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Revisão de Charcot 2018-1
Revisão de Charcot 2018-1
ä Charcot neuroarthropathy (CN) is a systemic disease that generates pathological changes in the musculoskeletal
system, causing fractures, dislocations, and deformities involving the foot and ankle.
ä A common underlying cause of CN is diabetes mellitus; a substantial risk of ulceration and deep infection
contributes to a 15% rate of major amputation (i.e., at the level of the ankle or higher).
ä The goal of both nonsurgical and surgical treatment of CN is to obtain a stable, plantigrade foot free from
ulcerations; an equally important goal is to manage the patient’s diabetes and obesity.
ä The primary indications for surgery are recurrent ulceration, substantial deformity, deep infection, and pain;
common complications include nonunion, wound breakdown, and infection.
ä The principles of thorough joint preparation, deformity correction, and minimizing soft-tissue trauma are essential
when arthrodesis is performed for CN, which requires more robust (internal and/or external) fixation and prolonged
off-loading.
Charcot neuroarthropathy (CN) is a systemic disease that proximately 50% of patients require at least 1 operation of the
causes weakening of the musculoskeletal system (ligaments and foot1,8. Ulceration and infection are common; CN is associated
bones), which ultimately leads to collapse, fracture, and joint with a rate of major amputation (i.e., at the level of the ankle or
destruction affecting structures under substantial stress, par- higher) of 15%1,10, which increases to 35% to 67% in patients
ticularly the foot1. Jean-Martin Charcot, for whom the disease with CN who present with an associated ulcer8,11. CN is
was named, described the entity in patients with tertiary considered a major risk factor for below-the-knee amputation
syphilis in 18682. CN has been associated with diabetes melli- in the diabetic population12.
tus, spinal cord injuries, tertiary syphilis, alcoholic peripheral
neuropathy, Charcot-Marie-Tooth disease, and idiopathic Pathophysiology
neuropathy3-5. CN is commonly associated with diabetes mel- The precise pathophysiology of CN has not been conclusively
litus, specifically diabetic neuropathy1. Diabetes mellitus affects determined because of its multifactorial etiology, variable pre-
29.1 million Americans (9.3% of the U.S. population), with 8.1 sentation, and rate of progression. Historically, the 2 dominant
million of these likely undiagnosed6. The worldwide incidence pathophysiological causes of CN included the neurotraumatic
and prevalence of diabetic CN are difficult to assess accurately, and neurovascular theories. The neurotraumatic theory hy-
but the annual incidence has been reported to range from 0.1% pothesized that repetitive microtrauma in an extremity that has
to 29% and the prevalence from 0.08% to 13%3. lost protective sensation causes an unregulated inflammatory
CN has a negative impact on the quality of life of patients, cascade, eventually resulting in the collapse and joint destruction
even when diagnosed early and managed appropriately7-9. Ap- observed in CN2,13-15. In contrast, the neurovascular theory
Disclosure: No funding was provided for this study. On the Disclosure of Potential Conflicts of Interest forms, which are provided with the online version
of the article, one or more of the authors checked “yes” to indicate that the author had a relevant financial relationship in the biomedical arena outside the
submitted work (http://links.lww.com/JBJS/E642).
Clinical Presentation
Diagnosis of CN is based primarily on a thorough history and
physical examination, with corroborating laboratory investi-
gations and diagnostic imaging. Patients typically present
with a warm, swollen, erythematous foot and/or ankle5,13,16,28-31.
The affected extremity is palpably warmer than the unaffected
foot and is 3° to 5°C warmer on the affected side (Fig. 3)28,32.
Despite the presence of peripheral neuropathy, 42 (76%) of 55
diabetic subjects in a longitudinal study of acute CN treatment
presented with pain28. Typically, less than one-half of patients
with CN are aware of their neuropathy. Approximately one-
third of patients report an inciting trauma11,30.
Differentiating CN from other inflammatory disease
processes is challenging because many patients present in a
manner that mimics inflammatory diseases. The diagnosis of
early CN, prior to the development of radiographic changes, is
often missed33. A careful history, physical examination, ap-
propriate investigations (primarily radiographs), and good
clinical acumen can obtain the diagnosis in the majority. Gout,
cellulitis, and osteomyelitis have overlapping symptoms and
physical findings16. The presence of ulceration, or a history of Fig. 4
ulceration, increases the odds of underlying osteomyelitis Acute CN with superimposed infection. Note the deep ulcer overlying the
(Fig. 4)5,34. An ulcer of >4.5 cm2 is associated with a threefold lateral aspect of the ankle.
