Over the last 40 years, there has been a significant increase in the incidence of testicular cancer. The epidemiologic evidence to understand this phenomenon is unclear, however exogenous estrogen exposure is thought to be a driver in the development of testicular cancer. This is of particular importance in the transgender population because utilization of exogenous estrogen therapy is an essential aspect of the transition process.We present the case of a 38-year-old Caucasian male to female transgender patient who presented with metastatic testicular cancer 15 months after initiating estrogen therapy. She presented to our emergency department with worsening back pain and fatigue. A clinical examination revealed a right-sided testicular mass. A computed tomography scan of her abdomen/pelvis identified a right groin lesion measuring 6.4 cm, a retroperitoneal mass causing right-sided hydronephrosis, an extensive deep vein thrombosis, and pathologic abdominal lymphadenopathy. Germ cell tumor markers revealed an alpha-fetoprotein of < 2.5 μg/L and a beta-human chorionic gonadotrophin of 2526 IU/L. Her lactate dehydrogenase was 5294 U/L. Medical oncology advised the discontinuation of hormonal therapy at this time. On the basis of elevation in germ cell tumor markers and the burden of disease, she was treated with four cycles of bleomycin, etoposide, and cisplatin chemotherapy. A decision to defer upfront radical inguinal orchiectomy was made due to not wanting to have an early interruption in anticoagulation.Following the completion of the chemotherapy, a 6 cm retroperitoneal mass persisted. Due to the location of the mass and surgical morbidity associated with excision, she was followed with positron emission tomography-computed tomography by Uro-oncology, with no evidence of recurrent disease 2 years since the time of diagnosis.While there are recognized risks associated with estrogen therapy less is known about the extent to which exogenous estrogen can serve as a driver of malignancy. With recent experimental evidence revealing a pro-growth impact of estrogen on human testicular cells, continued reporting of similar cases in the literature is imperative to see if a link between exogenous estrogen exposure and testicular cancer exists. Over the last 40 years, there has been a significant increase in the incidence of testicular cancer (TC). While the epidemiologic evidence to understand this phenomenon is unclear, testicular dysgenesis syndrome (TDS) is a theory that attributes the increased rate of disorders of male fertility  to a common error in gonocyte development that is in part driven by exposure to excess estrogen.This is of particular importance in the male to female transgender population, because utilization of sustained exogenous estrogen therapy is an essential aspect of the transition process. While there are recognized risks associated with estrogen therapy,  less is known about the extent to which exogenous estrogen can serve as a driver of malignancy such as TC.Here we discuss the case of a male to female transgender patient who developed TC after many months of exogenous estrogen therapy, review the biologic plausibility, and review the literature for similar cases for which there is only one case. Continued reporting of similar cases in the literature is imperative to see if a link between exogenous estrogen exposure and TC exists.A previously healthy 38-year-old Caucasian male to female transgender patient was placed on hormonal therapy for 15 months with estradiol 2 spironolactone while awaiting gender re-assignment surgery. There was no other previous past medical or surgical history. She was not taking any other medications and had a non-anaphylactic allergy to penicillins. She is married with two children and is employed as a truck driver. She never uses recreational drugs, has an infrequent alcohol intake (< 1/week), and is an ex-tobacco smoker. There was no family history of TC.9/L (absolute neutrophil count of 8.9), platelets of 446 × 109/L, sodium 130 mmol/L, potassium 4.2 mmol/l, creatinine 127 umol/L, with remaining extended electrolytes  and liver panel within normal limits. At the time of diagnosis, lactate dehydrogenase (LDH) was 5294 U/L and the tumor markers alpha-fetoprotein (AFP) and beta-human chorionic gonadotrophin (β-HCG) were < 2.5 μg/L and 2526 IU/L, respectively. Overall her clinical stage was IIIC .In early 2016, she developed progressive right-sided scrotal swelling, abdominal and back pain, fatigue, and weight loss of 7 kg. With ongoing pain, she presented to our Emergency Department in June 2016. On presentation her vital signs were 36.1 °C, heart rate (HR) 99 beats per minute, blood pressure (BP) 169/97, and 99% on room air with 18 respirations a minute. A clinical examination revealed no focal neurological deficits, normal cardiorespiratory examinations, palpable adenopathy in the left supraclavicular fossa but no palpable axillary or inguinal adenopathy. Her abdomen was soft and non-tender, there was no organomegaly, and a genitourinary examination revealed a large right-sided testicular mass. A computed tomography (CT) scan of her abdomen/pelvis identified a right groin lesion measuring 6.4 × 4.9 × 6.3 cm, and a retroperitoneal mass (11.5 × 10.6 × 17.4 cm) causing right-sided hydronephrosis see Fig. . FurtherShe was seen by the Thrombosis and Interventional Radiology services; she was started on dalteparin for anticoagulation, and a nephrostomy tube was inserted on the right side.While it would be standard practice to pursue a radical orchiectomy prior to the initiation of chemotherapy this was deferred in this case for a variety of reasons. First, it was felt that an early initiation of chemotherapy was indicated to reduce the high burden of disease as she was highly symptomatic and unstable. Second, given the extensive clot burden, there were concerns with the early interruption of anticoagulation for a surgical procedure. Last, although the left-sided neck disease could have been biopsied for diagnosis, it was felt to be too risky given its location relative to vascular structures.Immediately following this, Medical Oncology initiated treatment with chemotherapy using bleomycin, etoposide, and cisplatin (BEP) and advised our patient to discontinue hormonal therapy.Given the overall burden of disease, a decision was made to pursue four cycles of chemotherapy with BEP which was given between June 2016 and October 2016. Tumor markers following the completion of treatment were AFP 2.7 μg/L, β-HCG 2.4 IU/L, and LDH 247 U/L.She then underwent a radical right inguinal orchiectomy in October 2016 (preoperatively she received 2 grams of cefazolin intravenously) with pathology revealing scarring related to treatment effect but no evidence of malignancy. Re-staging CT scans revealed a residual retroperitoneal mass measuring 6.1 cm × 2.4 cm and persistent thrombosis of the inferior vena cava IVC; Fig .With a residual mass, she was referred to Uro-oncology in November 2016 for consideration of a retroperitoneal lymph node dissection. Ultimately, the decision was made to follow this residual mass with serial positron emission tomography (PET) and CT scans. The basis for this was twofold. First, the elevated β-HCG, the normal AFP, and overall clinical picture suggested that this was a seminoma, and second, the location of the lesion would result in a surgical risk and morbidity that would outweigh any potential benefit. Since then routine scans have been completed every 3 months with no evidence of disease recurrence.In November 2016, following the completion of chemotherapy, because of the persistence of thrombosis on imaging, and our patient’s wish to restart therapy with estrogen  she was switched from dalteparin to rivaroxaban 20 mg orally once a day. She was counseled on the risk of thrombosis with the resumption of exogenous estrogen therapy, and there was a discussion surrounding the experimental evidence suggesting a possible link between estrogens and TC.As of June 2018, she has shown no evidence of recurrence; her AFP and β-HCG remain within normal limits. She undertook orchiectomy of the remaining testicle in August 2017, which showed no evidence of malignancy or other significant pathology. She continues with indefinite anticoagulation given her ongoing use of exogenous estrogen.In summary, we have presented a case of a previously healthy 38-year-old male to female transgendered patient who, after being on exogenous estrogen therapy for 15 months, presented with a stage IIIC germ cell tumor. She was treated with BEP × four cycles, after which she underwent a radical inguinal orchiectomy which revealed no evidence of residual malignancy. Restaging CT scans following the completion of chemotherapy revealed a 6 cm retroperitoneal mass which was not amenable to surgical resection. As such, she was followed with serial imaging with PET/CT scans every 3 months. It is now over 2 years since the time of diagnosis, and she has no evidence of disease recurrence.TC is the most common malignancy in men aged 15–40. Over 95% are germ cell tumors, of which the main classification is seminoma and non-seminoma [Patients with TC will often present with a mass or lump in the testis. Signs and symptoms suggestive of loco-regional/metastatic disease include swelling of the lower extremities, back pain, dyspnea, or hemoptysis.In patients where TC is suspected, baseline investigations should include routine bloodwork with AFP, β-HCG, and LDH. Imaging studies should include a scrotal ultrasound and CT of the chest/abdomen/pelvis . Additional investigations such as a bone scan or CT scan of the head are indicated if there are symptoms suggestive of disease in these locations.On completion of these investigations, patients should be assessed for consideration of orchiectomy . OrchiecIn patients with localized disease, tumor markers should decline as predicted by their half-lives .In the metastatic setting, defining the tumor as seminoma versus non-seminoma, the level of tumor markers, and the sites of metastases helps to stratify risk and define the approach to therapy  may remain following chemotherapy. While a “residual mass” has not been defined in terms of size, consensus guidelines do recommend surgical resection of amenable masses > 1 cm. In almost 50% of the cases, these resected masses demonstrated post treatment necrosis while 10% demonstrated viable cancer cells. While there is no reliable way to predict the likely pathology of these residual masses, favorable factors include absence of teratoma in the primary tumor, large shrinkage of the mass with chemotherapy, and residual size < 1 cm .In metastatic seminomas, residual masses are classified as > 3 cm or < 3 cm. For lesions greater than or equal to 3 cm, fluorodeoxyglucose (FDG)-PET should be obtained for further assessment, while for masses < 3 cm, PET scans are optional. If the PET scan is positive, the treatment of choice would be surgical resection. Radiation therapy may also be considered, although the benefit of this would be less clear . In our in situ after birth. These cells remain dormant until there is a change in their local microenvironment, attributable to changes in (1) androgen levels, (2) excessive estrogens, or (3) exogenous hormonal imbalance [Over the course of the last few decades, the overall incidence of TC has increased and has doubled over the last 40 years in many regions in the world . One thembalance . These cWith this in mind, a 1987 study that assessed the histology of testicles in male to female transgendered patients following exposure to exogenous estrogens found atrophy of the seminiferous tubules, cellular hypertrophy, and hyperplasia of the rete testis, and to a lesser extent of the ductuli efferentes, and of the epididymal ductal epithelium [Furthermore, a recent study has demonstrated a pro-growth impact of estrogens on human testicular germ cells, mediated through the activation of an extracellular regulated kinase and protein kinase A .Our review of the literature reveals one other case of a male to female transgendered patient who, after being on estrogen therapy for 22 years, developed TC that required surgery and four cycles of BEP chemotherapy .Continued reporting of similar cases is imperative to understand any potential putative link between exogenous estrogen exposure and the development of TC. While there is a paucity of data to support or refute this suggestion, completing a screening scrotal ultrasound in male to female transgendered patients prior to being placed on estrogen therapy may be useful and is non-invasive and inexpensive. If a patient is planned to undergo prolonged hormonal therapy, regular interval ultrasounds  could be considered.To the best of our knowledge, this is only the second case in the literature whereby a male to female transgender patient developed TC while on exogenous estrogen therapy. While it is not possible to determine if estrogen is exerting a carcinogenic effect in this setting, continued reporting of similar cases is imperative to assess for this possibility.


