[Thoracoscopic resection of recurrent atypically located parathyroid adenoma of anterior mediastinum in a patient with hyperparathyroidism undergoing renal replacement therapy]. / Torakoskopicheskoe udalenie retsidivnoi atipichno raspolozhennoi adenomy okoloshchitovidnoi zhelezy perednego sredosteniya u patsientki s giperparatireozom, nakhodyashcheisya na zamestitel'noi pochechnoi terapii.

We present successful surgical treatment of a patient with chronic kidney disease (CKD) and hyperparathyroidism undergoing renal replacement therapy. At baseline, parathyroidectomy via cervical access was performed for parathyroid adenomas. After 6 years, clinical and laboratory relapse of disease required thoracoscopic resection of atypically located anterior mediastinal adenoma. This case demonstrates that this disease is one of the most difficult in modern medicine requiring a special approach in diagnosis and treatment. Patients with CKD and hyperparathyroidism need for follow-up, control of total and ionized serum calcium, inorganic phosphorus and parathormone, osteodensitometry, ultrasound and scintigraphy of thyroid and parathyroid glands, and, if necessary, CT or MRI of the neck and chest organs.

Pilar Cyst of the Dorsal Hand: A Rare Presentation of a Common Cyst.

Pilar cysts are common benign cysts of follicular origin that typically arise in areas of skin containing dense hair follicles such as the scalp. Here we describe a unique case of a young woman who was found to have a pilar cyst on the dorsum of her hand, a rather atypical location given the relative lack of pilosebaceous units. This case illustrates the variability in pilar cyst presentation and the importance of considering a pilar cyst in the differential diagnosis of a patient presenting with a tumor of the dorsal hand.

Lymphoplasmocytic plaque in children: A report of an atypical location.

Lymphoplasmocytic plaque in children (LPC) is a rare and distinctive skin disorder primarily affecting the pediatric population. Characterized by its unique histopathological features, the condition manifests as well-defined plaques with a predominance of lymphocytes and plasma cells infiltrating the dermis. Despite its limited prevalence, recognizing this entity is crucial for accurate diagnosis and appropriate management of affected patients. We report the case of a 10-year-old male presenting with LPC in the extensor surface of the upper arm, a rarely reported location, treated with both topical and intralesional corticosteroids resulting in partial improvement.

Typical Zollinger-Ellison syndrome-atypical location of gastrinoma and absence of hypergastrinemia: A case report and review of literature.

BACKGROUND: Zollinger-Ellison syndrome (ZES) results from hypersecretion of gastrin from pancreatic or duodenal neuroendocrine tumors, commonly referred to as gastrinomas. The high levels of gastrin lead to a typical presentation involving watery diarrhea and multiple ulcers in the duodenum. Here, we have presented the rare case of a patient with ZES and absence of hypergastrinemia as well as an atypical location of gastrinoma. CASE SUMMARY: A 72-year-old woman presented with the typical clinical manifestations of ZES, including upper abdominal pain, significant watery diarrhea, and acidic liquid vomitus. Surprisingly, however, she did not have an increased level of serum gastrin. In addition, there was no evidence of gastrinoma or any other ulcerogenic tumor. Esophagogastroduodenoscopy was conducted to examine the upper digestive tract. Revised diagnoses were considered, and an individualized treatment plan was developed. The patient responded to antacid medication while experiencing intermittent, recurring bouts of ZES. 18F-AlF-NOTA-octreotide positron emission tomography (18F-OC PET)/computed tomography (CT) helped locate the tumor. Postoperative pathology and immunohistochemistry results suggested that the tumor was a gastrinoma located at an unconventional site. CONCLUSION: This present case study demonstrates the possibility of ZES-like manifestation in patients with absence of hypergastrinemia. 18F-OC PET/CT is a relatively new imaging technique that can be applied for diagnosing even tiny gastrinomas that are atypical in terms of location.

Intracranial cholesteatoma in the thalamus: Uncommon location and atypical imaging findings.

