Orbital Giant Cell Reparative Granuloma: A Case Report.

Giant cell reparative granuloma has a very low incidence and is thought to be a response to trauma. While there have been only a few reported cases of orbital giant cell reparative granuloma, we recently observed such a case and analyzed 16 previously reported cases of this type. It is important to note that further investigation is necessary to fully understand the relationship between giant cell reparative granuloma and trauma.

Response of Central Giant Cell Granuloma of the Jaw to Imatinib.

Central giant cell granuloma of the jaw (CGCJ) can be locally aggressive and result in facial and dental deformity. A child with CGCJ was treated surgically and with denosumab with a response but life-threatening toxicity. Imatinib, a tyrosine kinase inhibitor, was prescribed based on clinical similarities between CGCJ and cherubism, for which Imatinib has been effective. Within 2 months, a computed tomographic scan showed significant ossification, which increased over the following 8 months. This case suggests that tyrosine kinase inhibitors may be an effective option, and one with limited toxicity, for CGCJ.

Giant cell epulis.

Giant cell epulis (peripheral giant cell granuloma) typically appears as a reactive benign lesion in the oral cavity in areas following local irritation or chronic trauma. Here we describe the case of a 45-year-old male patient who presented with the chief complaint of a large gingival mass in the anterolateral maxilla. There had been progressive growth within the past few months, with increased painless discomfort during mastication. The patient also reported bleeding during interdental cleaning. A full physical work-up led to the suspicion of giant cell epulis alongside other differentials including mucosal hemangioma and squamous cell carcinoma, with unremarkable laboratory values. Imaging including computed tomography showed signs of previous insertion of metal implants on either side of the lesion alongside mucosal hyperplasia. A confirmatory biopsy was taken and showed multiple giant cells on a reactive bed of stroma, in line with the diagnosis of giant cell epulis. Oral inflammatory conditions such as giant cell epulis have greater chances of local recurrence and, therefore, careful investigation with timely and accurate diagnosis is imperative for appropriate early treatment. Complete surgical excision should then be employed to prevent relapses, as incomplete removal can lead to further recurrence. Identification and eradication of potential sources of irritation should also be considered when treating the patient, to avoid further recurrence.

Pediatric Aggressive Benign Mandibular Tumors: Clinical Features and Management.

INTRODUCTION: Aggressive benign mandibular tumors are uncommon in the pediatric population, and there is few publishing in the literature specifically dealing with them. Aggressive tumors can be defined based on known biologic behavior and/or histologic type and/or clinical characteristics. AIM OF THE STUDY: To review the clinical features and management of lower jaw pediatric aggressive benign tumor. PATIENTS AND METHODS: Medical records review of pediatric patients presented with aggressive benign mandibular tumors to the Maxillofacial and Plastic Surgery Department, University of Alexandria, Egypt between 2011 and 2019. RESULTS: Fifty-eight patients were included in this study, aged between 2 and 16 years (average = 11.8). Ameloblastoma was the commonest pathological diagnosis (n = 18) followed by central giant cell granuloma (n = 11) and juvenile ossifying fibroma (n = 10). Patients with central giant cell granuloma were treated by en-block resection (n = 4) or curettage after interferon alfa injection (n = 7). All other benign tumors were treated by en-block resection. The length of follow-up ranged from 1 to 8 years. Successful reconstruction was accomplished in 45 patients (88.2%). CONCLUSIONS: Aggressive lesion should be treated in an aggressive manner and immediate reconstruction is advocated. However, pharmacotherapy combined with enucleation is a more conservative approach for management of aggressive central giant cell tumors.

Can Methotrexate be a successful treatment for unresponsive generalized annular elastolytic giant cell granuloma? Case report and review of the literature.

Annular elastolytic giant cell granuloma (AEGCG) is a rare granulomatous skin disorder, characterized by erythematous plaques with elevated borders and hypopigmented center, occurring mainly on sun exposed-skin. Histologically it presents with elastophagocytosis and elastolysis. There is no established first line treatment for AEGCG, especially for the generalized form. In a small number of cases, antimalarial drugs and tranilast, associated to topical or oral steroids, have been proposed to treat generalized AEGCG with partial benefits. We herein present the case of a patient with AEGCG aged 74 years, who was unresponsive to classical therapies, and then successfully treated with methotrexate.

Giant cell reparative granuloma of the phalanx in a violinist.

