Ym. Robin et al., Renal tubular dysgenesis-like lesions and hypocalvaria - Report of two cases involving indomethacin, PATH RES PR, 196(11), 2000, pp. 791-794
We describe a case of twins with twin-to-twin transfusion syndrome (TTS) wh
o were found to have renal tubular dysgenesis (TRD)-like lesions and hypoca
lvaria attributed to indomethacin treatment of the mother for acute polyhyd
ramnios. History of pregnancy, postnatal clinical course, pathological find
ings of the kidneys, and the skulls are presented and discussed. These find
ings include incompletely differentiated proximal tubules in the kidneys an
d hypoplastic calvaria in both twins. The renal tubular lesions were more m
arked in the donor than in the transfused twin, probably due to the greater
degree of ischemia in that twin. This seems to be in favor of a vascular e
tiology of the renal defects. However, the fact that similar renal lesions
and hypocalvaria were also present in the transfused twin seems to indicate
that indomethacin played a role in their onset. This so-called kidney-skul
l connection has never been reported in conjunction with indomethacin thera
py.