Gumber P et al. Garre’s Sclerosing Osteomyelitis.

(e) ISSN Online: 2321-9599

(p) ISSN Print: 2348-6805

CASE REPORT
GARRE’S SCLEROSING OSTEOMYELITIS – A
CASE REPORT
Parvind Gumber1, Asmita Sharma2, Kanchan Sharma3, Sonal Gupta1, Bindu Bhardwaj4, Kamal Kant Jakhar5
1
Senior lecturer, Dept of Oral Pathology & Microbiology, 2Senior lecturer, Dept of Pedodontics & Preventive
Dentistry, Mahatma Gandhi Dental College & Hospital, Jaipur, 3Senior lecturer, Dept of Oral Pathology &
Microbiology, Rajasthan Dental College, Jaipur, 4Professor, Department of Oral Surgery, Mahatma Ghandhi
Dental College and Hospital, Jaipur, 5PG student, Dept of Oral Pathology & Microbiology, Maharaja Ganga
Singh Dental College, Sri Ganganagar, Rajasthan, India
ABSTRACT:
Garre’s sclerosing osteomyelitis is a specific type of chronic osteomyelitis that primarily affects children and
adolescents. This disease is well described in dental literature and is commonly associated with an odontogenic
infection resulting from dental caries. This paper describes a case of Garre’s osteomyelitis in a 7-year-old-boy,
in whom the condition arose following periodontal infection due to pericoronitis in relation to permanent
mandibular left first molar. Clinically the patient presented with bony hard, non-tender swelling and the
occlusal radiograph revealed pathognomic feature of “onion skin” appearance. The elimination of infection was
achieved by gingival curettage and the complete bone remodeling was seen radiographically after three months
follow-up.
Key words: Garre’s osteomyelitis, periostitis ossificans, chronic osteomyelitis, gingival curettage.

Corresponding author: Dr. Parvind Gumber, Senior lecturer, Dept of Oral Pathology & Microbiology,
Mahatma Gandhi Dental College & Hospital, Jaipur, Rajasthan, India

This article may be cited as: Gumber P, Sharma A, Sharma K, Gupta S, Bhardwaj B, Jakhar KK. Garre’s
Sclerosing Osteomyelitis – A Case Report. J Adv Med Dent Scie Res 2016;4(2):78-83.

I
NTRODUCTION used to describe this reactive proliferation of the
Garre’s Osteomyelitis is named after Carl periosteum. These include proliferative periostitis of
Garre, who first observed the condition in Garre, Garre’s osteomyelitis, periostitisossificans,
1893. He described a massive focal non-suppurative ossifying periostitis, osteomyelitis
thickening of the periosteum of bones, with sicca, osteomyelitis with proliferative periostitis, and
peripheral reactive bone as a result of irritation or perimandibular ossification.5
attenuated infection. The condition was first In the maxillofacial region, the mandible is most
observed in the tibia, and was most frequently found frequently involved.5-7 Most reported cases are
on the anterior surface.1-4Berger (1948) reported a unifocal and unilateral. It has been noted that the
case of perimandibular ossification with all the highest incidence occurs in individuals less than 25
features of Garre’s osteomyelitis, although it was years of age. Eversoleet al. (1979), in a report of 29
Pell et al (1955) who first reported a case of Garre’s cases, found that the median age was 10.9 years
osteomyelitis, involving the mandible in a 12-year- (range 2.5-31 years).4,7 When it affects the jaw, this
old Negro boy, caused by a deep carious lesion in generally originates from an infection of low
the first molar.4 This disorder is characterized by virulence, such as dental decay, mild periodontitis,
chronic non-suppurative osteomyelitis with periodontal defect, pericoronitis, developing tooth
proliferative periostitis, which is caused by inert follicle, unerupted teeth, dental eruption or previous
stimulation from low grade infection that primarily dental extraction in the lesion area or a consequence
affects children and adolescents and frequently of infection of the underlying soft tissue that later
occurs in infants. Since then, many terms have been involved the deeper periosteum, untreated fracture,
78
Journal of Advanced Medical and Dental Sciences Research |Vol. 4|Issue 2| March - April 2016
Gumber P et al. Garre’s Sclerosing Osteomyelitis.

