Intercostal lung herniation is a rare condition that may be congenital (20%) or acquired (80%). The isolated congenital form is exceptional, with one case reported in the literature.We report a case of a 10-year-old French boy of Algeria origin, born with intermittent swelling of his right hemithorax. The swelling and pain gradually increased with age. A clinical examination revealed a localized swelling of his right hemithorax at the level of the midclavicular line and the fifth intercostal space. The swelling increased in size during respiratory movements and enlarged with Valsalva maneuvers. The intercostal lung hernia was treated by thoracoscopy.This is the second case of isolated congenital intercostal pulmonary hernia reported in the French and English literature. It is the first to be treated by thoracoscopy. Based on this case we performed a review of the diagnosis and therapeutic aspect of pulmonary hernias.The online version of this article (10.1186/s13256-019-2142-4) contains supplementary material, which is available to authorized users. A lung hernia is rare, particularly among children , 2. In 8A consensus has not been reached among physicians regarding the management of lung hernias , 7. ThisThis clinical case is exceptional because of its isolated congenital nature. All these reasons justified our sharing of our experience because for this rare pathology there is no codified treatment.This case report describes the case of a 10-year-old boy of Algeria origin living in Ile-de-France. Due to extremely painful thoracic swelling, the boy’s parents brought him in September 2012 to our hospital. This small nodular swelling was apparent from birth and progressively increased in volume. The chest pain appeared when the boy was 10-years old and his parents consulted for a treatment request at the pediatric hospital Robert Debré. The boy failed to receive treatment prior to this. He had never experienced thoracic trauma and he did not possess any known prior medical pathology. He had no risk of exposure to toxins in his environment. Moreover, no similar case was noted in his family and there is no hereditary disease and no consanguinity between parents.An initial clinical examination showed a well-developed child with no other physical abnormalities. His weight was 41.7 kg, blood pressure 85/140 mmHg, pulse rate 60 pulses/minute, respiratory rate 15 cycles/minute, and temperature 37 °C. His Glasgow Coma Score was 15/15. His cognitive functions were preserved. Sensitivity, motor skills, and osteotendinous reflexes were preserved in his limbs. There was no motor coordination disorder. There was no sphincter deficit. However, a clinical examination revealed a swelling of the right hemithorax 5 cm × 2.5 cm) on the midclavicular line and the fifth intercostal space. His respiratory movements caused the swelling to vary and enlarge with Valsalva maneuvers. Pulmonary and cardiovascular auscultation was normal. We diagnosed a congenital intercostal lung hernia based on the clinical information. A standard X-ray of his chest showed no anomaly for his lungs and thoracic wall  was asymptomatic at first. It was at the age of 10, when chest pain appeared, that the parents consulted for a treatment request. Except for the swelling of the right hemithorax, the clinical examination was normal.We chose thoracoscopic surgical treatment. This case is unique and the surgical indication is the first case reported in the literature. The sharing of our experience could allow clinicians to discover or better know this pathology.et al. about an 7-year-old child in 2004 [In 1946, Maurer and Blades defined a pulmonary hernia as a protrusion of the pulmonary tissue enveloped by the pleura through a defect of the thoracic wall , 9. ThisOur case report is the second case of ICILH in the literature. Information from a clinical examination was sufficient to make the diagnosis of intercostal lung hernia.ICILH can be silent and undiscovered during a clinical examination or it can show symptoms if the size is big . When smAs some authors reported, a standard X-ray of the thorax is useful to look for associated lesions , 8. Our Regarding the treatment, for small hernias, the indications are not codified; there are many controversies. One group of authors advocated abstention or a simple strapping while waiting for spontaneous resolution , 13–15. We think it is not an emergency and when a small-sized hernia is discovered early, it should just be monitored. Surgery will be indicated in the case of symptoms such as pain, a persistent cough, an episode of strangulation, or hemoptysis , 2. A coThe intervention can consist of a thoracotomy , 13. Theet al. in 1999 [Treatment of an intercostal hernia by thoracoscopy was described by Van Den Bossche  in 1999 . This me in 1999 .We report here the first case of treatment by thoracoscopy of an ICILH. It allowed an optimal exploration of the endothoracic wall and the pulmonary parenchyma to look for associated anomalies. The defect of the wall was easily reachable for repair as reported by other authors .The closing of the defect in ICILH can be made through a simple suture or, in cases of large collars, by a transplant of fascia lata or prosthesis as in our patient .Related to the evolution, the results are satisfactory in the different series or reported cases. Recurrences are rare, but a long-term follow-up seems to be necessary.We agree with other authors that in large pulmonary hernias with or without symptoms, the complications of not treating are greater than complications from treatment .Congenital Intercostal lung hernia is a rare pathology and the isolated congenital form is exceptionally rare. We report the second case of thoracic ICILH in the literature. The diagnosis was made clinically.Clinical monitoring is recommended in cases of small asymptomatic hernia, but if the hernia becomes larger with or without symptoms, surgical treatment is indicated. We performed the first thoracoscopy for ICILH. It allowed a simple and efficient surgical treatment, without the inconveniences of thoracotomy.Additional file 1:Video 1. Begining of polytetrafluoro-ethylene mech insertion for closing the defect of intercostal hernia in thoracoscopy. (MP4 2866 kb)Additional file 2:Video 2. Sequence 2 of prothesis fixation on intercostal hernia. (MP4 34887 kb)Additional file 3:Video 3. Sequence 3, showing the following fixation of the prosthetic plate on the intercostal hernia. (MP4 48585 kb)Additional file 4:Video 4. Sequence 4 of prothesis fixation on intercostal hernia. (MP4 24653 kb)Additional file 5:Video 5. Sequence 5, end of the first semicontinuous suture of prothesis on intercostal hernia. (MP4 113395 kb)Additional file 6:Video 6. Sequence 6, performing of the second semicontinuous suture of prothesis on intercostal hernia. (MP4 136579 kb)Additional file 7:Video 7. End of the intercastale hernia repair. (MP4 87975 kb)Additional file 8:Video 8. Final thoracoscopic appearance of the intercostal hernia repair by prosthesis. (MP4 1642 kb)


