Bilateral persistent hyperplastic primary vitreous (PHPV) is a rare ocular disorder. Its clinical manifestations include bilateral corneal haziness, microphthalmia, and cataract. It is the second most common cause of leukocoria after retinoblastoma. Most cases of PHPV are unilateral. The typical imaging features of PHPV comprise bilateral echogenic masses and a fibrous cord extending from the posterior surface of the lens to the optic disc. In this report, we present a case of bilateral PHPV in an infant who presented with bilateral corneal haziness and watery discharge. A detailed ocular examination and knowledge about its features on imaging can lead to a timely and accurate diagnosis of the condition. Bilateral persistent hyperplastic primary vitreous (PHPV) is a rare vitreoretinal disorder. The most common clinical manifestations of the condition are leukocoria, microphthalmia, and cataract . It is aPHPV is one of the most important differential diagnoses of retinoblastoma . It may Our patient was a 20-day-old female infant who presented to an ophthalmology clinic with complaints of bilateral corneal haziness and watery discharge. The patient had been born at term by uneventful spontaneous vaginal delivery. There was no antenatal and family history related to presenting complaints. Systemic examination was normal. On ophthalmological examination, pupillary reactions were found to be normal. Bilateral corneal haziness was present. Bilateral pupillary reflexes were positive, and the size of the left cornea was relatively smaller than that of the right one. On slit-lamp examination, posterior sub-capsular opacification was observed along with a fibro-vascular thread-like extension from the posterior surface of the lens. At this stage, we were suspecting retinoblastoma.We referred the patient to the department of radiology for further evaluation. The radiologist performed an ultrasound B scan for the identification of the pathology. Ultrasound showed echogenic masses in the posterior segment that were extending from the posterior surface of the lens to the optic disc. Doppler study showed the presence of hyaloid vasculature Figure .We also observed reduced axial length of both eyes. The radiologist performed contrast CT scans. CT scans revealed posterolental masses along with a hyper-dense thread-like extension in the posterior segment of both eyes. It was representing hyaloid arteries. We did not observe any intra-lesion calcification Figure .After correlating the clinical presentation and imaging findings, we made a diagnosis of bilateral PHPV. The ophthalmologist performed an anterior capsulotomy with lens aspiration. A posterior capsulotomy was also done to remove the central plaque. For the removal of primary vitreous and persistent hyaloid vasculature, an anterior vitrectomy was performed. There were no intra-operative or postoperative complications. We followed up with the patient after four weeks. There was no ocular abnormality.We reported a case of bilateral PHPV in a 20-day-old infant. She had no familial, antenatal, or postnatal history related to the disease. There was no history of trauma as well.As embryonic development progresses, the primary vitreous and hyaloid vasculature are replaced by avascular secondary vitreous. This progressive regression starts in the 20th week of gestation and completes at birth . Rarely,The most common clinical signs and symptoms of PHPV include bilateral corneal haziness with watery discharge, microphthalmia, and decreased vision . Based oOcular ultrasound and CT scan are the two gold standard imaging modalities that have proven to be very significant in the diagnosis of PHPV. Ocular ultrasound reveals an echogenic cord extending from the posterior surface of the lens to the optic disc. It also shows the decreased axial length of the globe. Ocular ultrasound can also help in ruling out any calcification . CT scanIn a patient with bilateral leukocoria, the differential diagnosis includes retinoblastoma, PHPV, retinopathy of prematurity, vitreoretinal dysplasia, and ocular toxocariasis . ImagingThe management of PHPV depends on the age of the patient at the time of diagnosis and extension of the pathology. Mild cases can be managed conservatively due to their benign nature. Moderate and severe cases that are diagnosed late require surgical interventions. These interventions include anterior and posterior capsulotomy along with anterior vitrectomy .Bilateral PHPV is the second most common cause of leukocoria after retinoblastoma. Detailed ocular examination along with imaging can help in the early diagnosis of the disease. Prompt recognition, along with surgical intervention in severe cases constitutes the key to a successful outcome.


Vascular lesions of the hand are common and are distinct from vascular lesions elsewhere because of the terminal vascular network in this region, the frequent hand exposure to trauma and microtrauma, and the superficial location of the lesions. Vascular lesions in the hand may be secondary to local pathology, a proximal source of emboli, or systemic diseases with vascular compromise. In most cases, ischaemic conditions are investigated with Doppler ultrasonography. However, computed tomography angiography (CTA) or dynamic contrast-enhanced magnetic resonance angiography (MRA) is often necessary for treatment planning. MR imaging is frequently performed with MRA to distinguish between vascular malformations, vascular tumours, and perivascular tumours. Some vascular tumours preferentially affect the hand, such as pyogenic granulomas or spindle cell haemangiomas associated with Maffucci syndrome. Glomus tumours are the most frequent perivascular tumours of the hand. The purpose of this article is to describe the state-of-the-art acquisition protocols and illustrate the different patterns of vascular lesions and perivascular tumours of the hand. Vascular lesions of the hand are common and are distinct from vascular lesions elsewhere.At the level of the hamatum, the ulnar artery is located in front of the tip of the hamulus, where it is exposed to shocks on the palm of the hand.Computed tomography angiography of the hand is performed with two successive acquisitions after injection of iodinated contrast medium.An artery occlusion may be missed with magnetic resonance imaging if magnetic resonance angiography is not performed.Vascular lesions of the hand are common and are distinct from vascular lesions elsewhere because of the terminal vascular network in this region, the hand’s frequent exposure to trauma and microtrauma, and the superficial location of the lesions. Vascular lesions in the hand may be secondary to local pathology , a proximal source of emboli (e.g. dissections or aneurysms), or systemic diseases with vascular compromise (e.g. rheumatic and vaso-occlusive diseases).Clinical history and direct observation remain the best approach for diagnosis of vascular lesions in the hand. Symptoms may suggest emboli, vessel occlusion, a vascular malformation, or a tumour. In most cases, Doppler ultrasonography (US) is the first imaging modality to be performed. Computed tomography angiography (CTA) or magnetic resonance imaging (MRI) is indicated to improve lesion characterisation and treatment guidance , 2. MRI In this article, we will review the relevant vascular anatomy, describe the state-of-the-art acquisition protocols, and illustrate the different patterns of vascular lesions of the hand in adults, while avoiding common diagnostic pitfalls.The vascularisation of the hand is mainly ensured by the radial and ulnar arteries  . The UA The RA and the UA feed several arches that are responsible for the vascularisation of the carpus; the main ones are the superficial palmar arch (SPA) and the deep palmar arch (DPA). The SPA is mainly fed by the UA and ensures at least the vascularisation of the last three or four fingers. The SPA is covered by skin and the palmar fascia. Its most distal part projects between the proximal and distal palmar creases.The SPA may or may not vascularise the thumb.A persistent median artery is observed in about 10% of cases; its size is extremely variable. It may be barely perceptible or, conversely, equal to the diameter of the RA. Its origin is also variable. It is often associated with a bifid median nerve; in that case, it travels between the two nerve trunks .The SPA can be supplied by the RA and/or the persistent median artery.There are many anatomical variations of the SPA Fig.  12, 15], 1512, 1The DPA is mainly supplied by the RA and ensures vascularisation of the first two fingers. The DPA travels to the palmar surface of the bases of the metacarpal bones deep to the deep flexor tendons. It gives rise to four palmar metacarpal branches. The first palmar metacarpal artery (FPMA) ensures vascularisation of the thumb and feeds the palmoradial digital artery of the index finger. The palmar metacarpal arteries of the second, third, and fourth spaces each receive a perforating branch originating from the corresponding dorsal interosseous artery before anastomosing with the corresponding common digital artery originating from the SPA. These arteries are then divided into palmar collateral arteries.The thumb arterial system may be supplied by the DPA, the SPA, and the dorsal system Fig. . The FPMCTA and MRI are both efficient methods to evaluate hand vessels and lesions with a vascular origin. However, advanced CT techniques, such as dynamic CTA and SHR-CTA, have increased the frequency with which CTA is performed Table . In any (A)CTASingle scan upper limb CTA can be performed with sufficient quality up to the distal portion of the RA and UA but no further. This is because it is difficult to estimate the ideal timing to achieve satisfactory enhancement of both the proximal and distal arteries and digital artery evaluation requires optimised spatial resolution, which is not achievable with an upper limb run-off Fig. .Fig. 3CoCTA of the hand if possible is performed in prone position with flat hand on the table with fingers extended (superman position) in order to place the hand as much as possible in the centre of the scanner to increase spatial resolution Figs.  and 3. AOptimal injection and acquisition parameters are critical when imaging the hand . IodinatIn helical mode, two successive acquisitions are carried out after ICM injection, in the same direction from the proximal to distal location. Even with a delay after contrast injection over 10 s, the contrast bolus can be outrun during the first acquisition. A double acquisition allows a better distinction between arteries and veins, as well as between delayed opacification and occlusion of the digital arteries. A late acquisition may also be useful for detecting arterial wall thickening and enhancement in cases of occlusion or an inflammatory arterial wall disease. A low-dose technique with 80 or 100 kVp increases the contrast at the enhanced vessels and reduces patient exposure .SHR-CTs are novel commercial systems with 0.25-mm detector elements in the in-plane and longitudinal directions. Images can be reconstructed with high matrix sizes up to 2048 × 2048) using deep-learning reconstruction algorithms. SHR improves the quality of both native images and multiplane reformats, especially the depiction of small vessels, particularly the distal portion of the digital arteries Fig. : Video 148 × 2048Dynamic CTA simultaneously yields an anatomic and a functional vascular evaluation , 11, 18.Post-processing is required to visualise and interpret SHR-CTA datasets and to communicate findings to referring physicians , 22, 23.Finally, the radiation dose to the patient remains low, ranging from 0.05 to 0.3 mSv, due to low-dose techniques and a low hand radiosensitivity at the level of the hand .