Castleman's disease in two patients with spondyloarthritis treated by Tumor Necrosis Factor-alpha antagonists

Castleman's disease is a rare lymphoproliferative syndrome involving several anatomical and clinical entities. We report two cases of Castleman's disease in patients with spondyloarthritis (with and without psoriasis) treated with Tumor Necrosis Factor-alpha blockers. During follow-up, imaging examinations, carried out for disease assessment, fortuitously revealed a pelvic mass close to the psoas or an atypical cervical adenopathy. Both lesions were in moderate hypermetabolism on Positron emission tomography–computed tomography. After surgical excision and histological analysis, Castleman's disease was diagnosed without signs of malignancy. To the best of our knowledge, these are the first cases of Castleman's disease diagnosed in a context of spondyloarthritis treated with Tumor Necrosis Factor-alpha blockers. The relationship between these two pathologies and the link with Tumor Necrosis Factor-alpha blockers agents has not been explored in the literature until now.     

Angiofollicular lymph node hyperplasia resembling a spinal nerve sheath tumor: a rare case of Castleman's disease

Background contextAngiofollicular lymph node hyperplasia (Castleman's disease) is a lymphoproliferative disorder of unknown etiology. Although uncommon, the localized form of this disease can manifest in the central nervous system, typically as a meningeal-based intracranial lesion. Castleman's disease involving the spine is exceedingly rare. This represents only the second reported case of a patient with Castleman's disease whose presentation mimicked that of a spinal nerve sheath tumor.PurposeWe report a rare case of angiofollicular lymph node hyperplasia that mimicked a spinal nerve sheath tumor and was treated with gross total resection.Study designCase report.Patient sampleA 31-year-old female with angiofollicular lymph node hyperplasia presenting with a paraspinal mass.Outcome measuresThe patient's outcome was based on clinical history, physical and radiographic examinations.MethodsA 31-year-old woman with a mediastinal mass previously diagnosed as sarcoid via biopsy presented with the new onset of radicular pain and radiographic enlargement of her mass. Magnetic resonance imaging demonstrated a 4.3×5.7×6.0 cm homogenously enhancing soft-tissue lesion that emanated from and widened the T3/T4 neural foramen on the left. The differential diagnosis based on the location of the lesion and imaging characteristics included schwannoma, neurofibroma, paraganglioma, sarcoid, and lymphoma. Gross total resection was performed via thoracotomy. Histological examination identified angiofollicular lymph node hyperplasia (Castleman's disease) of the hyaline-vascular subtype. The patient did not receive adjuvant chemotherapy or radiation therapy.ResultsThe patient had resolution of her symptoms without any clinical or radiographic evidence of recurrence at 1-year follow-up using magnetic resonance imaging with and without contrast.ConclusionsCastleman's disease is a rare pathologic entity that should be considered in the differential diagnosis of a patient with a paraspinal mass. Spine surgeons should be aware of this diagnosis as it has treatment and follow-up implications that differ from the neoplasms it can mimic. Complete surgical excision is an effective treatment for solitary lesions. Screening for concurrent and future malignancies in these patients is prudent.     

Management of benign retroperitoneal schwannomas: a single-center experience

Background

We describe the presentation, diagnostic pathway, and management of 28 patients with benign retroperitoneal schwannomas.

Methods

A prospectively kept soft-tissue tumor database was reviewed to identify patients who presented to the Royal Marsden Hospital with retroperitoneal schwannomas.

Results

From 2001 to 2009, 28 patients presented with retroperitoneal schwannomas. In 13 patients, tumors were identified incidentally, 8 patients presented with pelvic or abdominal symptoms and 7 patients presented with a palpable mass. Seventeen occurred in the pelvis and 11 occurred in the abdominal retroperitoneal space. The median age was 47 years and 21 patients were women; the mean follow-up period was 39 months. Twenty patients underwent resection and 8 patients were managed conservatively with radiologic surveillance. An initial histologic diagnosis was accomplished in 19 of 28 patients. Complete resection was achieved in 17 of 20 patients and 3 patients with pelvic schwannomas underwent a subtotal resection. Resected tumor size ranged from 5 to 23 cm (median, 9 cm), and weighed between 64 and 2,300 g (median, 500 g). There was no surgical mortality. In the 3 subtotal resected tumors, no progression of residual disease or malignant transformation has been noted on follow-up imaging. All 8 patients in the surveillance group had a histologic diagnosis and typical radiologic findings.

