Maxillary Mass Research Articles

Second malignant neoplasm in a treated Ewing sarcoma patient: A case report

Ewing sarcoma (ES) is an aggressive tumor commonly seen in children and young adults. Late effects of ES therapy include the second cancers, a tragic outcome for survivors of such young age. We report a case of a 3-year-old male child who presented with a right maxillary mass extending to the orbit. Fine-needle aspiration cytology and tru-cut biopsy showed features suggestive of a small round cell tumor. Immunohistochemistry confirmed the diagnosis of ES/primitive neuroectodermal tumor. The patient was started on non-metastatic Ewing family tumor protocol with intensity-modulated radiotherapy and responded well to therapy. He was followed up regularly, but after 5 months of initial diagnosis, his peripheral blood smear showed atypical cells resembling blasts. His bone marrow aspiration (BMA) and biopsy showed marrow infiltrated by hematolymphoid malignancy. Based on the positivity of the blasts for myeloperoxidase and non-specific enolase in cytochemistry on the BMA and flow cytometric analysis, a diagnosis of acute myeloid leukemia with monocytoid differentiation was made. We report this case to emphasize the role of the pathologist in differentiating the second malignancies from recurrence of the first malignancy or therapy-related changes, especially in the case of an equivocal morphological picture.

Three-Dimensional Printing-based Reconstruction of a Maxillary Bone Defect in a Dog Following Tumor Removal.

Three-dimensional (3D) printing has been applied extensively not only in human, but also veterinary medicine. However, the technique is still used in the clinical area for a surgical plan or education prior to surgery. Thus, we report a case of reconstruction after tumor removal surgery with the use of a 3D-printed scaffold. A 12-year-old female mixed dog had a left caudal maxillary mass. Based on computed tomography images, a defect was confirmed on the maxillary bone due to the oral mass, and a surgical plan was designed to remove the oral mass and graft the 3D printed scaffold. Customized polycaprolactone/ beta-tracalciumphosphate (PCL/β-TCP) scaffold was fabricated using the micro-extrusion-based 3D printer. In the operation, after the removal of the oral mass, the scaffold was grafted onto the defect site. At follow-up, 8 months after surgery, the result was successful without any special problems in the periodic CT scans and oral examinations. This case is believed to be the first case of reconstruction by using a 3D printed scaffold in the maxillary bone defect, and this 3D printing technique is thought to be very helpful for veterinary patients with bone defects and several other diseases.

안면 비대칭을 동반한 선종양치성종 1예

Adenomatoid odontogenic tumor is an uncommon benign odontogenic tumor, most often associated with an unerupted maxillary canine. The tumor is usually asymptomatic due to its slow growth. However, since facial deformities can result from the tumor, early detection and treatment are necessary. A 9-year-old girl visited our department due to an incidental finding of a maxillary mass during a brain magnetic resonance imaging examination. The right maxilla of the patient was protruded and facial asymmetry had developed. The mass was excised via the canine fossa approach and diagnosed as an adenomatoid odontogenic tumor. We report this rare case along with a literature review. (J Clinical Otolaryngol 2017;28:256-260)

Abstract CT100: First-in-human phase 1 trial of pyrimidine anti-metabolite FF-10502-01 in patients with advanced cancer

