Delusional parasitosis is a rare psychiatric disorder which often presents with dermatological problems. Delusional parasitosis, which involves urethral self-instrumentation and foreign body insertion, is exceptionally rare. This is the first case report to date that provides a detailed presentation of the urological manifestation of delusional parasitosis with complications associated with repeated self-instrumentation and foreign body insertion, resulting in stricture formation and requiring perineal urethrostomy.
A 45-year-old Irish man was electively admitted for perineal urethrostomy with chronic symptoms of dysuria, haematuria, urethral discharge, and intermittent urinary retention. He reported a 4-year history of intermittent pain, pin-prick biting sensations, and burrowing sensations, and held the belief that his urethra was infested with ticks. He also reported a 2-year history of daily self-instrumentation, mainly injecting an antiseptic using a syringe in an attempt to eliminate the ticks. He was found to have urethral strictures secondary to repeated self-instrumentation. A foreign body was found in his urethra and was removed via cystoscopy. On psychiatric assessment, he displayed a fixed delusion of tick infestation and threatened to surgically remove the tick himself if no intervention was performed. The surgery was postponed due his mental state and he was started on risperidone; he was later transferred to an acute in-patient psychiatric unit. Following a 3-week admission, he reported improvement in his thoughts and distress.
Delusional parasitosis is a rare psychiatric disorder. Self-inflicted urethral foreign bodies in males are rare and have high comorbidity with psychiatric disorders; hence, these patients have a low threshold for referral for psychiatric assessment. The mainstay treatment for delusional parasitosis is second-generation antipsychotic drugs.
Delusion of parasitosis; psychosis; delusional parasitosis
Malignant schwannomas or neurofibrosarcomas are rare nerve tumors of unknown etiology. These neoplasms are highly aggressive with a marked propensity for local recurrence and metastatic spread. Their management continues to be a challenge for pathologists and surgeons. Maxillofacial locations are very exceptional. We report the case of a patient with unusual malignant schwannoma of the infratemporal fossa discovered at a late evolving stage.
A 56-year-old woman, of Moroccan nationality, presented to our hospital in 2013 with a large right-sided hemifacial swelling that had evolved over the previous 4 months, with a limitation of mouth opening, nasal obstruction and episodes of epistaxis. A CT scan and MRI showed a large and invasive tumor occupying her right infratemporal fossa and maxillary sinus, with sphenoidal, ethmoidonasal, nasopharyngeal and intraorbital extension. A nasal endoscopic biopsy was performed. Immunohistochemical examination concluded a diagnosis of malignant schwannoma, and a palliative radiotherapy was decided; however, our patient died 10 days later.
Malignant schwannoma of paranasal sinuses and the anterior skull base is a rare tumor that involves a high rate of local invasion. The prognosis is poorer compared to that occurring in the trunk and extremities.
Malignant schwannoma; Infratemporal fossa; Paranasal sinuses; Immunohistochemistry
Congenital duodenal diverticula are a rare anomaly. The discovery of one in association with an ingested foreign body has only been reported on one previous occasion. In this challenging presentation, the presence of the coin led to the correct diagnosis. Patients with congenital duodenal anomalies may present a number of associated abnormalities. Interestingly, after the discovery of his intraluminal duodenal diverticulum, we searched and found that our patient presented a number of associated pathologies, as described in the literature.
Our patient was a 36-year-old man, Caucasian, a kidney transplant recipient who presented with abdominal pain, vomiting and fever after an episode of pancreatitis. Because of a history of behavioral problems associated with intellectual impairment, including a compulsion to swallow coins during childhood, an abdominal radiograph was performed. Surprisingly, the radiograph revealed a radiopaque shadow in the central abdominal area. The findings of the ultrasound examination and computed tomography scan were suggestive of dilated biliary and pancreatic ducts. We performed an endoscopic retrograde cholangiopancreatography, which led to confirmation of the suspected coin above an obstructing intraluminal duodenal diverticulum with associated biliary ductal dilation. Upon retrieval of the coin, it was found to be a 1975 copper two-cent piece out of circulation in Australia for a large number of years.
Foreign body retention in the gastrointestinal tract in an adult could be a sign of underlying mechanical pathology. Intraluminal duodenal diverticulitis can have a varied presentation, including life-threatening complications. Awareness should be raised of the conditions associated with congenital duodenal anomalies in adults, including renal, hepatobiliary and cardiac defects, many of which were present in our case.
Duodenal diverticulum; Pancreatitis; Foreign body; Endoscopic retrograde; Cholangiopancreatography; Coin
Genital self-mutilation is listed as a symptom of borderline personality disorder. The type of injury varies from simple skin laceration to total amputation of the penis and testicles. These injuries are urological and surgical emergencies.
