Mycobacterium haemophilum is a rare and emerging nontuberculous mycobacteria (NTM). It normally causes localized or disseminated systemic diseases, particularly skin infections and arthritis in severely immunocompromised patients. There have been 5 cases of M. haemophilum ocular infections reported in the literature. Only 1 case presented with scleritis with keratitis. Here, we reported 2 cases of M. haemophilum scleritis. One of them was immunocompetent host and had keratitis with radial keratoneuritis as a presenting sign.Case 1: A 52-year-old Thai female with rheumatoid arthritis presented with scleritis. Conjunctival scraping was carried out and the culture result was positive for M. haemophilum. Despite receiving systemic and topical antibiotics, her clinical symptoms and signs worsened. Surgical debridement was performed. After surgery, the lesion was significantly improved and finally turned to conjunctival scarring. Case 2: A 32-year old healthy Thai male without underlying disease presented with nodular scleritis and keratouveitis with multiple radial keratoneuritis. Surgical debridement of the scleral nodule was performed. Initial microbiological investigations were negative. Herpes ocular infections was suspected. Topical antibiotics, oral acyclovir, low-dose topical steroids and systemic steroids were started. The scleral inflammation subsided but later the keratitis relapsed, requiring corneal biopsy. Histopathology of the specimen revealed acid-fast bacteria and M. haemophilum was identified by polymerase chain reaction (PCR) and sequencing. The diagnosis of Mycobacterial keratitis was made. Although using the combination of systemic and topical antibiotics, his clinical status progressively deteriorated. Multiple therapeutic penetrating keratoplasties were required to eradicate the infection. No recurrence was found during the 1-year follow-up in both cases.M. haemophilum can cause scleritis and keratitis, even in immunocompenent host. Radial keraoneuritis is first described in M. haemophilum keratitis. NTM keratitis should be considered in the differential diagnosis of patients with radial keratoneuritis. Increased awareness and early diagnosis using appropriate culture conditions and molecular techniques are important for the proper treatment of this infection. Prompt surgical intervention appears to be vital for successful management of M. haemophilum scleritis and keratitis. Mycobacterium chelonae, M. abscessus, and M. fortuitum groups [M. avium complex, M. szulgai, M. avium, M. gordonae, M. haemophilum, M. kansasii, M. flavescens, M. marinum, M. nonchromogenicum, M. triviale, M. asiaticum or NTM of unknown species have also been reported to infect human eyes [Nontuberculous mycobacteria (NTM), also known as atypical mycobacteria or environmental mycobacteria, are aerobic, non-motile, non-spore forming bacilli that are naturally found in water, soil, food, and air . Althougm groups , 3. Howeman eyes , 3.M. haemophilum is a slow grower and traditionally classified as Runyon group III. It mainly causes cutaneous and subcutaneous infections, septic arthritis, osteomyelitis, pneumonia, and disseminated infection in immunocompromised hosts. Cervicofacial lymphadenitis is another common manifestation in immunocompetent children [M. haemophilum ophthalmic infections reported in the literature. Two cases presented with endophthalmitis [M. haemophilum scleritis. One of them also had keratitis with radial keratoneuritis as a presenting sign. Written informed consent was obtained from both patients to report their clinical data.children . This orhalmitis , 6, 1 cahalmitis , 1 case halmitis , and 1 chalmitis . Here, wA 52-year-old Thai female who presented with redness and irritation in her right eye for 1 month was referred to our hospital after 1 week of administration of topical ofloxacin and a combination of chloramphenicol and dexamethasone eye drops without remission. She had underlying rheumatoid arthritis which was well controlled with tofacitinib, methotrexate, and naproxen. She did not have any history of previous ocular trauma or surgery. Her best corrected visual acuity (BCVA) was 20/20 in both eyes. Slit-lamp biomicroscopy revealed multiple conjunctival pustules and severe, sectorial scleral inflammation with overlying conjunctival epithelial defects, extending from the 8 to 10 o’clock position and minimal punctate epithelial erosions at the inferior cornea , acid-fast and modified acid-fast stains. It was also cultured on blood agar, chocolate agar, Sabouraud’s dextrose agar, thioglycollate broth, Löwenstein-Jensen (LJ) medium and Mycobacteria Growth Indicator Tube . The specimens were also submitted for polymerase chain reaction (PCR) testing. The smears demonstrated no organism. Empirical treatment was started with topical 0.5% moxifloxacin eye drops 2 hourly and oral moxifloxacin (400 mg) once daily.The clinical picture did not improve over the following 2 weeks and the initial cultures were all negative. New and larger pustules appeared in the same area. Surgical debridement consisting of conjunctival dissection, de-roofing the pustular lesions and scraping the scleral base of the lesions was performed. Postoperatively, topical 5% amikacin and 0.5% moxifloxacin eye drops were given every hour.M. tuberculosis complex was evaluated using the M. tuberculosis complex specific antigen test  [M. tuberculosis complex, the mycobacterial strain was identified using pan-mycobacterial 16S rRNA gene PCR [M. haemophilum. In vitro antimicrobial susceptibility testing demonstrated that the isolate was sensitive to multiple antimicrobial agents, such as amikacin, rifabutin, rifampicin, clarithromycin, ciprofloxacin, moxifloxacin, and linezolid; and had intermediate susceptibility to doxycycline. The patient was placed on systemic antibiotics including oral rifampicin, clarithromycin, ciprofloxacin, and amikacin; and topical antibiotics including 0.5% moxifloxacin, 5% amikacin and 1.5% azithromycin. Oral tofacitinib and methotrexate were replaced with hydroxychloroquine for her underlying rheumatoid arthritis to minimize the adverse effects of immunosuppression.Three weeks later, mycobacterial isolate was detected in standard MGIT. The presence or absence of MD, USA) . When negene PCR . At the gene PCR . After tgene PCR . The resTwo days after intensive anti-nontuberculous mycobacterial treatment, markedly diffuse conjunctival hyperemia and edema as well as central corneal epithelial defect without infiltration developed Fig. b. Toxic A 32-year old healthy Thai male presented with pain and redness in the left eye for 5 weeks. He denied any history of previous ocular trauma, surgery, or contact lens wear. His BCVA was 20/20 in both eyes. Intraocular pressure (IOP) was 15 mmHg in the right eye but increased to 41 mmHg in the left eye. Slit-lamp biomicroscopy of the left eye demonstrated conjunctival congestion with a 2 × 2 mm scleral nodule in the temporal quadrant adjacent to the limbus, with dilatation of the superficial and deep episcleral vessels  5 times a day were added. Symptoms and signs gradually improved and all medications were slowly tapered. However, scleral inflammation relapsed after cessation of steroid therapy, once-daily dosing of topical steroids was then maintained.Mycobacterium tuberculosis. The PCR results were all negative. A presumptive clinical diagnosis of recurrent herpes simplex keratouveitis was made and topical steroids and oral acyclovir therapy were restarted.Six months later, the patient developed central corneal edema overlying an area of medium-sized, non-pigmented KPs, with some degree of superficial, gray-white, dry stromal infiltrates Fig. a. There M. haemophilum. Anti-nontuberculous mycobacterial therapy including hourly topical 0.5% moxifloxacin, 5% amikacin and 1.5% azithromycin; and oral rifampin, clarithromycin, amikacin and moxifloxacin were begun.One week after treatment, he developed multifocal paracentral stromal infiltrates at varying depths without epithelial defect, along with recurrent radial keratoneuritis and worsening of the anterior chamber inflammation Fig. b. ConjunDespite aggressive treatment, the corneal lesions became enlarged and rapidly progressed with dense stromal infiltrates and 2 mm hypopyon Fig. c-d. TherM chelonae (17), M. fortuitum (8), M. abscessus (1), M. marinum (1), and M. haemophilum (1) [M. haemophilum ocular infection was first described in 2007 in a man with graft-versus-host disease who presented with filamentary keratopathy and conjunctival mass and another man with post-cardiac transplantation who presented with purulent endophthalmitis [M. haemophilum ocular infection. Here, we present 2 cases of M. haemophilum scleritis and one of them also had keratitis with radial keratoneuritis as a presenting sign. The details of all 7 cases were summarized in Table To date, 15 reports of NTM scleritis have been published in the literature. The infections were caused by helonae 1, M. fortilum (1) –26. M. hortuitum , M. abscM. haemophilum ocular infection is challenging. The majority of patients with this infection had underlying immunocompromised conditions, including diabetes mellitus, use of immunosuppressive agents, and young age [M. haemophilum scleritis did not have predisposing surgeries, such as scleral buckling, pterygium excision, cataract surgery, intravitreal injection, and pars plana vitrectomy. Conversely, almost all cases of other NTM scleritis were directly preceded by surgical interventions [Diagnosis of oung age –9. Howevventions . TherefoM. haemophilum scleritis (4 and 5 weeks in our cases) appeared to be similar to that of other NTM scleritis, which generally varied, ranging from 1.5 weeks to 172 months [M. haemophilum scleritis compared to other infectious scleritis [M. haemophilum is often difficult and delayed because M. haemophilum is a slow-growing acid-fast bacilli and requires special culture techniques and media different from those of most pathogenic NTM species [M. haemophilum can be incorrectly identified as other organisms. Furthermore, some cases were misdiagnosed as viral infection or immune-mediated disease, leading to unnecessary use of steroids similarly to our second case [The insidious onset of 2 months . There wcleritis , 27. Add species . Thus, Mond case .Mycobacterium species in Case 1. Essentially, M. haemophilum is unique among Mycobacterium species owing to its specific growth requirements [M. haemophilum in conventional liquid MGIT medium at 35 °C in Case 1 was likely supported by the sufficient iron provided by blood from the biopsied conjunctival tissue [It is nevertheless intriguing that microbiological diagnosis was achieved by routine culture methods for irements . It prefirements , 29. AccM. haemophilum keratitis in our second patient presented with radial keratoneuritis which has never been reported in any case of NTM keratitis. Consequently, besides Acanthamoeba keratitis, Pseudomonas keratitis, fungal keratitis and Pythium keratitis [NTM keratitis is more common than NTM scleritis , 3. Typieratitis –33, NTM M. fortuitum (3), M. chelonae (1), M. marinum (1), and M. haemophilum (1) were responsible for 6 previously reported cases of sclerokeratitis. Four cases were associated with cataract surgery [NTM keratitis might extend to or start at the corneoscleral junction, resulting in sclerokeratitis , 17, 18.M. haemophilum [M. haemophilum including acid-fast staining, culturing at two temperatures with and without iron-supplemented media, and molecular detection is suggested [M. haemophilum-specific culturing at 30 °C in a liquid medium using hemin supplementation and on LJ medium supplemented with iron citrate [M. haemophilum [Therefore, increased awareness and early diagnosis of this infection are crucial for the proper treatment. Despite using the suitable culture condition, it may take up to 8 weeks to observe visible growth of mophilum . Moleculmophilum , 9. Hencuggested . The rec citrate . In addimophilum .M. haemophilum, previous studies showed that slow-growing NTM were susceptible to anti-tuberculosis drugs, whereas rapid-growing NTM were susceptible to macrolides, fluoroquinolones, and aminoglycosides [M. haemophilum [The treatment of NTM scleritis and keratitis needs a 2-pronged approach, which is appropriate antibiotic therapy plus judicious surgery to eradicate infection from affected tissues . Becauseycosides . Recentlmophilum –7. Topicmophilum , 30. MulM. haemophilum scleritis and keratitis. These 2 cases highlight the rare but emerging pathogen that can infect the eye in several different ways. This is also the first report of radial keratoneuritis in NTM keratitis. Real-time PCR assay is a valuable tool for the early detection of the organism.To the best of our knowledge, little information has been published regarding diagnosis and treatment of M. haemophilum can cause scleritis and keratitis, even in immunocompenent host. Radial keraoneuritis could be observed in NTM keratitis. The diagnosis of M. haemophilum ocular infection may be underdiagnosed because of unawareness among clinicians and challenges in identification of the organism in routine laboratories. The clinician’s vigilance must be increased to early recognize and promptly manage this infection. Consultation with a microbiologist and infectious disease specialist is essential to ensure accurate and timely diagnosis and effective treatment. Early surgical intervention appears to be vital for successful management of M. haemophilum scleritis and keratitis.In conclusion,


