Abstract
A 26-year-old woman developed acute compartment syndrome (ACS) of her right hand secondary to reperfusion syndrome. She suffered an out-of-hospital cardiac arrest following a pregabalin overdose. Attending paramedics mistakenly gave intra-arterial epinephrine into her right brachial artery. On resolution of her brachial artery spasm, she developed a reperfusion injury to her right hand and subsequently ACS. A four-incision fasciotomy with carpal tunnel decompression was performed and was successful in reversing focal ischaemia and an irreversible functional deficit. This case demonstrates an unusual case of hand ACS secondary to temporary limb ischaemia and reperfusion syndrome following iatrogenic intra-arterial epinephrine administration. We also summarise the current available literature on ACS of the hand including the aetiology, treatment and use of an intracompartmental monitor.
Keywords: orthopaedics, orthopaedic and trauma surgery
Background
Acute compartment syndrome (ACS) is an emergency in which intracompartmental pressure can lead to tissue hypoperfusion and irreversible tissue damage if left untreated. The diagnosis is made based on the associated signs and symptoms including pain out of proportion to the injury, pain on passive stretch of muscles in the affected compartment, swelling and in the late stages, sensorimotor deficit and loss of peripheral pulse.1 Intracompartmental pressure measurements can be used in times of uncertainty to help guide the decision regarding performing fasciotomies.2 ACS is most commonly seen in the leg and forearm following trauma3 and is rarely seen in the hand. However, it can lead to devastating loss of function if missed or managed ineffectively. There is uncertainty regarding the spectrum of aetiologies causing hand ACS and how this typically presents.
In this case report we present an unusual case of hand ACS which had good outcomes following four-incision fasciotomies and carpal tunnel decompression. We also summarise the current available literature on ACS of the hand including the aetiology, treatment and use of an intracompartmental monitor. The patient gave informed consent for submission and publication of the data.
Case presentation
A 26-year-old woman with a history of asthma, and borderline personality disorder suffered an out-of-hospital cardiac arrest following an overdose of pregabalin. Paramedics were summoned at noon when her partner first noticed her to be unresponsive and snoring. Initial cardiac tracing showed pulseless electrical activity and cardiopulmonary resuscitation was commenced. No direct-current shocks were given. A cannula was placed into her right antecubital fossa (ACF) and 2 mg of epinephrine and 400 µg of naloxone were administered. She had approximately 20 min of down time followed by spontaneous return of circulation and was with a Glasgow Coma Scale 3 on arrival into the emergency room. She was intubated and ventilated and a right internal jugular central venous catheter and left femoral arterial line were sited. Her background included five previous paracetamol overdoses, alcohol consumption of 6–8 units per day and smoking 1 g of cannabis and 10 cigarettes per day. She had no known allergies.
Approximately 6 hours later, it was noted that her hand had delayed capillary refill and was pale, cold. Peripheral pulses were intact. Doppler ultrasound revealed triphasic flow through the brachial, radial and ulnar arteries. The right ACF cannula was removed with high pressure, arterial flow from the puncture site noted and compression bandaging required to stem the bleeding. No further surgical review was planned at this stage and she was transferred to the intensive care unit (ICU).
Investigations
The patient required norepinephrine to maintain a mean arterial pressure of >65 mm Hg. Her initial acute kidney injury began to improve in the ICU including a reduction in her potassium from 6.2 mmol/L to 2.5 mmol/L, however her lactate remained high (5.9 mmol/L). Her initial creatine kinase in the emergency room was 7500 U/L which increased to 47 000 U/L the following day and the nurses noted the patient’s hand appeared blue and mottled. Radial and ulnar pulses were still palpable. At 07:00, the orthopaedic registrar was called to review the patient. The hand was cyanotic and cool to touch (figure 1A, B). There was a firm, fullness to the thenar and hypothenar eminences and the dorsum of the hand. A clinical diagnosis of ACS was made, and the patient was immediately transferred to the operating room for urgent fasciotomies. The decision to decompress was with the knowledge the hand was not displaying these features at 23:00 the night before; a time less than 12 hours. No further studies were obtained.
Figure 1.