Agenesis of the dorsal pancreas is very rare. Less than 70 cases have been reported to date. Some of these cases had an association with a tumor. The literature of agenesis of the dorsal pancreas and agenesis of the dorsal pancreas-associated pancreatic neoplasia is limited. Here we report the second case of a pancreatic neuroendocrine tumor in a setting of agenesis of the dorsal pancreas.A 71-year-old man, originally from North Africa, with a history of insulin-dependent diabetes mellitus, presented with a 2-month history of nonspecific abdominal symptoms. Contrast-enhanced computed tomography demonstrated an almost 3 cm round, quite well-defined and homogeneous tumor formation in the area between the neck and absent body and tail of his pancreas. The mass was confirmed by endoscopic ultrasound. Our patient underwent computed tomography-guided biopsy of the mass which provided proof of a neuroendocrine tumor. He underwent pancreas resection because of the presence of a neuroendocrine tumor. Seven months later his glycated hemoglobin increased from 6.9 to 8.7%.Diagnosis of agenesis of the dorsal pancreas is based on imaging techniques like computed tomography, magnetic resonance cholangiopancreatography, or endoscopic ultrasound. Endoscopic ultrasound-guided fine-needle aspiration can be helpful for the histological diagnosis of the tumor. The hypothesis of the association between pancreatic neoplasia and agenesis of the dorsal pancreas leads us to the suggestion that every patient with diagnosed agenesis of the dorsal pancreas should be observed with a focus on the early detection of potential malignancy. Pancreatic development is a complex process with a fusion of the ventral and dorsal bud. Development anomalies of the pancreas have been described but agenesis of the dorsal pancreas (ADP) is very rare. Around 60 cases of ADP have been published up to 2017. More than ten reported cases of ADP were associated with a pancreatic tumor.Here we report a rare case of ADP with a neuroendocrine tumor; only one case has been published before.A 71-year-old man, originally from North Africa, with a history of insulin-dependent diabetes mellitus, presented to our emergency department with a 2-month history of nonspecific abdominal symptoms: meteorism (bloating) and a subjective feeling of abdominal enlargement. Diarrhea, loss of appetite, weight loss, persistent fever, night sweats, headaches, anxiety, gastric ulcer disease, or skin rash were not reported. He did not report a neoplasm in the past and he had not undergone an abdominal examination before. He could not recall any episodes of pancreatitis or suffering from gall bladder stones. He had undergone a computed tomography (CT) scan a few days before which showed a tumor in the pancreatic head and he was referred to our clinic. Diabetes mellitus occurred 20 years ago and he was initially treated with orally administered anti-diabetic drugs for more than 12 years. Apart from diabetes and arterial hypertension he had no previous medical or surgical history. He has been treated with two different anti-hypertensives, two diuretics, acetylsalicylic acid, and insulin glargine. He reported an allergy to metformin. He did not smoke tobacco or drink alcohol.2). His lungs were clear to auscultation and percussion bilaterally. His heart examination was also normal. He had a soft, non-tender abdomen without any palpable masses. Icterus and lymphadenopathy were absent. His vital signs were normal.On general physical examination, he was conscious and oriented and in fair general condition. He appeared to be in a good nutritional state , hematocrit 25% (40–53), mean corpuscular volume (MCV) 78 fl (82–98), uric acid 10.2 mg/dl (< 7.0), creatinine 1.8 mg/dl (< 1.4), and random serum glucose 148 mg/dl. Transaminases and cholestasis parameter were normal. Glycated hemoglobin (HbA1c) was 6.9% (52 mmol/mol). The serum tumor markers carcinoembryonic antigen (CEA) and cancer antigen 19–9 (CA19–9) were in normal range. Serum pancreatic lipase was slightly elevated . The pancreatic elastase in stool was normal. There was no clinical evidence for exocrine pancreatic insufficiency.An ultrasound of his abdomen revealed a tumor of the uncinate process of the pancreas. The endoscopic ultrasound showed a mass in the area between the neck and the body of his pancreas, measuring approximately 24 mm in its largest dimension. The tumor compressed his splenic vein; however, there were no signs of infiltration. Contrast-enhanced CT demonstrated an almost 3 cm round, quite well-defined and homogeneous tumor formation in the area between the neck and absent body of his pancreas Fig. . The taiThe initial fine-needle aspiration was performed via endoscopic ultrasound. However, the pathological analysis did not detect tumor cells in the aspiration material.In the next step our patient underwent CT-guided biopsy of the mass. A histopathological examination Fig.  revealedOur patient underwent pancreas resection because of the presence of a neuroendocrine tumor. On macroscopic examination, the tumor was 1.8 cm in maximum diameter . There was no vascular invasion present. None of the three lymph nodes was metastatic . The active proliferative rate of Ki-67  was 5%.Well-differentiated neuroendocrine tumors of the pancreas usually show the characteristic nested, trabecular, or gyriform pattern known from neuroendocrine neoplasms of other organs. There are, however, architectural and cytological variations in some tumors, including abortive gland formation or oncocytic and clear cell changes.In our case, the differential diagnosis of the biopsy specimen was focused on a distinction from other epithelial pancreas neoplasms especially the solid variants of acinar cell carcinoma, solid pseudopapillary neoplasm, as well as pancreatoblastoma. The latter tumor types may also exhibit the nesting pattern observed in our case. Therefore, the intensive homogenous staining pattern for synaptophysin in our case provided an important argument for the diagnosis of a neuroendocrine tumor. This diagnosis was further corroborated in the resection specimen by the lack of features defining acinar cell carcinoma  or pancreatoblastoma or solid papillary neoplasm .68gallium-DOTATOC positron emission tomography (PET)/CT scan 4 months later, which did not show increased metabolic uptake. Seven months after pancreas resection his HbA1c increased from 6.9 to 8.7%. This led us to modify his diabetes medication to a combination including insulin glargine, insulin glulisine, and metformin.Our patient underwent a This section discusses ADP and presents a systematic review of cases of pancreatic tumors in patients with ADP. Dorsal agenesis of the pancreas is a very rare congenital pancreatic malformation. Pancreatic development is a complex process and results from the fusion of the ventral and dorsal bud. The ventral bud forms the head and the uncinate process while the dorsal bud forms the upper part of the head, isthmus, and body tail of the pancreas . Dorsal ADP is mostly asymptomatic, but common presenting symptoms include diabetes mellitus, abdominal pain, pancreatitis, enlarged pancreatic head, and, in a few cases, polysplenia .Raldh2) and gene H1xb9 or deficiency of retinoic acid in mice studies leads to ADP [The first description of this condition was published in 1911 in an autopsy finding . Since ts to ADP , 5.TablePatients with ADP are mostly asymptomatic and ADP is detected during an evaluation for an unrelated cause, so that most cases of ADP are incidental findings. The clinical manifestation of ADP varies extremely from abdominal pain, pancreatitis, and diabetes mellitus to exocrine insufficiency with steatorrhea .The association of pancreatic neoplasia and ADP has not been studied extensively, so the mechanism is unclear. Some theorized that ADP increases the risk of chronic pancreatitis, which in and of itself is a risk factor for pancreatic tumors. Our patient did not report any symptoms which can be associated with a chronic pancreatitis. An endoscopic ultrasound as well as CT scan did not show any sign of chronic pancreatitis. Gastroscopy before endoscopic ultrasound showed some ulcers, so that the hypothesis of a gastrin-producing tumor was favored. This hypothesis could not be confirmed by the immunohistochemistry of the tumor.While the diagnosis of ADP was made at autopsy or laparotomy in the past, with the increasing availability of newer imaging techniques the diagnosis of ADP increased in the last decade. Diagnosis of ADP can be made by ultrasound, CT, magnetic resonance cholangiopancreatography (MRCP), and endoscopic retrograde cholangiopancreatography (ERCP). In the differential diagnosis, periportal lymphadenopathy should be considered as well as anatomic variations , 8.We present a systematic review of pancreatic tumors associated with dorsal agenesis of the pancreas in Table Here we present the 17th case of an ADP associated with a tumor. It is the second case report of an ADP-related neuroendocrine tumor. Our patient presented nonspecific abdominal symptoms: meteorism (bloating) and a subjective feeling of abdominal enlargement. A diagnosis of ADP was made with imaging techniques (CT and ultrasound). A diagnosis of neuroendocrine tumor in this setting needs histopathological or cytological confirmation, which can be done by a CT-guided biopsy or fine-needle aspiration via endoscopic ultrasound. If the tumor has been confirmed, then surgery is needed to resect the tumor.The hypothesis about the association between pancreatic neoplasia and ADP leads us to the suggestion that every patient with diagnosed ADP should be observed with a focus on the early detection of potential malignancy.


Primary spinal epidural/extramedullary ewing sarcoma (ES) is a rare extraosseous lesion. Extraosseous ES has a similar demographic as osseous ES, primarily affecting adolescents and young adults and male propensity. Reported 5-year survival is 0% to 37.5% for spinal extraosseous ES.Two girls, 19 and 14 months old, presented with progressive lower extremity paraplegia and incontinence. Both had a compressive epidural/extramedullary mass without metastases and underwent decompression with multilevel laminectomy and tumor excision. Primary spinal epidural/extramedullary ES was diagnosed.Case 1 received 34 weeks of chemotherapy and radiation therapy, and case 2 received 14 cycles of chemotherapy and autologous stem cell rescue without radiation therapy. After more than 5- and 8-year follow-up, case 1 and case 2 are walking and disease-free, respectively.These cases are the youngest presentation reported for primary spinal epidural/extramedullary ES and suggest that toddlers have a better prognosis for survival than older children and adolescents. Extraosseous ES has a similar demographic as osseous ES, primarily affecting adolescents and young adults with a male sex propensity. We report two cases of primary spinal epidural/extramedullary ES in young female patients who presented with neurologic compromise.Ewing sarcoma (ES) is a malignancy of the bone and rarely soft tissues. Although osseous ES is the second most common primary bone tumor in pediatric patients,A 19-month-old white female patient presented with a 2-week history of refusal to bear weight, lower extremity pain, and acute onset of bowel and bladder incontinence. She was previously walking for 6 months. Her medical history was normal. Examination revealed no response to stimuli of her feet, decreased tone in her bilateral lower extremities and 3/5 strength with hip flexion and knee extension. She could not plantar or dorsiflex her ankles or toes. She exhibited 2/4 knee reflexes, absent ankle reflexes, negative Babinski signs, and no clonus. Rectal tone was absent with visible bladder incontinence.A total spine MRI with and without contrast revealed an epidural/extramedullary well-circumscribed mass extending from L3 to S1 with spinal cord effacement Figure , A and BHistopathologic examination revealed an undifferentiated small blue cell neoplasm Figure , A. Immu4 regimen B, and local radiation therapy with a total dose of 5580 cGy. She is currently walking and in remission at her most recent 5-year follow-up. Remaining deficits include neurogenic bowel and bladder, 4/5 strength with bilateral ankle dorsiflexion, diminished sensation in the S1 distribution, and absent knee and ankle reflexes. Pediatric Outcome Data Collecting Instrument Global Functioning Scale standardized and normalized scores were 84 and 36, respectively.Additional evaluation with bone marrow aspiration, positron emission tomography (PET) scan, and bone scan did not reveal evidence of metastatic disease. She underwent 34 weeks of chemotherapy per the AEWS1031 protocol,A 14-month-old white female patient presented with a 1-month history of progressive lower extremity weakness and urinary incontinence. She was previously walking for 3 months and regressed to inability to bear weight or stay seated due to trunk weakness. Her medical history was normal. Examination revealed weak withdrawal of her legs to noxious stimuli, minimal spontaneous leg movement, and flaccid tone. She exhibited 0/4 knee reflexes, 1/4 ankle reflexes, positive Babinski sign bilaterally, and bilateral ankle clonus. She had decreased trunk strength and sensibility below T10. Normal rectal tone was present.A total spine MRI with and without contrast revealed an epidural/extramedullary well-circumscribed mass extending from T7 to T12 with spinal cord displacement. Mass was isointense on T1, hyperintense on T2, and enhanced heterogeneously with contrast  confirm the diagnosis of ES.3 Case 1 and case 2 are walking and disease-free after 5- and 8-year follow-up, respectively. Although no conclusions can be made from two patients, it is suggestive that toddlers presenting with primary spinal extradural ES have a better prognosis for survival than older children and young adults.Prompt decompression was necessary for these two patients because of progressive neurologic compromise. These favorable outcomes are significant with respect to this tumor's characterization of aggressive growth, rapid progression, local recurrence with invasion of adjacent structures, and dismal prognosis. Reported 5-year survival is only 0% to 37.5% for extraosseous ES around the spinal canal.


The aim of this case report is to underline surgical strategies for complications in a case of a young man with fibrosarcoma of the bone treated with pelvic resection followed by reconstruction with massive bone allograft. A type I pelvic resection was performed as a radical resection of tumor followed by a biological reconstruction of iliac wing using frozen allograft. The iliac allograft was fixed in place using 4 screws. The immediate postoperative period was complicated with local sepsis of reconstructed site treated with pediculate omentoplasty. After 1 year from surgery, the X-ray exam showed an integrated allograft. After 20 years from the first surgery, the patient presented with the left hip pain of 3-month duration with mechanical pattern. The X-ray and CT exam showed the left hip arthritis and no signs of recurrence. A total hip arthroplasty with dual mobility cup and uncemented stem was performed. Despite the immediate postoperative local infection, the allograft was left in place and integrated after all. Omentoplasty could be a very useful technique in eradicating local infection, due to the immunogenic properties of the omentum. The allograft is still strong enough to give support for a hip arthroplasty at 20 years after implantation. The prognosis and survival in primary pelvic bone tumors are less favorable than in other locations due to the complexity of anatomy of the region and secondary to the difficulty of radical resection surgery, meaning an increased rate of recurrence . The goaA 24-year-old man presented himself with the left hip pain with nocturnal exacerbation and difficulty in walking of 6-month duration. On physical exam, a firm painful mass was palpable in the posterior part of the left iliac wing. The tumor was fixed in relation to the adjacent structures. Also, the pain was exacerbated by the motion and prolonged orthostatism. Laboratory testing showed elevated levels of erythrocyte sedimentation rate and alkaline phosphatase. The lung X-ray exam was normal. The pelvis X-ray showed a highly destructive osteolytic zone in iliac crest above the acetabulum, close to the sacroiliac joint . The CT Next, a type I pelvic resection was performed as a radical resection of tumor followed by a biological reconstruction of iliac wing using frozen allograft. The iliac allograft was fixed in place using 4 screws .Adjuvant chemotherapy was needed in 6 stages using doxolem, cyclofosfamide, and methotrexate. No neoadjuvant chemotherapy was used. The immediate postoperative period was complicated with local sepsis of reconstructed site treated with pediculate omentoplasty. After 1 year from surgery, the X-ray exam showed an integrated allograft. The walking was possible without crutches, and the patient returned to normal activity. The partial resorption of the graft was observed in the unloaded area . Still, The hip was free of pain, and a shortening of 2 cm was assessed. The imaging studies showed no recurrence of the disease. After 20 years from the first surgery, the patient presented with the left hip pain of 3-month duration with mechanical pattern. The X-ray and CT exam showed the left hip arthritis  and no sThe usual complications after pelvic resections are intraoperative bleeding, infection, nerve injuries, ureter, bladder, and bowel injuries; wound-healing complications, dislocation of prostheses, allografts resorption, lower quadrant hernia, bowel ischemia, and late venous thrombosis . IncreasPelvic bone tumors remain a great challenge for surgeon in terms of oncological resection and pelvic reconstruction. Despite the immediate postoperative local infection, the allograft was left in place and integrated after all. Omentoplasty could be a very useful technique in eradicating local infection, due to the immunogenic properties of the omentum. The allograft is still strong enough to give support for a hip arthroplasty at 20 years after implantation. Even if tumoral disease is cured, major complications are still possible any time after the first surgery of resection reconstruction, meaning that a close follow-up is necessary.


