Table 1 Clinical data of the study patients

Dilogo et al. [32]

8y/M

Severe bowing deformity

Left lower leg

X-ray: bowing, bubbled appearance, intracortical osteolytic lesions, no periosteal reaction;

MRI: sclerosis of the internal cortical surface

C-shaped bony spicules with immature bone trabeculae lined with active osteoblasts

Wide excision and MSCs transplantation

84 weeks follow-up without recurrence

Nagano et al. [8]

17y/M

Pain

Right front lower leg

X-ray and CT: osteolytic lesions;

Bone scintigraphy: focally increased radiotracer uptake in the bilateral tibia

IHC: expression of glucose transporter 1 (GLUT-1) and hexokinase II

-

-

Teo et al. [10]

Neonate/M

Swelling and deformity

Left lower leg

X-ray and MRI: extensive destructive lesion of the tibial shaft, with dysplastic congenital pseudoarthrosis of the lower fibula.

IHC: cytokeratin positivity

Osteotomy, physeal distraction, and Ilizarov technique

46 months

Jobke et al. [33]

Newborn/-

Swelling and pain

Left lower extremity

X-ray: central intraosseous translucent lesion within the proximal dia-metaphyseal region with circular cortical thinning and expansion with the neo-cortical formation

IHC: cytokeratin positivity

Conservative treatment

9 months

Karol et al. [34]

11.8y/F

Fracture

Left tibial and fibular

X-ray: Well-demarcated anterior cortical lytic lesion with sclerotic border in proximal 1/3 of the tibia

OFD

Closed reduction and application of a cast

Died

Segev et al. [35]

6y/M

Solid tumor and Deformity

Left tibia

X-ray: cystic lesion with a sclerotic reactive rim

OFD

6-10years: close follow-up;

10years: remove the lump;

11years: curetted and treated with cryosurgery, space was filled with PMMA

8 years

Kosuge et al. [36]

11y/M

Deformity

Left leg

X-ray: anterior apex bow to the tibia within which a multilocular lesion with mixed radiolucent and sclerotic foci was seen

OFD

Marginal excision of the lesion

-

Simoni et al. [37]

27y/M

Pain

Right leg

X-ray: a large focal area of cortical thickening. Multiple roundish, radiolucent lacunae, soap bubble appearance, no periosteal reaction;

CT: absence of a transitional zone and periosteal reaction. MRI: low signal intensity on both T1WI and T2WI

IHC: cytokeratin positivity

-

-

Yoshida et al. [15]

34y/M

Pain

Left upper arm

X-ray: bone tumor at the humeral shaft

OFD

Curettage, intraoperative anhydrous ethanol therapy, and artificial bone graft

7 years

Abraham et al. [38]

13y/F

Pain and swelling

Right leg

X-ray: eccentric expansile lytic lesion

OFD

Extraperiosteal excision, Autologous free fibular graft, and bone graft substitute

2 years

Gopinathan et al. [39]

14y/F

Pain and swelling

Left collar bone

X-ray: a diffuse periosteal thickening encircling the clavicle extending from its sterna end to lateral third;

CT: irregularity and sclerosis of the left clavicle along with heterogeneous ossification. MRI: altered heterogeneous T2 signal within the marrow of the clavicle

IHC: cytokeratin (AE1/AE3 + CK-1) positive

Excision of the lesion

-

Exner et al. [40]

38y/M

-

Left tibia

X-ray: multifocal, partially confluent osteolytic lesions

IHC: vimentin and pan-cytokeratin positivity

Observation

7 years

Goto et al. [13]

15y/M

Pain

Right elbow

X-ray: osteolytic lesions in the medial part of the proximal ulna, with thinning of the cortex and a sclerotic change around the osteolytic lesions

The lesion showed typical zonal architecture; the center of the lesion was predominantly fibrous; In addition, scattered woven bone was rimmed by plump osteoblasts

Five months after the surgery, the tumor recurred. Further surgery was not performed. At age 28 years, the patient had no pain, discomfort, or functional disturbance

13 years

Goto et al. [13]

6y/F

Contusion on the left elbow

Left ulna

X-ray: osteolytic lesions on the dorsal side of the left ulna. The cortex was thin, with medullary sclerosis around the lesion

-

Observation

16 years