Multiple Cutaneous Hemangiomas Research Articles

Diagnóstico clínico de hemangiomatose neonatal disseminada: um relato de caso

Hemangioma is the most common vascular tumor of childhood. It may present as multiple cutaneous hemangiomas on rare occasions, with or without visceral involvement. The condition of lesions restricted to the skin is called benign neonatal hemangiomatosis, and in the case of visceral lesions associated with cutaneous lesions, the denomination is disseminated or diffuse neonatal hemangiomatosis (DNH). For the diagnosis of DNH, three criteria must be met: onset in the neonatal period, involvement of three or more organs, and absence of malignancy in hemangiomas. ASAS, a full-term, vaginally delivered newborn, weight: 3560g, length: 52cm, head circumference: 35cm, APGAR: 9/9. Maternal history of syphilis during pregnancy - mother adequately treated in pregnancy. At birth, she presented with purpuric lesions of variable size on the chest, limbs, feet, and hands, which discolored under digital pressure; larger lesions with relief, not scaly. He developed severe heart failure and was admitted to the Neonatal Intensive Care Unit (NICU) in a secondary hospital in the Federal District. Imaging tests were performed: Computed tomography of the skull, thorax, and total abdomen, which revealed the presence of a possible hemangioma in the left frontal lobe, telangiectasias in the lungs, and multiple hepatic hemangiomas. He had a clinical diagnosis of DNH. The case described corroborates the fact that knowledge about the disease is relevant, as early diagnosis can lead to more accurate approaches that may reduce morbidity and mortality from the disease.

Diagnosis of infantile subglottic hemangioma and the effect of oral propranolol

ObjectivesTo investigate the clinical characteristics of infantile subglottic hemangioma (SGH), and to observe the safety and efficacy of propranolol in the treatment of SGH. MethodsThe data of 21 children diagnosed with SGH and treated with propranolol in our hospital from March 2013 to January 2021 were retrospectively analyzed and followed up. ResultsAmong the 21 cases, there were 7 males and 14 females. SGH was found 11 left-sided, 9 right-sided and 1 bilateral-sided. The clinical manifestations included stridor (13/21), respiratory distress (6/21), barking cough (5/21), feeding difficulty (4/21), three concave sign (4/21), cyanosis (2/21) and hoarseness (1/21). 8 patients had multiple cutaneous hemangiomas. The age of presentation ranged from 1 to 8 months, with a median of 1.1 months. 18 cases (85.7 %) had a history of misdiagnosis, 14 bronchitis/pneumonia, 5 laryngomalacia, 2 laryngeal obstruction and 1 asthma. The median ages at diagnosis were 3 months, with a range of 1.2–28 months. The treatment duration ranged from 6 to 25.6 months, with an average of (14.3 ± 4.9) months. Age at termination of treatment ranged from 9 to 38 months, with a median of 18.6 months, and only 2 cases were beyond 2 years old at that time. No adverse side effects from propranolol therapy occurred and all 21 cases were cured. ConclusionsWe advocate a strong index of suspicion for SGH presenting with respiratory symptoms under 2 years old who has poor effect or repeated condition after routine treatment. Laryngoscopy combined with contrast-enhanced CT can confirm the diagnosis of SGH. Oral propranolol is safe and effective, and that early diagnosis and intervention of propranolol without further delay are crucial to the successful management. We advocate continue propranolol treatment beyond 18 months of age, furthermore, 2 years old may be the best time for therapy termination.

Clinical evaluation of transcatheter arterial embolization combined with propranolol orally treatment of infantile hepatic hemangioma.

To assess the efficacy of transcatheter arterial embolization (TAE) plus propranolol treatment for infantile hepatic hemangioma (IHH). A retrospective study of symptomatic IHH and hemodynamic changes in IHH was conducted between 2016 and 2019. There were five boys and seven girls with diffuse lesions (n = 7) and multifocal lesions (n = 5). Hepatomegaly and abdominal distension (n = 6) were the predominant clinical presentations. Seven patients (58.3%) had multiple cutaneous hemangiomas. Pulmonary arterial hypertension, heart failure (n = 4), and hypothyroidism (n = 4) were observed. A total of 17 TAE procedures were performed in 12 IHH cases, with a technical success rate of 100%. All patients received standard propranolol orally, and one patient was orally administered metacortandracin. Two patients died of heart failure and multiple organ dysfunction caused by an enlarged liver. In addition, one patient was not reexamined after discharge. Of the remaining nine children, the average follow-up time was 10.78months (range 2-28months), and they all responded well to TAE combined with oral propranolol. TAE combined with propranolol is safe and effective for the treatment of IHH, demonstrating low complication rates.