Conclusions

An accurate preoperative diagnosis is important because the risks of complicated surgery in the retroperitoneum and pelvis for what is a benign lesion should be considered carefully. Cross-sectional imaging combined with a needle biopsy should in all cases provide the correct diagnosis. Management options include radiologic surveillance in asymptomatic patients or surgical resection in symptomatic patients.     

腹膜后局灶性Castleman病的临床分析并文献复习

目的分析总结腹膜后局灶性Castleman病(localized Castleman’s disease,LCD)的临床特点及诊治方法,以期对该病的诊断和治疗过程提供合理指导意见。方法回顾性分析2014年12月武汉大学人民医院收治的1例腹膜后LCD病人临床资料,并针对性复习相关文献。结果病人术前检查未明确诊断,手术行腹膜后肿瘤切除,术后病理切片检查确诊,未行辅助性治疗。1年后随访复查,病人恢复可,手术效果较好。结论腹膜后LCD术前诊断较困难,手术切除预后良好,术前充分评估可以为治疗方式的选择以及手术方式提供依据,提高手术成功率,减少副损伤及并发症。     

Curative surgery for locally advanced retroperitoneal mature teratoma in an adult. Case report

INTRODUCTIONPrimary teratomas of retroperitoneum are not usual in the adult population. These tumors most commonly seen at the gonadal and sacrococcygeal regions. Herein we describe a case of an 18-year-old female who had a benign cystic teratoma at the retroperitoneum.PRESENTATION OF CASEThe patient underwent an operation at another hospital following a misdiagnosis of hydatid cyst. The patient was referred to our hospital because of the detection of an unresectable tumor during her operation. A computerized tomography (CT)–angiography revealed a cystic mass, with a diameter of 14 cm which was invaded into the retrohepatic suprarenal inferior vena cava and also extended to the posterior aspect of the liver. Additionally the mass invaded the posterior wall of the inferior vena cava and the right renal vein. The tumor was completely resected with a vascular resection. The inferior vena cava was reconstructed with a 12 cm Dacron^® graft and the renal vein was implanted. The patient's postoperative period was uneventful.DISCUSSIONGerm cell tumors of retroperitoneum are usually seen in children, but there are also some reports of adult cases in the literature. Adult cases are especially seen in females. Imaging studies are paramount for diagnosis, preoperative strategy and safe surgical excision. CT scans and MRIs can identify various components of these tumors.CONCLUSIONEven though primary retroperitoneal teratomas are quite rare in adults. Preoperative radiology imaging and strategy is critical for performing a safe surgery. The gold standard treatment strategy for this neoplasm is the surgical resection.     

Severe stricturing Crohn's disease of the duodenum: A case report and review of surgical options

INTRODUCTIONDuodenal Crohn's disease is a rare clinical entity that occurs in 0.5–4.0% of patients with Crohn's disease. A unique case of Crohn's disease of the upper gastrointestinal tract characterized by multiple strictures within the duodenum and jejunum is described in our review.PRESENTATION OF CASEA 41-year-old male presented with a 2-month history of intermittent, crampy abdominal pain accompanied by nausea, bilious emesis, early satiety, anorexia and weight loss. Physical examination revealed fullness in the epigastric region. Imaging demonstrated strictures in the proximal and distal duodenum with dilatation of the intervening segments. There was also gross dilatation of the proximal jejunum, which was followed by a 9 cm strictured segment. There was no evidence of acute Crohn's disease. Although a Whipple's resection was initially considered as a form of operative intervention given the extent of disease within the duodenum, the discovery of unexpected disease intra-operatively presented a surgical dilemma. In this case, strictureplasty, surgical resection and bypass were used to treat the patient.DISCUSSIONDiffuse stricturing of the proximal gastrointestinal tract is a rare manifestation of Crohn's disease. Although imaging can aid in surgical planning, intra-operative decision-making to deal with unexpected findings will remain an important aspect of the management of this entity.CONCLUSIONThe fundamental goal of the surgical management of strictures secondary to Crohn's disease is to relieve obstruction while maximizing bowel conservation. A variety of operative techniques are currently described for the management of duodenal Crohn's disease and are reviewed in this case report.     

