Possible identity of diffuse sclerosing osteomyelitis and chronic recurrent multifocal osteomyelitis

Possible identity of diffuse sclerosing osteomyelitis and chronic recurrent multifocal osteomyelitis One entity or two

Yoshikazu Suei, DDS, a Keiji Tanimoto, DDS, PhD, b Akira Taguchi, DDS, PhD, a Toshikazu Yamada, DDS, PhD, a Kouji Yoshiga, DDS, PhD, c Takenori Ishikawa, DDS, PhD, d and Takuro Wada, DDS, PhD, e Hiroshima, Japan HIROSHIMA UNIVERSITY, SCHOOL OF DENTISTRY

On the basis of the findings of nine of our patients and our review of previously reported cases of diffuse sclerosing osteomyelitis and chronic recurrent multifocal osteomyelitis, we discuss the similarity of these two entities. Our nine patients had initially been given diagnoses of diffuse sclerosing osteomyelitis on the basis of their clinicopathologic findings. However, technetium 99m-MDP bone scans performed on four of them revealed multiple bone lesions leading to the diagnosis of chronic recurrent multifocal osteomyelitis. Furthermore, no clear difference between clinical features in the patients with multiple bone lesions and those in the patients with diffuse sclerosing osteomyelitis was found. We conclude that diffuse sclerosing osteomyelitis is an expression of chronic recurrent multifocal osteomyelitis. (ORAt SuaG ORAl. MED ORAL PATHOI. ORAl. RADIOL ENDOD 1995;80:401-8)

Diffuse sclerosing osteomyelitis (DSO) 1-10 is a wellknown entity that mainly affects the mandible and usually has a protracted course with recurrent exacerbations and remissions in spite of well-established therapeutic procedures for infection and inflammation such as antibiotic treatments, hyperbaric oxygen, muscle relaxation, curettage, and decortication. Chronic recurrent multifocal osteomyelitis (CRMO) u'4~ is another osteomyelitic lesion in which the clinical features of the affected bone are similar to those in DSO. We previously reported a case of CRMO (DSO), reviewed the literature, and noted the similarities between DSO and CRMO. 41 In this article, on the basis of the findings in nine patients with chronic nonsuppurative mandibular osteomyelitis that was initially diagnosed as DSO and treated in our hospital, we further evaluate the relationship of DSO and CRMO. MATERIAL A N D METHODS The diagnostic criteria for DSO of the mandible are as follows: the patient's complaint is pain and swell-

aResearch Associate, Department of Oral and Maxillofacial Radiology. bAssociate Professor, Department of Oral and Maxillofacial Radiology. CAssociate Professor, Department of Oral and Maxillofacial Surgery I. dprofessor and Chairman, Department of Oral and Maxillofacial Surgery II. eProfessor and Chairman, Department of Oral and Maxillofacial Radiology. Received for publication Aug. 16, 1994; returned for revision Nov. 29, 1994; accepted for publication Jan. 31, 1995. Copyright 9 1995 by Mosby-Year Book, Inc. 1079-2104/95/$5.00 + 0 7/12/63878

Fig. 1. Radiograph in occipitofrontal projection in patient 7. Periosteal bone formation is apparent (arrows). ing of the mandible without fistula formation; the radiographic findings for the affected site are compatible with osteomyelitis; the histologic specimen demonstrates chronic inflammation of bone; recur401

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Table I. Clinical data of nine patients Site of bone lesions detected Patient number

Age~sex

Duration of symptom (A + B---))

by x-ray

by Tc Mandible Righl 1st and 2nd ribs 5th lumbar vertebra Left 1st rib Frontal bone Mandible 5th lumbar vertebra

