Abstract
A 19-year-old healthy bodybuilder presented to the emergency department with gradually worsening pain in both his upper arms and shoulders and inability to fully flex his elbows. The haematological investigations revealed a markedly raised Creatinine Kinase (74,400 U/L) and myoglobinuria. The patient required an emergency surgical decompression of the pectoral and the anterior and posterior compartments of arms of both upper limbs with secondary closure after 48 h. The patient had an uneventful post-op and recovery of his functions with some initial restriction of full flexion of his left elbow and some weakness in his triceps, all of which gradually improved.
Keywords: compartment syndrome, pectoralis major, bilateral deltoid, bilateral triceps muscle, myoglobinuria, creatinine kinase, myoglobinuria, fasciotomy
Introduction
Compartment syndrome is a disorder of the musculoskeletal system characterised by an increase in the pressure within osteofascial compartments, which compromises the vascular supply resulting in ischaemic damage to the musculoskeletal tissues. It is commonly precipitated by trauma. Fasciotomy is a surgical procedure done to decompress the compartments under pressure. In an acutely established compartment syndrome i.e., if the onset is less than 8 h, the changes may be reversible. Beyond 8 h, sustained elevation of compartment pressure leads to necrosis of tissues. 1 This affects the renal circulation due to deposition of myoglobin released from the muscles. Surgical decompression of the closed osteofascial compartment relieves the pressure in the compartment and thus the perfusion to the muscles, nerves and bones is restored.
Case presentation
A 19-year-old male professional bodybuilder presented to the emergency department with pain in both the shoulders, pectoral area and both arms continuous for three days. Prior to the onset of symptoms, he was involved in heavy upper limb workouts in the gym. This involved bench-press of 8 sets with 10 repetitions per set with 10 s rest between the sets. He also had a push-up contest with one of his friends at the gym. The following day, the pain started insidiously and gradually progressed over the next 2 days. Paracetamol and ibuprofen did not relieve the pain. He described it to be 9/10 in severity when he presented to the emergency department. He also noted that he had been passing dark coloured urine. He was afebrile and had a good appetite. He was using anabolic steroids intermittently. There was no history suggestive of trauma or infection. He has a background of mild autism and mental health issues and indulged in excessive exercise to improve his mental health.
On clinical examination, the patient was tender over the pectoral, the deltoid and the triceps. The overlying skin was soft and supple. The triceps was tense bilaterally, left worse than the right. He had paresthaesia in the C5 and C6 dermatomes. 2 The MRC grading for motor power could not be satisfactorily done because of pain. The range of movement in bilateral wrists and fingers were unrestricted, however, the range of motion in the left elbow was between 30–80 degrees. Any passive flexion or extension beyond this was painful. Shoulder movements were also restricted due to pain. The radial pulse was well palpable with good flow. This case is important because of its bilateral involvement induced by excessive exercise.
Investigations
The patient was investigated with blood tests. The x-rays were normal, but the blood showed a significant increase in creatinine kinase (CK) levels to 74,400 U/L. Serum potassium level was found to be 4.6 mEq/L, the urine was dark coloured and a dipstick test of urine revealed myoglobinuria.
The diagnosis of compartment syndrome was established clinically. Magnetic resonance imaging (MRI) was available immediately and used as an adjunct to the clinical examination to assess the muscles and compartments involved and to plan surgical decompression. The intra-compartmental pressure monitoring was thought to be of no real diagnostic value due to the diffuse involvement and sub-acute presentation in this case and hence not used.
Treatment
The patient was admitted and intravenous fluids were started. Intravenous fluids are the first-line treatment for acute kidney injury known to occur because of tubular obstruction secondary to precipitation of protein casts. 3 It was decided to decompress the compartments and curtail the ongoing vascular insult to the musculature and neural tissue. This would also help limit the damage to the renal tissue due to myoglobinuria as decompression and surgical debridement with lavage would drastically reduce the levels of circulating myoglobin.
The surgical decompression was challenging given his build and the fact that both the anterior and posterior compartments of the arm bilaterally had to be approached at the same time. The patient was positioned in a beach chair position and both his upper limbs were draped free isolating them from the trunk. The procedure was carried out as an emergency under general anaesthesia. The arms were decompressed through the posterior midline approaches and conventional delto-pectoral extended to allow decompression of the pectorals. The deltoid and the pectoralis were decompressed by raising skin flaps along with the deep fascia. Pectoralis major and anterior deltoid were decompressed using the anterior incision whereas the middle and posterior deltoid was decompressed using the posterior incision. The radial nerve was identified and protected throughout the surgery. The muscle was noted to be pale and dusky in parts but was viable. No muscle tissue was excised. The incisions were left open with shoestring vessel loops.
