This case report describes a 26-year-old woman who developed chronic osteomyelitis of the tibia caused by Vibrio alginolyticus after her tibial fracture was contaminated with seawater. She underwent aggressive surgical debridement and bifocal osteosynthesis according to the Ilizarov technique. Bone cultures during surgery isolated V. alginolyticus. She was treated with ciprofloxacin and tetracycline and responded well without reactivation of the osteomyelitis. This represents the first reported case of chronic osteomyelitis caused by V. alginolyticus treated successfully with the Ilizarov technique.

1. 43(3):346-349,2002
CASE REPORT
Ilizarov Technique in the Treatment of Chronic Osteomyelitis Caused by Vibrio
alginolyticus
Vladimir Barbarossa, Nastja Kuèišec-Tepeš1, Eva Aldova2, Danijel Matek, Feodora Stipoljev3
Department of Orthopedic Surgery, Holy Ghost General Hospital; 1Department of Clinical Microbiology and
Hospital Infections, Holy Ghost General Hospital, Zagreb, Croatia; 2National Institute of Public Health, Praha,
Czech Republic; 3Cytogenetic Laboratory, Department of Obstetrics and Gynecology, Holy Ghost General
Hospital, Zagreb, Croatia
We present a case of 26-year-old woman with posttraumatic chronic osteomyelitis caused by Vibrio alginolyticus, con-
tracted after contamination of a tibial fracture with seawater. The patient underwent bone resection and bifocal
osteosynthesis according to Ilizarov and was treated with a combination of ciprofloxacin and tetracycline. The patient
responded in the same way to distraction osteogenesis as any other patient with chronic osteomyelitis with large de-
fects.
Key words: Ilizarov technique; osteomyelitis; Vibrio; Vibrio infections
Vibrio species is ubiquitous in brackish water unit, we found edema and purulent fistulous drainage
and saltwater habitats worldwide. Halophilic vibrios from the wound, but no fever. Weight bearing with
include V. parahaemolyticus, V. vulnificus, and V. tolerable pain was possible with the use of an an-
alginolyticus. Human infections are generally acqui- kle-foot orthosis.
red through consumption of contaminated seafood or X-ray findings and a computerized tomography
contamination of wounds by seawater or marine ani- scan revealed a bifocal defect and non-union of the
mals. Reported infections due to V. alginolyticus in- tibia.
clude conjunctivitis (1), otitis externa (2), peritonitis
in patients on ambulatory peritoneal dialysis (3), skin Blood tests showed leukocytosis of 16x109/L and
wound infections (4,5), and bacteremia and necro- increased erythrocyte sedimentation rate of 60/120
tizing fasciitis (6). V. alginolyticus is recognized as po- mm/h. C-reactive protein was 90 ng/L and values of
tential pathogen in soft-tissue infections, especially in liver enzymes were altered (aspartate aminotransfe-
cases when trauma was sustained directly in seawa- rase 48 U/L, alanine aminotransferase 26 U/L, gam-
ter. Severe infections have been described, with rap- ma-glutamil transferase 26 U/L). Serological tests on
idly evolving cutaneous necrosis and, in rare cases, hepatitis viruses showed anti-HAV and anti-HCV anti-
bone involvement requiring extensive surgical debri- bodies.
dement (7,8). Pre-existing liver disease, hemochro- Blood cultures and cultures of fistula fluid were
matosis or immunocompromised state is found in ap- taken on several occasions. Blood cultures were ster-
proximately 75% of such patients (6). ile. The fistula fluid cultures of methicillin-resistant
We report on a patient suffering from chronic Staphylococcus aureus were isolated, but no inflam-
osteomyelitis due to V. alginolyticus after wound con- matory cells were found in cytological samples.
tamination with seawater. Targeted therapy with intravenous vancomycin
was ineffective, and clinical and laboratory status
showed no change.