buccal bifurcation cyst, lateral inflammatory is fibrous, and contains lymphocytesand plasma cells
odontogenic cyst, non odontogenic infection and, either in focal areas of dispersed
rarely, no causative factor could be found.6-9 diffusely.11Microorganisms which are isolated in
Clinically, this reactive process accounts for the hard most cases are Staphylococci pyogenes, variety
swelling of the jaw and the subsequent facial aureus and albus, although various Streptococci and
asymmetry with which patients may present. The some mixed organisms can be associated.11
lesion is usually asymptomatic with no Typical evolution of this lesion can be attributed to
accompanying general and local signs of the fact that the high osteogenic potential in young
inflammation, although the clinical picture may vary patients allows an osteoblastic process which is
widely.6 Garre’s osteomyelitis presents a superior to the osteolytic one. This pattern is
characteristic radiographic feature, showing new identical to that of condensing osteitis, which is
periosteal proliferation located in successive layers frequently seen in the periapical areas of carious
to the condensed bone formation. This is a typical teeth, except that the proliferation of bone is of
radiographic feature of Garre’s osteomyelitis and is periosteal origin rather than endosteal.11
well known as “onion skin” appearance.6High The classification of PO of the mandible by Kawai
quality radiographs reveal radiopaque bone et al. is shown in Table 1. In this system of
laminations that are arranged roughly parallel to classification, Periostitis ossificans has been
each other and to the underlying cortical surface. classified into two types, each with two subtypes,
The periosteal reaction may be in single or multiple based on whether the original contour of mandible is
layers. These laminations vary from 1 to 12 in preserved or not. Type I lesions are of shorter
number, and radiolucent separations are often duration than type II. Both the subtypes of type I
present between the new bone and the original Periostitis ossificans occurs in the early stages of
cortex. The duplication is understood to be a result mandibular osteomyelitis. With adequate treatment
of periodic exacerbation and remission of the there can be complete resolution of PO type I cases;
infection, which causes repeated perforation of the however, if the disease continues, type I-1 may
outermost new bone with restriping of the progress to type II-2 and type I-2 to type II-1,
periosteum resulting in repetitive layering of bone.7,8 followed by type II-2. In type II cases where there
Periapical, occlusal and panoramic radiographs are has been loss of mandibular contour, deformity
often used for diagnosis of periostitis ossificans and remains even when normal bony architecture has
have a high diagnostic value. However, at the initial been restored and the disease process has been
osteomyelitis stage, there is no radiographic resolved. Mandibular osteomyelitis with bulbous
evidence and diffuse radiolucency begins to appear bony enlargement in young patients is referred as
with time. Computed tomography may also be used gross periostitis ossificans (GPO). Kawai et al.'s
and it is accurate for detecting not only typical classification of this more severe form of the disease
alterations inside the bone, but also periosteal is shown in Table 2. Type A is associated with
reactions and soft tissue involvement.7,8,10-12 carious tooth or followed extraction of tooth,
Histologically, there is considerable new reactive showing onion skin appearance. In 36·8% of types B
bone or osteoid tissue subperiosteally, with and C, no infectious source could be identified; it
osteoblasts surrounding many of the trabeculae, was suspected that it could be caused by a
which are frequently oriented perpendicular to the developing unerupted tooth or a dental follicle. Type
cortex. The connective tissue between the trabeculae D was seen in the chronic stage.8
Table 1: Classification of periostitis ossificans based on the radiographic appearance
TYPE 1 TYPE II
(original contours of mandible preserved) (original contour of mandible is lost)
Subtype 1 Single lamella seen as a radiopaque line of Newly formed bony enlargement with resorption of
periosteal new bone overlying the original original cortex and osteolytic areas usually visible.
cortex separated by a radiolucent line
Subtype 2 Visible hemi-elliptical newly formed bony Deformation with a homogeneously dense
enlargement, well outlined with a thin cortical osteosclerotic bone that made original cortex
surface located on the outer aspect of original discernible. This subtype occasionally showed
cortex producing an onion skin appearance. duplication of newly formed periosteal bone on the

79
Journal of Advanced Medical and Dental Sciences Research |Vol. 4|Issue 2| March - April 2016
Gumber P et al. Garre’s Sclerosing Osteomyelitis.

outer aspect of the deformed mandible.

