Necrotizing fasciitis of the thigh due to the colon cancer, especially during chemotherepy, has not been previously reported.A 67-year-old man admitted to the hospital was diagnosed with sigmoid colon cancer that had spread to the left psoas muscle. Multiple hepatic metastases were also found, and combination chemotherapy with irinotecan and S-1 was administered. Four months after the initiation of chemotherapy, the patient developed gait disturbance and high fever and was therefore admitted to the emergency department of our hospital. Blood examination revealed generalized inflammation with a high C-reactive protein level. Computed tomography of the abdomen and pelvis showed gas and fluid collection in the retroperitoneum adjacent to the sigmoid colon cancer. The abscess was locally drained under computed tomographic guidance; however, the infection continued to spread and necrotizing fasciitis developed. Consequently, emergent debridement was performed. The patient recovered well, and the primary tumor was resected after remission of the local inflammation.Necrotizing fasciitis of the thigh due to the spread of sigmoid colon cancer is unusual, but this fatal complication should be considered during chemotherapy for patients with unresectable colorectal cancer. Necrotizing fasciitis (NF) is a rare and life-threatening soft-tissue infection. Aggressive surgical management is required in the early stage in order to reduce the associated high mortality rate, which ranges from 20% to 40%; howeverNF is usually caused not only by trauma to the skin, such as that induced by insect bites, scratches, and abrasion, but also by surgical wounds in the perineum and lower extremities. Other lA 67-year-old man, who was healthy earlier, was referred to our hospital for a month-long history anorexia. On the basis of the results of a computed tomography (CT) scan and gastrointestinal endoscopy, the patient was diagnosed with unresectable sigmoid colon cancer that had spread to the retroperitoneum Figure ; multiplEscherichia coli and other anaerobic bacteria. The patient showed good postoperative recovery, and the primary tumor was resected 2 months after the first surgery. The operative findings indicated that the cancerous lesion and the tissues surrounding it were firmly attached to the left retroperitoneum. Multiple liver and peritoneal metastases were also detected. Palliative resection of the primary tumor was performed in order to prevent the recurrence of retroperitoneal inflammation. On the basis of the operative findings, the tumor was classified as a T4 (invading the psoas muscle), N1, and M1 (liver and peritoneum), and the patient was clinically diagnosed with stage IV cancer according to the definitions laid down by the International Union Against Cancer (UICC). The patient was given oxaliplatinm 5-fluorouracil, and folinic acid (modified FOLFOX6) therapy, but, he died due to cancer 8 months after the second surgery.Four months after the initiation of chemotherapy, he was readmitted to the hospital for dyskinesia of the left lower extremity and high fever. Blood examination data indicated leukopenia , and a high C-reactive protein (CRP) level (16.7 mg/dL). A CT scan showed fluid and gas collection in the retroperitoneum adjacent to the primary tumor Figure , althougNF is a serious soft-tissue infection that causes secondary necrosis of the subcutaneous tissues. It can occur in any region of the body but most commonly occurs in the abdominal wall, extremities, and perineum.It has been reported that NF has a high morbidity and mortality rate because of its acute and rapidly progressive course. The outcome of NF is rendered poor most importantly by delays in its diagnosis and surgical debridement. Thus, early diagnosis of necrotizing soft-tissue infections followed by administration of intravenous antibiotics and surgical intervention is the best way of decreasing the mortality associated with this aggressive infection. Clinical features of NF include high fever with chills, tenderness over the affected area along with changes in skin color, and palpable crepitus.It is well known that perineal NF, termed as "Fournier's gangrene," is caused by rectal cancer or periproctal abscess, and theIn the 3 previously reported cases, symptoms of NF preceded the diagnosis of colorectal cancer; thus, to our knowledge, this is the first reported case in which NF developed during chemotherapy for the treatment of colorectal cancer.In the present case, we inserted only the pig tail catheter immediately after the diagnosis of retroperitoneal abscess, because we thought that the patient may not tolerate the stress of radical surgery. However, we realized that this was a wrong strategy because NF developed eventually and additional debridement was required. Fortunately, the patient showed good postoperative recovery, however, we believe that NF, a serious complication, could have been avoided if the radical treatment had been initiated earlier.Recent advances in chemotherapy for colorectal cancer  have improved the median survival period of patients with unresectable colorectal cancer -15. PatiTherefore, chemotherapy is currently the first line of treatment for patients with unresectable colorectal cancer. Palliative resection of the primary lesion is rarely performed when there are no symptoms of primary cancer, such as intestinal obstruction or bleeding.Although there are several reports have stated that primary tumor resection contributes to prolonged survival in patients with incurable colorectal cancer,17, therSpecifically, a high incidence of bowel perforation and delayed wound healing have been observed in patients treated with bevacizumab. TherefoOur reported case highlights the importance of the removal of the primary tumor in an aymptomatic patient as an attempt to avoid concomitant serious complications.Retroperitoneal abscess and NF are rare complications of colorectal cancers that can potentially be fatal, particularly in patients who are immunocompromised because of chemotherapy. In the presence of these unclear risk factors, accurate and rapid clinical judgment and a careful consideration of balance between the risks and benefits are necessary before performing a palliative surgery.Colon cancer could be a cause of unexpected retroperitoneal abscess followed by NF of the thigh, and NF should be considered during the diagnosis of colon cancer. Early diagnosis and treatment can help reduce the mortality rate associated with NF.NF: necrotizing fasciitis; CT: computed tomography; CRP: C-reactive protein.The authors declare that they have no competing interests.YT participated in treatment of the patient, collected case details, literature search and draft the manuscript. SI participated in treatment of the patient and helped to draft the manuscript. MY, TH, DS, HT, YK, MS and MT participated in treatment of the patients. MO and HO participated in treatment planning of the patient and helped to draft the manuscript. All authors read and approved the final manuscript.Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.


Closing the pelvic peritoneum to prevent the small bowel dropping into the pelvis after surgery for locally recurrent rectal cancer is important to prevent adhesions deep in the pelvis or complications of adjuvant radiotherapy. Achieving this could be difficult because sufficient native tissue is unavailable; we report on the use of small intestine submucosa extra-cellular matrix mesh in the obliteration of the pelvic brim.We describe two cases in which submucosa extra-cellular matrix mesh was used to obliterate the pelvic brim following resection of a recurrent rectal tumour; the first patient, a 78-year-old Caucasian man, presented with small bowel obstruction caused by adhesions to a recurrent rectal tumour. The second patient, an 84-year-old Caucasian woman, presented with vaginal discharge caused by an entero-vaginal fistula due to a recurrent rectal tumour.We report on the use of submucosa extra-cellular matrix mesh as a pelvic sling in cases where primary closure of the pelvic peritoneum is unfeasible. Its use had no infective complications and added minimal morbidity to the postoperative period. This is an original case report that would be of interest to general and colorectal surgeons. Closing the pelvic peritoneum to prevent the small bowel falling into the pelvis following surgery for local recurrence of rectal cancer can be difficult because sufficient native tissue is unavailable. Small intestinal submucosa extra-cellular matrix mesh  has been used to repair hernias, but it has never been documented to be used for pelvic inlet closure.Our patient was a 78-year-old Caucasian man with a past surgical history of Hartman's procedure performed in February 2005 for a perforated recto-sigmoid tumour. Postoperatively he developed pulmonary embolism and partial abdominal wound dehiscence. The fascial dehiscence was treated with daily dressings followed by elective closure three months later. The patient was not offered adjuvant chemotherapy because of his poor general health. He was followed up as an outpatient; this included a normal carcinoembryonic antigen (CEA) level, colonoscopy through the stoma, and computed tomography (CT) scan of the abdomen and pelvis. In October 2006 he presented to the accident and emergency (A&E) department with a three-day history of generalized colicky abdominal pain associated with vomiting, constipation and abdominal distension.Examination showed tachycardia and dehydration. The abdomen was distended but soft with tenderness generally on deep palpation. There were exaggerated bowel sounds and digital examination through the colostomy showed no faecal impaction.Routine bloods were normal except a raised C-reactive protein (CRP) of 92.9 and urea of 11.5. Plain film of the abdomen (PFA) showed dilated loops of the small bowel. CT scan of the abdomen and pelvis demonstrated a solitary liver metastasis in the right lobe of the liver, dilated loops of small bowel, and a soft tissue mass in the region of the rectal stump suspicious of a local recurrence. Conservative treatment was tried initially to relieve the obstruction, but the patient's condition did not improve. An exploratory laparotomy was then performed and the loop of small bowel adherent to the recurrent rectal tumour was mobilised and resected. The tumour itself was unresectable. To prevent the small bowel from falling into the pelvis, SURGISIS mesh was sutured to the sacral promontory, lateral pelvic wall and symphysis pubis to close the pelvic brim, and a tube drain was left in the pelvic cavity. The patient recovered well from the surgery with no complications. Neither adjuvant chemotherapy nor radiotherapy was advised because of his frail general condition and he has been followed up by both the surgical and palliative care teams as an outpatient.Our patient was an 84-year-old Caucasian woman with a past surgical history of Hartman's procedure performed in 1993 for perforated sigmoid diverticulitis. The colostomy was reversed one year later. In 2005 she was diagnosed with a rectal tumour 10 cm from the anal verge. An anterior resection/Hartman's procedure was performed in May 2005. The tumour stage was Duke's B, but because of her age she was not considered for adjuvant therapy. In October 2006 she presented with per vaginal discharge of small intestinal contents.CT of the thorax, abdomen and pelvis showed pulmonary metastases and local recurrence at the rectal stump with an entero-vaginal fistula. An exploratory laparotomy was performed, resecting the recurrent rectal tumour and the involved segment of small intestine; continuity of the small bowel was restored with a side-to-side anastomosis. The pelvic brim was closed with SURGISIS mesh sutured as in the first case to the sacral promontory, lateral pelvic wall and the symphysis pubis. Postoperatively the patient did well and was discharged home; she was later reviewed by an oncologist who opted against adjuvant therapy because of her age and poor general health. She is followed up by both surgical and palliative care teams as an outpatient.A variety of abdominopelvic partitioning procedures designed to prevent the small bowel from coming into contact with the pelvic floor or the presacral area are described in the literature . These pIn our two cases, the pelvic peritoneum needed to be closed to exclude the small bowel from the recurrent tumour at the rectal stump and to facilitate postoperative radiotherapy. Because of previous pelvic surgery this was unfeasible and for that reason we used a prosthetic mesh that will encourage the growth of host tissues to prevent the small bowel from falling into the pelvis long after the mesh has dissolved. SURGISIS mesh is a three dimensional, acellular extra-cellular matrix comprised of collagen, noncollagenous proteins and other biomolecules including glycosaminoglycans, proteoglycans and glycoproteins harvested from porcine small intestine submucosa and made biocompatible using a patented process. It acts by providing a matrix for host connective and epithelial tissue growth and differentiation, resulting in a phenomenon called smart tissue remodelling as the implant and host tissue become indistinguishable . ExperimThere is evidence that SURGISIS has been used successfully to repair congenital diaphragmatic hernias  and it hThe technique we used to fix the mesh was similar to that described by Devereux et al  with minIn cases where primary closure of the pelvic peritoneum is not feasible, the use of SURGISIS as a sling to hold the small bowel out of the pelvis can be performed without infective complications and adds minimum morbidity during the postoperative period. The use of SURGISIS as a pelvic sling has not been reported before and we suggest that future studies should compare SURGISIS slings to other abdominopelvic partitioning procedures.SURGISIS: small intestinal submucosa extra-cellular matrix mesh; CEA: carcinoembryonic antigen; CT: computed tomography; A&E: accident and emergency; CRP: C-reactive protein; PFA: plain, film abdomen.Written informed consent was obtained from both patients for publication of this case report. A copy of the written consent is available for review by the Editor-in-Chief of this journal.The authors declare that they have no competing interests.OA reviewed patient charts and wrote the manuscript. SE critically reviewed the manuscript. Both authors read and approved the final manuscript.


