Sickle cell disease and jawbone osteomyelitis: case report and literature review | Journal of Oral Medicine and Oral Surgery

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Table I

Literature review of jawbone osteomyelitis in patient with sickle cell disease.

Author and year of case report	Number of cases	Age/gender	Sickle cell disease type/ Recent VOC of other localization	Affected jaw	Initial symptoms	Radiographic manifestation	Pathogen involved	Treatment
*Ryan et al.* (1971)** [6]	1	36/M	N/A N/A	Mandible	Abscess with mandibular third molar.	Loss of supporting alveolar bone	Normal flora plus Staphylococcus aureus	Antibiotherapy, Drainage, sequestrectomy
*Walker et al.* (1973)** [7]	1	21/M	Homozygous SS On admission, pain in the elbow region, lower back and hips (repeated admission for treatment of sickle cell crises)	Mandible	Tooth-ache, Tooth infection, Jaw pain, unilateral facial swelling, Trismus	No apparent change of inferior border or angle of mandible, osteoporosis of left mandible, Periodontal disease	N/A	Antibiotherapy, Drainage, Tooth extraction, Sequestrectomy
*Girasole et al.* (1977)** [8]	3	22/M 34/F 33/M		Mandible Mandible Mandible			Staphylococcus aureus Streptococcus viridans Pseudomonas spp.	Sequestrectomy and curetage Debridement and corticotomy Sequestrectomy and curetage
*Daramola et al.* (1982)** [9]	1	28/M	N/A N/A	Mandible	Painful facial swelling, purulent periodontal pockets, trismus	Patchy areas of radiolucency and radiopacity suggesting bone destruction and sequestrum formation, pathological fracture	Salmonella spp.	Antibiotherapy, sequestrectomy with loose teeth removal, bone cavity was saucerised and the wound packed open with Eusol on ribbon gauze
Hammersley (1984) [10]	1	29/W	Homozygous SS During an hospitalisation for a severe limbs crisis (≈ 2 VOC/year)	Mandible	Mandibular pain, Mental nerve anaesthesia	Alveolar bone only showed classic osteoporosis caused by extramedullary haemopoiesis as a result of severe haemolytic anaemia	N/A	Antibiotherapy, Pain relief in 8 days, Partial resolution of the anesthesia in 8 months (residual paraesthesia)
*Grodecki et al.* (1985)** [11]	1	19/W	N/A N/A	Mandible	Bilateral buccal and submandibular facial swelling, Trismus, lower lip hypoaesthesia, Periodontal infection of the 1st molar	Diffuse moth-eaten radiolucent areas in the area of the molars, and a bilateral proliferative periostitis on the buccal surfaces of the involved areas increasing the mandibular breadth.	Mixed oral flora / Salmonella tiphy	Drainage, Antibiotherapy, Sequestrectomy with teeth extraction under general anaesthesia (with preoperative exchange transfusion)
Iwu (1989) [12]	3	16/W 20/M 18/W	Homozygous SS (repeated admission for treatment of sickle cell crises) Homozygous SS Homozygous SS	Mandible Mandible Mandible	Facial pain and swelling, tooth mobility with whitish alveolar bone exposed	N/A Buccal expansion of the right ascending ramus and bilateral radiolucency of the body of the mandible. N/A	Negative N/A Negative	Antibiotherapy, teeth extraction with sequestrectomy of necrotic bone
*Patton et al.* (1990)** [13]	1	34/M	N/A 2 days after the onset of a sickle cell crisis	Mandible	Jaw pain, Facial swelling, Paraesthesia of inferior alveolar and mental nerve, teeth mobility and periodontal infection	Mandible lytic lesion in the molar area, Coarse trabeculation and osteoporotic bone character	Normal flora	Antibiotherapy, teeth extraction with debridement of necrotic bone
*Shroyer et al.* (1991)** [14]	1	22/M	Homozygous SS sickle cell crisis 7 days before his admission	Mandible	Facial pain and swelling, trismus, purulent pericoronitis of 3rd molar	N/A	Eikenella corrodens, Bacteroides melaninogenicus, Peptostreptococcus spp., mixed Streptococcus spp. and Staphylococcus spp.	Antibiotherapy, drainage, teeth extraction,resection of devitalized bone and muscles, hemimandibulectomy in 2^nd intention (infected relapse)
*Bishop et al.* (1995)** [15]	1	41/M	N/A During a recent stay in hospital for a sickle cell crisis (SCC) / Patient was not aware of any oral problems prior to the SCC	Mandible	Jaw pain, unilateral lower lip anesthesia, teeth mobility and periodontal infection, pericoronitis of 3rd molar	Well-defined multi-locular radiolucency in the molar area of the mandible	N/A	Antibiotherapy, Teeth extraction, multiple root canal treatments