Table 2: Classification of gross periostitis ossificans-GPO

TYPE A Showing an onion skin appearance, resulting from a non-vital tooth or following extraction
of a tooth
TYPE B Consolidation form shows fine bony spicules perpendicular to bone surface

TYPE C Consolidation form shows coarse trabeculation with wide marrow spaces

TYPE D Shows more osteosclerotic and osteolyic changes in the affected medullary bone and
disappearance of original cortex or loss of the original bone contour

Other diseases which present with similar features to earlier an asymptomatic swelling had appeared in
Garre’s osteomyelitis are infantile the left side of the lower face, which within a period
corticalhyperostosis (Caffey’s disease), of a few days enlarged and caused difficulty in
hypervitaminosis A, syphilis, leukaemia, Ewing’s opening of the mouth. The patient was taken to a
sarcoma and metastatic neuroblastoma.4,6,11 general practitioner who registered a sound erupting
This paper describes a case of 7-year-old child, in first permanent molar with no signs of periapical
whom the condition arose following a periodontal pathology. Oral amoxicillin was prescribed for 5
infection in first permanent molar. The lesion days and the swelling subsided until 3 months later
remained unresolved for a period of over 5 months when it reappeared. A second 5-day course of
as a result of misdiagnosis. The purpose of this antibiotics was given and the patient referred to a
report is to present a case of Garre’s osteomyelitis of pediatric dentist for the evaluation. Swelling
an unusual origin, because it resulted from a subsided after the second course of antibiotics and
localized periodontal infection in a newly erupted the patient visited the pediatric dentist after a month.
first permanent molar. Identification of the true An occlusal radiograph taken at that time showed a
cause and treatment through periodontal surgery buccal enlargement of the bone at the lateral side of
resulted in lesion resolution and resolved the the body of the mandible in the area of the second
diagnostic problem. primary and first permanent molars (Fig.1). A
tentative diagnosis of Garre’s osteomyelitis was
CASE REPORT made and a third course of antibiotic therapy was
A 7-year-old female was referred to the department prescribed for 5 days. The patient was well
of pediatric dentistry for evaluation and treatment of developed, well-nourished child in no acute distress
a hard mass involving the left side of the mandible. and with an uneventful past medical history.
According to the parents, approximately 5 months

Figure 1: shows preoperative radiographs of the patient

80
Journal of Advanced Medical and Dental Sciences Research |Vol. 4|Issue 2| March - April 2016
Gumber P et al. Garre’s Sclerosing Osteomyelitis.

Extraorally, there was a swelling overlying the left etiologic factor. Despite the absence of signs of
mandibular angle 2.0x1.0 cm in size not acute inflammation in the periodontal tissues,
causingobvious facial asymmetry. The swelling was examination with a periodontal probe showed an 8
hard, firm, non-fluctuant and not tender on mm periodontal pocket distal to the mandibular first
palpation. The overlying skin was normal. No permanent molar. The patient was subjected to the
cervical lymphadenopathy was present. basic hematological investigations.
The intraoral examination revealed a bony hard A full thickness buccal flap was elevated and
swelling extending from the left first primary molar gingival curettage performed with respect to 36. At
to the angle of the left mandible. The swelling was surgery the buccal bone plate appeared intact. Pieces
smooth and had eliminated the mucobuccal fold in of bony tissue were shaved using a chisel.A bony
the area of the first permanent molar. The overlying biopsy was performed and the sample was submitted
mucosa was in normal color and texture. The patient for histopathological evaluation. All the granulation
was in the mixed dentition with no signs and tissue was removed and the flap was repositioned
symptoms of carious lesions and periapical and sutured followed by application of periodontal
pathology associated with any tooth. pack. One week following curettage the patient was
The radiographic pictures were in accordance with called for follow-up. Histolopathology of the
the clinical impression in providing no evidence of biopsied tissue revealed reactive bone formation
periapical pathology. In occlusal radiograph of together with findings of chronic inflammation (Fig
mandible, a dull grey convex shadow was separated 2). Trabeculae of bone and osteoid tissue are visible
from the inferior border of the mandible by a thin, in a pool of fibrous connective tissue stroma with the
linear radiolucency (Fig.1). A prominent buccal osteoblasts bordering many trabeculae. Fibrous
cortex expansion extending from the involved area stroma displays irregular bundles of collagen fibers
showed absence of clear cut opacity suggestive of with abundant fibroblasts, fibrocytes and diffuse
osteolytic changes within the area of expansion. sprinkling of chronic inflammatory infiltrate
Radiographically, periosteal bone deposition predominantly lymphocytes and plasma cells.
alternating with areas of osteolysis was seen, hence Numerous endothelial cell lined blood capillaries are
suggestive of PeriostitisOssificans. The clinical and discernible along with extravasated Rbcs. Deeper
radiographic findings were considered to be section also shows L.S and T.S of muscle fibers.
indicative of Garre’s osteomyelitis. Odontogenic These findings were consistent with a diagnosis of
infection consequent to caries was excluded as an Garre’s osteomyelitis.

