Lymphangiography in Lymphangiomatosis of Bone

Abstract

radiographs are essential to demonstrate the intraosseous localization of the contrast material.
Lymphangiomatosis of bone seems to be due to lymphatic dysplasia with valvular insufficiency and
has therefore to be considered as a cong1mital malformation.
Lymphangiomatosis of bone has first been described as an entity by Bickel and Broders
in 1947. Haas and Reichelt (2) reported a family consisting of several blood relatives
with evidence of lymphangiomatosis of bone. However, lymphographic investigations
have been published in only four cases. Kittredge et al. (3) presented the case of a fifteen-
year-old boy with a lymphadenopathy in the left groin, but no lymphedema. The
plain roentgenograms showed multiple osteolytic lesions in the pelvis, hip and shoulder.
Histologically the defects were caused by dilated lymphchannels. The lymphograms
showed a disorganization of the lymphatic vessels in the thigh and pelvis, but the contrast
material did not enter the bone lesions.
Najman et al. ( 4) described a boy with a lymphangioma in the inguinal region accompanied
by lymphedema. Lytic bone lesions were found in the pelvis, limbs, chest and
the skull. Lower extremity lymphography showed dilated lyphatics, but no contrast
medium was located in the bone. Contrast material was injected into a bone cyst of
the skull from where it passed into adjoining cystic lesions.
Steiner et al. (6) performed a lower extremity lymphography on a boy with pleural
effusion and lytic bone defects in the humerus and the bony structures of the knee
joints, but no lymphedema. Abnormal lymphatic vessels in the mediastinum and thoracic
wall were demonstrated by lymphography. Although no contrast filling of the
lytic bone lesions was obtained, the histological examination confirmed the presence
of abnormal lymphatic channels in the bone.
Recently Nixon (5) demonstrated bone lymphangiomatosis lymphographically in a boy
with lymphedema and osteolytic lesions in the femur and tibia. Contrast material was
found within a cortical lesion of the femur 48 h following lymphography.