类似胆总管囊肿的胆总管横纹肌肉瘤：阻塞性黄疸的罕见原因

Nazmul Hassan; Rafiqul Islam; Kaniz Fathema; Maimuna Sayeed; Md. Wahiduzzaman Mazumder; ASM Bazlul Karim; Bishnu Pada Dey

doi:10.7499/j.issn.1008-8830.2007118

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中国当代儿科杂志 ›› 2020, Vol. 22 ›› Issue (12) : 1338-1343. DOI: 10.7499/j.issn.1008-8830.2007118

病例报告

类似胆总管囊肿的胆总管横纹肌肉瘤：阻塞性黄疸的罕见原因

Nazmul Hassan¹, Rafiqul Islam¹, Kaniz Fathema¹, Maimuna Sayeed¹, Md. Wahiduzzaman Mazumder¹, ASM Bazlul Karim¹, Bishnu Pada Dey²

作者信息 +

Rhabdomyosarcoma of the common bile duct mimicking choledochal cyst: a rare cause of obstructive jaundice

Nazmul Hassan¹, Rafiqul Islam¹, Kaniz Fathema¹, Maimuna Sayeed¹, Md. Wahiduzzaman Mazumder¹, ASM Bazlul Karim¹, Bishnu Pada Dey²

Author information +

文章历史 +

摘要

肝外胆道肿瘤罕见，其中以横纹肌肉瘤最常见。横纹肌肉瘤是一种软组织恶性肌肉骨骼肿瘤，是儿童非常罕见的一种胆总管恶性肿瘤。它通常表现为阻塞性黄疸和/或瘙痒。如果肿瘤没有局部浸润邻近组织，其影像学表现则类似胆总管囊肿。因此，确诊通常是在手术时或术前通过活检。早期诊断、与胆总管囊肿鉴别、尽早开始治疗对延长患者生存时间有重要意义。该文报道1例10岁男童，反复阻塞性黄疸和发热，术前诊断为胆总管囊肿，术后确诊为胆总管胚胎性横纹肌肉瘤。经手术切除、术后化疗，该患儿预后良好。故必须了解这种罕见的肿瘤可以类似先天性胆总管囊肿，并且在儿童阻塞性黄疸的鉴别诊断时应加以考虑。

Abstract

Extrahepatic biliary tract tumors are rare and among them rhabdomyosarcoma is most common. Rhabdomyosarcoma is a soft tissue malignant musculoskeletal tumor and is a very rare malignancy of the common bile duct in children. It usually presents as obstructive jaundice and/or pruritus. If there is no local invasion to the adjacent tissues, the radiological appearance of the tumor lesion is like a choledochal cyst. So the diagnosis is usually made at surgery or by preoperative biopsy. It is important to diagnose early and differentiate it from choledochal cyst and start treatment as early as possible for long time survival of the patient. This case report presented a case of a 10-year-old boy with recurrent onset of obstructive jaundice and fever preoperatively who was diagnosed as choledochal cyst and postoperatively as embryonal rhabdomyosarcoma of the common bile duct. After surgical resection and postoperative chemotherapy, the child had a good prognosis. So it is crucial to know that this rare tumor can mimic congenital choledochal cyst and it should be considered in the differential diagnosis of obstructive jaundice in children.

导出引用

Nazmul Hassan, Rafiqul Islam, Kaniz Fathema, Maimuna Sayeed, Md. Wahiduzzaman Mazumder, ASM Bazlul Karim, Bishnu Pada Dey. 类似胆总管囊肿的胆总管横纹肌肉瘤：阻塞性黄疸的罕见原因[J]. 中国当代儿科杂志. 2020, 22(12): 1338-1343 https://doi.org/10.7499/j.issn.1008-8830.2007118

Nazmul Hassan, Rafiqul Islam, Kaniz Fathema, Maimuna Sayeed, Md. Wahiduzzaman Mazumder, ASM Bazlul Karim, Bishnu Pada Dey. Rhabdomyosarcoma of the common bile duct mimicking choledochal cyst: a rare cause of obstructive jaundice[J]. Chinese Journal of Contemporary Pediatrics. 2020, 22(12): 1338-1343 https://doi.org/10.7499/j.issn.1008-8830.2007118

