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CASE REPORT
Year : 2015  |  Volume : 33  |  Issue : 4  |  Page : 344-346
 

Periostitis ossificans (Garrè's osteomyelitis): An unusual case


1 Department of Pedodontics and Preventive Dentistry, Jodhpur Dental College General Hospital, Jodhpur, Rajasthan, India
2 Department of Pedodontics and Preventive Dentistry, The Oxford Dental College, Hospital and Research Centre, Bommanahalli, Bangaluru, Karnataka, India
3 Department of Pedodontics and Preventive Dentistry, Pacific Dental College, Debari, Udaipur, Rajasthan, India

Date of Web Publication18-Sep-2015

Correspondence Address:
Dr. Deepesh Singh
Department of Pedodontics and Preventive Dentistry, Jodhpur Dental College, General Hospital, Jodhpur, Rajasthan - 342 001
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0970-4388.165718

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   Abstract 

Garrè's sclerosing osteomyelitis is a specific type of chronic osteomyelitis that mainly affects children and young adults and is commonly associated with odontogenic infection. The paper describes an unusual case of Garrè's osteomyelitis in an 11-year-old boy, in whom the condition arose following poor oral hygiene and periodontal problems in relation to permanent mandibular left first molar. Clinically the patient presented with bony hard, nontender swelling and the occlusal radiograph revealed pathognomic feature of "onion skin" appearance.


Keywords: Garrè′s sclerosing osteomyelitis, mandible, periodontitis, periostitis ossificans


How to cite this article:
Singh D, Subramaniam P, Bhayya PD. Periostitis ossificans (Garrè's osteomyelitis): An unusual case. J Indian Soc Pedod Prev Dent 2015;33:344-6

How to cite this URL:
Singh D, Subramaniam P, Bhayya PD. Periostitis ossificans (Garrè's osteomyelitis): An unusual case. J Indian Soc Pedod Prev Dent [serial online] 2015 [cited 2019 Sep 21];33:344-6. Available from: http://www.jisppd.com/text.asp?2015/33/4/344/165718



   Introduction Top


The word "osteomyelitis" originates from the ancient Greek words osteon (bone) and muelinos (marrow) and means infection of medullary portion of the bone. [1] Periostitisossificans (PO) is a type of chronic osteomyelitis, more popularly known as Garrè's osteomyelitis. Other names attributed to PO in the literature are nonsuppurative ossifying periostitis, osteomyelitis with proliferative periostitis, nonsuppurative sclerosing osteomyelitis, and chronic sclerosing inflammation of the jaw. [2]

Carl Alois Philipp Garrè was a Swiss surgeon and bacteriologist. He published an article in 1893 on the manifestations of osteomyelitis, hence the name Garrè's sclerosing osteomyelitis (sclerosing osteitis); a form of chronic osteomyelitis with proliferative periostitis. [3],[4] He was a student of Robert Koch and Theodor Kocher. In 1893, [5] Carl Garrè described a sclerosing form of osteomyelitis that caused distension and thickening of bone, but no suppuration, sequestration, or fistula formation. However, Garrè was not responsible for the description of the disease that now bears his name. In his original publication there is no mention of periostitis, periosteal duplication, or onion-skin appearance. Garrè himself had no access to pathologic specimens for microscopic examination. The term 'Garrè's osteomyelitis' as synonymous to PO may therefore be an improper designation and the term PO may be preferred. [2] Hardmeier, Uehlingen, and Muggli (1974) introduced the term primary chronic sclerosing osteomyelitis to differentiate the clinical presentation of chronic osteomyelitis from the clinical description given by Garrè; they also described the radiological appearances. [6] It was first described in mandible by Pell et al., in 1955. A review of literature in 1980 by Lichty and his associates revealed 22 reported cases involving the jaw bones. The condition is seen exclusively in children or young adults. Mandible is often affected than maxilla. [5] Primary chronic sclerosing osteomyelitis is a rare condition with insidious onset and the radiological appearances of expansion and sclerosis of bone, combined with the absence of signs of acute sepsis, often gives rise to a preliminary diagnosis of Ewing's sarcoma, osteosarcoma, or osteoid osteoma. [6]

