Oral and Maxillofacial Surgery, Medicine, and Pathology

Intraparotid lymph node toxoplasmosis diagnosis by fine needle aspiration cytology Publication date: Available online 31 May 2019 Source: Journal of Oral and Maxillofacial Surgery, Medicine, and Pathology Author(s): Hélder Domiciano Dantas Martins, Beatriz Araújo Suassuna Vaz, Larissa Suellen da Silva Lins, Brenna Louise Cavalcanti Gondim, Lucio Roberto Cançado Castellano, Paulo Rogério Ferreti Bonan Abstract Toxoplasmosis is a relatively common disease caused by the Toxoplasma gondii parasite. Its clinical manifestations are dependent on the immune status of the patient, and the correct diagnosis is established based on a combination of direct parasite visualization and serological assays for the detection of host antibodies acting against the parasite. This study presents a case of intraparotid lymph node toxoplasmosis diagnosed by fine needle aspiration cytology presenting bradyzoite- and tachyzoite-compatible structures associated with serology assays. After diagnosis, treatment comprised a triple antimicrobial scheme and follow-up for six months, and the patient reached total regression. Thus, it is concluded that FNAC associated to serology assays comprise adequate tools for toxoplasmosis diagnoses.

Orthognathic treatment in Greig cephalopolysyndactyly syndrome: A case report Publication date: Available online 28 May 2019 Source: Journal of Oral and Maxillofacial Surgery, Medicine, and Pathology Author(s): Tayfun Cıvak, Goksu Trakyali, Altan Varol Abstract Typical Greig cephalopolysyndactyly syndrome (GCPS) is characterized by preaxial polydactyly or mixed pre- and postaxial polydactyly, true widely spaced eyes, macrocephaly. Individuals with mild GCPS may have subtle craniofacial findings. The diagnosis of GCPS is based on clinical findings and family history. GLI3 is the only gene known to be associated with GCPS. It has an autosomal dominant inheritance. The literature fails in documentations of any treatment protocol for defective jaw relationship in these patients. Therefore, we report on a patient with GCPS presenting polysyndactyly, frontal bossing, high forehead, skeletal Class III deformity due to maxillary retrognathism and mandibular prognathism, treated with orthognathic surgery by means of double jaw surgery and orthodontic treatment with fixed appliances.

Benign fibro-osseous lesion and odontoma of the mandible: A report of a rare, hybrid lesion and review of literature Publication date: Available online 24 May 2019 Source: Journal of Oral and Maxillofacial Surgery, Medicine, and Pathology Author(s): Rekha Reddy, Rohan Jagtap, Deeba Kashtwari, Christopher C. Hambrook, Danielle L. Freburg-Hoffmeister, Mohammed N. Islam, Donald M. Cohen, Archana Shenoy, Indraneel Bhattacharyya Abstract This report documents a case of a 12-year-old male with a combined benign fibro-osseous lesion (BFOL) and odontoma. Radiographic imaging revealed an extensive, well-defined, multilocular, radiolucent lesion with small radiopaque entities in the left mandible. Microscopic examination revealed a central BFOL with associated odontomas. To the best of the authors' knowledge, this is the third report of a combined BFOL and odontoma and an extensive review of the English language literature on combined BFOL and odontoma cases is included. This case is important because it demonstrates the importance of diagnosing bone lesions with an accompanying radiograph. The initial biopsy was signed out as a BFOL with a comment that there was tooth-like material as well and a possibility that it represented an odontoma. The patient was operated on in our teaching hospital and the lesion completely excised with a diagnosis of dentigerous cyst. The hard tissue was grossed only as it appeared tooth-like. Seeing the final diagnosis, we requested decalcification of the hard tissue. Based on clinical, radiographic and histologic findings, the final diagnosis of BFOL and odontoma was made. Another reason for reporting this case is that the paucity of published cases of these combined lesions makes treatment decisions a challenge. Further future reporting of BFOL arising in association with odontomas with long-term follow-up information is paramount to better understand these entities vis-à-vis their nature and behavior.

End-to-end anastomosis of transected parotid duct in an emergency department: Clinical benefits from immediate repair and overview of treatment options Publication date: Available online 24 May 2019 Source: Journal of Oral and Maxillofacial Surgery, Medicine, and Pathology Author(s): Ioannis Papadiochos, Paschalis Strantzias, Georgios Mitsopoulos, Kalliopi Melaki Abstract Parotid duct injuries (PDIs) are not considered common complications of facial trauma. However, their associated morbidity may be increased by the formation of parotid sieloceles and fistulae. This article reports the case of a 31-year-old male patient who was presented to the emergency department of Evangelismos General Hospital of Athens, due to a sharp penetrating injury on left side of his face. Since transection of PD was clinically diagnosed, an end-to-end anastomosis was immediately carried out under local anesthesia. Within the early postoperative period, various angiocatheters of progressively increased diameters were used for stenting the repaired PD. After 10 months of follow-up, there were no clinical and ultrasonographic signs of sialocele or fistula formation. This case report aimed first to underscore the benefits of immediate surgical management in case of PDIs and second, to describe the postoperative management for maintenance of both PD patency and parotid gland function. PDIs in the context of emergency facial trauma are advisable to be timely recognized and treated immediately, by securing the saliva outflow to the oral cavity and maintaining a wide enough ductal lumen.

