PracticalDermatology

Infantile Hemangiomas with Minimal or Arrested Growth (IH-MAGs) are a subtype of hemangiomas with minimal to absent proliferative capacity that are uniquely erythrocyte type glucose transporter-1 (GLUT-1) protein positive. Only two cases have been reported in the literature of superimposed eczema associated with IH-MAG, both treated with systemic propranolol and topical agents including tacrolimus and hydrocortisone. This observational case report presents an instance where only topical mupirocin was successfully used to treat a bacterial infection overlying contact dermatitis with improvement of the infection as well as the eczema.

Infantile Hemangiomas

Infantile hemangiomas (IH), present at birth or developing within the first few weeks of life, are the most common benign vascular tumor of infancy.¹ The majority of hemangiomas are characterized by a rapid proliferative phase that usually continues up to five months of age followed by a more gradual and variable involution phase.¹

Infantile hemangiomas with minimal or arrested growth (IH-MAG) represent a unique subtype defined by minimal to absent proliferative capacity. Corella, et al. showed that IH-MAGs stain positive for erythrocyte type glucose transporter-1 (GLUT-1) protein, providing evidence that they are a distinct entity from other vascular tumors such as non-involuting and rapidly involuting congenital hemangiomas (NICH and RICH) or pyogenic granulomas.² In the medical literature, only two case reports have been published of superimposed eczema associated with IH-MAG.^3,4 Here we describe a case of IH-MAG with superimposed superficial skin infection as well as allergic contact dermatitis that we managed with topical mupirocin.

CASE

A five-month-old Hispanic boy with a past medical history of physiologic peripheral pulmonary stenosis presented for evaluation of hemangioma on his left inner thigh. His mother reported that the lesion was present at birth and had grown minimally in proportion to the child’s growth. One month prior, the patient’s mother noticed some breakdown and ulceration in a skin fold within the hemangioma that was painful to touch and produced scant foul-smelling red fluid. With the suspicion of a bacterial infection, their pediatrician prescribed a seven-day course of topical bacitracin zinc 500units/gm ointment twice a day. His mother reported no improvement or worsening of symptoms with treatment.

At the time of presentation, there was a 7x5cm blanching red patch on the left inner medial thigh with maceration along the skin fold and a healing erosion with fine scale-crust on the superior border (Figure 1). Punch biopsy was then performed, and topical ketoconazole was provided for prophylaxis while results were pending. The biopsy results showed GLUT-1 positivity uptake by thin-walled vessels in the papillary dermis indicating a superficial infantile hemangioma. It also showed coexistent subacute eczematoid dermatitis with eosinophils likely representing allergic contact dermatitis from bacitracin. Wound culture revealed a pansensitive superimposed bacterial infection with polymicrobial growth. The patient was then treated with topical mupirocin 2% twice a day for the infection and petroleum jelly-based ointment (Vaseline) for eczematous dermatitis. On follow-up three weeks later there was significant improvement in the dermatitis and superimposed bacterial infection (Figure 2). Instructions were given to continue current medications and pat the lesion dry after bathing. The patient will be followed for assessment of lesional growth or complications such as ulceration which might prompt the initiation of topical or oral beta blocker therapy.

Figure 1. Eczematoid dermatitis at initial presentation of IH-MAG on left inner thigh following seven-day course of topical bacitracin.

Figure 2. Resolving bacterial infection and eczematoid dermatitis at three week follow-up following initiation of topical mupirocin. The healing erosion is visible in the upper portion of the lesion.

DISCUSSION

Infantile hemangioma with minimal or arrested growth are a unique subgroup of IHs that tend to occur more commonly on the lower body as opposed to the head and neck predisposition of most IHs.⁵ While evidence seems to point towards IHs reaching 80 percent of their growth by five months, IH-MAGs may conclude their limited growth phase by three months of age.¹ Ulceration is the most common complication associated with hemangiomas but tends to occur at a lower rate in IH-MAGs, most likely due to the decreased proliferative potential.⁵ Lipoatrophy is another potential complication of IH-MAGs, reported to be as high as 13 percent, but further studies are needed to accurately understand the risks.⁶ Given the low complication rate and the lack of growth potential, active intervention in IH-MAGs is generally not necessary unless they are cosmetically disfiguring.⁷

