Congenital hemangioma exhibiting postnatal growth

Abstract

Congenital hemangiomas are vascular tumors that are fully formed at birth, typically without postnatal growth. Noninvoluting congenital hemangiomas (NICH) have a distinctive clinical, radiologic, and histopathological profile and lack of expansion or involution over time.Herein, we describe two cases of NICH with atypical postnatal growth. | 549 Pediatric Dermatology BRIEF REPORT and in this comprehensive study, the authors reported a slight enlargement of the lesion over the years in few patients. 2 No further investigation regarding the postnatal growth of NICH was reported, until the International Society for the Study of Vascular Anomalies 2018 meeting, where two retrospective case series, documenting significant postnatal growth in NICH in a total of 16 patients, were brought to our attention. 3,4 Cossio et al 3 collected 7 cases of NICH with postnatal growth at a mean age of 4.3 years, manifested by the development of red papules on the surface of the lesion (5), bleeding from the papules (2), and development of pyogenic granuloma in one case. We postulate that our first case illustrates a congenital hemangioma with postnatal growth including the development of additional coarse telangiectasias over the years, while the second case showed early postnatal growth leading us to question our initial diagnosis.The pathophysiology for the early or late expansion of NICH is unknown, although persistent high-flow vessels in NICH may in part explain the tardive growth we observed in this tumor. 5 Regardless, postnatal expansion of NICH does not represent a proliferative phase of this vascular tumor, and we believe that the postnatal growth of NICH may prove to be more frequent than classically taught.

Kaposiform hemangioendothelioma: current knowledge and future perspectives

“…NICHs persist in a stable state without growth or involution. However, a slight enlargement of NICHs over the years has recently been reported [91]. PICHs evolve from RICHs to persistent NICH-like lesions.…”

Cutaneous ultrasound and its utility in Pediatric Dermatology: Part II—Developmental anomalies and vascular lesions

“…Venous thrombi, which are never seen in infantile hemangiomas, have also been described in congenital hemangioma . The sonographically identifiable persistent vascularity within NICH might explain the recently reported postnatal growth of these tumors . In our experience, CH and IH cannot be differentiated sonographically.…”

“…Congenital hemangiomas (CHs) are uncommon benign vascular tumors that, unlike IH, are characteristically present and fully grown at birth, although postnatal growth of CH has recently been reported . They often manifest as a thickened plaque or as an exophytic mass with a pink to violaceous color, coarse telangiectasias, and a halo of blanched skin.…”

Expanding the phenotypes of congenital hemangiomas

“…Hua et al described eleven cases of CH with a disproportionate postnatal growth and coined the term TECH (tardive expansion congenital hemangioma) 4 . The high flows found in these vascular lesions have been proposed as the cause of this postnatal growth of NICH 5 . Activation of GNAQ has also been postulated as a possible influence on the growth of these tumors 6 .…”

“…Although PICH have not been studied at the molecular level, they are likely to have the same mutations 2 . Recently, several case series of CH with atypical presentations have been reported 3‐6 . We present five cases of CH that demonstrated an unusual evolution.…”