Unrelenting facial segmental hemangiomas: A case series of late growth and recurrent ulcerations

Faith, Esteban Fernández; Cordisco, Maria

doi:10.1111/pde.14253

Cited by 4 publications

(6 citation statements)

References 12 publications

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“…Two had been treated with oral propranolol, suggesting that beta blocker therapy for the initial IH may not completely protect against future dysesthesias. Notably, late ulceration up to several years of age has recently been reported and ulceration itself can cause pain 3 . However, none of our patients had signs of ulceration that would explain their symptoms.…”

Section: Discussionmentioning

confidence: 54%

“…Notably, late ulceration up to several years of age has recently been reported and ulceration itself can cause pain. 3 However, none of our patients had signs of ulceration that would explain their symptoms.…”

Section: Discussionmentioning

confidence: 60%

“…With or without medical intervention, some IHs leave behind fibrofatty residua, anetoderma, or persistent vascular markings or discoloration after the involution phase is complete. The majority of patients do not report symptoms associated with these residua; recurrent ulcerations even after involution are occasionally reported 3 . We report seven patients who described varying degrees of dysesthesias in sites of involuted IH in the absence of ulceration, including the IH characteristics, symptoms, and treatments rendered.…”

Section: Introductionmentioning

confidence: 89%

“…The majority of patients do not report symptoms associated with these residua; recurrent ulcerations even after involution are occasionally reported. 3 We report seven patients who described varying degrees of dysesthesias in sites of involuted IH in the absence of ulceration, including the IH characteristics, symptoms, and treatments rendered.…”

mentioning

confidence: 99%

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Persistent dysesthesias in involuted infantile hemangiomas: An uncommon complication in a common condition

Braun

Metry

Frieden

et al. 2021

Pediatric Dermatology

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Background/Objectives Infantile hemangiomas (IHs) are common benign vascular tumors of infancy. IHs tend to grow in the first few months of life and then gradually involute over years, often leaving fibrofatty residua or textural changes in their place. Classically, these lesions are painless throughout their entire natural history; however, we now report on seven patients with involuted IH with intermittent but persistent sensory symptoms. Methods This is a multicenter case series in which members of the Birthmarks Focused Study Group of the Pediatric Dermatology Research Alliance (PeDRA) and the Hemangioma Investigator Group contributed patients with IH and dysesthesias from their clinical practices. Charts were then reviewed to document clinical details. Results Seven patients were included, presenting at an average age of 14.6 years (range 3‐48 years) for complaints related to discomfort in the region of involuted IH. The majority (6/7) reported pain or tenderness to the area. One patient reported pruritus. All patients reported intermittent symptoms. The length of symptoms ranged between 4 months and 5 years. Treatment was attempted in 5/7 patients. Ice, oral propranolol, topical capsaicin, and intralesional triamcinolone partially improved symptoms. Conclusions Persistent cutaneous dysesthesias were present in seven patients, in most cases many years after completion of involution. Further research is needed to fully elucidate the pathophysiology and optimal treatments for this IH complication.

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Section: Discussionmentioning

confidence: 54%

Section: Discussionmentioning

confidence: 60%

Section: Introductionmentioning

confidence: 89%

mentioning

confidence: 99%

See 2 more Smart Citations

Persistent dysesthesias in involuted infantile hemangiomas: An uncommon complication in a common condition

Braun

Metry

Frieden

et al. 2021

Pediatric Dermatology

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“…Severe and recalcitrant ulcerations are a well‐recognized challenge in managing patients with IH, yet detailed reports on the subset of patients with AU are sparse and limited to case reports and small case series. These have been reported in different contexts, including IH with refractory ulcerations requiring multimodal therapy, worsening of ulceration in segmental IH following initiation or dose escalation of systemic beta‐blockers, and persistent or recalcitrant ulcerations in facial segmental IH in patients with PHACE syndrome 4–6 …”