Abstract Background: FF10502-01 is a synthetic pyrimidine nucleoside analogue that is structurally similar to gemcitabine with a substitution of sulfur for oxygen in the pentose ring. FF-10502-01 showed potent anti-tumor activity in preclinical studies. In Capan-1 and SUIT-2 pancreatic ca xenograft models, FF-502-01 achieved superior tumor growth suppression and survival, respectively, compared to gemcitabine (gem), with less toxicity at clinically relevant doses. In gem-resistant pancreatic PDX models, FF-10502-01, alone and in combination with nab-paclitaxel (nab-pac), had higher efficacy and tolerability than gem/gem+nab-pac. Methods: We conducted a standard 3+3, dose-escalation phase 1 trial with FF-10502-01 to determine safety, maximum tolerated dose (MTD), pharmacokinetics (PK), pharmacodynamics (PD) i.e. FF-10502 incorporation into peripheral blood cellular DNA, and preliminary antitumor activity. Eligibility criteria included age >18 years, solid tumors refractory to standard treatment and adequate organ function. FF-10502-01 was administered IV over 60 minutes on days 1, 8, and 15 every 4 weeks. Planned dosing cohorts included 8, 12, 18, 27, 40, 60, 90, 135 and 200 mg/m2. Supportive medications such as anti-nausea prophylaxis were allowed. Results: 22 patients (pts) have been treated in 6 dose cohorts of 8-60 mg/m2. The median number of cycles received was 2 (range 1 to >12). Pts with the following cancers were enrolled: pancreatic (5 pts); ovarian and cholangiocarcinoma (3 each); parotid gland, prostate, and sarcoma (2 each); and endometrial, squamous cell carcinoma of the head and neck, anal, colon-neuroendocrine, and unknown primary (1 pt each). 11 pts were male and 11 female, median age 63 years (range 21-80), average number of prior cytotoxic therapies 3 (range 1-7) and 9 pts with prior gemcitabine therapy. Common related adverse events were Gr 1/2 nausea (43%), rash (38%), fever (29%), fatigue (19%), and vomiting despite prophylactic ondansetron and dexamethasone (14%). One pt had Gr 3 nausea (a DLT). Cytopenias have been minimal: 2 pts with Gr 1 and 2 anemia; with no neutropenia or thrombocytopenia. No pts have required dose reduction for toxicity. To date, an MTD has not been identified and enrollment continues. A pt with chondroblastic osteogenic sarcoma (18 mg/m2) achieved an unconfirmed partial response (73% decrease) in a maxillary mass but developed progressive disease at another site. 3 currently active patients (1 each of acinar pancreatic, prostate, parotid gland), demonstrated durable stable disease for > 48, 40 and 28 weeks, respectively, at doses ranging from 8 to 27 mg/m2. FF-10502 plasma concentrations increased with dose. Additional PK and PD data will be presented. Conclusions: The pyrimidine nucleoside antimetabolite FF-10502-01 is well tolerated with prophylactic anti-emetics and demonstrated preliminary antitumor activity in heavily pretreated patients. Citation Format: Gerald Steven Falchook, Lindsay Bramwell, Lori Hannan, Deeksha Vishwamitra, Takayuki Yamada, Michele Rosner, David Wages, Thomas Myers, Linda Paradiso, Filip Janku. First-in-human phase 1 trial of pyrimidine anti-metabolite FF-10502-01 in patients with advanced cancer [abstract]. In: Proceedings of the American Association for Cancer Research Annual Meeting 2017; 2017 Apr 1-5; Washington, DC. Philadelphia (PA): AACR; Cancer Res 2017;77(13 Suppl):Abstract nr CT100. doi:10.1158/1538-7445.AM2017-CT100

Myofibroma in Infancy and Childhood.

Myofibromas are rare fibroblastic-myofibroblastic tumors in children. Their biological behavior is unpredictable and spontaneous regressions have been described. This is a retrospective review of clinical characteristics, treatment, and outcome of children diagnosed with myofibroma between 1999 and 2013 at Texas Children's Hospital. The median age at diagnosis of 42 patients was 37 months. Approximately two thirds of the patients were male. The median length of follow-up was 50.5 months (range, 0 to 165 mo). Thirty-eight patients (90%) had solitary lesions; 19 (50%) in the head and neck, 10 (26%) in the limbs, and 9 (24%) in the trunk. Twelve patients underwent a complete surgical resection. Of the 30 patients with positive margins, only 1 had tumor progression. Two patients had multicentric involvement, and 2 patients had generalized disease with visceral involvement. One patient with generalized disease and a progressive maxillary sinus mass was treated with vinblastine and methotrexate chemotherapy followed by complete surgical resection. All patients were alive at last follow-up. Myofibromas of childhood demonstrate clinical variability, and may spontaneously regress. Positive surgical margins do not adversely affect outcome. The rare patient with progressive unresectable disease may benefit from chemotherapy.