Giant cell reparative granuloma (GCRG) is a rare, pseudotumoural intraosseous lesion, considered a reactive injury after repeated trauma. Reactive lesions and benign bone tumours may show aggressive clinical and radiographic findings. Differential diagnosis must be performed in order to offer suitable treatment to the patient. Excisional biopsy and curettage of the lesion are the preferred methods of treatment. We present the first case of a GCRG of the distal phalanx of the left little finger in a professional violinist.

Pathologic evaluation of the solid variant of aneurysmal bone cysts with USP6 rearrangement with an emphasis on the frequent diagnostic pitfalls.

The solid variant of aneurysmal bone cyst (SVABC) is very uncommon and frequently misdiagnosed. We reevaluated and summarized the clinicopathologic features of 17 SVABCs and further discussed the use of this nomenclature to differ SVABCs from extragnathic giant cell reparative granuloma (GCRG) in the setting of the USP6 rearrangement era. The immunohistochemical markers included α-SMA, SATB2, AE1/AE3, Ki67, S100, CD68 and P63. USP-6 status was detected by fluorescence in situ hybridization using a break-apart probe. The 17 patients with SVABCs comprised 10 males and 7 females ranging in age from 4 to 70 years. The involved locations included the long bone (n = 11), hand (n = 4), rib (n = 1) and vertebra (n = 1). The lesions were characterized by proliferated spindle cells with scattered giant cells and hemorrhages with variable positive α-SMA, SATB2, CD68 and Ki-67 expression. All patients had USP6 rearrangements without H3F3A glycine 34 mutations. Our study reveals that SVABC shares similar clinical and histologic features with other bone lesions, which may lead to an erroneous diagnosis. The presence of an USP-6 rearrangement contributes to the diagnosis SVABC; SVABC and most of the previously documented extragnathic GCRGs may be considered within the umbrella of primary aneurysmal bone cysts.

Giant cell reparative granuloma of the scapula: report of a case and literature review.

Giant cell reparative granulomas (GCRGs) are non-neoplastic inflammatory lesions, usually observed in the maxilla, mandible or small bones of the hands and feet. These lesions present a wide range of morphology and the misinterpretation with other giant cell lesions can often occur. We report the case of a 47-year-old woman with GCRG in the left scapula, presenting some uncommon features: the location (scapula) and age at presentation, the lack of underlying bone disease such as Paget's disease or fibrous dysplasia, the large aggressive expansile aspect of the lesion. This was a therapeutic study, level IV (case series with no or a historical control group).

Pulse granuloma presenting as a complex pelvic cyst.

Pulse granuloma, a benign granulomatous lesion, develops due to foreign body reactions to food particles. This rare entity occurs occasionally in the abdominal cavity. However, it has never been reported as a pelvic mass or a recurrent case. A 37-year-old woman with a medical history of pelvic granulomatous lesions treated in Thailand, was admitted for abdominal pain. Imaging study revealed a pelvic complex cyst and disseminated nodules. Ovarian cancer or tuberculous peritonitis was suspected. The appearance at laparotomy was an inflammatory mass encased in adhesions; therefore, only biopsy was performed. The pathology diagnosis was pulse granuloma. The pathology report from Thailand was obtained only after her discharge, suggesting that the previous granulomatous lesion contained vegetable matter. This report demonstrates that pulse granuloma can present as a pelvic mass and may relapse. For complex cystic lesions with solid components in the pelvis, pulse granuloma should be considered as a differential diagnosis.

Treatment of Annular Elastolytic Giant Cell Granuloma With Topical Tretinoin.

Annular elastolytic giant cell granuloma, also known as actinic granuloma, is a rare skin condition with a chronic course that is often resistant to treatment. Literature is sparse, and only a handful of case reports are available to guide treatment decisions. Typical first line treatment options include topical and intralesional steroids, topical pimecrolimus, and cryotherapy. Resistant cases have been treated with cyclosporine, systemic steroids, antimalarials, and oral retinoids. In particular, acitretin and isotretinoin have shown success in three cases. However, these medications can have side effects and require frequent lab monitoring. We present a case of a 47-year-old woman with bilateral forearm lesions consistent with annular elastolytic giant cell granuloma who was successfully treated with topical tretinoin.

J Drugs Dermatol. 2017;16(7):699-700.

A recurrent central giant cell granuloma in a young patient and orthodontic treatment: a case report.

