rare case

罕见病例
  • 文章类型: Case Reports
    表皮样和皮样囊肿是良性的,通常缓慢生长的病变被分类为外胚层包涵囊肿。这些囊肿形成时,上皮残留物在第一和第二分支弓闭合期间被困住;然而,少数病例与创伤有关或与医源性有关。根据囊肿发育史和超声等影像学检查进行诊断。手术切除是避免并发症的选择。我们报告了一例17岁男性患者胸骨前区域皮样囊肿的病例报告。
    Epidermoid and dermoid cysts are benign, usually slow-growing lesions classified as ectodermal inclusion cysts. These cysts form when epithelial remnants become trapped during the closure of the first and second branchial arch; however, a few cases are related to trauma or are iatrogenic. Diagnosis is made based on the cyst development history and imaging such as ultrasound. Surgical excision is the treatment of choice to avoid complications. We report a case report of a dermoid cyst in the pre-sternal region in a 17-year-old male patient.
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  • 文章类型: Journal Article
    背景:硬脑膜海绵状畸形,尤其是在天幕上,非常罕见。在现有文献中,迄今为止,只有10例病例被描述。
    方法:作者介绍了一名46岁的男性患者的病例,该患者具有1厘米的幕下病变,可疑为脑膜瘤,在常规磁共振成像(MRI)中发现了眩晕。病变随访1.5年,信号和大小无变化。然而,患者担心病变并要求切除.切除成功,没有任何神经系统后遗症。然而,组织学评估显示海绵状畸形。术后计算机断层扫描和MRI显示完全切除。术前MRI特征,术中图像,还有一段视频,以及组织学评估,显示。关于文献讨论了该案例。
    结论:天幕海绵状畸形是极其罕见的模拟脑膜瘤;因此,他们需要被考虑在内。DOTATOC正电子发射断层扫描可能有助于在这些情况下进行区分。考虑到文献中报道的病例,在大肿瘤的情况下,术前血管造影和可能的栓塞可能是有帮助的。https://thejns.org/doi/10.3171/CASE24168.
    BACKGROUND: Cavernous malformations of the dura, especially of the tentorium, are exceedingly rare. In the available literature, only 10 cases have been described to date.
    METHODS: The authors present the case of a 46-year-old male patient with a 1-cm infratentorial lesion suspicious for meningioma that was found on routine magnetic resonance imaging (MRI) performed for vertigo. The lesion was followed for 1.5 years with no change in signal and size. Nevertheless, the patient was concerned about the lesion and requested removal. The removal was successful and without any neurological sequelae. However, histological evaluation demonstrated a cavernous malformation. Postoperative computed tomography and MRI showed complete removal. Preoperative MRI characteristics, intraoperative images, and a video, as well as histological evaluation, are shown. The case is discussed with respect to the literature.
    CONCLUSIONS: Cavernous malformations of the tentorium are extremely rare and mimic meningiomas; thus, they need to be taken into account. DOTATOC positron emission tomography may help to differentiate in these cases. Considering the cases reported in the literature, in cases of large tumors, preoperative angiography and possibly embolization may be helpful. https://thejns.org/doi/10.3171/CASE24168.
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  • 文章类型: Journal Article
    泪小管腺瘤(CA)是一种罕见的唾液腺良性肿瘤,主要影响老年女性,对上唇有强烈的偏爱。虽然CA通常出现在小唾液腺,它在腮腺的发生是非常罕见的。在这份报告中,我们在腮腺出现了一个独特的CA病例,增加了只有8个记录在案的文献。病人,一位57岁的亚洲男性,表现为持续8年的左腮腺无痛肿胀。临床检查和影像学检查发现了一个分叶状肿块,提示手术干预。病人接受了浅表腮腺切除术,切除组织的病理检查证实了CA的诊断,没有恶性肿瘤的迹象.此案例说明了与CA、特别是考虑到它在腮腺的罕见表现。准确的诊断依赖于手术活检,仔细的手术计划势在必行,尤其是考虑到面神经的距离.我们的案例强调需要提高对CA独特演示的认识,特别是在亚洲人口中。考虑到复发的可能性,长期随访至关重要。需要进一步的研究来阐明CA的生物学行为,并完善管理策略以获得最佳患者结果。
    Canalicular adenoma (CA) is a rare benign tumor of the salivary glands, predominantly affecting elderly females, with a strong predilection for the upper lip. While CA commonly arises in the minor salivary glands, its occurrence in the parotid gland is exceptionally rare. In this report, we present a unique case of CA in the parotid gland, adding to the scant literature with only 8 documented instances. The patient, a 57-year-old Asian male, presented with a painless swelling in the left parotid gland that had been persisting for 8 years. Clinical examination and imaging studies identified a lobulated mass, prompting surgical intervention. The patient underwent a superficial parotidectomy, and pathological examination of the excised tissue confirmed the diagnosis of CA, with no signs of malignancy. This case illustrates the diagnostic and management challenges associated with CA, particularly given its rare presentation in the parotid gland. Accurate diagnosis is reliant on surgical biopsy, and careful surgical planning is imperative, especially considering the proximity of the facial nerve. Our case underscores the need for heightened awareness of CA\'s unique presentations, particularly within the Asian population. Given the potential for recurrence, long-term follow-up is essential. Further research is needed to elucidate the biological behavior of CA and to refine management strategies for optimal patient outcomes.