A rare case of aggressive osteoblastoma in maxilla

AbstractOsteoblastoma is a benign osteogenic bone tumour. Despite having a benign character, a small subgroup referred to as aggressive or epithelioid osteoblastoma can exhibit locally aggressive behaviour. Herein, we report a 45‐year‐old man who presented with a rapidly growing palatal and maxillary mass that resulted in difficulty with respiration and oral feeding. Following radiological evaluation and incisional biopsy based on the prediagnosis of aggressive osteoblastoma, total maxillectomy was performed. Histopathological examination revealed an expansile bone‐forming tumour without any atypical mitosis, necrosis, pleomorphism or hyperchromasia and it was interpreted as aggressive osteoblastoma. The differential diagnosis includes low‐grade osteoblastoma‐like osteosarcoma, and there is an ongoing debate about the proper classification and diagnosis. Proper treatment should include surgical excision of the lesion, reconstruction of the surgical site and close follow‐up for possible recurrence. This case is a well‐demonstrated example of multidisciplinary approach to diagnosis and treatment of a rare and challenging phenomenon.

Mesenchymal Chondrosarcoma of Maxilla: A Rare Case Report

Chondrosarcoma (CS) is a malignant tumor of long and flat bone characterized by the formation of cartilage. Mesenchymal chondrosarcoma is a rare variety of chondrosarcoma .It is a biphasic tumor with areas of spindle cell mesenchyme along with chondroid differentiation in the connective tissue stroma. A 17 year old male presented to us as a painless mass in maxilla. Contrast enhanced computed tomography (CECT) showed a lytic expansile lesion in the right maxillary bone with foci of calcification within soft tissue lesion. Fine needle aspiration cytology (FNAC) and incisional biopsy was performed which confirmed the diagnosis of maxillary Mesenchymal chondrosarcoma .The patient underwent right subtotal maxillectomy with 2 cm margins. The review of literature shows that very lesser number of maxillary Mesenchymal chondrosarcoma cases were reported so far. Therefore an attempt is made to add this rare case of MC of maxillary alveolus in the Existing literature.

A rare cause of maxillary mass: juvenile ossifying fibroma

<p class="abstract">Juvenile ossifying fibroma (JOF) is a unique fibro-osseous neoplasm. It has 2 histopathological variants. Psammomatoid juvenile ossifying fibroma (PsJOF) and Trabecular juvenile ossifying fibroma (TrJOF) affecting the jaws of children. Only 20% of the patients are over 15 years of age. JOF is more common in maxilla than mandible. It presents as an asymptomatic progressive, rapid expansion of jaws. It has a recurrence rate of 30-58%. Surgery is the only cure for this kind of lesion and total excision should be the goal of the treatment. We present a case report of 12 year-old female patient with clinical, radiographic and histopathological features of Psammomatoid JOF which clinically admitted to our clinic as maxillary sinus mass.</p>

Organized Hematoma of the Maxillary Sinus: Rapid Progression and Complications

An organized hematoma of the maxillary sinus (OHMS), also known as a pseudotumor, hematoma, hematoma-like mass, and organizing hematoma, presents as an enlarging maxillary sinus mass which is associated with local bone destruction and which mimics a malignancy. However, it has been reported previously that cases of OHMS have benign clinical manifestations and do not recur or present with further complications after complete surgical evacuation. Herein, we report a case of OHMS which was complicated by facial and orbital abscesses. The OHMS showed marked enlargement and extensive bone destruction that had occurred during an 8-month period. We believe that this case will facilitate an understanding of the rapid progression of OHMS, as well as outline the risks posed by the condition. Case

Unusual Case of a Proptosed Eye: Isolated Right Maxillary Neurofibroma.