Central giant cell granuloma (CGCG) is an uncommon benign intraosseous lesion of the jaw, found predominantly in children and young adults below 30 years of age. The purpose of this article was to present a summary of the current literature and a case report of an 11-year-old boy diagnosed with an aggressive CGCG involving the anterior maxilla that was removed in 2004 and subsequently recurred almost 3 years later in 2006. The presenting features of the patient and the effect of combined surgical and orthodontic treatment for this condition are discussed. This case shows how the dentition was successfully maintained with conservative surgery and orthodontic treatment in spite of the extensive destruction of the supporting bone, and the importance of long-term follow-up. The report also reminds orthodontic practitioners that rare pathological conditions can occur in their child patient groups.

Ectopic parathyroid adenoma presenting as facial expansile growths.

BACKGROUND: Giant cell reparative granuloma of the facial skeleton is a rare presentation of hyperparathyroidism. METHODOLOGY: A 17-year-old girl presented with progressive expansile bony growths of the mandible and maxilla. The case was investigated using biopsies, blood tests, imaging, and surgery. RESULTS: Biopsies confirmed giant cell reparative granuloma. Blood testing revealed very high parathyroid hormone levels, and brown tumours due to primary hyperparathyroidism weresuspected. Surgical removal of an ectopic adenoma led to correction of the endocrine condition. CONCLUSIONS: To our knowledge, this is the first reported case of multiple brown tumours of the facial skeleton due to an ectopic parathyroid adenoma. Diagnostic pitfalls and therapeutic challenges are described.

Annular Elastolytic Giant Cell Granuloma and Temporal Arteritis Following Herpes Zoster.

A 70-year-old woman, who presented with pain and functional limitation of her right shoulder, without any previous traumatic incident, was evaluated in an emergency department. A shoulder x-ray was performed, and she received an intrajoint injection of an unspecified amount of triamcinolone. Forty-eight hours later, she noticed a papulovesicular and bullous eruption with a dermatomal distribution on her nape, supraclavicle, and left brachial and antebrachial regions (T1-T2-C5-C6-8). A diagnosis of herpes zoster (HZ) was made, for which she received valacyclovir 1 g every 8 hours for 10 days, pregabalin 75 mg every 8 hours, and topical acyclovir, with good response. The lesions and pain subsided, and results from general laboratory tests were normal or negative.

Kissing dentigerous cysts involving mandibular canines: report of unusual case with review of literature.

Dentigerous cysts are odontogenic developmental cysts, which mostly surround the crown of unerupted teeth, odontomas or supernumerary teeth. These cysts are mostly solitary in occurrence and their bilateral presentation is rare, especially in the absence of syndromes such as Maroteaux-Lamy, basal cell nevus or cleidocranial dysplasia. We present an unusual case of a 24-year-old female with nonsyndromic bilateral dentigerous cysts associated with unerupted mandibular canines that were diagnosed histopathologically as dentigerous cysts.

Peripheral Giant Cell Granuloma in a Dog.

Peripheral giant cell granuloma is considered rare in the dog with little known about the clinicopathologic features. There are few reports in the veterinary literature concerning this benign, reactive lesion, formerly known as giant cell epulis. In humans, the four most commonly described reactive epulides are focal fibrous hyperplasia (fibrous epulis), pyogenic granuloma, peripheral ossifying fibroma, and peripheral giant cell granuloma. This case report describes the diagnosis and surgical management of a peripheral giant cell granuloma in a dog.

Total spontaneous regression of a central giant cell granuloma after incisional biopsy: a four-year follow-up case report.