The ovary is a common site of metastasis from various organs. However, little is known about gallbladder carcinoma metastasizing to the ovaries and presenting as a primary ovarian tumor.We report two cases of a metastatic gallbladder carcinoma which mimicked a primary ovarian tumor in a 35-year-old and a 62-year-old North Indian woman. Clinically, both our patients presented with abdominal masses without obvious signs and symptoms related to gallbladder carcinoma. Radiology suggested the possibility of a primary ovarian tumor with chronic cholecystitis and cholelithiasis. The gross features also mimicked a primary malignant ovarian tumor in the first case and a benign mucinous neoplasm in the second case. Exact diagnoses could only be made after thorough sampling from both the ovaries and gallbladder.Gallbladder carcinoma with metastasis to the ovaries can mimic both malignant and benign primary ovarian tumors. Extensive cystic change in the ovary due to metastasis from gallbladder carcinoma has rarely been reported. A high index of suspicion and thorough sampling are essential to avoid misdiagnosis in such cases. Ovary is a relatively frequent site of metastasis from various organs especially pancreas and gastrointestinal tract. Rarely, the metastasis may precede detection of the primary site and may present as an ovarian tumor . MetastaA 35-year-old North Indian woman presented with abdominal pain and discomfort with loss of appetite and indigestion for one month. Systemic examination revealed abdominal distension and slight tenderness in her right hypochondrium along with palpable bilateral adnexal masses. There was no icterus, but mild elevation of serum bilirubin with normal liver enzyme levels. An ultrasound examination of her abdomen showed a diffusely thickened gallbladder with multiple calculi and bilateral large, solid-cystic adnexal masses suggestive of a primary ovarian malignancy with chronic cholecystitis and cholelithiasis. Her serum tumor marker CA-125 was raised . Our patient underwent total abdominal hysterectomy and bilateral salpingo-oophorectomy with cholecystectomy. On exploration during surgery the gallbladder was found to be inflamed and adherent to part of omentum, therefore extended omentectomy was performed with removal of pelvic and retro-pancreatic lymph nodes.Both her right and left ovaries were enlarged and measured 17 × 8 × 5 cm and 16 × 7 × 5 cm, respectively. External surface of both was nodular Figure  and slicBoth the masses showed a similar morphology on microscopy. Solid areas were composed of irregular glands and nests infiltrating the loose stroma Figure . The tumThe gallbladder had a gangrenous appearance with diffusely hemorrhagic and thickened wall covered with slough on both the serosal as well as mucosal aspect Figure . The lumA 62-year-old woman from a Northern part of India presented with complaints of pain and swelling in the abdomen and generalized weakness for a duration of four months. Routine biochemistry including liver function tests and hematological parameters were normal. A computed tomography (CT) scan of her abdomen showed two large masses arising from pelvis on either side of the uterus. The masses were reaching up to epigastrium and displacing gut loops anteriorly and towards right side. Both of them were largely cystic with well defined walls Figure . Her galBilateral ovarian masses were well encapsulated with right mass measuring 20 × 18 × 11 cm and left 18 × 13 × 10 cm. Capsular surface of both revealed evenly distributed multiple tiny pinhead size excrescences Figure . Cut surOn microscopy cystic spaces were lined by flattened epithelium and filled with acellular material Figure . On low The serosal surface of gallbladder was smooth and shiny. The lumen was impacted with a 1.3 cm diameter cholesterol stone. In the body region mucosa was ulcerated with variably thickened wall Figure . MicroscThe incidence of ovarian metastasis from different organs is nearly five to 15% . AlthougSimilar to the present report, a majority of such patients had non-specific abdominal or pelvic symptoms . Jaundice or other symptoms related to gallbladder carcinoma were observed in only few cases ,6,9. RadThe morphological features, on histology of metastasis, may mimic not only malignant but also a benign ovarian tumor as observed in our patients. In the first case, the gallbladder was gangrenous and no obvious growth was apparent on gross examination. Microscopically, only a few tumor glands were noticed in one of the sections taken from the gallbladder. The origin of these glands could not be traced from these initial sections. The gallbladder therefore was re-grossed. Repeat sections taken revealed a tumor diffusely involving the gallbladder wall with overlying dysplastic epithelium. This along with a bilateral tumor, multinodularity, infiltrative pattern and presence of uninvolved tissue supported the possibility of a metastatic carcinoma rather than a primary malignancy in the ovaries.The second case showed a full-fledged gallbladder malignancy. The ovarian masses, however, were completely cystic with no solid areas. The initial sections suggested possibility of a benign mucinous tumor. However, presence of focal atypia in the lining epithelium and a high index of suspicion, in view of presence of a gallbladder malignancy led to re-examination of the specimen. Tiny pinhead size elevations over the capsule Figure  and subc-/CK20+) in contrast to ovarian primaries (CK7+/CK20-/CK20+), its role in metastasis from gallbladder is limited because of similar profile to that of primary ovarian mucinous tumors [In the literature a variety of features have been emphasized Table  that mays tumors . A thoroOutcome in these cases is generally poor. However, adequate surgery with palliative treatment may prolong survival for few months. Therefore at the time of total abdominal hysterectomy and bilateral salpingo-oophorectomy with cholecystectomy presence of unusual findings such as a gallbladder mass, dense adhesions of the omentum and adjacent organs to the gallbladder, difficult dissection of the gallbladder from its liver bed should raise the suspicion of a carcinoma. A close evaluation of the extent of the disease should be carried out. Biopsy of any lymph node should be taken. Intra-operative ultrasound, intra-portal endoscopic ultrasound and frozen section all may be performed to assess the extent of the disease. In the presence of ascites, fluid should be obtained for cytology; otherwise, a peritoneal wash-out can be considered for cytology . ExternaGallbladder carcinoma should be added to the previously known list of origins of metastatic tumors to the ovary that can closely mimic primary ovarian mucinous tumors. Pathologists should maintain a high index of suspicion and adequate sampling should be done of ovarian masses especially if bilateral. In all bilateral mucinous tumors outer surface should be examined carefully for presence of tiny deposits. Knowledge of the extent to which gallbladder metastasis may mimic a primary ovarian tumor and its differentiating histological features may help in correct diagnosis and further management of the patient.Written informed consent was obtained from both the patients for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.The authors declare that they have no competing interests.YK designed, carried out acquisition and analysis of data and drafted the manuscript. AC and AB helped in drafting of manuscript and given their valuable suggestions, MG provided the images. All the authors read and approved the final manuscript.


Renal cell carcinoma has been increasing in incidence over the past two decades. Men are affected more than women and metastatic disease at presentation occurs in up to one third of patients. Metastasis can occur to virtually any organ, and involvement of multiple organs is not uncommon. To date, no reports have been found of metastatic disease without a renal primary. We present a case of renal cell cancer initially presenting as a subcutaneous mass with subsequent pancreatic and parotid gland metastases in absence of a primary renal source. In the United States, renal cell carcinoma (RCC) has incidence in excess of 30,000 cases, with 12,000 deaths every year from the disease . In contIn October, 2007 a 61-year-old woman presented to Saint Vincent's Medical Center with a 5 cm subcutaneous growth on her left upper extremity. Histological examination after surgical excision of the mass revealed a clear cell neoplasm consisting of polygonal cells with abundant clear cytoplasm, containing faint granular material. Immunohistochemical analysis demonstrated positive CD10 and AE1/AE3 staining. Pathologic interpretation of the mass was highly suggestive of metastatic RCC of the clear cell type. There were no lesions present anywhere else by physical examination or CT scan.The patient was closely followed in an attempt to locate a primary renal source of disease with multiple imaging studies negative for a renal primary or other sites of metastasis. However, repeat CT scan 9 months later revealed an asymptomatic pancreatic mass. Endoscopic evaluation was performed with endoscopic ultrasound and fine needle aspiration (EUS/FNA). The study demonstrated a 2-cm hyperechoic, well-defined lesion in the body of the pancreas. The remaining pancreatic parenchyma was otherwise normal without ductal dilation or evidence of pancreatitis. Histomorphological analysis of the core biopsy samples yielded similar findings to those of the upper extremity mass. Additionally, an immuno-profile was focally strong for both CD10 and PNRA, which was again highly suggestive of renal cell carcinoma. A central pancreatectomy was performed in August 2008 and tissue samples were positive for PRNA, Vimentin, and CD10, correlating strongly with RCC. The patient continued periodic surveillance to identify a renal primary and further metastasis at three month intervals. Six months later, physical exam revealed left parotid gland enlargement and an MRI revealed a 1.6 cm enhancing mass in the left parotid gland. No other lesions were found on surveillance PET/CT scan at that time. The patient had a superficial parotidectomy and again, pathological analysis demonstrated a clear cell carcinoma that was identical to the previous subcutaneous and pancreatic specimens. The sample was sent for expert verification at an outside institution, which corroborated our findings. Currently, the patient is doing well and is undergoing surveillance at 6 month intervals. To date, a renal primary has not been found.RCC has an annual incidence in excess of 30,000 cases in the United States and its greatest incidence occurs in males during the sixth decade of life . The natImmunohistochemical studies are helpful to distinguish metastatic from primary pancreatic tumors. According to the literature, 90-100% of pancreatic adenocarcinomas express CK7 as well as CK8,13,18, and 19 -13. EvenFor patients with solitary pancreatic metastases, surgical treatment should be recommended because it is more effective than other treatments such as radiation and chemotherapy. The mean survival reported in the literature is only 1.3 years following metastatic focus resection , but 5 yIn summary, this case describes the first documented report of metastatic RCC without the determination of a primary renal tumor. In the vast majority of cases, the primary renal lesion is found through subsequent radiographic surveillance, sometimes up to several years after the discovery of the initial metastatic lesion, which did not occur in our patient. The fact that this case presented with evidence of metastasis to three different sites  demonstrates the interesting ability of RCC to invade almost any organ and also reinforces the need for a thorough work-up to distinguish primary pancreatic neoplasm from another metastatic process. Although many cases are found incidentally from radiographic imaging obtained for other reasons, tissue sampling for pathological and immunohistochemical analysis is essential to help determine the tumor origin. When metastatic RCC is found, complete surgical resection should be the treatment of choice when medically feasible.The authors declare that they have no competing interests.Informed Consent was obtained from the patient for the publication of this case report.MW is the lead author and surgeon for this case. It is his patient being written about.WW is the lead pathologist, who did extensive research on this case. BC is the resident doctor who performed the literature search. JB is the GI doctor who aided in the review and correction of this article. FK is the GI doctor who performed the procedure. AC is the senior surgeon who assisted in the case and in the write up of the case.


Purpose. To present a case study involving the use of fast neutron            therapy to treat an extensive unresectable osteogenic sarcoma arising from            the left maxilla.            Patient. A 14-year-old male presented with a massive tumor producing            severe distortion of his facial structures. He had already received six courses of            chemotherapy, which had reduced his pain, but had not measurably reduced the tumor.            Methods. The patient was treated with 66 MeV fast neutrons to a dose                of 20.4 Gy in 13 fractions over 35 days.                Results. CT assessments indicate gradually increasing calcification                and noticeable reduction of soft-tissue disease in the frontal sinus, orbit and maxillary                antrum.There has been some recontouring of the facial structures.The boy conducts an                active life, has no pain, and feels well. He was 17 years old at the last follow-up.                Discussion. Fast neutrons have a greater biological effectiveness                than conventional photon beams. Their use has been associated with improved chance                for local control of unresectable disease.This case illustrates their effectiveness in                controlling an unusual and aggressive osteogenic sarcoma of the facial bone and sinuses.


Spermatic cord is a rare metastatic site of colorectal cancer. We herein report a case of spermatic cord metastasis of a previous undiagnosed multifocal colon adenocarcinoma, which was clinically presented as a strangulated groin hernia. Malignant lesions of spermatic cord, both primary and metastatic, are extremely uncommon. The proportion of metastatic neoplasms is very low, as these tumours account for less than 10% of malignant tumors of this site ,3.We herein describe the case of a 80-year-old male patient with multifocal colon adenocarcinoma, whose first presentation was a spermatic cord metastasis which was presented as strangulated inguinal hernia.An 80-year-old man with a known history of right inguinal hernia was admitted to our department complaining for intermittent pain at the right groin and multiple episodes of vomiting started approximately 48 hours earlier. Clinical examination revealed the presence of a painful mass at the right inguinal area, as well as a tender abdomen with decreased bowel sounds. Abdominal radiography showed the presence of multiple air fluid levels and distended small bowel loops. Based on the medical history of the patient and the clinical examination, the diagnosis of strangulated inguinal hernia was made and the patient was emergently transferred to the operating room for open repair. Through a right inguinal incision the inguinal canal was accessed, where a relatively large, but not incarcerated inguinal hernia was found. In addition a solid mass was observed in close relation to spermatic cord and spermatic vessels. A small piece of the tumor was excised and sent for frozen section examination which was positive for malignancy. The patient underwent right high orchiectomy Fig. , followeth century by Lesauvage [The first case of spermatic cord tumor was described in the middle of 19esauvage . Since tesauvage ,3. Colonesauvage , though esauvage .Primary malignant neoplasms spread to spermatic cord mainly via hematogenous or lymphatic route. However, other routes of spread have also been described, including i) retrograde extension through the vas, either along its lumen, or direct extension via the wall of the vas, and ii) transperitoneal seeding through a patent tunica vaginalis . In our To the best of our knowledge, this is the first described case of a patient with spermatic cord metastasis mimicking strangulated hernia. Incarcerated or strangulated inguinal hernia is a common diagnosis for patients presenting with a painful and non-reducible groin mass. However, a plethora of different pathological entities may mimic an incarcerated or strangulated inguinal hernia, including Meckel's diverticulum, inflamed appendix, diverticulitis, ovulating ovary, liposarcoma of spermatic cord, spermatic cord hematoma and pancreatic pseudocyst ,7. The cThough rare, metastatic lesion of spermatic cord should be included in the differential diagnosis of a painful groin mass, especially in high risk patients, in order to achieve a more targeted treatment.The authors declare that they have no competing interests.IG was the main surgeon and was major contributor in revising the manuscript critically for important intellectual content. GC, AK and KG analyzed and interpreted the patient data, reviewed the relative literature and were contributors in writing the manuscript.JM and KA were major contributors in revising the manuscript critically for important intellectual content.Written informed consent was obtained from the patient's relatives for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.


Ancient schwannomas are degenerate peripheral nerve sheath tumors that very rarely occur in the retroperitoneum. They generally reach large proportions before producing symptoms due to mass effect. We describe three cases of retroperitoneal ancient schwannomas and discuss the diagnosis and management of these tumors.Three female patients with retroperitoneal ancient schwannomas were reviewed. One patient presented with several weeks of upper abdominal pain and lower chest discomfort, whereas back pain and leg pain with associated weakness were predominant symptoms in the remaining two. Abdominal imaging findings demonstrated heterogeneous masses in the retroperitoneum with demarcated margins, concerning for malignancy. The patients successfully had radical excision of their tumors. Histological examination showed encapsulated tumors that displayed alternating areas of dense cellularity and areas of myxoid matrix consistent with a diagnosis of ancient schwannoma.A diagnosis of ancient schwannoma should be entertained for any heterogeneous, well encapsulated mass in the retroperitoneum. In these cases less radical surgical resection should be considered as malignant transformation of these tumors is extremely rare and recurrence is uncommon following excision. Ancient schwannomas are a rare variant of peripheral nerve sheath tumors, or schwannomas. The term "Ancient" refers to the histological degenerative features, which are acquired with increasing age in these tumors. Nuclear atypia is a common feature and often leads to the erroneous diagnosis of malignancy. They are slow growing and may produce vague local symptoms, but are usually diagnosed incidentally. They are an uncommon cause of a retroperitoneal mass, and are classically encapsulated, highly vascular and have a distinctive radiological appearance ,2. MaligWe describe three cases of retroperitoneal ancient schwannomas managed surgically at our institution over the last two years.en bloc resection of the distal pancreas, spleen and left adrenal gland. This mass had solid and cystic features  scan of the chest, abdomen and pelvis showed a large, well circumscribed, septated cystic lesion with a few scattered calcifications adjacent to the pancreas, measuring 12.0 × 13.0 × 12.0 cm, without any signs of metastases. The epicenter of the mass appeared to be the superior retroperitoneal region with displacement of the tail of the pancreas, kidney and stomach Fig . A presuures Fig . Histopaen-bloc low-anterior resection without complications. Pathology revealed an ancient schwannoma with spindle cells, cystic degeneration, atypical cells, and S-100 positive staining . This led to the discovery of a complex cystic pelvic mass, that was further characterized on CT of the pelvis Fig. . A well-ning Fig . At 18 men bloc resection of the mass with the left hemi-colon, left kidney and adrenal and a partial gastrectomy. The gross pathology specimen is shown  are benign soft tissue neurogenic tumors that arise from Schwann cells of peripheral nerve sheaths. The usually arise from sensory nerves, however, motor nerve origin is also reported. They most commonly manifest in the head and neck region and in the extremities . RetropeAncient schwannomas are a rare variant of schwannomas, originally described by Ackerman and Taylor in 1951 . They acThere is a tendency to confuse ancient schwannomas with malignant tumors on imaging and histology . On cytoOn histology, ancient schwannomas shows areas of cellularity and areas of myxoid matrix, as also observed in conventional schwannomas. There is, however, a relative loss of cellular regions, which tend to be fibrosed or sclerotic. These areas may degenerate into hematomas and cysts, leading to an overall decreased densitity. Nuclear palisades, seen in classic schwannomas, are absent and large intra-nuclear invaginations are characteristically present . The degWe described three cases of retroperitoneal ancient schwannomas, all with features concerning for malignancy and treated by radical excision. All three patients were symptomatic, with two presenting with back and leg pain and associated weakness. Malignant transformation of retroperitoneal schwannomas appears to extremely rare. If a diagnosis of ancient schwannoma is entertained based on imaging and histology, then consideration for less radical surgery may be appropriate in selected cases.Institutional review board approval was obtained to conduct to the case reviews. A copy of the approval documents is available for review by the Editor-in-Chief of this journal.The authors declare that they have no competing interests.HAC, MN, NJC and ETC collected the patient details. HAC and MN reviewed the literature. HAC wrote the paper with the assistance of MN, ETK, NJS, RC and KFS. JL examined the pathology specimens and provided histology details included in the manuscript. All authors were involved in the editing and reviewing of the initial document. KFS, MN, RC, ETC and NJG were involved in the study conception and design. All authors read and approved the final manuscript.


Generalized eruptive syringoma is a rare clinical presentation of a benign adnexal tumor that derives from the intraepidermal portion of the eccrine sweat ducts. It presents as successive crops of small flesh-colored papules on the anterior body surfaces. It generally occurs in the peripubertal period. Treatment of this benign condition is cosmetic only. A case of a 28-year-old female with an eight-year history of eruptive syringoma is presented. Syringomas are benign adnexal tumors of eccrine origin, with four principal clinical variants. In erupt4A 28-year-old white female presented with an eight-year history of a dermatosis involving the upper extremities, axillae, and the anterior chest. The patient was admitted to a private dermatology clinic in Isfahan, with a generalized eruption with mild pruritus during perspiration, of eight-year duration. The lesions appeared on the forearm first, which was followed by successive eruptions on the face, chest, upper abdomen, thigh and neck respectively. The patient denied any medical problems or the use of over-the-counter preparations. The review of systems was noncontributory. The lesions were treated as common wart, but they did not resolve. There was no one in the family who was similarly affected. There was no spontaneous resolution in the period of observation.Physical examination revealed multiple, skin to tan-colored, flat-topped papules, 1 to 3 mm in diameter, on the forearm, thigh, neck, anterior chest, upper abdomen Figures . The lesSyringoma is a benign adnexal tumor that derives from the intraepidermal portion of the eccrine sweat ducts. Syringomas first appear during puberty or the third or fourth decade.45 Althou4Our case was a 28-year old woman who had lesions of generalized eruptive form. The patient presented with an eight-year history of a dermatosis. The lesions appeared on the forearm first, which was followed by successive eruptions on the face, chest, upper abdomen, thigh and neck respectively. The patient had mild pruritus during perspiration. There was no spontaneous resolution in the period of observation.2 laser, with variable success; however, none eliminates the risk of recurrence and, therefore, treatment of syringoma is often frustrating.[Treatment of syringoma is cosmetic. Therefore, it should not be confused with the above referred differential diagnosis. Options are abundant and generally unsatisfactory, as they are located in the dermis and often numerous. Physical techniques such as excision, electrocoagulation and liquid nitrogen cryotherapy and dermabrasion yield poor cosmetic results.78 Oral i7strating. Most of strating.–12 One sstrating. UnfortunTreatment of syringoma is cosmetic. They are abundant and generally unsatisfactory. Treatment modalities have included dermabrasion, various methods of excision, cryosurgery, electrodesiccation, chemical peeling, and oral and topical retinoids.–10 Succe


Spontaneous splenic rupture considered a relatively rare but life threatening. The three commonest causes of spontaneous splenic rupture are malignant hematological diseases, viral infections and local inflammatory and neoplastic disorders. We describe a unique and unusual case of inflammatory myofibroblastic tumor of the tail of pancreas presented with massively enlarged spleen and spontaneous splenic rupture.A 19 years old male patient with no significant past medical history presented to emergency room with abdominal pain and fatigue. Massively enlarged spleen was detected. Hypotension and rapid reduction of hemoglobin level necessitated urgent laparatomy. About 1.75 liters of blood were found in abdominal cavity. A large tumor arising from the tail of pancreas and local rupture of an enlarged spleen adjacent to the tumor were detected. Distal pancreatectomy and splenectomy were performed. To our knowledge, we report the first case of massively enlarged spleen that was complicated with spontaneous splenic rupture as a result of splenic congestion due to mechanical obstruction caused by an inflammatory myofibroblastic tumor of the tail of pancreas. A review of the literature is also presented.Inflammatory myofibroblastic tumor of the tail of pancreas should be included in the differential diagnosis of the etiological causes of massively enlarged spleen and spontaneous splenic rupture. The most common causes of splenomegaly are liver diseases 33%), hematologic malignancies 27%), infections (23%), congestion or inflammation (8%), primary splenic diseases (4%) and others (5%) . Cirrhos7%, infec3%, hemat3, platelet count 271000/mm3. Other laboratory analysis including potassium, sodium, calcium, magnesium, phosphorus, blood urea nitrogen, creatinine, serum amylase, lipase, and liver chemistry were all within normal range. Five hours later, blood pressure dropped to 86/55 and reduction of hemoglobin level to 5.9 gr/dl was detected. These findings considered indications for urgent explorative laparotomy. Sudden massive bleeding may cause acute hypovolemic shock even without reduction in the hemoglobin level. The patient underwent an urgent explorative laparotomy. About 1.75 liters of blood were found in abdominal cavity. A large tumor arising from the tail of pancreas and local rupture of an enlarged spleen adjacent to the tumor were detected. Distal pancreatectomy and splenectomy were performed. The postoperative course was without any remarkable complications. Macroscopic pathology revealed a cystic mass measuring 8.2×6.5×6.0 cm in the tail of the pancreas and huge spleen measuring 23.5×15.5×6.3 cm  scan of the abdomen showed massive splenomegaly and a solid mass with hypodense area in the tail of the pancreas (Figure IMT is a histopathologic entity previously known as an inflammatory pseudotumor which was initially reported in 1990 in the pulmonary system . DiffereAlthough IMT occurs more frequently in the pulmonary system but it had been described in a wide variety of other organs . In a clIMT of the pancreas is rare. Only 27 cases of IMT located in the pancreas have been reported in English literature ,6,26-43.The presenting symptoms of the reported patient were abdominal pain and fatigue as a result of massively enlarged spleen and anemia due to substantial congestion of the spleen resulted from the location of the IMT in the tail of pancreas that cause a considerable obstruction of the blood drainage from the spleen. The abdominal pain and the anemia caused mainly by the huge palpable spleen rather than by the tumor itself. Anorexia, nausea, vomiting, weight loss or jaundice, were not included in the presenting symptoms of our patient. It was reported that one patient with IMT located in the body and the tail of the pancreas developed splenic vein thrombosis resulting in splenomegaly, thrombocytopenia and upper gastrointestinal hemorrhage from isolated gastric varices . The preThe tumor was located in the head of the pancreas in 57.7% of cases, whereas it was found in the body and the tail of the pancreas in 42.3%. IMT of the pancreas has a tendency to be a larger in size than that in the pulmonary system at the time of diagnosis ranging 1.5-13 cm . The sizComplete surgical resection is the treatment of choice for IMT without any need of chemotherapy and radiation therapy ,44,45. TBecause of a higher recurrence rate, approaching 25% in extrapulmonary IMT, our patient has been closely followed with no clinical or radiographic evidence of recurrence for 6 years following surgery.Inflammatory myofibroblastic tumor of the tail of pancreas should be included in the differential diagnosis of the etiological causes of massively enlarged spleen and spontaneous splenic rupture.The authors declare that they have no competing interests.KH participated actively in the diagnosis process, following up the patient, preparing, writing and revising the literature and the manuscript. HC is the pathologist that carried out the pathological diagnosis, edited and revised the figures' legends. FH participated actively in preparing, writing, editing, printing and revising the manuscript. HS participated actively in following up the patient, reviewing the literature, preparing, editing and revising the manuscript. All authors read and approved the final manuscript.