(B)MRIThe patient should be scanned in a comfortable position to avoid motion artefacts. A dedicated coil is essential to obtain high-quality images regardless of the field strength of the MRI system Fig. . A flexiIn most cases, MRA is not the only acquisition to be performed and the protocol also comprises conventional sequences with T1-weighted, fluid-sensitive sequences such as fat-suppressed T2-weighted MRI scans, and contrast-enhanced T1-weighted fat-suppressed sequences Table . These mThe technical aspects and specific protocols for MRA have been well described in the literature , 26, 27.The diagnosis of vascular lesions of the hand is often based on clinical history and clinical examination. Most of the pathologies require US examination which in many cases is sufficient to reach the diagnosis. However, CTA and MRA which provide distinct advantages over ultrasound may be indicated, and we will focus on their findings in the different entities. It should also be noted that incidental discovery of a vascular lesion on MRI is not uncommon and radiologists must be aware of this situation.This chapter is divided into two parts. The first part covers vessel injuries and ischaemia causing lesions. The second one describes the vascular malformations and the vascular and perivascular tumours affecting the hand.(A) Ischaemic phenomenaThe causes of hand ischaemia are varied  . TherefoRadial and ulnar arterial access is frequently used for catheterisation, and transradial access is increasingly used for coronary angiography and percutaneous coronary intervention . IatrogeUpper extremity arterial injuries account for up to 50% of all peripheral vascular injuries. Of these, brachial artery lacerations are the most common, followed by those of the RA and UA, while digital artery lacerations are the least frequent. Digital artery lacerations usually occur secondary to open puncture wounds to the hand, but these injuries may also result from severely displaced fractures, crush injuries, gunshots, and other penetrating wounds. Acute artery lacerations of the hand do not require a CTA, except in cases of associated complex bone lesions or multiple vascular lesions. CTA findings include active extravasation, luminal narrowing, lack of luminal contrast opacification, filling defect, arteriovenous fistula, and pseudoaneurysm Figs.  22, 35––35–37. SOcclusion of the digital arteries that results from acute blunt trauma is exceptional . Those dHHS is in most cases due to repetitive trauma to the UA at the level of the hamulus of the hamatum, leading to a stenosis, an occlusion, and/or an aneurysm of the UA. It is referred to as hammer syndrome because in most cases it originates from repetitive strikes and microtrauma to the ulnar side of the palm of the hand , 40. HowThe clinical manifestations depend on the location and extension of the lesions as well as the configuration of the vascular network. HHS can be asymptomatic, or it can generate Raynaud’s phenomenon and acute ischaemic manifestations with embolic digit pulp necrosis. Some clinical symptoms may be misleading because a UA aneurysm can be responsible for a mass effect or cause compression of the ulnar nerve or of its superficial (sensitive) branch Fig. : Video 3HHS is common in middle-aged, active, smoking men; it generally affects the dominant hand. HHS can be related to sports  or professional activities in which the hand is used to hit, push, or squeeze hard objects. HHS is recognised as an occupational disease in most countries; workers considered ‘at risk’ include metalworkers, car mechanics, lathe operators, machinists, miners, stonemasons, butchers, bakers, masons and carpenters, lumberjacks, and vibrating tool users.CTA and/or MRA may be indicated to confirm the diagnosis , 42. TheSurgical treatment is indicated when there is an aneurysm, ischaemic phenomena, or severe functional impairment resistant to medical treatment. It consists of removing the affected segment and reestablishing flow with venous or arterial bypass. Smoking cessation is obviously recommended, and occupational reclassification may be indicated –45.A so-called thenar hammer syndrome has been described and is exceptional. Repeated microtrauma to the palmar surface of the wrist is likely to generate lesions of the RA that are identical to those described for the UA in HHS. Playing volleyball has been reported to cause this condition.HAVS is a prevalent occupational disease that affects workers in multiple industries in which vibrating tools are used. The exact prevalence is unknown, but it has been estimated at 50% among exposed workers. HAVS mainly affects men over 35 years of age. It has three components: vascular, sensorineural, and musculoskeletal. Symptoms vary by intensity and duration of vibration exposure , 46. NeuHAVS is substantially underrecognised. This underdiagnosis is problematic because early recognition and management of this condition are crucial for preventing progression and improving prognosis. Management involves reduction of vibration exposure, avoidance of cold conditions, smoking cessation, and medication.The persistent median artery may be the site of an occlusion or an aneurysm that causes carpal tunnel syndrome Fig.  48]. Th. Th48]. Intra-arterial drug injections.Arterial complications result from inadvertent and often repeated arterial puncture during attempted venous access and can lead to acute vascular emergencies. Vascular complications are common in people who inject drugs. They can occur locally at the injection site or at a distant location and may be arterial or venous in nature . They caA multimodal imaging strategy is often required in the assessment of these vascular complications, typically involving a combination of ultrasound and CT Fig.  49]. Va. Va49]. Thromboangiitis obliterans, also known as Buerger’s disease, is a rare, non-atherosclerotic inflammatory disease of unknown aetiology. It typically affects young male smokers (younger than 45 years). Cannabis use has also been implicated. Although Buerger’s disease has a worldwide distribution, it is more prevalent in the Middle East and Far East than in North America and Western Europe .This disease primarily affects small- and medium-sized arteries and veins in the upper and lower extremities, usually beginning in the small distal vessels, resulting in distal ischaemia. As the disease progresses, it may involve more proximal arteries. The upper limbs are affected in 25% of cases , 53. ArtCTA, MRA, or angiography shows involvement of the small- and medium-sized vessels in the upper (and lower) extremities, including the palmar and plantar arches, the RA and UA, and the digital arteries of the fingers Fig. . The mosThe cornerstone of treatment of Buerger’s disease is strict abstinence from exposure to all tobacco-containing products. Vasodilator therapy with iloprost should be considered in acute phases, and bypass surgery may be indicated for limb salvage in critical ischaemia.SSc is a rare multisystemic disease that preferentially affects middle-aged women. Hand involvement is a major feature in patients with SSc, with severe Raynaud’s phenomenon in early stages, and skin thickening. Ischaemic digital ulcers may be responsible for major disability. Although rarely indicated for the evaluation of patients affected by SSc, vascular involvement as well as bone and other soft tissue lesions can be identified on CTA or MRA.Systemic vasculitides affecting the hand are mostly represented by polyarteritis nodosa (PAN). PAN is a systemic necrotising vasculitis that typically affects medium-sized arterial vessels but may also affect small-sized arterial vessels. PAN is usually diagnosed in middle-aged and older adults. Systemic symptoms can be the only complaints but almost every organ can be involved. Characteristic lesions of the hand include multiple short segment stenoses of the proper and common digital arteries, ectasia, and aneurysms with thickening of the vessels . Similar(B)Tumours and vascular malformationsHand tumours are encountered frequently. Of all soft tissue lesions throughout the body, 15% are seen in the hand , 7473, 7Angiosarcoma is an aggressive, malignant endothelial cell tumour of lymphatic or vascular origin. Angiosarcoma of the hand can be divided into primary cutaneous angiosarcoma and secondary angiosarcoma, associated with chronic lymphoedema (Stewart–Treves syndrome) or radiation therapy Fig. . They maGlomus tumours are rare benign tumours (1%–5% of hand tumours) that arise from the glomus bodies. A glomus tumour is a mesenchymal neoplasm composed of cells resembling the perivascular modified smooth muscle cells of the normal glomus body . The proGlomus tumours are mainly located in the nail bed (75%–90% of cases), but they can also develop in its matrix, on the side of the fingers, or in the palms of the hands. The average size is 5–6 mm and rarely exceeds 1 cm. Most of them are single, but multiple forms exist (2% of cases).Distal lesions of the nail bed are visible as a reddish-blue spot if the lesion is large enough. The typical clinical triad of localised tenderness, severe pain, and cold sensitivity is highly suggestive of a glomus tumour. More proximal sub-matrix lesions are hidden under the posterior skin fold but may result in longitudinal cracking of the nail bed if there is matrix compression . MRI is Angioleiomyoma is a rare benign vascular smooth muscle tumour arising from the tunica media of vessel walls. Angioleiomyoma affects a wide age range, peaking in the fourth through sixth decades of life. Approximately half of angioleiomyomas are painful. Imaging techniques show a small (less than 3 cm long), well-circumscribed oblong tumour in the subcutaneous fat layer and dermis. The MR pattern is nonspecific with variable enhancement on contrast-enhanced T1-weighted fat-suppressed sequence. However, enhancing vascular structures observed in at least 50% of the cases, on one or both ends of the mass, are suggestive of the diagnosis , 81.Myopericytoma is a rare and distinctive perivascular tumour generally arising in the dermis and subcutis in the distal extremities. Myopericytomas are mostly seen in adults. These tumours are painless. MRI shows a small (less than 2 cm), nodular, well-circumscribed subcutaneous tumour with a strong enhancement on contrast-enhanced T1-weighted fat-suppressed sequence. Myopericytomas are usually connected to a vessel and may develop within the lumen of a vessel.Vascular lesions of the hand have various origins. US may be the initial imaging modality, but CTA or MRA is also required, providing detailed vascular mapping, functional information, and precise determination of the extent of the lesions. This information is paramount to guide treatment.Additional file 1: Video 1. SHR-CTA of the hand (with GI) in a 69-year-old woman that shows the complex arterial network of the hand with a Type A incomplete superficial palmar arch.Additional file 2: Video 2. Dynamic CTA of an arteriovenous fistula fed by the ulnopalmar digital artery of the fourth finger in a 32-year-old patient.Additional file 3: Video 3. Dynamic CT showing a HHS in a 55-year-old manual worker exposed to different causative factors, including vibrating tools. Note that the delay between each phase is 5 seconds. It takes 35 seconds between the opacification of the RA and the common digital arteries at the level of the metacarpophalangeal joints.Additional file 4: Video 4. Dynamic CTA showing a pyogenic granuloma of the third finger in a 39-year-old woman characterised by increased vascularity of all the finger associated with a hypervascular exophytic soft tissue tumourAdditional file 5: Video 5. Dynamic CTA with bone subtraction showing a glomus tumour of the nail bed of the thumb in a 68-year-old man


