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斯皮格尔隐睾综合征:一种罕见临床病症的文献综述与病例报告

Spigelian-Cryptorchidism Syndrome: A Literature Review and Case Report of a Rare Clinical Entity.

作者信息

Kalantar Hamdah T, Naji Hussein, Naji Abdulla

机构信息

Medicine and Surgery, Mohammed Bin Rashid University of Medicine and Health Sciences (MBRU), Dubai, ARE.

Pediatric Surgery, Mediclinic Parkview Hospital, Dubai, ARE.

出版信息

Cureus. 2025 Jul 5;17(7):e87324. doi: 10.7759/cureus.87324. eCollection 2025 Jul.

DOI:10.7759/cureus.87324
PMID:40765591
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC12322183/
Abstract

Spigelian-cryptorchidism syndrome is a rare and diagnostically challenging condition in neonatology that is characterized by the association of an undescended testis with a Spigelian hernia, the latter usually without radiologic evidence. We report the case of a full-term male neonate delivered at 39 weeks and two days of gestation by kiwi-assisted vaginal delivery after a spell of fetal bradycardia. Antenatal imaging had shown persistent bilateral pelviectasis of the kidneys and an undescended right testis. Postnatally, the initial course was normal; however, clinical evaluation showed partially reducible sausage-shaped swelling in the right lower quadrant and an impalpable right testis. Ultrasonographic examination confirmed intra-abdominal location of the right testis and showed bilateral mild dilatation of the renal pelvis but no abdominal wall defect or hernia sac. Surgical assessment revealed a partially reducible mass, consistent with Spigelian hernia, and hence established the setting of Spigelian-cryptorchidism syndrome. The testis's anatomical separation from the inguinal canal and the lack of a discernible processus vaginalis favored a syndromic diagnosis. The case brings to light the inherent limitations of imaging in the detection of neonatal Spigelian hernia, the value of physical examination by skilled clinicians, and the imperative of a multidisciplinary diagnostic and therapeutic approach. Conservative monitoring was chosen, with surgical repair scheduled at 12 months of age, consistent with best practices at present. This case supports that in neonatal presentations with radiological uncertainty combined with strong clinical evidence, the diagnosis can be pursued critically and not delayed based on imaging. It also stresses the requirement to consider unexplained abdominal distension and non-palpable testes on a differential which includes this uncommon but clinically important syndrome.

摘要

半月线隐睾综合征是新生儿科一种罕见且诊断具有挑战性的病症,其特征为隐睾与半月线疝并存,后者通常无放射学证据。我们报告一例足月男婴病例,该婴儿在妊娠39周零2天时因胎儿心动过缓经猕猴桃辅助阴道分娩出生。产前影像学检查显示双侧肾脏持续肾盂扩张以及右侧睾丸未降。出生后,初期病程正常;然而,临床评估发现右下腹有部分可还纳的腊肠样肿胀,右侧睾丸触诊不清。超声检查证实右侧睾丸位于腹腔内,显示双侧肾盂轻度扩张,但未发现腹壁缺损或疝囊。手术评估发现一个部分可还纳的肿块,符合半月线疝,从而确诊为半月线隐睾综合征。睾丸与腹股沟管在解剖学上分离且未发现明显的鞘突,这支持了综合征的诊断。该病例揭示了新生儿半月线疝检测中影像学的固有局限性、熟练临床医生体格检查的价值以及多学科诊断和治疗方法的必要性。目前选择保守监测,计划在12个月大时进行手术修复,这与最佳实践相符。该病例支持,在新生儿表现中,若影像学存在不确定性但临床证据充分,可进行批判性诊断,而非基于影像学检查而延迟诊断。它还强调,在鉴别诊断中需考虑不明原因的腹胀和触诊不清的睾丸,其中应包括这种罕见但临床重要的综合征。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/650a/12322183/3ae17559430f/cureus-0017-00000087324-i03.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/650a/12322183/e43129d9b474/cureus-0017-00000087324-i01.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/650a/12322183/4acb2ad7fa7c/cureus-0017-00000087324-i02.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/650a/12322183/3ae17559430f/cureus-0017-00000087324-i03.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/650a/12322183/e43129d9b474/cureus-0017-00000087324-i01.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/650a/12322183/4acb2ad7fa7c/cureus-0017-00000087324-i02.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/650a/12322183/3ae17559430f/cureus-0017-00000087324-i03.jpg

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BMJ Case Rep. 2025 Jan 4;18(1):e261858. doi: 10.1136/bcr-2024-261858.
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Is there a link between Spigelian and inguinal hernias? A case series.Spigelian 疝与腹股沟疝之间是否存在关联?一项病例系列研究。
Hernia. 2024 Oct;28(5):1737-1746. doi: 10.1007/s10029-024-03061-5. Epub 2024 Jun 27.
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Association between general anesthesia in early childhood and neurodevelopment up to 4 years of age: the Japan Environment and Children's Study.
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J Anesth. 2024 Oct;38(5):609-621. doi: 10.1007/s00540-024-03359-9. Epub 2024 Jun 7.
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Spigelian-cryptorchidism syndrome: Lesson based on a case report.半月线隐睾综合征:基于一例病例报告的经验教训
Radiol Case Rep. 2024 May 23;19(8):3372-3375. doi: 10.1016/j.radcr.2024.04.080. eCollection 2024 Aug.
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