Patent paraumbilical and abdominal subcutaneous veins are found frequently as collaterals in patients due to portal hypertension mainly in liver cirrhosis.For evaluation of portal hypertension in a 72-year-old Caucasian man without liver cirrhosis, magnetic resonance imaging with gadolinium contrast-enhancement was performed and demonstrated a missing inferior vena cava. A blood return from the lower extremities was shown through enlarged collateral veins of the abdominal wall, vena azygos and hemiazygos continuation, and multiple liver veins emptying into the right cardiac atrium. We describe a rare case of abdominal subcutaneous wall veins as collaterals caused by a congenitally absent infrarenal inferior vena cava with preservation of a hypoplastic suprarenal segment.Knowledge of these congenital variations can be of clinical importance and it is imperative for the reporting radiologist to identify these anomalies as they can have a significant impact on the clinical management of the patient. Congenital malformations of the inferior vena cava (IVC) are rare. Patients are usually asymptomatic and this developmental anomaly is detected incidentally during abdominal surgery or radiologic evaluation. Patent paraumbilical and abdominal subcutaneous veins are found frequently as collaterals in patients due to portal hypertension, mainly associated with liver cirrhosis ,2. In thA 72-year-old Caucasian man reported no symptoms at a preoperative evaluation before hip joint replacement. Upon his admission, abnormal laboratory parameters included erythrocyte sedimentation rate (ESR) 29/73mm/hour  and total cholesterol 233 mg/dL ; all other routine laboratory parameters including those for liver function, were within normal limits. During clinical examination, a distended patent abdominal wall and paraumbilical veins were noted. An additional abdominal sonography revealed no signs of liver cirrhosis. During anamnesis, he reported a history of deep vein thrombosis in both lower limbs at age 50, and recurring thrombosis at age 62 of unknown etiology.Magnetic resonance imaging (MRI) of the abdomen with gadolinium contrast enhancement was performed and the infrarenal IVC could not be demonstrated Figure . The MRIThe hip replacement was performed successfully using anticoagulation with low-molecular weight heparin; he was subsequently anticoagulated with coumarin and compression stockings were prescribed. Although familial clustering of congenital anomalies of the IVC is known, his family members refused further investigations. Written informed consent was obtained for all procedures, which were in accordance with the Declaration of Helsinki and the recommendations of the local ethics committee.The umbilical vein is obliterated a few days after birth and is usually not part of the actual circulation. Patent paraumbilical and abdominal subcutaneous veins are found frequently as collaterals due to portal hypertension in liver cirrhosis or with abdominal and retroperitoneal tumors . CongeniThe IVC develops between the sixth and eighth week of embryonic life as a composite structure formed from the appearance, regression, and anastomosis of three paired embryonic veins: the posterior cardinal, subcardinal, and supracardinal veins. A normal IVC is composed of four segments which are hepatic, suprarenal, renal and infrarenal. Variations of IVC anatomy are classified in a system based on abnormal regression and/or persistence of various embryonic veins . We descThe course and number of collateral veins are variable and, due to inadequate collateral circulation, this results in venous stasis and an increased risk of DVT. Congenital anomalies of the inferior vena cava are described as a rare cause of thrombotic occlusions in iliac veins usually in patients 30 years of age and younger. In medical outpatients, the mean age of DVT (± SD) was reported as 59.1 ± 17.3) years, which compares well to the age DVT occurred and routine management for DVT was performed in this patient. As commonly recommended for DVT, oral anticoagulation in this patient was discontinued three to six months after both the first and second occurrence of thrombosis. However, after thrombosis is caused by absence of IVC, there is a reportedly high risk of recurrent thrombosis ,7..3 years,Nowadays, vascular anomalies can well be identified with venography, improved contrast-enhanced computed tomography, and high resolution magnetic resonance imaging methods ,8,10,11.Written and informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.The authors declare that they have no competing interests.WJS and RWL conceived of and designed the study. WJS and PR analyzed and interpreted the data. WJS drafted the article and, along with ET and SJW, critically revised it for important intellectual content. WJS, ET, SJW, RK and RWL approved the final article. WJS, RK and RWL provided administrative, technical, or logistic support. WJS collected and assembled the data. All authors have read and approved the final manuscript.


Sir,Ileosigmoid knot is an unusual clinical entity of small bowel obstruction in which the ileum wraps around the base of the sigmoid colon and forms a pseudo knot. We present a case of a 65-year-old man in whom the diagnosis of ileosigmoid knotting was made via emergency surgery.Our patient was a 65-year-old man who arrived at the Emergency Department and was admitted with profound shock. He was complaining of acute worsening abdominal pain, vomiting and constipation for 2 days. On admission, the patient was febrile at 38°C, with a pulse rate of 50/min and blood pressure of 70/50 mmHg. Abdominal examination revealed a mildly distended abdomen with diffuse tenderness and positive rebound tenderness. No abdominal mass was palpable. His rectal examination was negative for blood. We treated the patient with intravenous normal saline and ordered laboratory work-up for chemistry, coagulation profile and Hem studies.3) and blood urea of 0.8mg/ml. An abdominal X-ray showed small bowel air-fluid levels. A bedside sonography revealed free peritoneal fluid in the perihepatic and perisplenic spaces and between the distended intestinal loops. Because of the patient's instable status, a computerized tomography (CT) scan was deferred and the patient was taken to the operating room for an urgent laparotomy.Abnormal laboratory parameters were mild anemia (Hb: 11g/dl), with leucocytosis  was observed. A sigmoid loop was found tightly encircling the congested and dilated loops of the ileum forming a knot. The sigmoid was not necrotic and was not resected because of the condition of the patient. Three meters of the distal ileum was frankly necrotic. No perforation was visualized. The 3 m of distal ileum was resected with cecum. A termino-lateral ileo-colic anastomosis was performed because of the shortness of the small intestine. We planned an elective sigmoidectomy at a later date.Ileosigmoid knot is an uncommon lesion; however, it is a well-recognized condition in certain African, Asian and Middle Eastern nations. It was first reported by Parker in 1845. Its incidence in the general population is not known, but it is common in adult males and the peak incidence is in the fourth decade.[Although the basic requirements for an ileosigmoid knot are still controversial, most radiologists agree that they include a hypermobile small intestine with markedly elongated mesentery and a redundant omega-shaped sigmoid colon with a long mesocolon and a short attachment at the base of the mesentery. Alver performed a new classification for ISK. Type I: The ileum (active component) wraps itself around the sigmoid colon (passive component) in a clockwise or counterclockwise direction. Type II: The sigmoid colon (active component) wraps itself around a loop of the ileum (passive component) in a clockwise or counterclockwise direction. Type III: The ileocecal segment (active component) wraps itself around the sigmoid colon (passive component). In most cases, the ileum (active component) wraps around the sigmoid colon rather than the reverse. In our case, we noted an omega-shaped sigmoid encircling the small intestine, corresponding to Type II Alver's classification.The clinical features of ileosigmoid knot present with acute severe abdominal pain due to ischemia of the bowel associated with vomiting and abdominal distention and the in case of any delay in diagnosis, the general condition of the patient will deteriorate rapidly.Characteristic radiological features including a double closed loop obstruction, with the small intestinal loops in the upper left quadrant and the sigmoid loop in the right, are ofteThe mortality rate is high, 25-47%, mainly dWe conclude that the ileosigmoid knot is an unusual clinical entity of small bowel obstruction. It is a serious condition that rapidly progress to gangrene. Thus, early diagnosis and prompt treatment are essential despite difficulty in making a preoperative diagnosis.


Elephantiasis neuromatosa is the most impressive manifestation of neurofibromatosis type-1 (NF-1). We report a case of NF-1 who presented with elephantiasis neuromatosa of his right leg. Cross-sectional imaging not only assists in the correct diagnosis but also aids in imaging the vasculature of a plexiform neurofibroma, which is essential for proper surgical planning. Neurofibromatosis type 1 (NF-1), previously known as Von Recklinghausen disease, is a phakomatosis or neurocutaneous syndrome with autosomal-dominant inheritance, primarily affecting the development and growth of nerve cell tissues, with a frequency of approximately 1 in 3,000 births. PlexiforA 15-year-old male child with known NF-1 was referred to our radiology department for detailed assessment of a large disabling right leg plexiform neurofibroma that had recently undergone skin ulceration and had affected the patient’s gait. This slow-growing plexiform neurofibroma since childhood had resulted in gross limb enlargement and disfigurement , necessiConventional radiograph showed relative lengthening and marked soft tissue hypertrophy of the right leg. Osseous abnormalities included thinning of bones, erosion of distal articular surfaces and periosteal dysplasia . Gray scScreening MRI of the brain showed a well-defined lesion in the left basal ganglia, particularly involving the internal capsule, adjoining part of the globus pallidus and the thalamus. The lesion demonstrated mixed hypo- and hypersignal intensity on T1WI, high signal intensity on T2W/FLAIR sequences and no enhancement after contrast administration. The lesion typically did not exhibit mass effect, edema or contrast enhancement, suggesting non-neoplastic hamartoma .The patient underwent partial tumor resection and cosmetic repair of the right leg. He was also advised follow-up MRI of the brain for hamartomatous lesion, which normally shows spontaneous regression with time.The histopathologic evaluation of the resected mass revealed a non-capsulated lesion comprising a large number of irregular infiltrative spindle cells and several nerve segments of varying length embedded in subcutaneous fat. The nerves were surrounded by a myxoid stroma that extended into the papillary dermis. Many mast cells were also seen within the diffuse component of the tumor. However, no mitosis was detected. The findings were suggestive of diffuse plexiform neurofibromatosis .NF-1 is a hamartomatous disorder, with the genetic defect localized to the long arm of chromosome 17q11.2.Plexiform neurofibroma, found in up to 26% of patients with NF-1 is considered an uncommon skin tumor, usually presenting at birth or during the first several years of life. They are unencapsulated, poorly circumscribed tumors that diffusely infiltrate the nerve and the adjacent fat and muscle. As a result, neurofibromas are usually unresectable tumors, where tumor resection is impossible without sacrificing the nerve tissue. Fusiform enlargement of multiple nerve fascicles and branches is characteristic. Plexiform neurofibromas contain a mixture of Schwann cells, fibroblasts, reticulin and collagen fibers and a loose mucoid matrix interspersed between the axons of the parent nerve. They typically affect the trunk and extremities, but may also involve the head-neck and bladder. Associated bone dysplasia is often encountered secondary to chronic hyperemia or as part of the mesodermal dysplasia. Such tumors give rise to a variety of problems, including disfigurement and functional impairment.[On ultrasound, these masses are usually hypoechoic, well defined and elliptical, oriented along the long axis of the nerve. Posterior acoustic enhancement is very common 70%). Color Doppler reveals variable vascular patterns, ranging from moderate, irregular central to predominantly peripheral vascularity. Some may not have any demonstrable vascularity.0%. Color The typi394The radiological modalities most often used in analyzing neurofibroma include CT and MRI. Ultrasound and color Doppler has a very limited role in the evaluation of a large mass, extending outside the range of the probe. Although CT is rarely helpful in making a specific diagnosis, it can provide a precise evaluation of the bone lesion and the extent of the soft tissue lesion; however, it is by far inferior to MRI in soft tissue contrast resolution and the visualization of tissue planes. Dynamic contrast-enhanced 3D MR or CT angiography represents a recent advance in imaging with rapid acquisition of high-quality angiographic images that permit a free choice of imaging planes and phases delineating the arterial and venous supplies, visualization of the abnormal changes of the vasculature in the affected limb, important landmarks in surgical planning. It should be emphasized that MRI and MR angiography may assist not only in the correct diagnosis of neurofibroma but also in imaging the vasculature of a plexiform neurofibroma, which is essential for proper surgical planning.Treatment of diffuse and progressive plexiform neurofibroma is primarily surgery. However, complete resection of the tumor is not possible because of marked entanglement of the tumor with the nerves. Improved understanding of the molecular and cellular biology of the cells involved in the formation and growth of neurofibromas has lead to development of other forms of treatments, including drug therapies, whose role is yet to be defined.The common differential diagnosis in this case is other soft tissue tumors causing elephantiasis, such as filariasis, macrodystrophia lipomatosa, lymphangiomatosis, vascular malformation such as hemangioma and massive subperiosteal hematoma.There are many imaging modalities available today to study the peripheral nerves. It should be emphasized that MRI and angiography (MR or CT angiography) may assist not only in the correct diagnosis of neurofibroma but also in imaging the vasculature of a plexiform neurofibroma, which is essential for proper surgical planning.


Schwannoma is a rare tumor among pancreatic neoplasms. Schwannomas vary in size, and most of them are cystic, mimicking pancreatic cystic lesions. Generally, a definitive diagnosis is made at the time of histological analysis. The mainstay treatment is surgical resection.We report an unusual presentation of pancreatic schwannoma with abdominal pain and several episodes of cholangitis in a 54-year-old Caucasian (Iranian) man. The condition was not diagnosed pre-operatively and Whipple's procedure was performed.Pancreatic schwannoma is an important clinical entity to include in the differential diagnosis of pancreatic lesions. Pre-operative diagnosis is difficult but computed tomographic findings may be helpful. The tumor may also have atypical and rare presentations, such as cholangitis and weight loss. For benign tumors, simple enucleation is usually adequate, whereas malignant tumors require standard oncological resection. Schwannomas are uncommon neoplasms. They are sometimes also referred to as neurilemmomas and usually occur in the extremities, but can also be found in the trunk, head and neck, pelvis, and rectum . Benign A 54-year-old Caucasian (Iranian) man presented at our hospital with a history of intermittent epigastric pain and several episodes of cholangitis over the previous year. The pain ranged from moderate to severe and was associated with weight loss, nausea, vomiting, and intermittent jaundice. On physical examination, he was febrile  and mildly icteric. His abdomen was soft, non-distended, with no palpable mass, but mildly tender to palpation in the epigastrium.On laboratory tests, he showed mild direct bilirubinemia , but normal hemoglobin, liver function, and amylase. The tumor markers carbohydrate antigen (CA) 19-9 and carcinoembryonic antigen (CEA) were in the normal ranges.A dynamic computed tomographic (CT) scan demonstrated a 30 × 2.5 mm mass in the pancreatic head area. Upper gastrointestinal (GI) side-view endoscopy showed a normal papilla of Vater. Based on the patient's history, clinical findings, and CT images, he was taken to the operating theater for the Whipple procedure.With laparotomy, a mass was found in the head of the pancreas, without encasement of the superior mesenteric artery or portal vein. No mesenteric lymphadenopathy, peritoneal implants, or liver lesions were found. A classic pancreatoduodenectomy was performed. The pathological analysis showed a schwannoma consisting of spindle cells arranged in a palisading fashion Fig. . ImmunohTen months after the operation, the patient was completely symptom free.Less than 26 cases of pancreatic schwannoma have been reported in the literature. They vary in size from 1.5 cm to 20 cm in diameter. The majority of these tumors were found in the head and body of the pancreas. Generally, they were slow growing and originated from the peripheral epineurium of either autonomic sympathetic or parasympathetic fibers . More thNonspecific abdominal pain was the most commonly reported symptom but weight loss, jaundice, and GI bleeding were also reported . There iThe diagnosis of pancreatic schwannoma may be complicated by an inadequate amount of specimen or by deficiencies in the specimen-collecting techniques used. Fine-needle aspiration correctly diagnoses only one of eight histologically proven schwannomas . PancreaPancreatic schwannoma is an important clinical entity to include in the differential diagnosis of pancreatic lesions. Pre-operative diagnosis is difficult but CT findings may be helpful. The tumor may also have atypical and rare presentations, such as cholangitis and weight loss. For benign tumors, simple enucleation is usually adequate, whereas malignant tumors require standard oncological resection.Written informed consent was obtained from the patient for the publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.The authors declare that they have no competing interests.AMT, MH, BNH, and FS were all involved in the management of the patient, and in writing the case report. All authors have read and approved the manuscript.


Thymic seminoma is a rare clinical entity. We report a case of an 18-year-old male patient who presented with chest pain, dyspnea, dysphagia of 1-month duration. Contrast-enhanced computer tomogram of chest showed an anterior mediastinal mass. He was subjected to thoracotomy and excision of the mass. Histopathology examination revealed seminoma of thymus. He failed to follow up for a period of 8 months post-surgery as he was asymptomatic. He then presented with a recurrence of the tumor locally, along with metastases to the lung, para-aortic and peri-aortic lymph nodes. He was kept on cisplatin, bleomycin and etoposide. Seminoma of thymus should be regarded as a germ cell tumor rather than a true thymoma. It affects only young males. It grows very slowly and can become large before the appearance of symptoms. Differential diagnosis includes thymoma, lymphoma, ectopic thyroid, ectopic parathyroid, mesenchymal and neurogenic tumors. Chemotherapy plays a major role in the management of this neoplasm.An 18-year-old male patient presented in September 2006 with dull retrosternal and right-sided chest pain, dyspnea and dysphagia for solids of 1-month duration. Chest X-ray showed lobulated homogenous opacity occupying the right mid-zone and lower zone . There wCECT chest showed anterior mediastinal mass with extension into right side of chest . Image-gHe presented in June 2007 with dull-aching retrosternal and right-sided chest pain with exertional dyspnea and dysphagia for solids. On examination, there was impaired note to percussion over the right mammary and infra-axillary regions with diminished breath sounds. A 3 ×3 cm mass was palpable over the epigastric region. Both testes were normal. Chest X-ray showed superior mediastinal widening with homogenous opacity in the right mid and lower zone . CECT chContrast enhanced Computed tomography (CECT) abdomen showed extrinsic compression of liver by subpleural metastases with multiple para-aortic and peri-aortic lymphadenopathies . USG . After four courses of chemo, complete disappearance of the mediastinal mass and lung metastatic lesions and reduction of abdominal lymph nodes to sub-centimeter size were noted Figures –11.Malignant mediastinal germ cell tumors are uncommon, representing only 3% to 10% of tumors originating in the mediastinum. Other anGreat majority of mediastinal germ cell tumors occur in patients between 20 and 35 years of age. Mature cystic teratoma is the most common benign mediastinal germ cell tumor and affects both sexes equally. Seminoma and non-seminomatous germ cell tumors show male predilection.Mediastinal germ cell tumors arise as a consequence of abnormal migration of germ cells during embryogenesis. The cyto6p53 IHC was observed in 31% of mediastinal seminomas and 77% of testicular seminomas. Eight percent of mediastinal seminomas showed K-ras mutation , which is in contrast to 15% of testicular seminomas showing codon 12 mutations. Therefore, primary mediastinal seminomas appear to be different in their K-ras sequence and p53 immunostain profile from testicular seminomas. Codon mutation type may be useful in determining primary versus metastatic origin of a mediastinal seminoma.[The morphologic appearance is identical to that of its testicular counterpart at the light microscopic, immunohistochemical and ultrastructural levels.9 Focal wseminoma.The presence of fibrous septa infiltrated by lymphocytes and plasma cells, epitheloid granulomas, germinal centers, large amounts of cytoplasmic glycogen and an irregular skein-like nucleolus favors the diagnosis of seminoma. The true nature of the lesion may be obscured by the presence of a very prominent granulomatous reaction, reactive follicular hyperplasia, epithelium-lined cystic formation of thymic origin and fibrosis.It is immunoreactive for placental alkaline phosphatase, CD 117 and often CD 57, but negative for CD 45 and usually negative or focally positive for cytokeratin.Approximately 20% to 35% of seminomas are detected by routine chest radiography while still asymptomatic. The most common initial symptom is a sensation of pressure or dull retrosternal chest pain. Other symptoms include exertional dyspnea, cough, dysphagia, hoarseness of voice. Superior vena cava (SVC) syndrome develops in approximately 10% of the patients.At the time of diagnosis, only 30% to 40% of patients with mediastinal seminoma have localized disease. The lungs and other intrathoracic structures are the most common metastatic sites. The skeletal system is the most frequently involved extrathoracic metastatic site, while the retroperitoneum is an uncommon site of metastases in these patients with mediastinal seminoma.They appear as large anterior mediastinal mass without calcification in chest X-ray. A CT scan of the chest typically shows a large homogenous anterior mediastinal mass that obliterates the fat planes surrounding mediastinal vascular structures.Elevated serum levels of β human chorionic gonadotropin are detected in approximately 10% of mediastinal seminomas. A level of serum β human chorionic gonadotropin exceeding 100 ng/mL is unusual. Serum alpha fetoprotein level is always normal in pure mediastinal seminoma. Serum lactate dehydrogenase is also elevated in the majority of patients with mediastinal seminoma.If distant metastases or obviously unresectable intrathoracic tumor is present, a histological diagnosis should be made using the least invasive approach, because surgical therapy does not play a role in the initial treatment of these patients, and rapid initiation of definitive systemic therapy is essential. Patients with small tumors that appear resectable should undergo thoracotomy and attempted complete resection; postoperative radiotherapy 40 to 45 Gy is curative.Pure mediastinal seminomas are curable in majority of the patients. These tumors are highly sensitive to radiation therapy and to combination chemotherapy, and selection of treatment therefore depends on disease stage and size of mediastinal tumor.


