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Wataru Sumida, Kazuya Nagayabu, Kazuki Ota, Yuki Imai, Yasuyuki Ono, H ...
2025 年11 巻1 号 論文ID: cr.25-0182
発行日: 2025年
公開日: 2025/09/25
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INTRODUCTION: Aberrant right subclavian artery (ARSA) is the most common congenital anomaly of the subclavian artery, occurring in 0.2%–1.5% of the population. While typically asymptomatic, ARSA can cause dysphagia in older patients due to esophageal compression. In pediatric cases, it is less frequent and usually presents with chronic respiratory symptoms resulting from tracheal compression. Acute respiratory failure as a presentation of ARSA is extremely rare. This case report describes a pediatric patient with ARSA-associated esophageal stricture, leading to severe respiratory distress following food intake and requiring surgical treatment.
CASE PRESENTATION: A 1.8-year-old girl developed an inspiratory stridor after eating. Initial inhalation therapy provided temporary relief, but persistent respiratory distress prompted further evaluation. CT revealed an esophageal foreign body, which was successfully extracted using a balloon catheter. However, continued respiratory distress necessitated intubation and intensive care. Bronchoscopy and enhanced CT confirmed tracheal compression due to ARSA, along with esophageal stenosis. Despite initial medical management, recurrent respiratory distress and feeding difficulties required surgical intervention. A right thoracotomy was performed to reposition the aberrant artery anterior to the trachea, alleviating the vascular compression. Postoperatively, the patient made a full recovery, resumed a normal diet, and remained symptom-free at the 6-month follow-up.
CONCLUSIONS: Symptomatic ARSA is rare in pediatric patients but can cause severe respiratory distress due to esophageal and tracheal compression. In cases of recurrent or severe symptoms, surgical intervention is essential to prevent life-threatening complications. This case highlights the importance of considering ARSA in pediatric patients with unexplained respiratory distress, especially when associated with feeding difficulties.
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Tomohiro Minagawa, Kenji Watanabe, Yusuke Takashima, Toru Watanabe, Ta ...
2025 年11 巻1 号 論文ID: cr.25-0081
発行日: 2025年
公開日: 2025/09/25
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INTRODUCTION: Crohn’s disease (CD) can be complicated by complex fistulas and abscesses, and laparoscopic surgery for such cases is difficult. We report a patient with CD who developed a complex fistula and retroperitoneal abscess that were treated using single-incision laparoscopic surgery.
CASE PRESENTATION: A 34-year-old man presented to the hospital after collapsing and losing consciousness while at work. Blood tests showed a white blood cell count of 20000/μL and severe anemia. Abdominal CT showed a right retroperitoneal abscess, which was drained under CT guidance. After gastrointestinal bleeding and an anal fistula were observed, CD was suspected and confirmed by total colonoscopy. The patient was eventually discharged but readmitted 2 months later for abscess recurrence and a colocutaneous fistula. Once the inflammation had improved after 3 weeks of fasting and intravenous antibiotics, the patient underwent single-incision laparoscopic subtotal colectomy, duodenal fistula closure, and seton drainage.
CONCLUSIONS: Single-incision laparoscopic subtotal colectomy can be performed safely to treat CD-related abscesses and complicated fistulas after inflammation has improved.
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Yoshihito Kitamura, Masashi Miguchi, Ryo Nagao, Makoto Shinohara, Keig ...
2025 年11 巻1 号 論文ID: cr.25-0271
発行日: 2025年
公開日: 2025/09/25
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INTRODUCTION: Benign peritoneal multicystic mesothelioma (BPMM) is a rare mesothelial tumor with a high local recurrence rate and potential for malignant transformation. It predominantly affects women of reproductive age and is often associated with prior abdominal surgery or inflammation. Complete surgical resection is the standard treatment; however, tumor recurrence remains a concern. BPMM occurring outside the pelvis is extremely rare, and lesions requiring colectomy because of adherence to the gastrointestinal tract are infrequent.
CASE PRESENTATION: A 63-year-old woman presented with right lower abdominal pain. She had undergone cervical cancer surgery 20 years ago. Contrast-enhanced CT and MRI revealed multiple well-defined cystic lesions adjacent to the ascending colon. Colonoscopy revealed extrinsic compression, and 18F-fluorodeoxyglucose (18F-FDG)-PET/CT showed no abnormal uptake of 18F-FDG. Given the patient’s symptoms, a laparoscopic right hemicolectomy with intracorporeal anastomosis was performed. The cystic lesions were firmly adhered to the appendix, cecum, and ascending colon, requiring en bloc resection. Histopathological and immunohistochemical analyses confirmed a diagnosis of BPMM. The patient had an uneventful postoperative course and was discharged on POD 7. No recurrence was observed during the 1-year follow-up period.
CONCLUSIONS: A laparoscopic approach may be a feasible and safe option for complete resection of BPMM as it allows for magnified visualization and careful handling of cystic lesions, avoiding their intraoperative rupture. Given the high recurrence rate of BPMM, close postoperative surveillance is essential. This case illustrates the feasibility of laparoscopic resection for BPMM.
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Peipei Zhang
2025 年11 巻1 号 論文ID: cr.25-0300
発行日: 2025年
公開日: 2025/09/23
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INTRODUCTION: Primary cardiac schwannoma (PCS) is an exceptionally rare neurogenic tumor, with only 53 documented cases worldwide.
CASE PRESENTATION: We report the case of a 62-year-old woman with a 3-year history of paroxysmal palpitations, who subsequently developed progressive symptoms, such as chest tightness, dyspnea, and dizziness. Diagnostic evaluation revealed supraventricular tachycardia along with a 3.4 × 3.5 cm mass located at the coronary sinus ostium. Multimodal imaging—including echocardiography, CT, and MRI—revealed features characteristic of schwannoma, such as T1 hypointensity, T2 hyperintensity, and peripheral enhancement with internal septations. The patient underwent complete surgical resection of an encapsulated interatrial septal mass measuring 5.0 × 4.0 cm via a right atrial approach, with preservation of cardiac architecture and maintenance of compensatory venous drainage through an accessory ostium. Histopathological analysis confirmed a benign schwannoma exhibiting secondary degenerative changes, supported by positive SOX-10 and S-100 immunostaining and a low Ki-67 proliferation index. At the 3-year follow-up, the patient remained free of recurrence and exhibited no postoperative complications.
CONCLUSIONS: This case underscores the diagnostic challenges associated with PCS, particularly in the context of arrhythmias, and emphasizes the importance of multimodal imaging in the preoperative evaluation. The successful surgical resection demonstrates the importance of meticulous surgical planning and technique in managing these rare tumors. This report adds to the limited body of literature on PCS and reinforces the need for a multidisciplinary approach in the diagnosis and management of these complex cases.
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Takahisa Hirano, Michitaka Honda, Soshi Hori, Hirohito Kakinuma, Ryuya ...
2025 年11 巻1 号 論文ID: cr.25-0296
発行日: 2025年
公開日: 2025/09/23
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INTRODUCTION: Gastric neuroendocrine carcinoma (NEC) is a rare histological subtype of gastric malignancy. A small NEC component may coexist with gastric adenocarcinoma, making preoperative diagnosis challenging. Currently, no established treatment strategies exist for cases in which an NEC component is identified only in postoperative pathological findings.
CASE PRESENTATION: An 82-year-old man underwent esophagogastroduodenoscopy during a routine health checkup and was diagnosed with early-stage gastric cancer. The lesion was a superficial tumor, 30 mm in diameter, located on the lesser curvature of the cardia. Biopsy revealed well-differentiated adenocarcinoma. The patient subsequently underwent laparoscopic proximal gastrectomy with D1+ lymph node dissection. Histopathological examination of the resected specimen revealed adenocarcinoma with an NEC component, accounting for approximately 10% of the tumor. The pathological stage was T1bN1M0 (Stage IB), and the patient did not receive adjuvant chemotherapy. Five months after the surgery, the patient developed anorexia and weight loss. CT revealed multiple liver tumors. Diagnostic laparoscopy with excisional biopsy of the liver lesion was performed, revealing no peritoneal dissemination. Histopathological examination confirmed that the liver tumors were metastatic NEC, indicating hepatic recurrence of gastric NEC. Given the patient’s rapidly deteriorating condition and a performance status of 3–4, aggressive chemotherapy was deemed inappropriate, and palliative care was initiated. The patient died of the primary disease 8 months after surgery.
CONCLUSIONS: A small NEC component may be undetectable preoperatively. The prognosis of mixed tumors is dictated by the malignancy of the NEC component rather than its tumor burden. Therefore, clinicians should consider NEC-equivalent adjuvant chemotherapy and intensive surveillance.
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Ryo Karita, Yuki Onozato, Yuki Hirai, Toshiko Kamata, Hajime Tamura, H ...
2025 年11 巻1 号 論文ID: cr.25-0347
発行日: 2025年
公開日: 2025/09/20
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INTRODUCTION: Venous aneurysms in the anterior mediastinum are rare. While there are scattered reports on thoracic venous aneurysms, such as those involving the azygos or brachiocephalic veins, those involving the internal thoracic vein are exceedingly rare. We herein report the case of an internal thoracic venous aneurysm that was initially suspected of being a tumor and diagnosed intraoperatively.
CASE PRESENTATION: A 50-year-old woman underwent CT during hospitalization for cerebral infarction, which revealed a well-defined 14-mm nodular lesion in the anterior mediastinum. Contrast-enhanced CT performed 4 months later showed that the anterior mediastinal lesion had increased to 16 mm and exhibited slight contrast enhancement. 18F-fluorodeoxyglucose PET/CT demonstrated only a slight uptake in the anterior mediastinal lesion with no abnormal uptake elsewhere. Thymic epithelial tumors and cysts were included in the differential diagnosis, and video-assisted thoracoscopic surgery was planned. Intraoperative findings revealed a dark-red nodule beneath the mediastinal pleura. The right internal thoracic vein was observed to flow into the nodule and an outflow vessel draining into the right brachiocephalic vein was identified. Based on these findings, the lesion was diagnosed as an internal thoracic venous aneurysm. The right internal thoracic vein and outflow vessel were ligated, and the nodule was resected. A histopathological examination confirmed the diagnosis of venous aneurysm.
CONCLUSIONS: Although extremely rare, venous aneurysms can occur in the internal thoracic vein. This condition should be considered in the differential diagnosis of enhancing solid nodules of the anterior mediastinum.
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Ken Yonemitsu, Hiroaki Kasashima, Tatsunari Fukuoka, Mami Yoshii, Akih ...