We report two cases of penile self-mutilation precipitated by erotic and religious bizarre delusions.
Our first patient is a 24-year-old Moroccan man who visited our emergency room with a metallic ring at the root of his penis which had caused marked edema of his entire penis.
Our second patient is a 26-year-old Moroccan man evaluated in our emergency unit. A clinical examination revealed a wound at the dorsal side of his penis with complete transection of the dorsal vein and imperfect hemostasis.
The two patients were treated in our emergency unit after which a favorable clinical course was observed.
Cases of genital self-mutilation are urological and psychiatric emergencies, therefore it is important that surgical and psychiatric teams collaborate closely while managing cases of genital self-mutilation.
Behavior disorder; Genital self-mutilation; Schizophrenia
The pituitary gland is an uncommon site for metastases, in particular from rhabdomyosarcoma. Some authors have reported a recent increase in the incidence of metastases at infrequent sites, such as brain or bone, probably due to the expanded treatment options and the resulting improved survival. Treatment options are limited, but must be discussed and adapted to the patient profile.
We report the case of a 17-year-old Arabic man, diagnosed with alveolar rhabdomyosarcoma of the left shoulder, who, after several cycles of chemotherapy, presented symptoms and signs of pituitary dysfunction. To the best of our knowledge, it is the first case described.
Pituitary metastasis of rhabdomyosarcoma is a rare situation, which must be actively researched to have access to an optimal therapeutic approach.
Pituitary gland; Metastasis; Rhabdomyosarcoma
Mucinous cystadenoma of the liver is a rare (less than 5%) neoplasm. This condition is more common in young women and accounts for non-specific symptoms. Cyst adenomas commonly affect the intrahepatic system (90%) and are rarely found in the extrahepatic biliary system or affecting both the systems.
A 39-year-old Sinhalese woman presented with features of obstructive jaundice and was found to have a biliary neoplasm on imaging. In the absence of a definitive diagnosis despite extensive imaging she underwent preoperative endoscopic biliary drainage followed by a left hemihepatectomy with Roux-en-Y hepaticojejunostomy. A pathological examination of the specimen revealed an obstruction of the bile duct caused by a biliary mucinous cystadenoma affecting both the intrahepatic and extrahepatic systems.
Biliary mucinous cystadenoma rarely present with obstructive jaundice affecting both intrahepatic and extrahepatic ducts. Exhaustive investigation might not help in the diagnosis and may need to be treated based on clinical judgment. The definitive treatment modality is surgery due to its malignant potential. The operative procedure is technically demanding and is best performed at specialist centers to minimize complications.
Benign liver cyst; Liver cyst; Mucinous cystadenoma; Obstructive jaundice
Chromosomal aberrations of chromosome 16 are uncommon and submicroscopic deletions have rarely been reported. At present, a cytogenetic or molecular abnormality can only be detected in 55% of Rubinstein-Taybi syndrome patients, leaving the diagnosis in 45% of patients to rest on clinical features only. Interestingly, this microdeletion of 16 p13.3 was found in a young child with an unexplained syndromic condition due to an indistinct etiological diagnosis. To the best of our knowledge, no evidence of a microdeletion of 16 p13.3 with contiguous gene deletion, comprising cyclic adenosine monophosphate-response element-binding protein and tumor necrosis factor receptor-associated protein 1 genes, has been described in typical Rubinstein-Taybi syndrome.
We present the case of a three-year-old Malaysian Chinese girl with a de novo microdeletion on the short arm of chromosome 16, identified by oligonucleotide array-based comparative genomic hybridization. Our patient showed mild to moderate global developmental delay, facial dysmorphism, bilateral broad thumbs and great toes, a moderate size atrial septal defect, hypotonia and feeding difficulties. A routine chromosome analysis on 20 metaphase cells showed a normal 46, XX karyotype. Further investigation by high resolution array-based comparative genomic hybridization revealed a 120 kb microdeletion on chromosomal band 16 p13.3.
A mutation or abnormality in the cyclic adenosine monophosphate-response element-binding protein has previously been determined as a cause of Rubinstein-Taybi syndrome. However, microdeletion of 16 p13.3 comprising cyclic adenosine monophosphate-response element-binding protein and tumor necrosis factor receptor-associated protein 1 genes is a rare scenario in the pathogenesis of Rubinstein-Taybi syndrome. Additionally, due to insufficient coverage of the human genome by conventional techniques, clinically significant genomic imbalances may be undetected in unexplained syndromic conditions of young children. This case report demonstrates the ability of array-based comparative genomic hybridization to offer a genome-wide analysis at high resolution and provide information directly linked to the physical and genetic maps of the human genome. This will contribute to more accurate genetic counseling and provide further insight into the syndrome.