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Diagnostic Imaging
The initial workup of a patient suspected of having CN should
start with weight-bearing plain radiographs (standing antero-
posterior and lateral images of both feet and ankles)16,29,31. A
normal finding on a weight-bearing radiograph is uncommon,
but its presence does not exclude early CN; a high index of
suspicion is necessary, and clinical factors need to be consid-
ered43-45. If the diagnosis is delayed and treatment has not been
initiated, classic radiographic changes are likely to develop,
including fragmentation, demineralization, joint dislocations,
osseous debris, and joint space obliteration (Fig. 7)14,30,46. Ra-
diographs are useful for following disease progression through
the 3 stages of the Eichenholtz classification system: develop-
ment (fragmentation), coalescence, and reconstruction and
reconstitution (Table I)47,48. Notably, the Eichenholtz stages are
identical to the process by which our musculoskeletal system
responds to acute injury49,50. The main limitations of radio-
graphs are their low sensitivity and specificity for CN51. Ra-
diographic changes occur up to 2 years after diagnosis and
treatment for clinical resolution of acute CN52, reflect the end
stages of the disease, and can be considered a confirmation of
its presence; they are not a means for early intervention.
Fig. 5
Severe deformity (Fig. 5-A) and ulceration (Fig. 5-B) secondary to acute CN,
in the absence of either soft-tissue infection or osteomyelitis. This pre-
sentation can be misdiagnosed as acute infection and therefore managed
inappropriately.
Fig. 8
Figs. 8-A through 8-D A 65-year-old female patient with a 12-year history of type-II diabetes mellitus. Fig. 8-A A plantar hindfoot ulcer of the left foot, with a
circumference of 5 cm and a duration of 14 weeks. The patient had undergone local wound care with use of a removable cast-boot. Fig. 8-B The foot was
immobilized in a TCC with a metal stirrup, which allowed the patient to remain active while off-loading the foot and ankle. The cast was changed every 2 to
3 weeks. Fig. 8-C The TCC with stirrup was permanently removed after 9 weeks, and other than some central callus formation, the ulcer demonstrated
complete healing. Fig. 8-D The ulcer was fully healed, and the patient was able to bear full weight at 3 months following immobilization with the TCC.
recurrent ulceration (indicative of an unstable foot), substan- ports on surgical management of CN produced 1 small, pilot
tial axial malalignment, deep infection, and pain66. Surgical randomized clinical trial82; 1 prospective cohort study83; 2
procedures commonly performed for CN include exostectomy, retrospective cohort studies84,85; 21 prospective or retrospec-
Achilles tendon lengthening, arthrodesis, and osteotomy80,81. tive case series86-106; and 8 case reports107-114 (Table III). Fusion
Exostectomy is preferred in a stable foot (i.e., able to withstand rates ranged from 50% to 100%, with an overall fusion rate of
physiological forces) with an osseous prominence causing ul- 84.0% (489 of 582) across 28 studies83,85-97,99,100,102-107,109-114 when
ceration; if the foot is unstable, arthrodesis with deformity including stable fibrous unions, which are usually pain-free
correction is recommended. In cases of forefoot ulceration, and achieve the goal of deformity correction. Nonunion rates
Achilles tendon lengthening can be helpful in off-loading the across the same 28 studies ranged from 0% to 38%, with an
forefoot. The most common anatomic site of surgical inter- overall rate of 13.6% (79 of 582). The risk of amputation is
vention is the midfoot80. considerable, with below-the-knee amputations reported for
The literature on surgical management of CN largely 36 (5.8%) of 616 patients across 29 studies83,85-97,99-107,109-114.