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  • 文章类型: Case Reports
    背景:右侧膈疝是成人肠梗阻和绞窄的一种非常罕见的原因,这通常是一种先天性疾病。
    方法:A-55岁男性到我院急诊科就诊,主诉腹痛,恶心,呕吐,腹胀,发烧,便秘4天.在体检时,病人是原纤维,有毒,心动过速,和低血压。病人腹部扩张,肠鸣音夸张,腹部压痛,守卫,和刚性主要在右上象限。打击乐器有一定程度的节奏。直肠指检正常,没有大便受累的迹象。
    结论:膈疝患者常表现为内疝,监禁,阻塞,绞窄引起的缺血,或穿孔。患者可能出现呼吸道症状,如呼吸困难,胸部没有呼吸的声音,或腹部症状,如腹痛和肠扩张。
    结论:膈疝,这是一种罕见的情况,应包括在小肠梗阻的鉴别诊断中,以排除并发症。
    BACKGROUND: Right-side diaphragmatic hernia is a very rare cause of bowel obstruction and strangulation in adults, which is usually a congenital disorder.
    METHODS: A- 55-year-old male presented to the emergency department of our hospital complaining of abdominal pain, nausea, vomiting, abdominal distension, fever, and constipation for 4 days. On physical examination, the patient was fibril, toxic, tachycardic, and hypotensive. The patient had a distended abdomen with exaggerated bowel sounds, abdominal tenderness, guarding, and rigidity mostly in the right upper quadrant. There were some degrees of tempanicity on percussion. The digital rectal examination was normal with no evidence of impacted stool.
    CONCLUSIONS: Patients with a diaphragmatic hernia frequently present with manifestations of internal herniation, incarceration, obstruction, ischemia from strangulation, or perforation. The patient may present with respiratory symptoms such as dyspnea, absence of breath sounds in the thorax, or abdominal symptoms such as abdominal pain and bowel dilatation.
    CONCLUSIONS: Diaphragmatic hernia, which is a rare case, should be included in the differential diagnosis of small bowel obstruction to preclude complications.
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  • 文章类型: Case Reports
    血管周围上皮样细胞肿瘤(PEComas)是起源于血管周围上皮样细胞的罕见间充质肿瘤。在妇科系统中,子宫是受PEComas影响的最常见部位之一。大多数PEComas是良性的,患者通常预后良好。然而,恶性子宫PEComa罕见,需要更好的全面流行病学调查。迄今为止,有一些子宫PEComa报告病例。我们在此报告一例罕见的恶性PEComa发生在子宫体和子宫颈,可能伴有肺淋巴管平滑肌瘤病(PLAM)。此外,在PubMed和Medline数据库中抽取并收集55例恶性子宫PEComa。一方面,发病年龄,人口分布,临床表现,分析了转移部位和转移途径.另一方面,流行病学的总结,发病机制,诊断,给予子宫PEComa治疗。
    Perivascular epithelioid cell tumors (PEComas) are rare mesenchymal tumors originating from perivascular epithelioid cells. In gynecological system, the uterus is one of the most common sites affected by PEComas. Most PEComas are benign, and patients usually have a good prognosis. However, malignant uterus PEComa is rare, and better comprehensive epidemiological investigations are needed. To date, there are a few reported cases of uterus PEComa. We herein report a rare case of malignant PEComa occurred in the uterine corpus and cervix, possibly accompanied by pulmonary lymphangioleiomyomatosis (PLAM). In addition, 55 cases of malignant uterus PEComa were picked out and collected in the data base of PubMed and Medline. On the one hand, the age of onset, population distribution, clinical manifestations, metastatic sites and routes of metastasis were analysed. On the other hand, a summary of the epidemiology, pathogenesis, diagnosis, and treatments of uterus PEComa was given.
