Abstract
Background: Chronic hemophilic synovitis of the elbow usually leads
to enlargement and erosion of the radial head, resulting in mechanical
blockage of forearm rotation, synovial impingement, recurrent hemarthrosis,
and pain. The purpose of the present study was to evaluate the
intermediate-term results of radial head excision and synovectomy in a large
group of patients with hemophilia who had been managed at a single
institution.
Methods: Information on forty radial head excision and synovectomy
procedures that had been performed at our institution from 1969 to 2004 was
retrospectively collected. All but one of the operations had been performed in
patients with severe hemophilia. The mean age of the patients at the time of
the procedure was thirty-three years. Pain, limited range of motion, and
bleeding were the indications for surgery. The mean duration of follow-up was
7.7 years.
Results: Only one postoperative complication was observed: a
posterior interosseous nerve palsy that fully resolved by six months. No
additional surgical intervention for bleeding was required in sixteen of the
nineteen elbows in which bleeding was one of the indications for surgery. Of
the forty elbows, seven required a secondary surgical procedure at a mean of
five years after the excision of the radial head. Examination of the mean
range of motion at the time of the latest follow-up demonstrated a 63°
increase in the pronation-supination arc (p < 0.00001) but only a 2°
increase in the flexion arc.
Conclusions: Radial head excision in patients with hemophilia is an
effective procedure for improving forearm rotation and reducing pain and
bleeding frequency, with a low risk of complications.
Level of Evidence: Therapeutic Level IV. See Instructions
to Authors for a complete description of levels of evidence.
Hemophilia is an inherited, sex-linked recessive trait. Factor-VIII
deficiency (hemophilia A or classic hemophilia) accounts for 85% of cases and
has a prevalence of 1/5000 live male births. Factor-IX deficiency (hemophilia
B or Christmas disease) accounts for the other 15% and has a prevalence of
1/30,000 live male births. Twenty-five percent of new hemophilia A cases occur
in patients without any family history. These "sporadic" cases are
believed to be the result of a recent genetic mutation within the affected
family.
The clinical hallmark of hemophilia is intra-articular hemorrhage.
Following the knee, the elbow is the second most frequently affected joint in
patients with hemophilia. A single episode of hemarthrosis may give rise to
low-grade synovitis, which predisposes the joint to recurrent hemarthrosis and
ultimately to a cycle of chronic synovitis, inflammatory arthritis, and
progressive arthropathy. Chronic hemophilic synovitis of the elbow joint often
leads to enlargement of the radial head
(Fig. 1). The margins of this
hypertrophic radial head become rough and irregular, impinging against the
proximal ulnar facet. By virtue of its increased size and irregular shape, the
radial head acts as a mechanical block to forearm
rotation1,
generating synovial impingement, hemorrhage, and pain. Limited forearm
rotation can result in considerable impairment of activities of daily living,
such as eating, accepting change, and performing personal hygiene, especially
if the involved extremity is on the dominant side.
Radial head excision and synovectomy has been suggested as the treatment of
choice for hemophilic patients who present with pain, chronic hemarthrosis,
and disabling limitation of forearm rotation secondary to an enlarged radial
head and advanced arthropathy at the radiohumeral
articulation2. This
procedure has been shown to result in decreased pain and bleeding frequency
and increased forearm
rotation2-4.
However, the available literature is limited to few cases, with only
short-term follow-up.
The purpose of the present study was to evaluate the intermediate-term
results of radial head excision in a large group of hemophilic patients who
had been managed at a single institution.
From March 1969 to February 2004, fifty radial head excisions in forty-five
patients were performed at Orthopaedic Hospital, Los Angeles. Of the fifty
elbows, ten (in nine patients) were not included in the present analysis: four
patients (four elbows) lived outside the area and were lost to follow-up after
discharge from the hospital, and five patients (six elbows) had less than one
year of follow-up.