Masson's tumor or Masson's hemangioma, more precisely termed intravascular papillary endothelial hyperplasia (IPEH), is an uncommon benign vascular lesion of the skin and subcutaneous tissues which can be frequently confused with angiosarcoma. Although relatively rare, its accurate diagnosis is essential since it can clinically be similar to both benign and malignant lesions. We present a 39-year-old man with a round bulging arising from the left palm side of the hand with gradual growth in the last 5 months and on and off tenderness. The microscopic section demonstrated the papillary proliferation of endothelial cells in favor of Masson's hemangioma, which was sufficiently treated with excision alone. Intravascular papillary endothelial hyperplasia (IPEH) was first described by Pierre Masson as an intravascular papillary proliferation that forms within the lumen of an inflamed hemorrhoidal plexus, which was named vegetated intravascular hemangioendothelioma. Since then, various terms were considered for this pathology, such as hemangioendothelioma and reactive papillary endothelial hyperplasia or better known as Masson's hemangioma –3. It caA 39-year-old man with a complaint of a round mass on the palmar side of his left hand was referred to our clinic. The lesion had on and off tenderness and was gradually growing in size since 5 months ago. In our physical exam, there was a 4 × 3 cm, firm, round, and nontender lesion on the palmar side of the left hand over the second metacarpophalangeal joint, without a change in skin color or dense adhesions to surrounding soft tissues.With the impression of a benign hand lesion, without any further evaluation, the patient was scheduled for excisional surgery. Under local anesthesia, via a vertical incision over the lesion, the mass was enucleated easily, the wound was closed without any complications, and the lesion was sent for pathological evaluations.The gross examination of the lesion revealed an ovaloid gray-brown color cystic mass measuring 3.5 × 2 × 2 cm, in which the cut section revealed cystic mass filled with clot material. Microscopic examination showed dilated vessels containing papillary proliferation of plump endothelial cells without atypia overlying fibrous tissue Figures , and 3. IPEH constitutes about 2-4% of the benign and malignant vascular tumors of the skin and subcutaneous tissues . There iAs illustrated in our study, this entity is characterized by multiple small papillary proliferation of endothelial cells and thrombus formation at the center. The exact pathogenesis of IPEH is unknown, but an unusual organization of the thrombus after trauma is considered to play a key role , 18, 19.The main method for diagnosis is histological evaluation; however, IPEH can histologically simulate angiosarcoma, which makes distinguishing these two entities an important issue to avoid aggressive resection. Angiosarcomas are almost never confined to the vascular lumen; thus, the intravascular location of IPEH may assist in confirming the diagnosis. However, although rare, the extravascular subtype may result in a misdiagnosis. The differentials should also include pyogenic granuloma, Kaposi sarcoma, and other vascular conditions .  Table Simple excision is usually curative, although recurrence has been described . It is eIn conclusion, Masson's hemangioma can be misdiagnosed as a benign lesion as well as malignant neoplasm due to its clinical resemblance. Therefore, it is important to distinguish IPEH from other benign and malignant lesions, in particular, angiosarcoma to avoid unnecessary radiation and surgery. It can also be misdiagnosed as other conditions, such as a tendon cyst, on ultrasound evaluation. For these reasons, accurate diagnosis can solitarily be completed through histological examination.


Cocoon abdomen is a relatively rare abdominal disease characterized by the total or partial encasement of the small intestinal by a dense fibro-collagenous membrane.We reported an unusual case of idiopathic cocoon abdomen with congenital colon malrotation. Laparotomy and sac release were performed on the patient. The patient was no recurrence 6 months after operation. A literature review was also performed.Preoperative diagnosis of abdominal cocoon is difficult. A careful history, physical examination and appropriate radiology may be helpful in making a definitive diagnosis. If conservative treatment can’t relieve symptoms effectively, surgery is currently considered to be important in the management of this disease. Cocoon abdomen is a relatively rare abdominal disease in surgical emergency that was first described by Foo et al. in 1978 , 3. Due A 58-year-old male was admitted to the General Surgery Department with a 24-h history of paroxysmal colicky abdominal pain, abdominal distention, anus stop exhaust and defecation. The abdominal pain was persistent, and he had no nausea or vomiting. His symptoms gradually worsened. He had no previous medical history of abdominal pain, chronic diseases, chronic use of medication, infectious diseases like tuberculosis and abdominal surgery. He only had a history of tension-free repair for indirect inguinal hernia.The vitals were as follows: temperature 36 °C, pulse rate 96 beats per minute (regular), blood pressure 120/80 mmHg and respiratory rate 20 per minute. Abdominal examination showed a distended abdomen with tenderness. There were no palpable masses or gut sounds. The digital rectal examination was unremarkable.The laboratory examinations showed white blood cells 4.0 × 10^9/L, neutrophils 2.65 × 10^9/L, haemoglobin 13.2 g/dl, platelets 216 × 10^9/L, CRP 119.38 mg/L and PCT 0.32 ng/ml. Other lab findings were normal, including renal function tests, lactate and coagulation. Abdominal imaging examination was as follows: Erect abdominal X-ray detected hugely dilated small intestinal and multiple air-fluid levels in the rectum, which indicated the partial intestine obstruction  6, the patient complained of seriously abdominal pain, distension and vomiting. Bowel function was gradually recovered on POD 10 and the patient started oral intake on POD 11. He was discharged from the hospital on POD 14. A 6-month follow-up was carried out and no recurrence was detected.Cocoon abdomen is also called sclerosing encapsulating peritonitis (SEP) or chronic fibrocystic peritonitis . The etiThe clinical symptoms are nonspecific and variable ranging from asymptomatic to recurrent colicky abdominal pain, nausea, vomiting, abdominal distension, abdominal ascites, constipation, anorexia, malnutrition and weight loss. Preoperative diagnosis of cocoon abdomen is really challenge and most cases have been discovered incidentally during or after the operation. Imaging examinations might be helpful for preoperative diagnosis: 1. Erect abdominal x-ray: the main manifestation is intestinal obstruction for example, hugely dilated small intestinal or multiple air-fluid levels; however, sometimes normal phenomena are also presented and it is non-specificity. 2. Transabdominal ultrasound: dilated bowel loops enclosed within a membrane, peritoneal thickening and ascites are observed. 3.Barium meal films: the characteristic serpentine structure of the distal and inner cocoon dilated small intestine can be displayed. 4. Abdominal CT: the sign on CT is shown as a serpentine configuration of dilated small intestinal loops in a U-shaped cluster. Contrast-enhanced CT is the imaging modality of choice for the diagnosis of an abdominal cocoon  and PreoAt present, there is no consensus on the treatment of abdominal cocoon disease. For patients with mild symptoms, conservative treatment is advocated, including fasting, gastrointestinal decompression, electrolyte disturbance correction, traditional Chinese medicine, soap-suds or sodium phosphate enema. In addition, reasonable diet and good living habits can effectively prevent the aggravation of symptoms. For patients with severe symptoms, surgical treatment is the most effective method , 18.Surgical procedures included intestinal adhesiolysis and intestinal catheter separation. In idiopathic abdominal cocoon, a thin membrane surrounds the small intestinal and the capsule can be separated easily from the bowel. Excision of the small intestine is not routinely recommended unless necrosis of the small intestine occurs. On the contrary, it is usually quite difficult to separate due to a rather dense fibrous capsule with intestinal adhesion in secondary abdominal cocoon. This could be explained that why the membrane would be quite adherent in the chronic ongoing infection or inflammatory process. In the presented case the sac can be separated through an avascular plane easily which coincided with idiopathic cocoon abdomen. During surgery, the relationship between intestinal canal and mass should be carefully observed to avoid unnecessary resection. Extensive adhesiolysis for achieving complete excision of the capsule should not be carried out, so as not to damage the intestine or small intestinal vessels which lead to intestinal fistula or necrosis . FurtherSevere complications, including intra-abdominal infection, intestinal fistula, short bowel syndrome, and bowel perforation in idiopathic cocoon abdomen are not observed. Early postsurgical activity is recommended, and the postoperative dietary guidance is also very important to their rehabilitation . The dieIn conclusion, preoperative diagnosis of abdominal cocoon is difficult. A careful history, physical examination and appropriate radiology may be helpful in making a definitive diagnosis. If conservative treatment can’t relieve symptoms effectively, surgery is currently considered to be important in the management of this disease. Adhesiolysis with excision of membrane is recommended. Long-term prognosis is excellent and recurrence is rare .


