vascular anomalies

血管异常
  • 文章类型: Journal Article
    背景:先前已经报道了内窥镜切除血管异常(VA)。然而,没有研究比较儿童内镜切除手术(ERS)和开放切除手术(ORS).我们旨在比较两种方法在小儿VA中的临床和美容结果。
    方法:在2018年6月至2023年6月之间,对138例接受ERS或ORS的儿科VA患者进行了回顾性分析。进行倾向评分匹配(PSM)以最大程度地减少选择偏差。采用基于患者满意度的瘢痕记忆评价量表(SCAR)和数字评定量表(NRS)进行美容评价。
    结果:在PSM年龄之后,病变深度,病变的大小,和手术部位,对72例患者(ERS=24,ORS=48)进行分析。接受ERS的患者手术时间更长(164.25±18.46vs.112.85±14.26分钟;P<0.001),估计失血较少(5.42±2.15vs.18.04±1.62ml;P<0.001),住院时间中位数较短(4.50[3.00-5.00]vs.6.00[5.00-6.00]天;P<0.001)。随访时间ERS组为8.04±1.23个月,ORS组为8.56±1.57个月。对于美学结果,ERS的总体SCAR评分中位数低于ORS(2[1-3]vs.5[4-5];P<0.001),和“疤痕扩散”的分量表,“\”色素沉着,“\”轨迹标记或缝合标记,“”和“总体印象”更好。NRS评分中位数较高(8[7-8]vs.6[5-6];P<0.001),疤痕长度较短(2.18±0.30vs.ERS组8.75±1.98cm;P<0.001)高于ORS组。两组总并发症发生率及复发率比较差异无统计学意义。
    结论:内镜手术是一种安全有效的选择,适用于四肢和躯干的小儿VA。它提供了改善美学结果和减少术后伤口愈合时间的优点。
    BACKGROUND: Endoscopic resection has been reported for vascular anomalies (VA) previously. However, there is no study comparing endoscopic resection surgery (ERS) with open resection surgery (ORS) in children. We aimed to compare clinical and cosmetic outcomes between two approaches in pediatric VA.
    METHODS: Between June 2018 and June 2023, 138 pediatric VA patients undergoing ERS or ORS were retrospectively reviewed. Propensity score matching (PSM) was performed to minimize selection bias. The Scar Cosmesis Assessment and Rating (SCAR) Scale and numerical rating scale (NRS) based on patient satisfaction were used for cosmetic assessment.
    RESULTS: After PSM for age, depth of lesion, size of lesion, and site of surgery, 72 patients (ERS = 24, ORS = 48) were analyzed. Patients undergoing ERS had longer operative time (164.25 ± 18.46 vs. 112.85 ± 14.26 min; P < 0.001), less estimated blood loss (5.42 ± 2.15 vs. 18.04 ± 1.62 ml; P < 0.001), and shorter median hospital stay (4.50 [3.00-5.00] vs. 6.00 [5.00-6.00] days; P < 0.001). The follow-up time was 8.04 ± 1.23 month for ERS group and 8.56 ± 1.57 month for ORS group. For aesthetic results, the median overall SCAR score in ERS was lower than that in ORS (2 [1-3] vs. 5 [4-5]; P < 0.001), and the subscales of \"scar spread,\" \"dyspigmentation,\" \"track marks or suture marks,\" and \"overall impression\" were better. The median NRS score was higher (8 [7-8] vs. 6 [5-6]; P < 0.001) and length of scars was shorter (2.18 ± 0.30 vs. 8.75 ± 1.98 cm; P < 0.001) in ERS group than those in ORS group. The incidences of total complications and recurrence showed no significant difference between two groups.
    CONCLUSIONS: Endoscopic surgery can be a safe and effective option for pediatric VA in the limbs and trunk. It offers the advantages of improving aesthetic outcomes and reducing postoperative wound healing time.
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  • 文章类型: Journal Article
    患有复杂罕见疾病的患者通常需要多位专家的护理。需要在初级护理和专科护理之间进行有效协调,以确保患者获得高质量的护理。先前的研究已经记录了初级保健临床医生提供专业护理转诊的重要性,以及专家帮助患者做出诊断的重要性。然而,对初级保健临床医生在罕见疾病患者的持续护理中的作用知之甚少。在目前的研究中,我们探讨了初级保健临床医师在罕见和复杂血管异常治疗中的作用.