A choristoma is a tumor-like outgrowth consisting of heterotopic, histologically mature tissue located at an anatomically unusual part of the body. Salivary gland choristoma at the gastrointestinal junction (GEJ) is an extremely rare entity with only one other case reported in the literature. In this report, we present the case of an 87-year-old female with long-standing gastroesophageal reflux disease (GERD) history who was incidentally found to have salivary gland choristoma at GEJ through an upper endoscopy-guided biopsy. We suggest that the finding of salivary gland choristoma at the GEJ could be metaplasia secondary to the patient’s long-standing history of GERD with esophagitis. Heterotopic salivary gland tissue (HSGT) has been most commonly described in the head and neck region including the middle ear, gingiva, hard and soft palate, and external auditory canal. Malignancies arising from HSGT of the head and neck region have also been reported in the literature ,2. The gAn 87-year-old female presented with acute-onset left lower leg pain and swelling. The patient’s past medical history was significant for gastroesophageal reflux disease GERD (she was on daily famotidine), hypertension, and chronic kidney disease. On initial evaluation, vital signs were within normal limits and stable. Her laboratory tests were remarkable for elevated serum creatinine at 1.66 mg/dl, reduced estimated glomerular filtration rate at 29 ml/min, and microcytic anemia with initial hemoglobin at 7.9 gm/dL and mean corpuscular volume at 75.3 fL. Further investigation revealed normal coagulation panel. The iron panel was consistent with iron deficiency anemia. Doppler ultrasound showed an acute non-occlusive deep vein thrombosis in the left common femoral and femoral and popliteal veins. She was started tentatively on a therapeutic dose of enoxaparin.During the hospitalization, the patient underwent a gastrointestinal evaluation for acute on chronic anemia due to suspected gastrointestinal bleeding. Upper endoscopy showed a large hiatal hernia, reflux esophagitis mainly in the lower one-third of the esophagus, and a small nodularity at the GEJ Figure . Biopsy The human salivary glands are composed of three major glands  and numerous minor glands that take part in food digestion, lubrication, and immune defense. It is suggested that the parotid gland originates from the oral ectoderm, while the submandibular, sublingual, and intraoral minor salivary glands derive from the foregut endoderm .Salivary gland choristoma at the GEJ is an extremely rare finding. Karim et al. recently reported three cases of HSGT at the GEJ, and only one of them was in the form of choristoma. All three cases presented in their report had a history of GERD, with one of the HSGT cases demonstrating grade-3 esophagitis and the other HSGT case having Barret’s esophagus with high-grade dysplasia . SimilarRepeated exposure of the esophagus to gastric contents through reflux triggers protective mechanisms in the esophagus resembling salivary gland function. An increase in the rate of mucin release from salivary glands and esophageal mucosal and submucosal glands into the mucus-bicarbonate layer is seen with decreasing pH associated with GERD . ExposurIt is well known in the literature that intestinal columnar metaplasia at the GEJ can occur secondary to chronic acid exposure and inflammation in the setting of GERD. The origin of these intestinal glandular cells remains unknown. The presence of multipotent stem cells or the intrinsic ability of native squamous epithelium, submucosal glands, squamocolumnar junction cells or circulating bone marrow stem cells to dedifferentiate are proposed mechanisms . The latIn this case report, we aimed to enhance the understanding of clinicians about the pathophysiology of glandular metaplasia in response to chronic inflammation at the GEJ. Development in these biological mechanisms could also contribute to studies investigating cell-based therapy for salivary gland regeneration post-radiation therapy and in patients with Sjogren’s syndrome .https://insights.ovid.com/crossref?an=00000434-201910001-01841).This work has been already presented as an abstract (We reported an extremely rare case of salivary gland choristoma at the GEJ. Based on our literature review, this finding could be a metaplastic change in the setting of chronic inflammation as a result of reflux esophagitis. However, the biological and clinical significance of the salivary gland choristoma at the GEJ is yet to be investigated. Further studies are warranted to examine the association between these two.


Vascular malformation (VM) consists of a group of tumors that emerge from vascular origin caused by vascular angiovascular or lymphoproliferation. Arteriovenous malformations (AVMs) contribute high-flow, creating direct vein artery contact without regular capillary network. AVMs are present at birth or in congenital. Acquired AVMs occur later in life due to hormonal changes or trauma, and acquired AVMs in oral cavity are very rare. AVMs are persistent and progressive in nature when present, can represent a lethal, causing significant blood loss, and an incomplete resection frequently leads to a recurrence of the lesion. We present a rare case of a congenital AVM diagnosed on retinue dental checkup using color doppler ultrasonography (USG) in a 62-year-old man from South India. Arteriovenous malformations (AVMs) are the lesions with direct communications between endothelial‑lined artery/arteries and vein/veins bypassing the capillary bed . Head anArteriovenous malformation is congenital in nature and consists of a fistulae-tangle of arteries and veins, where the central component is a nidus. Hemorrhage is the most common symptom but other symptoms include headache, seizures, stroke-like symptoms, and ischemic stroke. Within the literature, cognitive results are rarely published, with some indications of a progressive course among case series reports . Hence, A 62-year-old male patient presented to the Department of Oral Medicine and Radiology for the retinue dental checkup. Extra oral examination revealed swollen lips, history revealed that it started at birth and slowly progressed, asymptomatic, no history of bleeding or discharge Figure .The intraoral clinical examination revealed over all swelling of the both upper and lower lips, with normal color, measuring 2-3 cm thickness of the lips; on palpation it was painless, fluctuant, nonreducible, and compressible with the presence of pulsatility. Furthermore, on chair side diascopy test blanching was noted  are rare but persistent and progressive in nature, and can be a fatal benign disease , 7. HemaThe AVMs present a therapeutic challenge because of their hemodynamic characteristics and their growth modality . They haThe AVMs are fairly uncommon in the area of the head and neck. Because of their potential for uncontrollable bleeding, they may pose as a dental emergency while performing dental procedures such as tooth extraction, biopsy, or during the normal primary tooth exfoliation. They can cause troubling hemodynamic manifestations such as venous obstruction, distal ischemia, and high-performance cardiac failure. Therefore, these cases must be diagnosed early and treated promptly. A clear clinical background with imaging usually delineates the lesion well and provides an opportunity to make a cautious intervention decision. The surgical treatment is complicated and involves careful preparation and multidisciplinary approach.


Pulmonary hamartomas are usually solitary, nodular benign lesions in the parenchyma of the lung. They are rarely situated in endobronchial areas, and very few cases are reported with the mediastinum. A 56-year-old female patient got a CT-scan conducted due to coughing and breathlessness and was diagnosed with a nodular lesion in the medial mediastinum. The lesion was operated: it measured up to 4 cm in the largest diameter, had a smooth surface, was of rather soft but elastic consistency, and was extirpated. At pathology, on cut section, it was yellowish and lobular, and with a mixture of cartilaginous, fibrous and adipose tissues with some smooth muscle cell fibers and myxoid areas.The diagnosis of pulmonary hamartoma was made with atypical medial mediastinal localization. This rare presentation could pose some differential diagnostic problems in the clinical diagnosis of more frequent primary and metastatic malignant diseases. A hamartoma is a benign growth or focal malformation of an abnormal mixture of cells and tissues that is normally found in the area of the body where growth occurs, but in a disorganized manner. Although they are considered developmental malformations, many hamartomas have clonal chromosomal aberrations that are now considered to be neoplastic. In favor of the malformation theory, stands the fact that they have the same growth rate as the surrounding tissue. Hamartomas could occur in many parts of the body, and usually, as an asymptomatic mass, they are diagnosed as incidentalomas (found on an imaging study for some other reason).They are closely related to choristoma, which is defined as a mass of tissue growing at the abnormal site. For both lesions, the underlying reasons for their occurrence are still not completely understood.The most common place for hamartoma is the lung, where it comprises about 5-8% of all solitary nodules and up to 75% of all benign tumors. By definition, histological components of hamartoma are at least two mesenchymal tissues, mainly cartilage, mixed with adipose cells or some other cell types (fibrous or smooth muscle), and bronchial epithelium . HamartoLung hamartomas are more frequent in men than in women, with the peak incidence of diagnosis in the 50s and 60s, and although usually asymptomatic, they may present some difficulties in smokers .A 56-years-old female patient who was previously a heavy smoker for 15 years ceased for almost the last 20 years due to proven pollen allergy. Following the exacerbation of cough and breathlessness, a CT-scan was conducted, and a single mass in the medial mediastinum was diagnosed. The mass was situated in the right side of the thorax, close to the mediastinal pleura at the level of superior vena cava; it was oval and well circumscribed. It was hypodense, had an inhomogeneous appearance, and with measured Hounsfield values from +10 up to -10. The greatest dimensions measured were 38 x 30 mm; it was extrapulmonary and in contact with pericardium in its caudal part . This important lesion must be considered in differential diagnosis with other solitary lesions, most importantly, the malignancy. On plain X-ray examination, there are not enough characteristic signs to make a correct diagnosis, so the CT-scan is advised. The popcorn-like calcifications are an important clue for diagnosis . They arBesides the potential harmful effect on the lung tissue, another importance of hamartomas is their association with some diseases, which could have a serious impact on the patients’ health. In one broad study of 24 cases, an unexpectedly high number of patients had some congenital abnormality in other organs; all of them had at least one benign tumor, and in five, a malignant tumor was diagnosed . The occThe most common localization of hamartoma is lung tissue. The mediastinal hamartoma is quite rare, in either the posterior or the anterior part -8.This case is unusual because the previously performed lung imaging examination  did not diagnose any sign of this lesion. Also, the unusual presentation in the medial mediastinum makes the case atypical. Theories about a possible migration of intraparenchymal hamartoma through visceral pleura toward mediastinum are posed . If we aAlthough a pulmonary hamartoma is not a difficult diagnostic problem at pathology, it must be included in the clinical and radiological differential diagnosis of solitary nodules in the lung parenchyma, and even within the mediastinum.


