Central xanthoma of the jaw in association with Noonan syndrome

Olson, Nicholas; Addante, Rocco R.; Abreu, Francine B. de; Memoli, Vincent A.

doi:10.1016/j.humpath.2018.04.020

Cited by 10 publications

(25 citation statements)

References 14 publications

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“…Mean age was 27.8 ± 14.3 years for individuals with lesions in the mandible and 48.5 ± 8.5 years for individuals with lesions in the maxilla. Only four cases reported the time of lesion evolution, including 1 day, 2, 3 and 54 months, respectively . Asymptomatic individuals (n = 16, 51.6%) were the majority, followed by patients complaining of painless swelling (n = 9, 29.0%).…”

Section: Resultsmentioning

confidence: 99%

“…After the elimination of 192 duplicates, the parameters for inclusion and exclusion were applied to 442 references. A total of 16 articles reporting 31 cases of IOX of the jaws were selected . The studies included in the present literature review were case reports or case series.…”

Section: Resultsmentioning

confidence: 99%

“…The studies included in the present literature review were case reports or case series. The articles were from the US, Canada, Brazil, Sweden, the Netherlands, Spain, the UK, Japan and Australia …”

Section: Resultsmentioning

confidence: 99%

“…Twelve studies carried out immunohistochemistry analysis as a tool to confirm diagnosis, representing a total of 26 cases (83.9%). Twenty different markers were evaluated, and the most commonly used being CD68+ (n = 24), S‐100‐ (n = 14), CD1a‐ (n = 10) and CD163+ (n = 10).…”

Section: Resultsmentioning

confidence: 99%

“…Thus far, few cases of IOX of the jaws with enough clinicopathologic and radiographic details to confirm the diagnosis have been documented in the literature . Since case series of IOX of the jaws are infrequent in the literature, in order to provide consistent and well‐documented reports that may serve as a basis for a better understanding of the lesion, we describe here a series of five new cases of IOX of the mandible in a multi‐institutional study, with emphasis on clinicopathological, radiographic and immunohistochemical data of this condition as compared with reports detected in a literature review.…”

Section: Introductionmentioning

confidence: 92%

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Intraosseous xanthoma of the mandible: A multi‐institutional case series with a literature review

Arruda

Almeida

Abreu

et al. 2019

J Oral Pathology Medicine

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Background The aim of the present study was to report the clinicopathologic, radiographic and immunohistochemical features of five South American cases of intraosseous xanthomas of the mandible and to compare them to those detected in a literature review. Methods Clinical data were collected from the records of three Oral and Maxillofacial Pathology services in South America and compared with those compiled from a literature review based on a search of three electronic databases (PubMed, Web of Science and Scopus). All cases were evaluated by haematoxylin and eosin staining and immunohistochemistry for CD68 and S‐100. Results The series comprised four females (80%) and one male (20%) with a mean age of 23.3 ± 10.9 years (range: 13‐45 years). In four cases, there was involvement of the posterior region of the mandible (80%). The lesions presented radiographically as unilocular (60%) radiolucencies with punched‐out margin (80%). All cases predominantly consisted of CD68‐positive and S‐100‐negative xanthomatous cells. No recurrences were observed after curettage, with a median follow‐up of 27 months. Conclusion Intraosseous xanthoma of the jaws is a rare benign disorder. We report here five additional cases affecting the mandible, for a total of 36 cases of the jaws reported in the literature. Overall, this lesion has predilection for posterior sites of the mandible of asymptomatic young adults.

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Section: Resultsmentioning

confidence: 99%

Section: Resultsmentioning

confidence: 99%

Section: Resultsmentioning

confidence: 99%

Section: Resultsmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 92%

See 3 more Smart Citations

Intraosseous xanthoma of the mandible: A multi‐institutional case series with a literature review

Arruda

Almeida

Abreu

et al. 2019

J Oral Pathology Medicine

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Primary Intraosseous Xanthomas of the Jaws: A Series of Six Cases Including an Example with Formation of Apoptosis-Related Hyaline Globules, So-Called “Thanatosomes”