(A, B) Focal hand ischaemia prior to emergency fasciotomies.
Treatment
Fasciotomies were performed over the thenar and hypothenar eminences as well as on the dorsum of the hand in line with the index and ring finger metacarpals (figure 2A, B). A carpal tunnel release was also performed. There was significant muscle bulging out of the thenar and hypothenar eminences however the muscle looked healthy. There was gross oedema over the muscle bellies within the dorsal incision and the muscle appeared slightly discoloured however, it appeared alive. The carpal tunnel incision was closed. Immediate improvement in the colour and capillary refill time of the patient’s hand was noted. The hand was dressed, and the patient moved back to ICU. They returned to theatre 24 hours later for debridement of some muscle necrosis of the dorsum of the hand. The patient was brought back to theatre a third time, 72 hours post-fasciotomies, for closure of the wounds except the most radial of the dorsal wounds. This healed by secondary intention.
Figure 2.
(A, B) Fasciotomy wounds 48 hours following surgery.
Outcome and follow-up
The hand was pain-free on discharge from hospital with some residual intrinsic tightness and grip strength of medical research council grade 4+. The patient was referred for outpatient hand therapy following discharge and the deficits had resolved when reviewed in the clinic at 3 months following surgery. The patient was followed up in a telephone clinic at 9 months post-surgery and was actively looking for employment and did not have any concerns regarding her current hand function.
Discussion
This case demonstrates an unusual case of ACS of the hand secondary to a reperfusion injury after relaxation of a brachial artery spasm which was caused by intra-arterial epinephrine during resuscitation. Ideally, fasciotomies would have been performed sooner however, this case highlights the rarity of hand ACS which few physicians or surgeons have encountered in their practice. It is for this reason that we have reviewed the current available literature reporting on hand ACS, and this can be seen in table 1.
Table 1.
The aetiology, age and treatment of compartment syndrome of the hand in the current literature. If more than two cases were included the age was represented as the mean and range
| Author | Year | No. of cases | Aetiology | Age | Treatment | Isolated compartment | Compartment monitor |
| Abidin and Jalaluddin9 | 2008 | 1 | Henoch-Schönlein purpura | 9 | Fasciotomy | No | No |
| Abdul-Hamid10 | 1987 | 1 | Exertional | Unknown | Fasciotomy | First dorsal interosseous | Unknown |
| Basaran et al11 | 2009 | 1 | Human bite | 41 | Fasciotomy | Not specified | No |
| Belzunegui et al12 | 2011 | 1 | Extravasation of contrast | 50 | Fasciotomy | No | No |
| Chokshi et al13 | 1998 | 1 | Reflex sympathetic dystrophy | 40 | Fasciotomy | No | Yes |
| Cosker et al14 | 2004 | 1 | Suction | 68 | Fasciotomy | Not specified | Yes |
| D'Asero et al15 | 2010 | 1 | Extravasation of contrast | 80 | Fasciotomy | Not specified | No |
| de Blacam et al16 | 2012 | 1 | Crush | 56 | Fasciotomy | No | No |
| Del Piñal et al7 | 2002 | 11 | Closed crush injury | 35 (20–57) | Fasciotomy | Varied | No |
| Dolan et al17 | 2012 | 1 | Metacarpal fractures | 44 | Fasciotomy | No | No |
| Dwyer et al18 | 2017 | 1 | Exertional | 44 | Fasciotomy | Not specified | Yes |
| Funk et al4 | 1999 | 2 | Intra-arterial drug | 24, 28 | Fasciotomy | Not specified | No |
| Gainor19 | 1984 | 1 | Flexor digitorum superficialis rupture | Unknown | Unknown | Not specified | Not specified |
| Gallagher and Ruiter20 | 2015 | 1 | Spontaneous arterial haemorrhage | 70 | Fasciotomy | Not specified | No |
| Horlocker and Bishop21 | 1995 | 1 | Brachial artery cannulation | 16 | Fasciotomy | Not specified | Yes |
| Hung et al22 | 1988 | 1 | Necrotising fasciitis | 52 | Amputation | Not specified | No |