    采用半结构化的定性访谈方法,对34名家长和25名来自倡导组的复杂血管异常患者进行了调查。我们询问了参与者的诊断,护理经验,与临床医生沟通。我们使用主题分析来确定说明初级保健临床医生角色的主题。
    PCC角色的特征在于四种行为。支持行为包括更多地了解血管异常,并询问参与者从专家那里获得的护理。促进包括提供转介,订购测试,参与解决问题。干扰包括未能提供转介或帮助参与者协调护理,订购不正确的测试,或者提出不恰当的建议。忽视包括狭隘地关注初级保健需求,而不关注血管异常。
    结果揭示了改善血管异常患者的初级保健的机会。忽视和干扰行为进一步促进了血管异常患者的初级和专科护理之间的划分,并阻止了患者接受全面的初级护理。支持和促进行为传达了对血管异常护理的真正兴趣,并致力于帮助患者和父母。
    UNASSIGNED: Patients with complex rare disorders often require the care of multiple specialists. Effective coordination between primary and specialty care is needed to ensure patients receive high-quality care. Previous research has documented the importance of primary care clinicians providing referrals to specialty care and the importance of specialists in helping patients reach a diagnosis. However, little is known about primary care clinicians\' roles in the ongoing care of patients with rare disorders. In the current study, we explored the role of primary care clinicians in the care of rare and complex vascular anomalies.
    UNASSIGNED: Data were collected using semi-structured qualitative interviews with 34 parents and 25 adult patients recruited from advocacy groups for patients with complex vascular anomalies participated. We asked participants about their diagnosis, care experiences, and communication with clinicians. We used thematic analysis to identify themes illustrating the roles of primary care clinicians.
    UNASSIGNED: PCC roles were characterized by four behaviors. Supporting behaviors included learning more about vascular anomalies and asking participants about the care they received from specialists. Facilitating included providing referrals, ordering tests, and engaging in problem-solving. Interfering included failing to provide referrals or help participants coordinate care, ordering incorrect tests, or making inappropriate recommendations. Disregarding included focusing narrowly on primary care needs and not showing concern about the vascular anomaly.
    UNASSIGNED: The results reveal opportunities to improve primary care for patients with vascular anomalies. Disregarding and interfering behaviors furthered the division between primary and specialty care for patients with vascular anomalies and prevented patients from receiving comprehensive primary care. Supporting and facilitating behaviors convey genuine interest in the care of the vascular anomaly and a commitment to helping the patient and parent.
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  • 文章类型: Journal Article
    以前从未探索过与普萘洛尔治疗婴儿血管瘤(IH)相关的治疗负担(BOT)。修改后的验证问卷,治疗负担问卷,和一对一半结构化访谈用于评估普萘洛尔用于IH的BOT。在80名护理人员中,总体负担得分非常低,为10分之1.2;访谈的主题分析将主题分为行政管理,监测,金融,和相关的异常。普萘洛尔治疗IH的BOT非常低,但可以通过提供与喂养频率和低血糖风险相关的基于年龄的风险分层来进一步降低。关于睡前剂量时间的务实建议,减少生命体征监测的频率。
    The burden of treatment (BOT) related to propranolol treatment for infantile hemangiomas (IH) has never previously been explored. A modified validated questionnaire, the Treatment Burden Questionnaire, and one-on-one semi-structured interviews were used to assess the BOT for propranolol for IH. Out of 80 caregivers, the overall burden score was very low at 1.2 out of 10; thematic analysis of interviews grouped themes into administration, monitoring, financial, and associated anomalies. The BOT of propranolol for IH is very low but could be reduced further by offering age-based risk stratification related to feeding frequency and risk of hypoglycemia, pragmatic advice around timing of doses before sleep, and reducing frequency of vital sign monitoring.