comprises Level-IV studies of small case series without a Wound breakdown is often reported83,85,91,101,102, and infections
control group80. A PubMed search of English-language re- (superficial or deep, and/or around hardware or along pin
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Mean
Level Follow-
of No. of Patients Region up Outcomes and
Authors Study Design Evidence† (No. of Feet) of Foot Type of Surgery (mo) Complications
82
Wang et al. Pilot I 12 (12) in op. Midfoot Medial column 12 Postop. reulceration:
(2015) randomized group and 9 (9) arthrodesis with 0/12 in op. group and
clinical trial controls (TCC internal fixation and 3/9 (33%) in control
only) followed by TCC, group; tibial nerve
versus TCC alone conduction: same as
baseline in op. group,
and decreased
compared with baseline
in controls
83
ElAlfy et al. Prospective II 27 (27) Hindfoot Ankle arthrodesis 31 Good-to-excellent AOFAS
(2017) cohort with Ilizarov external Ankle-Hindfoot scale
fixation (n = 14) or results for 21/27
intramedullary nailing patients (78%);
(n = 13) complications in external
fixation group: 2/14
(14%) had nonunion; 8,
pin-track infections; 2,
revisions of loose pins; 3,
surgical wound infections;
and 1, wound breakdown;
complications in
intramedullary nailing
group: 3/13 (23%) had
nonunion; 2, removal and
replacement of backed-
out distal locking bolt;
and 1, superficial wound
infection
Ramanujam Retrospective III 60 (60) had Midfoot; Tarsometatarsal >6 7/116 (6%) had
84
et al. (2016) cohort (case Charcot with hindfoot arthrodesis; medial amputations, including
control) study ulceration and/or and/or lateral column 5/60 (8%) cases and
osteomyelitis; 56 arthrodesis; subtalar, 2/56 (4%) controls
(56) had Charcot double, or triple
without ulceration arthrodesis; ankle
or osteomyelitis arthrodesis; and
(control) tibiotalocalcaneal
arthrodesis; all using
circular external
fixation
85
Wukich et al. Retrospective III 177 (202) had Midfoot; 18 had soft-tissue NA 38/162 (23%) had
(2017) cohort study op. treatment; 68 hindfoot surgery; 22, primary nonunion; 18/180 (10%)
(78) had nonop. amputation; 162, subsequently required
treatment osseous surgery: transtibial amputation;
treatment of 66/180 (37%)
osteomyelitis, developed wound
exostectomy, dehiscence or op. site
osteotomy, and/or infection postop.
arthrodesis, with
internal and/or
external fixation
107
Aikawa et al. Case report IV 3 (3) Hindfoot Tibiocalcaneal fusion 21 3/3 (100%) had union;
(2016) with locking plate no intraop. or postop.
complications
Capobianco Case report IV 1 (1) Midfoot Midfoot deformity 12 Fully ambulatory
108
et al. (2010) correction using
medial column
locking plate and
screws and Ilizarov
circular external
fixator
continued
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Mean
Level Follow-
of No. of Patients Region up Outcomes and
Authors Study Design Evidence† (No. of Feet) of Foot Type of Surgery (mo) Complications
109
Cullen et al. Case report IV 4 (4) Midfoot Medial column fusion 18.5 1/4 (25%) had wound
(2013) using midfoot fusion infection and 1/4 (25%)
bolt had revision (implant
removal) at 13 mo, due to
distal migration of medial
column fusion bolt
Dalla Paola Prospective IV 45 (45) with Hindfoot Debridement and NA 39/45 (87%) had stable
86
et al. (2009) case series Charcot and single or double- ankle fusion and a
osteomyelitis stage arthrodesis plantigrade foot; 2/45
with external fixator (4%), revision
stabilization with
intramedullary nail; and
4/45 (9%), below-the-
knee amputation
Dalla Paola Prospective IV 18 (18) Hindfoot Ankle arthrodesis, 14 14/18 (78%) had
87
et al. (2007) case series tibiocalcaneal complete osseous union;
arthrodesis using a 4/18 (22%), fibrous
retrograde nail union, and were
ambulatory with a custom-
molded ankle foot
orthosis; no amputations
Early and Retrospective IV 18 (21) Midfoot Tarsometatarsal 28 2/21 (10%) had
89
Hansen (1996) case series fusion, subtalar nonunion; 2/21 (10%),
fusion, triple below-the-knee
arthrodesis, or amputation; 1/21 (5%)
pantalar fusion died (myocardial
infarction on 3rd postop.