Section: Discussionmentioning

confidence: 99%

Clinical characteristics of infantile hemangiomas with aggressive, persistent, and destructive ulceration

Gautam,

Frieden,

Shah

et al. 2023

Pediatric Dermatology

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Background/ObjectivesUlceration is a common complication of infantile hemangioma (IH). Severe, persistent ulceration occurs in a minority of patients. This study aims to characterize the clinical features of IH with aggressive ulceration (AU).MethodsMulticenter retrospective study of clinical features of IH with AU.ResultsThirty‐five patients with AU were identified and included in the study. The majority of AU occurred in segmental IH (23/35, 65%). Segmental IH with AU were large (≥10 cm2; 16/23, 69%, p < .001) with a thin (<3 mm) superficial component (16/23, 69%, p < .001). Localized IH with AU had a thick (>3 mm) superficial component (11/12, 92%, p < .001).All diaper area IH with AU (9/35) were segmental with thin superficial component (100%, p = .02). IH with AU in the head/neck (10/35) were more commonly localized (67%) and mixed (62.5%), while segmental, thick superficial morphology was more common on trunk (9/35) and upper extremities (7/35).ConclusionsIH resulting in AU differ in clinical features by anatomic site. Those in the diaper area are nearly always segmental with thin superficial component, whereas other sites tend to be localized, mixed, with thick superficial component. These distinct phenotypes may prove useful in the clinical setting for physicians to identify patterns of IH ulceration with increased risk of aggressive, persistent ulceration.

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Diagnostic Techniques for Infantile Subglottic Hemangiomas: A Scoping Review

Ezeh,

Tesema,

Hasnie

et al. 2024

The Laryngoscope

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ObjectiveInfantile subglottic hemangioma (SGH) poses a risk of airway compromise if untreated. Traditionally, operative endoscopy (OH) diagnoses SGH, but since the discovery of beta‐blockers' efficacy in treating infantile hemangiomas (IHs) in 2008, and advances in endoscopic technology, nonoperative methods have emerged. This review identifies endoscopic practices for diagnosing and monitoring infantile SGH during the oral beta‐blocker treatment era.Data SourcesA comprehensive literature search in October 2022 and August 2023 covered PubMed, Embase, Cochrane Library, SCOPUS, and Web of Science.Review MethodsThe search was limited to English‐language studies published since 2008, considering this when propranolol was demonstrated as an effective treatment option for IH. The articles were screened for relevance based on predefined inclusion and exclusion criteria.ResultsAfter inclusion and exclusion criteria, sixty final studies were identified, describing 240 cases of infantile SGH. Most children were diagnosed using OE alone (73.3%; n = 176/240), 23.3% (n = 56/240) using office‐based laryngoscopy procedures (OBPs) followed by OE, 3.3% using OBP alone (n = 8/240). There were no reported diagnostic endoscopy‐related complications. Twenty‐nine studies described using endoscopy plus diagnostic imaging to either confirm an SGH lesion, characterize the extent of disease spread, or rule out other causes of presenting symptoms. The proportion of infants diagnosed with OE alone decreased from 2008 to 2023.ConclusionOperative endoscopy remains the SGH diagnostic standard, but OBP adoption is increasing. Further research is needed to determine the optimal SGH diagnosis and management approach.Level of EvidenceNA Laryngoscope, 2024

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Unrelenting facial segmental hemangiomas: A case series of late growth and recurrent ulcerations

Abstract: A 23-day-old girl was referred for evaluation of a facial vascular "stain." Physical examination showed a segmental IH with minimal

Cited by 4 publications

References 12 publications

Persistent dysesthesias in involuted infantile hemangiomas: An uncommon complication in a common condition

Persistent dysesthesias in involuted infantile hemangiomas: An uncommon complication in a common condition

Clinical characteristics of infantile hemangiomas with aggressive, persistent, and destructive ulceration

Diagnostic Techniques for Infantile Subglottic Hemangiomas: A Scoping Review

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