Colonic lipomas are uncommon and usually asymptomatic tumors. A 30-year-old woman with abdominal pain lasting 10 days was admitted to the surgical clinic. Her physical examination revealed sensitivity on the right upper quadrant and her bowel sounds were normal. A lesion and invagination findings in the colon were found in the ultrasound examination and CT was performed. CT scan revealed a lipoma and invagination in the colon and the patient has undergone surgery. Pathological diagnosis of the lesion was reported as submucosallipoma. In this case report, we present clinical and radiological findings of a submucosal colonic lipoma causing intussusception. Lipomas are benign nonepithelial adipose tumors that can be seen throughout the gastrointestinal tract, although they occur most frequently in the colon , 2. SiceA 30-year-old female patient was admitted to General Surgery outpatient clinic with complaints of abdominal pain which lasted for about 10 days and worsened during last 2 days. Abdominal pain was on both upper quadrants, especially on the right upper quadrant. Apart from abdominal pain, there was not any significant complaint . The patient's physical examination revealed normal blood pressure, pulse and body temperature and no abdominal distension. Tenderness was present on right upper quadrant, however, there was no rebound and bowel sounds were normal. Cardiac and pulmonary examination revealed no pathology. Leukocyte count (9.46x109/L), hemoglobin, hematocrit values and CRP (C reactive protein) and all other biochemical parameters were within normal limits in the laboratory tests performed. Whole abdominal ultrasound revealed an increase in the diffuse wall thickening of the 10 cm-intestinal segment in the right upper quadrant, presumably belonging to colon and an intraabdominal echogenic mass related with this segment in the upper quadrant, midline level. Upon this finding, intravenous contrast-enhanced computed tomography examination was performed. Iliocecal junction was observed in the upper abdomen by CT scan. The colon was the herniated towards the ascending colon pouch. Intussusception at the ileocecal junction and a heterogeneous mass with fat density measuring 40x53x55 mm were observed in this region. There were dense septal appearances in the mass . AccordiColonic lipomas are uncommon, slow growing, non-epithelial benign adipose tumors of the gastrointestinal tract , 7, 8. CLipomas larger than 4 cm are considered giant lipomas and 75% of them are symptomatic . There wColocolic intussusception, one of the causes of acute abdomen, is a clinical picture that is usually seen in childhood. Although it is seen rarely in adults, it is mostly due to malign tumors. However, large, colonic lipomas can also cause colocolic intussusception and lead to acute abdomen. For this reason, it should be kept in mind that patients presenting with ileus may have intussusception due to colonic lipoma.The authors declare no competing interests.


Burkitt lymphoma (BL), a highly aggressive B-cell non-Hodgkin lymphoma (NHL), usually presents in children and young adults with large extranodal masses involving jaw bones, gastrointestinal tract, and central nervous system. The three main subtypes of BL are endemic, sporadic, and immunodeficiency variant. Extranodal involvement is common in each variant of BL, although muscle tissue involvement is distinctly rare. Mode of spread may be hematogenous or via direct extension of the primary tumor. In this report, we present a case of a 41-year-old male who presented with a palpable mass in the buttock leading to foot drop as the initial manifestation of BL. An exhaustive review of the literature failed to discover any previous reports of BL occurring in this location. Lymphoma is defined as a neoplastic proliferation of lymphoid cells at different stages of differentiation and affects lymph nodes, but may infiltrate into the bone marrow, spleen, and the thymus. Lymphoma is ranked as the seventh most common malignancy in both males and females combined. Extranodal involvement is recognized as 25%-40% of cases of lymphomas and almost any organ can be involved. Extranodal lymphoma, by definition, involves sites other than lymph nodes, spleen, thymus, and the pharyngeal lymphatic ring . Burkitt A 41-year-old Ukrainian male with a past medical history of acquired immunodeficiency syndrome (AIDS) and syphilis presented to the emergency department with a complaint of sudden-onset left buttock pain for one day. The pain was described as severe, progressive, continuous, radiating to the left foot, associated with numbness and burning sensation of the left foot and foot drop. The patient also reported a palpable mass in the left buttock. The patient was diagnosed with human immunodeficiency virus (HIV) seven years ago and his most recent cluster of differentiation (CD)4 count was 20; he was not receiving treatment for AIDS. He was also a diagnosed case of latent syphilis for 12 years. His past surgical and family history was non-contributory. He denied smoking as well as alcohol and illicit drug abuse. The patient was sexually active with one male partner. The patient denied any fever, back pain, urinary/bowel habit changes, swelling in any other part of the body, history of trauma, sexual dysfunction, and appetite/weight changes.Pertinent findings during the physical examination of the lower limbs include diminished deep tendon reflexes (DTRs) of the left lower extremity, with a static response of plantar along with left foot drop. Sensation to touch was diminished on the left side in the L5, S2, and S3 dermatomal areas and was completely absent below the left ankle. The motor and sensory examination of the right lower extremity was completely normal. Local exam of the left buttock showed a firm, fluctuant, freely mobile mass measuring 4 x 4 cm with no overlying skin changes.The baseline investigations were unremarkable except hemoglobin (Hb): 9 g/dl, blood urea nitrogen (BUN): 18 mg/dl, and serum creatinine (Cr): 1.25 mg/dl. A left hip X-ray revealed a soft tissue density. Computed tomography (CT) scan of the pelvis confirmed a solid mass. A biopsy of the left buttock mass was performed which showed high-grade BL, a type of NHL  classification of lymphoid neoplasms as endemic, sporadic, and immunodeficiency-associated variants. Despite the presence of a considerable overlap, each variant displays unique genetic and clinical features. The endemic form is most commonly observed in children aged four to seven years in equatorial Africa, commonly involving the jaw and kidneys, although ileal, cecal, breast, and ovarian involvement have also been reported. This variant has been termed as endemic owing to its remarkably high incidence in equatorial Africa (50 times higher than in the United States) and the identical geographic distribution of this variant and endemic malaria. In contrast, the sporadic variant has not been associated with any specific geographic or climatic distribution, mostly presenting as an abdominal tumor. This clinical variant accounts for 1%-2% of all adult lymphomas in the United States and Western Europe .The immunodeficiency variant of BL, as seen in our patient, is commonly observed in the setting of HIV infection; however, unlike other HIV-related lymphomas, it has been reported in patients with CD4 counts above 200 cells/μL and those with no opportunistic infections . Adult pThe hallmark of BL is the overexpression of c-myc, most commonly the result of translocation 8;14), which causes the juxtaposition of the myc gene from chromosome 8 with the immunoglobulin (Ig) H region on chromosome 14 . The abe;14, whicNumerous staging systems have been formulated for BL due to increasing frequency of extranodal disease. Ann Arbor system is commonly referenced in adult trials, however, some researchers find this system inadequate due to its inability to fully describe the extent of extranodal involvement. Therefore, some trials prefer St Jude or Murphy staging schema. Notably, this staging system recognizes Burkitt leukemia as a separate entity, unlike the current WHO classification . During With the use of aggressive chemotherapy, the survival rates of patients with BL have improved particularly for children and adolescents, with a five-year survival rate of 85%-90% . In contThe present case, to the best of our knowledge, is the first to document buttock mass with foot drop as the initial manifestation of BL. BL involving such atypical sites can pose a diagnostic challenge to clinicians. Immunodeficiency variant of BL should be kept in mind in an HIV-positive patient presenting with a suspicious mass in an unusual site (such as the buttock as in this case), and a thorough evaluation including imaging and biopsy should be carried out.