day); 1/21 (5%) had
superficial infection
90
Ettinger et al. Retrospective IV 12 (12) Hindfoot Tibiocalcaneal >12 AOFAS Ankle-Hindfoot
(2016) case series arthrodesis, with scale improved by a
external fixation (n = mean of 42.4 points; in
7), intramedullary nail intramedullary nail group:
(n = 4), or dorsal 4/4 (100%) had fusion; in
plaster cast (n = 1) external fixation group:
4/7 (57%) had clinical
and radiographic fusion
and 3/7 (43%), moderate
radiographic fusion with
stable asymptomatic
pseudarthrosis
91
Grant et al. Retrospective IV 44 (50) Midfoot; Achilles tendon 30 7/50 (14%) had
(2009) case series hindfoot lengthening, nonunion; 2/50 (4%),
arthrodesis (Lisfranc, amputation; 13/50
midtarsal, and (26%), pin-track
midfoot) with internal infections; 9/50 (18%),
fixation and external wound dehiscences;
fixation for 8/50 (16%),
compression osteomyelitis; and 1 had
deep vein thrombosis
92
Herscovici et al. Retrospective IV 12 (12) Hindfoot Pantalar arthrodesis 46 Posttraumatic arthritis
(2011) case series posttraumatic using an cohort: 2/12 (17%) had
arthritis; 8 (8) intramedullary nail ankle nonunion and 1/12
Charcot (n = 11) or plates (8%), subtalar malunion;
and/or screws (n = 9) Charcot cohort: 3/8
(38%) had ankle
malunion; 2/8 (25%),
deep infection; and 1/8
(13%), superficial
infection
continued
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Mean
Level Follow-
of No. of Patients Region up Outcomes and
Authors Study Design Evidence† (No. of Feet) of Foot Type of Surgery (mo) Complications
continued
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Level Follow-
of No. of Patients Region up Outcomes and
Authors Study Design Evidence† (No. of Feet) of Foot Type of Surgery (mo) Complications
88
Richter et al. Prospective IV 47 (48) Midfoot; Variety of procedures 12 47/48 (98%) had union;
(2015) case series and to correct deformity, 1/48 (2%), nonunion;
hindfoot using a midfoot and 3/48 (6%), surgical
fusion bolt revision due to loss of
correction; 5/48 (10%),
amputation (2 below-
the-knee, 1 midfoot,
and 2 forefoot); 3/48
(6%), bolt loosening;
10/48 (21%), wound-
healing problem; and
8/48 (17%), deep
wound infection
Sammarco and Retrospective IV 26 (27) Forefoot; Arthrodesis with 27.9 2 patients required
99
Conti (1998) case series midfoot; internal fixation: further reconstructive
hindfoot reduction of rocker- surgery; 2 patients
bottom deformity; required screw removal;
triple arthrodesis; no deep infections; no
ankle arthrodesis amputations; and 6/27
(22%) had nonunion
Sammarco Retrospective IV 22 (22) Midfoot Midfoot arthrodesis 52 16/22 (73%) had full
100
et al. (2009) case series with osseous union; 5/22 (23%),
resection and partial union (a single
osteotomy joint did not unite in an
otherwise stable foot);
1/22 (5%), nonunion; 1,
soft-tissue infection;
and no amputation
101
Schon et al. Retrospective IV 50 ankles; 22 Forefoot; Tibiocalcaneal NA In tibiocalcaneal fusion
(1998) case series hindfeet; 131 midfoot; arthrodesis (n = 6); group: 2/6 (33%) had
midfeet; and 18 hindfoot hindfoot fusion (n = solid union; 2/6, stable
forefeet 11); midfoot ORIF (n = pseudarthrosis; and
4); midfoot biplanar 2/6, below-the-knee
osteotomy and amputation; in hindfoot
arthrodesis (n = 37); fusion group: 1/11 (9%)
forefoot cheilectomy had below-the-knee
(n = 4); forefoot amputation; in midfoot
resection fusion group: 12/37
arthroplasty (n = 2); (32%) had wound
and complications; and in
metatarsophalangeal forefoot group, no
joint fusion (n = 1) complications
102
Shah and De Retrospective IV 11 (11) Hindfoot Tibiotalar arthrodesis 38 In internal fixation group:
(2011) case series using intramedullary 0 had nonunion; 1/5
nails (n = 5) or (20%), wound breakdown;
external fixator (n = 6) 1/5, superficial infection;
and 1/5, removal of distal
interlocking bolt after
osseous union; in
external fixation group:
2/6 (33%) had union;
2/6, stable fibrous
nonunion; 2/6, nonunion;
4/6 (67%), revision of
external fixation; 1/6
(17%), below-the-knee
amputation; 3/6 (50%),
wound infection; and 2/6
(33%), wound breakdown
continued
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Authors Study Design Evidence† (No. of Feet) of Foot Type of Surgery (mo) Complications
*TCC = total contact cast, AOFAS = American Orthopaedic Foot & Ankle Society, NA = not available, FAAM = Foot and Ankle Ability Measure, SF-36 = Short Form-36, ORIF =
open reduction and internal fixation, and MTPJ = metatarsophalangeal joint. †Level of evidence assigned according to the table, “Levels of Evidence for Primary Research
Question” by The Journal of Bone & Joint Surgery (revised January 2015), which is available at http://journals.lww.com/jbjsjournal/Pages/Journals-Level-of-Evidence.
aspx, accessed November 3, 2017. ‡1 patient experienced multiple complications: osteomyelitis, tibial stress fracture, and malunion.
tracks) are common66,80,83,85,88,89,91,92,94,96,100,102,104,106,109. In part, these press soft tissues, use of graft material to fill ‡50% of the
complications may reflect the advanced stage of CN at the time fusion space116, extension of fixation past the injury zone into
of surgery. solid bone, and use of locking plate technology may help to
increase union rates and decrease soft-tissue complications115.
