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Volume 62, Issue 5
Displaying 1-10 of 10 articles from this issue
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Preface
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[in Japanese]2016 Volume 62 Issue 5 Pages 221
Published: May 20, 2016
Released on J-STAGE: July 20, 2016
JOURNAL FREE ACCESSDownload PDF (616K)
Case reports
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Seirin OH, Haruhiko TERAI, Yoichiro NAKAJIMA, Kimiko TAKUBO, Nozomu FU ...2016 Volume 62 Issue 5 Pages 222-225
Published: May 20, 2016
Released on J-STAGE: July 20, 2016
JOURNAL FREE ACCESSAcquired hemophilia A is a very rare disease caused by the acquisition of factor Vlll inhibitor. We report a case of acquired hemophilia A incidentally diagnosed in a patient who had bleeding after tooth extraction. A 77-year-old man who complained of bleeding after tooth extraction presented at our hospital. Hematological examination revealed a prolonged activated partial thromboplastin time (APTT), decreased coagulation activity of factor Vlll, and high level of factor Vlll inhibitor. Consequently, acquired hemophilia A was diagnosed. The patient received corticosteroids and bypass therapy. Four months later, the APTT and factor Vlll activity returned to the normal level. Although this is a very rare case, we should suspect hematologic disease, including acquired hemophilia A, when we encounter patients with bleeding after tooth extraction.View full abstractDownload PDF (479K) -
Takashi WATANABE, Nobuyoshi TOMOMATSU, Misa HOSOKI, Yoshinori INABA, K ...2016 Volume 62 Issue 5 Pages 226-231
Published: May 20, 2016
Released on J-STAGE: July 20, 2016
JOURNAL FREE ACCESSThe main stigmata of the nevoid basal cell carcinoma syndrome (NBCCS) are developmental abnormalities, including palmar or plantar pits, bifid ribs, calcification of the falx cerebri, and a high susceptibility to several neoplasms, such as basal cell carcinomas of the skin, keratocystic odontogenic tumors (KCOTs) of the jaw, and medulloblastomas. NBCCS is a hereditary disorder caused by mutations in human patched-1 gene (PTCH1) and transmitted in an autosomal dominant manner. Three cases of NBCCS in a maternal relationship are reported. We analyzed the PTCH1 gene in 2 patients (siblings) and detected identical deletion mutations (germline mutations). In patients with NBCCS, the KCOT-causing mutation is usually inherited through the germline. Thus the susceptibility to KCOT is present in every cell of the body. Consequently, KCOT associated with NBCCS shows a trend toward multiplicity. The early detection of KCOT during follow-up using computed tomography and other diagnostic imaging techniques allowed minimally invasive surgical treatment and good outcomes.View full abstractDownload PDF (758K) -
Hideo TANAKA, Hirohito TAKEUCHI, Momotoshi SHIGA, Kenji HASHIMOTO2016 Volume 62 Issue 5 Pages 232-237
Published: May 20, 2016
Released on J-STAGE: July 20, 2016
JOURNAL FREE ACCESSAtresia of the submandibular duct is a rare condition associated with congenitally imperforate duct orifices. Cystic swelling of the oral floor caused by mucous-filled dilation of the submandibular duct is sometimes similar to ranula. We present a case of atresia of the submandibular duct that we diagnosed as congenital ranula at initial presentation. A 5-month-old female infant was referred to our hospital because of a swelling under the tongue, first noted at birth. Intraoral examination showed a translucent, fluctuant cystic mass, 15 mm in diameter, arising in the left side of the mouth floor. In addition, no flow of saliva could be detected. Magnetic resonance imaging revealed a serpentine tube-form structure extending from the oral floor to the submandibular gland, which corresponded to the trajectory of Wharton’s duct. Marsupialization with sialodochoplasty was performed with the patient under general anesthesia. Histological examination showed that the roof of the cystic lesion was lined with ductal epithelium. The postoperative course has been uneventful, with no evidence of recurrence.View full abstractDownload PDF (628K) -
Junko YOSHIZUMI, Wataru KUMAMARU, Yasutaka KUBOTA, Risa YANAI, Tamotsu ...2016 Volume 62 Issue 5 Pages 238-243
Published: May 20, 2016
Released on J-STAGE: July 20, 2016
JOURNAL FREE ACCESSLangerhans’-cell histiocytosis (LCH) is a relatively rare proliferative disorder of Langerhans’ cells with unknown etiology 1～3）. LCH can occur throughout the body, and cases have been reported in the skin, liver, spleen, lung, lymph nodes, and bone. We present a case of LCH in a 1-year-old boy who presented with fever and swelling of the right cheek. Radiographic examinations revealed expansion of the mandibular molar region with destruction of the cortex. Because such findings might mimic a malignant tumor, we made a clinical diagnosis of mandibular tumor. After confirming the pathological diagnosis, chemotherapy was performed according to the protocol of the Japan LCH study group. No recurrence was noted during a 3-year follow-up period. However, in infants, hypoplasia of the teeth can occur as a side effect of chemotherapy or a direct effect of the tumor itself. Long-term follow-up in cooperation with pediatricians and pedodontists is recommended for infants with LCH.View full abstractDownload PDF (736K) -
Michiyo ONISHI, Akihiro MIYAZAKI, Jun-ichi KOBAYASHI, Megumi UEDA, Mak ...2016 Volume 62 Issue 5 Pages 244-249
Published: May 20, 2016
Released on J-STAGE: July 20, 2016