1

40/F

Mandible Right 1st and 2nd ribs 5th lumbar vertebra

2

73/M

3

60/M

Mandible 5thlumbarvertebra Left femur Mandible

4

21/M

Mandible

5

50/F

Mandible

6

66/F

Mandible

6 + 71---r

7

23/M

Mandible

6 + 13

8

38/F

Mandible

7 + 12---~

9

23/F

Mandible

3 + 28

Mandible Sternum Mandible 2nd lumbar vertebra Mandible

8 + 41--->

12 + 49--~

1 ~ 11 0 + 62--~ 2 + 24--*

Treatment of mandible Antibiotics Decortication Partial resection

Antibiotics Saucerization Decortication Antibiotics Decortication x 2 Antibiotics, HBO Saucerization x 2 Antibiotics, HBO Decortication Partial resection Antibiotics Saucerization x 3 Decortication Partial resection Antibiotics Saucerization x 2 Antibiotics Saucerization Antibiotics Sancerization Decortication

Tc. Technetium 99m-MDP bone scintigraphic examination t-: Not Done). A + B. Symptom duration tmonths) before and after admission, respectively. ---~. Symptom remained present at the last follow-up. HBO. Hyperbaric oxygen treatment,

rent exacerbations of the symptoms are observed in spite of long-term antibiotic therapy; and no cause is clearly identified. A review of the records of patients treated between 1985 and 1993 yielded nine patients with DSO of the mandible. For these patients, we reviewed the clinical f'mdings; radiographic findings for and site of the lesion; laboratory, histologic, and microbiologic findrags (results of aerobic and anaerobic culture and gram stain); and the response to the treatment and duration of the symptoms. These findings were compared with those in previously reported DSO cases and CRMO cases. RESULTS The clinical data for the nine DSO patients (four men and five women: age range, 21 to 73 years) are summarized in Table I. Patient 1 has previously been described in the literature. 4T

Clinical findings All patients complained of a painful swelling at the affected site of the mandible without fistula or abscess formation. Recurrent exacerbations and re-

missions of the symptoms had occurred at least three times in all patients. Trismus, mild to severe, was observed in seven patients. The duration of the symptoms after the start of the treatment was more than 11 months. Symptoms other than those for the mandible were confirmed only in patient 1. who had recurrent swelling and pain of the right upper chest.

Radiographic findings and site of the lesion Radiographic examination was performed in all patients with the use of panoramic, periapical, occlusal, and occipitofrontal radiographs, supplemented with technetium 99m-MDP bone scintigraphic scan (Tc bone scan) in five patients (patients 1 to 5). Osteomyelitic lesions of the mandible were found in all patients. In patients 3 and 5. radiographic abnormalities were not observed on plain radiographs at the time of initial examination but became apparent later. Sclerotic and osteolytic changes with periosteal bone formation were observed in all patients. However. the degree of each type of radiographic change varied among the patients and with the course of the disease.

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Fig. 2. Panoramic radiograph of patient 4 about 5 years after first examination. Left mandible shows severe sclerotic change and reduction of bone volume.

Fig. 3. Panoramic radiograph of patient 2 at first examination. External resorption is seen (arrow). In all patients 40 years old or younger, except one (patient 8), periosteal reaction was more apparent in the early stage (Fig. 1). In the chronic stage, sclerotic change became more apparent in almost all patients (Fig. 2). External erosion of the mandible and reduction of the mandibular volume was observed in seven patients (Fig. 3). All lesions had initially affected the molar or angle region of the mandible with later spreading. The lesion extended beyond the midline of the mandible to the contralateral side in five patients. Involvement of the mandibular condyle was observed in six patients. Four of the five patients who underwent Tc bone scan showed abnormal accumulation of radiopharmaceuticals in not only the mandible but also other bones (Figs. 4 and 5). Bone lesions located at sites other than the mandible were observed in the frontal bone of the skull, ribs, sternum, and 2nd and 5th lumbar vertebrae. In patients 3 and 4, Tc bone scan was performed twice, at an interval of 6 months and 3 years 8 months, respectively. In patient 3, the lesion of the sternum was observed at both examina-

tions. In patient 4, however, the vertebral lesion was detected only at the second examination. In patients 141 and 2, osteomyelitic change was also observed on plain radiographs (Fig. 5). In patient 2, an osteomyelitic lesion of the condyle of the left femur was detected on the plain radiograph during the follow-up, period. The mandibular lesions in the four patients with multiple bone lesions showed no apparent difference from those in the 5 patients with single lesions.

