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Management of inferior patellar sleeve fracture by ORIF with ortho cord suture: A case report. 骨科脊髓缝合ORIF治疗下髌骨套筒骨折1例。
IF 0.6 Q4 SURGERY Pub Date : 2025-01-13 DOI: 10.1016/j.ijscr.2025.110875
Dinesh Kumar Pandit, Bishal Regmi, Deeksha Subedi, Deepak Jha

Introduction: Patellar sleeve fracture is a rare fracture common in children between the age of 8 and 16 years but rare in adults. We report a case of 22 year old male with inferior patellar sleeve fracture following motorbike accident.

Case presentation: The case study presents a twenty-two year old boy who sustained inferior patellar Sleeve fractures following motor bike accident. The patient presented to ER with pain and swelling along with abrasion. Diagnostic workup, including blood test and imaging, confirmed the diagnosis, revealing elevated inflammatory markers and evidence of inferior patellar sleeve fracture on X-ray. The fracture was managed by Open reduction and internal fixation with ortho cord suture.

Discussion: Sleeve fractures of the patella in children usually occur in the inferior pole of the patella, very rarely in the superior pole. However, sleeve fractures of the patella mainly affect the superior pole in adults. The mechanism of sleeve fracture is a rapid contraction of the quadriceps on a flexed knee. Sleeve fractures usually occur in individuals involved in explosive acceleration activities such as jumping, although high-energy sports such as skateboarding are now also considered common causes Clinically, treatment options including conservative and surgical treatment depends on the severity of bone displacement and the extensor mechanism of the knee.

Conclusion: Inferior patellar sleeve fracture is rare in adults in comparison to the children. The fracture is confirmed through X-rays AP and Lateral view. Thus, diagnosed fracture is treated surgically via open reduction and internal fixation.

髌骨袖骨折是一种罕见的骨折,常见于8至16岁的儿童,但在成人中很少见。我们报告一例22岁男性摩托车事故后髌骨下袖骨折。病例报告:病例研究报告了一个22岁的男孩,他在摩托车事故后持续的下髌骨袖骨折。患者就诊时伴有疼痛、肿胀和擦伤。诊断检查,包括血液检查和影像学检查,证实了诊断,显示炎症标志物升高,x线显示下髌骨套筒骨折。骨折采用切开复位内固定及正骨脊髓缝合。讨论:儿童髌骨套筒骨折通常发生在髌骨下极,很少发生在上极。然而,成人髌骨套筒骨折主要影响上极。套筒骨折的机制是膝关节屈曲时股四头肌的快速收缩。套筒骨折通常发生在参与爆发性加速运动(如跳跃)的个体中,尽管像滑板这样的高能量运动现在也被认为是常见的原因。临床上,治疗选择包括保守治疗和手术治疗取决于骨移位的严重程度和膝关节伸肌机制。结论:成人髌骨下袖骨折较儿童少见。骨折通过x线正位片和侧位片确认。因此,确诊的骨折通过切开复位和内固定手术治疗。
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引用次数: 0
Laparoscopic wedge resection of a descending duodenal gastrointestinal stromal tumor under endoscopic nasobiliary drainage guidance: A case report. 内镜下鼻胆道引流引导下腹腔镜楔形切除十二指肠降段胃肠道间质瘤1例。
IF 0.6 Q4 SURGERY Pub Date : 2025-01-13 DOI: 10.1016/j.ijscr.2025.110877
Woo Yong Lee

Introduction: Gastrointestinal stromal tumors (GIST), which occur anywhere in the gastrointestinal (GI) tract, typically occur in the stomach and small intestine but rarely in the duodenum. We present a case report wherein a descending duodenal GIST was treated with a limited, minimally invasive surgery after endoscopic nasobiliary drainage (ENBD) insertion.

Presentation of case: A 67-year-old woman visited our hospital with an incidentally discovered duodenal tumor. Gastroduodenoscopy revealed a duodenal subepithelial tumor (SET) measuring approximately 2 cm in descending duodenum. Endoscopic ultrasound revealed a well-circumscribed, inhomogeneous hypoechoic lesion measuring approximately 17 × 4.6 mm, thought to arise from the muscularis layer. Computed tomography (CT) revealed an inhomogeneous enhancing mass with central necrosis, measuring approximately 2.7 cm, in the descending duodenum. Pathological findings from the bite-on-bite biopsy showed c-kit and DOG-1 positivity and CD34 and desmin negativity, leading to a GIST diagnosis. Laparoscopic wedge resection with preoperative ENBD insertion was planned due to the risk of pancreaticobiliary duct (PBD) damage during surgery because the lesion was located near the ampulla of Vater (AoV) and minor papilla. Surgery was performed using laparoscopic wedge resection without PBD injury. The patient was discharged 10 days post-surgery without complications.

