Dentomaxillofacial Radiology (2001) 30, 293 ± 295

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Scintigraphic evaluation of chronic osteomyelitis of the mandible in

SAPHO syndrome
T Sato*,1, H Indo1, Y Kawabata1, R Agarie2, T Ishigami2 and T Noikura1
1
Department of Dental Radiology, Kagoshima University Dental School, Kagoshima, Japan; 2Department of Oral & Maxillofacial
Surgery, Kagoshima University Dental School, Kagoshima, Japan

A patient with SAPHO syndrome (synovitis, acne, pustulosis, hyperostosis and osteitis) was
evaluated by combined scintigraphy. 99mTc HMDP scintigraphy showed accumulation in the
sternum and lumbar vertebrae as well as the right mandible, whereas 67Ga citrate showed an
accumulation in the right mandible, but not in the sternum or lumbar vertebrae. These results
are consistent with chronic osteomyelitis in the mandible.

Keywords: osteomyelitis; radionuclide imaging; skin diseases; syndrome

Case report

A 65-year-old Japanese male attended with a di€use extent of accumulation agreed approximately with the
swelling of the right ramus and body of the mandible osteomyelitis on the panoramic radiograph (Figure
and severe limitation of opening. His past history 2a). Scintigraphy with 67Ga citrate was performed to
revealed slight pain and crepitation in the TMJ and a evaluate any in¯ammatory activity. The lesion in the
slight limitation of opening for 6 months. He had been right mandible showed relatively less intense accumu-
prescribed painkillers but his symptoms did not reduce. lation, consistent with moderately enhanced inflamma-
His acute symptoms had developed immediately prior tion (Figure 2b). Taken together, these radiographic
to his presentation to us. The patient also had and scintigraphic ®ndings, clearly indicated new bone
pustulosis on his hands and psoriasis on his feet. A
diagnosis of SAPHO syndrome (synovitis, acne,
pustulosis, hyperostosis, osteitis) was made. Conven-
tional radiography and combined scintigraphy were
performed to evaluate the bone lesions further.
A panoramic radiograph of the right side of the
mandible showed extensive bone destruction with a
moth-eaten appearance. Most of the cortical bone at
the inferior border in the right premolar and molar
regions and the mandibular canal had resorbed with
loss of trabecular structure in the body of the right
premolar and molar regions, ramus and coronoid
process. The right mandibular second premolar and
the molar teeth were missing, but the cause was
unknown (Figure 1). Scintigraphy with 99mTc HMDP
showed an intense accumulation in the body of the
right mandible, ramus and coronoid process, consis-
tent with greatly increased osteoblastic activity. The

Figure 1 Panoramic radiograph of the right side of the mandible

*Correspondence to: T Sato, Department of Dental Radiology, Kagoshima demonstrating erosion of the cortex (arrows), and loss of trabecular
University Dental School, 8-35-1 Sakuragaoka, Kagoshima 890-8544, Japan pattern, producing a moth-eaten appearance in a body, ascending
Received 12 March 2001; accepted 11 June 2001 ramus and coronoid process of the mandible
 SAPHO syndrome
T Sato et al
294

a b

Figure 2 (a) Scintigraphy with 99mTc HMDP demonstrating marked accumulation in the right mandibular body, ramus and coronoid process
(arrow) consistent with proli®c osteoblastic activity. (b) Scintigraphy with 67Ga citrate showed moderate accumulation in the right mandible
indicating less intense in¯ammation (arrow)

a b c

Figure 3 (a) Scintigraphy with 99Tc HMDP demonstrating uptake in the left clavicular notch (arrow) and sternal angle (arrow head). (b) The
accumulation of 99mTc HMDP between the 1st and 2nd lumbar vertebrae (arrow). (c) Plain radiograph of the lumbar vertebrae demonstrated
hyperostosis between the 1st and 2nd lumbar vertebrae (arrow)

formation and in¯ammation. The lesion of the right
mandible was therefore diagnosed as chronic osteo-
myelitis.
In addition, there was uptake of 99mTc HMDP in the
sternum and lumbar vertebrae (Figure 3a,b). The
accumulation in the sternum was localized to the left
clavicular notch and sternal angle, while in the lumbar
vertebrae it was between the ®rst and second lumbar
vertebrae. These scintigraphic ®ndings indicated osteo-
blastic activity. Plain radiography demonstrated hyper-
ostosis between the ®rst and second lumbar vertebrae
(Figure 3c), but not in the sternum. 67Ga citrate failed
Dentomaxillofacial Radiology
to show any accumulations in either the sternum or
lumbar vertebrae, consistent with little or no inflam-
matory activity. The sternum and lumbar vertebrae
were asymptomatic and there was no history of trauma
or backache. Following chemotherapy, the oral
symptoms resolved.
Discussion
Osteomyelitis in the mandible is usually caused by
odontogenic infection. However, some cases do not

show any speci®c causative organisms and are some-
times accompanied by extramandibular bone lesions or
skin lesions of the hands or feet, as in this case. We
believe that the patient in our case should be diagnosed
as SAPHO syndrome a€ecting the mandible. SAPHO
syndrome is a rare disease which was identi®ed as a
new clinical entity in 1987 by Chamot and co-workers.1
It is characterized by speci®c clinical and radiographic
manifestations of synovitis, acne, pustulosis, hyperos-
tosis and osteitis. Chronic recurrent multifocal
osteomyelitis (CRMO), and di€use sclerosing osteo-
myelitis of the mandible (DSO)2 ± 5 show similar
radiographic ®ndings. For the purpose of evaluation
of these in¯ammatory bone lesions, especially the
remote multifocal bone lesions, combined scintigraphy
with 99mTc HMDP and 67Ga citrate is very helpful6 ± 7
and frequently yields more information than either
agent alone because their mechanisms of concentration
are basically di€erent.8
Our patient exhibited most of the manifestations of
SAPHO syndrome, persistent osteomyelitis of the
mandible, extramandibular bone lesions without clear
in¯ammatory activity and skin lesions of the hands and
feet. With regard to the di€erential diagnosis between
the SAPHO syndrome, DSO and CRMO, the bone
lesion in DSO is con®ned in the mandible. Although it
might seem to distinguish DSO from CRMO and the
SAPHO syndrome,9 Kahn and co-workers2 considered
References
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SAPHO syndrome
T Sato et al
295
that it might be the mandibular form of a more di€use
SAPHO syndrome, because the bone lesions in both
entities are very similar and both are of unknown
etiology. On the other hand, CRMO is characterized
by multifocal in¯ammatory bone lesions. 67Ga citrate
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SAPHO syndrome.
In summary, combined scintigraphy with
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Tc HMDP and 67Ga citrate is a useful method to
evaluate osteoblastic and in¯ammatory activity in
bone. However, it is dicult to distinguish between
SAPHO syndrome, DSO and CRMO on the basis of
scintigraphy alone. The possibility that all three
diseases are a spectrum of the same systemic condition
should be considered.
Acknowledgements
The authors wish to thank Professor Kazumasa Sugihara,
First Department of Oral & Maxillofacial Surgery,
Kagoshima University Dental School for his advice and
Dr Miguel Federico Vazquez Archdale, Faculty of Fish-
eries, Kagoshima University for reading the manuscript.
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Dentomaxillofacial Radiology