Renal tubular dysgenesis-like lesions and hypocalvaria. Report of two cases involving indomethacin.
We describe a case of twins with twin-to-twin transfusion syndrome (TTS) who were found to have renal tubular dysgenesis (TRD)-like lesions and hypocalvaria attributed to indomethacin treatment of the mother for acute polyhydramnios. History of pregnancy, postnatal clinical course, pathological findings of the kidneys, and the skulls are presented and discussed. These findings include incompletely differentiated proximal tubules in the kidneys and hypoplastic calvaria in both twins. The renal tubular lesions were more marked 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 etiology 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-skull connection has never been reported in conjunction with indomethacin therapy.
Robin
Y M
YM
Service d'Anatomie Pathologique, H tel Dieu BP69, Clermont Ferrand, France.
Reynaud
P
P
Orliaguet
T
T
Lemery
D
D
Vanlieferingen
P
P
Dechelotte
P
P
D000328:Adult; D050701:E2F6 Transcription Factor; D017809:Fatal Outcome; D005260:Female; D005330:Fetofetal Transfusion / Q000209:etiology / Q000473:pathology*; D005865:Gestational Age; D006801:Humans; D007124:Immunoenzyme Techniques; D007213:Indomethacin / Q000009:adverse effects*; D007231:Infant, Newborn; D007687:Kidney Tubules, Proximal / Q000002:abnormalities*; D006831:Polyhydramnios / Q000150:complications / Q000188:drug therapy*; D011247:Pregnancy; D012097:Repressor Proteins / Q000032:analysis; D012886:Skull / Q000002:abnormalities*; D015149:Tocolytic Agents / Q000009:adverse effects*; D014157:Transcription Factors / Q000032:analysis
D050701:E2F6 Transcription Factor; C491744:E2F6 protein, human; D012097:Repressor Proteins; D015149:Tocolytic Agents; D014157:Transcription Factors; D007213:Indomethacin