Pulmonary sequestration is a congenital abnormality of the lower airway. It is characterized by a nonfunctioning mass of lung tissue that lacks normal communication with the tracheobronchial tree or pulmonary arteries and always receives its arterial blood supply from the systemic circulation. Most cases of extralobar pulmonary sequestrations (ELSs) are asymptomatic and found incidentally or in prenatal ultrasound screening.A 10-year-old boy had severe chest pain and vomiting for 2 days.ELS was diagnosed, and torsion of the ELS had developed as a complication.In video-assisted thoracoscopic surgery, the ELS was resected.The clinical symptoms were relieved the 2nd day after surgery and did not recur over a follow-up period of 3 months.In young patients with sudden abdominal pain or chest pain, in whom computed tomography shows a well-defined mass of homogeneous soft-tissue density in the thorax, ELS with torsion should be suspected. The presence of a feeding artery greatly supports the diagnosis of ELS, and the absence of this classic finding may indicate torsion of the pulmonary sequestration. ELSs are located outside the normal lung, have their own visceral pleura, are usually asymptomatic, and are found prenatally or incidentally. Symptomatic ELS caused by pedicle torsion is extremely rare, and the early recognition is difficult. We describe an unusual case of severe chest pain and vomiting caused by torsion of ELS in a 10-year-old boy.In pulmonary sequestration, a nonfunctioning mass of lung tissue lacks normal communication with the tracheobronchial tree, and it always receives its arterial blood supply from the systemic circulation. Pulmonary sequestrations are divided into the extralobar pulmonary sequestration (ELS) and intralobar pulmonary sequestration according to the relationship of the aberrant segmental lung tissue to the pleura.29/L), of which the neutrophils accounted for 81.1%; the myoglobin level was 38.12 ng/mL, and other values were within normal ranges. Because radiographs were available from the local hospital and in the emergency situation, we did not perform abdominal ultrasonography. Contrast medium-enhanced computed tomography (CT) of the chest and abdomen revealed a well-defined, nonenhanced mass with soft-tissue density (3.8 × 4 × 4.7 cm) in the left thoracic cavity above the diaphragm  adherent to the aortic wall and a small amount of hemorrhagic pleural effusion in the left pleural cavity. The parietal pleura near the mass appeared to be red. The pedicle that connected the mass to the mediastinum was short (1.0 cm in length), thin (0.6 cm in diameter), and twisted 180°. The blood supply to the mass arose from the thoracic aorta, and an accompanying vein drained to the intercostal vein. The mass was clipped and then resected at the pedicle. The pathological examination confirmed the diagnosis of ELS with infarction caused by torsion. The patient recovered very well and was discharged the second day after surgery. During the 3-month follow-up period, no complication or recurrence was observed on chest radiographs. The final diagnosis was an ELS infarcted as a result of torsion.  This study was approved by the Human and Ethics Committee for Medical Research at Sichuan University in accordance with the Declaration of Helsinki. Written informed consent was obtained from parents of patient involved in the study.3 ELS is less common than the intralobar type. In the majority of cases, ELS is on the left side, between the left lower lobe and diaphragm. ELS lesions have no bronchial connection to the normal proximal airway or normal lung, and infectious complications are also uncommon. ELS is usually asymptomatic and found prenatally or incidentally in young children, torsion, and infarction of ELS are extremely rare.Pulmonary sequestration is rare, with an estimated incidence of only 0.225% to 0.425%, and it accounts for only 0.15% to 6.4% of all congenital abnormalities of the lower respiratory tract.–15 (Table Only 11 cases of pulmonary sequestration with torsion have been reported in the literatureAll the adult patients experienced pain or discomfort mainly in the chest. Only 2 of the 8 pediatric patients had chest pain; abdominal pain was the most common symptom in the other 6. Of all 11 patients, 9 underwent unenhanced or contrast medium-enhanced CT preoperatively, and in only 1 case was a suspect feeding artery visible. Pleural effusion was present in all 11 patients. The preoperative diagnoses included diaphragmatic herniation, tumor, malformation, pulmonary sequestration, and unclear disease.Because the initial symptoms in children were mainly abdominal pain, it was either missed or delayed. The symptoms of ELS with torsion depend on the location of the lesion and whether the children could articulate their symptoms clearly. Young children with ELS and torsion near the diaphragm on the left pleural cavity usually complain of abdominal pain, rather than chest pain. Thus, ELS with torsion should be considered in children presenting with abdominal pain and a thoracic mass on imaging, despite the fact that torsion with ELS is rare. Angiographic appearance of a feeding artery is very strong evidence for the diagnosis of ELS. However, the most widely used tool still is unenhanced or contrast medium-enhanced CT, which were the first choices of imaging in the reported 11 cases.Imaging for diagnosis includes CT, magnetic resonance imaging, angiography, and ultrasonography. torsion can cause the supply artery not to appear on imaging, which causes difficulty in diagnosing ELS with torsion. Of the reported 11 patients, only 2 received the correct diagnosis preoperatively. Choe and Goo reported a case in which they found the feeding artery on axial T1-weighted magnetic resonance imaging, which helped establish the correct diagnosis of ELS with torsion preoperatively.The imaging features of CT include a homogeneous, well-defined mass of soft-tissue density in the thorax. Although the appearance of a feeding artery greatly supports the diagnosis of ELS,Thoracotomy or video-assisted thoracoscopic ELS resection is the standard treatment for ELS with torsion. Chest drainage is not always necessary, depending on the pleural effusion and the surgery.If a young patient presents with sudden abdominal pain or chest pain, and if CT shows a homogeneous, well-defined mass of soft-tissue density in the thorax, the clinicians should suspect ELS with torsion. The appearance of a feeding artery is, as mentioned, evidence of ELS, and the absence of this classic finding may indicate ELS with torsion. In addition, clinicians need to differentiate ELS from congenital pulmonary airway malformation, diaphragmatic hernia, tumor, and causes of symptoms.With modern imaging tools, it is easier to find the lesions; however, the preoperative diagnosis of ELS with torsion remains difficult. It is also important to identify the types of ELS in which torsion is most likely to occur; this would lead to earlier preventive ELS resection.LY wrote the first draft of the manuscript; GY performed the clinical practice. All authors reviewed the manuscript for important intellectual content and approved the final version to be published.Conceptualization: Gang Yang.Writing – original draft: Lei Yang.Writing – review & editing: Gang Yang.


Wunderlich syndrome is a rare clinical syndrome characterized by the sudden onset of spontaneous, nontraumatic hemorrhage into renal subcapsular and retroperitoneal region. We present the case of a 24-year-old hypertensive who presented with acute flank pain and was found to have perinephric hematoma. He was managed conservatively and the follow-up revealed complete resolution of the hematoma with no structural abnormality of kidney. His connective tissue disorder/vasculitis work up was also normal. Acute abdominal pain is a common presenting complaint in patients visiting the ED. Such cases often prove a diagnostic challenge to emergency physicians, especially if the patients are hemodynamically unstable, elderly, and women of childbearing age group . SpontanA 24-year-old male presented to our ED with complaints of right flank pain, with sudden onset, and rapid progression. The pain was localized to the right flank. There was no associated radiation of pain, fever, hematuria, or history of trauma. He also complained of two episodes of vomiting. He had been on regular medication for hypertension and chronic renal failure.Upon arrival, the patient was afebrile, blood pressure (BP) was 114/70, pulse rate was 88 beats/min, room air saturation 100%, and respiratory rate was 22 breaths/min. Systemic examination of the abdomen was soft with tenderness over the right lower quadrant, with a palpable mass over the renal angle. Another systemic examination was unremarkable. Blood investigations revealed hemoglobin (Hgb) 10.7 g/dL (baseline Hgb 13.7 g dL), creatinine 222 umol/L, urine microscopy blood 3+, and protein 3+  was performed, showing enlarged right kidney with perinephric fat stranding and isodense lesion in the perinephric space, showing a CT density of recent bleed [(45-50 Hounsfield unit (HU)] Figure .The patient was hospitalized for further workup and management and was started on analgesics and antibiotics. After two days of hospitalization, the patient underwent an MRI abdomen with contrast, showing a large perinephric hematoma (arrowhead) and a small hemorrhagic cyst (arrow) in the anterior renal cortex communicating with perinephric hematoma Figure .He was treated symptomatically. The patient was pain-free and hemodynamically stable. The urology team was consulted, who advised for conservative management and close observation. He did not have a further fall in Hgb or worsening of renal function. Further workup for other causes, like vasculitis, were negative.On the eighth day of admission, ultrasound imaging of the kidney revealed an estimate of 208 cc of perinephric fluid collection Figure . As the patient improved symptomatically, he was discharged with outpatient follow-up. Follow-up ultrasounds done in the first and third weeks after discharge demonstrated regression of hematoma. Follow-up ultrasound done after three months postdischarge demonstrated complete resolution of hematoma, and no evidence of malignancies was detected Figure .Wunderlich syndrome is a rare clinical syndrome characterized by a sudden onset of spontaneous, nontraumatic hemorrhage into the renal subcapsular and retroperitoneal region . This syClinical presentation is often characterized by a constellation of symptoms known as the Lenk’s triad -- acute onset flank pain, palpable flank mass, and hypovolemic shock . HoweverOther presentations include fever, nausea, vomiting, and hematuria. The most common underlying pathology in up to 60% of cases are neoplasms which mostly include renal angiomyolipoma and renal cell carcinoma . These nNo underlying etiology could be found in our patient, as seen in a few other reported Wunderlich syndrome cases. In our case, the initial presentation mimicked renal colic. Early diagnosis can be aided by accurate clinical assessment and appropriate imaging. The initial imaging of choice is an ultrasound abdomen, although abdominal CT has a 100% sensitivity -8.The MRI of the abdomen further confirmed our diagnosis. Exploratory laparotomy or nephrectomy is done for most patients. However, in our case, as no underlying malignancy was detected on initial CT, we chose to opt for conservative management with close clinical monitoring and follow-up imaging. This option can also be considered in stable patients with no evidence of malignancy on CT/MRI. This conservative approach can avoid the need for surgery.It would be prudent to remember Wunderlich syndrome as a differential diagnosis in patients presenting with unexplained abdominal pain to ED.