Intracranial cholesteatoma is an uncommon condition that can occur anywhere in the intracranial cavity, but its occurrence in the thalamus is exceedingly rare. We present a case of thalamic cholesteatoma with atypical imaging findings and review the previous literature to explore the pathophysiological basis of these findings. A 55-year-old male presented to the hospital with right upper limb weakness and right ear hearing loss for more than 2 years. Imaging studies of the brain were performed, revealing a mixed density mass in the left thalamic region with calcification at the margin. Magnetic resonance imaging revealed a mixed signal intensity on T1-weighted and T2-weighted images, diffusion limitation on diffusion-weighted images, and ring enhancement on enhanced T1-weighted images. The mass caused mild obstructive hydrocephalus due to compression of the 3 ventricles and midbrain aqueduct. Thalamic cholesteatoma is an uncommon and often misdiagnosed lesion due to its unusual location and imaging characteristics. The pathophysiological basis of the atypical imaging findings associated with intracranial cholesteatoma is not well understood but may be related to the keratinization of the cyst lining or the presence of cholesterol crystals. Thalamic cholesteatoma should be considered in the differential diagnosis of thalamic lesions, especially in cases with calcifications or cystic components. Further studies are needed to better understand the pathophysiology of intracranial cholesteatoma and its associated atypical imaging findings.

Isolated Elastofibroma of the Thigh: A Case Report.

A 55-year-old female presented with elastofibroma of the thigh. On presentation, she complained of a palpable, painful mass on the anterolateral right thigh that had been present for one year. She had a history of surgery for a right femur fracture. On MRI, a soft-tissue mass was seen in the vastus intermedius muscle, as a heterogeneous lesion with streaky fatty and fibrous components. The fibrous component was isointense to the muscle, and the fatty component had a high signal on both T1- and T2-weighted images. Histopathological analysis after biopsy established the diagnosis of elastofibroma.

Submental liposuction with VASER complicated with hidradenitis suppurativa in neck area: a case report.

Hidradenitis suppurativa (HS) is a chronic inflammatory disorder that is characterized by deep-seated painful nodules, classically in the intertriginous skin and apocrine gland-rich areas of the body such as the anogenital, axillary, inframammary and inguinal regions. This is a case of a 35-year-old female, who is known to have gluteal HS, she underwent neck liposuction procedure that was then complicated by anterior neck HS, which is considered as an atypical location. The patient received medical treatment with antibiotics and showed huge improvement. In addition, in patients who do not show response to medical therapy, surgical treatment is usually carried out by incising the area affected and leaving the wound open to be healed by secondary intention or covering it with a skin graft if the area is extensive.

Atypical mid rectal localization of squamous cell carcinoma: A case report and literature review.

INTRODUCTION AND IMPORTANCE: Squamous cell carcinoma occurring in the rectum is a very rare malignancy. When encountered in the gastrointestinal tract, it usually involves the esophagus or the anal canal. The rare incidence of rectal squamous cell carcinomas has raised quite a few questions on the hypothetical etiologies and prognosis. CASE PRESENTATION: In this report, we present a case of a 73 years old woman who presented a rare case of squamous cell carcinoma, at 8 cm from the anal margin. CLINICAL DISCUSSION: Optimal treatment sequence of such an uncommon disease is yet to be standardized, surgery was the gold standard management for rectal squamous cell carcinoma, but exclusive chemoradiotherapy is slowly but surely supplanting it. CONCLUSION: This case allows us to engage in discussions over the uncommon location of the rectal SCC and its current treatment management. The exclusive chemoradiation therapy has given excellent results becoming the gold standard treatment of this rare entity.

Paediatric-type diffuse high-grade gliomas in the 5th CNS WHO Classification.

As a relevant element of novelty, the fifth CNS WHO Classification highlights the distinctive pathobiology underlying gliomas arising primarily in children by recognizing for the first time the families of paediatric-type diffuse gliomas, both high-grade and low-grade. This review will focus on the family of paediatric-type diffuse high-grade gliomas, which includes four tumour types: 1) Diffuse midline glioma H3 K27-altered; 2) Diffuse hemispheric glioma H3 G34-mutant; 3) Diffuse paediatric-type high-grade glioma H3-wildtype and IDH-wildtype; and 4) Infant-type hemispheric glioma. The essential and desirable diagnostic criteria as well as the entities entering in the differential will be discussed for each tumour type. A special focus will be given on the issues encountered in the daily practice, especially regarding the diagnosis of the diffuse paediatric-type high-grade glioma H3-wildtype and IDH-wildtype. The advantages and the limits of the multiple molecular tests which may be utilised to define the entities of this tumour family will be evaluated in each diagnostic context.

Pigmented Villonodular Synovitis Presenting at an Atypical Site: A Case Report.

Pigmented villonodular synovitis (PVNS) is an idiopathic villous overgrowth and pigmentation of the synovial membrane of a single joint. It is an uncommon condition characterized by yellow or yellowish-brown colour due to deposits of cholesterol and hemosiderin, excessive secretion of yellowish-brown synovial fluid, and the formation of brownish chocolate synovial tissue. This condition commonly occurs at the knee joint at the age of 20-50 years. Here we present a case of a 75-year-old male with PVNS involving the lower third of the right thigh who came to the surgery department and was eventually referred to the orthopedic department on the basis of investigations performed. It is a case of PVNS at a unique location on the lower third of the right thigh. The swelling was painless initially, but the pain increased over a duration of 10 months. Clinically, the mass was suspected to be bursitis or lipoma with features of mild inflammation in the overlying skin. Magnetic resonance imaging (MRI) suggested a swelling of 100*70*40 mm in dimension with the possibility of PVNS. Ultrasonography (USG) of the mass and fine needle aspiration cytology (FNAC) supported the diagnosis of PVNS. An excisional biopsy of the swelling was submitted. Per-operatively, there were typical features of PVNS. The swelling was situated superficial to the iliotibial band. There was no defect or gap in the iliotibial tract, and the swelling didn't have any continuity to the knee joint. The occurrence of synovial tissue without any attachment to the joint or tendon is rare and hence reported.

Unusual suicidal penetrating heart injury by captive-bolt gunshot.

We present a case of a 56-year-old man who committed suicide using a captive-bolt gun, pressed against the left side of his chest. As the victim worked as a butcher, slaughtering livestock, he owned a captive-bolt gun. Just before committing suicide, he received a disturbing phone call from a person to whom he owed money. The autopsy revealed a 12-mm wound in the left pectoral region with two symmetrical, oval soot deposits. The wound extended through the fifth left rib, the pericardium, and the inferior portion of the anterior wall of the left ventricle. There was a partial-thickness tear in the left aspect of the interventricular septum with associated contusion. About 300 ml of the blood was found within the pericardial sack and about 1200 ml in the left pleural cavity. Atypical location (left chest vs. head), absence of previous suicide attempts and suicide note, and the apparent immediate provoking event suggest abrupt rather than premeditated suicide.

Pleomorphic adenoma in the external auditory canal: Arising at an atypical location.

Pleomorphic adenoma is a common, benign neoplasm typically located in the major salivary glands. The external auditory canal is an atypical location for a pleomorphic adenoma to originate. Wide surgical excision is essential in cases of pleomorphic adenoma due to its tendency to recur and change into malignancy. We report a case of pleomorphic adenoma emerging in the external auditory canal, which is an atypical location for it to originate. A 23-year-old male presented with swelling and decreased hearing in the left ear for 2 months. On examination, there was a smooth, pink, firm swelling in the cartilaginous part of external auditory canal. Pure tone audiogram and computed tomography of temporal bone were performed. The patient underwent excision biopsy and the diagnosis of pleomorphic adenoma was confirmed after histopathology. Long-term follow-up is recommended as pleomorphic adenoma has the potentiality to recur or transform into malignancy.

Extradigital Glomangioma of the Cutaneous Chest Wall.

Glomus tumors (GTs) are rare benign tumors as a result of hyperplasia of glomus body. GT most commonly involves the subungual areas and rarely involves extra-digital sites. The clinical presentation of a glomus tumor is a triad of symptoms consisting of pain, cold intolerance, and pinpoint tenderness. Even though glomus tumors are benign, they can infrequently be malignant. Despite their benign nature, these lesions can cause disabling symptoms, therefore proper diagnosis and treatment is important. In this report, we present a 35-year-old Saudi male with a painful lesion on the right side of the chest wall at the posterior axillary line for seven years, with recent progressive growth and symptoms. Diagnosis of extra-digital glomangioma of the chest wall in this patient was confirmed by histopathology. The patient was managed by complete surgical excision of the lesion with the resolution of pain and without recurrence.

Atypical Congenital Triangular Alopecia (Brauer Nevus): Case Report and Review of Literature in Occipital and Mid-Frontal Localizations.

INTRODUCTION: Brauer nevus, also known as congenital triangular alopecia (CTA) is a localized alopecia that rarely affects the occipital or mid-frontal region. CTA is a localized follicular hypoplasia, commonly misdiagnosed as alopecia areata. Although named congenital, onset in adulthood is possible. CASE PRESENTATION: We present a review of literature of eighteen atypical locations, providing 4 new cases to the 9 previously published, with particular attention to trichoscopy and histopathological descriptions in this exceptional presentation forms. DISCUSSION: Occipital and mid-frontal Brauer nevus are unusual findings with very few cases reported so far. Because of its rarity and atypical presentation, it can often lead to a misdiagnosis. A higher incidence in the male sex stands clear in these locations. Detection at birth is slightly more frequent than in the classical CTA. Histopathological and trichoscopy findings do not differ from the classical entity, although white or hypopigmented hairs do not seem to be an item present in the mid-frontal forms.

Atypical purpura location in a Sudanese infant with Henoch-Schönlein purpura.

Henoch-Schönlein purpura (HSP) is a self-limited systemic vasculitis seen most commonly in paediatric group with multi organ involvements. We report a case of an infantile female who presented with cough, diarrhoea and vomiting. Two days later, she suddenly developed an erythematous, non-pruritic rash involving feet, thighs, buttocks and face; the lesion spared the trunk. Joints pain and swellings were also noticed. Coronavirus disease-19 Polymerase chain reaction (PCR) test was negative. She was diagnosed with HSP based on the American College of Rheumatology and European League Against Rheumatism, and Paediatric Rheumatology European Society criteria. The rash faded spontaneously within 2 weeks. We conclude that the careful skin examination is crucial for diagnosis of HSP.

Extralobar sequestration with a pulmonary arterial feeding vessel.

Extralobar sequestrations are rare congenital malformations of the lung. They are usually located between the diaphragm and left lower lobe and receive their blood supply from the aorta. We report a case of extralobar sequestration in an atypical location with a pulmonary arterial feeding vessel. An 18-year-old woman presented with an abnormality on chest X-ray. Chest computed tomography revealed a lung field not communicating with the bronchus between the upper and lower right lung lobes. Three-dimensional reconstruction computed tomography demonstrated a feeding artery from the pulmonary artery draining into the pulmonary vein. We diagnosed her with extralobar sequestration and resected the sequestered lung using video-assisted thoracic surgery. Therefore, three-dimensional reconstruction computed tomography helped identify the abnormal blood vessels, and video-assisted thoracic surgery may be useful in the treatment of extralobar sequestration.

Is radiofrequency ablation safe and effective in treating osteoid osteomas? A prospective single-center study with atypical cases.