Benign Neonatal Hemangiomatosis.

Benign neonatal hemangiomatosis (BNH) is a rare, self-limiting subtype of infantile hemangiomas (IHs), in which infants with multiple cutaneous hemangiomas lack visceral involvement. Other subtypes of IHs exist that may mimic BNH and can be life-threatening depending on hemangioma location and size. At birth, a 29 5 / 7 -week preterm female presented with several pinhead-sized pink papules distributed throughout her body. At 10 days of age, the patient had 12 enlarged domed-shaped red papules in a generalized distribution throughout her body. Over several weeks, the number and size of the domed-shaped red papules continued to increase to a total of 26 located on the head, chest, abdomen, back, legs and arms. They were of firm consistency with both smooth and lobulated surfaces. A diagnosis of BNH was made after extensive workup did not reveal any extracutaneous hemangiomas. Due to the lack of extracutaneous involvement and low-risk location/size of hemangiomas in our patient, no interventions were pursued and an observation-only approach was implemented. The patient remained stable while followed up over 8 months, with the size of the hemangiomas only increasing slightly in proportion to the patient's natural body growth. Given the life-threatening nature of certain hemangioma subtypes, it is important to implement a proper workup and subtype diagnosis as early as possible in any infant with multiple hemangiomas.

Screening for infantile hepatic hemangioma in patients with cutaneous infantile hemangioma: A multicenter prospective study

Abdominalultrasonography has been proposed to screen for infantile hepatic hemangioma (IHH) in patients with multiple cutaneous infantile hemangiomas (IHs). The aim of this study was to establish the optimal cutoff point for the number of cutaneous IHs needed to screen for IHH. We performed a prospective, multicenter study to screen for IHH in patients younger than 9months who had multiple cutaneous IHs (n≥3) on ultrasonography. For comparison, a group of patients with 1 or 2 focal cutaneous IHs was also recruited. In total, 676 patients with at least 3 cutaneous IHs and 980 patients with 1 or 2 focal cutaneous IHs were enrolled. Thirty-one patients were found to have IHH. A higher number of cutaneous IHs was associated with an increased risk of IHH (R=0.973; P<.001). Receiver operating characteristic curve analysis showed that 5 cutaneous IHs was the optimal cutoff point to screen for IHH, with an area under the curve of 0.872 (P<.001; 95% confidence interval, 0.789-0.955). This was an uncontrolled study. Screening for IHH is recommended in patients younger than 9months who present with 5 or more cutaneous IHs.

Diffuse neonatal hemangiomatosis presenting as congestive cardiac failure - A case report

Infantile hepatic hemangioma has substantial arteriovenous shunting which may lead to cardiovascular compromise and hydrops fetalis. It may present as hepatomegaly since the entire liver is involved in most cases. As mortality is very high, a high index of suspicion is required to make a diagnosis and common complications arising out it, especially in the presence of cutaneous hemangioma. We present a 2-month-old baby born at term presented with features suggestive of sepsis with multiple cutaneous hemangiomas, and on evaluation, there was congestive cardiac failure, which was initially thought of cardiac origin but subsequently came out to be arteriovenous shunting of blood in liver.

Multiple pulmonary, hepatic and cutaneous hemangiomas treated with propranolol

Multiple pulmonary, hepatic and cutaneous hemangiomas treated with propranolol

Successful Propranolol Treatment of Multiple Infantile Hemangiomas with Predominant Neuroaxial Involvement: a Case Report

Infantile hemangiomas (IH) are the most common vascular tumor of infancy. Occurrences during the neonatal period are divided in multiple congenital, limited to skin involvement, or diffuse neonatal hemangiomatosis (DNH). DNH is considered to be a benign and rare disease, determined by the presence of multiple cutaneous and extracutaneous hemangiomas. The encephalic and medullary involvement of IHs is unusual and rarely reported. The authors report a case of infantile hemangioma (IH) as diffuse neonatal hemangiomatosis with predominant central nervous system involvement on a neonate, treated with propranolol. We discuss the use propranolol as main therapy and review the available data regarding IHs with neuroaxial involvement and its treatment.

Impact of Screening for Hepatic Hemangiomas in Patients with Multiple Cutaneous Infantile Hemangiomas.