2025 年11 巻1 号 論文ID: cr.25-0308
発行日: 2025年
公開日: 2025/09/19
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INTRODUCTION: Jejunal diverticulosis is a rare condition, often asymptomatic, but it can lead to serious complications such as diverticulitis or perforation. Management of perforated jejunal diverticula is challenging, particularly in patients with autosomal dominant polycystic kidney disease (ADPKD) who have undergone kidney transplantation and are receiving immunosuppressive therapy. Early diagnosis is often difficult due to nonspecific symptoms and the frequent presence of multiple diverticula, which increases the risk of postoperative complications.
CASE PRESENTATION: We present the case of a 61-year-old woman with ADPKD, who developed jejunal diverticular perforation 3 years after undergoing ABO-incompatible kidney transplantation. She initially presented with mild abdominal pain and was managed conservatively, but her condition worsened 1 month later with evidence of free air on CT. Emergency surgery revealed multiple jejunal diverticula with a perforation on the mesenteric side, and a double-barrel stoma was created to avoid anastomotic leakage. Four months postoperatively, endoscopic and fluoroscopic evaluation from both the oral and stoma sides enabled accurate identification and marking of the diseased segment. A second-stage surgery was successfully performed with segmental jejunal resection and stoma closure. Histopathology confirmed multiple true diverticula, including at the perforation site. The patient recovered well and was discharged approximately 1.5 months later.
CONCLUSIONS: This rare case of perforated multiple jejunal diverticula in a patient with ADPKD highlights the value of a two-stage surgical approach with preoperative endoscopic evaluation to enable safe and targeted resection in complex settings.
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Yuta Takeuchi, Seiichiro Inoue, Yuki Muta, Keisuke Sawada, Taisuke Hay ...
2025 年11 巻1 号 論文ID: cr.24-0006
発行日: 2025年
公開日: 2025/09/19
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INTRODUCTION: Umbilical cord cysts detected after the 2nd trimester of pregnancy are associated with a variety of complications, including omphalocele, and thus require careful monitoring. Congenital hepatic cysts are rare, as are reports of their coexistence with omphaloceles. Herein, we present an unusual case of an omphalocele complicated by a large hepatic cyst that was initially suspected to be an umbilical cord cyst during the fetal period.
CASE PRESENTATION: A male infant was delivered via caesarean section at 36 weeks of age, with ruptured membranes and cloudy amniotic fluid. Fetal ultrasonography at 16 weeks had previously revealed an omphalocele with intestinal prolapse, and at 20 weeks, an umbilical cord cyst was suspected. At birth, the herniation sac ruptured, and abdominal wall closure and cyst excision were performed. The cyst, initially thought to be umbilical in origin, was instead identified as a hepatic cyst connected to the liver. Pathological examination confirmed the diagnosis. The infant was further diagnosed with Beckwith–Wiedemann syndrome, and was ultimately discharged 32 days postoperatively, with no recurrence to date.
CONCLUSIONS: Although asymptomatic congenital liver cysts can often be managed conservatively with follow-up, we considered surgical intervention necessary in this case due to the presence of an omphalocele associated with suspected Beckwith–Wiedemann syndrome
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Yukiko Matsui, Takahide Toyoda, Yuki Sata, Terunaga Inage, Kazuhisa Ta ...
2025 年11 巻1 号 論文ID: cr.25-0269
発行日: 2025年
公開日: 2025/09/19
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INTRODUCTION: Minimally invasive surgery has recently become the standard approach for pulmonary lesion resection, and its usefulness has been reported. However, these techniques involve small incisions, making intraoperative palpation difficult. Therefore, a simple and minimally invasive method is required to identify these lesions. Herein, we present a case of thoracoscopic lung wedge resection in which intra-thoracic ultrasonography equipped with a new technology was successfully used for lesion localization.
CASE PRESENTATION: A male patient in his 60s presented with a right intrapulmonary nodule following chemoradiotherapy for oropharyngeal cancer. Surgical resection was planned for the suspected malignancy. The procedure was a single-port thoracoscopic wedge resection of the right upper lobe. Intraoperatively, intra-thoracic ultrasonography successfully identified a pulmonary lesion, eliminating the need for palpation. Histopathological examination confirmed squamous cell carcinoma and complete resection was achieved.
CONCLUSIONS: We successfully performed a single-port thoracoscopic pulmonary wedge resection using intraoperative thoracic ultrasonography for precise lesion localization. The ultrasound devices we used were equipped with advanced technology, enabling clearer images. This case highlights the potential utility of intra-thoracic ultrasonography in minimally invasive lung surgery, as it enables accurate lesion localization and successful resection.
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Tetsuhiro Urashima, Yusaku Tanaka, Kota Imanishi, Sachika Kinoshita, S ...
2025 年11 巻1 号 論文ID: cr.25-0455
発行日: 2025年
公開日: 2025/09/19
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INTRODUCTION: Brain metastasis from esophageal cancer is rare. In particular, recurrent brain metastasis following multimodal treatment, such as preoperative chemotherapy and surgical resection, is extremely uncommon.
We present a case of early brain metastasis from esophageal cancer despite achieving pathological complete response (pCR).
CASE PRESENTATION: A middle-aged man presented with dysphagia. Upper gastrointestinal endoscopy revealed stenosis caused by an ulceroinfiltrative tumor located in the middle of the thoracic esophagus. Targeted biopsies confirmed squamous cell carcinoma. Contrast-enhanced CT revealed circumferential, irregular wall thickening with contrast enhancement, showing infiltration into the left main bronchus and enlarged lymph nodes in the paraesophageal and the left supraclavicular regions. The diagnosis was middle thoracic esophageal squamous cell carcinoma cT3br N2M0 cStage IIIB (according to the Japanese Classification of Esophageal Cancer, 12th Edition). The patient underwent chemotherapy, including 5-fluorouracil, cisplatin, and pembrolizumab, as the combined positive score exceeded 10. Following this chemotherapy with an immune checkpoint inhibitor, the tumor had regressed, and targeted biopsies revealed no malignant findings. The post-chemotherapy diagnosis was ycT3rN0M0 ycStage II, and the patient subsequently underwent thoracoscopic esophagectomy. Surgical findings showed no evidence of tumor infiltration. Postoperative histopathological examination showed no residual tumor cells in either the esophagus or resected lymph nodes, corresponding to histological response of grade 3. However, the patient presented with depression 2 months after the surgery, and abnormal behavior was shown 3 months after the surgery. Although cranial CT and MRI revealed ring-enhancing lesions in the right cerebellar hemisphere and the right frontal lobe, there was no recurrence or metastasis other than in the brain. The patient underwent resection of the frontal lobe tumor and was diagnosed with brain metastasis of esophageal cancer. Stereotactic radiation therapy and pembrolizumab were started; however, the patient died 5 months after esophagectomy due to brain metastasis progression.
CONCLUSIONS: 5-Fluorouracil and cisplatin plus pembrolizumab therapy may allow conversion surgery in advanced esophageal cancer. However, even in patients who achieve pCR at the primary lesion, brain metastasis may occur after surgical treatment. Preoperative and postoperative surveillance for brain metastases is necessary in patients at high risk of distant metastasis, even if the local lesion is controlled.
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Kotaro Kimura, Yoshihiro Kinoshita, Naoya Okada, Takumi Yamabuki, Saki ...
2025 年11 巻1 号 論文ID: cr.25-0257
発行日: 2025年
公開日: 2025/09/17
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INTRODUCTION: Sarcomatoid carcinoma is a rare histological variant of carcinoma characterized by a mesenchymal-like morphology, often arising through the epithelial-mesenchymal transition. Although sarcomatoid carcinoma is occasionally observed in primary esophageal carcinosarcoma, its diagnosis at metastatic sites is rare.
CASE PRESENTATION: A man in his 60s was diagnosed with esophageal squamous cell carcinoma with synchronous liver and cutaneous metastases. A cutaneous nodule in the abdomen was surgically resected and pathologically confirmed to be metastatic squamous cell carcinoma. Owing to the unresectable nature of the tumor, the patient underwent 10 cycles of combined pembrolizumab and fluorouracil + cisplatin therapy, followed by 20 cycles of S-1 monotherapy owing to immune-related adverse events. This treatment resulted in a complete clinical response of the primary esophageal tumor and a significant reduction in most liver metastases. However, one liver metastasis in the left lateral segment exhibited progressive disease, requiring surgical resection. Pathological examination of the hepatic lesion revealed a well-demarcated lobulated tumor with necrosis and hemorrhage. Microscopically, the tumor comprised polygonal-to-short, spindle-shaped atypical cells with pleomorphism, frequent mitotic figures, and sinusoidal infiltration. Immunohistochemically, the hepatic lesion exhibited diffuse vimentin positivity, partial Cam5.2, and scattered INSM-1 expression, suggesting a mesenchymal transition of the carcinoma with partial neuroendocrine differentiation. CK AE1/AE3, CK5/6, and p40 were negative. p53 and BRM/SMARCA2 showed a complete loss of expression. A retrospective analysis of esophageal and cutaneous lesions revealed a progressive loss of BRM/SMARCA2 expression across different metastatic sites, supporting the hypothesis of epithelial-mesenchymal transition-driven transformation during hepatic metastasis. Despite surgical intervention, multiple hepatic recurrences were detected within 2 months postoperatively, highlighting the aggressive nature of sarcomatoid carcinoma and the limitations of surgery alone in controlling this disease.
CONCLUSIONS: This case highlights the rare phenomenon of histological transformation of squamous cell carcinoma to sarcomatoid carcinoma within a metastatic site, emphasizing the importance of surgical resection for both diagnosis and treatment. The loss of BRM/SMARCA2 may have contributed to the epithelial-mesenchymal transition-driven transformation and, together with neuroendocrine differentiation, may have played a role in the tumor’s aggressiveness. Histological reassessment of chemotherapy-resistant lesions is crucial for elucidating tumor evolution and optimizing future therapeutic strategies.
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Byonggu An, Tetsuya Abe, Yasumitsu Oe, Yuki Morimoto, Yuuki Imazato, H ...
2025 年11 巻1 号 論文ID: cr.25-0246
発行日: 2025年
公開日: 2025/09/17
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INTRODUCTION: Central airway obstruction (CAO) due to malignancy is an oncological emergency that requires prompt management. On the other hand, esophagobronchial fistula (EBF) is also a serious condition that can lead to aspiration pneumonia and difficulty with oral intake. Secondary involvement due to malignant lymphoma is a very rare cause of both CAO and EBF. Here, we report a case of esophageal bypass surgery for an EBF that became apparent after palliative irradiation for left main bronchial obstruction caused by mediastinal malignant lymphoma.