Forty radial head excision and synovectomy procedures were performed in
thirty-six patients who were followed for at least one year (mean duration of
follow-up, 7.7 years; range, 1.0 to 27.8 years). Clinical information on these
forty procedures was retrospectively reviewed after approval from our
institutional review board. Demographic data included age, the type and
severity of hemophilia, human immunodeficiency virus (HIV) status,
preoperative and postoperative range of motion, and postoperative
complications. The development of infection and/or the requirement of
additional procedures were identified.
The mean age of the patients at the time of surgery was 33.0 years (range,
15.6 to 71.9 years). Thirty-two patients (88.9%) had hemophilia A and four
patients had hemophilia B. Thirty-nine (97.5%) of the forty procedures were
performed in patients with severe hemophilia (<1% of factor VIII or IX
activity). Seven patients (19.4%) (seven elbows) were HIV-negative at the time
of surgery, and twelve patients (33.3%) (twelve elbows) were HIV-positive. In
seventeen patients (47.2%) (twenty-one elbows), the HIV status was unknown at
the time of surgery because the procedure had been performed between 1975 and
1982, before the enzyme-linked immunosorbent assay (ELISA) test for the human
immunodeficiency virus was
available5. All
seventeen patients tested positive in later evaluations and, for the purpose
of this report, were considered to have been HIV-positive at the time of
surgery.
Thorough preoperative medical and hematological evaluations, including
screening for clotting factor antibodies or inhibitors, were performed for all
patients. None of the patients had inhibitors to clotting factors at the time
of surgery. Clotting factor replacement was infused preoperatively to a
minimum level of 100% of normal, was maintained at 60% of normal for ten to
fourteen days postoperatively, and then was infused to obtain a level of 30%
of normal prior to physical therapy sessions for six to nine weeks. The
indications for surgery included pain (forty elbows; 100.0%), limited range of
motion (thirty-nine elbows; 97.5%), and chronic bleeding (nineteen elbows;
47.5%). Chronic bleeding was defined as three or more bleeding episodes per
month.
Anteroposterior, lateral, and oblique radiographs of the affected elbow
were made prior to the procedure. The status of the radiohumeral and
ulnohumeral joints was assessed, and the radiographic severity of the
hemophilic arthropathy was evaluated with use of the Pettersson
score6 (see
Appendix). The mean preoperative Pettersson score (and standard deviation) for
the radiohumeral joint was 10 ± 2.9 points (range, 5 to 13 points). The
mean preoperative Pettersson score for the ulnohumeral joint was 10 ±
2.8 points (range, 4 to 13 points). The morphology of the humeral trochlea was
carefully examined in order to determine the presence or absence of trochlear
groove deepening (Fig. 2),
which can result in ulnohumeral impingement and a reduced flexion-extension
arc. In selected cases with trochlear groove deepening and ulnohumeral
impingement, partial resection of the coronoid process was also performed.
Operative Technique
The operative procedures were performed under tourniquet control with
standard surgical techniques. Universal precautions as recommended by the
Centers for Disease Control and the American Academy of Orthopaedic Surgeons
for the prevention of transmission of HIV or hepatitis-C infection to
health-care personnel were
followed7,8.
For the purpose of radial head excision, the patient was placed in the
supine position on a standard operating table. The affected upper extremity
was placed across the patient's chest, with use of an arm rest oriented
parallel to the operating table to support the elbow. Preoperative
prophylactic antibiotics were administered intravenously. A tourniquet was
used in all cases. A standard Kocher lateral approach to the elbow was used.
Careful attention was given to avoid excessive muscle retraction and secondary
tension on the radial nerve. If necessary, the triceps was elevated from the
distal part of the humerus to gain exposure to the posterior aspect of the
ulnohumeral joint for débridement and synovectomy. Small reverse
retractors were used to facilitate the exposure of the proximal part of the
radius. A transverse osteotomy of the radial neck was performed just distal to
the ulnar facet with use of an osteotome or an oscillating saw.
It has been long recognized that the synovial tissue in patients with
hemophilic arthropathy is hypertrophic, highly vascular, and prone to
impingement between the articular surfaces, thereby increasing the likelihood
of bleeding
episodes9-12.
