Author Affiliations: Departments of Dermatology (Drs Goujon, Beer, and Vabres) and Pediatrics and Réseau Périnatal de Bourgogne (Drs Gay, Sandre, and Gouyon), University Hospital of Dijon, University of Burgundy Medical School, Dijon, France.
Anetoderma of prematurity was described by Prizant et al1 in very-low-birth-weight infants in neonatal intensive care units (NICUs). This recent description probably reflects improvement in care of premature neonates who were previously unable to survive. The mechanism for the development of anetoderma is unknown, but the role of monitoring leads has been suspected. To further delineate this clinical condition, we have studied 11 additional cases.
All cases of anetoderma of prematurity seen in a single NICU (University Hospital, Dijon, France) from 1999 to 2006 were retrospectively studied. Anetoderma was diagnosed clinically.
Gestational age and birth weight ranged from 25 to 30 weeks and from 725 to 1250 g, respectively. All neonates had pulmonary diseases and required assisted ventilation. Ten developed bronchopulmonary dysplasia and received oral steroid treatment. Three had severe digestive tract complications (necrotizing enterocolitis or ileal perforation). Nine were treated with indomethacin for patent ductus arteriosus, and 4 of these needed surgical closure. The median duration of hospitalization in the NICU was 125.0 days vs 99.5 days in 30 control neonates matched for gestational age (P = .001).
Twin pregnancies occurred in 5 cases, but no co-twins were affected with anetoderma. In these twin pairs, the affected twin had the lower birth weight in 2 instances, and the higher in 3 instances. The incidence and severity of pulmonary or digestive tract diseases were similar in twins without anetoderma.
Localized, rounded flat, atrophic skin patches 5 to 20 mm in diameter were first noted between age 6 weeks and 5 months (Figure 1B and Figure 2A). Five infants had previously been examined for ecchymoses without atrophy or necrosis (Figure 1A) at the sites where monitoring leads had been applied. On follow-up, all ecchymoses turned into atrophic patches within a few days. Previous placement of monitoring leads at the site of atrophic patches was noted in 8 cases. All lesions were ventrally located: in the subclavicular areas on the chest in 8 cases (Figure 1) and in paraumbilical areas on the abdomen in 6 (Figure 2).
The periumbilical region of patient 3. A, Initial ecchymosis (age 4 months). B, Late atrophic patches (age 16 months).
The left subclavicular region of patient 5. A, Ecchymotic macules (age 6 weeks). B, Atrophic patches (age 9 months). C, Anetoderma with herniation (age 24 months).
Follow-up data were available for 8 infants. All atrophic patches evolved into typical herniated anetoderma (Figure 2B and C). No self-healing or improvement occurred. In 1 child aged 7 years, persistent anetoderma lesions on the upper chest caused disfigurement and led to surgical excision.
Eight cases were observed in a 2-year period between 1999 and 2001. At that time, because the pressure of the monitoring leads was suspected to be a causative factor, a preventive approach was implemented in the NICU. Monitoring leads were always applied on the ventral side when the infants were lying supine and on the dorsal side when lying prone, thus avoiding pressure from the leads onto the skin caused by the infants' weight. From that day on, only 3 additional cases were seen in the following 5-year period between 2001 and 2006.
Anetoderma of prematurity has rarely been reported.1- 4 Although ascertained at a single institution, the number of cases in our series, to our knowledge, is the highest ever reported. This suggests that the condition has previously been underreported. Birth weight does not appear to play a significant role,2 as suggested by our 5 discordant twin pairs. Most cases developed at the site of monitoring leads, where ecchymoses had sometimes been noted. From this we conclude that monitoring leads likely play a causative role, although the precise mechanism remains unknown.1 The hypothesis of local hypoxemia due to pressure on immature skin is strengthened by the decreased incidence of anetoderma following postural prevention. However, another explanation might be excessive traction on the skin when adhesive electrodes are removed, insufficient for skin tearing but causing either subclinical dermal damage or visible ecchymosis leading to anetoderma. Indeed, involvement of the periumbilical area suggests the role of adhesive tapes used for attachment of umbilical arterial lines. Hence, anetoderma of prematurity likely results from unnoticed minor iatrogenic trauma in the NICU, where cutaneous injuries are among the most common iatrogenic events. The prolonged duration of hospitalization supports this hypothesis. Anetoderma of prematurity is thus acquired and should be differentiated from congenital anetoderma, which is of unknown origin and has also been described in premature neonates.5
Because anetoderma patches tend to persist, they might cause long-lasting disfigurement on visible sites such as the upper chest. Thus, they should be prevented by avoiding placement of leads in this area. When anetoderma patches are first encountered later in childhood, their diagnosis may be difficult, and a neonatal history of extreme prematurity and a NICU stay can be a helpful clue.
Correspondence: Dr Goujon, Dermatologie, CH William Morey, BP 120, 71321 Chalon-sur-Saône, France (firstname.lastname@example.org).
Accepted for Publication: September 30, 2009.
Author Contributions: Dr Goujon had full access to all the data in the study and takes responsibility for the integrity of the data and the accuracy of the data analysis. Study concept and design: Goujon, Gay, and Vabres. Acquisition of data: Goujon, Beer, Gay, and Sandre. Analysis and interpretation of data: Goujon, Beer, Gouyon, and Vabres. Drafting of the manuscript: Goujon. Critical revision of the manuscript for important intellectual content: Beer, Gay, Sandre, Gouyon, and Vabres. Statistical analysis: Goujon and Vabres. Administrative, technical, and material support: Beer and Gay. Study supervision: Beer, Gouyon, and Vabres.
Financial Disclosure: None reported.
Additional Contributions: Philip Bastable, PhD, reviewed the manuscript.
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