-
Sae Aratani, Takeshi Matsunobu, Masashi Nakaishi, Akira Shimizu, Tetsu ...
Article ID: JNMS.2025_92-505
Published: 2025
Advance online publication: March 22, 2025
JOURNAL
FREE ACCESS
ADVANCE PUBLICATION
Immune checkpoint inhibitors (ICIs) have revolutionized the treatment of many cancers, including cancers of the head and neck. Despite the promising therapeutic efficacy of ICIs, immune-related adverse events (irAEs) are a major concern. Acute tubular injury and interstitial nephritis are the most common irAEs involving the kidneys. The present patient was diagnosed as having advanced papillary squamous cell carcinoma of the head and neck. After failure of the initial treatments, including chemotherapy, nivolumab (programmed death-1 inhibitor) was introduced. Shortly after initial administration of nivolumab, the patient developed acute kidney injury with hematuria and proteinuria. A renal biopsy and his clinical course indicated a diagnosis of ICI-related IgA nephropathy. Although glomerular involvement in irAEs is rare and challenging to treatment, the present patient was successfully treated with steroids, which improved kidney function and led to complete remission, as confirmed by urinalysis.
View full abstract
-
Takeshi Matsunobu, Takaomi Kurioka, Kimihiro Okubo
Article ID: JNMS.2025_92-601
Published: 2025
Advance online publication: March 22, 2025
JOURNAL
FREE ACCESS
ADVANCE PUBLICATION
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.
View full abstract
-
Akiko Yamazaki, Masahiro Mishina, Masanori Sakamaki, Takehiko Nagao, K ...
Article ID: JNMS.2025_92-602
Published: 2025
Advance online publication: March 22, 2025
JOURNAL
FREE ACCESS
ADVANCE PUBLICATION
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.
View full abstract
-
Takeshi Matsunobu, Hirotaka Suzuki, Akira Shimizu, Koji Sakamoto, Kimi ...
Article ID: JNMS.2025_92-603
Published: 2025
Advance online publication: March 22, 2025
JOURNAL
FREE ACCESS
ADVANCE PUBLICATION
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.
View full abstract
-
Takeshi Matsunobu, Kimihiro Okubo, Koichiro Saito
Article ID: JNMS.2025_92-604
Published: 2025
Advance online publication: March 22, 2025
JOURNAL
FREE ACCESS
ADVANCE PUBLICATION
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.
View full abstract
-
Takeshi Matsunobu, Hirotaka Suzuki, Kimihiro Okubo
Article ID: JNMS.2025_92-606
Published: 2025
Advance online publication: March 22, 2025
JOURNAL
FREE ACCESS
ADVANCE PUBLICATION
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.
View full abstract
-
Hirotoshi Kubokura, Yuuya Tomioka, Junichi Okamoto, Naoyuki Yoshino, J ...
Article ID: JNMS.2025_92-607
Published: 2025
Advance online publication: March 22, 2025
JOURNAL
FREE ACCESS
ADVANCE PUBLICATION
Objectives: 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.
View full abstract
-
Takeshi Matsunobu, Hirotaka Suzuki, Naoko Sakuma, Ryuji Ohashi, Kimihi ...
Article ID: JNMS.2025_92-608
Published: 2025
Advance online publication: March 22, 2025
JOURNAL
FREE ACCESS
ADVANCE PUBLICATION
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.
View full abstract
-
Kotoko Arai, Koji Sakamoto, Masashi Nakaishi, Atsuko Sakanushi, Ayaho ...
