Congenital infantile fibrosarcoma in a very low-birth-weight infant

Mukai, Makio; Sameshima, Hiroshi; Kodama, Yasuo; Yamashita, Rie; Kaneko, Masatoki; Ikenoue, Tsuyomu; Matsufuji, Hiroshi; Kondo, Kazuhiro; Chijiiwa, Kazuo

doi:10.1016/j.jpedsurg.2011.11.052

Cited by 8 publications

(7 citation statements)

References 17 publications

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“…All 4 experienced severe tumor hemorrhaging in utero, and 1 fetus died from hemorrhagic complications despite prompt treatments in response to the rapid deterioration of the fetal status. [12][13][14]16 The histology of all these cases was IF (Table 1). It was hypothesized that the rapidly growing and highly vascular nature of IF accounted for bleeding diathesis.…”

Section: Discussionmentioning

confidence: 99%

“…When the cases are limited to soft tissue sarcomas, to the best of our knowledge, 8 cases have been reported in PubMed-listed journals from 1989 to 2020 (Table 1). [10][11][12][13][14][15][16][17] Among these cases, 4 (50%) were not prenatally diagnosed as soft tissue tumors. 10,11,15,17 The neonates in these 4 cases suffered from severe hemorrhaging after vaginal delivery, and 1 died of hemorrhagic shock.…”

Section: Discussionmentioning

confidence: 99%

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Primitive Myxoid Mesenchymal Tumor of Infancy With Fatal Hemorrhage In Utero: A Case Report and Literature Review

Haga

Motojima

Masuda³

et al. 2022

Journal of Pediatric Hematology/Oncology

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Primitive myxoid mesenchymal tumor of infancy (PMMTI) is a rare soft tissue sarcoma in childhood. We present the case of a newborn male who experienced a severe hemorrhage in utero from the tumor on the scalp. He died at the age of 24 hours owing to hemorrhagic shock. The tumor was posthumously diagnosed as PMMTI. A literature search indicated that cases of severe hemorrhage from soft tissue sarcomas in utero or at birth are limited to infantile fibrosarcoma. This is the first case of PMMTI with massive hemorrhage. Clinicians must be aware of hemorrhagic complications of PMMTI.

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Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Primitive Myxoid Mesenchymal Tumor of Infancy With Fatal Hemorrhage In Utero: A Case Report and Literature Review

Haga

Motojima

Masuda³

et al. 2022

Journal of Pediatric Hematology/Oncology

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“…Although rare, patients with a prenatal diagnosis of IFS have been reported [12][13][14][15][16][17]. These cases were detected on routine US in the third trimester in asymptomatic pregnancies or on US performed due to maternal abdominal distension, and reduced or absent fetal movements [12,13,15].…”

Section: Discussionmentioning

confidence: 99%

Clinical and Molecular Characteristics, Management and Outcome of Infantile Fibrosarcoma: A Retrospective 18-Year Single-Institution Review

Marina¹,

Belen²,

Jessica³

et al. 2022

SCR

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Infantile fibrosarcoma (IFS) is the most common non-rhabdomyosarcoma soft-tissue sarcoma in children under 1 year of age, with local aggressiveness, but a high cure rate with conservative treatment. We report our experience with 15 patients with histological diagnosis of IFS treated at our Institution from January-2003 to December-2020. The median age was 6.72 months (range: 0-36); 66% were males. The extremities were the most common tumor site (66.7%). The tumor size was > 5 cm in 10 patients. Only one patient had metastatic disease (lung). Molecular studies were performed in 14 patients and were positive in ten: nine ETV6/NTRK3 and one RAF1. Initial non-mutilating surgery was performed in six patients. A delayed surgery could be performed in 4/8 patients after neoadjuvant chemotherapy with Vincristine - Actinomycin-D, and in one patient a watch-and-wait strategy was adopted. With a median follow-up of 58.67 months, all patients are alive except for the patient with metastatic disease. The 5-year overall and event-free survival were 93.5% and 86.5% respectively. Although IFS is locally aggressive, it has a high rate of overall survival due to its good response to chemotherapy and non-mutilating surgery even with involved margins. Advances in molecular genetics have improved diagnosis and refined classification.

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“…At the age of 2.5 years, the infant was healthy without evidence of recurrence. Because cases with congenital infantile sarcoma usually have a poor prognosis, termination of pregnancy at an appropriate time, or postnatal early treatment to improve outcome are discussed ( 25 ).…”

Section: Discussionmentioning

confidence: 99%

Cardiac Myosarcoma in a Newborn Infant—A Case Report and Literature Review

Vakrilova

Marinov

Hitrova‐Nikolova

et al. 2021

Front. Cardiovasc. Med.

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Background: Malignant cardiac neoplasms are extremely rare in neonates. Prenatal diagnosis is often not available. Initial symptoms can mimic non-cardiac diseases. We present a pre-mature newborn, birth-weight 2,480 g, 34 gestational weeks, which underwent cardiac surgery due to a cardiac tumor.Case Summary: This was a 3–rd pregnancy after two spontaneous abortions to a mother with thrombophilia, diabetes, hydramnios, and retroplacental hematoma. The baby was admitted to NICU with transitory respiratory failure and inborn infection; hence oxygen-supplementation and antibiotics were initiated. On day 11 a deterioration with tachypnea, high oxygen requirements, significantly increasing C-reactive protein values were noted. Chest radiographs were unremarkable. On day 18 a life-threatening condition with clinical symptoms of shock was identified. Echocardiography showed a large tumor formation in the right atrium, reduced blood flow in the right ventricle and pulmonary artery. On day 19 cardiac surgery was performed: a large tumor with a myxomatous appearance that occupied the cavity of the right atrium and infiltrated the annulus of the tricuspid valve was extirpated. The front wall was restored with a patch. Hemodynamics was temporarily stabilized. On the following day, ventricular fibrillation and asystole occurred. Despite life support efforts, the outcome was lethal. The histological result confirmed leiomyosarcoma of the right atrium and right ventricular hypotrophy.Conclusion: Inborn cardiac sarcomas are extremely rare. The prognosis is poor. Due to fast progression in the third trimester, they can be missed by fetal echocardiography in earlier pregnancy. The postnatal clinical course is aggressive, not specific until invasive infiltration or obstruction by the tumor mass occurred. In our case, the sarcoma mimicked an inborn infection, followed by nosocomial infection and septic shock. Despite low incidence, cardiac tumors should be kept in mind and echocardiography should be conducted if there are unclear symptoms with progressive deterioration during the neonatal period.

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Congenital infantile fibrosarcoma in a very low-birth-weight infant

Cited by 8 publications

References 17 publications

Primitive Myxoid Mesenchymal Tumor of Infancy With Fatal Hemorrhage In Utero: A Case Report and Literature Review

Primitive Myxoid Mesenchymal Tumor of Infancy With Fatal Hemorrhage In Utero: A Case Report and Literature Review

Clinical and Molecular Characteristics, Management and Outcome of Infantile Fibrosarcoma: A Retrospective 18-Year Single-Institution Review

Cardiac Myosarcoma in a Newborn Infant—A Case Report and Literature Review

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