Discussion: Descending duodenal GIST is difficult to operate on with minimally invasive surgery. However, if the size is not excessive and the PBD is not involved, minimal and limited surgery is possible after ENBD insertion.

Conclusion: We report the first case of limited and minimally invasive surgery followed by ENBD insertion in a rare descending duodenal GIST.

胃肠道间质瘤(GIST)可发生于胃肠道的任何部位,多发生于胃和小肠,很少发生于十二指肠。我们提出一个病例报告,其中十二指肠下行间质瘤是在内镜鼻胆道引流(ENBD)插入后用有限的微创手术治疗。病例介绍:一名67岁妇女因偶然发现十二指肠肿瘤来我院就诊。胃十二指肠镜显示一十二指肠上皮下肿瘤(SET),位于十二指肠降部约2厘米。内窥镜超声显示一边界清晰、不均匀的低回声病变,大小约为17 × 4.6 mm,认为起源于肌层。计算机断层扫描(CT)显示一不均匀强化肿块伴中央坏死,约2.7 cm,位于十二指肠降部。咬对咬活检的病理结果显示c-kit和DOG-1阳性,CD34和desmin阴性,导致GIST诊断。由于病变位于壶腹(AoV)和小乳头附近,手术中存在胰胆管(PBD)损伤的风险,因此计划进行腹腔镜楔形切除术并术前插入ENBD。手术采用腹腔镜楔形切除术,无PBD损伤。术后10天出院,无并发症。讨论:十二指肠下行间质瘤是微创手术的难点。然而,如果尺寸不过大且未累及PBD,则可以在ENBD置入后进行最小限度的手术。结论:我们报告了一例罕见的十二指肠下行间质间质瘤行有限微创手术后植入术的病例。
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引用次数: 0
Successfully enucleation of a rare pancreatic schwannoma: A case report.
IF 0.6 Q4 SURGERY Pub Date : 2025-01-13 DOI: 10.1016/j.ijscr.2025.110865
Ben Abdessalem Abdelaziz, Yassine Kallel, Jbir Ichraf, Hazem Beji, Emna Chalbi, Hassen Touinsi

Introduction and importance: Pancreatic schwannoma (PS) is an extremely rare benign tumor also known as neurilemoma or neuroma. The majority of PS develop cystic lesions, and its preoperative diagnosis is challenging due to its tendency to mimic other lesions of the pancreas. Herein, we reported a case of body PS incidentally discovered in an 81-year-old male, which was successfully treated through enucleation.

Case presentation: We report the case of a 81-year-old man. Who presented with a well-defined polycystic-tumor of about 2cm at the pancreatic body, overdrawn by a computed tomography scan, incidentally discovered. The patient underwent a laparotomy, and we performed an enucleation of the tumor successfully. Histopathological examination revealed spindle-shaped cells. Immunohistochemically studies showed S100-protein expression, confirmed a body pancreatic schwannoma. The postoperative course was uneventful.

Clinical discussion: Schwannomas are rare mesenchymal-tumors, with PS accounting for only 1 % of cases. They are often difficult to diagnose as they mimic other pancreatic tumors. Imaging techniques like CT, MRI, and EUS-FNA aid in detection, but definitive diagnosis requires histological and immunohistochemical analysis. Treatment is typically enucleation for benign tumors, but larger or malignant tumors may need more extensive resections. Due to diagnostic challenges, aggressive surgeries are common. Prognosis is generally favorable, but regular follow-ups are recommended to monitor for recurrence.

Conclusion: Although pancreatic schwannoma is rare, it should be included in the list of differential diagnoses of pancreatic masses, both solid and cystic. Both enucleation and radical surgical resections have revealed great therapeutic efficiency with a well prognosis without recurrences.

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引用次数: 0
Carcinosarcoma of the fallopian tube: A rare case with rapid metastases and port-site metastasis.
IF 0.6 Q4 SURGERY Pub Date : 2025-01-13 DOI: 10.1016/j.ijscr.2025.110889
Takuya Kuboya, Kaoru Kato, Junichi Sasaki

Introduction and importance: Fallopian tube cancer, particularly the carcinosarcoma subtype, is a rare malignancy posing diagnostic challenges.

