Puiseux Chloé, Bretonnier Maxime, Proisy Maia, Chappé Céline, Denizeau Philippe, Riffaud Laurent
Department of Pediatric Oncology, Rennes University Hospital, Rennes, France.
Department of Pediatric Neurosurgery, Rennes University Hospital, Rennes, France.
Childs Nerv Syst. 2021 Mar;37(3):1017-1020. doi: 10.1007/s00381-020-04785-2. Epub 2020 Jul 3.
Dysplastic gangliocytoma of the cerebellum (DGC), also called Lhermitte-Duclos disease, is a rare lesion of the posterior fossa consisting of a diffuse hypertrophy of the cerebellar cortex. DGC frequently presents in young adults and rarely in childhood. Only 3 cases have been previously described in newborns. We present an uncommon case of DGC which was diagnosed in utero. The radiological presentation prenatally and at birth was similar to a heterotopic neuroglial brain tissue. MRI aspects evolved from T1/T2 isointense signals to hypoT1 and hyperT2 signals at the age of 1 year. The girl was then operated on total removal of the lesion which was performed with no postoperative complication. Genetics did not demonstrate any germline PTEN mutation or family history suggesting Cowden disease. Two years later, the child was doing well and MRI confirmed complete resection. This case illustrates the difficulties of diagnosing intracranial lesions in foetuses and newborns. Physicians caring for pregnant women and pediatrics should be aware that neoplasm-like lesions such as DGC may present as hamartomas. Surgical resection could then be discussed whenever possible.
小脑发育异常性神经节细胞瘤(DGC),也称为Lhermitte-Duclos病,是一种罕见的后颅窝病变,由小脑皮质弥漫性肥大组成。DGC常见于年轻人,儿童罕见。此前仅有3例新生儿病例的报道。我们报告1例罕见的在子宫内诊断出的DGC病例。产前及出生时的影像学表现类似于异位神经胶质脑组织。1岁时,MRI表现从T1/T2等信号演变为T1低信号和T2高信号。随后该女孩接受了病变全切手术,术后无并发症。遗传学检查未发现任何种系PTEN突变或提示考登病的家族史。两年后,患儿情况良好,MRI证实病变已完全切除。该病例说明了胎儿和新生儿颅内病变诊断的困难。照顾孕妇和儿科患者的医生应意识到,像DGC这样的肿瘤样病变可能表现为错构瘤。只要有可能,就可以考虑手术切除。