Sex cord-stromal tumors of the ovary are uncommon. They behave unpredictably and often have a late recurrence, making counseling, management, and prediction of prognosis challenging.
A 52-year-old Moroccan woman with an sex cord-stromal tumors underwent a bilateral oophorectomy. The histology was unusual but was likely to be a luteinized thecoma with suspicious features for invasion. Seven years later, after a gastrointestinal bleed, a metastasis within the small bowel mucosa was detected. This represents probable isolated hematogenous or lymphatic spread, which is highly unusual, especially in the absence of concurrent peritoneal disease.
To the best of our knowledge, this is the second reported case of an sex cord-stromal tumors recurring in small bowel mucosa and mimicking a primary colorectal tumor. This highlights the diverse nature and behavior of these tumors.
Epithelioid hemangioma is a rare vascular tumor found in the penis. It is essential to avoid misdiagnosis with Peyronie's disease and penile cancer, as management differs significantly.
We present a case of epithelioid hemangioma of the penis in a 50-year-old Caucasian man. We also review the literature to evaluate the incidence of benign vascular anomalies of the penis and their management.
Epithelioid hemangioma of the penis should be considered in the differential diagnosis of patients presenting with painful penile lumps. A thorough histological and immunohistochemical examination is required to make the diagnosis. Optimal management is complete local excision and periodic physical examination for local recurrence.
Spontaneous biloma formation is a very rare condition, which mandates immediate treatment.
An 80-year-old Caucasian man was referred to our department with a diagnosis of intra-abdominal collection located in his right upper quadrant. Further radiological examination demonstrated multiple calculi in his gallbladder and common bile duct. Our patient underwent endoscopic retrograde cholangiopancreatography and the stones in the common bile duct were extracted. Percutaneous drainage of the abdominal collection revealed a spontaneous biloma formation. Continuous drainage of bile persisted for one week, so endoscopic retrograde cholangiopancreatography was repeated and a 10Fr stent was placed; subsequently the biliary leak ceased and our patient was discharged. A control abdominal computed tomography did not show any residual fluid collection.
Spontaneous biloma formation is a very rare incidence; awareness is necessary for prompt recognition and treatment.
We report the case of a true hermaphrodite with testicular seminoma with resulting metastases to the inguinal lymph nodes eight months after radical orchidectomy. This is an unusual presentation of testicular cancer and, to the best of our knowledge, the first report of this kind in the literature.
A 45-year-old Caucasian true hermaphrodite, raised as a male, developed a testicular seminoma. He had undergone a left orchidopexy at the age of 10 for undescended testes. Metastases from testicular tumors to inguinal lymph nodes are a rare occurrence. It has been suggested that previous inguinal or scrotal surgery may alter the pattern of nodal metastasis of testicular cancer. We review the literature to evaluate the incidence of inguinal lymph node involvement in early stage testicular cancer and discuss possible routes of metastases to this unusual site. We also discuss the management of the inguinal lymph nodes in patients with testicular tumors and a previous history of inguinal or scrotal surgery, as this remains controversial.
Inguinal lymph node metastases from testicular cancer are rare. A history of inguinal or scrotal surgery may predispose involvement of the inguinal nodes. During radical inguinal orchidectomy, the surgeon should be careful to minimize the handling of the testis and ensure high ligation of the spermatic cord up to the internal inguinal ring to reduce the risk of inguinal lymph node metastasis.
Echinococcosis, or hydatid disease, is endemic in some regions of the world, and has been a common pathology of surgical wards in Kosovo. Primary hydatid cyst of the gallbladder is an unusual and very rare localization of hydatid disease. So far, only five cases that fulfill the criteria of primary gallbladder hydatidosis have been published in the English medical literature.
We report a case of a 39-year-old Kosovan Albanian woman referred to the Abdominal Surgery Division of the University Clinical Center of Kosovo for "a calcified hydatid cyst of the liver with gallbladder involvement". Her history was significant for chronic right upper quadrant pain, characterized as intermittently colicky pain, accompanied by nausea. The patient underwent right subcostal laparotomy. Intra-operatively, a calcified primary hydatid cyst of the gallbladder was found. Its pericyst was tightly attached to the liver. Complete pericystectomy with cholecystectomy followed. The histopathology confirmed the presence of calcified hydatid cyst of the gallbladder, and that the cyst had developed entirely extra-mucosally. Five year follow-up showed no recurrence of disease.
Primary hydatid cyst of the gallbladder is a very rare clinical entity. