Alkhrait Samar, Ali Munira, Kertowidjojo Elizabeth, Romero Iris L, Hathaway Feighanne, Madueke-Laveaux Obianuju Sandra
Department of OBGYN, University of Chicago Medicine, Chicago, IL 60637, USA.
College of Medicine, University of Illinois, Chicago, IL 60607, USA.
J Clin Med. 2023 Aug 22;12(17):5436. doi: 10.3390/jcm12175436.
Uterine leiomyomas or uterine fibroids are the most common benign soft tissue tumor in reproductive-aged women. Fumarate hydratase deficient (FH-d) uterine fibroids are a rare subtype that is diagnosed only on pathologic evaluation. FH-d uterine fibroids may be the first indicator of hereditary leiomyomatosis and renal cell cancer (HLRCC) syndrome. Therefore, identifying and understanding the clinical implication and diagnosis of FH-d uterine fibroids is critical for early diagnosis of HLRCC. This case series investigates the uncommon yet significant condition of FH-d uterine fibroids. We examined the clinical manifestation, diagnostic imaging, and histopathological characteristics of FH-d uterine fibroids in five cases identified at our institution over the last ten years. All diagnoses were confirmed by pathologic evaluation after surgical treatment. Gynecologists and pathologists play a critical role in the early diagnosis of FH-d uterine fibroids and must recognize the relevant clinical and pathologic findings that raise suspicion about this diagnosis. The detection of these cases is largely dependent on the pathologist's ability to recognize unique histopathologic features. Once these characteristics are identified, it should prompt a referral to a gynecologist to consider conducting germline genetic testing. The management of FH-d uterine fibroids necessitates a multidisciplinary approach, including proper genetic screening and regular surveillance, especially for renal tumors.
子宫平滑肌瘤或子宫肌瘤是育龄期女性最常见的良性软组织肿瘤。富马酸水合酶缺陷(FH-d)型子宫肌瘤是一种罕见的亚型,仅通过病理评估才能诊断。FH-d型子宫肌瘤可能是遗传性平滑肌瘤病和肾细胞癌(HLRCC)综合征的首个指标。因此,识别和了解FH-d型子宫肌瘤的临床意义及诊断对于HLRCC的早期诊断至关重要。本病例系列研究了FH-d型子宫肌瘤这种不常见但意义重大的病症。我们检查了过去十年在我们机构确诊的5例FH-d型子宫肌瘤的临床表现、诊断性影像学检查及组织病理学特征。所有诊断均在手术治疗后经病理评估得以证实。妇科医生和病理学家在FH-d型子宫肌瘤的早期诊断中起着关键作用,必须认识到那些引起对该诊断怀疑的相关临床和病理表现。这些病例的发现很大程度上取决于病理学家识别独特组织病理学特征的能力。一旦识别出这些特征,就应促使转诊至妇科医生处,考虑进行种系基因检测。FH-d型子宫肌瘤的管理需要多学科方法,包括适当的基因筛查和定期监测,尤其是针对肾肿瘤。