研究者総覧

Carbamazepine is widely used as an anticonvulsant or antipsychotic agent. As a sodium channel blocker, it provides therapeutic benefit in patients with trigeminal neuralgia and may be used for the differential diagnosis of pain. We report a case of marked thrombocytopenia during carbamazepine administration in a patient with idiopathic trigeminal neuralgia. A 37-year-old woman presented with acute intermittent electric shock-like pain when pressure was applied to the left mental foramen, or when cold air was applied to the left lower lip. We observed no sign of dental infection. No obvious vascular contact in intracranial lesions or the trigeminal nerve root entry area was observed in MR imaging. We initiated carbamazepine (200 mg/day) therapy for a clinical diagnosis of idiopathic left trigeminal neuralgia (tertiary branch). The patient developed petechial hemorrhages on the face, neck, and extremities 16 days after carbamazepine administration, and blood test results revealed significant thrombocytopenia (her platelet count was remarkably low, at 1.5×10⁴ /μ L). We suspected carbamazepine-induced thrombocytopenia. The medication was discontinued, resulting in a gradual disappearance of the petechial hemorrhages and recovery of the platelet count to 23.7×10⁴ /μ L six days after carbamazepine cessation. Subsequently the left-sided mental foramen pain was controlled with pregabalin administration (150 mg/day), without any adverse effects.

Fanconi anemia is an inherited genetic disorder characterized by progressive pancytopenia, transition to myelodysplastic syndrome, acute myeloid leukemia, body malformation, and increased risk of certain solid cancers. Among the solid tumors, there is a high incidence of squamous cell carcinoma of the head and neck. Here, we report a case of advanced lower gingival cancer in a patient with Fanconi anemia.

　A 30-year-old man was referred to our hospital because he was aware of a painless mass arising in the right mandibular gingiva. In his medical history, the patient was given a diagnosis of Fanconi anemia at the age of 6 years because of pancytopenia. The patient achieved hematological recovery after allogenic hematopoietic stem cell transplantation following a preparative regimen consisting of total body irradiation and cyclophosphamide. The right mandibular tumor (36×27 mm) was diagnosed as lower gingival cancer. The patient underwent bilateral neck dissection, segmental mandibular resection, and total resection of the lower lip (squamous cell carcinoma, pT4aN2cM0). Reconstructive surgery comprised placement of a reconstructive titanium plate, a pectoralis major musculocutaneous flap, and local rotational flaps.

　Postoperative adjuvant treatment was not administered. 30 months postoperatively, there was no new lesion, recurrence, or metastasis.

Leiomyomatous hamartoma mainly occurs in the liver, spleen, kidney and lung. It is rarely found in the oral region.

　We report a case of leiomyomatous hamartoma arising in the hard palate of a 6-month-old female infant. Six months after her birth, her parents consulted the obstetrician about a tumor of the hard plate and were referred to our department. Although it had been recognized since her birth, she could suck milk without any problems. The results of medical examination revealed that there was no problem in general condition. The tumor was an elastic soft, and cord-like mass that measured about 15 × 3 mm and was localized in the incisive papilla. The primary clinical diagnosis was a benign tumor of the hard palate. An excisional biopsy was carried out in the outpatient clinic by using local anesthesia. Immunohistochemical analysis showed positive reactions for α-smooth muscle actin (α- SMA) and HHF35, which showed the growth of smooth muscle. A histological diagnosis of leiomyomatous hamartoma was established. Postoperative observation has revealed no recurrence as of 13 years after the operation.