CASE PRESENTATION: A 76-year-old man was referred to our hospital with a chief complaint of dyspnea and dysphagia. Contrast-enhanced chest CT revealed a 10-cm irregularly shaped space-occupying lesion (SOL) in the middle mediastinum, involving the left main bronchus and esophagus. The left main bronchus was obstructed by the SOL, and the presence of gas within the lesion suggested the formation of a fistula between the tumor and the bronchus. Bronchoscopy revealed almost complete obstruction of the left main bronchus due to an ulcerative tumor. Upper gastrointestinal endoscopy revealed a deeply excavated esophageal ulcer, raising suspicion of a fistulous connection with the tumor. Although fistula formation between the tumor and the bronchus and/or esophagus was suspected, due to the oncological emergency of CAO presumably caused by mediastinal-type lung cancer, palliative irradiation and intravenous administration of betamethasone sodium phosphate were initiated prior to obtaining a pathological diagnosis. The tumor shrank remarkably after the treatment. However, as the tumor regressed, an EBF became apparent. Given the patient’s strong desire to resume oral intake and the need for a definitive diagnosis, we planned a surgical intervention (esophageal bypass surgery with lymph node sampling). The patient’s postoperative course was uneventful. He resumed oral intake on POD 9, and was discharged on day 20. Histopathological examination of the lymph nodes revealed diffuse large B-cell lymphoma. The patient began chemotherapy for malignant lymphoma 2 months after surgery.
CONCLUSIONS: Esophageal bypass surgery is a valuable option for patients with an EBF, as it improves QOL and enables subsequent chemotherapy.
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Shinichiro Shiomi, Takashi Fukuda, Shintaro Nozu, Daiji Oka, Yusuke Yo ...
2025 年11 巻1 号 論文ID: cr.25-0217
発行日: 2025年
公開日: 2025/09/17
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INTRODUCTION: The optimal follow-up period and appropriate examinations for patients with esophageal squamous cell carcinoma (ESCC) who have remained disease-free for 5 years remain controversial.
CASE PRESENTATION: A 73-year-old man was diagnosed with ESCC and underwent curative esophagectomy. Pathological examination revealed a superficial tumor without lymph node metastases, despite lymphatic and vascular involvement. The patient underwent routine postoperative follow-up at our institution and showed no signs of recurrence until the 5th postoperative year. However, his serum p53 antibody titer increased 5 years postoperatively, and careful follow-up using imaging modalities, including CT, was scheduled. No lesions suspected of recurrence were noted over the next 2 years until bilateral pleural effusion was detected on CT in the 7th postoperative year. Cytological examination of the pleural effusion revealed pleural-seeded ESCC cells.
CONCLUSIONS: Although several cases of late recurrence (>5 years) have been previously reported, most have a deeper infiltration depth than that of T2 or pathologically positive lymph nodes. However, patients with lymphatic/vascular involvement, even those with pT1bN0 ESCC, require careful surveillance using imaging modalities and laboratory tests, given the possibility of late recurrence occurring beyond 5 years.
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Ayano Inao, Keisuke Noda, Tetsuro Tominaga, Toshio Shiraishi, Shintaro ...
2025 年11 巻1 号 論文ID: cr.25-0414
発行日: 2025年
公開日: 2025/09/13
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INTRODUCTION: Medullary carcinoma of the colon was formerly classified as poorly differentiated adenocarcinoma. The prognosis is relatively good, with a high degree of microsatellite instability and a predilection for the right colon.
CASE PRESENTATION: The mean age of the 4 patients was 69 years (range, 47–90 years), with 2 males and 2 females. Preoperative biopsy results showed 3 cases of moderately differentiated adenocarcinoma and 1 case of poorly differentiated adenocarcinoma. All cases were right-sided colon cancer, with 3 cases of ascending colon cancer and 1 case of transverse colon cancer. One case was classified as cT3, 2 as cT4a, and 1 as cT4b. One case was cN-negative, and 3 were cN-positive. No cases were cM-positive. The approach was laparoscopic in 3 cases and robot-assisted surgery in 1 case. Postoperative complications included postoperative ileus in 1 case. Pathological staging was Stage II in 3 cases and Stage III in 1 case. All cases showed high microsatellite instability, and no adjuvant therapy was administered. All patients remain under observation, with no recurrences identified at the time of writing.
CONCLUSIONS: Medullary carcinoma is rare but has been increasing in recent years. This pathology shows characteristic histological features and immunophenotypes, and accurate diagnosis is important because of its effect on postoperative follow-up.
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Keiichiro Yokota, Hiroyuki Kitagawa, Kohei Araki, Kento Shinno, Tsutom ...
2025 年11 巻1 号 論文ID: cr.25-0424
発行日: 2025年
公開日: 2025/09/12
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INTRODUCTION: Esophageal sarcomatoid carcinoma (ESC) is a rare malignancy characterized by both carcinomatous and sarcomatous components, with no established perioperative chemotherapy standards.
CASE PRESENTATION: We report a case of advanced ESC (T3N1M0, Stage III) treated with 2 courses of neoadjuvant docetaxel, cisplatin, and fluorouracil (DCF) chemotherapy, followed by robotic-assisted thoracoscopic esophagectomy. Histopathology revealed the complete disappearance of spindle cell components, although residual squamous cell carcinoma was found in the flat mucosa. The patient remained recurrence-free for 26 months postoperatively without adjuvant therapy.
CONCLUSIONS: This case suggests that preoperative DCF chemotherapy may be effective for ESC, especially when combined with complete surgical resection, highlighting the importance of addressing both the sarcomatoid and squamous components for long-term survival.
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Ayuna Sato, Yukiko Matsui, Takahide Toyoda, Yuki Sata, Terunaga Inage, ...
2025 年11 巻1 号 論文ID: cr.25-0439
発行日: 2025年
公開日: 2025/09/10
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INTRODUCTION: Multimodal treatment is required for advanced thymic epithelial tumors. However, to date, no pharmacotherapy has been established. Herein, we present 2 cases in which preoperative cisplatin, doxorubicin, and methylprednisolone (CAMP) therapies were effective.
CASE PRESENTATION: Case 1: A woman in her 70s was diagnosed with clinical stage IVA thymoma with intrathoracic dissemination. The patient received 2 cycles of induction therapy with CAMP, resulting in a partial response. Robot-assisted thoracoscopic resection of a mediastinal tumor and pleural lesions was successfully performed. Pathology revealed a type B2 thymoma (ypT1aN0M1a, stage IVA). Case 2: A woman in her 70s was diagnosed with clinical stage IIIA thymoma invading the upper lobe of the left lung. Two cycles of CAMP therapy resulted in sufficient tumor shrinkage to allow resection of only the upper division of the left lung along with resection of the thymoma, thereby avoiding lobectomy. Pathology confirmed type B2 thymoma (ypT3N0M0, stage IIIA), and adjuvant radiotherapy was administered postoperatively.
CONCLUSIONS: CAMP therapy was effective as induction chemotherapy in 2 cases of advanced thymoma, allowing for less invasive surgical approaches. Following this regimen, minimally invasive surgery was performed in one case, and the extent of pulmonary resection was reduced in the other. Therefore, this regimen may offer functional preservation and serve as a useful option for multimodal treatment.
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Ryuji Nakamura, Katsuhiro Okuda, Kensuke Iguchi, Shin Hosokawa, Emi Ha ...
2025 年11 巻1 号 論文ID: cr.25-0383
発行日: 2025年
公開日: 2025/09/09
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INTRODUCTION: Mediastinal non-seminomatous germ cell tumors (NSGCTs) are rare tumors. Neoadjuvant chemotherapy followed by complete surgical resection of residual masses is recommended, and is often performed through a median sternotomy or thoracotomy with regard to the influence of induction chemotherapy and tumor size. We herein report 2 cases of mediastinal NSGCT that were surgically resected by robot-assisted thoracoscopic surgery (RATS) using the subxiphoid approach.
CASE PRESENTATION: Case 1: A 23-year-old man was diagnosed with an anterior mediastinal mass measuring 95 × 73 × 73 mm while undergoing an examination due to fever. He was diagnosed with a yolk sac tumor based on percutaneous needle biopsy. After 4 cycles of neoadjuvant chemotherapy, the patient underwent tumor resection combined with wedge resection of the left upper lung via a robot-assisted subxiphoid approach. It was particularly difficult to identify the left phrenic nerve because of stiff adhesions and thickening of the tissue. Therefore, we decided to perform dissection of tissue other than that surrounding the left phrenic nerve. Subsequently, the thymus and tumor were flipped into the left thoracic cavity, and the left phrenic nerve was easily identified and preserved from the pericardial side. The patient was discharged without any postoperative complications. Case 2: An 18-year-old man was diagnosed with a yolk sac tumor measuring 86 × 68 × 150 mm during an examination to investigate intermittent right chest pain. After 4 cycles of neoadjuvant chemotherapy, the patient underwent anterior mediastinal tumor resection via a robot-assisted subxiphoid approach. The tumor was close to the right pulmonary hilum, and the inflammation was so intense that it was difficult to identify the right phrenic nerve. In this case, indocyanine green fluorescence imaging was helpful for identifying the right phrenic nerve. The tumor was completely resected. The patient was discharged on POD 6 without any postoperative complications.
CONCLUSIONS: We report 2 cases of mediastinal NSGCT after induction chemotherapy that were completely resected using RATS. The use of techniques to accurately identify the phrenic nerve and the advantages of robot-assisted surgery via the subxiphoid approach enabled safe and minimally invasive surgical procedures.
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Hiromitsu Imataki, Masaoki Hattori, Akihiro Hirata, Akihiro Tomida, Ju ...
2025 年11 巻1 号 論文ID: cr.25-0202
発行日: 2025年
公開日: 2025/09/09
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INTRODUCTION: Reports of fistulas in the common bile duct caused by pancreatic pseudocysts have increased. However, to the best of our knowledge, no prior report has described pseudocysts filled with microbiliary stones that were difficult to differentiate from neoplastic lesions.
CASE PRESENTATION: A 64-year-old man presented with groove pancreatitis attributable to heavy alcohol consumption and a hypovascular mass in the groove area with duodenal bleeding. The lesion, which was initially considered a pancreatic groove tumor with groove pancreatitis, was treated with subtotal stomach-preserving pancreaticoduodenectomy. However, a post-surgical pathological analysis revealed that it was a pancreatic pseudocyst in the groove area containing bilirubin calcium stones and pancreatic stones.
CONCLUSIONS: Cases comprising fistula formation in the common bile duct are rare. A pancreatic pseudocyst that formed a fistula with the common bile duct was suspected in the present case. This case was unique because the pseudocyst was filled with microbiliary stones. This report highlights the difficulty in differentiating a pseudocyst filled with bilirubin calcium stones from a neoplastic lesion and underscores the importance of the accurate diagnosis and management of this rare pathology.
