A case of an elderly female with diffuse hepatic hemangiomatosis complicated with multiple organic dysfunction and Kasabach–Merritt syndrome

Shimizu, Yasuhiko; Komura, Takuya; Seike, Takuya; Omura, Hitoshi; Kumai, Tatsuo; Kagaya, Takashi; Ohta, Hiroyuki; Kawashima, Atsuhiro; Harada, Kenichi; Kaneko, Shuichi; Unoura, Masashi

doi:10.1007/s12328-018-0871-3

Cited by 11 publications

(14 citation statements)

References 19 publications

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“…Histologically, Kaposi’s hemangioendothelioma has the characteristics of both hemangioma and Kaposi sarcoma and is accompanied by Kasabach–Merritt syndrome [ 8 ]. MRI T1 image shows low or iso-signal lump-like soft tissue with unclear boundaries.…”

Section: Discussionmentioning

confidence: 99%

Pancreaticoduodenal and choledochal hemangiomatosis with vascular variation in a child: a rare disease with challenge starts from diagnosis—a case report

2022

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Background Visceral hemangiomatosis is a benign tumor (rarer than hemangioma) that has not been reported to occur in the pancreas, duodenum, or choledoch. It can be easily confused with other pancreatic tumors or choledocholithiasis. Herein, we describe a case of a child with pancreaticoduodenal and choledochal hemangiomatosis and the key characteristics for the accurate diagnosis of pancreatic tumors based on previous reports and our findings. Case presentation We report a case of a 2-year and 9-month-old child who presented with repeated and fluctuating jaundice for 3 months with a history of endoscopic stone removal in a local hospital, following the diagnosis of choledocholithiasis. An abdominal computed tomography revealed a previously undiagnosed pancreatic head tumor and celio-mesenteric trunk (a rare vascular variation). This was misdiagnosed as a pancreatic neuroendocrine tumor. Since the patient’s parents refused FNA biopsy and insisted on surgery, pancreaticoduodenectomy was performed; however, postoperatively, the child was correctly diagnosed with pancreaticoduodenal and choledochal hemangiomatosis. Although the patient was in good condition and had gained 4 kg in weight 3 months postoperatively, pancreaticoduodenectomy could have been avoided if an accurate diagnosis had been established before or during the operation. Conclusion Our report highlights the difficulty in diagnosing visceral hemangiomatosis. Radiologists, endoscopists, and surgeons should consider this possibility in cases of repeated and fluctuating jaundice that cannot be explained by choledocholithiasis alone.

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Section: Discussionmentioning

confidence: 99%

Pancreaticoduodenal and choledochal hemangiomatosis with vascular variation in a child: a rare disease with challenge starts from diagnosis—a case report

2022

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“…Though the duration of follow-up in reported cases varies, the prognosis of DHH treated with resection is good with no recurrence in five cases and improvement in all symptomatic cases. In unresectable cases, anti-VFGF agents, radiation therapy, and arterial embolization have been reported to improve symptoms, but it is difficult to control the disease in a rapidly progressing case [16][17][18][19]. Although CH is a benign tumor, hemangiomatosis is severe as the CH grows, and KMS may also occur [18].…”

Section: Discussionmentioning

confidence: 99%

“…In unresectable cases, anti-VFGF agents, radiation therapy, and arterial embolization have been reported to improve symptoms, but it is difficult to control the disease in a rapidly progressing case [ 16 – 19 ]. Although CH is a benign tumor, hemangiomatosis is severe as the CH grows, and KMS may also occur [ 18 ]. Extensive DHH is difficult to resect considering remnant liver volume and no residual lesions, so the timing of resection should be carefully considered.…”

Section: Discussionmentioning

confidence: 99%

A rare case of cavernous hemangioma accompanied with diffuse hepatic hemangiomatosis

