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Takeshi Matsunobu, Takaomi Kurioka, Kimihiro Okubo
2025Volume 92Issue 6 Pages
430-434
Published: December 20, 2025
Released on J-STAGE: January 10, 2026
Advance online publication: March 22, 2025
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Cystic diseases of the salivary gland include mucous cysts and plunging ranula; cysts in the parotid region are rare. In this report, we describe a case of a first branchial cyst in the parotid region. The cyst extended into the parapharyngeal space and was repeatedly infected. The patient was a 35-year-old woman who presented to our hospital with a mass on the left lower ear. Imaging findings revealed a cystic lesion in the parotid region that extended to the parapharyngeal space. A yellowish slurry was aspirated on a percutaneous fine-needle aspiration biopsy. Cytology revealed a class II tumor. The patient initially showed signs of infection and was treated with intravenous antimicrobial agents. After the infectious inflammation had resolved, surgery was performed to resect the cyst. The infection did not recur postoperatively. A large first branchial cyst extending into the parapharyngeal space, which communicates with Stensen's duct, is rare. Care must be taken during surgery because of the complicated positional relationship between the first branchial cyst, parotid gland, and facial nerve.
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Akiko Yamazaki, Masahiro Mishina, Masanori Sakamaki, Takehiko Nagao, K ...
2025Volume 92Issue 6 Pages
435-439
Published: December 20, 2025
Released on J-STAGE: January 10, 2026
Advance online publication: March 22, 2025
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This case report highlights a rare occurrence of paraplegia following lumbar puncture (LP) in the presence of cervical disc herniation during cerebrospinal fluid (CSF) drainage. The patient was an elderly woman undergoing investigation for suspected neoplastic meningitis who experienced sudden acute paraplegia on the day following the LP procedure. LP is often contraindicated in patients with intracranial lesions. If patients exhibit myelopathy symptoms, such as increased lower extremity reflexes and positive Hoffmann and Trömner's signs without intracranial lesions, a thorough evaluation for occupying lesions, including cervical spondylotic myelopathy, is recommended before LP is carried out.
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Takeshi Matsunobu, Hirotaka Suzuki, Akira Shimizu, Koji Sakamoto, Kimi ...
2025Volume 92Issue 6 Pages
440-445
Published: December 20, 2025
Released on J-STAGE: January 10, 2026
Advance online publication: March 22, 2025
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First reported by Kussmaul in 1879, sialodochitis fibrinosa is a disease of the parotid and/or submandibular glands that is characterized by pain and diffuse swelling. It is believed to be caused by obstruction of the salivary duct by mucofibrinous plugs and is often misdiagnosed as other types of sialadenitis, such as Sjögren's syndrome, or sialadenitis caused by bacterial or viral infection. Although specific diagnostic criteria have not been defined, the typical pathological finding of sialodochitis fibrinosa is infiltration of lymphocytes and eosinophils into the interstitium around the salivary ducts. Sialendoscopy, a minimally invasive endoscopic technique, has recently been used to diagnose and treat diseases of the salivary glands. This new technique can be used to irrigate and dilate the salivary gland ducts under direct vision. We successfully used sialendoscopy to diagnose and treat sialodochitis fibrinosa, making this the first reported use of this technique for salivary gland biopsies. Sialendoscopy is a modern approach to salivary gland disorders that was helpful for understanding, diagnosing, and managing sialodochitis fibrinosa.
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Takeshi Matsunobu, Kimihiro Okubo, Koichiro Saito
2025Volume 92Issue 6 Pages
446-449
Published: December 20, 2025
Released on J-STAGE: January 10, 2026
Advance online publication: March 22, 2025
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Suspension laryngoscopy is commonly used in diagnostic and operative procedures involving the larynx. This study presents a rare case of asystole during suspension laryngoscopy. A 58-year-old woman with an unremarkable medical history underwent suspension laryngoscopy for a left polypoid vocal cord under general anesthesia. Anesthesia induction and tracheal intubation were performed without complications. However, upon inserting a direct laryngoscope and exposing the larynx, her heart rate gradually decreased and asystole occurred. The direct laryngoscope was immediately removed, and atropine sulfate was injected. Spontaneous circulation was restored after 20 s of asystole. The direct laryngoscope was then re-inserted, and the operation was successfully completed without complications. Asystole was attributed to stimulation of the pharyngolaryngeal mucosa through direct laryngoscope-induced vagal reflex. This case highlights the need for surgeons and anesthesiologists to be aware of this rare but potentially life-threatening complication and emphasizes the importance of vigilant electrocardiography monitoring during direct laryngoscope adjustment.