Duodenal Diaphragm in adults is very uncommon, caused by congenital and acquired changes. It is reported that acquired duodenal diaphragm is related to the long-term use of nonsteroidal anti-inflammatory drugs.We report an adult presentation of duodenal diaphragm in a 77-year-old woman, suffered from acute cholangitis and choledocholithiasis. She was performed endoscopic retrograde cholangiopancreatography (ERCP) procedure to remove the stone in common bile duct (CBD). After the stenosis ring dilated by endoscopic balloon dilatation, ERCP procedure was applied, and the CBD stone was removed successfully.Duodenal diaphragm is difficult to diagnose in clinic. Although the patient in this case had relatively mild symptoms of incomplete upper hemi-abdominal obstruction, these symptoms could be obscured by the emergency acute upper abdominal pain with fever as clinical manifestations of acute cholangitis. Duodenal diaphragm usually presents in infancy or childhood with congenital etiology, and it is rare in adults. We report an adult presentation of duodenal diaphragm in a 77-year-old Chinese woman, who underwent endoscopic retrograde cholangiopancreatography (ERCP) procedure to remove the stone in common bile duct (CBD). The occurrence of adult duodenal diaphragm in current case may associate with long-term application of nonsteroidal anti-inflammatory drugs (NSAIDs).9/L and serum amylase level was normal. Emergency abdominal CT scan represented that there was a stone in lower part of CBD and a muddy stone in gallbladder . She also had a history of progressive epigastric fullness for half a year, accompanied with bloating, nausea and occasional emesis after meals, with her body weight decreased for 7.5 kg. Recently, she had three episodes of acute abdominal pain, chills and fever. She have received norfloxacin orally at local clinic, without looking for professional help. Physical examinations revealed a febrile (body temperature 37.6 ℃), and extremely emaciated old woman weighed 34 kg (body mass index 14.5) without jaundice. Abdomen was soft with no tenderness or palpable mass. Her serum albumin level was 22 g/L, and she was anemic with hemoglobin 36 g/L; her white blood cell count was 15.9 × 10The next day of admission, the patient signed the consent form of ERCP and gastroscopy procedure, which did not contain the item of biopsy. Then the ERCP procedure was performed on her after her vital signs were stable. Unfortunately, this patient needed for the urgent intervention at that time, thus we did not have enough time to perform the differential diagnostic procedures. Gastroscopy showed an irregular ulceration in the anterior pyloric area Fig. b, and a 9/L.We inserted a yellow wire via the stenotic hole to explore whether it was a fistular passway to adjacent organs or the gut Fig. d. By usiWe concluded the final diagnosis as follows: acute cholangitis, choledocholithiasis, gastric ulcer, iron-deficiency anemia, duodenal diaphragm, hypoproteinemia, rheumatoid arthritis and severe malnutrition. Her gastric ulcer, severe anemia and malnutrition might be caused by long term NSAIDs ingestion and food retention due to the upper hemi-abdominal obstruction. Duodenal diaphragm may result from long term NSAIDs usage. We attempted to contact her for a 3-month follow-up after her discharge, but we received no response till now.The presence of duodenal membrane, which is an entity usually identified in infant or children with congenital etiology, is clinically characterized by postprandial vomits of acid and bilious food contents, accompanied by upper hemi-abdominal distention with visible peristalsis and borborygmus. Developmental delay is an important parameter, associated to several conditions such as Down syndrome, prematurity, situs inversus and coexisting extrinsic abnormalities –8.It is reported that acquired duodenal diaphragm is related to long-term use of NSAIDs . InitialNSAIDs could contribute to diaphragm-like strictures and ulceration in the small bowel and colon, which typically presents as anemia and bowel obstruction. Diaphragm disease was firstly described by Lang et al. . He repoWhile the symptoms and adverse events are well documented, the detailed pathogenesis of bowel diaphragms remains elusive at present. Going et al.  suggesteSeveral possible etiologies of duodenal strictures should be differentiated, including potassium-induced stricture or neoplastic, ischemic, inflammatory , and infectious  causes, all of which could be excluded by clinical and pathologic grounds.Many open or laparoscopic procedures have been applied in the surgical treatment of duodenal diaphragm, however, longitudinal duodenotomy with excision of the diaphragm and transverse closure of the duodenotomy is the most prevalent approach for congenital duodenal diaphragm (CDD) treatment , 5. WithAltogether, NSAIDs may cause numerous side effects, most seen in the gastrointestinal (GI) tract. NSAID-induced gastrointestinal ulcer is the main complication. Acquired duodenal diaphragm is a very rare malformation, it should be suspected in the cases of abdominal distension and malnutrition. Endoscopy could be used to identify the diagnosis. Endoscopic balloon dilation is a simple and effective method to treat this condition.


Introduction. Intra-abdominal testicular torsion is a rare event. We report hereby our experience of the management of a spermatic cord twist on intra-abdominal testis discovered during an acute surgical abdomen. Case Presentation. This was a 42-year-old patient admitted to the emergency department for abdominal pain that had been evolving for a week. The physical examination showed tenderness and guarding in the left iliac fossa with an empty ipsilateral hemiscrotum. Complementary examinations led to the discovery of an intra-abdominal left-lateral mass. The laparotomy found a whitish mass with areas of infarction, which was resected. Anatomopathological examination of the operative specimen identified it as a testis with atrophy of germ cells and necrotic areas without evidence of malignancy. Conclusion. Intra-abdominal testicular torsion should be considered in case of patients with an acute surgical abdomen with vacuity of one of the bursae. The cryptorchidism is defined as an undescended testis into the scrotum –5. It af3 and a normal hemoglobin level. Ultrasound revealed a heterogeneous left-vesical mass of 43 mm × 32 mm in diameter (βHCG (choriogonadotrophic hormone), αFP , and LDH (Lactodeshydrogenase), measured postoperatively were normal. Postoperative recovery was uneventful. The patient was discharged at postoperative day 5.A 42-year-old man, married with two children, was admitted to the emergency department for left iliac fossa pain radiating to the flank, which had been evolving for a week without fever. The pain was previously dull, little sensitive to the usual analgesics, with periods of remission. It progressively worsened. After initial care in a peripheral health facility without improvement, the patient was referred to the regional hospital. Clinical examination noted left iliac fossa tenderness with guarding, free hernial orifices, and a vacant left hemiscrotum with a right normal testis; the rectal examination found lateralized pain on the left side of the Douglas pouch. The rest of the somatic examination was unremarkable. The blood count indicated leukocytosis at 12,000 elements/mmdiameter . After adiameter . The peddiameter . ResectiIntra-abdominal testicular torsion is a rare event , 9, 10. It is well established that both cryptorchidism and testicular ectopia increase the risk of malignant degeneration. The risk of cancer is 35–40 times higher compared to the normal subject due to higher abdominal temperature than scrotal one , 9. IndeThe torsion of the testis in intra-abdominal position may be asymptomatic or may lead to nonspecific signs including irritability, nausea, and vomiting, constipation, atypical abdominal pain, hematuria, or even shock, which can be misdiagnosed as appendicitis or diverticulitis , 11, 12.The management of testicular torsion on an intra-abdominal testis remains controversial when the testis is viable; some authors advocate orchiectomy because of the high risk of malignant degeneration , 10, 15,In our Nigerien context with limited resources, the discovery of an intra-abdominal testis is often fortuitous, or in an acute surgical abdomen chart and in adulthood, an orchiectomy should be the treatment of choice, as recommended by experts .Intra-abdominal testicular torsion is rare and often presents as a misleading clinical picture. However, this diagnosis should be considered in a subject with acute abdominal pain with one or two vacant bursae. The orchiectomy remains the recommended treatment because of the significant risk of malignant degeneration that increases with age.