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  • 文章类型: Case Reports
    同侧近端,轴,股骨远端骨折极为罕见.治疗同侧股骨多层骨折可能具有挑战性和争议性。关于应首先修复的骨折类型的顺序和应使用的植入物的类型,仍然存在未回答的问题。
    方法:一名29岁的男性患者在一次摩托车事故后在急诊科进行了评估。患者左下肢明显变形,并抱怨左大腿疼痛。术前X线片显示,左大腿同侧股骨多节段骨折涉及股骨基颈骨折(AO/OTA31-B3),股骨横骨干骨折(AO/OTA32-A3)和关节外股骨髁上骨折(AO/OTA33-A2)。
    首先,我们进行了股骨近端防旋髓内钉,以稳定年轻成人股骨颈骨折并降低股骨头骨不连和缺血性坏死的发生率.固定轴和股骨远端骨折的下一步是执行股骨远端锁定钢板。实施这些程序后,EQ5D和Harris髋关节评分问卷显示出改善。
    结论:同侧股骨多节段骨折在治疗方面存在挑战和争议。在这种情况下,股骨近端防旋髓内钉和股骨远端锁定钢板是可行的选择,因为损伤的情况和较高的负面影响风险。在所有骨折都固定好之后,密切监测髋关节和膝关节以避免僵硬或挛缩是很重要的。
    UNASSIGNED: Ipsilateral proximal, shaft, and distal femur fractures are extremely uncommon. It might be challenging and contentious to treat ipsilateral multi-level femur fractures. There are still unanswered questions regarding the order of fracture types that should be repaired first and the type of implant that should be used.
    METHODS: A twenty-nine-year-old male patient was assessed at the emergency department after a motorcycle accident. The patient had a clearly deformed left lower extremity and was complaining of pain in the left thigh. Preoperative radiographs revealed ipsilateral multi-level femur fracture on the left thigh involved basicervical fracture of femur (AO/OTA 31-B3) with transverse shaft fracture of femur (AO/OTA 32-A3) and extra articular supracondylar femur fracture (AO/OTA 33-A2).
    UNASSIGNED: First, we performed proximal femur nail antirotation in order to stabilize the fracture of the femur neck and reduce the incidence of nonunion and avascular necrosis of the femoral head in young adults. The next step to fix the shaft and distal femur fracture was to perform the distal femur locking plate. The EQ5D and Harris Hip Score questionnaires showed improvement after implementing these procedures.
    CONCLUSIONS: Ipsilateral multi-level femur fractures have challenges and controversies in their management. In this situation, proximal femur nail antirotation and distal femur locking plates are viable options due to the condition of the injury and the higher risk of negative effects. After all fractures have been fixed, it is important to closely monitor the hip and knee joints to avoid stiffness or contracture.