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Po-Jen Hsu, Chih-Hao Chen, Shinn-Kuang Lin
2025Volume 92Issue 6 Pages
450-456
Published: December 20, 2025
Released on J-STAGE: January 10, 2026
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Cerebral venous thrombosis is common in individuals using contraceptives. However, the potential existence of an underlying occult prothrombotic condition is frequently overlooked. We report a case of a 46-year-old woman who experienced acute cerebral venous thrombosis, primarily affecting the deep cerebral veins, after a month of contraceptive use. The distinctive "unicorn" pattern observed in the attenuated vein signs on the sagittal plane of non-contrast brain computed tomography served as crucial indicators for diagnosing deep cerebral vein thrombosis. The patient's platelet counts slightly increased during initial admission. However, a gradual increase in the platelet count coupled with the presence of the JAK2 V617F mutation over the subsequent 2 years led to the diagnosis of essential thrombocythemia. After the initiation of cytoreductive treatment, the platelet counts rapidly decreased to the normal range. Myeloproliferative neoplasms (MPNs) should be considered in individuals with cerebral venous thrombosis. The JAK2 V617F mutation is a valuable target for MPNs screening. Clinicians can choose a more appropriate course of treatment and lower the risk of recurrent thrombosis if they promptly detect other prothrombotic states in the patient.
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Takeshi Matsunobu, Hirotaka Suzuki, Kimihiro Okubo
2025Volume 92Issue 6 Pages
457-462
Published: December 20, 2025
Released on J-STAGE: January 10, 2026
Advance online publication: March 22, 2025
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Middle ear anomalies are uncommon in persons with intact auricles and external auditory canals. Most reported cases have been sporadic, and only a few were inherited. Every anomaly can be explained by embryonic ear development. Here, we report a case of bilateral congenital ossicular anomalies in a 7-year-old girl without anomalies of the external ear canal or tympanum. Her mother and two maternal aunts had the same congenital incudostapedial disconnection. A school examination revealed bilateral hearing loss (53.3 dB in the right ear and 51.7 dB in the left ear) indicative of bilateral conductive hearing loss. Her mother and two maternal aunts also had bilateral conductive hearing loss. Surgery on her left ear revealed the absence of the long limbs of the incus and incudostapedial disconnection. An interposition was performed between the crura of the stapes, the handle of the malleus, and the body of the incus. Postoperatively, hearing levels improved in both ears. Although the heredity pattern is unclear, we identified four individuals in the same family with the same bilateral anomalies, suggesting a hereditary origin.
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Hirotoshi Kubokura, Yuuya Tomioka, Junichi Okamoto, Naoyuki Yoshino, J ...
2025Volume 92Issue 6 Pages
463-467
Published: December 20, 2025
Released on J-STAGE: January 10, 2026
Advance online publication: March 22, 2025
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Background: Catamenial pneumothorax (CP), the most common manifestation of thoracic endometriosis syndrome, is a rare form of primary spontaneous pneumothorax that occurs in women of reproductive age. Although CP is usually treated surgically or with hormonal therapy, there is no standard treatment and recurrence is common. We performed video-assisted thoracoscopic surgery (VATS) for five patients with CP from 2014 to 2023. Methods: Our surgical basic procedure (BP) was as follows. VATS was performed by using one port site for the camera and a 5-cm mini-thoracotomy. Perforated holes or blueberry spots were detected on the diaphragm, and lesions were ligated with an endoscopic loop. After reefing by ligation, the diaphragm was covered with a polyglycolic acid sheet. Three methods were used: (A) BP only (one case); (B) BP with tetracycline plus OK432 (two cases); (C) BP with tetracycline (two cases); because the use of OK432 (an anti-cancer agent) was disallowed in 2019 in our Institutional Review Board rules for patients with benign disease. Results: Age ranged from 33 to 45 years (mean, 38.6±5.1 years), and CP occurred on the right side in all cases. Recurrence occurred in only two of the five cases (one case each for procedures B and C). Conclusion: Our procedure (diaphragm reefing by ligation and covering with a polyglycolic acid sheet) was a more convenient and more effective treatment for CP.
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Takeshi Matsunobu, Hirotaka Suzuki, Naoko Sakuma, Ryuji Ohashi, Kimihi ...
2025Volume 92Issue 6 Pages
468-473
Published: December 20, 2025
Released on J-STAGE: January 10, 2026
Advance online publication: March 22, 2025
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Microtia is a congenital disorder characterized by an anomaly in the auricle. It is often associated with atresia, stenosis, or obstruction of the external auditory canal. In cases of microtia, the incidence of a normal external auditory canal, congenital external auditory canal stenosis, and congenital complete closure of the external auditory canal (aural atresia) is reported to be 8%, 8%, and 84%, respectively. Congenital stenosis of the external auditory canal can sometimes be accompanied by intractable otorrhea and otalgia, raising the possibility of complications of external auditory canal cholesteatoma. Here, we report a case of an adult with microtia and external auditory canal stenosis who presented to our clinic with complaints of otalgia. A large external auditory canal cholesteatoma was found in the patient's left ear. Although cholesteatoma is common in cases of canal stenosis, its extensive spread within the temporal bone is quite rare. A temporal bone-targeted computed tomography scan revealed a soft tissue shadow in the left external auditory canal with distensible expansion and bony destruction in the upper, anterior, and posterior walls of the external auditory canal. In patients with microtia who experience severe aural pain, the possibility of latent extended cholesteatoma should be considered.