Case presentation: Our patient was an 83-year-old, nulligravida woman, presented to our outpatient clinic with one month of pelvic pain. On examination, a pelvic mass was detected. MRI suggested a cystic tumor in the fallopian tube, and pyosalpinx was initially suspected. Laparoscopic surgery was performed. However, she was later diagnosed with carcinosarcoma of the fallopian tube, according to the pathological diagnosis. The disease rapidly recurred and metastasized, including the development of Port-Site Metastasis (PSM). The patient underwent PSM excision surgery based on her request, which contributed to her mental well-being.

Clinical discussion: Diagnosing fallopian tube cancer is difficult, and there is often a misdiagnosis risk during surgery, leading to potential errors in treatment. MRI can help identify fallopian tubal cancer, but its features can also resemble benign conditions. Factors like infertility and chronic inflammation can increase the suspicion of cancer. Preventive measures for PSM during laparoscopic surgery are crucial. While it's unclear if removing port-site metastases improves the prognosis, it may benefit the patient's mental health.

Conclusion: Our findings underscore the importance of considering the possibility of malignancy when treating tubal tumors. We should take preventive measures for PSM during laparoscopic surgery. PSM excision surgery could be an option to enhance patient's mental health.

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引用次数: 0
A preventable medical error: Successful management of iatrogenic Zenker's diverticulum perforation. 一个可预防的医疗错误:医源性Zenker憩室穿孔的成功治疗。
IF 0.6 Q4 SURGERY Pub Date : 2025-01-12 DOI: 10.1016/j.ijscr.2025.110859
Ali Alrahil, Essam Moumena, Mazen Aljanadi, Mhd Saeed Nassar, Tammam Hasan

Introduction and significance: Zenker's diverticulum is a rare condition characterized by a false diverticulum, as a true diverticulum involves herniation of all wall layers outward. Dysphagia, difficulty in swallowing, is the most common symptom. Diagnosis is primarily made through X-ray studies using contrast material during swallowing. Treatment options include open surgery or endoscopy.

Case presentation: We present the case of an adult male who experienced progressive dysphagia for approximately two years. A diagnosis of Zenker's diverticulum was confirmed by radiography following unsuccessful endoscopic treatment and a subsequent perforation of the diverticulum. The patient underwent successful open surgery after adequate preoperative preparation.

Clinical discussion: Zenker's diverticulum is an uncommon cause of pharyngeal dysphagia, but it should be considered in the differential diagnosis due to the importance of early intervention and prevention of malignancy. The endoscopic approach carries a significant risk of perforation, necessitating careful treatment and close monitoring during and after the procedure.

Conclusion: The endoscopic approach is considered the best treatment method for Zenker's diverticulum and should be used with caution to avoid complications, especially perforation, which makes open surgery the most appropriate treatment in this case.

简介及意义:Zenker憩室是一种罕见的以假憩室为特征的疾病,因为真憩室包括所有壁层向外突出。吞咽困难是最常见的症状。诊断主要通过吞咽时使用造影剂进行x线检查。治疗方案包括开放手术或内窥镜检查。病例介绍:我们提出的情况下,一个成年男性经历进行性吞咽困难大约两年。Zenker憩室的诊断是在不成功的内窥镜治疗和随后的憩室穿孔后通过x线摄影证实的。在充分的术前准备后,患者接受了成功的开放手术。临床讨论:Zenker憩室是咽部吞咽困难的罕见病因,但早期干预和预防恶性肿瘤的重要性,在鉴别诊断中应予以考虑。内窥镜入路有明显的穿孔风险,需要在手术期间和手术后仔细治疗和密切监测。结论:内镜下入路是Zenker憩室的最佳治疗方法,应谨慎使用,避免并发症,尤其是穿孔,开放手术是本病例最合适的治疗方法。
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引用次数: 0
Mesenchymal chondrosarcoma of maxilla misdiagnosed as a benign fibrous lesion: A rare case report from Syria. 上颌骨间充质软骨肉瘤误诊为良性纤维病变:叙利亚罕见病例报告。
IF 0.6 Q4 SURGERY Pub Date : 2025-01-12 DOI: 10.1016/j.ijscr.2025.110874
Abdulrahman Abdo Shouman, Kamilla A Mohammed, Hadeel M Kaadeh, Yehia M Haphian, Mohamad M Hamzeh

Introduction: Mesenchymal chondrosarcoma (MC) is a high-grade variant of chondrosarcoma, essentially composed of poorly differentiated spindle cells interspersed with areas of cartilage or chondroid matrix. MC is extremely rare; it only accounts for 0.1 % of head and neck tumors and for only 1 % of all chondrosarcomas (CSs).

