Successful Treatment of Relapsed Multifocal Nonvisceral Infantile Myofibromatosis

Roßbach, C.; Tannapfel, Andrea; Troebs, Ralf-Bodo; Hirsch, Wolfgang; Koerholz, Dieter

doi:10.1080/08880010500278780

Cited by 10 publications

(4 citation statements)

References 14 publications

Supporting

Mentioning

Contrasting

Order By: Relevance

“…Myofibromatosis with visceral involvement results in death in about 70% of patients. Chemotherapy or interferon may be effective in some cases [161][162][163][164].…”

Section: Infantile Myofibromatosismentioning

confidence: 99%

Fibroblastic and Myofibroblastic Tumors in Children and Adolescents

2012

View full text Add to dashboard Cite

Fibroblastic and myofibroblastic tumors in children and adolescents are a relatively common group of soft tissue proliferations that range from reactive to hamartomatous to neoplastic, with a full spectrum of benign, intermediate, and malignant neoplasms. These lesions are diagnostically challenging because of morphologic and immunohistochemical overlap, despite significant clinical, genetic, and prognostic differences. The fibromatoses are a major subgroup, and all types of fibromatoses can occur in the 1st 2 decades of life. Intermediate and malignant fibroblastic-myofibroblastic tumors are an important group that includes variants of fibrosarcoma and other tumors with recurrent cytogenetic or molecular genetic abnormalities and low metastatic potential. Pathologic examination is enhanced by adjunct techniques, such as immunohistochemistry, cytogenetics, and molecular genetics, although morphology provides the ultimate criteria for a specific diagnosis. This article reviews the clinicopathologic features of fibroblastic and myofibroblastic tumors with an emphasis on the unique aspects of these neoplasms in children and adolescents, the use of diagnostic adjuncts, and differential diagnoses.

show abstract

“…Myofibromatosis with visceral involvement results in death in about 70% of patients. Chemotherapy or interferon may be effective in some cases [161][162][163][164].…”

Section: Infantile Myofibromatosismentioning

confidence: 99%

Fibroblastic and Myofibroblastic Tumors in Children and Adolescents

2012

View full text Add to dashboard Cite

show abstract

“…Recurrent IM is rare and it has been reported in 17 patients in the literature (Table 3). 14,26,[34][35][36][37][38][39][40][41] Noteworthy is the high recurrence received chemotherapy of various combinations 34,38 or spontaneously regressed. 14,41 As demonstrated by our Patient 2, recurrence can be treated with the same chemotherapeutic regimen used for the treatment of the initial disease.…”

Section: Discussion and Literature Reviewmentioning

confidence: 99%

“…Recurrent IM is rare and it has been reported in 17 patients in the literature (Table 3). 14,26,34–41 Noteworthy is the high recurrence rate reported by Beck et al in a series of head and neck lesions 34 . This high rate of recurrence reflects an unfavorable location of tumors that were only subtotally resected and needed further surgical intervention when they regrew.…”

Section: Discussion and Literature Reviewmentioning

confidence: 99%

Diverse presentation and tailored treatment of infantile myofibromatosis: A single‐center experience

Manisterski¹,

Benish²,

Levin³

et al. 2020

Pediatric Blood & Cancer

View full text Add to dashboard Cite

Background: Infantile myofibromatosis (IM) is a rare benign fibrous tumor with diverse clinical presentations and treatments, such as watchful waiting, surgical excision, and low-dose chemotherapy. Procedure: Clinical presentation and tailored treatment of five infants with solitary and generalized IM are described, together with a review of the literature. Results: Three patients underwent total-body magnetic resonance imaging (MRI) at diagnosis and during follow up, which revealed disease extension that aided in designing treatment. Visceral involvement included central nervous system, cardiac, gastrointestinal, muscle, bone, and subcutaneous tissue lesions. The patient with the solitary form of IM was followed up without treatment and had spontaneous improvement. Patients with the multicentric form received intravenous low-dose methotrexate and vinblastine chemotherapy. One patient who received oral methotrexate due to cardiac involvement and unfeasible central line access had excellent results. Recurrence was successfully treated by the same methotrexate and vinblastine regimen as that administered at diagnosis. Conclusions: We suggest screening all patients with one or more IM lesions by means of total body MRI due to its inherent superior soft tissue resolution. Total-body MRI may also be used for routine follow up. Oral methotrexate can be administered successfully in patients that lack central line access, and recurrent lesions can be treated with the same chemotherapeutic combination as that given at diagnosis. Long-term follow up is needed, since recurrence could appear years after initial presentation of the disease.

show abstract

“…In a more recent review, Levine et al summarized 30 cases of multicentric IM with or without visceral involvement 6 . Review of these cases revealed only 1 in which involvement was only osseous; all others cases had lesions that involved skin and soft tissue or organs 20 .…”

Section: Discussionmentioning

confidence: 99%