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Abstract
For assessing the clinical
features and outcome of treatment in brucellar spondylitis, this
study was conducted on 38 cases with brucellar spondylitis from
April 1998 through December 2004. Twenty-five (65.8%)
cases were males. The mean age of the patients was 45.9(±17)
years. Back pain, sweating, fever and arthralgia, were the most
common clinical manifestations. The disease was acute in 19
(50%), sub- acute in 16 (42.1%) and chronic in 3 (7.9 %) of the
cases. Brucellar spondylitis was seen in 31 (81.6%) cases in
lumbar and lumbo-sacral, in 2 (5.3%) cases in dorsal and in 3
(7.9%) cases in cervical regions. Twelve cases were treated with
doxycycline plus Rifampicillin for 4 months. Relapse was seen in
2 (16.6%) cases. Relapse was seen in 1 (9.1%) of the eleven
cases who were treated with cotrimoxasol plus Rifampicillin for
4 months. Relapse was not seen in fifteen cases treated with
streptomycine for 14 days plus doxycycline and Rifampicillin for
4 months. Brucellar spondylitis may develop in any site of the
spine mostly in lumbar region. Streptomycin for 14 days and
doxycycline plus Rifampicillin for four months is a better
regimen for therapy of brucellar spondylitis.
Keywords: Brucellar spondylitis,
clinical manifestations, treatment
J.Orthopaedics 2006;3(1)e2
Introduction:
Brucellosis is still an important public
health problem and is endemic in many countries throughout the
world including Iran 1-3. Osteoarticular disease is the most
common complication of brucellosis and has been described in
10-85% of patients 1,4. Spondylitis is the most prevalent and
important clinical form of osteoarticular involvement in adults
with infection due to brucella species 5-7
Brucellosis is a relative common cause of
vertebral osteomyelitis in endemic areas 8-11. This complication
was reported between 8-13.8% 5,7,12,13. Spondylitis may be
complicated by potentially devastating neurological defects that
must be considered in endemic areas 14,15.It may be difficult to
diagnosis and can be complicated by potentially devastating
neurological or vascular conditions 16,17. Treatment of
brucellar spondylitis remains controversial regarding the
selection of antibiotics, the duration of treatment and the role
of surgery .It is often debilitating and difficult to treat and
there is no consensus on the preferred combination of antibiotic
14. The purpose of this study was to assess the clinical
manifestations and outcome of treatment in 38 cases of brucellar
spondylitis in Babol, northern Iran.
Material and Methods :
From April 1998 through December 2004,
thirty-eight cases of brucellar spondylitis were diagnosed and
followed prospectively at the General Hospital of Yehyanejad,
Babol, north of Iran. This hospital serves more than 500000
people living our region and department of infectious diseases
serves more than two million people living in the west of the
province Mazanderan, Iran. The
 diagnosis
of brucellosis was established by standard tube agglutination
test (STAT) with a titre ≥1: 320 and a titre in
2-mercaptoethanol (2-ME) ≥1: 160 for patients with clinical
signs and symptoms compatible with brucellosis. For spondylitis,
the radiographic findings were recorded by determining
epiphysitis of the anterosuperior angle of the vertebra,
narrowing of the disc space, erosion, sclerosis, vertebral
collapse and osteomyelitis. Diagnosis of spondylitis was
confirmed by magnetic resonance imaging (MRI). We also assessed
CBC, hemoglobin, erythrocyte sedimentation rate and C-Reactive
Protein (CRP) in all cases, and findings compared with the
normal ranges in references 18. Clinical type (the onset of
clinical symptoms and signs to diagnosis) was classified as
acute (less than two months) and sub- acute (more than two
months to on year) and chronic (more than one year). Three
regimens of therapy were given to the patients as following:
Doxycyclie plus Rifampicillin to all patient admitted from April
1998 to September 2000,Co-trimoxasol plus Rifampicillin to all
patients from October 2000 to April 2001 and Streptomycin
1gr/day for two weeks plus doxycycline and Rifampicillin was
given to all patients from May 2001 to December 2004. The dosage
of doxycycline was 100 mg twice daily, and dosage of co-
trimoxasol was 6 mg/kg/day of the trimethoprime component
divided into 2 doses and the dosage of Rifampicillin was 15mg/kg
once a day and streptomycin 1gr/IM/day. All of these
combinations were given for four months.
Patients were assessed every 20 days during
therapy and reassessed every three months interval after
completion of therapy as well as whenever clinical symptoms
reappeared. Duration of follow - up for all cases was one year.