The presence of koilocytic cells in the superficial epithelium led to immunohistochemical investigations with p16 antibody, which showed strong nuclear positivity and slight cytoplasmic positivity in >50% of the cells with >25% confluency. However, it was not possible to confirm the presence of HPV infection with further investigations due to logistic reasons. The lesion recurred twice within a short time despite the surgical resection following the first recurrence. Thus, this paper presents a case of proliferative verrucous leukoplakia, which demonstrated a significant resistance to routine treatment protocols recommended in the management of such lesions.This report describes a case of proliferative verrucous leukoplakia (PVL) of the gingiva with no discernible aetiology, which presented in a 36-year-old female. The initial nonscrapable gingival lesion was treated with CO White lesions in the oral cavity could represent a variety of aetiological factors including increased or abnormal keratin production in the oral epithelium, which is denoted by the term “keratosis” .In addition to keratotic lesions that occur due to tobacco use, they may also occur due to frictional, chemical, or thermal irritations as well as due to inflammation. Frictional keratosis is usually seen in areas of recurring mild mechanical trauma or irritation while chemical keratosis may occur as the result of the compounds in smokeless tobacco, certain toothpastes, inappropriately used acidic medications, and alkaline liquids , 3. UsuaOral leukoplakia (OL) is defined as a “white plaque of questionable risk having excluded (other) known diseases or disorders that carry no increased risk for cancer” . ProlifeRecently, Woo et al. have reported a subset of lesions, with the majority clinically presenting as OL, harbouring high-risk HPV subtypes and positive for p16 . In addiThis case report describes a rare case of early stage of PVL with no apparent aetiology that developed in a relatively young patient.A 36-year-old female presented with an asymptomatic whitish lesion, on the gingiva in relation to 13, 14, and 15. The lesion had existed for more than one year in duration which had initiated during her second pregnancy. She was otherwise healthy except for having mild hypochromic microcytic anaemia due to being a carrier for thalassemia trait. However, her serum ferritin levels were within the normal range. Her plaque control measures revealed brushing twice daily with a fluoridated toothpaste although supplementary tools were not used. Furthermore, the patient did not practice any risk habits such as betel chewing, smoking, or alcohol consumption.Intraoral examination revealed a linear, plaque-like whitish lesion on the palatal and buccal free gingiva of 13, 14, and 15 . No verrShe had no other significant extraoral or intraoral findings, and the radiological examination in relation to the site was normal . As baseThe histopathological analysis revealed a hyperorthokeratinized stratified squamous epithelium which showed cytological atypia in the lower part amounting to mild epithelial dysplasia with numerous koilocytes in the stratum spinosum Figures  and 4.Furthermore, due to the presence of koilocytosis, immunohistochemical evaluation was carried out with p16 antibody which clearly revealed positivity in >50% of the cells with >25% confluency . It is aHowever, as p16 is only a surrogate marker, it was not possible to confirm the high-risk HPV infection as the etiological factor for the present lesion due to the absence of adequate amount of fresh tissue. Therefore, considering the histological findings together with the clinical features, early stage of PVL was derived as the possible diagnosis.Thus, following informing the patient regarding the findings, she was referred for HIV and gynecological screening. The HIV test was negative, and she had no abnormalities detected with the gynecology screening.However, despite the thorough probing into the possible causative factors including behavioural risk factors and habits, no aetiological factor for this oral lesion was revealed, except for p16 positivity, which could not be confirmed as due to a high-risk HPV infection with further investigations.2 laser was carried out following improving the oral hygiene. Nevertheless, the lesion reappeared within few weeks at the same sites which led to perform a surgical resection with an adequate margin in the second attempt.For the management of the lesion, resection of the lesion with adequate margins using COHowever, the lesion recurred similar to the initial presentation showing its resistance to excision. Thus, within a period of nearly six months, it recurred thrice despite electrosurgical interventions. Considering the diagnosis of PVL, currently, the patient is being followed up with short recall intervals and the lesion remains same with no significant change in size compared to the initial presentation.As the lesion described in the present report is a rare occurrence, difficulties were encountered when naming the lesion, especially with references to the terms “keratosis” and “leukoplakia.” According to literature, similar lesions have been named as linear gingival keratosis  as well In frictional keratosis histology of the spinous layer often demonstrate intraepithelial edema and occasional vacuolated cells with pyknotic nuclei resembling koilocytes . The dia2 lasers for oral lesions, particularly less intraoperative bleeding, minimal damage to adjacent tissue, delayed acute inflammatory reaction, and reduced myofibroblast activity, leading to reduced wound contraction and scarring [When considering the management, PVL is commonly treated with conventional surgery, electrocautery, laser ablation, and cryosurgery . Except scarring . In addiscarring , 4, 12, PVL is a progressive disease and may not respond to traditional treatment  similar When there is no obvious aetiology for oral PVL, the lesions may be considered as leukoplakia and managed accordingly . FurtherThe key feature in the case presented was the high recurrence rate showed despite the various treatment methods. Therefore, the patient should be followed up with close monitoring to detect any further increase in dysplastic changes which could follow with the presence of koilocytic cells and with PVL lesions. In addition, occurrence of early PVL lesions as gingival keratosis should be further explored.


Abdominal ectopic pregnancies are rare category of ectopic pregnancies.We describe a ectopic pregnancy implanted on the surface of diaphragm and highlight the difficult clinical course of an abdominal ectopic pregnancy managed with strategic procedural intervention.A 33-year-old Chinese woman with abdominal pain was diagnosed with ectopic pregnancy implanted in the liver according to CT and advised to transfer the patient to tertiary referral center. The patient had a significant increasing abdominal pain and intense reflex sensitivity in the right shoulder when she presented to our hospital. Laparoscopic surgery was decided immediately with the impression of ruptured abdominal pregnancy and hemorrhagic shock. It was found that ectopic pregnancy was implanted on the diaphragmatic surface.The pregnant tissue was completely resected from the diaphragm and bipolar electrocoagulation was used to control bleeding. The patient was stable and discharged 3 days after surgery in a good condition.CT or MRI should be considered as an alternative to TVS in the management of ectopic pregnancy patients with unusual presentations. A thorough observation of the entire pelvis and upper abdomen during laparoscopic exploration is crucial for diagnosis. Abdominal ectopic pregnancies are rare category of ectopic pregnancies, which occur most frequently in the douglas of pouch, omentum and organs such as liver, spleen and bowel and large vessels –4 are asA 33-year-old chinese woman with a history of previous cesarean section was referred to her local emergency room with 8 weeks’ delay of menstruation and frequent increasing pain in the right upper quadrant of her abdomen and intense reflex sensitivity in the right shoulder for a duration of one day. In her medical history there was no record of use of an IUD, endometriosis, pelvic inflammatory disease, tubal surgery, intrauterine device, or previous ectopic pregnancy. Laboratory evaluation showed quantitative hCG of 3129.94 IU/L and hemoglobin of 10.3 g/dL. TVS examination demonstrated no evidence of intrauterine pregnancy, a normal bilateral adnexa and a large amount of free fluid in the abdomen. Considering the above factors, a CT scan of the abdomen and pelvis was performed. A 90-mm-long mixed hypodense mass was evident on the upper surface of the right liver lobe. She was diagnosed with ectopic pregnancy implanted in the liver and advised to transfer the patient to tertiary referral center. The patient traveled to our hospital and presented to the emergency room with increasing abdominal pain and weakness. On examination, her pulse was 109beats/minute and blood pressure was 90/50 mmHg. Diagnostic abdominocentesis was performed and found blood uncoagulable. Given the concern for ruptured abdominal pregnancy and hemorrhagic shock, she was taken laparoscopic surgery immediately. The laparoscopy revealed a significant hemoperitoneum (of approximately 1500 ml of blood). The uterus and ovaries appeared normal, with a corpus luteum cyst in 50 mm on the left ovary. There was no evidence of bleeding from the pelvic organs while a mount of free blood appearred aroundding perihepatic and spleen. The upper abdomen was inspected and a active bleeding was discovered from the mass with the size of 80 mm* 50 mm on the surface of diaphragm  are implanted in the fallopian tube, while 0.9–1.4% of ectopic pregnancies are implanted in the abdominal cavity 7% are im, 11 thatEctopic pregnancy should be considered in all reproductive-age females presenting with abdominal pain or vaginal bleeding. If the patient is stable, the diagnosis is based on the detection of the dynamic changing of beta subgroup of hCG in blood as well as on TVS and physical examination findings . In patiAccording to the literature, more than 1% of ectopic pregnancies are associated with undetectable levels of hCG . DaniiliThere is no standard on the optimal management of abdominal pregnancy. Management approaches reported in the literature vary from medical treatments to surgery. Medical therapy with MTX has been reported successful for unruptured abdominal pregnancy , 18. HowCT or MRI could be another choice instead of TVS for the difficult patient. A thorough observation of the entire pelvis and upper abdomen during laparoscopic exploration is crucial for diagnosis. It is important to adequately assess the effectiveness and safety of management choice.


In this article, we report an unusual case of a malignant mesothelioma of the testis, presenting as hydrocele. The patient has a known medical history of trauma and occupational exposure to asbestos. The clinical features of this injury are discussed together with its immunohistochemistry. Surgical intervention is discussed due to the nature of this pathology. Indeed, the tunica vaginalis originates from the same embryologic source as the visceral pleura, peritoneum, and pericardium.5 Mesotheliomas most commonly arise from the pleural or peritoneal cavity and less than 5% of cases arise from tunica vaginalis of the testis.6Malignant mesothelioma of the tunica vaginalis of the testis is an exceedingly rare neoplasm but often fatal type of testicular malignancy that is often diagnosed only after surgery is performed. Since the description of the first case in 1957, less than 100 cases have been reported,8 asbestos exposure remains the risk factor most highly associated with this malignancy.9 Jones et al10 have found a positive occupational history in 41% of 27 reviewed cases, and Plas et al9 found a positive history of exposure in 34.2% of patients with mesotheliomas of the tunica vaginalis. In this article, we report a case of testicular mesothelioma of the tunica vaginalis that presented as a hydrocele, with a patient history of trauma and occupational exposure of asbestos.Most patients initially present with painless scrotal enlargement, hydrocele being the most common presentation. Although potential risk factors such as chronic hydrocele, trauma, recurrent epididymitis, and herniorrhaphy have been noted and associated with malignant mesothelioma of the testes,We present the case of a 63-year-old male of African origin (Kenya) with an occupational work exposure to boilers, and with a known medical history of uncontrolled hypertension, diabetes mellitus type 2, brain aneurysm that developed after a motorcycle accident, and a large left testicular hydrocele (8 × 3 cm) developing for over 1 year. The patient was seen at another hospital with complaints of testicular enlargement but did not receive medical treatment at that time. More recently, the patient underwent a spectral and color Doppler ultrasound revealing a large complex left hydrocele with a 1.7 cm regular, heterogeneous solid structure within the upper anterior aspect possibly representing the left testis. Surgery was then performed a month later, 1300 cc of straw-colored fluid was noted, and the hydrocele sac and left testicular nubbin were removed via scrotal orchiectomy. After pathological review and consultation, the specimen was confirmed to be malignant mesothelioma, epithelioid type. The tumor demonstrates predominantly an exophytic papillary growth although in areas it is solid and invasive into the underlying tunica vaginalis. The neoplastic cells are epithelioid with cuboidal to oval nuclei and eosinophilic cytoplasm -3. By imFollow-up for positron emission tomography/computed tomography scan did not reveal any hypermetabolic foci except for a small 9 mm hypodense nodule in the right lobe of thyroid gland.Radical excision is planned and chemotherapy planned after more extensive surgery.9 The use of adjuvant therapy has not been studied well and remains inconclusive. Studies have repeatedly found that aggressive surgical management is the most important therapeutic approach in reaching complete remission and that chemotherapy or radiotherapy have not shown a clear benefit altogether. da Fonseca et al reported a case of malignant mesothelioma of testis treated postsurgically with pemetrexed and cisplatin with very dismal effect.11 Patient passed 24 days after chemotherapy.Radical inguinal orchiectomy with complete histologic examination and follow-up for an adequate duration is accepted as the optimal primary treatment option for malignant mesothelioma of the tunica vaginalis. Recurrence rate after radical orchiectomy ranges from 10.5% to 11.5% versus conservative surgical management such as local excision of the hydrocele having a 36% recurrence rate.In patients presenting with scrotal masses, and whether a history of exposure to asbestos is present or not, malignant mesothelioma should be a part of the differential diagnosis. Ultrasonography can give substantial information, and in case of suspicious cases cytoanalysis of the hydrocele fluid may be diagnostic. Once the diagnosis is established, usually in the postsurgical setting by pathology, aggressive surgical management should be the first line of treatment.