Case presentation: A 21-year-old man presented with a medical history of a painful irritation at the dextral maxillary region, presented as a mass at the vestibule of the oral cavity near the upper molars, and had been misdiagnosed as a benign fibrous lesion and excised without performing a biopsy. Magnetic resonance imaging (MRI) revealed an invasive lesion filling the right maxillary sinus and penetrating the orbital floor. A biopsy was then performed and revealed an MC.

Clinical discussion: The patient underwent a wide surgical resection, except for the infraorbital region, in which the tumor was surrounded by a fibrous capsule separating it from the anatomical structures of the eye. Due to the lack of wide resection in the orbital floor area (to preserve the eyeball), we applied the chemotherapy that was done with cisplatin and doxorubicin.

Conclusion: Confirmed diagnosis by biopsy and treatment, both surgical and chemical, with frequent follow-up are decisive factors in progressing MC.

间充质软骨肉瘤(MC)是软骨肉瘤的一种高级别变体,主要由散布在软骨或软骨样基质区域的低分化梭形细胞组成。MC极为罕见;它只占头颈部肿瘤的0.1%,占所有软骨肉瘤(CSs)的1%。病例介绍:一名21岁男性,病史为上颌右侧疼痛刺激,表现为上磨牙附近口腔前庭肿块,误诊为良性纤维病变,未行活检切除。磁共振成像(MRI)显示一浸润性病变填满右上颌窦并穿透眶底。临床讨论:患者接受了广泛的手术切除,除了眶下区域,肿瘤被纤维囊包围,将其与眼睛的解剖结构分开。由于缺乏眶底大面积切除(以保护眼球),我们采用顺铂和阿霉素联合化疗。结论:活检确诊、手术及化疗治疗、频繁随访是恶性肿瘤进展的决定性因素。
{"title":"Mesenchymal chondrosarcoma of maxilla misdiagnosed as a benign fibrous lesion: A rare case report from Syria.","authors":"Abdulrahman Abdo Shouman, Kamilla A Mohammed, Hadeel M Kaadeh, Yehia M Haphian, Mohamad M Hamzeh","doi":"10.1016/j.ijscr.2025.110874","DOIUrl":"https://doi.org/10.1016/j.ijscr.2025.110874","url":null,"abstract":"<p><strong>Introduction: </strong>Mesenchymal chondrosarcoma (MC) is a high-grade variant of chondrosarcoma, essentially composed of poorly differentiated spindle cells interspersed with areas of cartilage or chondroid matrix. MC is extremely rare; it only accounts for 0.1 % of head and neck tumors and for only 1 % of all chondrosarcomas (CSs).</p><p><strong>Case presentation: </strong>A 21-year-old man presented with a medical history of a painful irritation at the dextral maxillary region, presented as a mass at the vestibule of the oral cavity near the upper molars, and had been misdiagnosed as a benign fibrous lesion and excised without performing a biopsy. Magnetic resonance imaging (MRI) revealed an invasive lesion filling the right maxillary sinus and penetrating the orbital floor. A biopsy was then performed and revealed an MC.</p><p><strong>Clinical discussion: </strong>The patient underwent a wide surgical resection, except for the infraorbital region, in which the tumor was surrounded by a fibrous capsule separating it from the anatomical structures of the eye. Due to the lack of wide resection in the orbital floor area (to preserve the eyeball), we applied the chemotherapy that was done with cisplatin and doxorubicin.</p><p><strong>Conclusion: </strong>Confirmed diagnosis by biopsy and treatment, both surgical and chemical, with frequent follow-up are decisive factors in progressing MC.</p>","PeriodicalId":48113,"journal":{"name":"International Journal of Surgery Case Reports","volume":"127 ","pages":"110874"},"PeriodicalIF":0.6,"publicationDate":"2025-01-12","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143014192","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Duplication of the gallbladder: An intraoperative finding during laparoscopic cholecystectomy. 胆囊重复:腹腔镜胆囊切除术的术中发现。
IF 0.6 Q4 SURGERY Pub Date : 2025-01-12 DOI: 10.1016/j.ijscr.2025.110884
Deshan Mario Gomez, Arulprashanth Arulnathan, Dharmabandhu Nandadeva Samarasekera, Duminda Subasinghe

Introduction: Duplication of the gallbladder is a rare congenital malformation associated with the development of cholelithiasis. It increases the risk of iatrogenic bile duct injury during cholecystectomy and can lead to symptom recurrence if missed. Although preoperative imaging is helpful, detection rates are around 50 %.