Hepatic hemangiomas are often found in association with multiple cutaneous infantile hemangiomas. Screening abdominal ultrasonography has been recommended for patients with five or more cutaneous lesions. We sought to determine whether hemangiomas found through screening had improved clinical outcomes. Patients entered into our hepatic hemangioma registry between 1995 and 2012 were reviewed. Seventy-two patients with multiple cutaneous and hepatic hemangiomas were identified; 43 (60%) were detected through screening. The median age at diagnosis was 41 days for screened patients and 53 days for those not screened. Screening detected 40 (93%) multifocal and 3 (7%) diffuse hemangiomas, compared to 18 (62%) and 11 (38%), respectively, in the nonscreened group. Patients identified by screening had lower incidences of congestive heart failure and hypothyroidism and were less likely to receive treatment for their hemangiomas. The mortality rate in the children not screened was 28% (n = 8). None of the patients found by screening died (p < 0.001). Multivariate analysis of treated patients demonstrated that screening was a significant predictor of reduced mortality (p = 0.04). Hepatic hemangiomas found through screening ultrasonography are less likely to develop serious clinical sequelae. Although the reasons for this may include detection of hemangiomas that are less likely to progress to symptomatic disease, it appears that it also allows for earlier intervention for more concerning (e.g. diffuse) subtypes. Screening may allow for closer surveillance and earlier treatment before life-threatening progression in a subset of infants with liver hemangiomas, preventing complications and reducing mortality.

“Cyrano nose” associated with hepatic hemangiomas successfully treated with propranolol

Hemangioma of the nasal tip is commonly described as "Cyrano nose" and his treatment is extremely difficult because of its location and possible severe aesthetic complications like definitive nasal deformation. We describe a patient who presented at two months of age with a "Cyrano nose" associated with multiple hepatic and cutaneous hemangiomas, which completely resolved after therapy with propranolol. Treatment was well tolerated and aesthetic result was excellent.

Multiple cutaneous and hepatic hemangiomas in infants.

The objectives of the study were to determine the rate of hepatic hemangiomas in infants with cutaneous infantile hemangiomas that were screened by abdominal ultrasound; identify morphological subtypes and number of cutaneous infantile hemangiomas that are likely to suggest the presence of hepatic hemangiomas; and identify clinical history, physical findings, or laboratory abnormalities that may predict hepatic involvement. A retrospective study was conducted between 2000 and 2007 on 37 infants with cutaneous hemangiomas who underwent abdominal ultrasound for hepatic hemangiomas. Infants were classified into subgroups based upon the morphology of their cutaneous hemangioma(s). Data collected included clinical history, physical examination findings, sonographic findings, laboratory results, treatment(s) rendered, and clinical outcome. Eight of 37 infants (22%) had hepatic hemangiomas. Infants with miliary (30-100 pinpoint/small cutaneous hemangiomas), six or more small cutaneous hemangiomas, and one large with one or more small cutaneous hemangiomas were more likely to have concurrent hepatic hemangiomas. No other clinical findings were associated with hepatic involvement. Similar to other studies, our study found clinically asymptomatic hepatic hemangiomas in 22% of infants with multiple cutaneous infantile hemangiomas. No clinical findings studied were predictive of hepatic involvement.