Case Report
The patient underwent aggressive surgical debri-
A 26-year-old woman was referred to our ortho- dement with resection of 17 cm bone defect and bifo-
pedic unit in February 1998, after sustaining a com- cal osteosynthesis according to Ilizarov. After the re-
minuted fracture of the tibia in a car accident on May section of necrotic bone, we applied the technique of
30, 1997. After four surgical treatments performed in bone transport (Figs. 1-4).
another facility, she bathed in the sea during summer Ilizarov circular frame was applied before the re-
1997, following her surgeon’s recommendation. She section of necrotic bone. The intercalated (medium)
subsequently developed a chronic osteomyelitis and segment of bone was created by osteotomy of the cor-
non-union of the tibia fracture. On admission to our tical layer (corticotomy). The transport of the interca-
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2. Barbarossa et al: Ilizarov Technique for V alginolyticus Osteomyelitis Croat Med J 2002;43:346-349
lated fragment of tibia was gradually advanced dis-
tally (0.5 mm/24 h) together with the soft tissue. The
process was then continued with “relay-race”-like
transport, further distracting the distal segment to
achieve bifocal lengthening (Figs. 1 and 2). The treat-
ment lasted for 2 years. The integrity of the bone was
restored and the length of the leg preserved.
Bone cultures taken during necrotic bone resec-
tion and grown on blood agar yielded a gram-nega-
tive bacillus in swarming colonies producing al-
pha-hemolysis. Samples of resected bone were ana-
lyzed with standard microbiological procedures (Ta-
ble 1). Vibrio spp was isolated and determined at the
Figure 3. The same patient. A. Condition before our treat-
ment. B. The half-open method of treatment with Ilizarov
apparatus in progress. In central part of the image the bear-
ing (gap) of radically resected inflamed bone can be seen.
C. Condition before the apparatus was removed. The con-
tact between the intercalary and the target segment was ac-
complished.
Figure 1. Preoperative scheme of Ilizarov treatment of a pa-
tient with osteomyelitis caused by Vibro alginolyticus. A.
Red area in distal tibia indicates the site of planned resec-
tion, whereas the red area in the proximal part indicates the
site of planned cortical osteotomy (corticotomy). B. Arrow
indicates the direction of the middle (intercalated) segment
transport. C. Intercalated and target segment contact. Arrow
indicates the direction of further transport of the distal seg-
ment (“relay-race”-like transport). D. Final condition as
planned. Lengths of proximal and distal parts of planned re-
generated bone are designated in centimeters.
Figure 4. The same patient, three months after the apparatus
was removed.
Department of Clinical Microbiology and Hospital In-
fections, Holy Ghost General Hospital, with border-
line biochemical characteristics for V. vulnificus, V.
alginolyticus or V. fluvialis. It was not possible to per-
form Vibrio spp subidentification in Zagreb. The Na-
tional Institute of Public Health in Prague confirmed
further species identification of bone culture for V.
alginolyticus (Table 1, ref 9).
The disk-diffusion test of isolated microorgan-
isms showed their sensitivity to tetracycline, third
generation cephalosporins, quinolones, and amino-
glycosides. The patient received ciprofloxacin 500
Figure 2. The same patient. A, B. Before the treatment. mg every 12 h and tetracycline 500 mg every 6 h dur-
Anteroposterior and laterolateral radiograms of left tibia ing three weeks. The outcome of the treatment was
and fibula. C. After the radical resection. Ilizarov apparatus
was applied. The site of cortical osteotomy is indicated by
uneventful, without reactivation of osteomyelitis.
arrows. D. Arrow indicates bone transport in progress. E.
