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Oral Surgery, Oral Medicine, Oral... Jan 2022The purpose of this study was to describe an unusual case of intraoral juvenile xanthogranuloma (JXG) and to perform a systematic review to investigate the available... (Review)
Review
OBJECTIVE
The purpose of this study was to describe an unusual case of intraoral juvenile xanthogranuloma (JXG) and to perform a systematic review to investigate the available literature regarding oral JXGs.
STUDY DESIGN
We present a new case of oral JXG arising in a 36-year-old Italian woman and conducted a systematic literature review in PubMed, Web of Science, and Scopus, according to the PRISMA guidelines.
RESULTS
Our review of the English-language literature yielded 34 cases of oral JXG, which included our case report.
CONCLUSIONS
JXG is a non-Langerhans cell histiocytosis. Oral JXG has been reported, but it is a rare manifestation. Because of the rarity of oral lesions and possible variations in the clinical and histologic presentation, the correct diagnosis can be challenging, requiring a careful clinical and histopathologic evaluation with adjuvant immunohistochemical studies.
Topics: Adult; Female; Humans; Mouth Diseases; Xanthogranuloma, Juvenile
PubMed: 34736900
DOI: 10.1016/j.oooo.2021.09.015 -
Journal of International Society of... 2020This study aimed to review the success or remission of intralesional medicaments in the management of intraosseous lesions in the oral cavity.
AIMS AND OBJECTIVES
This study aimed to review the success or remission of intralesional medicaments in the management of intraosseous lesions in the oral cavity.
MATERIALS AND METHODS
A comprehensive search was performed in two databases (PubMed and Scopus). Research articles, case reports, case series, and clinical trials were included. Review articles, lesions not involving the bone, incomplete reporting, any other treatment other than intralesional medicaments to treat intraosseous bone lesions, publications without any treatment, and letter to editor were excluded. Data on remission (complete, partial, or no remission), details and regimen of the intervention, number of participants, and follow-up in months were recorded.
RESULTS
A total of 653 publications were available for title and abstract screening after the removal of duplicates. Seven articles were excluded, which were not in English. After title and abstract screening, a total of 88 publications were available for full-text screening. Fifty-five articles were included in qualitative synthesis. A total of 168 patients from 55 publications were evaluated. Minimum follow-up was 1 month and maximum was 264 months. More than two-third ( = 38) of the publications were case reports on single patient. More than two-third ( = 38) of the publications had complete remission.
CONCLUSION
Intralesional medications have shown variable success rates. Extensive lesions may undergo intralesional medications followed by surgical management.
PubMed: 32181219
DOI: 10.4103/jispcd.JISPCD_295_19 -
Neurosurgery Oct 2019Erdheim-Chester disease (ECD) is a rare, non-Langerhans cell histiocytosis. Up to 50% of patients develop central nervous system involvement, and a subset of these...
BACKGROUND
Erdheim-Chester disease (ECD) is a rare, non-Langerhans cell histiocytosis. Up to 50% of patients develop central nervous system involvement, and a subset of these patients can present with isolated tumor-like masses.
OBJECTIVE
To describe the skull base manifestations of ECD with an emphasis on aspects most pertinent to surgeons who may be referred such patients for primary evaluation.
METHODS
Scopus, Web of Science, and PubMed were searched from database inception to May 1, 2018 for articles reporting skull base ECD. An institutional retrospective analysis of all patients treated at the authors' institution since January 1, 1996 was also performed to supplement these data.
RESULTS
Of 465 retrieved articles, 18 studies totaling 20 patients met inclusion criteria. Institutional review identified an additional 7 patients. Collectively, the median age at diagnosis was 49 yr (interquartile range, 42-58) with a 4:1 male-to-female ratio. Patients frequently presented with diplopia (48%), headache (30%), dysarthria (22%), and vertigo or imbalance (22%), though trigeminal hypesthesia (11%), facial nerve paresis (7%), hearing loss (7%), and trigeminal neuralgia (7%) were also observed. ECD commonly mimicked meningioma (33%), trigeminal schwannoma (8%), neurosarcoidosis (8%), and skull base lymphoma (8%).
CONCLUSION
Discrete skull base lesions frequently mimic more common pathology such as meningioma or cranial nerve schwannomas. Medical therapy comprises the initial treatment for symptomatic skull base disease. Surgical resection is not curative and the utility of surgical intervention is largely limited to biopsy to establish diagnosis and/or surgical debulking to relieve mass effect.
Topics: Adrenal Cortex Hormones; Adult; Erdheim-Chester Disease; Female; Humans; Male; Middle Aged; Radiosurgery; Retrospective Studies; Skull Base
PubMed: 30828728
DOI: 10.1093/neuros/nyz027 -
Erdheim-Chester Disease With Extensive Pericardial Involvement: A Case Report and Systematic Review.Cardiology Research Apr 2020Erdheim-Chester disease (ECD) is a rare non-Langerhans cell histiocytosis characterized by systemic xanthogranulomatous infiltration. We described the case of a female...
Erdheim-Chester disease (ECD) is a rare non-Langerhans cell histiocytosis characterized by systemic xanthogranulomatous infiltration. We described the case of a female adult presenting with pericardial effusion. Pericardial infiltration is the most frequent cardiac manifestation of ECD and is the one discussed in this article. We found that the majority of patients with pericardial infiltration needed a cardiovascular procedure.
PubMed: 32256919
DOI: 10.14740/cr1025 -
Child's Nervous System : ChNS :... Aug 2023Eosinophilic granuloma (EG) is the most common form of Langerhans cell histiocytosis, presenting as a single osteolytic lesion of the calvarium. Its diagnosis is based...
INTRODUCTION
Eosinophilic granuloma (EG) is the most common form of Langerhans cell histiocytosis, presenting as a single osteolytic lesion of the calvarium. Its diagnosis is based on typical clinical and radiological features. While surgical resection has been the standard treatment for EG, growing evidence favors watchful waiting, as unifocal calvarial lesions appear to frequently undergo spontaneous remission. However, histopathological confirmations of this hypothesis are still very limited.
METHODS
Methods. Here, we report a case of EG with typical clinical and radiological features which, due to intervening circumstances, was resected in a delayed fashion. Moreover, we perform a systematic review of the literature on conservative management of EG.
RESULTS
In our case, histological examination showed ongoing bone regeneration with no traces of the disease. Through our literature review, we found 47 cases of calvarial EG managed with watchful waiting. No active intervention was required in 43 cases (91%). Four patients (9%) received surgery or chemotherapy due to the persistence/progression of symptoms or family request. Three reports other than ours documented spontaneous disease remission in surgically resected EG upon histopathological examination.
CONCLUSION
Our report provides further evidence that watchful waiting can be a reasonable option in the management of single calvarial EG.
Topics: Humans; Eosinophilic Granuloma; Conservative Treatment; Skull; Histiocytosis, Langerhans-Cell; Radiography; Remission, Spontaneous
PubMed: 36939904
DOI: 10.1007/s00381-023-05926-z