JOURNAL FREE ACCESSSynovitis-acne-pustulosis-hyperostosis-osteitis (SAPHO) syndrome is characterized by sterile osteomyelitis and pustular skin disease. Little is known about its pathogenesis, and no effective treatment methods have been established to date. We report the case of 69-year-old man with chronic sclerosing osteomyelitis of the mandible associated with SAPHO syndrome. After a previous surgical treatment, the patient’s lesion was resistant to antibiotics and hyperbaric oxygen therapy; therefore, teriparatide was administered. After 7 months of teriparatide treatment, the patient’s spontaneous pain disappeared, and computed tomographic and magnetic resonance imaging scans showed improvements in the bone structure and quality of the mandible. These findings suggest that suppression of bone remodeling occurs in SAPHO syndrome, similar to BRONJ.View full abstractDownload PDF (608K) -
Takeshi KUROSHIMA, Takehiko SATO, Masashi TAKANO, Masanobu SHINDOH, Sh ...2016 Volume 62 Issue 5 Pages 250-255
Published: May 20, 2016
Released on J-STAGE: July 20, 2016
JOURNAL FREE ACCESSCentral Mucoepidermoid carcinoma (cMEC) is uncommon entity and occurs commonly in the mandible, but rarely in the maxilla. Furthermore, the occurrence of cMEC in children and adolescents is extremely rare. An 18-year-old male patient was referred to our hospital because of halitosis and fistula of the palate. Radiological examination showed a cystic lesion in the right side of the maxilla with bone destruction. The histopathological diagnosis on biopsy was MEC. Partial maxillectomy was successfully performed. There has been no evidence of recurrence or metastasis as of 3 years after the operation. Real-time RT-PCR assay of paraffin-embedded tissue showed positivity of MEC-specific CRTC1-MAML2 gene fusions, suggesting a good prognosis.View full abstractDownload PDF (732K) -
Aki ABE, Ryosuke ABE, Tomoyuki AOMURA, Masaatsu YAGI, Emi NAKATA, Haru ...2016 Volume 62 Issue 5 Pages 256-261
Published: May 20, 2016
Released on J-STAGE: July 20, 2016
JOURNAL FREE ACCESSSynovial sarcoma (SS) is a malignant soft-tissue tumor that accounts for 5% to 10% of all soft-tissue sarcomas. SS typically arises in joint capsules and articular tendons and is uncommon in the head and neck and oral cavity. We report a case of SS arising in the submucosal region of the lower lip of a 49 year-old woman who visited our division in June 2010 because of a swelling in the right lower lip, developing 1 month previously. We performed a biopsy of the affected region with the patient under local anesthesia, and the pathological diagnosis was chronically inflamed tissue. However, the swelling and spontaneous pain continued for 1 month. The subsequent clinical diagnosis was a salivary gland tumor, and the tumor was resected with the patient under general anesthesia. Histopathological examination of the excised specimen revealed that the tumor consisted mainly of epithelial and spindle-cell components and exhibited the monophasic pattern of SS. An additional excision was performed with the patient under general anesthesia, and complete resection of the SS remnants was performed. The patient remains alive and has not had recurrence or metastasis as of 4 years after surgery.View full abstractDownload PDF (734K) -
Hiroe KAKEHASHI, Yasuhisa KAMIKASEDA, Shintaro KAWANO, Seiji NAKAMURA2016 Volume 62 Issue 5 Pages 262-266
Published: May 20, 2016
Released on J-STAGE: July 20, 2016
JOURNAL FREE ACCESSAcquired hemophilia is a rare disease caused by the acquisition of factor Vlll inhibitor. There is a sudden onset of a critical bleeding episode, and the reported incidence is 1.48 cases per million persons per year. We report a case of acquired hemophilia diagnosed incidentally on postoperative bleeding after tooth extraction. A 74-year-old woman was referred to our hospital because of bleeding after tooth extraction. Hematological examination revealed a prolonged activated partial thromboplastin time (APTT) and decreased coagulation activity of factor Vlll. In addition, the presence of factor Vlll inhibitor was detected. On the other hand, the platelet count (PT) and prothrombin time were normal. The patient was given a diagnosis of acquired hemophilia by a hematologist and was admitted to the hospital on the same day. On hospital day 1, hemostatic therapy and immunotherapy with corticosteroids were begun. On hospital day 23, the APTT returned to the normal range, and symptoms improved. The patient was discharged and is being treated on an outpatient basis in our hospital. Corticosteroids were gradually decreased by concurrently administering a cyclosporine preparation, and the patient’s condition has improved satisfactorily since the initial diagnosis 6 years ago.View full abstractDownload PDF (569K) -
Haruyuki SHINOHARA, Masashi SUGIURA2016 Volume 62 Issue 5 Pages 267-270
Published: May 20, 2016
Released on J-STAGE: July 20, 2016
JOURNAL FREE ACCESSA 69-year-old woman was referred to our department because of trismus. Physical examination showed a normal temporomandibular disorder, and she was prescribed a muscle relaxant and analgesic and went home. However, her symptoms became worse, and she was re-examined 2 days later and had severe trismus, stiffness of the masseter muscles, neck pain, and backache. She had a scab from a minor injury on the left knee. Tetanus was diagnosed on the basis of the clinical course and symptoms. The patient was immediately admitted to the intensive care unit. She was given anti-tetanus human immunoglobulin, tetanus toxoid, and antibiotics. The patient recovered without tracheotomy or tracheal intubation. We isolated Clostridium tetani from the scab on her knee, and tetanus was clinically and biologically diagnosed. Isolation of Clostridium tetani is very rare, and to our knowledge this is a rare case in which Clostridium tetani was isolated from a scab. Even if the wound shows signs of healing, tetanic symptoms do not improve, and such wounds should always be débrided for therapeutic purposes.View full abstractDownload PDF (560K)
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