Central giant cell granuloma (CGCG) of the jaws represents a localized and benign neoplastic lesion sometimes characterized by aggressive osteolytic proliferation. The World Health Organization defines it as an intraosseous lesion composed of cellular and dense connective tissues that contain multiple hemorrhagic foci, an aggregation of multinucleated giant cells, and occasional bone tissue trabeculae. The origin of this lesion is uncertain; however, factors such as local trauma, inflammation, intraosseous hemorrhage, and genetic abnormalities have been identified as possible causes. CGCG generally affects those younger than 30 years and occurs more frequently in women (2:1). This lesion corresponds to approximately 7% of all benign tumors of the jaws, with prevalence in the anterior region of the jaw. Aggressive lesions are characterized by symptoms, such as pain, numbness, rapid growth, cortical perforation, root resorption, and a high recurrence rate after curettage. In contrast, nonaggressive CGCGs have a slow rate of growth, may contain sparse trabeculation, and are less likely to move teeth or cause root resorption or cortical perforation. Nonaggressive CGCGs are generally asymptomatic lesions and thus are frequently found on routine dental radiographs. Radiographically, the 2 forms of CGCG present as radiolucent, expansive, unilocular or multilocular masses with well-defined margins. The histopathology of CGCG is characterized by multinucleated giant cells, surrounded by round, oval, and spindle-shaped mononuclear cells, scattered in dense connective tissue with hemorrhagic and abundant vascularization foci. The final diagnosis is determined by histopathologic analysis of the biopsy specimen. The preferred treatment for CGCG consists of excisional biopsy, curettage with a safety margin, and partial or total resection of the affected bone. Conservative treatments include local injections of steroids, calcitonin, and antiangiogenic therapy. Drug treatment using antibiotics, painkillers, and corticosteroids and clinical and radiographic monitoring are necessary for approximately 10 days after surgery. There are only a few cases of spontaneous CGCG regression described in the literature; therefore, a detailed case report of CGCG regression in a 12-year-old boy with a 4-year follow-up is presented and compared with previous studies.

Biopsy of different oral soft tissues lesions by KTP and diode laser: histological evaluation.

INTRODUCTION: Oral biopsy aims to obtain clear and safe diagnosis; it can be performed by scalpel or laser. The controversy in this latter application is the thermal alteration due to tissue heating. The aim of this study is the histological evaluation of margins of "in vivo" biopsies collected by diode and KTP lasers. MATERIAL AND METHODS: 17 oral benign lesions biopsies were made by diode 808 nm (SOL, DenMatItalia, Italy) and KTP 532 nm (SmartLite, DEKA, Italy). Samples were observed at OM LEICA DM 2000; margin alterations were evaluated through Leica Application Suite 3.4. RESULTS: Epithelial and connective damages were assessed for each pathology with an average of 0.245 mm and a standard deviation of ± 0.162 mm in mucoceles, 0.382 mm ± 0.149 mm in fibromas, 0.336 mm ± 0.106 mm in hyperkeratosis, 0.473 mm ± 0.105 mm in squamous hyperplasia, 0.182 mm in giant cell granuloma, and 0.149 mm in melanotic macula. DISCUSSION: The histologic aspect of lesions influenced the response to laser, whereas the greater inflammation and cellularity were linked with the higher thermal signs. Many artifacts were also associated to histologic procedures. CONCLUSION: Both tested lasers permitted sure histologic diagnosis. However, it is suggested to enlarge biopsies of about 0.5 mm, to avoid thermal alterations, especially in inflammatory lesions like oral lichen planus.

Computed tomography and magnetic resonance imaging of maxillofacial lesions in renal osteodystrophy.

PURPOSE: This study aims to describe the computed tomography (CT) and magnetic resonance (MR) imaging appearance of maxillofacial lesions in renal osteodystrophy. PATIENTS AND METHODS: We retrospectively reviewed the CT and MR imaging of maxillofacial region in 9 patients (6 females and 3 males with mean age of 31 yr) with renal osteodystrophy. They presented with facial swelling (n = 6), facial disfigurement (n = 2), and oral cavity mass (n = 1). They underwent CT and MR imaging of the maxillofacial region. RESULTS: Brown tumors (n = 6) were seen in the mandible (n = 4) and maxilla (n = 2). They appeared as mixed lytic and sclerotic (n = 4) and sclerotic (n = 2) lesions at CT. The lesions appeared as hypointense at T1-weighted images and of mixed signal intensity at T2-weighted images with intense contrast enhancement (n = 6). Uremic leontiasis ossea (n = 2) appeared at CT as diffuse hyperostosis with protruded maxilla and obliterated sinus. At MR imaging, there was expansion of the maxilla with obliteration of the maxillary sinuses and protrusion of the mandible. The lesion exhibited low signal intensity at T1-weighed images. At T2-weighted images, the lesion showed low signal intensity with small hyperintense lesions. Dystrophic calcification (n = 2) was seen in the parotid and the check. CONCLUSION: We concluded that CT and MR imaging are helpful for diagnosis and treatment planning of maxillofacial lesions of patients with renal osteodystrophy.