参考文献

[1] Ali S, Russo MA, Margraf L. Biliary rhabdomyoscarcoma mimicking choledochal cyst[J]. J Gastrointestin Liver Dis, 2009, 18(1):95-97.
[2] Raina S, Kumar N, Jad B, et al. Botryoid rhabdomyosarcoma of biliary tree:a diagnostic puzzle[J]. IOSR-JDMS, 2015, 14(8):94-96.
[3] Zampieri N, Camoglio F, Corroppolo M, et al. Botryoid rhabdomyosarcoma of the biliary tract in children:a unique case report[J]. Eur J Cancer Care (Engl), 2006, 15(5):463-466.
[4] Enzinger FM, Weiss SW. Rhabdomyosarcoma[M]//Enzinger FM, Weiss SW. Soft tissue tumours. 4th ed. St Louis, Missouri:Mosby, 2001:790.
[5] Wilks S, Moxon W. Lectures on Pathological Anatomy[M]. 2nd ed. Philadelphia:Lindsay and Blakiston, 1875:465-466.
[6] Kinariwala DJ, Wang AY, Melmer PD, et al. Embryonal rhabdomyosarcoma of the biliary tree:a rare cause of obstructive jaundice in children which can mimic choledochal cysts[J]. Indian J Radiol Imaging, 2017, 27(3):306-309.
[7] Tireli GA, Sander S, Dervisoglu S, et al. Embryonal rhabdomyosarcoma of the common bile duct mimicking choledochal cyst[J]. J Hepatobiliary Pancreat Surg, 2005, 12(3):263-265.
[8] Lad SK, Balasubramanian M, Kavishwar V, et al. Botryoid rhabdomyosarcoma of the common bile duct[J]. Bombay Hosp J, 2010, 52:108-110.
[9] Ruymann FB, Takeuchi A, Boyce HW. Idiopathic, recurrent cholestasis[J]. Pediatrics, 1970, 45(5):812-820.
[10] Levy CF, Wexler LH. Rhabdomyosarcoma and Other Soft-Tissue Sarcomas[M]//Lanzkowsky P, Lipton JM, Fish JD. Lanzkowsky's Manual of Pediatric Hematology and Oncology. 6th ed. USA:Academic Press, 2016:505-523.
[11] Parham DM. Pathologic classification of rhabdomyosarcomas and correlations with molecular studies[J]. Mod Pathol, 2001, 14(5):506-514.
[12] Baker KS, Anderson JR, Link MP, et al. Benefit of intensified therapy for patients with local or regional embryonal rhabdomyosarcoma:results from the Intergroup Rhabdomyosarcoma Study IV[J]. J Clin Oncol, 2000, 18(12):2427-2434.
[13] Crist WM, Anderson JR, Meza JL, et al. Intergroup rhabdomyosarcoma study-IV:results for patients with nonmetastatic disease[J]. J Clin Oncol, 2001, 19(12):3091-3102.
[14] Arnaud O, Boscq M, Asquier E, et al. Embryonal rhabdomyosarcoma of the biliary tree in children:a case report[J]. Pediatr Radiol, 1987, 17(3):250-251.
[15] Mathew M, Narula MK, Chadha R. Case report:sarcoma botryoides of the common bile duct[J]. Indian J Radiol Imaging, 2001, 11:101-102.
[16] Sanz N, de Mingo L, Florez F, et al. Rhabdomyosarcoma of the biliary tree[J]. Pediatr Surg Int, 1997, 12(2-3):200-201.
[17] Nemade B, Talapatra K, Shet T, et al. Embryonal rhabdomyosarcoma of the biliary tree mimicking a choledochal cyst[J]. J Cancer Res Ther, 2007, 3(1):40-42.
[18] Spunt SL, Lobe TE, Pappo AS, et al. Aggressive surgery is unwarranted for biliary tract rhabdomyosarcoma[J]. J Pediatr Surg, 2000, 35(2):309-316.
[19] Kumar V, Chaudhary S, Kumar M, et al. Rhabdomyosarcoma of biliary tract-a diagnostic dilemma[J]. Indian J Surg Oncol, 2012, 3(4):314-316.
[20] Sharma AK, Lahoti BK, Aggarwal R, et al. Obstructive jaundice in children may be due to a malignant tumour of the common bile duct[J]. Pediatr Surg Int, 1995, 10(7):496-497.
[21] Majmudar B, Kumar VS. Embryonal rhabdomyosarcoma (sarcoma botryoides) of the common bile duct:a case report[J]. Hum Pathol, 1976, 7(6):705-708.
[22] Davis GL, Kissane JM, Ishak KG. Embryonal rhabdomy-osarcoma (sarcoma botryoides) of the biliary tree. Report of five cases and a review of the literature[J]. Cancer, 1969, 24(2):333-342.
[23] Verstandig A, Bar-Ziv J, Abu-Dalu KI, et al. Sarcoma botryoides of the common bile duct:preoperative diagnosis by coronal CT and PTC[J]. Pediatr Radiol, 1991, 21(2):152-153.
[24] Patil KK, Omojola MF, Khurana P, et al. Embryonal rhabdomyosarcoma within a choledochal cyst[J]. Can Assoc Radiol J, 1992, 43(2):145-148.
[25] Mathew D, de Lima H, Mahomed N. Embryonal rhabdomyosarcoma of the biliary tree in a paediatric patient-a rare cause of obstructive jaundice[J]. SA J Radiol, 2019, 23(1):1662.
[26] Morotti RA, Nicol KK, Parham DM, et al. An immuno-histochemical algorithm to facilitate diagnosis and subtyping of rhabdomyosarcoma:the Children's Oncology Group experience[J]. Am J Surg Pathol, 2006, 30(8):962-968.
[27] Breneman JC, Lyden E, Pappo AS, et al. Prognostic factors and clinical outcomes in children and adolescents with metastatic rhabdomyosarcoma-a report from the Intergroup Rhabdomyosarcoma Study IV[J]. J Clin Oncol, 2003, 21(1):78-84.