Case presentation: A 36-year-old healthy female with symptomatic gallstone disease and ultrasonographic evidence of cholelithiasis was scheduled for elective laparoscopic cholecystectomy. Following standard four port entry and dissection of pericholecystic adhesions to duodenum, a V shaped duplicated gallbladder with calculi, and a common cystic duct and artery was noted. The cystohepatic triangle was dissected, critical view of safety demonstrated and the common cystic duct and artery were divided between clips.

Discussion: Variations in gallbladder morphology has been described by Gross, Boyden and Harlaftis classification. Type 1 (bilobed gallbladder) has a common embryological origin with an invaginating septum separating the lumens while type 2 (double gallbladder) has a double embryological origin with separate gallbladders and their own cystic ducts. Many cases go undetected or are mis-identified as choledochal cyst, diverticulum or Phrygian cap. MRCP is the imaging modality of choice for suspected duplicate gallbladder.

Conclusion: Careful dissection of the cystohepatic triangle with attention to critical view of safety is important in all cases as numerous variations in hepatobiliary anatomy have been described. Surgical awareness of these variations and good surgical technique will promote safe laparoscopic cholecystectomy even in the most unexpected of encounters.

简介:胆囊重复是一种罕见的先天性畸形与胆石症的发展。它增加了胆囊切除术中医源性胆管损伤的风险,如果遗漏,可能导致症状复发。尽管术前成像是有帮助的,但检出率约为50%。病例介绍:一名36岁健康女性,有症状的胆囊结石疾病和超声证据表明胆石症计划择期腹腔镜胆囊切除术。在标准的四口入路和十二指肠胆囊周粘连的剥离后,发现一V形重复胆囊伴结石,一总囊管和动脉。解剖囊肝三角,显示安全的关键观点,并将囊管和总动脉分开夹。讨论:胆囊形态的变化已被Gross, Boyden和Harlaftis分类所描述。1型(双叶胆囊)有一个共同的胚胎起源,有一个内陷的间隔分隔管腔,而2型(双胆囊)有两个胚胎起源,有独立的胆囊和它们自己的胆囊管。许多病例未被发现或被误诊为胆总管囊肿、憩室或弗里盖。MRCP是疑似重复胆囊的首选成像方式。结论:仔细解剖囊肝三角,注意安全的关键观点,在所有情况下都是重要的,因为肝胆解剖的许多变化已经被描述。外科意识到这些变化和良好的手术技术将促进安全的腹腔镜胆囊切除术,即使在最意想不到的遭遇。
{"title":"Duplication of the gallbladder: An intraoperative finding during laparoscopic cholecystectomy.","authors":"Deshan Mario Gomez, Arulprashanth Arulnathan, Dharmabandhu Nandadeva Samarasekera, Duminda Subasinghe","doi":"10.1016/j.ijscr.2025.110884","DOIUrl":"https://doi.org/10.1016/j.ijscr.2025.110884","url":null,"abstract":"<p><strong>Introduction: </strong>Duplication of the gallbladder is a rare congenital malformation associated with the development of cholelithiasis. It increases the risk of iatrogenic bile duct injury during cholecystectomy and can lead to symptom recurrence if missed. Although preoperative imaging is helpful, detection rates are around 50 %.</p><p><strong>Case presentation: </strong>A 36-year-old healthy female with symptomatic gallstone disease and ultrasonographic evidence of cholelithiasis was scheduled for elective laparoscopic cholecystectomy. Following standard four port entry and dissection of pericholecystic adhesions to duodenum, a V shaped duplicated gallbladder with calculi, and a common cystic duct and artery was noted. The cystohepatic triangle was dissected, critical view of safety demonstrated and the common cystic duct and artery were divided between clips.</p><p><strong>Discussion: </strong>Variations in gallbladder morphology has been described by Gross, Boyden and Harlaftis classification. Type 1 (bilobed gallbladder) has a common embryological origin with an invaginating septum separating the lumens while type 2 (double gallbladder) has a double embryological origin with separate gallbladders and their own cystic ducts. Many cases go undetected or are mis-identified as choledochal cyst, diverticulum or Phrygian cap. MRCP is the imaging modality of choice for suspected duplicate gallbladder.</p><p><strong>Conclusion: </strong>Careful dissection of the cystohepatic triangle with attention to critical view of safety is important in all cases as numerous variations in hepatobiliary anatomy have been described. Surgical awareness of these variations and good surgical technique will promote safe laparoscopic cholecystectomy even in the most unexpected of encounters.</p>","PeriodicalId":48113,"journal":{"name":"International Journal of Surgery Case Reports","volume":"127 ","pages":"110884"},"PeriodicalIF":0.6,"publicationDate":"2025-01-12","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142985199","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Retiform hemangioendothelioma: A case report and literature review. 网状血管内皮瘤1例报告并文献复习。
IF 0.6 Q4 SURGERY Pub Date : 2025-01-12 DOI: 10.1016/j.ijscr.2025.110869
Lin Song, Dule Xing, Zhixin Cao, Yuanyuan Zong, Dongsheng Hou

Introduction and importance: Retiform hemangioendothelioma(RH) is a rare vascular tumor affecting patients over a wide age range without a gender predilection; only about 50 cases have been described so far.

