Melanosis Coli is described as black or brown discolouration of the mucosa of the colon. Its a benign condition, which arises from anthraquinone laxative abuse and has no symptoms of its own. The main importance of diagnosing Melanosis Coli correctly lies in the fact that if its extensive, there may be difficulty in differentiating it from ischemic colitis.We present a case of extensive Melanosis Coli involving the whole of large bowel that appeared gangrenous. A sub total colectomy was performed on presumed diagnosis of ischemic bowel.This report reminds the clinicians that extensive Melanosis Coli may mimic ischemic colitis and thus must be considered as a differential diagnosis. Melanosis Coli is a common condition characterized by brown or black pigmentation of colonic mucosa. The condition itself is asymptomatic and the diagnosis is usually made on incidental endoscopic or histological findings. The importance of correctly diagnosing Melanosis Coli lies in the fact that if extensive, it can mimic ischemic mucosa and thus pose a diagnostic dilemma. This clinical situation has been reported earlier in a supposedly necrosed stoma ,2. We pr3/dland a CRP of 40. An abdominal CT scan revealed a marked gaseous distention of large bowel with no evidence of stricture or mass lesion. The patient underwent an emergency operation. On laparotomy the large bowel was quite distended and looked akinetic. The mucosa appeared dusky and necrotic from the outside. This abnormal appearance started from caecum and involved the large bowel all the way to the rectosigmoid junction. The pulses, however, were present in the major vessels and the serosa looked moist. There was no obstructing lesion distally and the small bowel was normal. The macroscopic appearance of dusky large bowel mucosa taken together with a history of AF led us to the diagnosis of ischemic colitis. A sub-total colectomy was performed with an end ileostomy. (Figure th postoperative day. The histopathology revealed extensive Melanosis Coli with spread to the pericolonic lymph nodes. (Figure A 63 years old woman presented with a two days history of worsening abdominal pain and distention. She had a past history of deep venous thrombosis, atrial fibrillation [AF] and long standing constipation. Her medications included various herbal laxatives and warfarin. On examination she had quite a distended abdomen with diffuse abdominal tenderness. The blood investigations showed an abnormal white cell count of 14 × 10. Figure  There waMelanosis Coli occurs because of the deposition of a brown black pigment called lipofuscin in the lamina propria of the colon . The iniA variable incidence of Melanosis Coli has been reported in various studies ,5 with sIn Melanosis Coli the pigmentation doesn't affect the small bowel . The posMelanosis Coli is a benign reversible condition with no malignant potential. The main importance of recognizing this condition correctly is to differentiate it from some of the other more sinister pathologies like ischemic bowel mucosa as highlighted by our case. As per our literature search of English language articles this is the most extensive case of MC ever reported and the only one in which a colectomy was performed on the basis of its appearance.The histopathology of the specimen not only showed that the there was extensive Melanosis Coli but it had also spread to the pericolonic lymph nodes, again an unusual finding. This is only the sixth case of Melanosis Coli reported in literature where the pigmentation from the bowel had spread to the regional lymph nodes .It's not easy to differentiate Ischemic Colitis from Melanosis Coli when the later is extensive because of various reasons. Firstly, it's very difficult to clinically differentiate Melanosis Coli from Ischemic Colitis when you are doing an operation in emergency circumstances as both conditions can co-exist. Secondly when you encounter this situation in an operation, there aren't any surgical maneuvers or quick tests that would help differentiate between the two. Tests like frozen section of a lymph node or a peroperative colonoscopic biopsy may not be feasible out of hours because of unavailability of a histo-pathologist and time considerations in emergency operations.The only practical thing which may be of help in such a situation is the awareness that Melanosis Coli can present like this and with that knowledge one can make an informed decision whether there is enough justification to take the colon out or to look for an alternate diagnosis. One of the reasons due to which we went ahead and did the colectomy was the fact that the appearance of the colon was that of ischemic bowel and we didn't know if anything else could also look like that.This report reminds the clinicians that extensive MC may mimic ischemic colitis and thus must be considered as a differential diagnosis.


A presacral mass can present a diagnostic dilemma for the surgical oncologist. Differential diagnoses include congenital causes such as teratoma or chordoma, neurological causes such as neurilemoma or neurofibroma or other malignancies such as lymphoma or sarcoma. Diagnosis usually requires imaging such as CT and MRI and tissue biopsy. We present an unusual cause of a presacral mass being extramedullary haematopoiesis, found incidentally in a 71 year old female. Extramedullary haematopoiesis is defined as the production of myeloid and erythroid elements outside of the bone-marrow. This diagnosis is extremely rare in the presacral area especially in a patient with no haematological abnormalities. A review of the literature is presented. Extramedullary haematopoiesis (EH) is defined as the production of myeloid and erythroid elements outside of the bone-marrow. It is usually a mechanism to compensate for haemolytic anaemia such as spherocytosis or thalassaemia, or as a response to abnormal bone-marrow function seen in disorders such as myelofibrosis or leukaemia -4.Approximately only 5% of cases of EH occur outside of the liver and spleen. Of these, the most common sites are surrounding the vertebral column , lymph nodes, retroperitoneum, lung and pleura . Other sThe differential diagnosis of a presacral mass can be categorized as congenital (65%), neurogenic (12%), osseous (11%) or miscellaneous (12%). Congenital causes are the most common and include causes such as epidermoid cyst and teratoma, especially in females and chordomas especially in males. Developmental cysts tend to occur in middle-age, are associated with anorectal malformation and sacral bone defects and are generally benign. They are usually asymptomatic, but can become infected, causing pain and discomfort. Other symptoms are associated with mass effect, occasionally causing bowel and bladder symptoms. TeratomNeurogenic causes of a presacral mass include neurilemomas (Schwanommas) or neurofibromas. Neurilemomas are rare and present with nonspecific symptoms and radiological findings. They can look cystic on imaging. Full excision is recommended as they have a high recurrence with other forms of treatment. They tend to be benign with minimal metastatic potential.Lymphoma, sarcoma and giant cell tumours account for other more common causes . Solid lA 71 year old female was referred for investigation of a presacral mass found incidentally during a staging CT figure  of a recHer CT scan revealed a suggestion of an ill-defined presacral mass, reported as possible oedema. The subsequent staging MRI clarified a 3.3 × 1.9 × 2.3 cm presacral mass at the level of S4/S5. Evidence of bony erosion could not be seen.Initially, differential diagnoses for this mass included chordoma, schwannoma and plasmacytoma. The mass was further investigated with a whole body thallium study and a CT-guided biopsy figure . The thaThis remarkable diagnosis was further investigated with the help of a haematologist. A full history found no personal or family history of bleeding disorders or anaemia. Full blood examination and liver function tests revealed no abnormality. A bone marrow biopsy was taken which revealed mildly hypercellular marrow, showing a mild increase in trilineage haemopoiesis. This was likely to be reactive. A cytogenetics study was organized to differentiate reactive marrow from early myelodysplasia. No cytogenetic abnormality was detected.The case was discussed at a multi-disciplinary meeting and a decision was made that this mass was benign in nature. No further investigations or treatment was deemed necessary. The patient subsequently underwent an uncomplicated resection of her rectal carcinoma. This is only the second case reported in the literature of a prescaral site of extramedullary haematopoiesis with no obvious underlying cause.In the literature, the majority of the cases of presacral EH were found incidentally (Table A CT-scan is a common preliminary investigation. EH usually appears as heterogeneous lobulated solid mass with smooth margins. Its density is similar to that of soft-tissue, slightly denser than fluid ,4,16. OtAn MRI of presacral EH often reveals a characteristic well encapsulated tumour with a slightly higher signal intensity than muscle on T1 and T2 weighted imaging, consistent with fatty tissue . There iNuclear imaging using a whole body scan post intravenous administration of Tc-99m sulfur colloid revealing increased uptake within the site of the mass is also consistent with EH,15. ThisWith any mass, a tissue sample is usually considered essential to correct diagnosis. The presacral location presents a technical challenge to obtaining such a sample. Possible complications associated with a biopsy of this region include infection and bleeding, especially resulting from insult to the middle sacral artery. Hence, Literature regarding the management of such a condition is limited due to its rarity. Of the reported cases 5 were treated conservatively and monitored, with none reporting an adverse outcome -5. Four The prognosis of presacral EH is highly reliant on the underlying cause. All cases with reported follow-up had a satisfactory outcome, with those requiring excision for symptoms remaining symptom free, and thoExtramedullary haematopoiesis as a diagnosis for a presacral mass is rare. It is usually asymptomatic and hence typically discovered incidentally. It is heavily associated with haematological disorders, especially myleodysplasia and thalassaemia. A tissue biopsy is generally performed to differentiate this diagnosis from other, more sinister conditions that are more common in this region. Once a diagnosis is made, it is considered safe to treat these lesions conservatively in an asymptomatic patient. In a symptomatic patient, the mass may either be excised or radiotherapy used to shrink its presence.The authors declare that they have no competing interests.SB researched the case and completed a literature review and wrote the initial draft of this case report. MB helped with the literature review and the discussion and edited the final draft of this case report. JS provided invaluable information on the pathology and its significance and was instrumental in describing the pathological findings. PC provided clinical information regarding the patient and the case and edited the final draft of this case report. All authors read and approved the final manuscript.Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.