Patients with lupus anticoagulants are at high risk of systemic arterial and venous thrombosis and arterial stroke. We present an unusual case of a young man presenting inguinal pain. Doppler ultrasound revealed spermatic vein thrombosis on the left side. Hematologic workup revealed positive lupus anticoagulant. The patient was treated with therapeutic heparin. Spermatic vein thrombosis is a rare event which can be difficult to diagnose. Multiple predisposing factors have been associated with Spontaneous Spermatic vein thrombosis, like malignant tumors, coagulopathies and varicocele . We presA 27-year-old male, with no previous health problems, was admitted with a 1-week history of painful in the left inguinal region, without fever or any associated signs. He denied any etiologic factors like trauma, surgeries, severe exercise or thrombogenic factors. Inguinal region, scrotum and prostate were normal to palpation. Initial laboratory test, were normal. Doppler ultrasonography of left inguinal region demonstrated an expanded vein which contained thrombus  with no Spermatic vein thrombosis is a rare pathology, it can clinically simulate an incarcerated hernia . In addiSpermatic vein thrombosis is particularly rare disease. Ultrasound should be the first line examination to avoid exploratory surgery.


Solitary fibrous tumors are rare neoplasms that originate from mesenchymal tissues and have been found to occur in any site, including the spine and liver. Although most of solitary fibrous tumors have benign features, only 10–20% are malignant and prone to metastasis. No previous reports have described the malignant and metastatic Solitary fibrous tumor arising in both of the liver and thoracic vertebrae. In this article, we present the case of a 60-year-old woman who underwent gross total resection of a meningeal tumor in 2007. She presented 10 years later with a thoracic vertebral mass that caused relentless pain and a lesion in the right lobe of liver. She underwent marginal excision of the T3 tumor with T2-4 pedicular screw fixation in March 2017, then right hemi-hepatectomy was performed to remove the liver lesion in June 2017. Both of the lesions were confirmed to be a metastatic and malignant tumor after surgery. The literature lacks randomized controlled trials and large studies that define the natural history of malignant solitary fibrous tumors and recommendations of precise management plan for the disease. However, the best choice for treatment is gross total resection, which probably provide the optimal treatment to achieve long-term disease-free survival. Solitary fibrous tumors (SFTs) are rare mesenchymal tumors derived from mesenchymal tissues and were first reported by Klemperer and Rabin in 1931 –4. AccorThe patient is a 60-year-old woman who presented to our hospital with new-onset upper back pain. The patient complained of upper back pain for 6 months accompanied by left intercostal neuralgia, which was refractory to analgesic treatment. The patient underwent gross total resection of a meningeal tumor 10 years ago. The pathology result was a hemangiopericytoma with a high proliferation index that was classified as WHO grade 2. The patient received twenty-seven cycles of adjuvant radiotherapy after surgical resection. Then, the patient had an excellent post-operative course, and yearly follow-up computed tomography (CT) scans confirmed a complete resection and no recurrence of the tumor until March 2017. Ten years after resection of the meningeal tumor, her upper back pain started. First of all, the patient admitted to another hospital, the positron emission tomography/computed-tomography (PET-CT) showed the lesion at the left T3 vertebral body and in the right lobe of the liver without other sites metastasis. The diagnosis of metastatic SFT was suggested by immunohistochemistry of the T3 mass after CT-guided percutaneous thoracic puncture biopsy was performed. The immunohistochemical study demonstrated CD34(+) and STAT6(++). Then, abdominal and thoracic magnetic resonance imaging (MRI) were performed. The abdominal MRI revealed an enhanced lesion in the right lobe of the liver, with a size of ~2.7 × 2.8 × 3 cm . The patSFTs are rare neoplasms that originate from the pleura. However, SFTs have been found to occur in any site, including the spine. Spinal SFTs are always classified as intramedullary (58%), intradural extramedullary (24%), or extradural (18%) , 10. MorHistopathologically, typical benign SFTs contain clear tumor cell boundaries and dilated branched blood vessels. Moreover, typical benign SFTs are always present with a fusiform pattern, scattered among the collagen fiber bundles in different forms. In addition, 80% of extra thoracic SFTs always present with atypical histological features, including increased cellularity, a large number of nuclear pleomorphisms, focal necrosis, and four or more mitoses per ten high-power fields (HPFs). Most SFTs usually exhibit positive staining for mesenchymal and vascular endothelium-related antigens, such as CD34, CD99, Bcl-2, and vimentin, whereas these tumors exhibit negative staining for SMA, EMA, and GFAP. Because these biomarkers are non-specific for the diagnosis of SFTs, STAT6 was recently found to be a typical tumor marker for the diagnosis of SFTs , 12. TheFurthermore, as reported by Melone et al., high-grade SFTs recur 90% of patients, whereas the rate of recurrence for low-grade SFTs was merely 27% . StatistIn terms of the management of metastatic SFTs, there is still no consensus because of the lack of sufficient research with long-term follow-up in the literatures. The management of metastatic SFTs typically consists surgery with or without radiotherapy. However, surgical excision still remains the first choice of treatment. Son et al. reported a case of malignant SFT which was resected but a local recurrence developed rapidly . MeanwhiThe literature lacks randomized controlled trials and large studies that define the natural history of malignant SFTs and recommendations of a precise management plan for the disease. Further studies are needed to understand the long-term outcome and clinical features for the diagnosis of the rare clinical situation. However, the best choice for treatment is gross total resection, which seems to provide the optimal treatment to achieve long-term disease-free survival.The original contributions presented in the study are included in the article/supplementary materials, further inquiries can be directed to the corresponding author/s.Written informed consent was obtained from the individual(s) for the publication of any potentially identifiable images or data included in this article.MM and LZ contributed equally to the writing of the manuscript and designed the figures. CH and XY collected clinical data and followed the patient during the clinical course. SH revised the article. HY and QZ interpreted the radiological images. DS reviewed and approved the final version of this article. All authors contributed to the article and approved the submitted version.The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.


A retroperitoneal abscess caused by duodenal perforation is a relatively rare disease clinically. We report the case of a patient with a local high-density shadow at the head of the retroperitoneal pancreas.9/L and a neutrophil absolute value of 18.81 109/L, and a computed tomography scan showed an irregular soft tissue mass with uneven enhancement of the cystic wall in the retroperitoneal space. Gastroscopy showed that there was a fistula in the anterior wall of the duodenal bulb. Endoscopic anastomosis clip system (over-the-scope clip) of the duodenal fistula was performed successfully. After the operation, nasal feeding was provided with a nutrition tube, and empiric anti-infection, acid-inhibiting, and stomach-protecting treatments were administered. Our patient’s body temperature gradually returned to normal, and his abdominal pain decreased.A 28-year-old Chinese man presented with fever and abdominal pain after overeating and heavy drinking. On physical examination, he had mild tenderness in his upper abdomen. Laboratory examination results showed a white blood cell count of 24.06 10A retroperitoneal abscess caused by duodenal perforation can be diagnosed by clinical symptoms and abdominal computed tomography imaging. The choice of treatment should be based on accurate and timely clinical and imaging data. Helicobacter pylori infection and non-steroidal anti-inflammatory drugs (NSAIDs) [Clinically, retroperitoneal abscesses due to duodenal perforation are relatively rare. Altemeier  reported(NSAIDs) . In cont(NSAIDs) . The patA 28-year-old Chinese man had consumed a large amount of barbecued food and alcohol 7 days before admission to our hospital. He had felt abdominal pain after waking the next day, mainly in the upper abdomen. The position of the pain could not be described, and he gradually felt better without special treatment. He had had fever, abdominal pain, and pharyngeal pain 3 days before hospitalization, with his highest temperature reaching 41 °C. Our patient’s temperature then dropped after anti-infection treatment at his local clinic. One day before admission, his abdominal pain and fever reoccurred, mainly in the lower xiphoid process and upper abdomen. The abdominal pain, which was intermittent lacerating pain accompanied by back pain, was persistent and could not be relieved. He had nausea and vomiting, and the vomitus was the stomach contents. Our patient was then transferred to our hospital, and a physical examination revealed a body temperature of 38 °C, heart rate of 100 beats per minute, mild tenderness in the upper abdomen, mainly in the lower xiphoid process and left abdomen, no rebound pain, negative Murphy’s sign, and no pain on percussion in the liver and kidney areas. Laboratory data are shown in Table H. pylori infection), Crohn’s disease, enteritis caused by drugs or radiation, foreign bodies (such as fish bones), and trauma [A retroperitoneal abscess is a relatively uncommon clinical disease, the main causes of which are pancreatitis, appendicitis, perforations of the colon and rectum , and comd trauma –4. This d trauma .Nevertheless, retroperitoneal abscesses due to duodenal perforation are rare. A total of 13 cases of retroperitoneal abscesses caused by duodenal perforation have been reported in nine articles –16 from Abdominal CT should be the first choice in the diagnosis of a retroperitoneal abscess. In the 13 reported cases, patients in the earlier stages did not undergo CT examination due to condition limitations, and only four patients underwent CT examination. The lesions of the four cases were all located in the retroperitoneum, they were solid lesions or cystic-solid lesions with blurred margins, and there was gas in some lesions. In this case, the location of the lesion was near the head of the pancreas because the perforation was located at the duodenal bulb. It was a cystic-solid lesion with punctate high-density shadows. Because of the interference of the punctate high-density shadows, the lesion was initially diagnosed as a mixed infection of tuberculosis and inflammatory exudation. The high-density shadows may have formed because the foreign body, which contained bone or other high-density substances, entered the retroperitoneal space through the duodenal fistula and was partially encapsulated by inflammatory tissue to form a retroperitoneal abscess.Studies –16 show A retroperitoneal abscess caused by duodenal fistula is a rare disease. Abdominal CT is the preferred examination method. The abscess can be diagnosed by typical clinical symptoms and imaging results, and the appropriate treatment should be chosen based on the patient’s condition and sufficient imaging data.