Relapse was said to occur when the indicative
clinical picture reappeared and reduced titer of STAT or 2 ME
after treatment, increased again. Symptoms or signs of the
disease that persisted at the end of treatment defined
therapeutic failure. X2- test and Fisher’s exact tests (when
appropriate) were used for categorical variables and Student’s
t-test was used to compare mean values. Differences with a P
value of < 0.05 were considered significant.
Results :
Sixteen patients underwent the procedure in
the last 2 years. All of them were referred from the orthopedics
department. All were done electively. Seven patients underwent
the procedure for defects in the region of the Achilles tendon –
either immediately for inadequate soft tissue cover, or for
later skin necrosis post repair. Nine patients underwent the
procedure for exposed tibia in the lower third of the leg.
Table 1: Clinical manifestation and laboratory test
results in 38 cases of brucellar spondylitis
|
|
Male
N=25 |
Female
N=13 |
Total |
|
|
|
|
|
|
Type of
disease: |
|
|
|
|
Acute |
14(56) |
5(38.5) |
19(50) |
|
Sub
acute |
8(32) |
8(61.5) |
16(42.1) |
|
Chronic
Back
pain |
3(12)
25(100) |
0(0)
13(100) |
3(7.9)
38(100) |
|
Fever |
19(76) |
9(69.2) |
28(73.7) |
|
Sweating |
20(80) |
11(84.6) |
31(81.6) |
|
Artheralgia |
9(36) |
4(30.8) |
13(34.2) |
|
Normal Hb |
17 (68) |
12(92.3) |
29(76.3) |
|
CRP+ |
17(68) |
12(92.3) |
29(76.3) |
|
Leucocytosis |
6(24) |
3(23.1) |
9(23.7) |
|
Predisp
factor |
15(60) |
6(46.2) |
21(55.3) |
|
Normal ESR |
20(80) |
10(76.9) |
30(78.9) |
CRP; C- Reactive Protein, ESR; Erythrocyte Sedimentation
Rate, Hb; Hemoglobin. There were not any differences in clinical
manifestations and test results between sexes (P>0.05%)
Out of the 16 patients, 2 were females and
the rest, males. The ages ranged from 14 years to 65 years, the
average being 36.8. Two people were diabetics and 2 had
peripheral vascular disease as demonstrated by color Doppler.
Table 2: Spinal involvement in 38 cases of brucellar
spondylitis
|
Vertebral
level
Lumbosacral |
Case
14 |
|
L1, L2 |
2 |
|
L2, L3 |
3 |
|
L3, L4 |
4 |
|
L4, L5 |
3 |
|
L5 |
3 |
|
L1, L2,
L3 |
1 |
|
Facet
Joint arthritis of L4 and L5 |
1 |
|
L3, L4,
T1 |
1 |
|
L1, L2,
L3, T9-T12 |
1 |
|
T12 |
2 |
|
C5-C6 |
2 |
|
C2 |
1 |
The sizes of the flaps ranged from 4 X 3 cm
to 12 X 10 cm. Three of the flaps underwent complete necrosis.
One flap had minimal rim necrosis. All the rest survived totally
and provided satisfactory cover. One patient complained of
excessive bulk and underwent flap thinning twice. There were no
cases of florid infection, hematoma or total graft loss at the
donor site.
Four of the cases were done under spinal
anesthesia and 1 under general anesthesia. Ten cases were done
under combined sciatic and femoral nerve block, which made the
prone positioning and the position changes needed during graft
harvest and inset of the flap, very simple.
Discussion :
During the study 823 cases of brucellosis
were admitted in our department and among them, 38 (4.6%) cases
had brucellar spondylitis. Twenty-five (65.8%) cases were males
and 13 (34.2%) were females. Mean age of the male patients was
42.7±16 years and for females was 51.5±18 years (P=0.716). The
total mean age of the patients was 45.9±17 years ranged 7 to 77
years. Twenty -eight cases (73.7%) were from rural areas. Eleven
(28.9%) cases had a history of consumed unpasturised cheese, 10
cases were husbandry and the remainders of 17 cases (44.7%) had
no predisposing factor for brucellosis. The mean days from the
onset of disease to diagnosis was 100±106 days ranged 7 days to
410 days. The disease was acute in 19 (50%) and sub- acute in 16
(42.1%) and chronic in 3 (7.9 %) of the cases. Risk factor for
brucellosis were reported from 43.3% of the patients. The most
common clinical symptoms and signs were back pain, sweating,
fever and arthralgia and were found in 38 (100%), 31 (81.6%), 28
(73.7%) and 13 (34.2%) of the cases, respectively. Lecucytosis
and normal sedimentation rate were seen in 9 (23.7%) and 30
(78.9%) cases, respectively. The distribution of the clinical
symptoms and signs of the patients between sexes are shown in
table 1.Single focal involvement of two vertebrae was seen in
twenty-eight cases (figure 1), single focal involvement of one
vertebra in 6 cases and single focal involvement of more than
two adjacent or non-continuous vertebrae in three cases (figure
2). One case had left facet Joint arthritis of L4 and L5.