Article ID: JNMS.2025_92-402
Published: 2024
Advance online publication: August 21, 2024
JOURNAL
FREE ACCESS
ADVANCE PUBLICATION
Nasogastric tube syndrome (NGTS) is a complication of NGT placement that can cause sore throat, bilateral vocal cord paralysis, and airway constriction. Although rare, this condition should be known by all physicians because it is sometimes fatal. We report a case of NGTS that was successfully diagnosed and debrided with a rigid curved laryngoscope. A 79-year-old man was referred to our department for evaluation of persistent pharyngeal pain and dysphagia after thoracic aortic aneurysm surgery. He had restricted bilateral vocal fold abduction, and the NGT had been placed for nearly 120 days. After induction of general anesthesia, the patient underwent laryngeal examination using a rigid curved laryngoscope, which revealed ossified cricoid cartilage in the postcricoid area, leading to confirmation of NGTS. The NGT was removed, and the protruding bony lesion was debrided. Subsequently, the patient underwent gastrostomy to improve his nutritional status. One month later, the postcricoid lesion improved, and the patient was able to tolerate an oral diet. The pathophysiology of NGTS is thought to involve ulceration of the postcricoid mucosa and injury of the posterior cricoarytenoid muscle by contact with the NGT. Although the treatment has not been established, early removal of the NGT is important. In the present case, the rigid curved laryngoscope was useful for resection of the bony lesion, which contributed to early epithelialization and symptom improvement.
View full abstract
-
Yutaro Tobita, Naka Shiratori, Yusuke Nishio, Kenji Nakamoto, Fumiki O ...
Article ID: JNMS.2025_92-501
Published: 2024
Advance online publication: August 21, 2024
JOURNAL
FREE ACCESS
ADVANCE PUBLICATION
A 76-year-old woman was referred to our hospital for evaluation of markedly high intraocular pressures (IOPs): ≥40 mmHg in both eyes. No intraocular inflammation, pseudoexfoliation, or glaucomatous optic neuropathy was observed on ophthalmological examination. The 24-hour IOP fluctuations, measured with a Goldmann tonometer in the sitting position at 8, 12, 16, 20, and 24 o' clock, were 22/17, 33/28, 41/33, 30/22, and 30/24 mmHg, respectively, and showed a peak in the afternoon. The patient was diagnosed with ocular hypertension, and microhook trabeculotomy (μLOT) (right eye with lens reconstruction, left eye initially pseudophakic) was performed. After μLOT surgery in both eyes (4 days postoperatively in the right eye and 1 day postoperatively in the left eye), IOP decreased in both eyes to 12/15, 11/14, 12/15, 10/11, and 10/10 mmHg, and the fluctuation range was suppressed. At 3 months postoperatively (measured at 8, 12, 16, 20, 24, and 4 o' clock), the effect was maintained at 14/15, 15/19, 14/19, 11/12, 13/14, and 13/13 mmHg, respectively, but was slightly attenuated. In a patient with marked 24-hour IOP fluctuation, μLOT was effective in reducing IOP values and fluctuation.
View full abstract
-
Yuna Kamioka, Kensuke Izumida, Eiji Ohtaka, Yoshiaki Hashimoto, Hajime ...
Article ID: JNMS.2025_92-502
Published: 2024
Advance online publication: August 21, 2024
JOURNAL
FREE ACCESS
ADVANCE PUBLICATION
A 38-day-old infant was referred to our hospital for evaluation of apnea, fever, and pyuria. Invasive bacterial infection, including meningitis, was suspected because of the presence of apnea. A contrast-enhanced CT scan revealed acute localized bacterial nephritis, and meningitis was ruled out. Gram-positive cocci and Gram-negative rods, ie, Enterococcus raffinosus and Escherichia coli, were isolated from a urine culture at the referring hospital. This case report describes the youngest case of E. raffinosus infection. Apnea was the main complaint, but the origin of fever was infant acute focal bacterial nephritis (AFBN) with mixed infection. In infants, bacterial infections, especially invasive bacterial infections, can result in poor outcomes and require careful evaluation and treatment. Furthermore, the possibility of AFBN should not be overlooked, because bacteriuria or leukocyturia may be absent and can flare up if antimicrobials are not administered for an adequate duration. Although ampicillin-susceptible E. raffinosus infection in our patient responded well to treatment, there have been reports of vancomycin-resistant enterococci, which highlights the importance of proper use of antimicrobial agents to avoid producing drug-resistant bacteria.
View full abstract
-
Satoshi Akita, Kei Ishimaru, Mitsunori Sato, Katsuya Watanabe, Hiroki ...
