Gastrointestinal Tract Granular Cell Tumor in the Pediatric Population: A Multicenter Experience.

IF 1.3 4区 医学 Q3 PATHOLOGY Pediatric and Developmental Pathology Pub Date : 2024-05-01 Epub Date: 2024-01-05 DOI:10.1177/10935266231220472
Muhammad Shaheen, Benjamin J Wilkins, Archana Shenoy, Kathleen Byrnes, Xiaoyi Tina Zhang, Iván A González
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Abstract

Background: Pediatric granular cell tumors (GCT) involving the gastrointestinal tract (GIT) are rare with limited case report/series reported to date.

Methods: Multicenter retrospective study of pediatric GIT GCT.

Results: A total of 10 cases were included in the study with a median age of 13.5 years (range: 7-18 years) and were predominantly female patients (60%). In half of the patients no significant medical history was present with the remaining 5 having Crohn disease (10%), eosinophilic esophagitis (EoE) (10%), Crohn disease and EoE (10%), growth hormone deficiency (10%), and aplasia cutis congenita (10%). The GCT median size was 1.3 cm (range: 1-1.6 cm) and were more commonly located in the esophagus (70%) followed by the stomach (20%) and rectum (10%). Most of the cases showed round/polygonal tumor cells with abundant granular cytoplasm, and none of the cases had nuclear atypia, increased mitotic activity, or tumor cell necrosis. None of our cases received specific therapy for GCT other than clinical follow-up, and none of the patients had evidence of local recurrence or metastatic disease.

Conclusion: We present our multicenter experience with GIT GCT, all cases had a benign course. Interestingly, 4 of the esophageal GCT cases (including 2 patients with EoE) showed an eosinophil-rich esophagitis in the underlying mucosa.

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儿科胃肠道颗粒细胞瘤:多中心经验。
背景:累及胃肠道(GIT)的小儿颗粒细胞瘤(GCT)非常罕见,目前报道的病例/系列有限:涉及胃肠道(GIT)的小儿颗粒细胞瘤(GCT)非常罕见,迄今报道的病例/系列有限:方法:对小儿胃肠道颗粒细胞瘤进行多中心回顾性研究:研究共纳入 10 例病例,中位年龄为 13.5 岁(7-18 岁),以女性患者为主(60%)。半数患者无明显病史,其余5人分别患有克罗恩病(10%)、嗜酸性粒细胞食管炎(EoE)(10%)、克罗恩病和EoE(10%)、生长激素缺乏症(10%)和先天性切口增生症(10%)。GCT 的中位尺寸为 1.3 厘米(范围:1-1.6 厘米),通常位于食道(70%),其次是胃(20%)和直肠(10%)。大多数病例的肿瘤细胞呈圆形/多角形,具有丰富的颗粒状胞质,无细胞核不典型性、有丝分裂活性增强或肿瘤细胞坏死。除临床随访外,我们的病例中没有人接受过针对 GCT 的特殊治疗,也没有人出现局部复发或转移性疾病:我们介绍了多中心的 GIT GCT 病例,所有病例均为良性病程。有趣的是,4 例食管 GCT 病例(包括 2 例 EoE 患者)的粘膜下层出现了富含嗜酸性粒细胞的食管炎。
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来源期刊
CiteScore
3.70
自引率
5.30%
发文量
59
审稿时长
6-12 weeks
期刊介绍: The Journal covers the spectrum of disorders of early development (including embryology, placentology, and teratology), gestational and perinatal diseases, and all diseases of childhood. Studies may be in any field of experimental, anatomic, or clinical pathology, including molecular pathology. Case reports are published only if they provide new insights into disease mechanisms or new information.
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