Infantile hemangioma of the upper lip and enamel hypoplasia: Two case reports

Ochando‐Ibernón, Gemma; Azaña-Defez, J.M.

doi:10.1111/pde.13988

Cited by 5 publications

(4 citation statements)

References 17 publications

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“…Lip hemangiomas (ulcerated or not) are rarely reported in PHACE syndrome [ 20 , 23 , 24 ]. A total number of 14 papers (out of 400) are reported in PubMed about the association of lip hemangiomas to PHACE syndrome [ 25 ].…”

Section: Discussionmentioning

confidence: 99%

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“Face(s)” of a PHACE(S) Syndrome Patient before and after Therapy: Particular Case Report and Review of Literature

et al. 2022

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A rare, uncommon disorder called PHACE(S) (P-posterior fossa anomalies, H-hemangioma, A-arterial anomalies, C-cardiac anomalies, E-eye anomalies, and S-sternal cleft) of unknown etiology was rarely reported. Children are susceptible to developing PHACE(S) syndrome from the moment they are born. It may be challenging for a physician to appropriately diagnose and treat children with PHACE due to the multifaceted nature of the disease and the extensive range of consequences that may be associated with it. A one-month-old newborn girl was admitted to hospital with extensive, multiple facial infantile hemangiomas, ulceration of the lower lip hemangioma-like lesion, cardiovascular, sternal, and neurological concomitant malformations. Five days following the initial application of the medication, systemic treatment with propranolol and topical treatment with silver sulfadiazine produced their first noticeable benefits. The lip ulceration was mostly healed and facial hemangioma started to regress. The regression continued under therapy and this effect persists for 6 months since Propranolol therapy ended. No cardiovascular or neurological clinical events have been registered during follow-up. The present case has three peculiarities: (1) high number of facial hemangiomas; (2) presence of subependymal cyst not yet reported in the literature associated with PHACE syndrome; and (3) lack of cardiovascular events during therapy knowing that these events frequently appear in PHACE syndrome patients.

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Section: Discussionmentioning

confidence: 99%

“…Because of the rarity of PHACE, the clinical trial began in 2009 and is expected to last until 2030 [ 17 ]. Dental root anomalies, enamel hypoplasia [ 18 , 19 ], and lip hemangiomas [ 20 ] are also uncommonly reported in patients with PHACE syndrome.…”

Section: Introductionmentioning

confidence: 99%

“Face(s)” of a PHACE(S) Syndrome Patient before and after Therapy: Particular Case Report and Review of Literature

et al. 2022

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“…As an example, a recent report documented that small indeterminate IHs or focal mixed perioral hemangiomas on the upper lip were associated with enamel hypoplasia, as seen in patients with PHACE syndrome. 11 Thus, indeterminate hemangiomas in certain high-risk locations can be associated with underlying anomalies and, depending on clinical context, may warrant evaluation for associated developmental anomalies such as those seen in PHACE syndrome. Additionally, IHs in high-risk locations that may appear at first glance to be focal could actually be indeterminate or partial segmental and carry an increased risk for complications.…”

Section: Discussionmentioning

confidence: 99%

“…While this might not alter current screening recommendations, this awareness could allow for further evaluations should specific signs or symptoms arise in individual patients. As an example, a recent report documented that small indeterminate IHs or focal mixed perioral hemangiomas on the upper lip were associated with enamel hypoplasia, as seen in patients with PHACE syndrome . Thus, indeterminate hemangiomas in certain high-risk locations can be associated with underlying anomalies and, depending on clinical context, may warrant evaluation for associated developmental anomalies such as those seen in PHACE syndrome.…”

Section: Discussionmentioning

confidence: 99%

Mapping of Segmental and Partial Segmental Infantile Hemangiomas of the Face and Scalp

et al. 2021

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IMPORTANCERecognizing segmental infantile hemangioma (IH) patterns is important for risk stratification and provides clues to pathogenesis. Previously, segmental hemangiomas were mapped to 4 facial regions, 3 corresponding to known facial metameres.OBJECTIVES To refine existing maps of facial segmental IHs, examine so-called indeterminate hemangiomas as they relate to known segmental patterns, and define a novel pattern of segmental scalp hemangiomas. DESIGN, SETTING, AND PARTICIPANTSThis retrospective cohort study was conducted at 4 pediatric dermatology centers (

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Infantile hemangioma. Part 1: Epidemiology, pathogenesis, clinical presentation and assessment

Bandera

Sebaratnam

Wargon

et al. 2021

Journal of the American Academy of Dermatology

108

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Infantile hemangioma of the upper lip and enamel hypoplasia: Two case reports

Abstract: Segmental infantile hemangiomas related to PHACE syndrome have recently been associated with enamel hypoplasia. We present two cases of solitary, localized upper lip infantile hemangioma with enamel hypoplasia of deciduous teeth.

Cited by 5 publications

References 17 publications

“Face(s)” of a PHACE(S) Syndrome Patient before and after Therapy: Particular Case Report and Review of Literature

“Face(s)” of a PHACE(S) Syndrome Patient before and after Therapy: Particular Case Report and Review of Literature

Mapping of Segmental and Partial Segmental Infantile Hemangiomas of the Face and Scalp

Infantile hemangioma. Part 1: Epidemiology, pathogenesis, clinical presentation and assessment

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