Discussion
In 2008, Léauté-Labrèze et al reported the efficacy of propranolol as a
treatment for IH. Numerous articles have since been published on this
treatment modality, with the majority reporting positive outcomes with
propranolol use and IH in their patient populations 9.
Propranolol has become the first-line treatment for problematic
hemangiomas, and its high efficacy and minimal adverse effects have been
extensively demonstrated. Additional treatment modalities include laser
therapy, surgical excision, and intralesional steroid injections. Pulse
dye laser is the most used in the management of IH and is effectively
used to remove the residual telangiectasias during involution and to
promote wound healing of ulcerated hemangiomas that are unresponsive to
medical management 10. Surgery should be considered in
some cases when it would lead to better results than the process of
involution or medical therapy, although timing remains
controversial1.
This study confirms previous findings that suggest IH are more
frequently associated with white non-Hispanic ethnicity and female sex.
In prior literature, about 60% of lesions involve the head and neck,
followed by 25% on the trunk and 15% on the extremities1,11,12. In our study, however, our cohort had a much
higher percentage of head and neck lesions, representing about 90% of
all IH. Morphologically, we found focal lesions were the most common in
our cohort, aligning with previous findings 12,13.
PHACE syndrome is characterized by vascular cutaneous and nerve lesions
of multiple systematic systems, which often cause structural or
functional impairments 14. Another study found that
PHACE syndrome was more frequently observed in patients with large
segmental facial hemangiomas 15. In our cohort, 10%
of patients had a diagnosis of PHACE syndrome and we found a
statistically significant association between head/facial lesions and a
diagnosis of PHACE syndrome. However, we did not find a statistically
significant association between PHACE syndrome and size distribution.
Additionally, the most common treatment indication was cosmetic
disfigurement with most patients initiating treatment before 6 months of
age. Although protocols for propranolol administration vary per
institution, the dosage administered is usually 1-3mg/kg daily, with
most physicians favoring 2mg/kg daily 16. This aligns
with the results of our study, with 98% of patients receiving a daily
dose of 2mg/kg. The mean length of propranolol treatment was about 11
months and 97% of patients responded to propranolol with 80% having a
nearly complete or complete response to an initial treatment cycle.
Surgical interventions for IH include resection or ablation with laser17 . Seven percent of patients in our study received
laser treatment with PDL or neodymium yttrium aluminum garnet (Nd:YAG),
and 10% received surgical excision in addition to propranolol
treatment. While the introduction of propranolol has decreased the need
for surgical excisions for IH, surgical intervention may still be
indicated in cases of ulceration, parent preference, patients with late
presentation, and patients with sequelae after IH involution18.
Regrowth after discontinuation of propranolol therapy occurs in as many
as 10-15% of patients 19 in some reports. Previous
studies have investigated the predictive factors for IH recurrence and
have found that most cases were due to partial regression, meaning that
recurrent lesions likely originate from the residual hemangioma cells3,20. Our study demonstrates similar findings with
almost 60% of patients requiring a second round of propranolol after
having a partial lesion regression response to initial propranolol
treatment. Additionally, the length of initial propranolol treatment for
the 14 patients requiring a second cycle was 8 months compared to 11
months in the entire cohort. Previous studies have recommended prolonged
monitoring of facial IH after initial propranolol treatment termination
to detect regrowth 21. Additionally, proliferation of
residual lesions is likelier when propranolol therapy is ended at
earlier stages compared to later stages, which is a plausible
explanation for our study results 19,22.
One interesting consideration our study proposes is a familial history
of hemangiomas or cardiac abnormalities as a risk factor for
necessitating a second propranolol cycle. A known risk factor for the
development of IH includes a family history of IH. Additionally,
previous studies have identified a higher frequency of cardiac
abnormalities among patients with IH in respect to the general
population. However, the correlation of these risk factors with the need
for a subsequent propranolol cycle remains novel. It is important to
acknowledge that the significance of these findings is limited by the
relatively small number of patients necessitating a second round of
propranolol therapy. Further exploration with larger cohorts is
essential to validate and fully understand the implications of these
potential risk factors in guiding treatment strategies for IH
recurrence.
The primary limitation of our study is its retrospective design, as
there may have been missing or unreported data. A larger patient cohort
and a multi-institutional approach are imperative for future
investigations to corroborate these findings. Additionally, the
prominence of head/face hemangiomas might be influenced by referral
biases inherent in a tertiary center setting.