Literature DB >> 24635971

Rapidly progressive congenital rhabdomyosarcoma presenting with multiple cutaneous lesions: an uncommon diagnosis and a therapeutic challenge.

Bharat Rekhi1, Sajid Shafique Qureshi2, Gaurav Narula3, Sumeet Gujral4, Purna Kurkure3.   

Abstract

Congenital rhabdomyosarcomas (RMSs) are rare tumors with variable clinical presentations. A 2 month-old, term male neonate (37 weeks, 4 days), weighing 3.2kg, born to a 24 year-old primigravida, by simple vaginal delivery presented with multiple erythematous papulonodular lesions over his trunk that progressed to his whole body, on the first day of delivery. Prior to conception, his mother was treated for polycystic ovarian disease. On the tenth day, his chest computed tomogram scans revealed multiple, heterogeneously enhancing, bilateral pleural-based soft tissue density nodular lesions, along with multiple soft tissue density lesions, involving skeletal muscles of all his body parts. Microsections from two biopsies (on 10th day and after 2 months) revealed a malignant round cell tumor with cells arranged in a diffuse, solid pattern, comprising embryonal and solid alveolar components. Immunohistochemically, the tumor cells were diffusely positive for desmin, myoD1 and myogenin. Diagnosis of embryonal and alveolar (mixed type) RMS was offered. Further molecular cytogenetic analysis was negative for PAX3-FKHR and PAX7-FKHR. The patient was induced on chemotherapy as per intergroup rhabdomyosarcoma study IV protocol. There was treatment response with near total remission after 8 weeks of treatment. Thereafter, new lesions started appearing that also disappeared after modification of the chemotherapy drugs. However, after 16 months, the baby died of brain metastasis. The present case forms the fourth case report of an aggressive form of a congenital RMS with extensive cutaneous involvement and brain metastasis. A review of previously diagnosed cases of congenital RMSs is discussed herewith.
Copyright © 2014 Elsevier GmbH. All rights reserved.

Entities:  

Keywords:  Congenital rhabdomyosarcoma; PAX3-FKHR; PAX7-FKHR; Pediatric sarcoma

Mesh:

Substances:

Year:  2014        PMID: 24635971     DOI: 10.1016/j.prp.2014.02.001

Source DB:  PubMed          Journal:  Pathol Res Pract        ISSN: 0344-0338            Impact factor:   3.250


  4 in total

Review 1.  Malignant Superficial Mesenchymal Tumors in Children.

Authors:  Philippe Drabent; Sylvie Fraitag
Journal:  Cancers (Basel)       Date:  2022-04-26       Impact factor: 6.575

2.  Congenital Rhabdomyosarcoma: a different clinical presentation in two cases.

Authors:  Ida Russo; Virginia Di Paolo; Carmelo Gurnari; Angela Mastronuzzi; Francesca Del Bufalo; Pier Luigi Di Paolo; Angela Di Giannatale; Renata Boldrini; Giuseppe Maria Milano
Journal:  BMC Pediatr       Date:  2018-05-15       Impact factor: 2.125

3.  Orbital rhabdomyosarcoma with skin metastasis: a case report.

Authors:  Fadwa Elomrani; Salima Touri; Imane Ouziane; Narjiss Berrada; Saber Boutayeb; Hind Mrabti; Basma Elkhannoussi; Hassan Errihani
Journal:  BMC Res Notes       Date:  2014-09-24

4.  Importance of histopathological analysis and molecular genetics in a rare neonatal case of rhabdomyosarcoma.

Authors:  Prudence Gramp; Tania Zappala; Lena Von Schuckmann; Diane Payton; Laura Wheller
Journal:  Australas J Dermatol       Date:  2022-05-02       Impact factor: 2.481

  4 in total

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