Gupta Aarushi, Sharma Swati, Mittal Abhenil, Barwad Adarsh, Rastogi Sameer
Department of Radiodiagnosis, ABVIMS and Dr. RML Hospital, New Delhi, India.
Department of Medical Oncology, Institute Rotary Cancer Hospital, All India Institute of Medical Sciences, New Delhi, India.
Radiol Case Rep. 2020 Dec 18;16(3):504-510. doi: 10.1016/j.radcr.2020.12.027. eCollection 2021 Mar.
Inflammatory myofibroblastic tumors (IMT) are rare soft tissue tumors of intermediate malignant potential with tendency for local recurrence. Although they can occur at all age groups, occurrence in infants is extremely unusual and their imaging characteristics are not well described. A 3-month-old female infant presented with gradually progressive abdominal distention without any fever or weight loss. She had a large ill-defined homogenous hypodense lesion of size 8.4 × 11.4 × 11.3 cm (APxTraxSag) in the abdomen showing mild delayed post contrast enhancement. She underwent exploratory laparotomy with gross total excision of mesenteric mass, histopathology of which was suggestive of IMT. She had recurrence within 6 months of complete resection with a well-defined heterogeneously enhancing lesion of size 1.8 × 1.8 × 2.3cm (APxTraxSag) in right paravesical region abutting the bladder without invasion with a similar lesion of size 4.4 × 2.1 × 3 cm (APxTraxSag) in left subdiaphragmatic region abutting superior surface of spleen (no invasion). Since, surgery in our patient would have entailed splenectomy and partial cystectomy, systemic therapy with ceritinib (anaplastic lymphoma kinase [ALK] inhibitor) was planned for her with which she had a near complete response after 2 months. A high index of suspicion is required to differentiate IMT from other common causes of mesenteric masses in children and role of radiologist is quintessential in this regard. Local recurrence with abutment but without invasion of surrounding structures points to the intermediate malignant pathology of IMT and may provide a clue to diagnosis. Systemic therapy is effective in patients who are ALK positive and destructive surgery should be avoided.
炎性肌纤维母细胞瘤(IMT)是一种罕见的具有局部复发倾向的中间恶性潜能软组织肿瘤。尽管它可发生于所有年龄组,但在婴儿中极为罕见,其影像学特征也未得到充分描述。一名3个月大的女婴出现逐渐加重的腹胀,无发热或体重减轻。她腹部有一个大小为8.4×11.4×11.3厘米(前后径×横径×矢状径)的边界不清的均匀低密度大肿块,增强扫描后呈轻度延迟强化。她接受了剖腹探查术,肠系膜肿块被完整切除,组织病理学检查提示为IMT。在完全切除后6个月内复发,右膀胱旁区域有一个边界清晰的不均匀强化肿块,大小为1.8×1.8×2.3厘米(前后径×横径×矢状径),紧邻膀胱但未侵犯,左膈下区域有一个类似的肿块,大小为4.4×2.1×3厘米(前后径×横径×矢状径),紧邻脾脏上表面(未侵犯)。由于对我们的患者进行手术需要行脾切除术和部分膀胱切除术,因此计划对她进行用色瑞替尼(一种间变性淋巴瘤激酶[ALK]抑制剂)的全身治疗,2个月后她有了近乎完全的反应。需要高度怀疑才能将IMT与儿童肠系膜肿块的其他常见病因区分开来,在这方面放射科医生的作用至关重要。局部复发但紧邻周围结构而未侵犯提示IMT的中间恶性病理特征,可能为诊断提供线索。全身治疗对ALK阳性患者有效,应避免进行破坏性手术。