Case presentation: We report a case of submandibular retiform hemangioendothelioma in a 58-year-old woman who had been diagnosed with RH 20 years ago and had experienced recurrence four times during the past 20 years. This will increase the limited number of such cases in the hope of gaining a better understanding of this rare type of tumor. The histological features of RH are characterized by arborizing blood vessels arranged in a testicular network pattern with endothelial cells arranged in a hobnail pattern. Immunohistochemistry revealed CD31, CD34, ERG, and other vascular markers.

Clinical discussion: Based on the morphological and immunohistochemical results, we diagnosed the patient with a rectiform hemangioendothelioma with local malignancy that transformed into angiosarcoma, with enlargement of the lymph nodes in the neck area, possibly indicating tumor lymph node metastasis. The patient underwent extended resection and completed 14 radiotherapy sessions. Follow-up at 3 months after surgery showed no recurrence.

Conclusion: RH is a low-grade malignant intermediate vascular tumor that is prone to recurrence and does not generally metastasize to distant sites. Patients with recurrent relapses may undergo malignant transformation or lymph node metastases. Treatment primarily relies on wide excision and adjuvant radiotherapy may be necessary when required.

介绍及重要性:网状血管内皮瘤(RH)是一种罕见的血管肿瘤,患者年龄广泛,无性别倾向;到目前为止,只有大约50例被描述。病例介绍:我们报告了一个58岁的下颌网状血管内皮瘤的病例,她在20年前被诊断为RH,在过去的20年里经历了4次复发。这将增加这类病例的有限数量,以期更好地了解这种罕见的肿瘤类型。RH的组织学特征是树突状血管排列成睾丸网状,内皮细胞排列成鞋钉状。免疫组织化学显示CD31、CD34、ERG和其他血管标志物。临床讨论:基于形态学和免疫组化结果,我们诊断患者为局部恶性转化为血管肉瘤的整形血管内皮瘤,颈部淋巴结肿大,可能提示肿瘤淋巴结转移。患者接受了延长切除并完成了14次放疗。术后3个月随访未见复发。结论:RH是一种低级别恶性中间血管肿瘤,易复发,一般不会远处转移。复发患者可发生恶性转化或淋巴结转移。治疗主要依靠广泛切除,必要时可能需要辅助放疗。
{"title":"Retiform hemangioendothelioma: A case report and literature review.","authors":"Lin Song, Dule Xing, Zhixin Cao, Yuanyuan Zong, Dongsheng Hou","doi":"10.1016/j.ijscr.2025.110869","DOIUrl":"https://doi.org/10.1016/j.ijscr.2025.110869","url":null,"abstract":"<p><strong>Introduction and importance: </strong>Retiform hemangioendothelioma(RH) is a rare vascular tumor affecting patients over a wide age range without a gender predilection; only about 50 cases have been described so far.</p><p><strong>Case presentation: </strong>We report a case of submandibular retiform hemangioendothelioma in a 58-year-old woman who had been diagnosed with RH 20 years ago and had experienced recurrence four times during the past 20 years. This will increase the limited number of such cases in the hope of gaining a better understanding of this rare type of tumor. The histological features of RH are characterized by arborizing blood vessels arranged in a testicular network pattern with endothelial cells arranged in a hobnail pattern. Immunohistochemistry revealed CD31, CD34, ERG, and other vascular markers.</p><p><strong>Clinical discussion: </strong>Based on the morphological and immunohistochemical results, we diagnosed the patient with a rectiform hemangioendothelioma with local malignancy that transformed into angiosarcoma, with enlargement of the lymph nodes in the neck area, possibly indicating tumor lymph node metastasis. The patient underwent extended resection and completed 14 radiotherapy sessions. Follow-up at 3 months after surgery showed no recurrence.</p><p><strong>Conclusion: </strong>RH is a low-grade malignant intermediate vascular tumor that is prone to recurrence and does not generally metastasize to distant sites. Patients with recurrent relapses may undergo malignant transformation or lymph node metastases. Treatment primarily relies on wide excision and adjuvant radiotherapy may be necessary when required.</p>","PeriodicalId":48113,"journal":{"name":"International Journal of Surgery Case Reports","volume":"127 ","pages":"110869"},"PeriodicalIF":0.6,"publicationDate":"2025-01-12","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143014158","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Retinal capillary hemangioma in a pthisical globe: Late sequelae in a case of Von Hippel-Lindau (VHL) disease presenting with endophthalmitis. 病理球形视网膜毛细血管瘤:以眼内炎为表现的Von Hippel-Lindau (VHL)病的晚期后遗症1例。
IF 0.6 Q4 SURGERY Pub Date : 2025-01-12 DOI: 10.1016/j.ijscr.2025.110867
Hala A Helmi, Rakan Alsaad, Hattan Alkhiary, Hind M Alkatan

