双胎妊娠的Pena-Shokeir综合征:一例罕见病例报告。

IF 0.8 Q3 MEDICINE, GENERAL & INTERNAL
Clinical Medicine Insights. Case Reports Pub Date : 2025-03-24 eCollection Date: 2025-01-01 DOI:10.1177/11795476251329986
Souhaila El Gazzane, Salahiddine Saghir, Mehdi Bahous, Mohamed Sellouti, Anass Ayad, Rachid Abilkassem
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引用次数: 0

摘要

Pena-Shokeir综合征(PSS)是一种罕见的常染色体隐性遗传病,其特征为畸形、喜足畸形、关节挛缩、宫内生长受限、羊水过多和肺发育不全。这种综合征有两种类型,根据不同的临床和遗传特征加以区分。PSS是一种潜在的危及生命的疾病,大多数病例预计将通过产前超声诊断。遗传咨询是至关重要的,告知家长复发的风险和管理策略,为未来的怀孕。我们报告一个双绒毛膜双羊膜(DCDA)双胎妊娠的PSS病例。尽管产前超声检查正常,但1名双胞胎在出生后被诊断出严重的颅面异常、肢体畸形和肺部并发症,与PSS一致。相比之下,第二个双胞胎表现出正常的生长发育,没有发现异常。据我们所知,这是第三例报道的双胎妊娠PSS病例,第二例涉及正常双胎。本病例旨在通过详细介绍与PSS相关的独特畸形和临床表现,并强调双胎妊娠的诊断挑战,为现有文献做出贡献。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
The Pena-Shokeir Syndrome in a Twin Pregnancy: A Rare Case Report.

Pena-Shokeir Syndrome (PSS) is a rare autosomal recessive disorder characterized by dysmorphic features, camptodactyly, arthrogryposis, intrauterine growth restriction, polyhydramnios, and pulmonary hypoplasia. Two types of this syndrome have been defined, differentiated by distinct clinical and genetic features. PSS is a potentially life-threatening condition, with most cases expected to be diagnosed prenatally via ultrasound. Genetic counseling is crucial to inform parents about recurrence risks and management strategies for future pregnancies. We report a case of PSS in a dichorionic diamniotic (DCDA) twin pregnancy. Despite normal prenatal ultrasounds, 1 twin was diagnosed postnatally with severe craniofacial anomalies, limb deformities, and pulmonary complications, consistent with PSS. In contrast, the second twin exhibited normal growth and development, with no anomalies identified. To the best of our knowledge, this is the third reported case of PSS in a twin pregnancy and the second involving a normal co-twin. This case aims to contribute to the existing literature by detailing the unique dysmorphic and clinical findings associated with PSS and emphasizing the diagnostic challenges in twin pregnancies.

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来源期刊
Clinical Medicine Insights. Case Reports
Clinical Medicine Insights. Case Reports MEDICINE, GENERAL & INTERNAL-
CiteScore
1.10
自引率
0.00%
发文量
57
审稿时长
8 weeks
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