Paragangliomas affecting the filum terminale are extremely rare, benign tumors. The literature yielded thirty-two cases of paraganglioma in this site.A 49 year-old-man, whose presenting symptoms were low back pain and left leg weakness, was diagnosed as having a paraganglioma of the filum terminale. The clinical, histological and radiological characteristics of this case, that brings the total number of cases described to 33, are discussed in the light of published data.This extremely rare pathology can usually be successfully treated by total surgical resection, which represents the gold standard. In the event of incomplete removal, assiduous long-term follow-up is mandatory. Paragangliomas affecting the filum terminale are extremely rare, benign tumours and only 32 cases have been reported in the international literature to date: their characteristics are summarized in Table This 49 year-old man was admitted with a 3-month history of increasing left leg weakness accompanied by progressive low back pain that radiated to the left leg. Painful symptoms were worsened by movement, coughing and upper body rotation. Sphincter dysfunction was absent. Neurological evaluation revealed tenderness in the region of the paravertebral muscles: a straight raise test of the left leg produced posterior thigh pain at 30°. Moderate weakness of the left leg was also observed and dorsal flexion of the foot in the area innervated by L5, revealed a 1/5 reduction of strength. Pinprick sensation was diminished but no dermatomal distribution was identified. Tendon reflexes, peri-anal sensitivity and anal tone were normal. Magnetic resonance imaging of the lumbosacral spinal segment, before and after Gadolinium administration, disclosed an L4-L5 oval shaped lesion measuring 3.3 × 2 × 1 cm. The lesion was intradural and extramedullary and was surrounded by bands of low signal intensity on T1 and T2-weighted images fig.. on the Paraganglioma is a neuro-endocrine tumour, it had a neuroectodermal origin and derives from the embryonic sympathetic and parasympathetic nervous system. The first authors to describe this pathological entity were Miller and Torack in 1970, denominating it a secretory ependymoma , whereasParaganglioma of the filum terminale is an extremely uncommon condition. It usually presents with low back pain and radicular pain and may be accompanied by a loss of strength in some cases. Magnetic resonance with Gadolinium administration gives useful information for surgical planning but is not diagnostic. The goal of surgical treatment is total resection of the lesion that guarantees cure in the majority of cases. Patients in whom complete removal is not feasible require assiduous long-term follow-up.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 journalThe authors have not been influenced by any financial or personal relationship with people or organizations in preparation of this studyAll authors have made substantial contributions to in the design of the article: AL was responsible for editing, English editing, correction, search of the literature, conception and design, and has contributed in surgical technique. RT was responsible for editorship of the manuscript and has contributed in surgical technique. NM was responsible for the search of the literature. PR was responsible for the search of the literature. MS was responsible for the oncology consulting. MA was responsible for the histology consulting and pathology examination. RD is the principal surgeon and was responsible for editing.


Benign mesenteric lipodystrophy is rare and often presents in a non-specific fashion. Imaging findings may mimic a range of malignant conditions, particularly malignant ovarian disease in women.We present the case of a 61-year-old Caucasian woman who was referred to the gynaecology service at our institution and was thought to have ovarian malignancy, and required a laparotomy. However, histopathological analysis unexpectedly revealed benign mesenteric lipodystrophy.Benign mesenteric lipodystrophy may mimic a range of conditions, particularly malignancy. Benign mesenteric lipodystrophy is a rare condition with just over 200 cases being noted in the worldwide literature . It was Its etiology is unclear. However, previous trauma, mesenteric ischemia or infection have been suggested as potential causes. Other documented possible associations with this disease include tuberculosis , pancreaA 61-year-old British Caucasian woman was initially referred to the gynecology service in our institution with low abdominal and pelvic pain. An initial clinical examination was unremarkable. However, a pelvic ultrasound scan was undertaken and revealed cystic masses within the pelvis. A potential diagnosis of ovarian malignancy was considered and computed tomography (CT) was then performed, which also suggested an intra-abdominal mass of possible ovarian origin Figure . These iA trial of tamoxifen 20 mg orally once daily was instituted in an attempt to reduce the size of the mesenteric masses. However, follow up CT of the abdomen did not shown any response after six months of therapy, so tamoxifen was discontinued. A follow-up small bowel study to exclude small bowel stricturing as a consequence of mesenteric fibrosis has not revealed any abnormality. Two years after the initial surgery, our patient remains well.Benign mesenteric lipodystrophy is rare and has been described in most detail in a three-case series by Durst , Kipfer A range of symptoms are described, including most commonly undiagnosed abdominal pain, and more commonly diagnosed signs of gastrointestinal obstruction. Fever, weight loss, abdominal mass or even a protein-losing enteropathy have been described. The frequency of differing symptoms is unclear because of the rarity of the condition but most reports suggest that the initial presentation is either with abdominal pain or as an asymptomatic finding at cross-sectional imaging. Biochemical tests are usually unhelpful, while hematological investigations may only show anaemia or a raised erythrocyte sedimentation rate (ESR) but are non-specific.et al. [CT may help in making the diagnosis of mesenteric lipodystrophy. There are a number of features on CT that may suggest mesenteric lipodystrophy. These include increased attenuation in the small bowel described by Seo et al.  as the 'et al. ,12.The CT findings are unfortunately not specific and can mimic other lesions of the mesentery including lymphoma, lipoma, edema , tuberculosis, carcinomatosis or, very rarely, mesothelioma . The extDiagnosis is usually during laparotomy, although it can also be done during biopsy at the time of laparoscopy or percutaneously ,14. Reseet al. suggest that immunohistochemistry using a panel of antibodies  may help differentiate the histological types [Histologically, the condition shows a progressive series of changes. However, the different histological stages often co-exist within the same specimen. Initially, the mesentry is infiltrated with lipid-filled macrophages within the fat-filled septa of the mesenteric adipose tissue, which is known as mesenteric lipodystrophy. As the condition progresses and inflammation supervenes, lymphocytes infiltrate the mesentery and lipid cystic necrosis can be identified. This is a change known as mesenteric panniculitis. Later, necrosis with associated fibrosis dominates. This is associated with shortening of the mesentery, and is called the retractile mesenteritis stage. Typically, these changes may be identified by H&E staining. However, in cases where there is diagnostic difficulty particularly, when differentiating mesenteric lipodystrophy from gastrointestinal stromal tumours.(GISTs) and mesenteric fibromatosis, Montgomery al types .Treatment of this condition depends on the stage of the disease: early changes are nearly always managed conservatively as the illness resolves in many individuals with the lipodystrophy phase without intervention. In the later panniculitis or fibrotic phases of the illness, a range of treatments have been investigated. To suppress inflammation, steroids, cyclophosphamide , azathioMesenteric lipodystrophy is a rare condition that can mimic a number of intra-abdominal conditions including ovarian pathology. It can be difficult to diagnose and is often only fully apparent at the time of laparoscopy or laparotomy. Histologically, it forms part of a continuum of inflammation and fibrosis and may often in the early stages resolve spontaneously. Although often unnecessary in the early stages, treatment may require immunosuppression or even resectional surgery in the later stages if the disease progresses.CT: computerised tomography; ESR: erythrocyte sedimentation rate; H&E: Haematoxylin and Eosin; GISTs: Gastrointestinal stromal tumours.Written informed consent was obtained from our patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the journal's Editor-in-Chief.The authors declare that they have no competing interests.JR and PB were involved in the direct clinical care of our patient and therapeutic planning and both authors contributed equally to the manuscript. Both authors read and approved the final manuscript


We report a case of a 20-year-old male who presented with symptoms and signs of intestinal obstruction. The patient reported no previous history of abdominal surgery or trauma while clinical and radiographic examinations were not diagnostic. An open laparotomy was subsequently performed and the intraoperative findings were consistent with a congenital band extending from the antimesenteric wall of the jejunum to the root of mesentery. The band was ligated and divided with an uneventful postoperative course. Congenital bands are extremely rare. Their exact incidence is still unknown and usually observed in childhood. This case, therefore, represents an unusual surgical problem in an older individual in which the diagnosis was clinically unexpected. Small bowel obstruction is the most common surgical disorder of the small intestine. Adhesions are by far the most frequent causes followed by hernias, tumors, intussusception, foreign bodies, gallstones, and inflammatory bowel disease.Obstruction by a congenital band is extremely rare and usually observed in childhood.This report presents a 20-year-old male with symptoms of intestinal obstruction, subsequently treated by ligation and division of a congenital band. This is the first report of a band running from the root of the mesentery to the jejunum.A 20-year-old male presented to the Emergency Department complaining of epigastric pain of 6-h duration. He also reported an episode of vomiting. There was no history of abdominal surgery or trauma. Abdominal palpation revealed moderate tenderness over the epigastric area without rebound tenderness or a palpable mass. On auscultation, there were active bowel sounds. Rectal examination was normal. At the time of arrival, he had a temperature of 37.0°C, a blood pressure of 130/80 mm Hg, and a pulse rate of 85 beats/min. Laboratory data showed an elevated lactate dehydrogenase of 241 IU/l and creatinophosphokinase of 507 IU/l, but no leukocytosis.Abdominal plain X-ray showed intestinal loops with air-fluid levels on the left side of the abdomen, which remained unaltered in a subsequent X-ray after 2 h . Due to et al. have recently reported in a series of eight patients that bands principally were located between ascending colon and terminal ileum followed by ligament of Treitz and terminal ileum; between the right lobe of liver and terminal ileum; and between the right lobe of liver and ascending colon.[et al. have reported a band extending from the iliac fossa to the sigmoid mesocolon,[et al. reported the presence of a jejuno-jejuno congenital band.[Congenital bands are a rare cause of intestinal obstruction in infancy and childhood. Their occurrence in adults is an extremely rare condition.2 Obstrucng colon. Lin et aesocolon, while Ittal band. As far atal band.6 In all Patients usually present with symptoms of intestinal obstruction, and despite the availability and wide use of modern imaging techniques, preoperative diagnosis is very difficult to establish. Plain films are nonspecific. Ultrasound scan might provide details of localized distended intestinal loops or indirect signs of peritonitis, but it is not specific while barium-contrast gastrointestinal series may provide clues to narrow the differential diagnosis. In the present case, plain abdominal X-ray revealed air-fluid levels located on the left side of the abdomen, which remained unaltered in a subsequent image after 2 hours.et al. have recently reported that laparoscopy may be safe and feasible in the diagnosis and treatment of a congenital band.[Concerning the management of a congenital band, surgical treatment is the cornerstone. Traditionally, laparotomy is indicated, whereas with the advent of minimally invasive surgery, laparoscopy has been proposed as an alternative. Wu tal band.In conclusion, the possibility of a congenital band must be included in the differential diagnosis of young patients with symptoms and signs of bowel obstruction and no history of abdominal surgery, trauma or clinical hernia, although this entity is very uncommon. This clinical situation requires early surgical intervention that will be diagnostic and therapeutic.