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  • 文章类型: Case Reports
    纵裂脊髓是一种罕见的先天性异常,在解剖学上表现为脊髓在一个或几个部分的水平上的纵向分裂。它通常在儿童中诊断和治疗。我们介绍了一名25岁的女性患者,该患者在经历了严重的下腰痛的创伤经历后进入神经外科诊所,在视觉模拟量表(VAS)上评估了7分。腰椎区域的磁共振成像(MRI)发现了L3-L5水平的1型脊髓纵裂,L4和L5椎骨变形和轻度脊柱侧凸的证据。因为缺乏神经缺陷,患者的治疗为保守治疗,包括10天摄入非甾体类抗炎药(NSAIDs),并结合肌松药和物理治疗.在第15天,患者在VAS上进行2分评估。在第3个月和第6个月进行了神经系统随访检查,并对患者进行了VAS1评估,这证明保守治疗对于没有神经系统缺陷的成年人的脊髓纵裂是成功的。
    Diastematomyelia is an infrequent congenital anomaly that anatomically presents with a longitudinal division of the spinal cord at the level of one or several sections. It is generally diagnosed and treated in children. We present a 25-year-old female patient who has entered the Neurosurgery clinic after a traumatic experience with severe low back pain evaluated with 7 points on the visual analog scale (VAS). Magnetic resonance imaging (MRI) of the lumbar area discovered evidence for diastematomyelia type 1 at levels L3-L5 with deformations in L4 and L5 vertebrae and mild scoliosis. Because of the lack of neurological deficiency, the patient\'s treatment was conservative and included a 10-day intake of nonsteroid anti-inflammatory drugs (NSAIDs) in combination with myorelaxant and physiotherapy. On the 15th day, the patient was evaluated with 2 points on VAS. Neurological follow-up examinations were conducted on the third and the sixth month and the patient was evaluated with VAS 1, which proves that conservative treatment is successful for diastematomyelia in adults without neurological deficiency.
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  • 文章类型: Case Reports
    放线菌病是由革兰氏阳性菌引起的慢性化脓性感染,放线菌,通常定殖在口腔中,泌尿生殖道和胃肠道。它会导致粘膜屏障受损的机会性感染,并经常被误诊为恶性肿瘤。我们报告了一例58岁的女性,患有严重的腹痛,左下象限有明显的触痛。计算机断层扫描与对比显示增厚的横向,下降到乙状结肠壁和强烈的对比增强,类似于结肠炎伴肠系膜炎。在剖腹手术中,我们发现了一个粘连性肿块,累及乙状结肠与邻近的小肠和腹膜。我们决定进行粘连松解术和哈特曼手术。文化结果是否定的,而乙状结肠活检显示放线菌菌落的特征性硫颗粒。给予头孢他啶和甲硝唑的静脉抗生素组合14天,然后完全缓解症状。组织病理学和细菌学检查是诊断放线菌病的关键。患者需要长期抗生素治疗,但由于术前诊断困难,通常需要手术。
    我们介绍了印度尼西亚罕见的腹内放线菌病病例。该病例根据体格检查在术前诊断具有挑战性,剖腹探查术显示假瘤或模仿晚期结肠肿瘤。
    Actinomycosis is a chronic suppurative infection caused by gram-positive bacteria, Actinomyces, which commonly colonize the oral cavity, urogenital tract and gastrointestinal tract. It causes opportunistic infection where the mucosal barrier is compromised and is often misdiagnosed as malignancy. We report a case of a 58-year-old female with severe abdominal pain and a palpable tender mass in the left lower quadrant. Computed tomography scan with contrast showed thickening of the transverse, descending to sigmoid colon wall and intense contrast enhancement resembling colitis with mesenteritis. At laparotomy, we found an adherent mass involving sigmoid colon with adjacent small bowel and peritoneum. We decided to perform adhesiolysis and Hartmann procedure. The culture result was negative, whereas the biopsy of sigmoid colon revealed characteristic sulfur granules of actinomycosis colony. Intravenous antibiotic combination of ceftazidime and metronidazole was administered for 14 days followed by complete resolution of symptoms. Histopathological and bacteriological examinations are keys to diagnose actinomycosis. Patients require long-term antibiotic therapy, but surgery is often required because preoperative diagnosis is difficult.
    UNASSIGNED: We presented the rare case of intra-abdominal actinomycosis in IndonesiaThe case was challenging to diagnose pre-operatively based on physical examination andExploratory laparotomy revealed pseudotumor mimicking advanced stage of colon neoplasm.