Synovectomy has proved to be an effective tool in the treatment of chronic
hemarthrosis12-17.
In this cohort, synovectomy was considered for patients who presented with
chronic bleeding as well as for those in whom the synovial membrane was
evidently thickened at the time of surgery. After excision of the radial head,
synovectomy of the radiohumeral joint and the lateral aspect of the
ulnohumeral joint was performed through the lateral approach.
Débridement of osteophytes from the coronoid and/or the olecranon was
performed to prevent mechanical impingement. Release of the ulnar groove with
transposition of the ulnar nerve was performed through a medial approach in
some patients with concomitant ulnar nerve symptoms. After the osteotomy, bone
bleeding was controlled with the application of bone wax to the exposed bone
surfaces. The tourniquet was deflated prior to wound closure in order to
achieve hemostasis. A 3.2-mm Hemovac drain (Zimmer, Warsaw, Indiana) was
routinely left in place. The muscle interval and the subcutaneous tissues were
then closed with use of simple, interrupted, synthetic absorbable sutures. The
skin was closed with use of running, subcuticular, monofilament nonabsorbable
sutures.
A compression dressing and plaster splint were applied with the forearm in
maximum supination for the first three to four postoperative days. Intensive
physical therapy, including active and passive motion, was started on the
fourth postoperative day. Physical therapy was performed twice a day while the
patient was in the hospital in order to maintain the forearm rotation that was
gained intraoperatively and to maintain at least the preoperative flexion arc.
After discharge from the hospital, physical therapy was continued three times
a week for at least eight weeks.
Of the thirty-six patients (forty elbows) in this series, twenty-four
(twenty-eight elbows) had died at the time of the present study, at a mean of
14.0 years (range, 1.0 to 39.8 years) after surgery. These patients died from
complications of acquired immune deficiency syndrome (AIDS) (ten patients),
hepatic carcinoma secondary to hepatitis-C infection (one patient), or
intracranial hemorrhage (three patients). For ten patients, the cause of death
was unknown. Four patients (four elbows) were lost to follow-up after a mean
of 5.7 years (range, 3.5 to 7.5 years), although extensive efforts were made
to locate them. The available clinical information on patients who had died or
had been lost to follow-up was used for the analysis. Eight patients (eight
elbows) returned to Orthopaedic Hospital, Los Angeles for a clinical
evaluation, including the Mayo Elbow Performance Index
(MEPI)18. The Mayo
Elbow Performance Index allows for the evaluation of pain, the flexion arc,
stability, and the ability to perform activities of daily living. For the
purpose of the present study, the index was modified to include the evaluation
of the arc of forearm rotation, with 20 points assigned to patients with a
rotation arc of 130°, 10 points assigned to those with a rotation arc of
70° to 129°, and 5 points assigned to those with a rotation arc of
10° to 69°. Therefore, the maximum obtainable score with the modified
Mayo Elbow Performance Index is 120 points.
Statistical analysis was performed with use of the Stata program
(StataCorp, College Station, Texas). A Student t test was used to compare
differences between groups, with use of a two-tailed level of significance of
0.05.
In addition to excision of the radial head, synovectomy was performed in
thirty-six elbows (90.0%), contracture releases were performed in one elbow,
ulnar nerve transposition was performed in one elbow, and resection of the tip
of the coronoid was performed in one elbow. During the same anesthetic
procedure, four patients underwent total knee replacement, one patient
underwent an arthroscopic synovectomy of the knee, and one patient underwent
interphalangeal fusions of the second, third, fourth, and fifth toes of one
foot.
For patients undergoing an isolated radial head excision with elbow
synovectomy, the mean duration of hospitalization was 10.1 days (range, three
to twenty-three days), with a mean requirement of 43.1 units of clotting
factor (range, 14.9 to 84.9 units) per kg per day and a mean estimated blood
loss of 34.8 mL (range, 0 to 150 mL). The duration of hospitalization has been
reduced in recent years, with a mean duration of 6.6 ± 2.4 days (range,
three to eleven days) for patients who underwent surgery in or after 1990 as
compared with a mean duration of 11.8 ± 5.2 days (range, four to
twenty-three days) for those who underwent surgery before 1990 (p =
0.006).
Recurrent chronic bleeding was observed in three (15.8%) of the nineteen
elbows in which bleeding was an original indication for excision of the radial
head and synovectomy. These three elbows underwent a secondary procedure (a
radioactive synovectomy) for bleeding control at a mean of 4.5 years (range,
2.9 to 7.5 years) after the radial head excision. No additional intervention
for bleeding was required in the remaining sixteen elbows.
There were minimal changes in the flexion-extension arc after excision of
the radial head. The mean elbow flexion contracture was 34.0° (range,
0° to 70°) preoperatively, 32.4° (range, 4° to 65°) in the
early postoperative period (within the first month after the operation) (p =
0.6), and 33.0° (range, 5° to 65°) at the time of the latest
follow-up (p = 0.8). The mean flexion arc was 76.0° (range, 25° to
135°) preoperatively, 79.6° (range, 50° to 116°) in the early
postoperative period (p = 0.5), and 78.4° (range, 25° to 115°) at
the time of the latest follow-up (p = 0.7).
In contrast, there was a significant increase in the rotation arc of the
forearm after excision of the radial head. The mean forearm pronation was
37.7° (range, 0° to 90°) preoperatively, 62.3° (range, 15°
to 90°) in the early postoperative period (p < 0.0001), and 66.9°
(range, 0° to 90°) at the time of the latest follow-up (p <
0.00001). The mean forearm supination was 30.4° (range, 0° to 90°)
preoperatively, 59.9° (range, 5° to 90°) in the early
postoperative period (p < 0.00001), and 64.0° (range, 0° to
90°) at the time of the latest follow-up (p < 0.00001). The mean
forearm rotation was 68.1° (range, 0° to 180°) preoperatively,
121.6° (range, 30° to 180°) in the early postoperative period (p
< 0.00001), and 131.0° (range, 0° to 180°) at the time of the
latest follow-up (p < 0.00001) (Fig.
3).
The modified MEPI score was obtained for eight patients postoperatively.
The severity of the arthropathy of the ulnohumeral joint in this subgroup of
patients, as assessed with the Pettersson score, was not significantly
different from that for the entire population (mean, 10 ± 2.1 points
[range, 6 to 13 points] compared with 10 ± 3.1 points [range, 4 to 13
points]). The mean modified MEPI score was 110 points (range, 100 to 115
points) (Table I). Six patients
had an excellent result (108 to 120 points), and two had a good result (90 and
107 points). No fair or poor results were observed. All of these elbows were
stable at the time of the latest follow-up. The six patients (six elbows) who
had an excellent result had no pain and no functional limitations of the
involved elbow during daily activities (including combing hair, feeding,
performing personal hygiene, and putting on shoes and shirts) despite having
limitations of the flexion-extension arc. Five of those six patients had
severe arthropathy at the ulnohumeral joint (Pettersson score, >10 points).
The two patients (two elbows) who had a good result had mild pain and a mild
limitation of flexion-extension range of motion. These two patients also had
severe arthropathy at the ulnohumeral joint (with Pettersson scores of 10 and
13 points). None of those eight patients had pain in the ipsilateral
wrist.
One patient had development of a posterior interosseous nerve palsy, which
was noted immediately after surgery. This patient had a massively enlarged
radial head that required more retraction than usual. The posterior
interosseous nerve palsy completely resolved within six months after the
operation. No other complications, including early or late postoperative
infections, were observed in this group of patients.
Of the forty elbows, seven (17.5%) underwent a secondary surgical procedure
at a mean of 5.0 years (range, 2.7 to 11.0 years) after excision of the radial
head. Three patients (three elbows) required a radiosynovectomy to control
bleeding. Three patients (three elbows) with advanced hemophilic arthropathy
presented with ulnar nerve symptoms that required ulnar nerve transposition at
a mean of 6.2 years (range, 2.7 to 11.0 years) after the index operation. In
these three patients, anteroposterior radiographs of the elbow that were made
prior to the transposition of the ulnar nerve demonstrated advanced
arthropathy of the ulnohumeral joint with the development of an exostosis on
the medial side of the ulna, perhaps made more prominent by slight valgus
angulation of the elbow (Fig.
4). This factor might have contributed to the development of ulnar
nerve symptoms. One patient (one elbow) with severe arthropathy of the elbow
that included advanced degeneration of the ulnohumeral joint underwent a total
elbow arthroplasty 5.2 years after the excision of the radial head.
We are not aware of any reports in the literature dealing specifically with
the excision of the radial head in patients with hemophilia. There have been
only scattered reports that have dealt, in general, with hemophilic
arthropathy of the
elbow2-4.
A decrease in pain and bleeding frequency as well as an increase in forearm
rotation2-4
has been observed in few patients in whom the radial head was excised. The
results of the present study, with a large number of patients and relatively
long-term follow-up, confirm the previously observed results.
A considerable reduction in the length of hospital stay has been observed
for patients with hemophilia who have undergone excision of the radial head
over the last fifteen years. This is in part a result of increased confidence
with the infusion of clotting factors. It is now our practice to allow early
discharge for patients who are reliable, proficient in self-infusing clotting
factor, and living within close proximity to the hospital in case a need for
an urgent visit to the Hemophilia Treatment Center arises.
In the present study, the complication rate associated with excision of the
radial head was low. Despite the high proportion of HIV-positive patients, no
early or late infections were observed. The only surgical complication that
was observed in this group of patients was a posterior interosseous nerve
palsy, which completely resolved within six months. As the radial head in
these patients is often large, retraction of the soft tissues around the
radial head and neck should be performed with extreme care in order to avoid
traction injury to the radial nerve. In some patients, the radial head may
need to be removed in segments.
The synovial tissue in patients with hemophilic arthropathy is
hypertrophic, highly vascular, and prone to impingement between the articular
surfaces9-12.
Synovectomy has proved to be an effective tool for the treatment of chronic
hemarthrosis in patients with
hemophilia12-17.
Open synovectomy is very successful for controlling hemarthrosis (with a
decrease in the frequency of bleeding of approximately 85%), but it requires
prolonged hospitalization and massive amounts of clotting factor replacement
and can be associated with extensive arthrofibrosis and limitation in range of
motion12-17.
Consequently, open synovectomy is rarely indicated as a primary treatment for
chronic hemarthrosis. Arthroscopic synovectomy has been shown to be nearly as
effective as open synovectomy for controlling
bleeding12-17,
but it also requires prolonged hospitalization and extensive use of clotting
factor replacement and it, too, can result in a postoperative decrease in
range of motion. Radiosynovectomy, an outpatient procedure in which a
radioisotope is injected into the affected joint, requires limited amounts of
clotting factor replacement and is highly effective for controlling bleeding
(with an observed reduction in the frequency of bleeding of >75% for more
than eight
years)12-17.
Radiosynovectomy is, therefore, our preferred method for the treatment of
chronic hemophilic hemarthrosis. However, in the case of a patient with
advanced hemophilic arthropathy of the elbow in whom excision of the radial
head is indicated to restore motion, an open synovectomy is a reasonable
adjunctive procedure.
Valgus instability has been described as a potential complication after
excision of the radial head for the treatment of a comminuted fracture,
especially in patients with insufficiency of the medial collateral
ligament19. In
general, patients with hemophilia who undergo excision of the radial head
should have a competent medial collateral ligament. Therefore, it is uncommon
to observe a considerable degree of valgus instability in this population. In
the present study, three patients with advanced arthropathy at the ulnohumeral
joint, in whom an exostosis of the medial side of the ulna had formed,
presented with late ulnar nerve symptoms that necessitated ulnar nerve
transposition. In those three patients, preoperative radiographs demonstrated
a slight valgus angulation of the elbow. It is likely that the combination of
a medial ulnar exostosis and slight valgus angulation of the elbow resulted in
increased tension on the nerve, with the consequent ulnar nerve symptoms.