We present a case of an incidentally detected cystic liver mass on CT scan, with histology showing biliary epithelium embedded in fibrous stroma and dysplastic features, consistent with an adenofibroma. This is only the third case described in the literature with malignant histology and the first case with angiographic imaging and subsequent management with preoperative embolization prior to surgical resection. We discuss the differential of the imaging findings and the features of this rare entity. Because of the risk of malignant transformation, consideration of this tumour is important to ensure early detection and ultimately resection for improved survival. We report a case of a 57-year-old female who was incidentally found to have a large hepatic mass on a CT scan performed for left flank and lower abdominal pain for 5 days. The patient had no history of jaundice, hepatitis or alcohol abuse. Her medical history included a prior blood transfusion.On clinical exam, the patient demonstrated modest left costovertebral angle tenderness. Her abdomen was soft, but she was tender in the left lower quadrant. Bowel sounds were somewhat decreased in the left lower abdomen. Laboratory data revealed a mild leukocytosis, and she was presumed to have diverticulitis and was treated with ciprofloxacin and metronidazole. Her symptoms worsened, and she presented to the emergency department the following day. At that time, her laboratory values had returned to normal.Laboratory work-up included liver enzymes, which were normal, as were her carcinoembryonic antigen and α-fetoprotein. Colonoscopy was also performed and was negative for malignancy., lobulated, heterogeneous, predominantly hypodense mass with irregular internal areas of enhancement. The mass was centred in the anterior segment of the right hepatic lobe, extending into the medial segment of the left hepatic lobe and deforming the liver contour  with dysplasia and architectural features concerning for malignant potential  and 3.2Right trisegmentectomy gross specimen revealed a 10 × 9.5 × 7.5-cm well-circumscribed mass comprising serous-filled cysts, multiloculated areas with spongy pink areas and fleshy-to-firm white–pink areas. There was a second lesion identical in appearance. Histologically, there were cystic, microcystic and tubular structures lined by cuboidal biliary epithelium embedded in fibrous stroma consistent with BAF. The fibroepithelial proliferation within the larger cystic spaces is characteristic of this entity . There wPortal venography pre-embolization showed normal-appearing lateral segment left portal vein (not shown). The medial left and anterior right portal vein branches were moderately displaced by the mass with external compression . PreoperWe report the imaging and pathological findings and management of a rare case of BAF of the liver. Based on dysplastic histological findings, this lesion was managed surgically. This is only the third case of reported malignant features and the first case treated with preoperative portal vein embolization.3; 7 cases of BAF have been reported in the pathology and gastroenterology literature, more commonly in women than men, with malignant progression/transformation documented in only 2 cases.BAF was first described in 1993 by Tsui et al11 There is a theoretical risk of seeding the tract. Fine needle aspiration cannot serve as an exclusive diagnostic method for malignant lesions because of false negatives. Improved diagnostic rate is obtained with cytology and histology. The risk of seeding the tract has been reported with an incidence of less than 0.009%.12 On histology, there is a resemblance to biliary hamartomas and bile duct adenomas. The large size would be atypical for biliary hamartoma. Nuclear accumulation of p53 and increased mitotic activity has been reported with premalignant lesions. Infiltration in the liver and the presence of metastasis are features suggestive of frank malignancy.6Patients may present with pain in the right upper quadrant from mass effect of the lesion. The location of this patient’s pain was atypical. Imaging findings have demonstrated a large subcapsular mass, occasionally multiple, in either cirrhotic or normal livers. Imaging features are non-specific, however, and biopsy is required. The role of biopsy in cystic lesions is debatable. If possible, it is advisable to target the wall or a nodular component to improve diagnostic yield. Yield can be lower in cystic or necrotic masses and give a false-negative cytological diagnosis because the aspirate is less cellular, with mostly macrophages.13 Because of the dual bloody supply, portal vein embolization does not cause complications associated with liver necrosis. Portal vein embolization has been performed successfully for liver and metastatic liver tumours. Various materials have been used for portal vein occlusion.14 Clinical management of this disease, including a recommended surveillance interval, has not been defined due to its rarity.Preoperative embolization is used to induce compensatory hypertrophy of the non-involved liver segment by causing an ischaemia in the area of the liver that will subsequently be removed by occluding its blood supply. Because the liver is able to tolerate a large resection and regenerate to ample size, portal vein embolization can lead to an increase in volume in the non-targeted liver segments.BAF is a very rare tumour of the liver.This lesion should be considered in patients with unusual complex cystic and solid masses of the liver.Malignant transformation can occur and early recognition, biopsy and, if indicated, resection, may improve survival.


Tumors of the heart are uncommon, and lymphangiomas are among the rarest of this group, with very few cases reported. These tumors consist of a benign slow-flow vascular malformation.We report a case of a man diagnosed incidentally with a pericardial mass and our surgical approach for its treatment.The pericardial mass of our case had no cleavage plane between the myocardium and the tumor. Because of this, rather than a total resection, with very high operative risk, a biopsy was preferred. The pathology specimen showed a benign tumor and the patient was referred to the cardiologist for regular follow-up.There is no specific investigation for diagnosing cardiac lymphangiomas, and though benign, they should be considered in the differential diagnosis of other malignant diseases of the mediastinum and definitive pathologic diagnosis is mandatory. Given the increased risk of uncontrolled bleeding related to percutaneous biopsy, the definite diagnosis is usually obtained with open surgical biopsy. The heaAlthough cardiac lymphangioma is of benign nature, it should be considered in the differential diagnosis of vascularized soft tissue masses together with other more aggressive pathologies, such as angiossarcoma, lymphoma and metastatic disease. Usually, the diagnostic approach is surgical, given the increased risk of bleeding related to percutaneous biopsy . DiagnosIn line with the SCARE criteria, we report a case of a man diagnosed incidentally with a cardiac mass and our surgical approach .2A 67-year-old male patient with no significant medical history presented to our institution to check a mediastinal enlargement incidentally found on routine chest X-ray. Echocardiographic examination revealed a solid mass surrounding the right cardiac chambers, and computed tomography of the chest confirmed the presence of a right lateralized 12 × 4 cm soft tissue mass beginning in the antero-superior pericardium recess down to the right atrium and right ventricle . The masThe subsequent techniques included a completely unremarkable coronariography with no signs of any neovascularization to the mass. The magnetic resonance imaging (MRI) confirmed the presence of an intrapericardial mass, with hypersignal in T2, localized in the anterior and superior pericardial recess, with inferior extension along the interatrial groove, and free wall of the right atrium and ventricle, surrounding the right coronary artery, but with apparent cleavage plane .Fig. 2MaMedian sternotomy approach was used to access the mass. The mass was completely adherent to the right atrium, right ventricle, and right coronary artery . Due to The patient had a normal post operative recovery and was discharged four days after the surgery. The patient was clinically well after one month.The pathology specimen showed a mass containing lymphoid tissue, and the immunohistochemistry stains were consistent with a cardic lymphangioma.3In this case, we have described a case of a pericardial mass for which the MRI suggested a cleavage plane between the myocardium and the tumor, which we were not able to confirm at surgery. Because of this, rather than a total resection with a very high operative risk, a biopsy was preferred due to the possibility of the tumor be benign.The pathology specimen showed a benign tumor and at 1-month follow-up, the patient was clinically well. He was referred to the cardiologist for regular follow-up with serial echocardiograms to monitor the behavior of the mass.4Lymphangioma is a very rare vascular malformation, hence there are very few cases reported in the literature . ConsequIn our case, as the patient was entirely asymptomatic and complete resection would have been a very complex and dangerous procedure, we opted for the surgical biopsy. Even in cases when the MRI does not show a visible cleavage plane, the safest, and more suitable option, is always a surgical biopsy because these masses tend to bleed easily. Therefore, a differential diagnosis with consideration of other malignant diseases is necessary, since there is no specific investigation to identify this particular tumor.The authors don’t have any financial and personal relationships that could influence their work.Nothing to declare.This is a case report. Informed consent was obtained from the patient and submitted to ethics committee of CHLN – Hospital Santa Maria according to protocol.Written consent was obtained from the patient for publication of this case and accompanying images. A copy of the written consent is available for review by the editor-in-chief of this journal on request.Ricardo Ferreira, Study conception and design, data collection, revision of the paper.Nadia Junqueira, Data collection and writing the paper.Joã, Writing and revision.Angelo Nobre, Revision and supervision.Not Applicable.Manuscript was approved by the Ethics Committee of Hospital de Santa Maria and Centro Académico de Medicina de Lisboa. The reference number is 365/18.A copy of the ethical approval is available for review by the editor-in-chief of this journal on request.Ricardo Ferreira.Not commissioned, externally peer reviewed.


The incidence of Bochdalek hernias in adults is much more than previously reported. Though most of these are asymptomatic. Tension gastrothorax is a rare complication of these. Literature about the endoscopic management of a tension gastrothorax is extremely sparse. In this case report, we describe how we combined laparoscopy and thoracoscopy in an emergency setting to manage a case of tension gastrothorax with an underlying Bochdalek hernia defect in an adult.A 27 year old male presented to our emergency with tension gastrothorax and a gastric volvulus with an underlying Bochdalek hernia defect, exhibiting the classical Borchardt’s triad. The patient underwent an emergency surgery, through a minimally invasive approach wherein the herniated contents were reduced, the gastric volvulus detorted and a repair of the diaphragmatic defect was performed. His post-operative course was uneventful.Tension gastrothorax, is a diagnostic challenge as an air-fluid level in the thorax on radiology with worsening respiration causes as many as 38% of these cases to be misdiagnosed as tension pneumothorax, hydro-pneumothorax, hemothorax, empyema, effusion or pneumonia. Various techniques of decompression have been described in literature but, employing those, in case of a misdiagnosis may have catastrophic outcomes.We strongly recommend employing a combined laparoscopic and thoracoscopic approach for an emergency repair of a tension gastrothorax in a hemodynamically stable patient as, it poses all the advantages of minimal access surgery and is available, at smaller centres, even in an emergency. The previously reported incidence of was 0.17%, erring on the conservative side. Though most of these are asymptomatic, symptoms and complications occur due to ventilatory compromise or luminal compression of abdominal viscera. Tension gastrothorax is a rare complication which is difficult to diagnose. An air-fluid level in the thorax on radiology with worsening respiration causes as many as 38% of these cases to be misdiagnosed as tension pneumothorax, hydro-pneumothorax, hemothorax, empyema, effusion or pneumonia  of tensi22.1A 27 year old gentleman presented to the emergency department at our institute, with a gradually worsening epigastric pain and retching without vomiting for one day. He had been breathless for two hours, without any precipitating trauma or history of similar episodes.On examination, he was anxious, tachycardic (136/minute), hypotensive (88/60 mm Hg) with an oxygen saturation of 84% in room air. His neck veins were visibly distended. He had a hyper-resonant note in the left hemithorax on percussion with absent breath sounds on auscultation with epigastric tenderness. Attempts to pass a naso-gastric tube were met with stiff resistance 30 cm from incisors.His chest radiographs showed a large air-fluid level in his left hemithorax, making a hydropneumothorax a probable diagnosis. The epigastric tenderness and inability to pass a naso-gastric tube were inconsistent with this diagnosis, which prompted a CT. The scan showed a vastly dilated stomach with a transition line between the antral and fundic air bubbles due to a volvulus . The sto2.2General anesthesia via single-lung ventilation was employed. With the patient in right lateral position, ports were placed. A diagnostic laparoscopy revealed a stretched left hemidiaphragm with convexity towards the abdomen. The stomach, spleen and left segment of transverse colon were missing, with a stretch on their vascular attachments. With an aim to reduce contents back into the abdomen, dissection commenced at the medial end of the posterolateral diaphragmatic defect. The omentum was reduced into the peritoneal cavity and adhesions to the colon were divided with ultrasonic shears. Due to an overdistended stomach, we were unable to reduce hollow viscera through the native defect. The defect was widened with a 2 cm incision medially, resembling a pendulum arm . This faThe abdominal viscera, particularly the spleen and stomach, tended to pull back into the thoracic cavity after reduction. This “memory” of herniated viscera combined with difficulty reaching the posterior edge of the defect prompted us to close the defect thoracoscopically. Isolated right-lung ventilation greatly aided the insertion of ports as depicted in The interval between arrival at hospital and surgery was 90 min. The operative time was 160 min. The patient was cared for in an intensive care unit for 2 days without need of ventilatory support. A CT scan on the 2nd postoperative day showed full expansion of the left lung without any evidence of herniation . He was 3The term “tension” refers to worsening pressures due to unidirectional air flow into the pleural space, with rapid clinical deterioration. Tension gastrothorax differs in causative pathology with fairly similar consequences as tension pneumothorax. The stomach prolapses into the thorax through a congenital or acquired diaphragmatic defect with pulmonary compression and ventilatory compromise. Due to rotation of stomach about its axis, its inlet and outlet are obstructed forming a “closed loop”. However, the acutely angulated gastroesophageal junction can function as a one-way valve allowing swallowed secretions and air to pass into the stomach causing progressively worsening distension. Like tension pneumothorax, gastrothorax causes worsening mediastinal and contralateral lung compression, which can culminate in cardiac arrest. This pathology was first described by Ordog et al. in 1984, in a case of traumatic rupture of the diaphragm . Five stAbnormal rotation of the stomach by more than 180 degrees is unusual, due to its numerous attachments. In congenital diaphragmatic hernia, these gastric attachments are lax or absent, increasing the risk of volvulus [Symptomatic Bochdalek’s hernias are rare in adults and acute tension gastrothorax, even more so. Due to its scarce occurrence, tension gastrothorax with an underlying Bochdalek hernia is not amongst the top differential diagnoses of a patient with respiratory distress and an air-fluid level on X-ray. Hydropneumothorax or tension pneumothorax are more likely, but hasty insertion of a chest tube in a misdiagnosed case can lead to hollow viscus or vascular injury with catastrophic outcomes. The clues in our patient were epigastric tenderness and an inability to perform naso-gastric intubation. The experience of our center in dealing with large diaphragmatic hernias further aided diagnosis. A chest X-ray improves diagnostic accuracy and one showing the tip of naso-gastric tube in an intrathoracic stomach or within the lower esophagus will confirm the diagnosis. But, a CT with coronal and sagittal images is the most useful form of imaging.Our case exhibited all three features of acute gastric volvulus - frequent unproductive retching, severe abdominal pain and inability to insert a nasogastric tube, originally described by Borchardt (Borchardt’s triad) . When un4Tension gastrothorax with an intra thoracic gastric volvulus, is a life threatening condition which can rapidly result in fatal cardiorespiratory distress and a cardiac arrest. Early diagnosis and surgery is extremely crucial in these cases. We strongly recommend employing a combined laparoscopic and thoracoscopic approach for an emergency repair in a young, hemodynamically stable patient.None.None.Our study is exempt from ethical approval.The patient consent has been obtained and will be sent to the Editor-in-Chief.Dr. Saurabh Gandhi helped draft the operative section of the manuscript and performed literature review.Dr. Ajay Bhandarwar was the lead surgeon on the case, helped draft the manuscript and reviewed it prior to submission.Dr. Nidhisha Sadhwani helped draft the manuscript discussion, edited images and is the corresponding author.Dr. Chintan Patel helped perform literature review and helped condense the manuscript to meet author guidelines.Dr. Amol Wagh was assistant operating surgeon and performed a literature review.Dr. Eham Arora helped draft the manuscript.Not applicable.Dr. Ajay Bhandarwar.Not commissioned, externally peer-reviewed.


Seed bezoars are a subcategory of phytobezoars, caused by consumption of indigestible vegetable or fruit seeds. We present the case of a 64-year-old male patient, who presented at the Emergency Department, complaining of constipation, tenesmus and rectal pain. History and digital examination revealed a rectal seed bezoar due to sunflower seeds, impacted in the lower rectum. The patient underwent manual disimpaction under general anaesthesia, after conservative measures failed. Seed bezoars represent a different pathophysiological process compared to fibre bezoars. They are usually found in the rectum of patients without predisposing factors, causing constipation and anorectal pain. History taking and digital rectal examination are the cornerstones of diagnosis, with manual disimpaction under general anaesthesia being the procedure of choice. Bezoars are retained concretions of indigestible foreign material that accumulate and conglomerate in the gastrointestinal tract lumen , 2. Mostrd postoperative day. At 30-day follow-up, the patient reported normal bowel movements and complete remission of anorectal discomfort.A 64-year-old man presented at the emergency department with a 24-hour history of faecal impaction, tenesmus and rectal pain. The patient reported consumption of large quantities of unshelled sunflower seeds over the past week. On admission, physical examination revealed a soft, non-tender abdomen, with no peritoneal signs. Digital rectal examination revealed a large phytobezoar with sharp edges, impacted in the distal rectum. Basic laboratory tests were all within normal range and an abdominal X-ray showed a faecal mass in the pelvis . Flexiblet al identified risk factors only in 7.2% of patients with seed bezoars, compared to 80.9% of patients with seedless faecal impaction [Bezoars can occur anywhere in the gastrointestinal tract, from the oesophagus to the rectum, however they are most commonly found in the stomach , 2. Recompaction , 4. Largmpaction -7. Wherempaction . Intestimpaction , 12. Whimpaction . Diagnosmpaction . In adulmpaction . Rectal mpaction . Dependimpaction , 16, 17.mpaction , 4. Morempaction , 4.Seed bezoars are a subgroup of phytobezoars, usually found in the rectum of patients without predisposing factors, causing constipation and non-specific abdominal or anorectal pain. Careful history and digital rectal examination are the sine-qua-non of diagnosis, in combination with rectoscopy and CT scans in selected cases. The majority of rectal seed bezoars can be manually disimpacted under general anaesthesia to minimise pain and discomfort. Intestinal seed bezoars on the other hand are usually found in the terminal ileum causing intermittent obstruction and require therefore surgical intervention.The authors declare no competing interests.