Laboratory findings The erythrocyte sedimentation rate was examined in eight patients (patients 1 to 8) and was found to be elevated by more than 20 ram/hour in six patients (patients 1, 2, 3, 4, 6, and 7) and by more than 10 mm/hour in 2 patients. C reactive protein was elevated in seven patients. Slight elevation of the white blood cell count and alkaline phosphatase was observed in patients 2 and 3, respectively. No other abnormal findings that continued during the treatment were observed.

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Fig. 4. A and B, Technetium bone scintigraph of patient 3. Abnormal accumulation of radiopharmaceuticals is seen not only in mandible but also in sternum (arrow).

Fig. 5. A, Technetium bone scintigraph of patient 2. Abnormal accumulation of radiopharmaceuticals is seen in fifth lumbar vertebra (arrow). B, Lateral view of lumbar vertebrae in patient 2. Sclerotic change is seen (arrow).

Histologic findings Histologic specimens were obtained from all patients and demonstrated nonspecific chronic inflammation with infiltration of plasma cells, lym-

phocytes, and granulation tissues (Fig. 6). Microabscess formation was observed in only one specimen that was obtained from the resected mandible of patient 5.

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Fig. 6. Photomicrograph of biopsy specimen from patient 2. Remodeling of bone trabeculae and chronic inflammatory infiltration of marrow spaces are seen. (Hematoxylin-eosin stain; original magnification x20.)

Fig. 7. Panoramic radiograph of patient 9 about 6 years after resolution of symptoms. Deformation of the fight mandible is seen.

Microbiologic findings Bacterial culture of the bone lesion was performed in seven patients. Growth of bacteria, such as Bacil-

lus species, c~-Streptococcus, and Streptococcus epidermidis, was detected in five patients. In two patients, no bacterium was cultured from the bone lesion.

Response to treatment and duration of symptoms In all patients, antibiotic treatment and decortication were performed. Antibiotics were administrated orally and parenterally to all patients. Parenteral antibiotics were used at the times of exacerbation. Two patients (patients 4 and 5) received hyperbaric oxy-

gen treatment (3 atmospheres absolute pressure, breathing 100% oxygen, 30 minutes a day for 40 days for patient 4 and 1 hour a day for 30 days for patient 5). Saucerization or decortication was applied when the symptoms were persistent and severe and did not respond to antibiotic treatment with nonsteroidal anti-inflammatory analgesic drugs. The site and area of the decortication had included both the clinically symptomatic region and the radiographically detected bone change area. Segmental resection with continuity defect of the mandible was done in three patients (patients 1, 5, and 6). The extent of the resection site had been selected by evaluation of plain radiographs,

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Table II. Characteristics of DSO and CRMO DSO

CRMO I

5-76. 37 years old Age (range, average) 1:1.3 Sex (M/F ratio) The mandible is affected Sites in all cases Multiple bone lesions occasionally reported Histologic Chronic inflammation conditions

Treatment

Antibiotics Steroidal and/or nonsteroidal anti-inflammatory drugs Hyperbaric oxygen treatment Curettage or decortication Muscle relaxation* Complications Not reported

0-55, 11 years old 1:3 Multiple bone lesions Mandible is occasionally involved Acute to chronic inflammatory change of bone at various stages Antibiotics Steroidal and/or nonsteroidal anti-inflammatory drugs Curettage or decortication Partial resection

Palmoplantar pustulosis Psoriasis DSO and CRMO

Clinical features

Radiographi c findings

Laboratory data

Prognosis

Swelling and pain without sinus tract formation Course is usually prolonged with recurrent exacerbation Osteomyelitic lesion Osteolysis and sclerosis with various amounts of periosteal bone formation Erythrocyte sedimentation rate usually elevated White blood cell count and C reactive protein occasionally elevated Other consistent abnormal data have not been found Difficult to assess Symptoms often continue more than several years and are observed at the last follow-up. Lesion heals after treatment or spontaneously with or without bone deformity

*Including instructions regarding soft diet and avoidance of parafnnctional habits, rotation exercise, occlusal splint therapy, myofeedback, and muscle relaxant drugs.

including panoramic, occlusal, periapical, and occipitofrontal radiographs. The intent of surgery had been resection of almost the entire region that showed the radiographic change. And it was confirmed at the time of surgery that the site involving the necrotic or granulation tissue was fully resected. Antibiotic treatment and hyperbaric oxygenation did not alter the course of the disease. Saucerization, decortication, and segmental resection often resulted in temporary disappearance of the symptoms, but the

symptoms recurred after various intervals, from a few weeks to about 1 year. in all patients. At the time of last follow-up, symptoms persisted in six patients, and reduction of the mandibular volume and deformity were observed in three patients (patients 2, 4, and 9). The symptoms disappeared in three patients (patients 3 . 7 , and 9) for 8 months to 9 years. However. in patients 3 and 7. slight bone resorption of the inferior border in the molar region was observed on panoramic radiographs more than 6 months after the symptoms had disappeared, and in patient 9, deformation of the mandible still remained 6 years after the symptoms had disappeared (Fig. 7). Patient 6 was affected by Basedow's disease. No other disease that affects the bone metabolism or immune mechanism was noted in any patient during the follow-up period. DISCUSSION Table II summarizes the main features of DSO and CRMO as described in the literature. 1-4~ No difference exists between the two entities in terms of the clinical, radiographic, and laboratory findings. Antibiotic therapy usually seems to have no effect on the course of the disease. The pathogenesis is not fully known in either disease, although the two differ in patient age, sex ratio, number of lesions, histologic findings, and complications. In comparison with DSO, C R M O usually occurs in younger persons, more predominantly in females, and affects multiple bones rather than predominantly the mandible. Acute inflammatory change of bone is sometimes observed on histologic specimens, 18, 37 and complication with palmoplanter pustulosis (PPP) or psoriasis has been reported.25, 28, 29, 39 CRMO often affects the long bone metaphyses,18, 23 but only in children. Therefore, in older patients, the number of lesions is decreased because of the disappearance of the metaphyses. Probst et al. 4~ also pointed out that the lesions were widely distributed in juvenile patients, whereas they were restricted to fewer locations in adult patients mainly as a result of the reduction of the number of long bone lesions. 4~ We consider that this iS one reason that DSO is usually reported as a single lesion occurring in relatively older patients compared with CRMO. Furthermore, our review of the reported C R M O cases* disclosed that more than 30% of the symptoms noted at the time of first examination were confined to only one site. In some of these cases, an additional lesion was noted later. 19 Unifocal osteomyelitis has been described in a few reports as the same entity as CRMO, because of the similarity of the clinical features of the affected *Refs; 11-15, 17-20, 27, 29-35, 38

