Aplasia Cutis Congenita in Surviving Co‐twins: Four Unrelated Cases

Cambiaghi, Stefano; Schiera, A.; Tasin, L; Gelmetti, Carlo

doi:10.1046/j.1525-1470.2001.1861998.x

Cited by 36 publications

(25 citation statements)

References 14 publications

(31 reference statements)

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“…The 2 structural defects noted in the case series, although not directly comparable with the cohort study results, were not consistent with any defects reported in the cohort group, and were attributable to alternative factors. Aplasia cutis is known to occur with increased frequency in the surviving member of a monozygotic twin pair (17), and chondrodysplasia punctata has been reported, from a case series, to be a fetal complication related to maternal lupus (18). Finally, there was no evidence of a specific pattern of 3 or more minor anomalies among those children with prenatal leflunomide exposure who underwent the dysmorphologic examination.…”

Section: Discussionmentioning

confidence: 99%

Birth outcomes in women who have taken leflunomide during pregnancy

Chambers

Johnson

Robinson

et al. 2010

Arthritis & Rheumatism

177

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Objective In preclinical reproductive studies, leflunomide was found to be embryotoxic and teratogenic. Women treated with leflunomide are advised to avoid pregnancy; those who become pregnant are advised to reduce fetal exposure through a cholestyramine drug elimination procedure. The present study was undertaken to investigate pregnancy outcomes in women who received leflunomide and were treated with cholestyramine during pregnancy. Methods Sixty-four pregnant women with rheumatoid arthritis (RA) who were treated with leflunomide during pregnancy (95.3% of whom received cholestyramine), 108 pregnant women with RA not treated with leflunomide, and 78 healthy pregnant women were enrolled in a prospective cohort study between 1999 and 2009. Information was collected via interview of the mothers, review of medical records, and specialized physical examination of infants. Results There were no significant differences in the overall rate of major structural defects in the exposed group (3 of 56 live births [5.4%]) relative to either comparison group (each 4.2%)(P = 0.13). The rate was similar to the 3–4% expected in the general population. There was no specific pattern of major or minor anomalies. Infants in both the leflunomide-exposed and non–leflunomide-exposed RA groups were born smaller and earlier relative to infants of healthy mothers; however, after adjustment for confounding factors, there were no significant differences between the leflunomide-exposed and non–leflunomide-exposed RA groups. Conclusion Although the sample size is small, these data do not support the notion that there is a substantial increased risk of adverse pregnancy outcomes due to leflunomide exposure among women who undergo cholestyramine elimination procedure early in pregnancy. These findings can provide some reassurance to women who inadvertently become pregnant while taking leflunomide and undergo the washout procedure.

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Section: Discussionmentioning

confidence: 99%

Birth outcomes in women who have taken leflunomide during pregnancy

Chambers

Johnson

Robinson

et al. 2010

Arthritis & Rheumatism

177

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“…Intra-uterine death of a twin may cause release of thrombotic material, promoting ACC in the surviving twin in the case of vascular shunts. This is usually associated with extensive placental infarctions, disseminated intravascular coagulation and other direct and usually severe injuries to the surviving twin, which we failed to fi nd in our index case (22,23) .…”

Section: Discussionmentioning

confidence: 61%

Aplasia cutis congenita in surviving co-twin after propylthiouracil exposure in utero

Löllgen

Calza

Schwitzgebel

et al. 2011

Journal of Pediatric Endocrinology and Metabolism

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Aim: Aplasia cutis congenita (ACC) has been observed after fetal exposure to the antithyroid drug methimazole (MMI), but not reported after propylthiouracil (PTU), the current antithyroid drug of choice during pregnancy. This occurrence has implications for patient information and causal research. Case report: We describe a surviving term co-twin to a mother with hyperthyroidism exposed to PTU from conception to 34 weeks of gestation presenting with ACC at birth. Discussion: The association between PTU exposure and ACC is clinically relevant and allows speculation on the etiology. A similar mechanism to the classical MMI-induced ACC is postulated, unless a vascular etiology suggested by a vanishing twin or maternal hyperthyroidism itself is causal. Coincidence of PTU exposure and ACC seems unlikely. Conclusion: ACC in a newborn after PTU exposure during pregnancy hitherto observed only after MMI strongly encourages further reports of similar cases that may remain clinically underdiagnosed or unreported. Such confi rmation could have signifi cant implications for maternal treatment of hyperthyroidism, common in women of childbearing age.

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“…The end result involves areas of fullthickness skin aplasia that may be the product of impaired vascular supply early in the second trimester. [12][13][14] The scars in the groin and axillae in our patient represent areas of aplasia that were able to heal over the ensuing months of gestation between the insult and birth.…”

Section: Commentmentioning

confidence: 73%