Unclear retroperitoneal tumors,an interdisciplinary challenge – A case report and review of the literature

Introduction and importanceUnclear retroperitoneal tumors impose major challenges for clinicians. Tumors can originate primarily from retroperitoneal tissue or secondarily invade into the retroperitoneum. While benign lesions also occur, malignant tumors are far more common. Clinical presentation depends on replacement or invasion of other organs and is therefore highly variable. The heterogeneous tumor composition makes a definitive preoperative diagnosis difficult. Surgical resection is the gold standard for treatment but often proves challenging due to frequent involvement of large retroperitoneal vessels.Case presentationWe present the case of a 70-year old woman diagnosed with a large, unclear retroperitoneal tumor. Initial clinical symptoms were increasing dyspnea and dysphagia in our clinic. Gastroenterologic and cardiologic workup was unremarkable. Computed Tomography (CT) revealed a large retroperitoneal mass in the right upper abdomen with severe displacement of the inferior vena cava and renal veins. The patient was scheduled for primary tumor resection. The procedure was challenging due to the vessel involvement and large blood pressure alterations during tumor mobilization. The post-op pathologic workup then revealed the rare finding of a completely resected paraganglioma. The post-surgical course was uneventful. One year after diagnosis, the patient is relapse-free.Clinical discussionAmong retroperitoneal tumors, paragangliomas and pheochromocytomas are rare tumor entities. Asymptomatic, sporadic disease is hard to identify preoperatively and can cause unexpected complications in the OR. An experienced team is crucial in achieving best short- and long-term outcomes.ConclusionThis case impressively shows the challenges of retroperitoneal tumors and the importance of interdisciplinary work in these cases.     

Massive per rectal bleeding following blunt abdominal trauma: First presentation of Crohn's disease

Introduction

Inflammatory bowel disease is a chronic and relatively common disorder with heterogeneous presentation. Peak incidence occurs in the second and third decades of life. We present a patient with Crohn''s disease whose first presentation was profuse bleeding/rectum following blunt abdominal trauma.

presentation of case

A 29 year old previously healthy man presented one hour after sustaining relatively mild abdominal trauma, due to fall onto the ball during a rugby match. He complained of abdominal pain and one episode of large fresh rectal bleeding. He was pale and distressed with hypotension, tachycardia and abdominal guarding & fresh blood on digital rectal examination. With a provisional diagnosis of intestinal injury he was taken to theatre. Right hemi-colectomy was done for a thickened and inflamed segment of distal ileum, a large adjacent mesenteric haematoma & mesenteric lymph nodes and blood in distal bowel. Histology confirmed the features of Crohn''s disease.

discussion

Crohn''s disease is unusual cause of massive lower gastrointestinal bleeding occurring in 0.9–6% of patients. Rectal bleeding associated with diarrhoea is relatively more common than massive bleeding. The presence of Crohn''s disease in young patients presenting like this is unlikely to be suspected and diagnosis could only be made after laparotomy.     

Castleman's disease arising from the chest wall

An 18-year-old man was referred to our hospital because of an abnormality on chest radiography. Computed tomography showed a sharply defined, 5 × 2 × 3?cm, densely enhanced mass in the left side of the chest wall. We performed tumor resection under video-assisted thoracoscopy, with minimal bleeding. Histologically, the tumor was shown to be Castleman's disease. This is the eleventh case of Castleman's disease arising from the chest wall reported over the last 20 years.     

Retroperitoneal unicentric Castleman's disease—A case report and review of literature

Introduction and importanceCastleman's disease is not so commonly diagnosed worldwide due to non-specific symptoms Clinical findings are variable.No definite blood investigation or any biomarkers are established to diagnose this disease. Radiological investigations do not play much role in diagnosing. It can be unicentric or multicentric. Etiological factors are not well understood except predilection of this disease towards immune-compromised persons. Surgery is considered as a prime modality to treat, if resectable.Case presentationPatient had recurrent abdominal pain in left lumbar region which was intermittent for last 6 months, dull aching with no aggravating and relieving factors. There was no history of abnormal bowel habits, urinary complaints, fever, night sweats, weight loss, rashes in body, joint pains and loss of appetite. On examination, there was a single, firm, non-tender, intra-abdominal, retroperitoneal mass of approximately size 10 1 7 cm in left lumbar region encroaching inferiorly towards left inguinal region. Digital rectal examination (DRE) and external genitalia were normal.Clinical discussionAll routine blood investigations along with relevant tumour markers were normal. Magnetic Resonance Imaging (MRI) abdomen showed a well-defined mass suggestive of stromal tumour or retroperitoneal sarcoma. Patient underwent surgery in which complete excision of the mass was done. Postoperative event was uneventful and currently doing well.ConclusionTake away lesson in this case report is that we should not presume all retroperitoneal mass as cancer or sarcoma, we have to think about other rare causes like Castleman's disease, if clinical picture, blood and radiological investigation are discordant.     

