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Journal of the American Academy of Dermatology
Volume 77, n° 5
pages 874-878 (novembre 2017)
Doi : 10.1016/j.jaad.2017.06.014
accepted : 3 June 2017
Original Articles

Wilms tumor screening in diffuse capillary malformation with overgrowth and macrocephaly–capillary malformation: A retrospective study
 

Caitlin M. Peterman, BS a, b, c, Sophie Vadeboncoeur, MD b, c, d, John B. Mulliken, MD c, e, Steven J. Fishman, MD c, f, Marilyn G. Liang, MD b, c,
a Tufts University School of Medicine, Boston, Massachusetts 
b Department of Dermatology, Boston Children's Hospital, Harvard Medical School, Boston, Massachusetts 
c Vascular Anomalies Center, Boston Children's Hospital, Harvard Medical School, Boston, Massachusetts 
e Department of Plastic and Oral Surgery, Boston Children's Hospital, Harvard Medical School, Boston, Massachusetts 
f Department of Surgery, Boston Children's Hospital, Harvard Medical School, Boston, Massachusetts 
d Department of Medicine, Dermatology Division, Hôpital Maisonneuve-Rosemont, Université de Montréal, Montreal, Quebec, Canada 

Correspondence to: Marilyn G. Liang, MD, Department of Dermatology, Boston Children's Hospital, 300 Longwood Ave, Boston, MA 02115.Department of DermatologyBoston Children's Hospital300 Longwood AveBostonMA02115
Abstract
Background

CLOVES (congenital lipomatous overgrowth, vascular malformations, epidermal nevi, and skeletal anomalies) syndrome is associated with regional bony and/or soft tissue overgrowth, capillary malformation, and an increased risk for Wilms tumor.

Objective

To evaluate the frequency of Wilms tumor in patients with 2 similar conditions: diffuse capillary malformation with overgrowth (DCMO) and macrocephaly–capillary malformation (M-CM).

Methods

Culling our Vascular Anomalies Center database, we retrospectively reviewed patients in whom DCMO and M-CM had been diagnosed and who were evaluated between 1998 and 2016 for possible development of Wilms tumor. Patients younger than 8 years of age at their last visit and not seen in more than 2 years were contacted for follow-up.

Results

The study comprised 89 patients: 67 with DCMO, 17 with M-CM, and 5 with an indeterminate diagnosis. No case of Wilms tumor was found in these groups.

Limitations

Some patients were younger than 8 years of age at last follow-up visit and the sample size was small.

Conclusion

Patients with DCMO do not appear to be at increased risk for Wilms tumor. Screening is probably unnecessary in DCMO unless there is associated hemihypertrophy. Although there were no cases in our cohort, there are 2 reports of M-CM associated with Wilms tumor in the literature.

The full text of this article is available in PDF format.

Key words : diffuse capillary malformation, macrocephaly–capillary malformation, overgrowth, Wilms tumor

Abbreviations used : BWS, CLOVES, CM, CVM, DCMO, KTS, M-CM, PROS, WT



 Ms Peterman and Dr Vadeboncoeur contributed equally to this work.
 Funding sources: None.
 Conflicts of interest: None declared.
 Preliminary data from this study were presented at the 42nd Annual Meeting of the Society of Pediatric Dermatology, Minneapolis, MN; July 16, 2016.
 Reprints not available from the authors.



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