This is the case of a 47-year-old male who developed mild dyspnea on great exertion. Routine transthoracic echocardiography (TTE) revealed a slightly thickened mitral valve with anterior leaflet prolapsed and mild incompetence. There was an echogenic image suggestive of an irregular and mobile tumor adhered to the ventricular face of the anterior mitral valve leaflet, measuring about 17 mm by 10 mm. The movement of the tumor contributed to a dynamic increase in the left ventricular outflow gradient, estimated at 33 mmHg. The transesophageal echocardiogram (TEE) performed 14 days later showed prolapse of the anterior mitral valve leaflet, which presents a movable filamentary mass, measuring 13 mm by 3 mm, located at its end on the ventricular surface, and which protruded during systole via the left ventricular outlet, causing dynamic obstruction with peak systolic gradient of 59 mmHg. Doppler revealed mild to moderate reflux . SurgeryWhat are the diagnostic possibilities and their characteristics?Regarding imaging techniques, were they the most suitable for diagnosis?What was decisive for surgical treatment? Does the surgical indication depend on the presence of symptoms?Is there a need for anticoagulation and association of arrhythmias in this type of tumor?. However, approximately 3.2% to 46.4% of patients with CM are asymptomatic-6. CM is a rare benign cardiac tumor, and its incidence is observed in approximately 0.5-1 case per 1 million people per year-10. Among primary cardiac tumors, myxoma is responsible for 50% of cases, whose average age range varies from 42 to 66 years,12. QUESTION A. The main diagnostic possibilities would be cardiac myxoma (CM), chordae rupture, papillary fibroelastoma, vegetation, and the redundant cusp itself. CM should be suspected in patients with chronic dyspnea, history of peripheral or systemic embolization, acute neurological disorder, and family history of CM. However, the three-dimensional (3D) echocardiogram provides a better characterization and morphological classification according to the microscopic appearance of the surface of these intracardiac masses, with good correlation with surgical and histopathological findings, but the need for greater training of operators, cost, and high dependence on image quality currently limit the wide application of 3D in the oncology scenario. Studies have shown that two-dimensional and 3D techniques are equally reliable, and that 3D TEE has an advantage in locating the disease-18. QUESTION B. The echocardiogram is the main modality of diagnostic imaging in the morphological evaluation of intracardiac tumors. TTE is defined as a first-line imaging modality for assessing valve anatomy, severity of mitral regurgitation, hemodynamics, and ventricular consequences. When TTE does not add value to the diagnosis, TEE is always advocated as an enhancement for additional diagnosis-21. Due to malignant potentials, timely surgical resection is guaranteed in all patients, without contraindication when making the diagnosis, as the tumor's behavior cannot be predicted and does not depend on how large and fragile it is.QUESTION C. For symptomatic patients, surgical exeresis is the treatment of choice, always trying to preserve the valve tissue and its function. In asymptomatic individuals, surgical management is controversial, with tumor mobility being the determining factor for surgical indication, as it is an independent predictor of embolization and death. Embolization is more likely to occur in mobile papillary cardiac tumors, which are smaller in size, and in patients who have preoperative atrial fibrillation, and it is necessary to treat embolic events with anticoagulation to avoid serious events such as stroke, an embolism in the peripheral vasculature, or an embolism in the pulmonary artery, caused by detached tumor tissue or mobilization of thrombotic deposits. Usually the occurrence of arrhythmias, mainly extrasystoles and atrial fibrillation, is due to some obstruction of the physiological blood flow,24.QUESTION D. The follow-up of asymptomatic patients who do not undergo surgery should include echocardiographic monitoring and anticoagulation therapy, although its effectiveness in protecting against embolic phenomena is controversialPrimary cardiac tumors are rare, with a prevalence between 0.0017% and 0.19% of the unselected autopsy studies. About 75% are benign tumors and almost half are myxomas. The rest are divided among rhabdomyomas, lipomas, and fibroelastomas.Due to its predominant involvement in cardiac valves, nonspecific symptoms, and characteristics of its shape and size, the chordae rupture, papillary fibroelastoma, vegetation, and the redundant cusp itself have become suggestive in this clinical case.It is important to note that obstruction of the heart valve caused by CM, for example, can lead to catastrophic consequences, including sudden death. A fragment of the tumor can embolize to the left or right coronary artery ostium and result in acute coronary syndrome, and surgical resection has a low rate of complications and mortality. This detail was also important for the decision for surgical treatment.


Scar endometriosis is an uncommon type of extra-pelvic endometriosis. However, it should be suspected in any woman of childbearing age complaining of a cyclic, painful nodule in a scar from a previous obstetric or gynecologic procedure, after excluding other differential diagnoses. The treatment of choice is surgical resection. We report two cases of scar endometriosis that appeared in two young ladies after cesarean sections, discovered by a parietal mass near the cesarean scars. Endometriosis is defined by the presence of ectopic functional endometrial tissue outside of the uterine cavity . It is tWe report two cases with cesarean scar endometriosis and refer to a recent review of the literature to discuss all the symptoms and radiology exams that may lead to earlier diagnosis and treatment of abdominal scar endometriosis.Case report 1 Case 1 was a 44-year-old female patient, married, and the mother of one child who had, as a surgical antecedent, a cesarean section performed three years prior to presentation. She described a three-month history of the appearance of cyclic hypogastric pain and pain in the right iliac fossa without irradiation and without favoring or sedative factors. The physical examination found a parietal mass at the right iliac fossa measuring 4 cm that was mobile with no inflammatory signs. Ultrasonography showed a subcutaneous right paramedian mass measuring 7 x 3 cm attached to the rectus abdominis muscle that was well-defined, hypoechoic, and heterogeneous with an arteriovenous vascularization on a color Doppler study. A complementary computed tomography (CT) scan with intravenous contrast reported a mass in the rectus abdominis muscle with spontaneously hypodense contents, enhancing heterogeneously, measuring 7 x 3 cm , we decided to perform a transfixing excision of the parietal mass, including a cutaneous flap, and performed an omentoplasty along the path of the biopsy with the repair of the fascial wound by a nonabsorbable plaque. The procedure went well, and the patient experienced no pain following the procedure. The histology results confirmed the diagnosis of parietal endometriosis. The patient experienced no recurrence over the next four years of follow-up.Case report 2A 31-year-old female patient, married and the mother of two children, presented with complaints of a painful hypogastric mass next to a cesarean section scar. Her past medical and surgical histories were negative, except that she had undergone two cesarean sections and the mass had appeared two years after her last cesarean section. Physical examination revealed a firm, tender, subcutaneous mass on the left side of the Pfannenstiel incision measuring 2 x 3 cm. A pelvic magnetic resonance imaging (MRI) scan with intravenous contrast revealed evidence of a subcutaneous fat mass of the left lateral pelvic wall, roughly rounded with spiculated contours, measuring 28 x 23 mm, and showing heterogeneous enhancement and 6% in the premenopausal age group . The patThere are two other theories:- The coelomic metaplasia theory by Meyer suggests that metaplasia of the coelomic epithelium was the origin of endometriosis . This th- The metastatic theory suggests that distant lesions are established by the hematogenous or lymphogenous spread of viable endometrial cells which logically explains the rare endometriotic lesions distant from the uterus . The ectopic endometrial cells stimulated by the osteoprotegerin will escape apoptosis, adhere to the underlying peritoneum, generate a new vascular supply, evade the immune surveillance system, and proliferate to build an endometriotic lesion. An increase in the concentration of hepatocyte growth factor and a decrease in the concentration of interferon gamma-inducible protein-10 in the peritoneal fluid of women with endometriosis may stimulate angiogenesis and the development of endometriosis . The mosThe manifestations of endometriosis are various in the form of dysmenorrhea, chronic pelvic pain, dyspareunia, and a mass in the abdominal incision during menses . AbdominThe most common treatment options for scar endometriosis include medical therapy and surgery . SurgicaMedical therapy has a low success rate and is associated with adverse effects. Based on a retrospective review of abdominal wall endometriosis made in China between 2003 and 2011, 63 of 229 patients diagnosed with abdominal wall endometriosis took hormonal treatment, such as oral contraceptives, gonadotropin-releasing hormone agonist (GnRHa), danazol, or progesterone. These drugs offer only a temporary alleviation of symptoms that are often followed by recurrence after cessation of drug intake . Local rBased on implantation theory, there are strategies for preventing the abdominal wall endometriosis, such as lifting the uterus outside of the pelvis before making the uterine incision, using separate needles for the uterine and abdominal closure, removing a functional corpus luteum at the time of the hysterectomy, using high-pressure irrigation, not using a sponge to clean the endometrial cavity, and suturing the uterine incision without endometrium . The strDespite the rarity of parietal endometriosis pathology, every surgeon must consider it when he is in front of a patient of reproductive age with a history of obstetric or gynecologic surgery and who has suffered from a parietal mass, infertility, or chronic pain. Non-invasive imaging modalities, like ultrasonography, CT scan, and MRI, are helpful to determine the location and extension of the masses, but they are unsatisfactory for confirming a diagnosis. Only the anatomopathological results will allow one to recognize endometriosis. Surgical treatment consists of the total excision of the mass and is still the gold standard treatment with a very low rate of recurrence if done properly. To conclude, awareness of the schedules and all the preventive rules in all gyneco-obstetrical surgery is the most important way to eradicate this disease.


Bilateral objective tinnitus is a rare accompanying manifestation of an underlying palatal tremor (PT). PT can either be secondary to a lesion in the triangle of Guillain and Mollaret, or it can present without a causal organic lesion. In this report, we present an unusual case of a young female with objective tinnitus revealing a PT. Tinnitus is a common otologic symptom and is defined as the conscious awareness of a sound in the absence of an external auditory stimulus . It can PT, previously known as palatal myoclonus, is a rare movement disorder presenting as continuous rhythmic spasms of the palatal muscles. These involuntary contractions present as “clicking” sensations in the ears . PT occuA 20-year-old female presented to our outpatient department complaining of intractable “clicking” tinnitus for the last two years. She had been in her usual state of health two years back when she had experienced an upper respiratory tract infection (URTI) for which she had taken antibiotics for one week, which had led to the resolution of the infection. One week after the resolution of her symptoms and discontinuation of antibiotics, she had developed clicking in both ears. She reported her symptoms to be persistent and bilateral (left > right). She denied any history of ongoing headache, blurring of vision, dysphagia, dysarthria, fever, weight loss, or any other neurological or auditory symptoms.An otologic examination revealed normal tympanic membranes and hearing was normal on audiological testing (pure tone and impedance audiometry). There were continuous, involuntary rhythmic contractions of the soft palate Video , and theShe had no significant past medical or family history. The contractions persisted during phonation and sleep. Her tinnitus was refractory to treatment with various antidepressants, antiepileptics, skeletal muscle relaxants, and physiotherapy. She underwent an MRI of the brain, which revealed no notable lesions. She has been advised to undergo a trial of botulinum toxin therapy and is currently awaiting the procedure.In this report, we presented a case of essential PT in a 20-year-old Asian female. The most recognized symptom to be reported with an underlying PT is tinnitus, often described by the patient as a clicking sound that is synchronous with the soft palate contractions when examined orally or nasendoscopically. Clicking tinnitus is a rare otologic symptom that has been attributed to various causes, such as a temporomandibular joint click, a patulous eustachian tube, middle ear myoclonus, and PT. Pulec and Simonton in 1961 proposed that the click sound is produced when there is a breaking of surface tension as the wall of the eustachian tube snaps open and shuts, mainly caused by a forceful contraction of the levator veli palatini or the tensor veli palatini .East et al.  conducteThe objective tinnitus of our patient persisted during sleep, which is a feature of symptomatic PT, which is often neurological in origin. However, due to the absence of any recognizable organic cause, it was labeled as essential palatal myoclonus. Nonetheless, one plausible explanation was that the tinnitus of our patient proceeded from an undiagnosed URTI. Björk  has repoVarious treatment options have been described for this condition. For essential PT, some of the recommended treatment options are anticonvulsants such as sodium valproate, carbamazepine, or clonazepam, and intra-lesional injections of therapeutic botulinum neuromuscular junction toxin into the insertion of the levator and/or tensor veli palatini muscles . ConversThis case report highlights the phenomenon of PT occurring in a patient after she has experienced a URTI, thereby implying a possible association between the two conditions. PT, though rare, should be part of the differential when clinicians encounter cases of bilateral objective tinnitus so that a prompt diagnosis can be made and patients can be offered appropriate treatment.