Leser-Trélat syndrome is characterized by the eruptive appearance of multiple seborrheic keratoses in association with underlying malignant disease. Usually, the sign of Leser-Trélat is associated with adenocarcinoma, most frequently of the colon, breast, or stomach, but also of the lung, kidney, liver, and pancreas. Herein, we present a case that we believe is the first report of the sign of Leser-Trélat in association with occult gastric adenocarcinoma and the anamnestic oncologic history of five other multiple primitive cancers. Epidermal growth factor receptor (EGFR) immunohistochemical expression analysis of multiple seborrheic keratoses revealed an intense membranous staining in the basal keratinocytes and in all the upper epidermal layers. Patients with the sign of Leser-Trélat should undergo a diagnostic screening programme for malignant disease along with patients with known Leser-Trélat syndrome who present with a recent acute and florid eruption of their seborrheic keratoses. We propose the importance of combining the molecular features of multiple seborrheic keratoses with EGFR immunohistochemistry analyses to determine the likelihood of Leser-Trélat syndrome and the consequent high risk of underlying multiple visceral malignancies. Skin manifestations are frequently observed in many internal malignancies. Cutaneous lesions, including benign neoplasms or pigmented lesions, can appear in the context of specific genodermatosis ,2, lentiSeveral cutaneous paraneoplastic syndromes may be associated with underlying tumours. They include musculoskeletal disorders , reactive erythemas (erythema gyratum repens and necrolytic migratory erythema), vascular dermatoses (Trousseau's syndrome), papulosquamous disorders , and disorders of hair growth (hypertrichosis lanuginosa acquisita) Table  4]. The. The4]. The sign of Leser-Trélat is characterized by the eruptive appearance of multiple seborrheic keratoses (SK) in association with underlying malignant disease. Two European surgeons, Edmund Leser and Ulisse Trélat, were the first to describe the sign, and although they gave no mention of the presence of seborrheic keratoses reporting a specific association with angiomas - that probably does not exist - their names continue to be linked to a different paraneoplastic association . In 1900Endogenous mediators of hyperproliferative skin disease such as epidermal growth factor (EGF), transforming growth factor-alpha (TGF-α), or amphiregulin that may function in a localized autocrine manner or be found in the circulation affecting the epidermis more diffusely, may induce some seborrheic keratoses ,10.Herein, we present a case that we believe is the first report of Leser-Trélat syndrome in association with occult gastric adenocarcinoma and the anamnestic oncologic history of five other multiple primitive cancers.A 83-year-old man was admitted to our hospital in August 2009 with a recent history of dyspnea, cough and dyspepsia. His medical history was characterized by sigmoidectomy and ileal-cecal resection for two synchronous adenocarcinomas T1N0M0) in August 2008; localized adenocarcinoma of the prostate diagnosed in 2007 treated with hormonotherapy and radiotherapy with radical intent; cutaneous basal cell carcinoma and squamous cell carcinoma removed about 10 years before. For the past 25 years, he had noted multiple asymptomatic lesions, diagnosed as multiple seborrheic keratosis, of the face and trunk and these had recently increased in size and number with generalized pruritis. On admission, dermatologic examination noted florid eruption of innumerable keratotic lesions of the face, neck and thorax, ranging in size from 3 to 15 mm  of the chest and abdomen revealed a 2.5 by 2.0-cm solid mass in the upper lobe of his right lung. Diagnosis of gastric adenocarcinoma was obtained at histological analysis by lung biopsy which revealed signet ring cells, and at clinical evaluation of his gastric symptoms . Contemporaneously, due to haematologic pancytopenia, the patient was studied by bone marrow biopsy (BOM) that revealed a diffuse infiltration of adenocarcinoma with signet ring cells at histological analysis. The patient died of disease progression in September 2009.The patient's sister had died of neck cancer at the age of 50 years. There was no history of malignancies in the patient's mother, father or other first-degree relatives.The clinical association of florid eruption of seborrheic keratoses and diagnosis of malignancy clearly identify Leser-Trélat syndrome. Heaphy et al.  suggest Our report appears to demonstrate that, even when the sign of Leser-Trélat is confirmed by the diagnosis of anamnestic or recent malignancies, patients affected by this syndromic disorder should be carefully monitored for different visceral and potentially multiple primitive malignancies. This instrumental surveillance strategy should be tailored taking into account either the novel sudden eruption of SK or the size and change in number of known SK. Regarding the type of associated malignancies, the sign of Leser-Trélat is usually associated with an adenocarcinoma, often in the stomach or colon, although squamous cell carcinoma, lymphoma, and leukaemias may also be observed. However, about 20% of patients reportedly have a lymphoma or a leukaemia rather than an adenocarcinoma. The distribution of the underlying cancer is similar to that of malignant acanthosis nigricans, that is, adenocarcinomas predominate, especially in the stomach and the colon.Seborrheic keratoses are exceedingly common and various authors have doubted the existence of anything other than a chance or coincidental association in the sign of Leser-Trélat . In otheThere is some evidence for a humoral link between various malignancies and the rapid onset of paraneoplastic syndrome. Ellis et al.  identifiImportantly, a question to consider is whether, given the relative frequency of such lesions, routine immunohistochemical (IHC) testing may be warranted for any multiple SK. We propose that EGFR immunohistochemical analysis indicates the presence of LTS only when some additional clinical information is present . In these settings, the SK sign should be carefully examined through IHC tests even when they appear in individuals without evidence of visceral malignancy. Systematic analyses of EGFR IHC expression in multiple SK of patients affected by LTS will provide more information on the role of concomitant hormonal alterations underlying multiple SK and visceral cancerogenesis.In the literature, only a few cases of LTS have been reported ,9,12. InPatients with sign of Leser-Trélat should undergo a diagnostic screening programme for malignant disease along with patients with known Leser-Trélat syndrome who present with a recent acute and florid eruption of their seborrheic keratoses. We propose the importance of combining the molecular features of multiple seborrheic keratoses with EGFR immunohistochemistry analyses to determine the likelihood of Leser-Trélat syndrome and consequent high risk of visceral cancer development.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.The authors declare that they have no competing interests.GP and GL were responsible for the clinical management of the patient, acquisition of data, and drafting the manuscript; LL was responsible for histologic diagnosis and EGFR immunohistochemical analysis and discussion; AG was involved in discussion of the manuscript; SS was supervisor of clinical management of the patient and interpretation of the data. All authors read and approved the final manuscript.


Intussuception is a rare cause of intestinal obstruction in adults. Diagnosis is often difficult due to the variable and sometimes episodic nature of symptoms. Surgery is the recommended treatment option in adults if the diagnosis is proven.We present a case of a 33 year old Caucasian female admitted with a small bowel obstruction and no history of previous abdominal surgery. Patient did not improve with medical management consisting of bowel rest and nasogastric tube decompression. Surgery was consulted and patient was taken to the operating room for a laparoscopic-assisted small bowel resection for a small bowel intussusception caused by a submucosal fibroma.Our case highlights the feasibility and potential benefits of laparoscopy in assisting the diagnosis and treatment of small bowel obstructions. Although fairly common in children, adult intussusception is relatively rare representing only 1% of patients with bowel obstructions . IntussuDiagnosis of adult intussusception is difficult secondary to the variable symptoms that can be acute, intermittent, or chronic. Computed tomography has proven to be a valuable diagnostic tool, with an accuracy rate of 78% [A 33 year old Caucasian female was admitted to the medicine service at our institution with an 18 hour history of abdominal pain, nausea, vomiting, and diarrhea. Patient had a strong family history of Crohn's disease including a sister that had onset of the disease at a similar age. Gastroenterology was consulted and a computed tomography scan was obtained showing edematous small bowel within the pelvis and a questionable internal hernia. Patient was treated medically for a small bowel obstruction including bowel rest, intravenous fluids, and nasogastric decompression without symptomatic relief. A repeat computed tomography scan was obtained four days after admission showing a possible transition point for her small bowel obstruction and mass like opacity possibly representing intussusception versus an internal hernia (Figure Surgery was consulted for evaluation and patient was counseled regarding risk, benefits, and alternatives of diagnostic laparoscopy, possible bowel resection, and laparotomy. The patient was taken to the operating room on hospital day five and underwent a diagnostic laparoscopy with specific attention to the right lower quadrant. Initial access was obtained via an open cutdown technique at the umbilicus. Three additional 5 mm ports were placed. Running of the small bowel was started at the cecum and progressed proximal from the ileocecal valve. Just proximal to the ileocecal valve an area of obstruction was identified, possible relating to an internal hernia Figure . This arThe patient tolerated the procedure well and was discharged home on postoperative day four with normal bowel function tolerating a regular diet. Patient was seen in follow-up doing well and without complaints Figure .Surgery has been the mainstay of treatment for adult intussusception owing to the high association with malignancy as the underlying cause ,8. AbdomRecently, minimally invasive techniques have been applied to the treatment of small bowel obstructions, specifically to the diagnosis and treatment of adult intussusception. Both laparoscopic and laparoscopic-assisted small bowel and colonic resections have been reported for both benign and malignant disease ,10. TherFirst, diagnostic laparoscopy may assist in the diagnosis of intussusception in cases such as ours where the diagnosis is suspected but not confirmed by preoperative workup. If the diagnosis is confirmed laparoscopically then appropriate surgical therapy and resection can be performed depending on the comfort level of the surgeon. Secondly, laparoscopy may aid in planning the incision if a laparoscopic-assisted or even laparotomy incision is required. In our case, we were unable to truly distinguish whether the obstruction in the right lower quadrant was due to intussusception or an internal hernia. However, the ability to mobilize the cecum and bring it up through a small periumbilical incision allowed for the appropriate diagnosis and ability to perform an extra-corporeal anastomosis. If laparoscopy had not been used in our case, then a much larger lower midline or even full laparotomy incision may have been needed to diagnosis and treat this distal small bowel intussusception.Our case highlights diagnostic laparoscopy and laparoscopic-assisted bowel resection as a potential and feasible tool in the treatment of small bowel intussusception. The ability to confirm diagnosis and plan targeted small incisions for treatment make laparoscopy a viable treatment option in patients suspected of having intussusception.The authors declare that they have no competing interests.DS and WH prepared the manuscript and reviewed the relevant literature. All authors were involved in clinical care and revising of the manuscript. All authors read and approved the final manuscript.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.


