Ahmed Nazmin, Ferini Gianluca, Haque Moududul, Umana Giuseppe Emmanuele, Scalia Gianluca, Chaurasia Bipin, Vats Atul, Rahman Asifur
Department of Neurosurgery, Ibrahim Cardiac Hospital and Research Institute (A Centre for Cardiovascular, Neuroscience and Organ Transplant Units), Shahbag, Dhaka 1000, Bangladesh.
Department of Radiation Oncology, REM Radioterapia srl, 95029 Catania, Italy.
Life (Basel). 2022 Apr 6;12(4):548. doi: 10.3390/life12040548.
(1) Introduction: Primary intraosseous osteolytic meningiomas (PIOM) are non-dural-based tumors predominantly presenting an osteolytic component with or without hyperostotic reactions. They are a subset of primary extradural meningiomas (PEM). In this study, we present a peculiar case with a systematic literature review and propose a new classification considering the limitations of previous classification systems. (2) Materials and Methods: Using a systematic search protocol in Google Scholar, PubMed, and Scopus databases, we extracted all case studies on PIOM published from inception to December 2020. A 46-year-old female patient form Dhaka, Bangladesh, was also described. The search protocol was performed according to the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) guidelines. (3) Results: Here, we present a 46-year-old female patient with PIOM who successfully underwent bifrontal craniotomy and gross total removal (GTR) of the tumor. At 6-month follow-up, no tumor recurrence was shown. Including our new case, 55 total cases from 47 articles were included in the analysis. PIOMs were in closer frequency among males (56.4%) and females (43.6%). The most common tumor location was the frontal and parietal calvarium, most commonly in the frontal bone (29.1%). Surgical resection was the predominant modality of treatment (87.3%); only 1.8% of patients were treated with radiotherapy, and 5.4% received a combination of surgery and radiotherapy. Gross total resection (GTR) was achieved in 80% of cases. Extracranial extension was reported in 41.8% of cases, dural invasion in 47.3%, and recurrence in 7.3%. Whole-body 68 Ga-DOTATOC PET/CT has also been reported as a useful tool both for differential diagnosis, radiotherapy contouring, and follow-up. Current treatments such as hydroxyurea and bevacizumab have variable success rates. We have also suggested a new classification which would provide a simple common ground for further research in this field. (4) Conclusions: Surgical resection, especially GTR, is the treatment of choice for PIOM, with a high GTR rate and low risk of complications and mortality. More research is needed on the differential diagnosis and specific treatment of PIOM.
(1) 引言：原发性骨内溶骨性脑膜瘤（PIOM）是一种非硬膜源性肿瘤，主要表现为溶骨成分，伴有或不伴有骨质增生反应。它们是原发性硬膜外脑膜瘤（PEM）的一个子集。在本研究中，我们报告了一例特殊病例，并进行了系统的文献综述，鉴于既往分类系统的局限性，提出了一种新的分类方法。(2) 材料与方法：我们使用谷歌学术、PubMed和Scopus数据库中的系统检索方案，提取了从开始到2020年12月发表的所有关于PIOM的病例研究。还描述了一名来自孟加拉国达卡的46岁女性患者。检索方案按照系统评价和Meta分析的首选报告项目（PRISMA）指南进行。(3) 结果：在此，我们报告了一名患有PIOM的46岁女性患者，她成功接受了双额开颅手术并实现了肿瘤的全切（GTR）。在6个月的随访中，未发现肿瘤复发。包括我们的新病例在内，分析纳入了47篇文章中的55例病例。PIOM在男性（56.4%）和女性（43.6%）中的发病频率相近。最常见的肿瘤部位是额顶颅骨，最常位于额骨（29.1%）。手术切除是主要的治疗方式（87.3%）；仅1.8%的患者接受了放疗，5.4%的患者接受了手术和放疗联合治疗。80%的病例实现了全切（GTR）。41.8%的病例报告有颅外扩展，47.3%有硬膜侵犯，7.3%有复发。全身68 Ga-DOTATOC PET/CT也被报道为一种在鉴别诊断、放疗靶区勾画和随访方面都有用的工具。目前的治疗方法如羟基脲和贝伐单抗的成功率各不相同。我们还提出了一种新的分类方法，可为该领域的进一步研究提供一个简单的共同基础。(4) 结论：手术切除，尤其是全切（GTR），是PIOM的首选治疗方法，全切率高，并发症和死亡率风险低。需要对PIOM的鉴别诊断和特异性治疗进行更多研究。