A 51-year-old Thai woman presented with bilateral leg edema and painful left inguinal mass for 6 months. Physical examination revealed matted bilateral inguinal lymph nodes up to 9 cm in size. Otherwise, physical examinations including skin were unremarkable. The result of the lymph node incisional biopsy is consistent with that of metastatic melanoma. The extensive investigation demonstrated multiple intra-abdominal and inguinal lymph nodes without detectable primary tumor. Palliative radiation and conventional chemotherapy were prescribed. The CT scan between treatments showed that the response was stable disease, but the following CT scan demonstrated a gradual decrease in size from August 2012 to November 2017 including the lesions outside radiation fields. Moreover, she developed vitiligo during a follow-up visit. The previous data reported the median overall survival among the patients who were treated with conventional chemotherapy ranging from 9.1 to 9.3 months and whose 5-year survival was less than 10%. This case represented a metastatic melanoma of unknown primary who achieved a durable response by conventional treatment. The clinical features including nodal-only disease, vitiligo, and abscopal effect of radiation were considered to be the favorable factors. An ie of age . Malignae of age , 2. Moste of age , 6. A 5-e of age , 4, 7. Te of age . Many hye of age , 9. ThisA 51-year-old Thai woman was hospitalized in July 2012 with edema at the left lower extremities and painful left inguinal mass for 6 months.Physical examination revealed matted bilateral inguinal lymph nodes up to 9 cm in size with hard consistency, erythema, and tenderness without fluctuation or ulcer. Marked swelling at both lower extremities was observed. There was no other superficial lymphadenopathy. Otherwise, physical examinations were normal.Incisional biopsy of the left inguinal lymph node revealed metastatic round cell tumor which is immunohistochemistry positive for vimentin, S100, and HMB-45. The immunophenotype is consistent with malignant melanoma .2 for 6 cycles. After completion of the planned chemotherapy, the symptom was slightly improved. The CT scan at the first 3 months showed that the response was stable disease, but the following CT scan demonstrated a gradual decrease in size over time from August 2012 to November 2017 (Therefore, primary tumors in the lower extremities, abdominal cavity, and anogenital organ were suspected. By complete skin examination, no cutaneous lesion was identified. Ophthalmoscopy, gastroscopy, colonoscopy, and cystoscopy were completely normal. Genital and pelvic examinations did not show any evidence of lesion. She denied previous abnormal or removal of cutaneous lesion. Computer tomography of the whole abdomen showed multiple enlarged lymph nodes throughout the abdominal and pelvic cavity up to 9.5 cm, along with compression of both iliac veins without an organ-specific lesion . CT chesber 2017 . During Systemic treatments of metastatic melanoma were developed for many decades since conventional chemotherapy has been considered a standard approach until the emergence of new drugs such as targeted therapy and immunotherapy over the last 10 years. The dramatic and durable response occurred by taking targeted therapy or immunotherapy but not by taking chemotherapy. In the patients who did not have access to those drugs, chemotherapy is a mainstay treatment. Previous data reported the median overall survival among the patients who were treated with conventional chemotherapy ranging from 7.7 to 16 months and whose 5-year survival was 8–18% .This case report represents a patient with metastatic melanoma of unknown primary with durable response by conventional chemotherapy and palliative radiation. When comparing prognosis, MUP tends to have a better prognosis than MKP as reported in previous studies –6, 8, 12Metastatic melanoma can achieve durable response by conventional chemotherapy and radiotherapy in patients with some clinical characteristics. The presence of the nodal-only disease, vitiligo, and effect of radiation seemed to be the favorable factors for better survival.


We describe a case of liraglutide-induced acute gastroparesis in a 52-year-old man with a history of well-controlled type 2 diabetes who presented with symptoms of gastric outlet obstruction. The patient responded markedly to conservative treatment with gastric suctioning, antiemetic and prokinetic therapy, and discontinuation of liraglutide with a resolution of his symptoms. This case highlights the importance of considering drug-induced gastroparesis as an etiology of unexplained upper abdominal pain, nausea, and early satiety, especially in the absence of mechanical obstruction. Gastroparesis is a syndrome of delayed gastric emptying without mechanical obstruction. The condition typically causes nausea, vomiting, early satiety, postprandial fullness, bloating, and/or upper abdominal pain . Common A 52-year-old man with a past medical history of hypertension, hyperlipidemia and well-controlled type 2 diabetes with no prior history of gastroparesis presented with nausea, abdominal distension, and pain of one-week duration. The patient initially reported symptoms of early satiety and excessive bloating, leading to nausea and progressive abdominal distension. He then developed more acute, severe epigastric and left upper quadrant pain. Upon arrival at a local emergency department, a nasogastric tube was placed with over 1 liter of fluids suctioned. This provided instant symptom relief but raised concern for gastric outlet obstruction. An abdominal CT scan at the time revealed markedly distended stomach with food/debris and normal caliber duodenum without an obvious lesion  was helpful to exclude a mechanical obstruction. Notably, by withholding further doses of liraglutide, dietary modification, and a brief course of antiemetics and metoclopramide, his symptoms completely resolved.Gastroparesis is a chronic disorder manifested by delayed gastric emptying without evidence of mechanical obstruction. In the United States, approximately 5 million patients suffer from some form of gastroparesis . Nausea In addition to the common etiologies mentioned earlier, rarer causes include certain neurological diseases, amyloidosis, and connective tissue disorders . Many caSymptoms of gastroparesis are nonspecific, as the condition can mimic dyspepsia, peptic ulcer disease, or gastric or small bowel obstruction . In suspThe pathophysiological mechanism of gastroparesis is not fully understood but involves abnormalities in multiple interacting cell types, including the extrinsic nervous system, enteric nervous system, interstitial cells of Cajal, smooth muscles and immune cells . Other cLiraglutide is a long-acting GLP-1 analog that activates the GLP-1 receptor in pancreatic beta cells to stimulate glucose-dependent insulin secretion while reducing inappropriate glucagon secretion from pancreatic alpha cells. Liraglutide also suppresses gastric emptying, leading to decreased food intake as a result of relaxation of the proximal stomach accompanied by increased tonic contraction of the antropyloric region . SubcutaThe management of gastroparesis predominantly focuses on symptom control. Initial therapy involves correction of fluid, electrolyte, and nutritional deficiencies. Once improved, patients are advised on dietary modification, including small frequent meals with limited fat and fiber content , 7. If oDrug-induced gastroparesis, specifically with liraglutide use, should be considered in patients presenting with significant abdominal distension, pain, and nausea once mechanical obstruction has been ruled out. It should be suspected in diabetic patients with recent liraglutide initiation, especially at higher doses. Management includes discontinuation of the offending medication, symptom control with antiemetics and prokinetic agents, nutritional support, and dietary modification.


Slow-growing intraventricular masses are sometimes imaging findings in asymptomatic patients. The neuroimaging characteristics frequently help making the correct diagnosis and the treatment decision. Subependymomas usually present as single lesions poorly vascularized, without invasion into adjacent brain parenchyma or cerebrospinal fluid dissemination. Ependymoma is considered the main differential diagnosis. We report two cases of this tumour who share the unusual location: The temporal horns. The lack of enhancement (or heterogeneous when present) and advanced neuroimaging techniques can be very useful in differentiating them from other lesions. A 59-year-old female with history of incidental finding of brain tumor during examination for labyrinthitis 18 years ago. Patient returned after that time for further investigation. Review of systems: asymptomatic. Past medical history of arterial hypertension and diabetes. No epileptic seizures were associated. There were no neurological deficits or any other significant findings on physical examination or laboratory studies.Head CT revealed an intraventricular expansile mass, which caused a right ventricular temporal horn expansion . Brain MA subtle hyperintensity in the adjacent brain parenchyma was noted in the temporal lobe, that might represent an edematous (or infiltrative) component .The Protons Spectroscopy with short TE demonstrated a decreased N-acetylaspartate (NAA) peak, suggesting neuronal viability loss, and an increased peak of myoinositol (MI), meaning astrogliosis. The Choline (Cho) peak remained normal, since there wasn’t an increase in the capillary density .Thus, a diagnosis of a primary glial low-grade central nervous system neoplasm (subependymoma) was considered as the most likely possibility.The patient insisted on a surgical approach and underwent a right frontotemporosphenoidal craniotomy. The lesion was poorly vascularized, and a complete macroscopic tumor removal was achieved.Post-operative tumor histopathology revealed a paucicellular neuroepithelial neoplasm, with small clusters of glial cells containing isomorphic vesicular nuclei, although varied sizes of nuclei were also present. The tumor cells were loosely arranged in a fibrillary matrix, with sparse microcysts and ependymal lining, typical features of subependymoma .Histological features, such as increased cellularity, nuclear atypia, necrosis and microvascular proliferation were analyzed, as well as immunohistochemistry was performed using antibodies for GFAP, Synaptophysin, S100 and EMA. There was a positive expression for S100 and GFAP. Ki-67 immunohistochemistry was also performed, and the proliferation index was low (Ki67 <1%). The final report considered it a Subependymoma (WHO grade I).The postoperative brain MRI showed complete resection of the intraventricular lesion and there was no evidence of disease at the last follow-up.A 22-year-old female with history of headache with sensation of pressure on the sides and across the forehead, and blurred vision. The headache would persist over a period of several hours. Her past medical history was otherwise unremarkable. No epileptic seizures were associated. She had no sick contacts or recent travel history. Her general and neurologic examinations did not show any neurological deficits. A brain MRI was performed for investigation and revealed the intraventricular lesion in the left temporal horn.T1 weighted imaging  3D Multi voxel spectroscopy demonstrated a NAA peak partially decreased, due to neuronal viability loss and an increased peak of myoinositol, meaning astrogliosis .A brain tumor surgical biopsy was performed in the left temporal region, without any procedural complications .Histopathology revealed a hypocellular neuroepithelial neoplasm, with clustering cells containing rounded vesicular nuclei and delicate chromatin. The tumor cells were dispersed in a loose fibrillary matrix, often arranged in perivascular pseudorosettes. There were no histological signs of malignancy. The features were typical of a subependymoma.The differential diagnosis for intraventricular lesions includes subependymoma, ependymoma, neurocytoma, and giant cell subependymal astrocytoma. When there is microcystic degeneration, the list should contain pilocytic astrocytomas, chordoid gliomas, myxoide meningioma and oligodendrogliomas. Immunohistochemistry may help, when it shows strong immunopositivity for GFAP and S-100 antigens.Here we discuss the three main differential diagnoses, starting with ependymoma. The imaging appearance may be similar, as both lesions are usually intraventricular, and calcification, hemorrhage, and cystic components can be seen, although other findings help in the differentiation process. Subependymomas frequently present in the fifth or sixth decade of life, and although ependymomas may occur at any age, the posterior fossa tumors tend to present more commonly in the pediatric age group. Intense enhancement is observed in ependymomas, as well as extraventricular extension to the brain parenchyma and the cerebrospinal fluid (CSF). Subependymomas may show no or subtle enhancement and there is usually no invasion into the adjacent brain parenchyma  nor CSF dissemination. Curiously, when there is overlap of these imaging features, the lesion tends to have an ependymoma-like clinical course, therefore it is graded on the basis of the ependymoma component. Those cases are referred to as mixed ependymoma–subependymoma, and represent the main problem in the histological diagnosis.1Another differential diagnosis is neurocytoma, which is typically seen in young adults (nearly 70% diagnosed between 20 and 40 years of age), emerging from the septum pellucidum or the ventricular wall, and lateral ventricles involvement is seen in half of the cases, near the foramen of Monro, while 15% are located in both the lateral and third ventricles. They may also contain calcifications and cysts within the lesion, with mild enhancement. Spectroscopy may be helpful, as subependymomas show a large MI peak (produced only by astrocyte cells), not seen in neurocytomas.Finally, giant cell astrocytomas are generally present in the context of a patient with tuberous sclerosis, at a younger age (most cases occurring in the first and second decades), with a mean age of 11 years. Imaging findings include calcifications and the typical location, virtually always located near the foramen of Monro.2–4 The most common locations are the fourth and lateral ventricles.5–7Subependymomas are slow-growing benign neuroepithelial neoplasms (WHO Grade I). They are usually incidental findings in middle-aged patients, during neuroimaging investigation for other reasons. No race or gender predilection is known.7Patients with subependymoma are frequently asymptomatic. When symptomatic, they usually present with hydrocephalus and the lesion is most often greater than 2.0 cm.5,6,8,9 . Recurrence is very rare.