Currently, the prevailing theory about the pathogenesis of hemangiomas is centered around local hypoxia promoting the release of growth factors such as hypoxia-inducible factor 1α, vascular endothelial growth factor, and stromal derived factor 1α leading to endothelial progenitor cell recruitment and aberrant vasculogenesis.⁸ Barnés, et al. explored this further, proposing that the shared immunophenotype of GLUT-1 immunostaining between IHs and fetal placenta cells indicates a placental origin to hemangiomas, most likely placental villi cells. This placental origin theory is further elucidated by the increase in incidence of IH patients whose mothers underwent chorionic villus sampling or had placental pathology, most notably placenta previa and preeclampsia.⁹ It has been hypothesized by Suh, et al. that the reason for minimal to absent growth potential in IH-MAGs may be related to their anatomic distribution. Compared to the head and neck, the lower body is accustomed to reduced blood flow which could lead to decreased release and response to endothelial progenitor cell growth factors and subsequently less vascular proliferation.⁵

To the best of our knowledge, there are only two other case reports of eczematous dermatitis associated with IH-MAG. They were both facial IH-MAGs. One was treated with topical 2.5% hydrocortisone ointment in addition to 1.5mg/kg/day of propranolol, and the other was treated with topical tacrolimus (Protopic) ointment as well as 2mg/kg/day propranolol.^3,4 Our case is unique in its concurrent eosinophilic eczematous dermatitis and superimposed bacterial infection. Drawing from our knowledge of the association between dermatitis and port-wine stains, the vascularity within IH-MAGs can lead to increased inflammation and cytokines, increasing the risk of dermatitis in the area.¹⁰ It has been further proposed that the increased temperature in blood-rich areas can create a more favorable environment for bacteria and yeast overgrowth and aggravate atopic dermatitis.³

Our patient’s IH-MAG was also located in an intertriginous area, thus leading to increased heat, moisture, and friction. This in addition to the suspected allergic contact dermatitis from the bacitracin could have led to further impairment of the skin barrier, worsening the bacterial infection and skin sensitivity. Conservative management of IH-MAGs should be explored on a case-by-case basis given their decreased risk of proliferation. That said, their increased vascularity and favored locations on the lower extremities may make them more predisposed to secondary eczematous reactions. In the case of our patient’s allergic contact dermatitis, eliminating the underlying cause (bacitracin) and treating the superimposed bacterial infection was sufficient to achieve symptomatic management.

The authors have no conflicts of interest relevant to this article to disclose.

1. Chang LC, Haggstrom AN, Drolet BA, et al. Growth characteristics of infantile hemangiomas: implications for management. Pediatrics. Aug 2008;122(2):360-7.

2. Corella F, Garcia-Navarro X, Ribe A, Alomar A, Baselga E. Abortive or minimal-growth hemangiomas: Immunohistochemical evidence that they represent true infantile hemangiomas. J Am Acad Dermatol. Apr 2008;58(4):685-90.

3. Roncone K, Kindley KJ, Zlotoff B. Eczematous dermatitis in an infantile hemangioma with minimal or arrested growth. Pediatr Dermatol. Mar 2018;35(2):261-262.

4. Alsaid RQ, Al-Musalhi BH. Progressing eczematous dermatitis in an infantile hemangioma with minimal or arrested growth treated with tacrolimus ointment. JAAD Case Rep. Jun 2020;6(6):572-573.

5. Suh KY, Frieden IJ. Infantile hemangiomas with minimal or arrested growth: a retrospective case series. Arch Dermatol. Sep 2010;146(9):971-6.

6. Bessis D, Bigorre M, Labrèze C. Reticular infantile hemangiomas with minimal or arrested growth associated with lipoatrophy. J Am Acad Dermatol. May 2015;72(5):828-33.

7. Ma EH, Robertson SJ, Chow CW, Bekhor PS. Infantile Hemangioma with Minimal or Arrested Growth: Further Observations on Clinical and Histopathologic Findings of this Unique but Underrecognized Entity. Pediatr Dermatol. Jan 2017;34(1):64-71.

8. Chang EI, Chang EI, Thangarajah H, Hamou C, Gurtner GC. Hypoxia, hormones, and endothelial progenitor cells in hemangioma. Lymphat Res Biol. 2007;5(4):237-43.

9. Barnés CM, Christison-Lagay EA, Folkman J. The placenta theory and the origin of infantile hemangioma. Lymphat Res Biol. 2007;5(4):245-55. doi:10.1089/lrb.2007.1018

10. Fonder MA, Mamelak AJ, Kazin RA, Cohen BA. Port-wine-stain-associated dermatitis: implications for cutaneous vascular laser therapy. Pediatr Dermatol. Jul-Aug 2007;24(4):376-9.