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Reeko Osada,1 Chiyako Oshikata,1– 3 Yuichi Kurihara,4 Kosuke Terada,1 Yuka Kodama,1 Yuga Yamashita,1 Ryo Nakadegawa,1 Hinako Masumitsu,1 Yuto Motobayashi,1 Hirokazu Takayasu,1 Nami Masumoto,1,3 Saki Manabe,1 Yingyao Zhu,4 Ryo Tanaka,4 Takeshi Kaneko,3 Aya Sasaki,5 Naomi Tsurikisawa1– 3 1Department of Respirology, National Hospital Organization Yokohama Medical Center, Yokohama, Japan; 2Department of Allergy and Respirology, Hiratsuka City Hospital, Hiratsuka, Japan; 3Department of Pulmonology, Yokohama City University Graduate School of Medicine, Yokohama, Japan; 4Department of Dermatology, Hiratsuka City Hospital, Hiratsuka, Japan; 5Department of Clinical Laboratory, Tokyo Dental College Ichikawa General Hospital, Ichikawa, JapanCorrespondence: Naomi Tsurikisawa, Department of Respirology, National Hospital Organization Yokohama Medical Center, 3-60-2 Harajuku, Totsuka-ku, Yokohama, 245-8575, Japan, Tel +81-45-851-2621, Fax +81-45-851-3902, Email User831328@aol.comAbstract: Monosodium glutamate (MSG), a salt form of a non-essential amino acid, is widely used as a food additive, particularly in Asian cuisines, due to its unique flavor-enhancing qualities. Type I allergic reactions to MSG have not previously been reported. Our patient, a 21-year-old woman, was 14 years old when she first noticed swelling of her tongue (but no oral itching, diarrhea, or abdominal pain) after eating various snack foods. Current skin prick testing elicited a weak positive reaction to MSG. We then performed an oral challenge test during which our patient ingested potato snacks. Subsequent histology showed telangiectasia of the buccal mucosa, interstitial edema in the subepithelial submucosa, and mast cell infiltration. Oral mucosal challenge tests using sodium glutamate confirmed oral swelling in this patient. This report is the first to confirm a case of type 1 allergy to MSG by combining pathology findings with the results of challenge testing.Keywords: food additive, mast cell, monosodium glutamate, food allergy

Background: Spontaneous anterior interosseous nerve (AIN) palsy is characterized by the sudden onset of upper limb pain followed by weakness of muscles mainly innervated by the AIN. Although this palsy is conventionally treated conservatively, interfascicular neurolysis to release hourglass-like fascicular constrictions has been recommended. The present study aimed to establish the clinical characteristics and treatment strategy for this condition., Methods: One hundred limbs with spontaneous AIN palsy from 24 Japanese institutions were treated either conservatively or with interfascicular neurolysis according to the patient's intention. Patients were followed periodically from 3 months to either recovery or ≥36 months after onset. Limbs recovering to manual muscle testing grade 4 or better in both the flexor pollicis longus and flexor digitorum profundus of the index finger were rated as Good recovery, with all other results classified as Poor recovery. We performed logistic regression analysis to determine the independent factors associated with Good recovery., Results: Good recovery was achieved in 39 of 49 limbs treated conservatively and 44 of 51 limbs by interfascicular neurolysis. All 26 limbs with conservative treatment displaying ≥1 grade of manual muscle testing improvement within 6 months after onset achieved Good recovery. All 23 limbs with Group I palsy, in which weakness was recognized only in muscles innervated by the AIN, obtained Good recovery with interfascicular neurolysis regardless of when it was performed. In the 59 limbs with no recovery at 6 months by conservative treatment, Good recovery was achieved in 30 of 36 limbs by subsequent interfascicular neurolysis and 13 of 23 limbs continuing conservative treatment. In these 59 limbs, interfascicular neurolysis and Group I palsy were significantly associated with Good recovery., Conclusions: Conservative treatment is recommended within 6 months following symptom onset, after which time interfascicular neurolysis is advisable for cases of no improvement., Competing Interests: Declaration of competing interest All authors declare that there is no conflict of interest associated with this study., (Copyright © 2024. Published by Elsevier B.V.)

This work demonstrates the effectiveness of vigorously stirred lanthanum oxide (La 2 O 3 ) suspensions in catalyzing Michael additions in water. These surfactant-free suspensions offer a counterintuitive yet highly efficient approach compared to traditional methods. Notably, the reactions are ineffective in the absence of water, suggesting a crucial role for the aqueous environment.

Diagnosis and treatment of pulmonary hypertension (PH) in patients with lung diseases (PH-LD) remain unestablished and pose significant challenges. In this report, we present a case of a 77-year-old patient with an indeterminate for usual interstitial pneumonia pattern along with chronic obstructive pulmonary disease, who developed groups 1 and 3 PH. Following diagnosis, upfront triple oral combination therapy (UTOCT) with macitentan, sildenafil, and selexipag was initiated. Stability in disease progression was achieved over 4 years with the addition of pirfenidone to address interstitial lung disease progression. To the best of our knowledge, this represents the first reported case of PH-LD, where disease control was maintained with the addition of pirfenidone to UTOCT. This case suggests that some patients with PH-LD, presenting with groups 1 and 3 PH, may benefit from combined UTOCT and antifibrotic agents, potentially improving symptoms and extending their prognosis., Competing Interests: None declared., (© 2024 The Author(s). Respirology Case Reports published by John Wiley & Sons Australia, Ltd on behalf of The Asian Pacific Society of Respirology.)

Introduction: Patients with chronic rhinosinusitis with nasal polyps (CRSwNP) and aspirin-exacerbated respiratory disease (AERD) have more severe sinus disease than those without AERD. CRSwNP associated with type 2 inflammation and AERD can be difficult to control with standard medical therapy and sinus surgery., Case Study: 74-year-old Japanese woman with chronic sinusitis since age 50 and asthma since age 60. At age 64, she began to experience asthma exacerbations and was started on short-term corticosteroid therapy with prednisolone. At age 70, she experienced urticaria, nasal congestion, and wheezing after taking an NSAID; based on an NSAID provocation test, we diagnosed the patient with AERD and CRSwNP. A diagnosis of severe eosinophilic chronic rhinosinusitis was also made based on the scoring system and algorithm used in the Japanese Epidemiological Survey of Refractory Eosinophilic Chronic Rhinosinusitis., Results: Treatment with benralizumab (30 mg), formoterol-fluticasone combination via pressurized metered inhaler (1000 μg), and leukotriene receptor antagonist improved the asthma symptoms and exacerbations so the short-term prednisolone was stopped; however, nasal congestion and olfactory dysfunction (hyposmia) persisted, and peripheral blood eosinophil count (peak, 1500 cells/μL) and fractional exhaled nitric oxide (peak, 42 ppb) became elevated. Swapping the benralizumab for monthly tezepelumab (210 mg) improved not only the asthma symptoms but also the nasal congestion, olfactory dysfunction, eosinophil count (<300 cells/μL), and fractional exhaled nitric oxide level [8ppb]., Conclusion: Changing from benralizumab to tezepelumab improved asthma symptoms, nasal obstruction, and olfactory dysfunction in elderly, female, Japanese patient with AERD and CRSwNP., Competing Interests: Have no conflicts of interest to disclose about this study., (© 2024 The Authors.)

