Diffuse neonatal hemangiomatosis in a very low‐birthweight infant treated with erythropoietin

Okuno, Takashi; Tokuriki, Shuko; Yoshino, Tomomi; Tanaka, Nanae; Ohshima, Yusei

doi:10.1111/ped.12517

Cited by 7 publications

(5 citation statements)

References 11 publications

(12 reference statements)

Supporting

Mentioning

Contrasting

Order By: Relevance

“…Several recent cases involving an excellent response of DHN to propranol. 10 In our case, the infant showed a positive clinical response to steroid treatment, with a gradual reduction in dyspnea and jaundice, and a restoration of normal liver function tests.…”

Section: Discussionsupporting

confidence: 48%

Unusual Case of Diffuse Neonatal Hemangiomatosis Case Report and Literature Review

El Mansoury,

El Yousfi,

Halfi

et al. 2024

Global Pediatric Health

View full text Add to dashboard Cite

Diffuse neonatal hemangiomatosis (DNH) is an infrequent condition characterized by the simultaneous occurrence of multiple cutaneous hemangiomas and the involvement of 3 or more organs. DNH is suspected when multiple hemangiomas are identified on the skin of the infant. Although it is benign in nature, DNH can lead to critical and life-threatening complications. Diagnosis primarily relies on clinical evaluation with a significant emphasis on imaging techniques. In this case report, we present an unusual pediatric case of diffuse infantile hemangioendothelioma, for which the investigative approach included ultrasound and CT scans. These imaging methods were instrumental in revealing the presence of lesions in the liver, thyroid, and brain, ultimately playing a pivotal role in making the diagnosis of DNH. A positive clinical and biological improvement was observed with corticosteroid treatment during a 3-month follow-up.

show abstract

Section: Discussionsupporting

confidence: 48%

Unusual Case of Diffuse Neonatal Hemangiomatosis Case Report and Literature Review

El Mansoury,

El Yousfi,

Halfi

et al. 2024

Global Pediatric Health

View full text Add to dashboard Cite

show abstract

“…A literature search revealed 15 other cases described with multifocal IHs and brain involvement [5,6,7,8,9,10,11,12,13,14,15,16,17,18]. Table 2 summarizes the data reported for these cases including our patient.…”

Section: Discussionmentioning

confidence: 84%

“…This high frequency of central nervous system involvement is unprecedented, and does not reflect the reports in the literature thereafter. Most of the infants described in this study and in other case reports [5,6,7,8,9,10,11,12,13,14,15,16,17,18] were symptomatic, with symptoms such as increased head circumference, flaccid paralysis, cranial nerve palsy, decreased rectal tone, areflexia, and others. Radiological findings in these cases include IHs, but also hemorrhages and hydrocephalus.…”

Section: Introductionmentioning

confidence: 86%

Screening for Brain Involvement in Infants with Multifocal Cutaneous Infantile Hemangiomas

Friedland¹,

Amitai²,

Zvulunov³

2017

Dermatology

View full text Add to dashboard Cite

Background: Multifocal (≥5) infantile hemangiomas (IHs) are known as a risk factor for extracutaneous involvement. Liver is the most commonly involved organ, but involvement of other systems has also been reported. This study aims to describe the characteristic findings in a group of infants with multiple cutaneous hemangiomas, with emphasis on intracranial involvement. Methods: A retrospective case series study was carried out in a pediatric dermatology unit of a tertiary pediatric medical center. Patients diagnosed with multiple cutaneous IHs from 2006 to 2015 were identified by a computerized search. Clinical data were retrieved from the medical charts. Results: A total of 60 infants (37 females and 23 males) were identified for analysis. Forty-four brain ultrasounds were recorded and reported as normal. One patient out of the 44 was later diagnosed with a small asymptomatic hemangioma seen on a brain MRI/MRA done for another indication. Conclusion: Brain hemangiomas may present as an asymptomatic incidental finding in infants presenting with multifocal cutaneous and liver IHs. The single case reported in our study emphasizes the low prevalence and the benign course expected. Therefore, routine ultrasound screening for brain involvement is probably unnecessary for this population.

show abstract

“…Approximately 16% of hemangiomas do not respond to steroid therapy, and b-blockers are considered the first-line systemic therapy [ 1 ]. Storch et al proposed several possible molecular targets for Propranolol treatment, depending on the timing of treatment [ 1 , 7 ]. Vincristine has been described for the treatment of life-threatening or corticosteroid-resistant hemangiomas.…”

Section: Discussionmentioning

confidence: 99%

“…Hydrocephalus was described in three cases [ 2 , 3 , 5 ]. Four of these cases utilized medical treatment, leading to a regression in the size and number of cutaneous and visceral hemangiomas [ 2 , 5 – 7 ]. In two cases, a surgical approach was used for the treatment of hydrocephalus [ 2 , 3 ] (Table 1 ).…”

Section: Discussionmentioning

confidence: 99%

A case report of hemorrhagic presentation of diffuse neonatal hemangiomatosis (DNH) associated to obstructive hydrocephalus: Medical and neurosurgical considerations

Pavanello,

Piro,

Roggero

et al. 2023

Childs Nerv Syst

View full text Add to dashboard Cite

Background Diffuse neonatal hemangiomatosis (DNH) is a rare disorder typically recognized at birth or during the neonatal period. DNH involves three or more organ systems, including the central nervous system (CNS). In these cases, serious complications such as hemorrhages and obstructive hydrocephalus can develop. Case report We present a case of DNH with intracranial hypertension and CNS hemorrhagic lesions on the mesencephalic aqueduct, resulting in triventricular hydrocephalus, treated with endoscopic ventriculocisternostomy (ETV) and medical therapy. Discussion DNH is a rare condition that can involve the CNS with serious complications. From a review of the literature, we highlighted only two cases of DNH with brain involvement treated surgically. We report the successful outcome of ETV, along with surgical considerations, imaging findings, and the complete resolution of cerebral and skin lesions following medical therapy. Conclusions Medical therapy is not standardized and must be individualized. In patients who develop severe neurological symptoms such as obstructive hydrocephalus, surgery may be considered to avoid neurological sequelae.

show abstract

Diffuse neonatal hemangiomatosis in a very low‐birthweight infant treated with erythropoietin

Cited by 7 publications

References 11 publications

Unusual Case of Diffuse Neonatal Hemangiomatosis Case Report and Literature Review

Unusual Case of Diffuse Neonatal Hemangiomatosis Case Report and Literature Review

Screening for Brain Involvement in Infants with Multifocal Cutaneous Infantile Hemangiomas

A case report of hemorrhagic presentation of diffuse neonatal hemangiomatosis (DNH) associated to obstructive hydrocephalus: Medical and neurosurgical considerations

Contact Info

Product

Resources

About