Neurofibroma is a slow growing benign tumour of the peripheral nerve sheath which is frequently associated with neurofibromatosis type 1 (Prakash et al., 2014). Isolated solitary occurrence of neurofibroma in the maxillary sinus is rare with only 29 reported cases in the literature. We present a rare case of a 70-year-old gentleman who was referred to ENT with a right maxillary sinus neurofibroma with extension into the right inferior orbit. He has significant proptosis, ptosis, and limitation in abduction of the right eye. He has a complicated past history of multiple neurofibromas which were completely excised. Craniofacial MRI shows a large mass filling the right maxillary antrum extending anteriorly into subcutaneous tissue towards nasal ala and posterolaterally into inferior temporal fossa and superiorly into orbit and cavernous sinus involvement. Biopsy of the right maxillary mass revealed cellular spindle cell tumour with wavy collagen bundles within myxoid stroma which is consistent with a neurofibroma. Patient's case was discussed in the skull-base MDT and he has been referred to a specialist center for surgical removal of the neurofibroma with reconstructive surgery. Despite the rarity of this disease, otorhinolaryngologist should consider a possibility of neurofibroma of the paranasal sinuses.

Metastasizing Ameloblastoma

A 27-year-old man presented with an 8 cm diameter left maxillary mass and an enlarged cervical lymph node at levels II to III. There was a reported history of a previous unspecified operation on the maxillary mass which had yielded a diagnosis of ameloblastoma. Total maxillectomy with modified radical neck dissection was subsequently performed.
 Microscopic examination of the maxillary mass shows epithelial islands and cords in a fibro-collagenous stroma. (Figure 1) The islands and cords are lined in the periphery by palisaded columnar cells with regular ovoid nuclei exhibiting reverse polarization. The nuclei are uniform with dispersed chromatin and no significant atypia. Towards the center of these islands are loosely arranged spindly to stellate cells (“stellate reticulum”). (Figure 2) Microscopic examination of the largest submitted lymph node shows an epithelial neoplasm with identical histologic features as the maxillary mass and a residual rim of lymphoid tissue at the periphery enclosed by the nodal capsule. (Figure 3) Similarly, there is neither atypia nor pleomorphism and only a few typical mitotic figures are seen in the nodal tumor. (Figure 4) These features support the diagnosis of a metastasizing ameloblastoma (MA).
 
 Metastasizing ameloblastoma is rare with only about 70 reported cases.1-5 Both the gnathic primary tumor and the metastatic foci have typical morphologies of a benign ameloblastoma with bland nuclei and absent to rare mitosis.6,7 There are no morphologic criteria that can predict this metastatic behavior. Thus, this diagnosis can only be made in retrospect upon the appearance of metastasis in a gnathic tumor that would otherwise have been diagnosed as a usual benign ameloblastoma.1-7 Suggested risk factors include rapid tumor growth, large primary tumor, delay in treatment, mandibular site, prior radiotherapy and chemotherapy.4,5 However, what is most consistent in the literature is the history of multiple recurrences and multiple surgical interventions.1,4,5
 
 The differential diagnosis is an ameloblastic carcinoma (AC). Both MA and AC are considered as the malignant counterparts of ameloblastoma.7 However AC is characterized by the presence of the usual cytologic features of a malignant neoplasm, such as nuclear pleomorphism, hyperchromasia and brisk mitotic activilty – features that are lacking in MA.6,7
 
 The literature on metastasizing ameloblastoma lists the lungs (70 to 88% of cases) and the cervical lymph nodes (15-37.8% of cases) as the most common sites of metastasis.1-5 Increased Ki-67 labeling and CD10 immunoreactivity have been reported to have significant correlation with recurrence.8,9 Whether these observations also apply to the risk of metastasis is not known due to the rarity of cases and hence the small subject populations of these studies. 
 
 As the risk of metastasis cannot be predicted by morphology, long-term follow-up appears prudent in all cases of ameloblastoma, especially if characterized by recurrences and prior surgical interventions.