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  • 文章类型: Journal Article
    高温要求A1(HTRA1)是丝氨酸蛋白酶家族的成员,包含四个结构域:IGFBP结构域,Kazal域,蛋白酶结构域和PDZ结构域。HTRA1编码一种丝氨酸蛋白酶,在脉管系统中广泛表达的分泌蛋白。HTRA1通过其蛋白水解活性调节广泛的生理过程,并参与多种血管异常相关疾病。本文综述了HTRA1在血管异常相关遗传性脑小血管病(CSVD)发生发展中的作用,年龄相关性黄斑变性(AMD),肿瘤和其他疾病。通过相关研究进展了解HTRA1在调节信号通路或重折叠中的作用,易位,细胞外基质(ECM)蛋白的降解,因此直接或间接调节血管生成,血管重塑,在血管稳态中发挥重要作用,进一步了解HTRA1在血管异常相关疾病中的作用机制对于HTRA1作为相关疾病的治疗靶点具有重要意义。
    High temperature requirement A1 (HTRA1) is a member of the serine protease family, comprising four structural domains: IGFBP domain, Kazal domain, protease domain and PDZ domain. HTRA1 encodes a serine protease, a secreted protein that is widely expressed in the vasculature. HTRA1 regulates a wide range of physiological processes through its proteolytic activity, and is also involved in a variety of vascular abnormalities-related diseases. This article reviews the role of HTRA1 in the development of vascular abnormalities-related hereditary cerebral small vessel disease (CSVD), age-related macular degeneration (AMD), tumors and other diseases. Through relevant research advances to understand the role of HTRA1 in regulating signaling pathways or refolding, translocation, degradation of extracellular matrix (ECM) proteins, thus directly or indirectly regulating angiogenesis, vascular remodeling, and playing an important role in vascular homeostasis, further understanding the mechanism of HTRA1\'s role in vascular abnormality-related diseases is important for HTRA1 to be used as a therapeutic target in related diseases.
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  • 文章类型: Journal Article
    全位反位(SIT)是一种罕见的疾病,以镜像解剖为特征,这可能在手术干预中带来独特的挑战和潜在的血管异常,尤其是胃癌患者。
    我们的目的是描述一例SIT患者的胃腺癌的罕见病例,并对现有的有关手术策略的文献进行全面回顾。血管异常,以及在不同的地理区域和技术方法中观察到的结果。
    对一例39岁男性SIT患者进行了全面检查,该患者成功进行了远端胃切除术并进行了D2淋巴结清扫术,并进行了大量文献综述。该评论涵盖47篇文章,整理49例诊断为SIT和胃癌的患者的手术入路和血管异常数据。
    患者在95分钟内接受了根治性远端胃切除术和BillrothII并进行Braun吻合术,发生最小的术中失血(100ml)。术后病理证实为中低分化胃腺癌(pT3N0M0),S-1辅助化疗6个月后无复发或转移迹象。文献综述显示约20%的报告病例血管异常,强调其手术意义。手术策略值得注意的变化,手术时间,失血,观察不同手术方式的并发症,全面了解此类案件的实际管理。
    尽管与SIT相关的固有挑战,通过精心的术前计划和了解血管异常,可以成功应用各种手术技术。这种不同的手术经验的汇编在许多记录的病例中寻求提供一个统一的资源,以完善手术策略和提高SIT胃癌患者的术后结果。强调了在这一生态位领域进行进一步研究的必要性。
    UNASSIGNED: Situs inversus totalis (SIT) is an uncommon disorder characterized by mirror-image anatomy, which can present unique challenges and potential vascular anomalies in surgical interventions, particularly in gastric cancer patients.
    UNASSIGNED: We aim to delineate a rare case of gastric adenocarcinoma in a SIT patient and conduct a thorough review of the existing literature concerning surgical strategies, vascular anomalies, and outcomes observed across varied geographic locales and technological approaches.
    UNASSIGNED: A thorough examination of a case involving a 39-year-old male SIT patient who underwent a successful distal gastrectomy with D2 lymph node dissection is presented alongside an expansive literature review. The review encompasses 47 articles, collating data on surgical approaches and vascular anomalies across 49 patients diagnosed with SIT and gastric cancer.
