Melorheostosis and central giant cell granuloma of the mandible in a 15-year-old girl

Anderson, Kenneth; Shintaku, Werner H.; Rosebush, Molly S.; Rawal, Yeshwant B.; Woodard, E. S.

doi:10.1016/j.oooo.2013.07.028

Cited by 5 publications

(16 citation statements)

References 15 publications

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“…Anderson et al compiled a list of 8 published reports in the English-language literature in November 2013 in Table I. These cases involved the cranio-facial region. [10] ("Current case" in the table, is the 9 th case reported by Anderson et al) Our case makes it the 10th such case. (Table 1) Per above table 10 , most cases were noted in females as a swelling in the maxilla or mandible with no associated pain or displacement of teeth.…”

Section: Discussionmentioning

confidence: 71%

“…[10] ("Current case" in the table, is the 9 th case reported by Anderson et al) Our case makes it the 10th such case. (Table 1) Per above table 10 , most cases were noted in females as a swelling in the maxilla or mandible with no associated pain or displacement of teeth. [10] Our patient is also a female and presents with nonsymptomatic involvement of multiple facial bones.…”

Section: Discussionmentioning

confidence: 71%

“…[3] Various theories have been proposed which include vascular disturbances, inflammatory and degenerative processes, defects in embryogenesis, and abnormalities in innervation. [3,4,10] The incidence of associated vascular anomalies is about 5%. [5] Melorheostosis is a rare sclerosing bone dysplasia which is characterized by a localized, diffuse thickening of the cortical bone.…”

Section: Discussionmentioning

confidence: 99%

“…It is most common in the diaphysis of long bones, and bones of hand and feet unilaterally. [3,5,6,10,13] Exclusive craniofacial skeleton involvement by melorheostosis is very rare. Literature search was conducted using PubMed, Query, Web of Science, and CINAHL databases using the following terms: melorheostosis, jaw, mandible, maxilla, craniofacial, and dripping candle wax.…”

Section: Discussionmentioning

confidence: 99%

“…[3,8] The incidence of this dysplasia has been reported to be 0.9 cases per million. [2,3,5,9,10] It does not show a gender or age predilection and in 40-50% of cases, it is evident usually by the age of 20 years. [2,5,10] Melorheostosis has been associated with abnormalities of mesodermal origin like glomus tumors, vascular nevi, hemangiomas, arteriovenous malformations, lymphangiectasis, lymphangiomas, neurofibromatosis, hypophosphatemic rickets, linear scleroderma etc.…”

Section: Introductionmentioning

confidence: 99%

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Osteosclerotic Bone Dysplasia of the Craniofacial Bones

Pannu

Katkar²,

Nair

2015

International Journal of Medical and Dental Sciences

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Melorheostosis is a rare sclerosing bone dysplasia which is characterized by a localized, diffuse thickening of the cortical bone mainly affecting diaphysis of long bones, and bones of hand and feet unilaterally. Involvement of the maxillofacial region is rare and the diagnosis is mainly based on radiographic features. We report a rare case of a 5 year old girl with Osteosclerotic Bone Dysplasia (Melorheotosis) of multiple craniofacial bones.

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Section: Discussionmentioning

confidence: 71%

Section: Discussionmentioning

confidence: 71%

Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

See 3 more Smart Citations

Osteosclerotic Bone Dysplasia of the Craniofacial Bones

Pannu

Katkar²,

Nair

2015

International Journal of Medical and Dental Sciences

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Cross‐Sectional Imaging Useful in Melorheostosis

et al. 2021

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Melorheostosis is a rare disease of bone overgrowth that is primarily diagnosed based on imaging studies. Recently, the association of different radiological patterns of the disease with distinct genetic cause was reported. Several case reports have described the radiological findings in patients with melorheostosis. However, the added value of cross-sectional imaging with CT and MRI beyond X-rays has not been investigated. The aim of the current study was to investigate this existing gap in knowledge. Forty patients with melorheostosis seen at the National Institute of Health Clinical Center were included in the study, and all their imaging studies were analyzed. The sequence of interpretation was X-ray followed by CT and then MRI. CT images were extracted from whole-body 18Fsodium fluoride positron emission tomography/CT studies. The information from CT reclassified the initial X-rays based radiological pattern in 13 patients. Additionally, CT comprehensively identified joint involvement and disease extent. In 76% of patients (n = 29) who underwent MRI, additional findings were noted, ranging from soft tissue edema to identification of soft tissue masses and incidental findings. MRI did not provide additional information on skeletal lesions beyond CT scans. However, it revealed the extension of soft tissue ossification into ischiofemoral space in four patients who complained of deep gluteal pain consistent with ischiofemoral impingement syndrome. In addition, MRI revealed soft tissue edema in 20 patients, 9 of whom had bone marrow edema and periosteal edema in the tibias consistent with shin splints. These findings suggest that select patients with melorheostosis should be evaluated with both CT and MRI, particularly patients in whom the distribution of pain does not correlate with the anatomic location of the disease in plain radiographs.

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