Burkitt's Lymphoma of Right Maxillary Sinus: Case Report

Introduction: Burkitt’s lymphoma (BL) is an endemic malignant neoplasm with a mandibular localisation, first described, in 1958, in African children. In paranasal BL, maxillary sinus is most commonly involved and spheno-ethmoidal sinuses are less commonly involved. The aim of this study, we describe here a rare case of BL in the right maxillary sinus. Case Report: A 23-year-old female presented with a history of right nasal obstruction after pregnancy. In maxillary CT scan a hetrogenous large solid mass showed in the right maxillary sinus which expands this sinus and extends into the right nasal cavity and right ethmoid. She underwent surgical biopsy. In pathology of right maxillary sinus mass biopsy consistent with high grad diffuse small non cleaved NHL. She treated with combination chemotherapy regimen of CODOX-MVAC with triple intrathecal four courses suggested for her. Then she treated with maintenance therapy with thioguanin and MTX and now she is alive for 3 years after last treatment without any new drug and off all treatment and only is in follow up. Conclusion: BL in the right maxillary sinus is very rare but we will have a more successful treatment if it is diagnosed early.

Primary Hyperparathyroidism Misdiagnosed For Giant-Cell Bone Tumor Recognized Following Surgical Resection of a Right (RT) Maxillary Sinus Mass: A Case Report

: Primary hyperparathyroidism is an endocrine disorder recognized by hyperfunction of parathyroid gland, which can result in persistent bone absorption and brown tumor. Facial involvement of brown tumor is rare and usually involves the mandible. Giant cell tumor ( GCT) is an expansile osteolytic bone tumor which is very similar in clinical, radiological and histological features to brown tumor. Herein, we present a 35-year-old woman with an 11-month history of gradually swelling of the right maxilla and buccal spaces began during pregnancy two years ago. No other clinical or laboratory problems were detected. Postpartum CT scan demonstrated a lytic expansile multi-septated mass lesion containing enhancing areas, which initially described as GCT of the right maxillary sinus following surgery. Four months later, gradual progressive swelling of the bed of tumor was recurred and revised pathological slices were compatible with GCT. Regarding patient recent paresthesia, repeated laboratory tests were performed. Finally, according to laboratory results (elevation of serum calcium and parathyroid hormone), ultrasonographic findings and radioisotope scan (Sestamibi), probable parathyroid mass and brown tumor of maxilla was diagnosed. Pathology confirmed hyperplasia of right inferior parathyroid gland. Our case was thought-provoking due to its interesting clinical presentation and unusual presentation of brown tumor in parathyroid hyperplasia.

Compound Odontoma of the Maxillary Sinus

Compound Odontoma of the Maxillary Sinus

Clinical Pathologic Conference Case 6: Proliferative Maxillary Mass

A 77-year-old male presented with an asymptomatic, exophytic mass of the left posterior maxilla. The patient had first noticed the mass 3 months before presentation. His medical history was significant for benign prostatic hyperplasia and cataracts. Medications included lisinopril, hydrochlorothiazide, nepafenac, and tamsulosin. The patient had been smoking at least one cigar a day for the past 40 years. Extraorally, the patient's left face showed fullness and asymmetry, which extended posteriorly to the cheek.

Treatment of a maxillary fibrosarcoma in an adult alpaca

An approximately 5-year-old sexually intact male alpaca was evaluated because of a right-sided maxillary mass that had recurred after previous surgical debulking. Clinical, radiographic, and CT examination revealed an approximately 1.5-cm-diameter soft tissue mass associated with expansile osteolysis of the maxillary alveolar bone, beginning at the level of the right maxillary third premolar tooth extending caudally to the level of the rostral roots of the second molar tooth. Right partial maxillectomy was performed, and histologic examination revealed an incompletely excised fibrosarcoma with osseous metaplasia. External beam radiation therapy to the tumor bed was initiated 1 month after surgery. Computerized planning was performed, and a total radiation dose of 48 Gy was prescribed in eleven 4.4-Gy fractions. Follow-up CT evaluations 6 and 58 weeks after radiation therapy was completed revealed no evidence of tumor recurrence. No clinical evidence of tumor recurrence was detected through 110 weeks after radiation therapy. The oral fibrosarcoma in the alpaca described here was successfully treated with surgical excision and adjuvant radiation therapy, resulting in excellent quality of life of the treated animal.