Introduction: Retinal capillary hemangioma (RCH) is a benign vascular hamartoma that can occur sporadically or as a manifestation of Von Hippel-Lindau (VHL) disease. If left untreated, it results in adverse ocular complications depending on its location and eventual visual loss.

Case presentation: We present a 50-year-old man who was a known case of VHL with history of left eye vision loss in the left eye at the age of 30 years. He underwent resection of a brain hemangioblastoma 10 years earlier, which was complicated by left facial nerve palsy. He presented with acute left eye pain, redness, and eyelid swelling. He had large corneal infiltrate with hypopyon and obscured fundus view. He was treated medically as a case of left endophthalmitis with no improvement. The eye was eventually eviscerated. Histopathological examination revealed acute necrotizing keratitis, osseous metaplasia, and long-standing RCH.

Discussion: Peripheral RCHs are the most encountered ocular vascular lesion in VHL disease. Other locations close to the optic nerve (juxtapapillary) result in complicated visual loss. The RCH in our VHL case was confirmed 20 years after the history of vision loss in the same eye. The patient also had confirmed surgically treated intracranial hemangioblastoma but didn't seek any ophthalmic care prior to his recent presentation with painful acute keratitis and endophthalmitis.

Conclusion: VHL has multiple organ involvement. Patients suspected or diagnosed with VHL should receive prompt health care counselling to ensure periodic eye examination for control of any intraocular vascular lesions to prevent visual loss.

视网膜毛细血管瘤(RCH)是一种良性血管错构瘤,可以零星发生或作为Von Hippel-Lindau (VHL)病的表现。如果不及时治疗,它会导致不良的眼部并发症,这取决于它的位置和最终的视力丧失。病例介绍:我们报告一位50岁的男性,他是一个已知的VHL病例,30岁时左眼视力丧失。10年前,他接受了脑血管母细胞瘤切除术,并伴有左侧面神经麻痹。他表现为急性左眼疼痛、红肿和眼睑肿胀。大面积角膜浸润伴低视,眼底视野模糊。他被当作左眼内炎病例治疗,没有好转。这只眼睛最终被掏空了。组织病理学检查显示急性坏死性角膜炎,骨性化生,长期RCH。讨论:外周RCHs是VHL疾病中最常见的眼部血管病变。靠近视神经的其他部位(乳头旁)导致复杂的视力丧失。我们的VHL病例的RCH是在同一只眼睛视力丧失20年后确认的。该患者也接受过颅内血管母细胞瘤的手术治疗,但在最近出现疼痛性急性角膜炎和眼内炎之前并未寻求任何眼科护理。结论:VHL累及多脏器。怀疑或诊断为VHL的患者应及时接受保健咨询,确保定期进行眼科检查,以控制任何眼内血管病变,防止视力丧失。
{"title":"Retinal capillary hemangioma in a pthisical globe: Late sequelae in a case of Von Hippel-Lindau (VHL) disease presenting with endophthalmitis.","authors":"Hala A Helmi, Rakan Alsaad, Hattan Alkhiary, Hind M Alkatan","doi":"10.1016/j.ijscr.2025.110867","DOIUrl":"https://doi.org/10.1016/j.ijscr.2025.110867","url":null,"abstract":"<p><strong>Introduction: </strong>Retinal capillary hemangioma (RCH) is a benign vascular hamartoma that can occur sporadically or as a manifestation of Von Hippel-Lindau (VHL) disease. If left untreated, it results in adverse ocular complications depending on its location and eventual visual loss.</p><p><strong>Case presentation: </strong>We present a 50-year-old man who was a known case of VHL with history of left eye vision loss in the left eye at the age of 30 years. He underwent resection of a brain hemangioblastoma 10 years earlier, which was complicated by left facial nerve palsy. He presented with acute left eye pain, redness, and eyelid swelling. He had large corneal infiltrate with hypopyon and obscured fundus view. He was treated medically as a case of left endophthalmitis with no improvement. The eye was eventually eviscerated. Histopathological examination revealed acute necrotizing keratitis, osseous metaplasia, and long-standing RCH.</p><p><strong>Discussion: </strong>Peripheral RCHs are the most encountered ocular vascular lesion in VHL disease. Other locations close to the optic nerve (juxtapapillary) result in complicated visual loss. The RCH in our VHL case was confirmed 20 years after the history of vision loss in the same eye. The patient also had confirmed surgically treated intracranial hemangioblastoma but didn't seek any ophthalmic care prior to his recent presentation with painful acute keratitis and endophthalmitis.</p><p><strong>Conclusion: </strong>VHL has multiple organ involvement. Patients suspected or diagnosed with VHL should receive prompt health care counselling to ensure periodic eye examination for control of any intraocular vascular lesions to prevent visual loss.</p>","PeriodicalId":48113,"journal":{"name":"International Journal of Surgery Case Reports","volume":"127 ","pages":"110867"},"PeriodicalIF":0.6,"publicationDate":"2025-01-12","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142985205","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Surgical management of the second cavernous lymphangioma in the tongue globally: A case report.
IF 0.6 Q4 SURGERY Pub Date : 2025-01-11 DOI: 10.1016/j.ijscr.2025.110863
Eiad Khouri, Jafar Hamdy, Mohammad Alsheekh Kadour

