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Research Letters |

Risk of Hepatic Hemangiomas in Infants With Large Hemangiomas FREE

Kimberly A. Horii, MD; Beth A. Drolet, MD; Eulalia Baselga, MD; Ilona J. Frieden, MD; Denise W. Metry, MD; Kimberly D. Morel, MD; Brandon D. Newell, MD; Amy J. Nopper, MD; Maria C. Garzon, MD; for the Hemangioma Investigator Group
[+] Author Affiliations

Author Affiliations: Section of Dermatology, Children's Mercy Hospitals and Clinics, Kansas City Missouri (Drs Horii, Newell, and Nopper); Departments of Dermatology and Pediatrics, Medical College of Wisconsin, Milwaukee (Dr Drolet); and Departments of Dermatology, Hospital de la Santa Creu I Sant Pau, Barcelona, Spain (Dr Baselga), University of California, San Francisco (Dr Frieden), Baylor College of Medicine, Houston, Texas (Dr Metry), and Columbia University, New York, New York (Drs Morel and Garzon).


Arch Dermatol. 2010;146(2):201-203. doi:10.1001/archdermatol.2009.391.
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Published online

While most infantile hemangiomas are benign and uncomplicated, a minority may have associated internal involvement that can lead to significant morbidity.1,2 Multiple cutaneous hemangiomas have been recognized as potential markers of underlying hepatic hemangiomas.14 Two recent retrospective studies suggest that infants with large (≥5 cm in diameter) and/or segmental cutaneous hemangiomas are also at risk for hepatic hemangiomas.2,3 These reports have led many clinicians to screen infants with large cutaneous hemangiomas for hepatic hemangiomas. An algorithm for the evaluation and management of hepatic hemangiomas in asymptomatic infants with multiple (≥5) cutaneous hemangiomas, based on retrospective data, has recently been published.4 However, controversy remains regarding the number of cutaneous hemangiomas that should serve as the threshold at which to perform such screening.5 Definitive guidelines regarding the workup for hepatic hemangiomas in asymptomatic infants with large cutaneous hemangiomas, particularly within the first few months of life, are lacking in the literature. The true prevalence of this association is also unknown because, to our knowledge, no prospective studies of infants with large cutaneous hemangiomas have been undertaken. To assess this risk, we report results from a multicenter, prospective study in which infants with large cutaneous hemangiomas were systematically evaluated for hepatic hemangiomas.

Infants aged between 1 and 6 months referred to a pediatric dermatologist with fewer than 5 and at least 1 large hemangioma (>30 cm2) were consecutively enrolled between 2006 and 2008 at 4 Hemangioma Investigator Group sites. This study was a nested study within a larger study looking at infantile hemangiomas with a risk of morbidity related to either size of the hemangioma or number (≥5). Institutional review board approval was obtained at each site. A standardized questionnaire was completed on each infant. The hemangiomas were classified based on morphologic characteristics and size, with size determined by measuring 2 perpendicular surface diameters with a flexible measuring tape. Physical examination and abdominal ultrasonography were performed on each infant.

Demographic and clinical characteristics are summarized in the Table. A total of 60 infants with at least 1 large hemangioma (>30 cm2) were enrolled. The mean (SD) hemangioma size was 73.6 (38.4) cm2. At the time of enrollment, all infants were clinically asymptomatic without signs of hepatomegaly, abdominal distention, or congestive heart failure. No hepatic hemangiomas were identified on ultrasonography.

Table Graphic Jump LocationTable Demographic and Clinical Characteristics of Infants With Large Hemangiomas

To our knowledge, this is the first attempt to prospectively assess the risk of hepatic hemangiomas in infants with large cutaneous hemangiomas, an association found in none of our patients. It is important to note that study recruitment occurred in pediatric dermatology clinics, which may explain why none of the patients presented with clinically symptomatic hepatic hemangiomas. The predominance of segmental hemangiomas in our report is not surprising because segmental hemangiomas typically have larger surface areas of involvement.6 The preponderance of term infants is explainable because unlike localized and multiple hemangiomas, which are more common in preterm infants, segmental hemangioma incidence is not affected by gestational age.7

Our findings suggest that infants with large cutaneous hemangiomas (>30 cm2) may not be at an increased risk for concomitant hepatic hemangiomas as initially reported in the literature and as seen in infants with multiple cutaneous hemangiomas. Hughes et al3 retrospectively reported the cases of 25 infants with solitary, large (≥5 cm) cutaneous hemangiomas, 3 of whom had clinically asymptomatic hepatic hemangiomas identified on abdominal ultrasonography (12%). Metry et al2 also noted the association of hepatic hemangiomas and solitary segmental cutaneous hemangiomas in a retrospective report of 4 cases of patients with segmental cutaneous hemangiomas and a literature review of 47 others; however, no prevalence could be estimated since all infants had visceral hemangiomas. The authors recommended that infants with solitary, segmental cutaneous hemangiomas be screened for visceral involvement when clinically indicated.

Based on the results of the present study, routine screening ultrasonography for hepatic involvement in asymptomatic infants with large cutaneous hemangiomas may not be necessary unless clinically indicated. However, because some infants may present with significant hepatic hemangiomas with few or no cutaneous hemangiomas, and until the true risk of this possible association is known, infants with large cutaneous hemangiomas should continue to be clinically monitored, especially during the first 6 months of life when the risk of complications associated with hepatic hemangiomas is considered to be the greatest. Signs or symptoms suggestive of hepatic hemangiomas that would indicate the need for further workup include a history of poor growth or feeding, tachypnea, cardiac murmur, abdominal distention, or hepatomegaly.

