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Horn Kolb syndrome (first report) following in utero exposure: case report A female fetus was prenatally diagnosed with Horn Kolb syndrome (acheiropodia) following in utero exposure to antihypertensives and nitroimidazoles; fetal death occurred. The 36-year-old mother, who was married to her first cousin, presented in her 23rd week of gestation with severe oligohydramnios and an inability to visualise the fetal stomach, kidneys and urinary bladder on ultrasound. She had received spironolactone 50mg and hydrochlorothiazide 50mg in a combination daily [route not stated] from 16 December to 15 January for mild hirsutism and hypertension, as well as oral ornidazole 500mg twice daily and vaginal metronidazole 500mg once a day from 16–23 December for a genital infection. Ultrasound examination revealed an amniotic fluid index of 0cm, an enlarged cisterna magna and periods of fetal arrhythmia. Upon examination of the extremities, the fetal radius, ulna, tibia, fibula, hands or feet could not be visualised [time to onset not stated]. Fetal death occurred within 1 hour of admission. The woman received misoprostol for pregnancy termination. Following delivery of the fetus, an autopsy revealed the distal portions of the fetal extremities had not developed, while other body structures were normal. X-ray analysis confirmed the diagnosis, demonstrating single hypoplastic ossification centres in the forearms and legs. Author comment: "Acheiropodia is an autosomal recessive inherited trait. The heterozygotes are phenotypically normal. Most affected individuals are the offspring of consanguineous matings. . . The possible involvement of teratogenic effects of the specifics of our case (cellular phone base station, spironolactone, hydrochlorothiazide, ornidazole and metronidazole) needs further investigation." Temur I, et al. The first case of Horn Kolb Syndrome in Turkey, diagnosed prenatally at the 23rd week of pregnancy: A very rare and unusual case far from the original geography. American Journal of Case Reports 13: 106-108, 13 Jun 2012. Available from: URL: http://www.amjcaserep.com/fulltxt.php?ICID=883025 803076212 - Turkey

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Editorial comment: A search of AdisBase, Medline and Embase did not reveal any previous case reports of Horn Kolb syndrome associated with spironolactone, hydrochlorothiazide, metronidazole or ornidazole. The WHO ADR database contained no reports of limb malformations associated with spironolactone, hydrochlorothiazide or ornidazole, and one report associated with metronidazole.

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Reactions 1 Sep 2012 No. 1417