*Podlesh et al.* (1996)** [16]	1	21/M	N/A Pain and fever consistent with sickle cell crisis (multiple admissions for painful crisis)	Mandible	Jaw pain, profound anesthesia of the mental nerve	Radiodensity associated with the apecies of the left mandibular first molar	Negative	Antibiotherapy, spontaneous improvement, no dental treatment because no symptoms
*Olaitan et al.* (1997)** [5]	16	12-30 13 M 3 F	11 Homozygous SS, 5 Heterozygous SC N/A	Mandible	Pericoronitis with impacted molar either bilaterally or unilaterally (n=12), carbuncle on the skin (n=1), uncertain aetiology (n=3).	N/A	Mixed (n = 5), Staphylococcus aureus (n = 4), Microaerophyllic streptococcus (n=1), Non-haemolytic streptococcus (n=1), Klebsiella species (n=1). none (n = 4)	Antibiotherapy, Sequestrectomy alone or in combination with extraction of wisdom teeth (n=12), Medical treatment only (complete healing in n=1/4)
*Borle et al.* (2001)** [17]	1	25/W	N/A N/A	Maxilla	Facial pain and swelling, con- gestion of the right maxillary gingiva, discharge of pus, and exposure of the bone from first premolar to third molar region	Large sequestrum of bone extending from right maxillary first premolar to the maxillary tuberosity	Pseudomonas aerugenosa	Antibiotherapy, Sequestrectomy
*Kavadia-Tsatala et al.* (2004)** [18]	6	22-34 3M 3F	N/A N/A	Mandible	Pain in maxillofacial region, no evidence of dental pathology or other infection	Intraosseous, radiopaque, quite homogeneous, adequately well-defined, multilobular lesions	N/A	N/A
*Watanabe et al.* (2013)** [19]	4	6-22 3M 1F	3 Homozygous SS, 1 Heterozygous SC N/A	Mandible	Jaw and facial pain, facial swelling	N/A	N/A	N/A
*DeBlieux et al.* (2014)** [20]	1	18/F	N/A During hospitalisation for a sickle cell crisis of her left leg. (monthly admissions to the hospital for sickle cell crisis)	Maxilla, Mandible	Facial swelling, trismus, paresthesia of the upper lip, dental carie, impacted 3rd molar	Numerous nonenhancing subperiosteal collections over the right maxilla, right body of the mandible, and left subcondyle	Negative	Intraoral decompression of the infarcts
*Araújo et al.* (2015)** [21]	1	28/M	Homozygous SS Hospitalization 1 month-prior for chest and femoral pain	Mandible	Pain, bilateral teeth mobility, gingiva recession of the lower molars	2 radiolucent symmetrical periapical lesions evolving both the first and the second lower molars, bilaterally	Streptococcus viridans	Antibiotherapy, Teeth extractions with sequestrectomy
*Al-Ismaili et al.* (2016)** [22]	3	23/M 16/M 25/M	N/A 3 weeks prior the patient had been admitted due to a sickle cell crisis VOC episode two weeks prior to the start of his symptoms. following his admission to a local hospital due to a severe VOC. (history of frequent hospital admissions)	Mandible Mandible Maxilla	Suppurative alveolitis of molar site, lower lip hypoaesthesia Pericoronitis	Area of radiolucency related to molar root	Actinomyces spp., Klebsiella pneumoniae and Moraxella catarrhalis Streptococcus anginosus Actinomyces spp.	Antibiotherapy, surgical curettage/ sequestrectomy of the mandible with removal of the involved teeth, bismuth iodine paraffin paste (BIPP) packing of surgical site
*Mahendran et al.* (2020)** [23]	2	34/M 38/F	N/A N/A Recent hospital admission for sickle crisis Three days prior, she was admitted for management of an acute painful vaso-occlusive crisis.	Mandible Mandible	Bilateral facial swelling and altered sensation of the lower lip, trismus	Irregular radiolucency in the mandible, buccal plate cortical pitting and alveolar bone destruction	N/A	Antibiotherapy, Oral bisphosphonates (risendronate/ oral alendronic acid)
*Chang et al.* (2021)** [24]	1	37/M	Hemoglobin S-Beta-Thalassemia N/A	Mandible	Facial pain and swelling	N/A	N/A	Antibiotherapy, hemimandibulectomy and reconstruction with a titanium prosthesis + radial forearm and adipofascial free flap
Present case (2024)	1	22/M	Homozygous SS 2 weeks after management of a vaso-occlusive hip crisis	Mandible	Facial pain and swelling, Paresthesia of the lower lip, pericoronitis of decayed 3rd molar, secondary abnormal teeth mobility	No suggestive radiological signs were found on the panoramic radiograph and CT scan	Staphylococcus aureus	Antibiotherapy, surgical curettage/ sequestrectomy of the necrotic bone with removal of the involved teeth

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