Spondylitis was seen in 31 (81.6%) cases in lumbar and
lumbodorsal, in 2 (5.3%) cases in dorsal and in 3 (7.9%) cases
in cervical regions (table 2). Among twelve cases who were
treated with doxycycline plus Rifampicillin, relapse was seen in
2 (16.6%) cases. Relapse was seen in 1 (9.1%)case of the eleven
cases who were treated with cotrimoxasol plus Rifampicillin.
No relapse was seen among fifteen cases who
were treated with streptomycine, doxycycline and Rifampicillin.
Conclusion:
Brucellosis is still endemic in Iran and
consumption of unsafe dairy product as well as occupational
exposure are the mean route of transmission of brucellosis both
in rural and urban residents like the reports of other
researchers in endemic regions 6,7,19,20. Osteoarticular
involvement is the commonest complication of brucellosis with
varying degrees of frequency 5-8,19.Brucellar spondylitis is one
of the most frequent complications of Brucellosis and frequently
seen in geographic areas of the world which brucella melitensis
is endemic 9-11,16, 21,22. The frequency of spondylitis in
brucellosis ranges from 2% to 53% in different studies
5,8,19,23,24.Brucellar spondylitis occurs most commonly in
adults aged between 50 and 60 years. The mean age of our series
of patients was much lower than the results of other studies
13,25. Because of the demographic features of human brucellosis
in Iran most patients in this study were males, and had
occupational risk factors, like the results of other studies in
endemic areas 6-7,12,13. Whereas in the study from Portugal and
Israel, the disease predominated in females and was mainly
caused by ingestion of unpasturized dairy product 26-28. The
most common clinical symptoms and signs in these series of
patients were back pain, sweating, fever and arthralgia and were
similar to the report of other researchers in endemic areas
10-13,23-25. In the present study, spondylitis occurred most
frequently in the lumbar region, followed by cervical and
thoracic locations. The pattern of the involvement of the spine
in our study was similar to the results of other researchers
13,15,20,25,28. But unlike the results of these studies,
paravertebral soft tissue swelling, epidural and paravertebral
abscess were not seen in our series of patients. Lack of these
complications in our patients may be due to early identification
of the patients and administration of appropriate treatment.
Involvement of the spine may be either focal or diffuse, with a
predilection to the lumbar region 28. Spondylitis with
neurobrucellosis was seen in one of our cases. Lopes et al
reported two cases of spondylitis with neurobrucellosis among 17
patients with brucellar spondilitis in Portugal 26.Arevalo
Lorido et al reported a case of brucellar spondylitis with
meningoencephalitis 29.In our series of cases, both lumbar and
dorsal and lumbar and cervical spondylitis were seen in 2 cases
and 1 case respectively. Other researchers also reported more
than two anatomic sites of the spine for involvement
13,30.Management of brucellar spondylitis remains controversial
regarding the selection of antibiotics, the duration of
treatment and the role of surgery. In our study, with
streptomycin 1 gr/day for 14 days plus doxycycline and
Rifampicillin for four months we have not seen any relapse.
Relapse was seen in two cases treated by doxycycline plus
Rifampicillin and in one case treated with cotrimoxasol plus
Rifampicillin for four months. A study from Turkey showed that
with regimens such as doxycycline plus Rifampicillin, ofloxacine
plus Rifampicillin for 45 days the relapse rates were 15% and
26%, respectively. They also treated 22 patients with
streptomycin 1 g/day i.m for 15 days and doxycycline 100 mg
every 12 h orally for 45 days plus rifampicin 15 mg/kg per day
in a single morning dose orally for 45 days (SDR). They had no
relapse with this regimen 31. Some recommended the use of a
combination of doxycycline and ciprofloxacin for a period of 3
months. Successful use of this combination in five patients with
spondylitis also was reported 14. In Summary, brucellar
spondylitis may occur in any site of the spine mostly in lumbar
region. Streptomycin for 14 days and doxycycline plus
Rifampicillin for 4 months is a better regimen for treatment
of brucellar Spondylitis.
Acknowledgement: The authors express their
appreciation to Mohammad Jafar Soleimani Amiri.Ph.D, for
performing the laboratory
tests.
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