Arthrodesis Axial screw placement, or “beaming” with a midfoot fusion
When arthrodesis is performed, more robust fixation and bolt (Fig. 9), is a less invasive method of obtaining rigid in-
prolonged immobilization and non-weight-bearing may im- ternal fixation and compression across extensive midfoot
prove fusion rates. The concept of “superconstructs” high- involvement117, and high fusion rates with low complication
lights the need to manage CN differently than standard rates have been reported88,100,103,109. These are judgments that
arthritic conditions115. Adequate bone resection to decom- are based on surgeon experience and patient-related factors,
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adhering to the basic surgical principles of good joint prep- Recommendation Grade*
aration and deformity correction.
The diagnosis of Charcot neuroarthropathy C
In the setting of ulceration or active infection, external (CN) is based on a thorough history and
fixation with ring fixators has been described with satisfactory physical examination.
outcomes118,119. Some authors have recommended the use of Advanced diagnostic imaging is usually not C
external fixation for feet with active infection, large soft-tissue required for the diagnosis and management of
defects, and poor bone quality and the use of internal fixation uncomplicated CN.
for osseous reconstruction of the foot with CN without a The goals of treatment of CN are to obtain a C
wound or active infection and good bone quality120. A recent stable, plantigrade, ulcer-free foot.
systematic review of 11 studies describing 126 feet with CN Nonsurgical management of CN includes C
found that internal fixation had a reduced ulceration rate, immobilization in a cast and off-loading of the
decreased risk for nonunion, and increased return to functional affected extremity.
ambulation compared with external fixation, but also had a Indications for surgery for CN include recurrent C
higher rate of overall complications, including amputation, ulceration, deep infection, severe deformity,
deep infection, and wound-healing complications121. Modern, and uncontrolled pain.
rigid, locking, low-profile plate-and-screw constructs have Adequate joint preparation, robust fixation, C
been designed for CN to address some of the shortcomings of and prolonged immobilization and off-loading
traditional internal fixation constructs, but these have yet to be are essential when arthrodesis is performed.
studied in a scientific method to support industry claims. 127
*According to Wright , grade A indicates good evidence (Level-I
Most patients with CN who require surgical interven- studies with consistent findings) for or against recommending in-
tion are in a late stage of diabetes and have advanced end- tervention; grade B, fair evidence (Level-II or III studies with con-
organ disease. Surgical stabilization of a foot with CN is often sistent findings) for or against recommending intervention; grade C,
considered a palliative intervention to allow the patient an poor-quality evidence (Level-IV or V studies with consistent findings)
for or against recommending intervention; and grade I, insufficient
improved level of function before mortality from myocardial or conflicting evidence not allowing a recommendation for or
infarction, renal failure, or stroke122. These factors should be against intervention.
considered before subjecting the patient to the prolonged
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success in foot and ankle arthrodesis, but not specifically in the sition to optimize all aspects of care for this debilitating chronic
setting of CN. disease. n
NOTE: The authors thank Dagmar Gross for assistance with the literature search and preparation of
the manuscript.
Overview
Charcot neuroarthropathy remains a debilitating disease that is
challenging to manage. Unfortunately, in a substantial number
of patients, the diagnosis of CN is a reflection of an advanced
Andrew Dodd, MD, FRCSC1
stage of the disorder associated with diabetes-related end-organ Timothy R. Daniels, MD, FRCSC2
disease. The goals of management are to obtain a stable,
plantigrade, ulcer-free foot, with minimal disruption to the 1Division of Orthopaedic Surgery, University of Calgary, Calgary,
patient (Table IV). This includes proper management of the Alberta, Canada
diabetes, realistic expectations, counseling, improved methods 2Division
of off-loading the extremity (e.g., a metal stirrup cast), ap- of Orthopaedic Surgery, St. Michael’s Hospital and University of
Toronto, Toronto, Ontario, Canada
propriate treatment decisions (nonoperative and operative),
and appropriate operative techniques. A multidisciplinary ap- E-mail address for T.R. Daniels: danielst@smh.ca
proach with involvement of the entire circle of care is optimal.
Fear of amputation often places the surgeon in a unique po- ORCID iD for T.R. Daniels: 0000-0003-0962-6977
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