It is uncommon to observe a clinically important enlargement of the radial
head in skeletally immature patients with hemophilia. Previous reports have
warned about the possibility of complications following excision of the radial
head in patients with open
physes20. Thus, the
excision of the radial head in hemophilic patients, if necessary, should be
delayed until skeletal maturity has been reached.
A considerable improvement in forearm rotation can be expected after
excision of the radial head in patients with hemophilia. This improvement in
forearm rotation usually allows the patient to recover the ability to carry
out activities that are critical for daily living, including eating, accepting
change, and performing personal hygiene, resulting in an improvement in the
quality of life. However, it is important to recognize that the
flexion-extension arc of motion of the elbow is not modified by the operation.
It is likely that arthrofibrosis (a phenomenon, commonly seen in patients with
hemophilia, in which the densely pigment-stained, hyperplasic synovial
membrane undergoes metaplasia to dense fibrous tissue) is responsible for the
observed lack in gain of flexion-extension arc of motion. Our early experience
with capsular releases in patients with severe hemophilic arthropathy of the
elbow was not encouraging. In the present study, capsular releases were
performed in one patient in an attempt to regain flexion-extension range of
motion, without success. Thus, expectations should be clearly discussed with
the patient, to ensure a full understanding of the goals of the operation: to
decrease pain, to decrease bleeding episodes, and to improve forearm rotation.
Future studies could be designed to evaluate the effectiveness of capsulectomy
combined with regional anesthesia and continuous passive motion in patients
with disabling loss of elbow flexion and extension.
Pain relief might be less predictable following excision of the radial head
in patients with generalized elbow arthropathy and severe damage to the
ulnohumeral joint. However, the majority of our patients for whom pain scores
were available reported no pain in the elbow after excision of the radial
head, despite the fact that there was advanced arthropathy of the ulnohumeral
joint. A patient undergoing radial head excision in whom the ulnohumeral joint
has advanced hemophilic arthropathy should be warned that it is difficult to
predict pain relief and that some pain might persist after the operation.
Total elbow arthroplasty has been used for the treatment of advanced,
global hemophilic arthropathy of the elbow, but only scattered reports,
including very few cases, are available in the
literature2,21,22.
Two recent case
reports21,22
included the results of twelve total elbow arthroplasties in hemophilic
patients who had a minimum duration of follow-up of two years. Six of the
twelve patients had complications, which included three infections, one
axillary vein thrombosis, one ulnar nerve palsy, and one case of persistent
pain. Although total elbow arthroplasty is an alternative, it carries an
increased risk of complications in hemophilic patients, especially infection,
and should be considered as a last resource. In patients with advanced, global
hemophilic arthropathy of the elbow, our choice remains to perform a radial
head excision and synovectomy with the goal of reducing pain and improving the
range of forearm rotation.
The present study has some limitations, including the fact that it is a
retrospective analysis. The limitations of retrospective analysis have been
demonstrated in the literature, including the difficulties in obtaining
accurate outcome information from records that were not set up for the purpose
of a specific
study23. In
addition, a large number of patients had died at the time of the present
analysis and, therefore, only a few patients returned for a current,
postoperative clinical evaluation, including the MEPI. Although clinical
information was obtained about all of the patients, it is impossible to
extrapolate the outcome information that was obtained with the MEPI for only
eight patients to the entire study population.
In conclusion, radial head excision is a simple and effective procedure
that can improve forearm rotation and decrease pain and bleeding frequency in
patients with hemophilic arthropathy of the elbow, with a minimal risk of
postoperative complications.
A table showing the Pettersson scoring system is available with the
electronic versions of this article, on our web site at
(go to
the article citation and click on "Supplementary Material") and on
our quarterly CD-ROM (call our subscription department, at 781-449-9780, to
order the CD-ROM). ?
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