Tibial radiogram after the Ilizarov apparatus was removed. Discussion
Anteroposterior view. Arrows indicate bone regenerates in
proximal and distal part. g – postoperative gap after radical Our patient is the first described case of chronic
resection; r – regenerated bone. osteomyelitis of the lower leg due to V. alginolyticus
347

3. Barbarossa et al: Ilizarov Technique for V alginolyticus Osteomyelitis Croat Med J 2002;43:346-349
dement of the underlying bone and the drainage of
Table 1. Metabolical characteristics of Vibrio spp. isolated the soft-tissue abscess 13 weeks after the original
from the patient‘s resected bone
Identification tests Vibrio sppa V. fluvialis V. vulnificus V. alginolyticusb
trauma. After doxycycline therapy, he had an un-
Oxidase + + + eventful recovery. Opal and Saxon (8) reported
Indole +/- + -/+ - intracranial infection due to V. alginolyticus in a
Citrate - +/- - +
ONPG c
- +/- -/+ - sailor with head injury. Osteomyelitis of the frontal
Hydrolysis of urea - - - - bone developed and the entire frontal plate of the in-
Hydrolysis of gelatin + +/- +/- + volved bone had to be excised. The patient was
Mobility + + +/- +
Arginine + + - - treated with chloramphenicol and was completely
Ornithine
Lysine
+
+/-
+
+/-
-
-
+
+
asymptomatic 6 months later. One patient with
H2O - - - - osteomyelitis due to V. vulnificus with total limb am-
Voges-Proskauer +
d e
-
d e
-
d e
putation was described in the study of Bisharat and
Acid production A g- A g- A g- Ad g-e
out of: coworkers (7). The patient had been injured while
glucose + + + + + + + + handling pond-cultivated fish. Osteomyelitis caused
arabinose - - - - + + - -
cellobiose - - + + - + by V. parahaemolyticus that lasted for 12 years was
lactose - - + - - - - - reported by Roland (15).
maltose + + + + + +
mannitol + + - + + + + + Most previously reported Vibrio spp. infections
salicin - - - - - - - - occurred during the summer months, when the num-
sucrose + + - + + + + +
Growth in NaCl: ber of bacteria was high. During the summer season
3% + at Eastern Adriatic coast in 1997, the water tempera-
6% +
8% + ture was above 21°C, which is the crucial tempera-
10% - ture for the growth of V. alginolyticus. Barbieri and
Lecithinase +
Lipase +
coworkers (16) also found V. alginolyticus as a pre-
Acetate - dominant species in two estuaries along the Italian
Novobiocin + (12 mm) Adriatic coast. Same authors found that the produc-
sensitivity
0 / 129 / vibriostatic + (18 mm) tion of toxins capable of causing cytoskeleton-de-
a
Determined at the Department of Clinical Microbiology and Hospital pendent changes was detected in a large number of
Infections, Holy Ghost General Hospital, Zagreb, Croatia.
b
Vibrio alginolyticus was identified at National Institute of Public Health, Vibrio strains. These findings indicate a significant
Prague, the Check Republic.
c
presence of potentially pathogenic Vibrio strains
O-nitrophenyl-ß-D-galactopyranosid. along the Adriatic coast.
d
Acid reaction.
e
No gas reaction. A history of contact with seawater or with a con-
taminated object should alert clinicians and microbi-
treated by Ilizarov technique. V. alginolyticus is sus- ologists to take into account a possibility of V. algino-
ceptible to a variety of antibiotics, including tetracy- lyticus infections. Recommending seawater bathing
cline, chloramphenicol, aminoglycosides, and the for recuperation in the presence of open wounds and
third generation of cephalosporins (10). Our patient liver damage is not advisable.
was successfully treated with combination of cipro- Surgical indication was made without knowl-
floxacin and tetracycline after radical and complete edge of V. alginolyticus tibial bone infection. We
removal of infected areas (Figs. 1-4). learned of the infection afterwards, after bone cul-
In cases of serious soft-tissue infections due to tures were grown. It is questionable whether we
Vibrio spp, there is often a rapid onset of cellulitis would have made a surgical indication if we had
with gangrenous changes of the skin and underlying known about chronic V. alginolyticus bone infection.
soft tissues. Surgical debridement of the involved ar- Howerver, our experience in treating the patient with
eas is the main therapy. In some cases, the amputa- V. alginolyticus osteomyelitis suggests that radical de-
tion of involved extremity is necessary. V. alginolyti- bridement followed by segmental bone transport us-
cus bacteremia was reported in association with se- ing Ilizarov technique is no less effective in dealing
vere burns (11), acute lymphocytic leukemia (12), with this unusual organism than with more common
and osteogenic sarcoma (13). Ho and coauthors (6) pathogens causing chronic osteomyelitis.
described a patient with cirrhosis secondary to hepa-
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