A case of a huge mandibular tumor composed of ameloblastoma and high-grade sarcoma Publication date: Available online 23 May 2019 Source: Journal of Oral and Maxillofacial Surgery, Medicine, and Pathology Author(s): Yusei Fujita, Hiromi Tamura, Shusuke Okunaga, Masakazu Hamada, Ken Wakabayashi, Hiroka Ando, Shiro Adachi, Tetsuro Sumi Abstract Ameloblastoma is a benign odontogenic tumor of the jaw. Sarcoma is a rare neoplasm that accounts for only about 1% of all tumors. Here, we report a quite rare case of simultaneous occurrence of an ameloblastoma and a high-grade sarcoma in the mandible of a 65-year-old man. He was referred to our hospital because of his difficulties in eating and drinking due to a remarkable swelling of the right mandible. He had first noticed the swelling about six years before and consulted a clinic one year before, but he had refused any examination or treatment. He underwent an incisional biopsy, and it revealed a high-grade spindle cell sarcoma with massive necrosis. Therefore, we suggested surgery, but he had never hoped any operation. His poor nutritional status gradually further declined, and he died due to massive hemorrhage from the tumor. In the autopsy, the histological findings revealed that the tumor was predominantly composed of high-grade spindle cell sarcoma and contained a small amount of epithelial component which was a granular cell variant of ameloblastoma. Metastatic tumors were seen in the bilateral lungs and the liver. They contained only the sarcomatous element.

Evaluation of therapeutic effects of teriparatide in a rat model of zoledronic acid-induced bisphosphonate-related osteonecrosis Publication date: Available online 23 May 2019 Source: Journal of Oral and Maxillofacial Surgery, Medicine, and Pathology Author(s): H. Ikeda, D. Yoshiga, S. Kokabu, W. Ariyoshi, H. Tsurushima, O. Sakaguchi, J. Tanaka, J. Kaneko, M. Habu, M. Sasaguri, E. Jimi, T. Nishihara, I. Yoshioka, K. Tominaga Abstract Osteonecrosis of the jaw is a complication in patients using bisphosphonate agents. In this study, we aimed to investigate the effects and mechanism of teriparatide (TPTD) in a rat model of bisphosphonate-related osteonecrosis (BRON). Osteonecrosis was induced by administration of zoledronic acid (ZOL). Four weeks after ZOL injection, the rats underwent a surgery in which drilling holes were made. These holes were filled with freeze-dried Aggregatibacter actinomycetemcomitans. After the four-weeks period, TPTD or saline (n = 9) was intermittently administered for four weeks. Later, rats were euthanized, and the mandible and femur bones were examined the amount of necrosis and bone regeneration. Serum receptor activator of NF-κB ligand (RANKL), osteocalcin (OC), and C-terminal crosslinking telopeptide of type I collagen (CTX) were also examined. TPTD administration reduced necrotic bone area of the mandibles and femurs in the BRON rat model and induced new bone formation. In addition, TPTD injection increased the number of osteoclasts. The suggested underlying mechanism is the induction of protein levels of RANKL by TPTD. Furthermore, the serum levels of bone metabolism biomarkers (OC and CTX) were upregulated in the TPTD injection group. In conclusion, ZOL has negative effects on osteoclasts. TPTD was found to be effective in eliminating the negative effects of ZOL. TPTD had positive effects in preventing bone resorption and promoting osteogenesis. In addition, TPTD improved osteoclastogenesis, which in turn led to the improvement of BRON.

A case of large developing odontoma, so-called ameloblastic fibro-odontoma, of the mandible Publication date: Available online 8 May 2019 Source: Journal of Oral and Maxillofacial Surgery, Medicine, and Pathology Author(s): Hideki Watanabe, Hitoshi Osano, Hisao Igarashi, Akihiro Dohi, Yoshiyuki Mori Abstract Large developing odontoma is a rare odontogenic tumor showing proliferation of both odontogenic epithelium and ectomesenchyme, with enamel and dentin formation. A case of large developing odontoma affecting the lower molar region to the mandibular ramus of a 14-year-old girl is reported. Panoramic and computed tomography images showed a clearly demarcated, multilocular radiolucency extending from the left lower molar region to the mandibular ramus, containing a tooth-like radiopacity. With the patient under general anesthesia, the mass was removed. The histopathological diagnosis was developing odontoma. There was no sign of recurrence at 1 year after tumor removal.