PO is a nonsuppurative type of osteomyelitis. The most common cause for PO is periapical infection of mandibular first molar. Radiographically PO is characterized by the presence of lamellae of newly formed periosteal bone outside the cortex, giving the characteristic appearance of 'onion skin'. [2] When it affects the jaw, this generally originates from an infection of low virulence, such as dental decay, mild periodontitis, dental eruption, or previous dental extraction in the lesion area. Clinically, this reactive process accounts for the hard swelling of the jaw and the subsequent facial asymmetry with which patients may present. [7]


   Case Report Top


An 11-year-old boy presented with a 3-month history of painless swelling on the left side of the mandible. Even after treatment using antibiotics and anti-inflammatory agents, there had been no significant reduction in the size of the swelling. The swelling kept subsiding to a little extent and then kept recurring. On examination there was a localized bony hard swelling of the left side of the body of mandible with thickening of the lower border. Skin and mucosa over the swelling was normal. Intraoral examination showed no carious tooth [Figure 1] and maxillary arch was also normal. Oral hygiene was very poor with grade 2 calculus and pocket depth extending upto 6mm depth with buccal cortical expansion adjacent to the teeth 36 and 37.
Figure 1: Intraoral examination revealed localized bony hard swelling of the left side of the body of mandible with no carious involvement


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Routine hematological investigations revealed normal results. A mandibular lateral occlusal view of left side was taken, which showed a fusiform bony expansion and diffuse radiopacity on the buccal aspect of the mandible extending from 36 to 37 regions. Two distinct radiopaque lamellae were visible outside the buccal cortex, with an osteolytic lesion evident on the buccal aspect in the 36 region. It also revealed a single radiopaque lamella below the lower border of the mandible and separated from the cortex by a fine radiolucent line in the 36 region. The case was diagnosed as PO [Figure 2]. Complete subgingival scaling with thorough curettage and root planning was performed at 36 region after flap reflection [Figure 3] and the patient was prescribed doxycycline and ibuprofen for 5 days along with chlorhexidine mouthwash. Review at 1 month showed marked reduction in the size of the swelling.
Figure 2: Occlusal radiograph showing two distinct radiopaque lamellae outside the buccal cortex, with an osteolytic lesion evident on the buccal aspect in the 36 region


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Figure 3: Complete subgingival scaling with thorough curettage and root planning was performed at 36 region after flap reflection


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   Discussion Top


The term PO was coined by Gorman in 1951. The mean age of patients with PO has been reported to be 13 years. [2],[8] There is no sex predilection for PO, although slight predilection in males has been reported, with a male/female ratio of 1.4:1. [8] In the present case, the Patient was an 11- year-old male diagnosed as having PO.

The etiological factors for PO include periapical infection of mandibular molars, periodontal infection, untreated fractures, developing tooth follicle, unerupted teeth, previous extraction site, pericoronitis, buccal bifurcation cyst, lateral inflammatory odontogenic cyst, and nonodontogenic infection. [8],[9],[10] In some cases there may not be any demonstrable etiological factor and these are termed idiopathic. [2],[10] It is a nonsuppurative inflammatory process, where there is peripheral subperiosteal bone deposition caused by mild irritation and infection. [7] The source of infection for PO in the present case was periodontal infection with deep periodontal pocket and subgingival calculus as a chronic inflammatory process.

For unknown reasons PO is most common in the mandible, and has been especially related to the left side of the mandible. [11] A previous study showed 82.7% of periostitis occurs along the lower border of mandible, 43% in buccal cortex, and 6.5% in lingual cortex. [2],[8] There was involvement of lower border of body of the mandible and the buccal cortex without involvement of the lingual aspect in the present case.

Terms like Garrè's osteomyelitis and osteomyelitis with periostitis are used to identify lesions with a large amount of periosteal reaction. But periosteal reaction can be seen in any type of osteomyelitis lesion, the amount of periosteal reaction depends on the activity of the osteoblastic cells in the periosteum. The degree and duration of the symptoms depend on various factors such as the virulence of the causative organisms, the presence of underlying diseases, and the immune status of the host. [7],[12] It is merely a periosteal inflammatory reaction to many nonspecific stimuli, leading to the formation of an immature type of new bone outside the normal cortical layer. [1]

