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伴有胆囊结石的大型透明血管型盆腔Castleman病的完整切除及治疗:1例罕见病例报告

Complete excision and management of large hyaline-vascular pelvic Castleman's disease with coexisting cholecystolithiasis: A rare case report.

作者信息

Jeo Wifanto Saditya, Haryono Samuel, Gultom Fajar Lamhot, Chaspuri Dismas Adiasa, Nikijuluw Rachmat Christian, Yoladifa Safira Farah

机构信息

Division of Digestive Surgery, Department of Surgery, Dr. Cipto Mangunkusumo General Hospital, Faculty of Medicine, Universitas Indonesia, Jakarta, Indonesia.

Department of Oncology Surgery, Dharmais Cancer Hospital, Jakarta, Indonesia.

出版信息

Int J Surg Case Rep. 2025 Mar;128:111019. doi: 10.1016/j.ijscr.2025.111019. Epub 2025 Feb 7.

Abstract

INTRODUCTION

Castleman's disease (CD) refers to scarce lymphovascular hyperplasia categorized as unicentric (UCD) or multicentric (MCD). Retroperitoneal UCD is uncommon (6.7 %) and frequently grows undetected, making diagnosis challenging. The hypervascular nature of hyaline-vascular variant and the average larger size of UCD may result in complete resection difficulty. We present a case of large pelvic UCD completely resected without previous prophylactic procedures or massive intraoperative bleeding with coexisting cholecystolithiasis, which follows the SCARE 2023 guidelines.

CASE PRESENTATION

A 45-year-old male was accidentally observed with pelvic mass suppressing ureter and right internal iliac artery in preoperative MRI for cholecystolithiasis and cholecystitis. Epigastric pain, vomiting, and jaundice due to the inflamed gallbladder had resolved before mass removal. Biopsy and immunohistochemistry (IHC) tests confirmed the hyaline-vascular CD. Complete resection per laparotomy was done due to lesion adherence and identified a 7x5x2.5 cm solid mass with cystic degeneration suppressing the ureter. After five months, the patient fully recovered with no symptoms.

DISCUSSION

Asymptomatic mass presented as lymphadenopathy on imaging should suggest CD. A prompt lymph node biopsy followed by a pre- or postoperative IHC test is important in diagnosing CD. Total pelvic mass excision with rectum preservation is feasible for larger (>5 cm) hypervascularized masses without remarkable bleeding and promotes recovery without adjunctive treatments.

CONCLUSION

Complete excision per laparotomy is accessible for the uncommon large pelvic retroperitoneal UCD-suppressing ureter.

摘要

引言

Castleman病(CD)是指一种罕见的淋巴血管增生性疾病,分为单中心型(UCD)和多中心型(MCD)。腹膜后UCD较为罕见(6.7%),且常常在未被察觉的情况下生长,这使得诊断颇具挑战性。透明血管型的血管丰富特性以及UCD通常较大的尺寸可能导致完全切除困难。我们报告一例巨大盆腔UCD病例,该病例在未进行先前预防性手术或术中大量出血的情况下被完全切除,同时并存胆囊结石,本病例报告遵循2023年SCARE指南。

病例介绍

一名45岁男性在因胆囊结石和胆囊炎进行术前MRI检查时意外发现盆腔肿块,该肿块压迫输尿管和右髂内动脉。因胆囊炎症引起的上腹部疼痛、呕吐和黄疸在肿块切除前已缓解。活检和免疫组织化学(IHC)检查证实为透明血管型CD。由于病变粘连,通过剖腹手术进行了完全切除,发现一个7×5×2.5厘米的实性肿块,伴有囊性变,压迫输尿管。五个月后,患者完全康复,无任何症状。

讨论

影像学上表现为淋巴结病的无症状肿块应怀疑为CD。及时进行淋巴结活检,随后进行术前或术后IHC检查对CD的诊断很重要。对于较大(>5厘米)的血管丰富肿块,保留直肠进行全盆腔肿块切除可行,且无明显出血,并在无需辅助治疗的情况下促进恢复。

结论

对于罕见的压迫输尿管的巨大盆腔腹膜后UCD,通过剖腹手术可实现完全切除。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5b51/11869881/164a4979082f/gr1.jpg

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