Primary leiomyosarcoma (LMS) of vascular origin is a rare lesion, and patients with LMS of vascular origin have poorer prognoses than patients with LMS of other origins. The inferior vena cava is the most commonly affected vessel and accounts for 60% of all vascular cases. However, LMS originating from the ovarian vein is extremely rare, and we are only aware of 15 reported cases. Therefore, we report our experience with a case of LMS originating from the right ovarian vein and review the related literature.18F-fluorodeoxyglucose positron emission tomography. Based on these findings, the differential diagnosis included a retroperitoneal tumor, such as a desmoid tumor, leiomyoma, LMS, and malignant mesothelioma. Operative findings confirmed that the mass had originated from the right ovarian vessels, and en bloc excision was performed for the mass and the right ovarian vessels. The final pathological diagnosis was LMS originating from the right ovarian vein, and the surgical resection margins were free from tumor cells. After histological findings confirmed the LMS diagnosis, the patient underwent adjuvant radiation therapy and has not exhibited signs of local recurrence or metastasis in the 6 months after surgery.A 71-year-old Japanese woman with no symptoms was admitted to our hospital because of abnormal findings in a routine abdominal ultrasonography check-up. Contrast-enhanced computed tomography of the abdomen revealed a well-defined, lobulated solid mass with a diameter of 5.5 cm in the right retroperitoneal space. The mass exhibited relatively low uptake during We encountered a 71-year-old woman with LMS originating from her right ovarian vein. The prognosis of vascular LMS is generally poor. Therefore, careful follow-up will be required for our patient. Leiomyosarcoma (LMS) is a mesenchymal tumor of smooth muscle origin. However, primary LMS of vascular origin is a rare lesion that represents < 1/100,000 of malignant tumors. Patients with LMS of vascular origin have poorer prognoses than patients with LMS of other origins , followe18F-fluorodeoxyglucose positron emission tomography (FDG-PET), the mass had relatively low uptake, with a maximum standardized uptake value (SUVmax) of 2.4  of the abdomen revealed no abnormal findings in the pancreas and gallbladder but revealed a well-defined, lobulated solid mass with a diameter of 5.5 cm in the right retroperitoneal space Fig. a, and on2.4 Fig. . Based oExploratory laparotomy was performed through a median incision, and the operative findings confirmed that the mass was attached and connected to the right ovarian vessels. The mass was slightly adhered to the ureter, but there were no signs of invasion to the other adjacent organs. Because the mass appeared to originate from the right ovarian vessels, en bloc excision of the mass and right ovarian vessels was performed. The surgical specimen consisted of a white solid tumor 5.5 cm × 4.5 cm)  and 9 (60%) cases, respectively.Three major growth patterns have been reported for retroperitoneal LMS: completely extravascular , completely intravascular , and a combination of extraluminal and intraluminal patterns  . Cases oBased on these presentations, contrast-enhanced CT is especially useful for establishing the preoperative diagnosis and for determining the tumor extent and boundaries before starting surgery . PreviouIn retroperitoneal sarcoma cases, 60% of resected adjacent organs were found to be microscopically invaded by the tumor . En blocMetastasis is identified at diagnosis in 12% of cases of vascular LMS that did not originate from the vena cava , althougIn the present case, careful follow-up will be required, despite the low mitotic rate of 10 mitoses/10 HPF, which would predict a good outcome based on the previous sentence.We encountered a 71-year-old woman with rare vascular LMS originating from the right ovarian vein. Although the prognosis of LMS originating from the ovarian vein is obscured by its rarity and short-term observation data, the prognosis of vascular LMS is generally poor. Therefore, careful follow-up will be required for our patient.


However metachronous carcinomas affecting small bowel are rarer.•Small Bowel adenocarcinoma (SBA) is a rare malignant neoplasm without specific signs and symptoms, and associated with late stage disease presentations.•Patients who develop a small or large bowel adenocarcinoma are at high risk for a second cancer at both sites. However data available to guide therapeutic decisions for those presenting one at small bowel are scarce, and the role of adjuvant therapy in patients who undergo curative resection is unclear.•Studies about strategies for detection at an earlier stage, optimal treatment and prognosis are mandatory for this disease. Small Bowel adenocarcinoma (SBA) is a rare malignant neoplasm without specific signs or symptoms. It’s been associated with late stage disease presentations. Midterm outcomes have suggested that after successful removal of colorectal carcinoma, there is higher risk for developing a further primary (metachronous) colorectal tumor. However when it comes to small bowel, metachronous carcinomas are unusual.A 46-year-old female who underwent an emergency Hartmann's procedure two years previously and treatment of adjuvant chemotherapy for adenocarcinoma of the sigmoid colon at stage IIB with loco-regional recurrence.The patient presented with bowel obstruction secondary to a small bowel intussusception, confirmed by computed tomography. An emergent exploratory laparotomy was performed and confirmed of an ileal tumor as the cause of small bowel intussusception and clinical bowel obstruction. Histopathology confirmed a primary small bowel mucinous adenocarcinoma with node metastasis .Patients who develop a small or large bowel adenocarcinoma have high risk of presenting a second tumor at both sites. Since data available to guide therapeutic decisions for patients presenting with small bowel metachronic tumors are scarce, the role of adjuvant therapy in patients who undergo curative resection remains unclear.The aim of this manuscript is present a case report of a patient admitted for a rare obstructive small bowel intussusception therefore underwent surgery for metachronic small bowel carcinoma from colorectal primary witch previously treated by surgery and adjuvant chemiotherapy. Studies about strategies for detection at an earlier stage, optimal treatment and prognosis are mandatory for this disease. AdenocaAfter successful colorectal cancer removal, there is high risk of developing a further primary (metachronous) large bowel tumor . However246-year-old female with past surgical history of Hartmann’s procedure on 2017 for abscessed and obstructive adenocarcinoma of the sigmoid. Histopathological study confirmed low-grade mucinous adenocarcinoma of sigmoid colon with involvement of all layers and perforation of the visceral peritoneum, pT4pN0. K-RAS gene mutation was present thus chemotherapy with capecitabine was completed. Hartmanns’s reversal procedure was performed months later.Thereafter 18-months-postoperative follow up appointment, computed tomography (CT) revealed iliac lymphatic recurrence. FOLFOX and FOLFIRI-aflibercept chemotherapy were provided. Abdominal CT control confirmed the persistence of lymphatic disease and target sign in small bowel suggesting intussusception as well.Forty-eight hours later, the patient presented to emergency department complaining of abdominal pain and distension, lack of elimination of flatulence and vomiting. An abdominal X-ray showed dilated bowel loops with air-fluid levels. Blood test revealed normal white cells level, and a serum-C-reactive protein of 206 mg/L. Abdominal CT evidenced a complete bowel obstruction secondary to small bowel intussusception . The lapAfter eight months on follow up, the patient had an elevation of tumor markers level. Abdominal CT showed left iliac lymph node disease, and PET Scan settled extra focus at retroperitoneum. The patient is about to start additional chemotherapy treatment.3Since SBA are exceptional tumors, the current evidence available remains hesitant. Therefore the specific clinical characteristics, the best treatment modalities and prognosis are not clear .Most of the adenomas in the small bowel have origin on duodenum, with malignancy incidence reported of 42%. Villous histopathology, growing size and higher grade of dysplasia increase the risk of malignancy . Some auOn clinical presentation, the most common symptoms of SBA are: abdominal pain, weight loss, nausea, vomiting, and gastrointestinal bleeding. Small bowel Intussusception is exceptionally in adults, with 90% of cases secondary to organic disease. Abdominal CT findings such as ‘target’ or ‘doughnut’ sign, may indicate the presence of small bowel intussusception, as previous case.SBA diagnosis can be assessed by enteroclysis in 90% of cases, whereas only 33% of them could be identified with barium follow-through study ,3. The oSBA staging is performed according to the American Joint Committee on Cancer (AJCC) guidelines, which is based on the TNM staging system . The maiSeveral factors have been associated with poor prognosis, the most important are late staging, lymph node involvement, poor histopathology differentiation, old age, duodenal primary, and positive margins ,3. Howev4Metachronous carcinomas affecting both large or small bowel are a very unusual condition. Poor prognosis is related to elderly patients in contrast to our young female patient, who despite her young age, developed colon carcinoma and later on, another primary SBA. We highlight the patient's inadequate response to chemotherapy treatment with a second gastrointestinal carcinoma and evolution to intussusception and finally obstruction, besides treatment. We consider our case an exceptional presentation that represents a challenge for any surgeon on diagnosis, treatment and follow-up of patients with GI adenocarcinoma.We declare no conflicts of interest.We have no any source of funding to declare.We obtained the patient’s informed consent besides our case report is exempt from ethnical approval in our institution.The authors have obtained the written and signed consent to publish the case.Author 1 GARANTOR - patient care, study concept or design, data collection, data analysis or interpretation, writing the paper, Images and revision.Author 2- patient care, study concept or design, data collection, data analysis, writing the paper, Images and revision.Author 3- patient care, study concept or design, data collection, data analysis, writing the paper, Images and revision.Author 4- patient care, study concept or design, data collection, data analysis, writing the paper, Images and revision.Author 5- patient care, study concept or design, data collection, data analysis, writing the paper, Images and revision.Author 6- patient care, study concept or design, data collection, data analysis, writing the paper, Images and revision.Author 7- patient care, study concept or design, data collection, data analysis, writing the paper, Images and revision.N/A.Yuhamy Curbelo-Peña, MD.Not commissioned, externally peer-reviewed.


We present a unique case of simultaneous rupture of the bladder and left renal pelvis after blunt trauma to the lower abdomen. To the best of our knowledge, this has not yet been reported in the literature. Another unusual aspect of this case was that the bladder rupture was bilateral, with both an extra- and intraperitoneal component. The management of this case was challenging. This involved an emergency laparotomy to repair the bladder tear, followed by a nephrostomy. This was followed by left ureteral stent insertion using a rendezvous technique. The case also highlights the role of expectant conservative management relating to the concurrent left renal pelvic rupture. He presented with abdominal pain and an inability to pass urine. He had no other significant past medical history apart from recently diagnosed hypertension.He was hemodynamically stable. His abdomen was distended and the lower abdomen was tender.9/L, hemoglobin of 16.1 gm/dL, and creatinine of 464 mmol/L . A noncontrast CT scan revealed a large collection of fluid in the peritoneal cavity. The bladder was grossly distended with evidence of extraperitoneal fluid  was inserted under ultrasonography guidance. The patient recovered well with good urine output from the nephrostomy and normalization of his renal function. A nephrostogram revealed a grossly dilated pelvicaliceal collecting system with large retroperitoneal extravasation of contrast. The ureter was not delineated on this study . A few dAn elective rendezvous procedure was planned after 4 weeks to allow the collection and inflammation to settle. A repeat CT confirmed reduction in the size of the retroperitoneal collection. During the rendezvous procedure, a Terumo hydrophilic guidewire was passed down to the bladder through an antegrade approach into the bladder and a stent (6F/26 cm) was secured in the left ureter . The ureReassessment after 4 weeks with a CT urogram showed satisfactory healing of both the bladder and upper tract injuries with no evidence of leak. This was confirmed on cystogram and retrograde study . The DouThis case highlights how significant blunt force to a chronically distended bladder in the presence of “high-pressure” chronic retention (and thus a dilated ureter and collecting system) can result in both concurrent lower urinary tract and upper urinary tract rupture.The nature of the injury involved the patient falling forward onto their chronically distended bladder, which took the impact of this fall. This resulted in bilateral bladder wall tears with simultaneous left pelvicaliceal rupture (“pop off phenomenon”) because of sideways transmission of force to the lateral walls of the bladder and also a transmission of this force up through the standing column of urine in the left hydroureter to the distended left renal pelvis .Although injury to the lower urinary tract required laparotomy and surgical repair, the upper tract injury in this case seems to have settled with a period of conservative management after insertion of a nephrostomy tube and subsequent rendezvous ureteral stent insertion.


Osteolipomas are uncommon variants of lipoma. These lesions have been usually reported to arise from the oral cavity, brain, and neck and scarcely from the knee and thigh. Intraspinal osteolipomas are rare. A single case of intraspinal osteolipoma has been reported in the cervical and thoracic spine in the literature. To our knowledge, there is no report of osteolipomas in the lumbar spine.  We report a very rare case of a solitary lumbar intraspinal osteolipoma, presenting as a cauda equina syndrome. The intraspinal osteolipoma was excised en bloc and fusion was performed as it required partial resection of the facet joint within 24 hours of presentation. He has since then improved neurologically, and there has been no recurrence of the lesion so far. The clinical presentation, radiological characteristics, treatment course, and histopathological features of this lesion along with the clinical outcomes and a pertinent literature review were done and have been discussed.  The heterogeneous signal intensity of the lesion in MRI differentiates it from other dural-based lesions, and this should raise suspicion of an osteolipoma, which warrants a CT. Although intraspinal osteolipomas are benign lesions and generally have good prognosis, they need to be removed en bloc as they may result in rapid neurological deterioration. Lipomas are the most common benign mesenchymal tumors . OsteoliA 36-year-old male presented to the outpatient department with a history of low backache for the past one year, associated with typical claudication symptoms, left-sided unilateral numbness, and paresthesia of the foot. He gave no history of radicular pain, but he complained of progressive reduction in claudication distance. He had experienced a sudden deterioration in gait along with urinary incontinence, following a trivial traumatic fall, one week before presentation. There was no history of constitutional features. On examination, he had a bilateral high-stepping gait due to foot drop. Symmetrical weakness of both the L4 and L5 roots (MRC grade 0/5) and partial weakness of the S1 root (MRC grade 3/5) was noted with nondermatomal sensory disturbances. Deep tendon reflexes of the lower limbs were absent bilaterally. Though anal tone was normal, saddle anesthesia was present. Postvoidal ultrasonogram of the urinary bladder revealed a residual urine volume of 250 ml, thus confirming a neurogenic bladder.Plain radiography of the lumbar spine did not show any gross feature of instability and was inconclusive . MagnetiAt 4 weeks, there was an improvement in his urinary symptoms, and by 12 weeks partial neurological recovery (MRC grade 3/5 motor power) in bilateral L4 and L5 was observed. The S1 root power increased by one grade (MRC grade 4/5). His gait improved thereafter, and he was able to return to his normal activities by 6 months with further neurological improvement by 1 grade in all roots.The gross specimen measured 2 cm × 1.5 cm × 1.5 cm. It was greyish white in color and firm to hard in consistency . It had Lipomas are ubiquitous and are the most common mesenchymal tumors comprising mature adipose tissue. Occasionally, lipomas may contain other types of mesenchymal tissue and such variants are named accordingly as fibrolipoma (connective tissue), angiolipoma (blood vessels), chondrolipoma (cartilage), and osteolipoma (bone). Osteolipomas are extremely rare variants and constitute only 1% of all lipomas; they are alternatively referred to as ossifying lipomas or osseous lipomas .Most of the reports in literature reveal their existence in oral cavities, head, brain, knee, and thigh, and there are only countable reports of osteolipoma related to the spine –9. TherePlain radiographs fail to reveal the lesion. MRI would demonstrate a mass occupying the spinal canal. A clarity of thought is essential in evaluating these patients, to differentiate it from the more common dural-based tumors. Heterogeneous signals within the lesion should raise the suspicion of the presence of both fat and bone in the lesion . CalcifyThis case represents the first report of an intraspinal osteolipoma of the lumbar spine presenting as a cauda equina syndrome. The characteristic signal intensity variations in different MRI sequences and the presence of osseous tissue in CT provide excellent aid in diagnosis. The histopathological presence of osteoid and mature adipose tissue confirms the diagnosis of benign osteolipomas and are best managed surgically by complete resection of the mass and usually carry good prognosis.


Sacral osteochondromas are rare tumors, and a handful of cases have been reported in the literature. The clinical manifestations of sacral osteochondral may vary from a painless mass to a complete neurological deficit. We report a case of sacral osteochondroma arising from S2-3 lamina causing difficulty in lying down in the supine position and sitting. Computed tomographic (CT) scan and magnetic resonance imaging (MRI) delineated the extent of the lesion and confirmed it to be a benign tumor. It was excised en bloc through a posterior midline approach. At two years follow-up, the patient was asymptomatic and the radiograph did not show any evidence of recurrence. To the best of our knowledge, this is the second case report on sacral osteochondroma, which caused postural difficulty in a young female. Bony growth in the sacrum can be either benign or malignant or may be a metastatic lesion. Because of the inaccessibility and proximity to the major neurovascular structures, surgical excision is often difficult. Approximately 10% of the benign tumors or pseudotumors are seen in the sacrum, and the common benign tumors are giant cell tumors 60%), aneurysmal bone cysts 4%), and osteoblastomas -4. Few c%, and os0%, aneurA 19-year-old young female presented with a painless, palpable bony swelling over her lower back region  were tabulated to gather information on sacral osteochondroma (Table Although X-ray is the primary modality of imaging, it may not truly reveal the extent of the lesion in sacral tumors. CT scan clearly delineates the lesion. MRI must be performed to look for cartilaginous cap thickness of the lesion and for malignant transformation. In case of neurological involvement, MRI will reveal the nerve impingement or canal compromise ,11.Surgical treatment of sacral osteochondroma is challenging due to its proximity to the sacral plexus, major blood vessels, pelvic organs, and sacroiliac joint. The excision of osteochondroma protruding into the spinal canal or sacral hiatus needs meticulous dissection as these nerve roots are mainly responsible for genitourinary and bowel/bladder function . En blocSolitary sacral osteochondromas with posterior growth may remain asymptomatic for a long time. Such tumors may attain a large size producing a mass effect on the overlying skin, thus making it difficult for the patient to lie down in the supine position. En bloc excision of the tumor provides immediate relief and less chances of recurrence.


Abdominal actinomycosis is a rare clinical entity and it is difficult to make the correct preoperative diagnosis because of its nonspecific clinical symptoms and varied radiological findings. The diagnosis is usually made after the patient undergoes an operation and tissue is available for pathological evaluation. When the diagnosis is made, the patient should be treated with the appropriate long-term antibiotics.A 69-year-old male patient was transferred to our hospital complaining of a palpable mass, painful abdominal discomfort, weight loss, and night sweating. After colonoscopy and radiologic studies, our presumptive diagnosis was intestinal lymphoma combined with ileocecal intussusception. He was found to have abdominal actinomycosis after surgery and successfully treated with ampicillin for six months.Pre-operative radiological imaging in abdominal actinomycosis is unlikely to allow a definitive diagnosis, but CT scanning is the single most useful imaging modality. Although we performed preoperative radiological studies, including CT, none led to a diagnosis of abdominal actinomycosis, we mistakenly considered the case as intestinal lymphoma combined with ileocecal intussusception.Physicians should include abdominal actinomycosis in the differential diagnosis when an abdominal mass presents an irregular, infiltrative growth pattern, even though ileocecal intussusception is an extremely rare presentation in abdominal actinomycosis. These organisms are not regarded as virulent pathogens but as opportunistic ones in humans, as they are normally present in healthy individuals. They become pathogens only in the presence of damaged or necrotic tissue; Actinomyces does not penetrate the normal mucosa and requires injury to cause disease [Abdominal actinomycosis is a chronic suppurative infectious disease caused by gram-positive  disease ,2.Abdominal actinomycosis is rare and its variation makes diagnosis difficult. Computed Tomography (CT) shows a mass with an extensive infiltration pattern that closely resembles a complicated gastrointestinal malignancy. Also, the pattern of spread through contiguous tissues is not unlike inflammatory diseases . TherefoThis work has been reported in line with the SCARE criteria .23, normocytic normochromic anemia of 9.1 g/dl, elevated CRP to 15.98 mg/dl, elevated ESR to 120 mm/h, elevated serum creatinine and BUN to 2.9 mg/dl and 27 mg/dl each. Serum levels of AFP, CEA, and CA-19-9 were normal.A 69-year-old male patient was transferred to our hospital because of the palpable mass in the right lower quadrant of the abdomen that had been present for one month. He had been well until three months previously when he began to suffer from a poor oral intake, painful abdominal discomfort, general weakness, weight loss, and night sweating. Around 30 and 35 years before the current admission, the patient had been admitted for renal disease of unknown etiology and appendectomy for acute appendicitis, respectively. The body temperature was 36.8 °C. On physical examination, the patient appeared relatively well. A transverse operative wound for the previous appendectomy was identified in the right lower quadrant of the abdomen, and an adult-fist-sized mass was palpated without peritoneal irritation. Laboratory examination revealed mild leukocytosis of 14,000/mmAbdominal ultrasonography showed results that could not exclude distal ileal inflammatory disease or lymphoma a. A contWith a presumptive diagnosis of intestinal lymphoma, an exploratory laparotomy was performed. A large mass was located in the right lower abdomen, and it adhered densely to the retroperitoneum and the bladder. The mass including the right colon was mobilized from the retroperitoneum, avoiding injury of the right ureter. Multiple intraoperative pathological examinations of the mass and adhered retrocecal lesions by frozen-sections revealed chronic inflammation without any evidence of malignancy in the morphology. We performed the right hemicolectomy with drainage. The permanent pathological examination confirmed ileocecal actinomycosis in which the characteristic finding of sulfur granules in the resected specimen was demonstrated . After s3Actinomyces israelii is the most common organism in human actinomycosis. Actinomyces was initially thought to be a fungus but is now known to be a filamentous, non-spore-forming, gram-positive bacillus which is microaerophilic or obligate anaerobe. The general growth pattern is the direct extension of an existing lesion regardless of natural anatomic barriers, but hematogenous or lymphatic spread, although rare, can occur if organisms penetrate the damaged mucosa . The patPossible predisposing factors of disease progression include trauma, previous surgery, appendicitis, bowel perforation, foreign bodies including intrauterine devices (IUD), or neoplasia . Among tPreoperative diagnosis of abdominal actinomycosis is rarely considered and is accurately established in less than 10% of cases. It is generally misconceived as a neoplasm, inflammatory bowel disease, diverticulitis, or tuberculosis both clinically and radiologically because of its characteristic infiltrative growth pattern . There aA. israelii in culture or demonstration of the specific “sulfur granules” microscopically. Unfortunately, the actinomycotic granules are found only in 50% of cases and routine bacteriological cultures of anaerobic organisms are negative for A. israelii [Definitive diagnosis of actinomycosis requires either isolation of the israelii . Althougisraelii ,10,14,15israelii ,4.Increasing evidence suggests that medical therapy alone is usually sufficient to cure actinomycosis, including extensive invasive disease, if an accurate diagnosis is made before surgery . Further4As described, the authors experienced an interesting case of abdominal actinomycosis combined with ileocecal intussusception. Ileocecal intussusception is an extremely rare presentation in abdominal actinomycosis. Nonetheless, physicians should include abdominal actinomycosis in the differential diagnosis when an abdominal mass presents an irregular, infiltrative growth pattern.The authors declare that they have no conflict of interest.None.No research ethics approval was necessary for this case report.Written informed consent was obtained from the patient for publication of this case report and accompanying figures.Young-Hoon Roh, Kyung-Do Byun, Mee-Sook Roh, Hong-Jo Choi: Data collection, writing original draft.Ki-Jae Park: study concept, methodology, writing reviewing and editing, advised and designed the report.All authors read and approved the final manuscript.N/A.Ki-Jae Park.Not commissioned, externally peer reviewed.


We herein report a unique case of sarcoidosis in a 44-year-old Caucasian female. The patient was initially evaluated for hematochezia and diagnosed with diverticulitis after computed tomography (CT) scan of the abdomen and pelvis. This imaging also incidentally showed a large mass abutting the esophagus. Further imaging of the mass revealed widespread lymphadenopathy in the thorax without any respiratory or classic B symptoms present. The remaining workup caused concern for possible lung or hematologic malignancy. Following biopsy of a thoracic lymph node, the patient was diagnosed with sarcoidosis. This patient met only one of the usual demographic criteria associated with sarcoidosis and none of the lab criteria. While sarcoidosis is typically a benign disease, this case exemplifies how it may appear as a more sinister entity and warrants extensive workup to rule out malignancy. Lymphadenopathy attributed to lymphoma, sarcoidosis, or metastatic lung cancer is the most common lesion to be found in the middle mediastinum. The highest incidence of sarcoidosis occurs in African Americans women between the ages of 30 and 39. For Caucasians, the incidence peaks from 40 to 49 years of age in both males and females . The higA 44-year-old Caucasian female with a significant past medical history of diverticulitis, hypertension, obesity, and tobacco use presented for evaluation of hematochezia and left lower quadrant abdominal pain for the past eight hours. A computed tomography (CT) of the abdomen and pelvis was obtained which showed acute diverticulitis along with an incidental right paraesophageal mass abutting the left atrium which was new from prior studies Figure . A transthoracic echocardiogram (TTE) was ordered to evaluate for possible cardiac compromise from the mass encroaching on the left atrium. The TTE showed a normal left ventricular ejection fraction of >65% with no compromise in the left atrial filling Figures -2B. A follow-up CT of the chest with contrast showed widespread mediastinal lymphadenopathy and bilateral hilar adenopathy concerning for malignancy, lymphoma, or sarcoidosis Figure . At this time a pulmonologist was consulted who recommended an excisional lymph node biopsy. CT surgery was consulted and performed the excisional biopsy via mediastinoscopy. The pathology exhibited effacement of the nodal architecture by multiple small granulomas with minimal necrosis or caseation indicative of sarcoidosis. Acid-fast bacillus and periodic acid-Schiff sections were negative for organisms and no polarizable foreign material was visualized. Hematology/Oncology was consulted and they recommended an outpatient positron emission tomography (PET) scan and bone marrow biopsy to evaluate for metastasis. The patient’s beta 2- microglobulin were elevated suggesting possible lymphoma. The patient denies weight loss, fevers, night sweats, chills, cough, chest pain, and shortness of breath. Her hematochezia resolved during the first day of her hospitalization. An esophagogastroduodenoscopy was performed which did not show any signs of active bleeding. Due to her acute diverticulitis, it was recommended that this patient should follow up with outpatient gastroenterology for colonoscopic evaluation in 4-6 weeks.This case illustrates how incidental findings are important to evaluate especially if there is concern for malignancy or compromised cardiac function. This patient was completely asymptomatic, not showing any of the typical B symptoms attributed to lymphoma. Evaluating the cardiac function of the patient with TTE was also vitally important as this mass was abutting the left atrium. If this mass had continued to grow, it could have eventually compromised left atrial filling leading to pulmonary edema and left-sided heart failure. Similar cases have been reported in the literature with initial concerns for malignancy later proving to be sarcoid in origin.One such case of a 65-year-old female presenting with dyspnea, fatigue, and weight loss was evaluated and determined to have probable lung malignancy based on lab work, imaging, and endoscopic evaluation . It was Another case that further demonstrates how our case does not meet any of the usual sarcoidosis demographic and lab criteria is of a 53-year-old female of African-Caribbean descent who presented with a chief complaint of vomiting and constipation with additional low mood and weight loss . InitialThis case illustrates the importance of thoroughly evaluating incidental findings. This patient had an atypical presentation of sarcoidosis that was only identified by imaging that was done for a different complaint that the patient was presenting with. By further evaluating these incidental findings, we were able to make the diagnosis of sarcoidosis. The patient did not present with the typical symptoms of sarcoidosis making the case that much more challenging in identifying the cause of widespread mediastinal lymphadenopathy as well as the mass that was abutting her left atrium.


Cancer is one of the most important leading cause of death in man and woman in the world. The occurrence of new cancer has become more frequent in recent years due to strict screening protocols and occupational and environmental exposure to carcinogens. The incidence of secondary malignancies has also increased due to close medical follow-up and advanced age. Herein, we report a case and its management diagnosed as synchronous peritoneal malignant mesothelioma and muscle-invasive urothelial carcinoma. A 71-year-old male presented with macroscopic hematuria and abdominal distension increasing gradually. A contrast enhanced computerized tomography demonstrated bladder mass and diffuse ascites with nodular peritoneal thickening and umbilical mass. He was treated with the multidisciplinary team working including urologist, medical oncologist and general surgeon. To our knowledge, this is the first case of peritoneal malign mesothelioma with synchronous muscle-invasive urothelial carcinoma. Because of the rarity of this condition, there is still no consensus on the definitive treatment protocols, yet. Individualized treatment with multidisciplinary close follow-up might improve the survival outcomes. On tectively . Transitectively . AdvanceA seventy-one-year-old male patient without any history of asbestos exposure has admitted to our hospital with complaints of macroscopic hematuria and abdominal distension increasing gradually. In his medical history, he underwent transurethral resection of the prostate due to benign enlargement 5 years ago. Physical examination revealed abdominal distention with flank and shifting dullness. An umbilical nodule was also noted. The urinalysis revealed pyuria and microscopic hematuria, but the urine culture was sterile. An abdominal ultrasonography showed bladder mass measured as 15mm and diffuse intra-abdominal free fluid collection. A computerized tomography (CT) demonstrated diffuse ascites with nodular peritoneal thickening and multiple lymph nodes over 10mm at the internal iliac, common iliac and paraaortic regions. An umbilical mass measured as 30mm was also seen on CT imaging . SurgicaPeritoneal mesothelioma is a rare variant of malignant mesotheliomas. The prognosis depends primarily on the type of tumor. Localized and well-differentiated PM tends to be clinically indolent with survival in the range of years and even decades. However, diffuse and high-grade tumors are more destructive and infiltrative with a median survival time of less than 1 year . SeveralTreatment alternatives for PM include observation, chemotherapy alone, cytoreductive surgery (CRS) alone, CRS combined with chemotherapy and CRS combined with HIPEC. One of the largest studies by Verma et al. suggested that combined modalities such as CRS/chemo and CRS/HIPEC had the most favorable results in terms of overall survival rates . On the Synchronous primary cancer with peritoneal malign mesothelioma is an extremely rare condition. There are only a few reports related to this topic in the current literature. Jacobsen et al. reported a case of PM in a patient with non-muscle invasive urothelial carcinoma . The othIn conclusion, to our knowledge, this is the first case published in the literature who have muscle-invasive bladder cancer with synchronous peritoneal malign mesothelioma. As in the presented case, precise pathologic examination with correct immunohistochemical staining is essential to confirm the diagnosis and exclude other types of malignancies. In addition, accurate clinical staging of these tumors is needed in order to plan treatment options. However, no curative or definitive treatment is determined at the current literature since its extremely rare condition. Such challenging cases must be addressed through direct communication in a multidisciplinary tumor board setting. Individualized treatment with multidisciplinary close follow-up schemes might improve the survival outcomes in these patients.


Objective: Unilateral ocular leukemic infiltration with acute angle closure is an infrequent complication of B-cell acute lymphoblastic leukemia (ALL-B). We present a clinical case of leukemic ocular infiltration as the sole manifestation of ALL-B relapse.Methods: Case descriptionResults: A 15-year-old female with a history of acute lymphoblastic leukemia in remission for 2 years and pulmonary tuberculosis treated in the past year presented with ocular redness and decreased visual acuity in the left eye (LE) with 5 days of evolution. Visual acuity was 20/20 in the right eye (RE) and absence of light perception in the left eye (LE). Biomicroscopy of LE showed a small hypopion, anterior chamber cells 4+, vitreous cells 3+, and a large white mass in the vitreous with associated vitreous hemorrhage in organization. In LE fundoscopy, the vitreous mass occupying most of the vitreous cavity and associated hemorrhage prevented retina visualization. B-scan ultrasound showed a multilobulated mass occupying virtually the entire vitreous cavity with associated choroidal detachment. Forty-eight hours later, she developed acute angle closure of LE with an IOP of 55 mmHg. A flow cytometric analysis of the anterior chamber and vitreous showed leukemic tumor cells. The microbiologic exam and PCR for Mycobacterium tuberculosis were negative. No other signs of relapse of the disease were identified after investigation by the oncology department. Rescue treatment of the underlying disease was started, with symptomatic improvement. Conclusion: Leukemic ocular infiltration can be the only manifestation of ALL-B relapse. Ophthalmic manifestations of leukemia have been described previously in literature and can occur in 9 to 90% of patients, mostly with retinal involvement , 2], , . HoweverWe present the case of a 15-year old girl with a history of B-cell acute lymphoblastic leukemia (t(1:19)) in clinical remission for two years. She was under maintenance chemotherapy with dexamethasone, mercaptopurine, and melphalan. In the past year, she was also suffering from pulmonary tuberculosis having completed 9 months of therapy. She presented in our ophthalmology emergency room with acute visual loss of the left eye (LE) and ocular redness for 5 days. Best corrected visual acuity in her RE was 20/20 and absence of light perception in her left eye (LE). Intraocular pressure upon presentation was 12 mmHg in RE and 22 mmHg in LE. In LE biomicroscopy, she presented with a very small hypopyon, small, inferior, keratic precipitates, anterior chamber cells 4+, vitreous cells 3+, and a dense white mass was visible in the vitreous cavity along with organized vitreous hemorrhage Figure 1 . The funMycobacterium tuberculosis. Ocular relapse of B-cell ALL was diagnosed. The patient was re-staged but there were no signs of relapse in the bone marrow, the central nervous system or any other systemic location. Radiotherapy (30Gy) and rescue chemotherapy (INTREALL HR) were started with symptomatic improvement and better IOP control even though the patient was still under topical hypotensive drugs. Final MAVC was absence of light perception and 8 months after presentation, the patient remained without any other signs of systemic relapse.The patient was medicated with an association of Timolol 1% and Dorzolomide 1% bid as well as topical Dexamethasone 1% qid. Two days later, she suffered clinical worsening with severe ocular pain, headache, and nausea. IOP was now 55 mmHg in LE and the biomicroscopy of LE showed a shallow anterior chamber. Acute angle closure was diagnosed, and we added topical Brimonidine 0.1% bid and Acetazolamide 500 mg qid to her therapeutic scheme. A flow cytometry analysis of the aqueous humor and vitreous revealed ALL-B cells sharing the immunophenotype with previously diagnosed leukemic cells (CD34–/CD10+/CD19+/CD20+ weak/CD38+). Microbiologic analysis was negative for bacteria and fungus as well as the PCR study for We present a case of unilateral leukemic infiltration as the sole manifestation of relapsed ALL-B. Though, infrequent, unilateral eye involvement as the first sign of leukemic relapse has been described previously in the literature , 5], , . Our patMycobacterium tuberculosis were negative and the finding of leukemic cells in the vitreous and anterior chamber was diagnostic. Our patient’s case also had the particularity of a challenging differential diagnosis given that our patient was under maintenance chemotherapy and had history of a tuberculosis infection in the previous year. Therefore, alternative diagnostic hypothesis such as endogenous endophthalmitis and ocular tuberculosis had to be excluded as well, especially given that the patient was clinically in remission. The microbiologic exam and PCR for Treatment of leukemic ocular infiltration comprises systemic chemotherapy, but if this is not an option or if the ocular disease is not responsive, ocular radiation may be combined , 6], ty, ty6], tFindings of ocular leukemic relapse should prompt a new systemic work-up by the oncology team. Even though our patient still has not developed any signs of systemic leukemic relapse, in some cases where ocular findings were the only sign of leukemic relapse, bone marrow or CNS relapse ensued in the following months .This case report highlights ocular leukemic infiltration as the first sign of leukemic relapse. A high suspicion index should be kept in these patients, even if in clinical remission for several years. Secondary acute angle closure is a less frequent presentation that can happen especially in cases with extensive intraocular infiltration.The authors declare that they have no competing interests.


Intercostal hemangioma is an extremely rare disease. It is difficult to distinguish intercostal hemangioma from other chest wall tumors. The difficulty in preoperative diagnosis may result in incomplete surgical resection and hence a high rate of recurrence.A 14-year-old boy who presented with asymptomatic right lateral chest wall mass with no history of trauma. Different radiological modalities were employed for diagnosis including computed tomography (CT) showed a soft tissue mass 6.5 × 4 × 5.6 cm in size abutting 5th and 6th ribs. Magnetic resonance imaging (MRI) revealed iso-intense signal in T1 and hyperintense signal in T2, that is higher than that of adjacent muscles in the inferolateral right chest wall which was compatible with intercostal hemangioma. The patient underwent surgery for excision of the mass. Through right posterolateral thoracotomy, there was a well-demarcated mass abutting 5th, and 6th ribs filling the right 5th interspaces. Histopathological examination confirmed the diagnosis of intercostal hemangioma.Chest wall hemangiomas are uncommon and mostly arise outside the rib cage. Hemangiomas rarely occur in the intercostal space, and most of these originate from intercostal muscles . Watson and McCarthy postulated two theories for the etiology of hemangiomas. The more widely supported theory claims that hemangiomas are of congenital origin. The other theory proposes a traumatic origin of hemangiomas.Complete surgical resection should not be compromised by the resultant chest wall defect to prevent recurrence. Difficu2In November 2017, an asymptomatic 14-year-old boy referred to the authors’ hospital for management of right lateral chest wall mass discovered incidently one month earlier. There was no history of trauma. Clinical examination was unremarkable. Plain chest radiography showed a smooth-walled 6.0 × 3.5 cm homogenous right lateral chest wall mass A. ComputMagnetic resonance imaging (MRI) revealed right extrapulmonary and extrapleural chest wall soft tissue mass. Tl-weighted MRI demonstrated a solid mass with a low signal intensity C, while The angiogram showed a sizeable vascular mass supplied by a branch of the internal mammary artery. That branch was subsequently embolized with Gelfoam pledgets with no further filling on the post-embolization arteriogram.®, DUALMESH®, W. L. Gore & Associates) (The patient underwent an exploratory video-assisted thoracoscopy (VAT) through the right 8th intercostal space anterior axillary line A and B. ociates) D.Fig. 2PThe surgical specimen measured approximately 6 × 4 × 3.5 cm, which included a hemorrhagic, highly vascular, mass that extended from the intercostal muscles to the extra-pleural space E. Cut se3Hemangiomas are benign slowly growing vascular tumors that account for 7% of all benign soft-tissue tumors in the whole body ,6. ChestThe preoperative diagnostic tools of chest wall tumors involve a detailed medical history, clinical examination, and radiological investigations. CT of the chest can detect the effect of the tumor on lung, ribs, and pleura, as well as phleboliths, which sometimes accompany hemangiomas . IntercoWide local excision with the sufficient tumor-free margin is recommended. Hemangiomas are prone to bleed spontaneously or after minor traumatic injury. Embolization of the feeding arteries ultimately followed by recurrence as the arteriovenous communications are small and numerous. Complete excision of the tumor is mandatory even after embolization to prevent recruitment of a collatera1 blood supply .The expected chest wall defect should not jeopardize wide local excision of the tumor. Incomplete excision may lead to recurrence, disturbed wound healing, and secondary bleeding . ReconstMethyl Methacrylate has been the best choice to reconstruct the sternum, ribs and chest wall, entirely or partially for many years. It is usually sandwiched between two layers of the mesh to strengthen the rigidity of the reconstruction. It is suitable for most of extensive anterior and lateral chest wall defects, prevents paradoxical movements and chest deformities. However, methylmethacrylate material is not permeable to fluids and therefore, are considered to increase pain and excessive chest wall rigidity, liable to fracture and has a higher risk of infection .Titanium plates are newly introduced material that seems to be an ideal prosthetic solution for chest wall reconstruction. It has resistance to corrosion, a low weight, resistance to traction, compatible with MRI, and biologically inert . Many au4This paper presented a case of intercostal hemangioma arising from the right lateral 5th intercostal muscles. A tentative diagnosis is feasible due to the characteristic imaging of the tumor. Complete surgical resection should not be compromised by the resultant chest wall defect to prevent recurrence.No conflict of interest to declare.No source of funding.Institutional Review Board approval. Case Report is presented anonymously.Patient is a minor, we have parental consent on behalf of the patient.Hatem Elbawab: Study concept, writing the paper.Farouk Alreshaid: Writing the paper.Asayil Alnasser: Editing the paper.Raja Husain: Communication, submission.Yasser Aljehani: Writing the paper.Tarek Hashem: pathologist, pathology report writing.Not Applicable .Hatem Elbawab.Not commissioned, externally peer-reviewed.