Background: Atypical fractures are caused by the combined effects of severe suppression of bone metabolism (SSBT) due to long-term bisphosphonate therapy and chronic repetitive bone microdamage. Atypical ulnar fractures (AUFs) due to SSBT are rare, and there is no standard treatment strategy for such fractures. The relevant literature was reviewed, and the treatment strategy for AUF is discussed., Methods: A systematic review was conducted. All studies on ulnar fractures in individuals with a history of bisphosphonate use were included, and the data were extracted and analyzed from the perspective of the therapeutic strategy., Results: Forty limbs of 35 patients were included. As for the treatment of AUF, 31 limbs were treated surgically, and conservative treatment with casting was performed for 9 limbs. The bone fusion rate was 22/40 (55.0%), and non-union was seen in all patients treated conservatively. There was a significant difference in the bone fusion rate between patients with surgical treatment and those with conservative treatment. The bone fusion rate of patients with parathyroid hormone (PTH) and surgery was 82.3% (14/17 limbs); the bone fusion rate with PTH and bone graft was 69.2% (9/13 limbs). However, there were no significant differences in the fusion rate in the groups with or without PTH, with or without bone grafting, or the combination of the two treatments. There was also no significant difference in the bone fusion rate in the groups with or without low-intensity pulsed ultrasound (LIPUS) treatment., Conclusions: Based on the literature review, surgery is necessary to achieve bone union, but surgery alone is not adequate to achieve bony union. Bone grafting and the administration of PTH and LIPUS may promote early bone fusion, but the present study did not show significant advantages of these additional treatments for bone union., (Copyright © 2023. Published by Elsevier B.V.)

A subscapular system free-flap is extremely useful for maxillofacial reconstruction since it facilitates the simultaneous harvesting of multiple flaps using one subscapular artery (SSA) alone. However, cases of aberrations in the SSAs have been reported. Therefore, the morphology of SSA needs to be confirmed preoperatively before harvesting the flaps. Recent developments in imaging, such as three-dimensional (3D) computed tomography angiography (3D CTA), facilitate obtain high-quality images of blood vessel images. Therefore, we examined the utility of 3D CTA in navigating the course of the SSA before harvesting subscapular system free-flaps. We examined the morphology and aberrations of the SSA using 39 sides of the 3D CTA data and 22 sides of Japanese cadavers. SSAs can be classified into types S, I, P, and A. Type S SSAs are significantly long (mean length = 44.8 mm). Types I and P SSAs have short mean lengths, measuring ≤2 cm in approximately 50% of cases. In type A, the SSA is absent. The frequency of types S, I, P, and A SSAs were 28.2%, 7.7%, 51.3%, and 12.8%, respectively. Type S can be advantageous for harvesting the SSA in subscapular system free-flaps, because it is significantly longer. In contrast, types I and P might be dangerous because their mean lengths are shorter. In type A, caution is needed not to injure the axillary artery because the SSA is absent. When surgeons need to harvest the SSA, presurgical 3D CTA is recommended., (© 2023 American Association of Clinical Anatomists and British Association of Clinical Anatomists.)

Treating ocular involvement in eosinophilic granulomatosis with polyangiitis (EGPA) can be challenging. We present the case of a 37-year-old woman with EGPA who had severe bilateral visual field defects. Laboratory results showed leukocytosis (17,500 WBC/μL, 25.8 % eosinophils), negative MPO-ANCA titer, and elevated PR3-ANCA level (33.2 IU/mL). Diffusion-weighted MRI revealed bilateral hyperintense occipital lesions, which were more prominent on the left. Her therapy initially included a steroid pulse, followed by daily prednisolone, but her visual field defects remained refractory. The addition of intravenous cyclophosphamide (5 courses) and intravenous immunoglobulin decreased her optic neuropathy and resolved her visual field defects., Competing Interests: The authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper., (© 2024 The Authors.)

Introduction: Many studies have reported a poor prognosis for eosinophilic granulomatosis with polyangiitis (EGPA) patients with cardiac involvement., Case Study: A woman developed EGPA at 37 years of age, with weight loss, numbness in the right upper and lower extremities, muscle weakness, skin rash, abdominal pain, chest pain, an increased peripheral blood eosinophil count (4165/µL), and necrotizing vasculitis on peroneal nerve biopsy. The patient was treated with prednisolone, immunosuppressants, intravenous immune globulin, and mepolizumab, but she experienced many relapses, with chest pain, abdominal pain, numbness, and paralysis, over a long period. The patient died from aspiration pneumonia at 71 years of age after undergoing left total hip arthroplasty for left hip neck fracture., Results: Autopsy showed bronchopneumonia in the lower lung lobes on both sides, as well as infiltration of inflammatory cells, including neutrophils and lymphocytes. There was no evidence of active vasculitis in either the lung or colon. At autopsy the heart showed predominantly subendocardial fibrosis and fatty infiltration, but no active vasculitis or eosinophilic infiltration., Conclusion: To our knowledge, there have been no autopsy reports of EGPA patients who have survived for 34 years with recurrent cardiac lesions. In this case, the cardiac involvement (active vasculitis and eosinophilic infiltration) had improved by the time of death.

Purpose: We investigated chronological changes in active range of motion (ROM) of the metacarpophalangeal (MCP) joint after zone VII extensor tendon reconstruction with wide-awake local anesthesia no tourniquet (WALANT) surgery., Methods: Reconstruction of the extensor tendons ruptured in zone VII of 82 fingers in 40 patients was performed using WALANT techniques. The mean patient age was 71.3 years. During surgery, we evaluated passive stretch distance and active contraction distance of the ruptured musculocutaneous unit. The sum of passive stretch distance and active contraction distance was defined as total excursion. In cases with total excursion ≥30 mm, we selected bridge tendon grafting for tendon reconstruction. In cases with total excursion <30 mm, end-to-side tendon transfer was performed. Active ROM of the MCP joint was measured before surgery; during surgery; 1, 3, and 5 months after surgery, and at the final follow-up., Results: Extension lag of the MCP joint was 49.2° before surgery, improving to 2.9° during surgery, deteriorating to 18.7° at 1-month, and improving again to 15.6° at 3-months, 13.6° at 5-months, and 10.5° at the final follow-up. Meanwhile, the active flexion angle of the MCP was 87.9° before surgery, 87.3° during surgery, 67.6° at 1-month, 76.0° at 3-months, 79.7° at 5-months, and 81.0° at the final follow-up. Extension and flexion angles at each time point remained nearly constant regardless of whether tendon grafting or tendon transfer was used., Conclusions: Extensor tendon reconstruction using WALANT surgery allowed intraoperative measurement of active ROM, confirming near-complete extension and flexion after reconstruction. The changes in ROM of the MCP joint during follow-up are an approximate indication of the progression of postoperative recovery., Type of Study/level of Evidence: Therapeutic Ⅳ., (Copyright © 2023 American Society for Surgery of the Hand. Published by Elsevier Inc. All rights reserved.)

Dupilumab-induced hypereosinophilia is mediated by blockade of the IL-4/IL-13 pathway, which reduces eosinophil migration from peripheral blood. The increase in peripheral blood eosinophils may lead to chronic eosinophilic pneumonia (CEP) and/or eosinophilic granulomatosis with polyangiitis, but a direct causal connection between dupilumab and eosinophilic lung diseases has not been established. A 33-year-old Japanese woman with bronchial asthma since age three was treated with fluticasone propionate plus salmeterol twice daily after several asthma exacerbations at age 17. Her course was complicated by CEP at age 33 which resolved without the need for systemic steroids. However, in the four months following resolution of her CEP, the patient had three asthma exacerbations, and a recurrence of CEP, with blood leukocytes of 8500/µL, of which 25.0% were eosinophils. She was treated with prednisolone 50 mg/day, but she could not continue this dose due to the onset of myalgia. Then she had relapsing CEP twice within three months. She was treated with prednisolone 15 mg/day for CEP, but she had persistent asthma for more than one month; dupilumab was added at 600 mg, followed by 300 mg every two weeks. In the first month of treatment with dupilumab, the patient's asthma symptoms resolved completely, and she had only one relapse of CEP. In 12 months of follow-up, she had neither an asthma exacerbation nor another relapse of CEP. Dupilumab may be a promising treatment for patients with refractory asthma complicated by recurring CEP and undesirable steroid side effects., Competing Interests: No author has any conflict of interest to disclose.