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  • 文章类型: Case Reports
    先天性鼻皮样和鼻窦囊肿(NDSCs)是罕见的先天性畸形,患病率为0.005%至0.0025%。早期诊断通常在生命的前三年进行,但在某些情况下,诊断可能会延迟。本病例研究阐述了一名22岁成人罕见的先天性鼻中线皮样囊肿的治疗过程。病人没有畸形的家族史,手术前颅内扩张也被排除。在手术过程中,开放鼻整形技术与截骨术一起使用。囊肿全部切除。总运行时间为6小时,术中或术后未观察到并发症。总之,病例介绍重点介绍了手术中可以使用的各种技术和方法,这些技术和方法以前没有练习过,以矫正畸形,同时也获得了良好的美学效果。
    Congenital nasal dermoid and sinus cysts (NDSCs) are rare congenital deformities with a prevalence rate of 0.005% to 0.0025%. Early diagnosis is usually made during the first three years of life, but in some cases, the diagnosis may be delayed. The present case study elaborates the treatment course of a 22-year-old adult with a rare congenital nasal midline dermoid cyst. The patient had no family history of the deformity, and intracranial extensions were also ruled out before surgery. Open rhinoplasty technique was used along with osteotomies during the surgical process. The cyst was removed in entirety. The total operating time was six hours, and no complications were observed during the intra-operative or post-operative period. In conclusion, the case presentation focuses on various techniques and methods that can be used during surgery that have not been practiced before to correct the deformity while achieving a good aesthetic result as well.
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    文章类型: Case Reports
    一名28岁的男子出现在我们的医院,左颈部和双侧腋下有一个快速增长的结节,有体重减轻的历史。自两年前以来,他一直在她的右宫颈结节,并进行了FNAC并诊断为肺外结核(EPTB),在左颈椎检查;肿块大小9厘米×7厘米,mobile,耳前淋巴结肿大3cmx3cm,淋巴结病锁骨上大小1cmx0,5cm。双侧淋巴结肿大Axila大小4cmx4cmx2xcm移动。其他体检是正常的。实验室试验Hb10,4d/dL,WBC14.250/mm3,LED78毫米/小时,D-二聚体1,81ug/mL,纤维蛋白原452mg/dL。HIV测试无反应性。HbsAg和HCV检测阴性。胸部CT扫描:前纵隔淋巴结扩大,直径0f2.9cm,右气管旁直径为1,2cm和1,1cm,和直径为1,3厘米的右肺门周围。和左肺门直径0,9。脾脏低密度病变测量2,3cm×1,6cm。患者接受了病理活检和免疫组织化学(IHC)活检,CD20+。CD3-,CD30+CD79a+,MUM1+,Ki6780-90%+,CD15-,BCL6+和BCL2+。对这个病人来说,我们开始了R-CHOP方案(利妥昔单抗375mg/m2(d1),环磷酰胺750mg/m2(d1),阿霉素50mg/m2(d1),长春碱1,2mg/m2(d1)和1泼尼松100mg(d1-d5)。我们介绍了PMGZL患者已获得完全缓解,尤其是化疗R-CHOP方案。
    A 28 -year-old man presented to our hospital with a rapidly growing nodule in the left cervical and bilateral axilla with a history of loss of weight. He has been experiencing a nodule in her right cervical since two years ago and had done FNAC and diagnosed extrapulmonary tuberculosis (EPTB), on Examination at left cervical colli; mass size 9cm x 7 cm, mobile, Lymphadenopathy at pre auricular size 3cm x 3cm, lymphadenopathy supraclavicular size 1cm x 0,5 cm. bilateral lymphadenopathy Axila size 4cmx 4cmx 2xcm mobile. The other physical exam was normal. Laboratory test Hb 10,4 d/dL, WBC 14.250/ mm3, LED 78 mm/hours, D-dimer 1,81 ug/mL, Fibrinogen 452 mg/dL. HIV test non-reactive. HbsAg and HCV test negative. CT-Scan Thorax: Enlarged Anterior mediastinal Lymph node with a diameter 0f 2.9 cm, right paratracheal with a diameter of 1,2cm and 1,1 cm, and right perihilar with a diameter of 1,3 cm. and the left perihilar diameter 0,9. And hypodense lesion of the spleen measuring 2,3cm x 1,6 cm. The patient underwent a biopsy with pathology biopsy and immunohistochemistry (IHC), CD 20+. CD 3-, CD 30+ CD79a +, MUM1 +, Ki67 80-90% +, CD15-, BCL6+ and BCL 2+. For this patient, we started an R-CHOP regimen (Rituximab 375 mg/m2 (d1), Cyclophopamid 750 mg/m2 (d1), Doxorubicin 50 mg/m2 (d1), Vincristine1,2 mg/m2 (d1) and 1 Prednisone 100 mg (d1-d5). We presented the patient with PMGZL has achieved a complete response, especially with chemotherapy R-CHOP regimens.
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