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.
{"title":"Large First Branchial Cyst Extending into the Parapharyngeal Space: A Case Report.","authors":"Takeshi Matsunobu, Takaomi Kurioka, Kimihiro Okubo","doi":"10.1272/jnms.JNMS.2025_92-601","DOIUrl":"10.1272/jnms.JNMS.2025_92-601","url":null,"abstract":"<p><p>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.</p>","PeriodicalId":56076,"journal":{"name":"Journal of Nippon Medical School","volume":" ","pages":"430-434"},"PeriodicalIF":1.4,"publicationDate":"2026-01-10","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143702252","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
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.
{"title":"Accurate Diagnosis of Familial Mediterranean Fever Improved Quality of Life for a Patient and Her Family: A Case Report.","authors":"Mami Kurihara, Toru Igarashi, Ryu Ishii, Hanako Tajima, Jun Hayakawa, Makoto Migita","doi":"10.1272/jnms.JNMS.2025_92-610","DOIUrl":"10.1272/jnms.JNMS.2025_92-610","url":null,"abstract":"<p><p>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.</p>","PeriodicalId":56076,"journal":{"name":"Journal of Nippon Medical School","volume":" ","pages":"480-485"},"PeriodicalIF":1.4,"publicationDate":"2026-01-10","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144509602","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
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.
{"title":"Aggravation of Pre-Existing Cervical Spondylotic Myelopathy with Limb Paralysis Following Lumbar Puncture: A Case Report.","authors":"Akiko Yamazaki, Masahiro Mishina, Masanori Sakamaki, Takehiko Nagao, Kazumi Kimura","doi":"10.1272/jnms.JNMS.2025_92-602","DOIUrl":"10.1272/jnms.JNMS.2025_92-602","url":null,"abstract":"<p><p>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.</p>","PeriodicalId":56076,"journal":{"name":"Journal of Nippon Medical School","volume":" ","pages":"435-439"},"PeriodicalIF":1.4,"publicationDate":"2026-01-10","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143702186","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
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.
{"title":"Familial Congenital Ossicular Anomaly: A Case Report.","authors":"Takeshi Matsunobu, Hirotaka Suzuki, Kimihiro Okubo","doi":"10.1272/jnms.JNMS.2025_92-606","DOIUrl":"10.1272/jnms.JNMS.2025_92-606","url":null,"abstract":"<p><p>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.</p>","PeriodicalId":56076,"journal":{"name":"Journal of Nippon Medical School","volume":" ","pages":"457-462"},"PeriodicalIF":1.4,"publicationDate":"2026-01-10","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143702220","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
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.
{"title":"Circulatory Management with Impella Assistance during Off-Pump Coronary Artery Bypass Grafting for Cardiogenic Shock: A Report of Two Cases.","authors":"Makiko Yamamoto, Yuka Ino, Keisuke Mori, Yuji Maruyama, Yosuke Ishii, Masashi Ishikawa, Atsuhiro Sakamoto","doi":"10.1272/jnms.JNMS.2025_92-609","DOIUrl":"10.1272/jnms.JNMS.2025_92-609","url":null,"abstract":"<p><p>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.</p>","PeriodicalId":56076,"journal":{"name":"Journal of Nippon Medical School","volume":" ","pages":"474-479"},"PeriodicalIF":1.4,"publicationDate":"2026-01-10","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144509603","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
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.
{"title":"Microtia with Coexistent Large External Auditory Canal Cholesteatoma: A Case Report.","authors":"Takeshi Matsunobu, Hirotaka Suzuki, Naoko Sakuma, Ryuji Ohashi, Kimihiro Okubo","doi":"10.1272/jnms.JNMS.2025_92-608","DOIUrl":"10.1272/jnms.JNMS.2025_92-608","url":null,"abstract":"<p><p>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.</p>","PeriodicalId":56076,"journal":{"name":"Journal of Nippon Medical School","volume":" ","pages":"468-473"},"PeriodicalIF":1.4,"publicationDate":"2026-01-10","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143702255","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2026-01-10Epub Date: 2025-03-22DOI: 10.1272/jnms.JNMS.2025_92-604
Takeshi Matsunobu, Kimihiro Okubo, Koichiro Saito
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.
{"title":"Vagal Reflex-Induced Asystole during Suspension Laryngoscopy: A Rare Complication.","authors":"Takeshi Matsunobu, Kimihiro Okubo, Koichiro Saito","doi":"10.1272/jnms.JNMS.2025_92-604","DOIUrl":"10.1272/jnms.JNMS.2025_92-604","url":null,"abstract":"<p><p>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.</p>","PeriodicalId":56076,"journal":{"name":"Journal of Nippon Medical School","volume":" ","pages":"446-449"},"PeriodicalIF":1.4,"publicationDate":"2026-01-10","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143702277","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2026-01-10Epub Date: 2025-03-22DOI: 10.1272/jnms.JNMS.2025_92-603
Takeshi Matsunobu, Hirotaka Suzuki, Akira Shimizu, Koji Sakamoto, Kimihiro Okubo
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.