Immune checkpoint inhibitors (ICIs) activate anti-tumor activity by inhibiting immune checkpoint molecules that suppress inflammatory T-cell activity. However, ICIs can initiate excessive immune responses, thereby causing immune-related adverse events (irAEs). ICI-associated uveitis (ICIU) is an irAE that affects the eyes. Although Vogt-Koyanagi-Harada disease (VKH)-like uveitis is a common form of ICIU, it is unclear which factors determine the ICIU form. We retrospectively reviewed the medical records of nine ICIU cases treated with ICIs for malignancies. We also performed HLA typing in seven cases to investigate the association between HLA and disease type. Fisher's exact test was used for the statistical analysis. Five of the ICIU cases were VKH-like ICIUs, and four were non-VKH-like ICIUs. No association was found between mean age, sex, primary disease, ICI, time to onset, and disease type. Four patients with VKH-like uveitis underwent HLA genotyping and were all positive for HLA-DRB1*04:05. All 3 patients with non-VKH-like uveitis were negative for HLA-DRB1*04:05. Statistical analysis showed that HLA-DRB1*04:05 was significantly associated with developing VKH-like ICIU (P = 0.029). In ICIU, HLA-DRB1*04:05 was associated with the pathogenesis of VKH-like uveitis, suggesting that ICI-associated VKH-like uveitis has a similar pathogenesis to VKH., (© 2023. Springer Nature Limited.)

Background: The mainstay of treatment for eosinophilic granulomatosis with polyangiitis (EGPA) is systemic corticosteroid therapy; some patients also receive intravenous immunoglobulins, other immunosuppressive agents, and biologics. Mepolizumab, an anti-interleukin-5 monoclonal antibody, induces remission and decreases the daily corticosteroid dose; however, the clinical efficacy of mepolizumab in EGPA and the prognosis with long-term treatment with this drug are unknown., Methods: Seventy-one EGPA patients were treated at Hiratsuka City Hospital, Japan, between April 2018 and March 2022. We administered mepolizumab for a mean of 2.8 ± 1.7 years to 43 patients in whom remission could not be induced by conventional treatment. After excluding 18 patients who had received mepolizumab for less than 3 years, we classified 15 patients into a "super-responder group" (the daily dose of corticosteroids or other immunosuppressant could be decreased, or the interval between IVIG treatments could be prolonged) and 10 patients into a "responder group" (neither of these changes was achievable). Eosinophil numbers, serum IgG levels, daily doses of corticosteroids and other immunosuppressants, Birmingham Vasculitis Activity Score (BVAS), and relapse frequency before and after mepolizumab initiation were determined., Results: Blood eosinophil count at diagnosis and the lowest serum IgG level before mepolizumab treatment were significantly higher in super-responders than in responders (p < 0.05). In super-responders, the prednisolone dose at last visit on mepolizumab treatment was lower than that before treatment (p < 0.01) and lower than that at last visit in the responders (p < 0.01). In both groups, peripheral blood eosinophil numbers and BVAS were lower after starting mepolizumab than before (p < 0.01). BVAS before mepolizumab (p < 0.05) and at last visit (p < 0.01) were lower in super-responders than in responders. Relapse rates every year after the start of mepolizumab were lower in super-responders than in responder groups (p < 0.01). In super-responders, relapse rates were lower during the 3 years following mepolizumab initiation (p < 0.01) and at last visit (p < 0.01) were significantly lower than after 1 year of treatment., Conclusion: Mepolizumab treatment of super-responders sustainably reduced the relapse rate., (© 2023. The Author(s).)