Brachial plexus schwannomas are extremely rare tumours of the head and neck region accounting for less than 5 % overall. Due to its rarity and anatomic complexity of the brachial plexus, schwannomas in this region present a diagnostic and surgical challenge to the surgeon.We present a case of a 56-year-old female who presented with a slow growing right sided neck swelling associated with pain and tingling in the distal end of the right forearm. According to FNAC, imaging studies results, a diagnosis of benign neurogenic tumour possibly schwannoma was made. After taking proper consent patient underwent surgical excision of the tumour. Postoperatively, patient developed numbness and tingling in right arm and stiffness at elbow joint, which is showing improvement after regular physiotherapy sessions. Although brachial plexus schwannomas are extremely rare head and neck tumours they should be kept as a differential diagnosis in patients presenting with supraclavicular neck swellings. These are potentially curable lesions. As such, detailed history and examination together with imaging studies is important in establishing a preoperative diagnosis for proper management. Brachial plexus schwannomas are extremely rare tumours of the head and neck region. Schwannomas are benign tumours arising from Schwann cells of peripheral nerves. Up to half of these occur in the head and neck region, only 5% of which arise from the brachial plexus. These tumours usually present as a painless palpable mass in the neck. We hereby present a case who presented to our out-patient department (OPD) with a slow growing right sided neck swelling which was eventually diagnosed as schwannoma of the brachial plexus. We also review such cases previously reported in literature. Our case highlights that due to the rarity and anatomic complexity of the brachial plexus, schwannomas in this region present a diagnostic and surgical challenge to the surgeon.A 56-year-old female presented to OPD with complaints of right sided supraclavicular neck swelling for last 2 years and associated pain and tingling in the right arm for past 1 year. There was no history of fever, trauma, systemic illness. On examination, a 5X4 cm spherical swelling was palpable in the right supraclavicular area which was non tender, not fixed to the overlying skin and it was more mobile in the horizontal direction than in the vertical direction. On neurological examination, muscle power in all muscles of the upper limb was found to be 5/5 with no signs of wasting and sensations were intact. Tinel sign was negative. Decision to take a FNAC of the swelling was taken which revealed spindle shaped cells and schwann cells which was suggestive of schwannoma. Furthermore, CECT neck was done which showed a heterogeneously enhancing solid cystic lesion in the right supraclavicular region medially extending into and causing widening of right C6-C7 neural foramen .MRI neck and chest were also ordered which showed a well-defined solid cystic lesion in right supraclavicular region which was isointense on T1WI and heterogenous enhancement was seen on post contrast scan which confirmed our diagnosis of Schwannoma most probably arising from the brachial plexus . After a thorough evaluation of the case, and taking proper consent from the patient after explaining all possible complications, the decision of undertaking excision of the tumour under general anaesthesia was made. A horizontal incision was given over the swelling and tumour was exposed. Trunks of the brachial plexus were seen passing around the tumour . The tumour was carefully excised without causing any damage to the trunk and sent for histopathological examination.The histopathology of the tumour specimen showed a characteristic cellular pattern of alternating regions containing compact spindle cells called Antoni type A areas and more loosely arranged, hypocellular zones called Antoni type B areas which was consistent with the diagnosis of Schwannoma .Hence, a final diagnosis of schwannoma of the Brachial plexus was made. In the postoperative period, numbness and tingling in the right arm along with stiffness at the elbow joint was noticed. Patient is being regularly followed up in the last 6 months and has shown improvement in stiffness, tingling and numbness with regular physiotherapy.Schwannomas or neurilemmomas are benign, well encapsulated, usually solitary tumours arising from the schwann cells of the peripheral nerves. Up to half of these arise in the head and neck region from cranial nerves, sympathetic chain, cervical nerve roots and brachial plexus. Brachial plexus tumours are extremely rare entities and form only 5% of all the upper limb tumours. The most common brachial plexus tumours are schwannomas and neurofibromas. The most common site of the brachial plexus tumours is head and neck. Head and neck region comprise of 25-45% of all the schwannomas of which only 5% arise from the brachial plexus .Schwannomas usually present as slow growing painless tumours. However, they may also be accompanied with symptoms secondary to nerve compression. Proper evaluation including detailed history and examination together with the imaging studies help in establishing preoperative diagnosis and avoiding undue misdiagnosis. MRI is usually the study of choice and schwannomas typically present as a well circumscribed mass having heterogenous and bright areas on T2WI with post contrast enhancement. CECT may further aid in diagnosis where most schwannomas are iso-dense to brain parenchyma.To highlight the importance of imaging studies in aiding the diagnosis of schwannoma, Ryu KH et al reported about a case who presented with a palpable neck swelling for one year. A differential of metastatic cervical lymphadenopathy was made. However, CT and MRI along with US guided core needle biopsy proved it to be a schwannoma .Rashid M et al also reported two cases of brachial plexus schwannoma involving C7 root presenting as supraclavicular neck swelling. One of their cases had previously been misdiagnosed as enlarged supraclavicular lymph node and an attempt of excisional biopsy from the same swelling had resulted in iatrogenic injury. Thus, they highlighted the importance of establishing a proper diagnosis of the lesion with the help of FNAC and imaging studies to avoid any misdiagnosis and hence mismanagement. Our case also presented in her fifth decade with a slow growing mass in her neck region. After a thorough history and examination, we decided to perform FNAC of the swelling, which was suggestive of schwannoma. MRI and CECT neck were also carried out to further help in aiding the diagnosis. Thus, FNAC and imaging studies are extremely important in preventing misdiagnosis and hence mismanagement which can have untoward complications. Surgical resection with preservation of the surrounding nerves is typically the treatment of choice as schwannomas are radioresistant. Neurological dysfunction, pain, rapidly growing tumours with suspicion of malignancy are usually the indications for surgical resection. Grossly, these tumours are round, oval or plexiform and can be yellow, grey, pink or tan. Histopatholgy, helps in confirming the diagnosis showing typical Antoni type A and B patterns with rows of nuclear palisading (verocay bodies).Unlike neurofibromas which are unencapsulated and usually envelop the nerve fascicles, schwannomas are generally well encapsulated and displace the nerve fascicles. However, while schwannomas can be enucleated from the nerve, post-operative neurological deficit is still common and as such preoperative counselling of patients regarding the same is extremely important in the overall management.Kumar A et al, in their case report, highlighted the importance of preoperative counselling regarding neurological deficit following surgical resection of brachial plexus schwannoma. They reported of a case in which the patient presented with an axillary swelling. The tumour was resected via an axillary approach and postoperatively the patient developed loss of sensation over index finger and thumb. Vishwanathan N et al also reported of a case of brachial plexus schwannoma, in which intra-operatively, the tumour was found to be splayed into very thin effaced sheet of neural tissue. Despite all efforts made microscopically, the upper trunk C5-C6 nerves had to be severed resulting in post-operative neurological deficit. The surgeons then planned a second stage nerve grafting with sural nerve within four weeks of the primary surgery. The patient was under follow up at the time of reporting  but were safely and completely resected using ultrasound-guided microsurgical excision techniques without any peri- or post-operative complications in any of the 11 patients .Kohyama S et al on the other hand described a patient who presented with a left supraclavicular swelling and left sided muscle weakness. Magnetic resonance imaging showed a continuous, multinodular, plexiform tumour from the left C5 to C7 nerve root along the course of the brachial plexus to the left brachia. Intraoperatively, an approximately 40 cm in length tumour, was seen causing extreme enlargement of median and musculocutaneous nerves. Both the nerves showed no response to electric stimulation. They resected the tumour together with a part of the musculocutaneous nerve for biopsy and performed latissimus dorsi muscle transposition in order to repair elbow flexion. Hence, even if during tumour resection a part of the nerve is resected or sustains injury, primary nerve repair using nerve graft or muscle transposition as used in this case, can help to prevent or reduce post- operative neurological sequelae .Before taking up our patient for surgical resection, a detailed counselling preoperatively regarding the surgery, the possible associated complications and its management was done. Under general anaesthesia, we performed complete surgical resection of the tumour with preservation of the nerve fibres. The patient however, developed postoperative stiffness, tingling and numbness, possibly due to the stretching of the nerve fibres during the surgical resection of the tumour. As we were certain that no nerve fascicles were injured during the resection of the tumour, we decided to keep the patient under regular follow up. The patient was also prescribed regular physiotherapy with which she has shown dramatic improvement.This article highlights the point that a patient presenting with a painless swelling in right supraclavicular region which can easily be misdiagnosed as enlarged lymph nodes in clinical practice could turn out to be a rare diagnosis of brachial plexus schwannoma as in our case report. This paper will help apprentice otolaryngologist that such a diagnosis should also be kept in mind while dealing with such swellings. Further this article also elaborates that how with proper work up and pre- operative counselling we can deal with such rare tumors in an amicable way. Regarding the incidence of the case, we would like to reiterate that schwannoma as an extracranial nerve sheath tumors rarely affects brachial plexus. Due to the rarity of such tumor and due to brachial plexus anatomic complexity, schwannomas in this region present a challenge for surgeons.To conclude, brachial plexus schwannomas should be kept as a differential in patients presenting with supraclavicular neck swellings. As these are potentially curable lesions, a detailed history and examination together with FNAC and imaging studies (MRI and CECT) is necessary in establishing a pre-operative diagnosis thereby reducing the chances of mismanagement. Pre-operative counselling regarding the surgery, its complications and the various options regarding management of neurological complications, if any, helps to improve patient compliance.