Ota¹,

Kamiyama

Kato³

et al. 2020

surg case rep

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Background Hepatic cavernous hemangioma (CH) is the most common hepatic benign tumor. Most cases are solitary, asymptomatic, and found incidentally. In symptomatic cases with rapidly growing tumors and coagulopathy, surgical treatment is considered. In rare cases, diffuse hepatic hemangiomatosis (DHH) is reported as a comorbidity. The etiology of DHH is unknown. Case presentation A 29-year-old female patient had a history of endometriosis treated with oral contraceptives. Hepatic CH was incidentally detected in the segment IVa of the liver according to the Couinaud classification. Follow-up computed tomography (CT) and ultrasound sonography showed the growth of the lesion and formation of multiple new lesions near the first. Enhanced CT and magnetic resonance imaging (MRI) revealed that the new lesions were different from CH. Although oral contraceptives were stopped, all lesions grew in size. Malignancy and possibility of rupture of these tumors were considered due to the clinical course, and we opted for surgical removal of the tumors. Left liver lobectomy and cholecystectomy were performed. Surgical findings were small red spot spreading and a mass in segment IV of the liver. Pathological examination revealed a circumscribed sponge-like tumor with diffuse irregular extension to the adjacent area. Both of the lesions consisted of blood-filled dilated vascular spaces lined by flat endothelium without atypia. The diagnosis was hepatic CH with DHH. The patient was discharged on postoperative day 12 uneventfully. Conclusion We report the successful resection of CH with DHH. The case findings suggest a relationship between oral contraceptive use and enlargement of CH and DHH. Although DHH has been poorly understood, a few previously published cases reported DHH occurrence in patients using oral contraceptives. In such cases, the decision to perform surgical resection should be made after careful examination.

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“…Although a huge cystic lesion was located in the abdominal cavity, the platelet count and coagulation function tests were within the reference ranges. Thus, Kasabach–Merritt syndrome was excluded[10]. We also excluded Maffucci's syndrome because no deformity or activity limitation of the extremities was found[11].…”

Section: Discussionmentioning

confidence: 99%

Peritoneal cavernous hemangiomatosis: A case report

Chen

Diao

et al. 2019

WJCC

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BACKGROUND Cavernous hemangiomatosis in the liver and spleen has been reported, but it occurs less commonly in the peritoneum. Here we report a case of peritoneal cavernous hemangiomatosis and share some valuable information about this disease. CASE SUMMARY A 57-year-old Chinese man had a huge abdominal mass with abdominal distention and a significant reduction of food consumption. An enhanced abdominal and pelvic computed tomography and positron emission tomography–computed tomography revealed multiple cystic masses on the peritoneum, greater omentum, small intestinal mesentery and the surface of the spleen, and a high maximum standardized uptake value of the largest cystic lesion. Exploratory laparotomy was performed, and multiple cystic masses were found on the surface of the peritoneum, greater omentum, mesentery of the small intestine, and surface of the liver and spleen. Dark red bloody cystic fluid was present in the cystic tumor. Pathological examination showed that in the stromal components, the irregular vascular wall was thin. The vessel lumen was interlinked, and the lumen was lined with flat endothelium. According to the intraoperative findings and pathologic results, the patient was diagnosed with peritoneal cavernous hemangiomatosis. CONCLUSION The possibility of peritoneal cavernous hemangiomatosis should be considered when multiple cystic masses are found in the abdominal cavity by preoperative examination.

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A case of an elderly female with diffuse hepatic hemangiomatosis complicated with multiple organic dysfunction and Kasabach–Merritt syndrome

Cited by 11 publications

References 19 publications

Pancreaticoduodenal and choledochal hemangiomatosis with vascular variation in a child: a rare disease with challenge starts from diagnosis—a case report

Pancreaticoduodenal and choledochal hemangiomatosis with vascular variation in a child: a rare disease with challenge starts from diagnosis—a case report

A rare case of cavernous hemangioma accompanied with diffuse hepatic hemangiomatosis

Peritoneal cavernous hemangiomatosis: A case report

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