Multiple cutaneous infantile haemangiomas and the risk of internal haemangioma

British Journal of DermatologyVolume 169, Issue 1 p. 188-191 Correspondence Multiple cutaneous infantile haemangiomas and the risk of internal haemangioma A.D. Vredenborg, A.D. Vredenborg Department of Paediatrics, Division of Paediatric DermatologySearch for more papers by this authorS.R. Janmohamed, S.R. Janmohamed Department of Paediatrics, Division of Paediatric Dermatology KinderHaven, Havenziekenhuis, Rotterdam, the Netherlands Department of Paediatric Surgery andSearch for more papers by this authorP.C.J. de Laat, P.C.J. de Laat Department of Paediatrics, Erasmus MC, University Medical Center, Rotterdam, the NetherlandsSearch for more papers by this authorG.C. Madern, G.C. Madern Department of Paediatric Surgery andSearch for more papers by this authorA.P. Oranje, A.P. Oranje Department of Paediatrics, Division of Paediatric Dermatology KinderHaven, Havenziekenhuis, Rotterdam, the Netherlands Department of Dermatology, Maasstad Ziekenhuis, Rotterdam, the NetherlandsSearch for more papers by this author A.D. Vredenborg, A.D. Vredenborg Department of Paediatrics, Division of Paediatric DermatologySearch for more papers by this authorS.R. Janmohamed, S.R. Janmohamed Department of Paediatrics, Division of Paediatric Dermatology KinderHaven, Havenziekenhuis, Rotterdam, the Netherlands Department of Paediatric Surgery andSearch for more papers by this authorP.C.J. de Laat, P.C.J. de Laat Department of Paediatrics, Erasmus MC, University Medical Center, Rotterdam, the NetherlandsSearch for more papers by this authorG.C. Madern, G.C. Madern Department of Paediatric Surgery andSearch for more papers by this authorA.P. Oranje, A.P. Oranje Department of Paediatrics, Division of Paediatric Dermatology KinderHaven, Havenziekenhuis, Rotterdam, the Netherlands Department of Dermatology, Maasstad Ziekenhuis, Rotterdam, the NetherlandsSearch for more papers by this author First published: 19 February 2013 https://doi.org/10.1111/bjd.12229Citations: 18 Correspondence: Arnold P. Oranje. E-mail: a.oranje@inter.nl.net A.D.V. and S.R.J. contributed equally to this work. Funding sources: none.Conflicts of interest: none declared. Read the full textAboutPDF ToolsRequest permissionExport citationAdd to favoritesTrack citation ShareShare Give accessShare full text accessShare full-text accessPlease review our Terms and Conditions of Use and check box below to share full-text version of article.I have read and accept the Wiley Online Library Terms and Conditions of UseShareable LinkUse the link below to share a full-text version of this article with your friends and colleagues. Learn more.Copy URL Share a linkShare onFacebookTwitterLinked InRedditWechat Citing Literature Volume169, Issue1July 2013Pages 188-191 RelatedInformation

Propranolol as a First‐line Treatment for Diffuse Infantile Hepatic Hemangioendothelioma

Propranolol as a First‐line Treatment for Diffuse Infantile Hepatic Hemangioendothelioma

Congestive heart failure with diffuse neonatal hemangiomatosis – case report and literature review

Vascular hemangiomas are considered to be a common finding among neonates and infants. The presence of five or more cutaneous hemangiomas may carry a higher incidence of multiple organ involvement An eleven week old female infant with high cardiac output heart failure and multiple cutaneous hemangiomas was referred to our clinic. Soon after birth she failed to gain weight and developed breathlessness. There was partial response to Captopril and Frusemide therapy, with development of a progressive cough. Echocardiogram and liver and brain ultrasound scans revealed heart chamber enlargement, multiple liver hemangiomas and an isolated cerebellar arterio-venous malformation (AVM). Supportive treatment for congestive heart failure combined with propranolol therapy resulted in rapid clinical response and recovery. The diagnosis of Diffuse Neonatal Hemangiomatosis in infants with failure to thrive and symptoms of heart failure raises the possibility of internal organ involvement. Augmentation of propranolol in such cases may cause regression of Hemangiomatosis with patient clinical improvement. We report our experience with a single patient of Hemangiomatosis induced heart failure which responded to propranolol combined with medication to control heart failure and failure to thrive prevention. This treatment should be further evaluated with special attention to potential adverse effects, tolerance and compliance.

Consumptive hypothyroidism in an Egyptian baby with benign neonatal hemangiomatosis: a case report

IntroductionBenign neonatal hemangiomatosis is a condition in which multiple cutaneous hemangiomas appear at birth or shortly thereafter; visceral complications are absent. Here, we report a case of a consumption hypothyroidism in an Egyptian baby with benign neonatal hemangiomatosis.Case presentationAn 8-month-old Egyptian boy with benign neonatal hemangiomatosis was referred to our institution for evaluation of developmental delay. Initial examination revealed a quiet baby who was able to sit only with support. He had hypotonia, a large anterior fontanelle, puffy eyes, cold extremities, hypothermia, bradycardia, and abdominal distension. An examination of his skin revealed more than 100 dome-shaped red-purple cutaneous hemangiomas that varied in size from 5 to 10mm on the back, the abdomen and the extremities without mucus membrane involvement. He had low serum free thyroxine concentration and triiodothyronine levels and high thyroid-stimulating hormone and reverse-triiodothyronine levels. A work-up that involved appropriate imaging ruled out visceral involvement. Based on the above mentioned data, a diagnosis of consumptive hypothyroidism due to benign neonatal hemangiomatosis was made. He was started on oral thyroid medication which was gradually increased to 90μg L-thyroxine daily (15μg/kg/day). After three months of treatment, he was able to sit alone without support and he had normal levels of thyroid-stimulating hormone and serum free thyroxine.ConclusionThyroid function should be assessed periodically in babies with benign neonatal hemangiomatosis, especially if symptoms of hypothyroidism appear or the size and number of hemangiomatosis increase rapidly. Moreover, high doses of L-thyroxine may be needed to achieve euthyroidism during the infancy.