Bilateral chronic subdural hematoma are not that common. It may be recurrent and rarely superimposed by acute bleed leading to rapid progression and poor clinical outcomes. We report the case of a seventy six years old lady with a history of traumatic subdural hematoma evacuated by trephination twenty years back, presenting at our hospital with a history of persistent headache and acute onset of several episodes of vomiting. A non-contrast head CT revealed bilateral chronic subdural hematoma with acute on chronic bleed on one side. Trephination was done initially unilaterally, but the symptoms persisted and bilateral trephination was performed. The patient developed bilateral pneumocephalus and chest infection post-surgery. Bilateral, recurrent subdural hematoma with acute superimposition of bleed is a rare entity that presents with signs of increased intracranial pressure as opposed to unilateral SDH. A single burr hole trephination can be an effective intervention in these cases. Although unilateral CSDH occurs in most patients, bilateral CSDH is relatively rare. Rapid escalation of bilateral CSDH has been documented, including the risk of recurrence.Bilateral, recurrent CSDH with superimposition of acute bleed is a rare condition. We hereby report a case of bilateral CSDH with acute bleed unilaterally in a 76-years old lady with a history of previous traumatic SDH and an ominous surgical history.A seventy-six years old lady presented to our Emergency Department with complaints of progressive worsening of headache eleven days in duration and acute onset of several episodes of vomiting since the past two days. She was referred from another center where she was treated symptomatically with no relief of either pain or vomiting. She reported no weakness or numbness of upper and lower limbs. There was no history of fever, abnormal body movements and loss of consciousness. She reported decreased appetite and alteration of sleep habit. She did not give any history of recent trauma or falls. She however gave a history of head surgery 20 years back for a subdural hematoma that she had due to a fall and a leg fracture 7 years ago which was fixed with plates. An interesting revelation was that the patient had remained unconscious for three months following her previous surgery for SDH evacuation.On examination, she was ill-looking. Her vitals were normal. Cardiovascular, respiratory and abdominal examination were normal with a blood pressure of 140/80 mm Hg , pulse rate of 80 beats per minute, a respiratory rate of 20 breaths per minute and oxygen saturation (Sp02) of 98%. She was afebrile, conscious with a Glasgow Coma Scale of 15 and normal neurologic exam, including that of cranial nerves. Tone, power and reflexes were normal in both upper as well as lower limbs. Sensations to all four limbs were also preserved. Palpation of spine revealed no tenderness or steps.Initial laboratory investigations including random blood glucose, hematological profile including that of clotting, liver and renal function tests and other basic tests were normal. A non-contrast CT scan of the head was ordered in the ER which revealed a small hyperdense area surrounded by hypodense areas indicating acute on chronic, right subdural hematoma with a small acute bleed around the temporal lobe and a chronic bleed around the temporoparietal region. The CT also showed a massive hypodense area indicating a large subdural hematoma on the left temporoparietal region, with a very slight midline shift to the right, to go along with a previous burr hole mark on the left temporal bone .A neurosurgical referral was made. She was monitored intensively and treated conservatively with no improvement of her symptoms. Surgery was then planned. A left parietal burr-hole craniostomy was done under local anaesthesia, the following day with aspiration of “machinery-oil” blood under pressure. The headache resolved. The next day after the surgery, the patient complained of headache on the side opposite to which the procedure was performed. The patient was closely monitored in the neurosurgical Intensive Care Unit. After the pain persisted for a day more, a repeat head CT was ordered. It revealed isodense to slightly hyperdense areas in the right temporo-parietal region with a slight midline shift to the left .A right parietal burr-hole craniostomy was performed under local anaesthesia which led to aspiration of dark brown blood under pressure. A repeat CT of the head showed resolution of the hematoma .Three days after the second procedure, the patient complained of frontal headache and nausea. The patient was febrile as well. Repeat CT showed a large bi-frontal pneumocephalus .A chest radiograph showed bilateral infiltrates. These problems were managed medically with high flow oxygen therapy and intravenous antibiotics and the patient recovered eventually . She was4Subdural hematoma is defined by the accumulation of blood between the dura and the arachnoid layers of the meninges. It usually occurs due to the rupture of bridging veins, resulting in collection of subdural blood. Subdural hematomas can be acute, subacute or chronic. Acute subdural hematoma is commonly associated with traumatic, high energy impact and usually presents within twenty four hours of the inciting event. In contrast, chronic subdural hematomas usually are associated with mild or insignificant impact and become symptomatic more than two weeks after the inciting event. Bilateral CSDH have a reported incidence varying from 16% to 20%.5 There are many identified risk factors of subdural hematoma, including cerebral atrophy due to ageing and in chronic alcoholics, anticoagulation, coagulopathy, decreased CSF pressure, aneurysmal rupture and arteriovenous malformation, neurosurgical procedures and infection.7Most of the cases of subdural hematoma are due to rupture of the bridging veins but about 20-30% cases are supposed to be due to arterial rupture.8 Acute SDH results in coma in about half of the cases and in about 12 to 38 percent of patients have a transient interval that is followed by progressive neurologic decline to coma.9 Chronic SDH usually present with insidious onset of headaches, light-headedness, cognitive impairment, apathy, somnolence. Focal neurologic deficits may be ipsilateral or contralateral to the side of the lesion and the symptoms may be transient or fluctuating. Bilateral CSDH are reported to show signs of increased ICP such as headache but less likely to show symptoms associated with brain shift such as hemiparesis and are associated with rapid progression and poor clinical outcomes.The clinical presentation of subdural hematomas vary greatly. Subdural hematoma is often referred as “great imitator” as it can mimic diseases such as stroke, dementia and parkinson's.11 While burr-hole craniostomy continues to be the most commonly used technique for the treatment of CSDH, successful treatment proves a challenging dilemma due to a 10-20% recurrence rate.12 Our patient had a history of a two month unconsciousness following her first burr hole trephination which we expected to be due to some anaesthetic used during that procedure. That is why more invasive methods were not recommended to her and trephinations were performed under local anaesthesia.Surgical evacuation of CSDH is recommended if it is symptomatic and associated with signs of increased ICP. The surgical evacuation of symptomatic SDH can be performed by different techniques, including burr hole trephination, craniotomy, and decompressive craniectomy.Conclusively, we can say that CSDH can present in the elderly without any inciting event or with minimal impact. Bilateral CSDH usually present with signs of increased intracranial pressure in contrast to signs of focal deficit and brain shifts seen with unilateral SDH. Unilateral evacuation may lead to expansion of the contralateral hematoma, requiring bilateral surgery. In the elderly population with an ominous surgical history, single burr hole trephination under local anaesthesia can be a better and safer alternative to more invasive procedures.


An umbilical metastasis from an internal visceral malignancy is defined as Sister Mary Joseph's nodule (SMJN), and, although a rare finding, it is a very poor prognostic indicator. We describe an interesting case of metastatic colon cancer with SMJN, emphasizing the significance of this classic finding.A 64-year-old female with a history of stage IV colon cancer with palliative right hemicolectomy and left hepatectomy presented to the hospital with nausea and abdominal discomfort. A computed tomography (CT) scan of the abdomen was performed, which showed small bowel obstruction secondary to metastatic tumor mass compressing the duodenum. She refused to undergo any chemotherapy or endoscopic intervention and was eventually discharged on hospice care.During the hospital stay an umbilical ulcerative lesion was also noted, which was violaceous, measuring 4.5 x 4 cm in size, firm in consistency with irregular borders, and tender to touch. Therefore, further history was obtained from the patient about it. Several months ago, she had developed localized swelling around the umbilicus, which gradually enlarged and ulcerated later. She eventually underwent the biopsy of that umbilical lesion, which confirmed it as SMJN with metastasis from the colonic primary. However, the patient refrained from the surgical intervention of the umbilical lesion.SMJN presents as a palpable periumbilical metastatic mass with diameter usually not exceeding 5 cm in size. It can be variable in color from violaceous to reddish brown. Once discovered, such lesions should be worked up with biopsy and imaging studies such as CT scan of the abdomen, as the nodule may be indicative of underlying malignancy or cancer recurrence. Its presence indicates a poor prognosis, with average survival time after diagnosis of SMJN of 10 months. Sister Mary Joseph's nodule (SMJN) is an umbilical metastasis from a visceral malignancy, and, although a rare finding, it is described to be an indicator of poor prognosis. The incidence of SMJN is 1%-3% in people diagnosed with intra-abdominal or pelvic malignancies [This case report was also presented as a poster at ACG 2020 P0634 (S1756): Sister Mary Joseph Nodule: Where Umbilicus Holds the Truth!A 64-year-old female with a medical history of stage IV metastatic colon cancer diagnosed in 2017 was evaluated at the hospital for suspected small bowel obstruction. At the time of initial diagnosis of cancer, she had refused palliative chemotherapy. In 2018, she was hospitalized for the first time because of gastric outlet obstruction and underwent palliative hemicolectomy and hepatectomy. The surgical pathology was positive for KRAS gene mutation, whereas the MMR gene was intact. She was started on cycles of FOLFOX  and Avastin® (bevacizumab) at that time.In early 2019, she was evaluated for newly developed localized abdominal swelling around the umbilicus with ulceration Figure . PalliatIn the current hospital admission, she presented with nausea, vomiting, bloating, and abdominal discomfort. Computed tomography (CT) of the abdomen with intravenous contrast was obtained, which demonstrated small bowel obstruction secondary to interval increase in the size of tumor burden and new hepatic metastatic lesions Figure . She wasUmbilical and periumbilical swellings can broadly be divided into benign and malignant lesions. The benign lesions can have broad differentials including hernia, cysts, and endometriosis. Malignant lesions can arise from primary tumors or secondarily to cancers in the abdomen and pelvis. Literature review suggests that these umbilical lesions can represent as primary tumor in 38% of the cases, endometriosis in 32%, and metastatic lesions in 30% . The priSMJN usually presents as a palpable periumbilical nodular mass with diameter usually not exceeding 5 cm in size. It can be variable in color ranging from violaceous to reddish brown and occasionally have skin ulceration with serous or purulent discharge . UmbilicIn cases of abdominal and pelvic malignancies, several mechanisms have been defined for possible spread of metastasis to the umbilicus giving rise to SMJN. These include direct hematogenous spread or lymphatic spread of the neoplasm. On rare occasions, seeding of malignant cells may also happen to the umbilicus during surgical manipulation of abdominal viscera . As SMJNManagement of SMJN is mainly palliative. Modalities such as surgical excision and chemotherapy have proven to have limited benefit. As seen in our clinical scenario, the colon cancer had aggressively metastasized, and surgical resection was not an option. Ultimately, the patient opted for palliative care with no further aggressive measures. Given her extremely poor prognosis, she was discharged to hospice with comfort measures only . SMJN isSMJN is a rare clinical finding that is described as a neoplastic mass in the periumbilical region. Its differentials include benign conditions such as cysts, hernia, endometriosis, and abscess. SMJN itself suggests underlying malignant process  and warrants further investigation including biopsy and imaging studies. It can be described as an indicator of poor prognosis in malignant process with limited survival expectancy, and therefore it is important to not miss this important and classic clinical finding in a patient.