Figure 2: shows photomicrographs of histopathological tissue.

81
Journal of Advanced Medical and Dental Sciences Research |Vol. 4|Issue 2| March - April 2016
Gumber P et al. Garre’s Sclerosing Osteomyelitis.

Figure 3: The post-operative radiographs of the patient

DISCUSSION of the pathologic material produced resolution of the

Garre’s osteomyelitis is a well-documented lesion, although antibiotic therapy alone had proved
pathologic entity in the dental literature. Because the inadequate to eliminate the infection. Extraction of
majority of the reported cases are sequel to an the tooth was not considered as a treatment of choice
odontogenic infection due to caries, the disease is once the causative factor had been identified.
most often associated with a deep carious lesion and Garre’s osteomyelitis most often exhibits typical
peirapical pathology.13 clinical and radiographic characteristics that help the
The periodontium has been reported by a number of clinician to reach a differential diagnosis. However,
authors as a potential source of infection for Garre’s this case suggests that where no obvious cases can
osteomyelitis where the inflammatory process may be found, one should be aware that the disease may
initiate and progress to a bony lesion. Loveman12 originate from the periodontal tissues, although this
reported a number of cases of mandibular is less common. Particularly in patients that present
subperiosteal swelling in children where no with a bony lesion indicative of Garre’s
periapical pathology was found. The only feature in osteomyelitis in the area of newly erupted first
common with the case reported here was the permanent molar, and where a necrotic pulp has
presence of an erupting permanent molar. Gorman12 been excluded as etiologic factor, the possibility of
presented a case of periostitis ossificans in the periodontal involvement should be explored. In such
mandible where the tooth responsible was an a case, a more conservative periodontal surgical
erupting second molar covered partially by a approach has definite advantages over tooth
gingival flap. The site of entry of infection was extraction and should be sufficient to treat the
thought to be pericoronal. There have been other disease.12
cases in the dental literature where the pathological A review of the literature has shown that this
process could not be explained by the factors more supposedly rare form of bone infection is becoming
commonly involved with Garre’s osteomyelitis. In more and more common.11Two reasons could
only one of these reports was there any mention of account for this growing incidence:
the periodontium as a potential site of infection.12 1. As a result of the increase of health and living
In our case, the absence of evidence of periapical standards, people are responding in an "anabolic
infection and the presence of a newly erupted first rather than catabolic manner."
permanent molar accounts for the presence of 2. The increased use of antibiotics has affected the
pericoronitis and favor the development of bacterial virulence of microorganisms, turning an osteolytic
infection.The history of the disease along with the process into aosteoblastic one. However, the abuse
diagnostic features justified a surgical exploration of of antibiotics could be harmful to the patient. In
the lesion. This verified the suspected etiologic many cases, the evolution into le Garre's
involvement of the periodontium. Surgical removal osteomyelitis could be prevented if the dentist had
82
Journal of Advanced Medical and Dental Sciences Research |Vol. 4|Issue 2| March - April 2016
Gumber P et al. Garre’s Sclerosing Osteomyelitis.