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Makiko Yamamoto, Yuka Ino, Keisuke Mori, Yuji Maruyama, Yosuke Ishii, ...
2025Volume 92Issue 6 Pages
474-479
Published: December 20, 2025
Released on J-STAGE: January 10, 2026
Advance online publication: June 26, 2025
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The combination of initial Impella therapy, Impella-supported coronary artery bypass grafting (ISCAB), and postoperative Impella therapy providing antegrade perfusion in myocardial infarction can prove effective. We investigated strategies for Impella stabilization in ISCAB, particularly during peripheral circumflex branch anastomosis. Case 1 was a 70-year-old man treated with an Impella 2.5, followed by urgent ISCAB on the day of hospitalization, for a left main trunk lesion. Use of an apical suction device to position the heart to expose an obtuse marginal branch caused Impella obstruction by applying suction to the left ventricular wall, interrupting revascularization; however, one revascularization was achieved. Case 2 was a 79-year-old man treated with an Impella CP for a three-vessel lesion until ISCAB 4 days later. The Impella was stabilized with appropriate positioning by adjusting the bed angle, minimal heart compression with a deep pericardial stitch without pulling on the cardiac apex, and sufficient preload, even during posterolateral branch anastomosis. Four revascularizations were eventually achieved. Multiple innovations to prevent Impella contact with the left ventricle as described herein improve ISCAB safety, particularly during peripheral circumflex branch anastomosis.
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Mami Kurihara, Toru Igarashi, Ryu Ishii, Hanako Tajima, Jun Hayakawa, ...
2025Volume 92Issue 6 Pages
480-485
Published: December 20, 2025
Released on J-STAGE: January 10, 2026
Advance online publication: June 26, 2025
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Periodic fever syndrome is diagnosed on the basis of duration of fever, associated symptoms, and blood and genetic test results. During a 6-month period, a 3-year-old girl experienced monthly fever (39°C) episodes persisting 5 to 12 days and two episodes of ankle arthralgia and skin rash during fever. No abdominal or chest pain was noted. Blood tests performed at the time of fever revealed elevated CRP levels and blood sedimentation rates. Urinalysis and bone marrow examination results were unremarkable. Genetic testing for hereditary autoinflammatory disease syndromes showed E84K MEFV gene mutations. After diagnosing atypical familial Mediterranean fever, oral colchicine alleviated the febrile attacks and improved family quality of life. Fever, arthralgia, and skin rash disappeared after oral colchicine, which is effective for atypical familial Mediterranean fever. During the 6 months before the diagnosis, periodic fever disrupted the daily lives of the entire family. The patient was absent from nursery school during fever. The patient's illness affected her mother's health. The mother was concerned about her child's unknown fever, which resulted in depression and the need for psychiatric medication. Colchicine resolved the febrile episodes, and resolution of the patient's symptoms alleviated her mother's depression. This case shows that genetic testing for periodic fever syndrome can improve family quality of life.
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Noriko Kubota, Kenji Nakamoto, Fumiki Okamoto
2025Volume 92Issue 6 Pages
486-491
Published: December 20, 2025
Released on J-STAGE: January 10, 2026
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Peripapillary hyperreflective ovoid mass-like structures (PHOMS) are a novel, incompletely understood finding in several optic nerve disorders. We describe PHOMS in two Japanese men. Both underwent general ophthalmologic examination as well as multimodal imaging, including visual field testing, fundus photography, spectral-domain optical coherence tomography (SD-OCT), OCT angiography (OCTA), B-scan ultrasonography, fundus autofluorescence (FAF) imaging, and fluorescence angiography. Case 1 was a 31-year-old man with normal pupillary reactions and normal visual acuity in both eyes. SD-OCT revealed PHOMS-oval hyperintense lesions-around the right optic disc. OCTA showed a vascular complex in the PHOMS, and FAF showed a blurry area of hypoautofluorescence at the optic disc. Case 2 was a 61-year-old man who, after undergoing several examinations, was diagnosed as having primary open angle glaucoma with a tilted disc. During a comprehensive examination, a color change was detected at the nasal side of the optic disc in his left eye; SD-OCT revealed PHOMS. One of these two Japanese patients with PHOMS had no other ophthalmic conditions, while the other had glaucoma with a tilted optic disc. Although SD-OCT and OCTA were essential in diagnosing PHOMS, multimodal imaging is required in order to rule out other disorders.
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