Rosai-Dorfman disease (RDD) is a rare medical condition with bilateral painless lymphadenopathy. We present the case of a young man diagnosed with a very unique presentation of Rosai-Dorfman disease.A 40-year-old African-American man presented with a firm, non-tender, progressive chest and neck mass appeared three months ago. Imaging of the neck demonstrated an 8.6-cm anterior neck subcutaneous soft tissue mass extending into the anterior mediastinum through the sternum with erosive changes in the sternum and the lesion is abutting the right common carotid artery and innominate vein and surrounds the medial aspect of the clavicles bilaterally. Ultrasound (US)-guided biopsy showed marked polytypic-appearing plasma cell proliferation associated with relatively prominent histiocytes with hemophagocytosis/emperipolesis and focal neutrophils. There were S100+ histiocytes; however, findings were not typical for RDD. As that biopsy was not diagnostic, incisional biopsy with adequate sampling was performed. Surgical pathology demonstrated a very abnormal infiltrate with prominent histiocytes including areas with the features of extranodal RDD. BRAF V600E immunohistochemistry (IHC) was negative. Modified radical neck dissection, proximal sternal resection and superior mediastinal nodal dissection surgery was recommended. However, the patient refused the procedure.Typical manifestations are lymphadenopathy with fever that our patient did not experience. Bone involvement happens in 5-10% of cases. There is not enough data about blood vessel invasion which make our case unique. Treatment plan is still controversial. Clinical monitoring is recommended if the symptoms are tolerable as regression has been reported in many cases (20-50%). Surgery is reserved for patients with vital organ involvement or extra-nodal disease. Rosai-Dorfman disease (RDD) is a rare benign medical condition that primarily manifests with bilateral painless lymphadenopathy, fever, and neutrophilia in adolescents and young adults. Concomitant lymphadenopathy with extranodal involvement occurs in 43% of cases. Isolated extranodal involvement is reported in 23% of patients, most commonly in the area of the head and neck . InvasioA 40-year-old African-American man presented with a firm, non-tender, upper chest and neck mass of sudden-onset three months ago that had been slowly growing since. He denied difficulty swallowing, vocal hoarseness, neck or chest pain and shortness of breath. He reported intentional weight loss of 30 lbs over the past three months. Computed tomography (CT) scan of the anterior neck demonstrated an 8.6-cm subcutaneous soft tissue mass extending into the anterior mediastinum through the sternum, which demonstrated erosive changes Figure . The lesUltrasound (US)-guided biopsy showed proliferation of marked polytypic-appearing plasma cells associated with prominent histiocytes, with hemophagocytosis/emperipolesis and focal neutrophils. These histological characteristics were partially compatible with Rosai-Dorfman disease, especially in accordance with the presence of S100+ histiocytes. However, these findings were atypical, and the architectural pattern could not be definitively assessed due to limited biopsy sizes. In order to make a proper determination, an incisional biopsy was performed. Surgical pathology showed an abnormal cellular infiltrate with prominent histiocytes, including areas with demonstrable features of extranodal sinus histiocytosis with massive lymphadenopathy Figure .In addition, there was discernible sclerosis, signs of acute inflammation, and a benign-appearing lymphoplasmacytic component. Many of these features suggest the mass represents extranodal Rosai-Dorfman disease. On immunohistochemistry, B-raf V600E was reported negative. Modified radical neck dissection, proximal sternal resection, and superior mediastinal nodal dissection surgery was suggested. However, the patient refused the surgery and did not receive any treatment for about six months.Rosai-Dorfman disease is a rare medical condition that is clinically characterized by bilateral painless cervical lymphadenopathy. Histologically, it is typified by alpha-1-anti-chymotrypsin+, S-100+, CD68+, and CD1a- histiocytes; this immunostaining pattern may help differentiate RDD from Langerhans cell histiocytosis and Erdheim-Chester disease . Rosai-DExtranodal manifestations of Rosai-Dorfman disease make up between 25-40% of cases and may present a diagnostic challenge to practicing clinicians; isolated extranodal RDD commonly mimics neoplastic entities in a variety of organ systems. The most frequent sites of extranodal involvement are the skin, bone, central nervous system, upper respiratory tract, and retro-orbital tissue . Our patThe pathogenesis of sinus histiocytosis with massive lymphadenopathy is not entirely clear. Recent studies have pointed to the role of intermediate recruiting monocytes, and alterations in their differentiation, in the development of RDD . In addiBiopsy is the most important tool in the definitive diagnosis of RDD . PatholoTreatment plan is still controversial. Clinical monitoring is recommended if the symptoms are tolerable, as regression has been reported in many cases 20-50%) . Cryothe0% . CryoThis case report reviewed an unusual presentation of Rosai-Dorfman disease. Invasion of the major vessels is exceedingly rare. Treatment of Rosai-Dorfman disease is controversial. Given the involvement of the right carotid artery in the presented case, surgery was offered as the best option.


Cutaneous myiasis is a condition that involves an infestation of fly larvae into human tissue, most commonly caused by Dermatobia (D.) hominis or the bot fly. While this is a condition most commonly seen in tropical regions of the globe due to increased travel to endemic regions, physicians must increasingly be aware of this as a potential diagnosis. In addition, there is minimal literature on cutaneous myiasis in the pediatric patient and its potential associated symptoms. This case report thus highlights a toddler that presented to our facility with a raised, erythematous scalp lesion and associated preauricular and occipital lymphadenopathy. Of note, the patient had a recent travel history to Belize, an endemic area where she was likely infected. As there are multiple other differentials for not only scalp swellings in the pediatric population, in addition to regional adenopathy, a high index of suspicion was needed to make the diagnosis. Ultrasound was the imaging modality used to visualize the fly larva, and surgical excision was the mechanism of treatment. Thus, this case highlights a unique presentation of cutaneous myiasis in a toddler and aims to add to the growing body of literature on a condition likely to be encountered by physicians at a greater frequency. Cutaneous myiasis is described as the infestation of human tissue by dipterous fly larvae -2. DermaA three-year-old female with a history of recent travel history to Belize and Guatemala presented with swelling and pruritus of the occipital scalp two weeks after returning to the United States. She was seen in the emergency room of a local hospital. During her trip, the patient, accompanied by her family, visited many forests, lakes, rivers, and sulfur hot springs. While in those countries, her mother reported multiple mosquito bites despite taking protective measures. The patient’s grandmother had similar localized swelling and pain on the lower extremity, which was incised and drained at the emergency department at an outside institution. The emergency department physician reported extracting an unknown larva from her grandmother’s wound and recommended that the patient be evaluated due to similar symptoms. At the emergency department of an outside facility, physical examination was remarkable for a 1x1 cm occipital scalp swelling associated with associated pruritus. Unsuccessful incision and drainage were performed and the patient was then transferred to our pediatric inpatient unit for further management.On presentation, the physical examination was significant for a 1 cm x 1 cm open laceration on the posterior scalp with surrounding swelling and erythema; no fluctuance or drainage of the wound was appreciated. The patient also presented with left-sided postauricular lymphadenopathy and bilateral occipital lymphadenopathy with the largest measuring 1x1 cm, firm, non-tender to palpation. Ultrasound was done to rule out other causes of scalp swelling  and to confirm the presence of a foreign body. Ultrasound results showed an echogenic structure in the subcutaneous tissue measuring 8x5x3 mm, which may represent the foreign body Figure -3.A layer of thick petroleum jelly was applied to the site, occluding the pore to deprive the larva of air. Surgery was then consulted for incision and drainage of the swelling. Under local anesthesia, a 3 cm x 1 cm incision was made on the skin of the posterior scalp; exploration of the wound was done to identify and remove the foreign body , which confirmed the diagnosis Figure .Among pediatric patients, case reports of myiasis caused by the human bot fly (D. hominis) are very rare; an extensive literature review revealed only around 10 cases reported worldwide. As in our patient, the most common site of bot fly myiasis is the scalp, a region not typically covered and amenable to the inciting insect bite required for inoculation -10.Exposure to D. hominis is almost exclusively found in those with a history of travel to South or Central America ,11. No dPatients with cutaneous myiasis typically present due to a raised papule with a central, necrotic opening and surrounding erythema ,8,11. PaOn examination, a mass underlying the lesion may be felt and many patients report the sensation of something moving beneath the skin -15. BesiAs it pertains to our patient, the presentation was unique in that the lesion was located on the occipital scalp, with accompanying multiple post-auricular and occipital lymphadenopathy. To our knowledge, this specific distribution of lymphadenopathy secondary to bot fly infection has not previously been reported in the literature. Postauricular lymphadenopathy is most commonly noted in bacterial or fungal infections of the parieto-temporal scalp, in addition to infections caused by rubella and roseola. Occipital lymphadenopathy in the pediatric population more commonly presents with localized infections such as tenia capitis, head lice, or impetigo . While tOnce suspicion of bot fly infection exists, imaging modalities may be used to identify the presence of the larva beneath the skin. While computed tomography (CT) and magnetic resonance imaging (MRI) have been used in identification, recent studies indicate that ultrasonography is a superior imaging modality -10. UltrOnce identified, the treatment of myiasis consists of extracting the botfly larvae. As most physicians in the United States are unfamiliar with myiasis, surgical extraction, as was done in our patient, is the most commonly used means of resolving bot fly infections reported in the literature . Simple Following the customs of residents of botfly endemic regions, botfly larvae may alternatively be removed without the need for invasive surgical intervention . Simple After surgical extraction, the wound should always be thoroughly cleaned, debrided of any necrotic tissue, and closed. The role of treatment with antibiotics after extraction is debated but does not appear to be routinely necessary . Rarely,The diagnosis of cutaneous myiasis is important to consider in pediatric patients presenting with cutaneous lesions similar in appearance to an abscess, with a history of recent travel to bot fly endemic areas. Generally, clinical suspicion for myiasis in pediatric patients in the United States is low because: (i) The botfly is not an endemic species, and without a specific history of travel to an endemic area , probability of exposure is almost non-existent, (ii) Presentation varies significantly in the size and location of lesions and associated symptoms, and (iii) Based on the literature review, the condition in children appears to be much rarer than in adults.Cutaneous myiasis secondary to bot fly infection is a rare clinical entity in the United States and, as such, is not typically included in the list of differentials for superficial cutaneous lesions in pediatric patients. Ultrasonography is recommended in identifying the presence of bot fly larvae, regardless of their size or stage of development. Although this condition can be easily treated via extraction of the larva through surgical means, increased knowledge of the condition demonstrates that non-surgical extraction is technically simple and prevents unnecessary invasive procedures, with a definitive cure. Although it may represent a rare clinical entity in any patient presenting with a cutaneous lesion of the scalp, with associated regional lymphadenopathy, myiasis should be included in the differential diagnosis. Overall, thorough travel history and a high index of clinical suspicion is necessary to prevent an unnecessary delay in diagnosis, with the continuation of lancinating pain and irritation, and undue frustration on behalf of the family.


Although duodenal neuroendocrine neoplasms (DuNENs) usually have indolent phenotypes, some DuNENs exhibit aggressive clinical manifestations. Tumor size > 1 cm, lymph node metastasis, and high grade have been associated with poor prognosis. However, preoperative risk evaluation is often difficult, because Ki-67 index on biopsy is frequently underestimated due to the intratumor heterogeneity. Here, we present a case of a subcentimeter DuNEN with a low Ki-67 index on endoscopic biopsy, who developed lymph node metastasis and high-grade liver metastasis.The patient was a 52-year-old female who presented an epigastric pain. Esophagogastroduodenoscopy revealed a duodenal submucosal lesion with a size of 8 mm. The endoscopic biopsy showed DuNEN with a Ki-67 index of 3.3% . We performed an open partial duodenectomy with adjacent lymph node dissection. Pathological examination of the resected specimens revealed a Ki-67 index of 13.5% (G2) in the “hot spot” and lymph node metastasis. A hepatic low-density area detected on preoperative contrast-enhanced computed tomography appeared to be a liver metastasis on postoperative gadoxetic acid-enhanced magnetic resonance imaging. Subsequently, we performed a laparoscopic partial hepatectomy. Pathological examination of the liver specimen showed a metastatic neuroendocrine tumor with a Ki-67 index of 27.5% (NET-G3). The patient has been alive for 14 months since the hepatectomy.This case shows the possibility of high malignant potential of DuNEN even if the primary lesion is < 1 cm and has a low Ki-67 index on biopsy. Neuroendocrine neoplasm (NEN) is a rare disease with an age-adjusted annual incidence of 6.98 per 100,000 population in the United States . AlthougThe patient was a 52-year-old female who presented an epigastric pain persisting for 6 months. Esophagogastroduodenoscopy showed a small submucosal lesion in the anterior wall of the duodenal bulb Fig. a. EndoscBecause the malignant potential was higher than expected on preoperative workup, the hepatic low-density area detected on preoperative CE-CT Fig. a, b was The patient was observed without adjuvant therapy, but multiple liver metastases appeared 1 year after the hepatectomy Fig. a–d. TherG1/G2 on preoperative biopsy, whereas the finding of EUS suggested slight invasion of the muscularis propria. Collectively, these preoperative findings did not strongly suggest high malignant potential. Hence, we did not emphasize the significance of the hepatic low-density area appreciated on preoperative CE-CT, and did not examine the patient by somatostatin receptor scintigraphy. However, the pathology showed that the primary DuNEN actually exhibited an infiltrative pattern extensively involving the muscularis propria, as shown in Fig. Although DuNENs usually have indolent phenotypes, tumor size > 1 cm, invasion of the muscularis propria, lymph node metastasis, and high grade have been associated with poor prognosis , 4, 9. TNotably, the present case exhibited a grade increase to G3 in the liver metastasis. This might be the result that the high-proliferative cells in the invasive front of the primary DuNEN metastasized to the liver. In a retrospective study from Italy including 108 DuNENs, G3 accounted for 4% and was associated with 30-fold increased risk of death and progression compared to G1 . NET-G3 We extensively searched previous case reports on subcentimeter primary gastrointestinal NET-G1/G2 which upgraded to G3 in the liver metastasis, and found one such case of rectal origin . In thatAlthough the present case showed a subcentimeter, marginal G1/G2 DuNEN on preoperative workup, the tumor did exhibit extensive infiltration and high proliferative activity in the invasive front and was upgraded to G3 in the liver metastasis. The present case alerts to the possible malignant potential of DuNEN even if the preoperative workup shows benign findings.


We present an extremely rare combination of biventricular outflow obstruction associated with atrioventricular septal defects and patent ductus arteriosus (PDA). Almost all the other published cases, including ours, were associated with other congenital cardiac lesions other than biventricular outflow obstruction. Most cases ended with poor outcomes. Our patient was a 55-day-old term female infant. She was managed by successful aortic balloon valvuloplasty with successful early outcome. Congenital heart diseases (CHDs) are common in infancy, accounting for one third of all congenital disease . In SaudThis article describes a case of a female infant patient who was diagnosed with biventricular outflow obstruction associated with VSD, atrial septal defects (ASD), and patent ductus arteriosus (PDA). To the best of our knowledge, this combination is unique in the medical literature. In this article, we are highlighting the importance of the accurate diagnosis of all described lesions, awareness of peculiar hemodynamic findings, and the importance of early intervention to avoid hemodynamic compromise. Failure to do so has the potential to lead to tragic outcomes .A 55-day-old term female infant with low birthweight (2 kg) was transferred to our tertiary cardiac center with a diagnosis of heart failure associated with a large VSD, moderate ASD, BAV, moderate PDA, and mild mitral regurgitation (MR). Upon arrival, the physical examination showed a nondysmorphic infant who was slightly tachypneic on nasal oxygen. Feeding was administered by a nasogastric tube. On cardiac examination, there was visible apical pulsation, no precordial bulge, and left parasternal thrill. On auscultation, the first and second heart sounds were normal. Grade 4/6 ejection systolic murmur was heard in the second and third left intercostal spaces, which were propagated all over the precordium.The echocardiogram showed Figures -4 a larg Left ventriculography was performed as part of cardiac catheterization and revealed a 4.2-mm aortic valve annulus. Balloon valvuloplasty of the aortic valve, a catheter-based intervention  was carried out Figure . SuccessPostcardiac catheterization, she was extubated, hemodynamically stable and without heart failure, then transferred to the pediatrics ward. Two days later, she was transferred to the pediatric ICU and was re-intubated due to lung collapse. Two weeks later, the patient’s condition deteriorated. There was bleeding from the mouth and in the endotracheal tube suggesting acute lung injury. During this period of respiratory compromise, the cardiac parameters were normal. Despite aggressive management by adrenaline, cryoprecipitate, and factor 7 replacement, the patient developed severe bradycardia, desaturation, and hypotension. After 30 min of cardiopulmonary resuscitation, which failed to revive her, the patient was declared dead.Biventricular outflow tract obstruction is rarely described in the medical literature; this diagnosis has been made in only 48 patients . RichterThe age of presentation in the 48 cases varied, ranging from the prenatal period to 55 years. It was speculated that an early presentation indicated severe disease. Almost all the other published cases in the literature were associated with congenital cardiac lesions: ASD was present in nine cases , 13-14, An evaluation was performed of the management of biventricular stenosis in the literature to identify the most optimal approach. Most cases published in the last century are characterized by poor outcomes, especially those prior to the era of catheter-based interventions. Specific management guidelines are not available in the medical literature due to the rarity of this condition. Clearly, evidence-based interventions rely on published case reports. Of the 48 cases, only nine of them were managed by balloon valvuloplasty. Ours was the 10th published case. In the current study, pulmonary valvuloplasty was not performed owing to the presence of a significant left-to-right shunt at three levels: ventricular, atrial, and great levels. The presence of native pulmonary valve stenosis is considered to be protective against pulmonary circulation overflow. Aortic valve PG dropped from 65 to 22 mmHg. This was accompanied by a significant improvement in the cardiac hemodynamic parameters and the patient’s general condition. The planned next step was to perform total repair: septal defects closure, PDA ligation, and pulmonary valvotomy. The patient's condition deteriorated because of sepsis with Gram-negative bacilli (Escherichia coli) despite the remarkable stable cardiac parameters. The current scenario is a strong indicator, for future cases to perform a meticulous search for associated extracardiac and systematic involvement. In our case, the deterioration in the infant’s condition is mainly attributed to sepsis, liver failure with increasing liver enzymes and deteriorated coagulation profile followed by pulmonary hemorrhage, and respiratory failure terminated by disseminated intravascular coagulation. Extracardiac involvement is significantly associated with CHD. This association is more pronounced with an increase in CHD complexity. Comparative studies carried out in our region on 2,020 cases, documented the involvement of other systems in one-third of CHD cases versus 10% in the controls. Anomalies of the central nervous system, hematological system, and gastrointestinal system were the most frequent associated extracardiac anomalies -18. MetiBiventricular outflow obstruction is rarely described. Of the 48 published cases, only nine of them were managed by balloon valvuloplasty. Ours was the 10th published case. To the best of our knowledge, this combination of biventricular outflow obstruction ASD, VSD, and PDA were first described. Future cases should be recognized early with meticulous search for extracardiac anomalies to avoid tragic hemodynamic outcomes.