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Naohiko Oki, Naritaka Kimura, Hideaki Shimizu, Hideyuki Shimizu
2025 年11 巻1 号 論文ID: cr.25-0329
発行日: 2025年
公開日: 2025/09/09
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INTRODUCTION: The main causes of right-to-left shunting (RLS) in patients with atrial septal defect (ASD) are pulmonary hypertension, right ventricular outflow tract obstruction, severe tricuspid regurgitation, and a large ASD resulting in equal bi-atrial pressures. Reports of a case of an unintentional connection of the inferior vena cava (IVC) to the left atrium (LA) discovered many years after the repair of ASD are rare.
CASE PRESENTATION: A 47-year-old male with a history of congenital ASD repair was found to have large RLS during examination of choledocholithiasis. Details of the former surgeries, performed twice for some reason, were unknown. He had cyanosis of fingers, but neither pulmonary hypertension nor right ventricular outflow obstruction. Transesophageal echocardiography, cardiac CT, and cardiac catheterization demonstrated a direct connection between the IVC and the LA as well as a residual ASD. The patient subsequently underwent successful surgical repair. It was speculated that the eustachian valve (EV) of the IVC had been wrongly taken as the lower margin of the defect in the first repair, and then a new ASD was created in the second surgery to maintain his hemodynamics. Though he had RLS with ASD, he subsequently lived a normal life for approximately 40 years, albeit with exertional dyspnea and mild cyanosis.
CONCLUSIONS: We report on a case of iatrogenic RLS after ASD closure. The surgeon must always check the intracardiac anatomy carefully and close the ASD without using other structures such as the EV.
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Sota Nakamura, Shohei Yoshiya, Kensaku Ito, Noriaki Yamamoto, Ippei Ka ...
2025 年11 巻1 号 論文ID: cr.25-0280
発行日: 2025年
公開日: 2025/09/06
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INTRODUCTION: Pleuroperitoneal communication (PPC) is an abnormal connection between the thoracic and abdominal cavities, allowing ascitic fluid to migrate into the pleural space and cause pleural effusion. Although PPC is a recognized complication in peritoneal dialysis patients, it is rarely reported as a postoperative complication.
CASE PRESENTATION: A 54-year-old female patient with a history of right hemicolectomy and right hepatectomy for ascending colon cancer and metastatic liver tumor developed persistent right pleural effusion 3 months postoperatively. She also had ovarian metastases, peritoneal dissemination, and malignant ascites. Despite treatment with diuretics and 2 attempts at pleurodesis, the pleural effusion persisted. A combined thoracoscopic and laparoscopic approach was performed. Intraoperatively, a diaphragmatic hernia with a pinhole defect was identified, suggesting migration of ascitic fluid into the thoracic cavity. Resection of the hernia sac and suture closure of the diaphragmatic defect were carried out. Postoperatively, the pleural effusion resolved, and her dyspnea improved.
CONCLUSIONS: In case of refractory pleural effusion following hepatectomy, PPC due to diaphragmatic defects should be considered, particularly in the presence of ascites. A combined thoracoscopic and laparoscopic approach is a viable surgical option for definitive treatment.
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Takeshi Kano, Ryugen Takahashi, Nobuhisa Akamatsu, Yujiro Nishioka, Yu ...
2025 年11 巻1 号 論文ID: cr.25-0264
発行日: 2025年
公開日: 2025/09/06
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INTRODUCTION: Steatotic liver disease (SLD) may develop in some patients after pancreaticoduodenectomy (PD), but no cases requiring liver transplantation (LT) have been reported to date. Here, we present two cases in which LT was performed for decompensated liver cirrhosis (LC) after PD.
CASE PRESENTATION: Case 1 was a 53-year-old man with obesity, metabolic-associated SLD (MASLD), and diabetes mellitus. The patient underwent PD for an intraductal papillary mucinous neoplasm. His liver function worsened and he developed decompensated LC 6 years later, eventually requiring LT. Due to poor mobility of the jejunal limb caused by severe adhesions and the presence of a pancreatojejunostomy, a choledochojejunostomy was performed at the more distal site of the common bile duct than usual. He developed hemobilia and biliary leakage but was discharged on POD 107. Liver function has been good for 2 years after LT without MASLD recurrence, although endoscopic treatment is periodically required for biliary stricture. Case 2 was a 46-year-old man with obesity, SLD, and a history of excessive alcohol consumption. The patient underwent PD for duodenal cancer. Five years later, he developed decompensated LC, which required living-donor LT. For biliary reconstruction, a new jejunal limb was created and elevated. He was discharged on POD 79. He has repeatedly developed cholangitis, but his liver function has been good for 6 years without SLD recurrence.
CONCLUSIONS: Steatohepatitis can worsen following PD and may lead to decompensated LC, ultimately requiring LT. Therefore, screening for steatohepatitis and its risk factors prior to PD is essential, and prophylaxis should be considered. LT after PD presents surgical challenges and biliary reconstruction with some procedural modifications.
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Naoto Nakamura, Yosuke Kasai, Kazuyuki Nagai, Asahi Sato, Kentaro Kado ...
2025 年11 巻1 号 論文ID: cr.25-0247
発行日: 2025年
公開日: 2025/09/05
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INTRODUCTION: von Hippel-Lindau (VHL) disease is an autosomal dominant hereditary disorder characterized by the development of tumor-like lesions in multiple organs. While central nervous system hemangioblastomas, pancreatic neuroendocrine tumors, and pancreatic cysts are commonly associated with VHL disease, there have been few reported cases of pancreatic hemangioblastoma in patients with VHL disease.
CASE PRESENTATION: A male patient in his 30s had been diagnosed with VHL disease and had been followed for cerebellar and spinal hemangioblastomas, and renal cell carcinoma, for which he had undergone several tumor resections, radiation therapy, and a ventriculoperitoneal shunt. A pancreatic head tumor deemed to be a neuroendocrine tumor on imaging findings exhibited a gradual increase in size from 12 to 33 mm for the past 2 years, but it had been monitored due to his comorbidities and declining daily living activities. Severe anemia was detected during his regular outpatient visit, and an emergency esophagogastroduodenoscopy revealed a submucosal tumor near the duodenal papilla with ulceration and active bleeding, making endoscopic hemostasis challenging. Dynamic contrast-enhanced CT showed active bleeding from the pancreatic tumor. Subsequently, emergency angiography was performed via the superior mesenteric artery, successfully embolizing vessels supplied by the inferior pancreaticoduodenal artery to achieve hemostasis. Due to concerns about rebleeding, we performed pancreaticoduodenectomy 1 month after the emergency angiography, during which we awaited the improvement of the patient’s overall condition. Microscopic findings of the tumor showed multinodular proliferation with hematoxylin-eosin staining, revealing cells with clear cytoplasm and abundant capillaries and dilated branching vessels within the nests. Immunohistochemical analysis demonstrated positivity for alpha-inhibin and S100, with partial positivity for carbonic anhydrase IX, leading to a diagnosis of pancreatic hemangioblastoma.
CONCLUSIONS: This paper reports a rare case of pancreatic hemangioblastoma arising in a patient with VHL disease. It is crucial to consider the possibility of pancreatic hemangioblastoma when treating pancreatic tumors in VHL disease patients.
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Tadakazu Ao, Eiji Shinto, Tenma Ichisawa, Koki Ichio, Takafumi Suzuki, ...
2025 年11 巻1 号 論文ID: cr.25-0314
発行日: 2025年
公開日: 2025/09/04
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INTRODUCTION: Solitary fibrous tumor (SFT) is a rare mesenchymal neoplasm that most commonly originates in the pleura but can also occur at extrapleural sites, including the abdominal cavity. Among these, primary SFT of the stomach is exceptionally rare. Due to overlapping clinical, endoscopic, and radiologic characteristics, distinguishing SFT from gastrointestinal stromal tumor (GIST) can be particularly challenging. Misdiagnosis may result in inappropriate treatment, such as unnecessary administration of imatinib. Therefore, accurate preoperative differentiation is essential for appropriate management.
CASE PRESENTATION: A 74-year-old man was incidentally found to have a submucosal gastric tumor during a routine health checkup and was referred to our hospital for further evaluation and treatment. Upper gastrointestinal endoscopy revealed a 30-mm subepithelial lesion on the greater curvature of the gastric fundus. Endoscopic ultrasound-guided fine-needle aspiration (EUS-FNA) demonstrated isolated and clustered cells with ovoid to spindle-shaped nuclei. Although not definitive, the combined endoscopic and cytological findings led to a preoperative diagnosis of suspected GIST, and laparoscopy and endoscopy cooperative surgery (LECS) was subsequently performed for local resection. Histopathological examination of the resected specimen revealed an irregular proliferation of spindle cells and nuclear immunopositivity for signal transducer and activator of transcription 6 (STAT6), leading to a final diagnosis of SFT of the stomach. According to Demicco’s risk stratification model, the tumor was classified as low risk. The patient underwent complete resection via LECS and has remained free of recurrence for more than 2.5 years postoperatively.
CONCLUSIONS: This case highlights the difficulty in differentiating SFT from GIST preoperatively and underscores the importance of obtaining sufficient tissue samples to allow for immunohistochemical analysis, particularly STAT6 staining. Recognizing gastric SFT as part of the differential diagnosis is critical to avoid misdiagnosis and ensure appropriate therapeutic decision-making.
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Koto Kawata, Hajime Otsu, Qingjiang Hu, Yasuo Tsuda, Yoshihiro Nagao, ...
2025 年11 巻1 号 論文ID: cr.25-0239
発行日: 2025年
公開日: 2025/09/04
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INTRODUCTION: Glycogen storage disease type IX (GSD type IX) is caused by a deficiency in phosphorylase b kinase (PHK) and is classified into hepatic (IXa–c) and muscular (IXd) subtypes. GSD type IXd leads to exercise intolerance, rhabdomyolysis, and myoglobinuria owing to impaired glycogen breakdown. It is a rare and mild metabolic disorder, with only 19 reported cases of PHKA1 mutations. To the best of our knowledge, this is the 1st report on the perioperative management of a patient with GSD type IXd.
CASE PRESENTATION: A 61-year-old male presented with a right inguinal hernia requiring surgical repair. He had experienced muscle weakness since the age of 53, which progressed to severe neck muscle atrophy by the age of 58. Genetic testing confirmed a PHKA1 mutation, leading to the diagnosis of GSD type IXd. He had previously undergone multiple surgeries without any complications. Given his underlying muscle weakness, totally extraperitoneal (TEP) inguinal hernia repair was performed to minimize postoperative pain and muscle damage. Postoperative monitoring revealed no rhabdomyolysis or myoglobinuria, and the patient was discharged without complications on POD 7.