Giant recurrent retroperitoneal liposarcoma initially presenting as inguinal hernia: Review of literature

INTRODUCTIONLiposarcomas comprise around 15% of soft tissue tumors. These tumors of mesodermal origin arise as single tumors, present one histologic type and diverse locations (including the retroperitoneum). Diagnosis of liposarcomas of retroperitoneum is difficult because of this unspecific presentation and in 50–100% of the cases there is recurrence from residual tissue.PRESENTATION OF CASEAn 86 year old male patient was admitted in 1996 due to a right and voluminous inguinal hernia. During the herniaplasty, a right paratesticular tumor was isolated and removed. The histologic exam revealed a well-differentiated liposarcoma. A CT scan was performed and a large abdominal mass was detected. The patient underwent a laparotomy and an incomplete resection of the tumor was achieved. After the surgery the patient remained asymptomatic during a long period. Nine years later, the patient underwent another laparotomy with partial removal of the giant recurrent retroperitoneal liposarcoma.CONCLUSIONThe purpose of this publication is to report the recurrence of giant retroperitoneal liposarcoma, which is an unusual presentation in surgery today. Furthermore, we would like to emphasize the long-term survival of this patient despite partial resection and the possibility of performing a re-resection in this type of cases.     

介入诊疗急性动脉性消化道大出血

目的探讨急性动脉性消化道大出血介入诊疗的临床应用价值。方法对39例急性动脉性消化道大出血患者行DSA检查,对38例出血征象阳性的患者中37例采用明胶海绵颗粒配合微弹簧圈进行介入栓塞治疗。结果 39例动脉造影中38例出血征象阳性,主要表现为对比剂外溢。接受栓塞治疗的37例中,止血成功36例(其中3例行第2次栓塞),均未发生与介入栓塞相关的严重并发症。最终2例患者转剖腹探查手术。结论介入技术是急性动脉性消化道大出血安全、有效的诊疗手段,能快速明确出血动脉及部位,并迅速有效控制出血。     

Laparoscopic resection of paraaortic or paracaval lesions: feasibility and outcome

Background

Laparoscopic retroperitoneal lymph node dissection of paraaortic and paracaval lymph nodes is used to stage nonseminomatous germ cell tumors. Primary tumors can arise from the retroperitoneum, and tumors from nonurologic malignancy also may metastasize to retroperitoneal lymph nodes. This study aimed to describe the authors’ experience with laparoscopic resection of these lesions.

Methods

A consecutive series of patients between January 2007 and June 2011 with paraaortic, aortocaval, or paracaval tumors with a maximum diameter smaller than 10 cm and confined to the abdomen were considered for laparoscopic resection. Data were collected on size and pathology of the lesions, anesthesia time, postoperative stay, and postoperative morbidity and mortality.

Results

In this study, 25 patients with a median age of 49 years were assessed for laparoscopic resection. Eight patients were considered unsuitable for a laparoscopic approach because of tumor location (n = 5), previous retroperitoneal surgery (n = 1), stoma (n = 1), or lesion not clearly visible on computed tomography (n = 1). Of the 17 patients undergoing laparoscopic resection, 1 was found to have diffuse peritoneal disease at laparoscopy, whereas another was converted to an open procedure due to bleeding. All the laparoscopic patients had an R0 resection. The median hospital stay was significantly shorter in the laparoscopic group (2 days) than in the open group (6 days) (P = 0.009). One patient in the laparoscopic group with a functioning paraganglioma and advanced cardiac disease died on postoperative day 7.

Conclusion

Laparoscopic paraaortic and paracaval surgery for primary and recurrent tumors of the retroperitoneum is feasible, with clear resection margin rates similar to that observed for open surgery.     

A case of incidentally detected Castleman's disease with retroperitoneal paravertebral localization.

Castleman's disease, or angiofollicular lymph node hyperplasia, is a fairly rare benign tumor of lymphoid origin. The expected localization is mediastinum and rarely retroperitoneum. An asymptomatic case is reported with multimodality imaging and postoperative findings. The lesion was detected incidentally on routine chest radiogram. Surgical exploration revealed a retroperitoneal mass and the mass was resected successfully through a thoracoabdominal flank incision. Castleman's disease should be included in the list of differential diagnosis of retroperitoneal masses which are mostly malignant tumors.     