Solid, fat-containing tumors of the testes are extremely rare with only a few cases having been reported so far, contrary to the more frequent occurrence of paratesticular lipomatosis. Testicular angiomyolipomas and gonadal involvement in Cowden's disease, although infrequently occurring, are known examples of fat-bearing testicular lesions. Hereby, we present an extremely rare case of intratesticular angiolipoma. Angiolipomas are benign tumors of the subcutaneous tissue commonly occurring in the trunk and the extremities. Histologically, they are characterized by ample vascularity and an excess of mature adipocytes. Definitive diagnosis is established by biopsy of the lesion.  A 35-year-old patient presented to our andrology outpatient clinic for fertility assessment. Physical examination of external genitalia revealed no significant pathology. Testicular ultrasound however depicted an isoechoic lesion on the upper pole of the right testis measuring 1.8 cm × 0.8 cm × 1 cm and exhibiting intense arterial flow. After sonographic and MRI investigation, the patient was referred for semen analysis and cryopreservation. Subsequently, the patient underwent testicular biopsy (frozen section biopsy) and right partial orchiectomy. Final histology reported a noninfiltrating testicular angiolipoma. No recurrences have been observed in the follow-up period.  Angiolipomas, which mainly occur in the trunk and extremities, are classified as infiltrating and noninfiltrating. The diagnosis is based on both clinical and histologic criteria, and the main method of treatment for both types is by surgical excision. The infiltrating type exhibits higher recurrence rates.  Angiolipomas commonly occur in the subcutaneous tissue and have been invariably treated by radical excision. Hereby, we report the first case of an intratesticular, noninfiltrating type angiolipoma which was treated by partial orchiectomy. Angiolipomas are benign tumors, commonly occurring in the subcutaneous tissue. They are either encapsulated or nonencapsulated fatty tumors and are distinguished from other lipomas by the excessive degree of vascular proliferation and abundance of mature adipocytes. They more often occur in the trunk and extremities, rarely in the maxillofacial area  and are A 35-year-old patient presented to our andrology outpatient clinic for fertility assessment. Physical examination of the external genitalia was insignificant. There was neither history of testicular trauma nor any sexually transmitted disease in his medical record.Testicular ultrasound revealed a solitary isoechoic, highly vascularized lesion of the upper pole of the right testis measuring 1.8 cm × 0.8 cm × 1 cm distinctly separated from the adjacent healthy parenchyma by a well-defined hypoechoic capsule . No addiβ-hCG, and aFP) and semen analysis fell within the normal range (Tables Hormonal testing (FSH, LH, Testosterone, e Tables  and 2.CT scan of the thorax, abdomen, and pelvis revealed no retroperitoneal lymphadenopathy or other abnormalities.MRI of the scrotum demonstrated a well-defined encapsulated mass, located in the upper pole of the right testis measuring 1.9 cm × 0.9 cm × 1.2 cm.After semen cryopreservation, the patient underwent surgical exploration. Intraoperative frozen section analysis reported a nonmalignant lesion, possibly an angiolipoma. A right partial orchiectomy was subsequently performed. The final histology reported a 1.9 cm × 0.9 cm × 1.2 cm lesion composed of adipocytes and small vessels with fibrous microthrombi, identified as noninfiltrating type angiolipoma. The tumor was circumscribed and partially encapsulated. Capillaries and mature adipocytes were observed. Few vascular channels contained microthrombi. Around the periphery of the tumor, the capillaries and the adipocytes entrapped the seminiferous tubules. This finding, which is also observed in testicular hemangiomas, perhaps predicts a more aggressive behavior of the tumor with higher possibility of recurrence in cases of partial orchiectomy Figures 2.Follow-up visits included physical examination, hormonal assessment, and scrotal ultrasound every three months for the first two years, every six months for the next two years, and subsequently on a yearly basis. The patient has currently been free of recurrence.Angiolipomas consist a variant of lipomas which was first described by Bowen in 1912  classifiBased on cytogenetic analysis, the karyotype of angiolipomas differs from that of other benign lipomatous tumors  which suAngiolipomas can easily be distinguished from other lipomas from their excess of vascular elements . There aJ. J. Lin and F. Lin classified angiolipomas into infiltrating and noninfiltrating types . The nonThe main treatment for both types is by complete excision. Noninfiltrating angiolipomas have no tendency to recur after surgical removal; therefore, a simple excision of the lesion will probably be curative. In the case of infiltrating angiolipomas, the recurrence rate is higher and a wide surgical excision is imperative , 12, 13.Angiolipomas are benign tumors, commonly occurring in the subcutaneous tissue. This is the first recorded case of an intratesticular, noninfiltrating type angiolipoma which was treated by organ-sparing surgery. Two years after the procedure, there were no recurrences. The rarity of the specific entity and the lack of published experience impede the construction of an evidence-based treatment algorithm. Every case report adds to the growing body of evidence.


R334Q). This variant is the most common mutation responsible for early onset sarcoidosis (EOS)/Blau syndrome, an autoinflammatory disease characterized by granulomatous inflammation of joints, skin and eyes. The early onset of symptoms and presence of multinucleated giant cells and granuloma in this case are in keeping with a diagnosis of EOS/Blau syndrome. The patient responded well to treatment with methotrexate and etanercept. This case extends the clinical spectrum of EOS/Blau syndrome, which should be considered for GCTTS and other unusual presentations of tendon inflammation in children, even in the absence of the characteristic triad of arthritis, dermatitis and uveitis.Giant cell tumor of tendon sheath (GCTTS) is characterized by diffuse proliferation of synovial-like cells and multinucleated giant cells along tendon sheaths. This benign tumor typically presents in the third to fourth decade of life and is exceeding rare in children. Here we describe a case of a 10-years-old girl with a history of soft tissue swelling involving the third digit of left hand, bilateral wrists and ankles. Pathology of the finger mass revealed abundant multinucleated giant cells consistent with GCTTS. Resection of the tendinous masses from the ankles also showed multinucleated giant cells along with chronic bursitis. She began to show features of polyarticular arthritis by age 7. Due to progression of arthritis, whole exome sequencing was performed and found a  Giant cell tumor of tendon sheath (GCTTS) is the second most common tumor of the hand, typically presenting in the third to fourth decade of life . Also knNOD2 responsible for early onset sarcoidosis , an autoinflammatory syndrome characterized by granulomatous arthritis, uveitis and dermatitis.GCTTS is rare in children with only one large case series of 29 patients describing this entity in the pediatric population . Most ofWe present a 10-years-old girl with a history of tissue swelling involving the third digit of left hand, bilateral wrists and ankles. At 2 years of age, she first developed soft tissue masses above both wrists and ankles without pain, warmth or redness. She did not receive any treatment and the soft tissue masses enlarged slowly. Two years later, she underwent surgery to remove the ankle masses and pathology was reported to be consistent with chronic bursitis. However, swelling gradually recurred near the surgical sites within 1 year.At 6 years of age, a new mass appeared on the palmar aspect of the left hand at the base of the third digit . The masWith progression of symptoms, the patient was referred to our Rheumatology Clinic for evaluation. Physical examination was notable for symmetric soft tissue masses along the anterior tibialis tendon of ankles and extensor tendons of the wrists . She hadMRI of wrists showed synovial thickening with abnormal signals around the tendon sheath, with evidence of wrist bone erosions . Ophthalde novo heterozygous mutation in NOD2  that was not found in either parent , a growth factor for monocytes/macrophages  is among the most common mutations found in EOS/Blau syndrome. More than 15 mutations in NOD2 are closely associated with the disease  or interleukin-1 , 18. EffIn summary, we present a young child with EOS that developed GCTTS and tendon inflammation early in the disease course in the absence of dermatitis or uveitis. This case suggests an intriguing link between GCTTS and EOS/Blau syndrome, two conditions that share the hallmark of multinucleated giant cells and tendon inflammation. EOS/Blau syndrome should be considered in children with GCTTS and unusual presentations of tendon inflammation even in the absence of the characteristic triad of arthritis, rash and uveitis.The datasets generated for this study are available on request to the corresponding author.The studies involving human participants were reviewed and approved by Ethics committee of Guangdong Second Provincial General Hospital. Written informed consent to participate in this study was provided by the participants' legal guardian/next of kin. Written informed consent was obtained from the individual(s) for the publication of any potentially identifiable images or data included in this article.SZ supervised the data collection and drafted the manuscript. PL designed the study and revised the manuscript. YH analyzed the data and drafted part of the manuscript. AW carried out the pathological analyses. TL designed the study, assigned the treatment protocol, and revised the manuscript. All authors approved the final manuscript as submitted and agree to be accountable for all aspects of the work.The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.