CONCLUSIONS: We successfully managed a patient with GSD type IXd perioperatively, without complications. Although this disease can cause rhabdomyolysis, the symptoms are often mild and may remain undiagnosed. Therefore, in patients with muscle weakness or elevated creatine kinase levels, careful surgical planning and perioperative monitoring are essential.
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Daiki Mori, Yusuke Ozaki, Dai Fukushima, Kohei Yamao, Ryosuke Usui, Hi ...
2025 年11 巻1 号 論文ID: cr.25-0326
発行日: 2025年
公開日: 2025/09/03
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INTRODUCTION: Thoracic vertebral fractures are commonly associated with aortic injury at the time of injury. However, delayed thoracic aortic injury during intensive care management of thoracic fractures has not been reported.
CASE PRESENTATION: An 83-year-old man was transported to our hospital after a collision with a car while riding a motorized bicycle. A fracture of the 10th thoracic vertebra was identified and treated with thoracic fusion on day 24. On day 34, a new pseudoaneurysm was found in contact with the vertebral fracture. Thoracic endovascular aortic repair (TEVAR) was performed on day 36, and disappearance of the pseudoaneurysm was confirmed on day 38.
CONCLUSIONS: The possibility of delayed thoracic aortic pseudoaneurysm should be considered when thoracic vertebral fractures are near the descending aorta. Early TEVAR may be effective for delayed aortic injury.
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Takeshi Miwa, Tomoyuki Okumura, Yoshihisa Numata, Mina Fukasawa, Nana ...
2025 年11 巻1 号 論文ID: cr.25-0204
発行日: 2025年
公開日: 2025/09/03
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INTRODUCTION: There are no reports of patients undergoing McKeown esophagectomy for esophageal cancer after undergoing pancreaticoduodenectomy for pancreatic cancer. We report the case of a patient who underwent subtotal esophagectomy and colon reconstruction after pancreaticoduodenectomy using the mesenteric approach.
CASE PRESENTATION: A 71-year-old male was diagnosed with advanced esophageal cancer. Four years prior to diagnosis, he underwent subtotal stomach-preserving pancreaticoduodenectomy using the mesenteric approach for pancreatic surgery, followed by Child’s reconstruction surgery. After undergoing 3 cycles of neoadjuvant chemotherapy with docetaxel, cisplatin, and 5-fluorouracil, the patient was scheduled for a subtotal esophagectomy. The middle colic artery was transected using the mesenteric approach, and the upper jejunum was utilized for Child’s reconstruction surgery. A 2-stage procedure involving McKeown esophagectomy and left-sided colon reconstruction was planned. The 1st stage of the procedure involved robot-assisted subtotal esophagostomy in the prone position, followed by cervical esophagostomy and gastrostomy. The patient underwent the 2nd stage of the surgery after approximately 1 month of parenteral nutrition via a gastrostomy tube. The transverse colon was mobilized and transected at the hepatic flexure. The left side of the mesocolon, which is fed by the left colic artery, was then pulled up through the antethoracic route. The right internal thoracic artery and vein were anastomosed to the marginal artery and vein of the transverse colon, respectively, for supercharge and superdrainage. Reconstruction involved esophago-colonic and colonic–gastric anastomoses. The patient was discharged without postoperative complications, and no signs of recurrence were observed at the 2-year postoperative follow-up.
CONCLUSIONS: Subtotal esophagectomy for esophageal cancer after subtotal stomach-preserving pancreaticoduodenectomy using a mesenteric approach and colon reconstruction can be safely performed in 2 stages. The optimization of pancreaticoduodenectomy for pancreatic cancer could improve the long-term survival of patients with 2nd primary esophageal cancer, for which radical esophagectomy is necessary.
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Takuro Fujita, Akihiko Soyama, Taiichiro Kosaka, Takanobu Hara, Hajime ...
2025 年11 巻1 号 論文ID: cr.25-0277
発行日: 2025年
公開日: 2025/09/03
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INTRODUCTION: Osler–Weber–Rendu syndrome, or hereditary hemorrhagic telangiectasia (HHT), is a rare autosomal dominant genetic vascular disease characterized by arteriovenous malformations, vascular wall fragility, dilatation, and rupture of the vessels with hepatic symptoms. As HHT with hepatic symptoms is recognized as the primary etiology for liver transplantation, liver transplantation with liver grafts from donors affected by HHT is extremely rare. Herein, we report a successful liver transplantation in a patient with biliary atresia who received a whole-liver graft from a young brain-dead donor with HHT.
CASE PRESENTATION: The patient was a 15-year-old girl with decompensated liver cirrhosis who underwent Kasai surgery for biliary atresia at 3 months of age. The donor was a female in her teens, diagnosed with brain death due to cerebral hemorrhage. Although the donor was diagnosed with Osler disease, she had no hepatic symptoms and normal liver function. CT did not reveal any apparent vascular malformations in the liver. A whole-liver transplant was performed using the donated liver. The patient recovered well in terms of liver function, without any hepatic-related symptoms.
CONCLUSIONS: Our experience with this patient may have important implications for liver transplantations from donors with HHT.
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Masaya Matsumoto, Kojiro Eto, Satoshi Ida, Hiroki Tsubakihara, Keisuke ...
2025 年11 巻1 号 論文ID: cr.25-0306
発行日: 2025年
公開日: 2025/09/02
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INTRODUCTION: Brain metastasis from gastric cancer is rare (0.5%) and often occurs with metastasis to other organs. We herein describe a very rare patient with a solitary brain metastasis from residual gastric cancer with no metastasis to other organs.
CASE PRESENTATION: The patient was an 82-year-old man who visited a previous institution for anemia. Upper gastrointestinal endoscopy revealed a type 2 tumor extending from the esophagogastric junction to the upper part of the residual gastric body. Biopsy revealed tubular differentiated adenocarcinoma, and he was referred to our institution. He had a history of distal gastrectomy for a gastric ulcer in his 30s. After contrast-enhanced CT, we diagnosed residual gastric cancer (cT4aN + M0 cStage III). After three courses of preoperative chemotherapy with S-1 plus oxaliplatin, the patient underwent open total resection of the residual stomach, lower esophagectomy, D2 dissection, and Roux-en-Y reconstruction and was discharged without postoperative complications. Six months after surgery, thoracic and abdominal contrast-enhanced CT showed no apparent recurrence. However, 1 month later, he began to experience speech difficulties and mobility issues, and head CT revealed a 3-cm tumor in the left frontal lobe. After whole-body contrast-enhanced CT and PET-CT, the brain tumor was confirmed as a solitary lesion with no metastasis to other organs. The patient underwent open brain tumor resection, and pathology diagnosed brain metastasis from residual gastric cancer. Postoperatively, he underwent radiation therapy (40 Gy in 8 fractions) to the tumor cavity. At the time of writing, 24 months have passed since the gastrectomy and 16 months have passed since the removal of the brain tumor, with no significant neurological damage or other evidence of distant metastasis.
CONCLUSIONS: We experienced an extremely rare case of a solitary brain metastasis after residual gastric cancer surgery. Our findings suggest that aggressive local treatments for brain metastasis, including surgical resection and radiotherapy, may contribute to improvements in symptoms and prognosis.
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Satoshi Takada, Shinichi Nakanuma, Renta Kobori, Takahiro Araki, Kazuk ...
2025 年11 巻1 号 論文ID: cr.25-0341
発行日: 2025年
公開日: 2025/09/02
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INTRODUCTION: Liver transplantation for polycystic liver disease (PLD) poses significant intraoperative risks due to the presence of a massively enlarged liver. We report a rare case of intraoperative pneumothorax and pneumatocele formation during total hepatectomy, which was successfully managed with a non-operative approach.
CASE PRESENTATION: A female patient in her 40s with a history of autosomal dominant polycystic kidney disease presented with progressive liver cyst enlargement (Gigot type III, Qian classification Grade 4), which led to decreased activities of daily living and intracystic hemorrhage. The patient underwent a deceased-donor liver transplantation. During mobilization of the liver from the right side of the diaphragm, the patient experienced sudden onset of pneumothorax. Incision of the diaphragm revealed a cystic structure containing a hematoma, suggesting pneumatocele formation. The pneumatocele was not resected during the ongoing operation; instead, thoracic drainage was performed as the primary intervention. Postoperatively, no air leakage was observed, and the thoracic drain was successfully removed on POD 12. The pneumatocele, which measured approximately 10 × 10 × 7 cm, showed no signs of infection, and was monitored without additional surgical intervention. On POD 19, a fever prompted further evaluation, and CT-guided cyst aspiration for culture was performed, which revealed no evidence of infection. Acute T-cell-mediated rejection was observed on POD 27, and a steroid pulse was administered, but even after that, the pneumatocele gradually decreased in size without any signs of infection.
CONCLUSIONS: A pneumatocele is an uncommon but important consideration during liver transplantation for PLD, potentially resulting from barotrauma related to abrupt changes in intrathoracic pressure during hepatectomy and mechanical ventilation. Considering the risk of infection in immunosuppressed patients, close monitoring is essential. On the contrary, surgical resection also carries the risk of pulmonary or bronchial fistulae; therefore, careful consideration is required. This case demonstrates that non-operative management with careful observation can be an effective strategy in selected patients.
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Taiki Matsuoka, Ikuko Shibasaki, Shunsuke Saito, Yusuke Takei, Hirotsu ...
2025 年11 巻1 号 論文ID: cr.25-0316
発行日: 2025年
公開日: 2025/08/30
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INTRODUCTION: Prosthetic valve endocarditis following aortic root replacement (ARR) typically necessitates redo-ARR, which involves complete graft removal, extensive aortic root dissection, and coronary reimplantation. This highly invasive procedure carries substantial surgical risk, including high operative mortality. In select high-risk patients without evidence of prosthetic graft infection, alternative surgical strategies may reduce procedural complexity and improve outcomes.
CASE PRESENTATION: Here, we report 3 cases of prosthetic valve endocarditis following the Bentall procedure, a common ARR technique, in older patients (mean age: 73.7 ± 3.5 years). All preoperative blood cultures were negative, and no signs of prosthetic graft infection were noted on CT. Due to advanced frailty (Clinical Frailty Scale scores of 7 or 8), conventional redo-ARR was deemed prohibitively high-risk. Risk assessment using the JapanSCORE showed a mean predicted mortality of 32.5% ± 21.0%, with combined mortality and morbidity of 63.7% ± 22.9%. Instead of redo-ARR, annular reconstruction using a bovine pericardial patch was performed, followed by redo aortic valve replacement. All patients underwent successful surgery with no postoperative reinfection. One patient required prolonged intensive care and was transferred to another facility for rehabilitation, while the other 2 recovered uneventfully and were discharged. During a mean follow-up of 26.3 ± 17.6 months, 2 patients died due to non-cardiac causes: one from pneumonia and the other from gastric cancer.
CONCLUSIONS: In high-risk patients without clear evidence of prosthetic graft infection, aortic valve replacement with annular patch reconstruction may represent a viable alternative to redo-ARR, particularly in settings where homografts are not readily available. This approach reduces operative complexity while maintaining structural integrity. Further studies are warranted to validate infection control criteria and assess long-term outcomes.
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Masaya Yamasaki, Yasuaki Kubouchi, Toho Wada, Wakako Fujiwara, Shinji ...
2025 年11 巻1 号 論文ID: cr.25-0428
発行日: 2025年
公開日: 2025/08/28
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INTRODUCTION: Pulmonary smooth muscle hyperplasia (SMH) is a rare benign tumor that presents CT imaging findings that require differentiation from those of primary lung cancer.
CASE PRESENTATION: The postoperative follow-up chest CT for gastric cancer in a 76-year-old Japanese man revealed an abnormal shadow. A 2.2-cm nodule with an unclear border and showing a tendency to grow was detected in the right lower lobe (S6), and suspected infiltration into the right upper lobe (S2). PET showed minimal accumulation of 18F-fluorodeoxyglucose in the nodule, with a maximum standardized uptake value of 1.0. A transbronchial lung biopsy showed no malignant findings. Due to the tumor’s progressive growth, surgical resection was performed. Intraoperatively, a tumor located in S6 with suspected partial invasion into S2 was observed, and a wedge resection from S6 to S2 was thus performed. A frozen section of the resected specimen revealed irregularly distributed atypical cells forming mildly irregular glandular structures, leading to a diagnosis of “suspected adenocarcinoma.” Robotic-assisted thoracoscopic surgery for a right S6 segmentectomy with combined wedge resection of S2 was performed. However, the final histopathological examination revealed spindle-shaped smooth muscle cells’ proliferation. The immunohistochemical analysis revealed positivity for α-SMA, desmin, and h-caldesmon, leading to a diagnosis of pulmonary SMH.
CONCLUSIONS: SMH is an extremely rare benign disease that can mimic lung cancer and may be considered among the possible differential diagnoses of solitary pulmonary nodules. A careful treatment strategy, including the choice of surgical procedure, is recommended to minimize the possibility of overtreatment.
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Yuki Hashimoto, Shinsuke Kikuchi, Yuya Tamaru, Tsutomu Doita, Keisuke ...
2025 年11 巻1 号 論文ID: cr.25-0231
発行日: 2025年
公開日: 2025/08/28
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INTRODUCTION: Abdominal compartment syndrome (ACS) is a serious complication that can occur after endovascular aneurysm repair (EVAR) for ruptured abdominal aortic aneurysm (rAAA). Prompt recognition and appropriate management are crucial to improve patient outcomes.
CASE PRESENTATION: An octogenarian with an 11-cm rAAA underwent emergent EVAR due to cardiovascular instability. Postoperatively, the patient developed ACS, necessitating open abdominal management (OAM) due to intestinal edema and retroperitoneal hematoma. Despite multiple surgical interventions, including aneurysmorrhaphy and removal of retroperitoneal hematoma, the patient experienced prolonged difficulty in abdominal closure. The following procedures were attempted for abdominal closure: 1) Dissection of skin and subcutaneous tissues from the rectus sheath on both sides. 2) Release of the external oblique muscle from the anterior layer of the rectus by longitudinally cutting the superficial fascia. 3) Closure of both rectus sheaths with horizontal mattress sutures and negative pressure wound therapy (NPWT). On POD 20, fluid accumulation from bleeding complicated abdominal closure. However, the abdominal wall was successfully closed by achieving hemostasis and using NPWT. Despite these efforts, the patient developed multiple organ failure, including respiratory and renal failure. Sepsis ultimately led to the patient's death on the 80th POD.
CONCLUSIONS: ACS following EVAR for rAAA significantly impacts patient prognosis. Specific techniques for abdominal closure, as described in this case, may help minimize the duration of OAM in challenging cases.
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Kimihiko Nakamura, Toru Inoue, Kazuhiro Ohya
2025 年11 巻1 号 論文ID: cr.25-0301
発行日: 2025年
公開日: 2025/08/26
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INTRODUCTION: The most common etiology of large bowel obstruction (LBO) is colorectal cancer. However, extrinsic compression may occur from cancer of other organs. Plasmacytoid urothelial carcinoma (PUC) is a rare subtype of urothelial carcinoma that can present aggressively as an intraperitoneal spread. This report describes a case of LBO caused by PUC, which initially presented a diagnostic challenge.
CASE PRESENTATION: A 68-year-old woman presented with hematochezia and abdominal pain. She had a history of recurrent bladder cancer, treated with chemotherapy and multiple transurethral resections of bladder tumor (TURBT). The third TURBT revealed invasive papillary urothelial carcinoma (UC; ≥pT2) with partial positivity suggestive of a plasmacytoid or lymphoma-like subtype, while the fourth showed noninvasive UC. CT revealed wall thickening and obstruction of the transverse colon near the hepatic flexure without enlargement of the lymph nodes. Colonoscopy showed luminal narrowing caused by edematous wall thickening, suggesting extrinsic compression. To establish a diagnosis, the patient underwent diagnostic laparoscopy, which revealed that the right side of the transverse colon and the greater omentum were firmly compressed together and immobile, with extensive peritoneal dissemination. An ileo-transverse colon bypass and excisional biopsy of the disseminated nodules were performed, leading to the diagnosis of PUC. Although the patient’s postoperative course was initially satisfactory and oral intake was resumed, duodenal constriction caused by tumor invasion subsequently developed, necessitating the initiation of parenteral nutrition. After undergoing 2 courses of pembrolizumab, pulmonary edema with pneumonia occurred. Despite best supportive care, the patient died 69 days after the operation.
CONCLUSIONS: PUC is a rare and aggressive subtype of bladder cancer that frequently metastasizes, including to the peritoneum, which may lead to LBO. Although colorectal cancer is the most common cause of LBO, considering alternative differential diagnoses and planning appropriate subsequent management are essential in such cases.
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Kotaro Suehiro, Hiroyuki Takasu, Seiko Fujino, Takasuke Harada, Makoto ...
2025 年11 巻1 号 論文ID: cr.25-0375
発行日: 2025年
公開日: 2025/08/26
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INTRODUCTION: Lymphedema is generally managed with conservative therapy. However, in cases of severe fibrosclerotic lymphedema, debulking surgery is required, although rarely. We present a case of massive lymphedema in the left calf complicated by severe skin fibrosclerosis that was successfully managed with debulking surgery.
CASE PRESENTATION: A 58-year-old woman presented to our clinic with bilateral leg swelling, which was particularly massive in the left calf. She could hardly walk independently and experienced cellulitis 2 to 4 times a year. The patient was admitted, and aggressive decongestion with compression therapy was attempted initially. However, this was unsuccessful due to severe skin hardening caused by abnormal dermal thickening. We then performed partial subcutaneous tissue resection and wrapping with the redundant skin, but this resulted in extensive skin necrosis. Finally, resection of the whole skin and subcutaneous tissue down to the deep fascia in the left calf was performed, followed by split-thickness skin grafting harvested from the left thigh. At present, one year after the surgery, the patient is capable of performing light exercise and has not experienced a recurrence of cellulitis.
CONCLUSIONS: When preoperative conservative therapy is unsuccessful due to severe skin fibrosclerosis, earlier surgical intervention, including debulking, is beneficial in the management of massive lymphedema.
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Yoshifumi Shimada, Takahiro Homma, Keitaro Tanabe, Tomoshi Tsuchiya
2025 年11 巻1 号 論文ID: cr.25-0409
発行日: 2025年
公開日: 2025/08/26
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INTRODUCTION: Lung cancer with chest wall (CW) involvement can develop metastases directly to the ipsilateral axillary lymph node (ALN) via lymphatic flow of the CW. Such metastatic ALNs should be evaluated as locoregional metastases, and multimodal treatment of patients with stage III lung cancer including surgery may be utilized.
CASE PRESENTATION: A 59-year-old man presented with a chief complaint of back pain and was diagnosed as having primary lung adenocarcinoma of the right upper lobe with CW involvement and an ipsilateral ALN metastasis (cT3N0M1b: IVA, 8th edition of the tumor, node, metastasis). We found no mediastinal lymph node (MLN) metastases, so we believed that the metastatic ALN involved metastasis directly from the primary lesion via lymphatic flow of the CW. Therefore, radical surgery after neoadjuvant chemoradiotherapy was chosen as the treatment. During the operation, we performed a right upper lobectomy combined with resection of the involved CW through a posterolateral incision. The right upper limb was then raised, and the scapula was displaced backward, which allowed us to dissect the right ALN.
CONCLUSIONS: Because complete resection can be achieved through intraoperative repositioning of the upper limb, surgical treatment may be utilized for patients with locally advanced lung cancer who have CW involvement and ipsilateral ALN metastasis when the ipsilateral ALN metastasis is believed to have developed from the involved CW rather than from the MLN.
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Takuma Mikami, Daisuke Akashi, Chikara Shiiku
2025 年11 巻1 号 論文ID: cr.25-0014
発行日: 2025年
公開日: 2025/08/26
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INTRODUCTION: There are many reports of late open conversion after endovascular aortic repair (EVAR). Herein, we report the case of an octogenarian patient with a giant ovarian tumor who underwent ovarian tumor resection and open conversion with graft replacement simultaneously via laparotomy.
CASE PRESENTATION: An 86-year-old woman underwent EVAR 7 years ago. After surgery, persistent type II endoleaks from the lumbar arteries were detected. The diameter of the aneurysmal sac gradually increased. Although we attempted to perform coil embolization of the lumbar arteries one year ago, the procedure was unsuccessful, and type II endoleaks persisted, further dilating the aneurysmal sac. One month earlier, the patient experienced lower abdominal pain, which was thought to be a symptom of an ovarian tumor. Surgery to remove the ovarian tumor was considered. Although open conversion was considered, laparotomy was difficult due to the presence of the giant ovarian tumor. Therefore, we decided to perform open conversion with aortic graft replacement in addition to salpingo-oophorectomy. Surgery was performed via a median laparotomy, and graft replacement of the infrarenal abdominal aortic aneurysm was performed after salpingo-oophorectomy. The postoperative course of the patient was uneventful, and she was discharged.