Wilkinson

Merkourea

Gopalakrishnan

et al. 2020

Head and Neck Pathol

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Primary intraosseous xanthomas of the jaws (PIXJ) are rare and predominantly affect the posterior mandible (86%) of normolipemic patients, with a mean age of 30 years and no gender predilection. Clinically, PIXJ exhibit indolent biologic behavior; curettage is considered treatment of choice. Only 36 PIXJ have been reported. Apoptosis-related hyaline globules (HGs), also known as "thanatosomes", have not been previously reported in PIXJ. Cases diagnosed as xanthoma of bone were retrieved. Six cases fulfilling currently accepted criteria were identified and their clinicopathologic and immunohistochemical properties are presented herein. Mean age for PIXJ was 21.8 years (range = 12-33) and F:M ratio = 2:1. All cases presented as well-demarcated, unilocular or multilocular radiolucencies. Microscopically, PIXJ featured sheets of lipid-laden macrophages with eosinophilic or foamy cytoplasm. A secondary fibroblastic population lacking storiform pattern was evident in two cases. Adipocytes (3/6), peripheral neurovascular bundles (1/6), bone fragments (3/6) and dystrophic calcifications (3/6) were observed enclosed by the xanthoma cells. Notably, one case exhibited numerous, spherical, eosinophilic HGs containing apoptotic nuclei. PIXJ were consistently CD68(+) and negative for CD1α and S100. CD45 decorated lymphocytes and the membrane of foamy histiocytes. Xanthoma cells stained for lysozyme and plasma proteins including alpha-1 antitrypsin (AAT), IgG and IgA in one probed case. HGs were lysozyme(+), AAT(+), IgG(+), IgA(+), PAS(+) and diastase-resistant, and fuchsinophilic with Masson's trichrome. PIXJ represent infrequent, solitary, mandibular lesions with a predilection for the second and third decade of life. Thanatosomes associated with cell injury and death can be present in PIXJ.

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Xanthoma of the Jaw Bones: Cases Series and Review of Literature

Venkat,

Islam,

Bhattacharyya

et al. 2024

Head and Neck Pathol

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Background Intraosseous xanthomas are rare benign lesions sometimes associated with excess lipid production. Xanthoma of the jaw bones (XJB) was first reported in 1964, and fewer than 50 cases have been reported in the English literature to date. The etiopathogenesis of XJB is highly suggestive of a reactive process or a metabolic condition. Method Seven cases of XJBs were retrieved from the archives of 4 oral and maxillofacial pathology services. Clinical, radiographic and histopathologic features of all these cases were retrospectively analyzed. Immunohistochemical (IHC) stains for S100 and CD68 were performed. Results All seven cases involved the mandible. Patients’ age ranged between 13 and 69 years with an evenly distributed female to male ratio. One patient had a medical history of hyperlipidemia, but the medical and dental histories of the others were unremarkable. For most cases, XJB was an incidental finding discovered during routine radiographic examination. Swelling and cortical expansion were noted in a few cases. Radiographically, cases typically presented as either well-defined multilocular or unilocular lesions, which were either radiolucent or mixed radiolucent/radiopaque. All the lesions were treated with surgical curettage and no recurrence was observed during subsequent follow-ups. Each of the seven cases exhibited sheets of foamy macrophages. The diagnosis is established by exclusion of entities with overlapping microscopic features and involved correlation with the clinical, histological, radiographic and IHC profiles. Immunohistochemically, all the cases expressed diffuse positivity for CD68 and were negative for S100. Conclusion XJB is a rare lesion of unknown etiology, which may mimic other benign or reactive jaw lesions. Due to its rarity and the potential diagnostic challenges it presents, clinicians must remain vigilant and consider CXJ in their differential when assessing radiolucent jaw anomalies.

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Central xanthoma of the jaw in association with Noonan syndrome

Cited by 10 publications

References 14 publications

Intraosseous xanthoma of the mandible: A multi‐institutional case series with a literature review

Intraosseous xanthoma of the mandible: A multi‐institutional case series with a literature review

Primary Intraosseous Xanthomas of the Jaws: A Series of Six Cases Including an Example with Formation of Apoptosis-Related Hyaline Globules, So-Called “Thanatosomes”

Xanthoma of the Jaw Bones: Cases Series and Review of Literature

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