Correspondence: Dr Horii, Section of Dermatology, Children's Mercy Hospitals and Clinics, 2401 Gillham Rd, Kansas City, MO 64108 (kahorii@cmh.edu).

Accepted for Publication: August 20, 2009.

Author Contributions: Drs Garzon, Horii, and Drolet had full access to all data in the study and take responsibility for the integrity of the data and the accuracy of the data analysis. Study concept and design: Drolet, Frieden, and Garzon. Acquisition of data: Horii, Drolet, Baselga, Metry, Morel, Newell, Nopper, and Garzon. Analysis and interpretation of data: Horii, Drolet, Frieden, Metry, and Garzon. Drafting of the manuscript: Horii, Drolet, and Garzon. Critical revision of the manuscript for important intellectual content: Horii, Drolet, Baselga, Frieden, Metry, Morel, Newell, Nopper, and Garzon. Horii and Drolet. Administrative, technical, and material support: Horii and Drolet. Study supervision: Frieden.

Hemangioma Investigator Group Members: Kimberly A. Horii, MD; Beth A. Drolet, MD; Eulalia Baselga, MD; Ilona J. Frieden, MD; Denise W. Metry, MD; Kimberly D. Morel, MD; Brandon D. Newell, MD; Amy J. Nopper, MD; Maria C. Garzon, MD; Sarah L. Chamlin, MD; Anita N. Haggstrom; Anne W. Lucky, MD; Anthony J. Mancini, MD; Dawn Siegel, MD; Kristen Holland, MD; Julie Powell, MD; Catherine McCuaig, MD; Denise Adams, MD.

Financial Disclosure: Drs Frieden and Drolet serve as consultants for Pierre Fabre Dermatology.

Funding/Support: This study was supported in part by a Katherine B. Richardson Grant, the American Skin Association, the Dermatology Foundation, and the Greater Milwaukee Foundation.

Role of the Sponsors: The sponsors had no role in the design and conduct of the study; in the collection, analysis, and interpretation of data; or in the preparation, review, or approval of the manuscript.

Trial Registration: clinicaltrials.gov Identifier: NCT00374335.

Additional Contributions: Ashley Sherman, MA, provided statistical expertise; Sia Vue, BA, helped with data collection; and our colleagues and staff at participating institutions also provided valuable assistance.

Christison-Lagay  ERBurrows  PEAlomari  A  et al.  Hepatic hemangiomas: subtype classification and development of a clinical practice algorithm and registry. J Pediatr Surg 2007;42 (1) 62- 67
PubMed Link to Article
Metry  DWHawrot  AAltman  CFrieden  IJ Association of solitary, segmental hemangiomas of the skin with visceral hemangiomatosis. Arch Dermatol 2004;140 (5) 591- 596
PubMed Link to Article
Hughes  JAHill  VPatel  KSyed  SHarper  JDe Bruyn  R Cutaneous haemangioma: prevalence and sonographic characteristics of associated hepatic hemangioma. Clin Radiol 2004;59 (3) 273- 280
PubMed Link to Article
Dickie  BDasgupta  RNair  R  et al.  Spectrum of hepatic hemangiomas: management and outcome. J Pediatr Surg 2009;44 (1) 125- 133
PubMed Link to Article
Lorette  GGeorgesco  GSirinelli  DAlison  DDelhommais  A Cutaneous immature hemangioma and hepatic angioma: there is no frequent association [in French]. Ann Dermatol Venereol 1996;123789- 790
PubMed
Chiller  KGPassaro  DFrieden  IJ Hemangioma of infancy: clinical characteristics, morphologic subtypes, and their relationship to race, ethnicity, and sex. Arch Dermatol 2002;138 (12) 1567- 1576
PubMed Link to Article
Garzon  MCDrolet  BABaselga  E  et al. Hemangioma Investigator Group, Comparison of infantile hemangiomas in preterm and term infants: a prospective study. Arch Dermatol 2008;144 (9) 1231- 1232
PubMed Link to Article

Figures

Tables

Table Graphic Jump LocationTable Demographic and Clinical Characteristics of Infants With Large Hemangiomas

References

Christison-Lagay  ERBurrows  PEAlomari  A  et al.  Hepatic hemangiomas: subtype classification and development of a clinical practice algorithm and registry. J Pediatr Surg 2007;42 (1) 62- 67
PubMed Link to Article
Metry  DWHawrot  AAltman  CFrieden  IJ Association of solitary, segmental hemangiomas of the skin with visceral hemangiomatosis. Arch Dermatol 2004;140 (5) 591- 596
PubMed Link to Article
Hughes  JAHill  VPatel  KSyed  SHarper  JDe Bruyn  R Cutaneous haemangioma: prevalence and sonographic characteristics of associated hepatic hemangioma. Clin Radiol 2004;59 (3) 273- 280
PubMed Link to Article
Dickie  BDasgupta  RNair  R  et al.  Spectrum of hepatic hemangiomas: management and outcome. J Pediatr Surg 2009;44 (1) 125- 133
PubMed Link to Article
Lorette  GGeorgesco  GSirinelli  DAlison  DDelhommais  A Cutaneous immature hemangioma and hepatic angioma: there is no frequent association [in French]. Ann Dermatol Venereol 1996;123789- 790
PubMed
Chiller  KGPassaro  DFrieden  IJ Hemangioma of infancy: clinical characteristics, morphologic subtypes, and their relationship to race, ethnicity, and sex. Arch Dermatol 2002;138 (12) 1567- 1576
PubMed Link to Article
Garzon  MCDrolet  BABaselga  E  et al. Hemangioma Investigator Group, Comparison of infantile hemangiomas in preterm and term infants: a prospective study. Arch Dermatol 2008;144 (9) 1231- 1232
PubMed Link to Article

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