active use of the limb.
59
Electromyography and nerve conduction study tests were unremarkable, and he had no cervical ribs
60
on chest X-ray. Magnetic resonance angiography (MRA) of his upper limbs revealed significant
61
impingement of both subclavian arteries upon elevation and external rotation.
62
EP
TE D
M AN U
SC
52
Figure 1: Patient MRA showing right subclavian artery (SCA) occlusion and left subclavian artery
64
stenosis.
65
AC C
63
66
He underwent right-sided anterior scalenectomy with division of his scalenus medius and first-rib
67
resection. The fibres of his scalenus medius muscle were found to be interdigitating with his brachial
68
plexus throughout all nerve roots. Post-operatively, he had normal sensation in his right upper limb
69
and shoulder girdle bar some numbness over his pectoralis major muscle, which was thought to be
70
neuropraxia. The winging of his scapula had resolved and he was able to return to work and back to
71
the gym.
ACCEPTED MANUSCRIPT A year later, he re-presented to clinic with a three-week history of severe pain at the medial border of
73
his left scapula that kept him awake at night, and subjective ipsilateral upper limb heaviness and
74
weakness. On examination, his scapula was not winged, but he had a positive Gerber’s Lift-Off Test
75
suggesting subscapularis paralysis. Again, Roos test was positive.
76
He underwent left-sided anterior and medius scalenectomy with first-rib resection. This time, the
77
fibres of his scalenus medius muscle passed posterior to the C5-6 trunks of the brachial plexus before
78
passing anterior to the trunks of C7, C8 and T1, causing compression at this point (Figures 2 & 3).
RI PT
72
SC
79
Figure 2: Intra-operative images of his left-sided thoracic outlet decompression, using the supraclavicular
81
approach.
M AN U
80
82
2
The Disabilities of the Arm, Shoulder and Hand (DASH) Questionnaire was used to assess his pre-
84
and post-operative upper limb function. Prior to his left thoracic outlet decompression, his DASH
85
score was 35. At his six-week follow-up appointment, his score was 13.4 and his subscapularis
86
weakness had resolved.
87
TE D
83
Conclusion
89
Roos described seven different types of congenital anomalies in his seminal paper on TOS in 1976 .
90
The discovery of fibres of scalenus medius are described best in this classification as Type 4.
91
However, this may not fully reflect our findings of scalenus medius interdigitating the trunks of the
92
brachial plexus, but highlights the different anatomies that present in this challenging operative area.
93
Isolated nerve injury is a rare clinical finding in TOS. Compression of the long thoracic nerve is not
94
unusual in shoulder pathology
95
cases of long thoracic nerve injury in TOS in the literature and these were treated with orthosis as
96
opposed to surgery . This was successful due to deformity of posture in the subjects. The anatomical
97
course of the long thoracic nerve having originated off the C5-7 nerve roots takes it anterior to
98
scalenus posterior muscle before coursing distal and laterally deep to the clavicle and superficial to
6
AC C
EP
88
3,5
but is limited in TOS. The authors were only able to identify two
4
ACCEPTED MANUSCRIPT 99
the first and second ribs. This classically is not involved in TOS but entrapment in these cases must have been present as decompression was successful.
101
The authors were unable to identify any cases of subscapularis paralysis due to TOS in the literature.
102
This may reflect a very low incidence or possibility that isolated nerve injuries are under reported. The
103
upper and lower subscapular nerves originate from the posterior cord of the brachial plexus and this
104
may have been the area of compression in this case.
105
Physicians must be aware of the possible anatomical abnormalities when investigating and treating
106
these patients. This case highlights congenital aberration in scalenus medius muscle anatomy as a
107
cause for two nerve palsies; in the long thoracic, and subscapular nerves. Clinicians presented with
108
patients with isolated nerve dysfunction should consider thoracic outlet syndrome in their differential
109
diagnoses.
M AN U
SC
RI PT
100
110 111
References
112
1. Gerber, C. and Krushell, RJ. Isolated rupture of the tendon of the subscapularis muscle. Clinical
114
features in 16 cases. J Bone Joint Surg Br. 1991 May; 73(3): 389-94.
TE D
113
2. Hudak, PL., Amadio, PC., and Bombardier, C. Development of an upper extremity outcome measure: the DASH (disabilities of the arm, shoulder and hand) [corrected]. The Upper Extremity
116
Collaborative Group (UECG). Am J Ind Med. 1996 Jun; 29(6):602-8. Erratum in: Am J Ind Med
117
1996 Sep; 30(3):372. The Institute for Work & Health are the copyright owners of the DASH and
118
QuickDASH.
AC C
EP
115
119
3. Scapular Winging: A Great Masquerader of Shoulder Disorders: AAOS Exhibit Selection.
120
Srikumaran, U., Wells, J., Freehill, M. et al., J Bone Joint Surg Am. 2014; 96: e122(1-13)
121 122 123 124
4. Nakatsuchi, Y., Saitoh, S., Hosaka, M., Uchiyama, S. Long thoracic nerve paralysis associated with thoracic outlet syndrome. J Shoulder Elbow Surg. 1994 Jan; 3(1): 28-33. 5. Freedman, J., Shankwiler, J. Nerve compression syndromes about the shoulder girdle. Current Orthopaedic Practice. 2008; 19(5): p524–529.
ACCEPTED MANUSCRIPT 125 126
6. Roos DB. Congenital anomalies associated with thoracic outlet syndrome. Anatomy, symptoms, diagnosis, and treatment. Am J Surg. 1976 Dec;132(6): 771-8.
127
RI PT
128 129 130
SC
131 132
M AN U
133 134 135
139 140 141 142 143 144 145 146 147
EP
138
AC C
137
TE D
136
ACCEPTED MANUSCRIPT Figure Legends
149
Figure 1: Patient MRA showing right subclavian artery (SCA) occlusion and left subclavian artery
150
stenosis. Intravenous contrast-enhanced T1-weighted MRA imaging performed as part of his
151
diagnostic work-up. The right-sided occlusion and left-sided stenosis suggests arterial-type thoracic
152
outlet syndrome, which did not completely explain the clinical symptoms and signs.
RI PT
148
153
Figure 2: Intra-operative images of his left-sided thoracic outlet decompression, using the supraclavicular
155
approach. Superficial dissection (A) shows the scalenus anterior and its relations to the phrenic nerve and
156
the nerve roots of the brachial plexus. Deep dissection (B) shows the scalenus medius fibres emerging
157
posterior to nerve roots C5 and C6 before merging with scalenus anterior.
AC C
EP
TE D
M AN U
SC
154
AC C
EP
TE D
M AN U
SC
RI PT
ACCEPTED MANUSCRIPT
AC C
EP
TE D
M AN U
SC
RI PT
ACCEPTED MANUSCRIPT
AC C
EP
TE D
M AN U
SC
RI PT
ACCEPTED MANUSCRIPT