PURPOSE: Osteoid osteomas are benign and nonprogressive lesions. The clinical presentation of osteoid osteoma is typical with night pain responsive to nonsteroidal anti-inflammatory agents. The typical radiological appearance is lucent nidus and adjacent reactive sclerosis. The most traditional management of osteoid osteoma is surgical removal which is associated with significant morbidity. This article aims to demonstrate our single-center experience of 55 patients with osteoid osteoma treated with percutaneous radiofrequency ablation (RFA). METHODS: Fifty-five symptomatic patients who were seen at our orthopedics outpatient clinics, diagnosed with osteoid osteoma, and referred to interventional radiology department between May 2015 and April 2019 were enrolled. The nidus size, pain numeric rating scale score, and intramedullary edema diameter before and after RFA were compared. Clinical and technical success, intervention-related complications, and need for subsequent ablation were recorded. RESULTS: Of the 55 patients, including 12 pediatric cases, 6 had atypical locations such as metatarsal, vertebra, and scapula, while 1 case had osteoid osteoma with multiple nidus. The mean age was 18.5 ± 9.6 years. Preintervention maximum nidus diameter, pain score, and edema diameter were significantly lower in postintervention measurements (p < 0.001). Technical success rate was 98.1%, and clinical success was 96.36%. Severe complication occurred in one patient with soft tissue burn and osteomyelitis. CONCLUSION: This is one of the largest series of osteoid osteoma treated with RFA done in a single center with atypical cases. In conclusion, RFA is safe and effective in both pediatric and adult patients with rare complications even in challenging cases with atypical locations.

[Atypical location of lenticular coloboma]. / Localisation atypique d'un colobome cristallinien.

We report the case of a 37-year old patient with no particular past medical history presenting to the Ophthalmology Department for lens correction. Examination of the right eye showed corrected visual acuity of 6/10e, refractive error of -1.50 (-6,25 to 56°); the examination of the anterior segment objectified lenticular coloboma in the superior temporal area with zonular deficiency ranging from 8h to 11h and opacity of the crystalline. The examination of the iris and fundus examination were normal. Left eye exam was normal. Colobomas are congenital malformations of the eye secondary to an abnormal closure of the foetal fissure. Lenticular coloboma is rare, secondary to uveal coloboma resulting in indentation of the lens periphery. It usually occurs in the inferonasal region. The peculiarities of our case study is the occurrence of lenticular coloboma in the superior temporal area. It is essential to search for opacity of the crystalline in patients with other colobomatous abnormalities and associated ocular malformations such as cataract, microphthalmia or ectopia of the crystalline.

Extranasopharyngeal angiofibroma revisited.

BACKGROUND: Angiofibromas in the head and neck region usually arise in the nasopharynx, but may also occur elsewhere. This study aims at evaluating the incidence and clinical features of extranasopharyngeal angiofibroma (ENA). MATERIAL AND METHODS: Systematic review of the literature (Medline® and Google™ ) up to 31 December 2015. RESULTS: 174 cases of ENA were retrieved from a total of 170 publications. In contrast to former publications and previous understanding, the nasal septum was by far the most common site of the disease. Four patients had a congenital lesion, the oldest patient was 87 years old (mean: 28.7 years; median: 23 years). Male gender was predominantly affected, but the sex ratio was more balanced (2.13:1) than in previous reports in the literature until 12/2015. The majority of patients presented with nasal obstruction, either in combination with epistaxis (25.8%) or other symptoms (12.6%). Symptoms had developed within 13.1 months on average (median: 4 months). Brisk bleeding resulted in 11 of 43 biopsy procedures. Surgical resection as first-line therapy was performed in 170 patients. A tumour regrowth within 12 months was registered in four patients. CONCLUSION: The increasing awareness of ENA and the willingness to publish case reports-not only in Medline-listed journals-resulted in a significant increase of published case reports lately. Although extremely rare, ENAs have to be taken into account in the differential diagnosis of unclear masses, particularly in adult patients presenting with a rapidly developing nasal obstruction resulting from a nasal septum tumour. Female gender or normal vascularity does not exclude the diagnosis. Transnasal resection is sufficient in most cases, and recurrences are rare. Pathologists as well as clinicians should consider ENA in their differential diagnosis of any mass of the upper airway.