Dermoid cysts usually occur later in the second decade of life; we present the approach of an unusual case of an infant who presented a cyst within the oral cavity, which is important because it can be confused with other pathologies. Dermoid cysts usually occur later in the second decade of life; we present the approach of an unusual case of an infant who presented a cyst within the oral cavity, which is important because it can be confused with other pathologies. It was referred from the pediatric service because she presented swallowing difficulties and an increased volume in the right submandibular region and on the floor of mouth with seven months of evolution Figure . On clinAn ultrasound was requested, in which we identified the right submandibular gland and adjacent to it, we observed an ovoid lesion, moving upwards and laterally with well‐defined edges and a thin wall. The interior showed heterogenicity with multiple small points. Afterward, we then requested the Doppler mode to assess the blood flow and showed abnormal vascular flow in the lesion. Later, an aspiration was performed, obtaining abundant material with cheese‐type appearance Figure .The surgical excision was made under general anesthesia with nasal intubation, while using an incision on the floor of the mouth Figure  and closThe histological examination confirmed a diagnosis of dermoid cysts Figure . The cys3An initial search was conducted by PubMed and Google Scholar through 2000 to 2019, based on a combination of the following medical subject headings: Dermoid, Cyst, Floor of the mouth. The inclusion criteria for the election of the reported cases were that they contain a histological study report which described the characteristics of the dermoid cysts (cystic lesion with stratified keratinized epithelium surrounded by a capsule of connective tissue containing skin appendages). The search produced a total of 52 articles, the patient´s ages when the lesion on the floor was detected were from newborn to 59 years old. When we categorized the patient´s ages, it is most often presented in the first three decades of life , 40% in females and 60% in males. But, after 30 years, the highest incidence is among men, 84.61%, and only 15.38% among women , the cyst was enucleated through intraoral approach; and 18 cysts were (30.5%) eliminated by an extraoral approach. In two cases, it was used as a mixed approach4The dermoid cysts of the floor on the mouth are rare lesions that originate more frequently in the first decades of life, and the case we present was detected and treated surgically in a female infant of 13 months. Schwanke et alThe differential diagnosis is made with ranula, mucocele, and Warthon duct obstruction, as well as congenital malformations of the floor of the mouth, such as hemangiolymphangiomas, thyroglossal duct cysts, branchial cysts, and ectopic thyroid tissue.Otherwise, SeahIn the case presented here, the lesion was diagnosed by ultrasonography and fine canula aspiration with the initial diagnosis of development cysts, which was confirmed by the histopathological study that also defined the type of cyst which according to the presence of sebaceous glands in the connective tissue capsule was diagnosed as a dermoid cyst, and the treatment employed in this case was an intraoral approach because it was above the geniohyoid muscle and an easy access. Some authors have mentioned that an intraoral approach is ideal because it gives a good view of the cyst, an easy access as well as esthetic results, even when the cyst is above the geniohyoid muscle.None declared.MEVC, CGLE, LAMP, and JJTF: examined and treated the patient, and wrote and edited the manuscript; MEVC and AGM: conceived and lead the idea for the case report; JFGC, CGTI, AGM, and MFRC: contributed to the design and editing the manuscript.


Pilonidal sinus is commonly seen at the sacrococcygeal region and few other sites, usually located at the midline, at areas where hairs collect or near protuberances or some adjacent rubbing surfaces. Its presence elsewhere is uncommon. We share an interesting case of a recurrent discharging sinus from the cheek bulge of a male which turned out to be a pilonidal sinus containing tuft of hairs on exploration and wide excision.A 37 years old hirsute male presents to us with a non-healing discharging sinus at the bulge of the cheek. Exploration after a course of antibiotics showed 2 subcutaneous cavities with tuft of hairs. The area was excised along with a margin, thorough irrigation and curettage was done and the wound was closed primarily; a Z-plasty was incorporated in the central part to break the resultant suture line. Histopathological examination was done to confirm the diagnosis and rule out an off-midline dermoid cyst or an underlying/coexisting malignancy. Post-operative course was uneventful. The patient has been recurrence free for 1.5 years and is satisfied with the nature of the scar.Pilonidal sinus of the cheek bulge is an extremely rare entity. Complete excision and clinical suspicion are important for cure of this nagging ailment, especially at unexpected areas. Pilonidal sinus (PNS) refers to a subcutaneous sinus which contains hair. It is an acquired condition caused by penetration of hair fragments into the skin. This hair containing cavity usually is asymptomatic and the only sign of its presence may be a small pit on the surface of the skin , 2. Due PNS is a relatively common condition mostly seen in hirsute males at the sacrococcygeal region in approximately 26/100,000 population .We present a case of a recurrent discharging sinus of the cheek which turned out to be a pilonidal sinus pathologically and not a dermoid, tubercular or a malignant cyst.On February 2019, a 36 year old hirsute male presented to us with a recurrent  discharging sinus over the right cheek which was operated twice previously. He did not have tooth ache or pain while moving the upper and the lower jaws. There was no significant medical or psychosocial history and his family support was considered satisfactory. There was no history of Tuberculosis (TB); similarly history of insect bite or pricks over the area was lacking. On examination, the right cheek contained a fairly good amount of normal looking hair and showed some signs of inflammation at the preauricular area around a small opening. The discharge from the opening was turbid, non-bloody, slightly foul smelling and did not contain any sebum, hair or yellowish granules. A provisional diagnosis of infected dermoid cyst was made and the patient discharged home with anti-inflammatory drugs and a 7 days oral course of Flucloxacillin and Metronidazole after radiologically ruling out Temperomandibular joint (TMJ), gums, teeth, parotid or maxilla/mandible as a potential underlying source of infection. The patient was planned for excisional biopsy with 2 mm margin after a week of local infection control  interconnected sinus cavities containing 2 rolls of hair and 2 tracks leading to the skin  may mimic Pilonidal Sinus , or moreIn the present case, there was a surgical history of repeated drainage of a cheek swelling about 3 months back which might be the cause of inadvertent entry of hairs in the wound. This cyst may have been misdiagnosed as an abscess and operated twice, which was not the definitive surgery. So the patient continued having discharge from the operative site and was finally diagnosed as pilonidal sinus after the last operation. The patient is satisfied with both the scar as well as the outcome of the surgery.The standard method of management of facial PNS is excision and primary closure which is usually performed under general or local anesthesia. This may differentiate facial PNS from PNS of other areas where many non-surgical modalities exists. The skin of the face is more lax allowing direct closure which may also eliminate a crease or groove. Leaving a wound over the cheek for secondary closure or having an underlying dead space results not only in a cosmetically inferior scar but also increases the chances of a recurrence. Adequately excised PNS of the face usually do not recur ; howeverNo preoperative pictures were available as the sinus was initially considered trivial and operation planned under local anesthesia as last case of the day (fear of infection and contaminating other cases). Patient was not physically available for late follow up and quantification of scar and satisfaction outcome was not done.Although PNS is very rare in the cheek bulge, it should be included in the differential diagnosis of subcutaneous nodule and chronic sinus when hair is seen inside the cyst cavity or when there is history of some surgical intervention. Wide excision of the sinus tract with a margin and primary or a delayed primary closure is recommended for a better cosmetic outcome.


Dedifferentiated liposarcoma is a rare, highly malignant, rapidly recurring tumor. When found in the spermatic cord, this tumor may deceptively present as an inguinal hernia, and due to its rarity, may not be readily identified.We report a case of a 46-year old man with dedifferentiated liposarcoma presenting as a slow-growing right inguinal bulge. His surgical management included ipsilateral orchiectomy and excision with wide margins. He received adjuvant focused radiation therapy.Liposarcoma of the Spermatic Cord is a rare and highly malignant tumor that rapidly recurs. Therefore, a high degree of suspicion, regular surveillance and imaging are justified. The incidence of malignant spermatic cord tumors is 0.3 cases per million, and of those, the most common type is liposarcoma . AlthougThis work has been reported in line with the SCARE criteria 2The patient is a 46-year-old male nonsmoker with no other medical problems. Two years prior to his current presentation, the patient was evaluated for right lower quadrant pain. Computed tomography (CT) image showed acute epiploic appendicitis of the cecum and a small fat-containing right inguinal hernia. No further treatment was pursued at that time. He had persistent symptoms, and two years later his primary care provider ordered an ultrasound, which showed a 3 × 3.4 × 3.6 cm complex heterogeneous mass superior to the bladder. Repeat CT scan confirmed a fat-containing right inguinal hernia and a solid mass just below the right inguinal canal, concerning for liposarcoma versus inflammatory process . The pat3Liposarcoma of the spermatic cord is a rare cause of inguinal hernia (0.1%), and most tumors of less than ten centimeters are found incidentally and not diagnosed preoperatively . In gene4Liposarcoma of the Spermatic Cord is a rare and highly malignant tumor that rapidly recurs. Therefore, a high degree of suspicion, regular surveillance and imaging are justified.Mr. Eric Wetzel or any member of his or her immediate family, has no funding or commercial associations  that might pose a conflict of interest in connection with the submitted article.Dr. Norair Adjamia or any member of his or her immediate family, has no funding or commercial associations  that might pose a conflict of interest in connection with the submitted article.Dr. Graal Diaz, or any member of his or her immediate family, has no funding or commercial associations  that might pose a conflict of interest in connection with the submitted article.Dr. Shawn Steen, or any member of his or her immediate family, has no funding or commercial associations  that might pose a conflict of interest in connection with the submitted article.Ms. Janet Hobbs, or any member of his or her immediate family, has no funding or commercial associations  that might pose a conflict of interest in connection with the submitted article.The authors have no source of funding to disclose.There is no ethical approval was obtained as it’s a case report but a written consent was taken from the patient.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.Eric Wetzel: study concept, design, writing the paper.Norair Adjamian: data collection, data analysis, interpretation, writing the paper.Graal Diaz: data collection, writing the paper.Shawn Steen: data collection, writing the paper.Janet Hobbs: editorial review, formatting and submitting the paperNA.Dr. Graal Diaz.Not commissioned, externally peer-reviewed.