thought about eliminating the causal factor rather 3. Neelima M. Osteomyelitis and osteoradionecrosis of
than just instituting some antibiotic therapy. the jaw bones. Textbook Oral MaxillofacSurg 608-11.
We all agree that, in the presence of an infected 4. BA Ferreira & AL Barbosa. Garre’s osteomyelitis: a
tooth, the microorganisms are responsible for the case report. International Endodontic Journal. 1992;
25: 165-68.
irritation causing the host's proliferative response.
5. Nakano H, Miki T, Aota K, Sumi T, Matsumoto K,
However, Thoma recalls that cultures from the bone Yura Y. Garré’s osteomyelitis of the mandible caused
of the tibia or femur are always sterile. Smith and by an infected wisdom tooth. Oral Science
Farman offer a diagram showing a sinus formation International. 2008; 5: 150-4.
with pus from the periapical area tracking toward the 6. Suma R, Vinay C, Shashikanth MC, Subba Reddy
surface of the bone. Rowe and Heslop11took a VV. Garre’ssclerosing osteomyelitis. J Indian
culture of such a sinus which proved to be sterile as SocPedod Prevent Dent. Supplement 2007; S30-33.
well. It seems that the culprit of the tissue irritation 7. Luciana O, Thais O, Daniela M, Alynne M, Ieda R,
is not the bacteria per se, but the product of their Paulo C. Computed tomography findings of
presence and degradation. The different toxins and periostitisossificans. Braz J Oral Sci. 2010; 9(1): 59-
62.
endotoxins, spread to surrounding tissues can
8. Kannan SK, Sandhya G, Selvarani R.
account for themaintenance of the chronic Periostitisossificans (Garrè’s osteomyelitis):
inflammation sites seen in microscopic radiographic study of two cases. Int J Paediatr Dent.
examinations. The young host cannot entirely 2006; 16: 59-64.
dispose of those byproducts, but can stimulate new 9. Rafel S, Andre FL, Paulo F, Andre C, Debora FC,
bone formation in an attempt to encapsulate the Eliete G and Nilce M. Proliferative periostitis with
lesion.11 A fine balance is maintained between the Dens In Dente: case report. Brasilia Med. 2009;
resistance of the host and the number and virulence 46(3):295-99.
of present organisms, such that infection can 10. Ebihara A, Yoshioka T, Suda H. Garrè’s osteomyelitis
continue at a low, chronic stage, invasive enough to managed by root canal treatment of a mandibular
second molar: incorporation of computed tomography
stimulate new periosteal bone formation but not
with 3D reconstruction in the diagnosis and
severe enough to induce bone resorption.14,15 monitoring of the disease. IntEndod J. 2005; 38: 255-
61.
CONCLUSION 11. Stephane S, Huan Pham. Garre’s osteomyelitis: a case
In conclusion, an abnormality of development of report. Pediatric Dentistry. 1981; 3(3): 283-86.
teeth can allow the entry of bacteria and result in 12. Oulis C, Berdousis E, Vadiakas G, Goumenos G.
periapical lesions. In this case, this persistent low- Garre’s osteomyelitis of an unusual origin in a 8-year-
grade infection caused a response of the periosteal old child: A case report. Int J Paediatr Dent. 2000; 10:
tissue known as proliferative periostitis. The 240-4.
proliferative periostitis is unusual because its 13. Ozdemir A, Guven G, Dilsia A, Sencimen M.
Diagnosis and treatment of mandibular extraoral sinus
development depends on the occurrence of a set of
of periodontal origin in a 9-year-old boy: A case
critically integrated conditions: that is a chronic report. J Indian SocPedod Prevent Dent. Supplement
infection in a young individual with a periosteum 2008; S76-78.
capable of vigorous osteoblastic activity and a 14. Malini NS. Osteomyelitis with Proliferative
equilibrium between the virulence of the infectious Periostitis: Conservative management of a case.
agents and the host resistance. Journal of Indian Dental Association. 2010; 4(9): 327-
REFERENCES 29.
1. Neville BW, Damm DD, Allen CM, Bouquot JE. Pulp 15. Eswar N. Garre’s osteomyelitis: a case report. J Indian
and periapical diseases. In: Neville BW, editor. Oral Soc Pedod Prevent Dent. 2001; 19: 157-9.
and maxillofacial pathology. 2nd edn. Rio de Janeiro:
W.B. Saunders; 2004. p.128-9.
2. Shafer WG, Hine MK and Levy BM. (1983) A
Textbook of Oral Pathology. 4thedn. Saunders.
Philadelphia.

Source of support: Nil Conflict of interest: None declared

This work is licensed under CC BY: Creative Commons Attribution 3.0 License.
83
Journal of Advanced Medical and Dental Sciences Research |Vol. 4|Issue 2| March - April 2016