The axial chirality of hexahydroxydiphenoyl (HHDP) groups contained in ellagitannins depends on the bridging position of d-glucose. The 4,6- O -HHDP group predominantly exhibits S- type chirality. This study elucidated the formation of the 4,6- O -( R )-HHDP group and subsequent axial isomerization by means of oxidative coupling of galloyl groups, which resulted in S chirality.

Purpose: We investigated the clinical outcomes of flexor tendon reconstruction for chronic rupture of the flexor tendon based on an evaluation of the voluntary active contraction distance (ACD) of the ruptured musculotendinous unit and changes in intraoperative total active motion (TAM) that could only be observed during wide-awake local anesthesia no tourniquet (WALANT) surgery., Methods: Reconstructions of 19 tendons of the flexor pollicis longus (FPL) and 18 tendons of the flexor digitorum profundus (FDP) were performed during WALANT surgery to evaluate the ACD of the ruptured musculotendinous unit and TAM observed during the surgery. Tendon grafting or tendon transfer was selected during the surgery based on ACD. TAM, pinch strength, and grip power were evaluated before the surgery, during the surgery, and at final follow-up, and they were surveyed based on Quick Disabilities of the Arm, Shoulder, and Hand (q-DASH) scores. The final outcomes of tendon grafting and tendon transfer were compared., Results: In FPL tendon reconstruction, tendon grafting was performed in 10 patients with a total PDD and ACD value greater than 30 mm, and tendon transfer was performed in 9 patients with the value less than 30 mm. In FDP tendon reconstruction, tendon grafting was performed in 8 patients and tendon repair in 2 patients with a total PDD and ACD value greater than 40 mm, and tendon transfer was performed in 8 patients with the value less than 40 mm. The TAM value, q-DASH score, pinch power, and grip strength were improved in all patients. In both the tendon reconstructions, intraoperative TAM was significantly increased compared with preoperative TAM but significantly decreased at final follow-up. No significant differences were identified in final follow-up TAM and the q-DASH scores between tendon transfer and tendon grafting., Conclusions: The great advantage of WALANT surgery is that surgeons can evaluate the ruptured musculotendinous unit and measure TAM during the surgery., Type of Study/level of Evidence: Therapeutic I., (© 2022 The Authors.)

Background: No anti-adhesive materials are currently in clinical use for orthopaedic surgery. We developed a hyperdry amniotic membrane (HD-AM) for easy storage and transplantation as amniotic membrane. The purpose of this study was to examine the application of HD-AM to reduce peritendinous adhesions without impairing tendon healing., Methods: We randomly divided 3 digits (2nd, 3rd, and 4th digits) from each rabbit into three groups: a tendon repair group; a tendon repair with HD-AM group (HD-AM group); and a control group (cast only). The effects of HD-AM on peritendinous adhesions and tendon healing were examined using microscopic, histological, and mechanical analyses in a rabbit flexor digitorum profundus tendon model., Results: Adhesions on macroscopic evaluation of the tendon repair site were significantly smaller in the HD-AM group than in the tendon repair group. Little adhesion formation or foreign body reactions were seen by on histologic evaluation in the HD-AM group. Range of motion following tendon repair was significantly better in the HD-AM group than in the tendon repair group. Maximal tensile strength required to pull the tendon from the site of adhesion was significantly smaller in the HD-AM group than in the tendon repair group. As for tendon repair site, no significant difference was seen between the tendon repair and HD-AM groups., Conclusions: HD-AM prevented peritendinous adhesion macroscopically, pathologically, and mechanically without impairing the sutured tendon. HD-AM has already been clinically applied in neurosurgery, ophthalmology, and otolaryngology, and clinical application as an anti-adhesive materials may be achieved in the future., Competing Interests: Declaration of competing interest The authors declare that they have no conflicts of interest., (Copyright © 2021 The Japanese Orthopaedic Association. Published by Elsevier B.V. All rights reserved.)