Occlusal radiographs taken for the same case at the same visit showed fusiform bony mass with two radiopaque lamellae outside the original buccal cortex, giving a characteristic onion-skin appearance. The affected area showed increased radiodensity, which may be attributed to osteosclerosis. Bony enlargement on the outer aspect of the original cortex of lower border without alteration of the contour was observed. Also seen was the duplication of cortical lining and homogenously dense osteosclerotic bone. Radiological differential diagnosis of PO includes infantile cortical hyperostosis, Ewing's sarcoma, and osteosarcoma; none of which fitted the clinical and radiographic features of the two cases reported here. Garrè's osteomyelitis should be distinguished from infantile cortical hyperostosis which occurs in young infants and involves a number of bones. The mandible is involved invariably along with clavicle and ulna. Once identified, the treatment procedure includes the removal of infected tooth curettage of the socket. Surgical recountouring of the cortical expansion is usually not required because the jaws are gradually remodeled during the growth period. [5]

PO in young people is, in general, curable with early diagnosis and adequate treatment; however, if the correct diagnosis is delayed by more than 6 months, it may progress into a persistent and deforming stage. Once the cause is removed, the bone will remodel itself gradually and the original facial symmetry will be restored. This remodeling of the bone may be helped by the overlying muscle pull, which is attached to it. Successful resolution may be more difficult in severe and long standing cases of PO. [2]

Dentists should be aware that the periodontium may be a potential source of infection for Garrè's osteomyelitis in children, particularly in the presence of ectopically erupting posterior teeth. In such cases, periodontal treatment should be sufficient to treat the disease and extraction of the tooth involved may not be necessary. [13]

 
   References Top

1.
Baltensperger M, Eyrich GK. Osteomyelitis of the jaws: definition and classification. In: Baltensperger M, Eyrich GK, editors. Osteomyelitis of the jaws. 1 st ed. Springer: Berlin Heidelberg 2009; p. 6, 13.  Back to cited text no. 1
    
2.
Kannan SK, Sandhya G, Selvarani R. Periostitis ossificans (Garrè′s osteomyelitis) radiographic study of two cases. Int J Paediatr Dent 2006;16:59-64.  Back to cited text no. 2
    
3.
Wood RE, Nortjé CJ, Grotepass F, Schmidt S, Harris AM. Periostitis ossificans versus Garrè′s osteomyelitis. Part I. What did Garrè really say? Oral Surg Oral Med Oral Pathol 1998;65:773-7.  Back to cited text no. 3
    
4.
Vienne P, Exner GU. Garrè sclerosing osteomyelitis. Orthopade 1997;26:902-7.  Back to cited text no. 4
    
5.
Mollan RA, Craig BF, Biggart JD. Chronic sclerosing osteomyelitis. An unusual case. J Bone Joint Surg Br 1984;66:583-5.  Back to cited text no. 5
    
6.
Eswar N. Garre′s osteomyelitis: A case report. J Indian Soc Pedod Prev Dent 2001;19:157-9.  Back to cited text no. 6
[PUBMED]    
7.
Suma R, Vinay C, Shashikanth MC, Subba Reddy VV. Garre′s sclerosing osteomyelitis. J Indian Soc Pedod Prev Dent 2007;25:S30-3.  Back to cited text no. 7
    
8.
Nortjé CJ, Wood RE, Grotepass F. Periostitis ossificans versus Garrè′s osteomyelitis. Part II: Radiologic analysis of 93 cases in the jaws. Oral Surg Oral Med Oral Pathol 1988;66:249-60.  Back to cited text no. 8
    
9.
Neville BW, Damm DD, Allen CM, Bouquot JE. Pulp and periapical diseases. In: Neville BW, editor. Oral and Maxillofacial Pathology. 3rd ed. Missouri: Saunders Elsevier; 2009. p. 148-55.  Back to cited text no. 9
    
10.
Benca PG, Mostofi R, Kuo PC. Proliferative periostitis (Garré′s osteomyelitis). Oral Surg Oral Med Oral Pathol 1987;63:258-60.  Back to cited text no. 10
    
11.
Felsberg GJ, Gore RL, Schweitzer ME, Jui V. Sclerosing osteomyelitis of Garrè (periostitis ossificans). Oral Surg Oral Med Oral Pathol 1990;70:117-20.  Back to cited text no. 11
    
12.
Suei Y, Taguchi A, Tanimoto K. Diagnosis and classification of mandibular osteomyelitis. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2005;100:207-14.  Back to cited text no. 12
    
13.
Oulis C, Berdousis E, Vadiakas G, Goumenos G. Garre′s osteomyelitis of an unusual origin in a 8-year-old child. A case report. Int J Paediatr Dent 2000;10:240-4.  Back to cited text no. 13
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]



 

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