    UNASSIGNED: The patient underwent curative distal gastrectomy and Billroth II with Braun anastomosis within 95 minutes, incurring minimal intraoperative blood loss (100ml). Postoperative pathology confirmed moderately to poorly differentiated gastric adenocarcinoma (pT3N0M0), with no signs of recurrence or metastasis after 6 months of S-1 adjuvant chemotherapy. The literature review revealed vascular anomalies in approximately 20% of reported cases, accentuating its surgical significance. Noteworthy variations in surgical strategies, operative times, blood loss, and complications across different surgical modalities were observed, providing a comprehensive view into the practical management of such cases.
    UNASSIGNED: Despite the inherent challenges associated with SIT, various surgical techniques can be successfully applied with meticulous preoperative planning and understanding vascular anomalies. This compilation of diverse surgical experiences across numerous documented cases seeks to provide a consolidated resource for refining surgical strategies and enhancing postoperative outcomes for gastric cancer patients with SIT, underscoring the imperativeness of further research in this niche domain.
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  • 文章类型: Journal Article
    血管畸形的临床和组织学诊断之间存在差异。
    为了评估国际血管异常研究学会(ISSVA)分类在根据临床和(免疫)组织学参数诊断良性血管异常中的有效性,关注淋巴分化和血管增生。
    对121例头颈部良性皮肤和软组织血管异常(不包括化脓性肉芽肿和老年血管瘤)患者进行回顾性研究,方法是对淋巴管进行多重免疫组织化学染色(D2-40),内皮血管,和增殖细胞(Ki67)。ISSVA分类后修订了临床和组织学诊断。
    最初,64个病灶被诊断为肿瘤,57个病灶被诊断为畸形。ISSVA分类后的修订诊断显示27个肿瘤,90畸形(22.2%淋巴管),和4个非ISSVA。免疫染色显示121例中24例(19.8%)淋巴分化,其中20人是畸形。121例中41例(33.8%)有增殖活性(Ki67+),其中8例为动静脉畸形。
    材料的质量和大小(活检与切除)和临床信息。
    通过结合其他免疫染色技术来评估淋巴分化和增殖活性,可以大大提高ISSVA分类后识别血管异常的组织学和临床联合方法的诊断准确性。特别是在确定血管畸形的发生。
    UNASSIGNED: Discrepancies have been noted between the clinical and histologic diagnosis of vascular malformations.
    UNASSIGNED: To evaluate the effectiveness of the International Society for Study of Vascular Anomalies (ISSVA) classification in diagnosing benign vascular anomalies based on clinical and (immuno) histologic parameters, focusing on lymphatic differentiation and vascular proliferation.
    UNASSIGNED: A retrospective study of 121 consecutive patients with benign skin and soft-tissue vascular anomalies located in the head and neck region (pyogenic granulomas and angioma senilis were excluded) by applying multiplex immunohistochemistry staining for lymph vessels (D2-40), endothelial blood vessels, and proliferating cells (Ki67). Clinical and histologic diagnosis was revised after the ISSVA classification.
    UNASSIGNED: Initially, 64 lesions were diagnosed as tumors and 57 as malformations. Revision diagnosis following the ISSVA classification revealed 27 tumors, 90 malformations (22.2% lymphatic), and 4 non-ISSVA. Immunostaining showed lymphatic differentiation in 24 (19.8%) of 121 cases, of which 20 were malformations. Proliferative activity (Ki67+) was found in 41 (33.8%) of 121 cases, of which 8 were arteriovenous malformations.
    UNASSIGNED: Quality and size of materials (biopsies vs resections) and clinical information.
    UNASSIGNED: The diagnostic accuracy of combined histologic and clinical approaches for identifying vascular anomalies following the ISSVA classification can be substantially enhanced by incorporating additional immunostaining techniques to evaluate lymphatic differentiation and proliferative activity, particularly in identifying the occurrence of vascular malformations.