Follow-up and outcomes
The patient was started empirically on broad-spectrum intravenous antibiotics and hydration with a strict elevation of the upper limbs. Postoperatively the patient was given broad-spectrum intravenous antibiotics for another 48 h, strict limb elevation and physiotherapy. IV fluid administration and concomitant fasciotomy greatly reduced the CK levels. On the 1st post-operative day, the CK levels dropped to 51,090 U/L in the morning and 37,800 U/L in the evening. The wounds were reviewed after 48 h in the theatre under general anaesthesia and after thorough lavage, it was possible to close the wounds without undue tension to the skin. The deep fascia was not closed. The CK levels dropped further to 13,266 U/L after which he was discharged and followed up as an outpatient. At the time of suture removal, he had weakness in his triceps secondary to the involvement of the deep compartment. After that, he did not attend any further appointments.
He re-presented 8 months later for some discomfort in his pectoralis major after a workout. There were no neurovascular deficits found at presentation. He was also noted to have regained the full range of motion in his upper limbs. Power in triceps was completely restored.
Discussion and conclusion
Clinical diagnosis of compartment syndrome is highly subjective. Pain out of proportion is usually the first sign followed by pain on passive stretch followed by paraesthesia. 4 Pulselessness occurs because of direct compression of the artery by the pressure when the compartment syndrome is well established. This is a late finding which can even be absent in an established compartment syndrome. 4
Fascia in the upper limb is comparatively thinner and is more expandable. 5 The number of compartments in the upper arm (flexor, extensor and deltoid) 6 are fewer and relatively large for the number of muscles contained as compared to that of the lower limb. Hence, they have room for distension. Sensory nerves are the first to be affected since they are more susceptible to compression compared to their motor counterparts. Nerve conduction studies in entrapment syndromes of the upper limb show that sensory nerve conduction is the earliest to show abnormalities. 7 This can explain why the sensory changes were first to appear in this case.
Anabolic steroids are used by bodybuilders to accomplish rapid muscle hypertrophy. They have been known to cause numerous neuro-muscular complications like upper limb mononeuropathies 8 and multi-compartment syndromes following trauma. 9 Mondelli et al. reported the mononeuropathies affecting thoracodorsal, dorsal scapular, suprascapular and medial pectoral nerves. 8 The mechanism of the mononeuropathies has been suggested secondary to pressure onto the nerves from hypertrophic muscles. Anabolic steroids lead to rapid muscle hypertrophy enclosed within a relatively inexpandable fascia which increases the compartmental pressures.
Tarkin et al. published a case of the pectoralis major and anterior deltoid compartment syndrome associated with rhabdomyolysis and renal failure in a professional tree climber. They have proposed that these muscles were affected as they were used repetitively to operate machinery. The vigorous repetitive activity led to myositis and compartment syndrome. 10 The mechanism of repeated cyclical loading and the muscle groups affected are similar to our case. A systematic review conducted by Scollan et al. listed the various aetiologies of compartment syndrome of the shoulder. Of the 17 cases, 10 of them were secondary to prolonged recumbence, either due to intraoperative positioning or substance abuse. 11 Other causes included reperfusion injury, burns, haematoma and intra-muscular injections like testosterone and vitamin B12.
The reference range for creatine kinase level in male athletes according to Mougios et al. is 82–1083 U/L. 12 The levels use in routine clinical practice for non-athletic individuals are lower. 13 In athletes the levels are usually higher because CK seeps out from the skeletal muscle spindle during exercise. Furthermore, the reference limits change according to sex, race and the type of sport involved. 12 In this case, the levels of CK were found to be 74,400 U/L on admission.
MRI is known to be one of the diagnostic tools available for compartment syndrome and it is particularly useful in sub-acute and chronic cases. Increased signal intensity in T2-weighted images of the affected muscles is usually seen in the acute phase. MRI has good sensitivity which is comparable to intra compartmental pressure (ICP) measurements however, direct compartmental pressure measurement is more specific for diagnosis.14–16 MRI was done in this case to aid clinical judgement, documentation of the extent of damage and in planning the surgery including the length of incisions and the compartments that need to be addressed.
ICP measurement is a quick and easy method to diagnose compartment syndrome and several instruments are available. Mubarak et al. demonstrated measurement by Wick catheter for diagnosis of compartment syndrome in upper and lower limbs. 17 If pressure was found to be greater than 30 mm of Hg, a fasciotomy was done. Whitesides apparatus and Stryker ICP Monitor System are well-accepted instruments. 18 Boody et al. compared the accuracy of measurement of the devices based on the type of needle tip transducer used and found that the slit catheter and 18-gauge sideport needle proved better than the 18-gauge straight needle. 18 In our case compartment pressure monitoring was not used due to the extent and diffuse involvement in several compartments.
Footnotes
Learning points: Pain out of proportion in the shoulder girdle in bodybuilders should be adequately investigated. A higher index of suspicion in patients who use anabolic steroids. Neurology of the limb should be thoroughly assessed as sensory changes are the first to appear.
Declaration of Conflicting Interests: The authors declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.
Funding: The authors received no financial support for the research, authorship and/or publication of this article.
Ethical Review and Patient Consent: Verbal consent has been obtained from the patient.
ORCID iD: A Kalaskar https://orcid.org/0000-0002-1073-8792
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