Adrenal schwannoma: A case report of an unusual incidentaloma

Introduction and importanceAdrenal schwannomas are extremely rare tumors often misdiagnosed. The patients are usually asymptomatic while some present with non-specific abdominal pain. Only a few cases are reported to date.Case presentationWe here present a case of a 55-year-old Nepalese man presented with nonspecific abdominal pain at our Outpatient Department (OPD) found to have mass on ultrasonography of abdomen. On further investigation with Contrast Enhanced Computerized Tomography (CECT) of the abdomen and pelvis, a well-defined heterogeneous adrenal mass of size (7.8 1 8.3 1 6) cm with foci of calcification was seen in the left retroperitoneum. The intraoperative finding of adrenal mass and histopathology of resected mass was suggestive of schwannoma arising from the adrenal gland which was further confirmed by immunohistochemistry.Clinical discussionAdrenal schwannoma can mimic tumors like pheochromocytoma, adrenal adenoma, cortical carcinoma, neuroblastoma, and other masses. Only 1–3% of schwannomas are retroperitoneal. Radiological findings of this tumor are non-suggestive. The histological section shows spindle cells with Antoni A and Antoni B regions while positive staining of S-100 protein in Immunohistochemistry.ConclusionThe diagnosis of adrenal schwannoma in the retroperitoneum is often challenging. The treatment of choice is surgical resection with a good prognosis.     

Benign schwannoma of the pelvic retroperitoneum. Report of a case and review of the literature

Benign schwannoma is a tumor arising from Schwann cells (forming the neural sheath of peripheral nerves). The retroperitoneal location is unusual (0.5-5% of cases). Most common locations are cranial nerves (especially the 8th pair) and, in peripheral nerve system the neck, mediastinum and extremities. To this date the known cases of benign retroperitoneal schwannoma are about 60, of which less than 20 in the pelvis. The low frequency of this tumor and the lack of specific instrumental signs and objective symptoms (since it develops in a deep and broad region as retroperitoneum) make presurgical diagnosis very difficult. It can be confirmed only during surgery and definitive histological examination. The information provided by ultrasonography, CT and MR help to limit diagnostic hypothesis, but they don't show any pathognomonic images. The resection of this tumor is the appropriate treatment, even though it is really a complex one. Prognosis is quite good since post-surgical recurrences are unusual. If they appear is probably because excision wasn't radical. Complete resection is the best treatment for retroperitoneal pelvic schwannoma and today it can be performed also by laparoscopy. Partial resection can be used when the mass is strongly connected to essential organs in order to prevent iatrogenic harms (neural deficit, vessel lesions); this may occur in 10% of cases. This paper describes a benign schwannoma with pelvic retroperitoneal location, incidentally discovered during a routine gynecological check up. The purpose of this study is to review current therapeutic and diagnostic techniques in retroperitoneal pelvic schwannoma (including a review of current literature) and to identify th problems that can be encountered in the differential diagnosis of this unusual disease from other neoplasms occurring in the same place.     

Primary retroperitoneal mucinous cystadenoma-A case study and review of the literature

INTRODUCTIONPrimary retroperitoneal tumours of mucinous type are extremely rare and can be further sub-divided into benign, borderline or cystadenocarcinoma. Prompt diagnosis of retroperitoneal tumours is important as the majority are malignant.PRESENTATION OF CASEOur case describes a 30 year old woman, presenting with a 3 month history of intermittent right iliac fossa pain. Abdominal examination demonstrated a mass palpable in the right iliac fossa. Ultrasonography of the abdomen demonstrated a cystic mass with a magnetic resonance imaging (MRI) scan of the pelvis further defining the lesion. Laparoscopy was performed to further evaluate and ultimately remove the retroperitoneal mass. Macroscopic and microscopic examination reported mucinous epithelium of endocervical type with no evidence of invasion. Findings were consistent with primary retroperitoneal mucinous cystadenoma.DISCUSSIONThis is the 19th reported case of a benign primary retroperitoneal mucinous cystadenoma in the English literature. The origin of mucinous cystadenomas in the retroperitoneum is widely debated with multiple theories suggested. Diagnosis of retroperitoneal tumours is important but difficult as serological investigations, ultrasonography, computed topography and magnetic resonance imaging, although useful, cannot allow a confident diagnosis.CONCLUSIONPrimary retroperitoneal mucinous cystadenoma is a benign tumour, however because of the malignant nature of the majority of mucinous retroperitoneal tumours they should be considered in the differential of chronic abdominal pain despite their rarity.     