Peripheral Odontogenic Fibroma as a diffuse mass in Maxilla: A Case Report

Peripheral odontogenic tumor (POdF) is one of the rare forms of benign gingival enlargements. Odontogenic fibroma occurs as central or as peripheral lesion. WHO classification presents detailed explanation of central odontogenic fibroma and briefly states that peripheral odontogenic fibroma is extra-osseous counter part of central odontogenic fibroma. Thus, most of the POdF is described in literature presented as single, focal swellings. Hence POdF may be mistaken for more common exophytic gingival lesions. In this article we report a rare case of POdF of the maxilla with clinical, radiological and histological features.

A Case of Maxillary Sinus Cholesteatoma Originating from the Retromaxillary Sinus Wall

Cholesteatomas are often observed in the middle ear or mastoid cavity. However, cholesteatomas in areas other than the middle ear or mastoid are extremely rare and thus are not often reported in the literature. We recently treated an 18-year-old female patient with an incidentally detected maxillary sinus mass. A paranasal sinus computed tomography examination revealed a cystic lesion in the left maxillary sinus infiltrating the posterolateral walls of the left maxillary sinus. An endoscopic operation was performed for definitive diagnosis. Histopathological examination of the mass confirmed the diagnosis of cholesteatoma. Despite the rarity of paranasal cholesteatoma in comparison with cholesteatomas in the tympanum, a few cases involving the frontal sinus have been reported. However, retromaxillary cholesteatoma is even less common than sinus cholesteatoma and is reported rarely. To our knowledge, this is the first reported case of a maxillary cholesteatoma originating from the retromaxillary area. For this reason, we present this case along with a literature review.

2. Double hit mantle cell lymphoma with blastoid morphology: a case report

About 40% of all B-cell lymphomas are characterized by the presence of recurrent reciprocal chromosomal translocations. The term ‘double-hit' lymphoma is mostly used for mature B cell lymphomas with a chromosomal breakpoint affecting the MYC locus along with another recurrent chromosomal breakpoint. These lymphomas are aggressive and usually show high proliferation rate. Mantle cell lymphoma is a mature B-cell lymphoma characterized by t(11;14) resulting in cyclin D1 over expression. Although rare, a MYC and cyclin D1 double-hit phenomenon can occur in mantle cell lymphoma and is associated with an adverse clinical course and blastoid morphology. We report a case of a 58 years old female with a left maxillary mass histologically showing features of blastoid mantle cell lymphoma. Fluorescence in situ hybridization (FISH) was positive for CCND1 and MYC gene rearrangements. Recognising ‘double-hit' lymphomas, through use of appropriate investigations is important for optimal management of patients with these highly aggressive tumours.

Tumeur maxillaire révélant une hyperparathyroïdie primaire

Primary hyperparathyroidism can infrequently present as a giant cell tumor of the jaw. We report the case of a patient presenting with a giant cell tumor leading to the discovery of a parathyroid adenoma. A 70-year-old male patient consulted for a stiffly swollen right jaw. The radiological assessment revealed osteolytic lesions of the right maxilla. The calcium and phosphate levels were disrupted and the parathyroid hormone level was high. A neck ultrasonography revealed a parathyroid adenoma. The patient underwent excision of the maxillary mass and of the parathyroid adenoma; the outcome was marked by normalization of calcium and phosphate levels. Giant cell tumors unusually reveal primary hyperparathyroidism and their maxillary location is exceptionally rare. The diagnosis is made on parathyroid hormone level and imaging. The treatment is surgical.