Intraocular metastatic tumors have been increasingly reported in the recent past. Unlike choroidal metastasis, metastasis to retina is very rare and so far has been reported in very few case reports only.A 56 year-old male who presented with a history of adenocarcinoma of the cecum and underwent lap colectomy for the primary cecal tumor, received adjuvant chemotherapy for a year after surgery and decided to stop. He was also diagnosed with metastasis to liver and lung at this time. He presented with left eye pain, pressure and decreased vision suspicious for retinal metastasis from cecal primary lesion, 2 years after initial diagnosis. A mass of 5 x 10 mm was found on ophthalmoscopic examination and on ultrasound of the eye, in spite of normal results of MRI of the orbit. Palliative radiation therapy of the left eye resulted in decreased eye pressure and improved vision.As retinal metastasis carries a poorer prognosis due to higher risk of spread to central nervous system, the diagnosis of retinal metastasis in case of gastrointestinal cancers patients who present with vision changes should be made urgently. These patients should be thoroughly investigated with a synergistic approach of opthalmoscopic examination, ultrasound of the eye along with other imaging modalities like MRI of the orbit and just not MRI of orbit. Immediate action in the form of surgical or radiation treatments of the metastatic tumors of the eye should be instituted early on for a better prognosis. Majority of the retinal metastasis spreads from cutaneous melanoma; lung primaries in males and breast primaries in females constitute a minor category.3 We present a case report of retinal metastasis from colon adenocarcinoma and then review the few reported cases.However, the colorectal cancer as the third common cancer in the United States and third leading cause of death of cancer in both genders warrants a special attention. A cecal adenocarcinoma metastasizing to retina is described in this case report. A 56 year-old male with history of alcohol abuse was admitted to ICU at Prince George's Hospital Center (PGH) on 05/12/2013 with severe symptomatic anemia and found to be guaiac positive. He underwent colonoscopy and biopsy of the cecal polyp was consistent with well differentiated adenocarcinoma. He underwent laparoscopic assisted right colectomy on 05/19/2013. The pathology report of the resected colon was consistent with well differentiated adenocarcinoma with focal mucinous differentiation arising from the cecum and invading through the muscularis propria and focally into the sub serosa (size 3.7 cm). The proximal, distal circumferential and mesenteric margins were negative for invasive carcinoma. There was no lymphatic vascular invasion and no lymph node metastasis at the time of resection . The pathologic staging of the primary colon cancer was T3 N0 Mx. He received adjuvant chemotherapy for primary colon cancer. In 01/2014 he was admitted to PGH for abdominal discomfort. He was evaluated in hospital for pancreatitis and further work up with MRCP on 01/16/2014 showed evidence of 3 lesions in liver and subsequent CT guided core biopsy of liver lesions showed metastatic mucinous adenocarcinoma consistent with colon cancer as the primary source. His PET-CT scan on 02/14/14 showed liver and lung metastases.The patient was advised to continue chemotherapy. However, the patient decided to hold off further chemotherapy and considered alternate regimens such as integrative oncology at Cancer Centers of Philadelphia. As per the medical records, patient stopped chemotherapy on 05/2014. Of note his DNA testing was negative for K-RAS and BRAF.Patient was admitted in PGH from 03/15/15- 03/17/15 for productive cough. He was again advised to start chemotherapy; however, he opted to hold off on therapy till he explores all his options. On 06/24/2015 he was seen by the ophthalmologist for significant decrease in vision and pressure sensation in the left eye for couple of months which he described as a shadow along the supra temporal area in that eye. Dilated fundus examination of the left eye and the B-scan of the eye showed clear vitreous with normal optic nerve and just adjacent to the nerve an elevated whitish retinal mass with subsequent serous retinal detachment inferiorly. The mass measured 5 mm in elevation and 10 mm in diameter and extending up to the optic nerve, inferiorly up to the fovea and beyond the arcades .There was no hemorrhage and vitreoretinal traction. Subsequently he had an Optical Coherence Tomography of the left eye  and was By this time he also had hepatic decompensation with ascites requiring paracentesis and DVT treated with Lovenox. He was sent to ED on 07/08/2015 for evaluation of the retinal tumor and for further work up and management. The inpatient MRI of the orbit was negative for any mass in the eye and orbit. He was referred to Radiation Oncologist for further course of action.He was seen by radiation oncologist on 07/09/2015. Given his existing metastatic disease burden, the recommendation was made to give a course of palliative radiation therapy to the left eye.The patient underwent CT simulation as part of the initial work up to develop a treatment plan and precisely identify and localize the treatment target  and 4. H1 Unlike metastasis to choroid, retinal metastasis is very rare.1 The current case presents an uncommon site of metastasis for colorectal cancer. Historically intraocular metastasis was considered a rare intraocular tumor; though recently it is considered more common.2,4,5 retinal metastasis from esophageal and pancreatic cancers are reported in few case reports. Utilizing OVID, PUBMED/MEDLINE and Google Scholar, we conducted a literature search to find out prior reported cases of retinal metastasis originating from colon cancer. Keywords used for the literature search included ‘colorectal neoplasms’, ‘metastasis to retina’/’retinal metastasis’ and ‘eye neoplasms’. The retinal metastasis from colon adenocarcinoma of the colon are reported in only 3 cases.4,7,8 First case was described by Kennedy et al.3 in 1956 in a 51 year-old male patient who presented with blurry vision in the right eye and on ophthalmoscopic examination was found to have 1/6 dd. Sized well circumscribed grayish white lesion in the macula extending into the vitreous. His past medical history was significant for intermittent rectal bleeding and underwent fistulectomy. He was referred to a proctologist who confirmed that the patient had annular carcinoma of the rectosigmoid junction. The patient immediately underwent rectosigmoidectomy and the pathology was consistent with adenocarcinoma of the rectosigmoid.In contrary to choroidal metastasis which occur 7% from visceral or gastrointestinal cancers,6 The patient had multiple tumors including sebaceous adenomas of face and neck status post excision, uterine leiomyoma, keratoacanthoma of the eyelid, squamous cell carcinoma of the forehead, adenocarcinoma of the breast status post mastectomy, adenocarcinoma of the colon and retinal tumor. The patient underwent enucleation of the eye for retinal tumor and the histopathology was consistent with adenocarcinoma. However, the source of primary tumor was not verified because of the unavailability of the breast adenocarcinoma samples for review.The follow-up ophthalmoscopic examination in 6 months from his initial visit showed increasing size of the tumor and new vessels coursing over the tumor prompting enucleation of the eye. The histopathology of the sections of the eye was consistent with that of the adenocarcinoma of the rectosigmoid. However, 3 months later the patient died most likely from metastatic disease and necropsy was denied. A second case was reported in a 74 year old woman with Muir-Torre syndrome; an autosomal dominant condition with skin lesions associated with visceral cancers.7 A 39 year-old male with adenocarcinoma of the cecum metastasizing to liver and lung underwent hemicolectomy. Six weeks after surgery, he received postoperative chemotherapy for 6 weeks and presented with complaints of vision changes in his left eye. Ophthalmologic examination revealed sub retinal hemorrhage in the eye. No mass was identified at that time. However, during his follow-up visit at 2.5 months, he was found to have a 2.8 mm x 5.3 mm x 6.6 mm retinal mass associated with exudative retinal detachment also involving the fovea. One month later, on dilated eye exam and ultrasonography, the mass was found to have increased in size significantly. He was still receiving chemotherapy for the primary cancer and underwent 3-port plana vitrectomy for the retinal tumor.7 The procedure took care of the entire retinal tumor en bloc due to confinement of the tumor to retina and not involving any surrounding structure. The histopathology of the tumor was consistent with the mucin-secreting adenocarcinoma of the colon primary. The patient received post-operative palliative radiation to the orbit and at 3 months follow-up visit he had a non-painful eye with normal intraocular pressure. His peripheral vision was regained subsequent to retinal reattachment procedure.7Most recently another case of adenocarcinoma of the cecum metastasizing to retina was described by Apte et al.When there is cancer metastasis to retina there is a high probability of the cancer spreading to central nervous system which carries poor prognosis. Hence, retinal metastasis should always be considered in the differential diagnosis of any colon cancer patient presenting with vision changes. Also, as demonstrated by our case, MRI of the orbit may not always be conclusive in diagnosing retinal tumors.9 This case forms a basis for difficult diagnosis of a rare clinical presentation. Clinical presentation of vision changes in a patient with history of colon/GI cancers should raise high index of suspicion for retinal metastasis and has to be evaluated by ophthalmoscopic examination involving dilated fundus exam, B-scan and OCT to arrive at a diagnosis and further assist in the treatment plan which is most often palliative radiation to the eye.


Extragastrointestinal stromal tumors (EGISTs) are relatively rare soft tissue neoplasms arising from the extra gastrointestinal tract. The current study presents a case of primary EGIST of the prostate observed in a 55-year-old male. Imaging studies showed a 10×10.5×9.5-cm prostate mass. On histological observation, the mass was separated from the rectum serosa and exhibited a high mitotic count (8/50 high-power fields). The results of immunohistochemical staining showed positive immunoreactivity for cluster of differentiation (CD)117 (c-kit), CD34 and DOG1 in the tumor. On mutation analysis, loss of heterozygosity of the c-kit gene was observed in the prostatic EGIST; however, the platelet-derived growth factor receptor-α (PDGFRA) gene was considered to be normal. Therefore, as EGIST of the prostate is rare, there is a requirement for the confirmation of the diagnosis to be based on immunohistochemistry and mutation analysis (of c-kit and PDGFRA). Gastrointestinal stromal tumors (GISTs) are the most common tumors of the gastrointestinal (GI) tract. GISTs originate from the interstitial cell of Cajal, an intestinal pacemaker cell in the gut. These cells are known to express the KIT gene (detected as the cluster of differentiation [CD]117 antigen), which is important for distinguishing GIST from other mesenchymal neoplasms . GISTs tA 55-year-old male presented to the Department of Urology at the Xiangya Hospital of the Central South University  with dysuria and urinary frequency that had persisted for approximately six months. The patient’s review of symptoms and medical history were otherwise unremarkable. A digital rectal examination revealed that the prostate was markedly enlarged with a smooth, bulging surface and unusual consistency on palpation. The patient’s prostate-specific antigen (PSA) level was 2.01 ng/ml; all other laboratory values were normal. Computed tomography (CT) showed an enlarged prostate measuring 10×10.5×9.5 cm, but the prostatic capsule was intact . MagnetiDuring surgery, the tumor was circumscribed and no associations with the neighboring organs and structures were identified. The tumor was easily separated from the adjacent structures and no enlarged pelvic lymph nodes were detected.Microscopically, the tumor consisted of spindle-shaped cells with 8/50 high-power fields (HPFs) of mitotic activity  and regiA mutation analysis of exons 9 and 11 of the c-kit gene and those of exons 12 and 18 of the platelet-derived growth factor receptor-α (PDGFRA) gene were examined. Microsatellite markers proximal to the c-kit gene were performed via polymerase chain reaction of the tumor tissue and normal specimens , howeverThe patient had an uneventful postoperative course. As an adjuvant to the postoperative molecular targeted chemotherapy, the patient was treated with an oral administration of 400 mg/day of imatinib (IM). No local recurrence or distant metastasis was observed at the 12-month follow-up.In accordance with the regulations of the Human Investigation Committee of the Central South University , written informed consent was obtained from the patient for publication of the current report and any accompanying images.GIST is a non-epithelial, mesenchymal tumor of the GI tract, occurring predominantly in the stomach and small and large intestines. A mutation in c-kit exons 9, 11, 13 and 17, and PDGFRA exons 12, 14 and 18 is responsible for activation of the gene signaling system, which results in uncontrolled phosphorylation and tissue growth  (9). EGIet al , high mitotic counts (8/50 HPFs) and extensive hemorrhagic necrosis of the tumor were the malignant features. Therefore, this tumor belongs to the high-risk group and the results indicated that IM was required.GISTs exhibit highly variable biological behavior. Although only 10–30% of GISTs are clinically malignant, GISTs harbor a slight malignant potential . TherefoIn conclusion, the current study presents a rare case of EGIST arising from the prostate and the following are proposed: i) It is crucial to note possible adhesion to the rectal wall during pathological examination; ii) immunohistochemistry, using antibodies against CD117 (c-kit) and CD34, is valuable for the diagnosis of EGIST; iii) DOG1 must be included in the routine diagnostic immunohistochemical panel as it allows proper classification of EGIST; iv) mutation analysis (of c-kit and PDGFRA) is potentially significant in the diagnosis and treatment of EGIST; v) IM therapy is recommended for high-risk patients following complete surgical removal of EGIST; and vi) long-term follow-up is necessary.


Abdominoperineal excision is performed in patients with locally advanced, low rectal carcinoma. Reconstruction of the dorsal vagina and perineum using the vertical rectus abdominis myocutaneous flap following extensive surgery results in favorable surgical outcome and quality of life. However, the rectus abdominis muscle, as part of the anterior abdominal wall, may develop fibrous lesions also as a transplant.A 39-year-old female patient with low rectal cancer and extensive colorectal polyposis was treated with neoadjuvant chemoradiotherapy followed by colectomy and abdominoperineal excision with resection of the dorsal vaginal wall and subsequent reconstruction of the perineum using the vertical rectus abdominis myocutaneous flap. At the 6-month follow-up, a suspected 2 × 2 cm tumor recurrence was detected in the transposed tissue and was subsequently surgically removed. Histologic examination concluded with fibromatosis. Genetic testing revealed a known disease-causing mutation in the adenomatous polyposis coli gene, confirming the diagnosis of familial adenomatous polyposis.Fibromatosis may affect the anterior abdominal wall, that is the rectus abdominis muscle, at the primary site or may develop in the muscle after its transposition into the perineum at pelvic reconstruction. Fibromatosis in the muscle flap after pelvic reconstruction may present a difficult diagnostic challenge for the multidisciplinary team. For low rectal cancer, abdominoperineal excision (APE) is usually the procedure of choice, and in locally advanced cases, the majority of patients require extralevatoric excision to achieve adequate circumferential resection margins . Tumor i2) on days of RT. The response evaluation showed excellent tumor volume response, but with similar organ involvement as at baseline, and she proceeded to surgery 8 weeks after CRT completion. Surgery involved total colectomy, APE with resection of the posterior vaginal wall and coccygeal bone with subsequent reconstruction of the perineum with right-sided VRAM flap, and a terminal ileostomy. After an uneventful recovery, she was discharged 15 days postoperatively. Histologic examination of the specimen showed a 35-mm large adenosquamous carcinoma removed with free resection margins and metastasis in one of four local lymph nodes (pT2N1). In addition, multiple dysplastic adenomas were present in the entire colon.A 39-year-old, healthy, non-smoking female patient with no family history of cancer was referred on suspicion of rectal cancer. Digital rectal examination and subsequent rigid proctoscopy revealed a low rectal tumor and multiple polyps in the rectum. Subsequent colonoscopy showed extensive polyposis of the entire colon. The tumor biopsy confirmed the presence of a rectal adenocarcinoma, while dysplastic changes were found in the polyp biopsies. No distant metastases were detected on the thoracoabdominal computed tomography (CT) scan, and baseline carcinoembryonic antigen (CEA) was 8 μg/L. Pelvic magnetic resonance imaging (MRI) revealed a locally advanced rectal cancer with extra-mesorectal growth into the rectovaginal septum and the lower posterior vaginal wall as well as a suspected growth into the anterior aspect of the coccygeal bone with multiple suspected malignant lymph nodes within the mesorectum (T4N2). Upon evaluation by our multidisciplinary team (MDT), she was scheduled for neoadjuvant chemoradiotherapy (CRT) followed by surgery. Neoadjuvant treatment was given as CT planned radiotherapy  with concomitant capecitabine , confirming this diagnosis.At the routine follow-up 6 months postoperatively, clinical examination revealed a 2 × 2 cm tumor located in the muscle tissue of the VRAM flap, and an early local recurrence was suspected. Pelvic MRI showed a 10 × 16 mm rich vascularized and cell dense lesion corresponding to the clinically detected tumor  is a benign, myofibroblastic proliferation that rarely occurs in the general population but frequently in one of the hereditary cancer predisposition conditions known as FAP or a variant of FAP called Gardner syndrome , 7. Near-catenin . Nuclear-catenin , 10. Desdon 1400 . Howeverdon 1400 , 13.Taking into consideration all of these factors, our patient was at high risk of developing fibromatosis. However, as the standard follow-up procedure for patients undergoing rectal cancer treatment is to focus on early detection of recurrence and metastasis, and furthermore considering the patient’s primary diagnosis, the MRI findings, the lesion site, and its rapid development, the initial conclusion was local recurrence. Fibromatosis as a differential diagnosis was thus not considered, and the MDT opted for direct surgical removal, as the lesion’s location and size were found to be appropriate for primary surgical removal. The use of PET-CT was discussed, but high-quality CT and MRI analysis did not reveal additional suspected malignant lesions and were considered sufficient for making treatment decisions. The histological examination, however, revealed fibromatosis in the VRAM flap in the perineum instead of the expected recurrence. It is noteworthy that no sign of fibromatosis in the abdominal rectus muscle was found on the CT prior to the primary surgery.Currently, the universal approach to management of desmoid tumors  favors the “watch and wait” approach rather than surgical removal. This is based on the observation that size stabilization or spontaneous regression may occur while surgery in itself may trigger the growth of new lesions , 15.In this case, no histological evidence (biopsy) was available to support such a decision, nor was the genetic evaluation implemented. The lesion was considered to be a suspected local relapse without distant metastases at the time of diagnosis and was dealt with according to national guidelines for treatment of recurrent rectal cancer.In this case, fibromatosis in transposed rectus abdominis muscle tissue imitated a rectal cancer recurrence. Although rare, but with pelvic reconstructive surgery becoming more frequent, this may be a relevant differential diagnosis for the MDT to consider, particularly in patients with FAP.Written informed consent was obtained from the patient for publication of the case report and any accompanying images. A copy of the written consent is available for review at the editor of this journal.


Colonic atresia and anorectal malformation are rare congenital anomalies individually. Few reports of the conditions combined in a single patient have been published in the literature. Neither colonic atresia, anorectal malformation or a combination of the disorders has previously been reported in the Haitian population.A 5-day-old female presented with feculent emesis, failure to pass stool since birth and an imperforate and stenotic anus. Exploratory laparotomy revealed colorectal atresia distal to a malformed cecum and a Wingspread low subtype anorectal malformation without any associated urogenital fistulae. Temporizing percutaneous ileal drainage was followed by second-stage anal perforation and dilation, ileal J-pouch and pull through.in utero have been implicated as the etiology of colonic atresia, simultaneous presence of anorectal malformation suggests a multifactorial cause. Investigation for multisystem abnormalities is warranted. Two-staged operative correction is considered the best treatment; however, long-term postoperative outcomes are uncertain.This is the first reported case of colonic atresia, anorectal malformation or the combination of the disorders among the Haitian population and one of only a handful of such cases reported worldwide. Although vascular accidents The coexistence of colonic atresia and anorectal malformation is a very rare occurrence and presents unique clinical and operative challenges. Investigation for additional congenital abnormalities is appropriate, and although two-stage operative correction is considered the best treatment, long-term outcomes are uncertain. CA represents 5–15% of all intestinal atresias, with an incidence as low as 1 in 66,000 live births  is a congenital anomaly in which part of the colon has not formed Worldwide, coexistence of these anomalies in a single patient has been reported only a handful of times  and moderate dehydration. Her abdomen was grossly distended without signs of peritonitis.In hospital, the patient presented with malnourishment (2.5 kg, 3Because external genitalia appeared anatomically correct; the anus was imperforate; and by using a probe, we could distinguish a stenotic anal canal, we classified the child as Wingspread low subtype ARM Wakhlu .Abdominal radiograph demonstrated extensive bowel dilatation. Attempt at barium enema revealed no recto-anal fistula.Cardiovascular examination revealed no evidence of cardiac anomalies with no murmurs, equal femoral pulses and appropriate pre- and post-ductal oxygen saturations.Electrolytic and fluid balance was optimized for surgical intervention and parenteral antibiotics were administered.Exploratory laparotomy revealed grossly intact gastrointestinal system from proximal stomach to patent ileocecal valve. The length of the small bowel was grossly adequate for nutrient absorption.However, distal to the ileocecal valve, five centimeters of a blind-ending colon existed with a fully-formed appendix and some bulbous anatomical features of a cecum. But there was no ascending, transverse, descending or sigmoid colon. We visualized the superior mesenteric artery but were unable to visualize the inferior mesenteric vessels. The colonic segment was not attached to the posterior abdominal wall and floated freely in the abdominal cavity Figure .Figure 1There was no rectum, no evidence of no intestinal-urinary or genitor-urinary fistulae and no gross anal musculature atresia—the pelvic bowl was an empty cul-de-sac at the base of which, the imperforate, stenotic anus could be palpated, confirming Wingspread low subtype ARM. There was no evidence of malrotation, other urogenital abnormality or abdominal wall defect.A percutaneous drainage tube was placed into the colonic segment and through the ileocecal valve into the terminal ileum. Post-operatively, the patient remained hemodynamically stable and received enteral nutritional support for two months.She then underwent a second-stage exploratory laparotomy, J-pouch creation, perforation and dilation of the anal opening and anal pull-through.The patient recovered from the second-stage operation without complications and is defecating effectively with plans for long-term monitoring and follow-up.Coexistence of CA and ARM has been reported only a handful of times in the literature (Goodwin et al. in utero are most commonly implicated in the etiology of CA; whereas, the etiology of ARM is widely considered multifactorial and may include participation of recessive genes (Nazer et al. Vascular accidents occurring Our patient’s mother did not smoke or use legal or illicit vasoactive drugs of any kind and had no family history of congenital anomalies. She denied first trimester menorrhagia and consanguinity. An impoverished woman, she had no access to adequate nutrition or prenatal medical care. As with previous cases of combined CA and ARM, etiology of our case remains unclear and is considered multifactorial.Presentation of CA can be delayed by a number of factors, including restricted access to care and lack of adequate diagnostic tools or clinical experience with the disorder (Levard and Boureau Presentation of the combined disorder of CA and ARM is unclear. However, as in our case, the classical features of bowel obstruction in neonates should raise suspicion for either or both underlying causes. Cardinal signs and symptoms of neonatal bowel obstruction include maternal polyhydramnios, bilious emesis, failure to pass meconium on first day of life and abdominal distension (Juang and Snyder Our patient’s abdominal radiograph revealed distended bowel loops consistent with intestinal obstruction. Although she was hemodynamically stable, the severity of imaging studies and obvious imperforate anus necessitated urgent operative intervention.in utero vascular accident of the inferior mesenteric artery may help explain the extensive CA; whereas, the ARM may be related to a genetic failure of anus formation.Laparotomy revealed CA and confirmed Wingspread low subtype ARM. While Wingspread high subtype ARM may involve rectal atresia, our case had no evidence of fistulae, presacral mass or anal musculature atresia also commonly associated with high Wingspread classification (Hamrick et al. Optimal surgical correction of combined CA and ARM is two-stage with an ostomy for decompression and evacuation of bowel content, followed by nutritional optimization before definitive correction Wakhlu . DependiIn a stable child with CA but without other abnormalities, complete recovery of bowel function can be expected (Mansoor et al. The coexistence of CA and ARM is a very rare occurrence and presents unique clinical and operative challenges. Investigation for additional congenital abnormalities is appropriate, and although multi-stage operative correction is considered the best treatment, long-term outcomes are uncertain.Written consent was obtained from the patient’s mother for the publication of this report and any accompanying images.


Cystic lymphangioma is a rare tumor of the lymphatic vessels that occurs more frequently in women. Location of this pathology can be diverse but most commonly occurs in the neck or axilla. Cystic lymphangioma originating from the adrenal tissue represents a very rare entity.We report here the case of a 38-year-old woman who was diagnosed with a cystic retroperitoneal mass. After further investigations, the patient was suspected to have a left adrenal cystic lymphangioma. She underwent successful open left adrenalectomy as curative treatment, and the diagnosis of cystic lymphangioma of the left adrenal gland was confirmed at histology. The postoperative course was uneventful.This case report and review of the literature bring new insights into the diagnostic difficulty and management of cystic lymphangioma of the adrenal gland. Cystic lymphangiomas are rare benign lesions of the lymphatic vessels . These tA 38-year-old woman known for anxiety disorder developed constant epigastric pain without radiation toward the back. She mentioned that the pain was not related to the food intake and scored it between four and six (out of ten) on a visual analog scale. She had multiple episodes of vomiting, no bowel movement problems, and no fever. Her past medical and surgical history was otherwise uneventful. She went to her general practitioner who first performed an ultrasonography (US) and a CT scan. These exams showed a cystic-like retroperitoneal mass on the left side measuring 13.4 × 7.2 × 5.2 cm. Laboratory tests were normal. No further exams were undertaken at this point. The pain slightly diminished with paracetamol and non-steroidal anti-inflammatory drugs. However, after a couple of months, the pain reappeared, and the patient was scheduled for a CT-guided puncture of this cyst.At first, the radiologist was not able to puncture the cyst due to a thick capsule. The cyst was finally punctured under US control but could not be completely evacuated due to technical problems . Cytology came back negative for malignant cells and was compatible with a cystic lymphangioma. Unfortunately, the puncture of the cyst did not help to relieve the patient’s symptoms. The patient then underwent a magnetic resonance imaging (MRI) to assess more precisely the location of this retroperitoneal cyst and its anatomic relations. T2-weighted turbo spin-echo MR-sequences showed a lesion compatible with a cystic lymphangioma originating from the left adrenal gland Figure . The cysen bloc’ left adrenalectomy was performed without rupture of the cyst. No drain was left in place. The postoperative course was uneventful, and the patient was discharged on postoperative day 5. The patient was seen at the outpatient clinic one month after surgery and described no more symptoms.Due to the size and position of the cyst, and to avoid a rupture of the cyst during laparoscopy, a left subcostal laparotomy was performed. During surgery, the adrenal origin of the cyst was confirmed, and an ‘The specimen consisted of an ovoid, cystic mass measuring 8.5 × 4.3 × 2.8 cm (Figure This article reports a rare case of symptomatic cystic lymphangioma originating from the left adrenal gland in a female patient successfully treated by complete surgical resection.The majority of intra-abdominal cystic lymphangiomas is located in the mesentery, in contrast to adrenal location that is very rare . No moreOf note, lymphangioma is the generic term for a tumor arising from the lymphatic vessels and is often found in children. Lymphangiomas have an endothelial origin. The exact pathogenesis is currently not completely elucidated, and whether lymphangioma of the adrenals is a real neoplasm remains unclear . The mosDifferential diagnosis of a retroperitoneal cystic-like lesion includes primary adrenal tumors, metastatic adenocarcinomas, angiosarcomas, multicystic mesotheliomas, or adrenal cysts . AdrenalCystic lymphangioma of the adrenals does not have a pathognomonic radiological presentation, but new imaging modalities bring useful information helping the diagnosis . As the Immunohistochemistry is an important tool to differentiate this pathological entity from other diagnoses. Lymphangiomas usually display D2-40, PROX1, and CD31 positivity and absence of CD34 and CKAE1/AE3 stains .Asymptomatic cystic lymphangioma discovered incidentally can just be followed clinically or with control imaging, as there is no risk of malignant degeneration . PuncturIn summary, cystic lymphangioma of the adrenal gland is a rare pathology that should be included in the differential diagnosis of cystic lesions of the adrenal glands. Its diagnosis can be difficult and challenging. MRI seems to be a good diagnostic modality to detect degeneration or intracystic hemorrhage. If the patient is symptomatic, definitive treatment is surgery.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.


Ectopic (accessory) adrenocortical tissue, also known as adrenal rests, is a developmental abnormality of the adrenal gland. The most common ectopic site is in close proximity to the adrenal glands and along the path of descent or migration of the gonads because of the close spatial relationship between the adrenocortical primordium and gonadal blastema during embryogenesis. Ectopic rests may undergo marked hyperplasia, and occasionally induce ectopic adrenocortical adenomas or carcinomas.A 27-year-old Chinese female patient who presented with amenorrhea of 3 months duration underwent computed tomography urography after ultrasound revealed a solitary mass in the left renal hilum. Histologically, the prominent eosinophilic tumor cells formed an alveolar- or acinar-like configuration. The immunohistochemical profile  indicated the adrenocortical origin of the tumor, diagnosed as ectopic adrenocortical adenoma. The patient was alive with no tumor recurrence or metastasis at the 3-month follow-up examination.The unusual histological appearance of ectopic adrenocortical adenoma may result in its misdiagnosis as oncocytoma or clear cell renal cell carcinoma, especially if the specimen is limited. This case provides a reminder to pathologists to be aware of atypical cases of this benign tumor. Although uncommon, an ectopic adrenal lesion should be included in the differential diagnosis of tumors involving the renal hilum. A misdiagnosis of this benign condition as a malignant renal tumor may have severe consequences for the patient, including unnecessary radical nephrectomy. Preoperative biopsy and appropriate immunohistochemical staining will assist in determining the origin and nature of the tumor and in avoiding intraoperative uncertainty. Ectopic adrenal rests have been reported from a variety of anatomic sites, including the celiac plexus, kidney, testis, epididymis, broad ligament, the canal of Nuck, hernial and hydrocele sacs, the mesoappendix, liver, lung, intradural space and brain –7. CortiA 27-year-old Chinese female complained of amenorrhea of 3 months duration. Her medical history and that of her family was unremarkable. Her body mass index (BMI) and blood pressure (BP) were within the normal range . Virilization and other clinical hormonal abnormalities were not noted, except for a borderline elevation of testosterone. Multislice spiral computed tomography (CT) urography revealed a well-circumscribed, round, soft-tissue mass with a maximum diameter of 2.7 cm in the left renal hilum, near the renal pelvis and atrophic bilateral adrenals. The CT value for the unenhanced mass was 35 HU; following contrast agent administration, it increased to 161 HU. Multislice spiral CT urography showed no abnormalities in kidney blood flow or renal perfusion. Renal function was normal, except for a slight delay in renal excretion ; the others were stained for immunohistochemistry using the streptavidin-peroxidase system  according to the manufacturer’s instruction. Commercially available, prediluted monoclonal antibodies against the following antigens were used to evaluate the specimen: pan-cytokeratin (AE1/AE3), vimentin, synaptophysin, CD56, neuron specific enolase (NSE), alpha-inhibin, Melan-A, CD34, S100, PAX8, chromogranin A, Ki-67 and anti-mitochondrial antibody . For the negative controls, the primary antibody was replaced with PBS.Grossly, the mass was solid, round and well circumscribed; its cut surface was yellow and brown. The mass measured 2.5 cm at its largest diameter and weighed 8 g. Neither necrosis nor hemorrhage was identified.Histologically, the tumor was well circumscribed and composed of cells mainly arranged in nests forming an alveolar- or acinar-like configuration Fig.  and surrThe tumor cells were markedly and diffusely positive for vimentin, alpha-inhibin Fig. , Melan-AAdrenocortical primordium (seen as a thickening of the coelomic mesothelium) is formed by the invagination of the coelomic epithelium, adjacent to the region where the gonadal blastema arises, on approximately day 30 of gestation. During gonadal migration, fragments of adrenocortical tissue may be scattered along the descending path and form ectopic adrenal glands , 8. AlthTumors of the renal pelvis are limited in their histological type, especially those in the soft tissue near the hilum. Urothelial and renal neoplasms should be considered first, whereas other rare neoplasms include pelvic lipomatosis or fibrolipomatosis, urinoma, angiomyolipoma, nephrogenic adenoma, solitary fibrous tumor, leiomyoma, neurofibroma and hemangioma . In our Adrenocortical adenoma must be distinguished from adrenocortical adenocarcinoma once an adrenocortical origin has been confirmed. Most adrenocortical adenocarcinomas are larger than adenomas, but they may overlap in size. Adrenocortical adenomas lack the histological features of malignancy, such as vascular invasion, necrosis, fibrous bands, capsular invasion, an increased mitotic rate, atypical mitoses and nuclear atypia. Nonetheless, distinguishing these benign tumors from malignant ones is difficult in some cases , 7, 11. The final problem was to distinguish this tumor from oncocytic adrenocortical neoplasm and adrenocortical oncocytoma. The neoplastic cells of oncocytoma are exclusively oncocytes, with an abundant, eosinophilic and granular cytoplasm. The cells are predominantly arranged in a diffuse or solid pattern, although foci with an alveolar configuration have been described –17. OncoEctopic adrenal rests may undergo remarkable hyperplasia in patients with Nelson syndrome or in association with increased adreno-cortico-tropic-hormone production. However, ectopic adrenal cortical adenomas and carcinomas arising from ectopic rests are extremely rare. Some ectopic neoplasms can lead to Cushing’s syndrome, hyperaldosteronism or virilization or feminization, while others are nonfunctioning or asymptomatic, with only biochemical evidence of hormone hypersecretion , 7, 11. This case provides a reminder to pathologists to be aware of atypical ectopic adrenocortical adenoma and to include an ectopic adrenal lesion in the differential diagnosis of tumors involving the renal hilum. The failure to distinguish this benign tumor from a malignant renal tumor may have severe consequences, including unnecessary radical nephrectomy. Preoperative biopsy and appropriate immunohistochemical staining can aid in determining the origin and nature of the tumor and thereby avoid intraoperative uncertainty.Ethical approval for this study was obtained from the Local Trials Committee of the China Medical University.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.


Multiple brain magnetic resonance imaging (MRI) studies showed enlarged ventricles but no masses. Immediately before surgery, a discrete cystic structure was visible on imaging (A 33-year-old male presented for resection of an intracranial mass after 1 year of recurrent headaches and dizziness. Cerebrospinal fluid testing had shown a lymphocytic pleocytosis (163 cells/mm


Rectal syphilis is a rare expression of the widely recognised sexual transmitted disease, also known as the great imitator for its peculiarity of being confused with mild anorectal diseases because of its vague symptoms or believed rectal malignancy, with the concrete risk of overtreatment. We present the case of a male patient with primary rectal syphilis, firstly diagnosed as rectal cancer; the medical, radiological, and endoscopic features are discussed below. In the last fifteen years, sexual transmitted diseases (STD) had a new increase in incidence; among them syphilis plays a primary role, above all in men who have sex with men (MSM) .Its anorectal localization is particularly rare and constitutes a challenge for the triad surgeon-radiologist-GI endoscopist because it is often misinterpreted as malignant rectal pathology .We present the case of a male patient firstly diagnosed for anorectal cancer and then discovered to be a form of anorectal syphilis.A 48-year-old HIV-positive white male was admitted in our proctology outpatient clinic due to proctorrhagia arisen a few weeks before. Digital rectal examination revealed a palpable mass extended for 3 cm on the right-anterior wall of the rectum, 5 cm away from the anal verge.Investigating patient's clinical history and his sexual preferences, we knew he had no relevant medical history, had an ongoing homosexual relationship, and denied recent unsafe sexual behaviour.An urgent colonoscopy was arranged and confirmed a single large ulcerated lesion, occupying one-third of the visceral circumference; biopsy specimens were sent for histological analysis.b = 800, 1000); two centimetric lymphadenopathies were also identified in the perirectal fat, a possible expression of nodal metastases. A Positron Emission Tomography reported a focal [F-18]FDG accumulation in the rectal wall, expression of a lesion with high glucose metabolism, and in five spots in the mesorectal space, suggestive for nodal spread.The patient underwent a complete staging in the suspect of rectal malignancy; the computed tomography was negative for metastases to thoracic and abdominal parenchymal organs. The MRI showed a pathological, 2 cm deep thickening of the rectal wall, starting at 4 cm from the anal verge and extended proximally for about 6 cm, with a possible infiltrative behaviour to the mesorectal fat and significant postcontrast signal enhancement but no hyper intensity in the diffusion-weighted images ; there was no hyperintensity of the suspected neoplastic lesion, that is, a classical MRI sign of both rectal adenocarcinoma and mucinous carcinoma FDG accumulation in the rectal wall and nodes in mesorectal fat is common to lesions with high glucose metabolism , 11 Fig.Histological examination and dark field microscopy on fresh specimen, together with a positive serological test like VDRL, are fundamental for the definitive diagnosis.Haematoxylin-Eosin stain shows chronic aspecific inflammation concerning tissues, vessels, and perivascular space, with presence of plasma-cells and giant multinucleated cells; Warthin-Starry stain allows direct visualization of spirochetes in bioptic specimen .Once the diagnosis has been clarified, the therapy consists of 2.4 million units of intramuscular Penicillin G-Benzathine a week for a minimum of three weeks. Regression of lesions and infectious disease is usually rapid and complete, without residual manifestations .In conclusion the incidence and prevalence of syphilis and in particular its anorectal form rose significantly in the last 15 years alone or associated with other STD or HIV, mainly between men who have sex with men. It is also called the great imitator because of the wide range of proctologic diseases that must be excluded in order to have a precise diagnosis, firstly rectal neoplasm.For this reason we believe that endoscopist, surgeon, and radiologist should deeply investigate patient's sexual habits and routinely perform STD screening to avoid unnecessary diagnostic procedures and overtreatments and to provide the anatomopathologist the most complete picture of the patient in order to obtain the best answer.


Malignant schwannomas or neurofibrosarcomas are rare nerve tumors of unknown etiology. These neoplasms are highly aggressive with a marked propensity for local recurrence and metastatic spread. Their management continues to be a challenge for pathologists and surgeons. Maxillofacial locations are very exceptional. We report the case of a patient with unusual malignant schwannoma of the infratemporal fossa discovered at a late evolving stage.A 56-year-old woman, of Moroccan nationality, presented to our hospital in 2013 with a large right-sided hemifacial swelling that had evolved over the previous 4 months, with a limitation of mouth opening, nasal obstruction and episodes of epistaxis. A CT scan and MRI showed a large and invasive tumor occupying her right infratemporal fossa and maxillary sinus, with sphenoidal, ethmoidonasal, nasopharyngeal and intraorbital extension. A nasal endoscopic biopsy was performed. Immunohistochemical examination concluded a diagnosis of malignant schwannoma, and a palliative radiotherapy was decided; however, our patient died 10 days later.Malignant schwannoma of paranasal sinuses and the anterior skull base is a rare tumor that involves a high rate of local invasion. The prognosis is poorer compared to that occurring in the trunk and extremities. Malignant schwannomas or neurofibrosarcomas are rare nerve tumors with very poor prognosis; maxillofacial locations are very exceptional . These tA 56-year-old woman, of Moroccan nationality, a chronic cigarette smoker consuming an average of one pack a day for 38 years, was hospitalized for a large right-sided hemifacial swelling. Her early symptoms dated from 4 months previously, by the appearance of straight facial neuralgia, treated as dental neuralgia without any improvement. The evolution was marked by rapid aggravation of the pain, which then became disabling, and the emergence of a right-sided maxillary swelling rapidly increasing in size and accompanied by a limitation of mouth opening, and a nasal obstruction with episodes of epistaxis.A clinical examination determined a painful and inflammatory right-sided hemifacial swelling, right exophthalmus without notion of decreased visual acuity, and hypoesthesia at the second and third trigeminal nerve areas. An endonasal examination detected a tumor mass in her right nasal cavity, friable and bleeding at contact. An oral examination, very difficult because of the limited mouth opening, showed an erosion of the hard palate. Cervical lymph node areas were free and an endoscopy examination of the upper aerodigestive tract did not show any lesions. Otoscopy and audiometry test results were normal. Our patient was hospitalized and placed immediately on morphine.A computed tomography (CT) scan revealed the presence of a large heterogeneous mass occupying her infratemporal fossa as well as the masticator space and right maxillary sinus, lysing the walls of the maxillary sinus, the ascending branch of the mandible and the orbital floor with ethmoidonasal, parapharyngeal and intraorbital extension.A magnetic resonance imaging (MRI) scan showed an aggressive and invasive tumor occupying her right infratemporal fossa and maxillary sinus with irregular boundaries, heterogeneous intermediary signal at T1 and T2 weight, with sphenoidal, ethmoidonasal, nasopharyngeal and intraorbital extension. This process infiltrated the jugal and temporozygomatic soft parts, with heterogeneous contrast enhancement and areas of necrosis Figs. , 5 and 6A nasal endoscopic biopsy was performed. A pathological examination was in favor of a malignant process, poorly differentiated and invasive Fig. . An immuMalignant schwannoma, also called neurofibrosarcoma, neurogenic sarcoma or malignant tumor of the peripheral nerve sheath , is a tumor of the peripheral nervous system that develops in the nerve sheath at the expense of Schwann cells . This tuIt accounts for approximately 5 to 10 % of all soft tissue sarcomas and has a strong association with NF-1 , 3, alsoMPNSTs represent only 2 to 6 % of head and neck sarcomas , evolutiThe origin of most MPNSTs of the nose, the paranasal sinuses and the anterior skull base is presumed to be the ophthalmic and maxillary divisions of the trigeminal nerve and its terminal branches, as well as autonomic ganglia , 5.These tumors primarily affect adults aged between 20 and 50, with an average of 40 years of age, without sex predominance .Clinically, MPNSTs generally are presented as a progressively enlarging and painful mass in the paranasal sinus or anterior skull base. Malignant schwannomas may become clinically apparent with unilateral nasal obstruction, hyposmia, epistaxis, atypical pain, hypoesthesia or localized swelling of the facial and orbital region, mucopurulent rhinorrhoea and headache .Exophthalmus, epiphora and progressive visus reduction are less frequently described and are principally related to neoplasms of the orbital compartment. Intracranial extension has also been reported , 6.High-resolution CT scanning is considered to be an adequate imaging investigation for schwannomas of paranasal sinuses . CT, howMicroscopically, MPNSTs are highly cellular, focally polymorphic tumors compounded of spindle cells arranged in bundles or fascicles with high mitotic rates, indistinct cytoplasmic borders and a variable degree of nuclear pleomorphism , 9.MPNSTs can be histologically similar to other malignant tumors, particularly malignant melanoma and other spindle cell sarcomas. Currently, immunohistochemical analysis especially, but also the recognition of Schwann cells by electron microscopy, help identify MPNSTs , 10. In A literature review by Pfeiffer, revealed that of 33 well-documented cases of MPNSTs localized in the anterior skull base or the paranasal sinuses published since 1971 , only niManagement of MPNSTs has often been reported to be challenging, especially in the head and neck, so early diagnosis is indispensable. The recommended treatment of MPNSTs is surgical extirpation including wide margins, and involved nerves should be followed proximally in an attempt to obtain clear margins. Regional nodal dissection is not recommended. The surgical approach depends upon the location and the extent of the tumor .The role of radiotherapy and chemotherapy in the treatment of this tumor is still controversial. MPNST has traditionally been described as being highly radioresistant, but, some recent reports recommend the use of postoperative radiotherapy, but there is no clear evidence of definite advantage , 11.For our patient, the location of the tumor in the infratemporal fossa and the large infiltration of neighboring structures made it difficult to perform surgery, and treatment was limited to radiotherapy.MPNST is a very aggressive tumor that spreads via direct perineural invasion and the hematogenous route. The local recurrence rate is about 54 % and the rate of distant metastases to lung and bone is about 65 %. Lymph node involvement is not common .The prognosis of MPNSTs of the head and neck is relatively poorer than that of MPNSTs of the extremities and the trunk with documented 5-year survival rates from 15 to 35 % , 11, theMPNST of the paranasal sinuses and the anterior skull base is a rare tumor that involves a high rate of local invasion, distant spread, and local recurrence.The management of this aggressive neoplasm in this anatomical location remains an interdisciplinary challenge. A good outcome is mainly a function of local control by means of surgical resection. The prognosis is poorer compared to that occurring in the trunk and extremities.Written informed consent was obtained from the patient’s next-of-kin 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.


A 45-year-old man with a left testis tumor with a 25 mm para-aortic lymph node swelling, multiple bilateral pulmonary metastases, bilateral pulmonary embolism, and inferior vena cava (IVC) thrombus underwent a radical orchidectomy in our institution. The thrombus extended from the left gonadal vein to the left renal vein to the IVC. The fluorine-18 fluorodeoxyglucose (f-FDG) positron emission tomography (PET) computerized tomography (CT) demonstrated a hypermetabolic focus in the retroperitoneum and in the IVC thrombus. Before orchidectomy only lactate dehydrogenase (LDH) was high but all the serum tumor markers increased postoperatively. The tumor was staged pT1N2M1aS1, which was an intermediate prognosis, based on the International Germ Cell Cancer Collaborative Group consensus (IGCCCG). After 4 courses of bleomycin, etoposide, and cisplatin (BEP) chemotherapy the patient's tumor markers normalized and the thrombus disappeared. There was only one residual retroperitoneal lymph node M1. Retroperitoneal lymph node dissection was performed. The pathological examination revealed only necrotic tissues. The patient has been disease-free since surgery. Tumor thrombus is a rare complication of testis cancer. Extension into the inferior vena cava (IVC) and/or the right atrium needs appropriate treatment including chemotherapy and surgery. We report a case of testicular cancer with inferior vena cava tumor thrombus and multiple metastases and we discuss the different options of management after diagnosis.A 45-year-old man presented with a 3-month history of left scrotal pain initially diagnosed as an epididymitis. Physical examination and scrotal US revealed a left testis tumor. Computerized tomography (CT) scan of the chest, abdomen, and pelvis demonstrated a 25 mm para-aortic lymph node swelling, multiple bilateral pulmonary metastases, bilateral pulmonary embolism, and inferior vena cava (IVC) thrombus . The thrWe performed a fluorine-18 fluorodeoxyglucose (f-FDG) positron emission tomography (PET) computerized tomography (CT) in order to characterize the thrombus . It demoμg/L (≤7), human chorionic gonadotrophin (HCG) 2,9 UI/L (≤5), and LDH 440 UI/L .Serum tumor markers were normal except lactate dehydrogenase (LDH): alpha-fetoprotein (AFP) 5,2 The patient underwent left inguinal orchidectomy and pathological examination revealed embryonal carcinoma with intratubular germ cell neoplasia (ITGCN). The rete testis was involved by tumor. The epididymitis, the tunica albuginea, and the spermatic cord were free of tumor.μg/L, 2,9 UI/L, and 440 UI/L to 9,3 μg/L, 14,8 UI/L, and 486 UI/L.Postoperatively, serum tumor makers were increasing: AFP, HCG, and LDH, respectively, from 5,2 The tumor was diagnosed as a nonseminomatous germ cell tumor (NSCGT) with a clinical stage of pT1N2M1aS1, which was an intermediate prognosis, based on the International Germ Cell Cancer Collaborative Group consensus (IGCCCG).Chemotherapy was started with the bleomycin, etoposide, and cisplatin (BEP) regimen for four courses and therapeutic anticoagulation was started. Placement of an IVC filter was impossible because the upper limit of the thrombus was too close to the hepatic vein.After 4 courses of chemotherapy the patient's tumor markers normalized and the thrombus disappeared. There was only one residual retroperitoneal para-aortic lymph node on the CT scan whitch was hypermetabolic on the 18 fDG PET CT.Retroperitoneal lymph node dissection was performed. The pathological examination revealed only necrotic tissues. The patient has been disease-free at 8 months since surgery.Involvement of the inferior vena cava (IVC) by a testicular tumor is a rare event. Two autopsy series of patients with testicular germ cell tumors have suggested IVC involvement in 3% and 11% of patients . HusbandThe diagnosis of IVC thrombus can be performed by CT scan and by magnetic resonance imaging (MRI) . The 18FThere are two mechanisms by which the IVC may be involved. The first is due to the spread of the tumor by direct invasion of the spermatic vein and then of the vena cava. That explains why IVC invasion more frequently occurs with right side tumors because of the direct insertion of the right gonadal vein into the IVC. The second explanation is lymphatic spread and direct invasion from paracaval metastatic sites secondary to the development of lymphatic-venous shunting in severe lymphatic disease. Thus, bulky retroperitoneal disease is a major risk factor for IVC tumor thrombus.The most significant potential complication of IVC thrombosis is the risk of pulmonary  embolism which may lead to death. Bredael et al. reported that 9% of testicular cancer patients died of pulmonary embolism in autopsy cases . DespiteWhen the tumoral thrombus is extending to the heart such as the case report by Savarese et al., the risk of embolism is very high . There iFor patients with an IVC thrombus without cardiac involvement, surgery could be performed after chemotherapy with or without vena caval filter. While reviewing the literature over the past 30 years, we find only 2 cases reporting involvement of the IVC by testicular neoplasm treated by chemotherapy and anticoagulation without any vena cava resection , 11 and The histopathological composition of intraluminal vena caval thrombosis reflects the pathological features present in the testis, most frequently embryonal carcinoma component  , or is dTestis cancer with an IVC tumor thrombus is a rare event. Aggressive surgical approach before or after chemotherapy is the best therapeutic option for such patients. Most tumor thrombus rarely regresses totally after chemotherapy and thrombus surgical resection is commonly deployed.


Morgagni hernia is a congenital diaphragmatic defect that rarely presents with symptomatic findings in adults. The presence of one diaphragmatic defect may decrease the occurrence of a separate diaphragmatic defect. Appendicitis may be a unique presentation of incarcerated bowel in a Morgagni defect.Review of recent literature and presentation of a patient with Morgagni defect. Only five cases of simultaneous Morgagni hernia and paraesophageal hernia have been described in the English-language literature since 1958. Here, we report the first case of acute appendicitis within an incarcerated right Morgagni hernia in a 76-year-old patient who also had a paraesophageal hernia.This case illustrates that there is no role for watchful waiting in the management of Morgagni Defects when diagnosed in adult patients. The coexistence of two non-traumatic diaphragmatic hernias is rare; only five cases of combined Morgagni and paraesophageal hernias have been described in the English-language literature –5. FocalParaesophageal hiatal hernias account for approximately 14% of hiatal hernias and occur most commonly in adults . The defIn all five cases of simultaneous Morgagni hernia and paraesophageal hernia, the paraesophageal defect led to the main symptomatic complaint. Surgical intervention focused on repair of the dual diaphragmatic defects, with one case citing the incidental finding of Morgagni defect at time of paraesophageal repair. In this case report, we describe the surgical procedure and outcome of a patient with acute appendicitis within an incarcerated right Morgagni hernia combined with a paraesophageal hernia.The patient was a 76-year-old man with abdominal pain who was transferred to our hospital in August, 2013 from another institution. He had experienced 48 hours of obstipation, and computed tomography of the abdomen was concerning for an incarcerated Morgagni’s hernia. He had a 10-month history of intermittent abdominal discomfort after eating large meals. Evaluation at another institution in 2012 had revealed a right Morgagni’s hernia that was managed non-operatively. He also had a longstanding history of gastroesophageal reflux disease and a known moderate-sized paraesophageal hernia.A physical examination revealed decreased breath sounds in the base of the right lung and diffuse abdominal tenderness with guarding. There was focal tenderness at the right lateral chest wall. Computed tomography (CT) scans of the chest, abdomen and pelvis obtained for operative planning showed a large right Morgagni hernia containing small bowel, large bowel, and appendix with interval development of acute appendicitis when compared to CT scans obtained at the referring facility Figures  and 2. AWe chose an upper midline laparotomy over a laparoscopic approach, given the possibility of ischemic or necrotic bowel. There was a 4 cm by 6 cm anterior defect 5 cm right of midline, anterior to the liver Figure , containThe Morgagni hernia contents were reduced and an appendectomy was performed. The paraesophageal hernia was reduced, the hiatal defect was repaired and a Nissen fundoplication was performed. The Morgagni defect was repaired primarily with interrupted prolene sutures without removal of the hernia sac Figure .Figure 1Clostridium difficile colitis treated with oral vancomycin, and ileus that resolved with nasogastric decompression and bowel rest. He was discharged home on postoperative day 14. The final pathology report confirmed acute appendicitis and periappendiceal inflammation. At his follow-up visit, the patient was doing well and eating a normal diet without dysphagia or persistent gastroesophageal reflux.Postoperatively, the patient’s course was complicated by a pulmonary embolism that required anticoagulation with a direct thrombin inhibitor because of heparin resistance, new onset atrial fibrillation secondary to volume overload that improved with aggressive diuresis, This case is striking because of the combination of two diaphragmatic defects, together with an appendiceal pathology. We are not aware of another such case in the English-language literature. Our patient’s chronic, intermittent symptoms, and ultimate trajectory towards incarcerated bowel and acute appendicitis within the hernia, supports the existing teaching that there is no role for watchful waiting in the case of Morgagni hernia. Surgical repair of Morgagni hernia is indicated both in asymptomatic and symptomatic patients to avoid morbidity due to incarceration or ischemic changes of abdominal contents in the chest –8.Almost any non-retroperitoneal abdominal organ can be found within a Morgagni hernia, but the contents most commonly found during surgical exploration are omentum exclusively (31%), omentum and colon (29%), stomach (15%) and small bowel (11%) . The def, a review of 298 cases of adult Morgagni hernia revealed only 28% patients were asymptomatic at the time of presentation [Most Morgagni hernias come to clinical attention in childhood. It is a common belief that adult patients often are typically asymptomatic at the time of presentation, with the defect discovered incidentally on imaging during workup for other problems , 9. Howeentation . Most frOnce a Morgagni hernia is diagnosed, surgery is indicated because of the risk of incarcerated omentum, small bowel, colon or other complications such as intra-abdominal pathology mistaken for primarily thoracic pathology –8. In a Since the 1990s, thoracoscopic and laparoscopic methods of Morgagni hernia repair have been described as having shorter hospital stays and fewer complications than with laparotomy or thoracotomy, as well as successful long-term outcomes . HoweverIn conclusion, the combination of two diaphragmatic hernias and appendicitis make this case unusual, and provide further support for the existing teaching that there is no a role for watchful waiting in the case of Morgagni hernia. Instead, asymptomatic and symptomatic patients require surgery to avoid morbidity as a result of incarceration or compromise of abdominal contents in the chest.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 of this journal. No ethics committee approval required for clinical case report.


Schwannoma is a benign tumour of peripheral nerve sheath. It usually arises from head, neck, and trunk. Retroperitoneal schwannoma is a rare entity, accounting for only 0.3–3% of total schwannomas. Majority of retroperitoneal schwannomas reported in literature have a diameter of 5 to 15 cm. Preoperative diagnosis is difficult due to low frequency, nonspecific clinical course, and nonspecific imaging features. Histology usually provides definitive diagnosis. Schwannomas are solitary, well-circumscribed, and noninvasive, so complete surgical excision provides good result. We report a case of a 23-year-old male, who presented with progressive abdominal distension and intermittent episodes of intestinal obstruction. CECT was suggestive of huge solid-cystic mass in abdominopelvic region. Image guided percutaneous aspiration revealed around 1 litre of frank pus and FNAC was suggestive of abscess. Exploratory laparotomy revealed a large 32 × 28 × 26 cm mass with solid and cystic components containing 1 litre of pus. Histological features of tumour were suggestive of benign schwannoma and immunohistochemistry for S-100 was positive. Postoperative recovery was uneventful. We report this case of a retroperitoneal schwannoma because of giant size, rare location, unusual presentation, and diagnostic dilemma. Schwannoma is a benign, slow growing tumour originating from the Schwann cell. Majority of them arise from cranial nerves or nerves of upper extremities . We repoA 23-year-old male presented with history of abdominal distension since one year, which was insidious in onset and rapidly progressive. There was history of intermittent episodes of intestinal obstruction for the last 6 months. The patient was cachectic and febrile and was having tachycardia. There were no features suggestive of Von Recklinghausen's disease. Abdominal examination revealed an enormous rounded mass, which filled the entire abdomen. It was immobile, nontender, and variegated in consistency. Boundaries of mass were indistinct as superiorly it was extending below costal margins and inferiorly into the pelvis.Routine blood investigations were within normal range except haemoglobin (6 gm%). CECT (contrast enhanced computed tomography) features were suggestive of a large, lobulated, relatively well-defined, heterogeneously enhancing solid-cystic mass in abdominopelvic region . Mass waExploratory laparotomy through midline revealed a large 32 × 28 × 26 cm mass with solid and cystic components occupying whole abdomen. The mass contained around one litre of pus. Bowel loops were pushed to left upper quadrant of abdomen. Proximal right ureter was dilated and distal part was collapsed and displaced anteriorly over the mass . Pelvic Postoperative recovery was uneventful. Patient is doing well and no recurrence or sensory neurological deficits were noted even after 1 year of follow-up.Benign schwannomas are usually slow growing tumours and predominantly occur in females  between the 3rd and 5th decades of life [We found only 3 case reports of tumour of this size after extensive search. Foote et al. reported a giant retroperitoneal schwannoma of 43 × 40 × 20 cm in 1963 . UsuallyThe symptoms due to benign retroperitoneal schwannomas are nonspecific and are usually associated with compression of adjacent structures . The mosCECT and magnetic resonance imaging (MRI) are widely used as imaging methods for evaluation of retroperitoneal soft tissue tumours. MRI is usually regarded as the imaging modality of choice for most soft tissue lesions. On CECT, schwannoma appears as a well-defined, homogeneous mass with rim enhancement of the fibrous capsule . The degDefinitive diagnosis is based on histological analysis of biopsy specimens. Histological studies show different structural areas identified as Antoni A and Antoni B. The A areas express a solid hypercellularity and the B areas are hypocellular . IHC stuSchwannomas are well-circumscribed, firm, smooth-surfaced tumours. Treatment of choice is local excision as they are noninvasive. After complete excision, tumour does not recur so adjuvant therapy is not required. Identification of the originating nerve is not always possible and a minor degree of neurological impairment may be present afterward . In our


Extramedullary myeloid tumors (EMMTs) are the tumors of myeloid cells. These tumors may occur in all of the organs of the body, but some localizations are rare. Pancreatic involvement of EMMTs is a rare entity. Here we report a case of EMMT of the pancreas 4 years after allogeneic stem cell transplantation and we review the existing data about EMMTs involving the pancreas. Extramedüller miyeloid tümörler (EMMT) miyeloid hücrelerin neoplazileridir. Bu tümörler bütün organlarda oluşabilir fakat bazı lokalizasyonlarda EMMT saptanması nadirdir ve pankreasın EMMT ile tutulumu da nadirdir. Burada allogeneik kök hücre naklinden 4 yıl sonra gelişen pankreas EMMT’ü sunulmuş ve mevcut bilgi gözden geçirilmiştir.Granulocytic sarcoma is the tumor of immature myeloid cells. It was first defined by Burns about 100 years ago . It may A 37-year-old man was admitted to the hospital at December 2010 with fatigue, abdominal pain, and a 10-kg weight loss over 6 months. The pain was localized to the epigastrium and increased following food intake. Upper endoscopic examination 4 months prior to admission showed no abnormality. One month later he noted dark urine and jaundice. Abdominal ultrasonography showed a hilar mass of the pancreas. Additionally, there was intra/extrahepatic bile duct dilatation, hydropic gallbladder, and splenomegaly. Upper abdominal MRI and MR angiography showed a mass lesion localized at the head of pancreas, invading the superior mesenteric vein and bilateral renal veins with intra/extrahepatic bile duct dilatation. In October, a cholecystectomy was performed. Frozen section biopsy taken from the pancreas revealed chronic pancreatitis. After surgery, he was referred to our department and was hospitalized.He had a past medical history of intermediate-risk AML 5 years prior to admission. He had received 1 course of induction treatment  and 3 courses of postinduction treatment (high-dose cytosine arabinoside), and then an allogeneic stem cell transplant from his sister 4 years prior to admission. The conditioning regimen was Bu 6.4/Flu 120/ATG 20. Cyclosporine had been given for 11 months after transplantation with no subsequent immunosuppressive treatment. His family and social histories were unremarkable. Informed consent was obtained.Vital signs were within normal limits upon physical examination. There was jaundice, ascites, and abdominal scars associated with surgery. The liver and spleen were palpable 2 and 3 cm below the right and left costal margins, respectively, and there was (+++) pretibial edema.Laboratory findings were Hb of 11.1 g/dL, WBC 4x109/L, platelet count 194x109/L, blood urea nitrogen/creatinine 3.9/0.4 mg/dL, total/direct bilirubin 6.4/1.9 mg/dL, lactate dehydrogenase 546 IU, aspartate aminotransferase/alanine aminotransferase 48/87 IU, alkaline phosphatase 2348 IU, total protein/albumin 5.4/1.9 mg/dL, ferritin 1397 ng/mL, PT-INR 1.3, and thyroid function tests within normal limits. Hepatitis antibodies for HBV and HCV were found to be negative. Ascites cytology was negative for tumor cells. Other tumor markers were CEA, 1.1; CA19-9, 9.26; AFP, 10; and CA-125, 352 IU.PET/CT showed a huge mass involving the pancreas and invading surrounding structures .Upon follow-up, the most important problem of the patient was dyspeptic complaints. Upper gastrointestinal endoscopic examination was repeated and enterogastric reflux, F1 varices, portal hypertensive gastropathy, and a duodenal mass obstructing the lumen and diverticula were detected. Histopathological exam of the biopsy taken from the duodenal mass revealed myeloid sarcoma  with strongly positive myeloperoxidase . MoleculNearly all of the organs have been reported to be involved with EMMTs. The most commonly involved organs are the skin, lymph nodes, soft tissues, bone, and periosteum. The testes, kidneys, orbit, ovaries, uterus, bladder, gingiva, and stomach are the other sites involved in EMMTs . PancreaEMMTs are not rare after allogeneic transplantation and their most common localizations are the breast, gastrointestinal tract, skin, spine, central nervous system, and testes . LookingPancreatic involvement of EMMT developed in our case after allogeneic transplantation. At the beginning, pancreatic cancer had been suspected in the department of surgery. However, the history of AML and allotransplantation suggested EMMT. Histologic confirmation of this entity was made by duodenoscopic mass biopsy. Although the first endoscopic exam was negative when the patient was admitted for pancreatic mass, the endoscopic exam was repeated and biopsy of the then-detected mass showed myeloid sarcoma; this was confirmed by myeloperoxidase stain. There was no evidence of bone marrow relapse in our case. In conclusion, although rare, EMMT may be seen in the pancreas and may mimic pancreatic cancer. Pancreatic EMMT may be seen either in isolation or accompanying myeloid malignancies, and also following allogeneic stem cell transplantation.Conflict of Interest StatementThe authors of this paper have no conflicts of interest, including specific financial interests, relationships, and/ or affiliations relevant to the subject matter or materials included.


The treatment for giant cell tumors of the tendon sheath is surgical therapy, but surgical recurrence rates were reported to be as high as 50% in some cases. Therefore, complete radical excision of the lesion is the treatment of choice. If the tumor originates from the joint, it is important to perform capsulotomy. Here, the authors report the first case of successful treatment of a localized intra-articular giant cell tumor in the wrist by arthroscopic resection.A 28-year-old right-handed woman visited the clinic because of left wrist ulnar-side pain, which had been aggravated during the previous 15 days. Vague ulnar-side wrist pain had begun 2 years ago. When the authors examined the patient, the wrist showed mild swelling on the volo-ulnar aspect and the distal radioulnar joint, as well as volar joint line tenderness. She showed a positive result on the ulnocarpal stress test and displayed limited range of motion. Magnetic resonance imaging revealed an intra-articular mass with synovitis in the ulnocarpal joint. Wrist arthroscopy was performed using standard portals under regional anesthesia. The arthroscopic findings revealed a large, well-encapsulated, yellow lobulated soft-tissue mass that was attached to the volar side of the ulnocarpal ligament and connected to the extra-articular side. The mass was completely excised piece by piece with a grasping forceps. Histopathologic examination revealed that the lesion was an intra-articular localized form of a tenosynovial giant cell tumor.At 24-month follow-up, the patient was completely asymptomatic and had full range of motion in her left wrist, and no recurrence was found in magnetic resonance imaging follow-up evaluations.The authors suggest that the arthroscopic excision of intra-articular giant cell tumors, as in this case, may be an alternative method to open excisions, with many advantages. Giant cell tumors of the tendon sheath have a favorable prognosis because they are not considered to be malignant lesions.5 These tumors have less frequent association with joints. When association with a joint occurs, the tumor usually grows outside and extends into or near the joint. Here, we report the first case of successful treatment of a localized intra-articular giant cell tumor in the wrist by arthroscopic resection.Giant cell tumor of the tendon sheath is the second most common tumor found in the hand. It occurs almost exclusively in the hand, though it has also been reported in other parts of the body.The patient signed informed consent for the publication of this case report and any accompanying images. Ethical approval of this study was waived by the ethics committee of Chonbuk National University Hospital because it was a case report and there were fewer than 3 patients.A 28-year-old right-handed woman visited the clinic because of left wrist ulnar-side pain, which had been aggravated during the previous 15 days. Vague ulnar-side wrist pain had begun 2 years ago, and she reported that she was an office worker and had worked with a computer 5 days a week for 8 hours a day for the last 7 years. Her height was 163 cm and weight was 48 kg. She was diagnosed as having tenosynovitis at another clinic and was treated with physical therapy, medication, and steroid injection, but her symptoms did not improve. The patient had no trauma history or evidence of systemic diseases.When we examined the patient, the wrist showed mild swelling on the volo-ulnar aspect and the distal radioulnar joint, as well as volar joint line tenderness. She showed a positive result on the ulnocarpal stress test and displayed limited range of motion. The flexion and extension angles of the left wrist were 60° and 30°, respectively (with 70° and 60° in the right). The radial and ulnar deviation angles in the left wrist were 20° and 30°, respectively (with 20° and 45° in the right).Initial plain wrist radiograph showed pressure bone erosion with sclerotic margin in the triquetrum and mild soft-tissue swelling on the ulnar side Fig. . MagnetiPostoperatively, the patient was placed in a short arm splint. Two weeks after surgery, the patient was seen back at the clinic for a wound check, suture removal, and further immobilization in a removable short arm brace for an additional 4 weeks. After that, she was seen back at the clinic every 2 weeks to check the condition of the left wrist. At 6 weeks after the surgery, gradually increasing the active range of motion of left wrist was recommended. At 8 weeks after the surgery, there was no pain or swelling in her left wrist, and the flexion and extension angles were 60° and 30°, respectively. After that, she was seen back at the clinic every 6 months to check the condition of the left wrist. At the 14-month follow-up, the wrist showed no swelling on the volo-ulnar aspect and no joint line tenderness. The flexion and extension angles were 75° and 75°, respectively, and the radial and ulnar deviation angles were 35° and 35°, respectively. The wrist grip strength was 16 kg (20 kg in the right). The visual analogue scale for pain was 0 at rest .At 24-month follow-up, the patient was completely asymptomatic and had full range of motion in her left wrist, and no recurrence was found on plain radiograph or magnetic resonance image follow-up evaluations Fig. .6 Even higher occurrence rates are typically observed upon subsequent re-excision.6 Therefore, complete radical excision of the lesion is the treatment of choice. If the tumor originates from the joint, it is important to perform capsulotomy to inspect the joint and debride any pigmented tissue within the joint. The requirement for a large incision carries the risk of infection, necrosis, or a mechanically alterable scar. In some patients, this type of complete excision may also involve the sacrifice of certain tissues, such as tendon or portions of the bone in order for the lesion to be sufficiently eradicated. Therefore, in cases of intra-articular type tumors, removal can be accomplished noninvasively through arthroscopy without resection of the affected joint. This can reduce complications and allow for rehabilitation to be started early.The treatment for giant cell tumors of the tendon sheath is surgical therapy, but surgical recurrence rates were reported to be as high as 50% in some cases, and as low as 5% after primary excision.12 These reports have described the advantages of using this procedure.10 In addition, a comprehensive assessment of the joint and associated disorders can be made with simultaneous treatment during arthroscopy. The benefits include small, nonproblematic scars, reduced postoperative pain, and earlier rehabilitation. Although arthroscopic wrist surgery emerged from the stage of ablative surgery to become reparative surgery, it is moving toward the territory of reconstructive surgery.13 To the best of our knowledge, there have been no other reports of the arthroscopic resection of giant cell tumor in the wrist to date.Many articles have reported the successful treatment of tenosynovial giant cell tumors in the knee, ankle, and hip with arthroscopy.In the case of our patient, a portion of the tumor was located at the ulnocarpal joint, and the rest of the tumor was located in the extra-articular area, so it was very difficult to completely remove with the arthroscopic technique alone. The giant cell tumor was connected, so the intra-articular portion of the tumor, however, was removed by grasping forceps, and the extra-articular portion of the tumor was removed by pulling it into the joint. During the operation, the assistant pushed the extra-articular portion of the tumor into the joint with finger pressure, so it could be removed via arthroscopy.We suggest that the arthroscopic excision of intra-articular giant cell tumors, as in this case, may be an alternative method to open excision, with many advantages.


A 29-year-old male patient with a functioning pituitary macroadenoma is discussed. The pituitary mass was detected by MRI after the patient presented with sudden onset of headache, suggestive of an apoplectic event. The headache resolved with analgesic medications. Within a follow-up period of one week, the pituitary mass had spontaneously regressed to nearly half its original size without any therapy. The patient never reported any visual complaints and displayed no signs of hypopituitarism. Elevated prolactin levels were present. Seven weeks after the initial event, the pituitary mass showed continued regression on MRI. Prolactin levels remained elevated. This case provides a unique look at the rapid spontaneous regression of mass effect that may occur following apoplexy of a pituitary adenoma. Apoplexy into a prolactinoma is not rare and is more likely to occur in macroprolactinomas. In those that hemorrhage occurs, the majority of patients are women with an average age at diagnosis of 31, ranging from 25 to 37. Men have an average age at diagnosis of 41, ranging from 32 to 54 [A 29-year-old man presented with history of fatigue and sudden onset of headache on the left side of his head. The fatigue had been present for roughly six months. The headache occurred more recently. The patient reported that the headache felt like a migraine and was severe enough that he went to the emergency room. An MRA was done which was negative. CT of the head revealed a questionable enlarged pituitary gland. An MRI without contrast was then done which showed a mass, 2.5 cm in maximal diameter , arisingA week later, after being evaluated and treated in the ER, the patient was noted to have an elevated prolactin level of 120 ng/mL . Another MRI that was done, with contrast, revealed a pituitary mass with heterogeneous enhancement . There wWith the spontaneous regression of the pituitary mass and no visual deficits on clinical exam, transsphenoidal resection of this mass was not recommended. To establish a baseline of the pituitary mass and assess any changes following the start of medical treatment, another MRI was performed. This MRI, done seven weeks after presenting to the ER, showed again further reduction in the size of the pituitary mass with it now measuring 8.1 mm in maximal diameter  comparedThe start of the spontaneous regression of pituitary adenomas following apoplexy has not been clearly defined nor has the time it takes for complete resolution been clearly defined. This particular case shows that spontaneous regression can occur as early as one week following an apoplectic event. It also reveals the rate of reduction for this particular pituitary macroadenoma. At the rate it was decreasing, the mass could have potentially resolved on its own without any further treatment. This case contributes to the literature that presents spontaneous regression of pituitary adenomas.A few of the cases showing pituitary adenomas regressing without any treatment are discussed here. Kachhara et al. present the case of a 42-year-old male with history of sudden onset of headache with associated vomiting. The patient was only treated with analgesic medications, and his symptoms resolved in one week. CT scan revealed a contrast-enhancing sellar-suprasellar mass with no extension. An MRI performed six weeks later showed complete resolution of the pituitary adenoma. The patient's pituitary macroadenoma had completely resolved with no treatment and no evidence of hypopituitarism . Rainov In our particular case where the patient potentially has an inefficient macroprolactinoma, the mass was shown to spontaneously shrink in one week without any treatment. Although complete resolution did not occur before the decision to start cabergoline, our case provides a unique opportunity to track the spontaneous reduction in size of a pituitary mass following an apoplectic event. This case also allows us to observe how soon the reduction can occur. In one week, the patient's pituitary mass decreased from 2.5 cm  to 1.5 cPituitary adenomas can be asymptomatic, and pituitary tumor apoplexy may be the first indication that an adenoma is present. Pituitary tumor apoplexy is thought to be due to a rapidly growing tumor that exceeds its vascular supply while another hypothesis involves the compression of portal vessels by the growing mass leading to ischemic necrosis with or without hemorrhage . ApoplexSurgical intervention is indicated in a majority of pituitary apoplexy cases, but conservative management can be used for select cases. Correction of electrolyte imbalances, hemodynamic stabilization, and corticosteroid repletion are among the most important initial interventions . None ofStalk effect typically presents with prolactin levels of 30–200 ng/mL, and this elevation is attributed to diminished dopaminergic inhibition . With fiThe regression and disappearance of pituitary adenomas following apoplexy are commonly reported after patients are medically or surgically treated. However, the rapidity of resolution is unknown. Apoplexy has been shown to decrease the production of pituitary hormones released by functioning pituitary adenomas, leading to spontaneous resolution of symptoms and in some cases leading to hypopituitarism , 12.The subsiding of hormonal effects associated with acromegaly and Cushing's disease has been reported , 14. We


Gastrointestinal (GI) perforation accounts for over 90% of acute abdomen and pneumoperitoneum. The presence of pneumoperitoneum secondary to spontaneously perforated pyometra is an interesting yet confusing finding given the absence of gastrointestinal (GI) perforation, because pyometra is more common in postmenopausal women. We report an instructive case of diffuse peritonitis caused by spontaneous perforation of pyometra. A 70-year-old postmenopausal female was admitted to surgical emergency with signs of diffuse peritonitis. After resuscitation, an emergency laparotomy was performed because of suspicion of GI perforation. At laparotomy, about 2,000 mL of purulent fluid was found to be present in peritoneal cavity, while GI tract was intact. A rent with a diameter of 5 mm was found on anterior fundus of uterus. A total abdominal hysterectomy with a bilateral salpingo-oophorectomy was performed. Despite intensive care and a course of antibiotics, the patient died of multiple organ failure resulting from sepsis on postoperative day 16. Our case illustrates the importance of clinical knowledge of acute gynecological diseases, which are not uncommonly encountered by the general surgeon. Moreover, good appreciation of pelvic anatomy and close collaboration with gynecology and GI surgery colleagues is essential as operative intervention is often required. Pyometra is defined as buildup of pus  in the uterine cavity , 2. It i6/L and C-reactive protein at 32.39 mg/dL . Urgent computed tomography (CT) of the abdomen reported the presence of fluid and free intraperitoneal air mostly in the upper abdomen  3 years ago. Her gynecological history was unremarkable, and there was no history of postmenopausal bleeding or vaginal discharge. On the physical examination, her abdomen was very tender and was distended and showed muscle rigidity. Rebound tenderness was absent. Bowel sounds were hypoactive. There was no palpable mass. Her blood pressure was 81/43 mmHg and pulse rate was 110 beats/min. Laboratory studies demonstrated a white blood cell count of 22,350 × 10 abdomen , in addi Bacteroides fragilis, Escherichia coli, and Eubacterium.Following prompt resuscitation and intravenous antibiotics , laparotomy was performed, which confirmed the presence of free air and pus as well as an inflamed small intestine. Two thousand mL of pus was found to be present in peritoneal cavity. The rest of alimentary tract, gall bladder, and liver were normal. During peritoneal lavage, we found a perforation with a diameter of approximately 5 mm over fundus of the uterus. Total abdominal hysterectomy, copious saline lavage, and tube drainage were performed as a joint procedure. No colonic resection was deemed necessary. The uterus was soft and slightly enlarged. Retrospective review of the CT reported, in addition to the previously documented findings, a fluid-filled uterus with free air in the anterior wall . HistoloPostoperatively, intravenous antibiotics  and sepsis treatment were continued and the patient was admitted to the intensive care unit with strict management of respiration and circulation. Her condition improved over time and she was transferred to the gynecological unit on postoperative day (POD) 4. However, she broke into decrease in blood pressure and saturation, pyrexia, and abdominal distension on POD 11. Although hepatic and renal dysfunctions were not revealed, sepsis and multiple organ failure were suspected. A contrast-enhanced CT showed the increased abdominal pus, and then vancomycin (1 g/day) and meropenem (1 g/day) were added to antibiotic therapy. Despite exhaustive clinical efforts, sepsis grew progressively worse and the patient died on POD 16.Accumulation of purulent material in the uterine is termed as pyometra. It is an uncommon condition occurring mainly in elderly postmenopausal women and due to impaired drainage of uterine cavity. Impaired drainage conditions include cervical canal stenosis, benign or malignant cervical lesions, and surgical complications –9. AlthoAbdominal pain, vomiting, and fever predominate as the presenting symptoms in spontaneously perforated pyometra while gynecological symptoms such as vaginal bleeding or discharge occur in less than 10% , 10. GenGI perforation is the cause of pneumoperitoneum in 85–95% of cases and requires surgical intervention as its definitive management , 11. Pre Escherichia coli and Bacteroides fragilis [The presence of free intraperitoneal and intrauterine air may be due to gas forming organisms such asfragilis , althougRuptured pyometra cases should be managed by total abdominal hysterectomy with bilateral salpingo-oophorectomy, thorough drainage and irrigation of pelvoabdominal cavity, postoperative intensive care support, and administration of broad-spectrum antibiotics –7, 12–15In summary, spontaneously perforated pyometra is an interesting yet confusing finding given the absence of gastrointestinal (GI) perforation, because pyometra is more common in postmenopausal women. It should be kept in mind as a differential diagnosis in postmenopausal women presenting with acute abdomen and pneumoperitoneum. An appreciation of acute gynecological disease is essential as many patients with gynecological pathologies may present with signs and symptoms mimicking those of GI origin. Despite the absence of gynecological symptoms in most cases, gynecological sonography may be the most convenient to illustrate fluid accumulation in utero. Correct diagnosis and proper treatment can reduce morbidity and mortality.


Introduction. Gallstone ileus (GSI) of the colon is an extremely rare entity with potentially serious complications including perforation. Case Presentation. An 88-year-old man presented to the emergency department with abdominal pain and distension. Clinical exam revealed signs of peritonism. Computed tomography (CT) revealed GSI of the transverse colon with a closed-loop large bowel obstruction (LBO) and caecal perforation. The patient underwent emergency laparotomy. A right hemicolectomy was performed, the gallstone was removed, and a primary bowel anastomosis was undertaken. A Foley catheter was sutured into the residual gallbladder bed to create a controlled biliary fistula. The patient recovered well postoperatively with no complications. He was discharged home with the Foley catheter in situ. Discussion. Gallstone ileus is a difficult diagnosis both clinically and radiologically with only 50% of cases being diagnosed preoperatively. Most commonly it is associated with impaction at the ileocaecal valve and small bowel obstruction. Gallstone ileus should also be considered as a rare but potential cause of LBO. This is the first reported case of caecal perforation secondary to gallstone ileus of the transverse colon. Successful operative management consisted of a one-stage procedure with right hemicolectomy and formation of a controlled biliary fistula. Most common mechanical bowel obstruction due to a gallstone occurs at the ileocaecal valve due to the reduced diameter and reduced active peristalsis . This haColonic obstruction due to a gallstone occurs rarely , 5. The An 88-year-old man presented to the Emergency Department in a tertiary Irish hospital with abdominal pain and distension having being referred by his general practitioner. His symptoms had occurred three days prior to presentation. There was no associated nausea or vomiting. The patient had no previous medical or surgical history and was on no regular medications. He had no previous known history of gallstones.An abdominal X-ray and erect chest X-ray were requested. His abdominal X-ray revealed a distended large bowel with a collapsed small bowel . His cheThe patient underwent a vertical midline laparotomy. The right colon was noted to be grossly distended with caecal ischemia and perforation. A seven-centimetre gallstone was noted to be causing obstruction at the proximal third of the transverse colon, where a cholecystocolic fistula communicating between the transverse colon and the gallbladder was identified Figures , and 5. Postoperatively the patient recovered well. The patient recommenced oral intake day three postoperatively and completed a ten-day course of intravenous antibiotics. He was discharged home with the Foley catheter in situ, where it was subsequently removed in surgical outpatients.It is acknowledged that GSI is a difficult diagnosis both clinically and radiologically with only 50% of cases being diagnosed preoperatively . Only 50With regard to imaging techniques, CT is recognized as having the greatest diagnostic accuracy with a sensitivity of 93% and specificity of 100% . In an eDebate exists regarding surgical options for GSI causing uncomplicated small bowel obstruction. Enterotomy alone with removal of the obstructing gallstone alone with expectant management is regarded as the most appropriate surgical management in an emergency setting , 9. SingThere is a paucity of reported cases involving colonic perforation. In 2009, van Kerschaver et al. reported on GSI causing necrosis and perforation of the rectosigmoid junction. A Hartmann's procedure was performed to treat the perforation and the obstruction. A subsequent cholecystectomy and closure of the cholecystocolonic fistula were planned to be performed at the time of restoration of the intestinal continuity . D'HondtGallstone ileus causing a LBO is a rare entity, and cases involving colonic perforation are rarer still. Previous reported cases have demonstrated colonic perforation at the site of mechanical obstruction. We report on a closed-loop LBO leading to remote perforation secondary to GSI at the transverse colon. Successful management included right hemicolectomy and controlled biliary fistula formation. Gallstone ileus should be considered in patients as a potential cause of bowel obstruction, even without previously known history of gallstones.