{"title":"Use of Sialendoscopy to Diagnose Sialodochitis Fibrinosa: A Case Report.","authors":"Takeshi Matsunobu, Hirotaka Suzuki, Akira Shimizu, Koji Sakamoto, Kimihiro Okubo","doi":"10.1272/jnms.JNMS.2025_92-603","DOIUrl":"10.1272/jnms.JNMS.2025_92-603","url":null,"abstract":"<p><p>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.</p>","PeriodicalId":56076,"journal":{"name":"Journal of Nippon Medical School","volume":" ","pages":"440-445"},"PeriodicalIF":1.4,"publicationDate":"2026-01-10","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143702257","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
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.
{"title":"Video-Assisted Thoracoscopic Surgery for Catamenial Pneumothorax: A Report of Five Cases Treated by Diaphragm Reefing with and without Chemical Pleurodesis.","authors":"Hirotoshi Kubokura, Yuuya Tomioka, Junichi Okamoto, Naoyuki Yoshino, Jitsuo Usuda","doi":"10.1272/jnms.JNMS.2025_92-607","DOIUrl":"10.1272/jnms.JNMS.2025_92-607","url":null,"abstract":"<p><strong>Background: </strong>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.</p><p><strong>Methods: </strong>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.</p><p><strong>Results: </strong>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).</p><p><strong>Conclusion: </strong>Our procedure (diaphragm reefing by ligation and covering with a polyglycolic acid sheet) was a more convenient and more effective treatment for CP.</p>","PeriodicalId":56076,"journal":{"name":"Journal of Nippon Medical School","volume":" ","pages":"463-467"},"PeriodicalIF":1.4,"publicationDate":"2026-01-10","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143702284","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
A 78-year-old man was diagnosed as having a submucosal gastric mass (diameter 4 cm). Preoperative findings from endoscopic ultrasound-guided fine needle aspiration suggested a diagnosis of gastric neuroendocrine neoplasm. Total gastrectomy with excision of a metastatic liver lesion and dissection of gastric lymph nodes was performed. Analysis of frozen sections indicated adenocarcinoma of the peritoneum, which suggested the possibility of a mixed neuroendocrine-non-neuroendocrine neoplasm (MiNEN), based on the presence of solid tubules, metastatic spread, and lymphovascular invasion. However, the smooth gastric mucosal surface, organoid architecture with rare atypia or necrosis, immunopositivity for neuroendocrine markers, a Ki-67 index of 21%, and the presence of somatostatin receptor 2 expression confirmed a final pathological diagnosis of grade 3 gastric neuroendocrine tumor (NETG3) with glandular formation. NETG3 with glandular formation can be difficult to distinguish from MiNEN because their histological features overlap. However, gastric NETG3 with glandular formation is distinguishable from MiNEN by the presence of a submucosal tumor with a histological organoid pattern without frequent mitoses and/or necrosis, immunopositivity for neuroendocrine markers, and the absence of an adenocarcinoma or neuroendocrine carcinoma component within the tumor, as determined by immunohistochemistry for somatostatin receptor 2 expression, Ki-67, and Rb1.
{"title":"A Case of Grade 3 Gastric Neuroendocrine Tumor with Glandular Formation: Diagnostic Process and Differentiation from Gastric Mixed Neuroendocrine-Non-Neuroendocrine Neoplasm.","authors":"Makiko Kurata, Takuma Tajiri, Masataka Ueda, Chie Inomoto, Tomoko Sugiyama, Takayoshi Suzuki, Hideki Izumi, Junichi Kaneko, Eiji Nomura, Naoya Nakamura, Yohei Masugi","doi":"10.1272/jnms.JNMS.2026_93-502","DOIUrl":"https://doi.org/10.1272/jnms.JNMS.2026_93-502","url":null,"abstract":"<p><p>A 78-year-old man was diagnosed as having a submucosal gastric mass (diameter 4 cm). Preoperative findings from endoscopic ultrasound-guided fine needle aspiration suggested a diagnosis of gastric neuroendocrine neoplasm. Total gastrectomy with excision of a metastatic liver lesion and dissection of gastric lymph nodes was performed. Analysis of frozen sections indicated adenocarcinoma of the peritoneum, which suggested the possibility of a mixed neuroendocrine-non-neuroendocrine neoplasm (MiNEN), based on the presence of solid tubules, metastatic spread, and lymphovascular invasion. However, the smooth gastric mucosal surface, organoid architecture with rare atypia or necrosis, immunopositivity for neuroendocrine markers, a Ki-67 index of 21%, and the presence of somatostatin receptor 2 expression confirmed a final pathological diagnosis of grade 3 gastric neuroendocrine tumor (NETG3) with glandular formation. NETG3 with glandular formation can be difficult to distinguish from MiNEN because their histological features overlap. However, gastric NETG3 with glandular formation is distinguishable from MiNEN by the presence of a submucosal tumor with a histological organoid pattern without frequent mitoses and/or necrosis, immunopositivity for neuroendocrine markers, and the absence of an adenocarcinoma or neuroendocrine carcinoma component within the tumor, as determined by immunohistochemistry for somatostatin receptor 2 expression, Ki-67, and Rb1.</p>","PeriodicalId":56076,"journal":{"name":"Journal of Nippon Medical School","volume":" ","pages":""},"PeriodicalIF":1.4,"publicationDate":"2025-11-17","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145535015","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}