Pleomorphic adenomas are benign tumors of the salivary glands that mainly affect the lower pole of the superficial lobe of the parotid gland. The term “pleomorphic” refers to the epithelial and connective origin of the mass. The clinical presentation is typically that of asymptomatic swelling which increases in volume. Therapy consists in surgical removal of the tumor mass by parotidectomy with nerve preservation.This clinical case describes an interesting case of pleomorphic adenoma of the parotid gland in a 62-year-old patient. The patient presented with a long history of an asymptomatic mildly worsening swelling of the left parotid region. The peculiarity of the clinical case is the dimension of the adenoma (9x5x9 cm) and the presence of a thyroid incidentaloma (TI), consisting of a thyroid multinodular goiter composed of nodules, the largest of which measured 8 cm in diameter. This mass dislocated the laryngotracheal axis, compressed the larynx and caused the reduction of the respiratory space, making orotracheal intubation difficult and determining the need to perform a tracheotomy.Benign pleomorphic adenomas can potentially reach large sizes if untreated. Socio-economic problems may be the reason for late diagnosis. Salivary gland tumors represent 1–3% of head and neck cancer, they mainly affect the parotid gland and 70–80% are adenomas (1). Among benign neoplasms, the most frequent histotype is pleomorphic adenoma (PA). PA is more common in women with a male:female ratio of 1:2 and is more prevalent between the fourth and sixth decade of life (2–6).PA is a mixed tumor that originates from epidermoid and myoepithelial cells; it includes mesenchymal tissues  and epithelial tissues usually with adenomatous appearance , hence the term “mixed tumor” . PA is characterized by a slow but progressive growth and, if untreated, can reach a significant size (9), similarly to other benign head and neck masses such as lipoma (10). The diagnosis of PA is based on both clinical and radiological exams, including imaging techniques such as B-mode Ultrasound, Magnetic Resonance Imaging (MRI) and Computed Tomography (CT), followed by fine needle aspiration (FNA) for preoperatory cytological diagnosis .PA can undergo malignant transformation to carcinoma ex-pleomorphic adenoma (13–15); rarely it metastasizes without malignant transformation (16).Treatment of PA is surgical; recurrence may occur mainly due to intraoperative multifunctional cellular effusions and cellular dissemination caused by difficulty in completing removal of the pseudocapsule (17). Historically, the incidence of recurrence was estimated between 20% and 40%; today, with the most modern parotidectomy techniques, recurrence is estimated around 2.5% (18–20). The management of recurrent pleomorphic adenoma of the parotid gland is challenging because the tumor is often multinodular and can be associated with lesions to the facial nerve .A 62-year-old female patient was admitted to the Otolaryngology Department of our university hospital reporting a painless swelling of the parotid region appeared several years earlier and slowly increased in volume.The patient was suffering from high blood pressure, severe obesity (BMI: 37), permanent atrial fibrillation and iatrogenic hypothyroidism. The patient regularly assumed nebivolol, furosemide and levothyroxine. In 1999, the patient had undergone a left hemithyroidectomy for thyroid nodularity.Physical examination showed facial symmetry with normal movements of the face and eyes. The swelling in the left parotid region had a multilobular appearance and an elastic consistency; it was not painful on palpation and the overlying skin were hyperemic.Fibrolaryngoscopy showed a paralysis of the left hemilarynx, probably due to the previous thyroid surgery; respiratory space was reduced. The patient underwent CT scan with contrast agent that detected a solid hypervascularized mass in the superficial lobe of the left parotid gland measuring 9x5x9 cm  according to the 2014 Italian Consensus for Classification and Reporting of Thyroid Cytology classification (22).Patient underwent a combined parotidectomy and thyroidectomy ; a tempoHistological examination of the parotid specimen revealed a capsulated, solid and cystic mass characterized by the presence of a wide squamous component containing partially calcified corneal cysts and by multifocal proliferation of epithelial cells, which formed structures that resembled glandular lumina and spindle-shaped myoepithelial cells bearing pale cytoplasm. A hyaline and chondromyxoid extracellular matrix was present. There was evidence of multiple granulomatous inflammation foci containing multinucleated giant cells, even within the capsule. The features of chondromyxoid foci and glando-duttal differentiation and the low growth rate (ki67 <5%) confirmed the diagnosis of PA with extensive squamous metaplasia. The histological exam also revealed reactive nodes, negative for tumoral invasion. Histological examination of the thyroid mass showed multiple colloid nodules in both lobes, characteristic of multinodular goiter (23–25). The parenchyma had histological features of macro and microfollicular stroma, with areas of focal necrosis sclerosis and inflammatory infiltration.The patient had no postoperative complications; tracheostomy was removed 2 weeks after surgery and showed no recurrence of disease at regular follow up visits .This case report shows an obese patient with a large PA who presented in a late stage of the disease. In addition, the patient presented a contextual finding of a TI, a MG whose largest nodule reached 8 cm in diameter. The presence of vocal fold paralysis probably due to previous hemithyroidectomy, the very voluminous MG and the displacement of the laryngotracheal axis complicated the surgical intervention.PA or benign mixed tumor is the most common salivary gland neoplasm; in 80% of cases it involves the parotid gland, in 8% the submandibular gland, and in 6% the minor salivary glands (5). Generally, PA involves the lower pole of the superficial lobe of the parotid gland, is not painful and presents solid consistency . Facial nerve palsy is a rare occurrence (27), and was not found in this patient despite the large size of the parotid mass.Although encapsulated, in some locations the capsule is not fully developed and expansile growth produces protrusions into the surrounding gland, which may lead to recurrences if the tumor is enucleated. In the present case, a radical parotidectomy was performed with removal of the pseudocapsule; no recurrence of the parotid mass was found during follow up.The dominant histologic feature found in this case of PA is the great heterogeneity. The epithelial elements resembling ductal cells or myoepithelial cells are arranged in duct formations, acini, irregular tubules, strands, or sheets of cells. These elements are typically dispersed within a mesenchyme-like background of loose myxoid tissue containing islands of cartilage and, rarely, foci of bone. Sometimes, the epithelial cells form well-developed ducts lined by cuboidal to columnar cells with an underlying layer of deeply chromatic, small myoepithelial cells. In other cases, there may be strands or sheets of myoepithelial cells. Islands of well-differentiated squamous epithelium may also be present. In most cases there is no epithelial dysplasia or evident mitotic activity. There is no difference in biologic behavior between the tumors composed largely of epithelial elements and those composed largely of seemingly mesenchymal elements (28).Malignant transformation occurs in nearly 3% of cases, is usually linked a long-standing, untreated condition and has to be suspected in case of pain, ulceration, spontaneous bleeding, superficial and/or deep tissue invasion, rapid growth and facial nerve alterations .In conclusion, the present case shows a peculiar situation in which a PA and TI were found in the same patient at a very late stage. PA may reach significant sizes if untreated, and socio-economic problems can be a reason for late diagnosis and treatment.