We present a histologically proven mucinous adenocarcinoma of the colon in a 40 year old female from Gulu, Northern Uganda. Her elder sister died at 25 years with advanced adenocarcinoma of colon similarly with her mother who died of the same illness 10 years apart. Using the Amsterdam criteria for the diagnosis of the carcinoma of the colon, this is descriptive of Hereditary Non Polyposis Colorectal Carcinoma (HNPCC). Blood examinations revealed microcytic hypochromic anaemia. The Renal and Liver function parameters were essentially normal. The abdominal ultrasonography showed an ill-defined mass in the right hypochondrial region which was heterogeneous with central echogenicity approximately 7.2cm wide and with no intra-abdominal lymphadenopathy or ascitis. At laparotomy, the sonographic findings were confirmed with a demonstrable mass in the hepatic flexure of the colon with hyperemic areas on its serosa. Macroscopically, there was an annular fungating mass with a central necrosis in the hepatic flexure measuring over 7.0cm. Histology of the colonic tumour showed a mucinous adenocarcinoma of the colon (Duke's B). This finding highlights the occurrence of colonic adenocarcinoma in the young person in Northern Uganda, a finding which draws the attention of the medical community towards having a higher index of suspicion for carcinoma of the colon in patients with similar presentation. There is paucity of literature on the incidence and prevalence of carcinoma of the colon in Northern Uganda. However, according to the Kyadondo Cancer Registry (KCR) in Kampala; its incidence in Uganda is 9.0%  Table 1Table 1 aoC. She had moderate pallor but not jaundiced, she had moderate dehydration, but with no palpable lymphadenopathy.A 40 year old female, a wife to a senior Medical Doctor in Northern Uganda presented to Gulu Regional Referral Hospital Surgical Out-patient Department (SOPD) with a dull aching abdominal pain in the right hypochondrial region which had lasted 7 months and an abdominal mass in the same area for 5 months. The abdominal pain was not colicky in nature but progressive and radiated to epigastric region and relieved only temporarily and partially by some analgesics. She reported passage of dark colored stool and associated progressive weight loss over the period. There was no reported history of vomiting, diarrhoea or passage of mucoid stool and neither were these symptoms associated with abdominal distension, vomiting or early morning diarrhoea but associated with flatulence which she described as of “rotting smell ”. She reported a history that her mother and elder sister passed on due to a similar illness but for the elder sister, she died when she was 25 years old having been diagnosed with an advanced adenocarcinoma of the colon at one of the hospitals in Northern Uganda. Her mother too died due to a similar condition but at the time of her death she was middle aged. She denies any history of smoking cigarettes or drinking alcohol in her entire life but reports that they normally used firewood for cooking since childhood. She had moved to several health facilities in Gulu seeking medical treatment and she was being treated for Peptic Ulcer Disease (PUD) for which she only got mild relieve from the pain and discomfort. At the time of her admission she had a booklet of medical forms which when combined could be measured at about 10cm in thickness. In all, the clinicians continued to diagnose and treat her with Peptic Ulcer Disease (PUD) and continued to prescribe antacids and other proton pump inhibitors for her treatment. The mass was even biopsied under ultrasound guided biopsy in one of the major hospitals in the region for which the histology results showed a non-specific inflammation. On general examination, she was a young woman in fair general condition, moderately wasted, afebrile to touch with an axillary temperature of 363], serum creatinine, blood urea and nitrogen levels) were all within normal ranges  and renal function parameters  of the hepatic flexure of the colon. His elder sister died at 25 years with advanced carcinoma of the colon and the mother too died of carcinoma of the colon. According to the Amsterdam criteria, hereditary non polyposis colorectal carcinoma (HNPCC) is the diagnosis. The need to educate health workers about the increasing incidence and prevalence of colorectal carcinoma calls for higher index of suspicion whenever they are confronted with another of such a patient.The authors declare no competing interests.


Multicystic peritoneal mesothelioma (MCPM) is a particularly rare and benign neoplasm that arises from the peritoneum in reproductive aged females. Its etiopathogenesis is still unclear. The current prevailing theory supports the idea that the tumor is the result of an excessive inflammatory process. Because of a lack of clinical and imaging presentation, the diagnosis is intricate, and heavily relies on case reports and short studies. A histological analysis with immunohistochemistry is required for a definitive diagnosis. To date, there is no standard treatment recommended for MCPM. However, some studies suggest proceeding with a cytoreductive surgery and a hyperthermic intraperitoneal chemotherapy combining CISPLATIN and DOXORUBICIN, due to a high incidence of recurrence rate after medical treatment or surgery alone and potential malignant transformation. Peritoneal mesothelioma is a particularly rare disease characterized by peritoneal malignancy occurring in the mesothelium (lining cells) of the pleural, peritoneal and pericardial cavities, and tunica vaginalis covering the testes. Seven histological types have been identified thus far: low-grade mesothelioma, well differentiated papillary, multicystic, epithelioid, sarcomatoid, biphasic (combining epithelioid and sarcomatoid), and deciduoïd . MulticyAs the pathogenesis is still unclear, current understanding of the disease is relies heavily on case reports and short series. In contrast to malignant peritoneal mesothelioma, the prevalence of the tumor has no association with asbestos exposure. The prevailing theory suggests that MCPM may be a peritoneal reaction secondary to a chronic irritant with mesothelial cell entrapment, reactive proliferation, and cystic formation . ChronicIn addition, some studies suggest that female sex hormones play a role in its pathogenesis. This hypothesis is supported by the fact that MCPM occurs mainly in females of reproductive age. High CA 19.9 serum concentration has been associated with the diagnosis of MCPM . FinallyThe diagnosis is usually difficult to establish based on clinical and imaging data. The majority of patients are typically asymptomatic until the tumor is large enough to cause a mass effect on organs. The most common presenting symptoms are non-specific such as abdominal pain, tenderness, palpable mass, or weigh loss. The lesion shows a strong predilection for the pelvic peritoneum, which may adhere to the rectum, bladder, or uterus. Consequently, the other symptoms may be: dyspareunia, dysuria, intestinal obstruction, urinary symptoms, although these symptoms remain non-specific .The preoperative diagnosis is challenging. Ultrasonography (US) is often the first examination requested in case of abdominal pain, due to its accessibility and the absence of irradiation source. Multiloculated anechoic cysts with liquid content can be observed on US. A “spider-in-web” sign can be described as an ovary encased by cystic mesothelioma of peritoneum .CT scan is used to evaluate the location and the extent of the cystic mass, where lesion appears as a low-density, multi-loculated, and thin-walled multicystic mass . HoweverMRI is considered the best imaging technique. MCPM appear as hypointense lesions on T1 and hyper to intermediate intense on T2, with a mild contract enhancement of the wall  Figure A, B. HowLymphangioma has the most important differential imaging diagnosis, but it occurs mainly in the pediatric population and do not have a gender or regional predilection .However, a definitive diagnosis requires histology as well, and the intraoperative examination is unnecessary since the diagnosis requires immunohistochemistry.Laparoscopy is the most efficient procedure to obtain the definitive diagnosis of MCPM and to analyze peritoneal extension of the disease, using large surgical biopsies. Macroscopically, the tumor is composed of multiple mesothelial and thin-walled translucent cysts ranging from several millimeters to 20 centimeters, arranged in a grape-like form. Cysts are mostly composed of clear serous fluid and occasionally mucinous, gelatinous, or hemorrhagic fluid A, B, C. The differential diagnosis of MCPM includes benign and malignant cystic or multicystic abdominal tumors such as cystic lymphangioma, endometriosis , cystic Immunohistochemistry is a necessary step for the definitive diagnosis of MCPM , cytokerOver the last decade, the most common treatment has consisted of a complete resection of the tumor. However, the recurrence rate after resection is approximately 50% after a period of 3 to 27 months (mean 32 months) . No riskAlternative treatments and observation such as hormonal therapy, sclerotherapy, and potassium-titanyl-phosphate laser vaporization have been proposed in the recent studies. Hormonal therapy with anti-estrogen drugs like tamoxifen  and GnRHDue to the high risk of recurrence rate and potential malignant transformation, some studies propose an aggressive approach using CRS with extended peritonectomy associated with HIPEC. The HIPEC protocol usually combines CISPLATIN and DOXORUBICIN. Baratti et al. published the first multi-institutional study with 12 MCPM patients treated by CRS and HIPEC, and showed no recurrence over a follow-up period of 64 months . In addiThe use of the combined laparoscopy CRS – HIPEC may be a promising alternative approach to treat MCPM. Passot et al. included in their study patients with low-grade PMP or MCPM, and compared 8 patients treated by laparoscopy with 8 patients treated by laparotomy. No recurrence has been reported after a median follow-up of 9, 5 months. This cohort study suggests that laparoscopy CRS-HIPEC should be reserved for selected patients with borderline tumors (like MCPM and low-grade PMP) or PCI  of 10 or less . MoreoveIn summary, MCPM is a rare benign neoplasm that is associated with no well-defined symptoms, typical clinical, or imaging data. Consequently, the diagnosis remains unclear and the definitive diagnosis requires histology and immunohistochemistry. MCPM has a high risk of recurrence and a significant potential of malignant transformation. Hence, the definition of low-grade or borderline tumors seems more appropriate to describe the lesion. Therefore, a combination of CRS and HIPEC in specialized center is the recommended strategy to decrease the risk of recurrence and increase patient’s overall survival.