Benign tumors are a rare cause of gastrointestinal hemorrhage of which angioleiomyomas constitute a very small minority. They have been reported in literature to present with volvulus, bleeding or intussusceptions.An interesting case of a patient presenting with gastrointestinal bleeding from an underlying angioleiomyoma is discussed along with its management options.Angioleiomyoma though rare can be managed successfully by surgical and/or minimally invasive endovascular procedures. Angioleiomyoma of the small bowel is an extremely rare benign tumor. This is the first reported case in English literature, where a patient presenting with gastrointestinal bleeding had an underlying angioleiomyoma.A 58-year-old woman presented with a one-day history of malena and light-headedness. She did not have any known co morbidities. Apart from being anaemic she was haemodynamically stable and her general physical and abdominal examination unremarkable. Hematological examination revealed low hemoglobin levels of 7.3 g/dl with normal clotting profile. After initial resuscitation, an esophagogastroduodenoscopy (OGD) was performed which excluded any cause of upper gastrointestinal bleed. This was followed by colonoscopy, which although did confirm the presence of blood within the gastrointestinal tract but did not localize the site of bleeding. However, the patient's hemoglobin level continued to fall over the next two days, which was treated with a transfusion of six units of packed red cell.Subsequently, she underwent selective superior mesenteric angiography, which demonstrated a large hyper vascular mass with an extensive contrast blush, near the terminal ileum in the right iliac fossa Figure . It was th postoperative day.She thereafter underwent laparotomy through midline incision, which identified vascular solid tumor along the anti-mesenteric border of the ileum about 30 cm from the ileo-caecal junction, measuring 7 × 7 cm Figure . A segmeThe histopathology of the resected tumor showed a submucosal tumor compressing the mucosal lining causing ulceration. Sectional analysis of the tumour showed thick bands of muscle with vascular spongy stroma. Microscopic examination Figure  showed tBenign tumors are a rare cause of gastrointestinal hemorrhage, being responsible for fewer than 1% of all cases  distinguVascular leiomyomas are well-defined benign smooth muscle tumors with prominent abnormal thick walled venous channels . The preFour histological subtypes of angioleiomyomas ,7 have b• Capillary or solid angioleiomyomas having a rich smooth muscle cell stratification surrounding and holding a few thin vascular channels,• Venous angioleiomyomas characterized by more numerous and thicker vascular channels than found in capillary angioleiomyomas,• Cavernous angioleiomyomas having widened vascular channels surrounded by a thin layer of smooth muscle cells,• Combined capillary and venous angioleiomyomasOur patient had a mixed vascular angioleiomyomas with positive immunohistochemical staining with Desmin for smooth muscle cells. However, staining with Desmin is not necessary for such tumors as suggested by Hasegawa , LundgreOne peculiar feature of the tumor found in our patient was its large size measuring 7 × 7 cm. Vascular leiomyomas are usually < 2 cm  althoughThe operative management is usually surgical involving resection of the affected bowel segment with end-to-end anastomosis as was done in our patient. Embolization of the feeding vessel can be performed as suggested by Cho and colleagues  to contrAngioleiomyoma though rare can be managed successfully by surgical and/or minimally invasive endovascular procedures.The author(s) declare that they have no competing interests.US has been involved in the conception and design of the case report ST, JV, AJ, have been involved in the acquisition of data, analysis and interpretation of data, drafting the manuscript, revising it critically for important intellectual content. All authors read and approved final manuscript.


The tendon sheaths constitute an uncommon target of extra-articular tuberculosis.We present a rare case of tuberculous tenosynovitis of the wrist involving the extensor tendon with rupture of the extensor pollicis longus tendon in a 55-year-old Indian man.Prompt surgical debridement and tissue diagnosis are essential for the diagnosis and treatment of this type of infection. With an accurate and timely diagnosis, appropriate surgery and antituberculous treatment may eradicate these infections and prevent complications. Extrapulmonary tuberculous involvement of the musculoskeletal system is uncommon, accounting for only 10% of tuberculosis (TB) cases . The diaA 55-year-old Indian man employed in the armed forces presented to us with a 5-month history of progressive painful swelling of the right wrist and inability to extend his right thumb. The swelling gradually increased in size. The pain, a mild, dull aching, had started insidiously, and gradually worsened with time. There was no associated history of fever, loss of weight or appetite, night sweats, malaise or fatigue. There was no history of trauma, pain in other joints of the body, morning stiffness of the back or hand joints, or continuous use of vibratory tools. The patient had no history of previous illness suggestive of diabetes mellitus, injuries or surgery.Physical examination revealed a well-looking man with a right wrist swelling. The swelling was non-tender, doughy in consistency, approximately 8 × 5 cm, non-compressible, extending proximal and distal to the wrist with positive cross-fluctuation. It was mobile at right angles to the plane of the wrist joint but not longitudinally present on the dorsolateral aspect of the wrist. Movements of the wrist were only limited and slightly painful at the extremes with crepitation felt in the swelling on movement of the joint. Extension of the thumb with palm down on a table was limited and weak at the metacarpophalangeal (MCP) and interphalangeal (IP) joints indicating clinical signs of extensor pollicis longus rupture. There was no clinical evidence of chest infection or other joint involvement.Laboratory findings showed an erythrocyte sedimentation rate of 32 mm/hour and serum C-reactive protein was 12 mg/dl. A diagnosis of ganglion was established on fine needle aspiration cytology. Findings on chest X-rays were normal. Right wrist joint X-ray showed no significant findings. Magnetic resonance imaging (MRI) of the right wrist joint revealed tenosynovitis of the extensor tendons Figure .The patient was operated on under local anesthestic and a curvilinear incision was made over the prominence of the swelling. Extensive drainage, widespread surgical debridement, irrigation, and synovectomy were performed. During the operation, multiple large melon seed bodies along the extensor tendons with adherent synovitis were found. The tendon of the extensor pollicis longus was found to be attached with a streak of fibrous tissue. The margins of the tendon were freshened and debrided and tendon transfer with extensor indices was performed. A below-elbow slab was applied with the wrist, MCP and IP joints of the thumb, in extension for 4 weeks. Intermittent mobilization with a splint during rest was applied for the following 2 weeks. Subsequently, the patient was freed of splintage and unhindered occupational activity was allowed after 3-4 months when the power in his thumb had reached near normal. Resected synovium and tissue were sent for histopathology.Mycobacterium tuberculosis polymerase chain reaction (PCR) was positive.Biopsy showed hyperplasia of the synovial lining epithelium. The underlying tissue exhibited vascular proliferation, acute and chronic inflammatory infiltrates, fibrin deposits and multiple epithelioid cell granulomas with minimal necrosis Figure . Ziehl-NM. tuberculosis was confirmed. The patient was prescribed 300 mg of isoniazid with 10 mg of pyridoxine, 800 mg of ethambutol, 750 mg of pyrazinamide twice daily and 600 mg of rifampicin daily for 2 months. He was evaluated monthly. The lesion was completely stable and no sign of infection was detected. Consequently, treatment with isoniazid and rifampicin was planned for an additional 10 months. Postoperatively, the patient had good extension of his thumb and was able to return to the high occupational demand of the armed forces.Antituberculous treatment was started as soon as the identification of Primary tuberculous tenosynovitis is a rare condition. Tuberculous tenosynovitis selectively targets the wrist and volar aspect of the hand, where it accounts for 5% of cases of osteoarticular TB . InvolveThe precipitating factors include trauma, overuse of the joint, old age, low socioeconomic status, malnutrition, alcoholism, immunosuppression and steroid injections. The right hand and wrist are the most common sites of involvement of tuberculous tenosynovitis and men are more affected than women tuberculous tenosynovitis of the wrist. Patients usually present with an insidious, slow-growing, sausage-like mass along the inflamed tendon with no or little pain. Patients may present with discharging sinus and carpal tunnel syndrome ,9. TendoThere are three histological forms of tuberculous tenosynovitis as a result of the long duration of the disease, the resistance of the individual, and the varying virulence of the microorganism. In the earliest stage, the tendon is replaced by vascular granulation tissue. Later on, the sheath is obliterated by fibrous tissue. Fluid is confined within the sheath and rice bodies may appear due to caseation. In the end, the tendon may consist of only a few strands of tissue and may rupture spontaneously. If healing by fibrous tissue formation fails to curtail the pathologic process, extensive caseation and granulation occur. This may lead to sinus formation and superimposed secondary infection ,10-12. ALaboratory findings are generally negative, except for the erythrocyte sedimentation rate, which is usually increased . In the The currently recommended 6-month course is often inadequate and is commonly extended to 9 or 12 months. Extensive curettage, lavage and synovectomy should be performed. Surgery is essential, but the extent of surgical debridement is still debatable. Some authors advocate surgical debridement with complete excision of the tendon sheath while others advocate decompression of the tendon sheath without excision and debridement of the surrounding tissue ,11,12,16Tuberculosis of the tendon sheaths of the wrist is rare. Delayed diagnosis is common due to slow progression and numerous differential diagnoses, which often leads to complications. Early radical excision of the infected tissues combined with antituberculous multidrug therapy gives good functional results and prevents recurrence.TB: tuberculosis; EPL: extensor pollicis longus; MCP: metacarpophalangeal; IP: interphalangeal; MRI: magnetic resonance imaging; AFB: acid-fast bacilli; PCR: polymerase chain reaction.The authors declare that they have no competing interests.HL, SN, and VKJ wrote the case presentation and performed the literature review. VKJ and RK assisted in reviewing the radiology and provided images. HL and DM assisted in diagnosis. All authors read and approved the final manuscript.Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.