CONCLUSIONS: We performed a salpingo-oophorectomy and late open conversion with graft replacement after EVAR in an octogenarian patient with a giant ovarian tumor and achieved favorable results.
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Shin-nosuke Watanabe, Daisuke Kimura, Kengo Tani, Takahiro Sasaki, Shu ...
2025 年11 巻1 号 論文ID: cr.25-0150
発行日: 2025年
公開日: 2025/08/22
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INTRODUCTION: Neurogenic tumors commonly develop in the posterior mediastinum in both pediatric and adult patients. In patients with neurofibromatosis type 1, distinguishing benign schwannomas from malignant peripheral nerve sheath tumors is challenging. In this study, we aimed to present the surgical management of a giant schwannoma that required differentiation from a malignant peripheral nerve sheath tumor.
CASE PRESENTATION: A 15-year-old boy presented with a large right mediastinal mass discovered on chest radiography at high school admission. Subsequent contrast-enhanced CT of the chest showed the development of a large tumor (16.0 × 12.5 × 11.8 cm) occupying approximately two-thirds of the right thoracic cavity, with atelectasis of the lower lobe of the right lung. The patient was histopathologically diagnosed with a benign schwannoma associated with neurofibromatosis type 1 through a thoracoscopic biopsy of the tumor and had received oral selumetinib at 50–70 mg/day for 11 months. Surgical excision was performed because of tumor progression and suspected malignant transformation. Right posterolateral thoracotomy with the opening of the 6th intercostal space was performed by extending the anterior skin incision along the abdominal rectus muscle and separating the 6th costal cartilage and diaphragmatic margin along the chest wall. The tumor was completely removed by resecting numerous adhesions between the tumor and the surrounding tissues and coagulating several nutrient vessels that flowed into the tumor, while resecting the lower lobe of the lung. The postoperative course was uneventful. The pathological examination revealed no malignancy. Subsequent contrast-enhanced CT of the chest revealed no residual tumors.
CONCLUSIONS: Posterolateral thoracotomy with the separation of the costal cartilage and diaphragmatic margin along the chest wall could achieve safe surgery for a giant mediastinal schwannoma.
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Kozue Matsuishi, Taisuke Yagi, Ryota Omura, Tomo Horinouchi, Toshihiko ...
2025 年11 巻1 号 論文ID: cr.25-0268
発行日: 2025年
公開日: 2025/08/22
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INTRODUCTION: Gastric antral vascular ectasia (GAVE) is a condition characterized by clusters of dilated capillaries in the gastric antrum, leading to gastrointestinal bleeding. Although massive hemorrhage is rare, some cases present with recurrent minor bleeding, which can make endoscopic hemostasis challenging. Here, we report a case of GAVE that was refractory to argon plasma coagulation (APC) and required surgical intervention.
CASE PRESENTATION: A 74-year-old man with end-stage renal disease who was on hemodialysis was referred to our hospital for evaluation of refractory anemia. Upper gastrointestinal endoscopy revealed the characteristic “watermelon stomach” appearance, with radially and longitudinally distributed dilated capillaries in the gastric antrum, leading to a diagnosis of GAVE. Despite undergoing APC 4 times, his anemia persisted. Given the refractory nature of the condition, surgical intervention was considered. To preserve the stomach, we initially performed laparoscopic gastric antral devascularization to reduce the blood flow to the affected area. Intraoperatively, dilated marginal vessels were observed along the greater curvature of the gastric antrum. The marginal artery was ligated along the greater curvature from the watershed area to the pylorus and pyloric ring. Indocyanine green (ICG) fluorescence imaging revealed delayed enhancement in the marginal artery resection area, indicating reduced perfusion. However, after a 2-month postoperative observation period, no improvement in the anemia was observed, and follow-up endoscopy revealed no significant changes in the gastric antral lesions. Consequently, a laparoscopic distal gastrectomy was performed. Following the procedure, the anemia stabilized, and the postoperative course was uneventful.
CONCLUSIONS: Gastric antral devascularization was ineffective for the treatment of GAVE, even when combined with ICG blood flow assessment. For refractory GAVE unresponsive to endoscopic therapy, a distal gastrectomy appears to be the most effective treatment.
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Makoto Shinohara, Takuya Yano, Manabu Shimomura, Hiroshi Okuda, Shinta ...
2025 年11 巻1 号 論文ID: cr.25-0241
発行日: 2025年
公開日: 2025/08/21
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INTRODUCTION: Surgical site infection (SSI)-associated wound dehiscence offers management challenges, often requiring frequent and prolonged wound care to achieve healing. Dehiscence may result in evisceration, requiring careful attention to infection management and organ protection, leading to extended hospitalization, poor cosmesis, increased costs, and higher risks of incisional hernias, all of which reduce patient satisfaction. Herein, we outline 3 cases in which the combination of negative pressure wound therapy with instillation and dwell time (NPWTi-d) and deep cavity wound dressing and protective agent (Sorbact) enabled safe and early wound healing.
CASE PRESENTATION: Case 1: A 76-year-old man underwent open ileocecal resection for bowel obstruction secondary to cecal cancer. On POD 3, an SSI was observed, and on POD 6, bowel evisceration resulting from wound dehiscence occurred, necessitating reoperation for suture closure. NPWTi-d was initiated 2 days after reoperation. On POD 9, extensive necrotic tissue was observed at the wound base, and Sorbact was applied while continuing NPWTi-d. On POD 34, favorable granulation tissue formation was noted, and skin closure was performed. Case 2: A 94-year-old man underwent a Hartmann procedure for a sigmoid colonic perforation. On POD 10, wound erythema and purulent discharge were noted, leading to wound opening. As a result, NPWTi-d was initiated. Partial fascial dehiscence with bowel exposure was observed on POD 19. Extensive necrotic tissue was present; thus, Sorbact was applied, and NPWTi-d was continued. On POD 38, granulation tissue formation was deemed favorable, and NPWT was discontinued. Case 3: A 77-year-old man underwent a Hartmann procedure for rectal cancer perforation. On POD 9, an SSI was noted, and wound irrigation was initiated. On POD 13, partial fascial dehiscence with bowel exposure was observed, with necrotic tissue at the wound base. Sorbact was applied, and NPWTi-d was initiated. Gradual granulation tissue formation was achieved, and NPWT was discontinued on POD 40, followed by skin closure.
CONCLUSIONS: Wound dehiscence resulting from SSI markedly impairs patients’ quality of life and presents a major therapeutic challenge. The combination of NPWTi-d and Sorbact enabled safe and effective treatment for refractory wounds.
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Takamitsu Hayakawa, Mikako Mitake, Hirohisa Inaba, Mayumi Kobayashi, Y ...
2025 年11 巻1 号 論文ID: cr.25-0287
発行日: 2025年
公開日: 2025/08/20
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INTRODUCTION: Bronchogenic cysts are congenital, benign cystic lesions that develop in the mediastinum. Many patients are asymptomatic, and conservative observation is often chosen in clinical practice. However, delayed surgical resection following cyst enlargement and compression of the adjacent membranous portion of the trachea can result in perioperative challenges.
CASE PRESENTATION: We report the case of a 53-year-old woman who had been under observation for 10 years for an asymptomatic mediastinal mass. The mass enlarged gradually and caused persistent cough along with obstructive ventilatory impairment. Chest CT revealed a 5.5 cm mass compressing the membranous trachea, resulting in tracheal stenosis. MRI revealed a homogeneously high T2 signal within the mass, suggesting a simple cystic nature. PET showed no accumulation of fluorodeoxyglucose in the mass, indicating no malignancy. Based on preoperative diagnosis of a bronchogenic cyst, the patient underwent video-assisted thoracoscopic surgery. Tracheal intubation using a double-lumen tube was challenging due to the tracheal stenosis. Moreover, the membranous trachea compressed by the cyst exhibited white degeneration, suggesting thinning and fragility. Intraoperatively, due to firm adhesion to the membranous trachea, a part of the cyst wall was intentionally left in place to avoid tracheal injury. The inner lining of the residual cyst was cauterized to prevent recurrence. Bronchoscopic findings on POD 7 showed that white degeneration of the membranous trachea remained. Histopathological examination revealed ciliated columnar epithelium and cartilage on the cyst wall, confirming the diagnosis of a bronchogenic cyst.
CONCLUSIONS: Long-term observation of mediastinal bronchogenic cysts can lead to degeneration and thinning of the membranous trachea, increasing the risk of tracheal injury and incomplete resection during surgery. Therefore, the absence of symptoms should not justify delaying surgical intervention. Preoperative assessment for coexisting malignancy and tracheal abnormalities can support surgical decision-making to ensure a safe procedure.
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Yuko Kijima, Munetsugu Hirata, Yumika Nakazawa, Kazuya Shimmura, Naoki ...
2025 年11 巻1 号 論文ID: cr.24-0187
発行日: 2025年
公開日: 2025/08/19
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INTRODUCTION: Nipple-sparing mastectomy (NSM) has been increasingly used therapeutically for breast cancer patients in whom the nipple-areolar complex is not involved, being associated with better esthetic results and QOL than skin-sparing mastectomy. Robotic nipple-sparing mastectomies (R-NSM) using da Vinci SP surgical system (Intuitive Surgical, Sunnyvale, CA, USA) with immediate breast reconstruction (IBR) has been reported as a suitable surgical treatment for early breast cancers. We present a patient who underwent R-NSM and IBR, with excellent results. To our knowledge, this is the 1st reported case of R-NSM and IBR using the da Vinci SP surgical system in Japan.
CASE PRESENTATION: We performed R-NSM for a 41-year-old Japanese woman with cTisN0M0 breast cancer. NSM and IBR to place a tissue expander (TE) into the post-pectoral pocket were performed using the da Vinci SP surgical system with the double bipolar method.
CONCLUSIONS: This is the 1st reported case of R-NSM using da Vinci SP surgical system in Japan.
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Kentaro Goto, Masato Narita, Ryoya Yamaoka, Koki Moriyoshi, Hiroaki Ha ...
2025 年11 巻1 号 論文ID: cr.25-0320
発行日: 2025年
公開日: 2025/08/19
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INTRODUCTION: For inguinal hernia repair, the plug-and-patch technique is commonly employed. Although abdominal wall hernias are occasionally treated with plugs, their safety remains uncertain. Herein, we report a surgical case of enterocutaneous fistula occurring 18 years after incisional hernia repair using the plug-and-patch technique.