| Jue et al23 | 2017 | 1 | Transradial cardiac catheterisation | 64 | Fasciotomy | Thenar | Yes |
| Kies et al8 | 2004 | 79 | Multiple causes | 43 | Fasciotomy | Not specified | Not specified |
| Lee et al24 | 2012 | 1 | Exertional | 34 | Fasciotomy | Thenar | Yes |
| Malik et al25 | 2011 | 1 | Hereditary angioedema | 13 | Fasciotomy | No | No |
| Martin and Treharne26 | 2016 | 1 | Birth | 1 month | Fasciotomy | No | No |
| Mehta et al27 | 2018 | 1 | Crush | 44 | Amputation | No | No |
| Mull et al28 | 2015 | 1 | McArdle disease | 40 | Fasciotomy | Volar only | Yes |
| Neth29 | 2019 | 1 | Unknown | 67 | Fasciotomy | Thenar | Yes |
| Onal et al30 | 2015 | 1 | Radial artery cannulation | 67 | Amputation | Not specified | No |
| Orta et al5 | 2018 | 1 | Exertional | 30 | Incobotulinum A toxin | First dorsal interosseous | No |
| Ouellette and Kelly6 | 1996 | 19 | Intravenous injections (11), gunshot (2), crush (3), arterial line complication (2), postoperative wrist arthrodesis (1) | Unknown (0–67) | Fasciotomy | Mixed | Yes |
| Pentz et al31 | 2018 | 1 | Protein S deficiency | 48 | Fasciotomy | No | Yes |
| Petratos et al32 | 2011 | 1 | Wasp sting | 6 | Fasciotomy | No | No |
| Phillips et al33 | 1986 | 1 | Exertional | 21 | Fasciotomy | First dorsal interosseous | Yes |
| Quigley et al34 | 1981 | 1 | Extravasation of intravenous fluid | 25 | Fasciotomy | No | No |
| Reichman35 | 2016 | 1 | Carpal fracture/dislocation | 37 | Fasciotomy | No | Yes |
| Rios-Alba and Ahn36 | 2015 | 1 | Elastic bandage | 11 months | Fasciotomy | No | Yes |
| Sawyer et al37 | 2010 | 1 | Wasp sting | 5 | Fasciotomy | No | Yes |
| Seiler et al38 | 1996 | 1 | Extravasation of intravenous fluid | 44 | Fasciotomy | Not specified | Yes |
| Selek et al39 | 2007 | 1 | Extravasation of contrast | 70 | Fasciotomy | Dorsal only | No |
| Sharma et al40 | 2013 | 1 | Crush | 38 | Fasciotomy | Not specified | No |
| Shin et al41 | 1996 | 1 | Pool suction | 19 months, 24 months | Fasciotomy | No | Varied |
| Silveri et al42 | 1997 | 1 | Rupture of transverse carpal ligament | Unknown | Fasciotomy | No | Not specified |
| Söderberg43 | 1996 | 2 | Exertional | 32, 20 | Fasciotomy | No | Yes |
| Song et al44 | 2015 | 1 | Transradial cardiac catheterisation | 84 | Spontaneous rupture | Dorsal only | No |
| Stavrakakis et al45 | 2018 | 1 | Extravasation of contrast | 72 | Fasciotomy | No | No |
| Stein et al46 | 2003 | 1 | Extravasation of contrast | 51 | Unknown | Not specified | Not specified |
| Styf et al47 | 1987 | 4 | Exertional | 29 (20–39) | Fasciotomy | First dorsal interosseous | Yes |
| Sveen et al48 | 2018 | 1 | Restrictive splint | 17 months | Fasciotomy | No | No |
| Talbot and Rogers49 | 2011 | 2 | Extravasation of intravenous fluid | 10 months | Fasciotomy | No | Yes |
| Tanagho et al50 | 2015 | 1 | Systemic sclerosis | Unknown | Fasciotomy | Not specified | No |
| Tetreault et al51 | 2018 | 1 | Birth | Neonate | Fasciotomy | Not specified | Not specified |
| Thomas et al52 | 1994 | 1 | Thrombolysis | 35 | Fasciotomy | No | No |
| Tucker and Josty53 | 2005 | 1 | Snake bite | 42 | Fasciotomy | Thenar | Yes |
| Varacallo et al54 | 2018 | 1 | Propofol extravasation | 52 | Fasciotomy | No | Yes |
| Venditto et al55 | 2017 | 1 | Paediatric hereditary angioedema | 13 | Fasciotomy | No | Yes |
| Werman et al56 | 2013 | 2 | First metacarpophalangeal joint fracture/dislocation | 16, 26 | Fasciotomy | Thenar (in one case) | Yes (in one case) |
| Whatling and Galland57 | 1999 | 1 | Post-brachial embolectomy | 72 | Fasciotomy | Thenar, first dorsal interosseous | No |
| Wirth et al58 | 2008 | 2 | HIV-induced vasculitis | 41, 49 | Fasciotomy | No | Yes |
| Yurdakul et al59 | 2014 | 1 | Extravasation of contrast | 60 | Fasciotomy | Dorsal only | No |