Adrenocortical cancer is a very rare tumor with a poor prognosis. About half of them are hormone-secreting tumors. In most cases, hormonal investigations reveal an excess secretion of steroids, mostly cortisol and androgens. A 54-year-old lady presented with history of pain in left shoulder and leg for 6 months and features of virilization. CT-guided fine-needle aspiration cytology of an abdominal mass revealed the presence of a carcinoma of the left adrenal cortex. A whole-body radionuclide bone scan revealed increased uptake in the left clavicle and left femur. The patient has received palliative radiotherapy for the skeletal lesions and 3 cycles of palliative chemotherapy at present. Carcinoma arising from the adrenal cortex is a very rare malignancy with an extremely poor prognosis. It has aA 52-year-old female patient presented to us with pain in the left thigh and inability to walk for the past 3 months. She had also noticed a painless hard lump over the left clavicular region for the last 6 months. On further inquiry, it was revealed that the lady had gradually developed temporal alopecia, appearance of fine facial hair, as well as hair on the arms and upper back. There was also a deepening and hoarseness of her voice for the last 3 years. She gave no history of any flushing attacks, diarrhea, sweating or palpitation. The patient has 6 children and had attained menopause 6 years back. There was no significant past medical or surgical history or history of intake of any medication.On examination the patient had features of hirsutism, suggested by facial appearance resembling that of a male with facial hair, with hair also present on her upper limbs and back . She als+ (139 mmol/L), K+ (4.1 mmole/L) were not raised either, but serum 17 dehydroepiandrostenedione (17 DHEA) was >10 ng/ mL . Twenty-four-hour urinary cortisol was marginally raised, 227.87 μg per 24 hours .Results of hematological tests were within normal limits. Liver enzymes were not raised . Serum urea and creatinine were within normal limits. Fasting blood sugar was 116 mg%. Serum NaSkiagram of postero-anterior view of the chest revealed a fracture of the left clavicle . ComputeUltrasonography (USG)-guided fine-needle aspiration cytology (FNAC) from the left adrenal swelling showed malignant cells, possibly adrenocortical carcinoma . FNAC fr2 Day 1 and Inj. doxorubicin @ 40 mg/m2 Day 1 every 21 days. A repeat CT scan of the abdomen at the end of 3 cycles of chemotherapy showed no response of the tumor to this regime.The patient was treated by giving external radiation to the areas with skeletal metastases. The left clavicular lesion was treated by a direct AP field to a dose of 2000 cGy in 5 fractions. The left femoral lesion was treated using the same dose. External radiation was given by Co 60 (Theratron 780C). Due to the presence of multiple hepatic metastases involving both lobes of the liver and after consultation with the concerned surgeon, it was decided that operative intervention would not be feasible. After completion of radiotherapy, the patient received 3 cycles of chemotherapy with Inj. cisplatin @ 80 mg/mp53 gene are common and may be used as an optional diagnostic test. About 10% of malignant tumors produce features of masculinization due to testosterone production, whereas the rest secrete cortisol, aldosterone or nothing. Excessive cortisol secretion gives rise to features of Cushing′s syndrome, whereas excess mineralocorticoids lead to mild increase in blood pressure. Co-secretion of cortisol and androgen is the most common presentation and is highly suggestive of malignancy.[Carcinoma of the adrenal cortex is a very rare malignancy and constitutes about 0.05% to 0.2% of all malignancies and has a prevalence of 4-12 per million of adult population. These carcinomas account for 0.2% of all cancer deaths. Adrenal cortex tumors may be functioning or nonfunctioning. About 50% of such tumors are functioning ones, with most presenting with Cushing′s syndrome.[lignancy. In our clignancy.Clinically, virilization suggests malignancy. Patients55Imaging studies such as CT scan/MRI also help to detect the nature of adrenocortical tumors to some extent by not only stating the size but also by the appearance on CT scan. MeasurinACCs are classified by the Macfarlane classification or Sullivan modification of Macfarlane classification and the TMN classification is an adaptation of the aforesaid staging system. Among the various prognostic factors, tumor staging is one of the important clinical parameters which affect prognosis of ACCs. Also theThe mainstay of treatment of not only primary ACCs in stage I to III disease but also in selected cases of locally recurrent, residual disease and in limited metastatic lesions is surgical resection. R0 resecThere is evidence that radiotherapy may be used as palliative treatment in bone, brain metastases, as well as in postoperative recurrences. RadiotheWe had advised our patient to take mitotane along with palliative chemotherapy. As she was unable to afford mitotane, she received 3 cycles of chemotherapy with doxorubicin and cisplatin, but to no effect. Though mitotane (a synthetic derivative of the insecticide DDT) has been used in trials as an adrenolytic agent, its toxicities have precluded its usage in clinical practice. Mitotane has been recommended in patients that have undergone complete surgical resection, as the rate of recurrence is quite high in ACCs (50%-60%) and use 2Single-agent chemotherapeutic drugs used in adrenocortical carcinomas give a low response rate for a limited duration. Certain combinations4 like ciPrognosis in advanced ACCs is very poor, with a 5-year survival of 10% to 25% and a mean survival of 5 to 18 months. At the end of 6 months from diagnosis, our patient is still alive with subjective improvement of her symptoms.Present treatment modalities are often disappointing in the management of advanced cases of adrenocortical carcinomas, and it is hoped that further molecular studies of this rare tumor may offer new treatment options.


Capillary hemangiomas are the most common periocular and orbital tumors of childhood that typically arise in infancy. Though the diagnosis is frequently made on clinical examination, various diagnostic modalities may be helpful in initial evaluation and follow-up. Tests may be necessary in diagnosing suspect cases or aid in the differentiation of potential malignant tumors. In the vast majority of cases these tumors undergo spontaneous involution without sequelae. However, some periocular and orbital capillary hemangiomas require intervention to prevent serious complications. Other tumors require treatment to lessen the surgical burden for cosmetic repair. When treatment is necessary, there are a number of therapeutic options available. As there is no standard, potential risks and benefits must be discussed with the family and treatment should be specific in each case. A complete understanding of the natural history of the tumor, indications for treatment, and response to different therapies is imperative in managing this common lesion. Capillary hemangiomas are the most common eyelid and orbital tumors of childhood. This tumor has been referred by many names , but is most commonly called ‘capillary hemangioma.’ This term is most common as it accurately describes the capillary unit structure of endothelial cells surrounded by pericytes.12These tumors can present as small isolated lesions, or large masses that can cause visual impairment, systemic effects, or exist as part of a syndrome. Many capillary hemangiomas can be diagnosed on examination, but occasionally identification may require the use of ultrasonography, computed tomography or magnetic resonance imaging for accurate diagnosis. Understanding of this tumor, its natural history, and the indications for treatment are necessary for management of the tumor and to help the patient's family understand the disease process.Capillary hemangiomas are among the most common tumors in infants, and the most common eyelid and orbital tumors of childhood.11et al classified hemangiomas into three categories based on the color of the skin associated with the hemangioma: classic superficial strawberry nevus; subcutaneous hemangiomas that appear bluish or purple through the overlying, unaffected skin; and deep orbital tumors that present with proptosis without observable skin discoloration.1The exact etiology of capillary hemangiomas remains unclear, but likely involves a combination of angiogenesis and vasculogenesis. The natural history of capillary hemangiomas has been well documented over the past century. The tendency for these tumors to spontaneously involute is well accepted. Capillary hemangiomas typically present during the first few months of life, with 33% to 55% present at birth.et al described a case series in which 25% had the classic strawberry nevus and a subcutaneous mass, 68% had the bluish-purple, ‘spongy’ subcutaneous mass, and 7% had no skin discoloration or visibly notable mass, but had deep orbital involvement.Haik Enlargement of the capillary hemangiomas can often be observed when the patients cry. Crying simulates the valsalva maneuver, which raises intrathoracic pressure, and theoretically forces deoxygenated blood into the capillary spaces of the hemangioma causing enlargement and deepening of the color. Haik's series noted this change in 46 of the 101 patients, so although this sign can be observed, it is not pathognomonic for capillary hemangiomas.6The most common ocular complication of periocular capillary hemangiomas in infants is visual loss secondary to amblyopia. Case reviews have reported between 40 to 60 percent incidence of amblyopia with periocular capillary hemangiomas.688The strikingly high incidence of amblyopia in some periocular infantile hemangiomas may be related to the onset of the tumor within the first few months of life. In numerous studies, it has been shown that young children may be susceptible to visual deprivation amblyopia in the first 7 years of life, though the sensitivity is likely greatest in the early months of life. Animal research models demonstrate severe amblyopia after only 1-4 weeks of visual axis occlusion.1et al demonstrated there was a decrease in the amount of anisometropic astigmatism with reduction in the size of the capillary hemangioma.et al, performed the largest review of periorbital capillary hemangiomas and studied the risk factors for amblyopia.14Previous studies have debated whether there is proven change in refractive error with treatment of the hemangioma. Robb suggested that there was no influence on refractive error by treating hemangiomas.Capillary hemangiomas have been associated with hematologic, cardiac, respiratory and infectious disorders. Large hemangiomas are subject to superficial ulceration, and may rarely cause significant hemorrhage or become secondarily infected with resultant sepsis.1Kasabach-Merrit syndrome is a rare, but feared hematologic abnormality associated with extensive capillary hemangiomas. This disorder was first described in 1940, and is characterized by platelet and/or fibrinogen entrapment and consumption coagulopathy. Numerous cases have been reported, however it is rarely associated with isolated facial hemangiomas. In Kasabach-Merrit syndrome, a critical thrombocytopenia occurs from platelet sequestration within the tumor's vascular channels. Clinically, thrombocytopenic purpura is visible, along with large ecchymoses around the hemangioma or elsewhere on the body. Kasabach-Merrit syndrome can lead to sudden, massive hemorrhage with resultant cardiovascular collapse. Treatment of this syndrome consists of systemic steroids and thrombocyte-replacement therapy, with a 50% response rate. Recalcitrant cases can be further treated with radiotherapy, surgical removal or embolization, however this syndrome has a high mortality rate.1Microangiopathic hemolytic anemia (MAHA), has also been described with large capillary hemangiomas of the periocular region, digestive tract, and the liver. In MAHA, the erythrocytes are destroyed from coagulation, or are sheared or fragmented by high pressure forcing them through the abnormally small vessels of the hemangioma.3Periocular capillary hemangiomas may occur simultaneously with oral, nasal, subglottic, and paratracheal hemangiomas. These hemangiomas can pose a threat by compressing the adjacent trachea or larynx, thus causing respiratory distress and asphyxiation, or can compress the esophagus, leading to secondary aspiration. There may be a risk from uncontrolled bleeding from these hemangiomas, leading to potential aspiration or massive hemorrhage.15While capillary hemangiomas can largely be diagnosed through clinical examination, some tumors are not easily distinguished. The differential diagnosis includes rhabdomyosarcoma, lymphangioma, chloroma, neuroblastoma, orbital cysts, and cellulitis, hence additional diagnostic testing may be necessary.Plain radiography is not particularly helpful in the diagnosis of capillary hemangiomas. These tumors are not easily seen or clearly identified on plain film. Soft tissue enlargement can be seen, but bony structures will not show evidence of bony erosion. If there is rapid growth of the orbital tumor during development, orbital enlargement can be seen. However, none of these findings are diagnostic for capillary hemangioma.Ultrasonography scan may show an irregular mass with variable reflectivity which blends into surrounding orbital structures. With the use of A-scan ultrasonography, areas of low internal reflectivity, moderate reflectivity, and high reflectivity may be identified. The low areas represent the areas of solid, hypercellular endothelial proliferation, whereas the moderate echoes represent ecstatic vascular channels, and the high reflectivity corresponds to fibrous septae separating the tumor lobules. Orbital involvement and monitoring of tumor size can be accomplished with the use of ultrasonography.12Computed tomography (CT) is another modality for monitoring tumor growth. Capillary hemangiomas often appear as homogeneous soft tissue masses of the orbit, without destruction of orbital bone. The tissue mass occurs in the anterior orbit, or in the extraconal space with ‘finger-like’ posterior extension. This mass can be well defined or irregular and infiltrating the surrounding tissue.On Magnetic Resonance Imaging (MRI) studies, capillary hemangiomas may appear as well-circumscribed densely lobulated tumors. On T1-weighted MRI, the capillary hemangioma is of intermediate signal intensity, but it is moderately hyperintense on T2-weighted imaging due to the slow blood flow through the vascular channels. The tumor mass may enhance following gadolinium contrast injection, so images should have fat suppression on T1 weighted images to better visualize the tumor.13Angiography is rarely used as a diagnostic tool for capillary hemangiomas, as both venography and arteriography are technically difficult, and not without complications. Arteriography is rarely used diagnostically, but may be useful in rare situations to identify feeder vessels for possible ligation or embolization in life-threatening hemangiomas that are non-responsive to less invasive therapies.Although capillary hemangiomas can often be diagnosed with less invasive techniques, tissue biopsy may be occasionally required, especially in the case of proptosis without superficial involvement. In these cases, the differential diagnosis may include other tumors in this age group, such as lymphangioma, metastatic neuroblastoma, or rhabdomyosarcoma. As all surgeries carry inherent risks of infection, hemorrhage, diplopia, or visual loss, biopsy diagnosis is reserved for cases that cannot be diagnosed with less invasive techniques.Indications for therapy of capillary hemangiomas are important to note, as the natural course for this tumor is spontaneous involution. Visual indications include occlusion of the visual axis, amblyogenic anisometropia, optic nerve compression, or significant proptosis causing exposure keratopathy. Dermatologic indications for treatment are maceration and erosion of the epidermis caused by severe hypertrophy of the epidermis and subcutaneous tissues. Systemic indications for treatment include: obstructive , hematologic (thrombocytopenia or hemolytic anemia), and cardiovascular (high-output congestive heart failure).1Careful discussion with the family is imperative when contemplating any treatment. It is important to carefully describe the risks and benefits of each therapy, including vigilant monitoring, vascular occlusion, excision, radiation, intralesional or systemic corticosteroid, interferon, laser, or the newly introduced propranolol therapy.It has been well documented by several published reports that adenexal capillary hemangiomas can result in severe and permanent visual loss.11Strabismus surgery is usually delayed until amblyopia therapy is completed, and the hemangioma shows signs of regression. In doing so, there is less potential for hemorrhagic complications, and improved predictability of surgical outcomes. Frequently, surgery is not necessary, as the strabismus often resolves as the tumor regresses.1Surgical removal is usually reserved for cases where more conservative therapy has failed, and tumor removal is necessary. The highly vascular tumor may create a significant higher risk of intraoperative bleeding. Surgery can reduce tumor size by compromising the tumor feeder vessels, constriction and sclerosing of the small vascular channels, or primary excision of the entire mass can be performed. Historically there have been attempts to stimulate the natural involution of capillary hemangiomas by destroying the small vascular channels and stimulating thrombosis through the use of compression, cryotherapy, diathermy, or injection of sclerosing agents or even boiling water. These techniques may accelerate the involutional process, but may also end in more pronounced cosmetic and functional deformities.16Actual excision of the tumor is sometimes effective for tumors that are small and non-responsive to pharmacologic therapy, or remain after the natural involutional. Primary surgical excision during the proliferative phase, or for large tumors is not ideal, as these tumors tend to have greater infiltration, are less encapsulated, and carry higher risk of bleeding associated complications. If surgical excision is absolutely necessary, it is important to obtain preoperative imaging, including MRI, MRA, and possibly an angiogram in order to identify the vascular channels of the tumor. As these large hemangiomas can infiltrate surrounding structures, there is the complication of unintended excision of surrounding orbital structures. The surgeon must also be prepared for the possibility of considerable blood loss especially in pediatric cases with limited blood supply, hence blood-typing is necessary. Due to the potential for such severe complications, surgical excision is best utilized very early, with small lesions, or to repair remaining deformity after involution of the tumor.Radiation therapy can be effective in the management of certain capillary hemangiomas. Microembolisms can be created in the tumors by low-level radiation, which can speed the regression of the mass. Involutional response usually occurs in 1-2 weeks, and treatment may be repeated for further resolution. Cumulative doses must be monitored as historically, large doses of radiation with poor delivery or shielding could cause radiation-induced complications of the skin, eyelids, eye and orbit. Current radiologic technology makes direct radiologic damage rare, however radiation induced cataract can still occur with 200-600 rads if the anterior segment is not adequately shielded.1Carbon dioxide, argon, neodymium-YAG, and flash-lamp pumped-dye laser have all been utilized in the treatment of capillary hemangiomas.113Zarem and Edgerton first reported the use of systemic corticosteroids in the successful treatment of large capillary hemangiomas of seven infants in 1967.118The exact mechanism of action of systemic corticosteroids is speculative. It has been proposed that steroids have an anti-anabolic effect on the immature vascular tissue of capillary hemangiomas.1Due to the systemic risks of long term oral steroid use, intralesional corticosteroid injection is more commonly used. A recent study of American Association for Pediatric Ophthalmology and Strabismus members, reported that intralesional steroid injection is the most common treatment and also the most frequent initial therapy.Complications from local corticosteroid therapy are reportedly low and apparently vary with frequency, quantity, and method of injection. The most serious and feared complication is central retinal artery occlusion (CRAO).Other complications following intralesional corticosteroid injection include eyelid necrosis, subcutaneous deposits, focal hypopigmentation and fat atrophy. Transient adrenal suppression and cushingoid symptoms have been reported.24Recombinant interferon alfa, and angiogenesis inhibiting agent, has been used successfully as a secondary agent for life-threatening hemangiomas that have failed corticosteroid therapy.1Due to the serious side effect of spastic diplegia, vincristine replaced Interferon α as the second-line treatment for large capillary hemangiomas resistant to corticosteroids. Vincristine is a microtubule inhibitor, and is also an immunosuppressant used in treating thrombotic thrombocytopenic purpura or chronic idiopathic thrombocytopenic purpura. It has be shown to be efficacious in the treatment of Kasabach-Merritt syndrome with non-orbital capillary hemangiomas.The use of an alkylating agent for the treatment of capillary hemangioma was first reported by Rush in 1966, with subsequent case reports verifying the efficacy of cyclophosphamide for life-threatening and corticosteroid resistant hepatic hemangiomas.18in situ, and actinic keratosis. Imiquimod has been found successful in treating superficial capillary hemangiomas of the scalp, eyebrow and forehead. This is a new agent with future potential however, safety or efficacicy for ophthalmic use is still under investigation.10Imiquimod is an immune-response modifier that induces production of cytokines from keratinocytes and monocytes to cause regression of numerous dermatologic conditions, including warts, superficial basal cell carcinoma, squamous cell carcinoma et al serendipitously observed a dramatic decrease in the size of cutaneous capillary hemangiomas in an infant being treated with propranolol for a cardiac indication. In follow up report, they observed improvement or regression of the hemangiomas in 31 of 32 patients. One patient discontinued treatment because of wheezing.42812315Systemic propranolol is a new treatment modality introduced by Leaute-Labreze in 2008.Capillary hemangiomas are the most common tumors in infants, and the most common eyelid and orbital tumors of childhood. The diagnosis is usually made clinically, although other diagnostic modalities are occasionally employed. Most tumors undergo spontaneous involution, however, some tumors require intervention to prevent serious complications. Other tumors require treatment to decrease the surgical burden for cosmetic concerns. There are many treatment options available for the practicing clinicians, and there is no exact standard for these treatment modalities. More recently, systemic propranolol has been found to be safe and effective in the treatment of eyelid and orbital hemangiomas. A complete understanding of the natural history of the tumor, indications for treatment and response to different therapies is vital to properly treat these common lesions.


We report a case of malignant nodular hidradenoma in a middle-aged man, who presented with a nodular swelling in the eyelid. The tumor was similar to its benign counterpart but had additional features such as surface ulceration, numerous mitiotic figures, and an infiltrative growth pattern. Malignant forms of hidradenomas are unusual and the possibility this variant should be considered in the differential diagnosis of eyelid tumors. Nodular hidradenoma is a cutaneous neoplasm that can occur at various sites. It has been referred to as clear cell hidradenoma or eccrine sweat gland adenoma.A 51-year-old man presented to our institute with an enlarging, painless, nodular mass in the right upper lid that began 4 months prior with rapid increase in size over the last 3 months. The mass became significantly prominent over the last month prior to presentation resulting in discomfort, ulceration, and bleeding on manipulation. Physical examination revealed a solitary mass in the middle third of the right upper eyelid, overhanging the lid margin that was 9 mm × 12 mm in size, pinkish, firm, fleshy with small-dilated blood vessels on the surface with central ulceration and crusting . The masA complete, wide excision of the nodular mass with a 3 mm clear margin of healthy surrounding tissue was performed along with primary closure . The patThe specimen submitted to the pathology department comprised of a nodular mass 1.5 cm in diameter. The cut surface was yellowish with few cavities.Histopathologic examination revealed a tumor in the dermis with surrounding fibrous capsule. Two types of tumor cells were noted with predominance of one cell-type which was polyhedral to round in configuration with round to oval nuclei and prominent nucleoli surrounded by faintly basophilic cytoplasm. The second cell-type was clear, round to oval with eccentric nucleus. The tumor cells were arranged in lobules and separated by delicate fibrous collagenous tissue . The lobTumors arising from the sweat glands of the eyelids are uncommon, and the differences in their structure have given rise to much confusion. A wide variety of descriptive terms have been applied to sweat gland tumors.6Histologically, sweat glands may be either eccrine or apocrine in nature. Eccrine glands are present throughout the skin but are most abundant in the palms, soles, and axillae. In the eyelid, eccrine glands are present at the lid margin and in the surface dermis. Apocrine glands are found in relatively fewer regions of the body, mainly the axillae, around the nipples, the anogenital region, and occasionally a small number on the abdomen and chest.7Eccrine hidradenomas are also referred as nodular hidradenoma or clear cell hidradenomas arise from the eccrine sweat glands. This nomenclature is based on electron microscopy and histochemical studies.9The differential diagnosis includes primary skin tumors with follicular, sebaceous, or sweat gland differentiation. Hidradenomas can mimic cutaneous metastatic disease from clear cell tumors such as renal cell carcinoma.11The hidradenoma in our case had similarities to its benign counterpart but exhibited ulceration, brisk mitotic figures, and an infiltrative growth pattern suggestive of a malignant lesion. Malignant hidradenomas are most often malignant from inception as seen in our case. Some malignant hidradenomas develop from their benign counterparts. These tumors in contrast to the benign forms, tend to invade the surrounding tissue and have a high incidence of recurrence and distant metastasis.9Malignant hidradenoma are distinctly unusual and are extremely rare in the eyelids. The possibility of sweat gland tumors should be considered in the differential diagnosis of eyelid tumors to ensure early detection. Management involves complete excision of the lesion.


Castleman disease (CD) is a rare benign disorder characterised by hyperplasia of lymphoid tissue that may develop at a single site or throughout the body. The etiology of this disorder is unclear, although the histopathological presentation can be differentiated into a hyaline vascular variant, a plasma cell variant and a mixed variant. Clinically, it has been recorded that 3 manifestations of CD are characterized: a localized unicentric type, a generalized multicentric type and a mixed form. Surgery remains the main treatment for resectable unicentric CD, since removal of the large node is possible without further complications. No consensus has been reached concerning the most adequate treatment for irresectable unicentric CD.Case report of a 67 year old woman.This report, describes the case of a 67-year-old woman with unicentric Castleman disease located in the right lower abdomen. The patient had symptoms of fatigue, dyspnoea and pain in the right lower abdomen. Computed tomography (CT)- examination revealed a tumour, which had grown to form a close relationship with the common iliac vessels and the sacral bone. A Laparotomy procedure revealed that the tumour was an irresectable mass. Neo-adjuvant radiotherapy (40 Gy) was administered in order to downsize the tumour. Six weeks later a new CT-scan revealed a major reduction of the tumour, which enabled a successful radical resection of the tumour to be performed. Histopathological analysis of the tumour showed the hyaline vascular type of CD.Neo-adjuvant radiotherapy should be considered in case of an irresectable unicentric CD. In 1954, Benjamin Castleman described an unusual benign disorder that was characterized by hyperplasia of lymphoid tissue  publisheThe first being a hyaline vascular type (HV), characterized by lymphoid follicular hyperplasia with involuted germinal centres, which are partly or totally replaced by deposit of hyaline material and transfixed by a radially penetrating vessel, characterized as a 'lolly pop' structure. The second variant was defined as the plasma cell type (PC) with a follicular hyperplasia of hyperplastic, large germinal centres in which the interfollicular areas were occupied by large sheets of plasma cells.Clinically a broad spectrum of manifestations of CD are described, ranging from an asymptomatic localized lymphadenopathy to a severe symptomatic multifocal or generalized lymphadenopathy . A commoCD has to be treated because of its progressive course associated with local involvement of surrounding structures or because of the systemic effects associated with the multicentric form such as fever, weight loss, excessive sweating, hemolytic anaemia, splenomegaly, oedema and neuropathy.CD can also be associated with HIV infection, POEMS syndrome, amyloidosis, renal insufficiency and increased risk of lymphoma ,9-11, thSurgery is considered to be the most adequate therapy for unicentric resectable cases of CD, as it seems to be curative in almost all of the cases ,6,12-20.No consensus has been reached concerning the most adequate treatment for irresectable CD. To our knowledge, neo-adjuvant radiotherapy to downsize primary irresectable CD in order to achieve a radical surgical resection has not been described yet. This paper reviews the associated literature concerning the treatment of CD and describes a case history of an irresectable unicentric localisation of CD which was treated with neo-adjuvant radiotherapy and subsequent radical resection.A 67-year-old woman with a history of hyperthyroidism, proptosis and anaemia was referred to the internal medicine department with complaints of weight loss, fatigue, dyspnea and pain in the right lower abdomen. Physical examination revealed a painful palpable mass in the right lower quadrant of the abdomen.Ultrasonography of the abdomen showed a multicystic, solid structure in the right lower abdomen. Subsequent Computed Tomography (CT) imaging of the abdomen showed right ventrally of the body of the first sacral vertebrum a partly solid, partly cystic tumour of 71 × 51 mm with oedematous infiltration of the surrounding adipose tissue and a close relationship with the common iliac artery and vein figure . Since tThis report documents a unique case of a primary irresectable case of the HV unicentric type of CD which was treated with neoadjuvant radiotherapy and subsequent successful radical resection. To our knowledge, there is no report in the literature describing neoadjuvant radiotherapy as part of the treatment of unicentric CD.The etiology of Castleman's Disease is unknown. However, CD is associated with other disorders such as HIV infection, POEMS syndrome, amyloidosis, renal insufficiency and increased risk of lymphoma ,9-11. ThPrimary radiotherapy has been described in numerous case reports and small case series as one of the strategies for the treatment of both unicentric and multicentric forms of CD. Keller et al  however With respect to the dose of radiotherapy, no correlation can be observed between dose and response. Most patients were treated with a dose between 40 and 50 Gy, however patients with a complete response received a dose between 12 Gy and 50 Gy.The evidence in the current literature reveals that radical surgery results in excellent rates of cure ,6,12-19.Surgery was performed six weeks after the last radiotherapy dose. The rationale for this interval is based on the experience gained from treatments with other neoadjuvant strategies in solid tumours .Different types of therapy have been described for the treatment of patients with multicentric CD. However the treatment strategy for this variant of CD is not within the scope of this article. Nevertheless, the literature reveals that primary radiotherapy can also achieve a remission of symptoms ,28,30,32The rationale of our additional intra-operative radiotherapy (IORT) boost was based on the close relationship with the sacral bone and iliac vessels and was analogous to our management of other locally advanced solid tumours like rectal cancer and soft tissue sarcomas -40.In this case report it is demonstrated that neo-adjuvant radiotherapy in case of locally advanced irresectable unicentric CD facilitates a radical resection. Therefore, analogous with the treatment of other locally advanced solid tumours, neo-adjuvant radiotherapy with a dose of 40-50 Gy and a subsequent resection 6 weeks later should be considered if an irresectable unicentric variant of CD is encountered.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.The authors declare that they have no competing interests.IAC, MMS, GAP have made substantial contributions to conception and design, and acquisition of data and analysis and interpretation of data. IAC, MMS, T, MLM, JP, GAP have been involved in drafting the manuscript or revising it critically for important intellectual content. All authors read and approved the final manuscript.