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  • 文章类型: Multicenter Study
    目的:接受口服西罗莫司的血管异常(VA)患者可能存在感染性并发症的高风险。已提倡使用甲氧苄啶-磺胺甲恶唑(TMP-SMZ)进行抗生素预防。然而,关于这个主题的证据分析很少。本研究评估了预防性TMP-SMZ对接受西罗莫司单药治疗的VA患者感染发生率的影响。
    方法:回顾性研究,从8月开始对所有接受西罗莫司治疗的VA患者进行多中心图表审查,2013年1月,2021年。
    结果:在2017年1月之前,112例患者接受了西罗莫司治疗,没有预防抗生素。在随后的时期,195名患者在西罗莫司治疗期间接受TMP-SMZ治疗至少12个月。在西罗莫司治疗的最初12个月中,至少有一次严重感染的患者百分比在两组之间没有差异(差异,1.1%;95%CI-7.0-8.0%)。我们观察到组间个体感染或总不良事件的发生率没有差异。由于不良事件导致的西罗莫司停药率在组间没有显著差异。
    结论:我们证明预防性TMP-SMZ不会降低接受西罗莫司单药治疗的VA患者的感染发生率或改善耐受性。
    Patients with vascular anomalies (VAs) who receive oral sirolimus may be at high risk of infectious complications. Antibiotic prophylaxis with trimethoprim-sulfamethoxazole (TMP-SMZ) has been advocated. However, there have been few evidence-based analyses on this topic. This study assessed the effect of prophylactic TMP-SMZ on the incidence of infections in VA patients receiving sirolimus monotherapy.
    A retrospective, multicenter chart review was performed on all VA patients receiving sirolimus treatment from August, 2013 to January, 2021.
    Before January 2017, 112 patients were treated with sirolimus without antibiotic prophylaxis. In the subsequent period, 195 patients were treated with TMP-SMZ for at least 12 months during sirolimus therapy. The percentage of patients with at least one serious infection during the initial 12 months of sirolimus treatment did not differ between the groups (difference, 1.1%; 95% CI - 7.0-8.0%). We observed no difference in the incidence of individual infection or total adverse events between the groups. The rate of sirolimus discontinuation due to adverse events did not differ significantly between groups.
    We demonstrated that prophylactic TMP-SMZ does not decrease the incidence of infection or improve tolerance in VA patients receiving sirolimus monotherapy.
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  • 文章类型: Journal Article
    背景:这项研究描述了儿科患者毛细血管静脉畸形的治疗方案,并报告了2014年至2022年在巴里AldoMoro大学齿科的诊断和治疗病例的流行病学。
    方法:作者通过表面直径(<1厘米,1-3厘米,>3cm)和超声深度延伸(≤5mm,>5毫米)。所有患者均接受脉冲模式二极管激光经粘膜光凝(8-12W/cm2);那些患有宽(>3cm)和深(>5mm)畸形的患者接受了病灶内光凝,(13W/cm2)。根据患儿的依从性和病变范围接受全身麻醉。随访持续了6个月。
    结果:共有22名女性和14名男性(年龄范围4-18岁)出现了63例毛细血管静脉畸形。5名Sturge-Weber综合征患者,七个遗传性出血性毛细血管扩张症,5例血管瘤病表现出多种畸形。作者没有发现术中或术后并发症。17例病灶>1cm和>5mm深的患者需要多次激光治疗才能愈合。
    结论:当前研究的结果支持二极管激光光凝作为治疗儿科患者口腔内和口周毛细血管静脉畸形的金标准。
    BACKGROUND: This study describes the management protocol for capillary-venous malformations in pediatric patients and reports the epidemiology of diagnosed and treated cases at the Unit of Odontostomatology of the Aldo Moro University of Bari from 2014 to 2022.
    METHODS: The authors classified the intraoral and perioral capillary-venous malformations by superficial diameter (<1 cm, 1-3 cm, >3 cm) and ultrasonographical depth extension (≤5 mm, >5 mm). All patients underwent pulsed-mode diode laser transmucosal photocoagulation (8-12 W/cm2); those with malformations that were wide (>3 cm) and deep (>5 mm) received intralesional photocoagulation, too (13 W/cm2). The children received general anesthesia based on their compliance and lesions\' extension. The follow-up lasted six months.
    RESULTS: A total of 22 females and 14 males (age range 4-18 years) presented 63 capillary-venous malformations. Five patients with Sturge-Weber syndrome, seven with hereditary hemorrhagic telangiectasia, and five with angiomatosis showed multiple malformations. The authors found no intraoperative or postoperative complications. Seventeen patients with lesions >1 cm and >5 mm deep required multiple laser sessions to heal.
