A Case of Juvenile Trabecular Ossifying Fibroma Arising from the Maxilla in a 2-Year-Old Child.

IF 0.9 4区 医学 Q4 MEDICINE, RESEARCH & EXPERIMENTAL
Yonago acta medica Pub Date : 2025-02-14 eCollection Date: 2025-02-01 DOI:10.33160/yam.2025.02.008
Makoto Kawasaki, Rieko Doi, Nobuyuki Fuji, Takayuki Tamura, Isamu Kodani
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引用次数: 0

Abstract

An ossifying fibroma is a fibroosseous lesion from the maxilla and mandible. Its histopathological subtypes include juvenile trabecular ossifying fibroma (JTOF) and juvenile psammomatoid ossifying fibroma. It commonly affects young individuals and is invasive in nature. A 2-year-old girl with a 2-week history of swelling in the left infraorbital region and a reduced opening of the left eye was referred to our department. Clinical examination revealed a diffuse bone-like swelling extending from the left infraorbital region to the left buccal region. Computed tomography revealed a well-defined 2-cm lesion, and magnetic resonance imaging revealed moderate-to-high signal intensity on T2-weighted imaging. A definitive diagnosis could not be arrived at based solely on imaging findings. A biopsy was initially planned; however, the tumor was extracted because it was relatively easy to separate from the surrounding bone. Histological findings showed fascicular and spiral hyperplasia of the tumor cells containing spindle nuclei and cytoplasm. Fibroma-like stroma, immature chondrocytes, and mature trabecular osteogenesis are also observed. Osteoblast-like or partially chondrocytic sequences were observed at the margins of hard tissue. Thus, a definitive diagnosis of JTOF was established. Although the tumor was extracted only in the present case, JTOF has a high recurrence rate; thus, resection of the surrounding bone is recommended following a definitive diagnosis. In this case, the period from onset to surgery was short, and early treatment allowed the tumor to be removed before it invaded the surrounding bone; therefore, extraction alone prevented recurrence. It can be distinguished from fibrous dysplasia, which has similar pathological findings, based on the observation of osteoblast-like structures at the edge of the trabecular bone. Moreover, the negative guanine nucleotide-binding protein alpha-stimulating activity of polypeptide 1 further confirmed JTOF. No signs of recurrence were observed in this case at 11 years and 9 months postoperatively.

2岁儿童上颌骨发生幼年小梁骨化性纤维瘤1例。
骨化纤维瘤是一种来自上颌骨和下颌骨的纤维骨性病变。其组织病理学亚型包括幼年型小梁骨化纤维瘤(JTOF)和幼年型沙沫样骨化纤维瘤。它通常影响年轻人,具有侵袭性。一名2岁女童,左眶下区肿胀2周,左眼开口缩小,被转介至我科。临床检查发现弥漫性骨样肿胀,从左侧眶下区延伸至左侧颊区。计算机断层扫描显示明确的2厘米病变,磁共振成像显示t2加权成像中至高信号强度。一个明确的诊断不能仅仅基于影像学发现。最初计划进行活检;然而,由于肿瘤相对容易与周围骨骼分离,因此我们将其取出。组织学发现肿瘤细胞呈束状和螺旋状增生,含有梭形核和细胞质。纤维瘤样基质、未成熟软骨细胞和成熟小梁成骨也被观察到。硬组织边缘可见成骨细胞样或部分软骨细胞序列。因此,确定了JTOF的明确诊断。虽然本例仅切除肿瘤,但JTOF复发率高;因此,在明确诊断后,建议切除周围的骨。在这种情况下,从发病到手术的时间很短,早期治疗可以在肿瘤侵犯周围骨骼之前将其切除;因此,单纯拔牙可预防复发。通过观察骨小梁边缘的成骨细胞样结构,可将其与具有相似病理表现的纤维性发育不良区分开来。此外,多肽1的负鸟嘌呤核苷酸结合蛋白α刺激活性进一步证实了JTOF。该病例术后11年9个月无复发迹象。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
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来源期刊
Yonago acta medica
Yonago acta medica MEDICINE, RESEARCH & EXPERIMENTAL-
CiteScore
1.60
自引率
0.00%
发文量
36
审稿时长
>12 weeks
期刊介绍: Yonago Acta Medica (YAM) is an electronic journal specializing in medical sciences, published by Tottori University Medical Press, 86 Nishi-cho, Yonago 683-8503, Japan. The subject areas cover the following: molecular/cell biology; biochemistry; basic medicine; clinical medicine; veterinary medicine; clinical nutrition and food sciences; medical engineering; nursing sciences; laboratory medicine; clinical psychology; medical education. Basically, contributors are limited to members of Tottori University and Tottori University Hospital. Researchers outside the above-mentioned university community may also submit papers on the recommendation of a professor, an associate professor, or a junior associate professor at this university community. Articles are classified into four categories: review articles, original articles, patient reports, and short communications.
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