This is the first case to describe ACS of the hand secondary to intra-arterial epinephrine during resuscitation and explore the theory of reperfusion syndrome. Funk et al reported two cases of hand ACS following injection of heroin into the radial and brachial artery.4 Both cases made full recoveries following fasciotomies. It is well documented that intra-arterial epinephrine into a peripheral limb leads to vascular spasm. At the level of the brachial artery, no collateral blood is available to supply distal perfusion to the limb. In the case presented, this ischaemic period may have been up to 6 hours followed by spontaneous reperfusion.
A review of the literature demonstrated causes for hand ACS are most frequently attributed to extravasation of intravenous fluid, contrast or medication from the venous vasculature of the hand. Trauma was a common aetiology and this included metacarpal fractures, a fracture/dislocation of the carpus and crush injuries to the hand. Interestingly, a number of case reports of exertional compartment syndrome of the hand have been reported. These typically affect the first dorsal interosseous but have also been reported in the thenar eminence. The diagnosis of exertional hand compartment syndrome can be susceptible to delay secondary to its rarity and unfamiliarity with clinicians. Treatment typically included fasciotomies however, one case in the literature was successfully treated with Incobutulinum A toxin.5
Included in the literature review were three larger case series. The first was performed by Ouellette and Kelly6 and reported a series of 19 patients with hand ACS of which 11 were caused by extravasation of intravenous fluid, 3 were crush injuries, 2 gunshot wounds, 2 arterial line complications and 1 following wrist arthrodesis. Del Piñal et al7 retrospectively reviewed 11 cases of hand ACS secondary to closed crush injuries over a 5-year period. They found the injury to be sustained entirely by men involved with manual labour. Six had the ‘generalised form’ affecting all compartments, three had the ‘localised form’ affecting the thenar and first web space muscles, two cases also affected the second web space and two cases were the atypical form which affected the second and third web spaces and the hypothenar eminence. Following treatment 9 of the 11 patients returned to manual labour. The final study by Kies et al8 included five cases over a 15-year period sustained following operative procedures where the mechanism was thought to be secondary to crushing of the hand with the draw sheets when the patient was positioned.
Learning points.
This case demonstrates an unusual cause for acute compartment syndrome (ACS) of the hand following reperfusion syndrome secondary to brachial artery spasm and a requirement for high degrees of clinical suspicion when reviewing patients with suspected acute ischaemia of the hand.
Four compartment fasciotomies and carpal tunnel release can be a successful treatment option if performed in a relatively timely manner.
The current literature would suggest that the most common causes of hand ACS are secondary to extravasation of intravenous fluids and crush injuries.
Footnotes
Contributors: PR was involved in the conception and design, acquisition of data or analysis, interpretation of data, writing up of the article. JML was involved in the acquisition of data or analysis, interpretation of data and editing the article. IB is the senior author of the study and was involved in the editing and final approval of the version published.
Funding: The authors have not declared a specific grant for this research from any funding agency in the public, commercial or not-for-profit sectors.
Competing interests: None declared.
Patient consent for publication: Obtained.
Provenance and peer review: Not commissioned; externally peer reviewed.
References
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