Differential diagnosis with Von Meyenburg complex, biliary adenoma, biliary cistadenoma, congenital biliary cystsy, and hepatic benign cystic mesothelioma is provided. The occasionally reported expression of p53 in biliary adenofibroma has suggested that this tumour could represent a premalignant lesion. The absence of both cytological atypia and p53 immunoreactivity in our case confirms that BAF is a benign tumour with an indolent clinical behaviour. However, a careful histological examination of BAF is mandatory because malignant transformation of the epithelial component has been documented in two cases.We herein report the clinicopathologic features of a rare case of biliary adenofibroma (BAF) of the liver in a 79-year-old man. Grossly, tumour presented as a well-circumscribed, 5.5-cm mass with a solid and microcystic appearance. Histological examination was typical of biliary adenofibroma, showing a proliferation of variable-sized tubulocystic structures embedded in a moderately cellular fibrous stroma. Immunohistochemistry, revealing immunoreactivity of the epithelial component to cytokeratins 7 and 19, was consistent with a bile duct origin. Notably, the stromal cells had a myofibroblastic profile, showing a diffuse and strong expression of vimentin and  Two of t origin . Althougt origin , 3 with We herein report the clinicopathological features of a rare case of liver BAF with a benign clinical course after a 7-year follow-up period. Differential diagnostic and histogenetic considerations are discussed.A 79-year-old man complained of a vague abdominal pain. Ultrasound examination and computerized tomography revealed a solid 5.5 cm mass in the right lobe of liver. Blood tests, including alpha-fetoprotein, were unremarkable. No lymphadenopathy was present. A partial liver resection was performed. No postoperative complications were noted. The patient is well with no recurrence after a 7-year follow-up period.μm, and stained with hematoxylin and eosin. Immunohistochemical studies were performed with the labeled streptavidin-biotin peroxidase detection system using the Ventana automated immunostainer . The antibodies tested are summarized in Surgical specimen was submitted for histological examination in neutral-buffered 10% formalin, dehydrated using standard techniques, embedded in paraffin, cut to 5 Grossly, surgical specimen consisted of liver parenchyma (14 × 10 × 4 cm) containing a 5.5-cm nodular mass merging from the liver capsule. The cut surface of the mass showed an unencapsulated, well-circumscribed, and firm lesion with a solid and microcystic appearance. Cysts ranged in size from 0.1 to 0.5 cm. Histologically, at low magnification, the tumour was composed of a proliferation of nonmucin-secreting, tubulocystic structures, embedded in a moderately cellular fibrous stroma . Both tuα-smooth muscle actin  suggesting a bile duct origin [α-smooth muscle actin. Notably, the cellular stromal component of liver BAF has been reported to be fibroblastic rather than myofibroblastic in nature, because only scattered α-smooth muscle actin+ cells have been identified [The present case has the morphological characteristic and immunohistochemical features of liver BAF. Grossly, tumour presented as an unencapsulated, well-circumscribed, 5.5 cm solid-cystic nodule. Histologically, it was composed of a tubulocystic proliferation of variable-sized bile ducts with an immunohistochemical profile (CK7t origin . The lumt origin . The epientified . As betaentified , we evalDifferential diagnosis of BAF includes von Meyenburg complex, bile duct adenoma, biliary cystadenoma, biliary cysts, and benign cystic mesothelioma. BAF generally presents as a large-sized nodule with a solid microcystic appearance, whereas von Meyenburg complex can be incidentally found either as single or multiple subcapsular nodules of small size, usually less than 5 mm in diameter . This coAs far histogenesis of BAF is concerned, it is noteworthy that histological features similar to BAF and von Meyenburg complex have been obtained in animal-model of aflatoxin-induced cholangiocarcinoma . These e


We report on a 62-year-old male, who had resection of a large ductal carcinoma in the body and tail of thepancreas. Four months later a metastasis was removed from the abdominal scar, and 14 months later anothermetastasis was removed from the anterior wall of the stomach. Moreover, he had a left mastectomy followedby radiation therapy for a primary ductal carcinoma and a transurethral resection of the prostate because ofbenign hyperplasia. A minor focus of primary highly differentiated adenocarcinoma was found in the chips.More than 6 years later, the patient is without any signs of recurrences.


Apocrine hydrocystomas arising in the external auditory canal are very rare. In this report a clinical case of apocrine hydrocystoma located in the cartilaginous part of the external auditory canal is presented.A 64-year-old Caucasian female patient presented with a solitary nodule, located in the outer part of the external ear canal after repeated episodes of external otitis. For diagnostic purposes, computerized tomography was used. The patient underwent an excisional biopsy of the mass via an intra-aural incision and the surgical specimen was sent for histopathologic examination.An apocrine hidrocystoma inside the auricular canal is uncommon. It can cause recurrent external otitis and conductive hearing loss and should be treated with wide local excision and reconstruction of the external auditory canal for diagnostic and therapeutic purposes. The lateral one-third of the external auditory canal is cartilaginous and covered with a thin epithelium, firmly bound to the perichondrium and with scant subcutaneous tissue . Hair foHidrocystomas are rare, benign skin adnexal neoplasms, sometimes presenting as cystic lesions . AccordiWe describe a patient with an apocrine hidrocystoma in the auricular canal. In the English literature, very few cases of apocrine hidrocystomas originating in the external auditory canal have been reported. A single, related case has been identified in the PUBMED database  of an apA 64-year-old Caucasian female presented to our department with slowly developing left side hearing loss and recurrent external otitis. Physical examination revealed a tender solitary, skin-colored, dome-shaped nodule arising from the inferior and posterior wall of the left external auditory canal that narrowed the external auditory meatus and canal figure . Pure toThe patient underwent, under local anesthesia, an excisional biopsy of the mass via an intra-aural incision. The mass was removed while preserving the osteocartilaginous canal wall and the integrity of the underlying tympanic membrane. The external auditory canal was packed with a Merocel wick to support the skin and to prevent canal stenosis for two weeks. The patient made an uneventful recovery and there was no recurrence during two years of follow-up.Histopathologic examination of paraffin sections of the specimen, stained with haematoxylin-eosin figure , revealeSkin adnexal neoplasms exhibit morphological differentiation towards one or more types of adnexal structures found in normal skin  and comprise a wide spectrum of benign and malignant tumours, including cylindromas, spiradenomas, trichoepitheliomas, trichoblastomas, sebaceous adenomas, sebaceomas, sebaceous carcinomas, syringomas, microcystic adnexal carcinoma, papillary eccrine adenomas, tubular apocrine adenomas, hidradenomas, poromas, digital papillary adenomas/adenocarcinomas, adenoid cystic carcinomas, syringoid/eccrine carcinomas, apocrine (axillary) carcinomas, eccrine and apocrine hidrocystomas, and admixtures of these entities . Most adMcCalmont  offered Most adnexal tumors are benign, but many have definite recurrence and malignant potential if incompletely excised. Therefore, the presence of any atypical or malignant features, such as asymmetric growth, jagged/infiltrative borders, irregular arrangement of neoplastic cells, cytonuclear atypia, increased mitotic activity, tumour necrosis, and superficial ulcerations in a given case should be accessed, and complete excision for lesions that are only partially sampled is recommended to facilitate complete evaluation and to prevent future recurrence and rare malignant transformation ,7.Both benign and malignant tumors grow along the path of least resistance, extending laterally out the meatus or medially through the tympanic membrane and into the middle ear . InvasivWe have described an unusual appearance of an apocrine hidrocystoma inside the auditory canal of a female patient that caused occlusion, conductive hearing loss and secondary otitis externa. Although diagnosis relies on biopsy with histologic examination, diagnostic imaging by CT-scan is necessary for defining the characteristics, extension of the lesion and for planning the therapeutic approach. Localized excisional biopsy is the treatment of choice for external auditory canal apocrine hydrocystomas via intra aural or rertoauricular approach while preserving the adjacent structures.ENT: ear, nose, throat; CT: computerized tomography; FNA: fine needle aspiration; PAS: periodic acid Schiff; CEA: carcinoembryonic antigen; EMA: epithelial membrane antigen; GCDFP-15: gross cystic disease fluid protein 15; HMWK: high molecular weight keratin; LMWK: low-molecular weight cytokeratin; SMA: smooth muscle actinWritten informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.The authors declare that they have no competing interests.CES was responsible for diagnosing and performing surgery on the patient. DGI drafted and prepared the manuscript. CEP was responsible for review of the literature. EID reviewed the patient's medical record in order to collect all the available information. JGB was involved in revising the article for intellectual details. AF performed the histologic examination of the surgical specimen. All authors read and approved the final manuscript.