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sites.23, 25 Probst et al. 4~ suggested that a single lesion and a p r o l o n g e d course should suggest the p o s s i b i l i t y o f C R M O . S o m e patients with m a n d i b u l a r D S O , like s o m e o f our patients, were reported to h a v e a s y m p t o m a t i c bone lesions a t other sites such as the m a x ilia, z y g o m a , t e m p o r a l bone, and s t e r n o c l a v i c u l a r articulation.3, 5, 8, 10 B e c a u s e the Tc b o n e scan is nonspecific w h e n the a b n o r m a l a c c u m u l a t i o n is detected, possibilities o f post-traumatic c o n d i t i o n (fracture) and m e t a s t a t i c bone disease m a y be suspected. H o w ever, in our patients, there was no history o f trauma, and plain r a d i o g r a p h s in patients 1 and 2 s h o w e d inf l a m m a t o r y changes o f bone. F u r t h e r m o r e , a longterm f o l l o w - u p o f m o r e than 10 m o n t h s without any additional s y m p t o m s did not support the hot spot as metastatic disease. A l t h o u g h h i s t o l o g i c investigation h a d n o t b e e n done, it was suggested that the lesions not in the m a n d i b l e were the same basic d i s e a s e process as the m a n d i b u l a r lesion. W e b e l i e v e that m u l tiple b o n e lesions in p r e v i o u s l y r e p o r t e d D S O cases m i g h t have b e e n f o u n d if a Tc bone scan h a d b e e n done. H i s t o l o g i c e v a l u a t i o n o f C R M O lesions m a y r e v e a l m i c r o a b s c e s s formations, 37 but this feature has not b e e n r e p o r t e d in D S O . Dental clinicians often enc o u n t e r o s t e o m y e l i t i c lesions o f j a w bones, w h i c h m a y often be treated initially with antibiotics, incis i o n , and curettage before biopsy. In our patients, b i o p s y was p e r f o r m e d m o r e than 4 m o n t h s after the first treatment, It is likely that m a n y lesions are in the chronic stage at the time o f histologic e x a m i n a tion. J a c o b s s o n 8 also p o i n t e d out that the p o s s i b i l i t y o f D S O has s e l d o m been considered, s i m p l y b e c a u s e o f the rarity o f the disease. M o s t early cases were o v e r l o o k e d , a l l o w i n g the disease to g r a d u a l l y progress to the chronic stages, with r e p e a t e d e p i s o d e s o f pain, swelling, and trismus. H o w e v e r , in patients with C R M O , b i o p s y is usually one o f the first options c h o s e n to differentiate the lesion f r o m other lesions such as histiocytosis X, l e u k e m i a , and bacterial osteomyelitis. 23, 24 A t the s a m e time, w e m u s t note that abscess f o r m a t i o n has s e l d o m been obs e r v e d h i s t o l o g i c a l l y and that abscess or fistula form a r i o n has not b e e n noted clinically in any C R M O case. T h e strategy o f treatment differs in the two diseases. In a l m o s t all D S O cases, the authors 2, 3, 5, 8 rep o r t e d that they a t t e m p t e d to cure the d i s e a s e by various curative m e a n s such as a d m i n i s t r a t i o n o f antibiOtiCS, m u s c l e relaxation, h y p e r b a r i c o x y g e n a t i o n , and decortication. O n the other hand, in m o s t C R M O cases, the authors 11, 12 r e c o m m e n d e d e x p e c t a n t treatm e n t with the use o f steroidal or n o n s t e r o i d a l anti-inf l a m m a t o r y drugs and indicate that the g o a l o f the treatment should b e the alleviation o f the s y m p t o m s

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w h i l e a w a i t i n g the r e s o l u t i o n o f the disease process. In b o t h entities, steroid a d m i n i s t r a t i o n was v e r y effective in r e l i e v i n g the s y m p t o m s . 8, 27, 39 S p o n t a n e ous d i s a p p e a r a n c e o f the s y m p t o m s is r e p o r t e d in m a n y C R M O cases 17, 21, 31 and in s o m e D S O cases 1~ as well. S o m e patients with D S O m a y be effectively treated c o n s e r v a t i v e l y with the use o f analgesics and a n t i - i n f l a m m a t o r y drugs until the r e s o l u t i o n o f the symptoms. P P P or psoriasis has b e e n r e p o r t e d as a c o m p l i c a tion in C R M O . V a n H o w e 21 r e v i e w e d 50 C R M O cases and r e p o r t e d that P P P was o b s e r v e d in 10%. T h e s e skin lesions h a v e not b e e n d i s c u s s e d in reports d e s c r i b i n g D S O , although c o m p l i c a t i o n with P P P or psoriasis has b e e n r e p o r t e d in s o m e D S O cases. 7, 9 F u r t h e r m o r e , M a l m s t r r m et al. 9 p o i n t e d out that the f r e q u e n c y o f H L A antigen B13 was high in their patients with D S O and it was m o r e c o m m o n a m o n g p a tients with psoriasis. T h e findings in all nine o f our patients were consistent with those o f D S O lesion o f the m a n d i b l e , but, at the s a m e time, w e c o n s i d e r that the m u l t i p l e b o n e lesions in patients 1, 2, 3, and 4 should be d i a g n o s e d as C R M O . F u r t h e r m o r e , there were no a p p a r e n t differences a m o n g the patients, and w e therefore c o n c l u d e that D S O is an e x p r e s s i o n or t y p e o f C R M O . REFERENCES

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Reprint requests: Yoshikazu Suei Department of Oral and Maxillofacial Radiology Hiroshima University, School of Dentistry 1-2-3, Kasumi, Minami-ku, Hiroshima, 734 Japan