Introduction and importance: Oral Lymphangioma is a rare benign tumor that arises from the lymphatic vessels in oral cavity. It is a development defect in the lymphatic system, which is less common than other types of vascular anomalies, and the incidence in the general population is very low, especially in the oral cavity. It typically presents as soft, painless swelling, often found on the tongue, lips or floor or the mouth. The lesions can vary in size and may have a translucent appearance, sometimes resembling a cyst.

Case presentation: A 13-year-old male complaint of congenital diffuse, painless swelling of the lateral side of the tongue. The swelling was soft and compressible and involved half of the tongue. Incisional biopsy from the lesion can confirm the diagnosis by showing dilated lymphatic vessels histologically.

Clinical discussion: Oral lymphangioma is a rare tumor found in the oral cavity, occurring at various sites but most commonly on the tongue. This often leads to macroglossia, presenting as a compressible, painless swelling that typically necessitates surgical excision when it causes functional issues.

Conclusion: Oral lymphangioma generally has a good prognosis, especially when treated appropriately. However, they can recur if not completely excised.

{"title":"Surgical management of the second cavernous lymphangioma in the tongue globally: A case report.","authors":"Eiad Khouri, Jafar Hamdy, Mohammad Alsheekh Kadour","doi":"10.1016/j.ijscr.2025.110863","DOIUrl":"https://doi.org/10.1016/j.ijscr.2025.110863","url":null,"abstract":"<p><strong>Introduction and importance: </strong>Oral Lymphangioma is a rare benign tumor that arises from the lymphatic vessels in oral cavity. It is a development defect in the lymphatic system, which is less common than other types of vascular anomalies, and the incidence in the general population is very low, especially in the oral cavity. It typically presents as soft, painless swelling, often found on the tongue, lips or floor or the mouth. The lesions can vary in size and may have a translucent appearance, sometimes resembling a cyst.</p><p><strong>Case presentation: </strong>A 13-year-old male complaint of congenital diffuse, painless swelling of the lateral side of the tongue. The swelling was soft and compressible and involved half of the tongue. Incisional biopsy from the lesion can confirm the diagnosis by showing dilated lymphatic vessels histologically.</p><p><strong>Clinical discussion: </strong>Oral lymphangioma is a rare tumor found in the oral cavity, occurring at various sites but most commonly on the tongue. This often leads to macroglossia, presenting as a compressible, painless swelling that typically necessitates surgical excision when it causes functional issues.</p><p><strong>Conclusion: </strong>Oral lymphangioma generally has a good prognosis, especially when treated appropriately. However, they can recur if not completely excised.</p>","PeriodicalId":48113,"journal":{"name":"International Journal of Surgery Case Reports","volume":"127 ","pages":"110863"},"PeriodicalIF":0.6,"publicationDate":"2025-01-11","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143030153","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
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International Journal of Surgery Case Reports
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