    CONCLUSIONS: The results of the current study support diode laser photocoagulation as the gold standard for the treatment of intraoral and perioral capillary-venous malformations in pediatric patients.
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  • 文章类型: Journal Article
    目的:复杂的血管异常(VA)在常规治疗中可能难以控制和控制,并可能导致致死性结局。我们进行了一个前瞻性的,多中心II期试验,以评估西罗莫司在患有复杂VAs的儿科患者中的疗效和安全性。
    方法:符合条件的患者需要年龄为0至14岁,并且患有复杂的VA。患者每日口服西罗莫司治疗12个月。主要终点是反应,这是使用顺序体积磁共振成像测量的。次要终点是疾病严重程度评分和生活质量。
    结果:在意向治疗基础上登记的126名患者中,98人(77.8%)对西罗莫司有客观反应,病变体积减少≥20%。与动静脉畸形相比,对于常见的淋巴畸形患者,反应率更高(>80%),静脉畸形,kaposiform血管内皮瘤,合并有突出的静脉和/或淋巴成分的畸形(P<0.05)。83.3%和79.4%的患者获得了疾病严重程度评分和生活质量的改善,分别。最常见的不良事件是47例患者的粘膜炎。更严重的不良事件包括3例患者的可逆性4级肺炎和1例患者的4级上呼吸道感染。所有这些不良事件被认为至少可能与治疗有关。
    结论:西罗莫司对于患有各种类型的复杂血管腔的儿科患者是一种明显有效的选择。需要密切监测可能的不良事件。本试验的结果是未来使用新治疗方法进行前瞻性研究的基础。
    OBJECTIVE: Complicated vascular anomalies (VAs) can be intractable and uncontrollable using conventional treatment and can result in lethal outcomes. We undertook a prospective, multicenter phase II trial to evaluate the efficacy and safety of sirolimus in pediatric patients with complicated VAs.
    METHODS: Eligible patients were required to be aged 0 to 14 years and to have a complicated VA. The patients were treated with daily oral sirolimus for 12 months. The primary endpoint was the response, which was measured using sequential volumetric magnetic resonance imaging. The secondary endpoints were the disease severity score and quality of life.
    RESULTS: Of 126 patients enrolled on an intention-to-treat basis, 98 (77.8%) had had an objective response to sirolimus, with a ≥20% decrease in lesion volume. Compared with those with arteriovenous malformations, the response rates were higher (>80%) for patients with common lymphatic malformations, venous malformations, kaposiform hemangioendothelioma, and combined malformations with a prominent venous and/or lymphatic component (P < .05). Improvements in the disease severity score and quality of life were obtained in 83.3% and 79.4% of patients, respectively. The most common adverse event was mucositis in 47 patients. More serious adverse events included reversible grade 4 pneumonitis in 3 patients and grade 4 upper respiratory infection in 1 patient. All these adverse events were considered at least possibly related to the treatment.
    CONCLUSIONS: Sirolimus is an apparently effective option for pediatric patients with various types of complicated VAs. Close monitoring of possible adverse events is required. The results from the present trial are the basis for future prospective studies using new therapeutic approaches.
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  • 文章类型: Journal Article
    Vascular malformations (Vascular Malformation) of soft tissue are a consequence of abnormal development of vascular elements during embryogenesis. They are named after the vessel responsible. Their presence at birth raises the hypothesis of an intrauterine etiology of Vascular Malformation, but the influence of other factors may play an important role in their appearance, localization, and size. The literature suggests that treatment of Vascular Malformation is rarely surgical. Surgery is applied in cases of voluminous malformations and in cases of possible hemorrhagic complications. Meanwhile, patient outcomes have improved with new discoveries in laser technology for the treatment of Vascular Malformation, especially those of capillaries and the results are, in most of the cases, quite satisfactory and without significant complications. After institutional review board approval, we collected and analyzed the medical records of 90 patients diagnosed with Vascular Malformation, between January 2015 and December 2019 at the service of Burns and Plastic Surgery, \"Mother Teresa\" University Hospital Center of Tirana, Albania and from another private clinic in Tirana. Data were collected from the medical records on patient\'s charts. The aim of this study is to analyze different treatment modalities and the outcomes achieved for each group.
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