Article ID: JNMS.2025_92-504
Published: 2024
Advance online publication: August 21, 2024
JOURNAL
FREE ACCESS
ADVANCE PUBLICATION
A rectovaginal fistula (RVF) is an abnormal tract between the rectum and vagina, which requires surgical intervention in many cases. Although there are many different therapeutic approaches for RVF depending on the patient's' condition, there are no established guidelines for the care of RVF. This study aimed to evaluate the results of laparoscopic colostomy in advanced cancer patients with RVF, and the safety and efficacy of this surgery. In this study, seven female advanced cancer patients with RVF were hospitalized and successfully treated with laparoscopic colostomy from 2015 to 2018 at our university hospital. Their data were retrospectively evaluated from their medical records. The early use of diverting stomas facilitated timely resumption of cancer treatment and enabled early treatment with chemotherapy or radiotherapy. Although vaginal stool leakage affected three patients, all patients recovered, experiencing neither pain nor infection during their cancer treatment. While colostomy was physically and mentally taxing for the patients, it improved the infection and pain caused by the RVF. We conclude that the early use of diverting stomas had two effects: a significant improvement in infection management and facilitation of the rapid resumption of cancer treatment.
View full abstract
-
Harumi Morioka, Masaru Yanagihashi, Mikito Toda, Takanori Ikeda, Masaf ...
Article ID: JNMS.2025_92-202
Published: 2024
Advance online publication: June 18, 2024
JOURNAL
FREE ACCESS
ADVANCE PUBLICATION
Prosopagnosia is a cognitive disorder in which facial recognition is severely impaired despite normal vision and intelligence. Prosopagnosia was first reported in the 1800s, but its cause remains unclear. Although other neurological symptoms are often present, some patients have pure prosopagnosia. The bilateral occipital lobes are believed to be associated with symptoms. Recent brain imaging techniques have identified the right fusiform gyrus (rFG), located at the junction of the right occipital temporal lobe, as the affected region. In this report, we present a case of associative prosopagnosia with no concomitant symptoms in a 76-year-old man. Brain magnetic resonance imaging detected a subcortical hemorrhage in the right temporal lobe. Using tractography based on diffusion tensor imaging, we visualized atrophy of the right inferior longitudinal fasciculus (ILF). This is the first time tractography has been used to show a clear association between associative prosopagnosia and ILF damage projecting from the rFG.
View full abstract
-
Masatsugu Ishii, Kakusho C Nakajima-Ohyama, Hayato Saito, Tomoyuki Ohy ...
Article ID: JNMS.2025_92-301
Published: 2024
Advance online publication: June 18, 2024
JOURNAL
FREE ACCESS
ADVANCE PUBLICATION
Schizophrenia develops during adolescence. Maternal infections during the fetal period increase the incidence of schizophrenia in children, which suggests that the pathogenesis involves neuroinflammation. Here, we report a case of new-onset schizophrenia in a 16-year-old boy after COVID-19. After developing COVID-19, he entered a catatonic state 4 days later and was hospitalized. Benzodiazepines alleviated his catatonia, but hallucinations and delusions persisted. Encephalitis and epilepsy were excluded by magnetic resonance imaging (MRI), encephalography, and cerebrospinal fluid examination. Psychosis persisted after the virus titer declined and the inflammatory response subsided. Moreover, the patient exhibited delusions of control—a Schneider's first-rank symptom. Schizophrenia was diagnosed, and olanzapine improved his symptoms. He had a brief history of insomnia before COVID-19 but his symptoms did not satisfy the ultra-high-risk criteria. However, COVID-19 may have facilitated development of schizophrenia through neuroinflammation and volume reduction in the gray matter of the right medial temporal lobe. This case demonstrates that infectious diseases in adolescents should be carefully managed, to prevent schizophrenia.