Lingual ASPS is extremely rare and aggressive tumor. rearrangement is typically detected in ASPS patients using FISH analysis.To present the clinical, histopathological, and radiological features of lingual Alveolar Soft Part Sarcoma.A 30‐year‐old male presented with a painless, slowly growing mass of the tongue. Initial impression was of benign vascular lesion. Later, the patient became symptomatic as the mass progressed in size, which necessitated further investigations.A lip‐split, mandibulotomy was performed for the excision of the tumor and revealed an alveolar soft part sarcoma with PAS‐positive, diastase resistant intracytoplasmic granules. However, molecular analysis using FISH was negative for TFE3 rearrangement. Patient underwent partial glossectomy with postoperative radiotherapy.Clinical and pathological correlation of ASPS is very useful to reach a proper diagnosis. The mass was slowly progressing in size. There were no other associated symptoms. Clinical examination revealed a nontender swelling on the left posterior part of tongue. The mass was firm in consistency, oval in shape, and had an intact mucosal covering, not associated with ulceration, and was not attached to any underlying tissue. Neck examination revealed palpable ipsilateral submandibular and jugulodigastric lymph nodes. A clinical diagnosis of hemangioma was suspected. CT angiogram revealed a left sided highly vascular lesion. Three months later, the patient came back with symptomatic dysphagia, chewing difficulty, and difficulty in breathing especially while sleeping, and CT scan showed a slight increase in the size of the mass.Since the possibility of tongue cancer could not be ruled out in the presence of alarming compressive symptoms, incisional biopsy was planned. During the procedure, a significant bleeding was encountered which was difficult to be controlled with diathermy, local anesthesia, pressure packing and surgicel. Thus, it was decided to abort the procedure after hemostasis was completely secured, and the patient was extubated without any complications. Following incisional biopsy, selective embolization of the left lingual artery achieved a near complete devascularization of the mass. Then the patient was sent back for lip‐split, mandibulotomy for access and excision of the left tongue tumor. Suturing on the muscle and in the mucosa was performed with 2‐0 Vicryl and 3‐0 Vicryl and the defect was closed primarily. Postoperative period was unremarkable.Three weeks after excision of the tumor, radiological evaluation was performed using PET/Scan and neck MRI. PET/Scan showed a moderate hypermetabolism at the site of surgical site and at the level of 2A lymph nodes with no evidence of distant metastasis. Neck MRI showed postsurgical changes of the tongue with enhancing left upper cervical lymphadenopathy.Following the excision of the tumor, the specimen was sent to the Anatomic Pathology Department. Grossly, it was a cauliflower‐like ulcerating mass lesion which was 4.8 cm in greatest dimension. Histopathological examination revealed a malignant neoplasm formed of large, polygonal cells with clear to lightly esinophilic cytoplasm and hyperchromatic nuclei arranged in a pseudoalveolar pattern with prominent capillary vasculature. Perineural invasion and extensive lymphovascular invasion were present. PAS‐positive, diastase resistant intracytoplasmic granules were present Figure .Immunohistochemical studies ruled out mimickers Figure . Tumor cAs the histopathology report showed involved surgical margin, patient agreed to have further resection of the lesion through partial glossectomy. Further excision revealed multiple residual foci of ASPS and a close deep resection margin. Left hemiglossectomy plus reconstruction of the left side of the tongue and the floor of the mouth with radial forearm free flap were performed. Adjuvant radiotherapy was completed 4 months postoperatively. A follow up period of 1 year was unremarkable except for multiple bilateral lung micronodules that remained unchanged on successive evaluation with chest imaging. Patient was stable and fully able to perform daily activities without restriction.3Alveolar soft part sarcoma (ASPS) mainly affects young adults and comprises less than 1% of all soft tissue sarcomas.It is still controversial whether ASPS is of neuroendocrine or myogenic origin. Initially, ASPS was thought to be a malignant variant of granular cell myoblastoma which comes from a neuroendocrine origin. However, recent immunohistochemical studies have proven the absence of neuroendocrine differentiation.ASPS could be confused with hemangioma since both look similar in term of clinical and radiological pattern. Unlike hemangioma, ASPS is not present at birth and the growth pattern of the lesion is typically slow. Hemangioma appears as a well‐defined hyperattenuated lesion in CT without contrast and appears homogeneously intense in CT with contrastHistologically, ASPS often looks benign in a deceptive manner. Cells are typically of uniform size and shape with eosinophilic cytoplasm and prominent nucleolus without atypical mitosis or nuclear pleomorphism. ASPS characteristically appears in a pseudoalveolar pattern as alveolar cells get necrotic centrally and lose their cohesiveness Figure .2 PosiRecently, it was proposed that the unbalanced translocation, del (17) t , which results in the formation of ASPL‐TFE3 transcript fusion detected on tumor cell, could specifically explain the tumorigenesis of ASPS.Although FISH analysis remained extremely sensitive and specific to detect Xp11.23 translocation associated with ASPS and Renal cell carcinoma (RCC), the possibility of chromosomal abnormalities could not be ruled out by FISH analysis alone.Complete Surgical excision of the primary tumor is the treatment of choice to prevent local recurrence. Bleeding might be encountered, especially if the tongue lesion is located posteriorly.4Lingual ASPS is an extremely rare tumor. To the best of our knowledge, this is the first case to report ASPS of the tongue in Saudi Arabia. Clinically and radiologically, ASPS can be misdiagnosed as hemangioma. The nature of tumor location and behavior made the diagnosis and management of this case more challenging. Therefore, careful evaluation of any hypervascular growing mass of the tongue should be done.Histologic findings provide a reliable diagnostic method. Although molecular methods provide a great support to the diagnosis of ASPS, the absence of TFE3 fusion gene does not always rule out ASPS. Preoperative embolization for ASPS might be useful to avoid the risk of extensive bleeding during surgery.Informed consent was obtained to publish this report.The authors declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.Saeed Alshahrani: wrote and edited the manuscript.Abdullatif Khan: contributed to the writing and literature review.


Onco‐TESE is a useful strategy for cancer patients with a congenital single testis who wish to preserve their fertility. Onco‐TESE is a useful strategy for cancer patients with a congenital single testis who wish to preserve their fertility. The patient underwent orchidectomy and simultaneous ipsilateral oncological‐testicular sperm extraction, which collected viable sperm for cryopreservation. The sperm was used for intracytoplasmic sperm injection, and a healthy child was ultimately born.Testicular germ cell tumors (TGCTs) are most commonly found in men who are 15‐35 years old and account for 1.0‐1.5% of neoplasms in men.Although the American Society of Clinical Oncology recommends sperm cryopreservation to preserve fertility in this setting, few patients receive infertility treatment,2A 26‐year‐old Japanese man noticed painless enlargement of his right scrotum and visited the urology department at a nearby hospital. He had been diagnosed with cryptorchidism during childhood and had undergone laparotomy, which failed to identify his left testis and he was subsequently diagnosed with a left testicle defect. At his presentation, the right testis was swollen to a diameter of 5 cm and ultrasonography confirmed a 5 cm hypoechoic mass in the right testis. Plain computed tomography revealed a uniform approximately 5 cm mass inside the scrotum with no obvious metastasis. Laboratory tests revealed high concentrations of human chorionic gonadotropin (HCG: 16.6 mIU/mL) and human chorionic gonadotropin‐β (HCG‐β: 0.46 ng/mL), with nearly normal results for α‐fetoprotein (2.3 ng/mL), luteinizing hormone (2.1 mIU/mL), follicle‐stimulating hormone (4.7 mIU/mL), and testosterone (470 ng/dL).The patient had a fiancé and wished to cryopreserve his sperm, although azoospermia was identified based on twice semen tests that were performed at the assisted reproductive therapy clinic. His karyotype and Y chromosome were normal and without microdeletions. He was referred to our hospital and underwent radical orchiectomy with planned sampling of any non‐cancer seminiferous tubule tissue to collect sperm if possible. The orchiectomy revealed that most of the testicle was occupied by a gray‐white mass without bleeding or necrosis Figures  and 2, aHistopathological examination revealed tumor cells that contained lymphocytes and clear cytoplasm, which supported a diagnosis of pure seminoma (pT1N0M0) Figure . SpermatThe patient's postoperative course was uneventful, with normalization of his HCG and HCG‐β concentrations. No adjuvant therapy was performed, although close follow‐up was performed using imaging and blood tests. No obvious tumor recurrence has been observed, and the patient is receiving continuous testosterone supplementation because of low testosterone concentrations after the orchiectomy. The patient subsequently married his fiancé, who had normal gynecological findings based on hysteroscopy, transvaginal ultrasonography, and blood tests. Thus, ICSI was performed using the cryopreserved sperm, the wife became pregnant, and a healthy boy was born on gestational week 39 + 3 .3In this case, the patient was preoperatively diagnosed with azoospermia, and onco‐TESE was the only option to preserve his fertility because the congenital single testis needed to be removed. Testicular tumors are generally associated with decreased spermatogenesis, with patients having approximately 33% of the sperm concentration and 17% of the total sperm count, relative to men in the general population.The reported frequency of congenital single testis diagnosed during surgery for cryptorchidism is 4% in Europe and the United States.4We performed Onco‐TESE during radical orchiectomy and were able to preserve the fertility of a patient with a congenital single testis. This technique successfully retrieved sperm that were cryopreserved and used for ICSI, which ultimately resulted in the birth of a healthy child. Therefore, Onco‐TESE is a useful strategy for cancer patients with a congenital single testis who wish to preserve their fertility.None declared.All authors: read and reviewed the manuscript. HM: performed the orchidectomy, Onco‐TESE, and postoperative follow‐up. TI and HA: reviewed the manuscript. HO: referred the patient to our clinic and successfully performed ICSI with the cryopreserved sperm.


Primary intraosseous osteolytic meningiomas are not common, and total surgical removal is the best treatment option if the location allows that; however, long‐term follow‐up is recommended since it may recur. Primary intraosseous osteolytic meningiomas are not common, and total surgical removal is the best treatment option if the location allows that; however, long‐term follow‐up is recommended since it may recur. A 45‐year‐old obese woman presented with headache only for six months, and skull lesion was diagnosed. Total surgical removal was done, and the histopathology result revealed meningioma. Primary intraosseous meningiomas are usually benign, and their growth correlates with their histological grade and can show malignant changes more than its intracranial equivalent. Total surgical resection is the treatment of choice with safe margins. A long‐term follow‐up period is recommended even if total resection is carried out.Meningiomas are the most frequent intracranial benign neoplasia; they arise from arachnoid granulations and represent approximately from 14% to 20% of intracranial tumors.In this article, we present a case of intraosseous osteolytic meningioma and describe the steps of management.2A 45‐year‐old obese woman with history of diabetes mellitus and hypertension presented with headache for six months of duration. Neurological examination was normal, and there was no scalp mass or deformity. Computed tomography scan (CT scan) revealed hypodense (osteolytic) lesion occupying the right temporoparietal region with destruction of inner and outer table, differential diagnosis was suspected and includes fibrous dysplasia, aneurysmal bone cyst, and primary intraosseous meningioma. She underwent magnetic resonance imaging (MRI) that revealed hypointense in T1 and hyperintense in T2 with homogenous contrast enhancement lesion in the right temporoparietal region with mild compression of the right ventricle, which most likely represent a meningioma. Surgical resection was planned; a large question mark skin incision was made, after periosteal dissection and reflection of temporalis muscle; en bloc resection of tumor with safe margins was done; dura was intact; we control the feeders of the tumor, which were mainly from middle meningeal artery branches, wound closed in layers; and cranioplasty was postponed for another time to shorten the operation time due to patient medical condition (Figure 3This neoplasm has no gender predilection, and it has two peaks of incidence in the second decade and between the fifth and seventh decades with a median age of 40‐49 years,4Primary intraosseous meningiomas are usually benign, and their growth correlates with their histological grade and can show malignant changes more than its intracranial equivalent. Total surgical resection is the treatment of choice with safe margins. If there is doubt about complete resection, the lesion should be followed with appropriate imaging studies. A long‐term follow‐up period is recommended even if total resection is carried out.The authors declare no conflict of interest; the manuscript is not published elsewhere.AD and SH: conceived and designed the study. GL: involved in overall supervision of the paper. All authors: read and approved the final manuscript.