Rare complication – skin atrophy – after systemic conservative therapy of infantile hemangioma

Infantile Hemangiomas still remain to be a challenge for the medical field, constantly raising questions about pathogenesis, risk factors, prognosis, evolution and management. Even though IHs are mostly harmless and „wait-and-see“ approach is reasonable (due to self-involution growth pattern), there are circumstances when intervention is crucial and compulsory [4]. The treatment of IH depends on different factors, like localization, stage and distribution type of the lesions, hemangioma type, presence of ulceration, systemic involvement, level of psychosocial condition of the child or parent [3]. Location of hemangiomas plays an important role in the selection of treatment approach. Any life threatening (airway) or function threatening (eyelid, periocular, lip, ear, nasal or ano-genital region) lesions are at increased risk of complications and early specific therapeutic intervention is needed [3, 5]. Active non-intervention tactic can be applied in majority of the remaining cases (especially when superficial, small, are covered with hair or cloth, small) which includes active observation of the infant during first few months and the education of the parent about course of the disease, intervention methods, complications and anticipatory guidance [3, 6]. When decision about the therapeutic intervention of IH is made, pre-treatment work-up should be executed. Before starting therapy via propranolol, children should be assessed for certain contraindications (propranolol allergy, heart failure, sinus bradycardia, bronchial asthma, hypotension, hypoglycemia, heart block, hypotension) [3]; Blood sugar levels, blood pressure and heart rate should be monitored and electrocardiogram should be performed as well. Pre-treatment investigations before initiation of steroid therapy comprises of monitoring anthropometric parameters and blood pressure, ruling out of primary immunodeficiency and active infection, performing of CBC, serum biochemistry tests, stool microscopy and chest x-ray [3].

Since 2008, the approach by Laute-Labreze, which includes the use of Propranolol as a treatment option of infantile hemangiomas, became first-option therapy in most of the cases because of its few adverse effects and favorable outcomes [6].

The serious side-effects of propranolol treatment such as dyspnea, hypoglycemia and bradycardia are well known [7‐10]. There also has been an association with certain medical skin conditions like psoriasis and beta-blockers [11]. It is also known that treatment with local injections can end in soft tissue atrophy [12]. We could not identify literature that implicated systemic beta-blockers for skin atrophy lesions. The skin lesions, such as skin atrophy, ulceration and hyperpigmentation have been reported after pulsed dye laser treatment as well [13].

Nowadays, skin atrophy is a huge challenge in dermatology. For example, there are many procedures that can be used in the management of atrophic acne scars: Resurfacing procedures (Chemical peeling, dermabrasion), lifting procedures (subcision, fillers), excisional techniques (punch grafting, elliptical excision) and others (skin needling, combinations techniques etc.) [14]. Skin atrophy is one of the complications of topical corticosteroid use as well [15]; Although it should resolve on its own after cessation of therapy, which can take months [15].

Topical and systemic beta-blockers have been associated with good results in regards to treatment of atrophic scars, keloids etc. [16].

One of the interesting components of the case is that we are unsure what was the thing that triggered development of the skin defect: was it systemic administration of propranolol, was it somehow linked to initial topical timolol solution, or was it linked to involution of the lesion, which due to abrupt borders of the IH started to form such a sequela.

There have been multiple reports indicating that involution of IH may lead to various types of lesions, including telangiectasia, fibrofatty tissue, and anetodermic skin. However, the specific factors contributing to this development are still not fully understood [17, 18]. Two studies have focused on the complications observed in a total of 358 patients: 187 patients who went untreated [17] and 171 patients who received treatment (oral propranolol) [18]. Among the untreated patients, an alarming 54.9% developed significant sequelae, while only 26.9% of the treated patients experienced such complications. This difference is considered statistically significant (chi-squared − 30.7, DF − 1, P < 0.0001), indicating that there is a lower chance of complications when using oral propranolol as a treatment. It is important to note, however, that based solely on this data, we cannot confirm or rule out oral propranolol as a definite causative agent for the observed outcomes, including our lesion at hand. Further research and investigation are needed to fully understand the relationship between oral propranolol and the development of these complications.

The other thing that makes us curious is the following: did the lesion start to heal itself or did the topical cream make a difference? The depth and size of the defect was clearly reduced (Figs. 1 and 2), however, since this was a single case and we did not compare it to a control, it would not be scientifically correct to estimate one or another. One thing we can say for sure is that further research is warranted, so that clinicians know what the appropriate management strategies for a lesion like this are, since the incidence of hemangioma is known to range from 0.97 to 1.97% [19].