View full abstract
-
Yutaka Furuta, Takahiro Masuoka, Ryuichiro Narishige, Amane Tateno
Article ID: JNMS.2025_92-302
Published: 2024
Advance online publication: June 18, 2024
JOURNAL
FREE ACCESS
ADVANCE PUBLICATION
Painless thyroiditis, which is rare in children, exhibits the characteristic sequence of hyperthyroidism, including aggressive and disruptive behaviors. Unlike subacute thyroiditis or Graves' disease, painless thyroiditis is challenging to diagnose because of its mild symptoms and minimal or absent physical findings. Moreover, aggressive and disruptive behaviors in children with psychiatric disorders may be misconstrued as exacerbation of underlying symptoms. The present patient was a 16-year-old male with adjustment disorder who presented to a pediatric psychiatric clinic for assessment of irritability. After 4 months, he developed aggressive and disruptive behaviors that prompted initiation of risperidone but without improvement. After 1 month, he reported palpitations and dyspnea. His neck was supple and non-tender without thyroid enlargement. Thyroid studies revealed elevated free T4 and T3 levels and suppressed thyroid-stimulating hormone level, suggesting hyperthyroidism. A radioactive iodine uptake test revealed a barely visible thyroid gland, consistent with thyroiditis. Painless thyroiditis, without thyroid tenderness, was diagnosed. We describe a case of painless thyroiditis in an adolescent patient with aggressive and disruptive behaviors that were initially attributed to worsening of an underlying adjustment disorder. Even when minimal or no signs of hyperthyroidism are present, painless thyroiditis should be considered in the differential diagnosis of children with aggressive and disruptive behaviors. Awareness of potential anchoring bias is also recommended to prevent its delayed diagnosis of such behaviors.
View full abstract
-
Haruka Ota, Toru Igarashi, Ryosuke Matsui, Hikaru Takeshita, Koji Hash ...
Article ID: JNMS.2025_92-303
Published: 2024
Advance online publication: June 18, 2024
JOURNAL
FREE ACCESS
ADVANCE PUBLICATION
Mixed connective tissue disease (MCTD) is characterized by mixed features of systemic lupus erythematosus, systemic sclerosis, and polymyositis/dermatomyositis and is rare in children. Here, we report a case of MCTD in a 10-year-old girl who presented at our hospital with arthralgia, Raynaud's phenomenon, and fatigue. Blood tests were positive for anti-U1-ribonucleoprotein (RNP) antibodies and for rheumatoid factors (RFs) IgG-RF and anti-galactose-deficient IgG. Levels of myogenic enzymes and hypergammaglobulinemia were elevated. Macrophages were prominent in bone marrow, with scattered phagocytic macrophages. MCTD was diagnosed based on the patient's symptoms and laboratory findings. Methylprednisolone pulse therapy combined with oral tacrolimus was administered, which led to resolution of symptoms. Three months after pulse therapy, arthralgia worsened and methotrexate was administered. Arthralgia improved but did not resolve. Magnetic resonance imaging performed to investigate the hip pain revealed a mature ovarian teratoma, which was surgically removed. Because the pain persisted and interfered with her daily life, she was treated with tocilizumab for joint pain relief, which decreased the pain level. Tocilizumab is a candidate for additional treatment of juvenile idiopathic arthritis-like arthritis associated with childhood-onset MCTD.
View full abstract
-
Yutaka Furuta, Naoto Matsuda, Hiroaki Wakakuri, Hideya Hyodo, Toshihik ...
Article ID: JNMS.2025_92-401
Published: 2024
Advance online publication: June 18, 2024
JOURNAL
FREE ACCESS
ADVANCE PUBLICATION
Anisakiasis is a parasitic infection affecting the human gastrointestinal tract. It is caused by the consumption of contaminated, raw or inadequately cooked fish or squid, which is typically used for making sushi and sashimi. Most cases involve gastric anisakiasis, whereas intestinal anisakiasis is rare. This report describes the case of a 63-year-old Japanese woman with a history of raw fish consumption who presented with acute-onset abdominal pain and vomiting. Abdominal computed tomography (CT) demonstrated thickened small bowel loops and ascites on the liver surface. The patient was admitted for supportive care. On the second day of hospitalization, contrast-enhanced abdominal CT revealed that the ascites had moved from the liver surface to the pouch of Douglas. On the fifth day of hospitalization, the patient was discharged with a substantial improvement in abdominal pain. Five days after the discharge, her eosinophil count was elevated, and parasitic disease was therefore suspected. Anti-Anisakis IgG/A and IgE (RAST) antibody levels were elevated, confirming the diagnosis of intestinal anisakiasis. A review of 51 reported cases of intestinal anisakiasis suggests that the presence of ascites and measurement of anti-Anisakis antibody titers are helpful for diagnosis in cases presenting with nonspecific abdominal symptoms after consumption of raw or undercooked fish.
View full abstract