Duodenocaval Fistula: A Late Complication of Retroperitoneal Irradiation and Vena Cava Replacement

Duodenocaval fistula (DCF), an unusual pathology, is associated with a 40% mortality rate in the 36 patients previously reported. Although migrating or ingested foreign bodies, trauma, and peptic ulcer disease are often described etiologies, 11 patients have been described who developed DCF after resection of retroperitoneal tumors, 9 of whom also had postoperative radiotherapy. We report two patients who developed DCF after resection of retroperitoneal tumors followed by radiation therapy. The first patient, a 56-year-old female, presented with upper gastrointestinal hemorrhage requiring transfusion caused by a duodenoprosthetic caval fistula 7 years after successful resection of a retroperitoneal leiomyosarcoma and replacement of the inferior vena cava followed by radiation and chemotherapy. The second patient, a 37-year-old male who had previously undergone resection of a retroperitoneal sarcoma followed by external radiotherapy, developed massive upper and lower gastrointestinal bleeding secondary to a duodenocaval fistula. The etiology, diagnosis, and treatment of DCF are analyzed with an emphasis on DCF following resection and irradiation of retroperitoneal tumors. In most patients, spontaneous DCF have occurred as a late complication of high-dose radiation for carcinoma of the right kidney or retroperitoneal structures. Presented at the Annual Meeting of the Southern California Vascular Surgical Society, Carlsbad, CA, April 11-13, 2003.     

Asymptomatic splenic infarct and retroperitoneal sepsis in patient with Wegener's granulomatosis

INTRODUCTIONWe present a patient with an unusual combination of surgical findings, complicated by both a medical condition and the treatment there of.PRESENTATION OF CASEA 52 year-old man with Wegener's granulomatosis presented with groin pain and sepsis while on immunosuppressive therapy. Large retroperitoneal abscesses were found on CT scanning, together with a collapsed left lower lobe of the lung, a complete infarct of the spleen, and evidence of diverticulosis. At the exploratory laparotomy, the infarcted spleen was removed and the retroperitoneal abscesses were drained via a separate lateral incision. Uncomplicated diverticular disease were also confirmed.DISCUSSIONThe left lower lobe collapse, infarcted spleen and diverticular disease were all potential sources of the sepsis, but none of them could be clearly linked to the abscesses. The splenic infarct and a post-operative myocardial infarct were likely related to his vasculitic disease.CONCLUSIONPatients with systemic vasculitis may present with unusual pathologies, and immunosuppressive treatment may also modify clinical presentation.     

Surgical resection for liver metastasis and local recurrence of pheochromocytoma 16 years after primary surgery: A case report

Introduction and importancePheochromocytomas arise from the adrenal medulla and are rare. Pheochromocytomas metastasize to bone, lung and liver, and surgery might be the curative treatment. However, few cases are detected when they are resectable lesions because of difficulty in diagnosis and rapid growth. We herein report a patient who underwent resection of liver metastasis and local recurrence of pheochromocytoma.Case presentationA 74-year-old woman visited our hospital for treatment for liver and retroperitoneal tumors. She had undergone left adrenal gland resection for pheochromocytoma 16 years earlier. Eleven years after primary surgery, breast cancer was diagnosed and resected. During the breast cancer follow-up, a liver tumor was identified with computed tomography. Breast cancer recurrence and metastasis were considered, so chemotherapy was administered first. However, the liver tumor gradually enlarged, and another lesion appeared in the retroperitoneum. The tumors were diagnosed as pheochromocytoma recurrence using ¹²³I-metaiodobenzylguanidine scintigraphy, and she underwent resection of the local recurrence and liver metastasis. She was discharged on postoperative day 25 without complications, and no evidence of recurrence occurred more than 3.5 years postoperatively.Clinical discussionAll pheochromocytomas have metastatic potential; however, there are no reliable markers to predict malignancy. Early detection of recurrence by regular imaging and complete resection are important in the treatment. If the recurrence was oligometastasis and tumor growth is slow, surgical resection may be eligible.ConclusionA favorable outcome resulted from complete resection for liver metastasis and local recurrence of pheochromocytoma.