Calcium pyrophosphate dihydrate crystal deposition of the temporomandibular joint: A case report and review of the literature Publication date: Available online 8 May 2019 Source: Journal of Oral and Maxillofacial Surgery, Medicine, and Pathology Author(s): Isao Miyamoto, Atsushi Kasamatsu, Kazuya Hiroshima, Nao Ishida, Shin Takeuchi, Toshikazu Takahara, Sho Ishida, Shusaku Yoshimura, Takao Baba, Yasuhiro Saito, Manabu Iyoda, Hideki Tanzawa Abstract Calcium pyrophosphate dihydrate crystal deposition disease (CPPDd) induces arthritis in the surrounding joints with symptoms, such as pain, swelling, and restricted joint movement. CPPDd occurs primarily in the major joints; however, the main manifestation of this disease in the temporomandibular joint (TMJ) is relatively rare. We describe a case of CPPDd in the TMJ of a 54-year-old Japanese woman who was referred to our hospital with the complaints of unpainful swelling in the right TMJ region and restricted mouth opening. Computed tomography (CT) and magnetic resonance imaging (MRI) showed 46 × 42-mm and 27 × 18-mm calcified masses in the right TMJ region. The masses were both outside and inside the mandibular condyle, both of which extended to the skull base. Since we suspected a tumor in the TMJ, the lesion was partially excised for biopsy with the patient under general anesthesia. The microscopic examination showed deposits of calcium pyrophosphate dihydrate crystals, which resulted in a histopathologic diagnosis of CPPDd. According to the suggestion of the neurosurgeon, we removed as much as possible of only the CPPDd tissue on the outside of the mandibular condyle via the preauricular approach to avoid damaging the cranial nerves. The patient was satisfied with the postoperative facial symmetry without limited mouth opening. The postoperative follow-up was uneventful.

Amelioration of limited mouth opening after treatment of primary biliary cholangitis: A case report Publication date: Available online 8 May 2019 Source: Journal of Oral and Maxillofacial Surgery, Medicine, and Pathology Author(s): Takeshi Kaneuji, Daigo Yoshiga, Wataru Ariyoshi, Ikuo Nakamichi, Hironori Tanimoto, Junpei Tanaka, Ikuya Miyamoto, Manabu Habu, Sho Mitsugi, Tatsuji Nishihara, Tetsu Takahashi, Kazuhiro Tominaga, Izumi Yoshioka Abstract Primary biliary cholangitis is a slow-progressing autoimmune disease of the liver characterized by the presence of portal inflammation and immune-mediated destruction of the intrahepatic bile ducts. Herein, we report a case of temporomandibular joint disorder that concurred with primary biliary cholangitis. A 54-year-old woman presented to us with severe pain on the left side of the temporomandibular joint and difficulty in mouth opening. We used pharmacotherapy, occlusal sprint, and arthrocentesis for the treatment; however, there was no improvement. Magnetic resonance imaging revealed malposition of the articular disk on the left side of the joint and joint effusion. Thus, we performed arthroscopic surgery under general anesthesia. Postoperatively, she developed impaired liver function and itchiness of the skin. A liver biopsy was performed by a hepatologist, and a diagnosis of primary biliary cholangitis was made, as suspected. The temporomandibular joint pain and trismus persisted postoperatively; however, these symptoms improved after primary biliary cholangitis therapy. Thus, we suggest a relationship between temporomandibular joint disorder and primary biliary cholangitis from this disease trajectory and suspect that interleukin-17 may play an important role in the pathogenesis of temporomandibular joint disorder.

Severe stomatitis caused by misuse of methotrexate in an elderly patient with chronic rheumatoid arthritis Publication date: Available online 4 May 2019 Source: Journal of Oral and Maxillofacial Surgery, Medicine, and Pathology Author(s): Kohta Yamada, Kei Tomihara, Manabu Yamazaki, Makoto Noguchi Abstract Methotrexate (MTX) is an antifolate that has been used as a chemotherapeutic agent for the treatment of neoplasm, such as leukemia and osteosarcoma. Moreover, MTX is also commonly used for treatment of chronic rheumatoid arthritis (RA). Here, we report a case of severe stomatitis in a patient with RA, which was caused by misuse of MTX. An 83-year-old female patient visited our department with the chief complaint of oral mucosal pain, accompanied by extreme fatigue. Severe stomatitis lesions were observed throughout her oral mucosa. Moreover, blood examination revealed pancytopenia. The patient reported a history of RA, and had been prescribed MTX since 2004. Although 6 mg MTX should be used twice per week, the patient mistakenly used MTX every day following her last consultation, for a period of 1 month. These findings led to a diagnosis of severe stomatitis, pancytopenia, and sepsis, all induced by misuse of MTX. Ultimately, she was treated with blood transfusion, antibiotics, and intravenous hyper alimentation, as well as administration of granulocyte colony-stimulating factor. MTX for the treatment of RA should be carefully managed to limit the risk of its misuse, especially in elderly patients, because of the severity and potential lethality of symptoms.

Alexandros Sfakianakis
Anapafseos 5 . Agios Nikolaos
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alsfakia@gmail.com