Lavell Anton, Jones Christopher W, Wong Daniel, Counsel Peter, Carey-Smith Richard
Department of Radiology, Fiona Stanley Hospital, Murdoch, WA, 6150, Australia.
Perth Orthopaedic Institute, Nedlands, WA, Australia.
Skeletal Radiol. 2017 Oct;46(10):1405-1413. doi: 10.1007/s00256-017-2689-3. Epub 2017 Jun 16.
Ossifying subperiosteal haematoma is an exceedingly rare manifestation of Neurofibromatosis type 1 (NF-1). We report an interesting case of plexiform neurofibroma causing a rapidly growing tibial mass as a result of subperiosteal haemorrhage, in an 11-year-old girl with previously undiagnosed NF-1. This reflects a precursor of the more mature periosteal ossification seen in cases traditionally termed "subperiosteal cysts". A previously well young girl was referred by her general practitioner with an increasingly large, mildly tender, soft lump on the anterior aspect of her right tibia. Plain radiographs demonstrated soft tissue thickening overlying the anterior tibia, without appreciable periosteal ossification. Magnetic resonance imaging (MRI) illustrated a single central fluid-fluid level and periosteal elevation with saucerisation of the anterior tibial cortex and mild surrounding oedema. Histopathology revealed a large plexiform neurofibroma. Interestingly, this was associated with haemorrhagic change and a peripheral rim of florid reactive new bone formation. This unusual presentation was discussed at a multidisciplinary bone and soft tissue tumour meeting, where in combination with the clinical history of café au lait spots and positive family history, a consensus diagnosis of NF-1 was made. To date, there have only been limited case reports of this rare pathological process. In summary, this case report accounts an acute presentation of this rare osseous manifestation of NF-1, being the first to clearly demonstrate a timeline of subperiosteal haematoma with subsequent subperiosteal bone proliferation. The clinical reasoning and radiological features for such a presentation are also described.
骨化性骨膜下血肿是1型神经纤维瘤病(NF-1)极为罕见的一种表现形式。我们报告了一例有趣的病例,一名11岁此前未被诊断出患有NF-1的女孩,其丛状神经纤维瘤因骨膜下出血导致胫骨肿物迅速生长。这反映了在传统上称为“骨膜下囊肿”的病例中所见更成熟骨膜骨化的一种先兆。一名此前健康的年轻女孩被全科医生转诊,她右胫骨前方有一个越来越大、轻度压痛的软肿块。X线平片显示胫骨前方软组织增厚,未见明显骨膜骨化。磁共振成像(MRI)显示单个中央液-液平面以及骨膜抬高,胫骨前皮质呈碟形,周围有轻度水肿。组织病理学检查显示为一个大型丛状神经纤维瘤。有趣的是,其伴有出血性改变以及周边有活跃的反应性新骨形成。在一次多学科骨与软组织肿瘤会议上讨论了这种不寻常的表现,结合咖啡斑的临床病史和阳性家族史,作出了NF-1的共识诊断。迄今为止,关于这种罕见病理过程的病例报告仅有少数。总之,本病例报告描述了NF-1这种罕见骨表现的急性呈现,首次清晰展示了骨膜下血肿及随后骨膜下骨增生的时间线。还描述了这种表现的临床推理和放射学特征。