Prospective Study of the Frequency of Hepatic Hemangiomas in Infants with Multiple Cutaneous Infantile Hemangiomas

Prospective Study of the Frequency of Hepatic Hemangiomas in Infants with Multiple Cutaneous Infantile Hemangiomas

Generalized Haemangiomas with Visceral Involvement: Case Report

Haemangiomas are the most common benign tumours of infancy. Most cases of haemangiomas present as solitary lesions, but about 20% have more than one lesion. The presence of 3 lesions is uncommon and the presence of 4 or more lesions is rare. The presence of multiple cutaneous haemangiomas greatly enhance the chances that the child could have visceral haemangioma. We report an 11- month old infant with multiple cutaneous haemangiomas with visceral involvement complicated by congestive cardiac failure. Initially,he responded to treatment with prednisolone and antifailure drugs, but died later as a result of exacerbation. Some of the problems associated with managing such cases in a resource-poor country are highlighted.

Occult Androgenetic-Biparental Mosaicism and Sporadic Hepatic Mesenchymal Hamartoma

The incidence of hepatic mesenchymal hamartoma (HMH) is increased in patients with placental mesenchymal dysplasia (PMD), which appears to be caused by androgenetic-biparental mosaicism (ABM). We hypothesized that occult ABM might underlie cases of HMH with no known history of PMD. Formalin-fixed, paraffin-embedded HMH specimens from 10 such patients and liver specimens from 6 non-HMH controls were identified retrospectively from the surgical pathology records of a pediatric hospital. The relative abundance of maternal and paternal alleles was assessed by quantitative polymerase chain reaction amplification of polymorphic short tandem repeats and single nucleotide polymorphisms located on 15 different chromosomes. Androgenetic-biparental mosaicism was diagnosed in one patient based on global allelic imbalances at all informative loci. In that patient, the greatest imbalances were observed in stroma-rich portions of the hamartoma, with no significant imbalance in histologically normal liver or epithelium-rich portions of the hamartoma. A retrospective, unbiased review of the histology and clinical records from all 10 patients revealed no morphologic or clinical correlates to distinguish the affected patient, except that she had multiple cutaneous hemangiomas, which like HMH, appear to be more common in patients with PMD. Our findings suggest that other patients with apparently sporadic HMH, hemangioma, or other lesions seen more frequently with PMD may harbor occult ABM. Recognition of ABM may be important because its long-term consequences are unknown but may be significant.

Infantile intraspinal and extensive cutaneous hemangiomas

A 6-week-old girl presented with multiple cutaneous hemangiomas (figure 1). She had brisk lower extremity reflexes without other deficits. Imaging revealed extensive hemangiomas in liver and …

Prospective Study of the Frequency of Hepatic Hemangiomas in Infants with Multiple Cutaneous Infantile Hemangiomas

Multiple cutaneous infantile hemangiomas have been associated with hepatic hemangiomas. Screening of infants with five or more cutaneous infantile hemangiomas with abdominal ultrasound is often recommended. The aim of this study was to determine the frequency with which hepatic hemangiomas occur in infants with five or more cutaneous infantile hemangiomas compared to those with one to four cutaneous infantile hemangiomas and to characterize the clinical features of these hepatic hemangiomas. A multicenter prospective study of children with cutaneous infantile hemangiomas was conducted at pediatric dermatology clinics at Hemangioma Investigator Groups sites in the United States, Canada, and Spain between October 2005 and December 2008. Data were collected, and abdominal ultrasonography was performed on infants younger than 6 months old with five or more cutaneous infantile hemangiomas and those with one to four cutaneous infantile hemangiomas. Twenty-four (16%) of the 151 infants with five or more cutaneous infantile hemangiomas had hepatic hemangiomas identified on abdominal ultrasound, versus none of the infants with fewer than five (p = 0.003). Two of the 24 infants with hepatic hemangiomas received treatment specifically for their hepatic hemangiomas. Infants with five or more cutaneous infantile hemangiomas have a statistically significantly greater frequency of hepatic hemangiomas than those with fewer than 5. These findings support the recommendation of five or more cutaneous infantile hemangiomas as a threshold for screening infants younger than 6 months old for hepatic hemangiomas but also demonstrate that the large majority of these infants with hepatic hemangiomas do not require treatment.