Successful treatment of diffuse neonatal hemangiomatosis with propranolol: a case report

Zhou, Hui; Li, Qiao‐fei; Cao, Guangling; Hong-zhong, Ling; Dai, Huaping; Chen, Xiaohong

doi:10.1111/ddg.12389

Cited by 5 publications

(5 citation statements)

References 2 publications

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“…Moreover, these lesions regress after treatment with propranolol and oral corticosteroids and usually do not proliferate after the age of 6 months. 16 Morphologically, another differential diagnosis is with bacillary angiomatosis that, in contrast with our case, appears with an associated neutrophil infiltrate. In our cases, no systemic involvement was seen, but as it is reported to occur in half of the cases, investigation of this kind of manifestation is justified.…”

Section: Patientcontrasting

confidence: 58%

Congenital cutaneous pyogenic granuloma: Report of two cases and review of the literature

Schneider

Garcia

Aleixo³

et al. 2019

J Cutan Pathol

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Congenital cutaneous pyogenic granuloma is a rare benign vascular tumor with clinical and histopathological features similar to infantile hemangioma. It usually presents as a red, pedunculated and highly friable papule. On histopathological analysis, one can see a capillary vessel proliferation with lobular pattern and endothelial proliferation. The differential diagnosis is based on negativity of glucose transporter 1 (GLUT1) immunochemistry studies. We report two infants with congenital pyogenic granuloma, one with a unique cutaneous lesion and the other with multiple lesions affecting both skin and mucosal surfaces. These two cases highlight the importance of the differential diagnosis based on the GLUT1 immunochemistry analysis considering the distinct treatments required to these infant vascular tumors.

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Section: Patientcontrasting

confidence: 58%

Congenital cutaneous pyogenic granuloma: Report of two cases and review of the literature

Schneider

Garcia

Aleixo³

et al. 2019

J Cutan Pathol

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“…Propranolol, which has been utilized in the treatment of neonatal hemangiomatosis (12), has been used in the treatment of MLT, with inconsistent results (5,10). Although our patient did receive propranolol as well, it ultimately did not improve her liver failure or her GI bleeding, and she was found to have multiple GI lesions despite prior treatment with propranolol.…”

Section: Discussionmentioning

confidence: 66%

Orthotopic liver transplant for multifocal lymphangioendotheliomatosis with thrombocytopenia

Yang

Zhou

Alexopoulos

et al. 2016

Pediatric Transplantation

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An eight-yr-old female with a history of multifocal lymphangioendotheliomatosis and thrombocytopenia presented for MVT. The patient had multiple vascular lesions in the skin and stomach in infancy. Although her cutaneous lesions resolved with vincristine and methylprednisolone, her gastric lesions persisted. Eight yr later, she was diagnosed with portal hypertension and decompensating liver function despite therapy with bevacizumab, propranolol, furosemide, and spironolactone. Upon presentation, she was found to have a Kasabach-Merritt-like coagulopathy in association with multiple lesions in her GI tract and persistent gastric lesions. Although treatment with methylprednisolone and sirolimus normalized her coagulation factors and d-dimer levels, she never developed sustained improvement in her thrombocytopenia. Her liver function continued to deteriorate and she developed hepatorenal syndrome. Given better outcomes after OLT in comparison with MVT, she underwent OLT, with the plan to manage her GI lesions with APC post-transplant. Post-transplant, her liver function and coagulopathy normalized, and GI tract lesions disappeared upon screening with capsule endoscopy. The patient is doing well, without recurrence of either GI lesions or thrombocytopenia, at 18 months after transplantation.

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“…Its use in the treatment of infantile hemangiomas, the most common benign tumor of the skin, has been discovered accidentally and it was verified that propranolol administration is highly efficient in inducing tumor regression with very few adverse effects (40). Thus far, the beneficial effects of propranolol have been observed in the treatment of neonatal hemangiomatosis (51,52), placental chorioangioma (53) and CCM (13).…”

Section: Discussionmentioning

confidence: 99%

Propranolol therapy for cerebral cavernous malformations

et al. 2022

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Cerebral cavernous malformations (CCMs) are vascular malformations characterized by the abnormal growth of vascular structures in the central nervous system. However, the precise mechanism(s) responsible for the development of CCM vascular abnormalities remain poorly understood. Although the mechanisms of action of propranolol in CCM have not yet been fully explored it is not commonly prescribed, it has been shown to be effective in children and appears to play a protective role in the prevention of CCM-derived hemorrhage in adults. The present study performed in vitro and ex vivo assays in order to examine the effects of propranolol on endothelial cells (ECs). The percentage of CD14 + /CD31 + cells and the levels of VEGF in the peripheral blood (PB) of a child patient with CCM, with recurrent seizures and hemorrhages, who was maintained under propranolol therapy, were also analyzed. In addition to the effects of propranolol on differentiated ECs, and the decrease angiogenic-related features in vitro and ex vivo, it was observed that in the PB of this patient, propranolol administration decreased the percentage of circulating cells sharing monocytic and EC features (CD14 + /CD31 + cells), as well as the VEGF levels; this was concomitant with a good prognosis and with the reversion of CCM lesions. A decrease in VEGF levels by propranolol may also be involved in the impairment of the recruitment of CD14 + /CD31 + monocytes functioning as endothelial progenitor cells to sustain the vascular lesion. On the whole, the present study demonstrates that propranolol impairs angiogenesis in vitro and may thus be a useful tool for the clinical management of CCM. Moreover, the present study highlights the monitorization of the levels of CD14 + /CD31 + monocytes and VEGF levels as a useful tool for predicting the clinical efficacy of propranolol in patients with CCM.

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Successful treatment of diffuse neonatal hemangiomatosis with propranolol: a case report

Cited by 5 publications

References 2 publications

Congenital cutaneous pyogenic granuloma: Report of two cases and review of the literature

Congenital cutaneous pyogenic granuloma: Report of two cases and review of the literature

Orthotopic liver transplant for multifocal lymphangioendotheliomatosis with thrombocytopenia

Propranolol therapy for cerebral cavernous malformations

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