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类变形杆菌综合征:一种磷酸酶和张力蛋白同源物错构瘤肿瘤综合征的罕见表型。

Proteus-Like Syndrome: A Rare Phenotype of Phosphatase and Tensin Homolog Hamartoma Tumor Syndrome.

作者信息

Abu-Shaban Kamil, Bakri Kenan, Kihm Amber, Elsamaloty Mazzin, Elsamaloty Haitham

机构信息

Radiology, University of Toledo College of Medicine, Toledo, USA.

Radiology, University of Toledo Medical Center, Toledo, USA.

出版信息

Cureus. 2022 Apr 14;14(4):e24135. doi: 10.7759/cureus.24135. eCollection 2022 Apr.

Abstract

The phosphatase and tensin homolog (PTEN) hamartoma tumor syndrome (PHTS) is a collection of diseases stemming from mutations in the PTEN tumor suppressor gene and is characterized by variable expressivity and abnormal overgrowth in multiple body systems. Its clinical manifestations include, but are not limited to, lipomas, limb overgrowth, dermatologic lesions, and malignancy. The infrequency of occurrence and broadness of clinical presentation has made the diagnosis and differentiation of different subtypes of PHTS challenging. This case report describes a five-year-old patient with a history of autism and macrocephaly who presented to the emergency department with right lower quadrant (RLQ) pain concerning for appendicitis. A physical exam was significant for right leg hemihypertrophy. Imaging ruled out appendicitis but diagnosed two large right-sided abdominal lipomas. The patient was discharged with the recommendation to pursue genetic testing given the physical exam findings and history. Following confirmation of a PTEN tumor suppressor gene mutation, the patient continued to have increased frequency of abdominal pain, developed vision changes, and was diagnosed with a benign follicular thyroid nodule. Hemihypertrophy, recurrent unilateral lipomas, and a confirmed PTEN mutation are consistent with a diagnosis of Proteus-like syndrome, a rare subtype of PHTS.

摘要

磷酸酶与张力蛋白同源物(PTEN)错构瘤肿瘤综合征(PHTS)是一组源于PTEN肿瘤抑制基因突变的疾病,其特征为多系统的可变表达性和异常过度生长。其临床表现包括但不限于脂肪瘤、肢体过度生长、皮肤病变和恶性肿瘤。PHTS不同亚型的发生率低且临床表现广泛,使得其诊断和鉴别具有挑战性。本病例报告描述了一名5岁自闭症和巨头畸形患儿,因右下腹痛疑似阑尾炎就诊于急诊科。体格检查发现右腿半侧肥大。影像学检查排除了阑尾炎,但诊断出右侧腹部有两个大脂肪瘤。鉴于体格检查结果和病史,患者出院时被建议进行基因检测。在确认PTEN肿瘤抑制基因突变后,患者腹痛频率持续增加,出现视力变化,并被诊断出患有良性滤泡性甲状腺结节。半侧肥大、复发性单侧脂肪瘤和确诊的PTEN突变符合变形杆菌样综合征的诊断,这是PHTS的一种罕见亚型。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/4b0a/9107286/942956a02444/cureus-0014-00000024135-i01.jpg

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