A 44-year-old woman with fetal thalidomide syndrome and congenital pseudoainhum of the left big toe had a 5-year history of painful nonhealing ulcers in the left malleolar region. Venous Doppler ultrasonography showed hypoagenesis of the valve flaps of the deep and superficial venous circuit. To our knowledge, this is the first description of congenital pseudoainhum associated with fetal thalidomide syndrome. The coexistence of cutaneous ulcers in our case might be coincidental but may be related to a congenital valve defect (hypoagenesis) caused by thalidomide.
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References
1.
Teo SK, Denny KH, Stirling SD, et al: Effects of thalidomide on developmental, peri- and postnatal function in female New Zealand white rabbits and offspring. Toxicol Sci81(2):379-389, 2004.
2.
Matthews SJ, McCoy C.: Thalidomide a review of approved and investigational use. Clin Ther25(2): 342-395, 2003.
3.
D'Amato RJ, Loughnan MS, Flynn E., et al: Thalidomide is an inhibitor of angiogenesis. Proc Natl Acad Sci U S A91(9):4082-4085, 1994.
4.
Stephens TD: Proposed mechanisms of action in thalidomide embryopathy. Teratology38(3):229-239, 1988.
5.
Stephens TD, Bunde CJ, Fillimore BJ: Mechanism of action in thalidomide teratogenesis. Biochem Pharmacol59(12):1489-1499, 2000 .
6.
Kruse FE, Joussen AM, Rohrschneider K., et al: Thalidomide inhibits corneal angiogenesis induced by vascular endothelial growth factors. Graefes Arch Clin Exp Ophthalmol236(6):461-466, 1998.
7.
Yabu T., Tomimoto H., Taguchi Y., et al: Thalidomide-induced antiangiogenic action is mediated by ceramide through depletion of VEGF receptors, and is antagonized by sphingosine-1-phosphate . Blood106(1):125-134, 2005.
8.
Dornier C., Cuny JF, Di Cesare MP, et al: Pseudoainhum in lamellar ichthyosis [in French]. Ann Dermatol Venereol128(10,pt1):1037-1039, 2001.
9.
Petter C.: Early foetal thromboses induced by thalidomide in mouse: possible explanation for teratogenicity. Experientia33(10):1384-1386, 1977.
10.
Graham RM, James MP: Pseudo-ainhum, angiodysplasia and focal acral hyperkeratosis. J R Soc Med78(suppl 11):13-15, 1985.
11.
Pianigiani E., Ierardi F., Taddeucci P., et al: Skin allograft in the treatment of toxic epidermal necrolysis (TEN). Dermatol Surg28(12):1173-1176, 2002.