Heterotopic pancreas (HP) is a rare disease commonly found incidentally on imaging studies, at endoscopy or at autopsy and can be associated with abdominal pain, vomiting, heart burn, gastric outlet obstruction, and even dysphagia in very rare cases. Heinrich’s classified HP into three groups, types1–3, with Heinrich’s type 3 HP the rarest and difficult to diagnose properly because it has only pancreatic ducts but has no islet and acini. The aim of this study is to report a case of gastric outlet obstruction caused by type 3 HP with gastroduodenal invagination with reference to the literature and diagnosed finally by immuno-histochemical analysis.The case presented is a 40-year-old male presenting with vomiting and abdominal pain. Computed tomography (CT) revealed a cystic mass in the upper abdomen and he was referred to the Tokushima University. Gastric fiber showed that the pedunculated mass originated from the stomach. An open distal gastrectomy was performed. Pathologically, there was small glands proliferation in the sub-mucosal (SM) layer which was membrane and cytoplasm (MUC)1 positive and muscle proliferation.This finding revealed the tumor as HP. Postoperative course was uneventful and the patient was discharged 12 days after surgery. The patient has remained well 12 months after surgery.HP should be considered in the differential diagnosis of SM tumors with gastroduodenal invagination even if this is a rare symptom. Submucosal (SM) tumors are tumors in the submucosa or muscularis propria of the gastric wall and are associated with many types of diseases. Gastrointestinal stromal tumors (GISTs) and leiomyomas are the most common gastric SM tumors. Heterotopic pancreas (HP) is also one of the differential diagnoses for SM tumors but is very rare. It is often difficult to differentiate SM tumors , 2, so sAs previously stated, HP is one of the SM tumors and is defined as the presence of pancreatic tissue outside its normal location and without anatomic and vascular continuity with the main body of the pancreas . UsuallyPathologically, HP is variably composed of pancreatic acini, ducts, or islets. Some cases contain all components of the pancreas, including ducts, acini, and endocrine islets, whereas others consist of pancreatic ducts only. Heinrich named the three types as types 1–3 respectively . HPs havA 40-year-old male presented with vomiting and abdominal pain. Computed tomography (CT) revealed a cystic mass in the upper abdomen and the patient was subsequently referred to the Tokushima University. Laboratory tests were as follows: leukocyte count 7400/μl; hemoglobin 8.6 g/dl; albumin 4.0 g/dl; amylase 91 IU/l; total bilirubin 0.3 mg/dl; carcinoembryonic antigen (CEA) 1.5 ng/ml; carbohydrate antigen 19-9 (CA19-9) 8 U/mL; DUPAN-II < 25 U/ml; and S-pancreas-1 antigen (Span-1) 7 U/ml. The CT in the axial view revealed a 6 cm low-density mass in the pylorus of the stomach and a coronal view confirmed gastroduodenal invagination (Fig. According to these findings, GIST, duplication cyst, malignant lymphoma, and gastric polyp could be stated as a differential diagnosis Table . In partThe resected specimen showed a solid, edematous nodule with small glands originating from the gastric antrum with some of the small glands and the stroma in the SM layer. Fig. . PatholoSM tumors are divided into a number of groups and GISTs and leiomyomas are the most common of these gastric SM tumors. HP is also one of the differential diagnoses. It is often difficult to differentiate SM tumors into the respective groups . Pang’s The SM location of pancreatic heterotopia is often evaluated using endoscopic ultrasound with the recognition of features distinguishing it from a GIST or leiomyoma, the major clinical and endoscopic differential diagnoses. Moreover, EUS-FNA is reported to be useful  for a prIt has been reported that the proper diagnosis rate of HP by CT is only 16.7% , while tPathologically, HP is variably composed of pancreatic acini, ducts, or endocrine islets. Some cases contain all components of the pancreas, including ducts, acini, and endocrine islets, whereas others consist of pancreatic ducts only. In 1909, Heinrich classified the HP into three types histologically: type 1, the most common, with all the components of the pancreas including acini, ducts, and endocrine islets; type 2, with acini and ducts and no endocrine islets; and type 3, with ducts alone . In the HP is usually asymptomatic , 13, 14 The HP tissue can undergo secondary changes and complications such as acute pancreatitis, pseudo-cyst formation, development of mucinous cysts, and pancreatic intra-epithelial neoplasia , 24–26. HP can show vertical progression and gastroduodenal invagination. When experimenting with such a tumor, consideration should be given to HP always being included as a part of differential diagnoses. When the diagnosis is difficult, the histological examination, in particular, immuno-histochemical analysis, for example MUC, can reveal pancreatic ectopia, which is a helpful diagnostic method.


Hepatocellular carcinoma (HCC) most commonly develops in patients with liver cirrhosis caused by hepatitis C and B virus. HCC is the most common cause of death in people with cirrhosis. The lungs, bone, and lymph nodes are frequent sites of metastasis of HCC.  We present a case of solitary skull metastasis as the first symptom of HCC. A literature review with regard to HCC skull metastasis was undertaken.  This patient had HCC based on chronic hepatitis C. He presented with an erosive lesion masquerading as a scalp lipoma resulting in hepatocellular carcinoma with retrograde reflux and sinus metastases. The lesion was ultimately resected, but the patient died within one week from liver failure. Seventeen similar cases were found in the literature. Median age of the patients was 58 years with 95% being male and 35% having viral hepatitis. Surgery was the preferred treatment with chemotherapy and radiation being other options. Outcome was acceptable but baseline liver disease is a limiting factor.  Cranial metastases of HCC are rare but should be considered in patients with chronic hepatitis C presenting with scalp skin lesions. Hepatocellular carcinoma (HCC) is the most common type of primary liver cancer in adults and is the most common cause of death in people with cirrhosis . It occuExtrahepatic metastatic spread of HCC occurs in about 50% of cases, most often to the lungs, lymph nodes, and bones. Prognosis of patients with distant metastases from HCC is unfavorable with a median survival time after diagnosis of 6.7 months . In receIn many patients with bone metastases, antineoplastic drug therapy, radiotherapy, and palliative therapy are selected to relieve pain, reduce neurological symptoms, and maintain quality of life. Surgery is indicated for a limited number of patients as long as their general condition permits. Many patients cannot undergo surgery because the metastatic lesion is present at a nonresectable site or multiple metastases are present. . In addiBodhu et al.  have repSuperior sagittal metastasis eroding through the skull masquerading as a clinical lipoma or other soft tissue mass is a rare occurrence. As such, a clinician requires a high degree of clinical suspicious and resultant radiographic evidence in order to make the diagnosis. Ultimately, tissue diagnosis is desirable in such cases, and excision of such lesions should be chosen over percutaneous biopsy due to risk of tumor spread in the biopsy track.Here, we present the case of a 54-year-old male patient who presented to the surgical clinic with a mobile subcutaneous mass of the vertex of the scalp clinically consistent with a lipoma that turned out to be an erosive metastatic sagittal sinus tumor. We performed a review of the literature with regard to patients with similar HCC lesions.The case is presented in detail. We conducted a literature review of all the cases between 2009 and 2019 using PubMed using the search words “skull metastases” and “hepatocellular carcinoma”. The following data was extracted: age, sex, clinical presentation, microscopic features, site of metastasis, dural involvement, bone involvement, treatment, outcome, time to expiration (if noted), and presence of viral hepatitis.A 54-year-old male with chronic hepatitis C was referred to our plastic surgery office for a scalp mass. His MELD (model for end-stage liver disease) score was calculated to be 19 just prior to our clinical encounter . His vitals were within normal range, and he had no ascites but did have an episode of encephalopathy the previous year which since resolved with improved liver functions. His multiple comorbidities included hypertension, hyperlipidemia, tobacco use, and stage II chronic kidney disease. He had not been diagnosed with any malignancy.The vertex scalp mass had been gradually increasing in size over the previous month. On inspection, it was noted to be 3 × 4 cm with no visible ulcerations or blisters on the overlying skin. On palpation, the mass was noted to be soft and moderately mobile but with some adherence at the base. The surrounding cranium was palpable except for the area directly under the mass. His cervical lymph nodes were noted to be normal across all subgroups. He had recent head imaging with a computed tomography (CT) scan after a motor vehicle accident 7 months prior. These images were reviewed, and no supracranial nor intracranial mass was noted. Furthermore, there was no sign of any erosive skull pathology.th rib metastases as well. Neurosurgery was consulted, and an en bloc tumor resection was performed by duroplasty and cranioplasty with bone cement. Microscopically, the tumor revealed a characteristic trabecular pattern with thickened cords of cells separated by vascular sinusoids, mimicking the cell plates and sinusoids of a normal liver (As a result, we planned to proceed with excision of the mass, clinically presumed to be a lipoma or cyst, in our clinic procedure room. Under local anesthesia and sterile conditions, an incision was made in the skin over the lesion, and a glistening grey capsule was encountered with a cranially adherent base. There seemed to be a small but present cranial defect. Given the unusual findings, the incision was closed without excision, and a CT and MRI scan of the head was obtained revealing a superior sagittal sinus-based tumor eroding through the cranium . Systemial liver . The tumal liver . The marTo date, 17 cases of HCC with skull metastases have been reported . The meaSkull metastasis of HCC is relatively rare, in contrast to the incidence of skull metastasis in the lung, breast, thyroid, and prostate cancers . In the In a review of 60 articles published between January 1966 and November 2005, Hsieh et al.  found thThere are previous reports that document the high frequency of HCC metastases to the head and neck region as an initial presentation of the disease , 23 AlthHCC with distant metastases markedly worsens the patient's prognosis. It is rarely associated with a metastatic spread to the sagittal sinus eroding to the skull. Diagnosing such a rare clinical entity is a challenging task and is made possible via a high clinical suspicion and resultant radiographic evidence. In patients with a history of liver cirrhosis, an index of suspicious must be maintained regarding the risk of head and neck tumors being the first presentation of metastatic HCC with the probable mode of spread being retrograde via vertebral or azygos veins. This case and our literature review illustrate that skull metastases should be considered in the clinical course of HCC because early diagnosis leads to prompt treatment which can improve patient survival.