An 18-year-old Turkish woman was referred with a 6-week history of rapidly enlarging cervical mass at the left side.She was diagnosed of ampullary carcinoma for which pancreatoduodenectomy was performed 14 months ago. In our patient with a history of malignancy, a rapidly enlarging neck mass was considered a metastasis to the neck. Tumor resection was performed. Histopathological examination revealed the metastasis of the precedent ampullary adenocarcinoma.Surgery does not improve survival for advanced metastatic ampullary cancer however, it can be mandatory in specific conditions as our patient. Periampullary neoplasms include carcinomas of the duodenum, ampulla of Vater, distal common bile duct and pancreas. They are considered collectively because of their similar clinical presentation and the difficulty in distinguishing them without examination of a resected specimen ,2. More An 18-year-old Turkish woman was referred from Oncology Department with a 6-week history of rapidly enlarging cervical mass at the left side of her neck which had been detected by the patient herself. She was diagnosed of ampullary carcinoma for which pancreatoduodenectomy was performed 14 months ago. Histopathological examination revealed well-differentiated adenocarcinoma in the ampullary region. Only one lymph node metastasis was present among 35 dissected lymph nodes. Additionaly, peripancreatic fat tissue invasion was also remarked. She has not a family history of pancreas cancer. Gemcitabine was started 1000 mg/m2 Day 1,8,15/every 28 days for ampullary cancer. After chemotherapy performed, she lost follow-up.Six months after the chemotherapy, she noticed a rapidly enlarging right neck mass. A 15 cm soft and painless mass involving the left antero-lateral aspect of the neck was palpable. The mass was apparent by inspection Figure . The masUltrasonography revealed a mixed solid- cystic mass, measuring 13 × 11 cm, with a hyperechogenic capsule. The lesion had a close relationship with the thyroid gland and its borders could not be easily defined. On T2 weighted axial MR images, a hyperintense, dominantly cystic mass with thin borders and papillary vegetations at the posterior wall was occupying the left cervical region. The lesion had displaced the trachea, the thyroid gland and also compressed the left common carotid artery and internal jugular vein. Smaller lesions with similar internal structure could also be detected at the posterosuperior and left posterolateral sides of the primary lesion Figure . On T1 wIn our patient with a history of malignancy, a rapidly enlarging neck mass was considered a metastasis to the neck. Since imaging studies suggested malignant tumor, a Tru-cut biopsy was performed. Cytologic findings did not provide any further information. After Tru-cut biopsy, severe compressive symptoms and stridor were occured. Those symptoms, accompanied by the enlargement of the mass, were suggestive of intralesional bleeding following biopsy Figure .The patient underwent sternotomy and the left brachiocephalic artery and vein, and left common carotid artery, and left subclavian artery were isolated for vascular control. At the neck exploration, the left cervical mass was voluminous, and adherent to surrounding structures including the carotid artery, internal jugular vein, and left vagal nerve. Left thyroid lobe was found normal in appearance. Left common carotid artery and vagal nerve were liberated from the tumor. Because the tumor invaded left internal jugular vein, it was ligated and divided. After mobilization of the tumor from the adjacent structures, tumor resection was completed Figure . She hadOverall, periampullary cancers account for 5% of all gastrointestinal tract malignancies, which can be divided into four groups of tumor entities: periampullary adenocarcinoma originates from the pancreatic duct (pancreatic carcinoma), the mucosa of the ampulla of Vater (ampullary carcinoma), the common distal bile duct segment  or the duodenum  ,2.Panreatic cancer occurs most frequently among the periampullary cancers, accounting for 3% of all gastrointestinal cancers. Carcinoma of the ampulla of Vateri is the second most common periampullary malignancy. Less frequent are distal cholangiocellular carcinoma and duodenal adenocarcinoma of the periampullary region (1). The prognosis of a 5-year survival rate between 1% and 10% for all periampullary cancers still remains a frustrating challenge. However, it has been shown that periampullary tumors, not arising from the pancreatic duct, have a much beter outcome [Morbidity and mortality from pancreatic cancer is conspicuously associated with metastasis; the most frequent sites of metastasis are local lymph nodes, lung, liver, adrenal glands, kidney and bones. Patients with pancreatic cancer have only very rarely been reported to develop metastatic lesions to the brain, skin, and larynx (3-7). Cervical lymph node and brain metastases from pancreatic cancer have rarely been seen and most of them were identified in advanced stage ,8. In ouWhile surgery does not improve survival for advanced metastatic ampullary cancer, it can be mandatory in specific conditions as our patient.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.The authors declare that they have no competing interests.MA and YE carried out the operation, AS and YE were contributor in writing the manuscript. SS participated in discussing of the manuscript. OM performed the histological examination of the mass. ARS performed the MRI image examination of the mass. All authors read and approved the final manuscript.


Solitary endobronchial papillomas (SEP) are rare tumors and most of them are described by case report. A misdiagnosis is common with viral related papillomas. A histopathological classification has recently permitted a major advancement in the understanding of the disease.We report a case of a mixed bronchial papilloma with an unusual endoscopic presentation. The literature was extensively reviewed to ascertain the unusual characteristics of the current case. A 39-year of age male was referred to our institution for the investigation of a slight hemoptysis. Routine examination was normal. A fibroscopy revealed an unusual feature of the right main bronchus. The lesion was a plane, non-bleeding, non-glistering sub-mucosal proliferation. No enhanced coloration was noticed. Biopsies revealed a mixed solitary bronchial papilloma. In situ HPV hybridization was negative. Endoscopic treatment (electrocautery) was effective with no relapse.This lesion contrasts with the data of the literature where papilloma were described as wart-like lesions or cauliflower tumors, with symptoms generally related to bronchial obstruction. We advise chest physicians to be cautious with unusually small swollen lesions of the bronchi that may reveal a solitary bronchial papilloma. Endoscopic imaging can significantly contribute to the difficult diagnosis of SEP by pulmonary physicians and endoscopists. Solitary endobronchial papillomas (SEP) are rare tumors. To date, less than 50 cases were described. Due to low frequency, the majority of data available in the literature are case reports. A major advancement was achieved in 1999 with a large histopathological review that led to a classification of the different sub-types related to solitary papilloma [Due to the rarity of SEP, we are reporting here a single case. The history of the patient was carefully analyzed. Extensive review of the published literature was made to compare this uncommon clinical and endoscopic presentation to generally described features.2. It was a plane, non-bleeding, non-glistering sub-mucosal lesion. No enhanced coloration appeared. The tumor was soft and biopsies were easy despite the tangential localization in the bronchus. Histopathological examination revealed a mixed papilloma. It was an endobronchial polypoid lesion, 2.5 cm long and 0.2 cm wide, made of thin fibrovascular cores with moderate lymphoplasmocytic infiltrates, and lined by squamous and glandular epithelium. Pseudo-stratified ciliated and non ciliated, cuboidal to columnar cells, with rare mucinfilled cells were mixed with acanthotic and focally keratinizing squamous epithelium. Squamous atypia, ranging from mild to severe dysplasia could be seen, but viral cytopathic changes were not reported. No glandular atypia nor necrosis were seen (figure A 39-year old man was referred to our institution for investigation of recent cough. He experienced a unique slightly bloody sputum. He was a mild current smoker (less than 15 packs/year). There were no baseline abnormal conditions, and physical examination was unremarkable. The chest-X-Ray was normal. The bronchoscopy revealed a swollen lesion at the internal proximal part of the main right bronchus  were also normal.Endoscopic treatment was proposed with a rigid bronchoscope  under general anesthesia. We used an electrocautery knife . The procedure was rapid and easy. A fiberoptic bronchoscopy was performed 7 days after the procedure showing a total destruction of the tumor with a small scar. Further endoscopic controls over 12 and 24 months did not reveal any relapse and the patient was considered to be cured.This case produced two interesting findings. First, solitary bronchial papillomas are rare tumors and the description of a new case enhances the understanding of this disease. Second, the endoscopic presentation of this mixed solitary papilloma is uncommon and contrasts with previous descriptions in the literature.Papillomas were recently classified and subdivided into 3 categories: 1 – squamous cell, 2 – glandular and 3 – mixed  but the P, as it includes risk factors related to viral infection [It is important to differentiate closed related entities. Condylomatous papillomas are related to viral infection and, may occasionally be found in larynx or airways . Howevernfection ,12 For anfection . Barzo eA major issue in SEP is the clinical presentation. The analysis of the literature shows protean symptoms and chest-X-Ray presentation. The 8 Japanese cases found 5/8 63%) patients with cough and wheezing % patient. Our patWe have little information on the endoscopic features for SEP. As the majority of SEP presented with radiographic abnormalities highly suggestive of a tumoral disease, bronchoscopy was the first diagnostic tool used. In 1965, Drennan and Douglas described a solitary papilloma of the bronchus (2 × 1.5 cm), but did not find any bronchoscopic abnormalities . This waAll the lesions described by Fliedler et al. were similar: polypoid, friable tan to red, glistering and, ranged in size from 0.2 to 2.5 cm. No significant differences were seen between histologic sub-types . An exteIt is difficult to compare our findings to the literature, as our findings seem to be unique. Our patient did not present with a common tumor. It was not a peduncule-like tumor but an infiltrative mode. The lesions arose the bronchus wall but cannot be considered as wart-like with normal bronchus mucosa coloration. Obviously, as shown on figure Endoscopic treatment was proposed here. Considering the absence of abnormalities on CT-scan examination and the small size of the tumor, this decision was acceptable. YAG-Laser therapy is commonly used . We prefIn conclusion SEP are rare tumors. It is currently important to discriminate real solitary pulmonary papillomas from others related papillomas and achieve to obtain a definite histological sub-type. Clinical, radiographic and endoscopic presentations are generally more related with usual tumor management. We advise chest physicians to be cautious with unusually small swollen lesions as described here, as they could relate to SEP. Biopsies are easy, and contrast with the difficulty of the pathologic examination. In case of a limited lesion, endoscopic therapy is mandatory.SEP: Solitary endobronchial papillomas; CEA: carcino embryonary antigen; SCC: squamous cell carcinoma; HPV: human papilloma virus; COPD: chronic obstructive pulmonary disease; CT-scan: computed tomography scanner.The authors declare that they have no competing interests.FP, CAB and AB designed and wrote the manuscript. JBN analyzed the radiologic data and reviewed the manuscript. MP and MF wrote the pathologic section and reviewed the manuscript.Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the editor-in-chief of this journalThe pre-publication history for this paper can be accessed here:


Polymorphous low-grade adenocarcinoma is a distinct neoplasm of the salivary gland composed of luminal and non-luminal tumor cells admixed in varying proportions. Its resemblance to lobular carcinoma of the breast had led to its earlier nomenclature of 'terminal duct carcinoma'. Most patients present with an asymptomatic mass in the hard palate. In rare cases, the mass can also occur in the tongue. We report an unusual case of polymorphous low-grade adenocarcinoma at the base of tongue.A 47-year-old Asian Caucasian woman presented with a painless swelling at the right lateral border of the tongue with an intact overlying mucosa. There were no other associated complaints. The lesion was excised and subjected to histopathological examination that revealed an interesting and unusual morphology of polymorphous low-grade adenocarcinoma.Polymorphous low-grade adenocarcinoma is a well-defined entity in the minor salivary glands. Its occurrence in the tongue is rare with very few cases reported in the literature. It is a malignant neoplasm with low aggressiveness and it is thus important to identify and treat it accordingly. Polymorphous low-grade adenocarcinoma (PLGA) is a malignant neoplasm with a low level of aggressiveness that occurs almost exclusively in the minor salivary glands, primarily those in the palate. We report a case of PLGA that arose at the base of the tongue in a 47-year-old woman. The tumor was resected through the oral cavity with wide margins. The patient recovered and remained disease-free at follow-up. This case shows that PLGA, which has a variable morphologic appearance, can occur at sites other than the salivary glands.A 47-year-old Asian Caucasian woman presented with a painless swelling over the right lateral border of her tongue that had gradually increased over the four months prior to presentation. It had an insidious onset and progressively increased in size. The patient had no history of discharge, bleeding or ulceration over the swelling. On examination, the swelling was 3 × 2 cm in size and was located along the lateral border at the junction of the anterior 1/3rd and posterior 2/3rd. It was firm in consistency and well circumscribed with all the margins felt clearly. The patient had no restriction in the movement of her tongue. There was no significant peripheral lymphadenopathy. The lesion was excised and sent for histopathological examination.On gross examination, it was discovered to be well-circumscribed lesion, 2 × 3 cm in size, firm in consistency and with a gray-white cut surface. Histological examination showed a relatively well-circumscribed tumor with focally infiltrative margins. The tumor cells were arranged in varied patterns: tubular, papillary, cords, and also in sheets Figure . The tumThe term polymorphous low-grade adenocarcinoma was first used in 1984 by Evans and Batsakis to describe a tumor of the salivary glands that had a variety of architectural patterns associated with cytologic uniformity as its primary histologic characteristic . In veryPLGA had been previously referred to as terminal duct carcinoma in view of its probable origin in the ductal system of the salivary glands . SimilarBecause of its morphologic pleomorphism, PLGA has often been misdiagnosed as pleomorphic adenoma or adenoid cystic carcinoma (ACC) . HoweverPLGA is an unusual tumor to occur at the base of the tongue. It is a low-grade aggressive neoplasm and it is important to recognize and distinguish it from other benign tumors known to occur at this site. The possibility of PLGA must be considered in cases of oral cavity tumors, such as the tongue.ACC: adenoid cystic carcinoma; PLGA: polymorphous low-grade adenocarcinoma; SMA: smooth muscle antigenWritten 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.The authors declare that they have no competing interests.KG and RG performed the histological examination of the tumor. They were also major contributors in writing the manuscript. GR analyzed and interpreted the patient clinical data and also carried out the excision of the lesion. All authors read and approved the final manuscript.


Although Meckel's diverticulum is the most common congenital abnormality of the gastrointestinal tract and modern imaging techniques are available, its diagnosis remains problematic.A technetium-99 scan was performed in an 18-year-old man with abdominal pain, vomiting and rectal bleeding to confirm the presence of a Meckel's diverticulum which was not diagnosed laparoscopically elsewhere. The technetium-99 scan was positive and a diagnostic laparoscopy was re-performed which revealed a Meckel's diverticulum that was subsequently resected.We suggest that a technetium-99m scan should be performed before laparoscopy in children and adolescents with suspected Meckel's diverticulum. A positive technetium-99m scan may significantly contribute to the laparoscopic definitive diagnosis and treatment of a bleeding Meckel's diverticulum. However, diagnostic laparoscopy should only be performed by experienced surgeons. Meckel's diverticulum (MD) is the most common congenital abnormality of the gastrointestinal tract, occurring in 1% to 3% of the population . AlthougAn 18-year-old man was referred to the accident and emergency department of our hospital with a 5-day history of abdominal discomfort, vomiting and fresh blood in his stools. The patient had passed bloody stools five times in the last 36 hours. The clinical evaluation revealed diffuse abdominal pain, localized mostly in the right lower quadrant with no signs of peritoneal irritation. The rectal examination was positive for blood. His vital signs were: blood pressure 110/70 mmHg, pulse rate 92/minute, respiratory rate 16/minute, and temperature 37.10 C. Physical examination of the other systems showed no abnormality. Laboratory tests returned the following values: white blood cell count 7.53 K/μL ; red blood cell count, 3.94 M/μL; hematocrit, 29.3%; hemoglobin, 11.3 g/dL; platelets, 239 K/μL; C-reactive protein, 0.1 mg/dl; erythrocyte sedimentation rate, 1 mm/hour; and normal biochemical parameters and urinalysis. The patient had a long history of recurrent abdominal pain. From the age of 12, he started occasionally having mild abdominal pain located mostly in the right lower quadrant and radiating into the back. He was not passing bloody stools. Three years previously, an ultrasound of the upper and lower abdomen was performed which revealed findings of ileitis. Therefore a computed tomography (CT)-scan of the abdomen with contrast was obtained which showed a thickened wall of the terminal ileum and a small amount of fluid at the lower limit of the cecum, inside the small pelvis. The possibility of the presence of an inflammatory disease of the bowel was considered and a colonoscopy with multiple biopsies was performed. The colonoscopy showed no macroscopic abnormalities of the large-bowel mucosa and the terminal ileum . However, histological findings were compatible with Crohn's disease. Although the patient received mesalazine, his clinical condition deteriorated and therefore a more detailed evaluation was performed including video capsule endoscopy, CT enteroclysis with barium meal and magnetic resonance imaging (MRI) of the abdomen. Nevertheless, these investigations showed no abnormality. An exploratory laparoscopy was undertaken for suspected MD without discovering any findings which could explain the patient's symptomatology and an incidental appendicectomy was performed.The patient was admitted to our hospital for further investigation. A Tc-99 m scan was performed which revealed an accumulation of pertechnetate in the abdomen laterally to the bifurcation of the iliac artery Figure . This fiet al.[According to a statement from the Mayo Clinic, "Meckel's diverticulum is frequently suspected, often looked, and seldom found". Therefore, the diagnosis of MD is difficult . There aet al. reportedTc-99m scan is a widely accepted imaging modality for the discovery of a MD, especially in children with lower gastrointestinal (GI) bleeding . The affDespite the availability of modern imaging techniques, diagnosis of MD is challenging. In a study of 17 patients, the diagnosis of MD was established in 8 (76%) patients with laparoscopy. Only 4 (50%) of these eight patients had positive Tc-99m scan findings . Howeveret al.[et al.[When the findings from the Tc-99m scan are uncertain for the diagnosis of a MD, a diagnostic laparoscopy should be performed. The safety and efficacy of diagnostic but also therapeutic laparoscopy are widely accepted . A greatet al. performel.[et al. advocatel.[et al.. In the l.[et al..MD should be included in the differential diagnosis of recurrent abdominal pain or rectal bleeding, especially in children and adolescent patients. The most helpful non-invasive diagnostic tool in the diagnosis of a bleeding MD is the Tc-99m scan that should precede laparoscopy in patients with lower GI bleeding. In cases with suspected MD, a positive Tc-99m scan in combination with laparoscopy, performed by experienced surgeons, can definitively confirm the diagnosis and treat the patient.CT: computed tomography; GI: gastrointestinal; HGM: heterotopic gastric mucosa; MD: Meckel's diverticulum; MRI: magnetic resonance imaging; Tc-99m scan: technetium-99 scan.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.The authors declare that they have no competing interests.MP conceived the study, performed the laparoscopic operation, followed up the patient, and wrote the manuscript. GV designed the study, contributed to the writing of the manuscript and drafted the final manuscript. MK contributed to the writing of the manuscript and followed up the patient. SG helped with study design and contributed to the writing of the manuscript.ET followed up the patient and reviewed the literature. CS helped with design of the study and contributed to the writing of the manuscript. All authors read and approved the final manuscript.


Esophageal squamous cell carcinoma (ESCC) is the most common histological subtype of cancer in the upper and middle esophagus and is characterized by a high rate of mortality. The incidence of esophageal cancer varies greatly among regions of the world and occurs at a high frequency in Asia and South America.In our department, a 51-year-old man was diagnosed with ESCC after presenting with extensive disseminated skin nodules. Biopsy of the nodules showed metastatic ESCC. Cutaneous manifestations of esophageal neoplasia are very rare and are mainly described for esophageal adenocarcinoma (EADC). Here we report a very uncommon case of extensive skin metastases of ESCC.Early biopsies of suspicious skin lesions are important and should be performed in patients with unclear symptoms such as weight loss or dysphagia and especially in patients with a history of cancer, since they can reveal the existence of a distant malignant disease leading to diagnosis and prompt therapy. Cancer of the esophagus is the ninth most common malignancy and ranks as the sixth most frequent cause of cancer death in the world, constituting 7% of all gastrointestinal cancers . PatientIn general, skin metastases from malignant tumors of the internal organs are rarely seen, with a frequency of between 0.7 and 9% -5. The oA 51-year-old man was admitted to our department with a four-week history of dysphagia, weight loss and nausea. He had a medical history of multiple sclerosis since April 2004 and a smoking history of 30 pack-years. The patient underwent esophagogastroduodenoscopy resulting in the diagnosis of esophageal carcinoma located in the mid-thoracic part of the esophagus. Histology of an endosonography-guided biopsy showed an intermediate grade ESCC according to the criteria of the American Joint Committee of Cancer (AJCC). Moreover, the patient presented with approximately 20 diffuse, painless and solid skin nodules that were about 1–3 cm in diameter, found all over his body surface including the scalp, upper extremities, axillae, back, chest and abdominal wall. According to the patient they had been growing rapidly over the previous four weeks, and he had noticed the first skin lesion more than two months earlier. Excisional biopsy of one representative prominent cutaneous formation on the abdominal wall was performed. On macroscopic inspection, the lesion was superficially ulcerated and measured 2 cm × 3 cm , pulmonary cancers (n = 36) and melanomas (n = 31) [n = 77) and breast cancer (n = 212). The esophagus was the primary site in only three cases, spreading mainly to the chest and abdomen [Due to the extreme rarity of cutaneous metastases from ESCC, there are only limited data in the literature regarding their incidence. Fereidooni and colleagues reported a solid facial skin metastasis of EACC . Two add(n = 31) . Looking(n = 31) . In this abdomen . Tharaka abdomen .Skin manifestations of ESCC are extremely rare and only a small number of cases with solid skin metastases have been reported. A case of ESCC with such diffuse and massive skin metastases, most likely indicating highly aggressive disease, has not been described previously. Our patient complained about these unusual cutaneous metastases before any of the more usual symptoms such as dysphagia or weight loss were manifested.The authors declare that they have no competing interests.GBI initiated the report and undertook the majority of the writing of the manuscript. AB made substantial contributions to the conception and design of the report and was involved in drafting the manuscript. GK, JM and SC made contributions to the conception and design of the report and was involved in drafting the manuscript. WF and CB made substantial contributions to the conception and design of the manuscript and revised it critically for important intellectual content. All authors read and approved the final manuscript.Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.