Shi Jing, Zhang Zekun, Li Yuqing, Latif Mahrukh, Gao Feng
Department of Radiology, The Second Hospital of Shijiazhuang City, 53 Huaxi Road, Shijiazhuang City, Hebei Province, 050051, China.
Skeletal Radiol. 2015 Jul;44(7):1033-8. doi: 10.1007/s00256-015-2121-9. Epub 2015 Feb 26.
Gorham's disease (GD) rarely occurs in the hand and wrist. Only nine cases of GD in the hand and wrist have been reported in the literature. The imaging technique used in all nine cases was mainly radiography. The natural history of GD is unpredictable. Spontaneous regression has been reported in a few cases. There is no consensus about the most efficient treatment of GD. Surgical resection and reconstruction with bone grafts and/or prostheses are used sparingly as bone grafts tend to be resorbed in most cases. We report a case of GD that involved the right hand and wrist in a 26-year-old male. The lesion displayed multiple areas of osteolysis in the metacarpals, carpals and proximal phalanx of the base of the thumb on radiography, while on CT and MRI, the osteolytic areas showed homogeneous density and signal intensity, similar to that of fluid. The patient was successfully treated by surgical reconstruction with autogenous iliac bone grafts. Gradual reossification in the osteolytic areas had occurred by the 3-year follow-up evaluation. This case merits special attention because of the unusual location of the involvement, unusual CT and MR imaging findings and unusual reossification in the osteolytic areas.
Gorham病(GD)很少发生于手部和腕部。文献中仅报道了9例手部和腕部的GD病例。这9例病例所采用的影像学检查主要是X线摄影。GD的自然病程难以预测。少数病例报道有自发消退的情况。对于GD最有效的治疗方法尚无共识。由于在大多数情况下骨移植往往会被吸收,因此很少使用手术切除并用骨移植和/或假体进行重建的方法。我们报告了1例26岁男性右手和腕部受累的GD病例。X线摄影显示病变在掌骨、腕骨和拇指基部近端指骨有多个骨质溶解区域,而在CT和MRI上,骨质溶解区域显示出均匀的密度和信号强度,类似于液体。该患者通过自体髂骨移植手术重建成功治愈。在3年的随访评估中,骨质溶解区域出现了逐渐的再骨化。由于受累部位不寻常、CT和MRI表现不寻常以及骨质溶解区域出现不寻常的再骨化,该病例值得特别关注。