One third of all cases of head and neck carcinoma (CA) concern the oral mucosa. The use of dental implants (DI) for dental rehabilitation is widely extended. However, a few studies have reported some cases with neoplasic alterations, among the tissue surrounding implants. Our aim was to analyze possible alterations at the bone-implant interface in patients with oral squamous cell carcinoma (SCC), providing new evidence that could relate or discard a possible link between these factors. We used, for the first time, different techniques, including electron microscopy and histology, to analyze the implant ´s surface and the surrounding tissue from four clinical cases with neoplasic alterations surrounding DI. Histologically, ample inflammatory tissue was found in direct contact with the implant surface. Surface analysis of this tissue, revealed titanium percentages. According to our study, no oncological relation with deterioration of the implant surface was found, although DI were constantly related with peri-implantitis, a chronic trauma of the oral mucosa that could involve a neoplastic factor. Key words:Dental implants, carcinoma, peri-implantitis. SucceCase ReportCase 1: An 83-year-old man underwent bilateral cervical functional dissection and right partial mandibulectomy at our facility due to squamous cell carcinoma of the gums (T4N0Mx), receiving adjuvant radiotherapy. During routine clinical surveillance, a new ulcerous lesion was detected, surrounding the DI placed for rehabilitation eight years earlier. An implantoplasty  was performed to eliminate contact between the implant and this lesion. Nevertheless, no improvement was noted after 6 months. A CT scan showed lytic lesions on the left mandibular body and lytic lesions on the cortical lingual side, surrounding the DI. A biopsy revealed well-differentiated invasive SCC and the patient underwent a segmental mandibulectomy, with a second radiotherapy regimen.Case 2: A 60-year-old man reported a two-month history of a verrucous lesion on the gums, associated with pain and bleeding to the touch; over the alveolar rim of the third quadrant, located distal to a DI at position 37. Left cervical lymphadenopathy was palpable, level IIA. A CT scan and orthopantomography showed lytic lesion. Biopsy showed well-differentiated SCC, clinical stage T4N1Mx. A bilateral functional cervical dissection with segmental mandibulectomy was performed. Adjuvant radiotherapy and chemotherapy were administered.Case 3: A 54-year-old woman with a mandibular dental prosthesis supported on DI with a two-month history of a gum lesion over the right mandible, and no cervical lymphadenopathies. CT scan showed bone resorption at mandibular symphysis adjacent to the gum lesion. After a biopsy confirmed the presence of SCC, we performed a bilateral cervical dissection with segmental mandibulectomy and micro-vascularized fibula free flap. Adjuvant radiotherapy was then given. Case 4: A 64-year-old man with diabetes reported an excrescent lesion over the mandibular vestibular gingiva, in contact with a dental prosthesis over two DI placed two years previously. A biopsy revealed poorly-differentiated SCC. A CT scan and orthopantomography showed a lytic bone area related to a soft tissue mass, corresponding to oncological stage pT4N0Mx, close to the DI. A bilateral cervical dissection and marginal mandibulectomy were performed, followed by adjuvant radiotherapy. Six months later the patient died due to metastatic spread of a lung’s adenocarcinoma, developed after oral CA.Fragments of the resected mandibles, including the implants, were analyzed by Scanning Electron microscopy (SEM) with a JEOL JSM-6490LV SEM. A new, clean and sterilized DI, never implanted, was used as a control.For histological evaluation the samples of the resected mandibles not containing the metal DI were decalcified, and embedded in paraffin. Sections were stained with Hematoxylin-Eosin. Samples containing the DI were not decalcified and stained with von Kossa’s Stain, to highlight bone, or with Toluidine Blue, to reveal soft tissues. All sections were observed and scanned with an Olympus VS120 microscope.Histological analysis confirmed the episodes of peri-implantitis. As shown in Figures Results show the control implant Fig.  left witet al., no association had been demonstrated. We have performed, for the first time, SEM surface analysis to reveal possible alterations of the DI or the host’s surrounding tissues. We detected no surface alterations on DI, exception made one case, in which decreased Ti values (up to 60%) were found. However, in this case the decrease in Ti was associated with the technique used for implantoplasty (Numerous carcinogenicity theories have been proposed, including corrosion of DI and release of Ti particles, that might be associated with cancer formation . Howevertoplasty . Histopaet al. (The relation between peri-implantitis and oncological lesions continues to be feeble . Althouget al.  have staIndeed, peri-implantitis, by definition, involves not only mucosal damage but also damage to the bone surrounding the implant as well . The asset al., including DI cases associated with oral cancer, though no direct etiological relation with DI was evident. Nevertheless, in their review, Singhvi et al., noted chronic trauma as a paramount factor in association with ill-fitting dentures. An inflammatory response associated with trauma represents a common factor with the peri-implantitis observed in our series. Likewise, Moergel et al., cited by Javed et al. in 2012, presented the largest series of cases with cancer near DI, emphasizing the role of peri-implantitis as a predominant clinical sign in 11 of 26 cases, including one from their own series and ten from other authors (Our findings are in consonance with those presented by Singhvi  authors . Peri-imThe setting of maximum risk could be a previously radiated patient with a genetic profile favoring CA lesions who undergoes dental rehabilitation with implants over bone lacking regenerative ability and where peri-implantitis is likely .After a complete review of the literature and the study of our clinical cases we conclude that, there is still no definitive evidence for DI as a direct risk factor for SCC. However, chronic irritators of the oral mucosa have been postulated as risk factors due to their presence in cases of oral cancer, and DI are frequently related with peri-implantitis, a chronic trauma of oral mucosa that is increasing. Given the presence of trauma and inflammatory processes close to DI; these could be contributing factors in oncological disease. Histological analysis confirmed the presence of peri-implantitis in all the cases studied. An extensive series of cases with a longer follow-up is advised to establish a possible direct etiologic or contributory agent for oral cancer.


Primary cutaneous adenoid cystic carcinoma is a rare slow-growing neoplasm, with limited literature reporting the involvement of the scalp. It has a tendency to recur locally; however, lymph node, distant pulmonary and bony metastases are exceptionally rare. We highlight the case of a 65-year-old female with primary cutaneous adenoid cystic carcinoma with distant pulmonary and bony metastases and the importance of imaging in diagnosing distant metastasis and perineural spread. In July 2012, a 65-year-old female presented with a subcutaneous nodule in the left postauricular region. She had no history of previous skin lesions or systemic tumour disease. Excision biopsy performed elsewhere showed benign skin adnexal tumour–eccrine cylindroma. She did not undergo any further treatment. In October 2014, the size of the lesion increased; non-contrast CT performed elsewhere demonstrated a mass lesion in the left temporal region posterior to the pinna with erosion of the underlying mastoid bone cortex. Fine needle aspiration cytology of the lesion showed epithelial neoplasm. Excision of the lesion with mastoid exploration was performed. Histopathological examination of the operated lesion showed features of malignant neoplasm with perineural invasion, and a diagnosis of adenoid cystic carcinoma (ACC) was made. Subsequently, the patient underwent external beam radiotherapy with a dose of 5000 cGy for a period of 3 months. In April 2015, the patient came to the dermatology department in our hospital, with complaints of tenderness and swelling in the left preauricular region; she was hard of hearing on the left side. Physical examination showed that the swelling was non-mobile and firm in consistency. Audiological evaluation revealed left conductive hearing loss. CT and MRI examination was requested to evaluate recurrence.T2 weighted fluid-attenuated inversion-recovery (FLAIR) hyperintense lesion in the pre- and postauricular region with diffusion restriction and apparent diffusion coefficient (ADC) value of 0.132 × 10−3 mm2 s−1 , indicating perineural spread via the left trigeminal nerve  is a rare skin tumour, with <50 cases reported.2 PCACC originating in the scalp was first reported by Boggio in 1975.3 Other reported locations include the tracheobronchial tree, oesophagus, lacrimal gland, ear canal, breast, uterine cervix, bartholins gland, prostate, mucosal gland of the upper respiratory tract and the skin. PCACC frequently arises in the scalp of middle-aged or elderly patients, with a slight predilection for women.4 Histologically, several skin tumours closely mimic PCACC, such as the adenoid cystic variant of basal cell carcinoma, mucinous carcinoma, dermal cylindroma and other primary cutaneous apocrine carcinomas. The lack of epidermal connection, absence of peripheral palisading of neoplastic cells, periodic acid Schiff-positive material and perineural space invasion favour the diagnosis of ACC over other entities. The positivity for carcinoembryonic antigen and S100 protein and negativity for p63 favours the diagnosis of ACC.5 ACC of the head and neck is a slow-growing tumour. The average duration of the tumour prior to diagnosis is 9.8 years with a tumour size ranging from 0.5 to 8 cm with an average size of 3.2 cm.5 Local recurrences are frequent and metastasis, which occurs in 21–54% of cases, tends to involve the lung, liver, bone and brain.8 PCACC has a 50% local recurrence rate; however, metastases to regional lymph nodes and distant organs are exceedingly rare. To the best of our knowledge, after extensive literature search, only a few cases of PCACC with distant metastasis have been reported and none with bony metastasis.9 Imaging plays a vital role; CT scan determines the spread and analyses the metastatic spread of the disease, as seen in this patient with pulmonary and bony metastases to the vertebra and the iliac bone. The treatment for primary cutaneous ACC consists of either wide local excision or Mohs micrographic surgery.14 Mohs micrographic surgery has been recognized as the treatment of choice with the highest reported cure rate and very low recurrence rate.15 This patient had a recurrence after wide local excision. This could be attributed to the high rate of perineural spread, which accounts for the high recurrence rate after local excision with minimal margins or without micrographic control. ACC has a higher predilection for perineural spread, that is, tumour dissemination along the tissues of nerve sheath. Recognizing perineural spread  showed an average ADC value of >1.5 × 10−3 mm2 s−1 and more aggressive tumours (grade II and III lesions) showed an average ADC value of <1.2 × 10−3 mm2 s−1. Since all tumours follow the same physiology, these ADC values can be used as a reference in this article. The mean ADC value obtained in this lesion was 0.132 × 10−3 mm2 s−1, favouring the malignant grade. This needs further studying.Furthermore, MRI also aids in assessing post-chemotherapy changes by measuring ADC changes following treatment. Sun et al21 However, our patient did not undergo PET-CT examination.Positron emission tomography (PET-CT) acts as a vital imaging modality for assessment and management of head and neck cancer. PET-CT is found to be superior to MR or CT imaging in the assessment of lymph nodes, distant metastasis and synchronous primary malignancy. PET-CT is useful for evaluating the response to treatment and has higher sensitivity and specificity than MRI in detecting recurrent disease.PCACC of the scalp is a rare tumour of the head and neck region with common local recurrence but rare distant metastatic spread. Imaging plays a vital role in diagnosis of the primary lesion, recurrence, local and distant metastasis and perineural spread. PCACC of the scalp is a rare skin tumour.It is a locally aggressive tumour with rare lung and bone metastases; hence, imaging of the thorax, vertebral column and pelvis must be performed in patients to rule out metastasis due to the disease. PET-CT is an effective modality in evaluating distant metastasis and is highly recommended.Head and neck tumours are responsible for tumour recurrence and affect the prognosis and long-term survival. MRI is the modality of choice to identify perineural spread, which could result in better planning of surgical approach to achieve tumour-free margin.Written informed consent was obtained from the patient for publication of this case report, including accompanying images.