CASE PRESENTATION: An 89-year-old woman presented with right lower abdominal discomfort and a skin ulcer and was admitted to our hospital. She had undergone an open appendectomy 64 years prior, followed by incisional hernia incarceration (leading to small intestinal resection) and suture hernia repair 20 years prior. The incisional hernia recurred 2 years postoperatively and was repaired using a mesh. Physical examination revealed a skin ulcer with purulent discharge and erythema on the right lower abdomen surrounding the surgical wound. Contrast-enhanced CT revealed an enterocutaneous fistula. Fasting, drainage, and antibiotic therapy were required before surgery. Laparoscopic resection of the intestinal loop involving the enterocutaneous fistula and the entire mesh was performed. The fascia was closed without a mesh. No hernia recurrence was observed after 39 months.
CONCLUSIONS: The use of plugs is simple but might not be suitable for incisional hernia repair.
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Ayaka Tachikawa, Kazushige Kawai, Akira Dejima, Sakiko Nakamori, Hirok ...
2025 年11 巻1 号 論文ID: cr.25-0386
発行日: 2025年
公開日: 2025/08/19
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INTRODUCTION: We report herein a rare case of Ewing sarcoma that metastasized to the mesenteric lymph nodes.
CASE PRESENTATION: The patient was a 40-year-old female with Ewing sarcoma of the 1st lumbar vertebra, which was treated with chemotherapy and stereotactic radiotherapy. No local recurrence or distant metastasis was observed during the first 3-year follow-up period after treatment. Three years later, she presented to the emergency department with muscle weakness. A 60-mm lesion in the right parieto-occipital lobe of the brain and a 40-mm tumor in the small bowel mesentery were detected. Emergency craniotomy confirmed a cerebral metastasis of the Ewing sarcoma. The patient subsequently underwent 6 cycles of ifosfamide (IFM) monotherapy, which reduced the mesenteric tumor to 10 mm in size. Surgical resection was performed with clear margins. Histopathological examination of the mesenteric lymph nodes confirmed the findings of the emergency craniotomy. The patient continues to receive IFM monotherapy as adjuvant chemotherapy. Although brain metastases developed at postoperative months 1, 6, and 10, no intra-abdominal recurrence was observed during the 1-year surveillance period.
CONCLUSIONS: While Ewing sarcoma can metastasize to isolated distant lymph nodes, oligometastases can be treated with surgical resection.
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Asuka Tomida, Motoki Yano, Tomohiro Setogawa, Ryotaro Katsuya, Chihiro ...
2025 年11 巻1 号 論文ID: cr.25-0196
発行日: 2025年
公開日: 2025/08/19
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INTRODUCTION: The most common anterior mediastinal tumors are thymic epithelial tumors, including thymoma, and other kinds of diseases are relatively rare. Here, we report our experience in treating a patient with amyloidosis of the thymus, a very rare type of mediastinal lesion.
CASE PRESENTATION: We experienced thymic amyloidosis mimicking a thymoma. A 66-year-old male underwent thymectomy for an anterior mediastinal tumor, which was incidentally pointed out with chest CT. The pathological examination revealed that eosinophilic unstructured substances were present around the atrophied thymic tissue, suggesting the presence of amyloid deposits. Thus, the postoperative pathological diagnosis was thymic amyloidosis. Thymic amyloidosis is extremely rare. Since other amyloid deposits were identified in the gastric mucosa of this patient, he was diagnosed with systemic amyloidosis, and chemotherapy was initiated. The patient had no progression of amyloidosis for 21 months after surgery.
CONCLUSIONS: To the best of our knowledge, only 8 cases of thymic amyloidosis, including the present case, have been reviewed in the literature. Although thymic amyloidosis is extremely rare, an unfavorable prognosis has occasionally been reported. In addition, thymoma and thymic amyloidosis have similar imaging findings, thus making it difficult to distinguish between them. Therefore, thoracic surgeons may need to be aware that thymic amyloidosis is included in the list of anterior mediastinal lesions.
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Takara Kinjo, Keishi Okubo, Masahiro Hamanoue, Miki Murakami, Takao Oh ...
2025 年11 巻1 号 論文ID: cr.25-0285
発行日: 2025年
公開日: 2025/08/19
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INTRODUCTION: Gastric neuroendocrine carcinoma (NEC) is a rare disease among gastric cancers, accounting for only 0.1%–0.6% of all cases. This disease is known to have a poor prognosis and a higher risk of recurrence compared to conventional gastric adenocarcinoma.
CASE PRESENTATION: At the age of 44, a 60-year-old female underwent a laparoscopic-assisted proximal gastrectomy for gastric cancer at a previous hospital. Neuroendocrine carcinoma was diagnosed following a postoperative pathological examination based on histological findings and immunostaining results. The patient was followed up without any recurrences. After 14 years, a follow-up contrast-enhanced CT revealed a 9-mm mass on the greater curvature side of the gastric antrum, which was suspected to be lymph node swelling at the previous hospital. After 8 months, she came to our hospital with abdominal discomfort and distention. The CT scan revealed a 55-mm mass, indicating an increase in the previously mentioned mass. At our hospital, the patient underwent open tumor resection. The pathological findings revealed a recurrence of gastric NEC. The patient has been recurrence-free for 6 months following surgery.
CONCLUSIONS: We present a case of gastric NEC with disseminated recurrence. To our knowledge, this is the first report of a disseminated case in which a recurrent lesion caused by omental dissemination grew in size and infiltrated a portion of the gastric serosa approximately 14 years after the initial surgery.
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Eri Nagasaki-Maeoka, Katsuyoshi Shimozawa, Masaru Ueno, Kanako Saiki, ...
2025 年11 巻1 号 論文ID: cr.25-0010
発行日: 2025年
公開日: 2025/08/14
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INTRODUCTION: Anaplastic sarcoma of the kidney (ASK) is a rare renal tumor, with fewer than 50 cases reported in the literature since 2007. ASK is pathologically characterized by the presence of cystic and solid areas consisting of spindle cells showing marked anaplasia. Recent studies have reported that the vast majority of patients with ASK have DICER1 variants, and that these tumors are part of the DICER1 syndrome, a hereditary cancer predisposition disorder. Herein, we report a pediatric case of this rare tumor, including pathological findings, DICER1 gene analysis of the tumor and peripheral blood samples, and the disease course.
CASE PRESENTATION: A previously healthy 2-year-old girl presented with gross hematuria and a mass in her right abdomen. She had a family history of tumor; her eldest maternal aunt had developed rhabdomyosarcoma, another maternal aunt had follicular thyroid cancer, and her maternal grandmother had a benign thyroid tumor. Imaging revealed a 10-cm tumor with conspicuous internal cystic structures in the right kidney. The patient underwent right nephrectomy, removing a tumor measuring 12.5 × 9 × 8 cm that contained cystic and solid parts. The tumor was composed of spindle-shaped cells with anaplastic changes. Finally, the diagnosis of ASK was established. The treatment regimen, in accordance with the therapy for clear cell sarcoma of the kidney or diffuse anaplasia type Wilms tumor, was administered. Based on the diagnosis of ASK and the family history of DICER1-associated tumors, DICER1 syndrome was suspected. Sequencing of the hotspot region (i.e., RNase IIIb domain) using tumor specimen and coding regions using blood sample in the DICER1 revealed somatic c.5125G>A (p.D1709N) and germline c.4458dupA (p.S1487Ifs*5). Two years have passed since the end of treatment, and the patient is alive and healthy without evidence of tumor recurrence.
CONCLUSIONS: Treatment with intensive chemotherapy (vincristine, doxorubicin, etoposide, and cyclophosphamide) and abdominal irradiation was effective as 2-year event-free survival was achieved. Since DICER1 syndrome causes a variety of rare cancers, particularly in infants and young adults, all surgeons and pediatric oncologists should be aware of the possibility of DICER1 syndrome.
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Jingjun Yang, Jianming Lu
2025 年11 巻1 号 論文ID: cr.25-0157
発行日: 2025年
公開日: 2025/08/14
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INTRODUCTION: For this case, when the preoperative diagnosis indicated malignant tumor with metastasis, intraoperative frozen section examination was performed to adjust the surgical plan accordingly. This approach helped avoid overtreatment, minimizing the patient’s pain and surgical trauma. This case holds educational significance.
CASE PRESENTATION: A 45-year-old female patient underwent open myomectomy 12 years ago for uterine fibroids and laparoscopic subtotal hysterectomy 10 years ago for the same condition. During this check-up, her CA125 was found to be elevated. Further examinations, including ultrasound, enhanced CT, and enhanced MRI, all suggested a pelvic malignant tumor with liver metastasis. The patient underwent tumor resection, and both intraoperative frozen section and routine histopathologic examination confirmed that the pelvic and subphrenic tumors (which had been considered as liver metastases preoperatively) were both conventional leiomyomas.
CONCLUSIONS: This case highlights that leiomyomas, when metastatic, are easily misdiagnosed as malignant tumors with metastasis, presenting a significant challenge for preoperative diagnosis. Clinicians should maintain a high level of suspicion in such cases to avoid overtreatment. In this case, the intraoperative frozen section played a crucial role in preventing unnecessary pelvic lymph node dissection.
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Yui Innami, Yuki Mizuno, Kentaro Yamada, Kei Takasawa, Yoshifumi Ito, ...
2025 年11 巻1 号 論文ID: cr.25-0111
発行日: 2025年
公開日: 2025/08/14
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INTRODUCTION: We report the case of a child who underwent splenectomy and developed marked thrombocytopenia after detorsion due to wandering splenic torsion.
CASE PRESENTATION: A 7-year-old boy who underwent laparoscopic inguinal hernia surgery developed sudden abdominal pain 2 days later. Contrast-enhanced CT revealed a poor contrast effect in the spleen, and emergency surgery was performed based on the diagnosis of splenic infarction. The spleen was not fixed to the retroperitoneum and was twisted at 720° around the splenic hilum. However, the color tone improved after detorsion of the spleen; therefore, the spleen was preserved. The next day, the patient developed spinal cord injury due to marked thrombocytopenia and epidural hematoma, and emergency hematoma removal surgery was performed. As the patient continued to depend on platelet transfusion, laparoscopic splenectomy was performed. The patient’s platelet counts rapidly increased after surgery. Since then, the patient has undergone treatment and rehabilitation for the spinal cord injury, and his neurological symptoms have improved.
CONCLUSIONS: Preserving the spleen is recommended for wandering splenic torsion, especially in children. In the present case, splenectomy was necessary because of rapid thrombocytopenia caused by increased splenic function after detorsion of the spleen; however, there have been no similar case reports in the past. This condition can be potentially dangerous and can lead to serious complications.
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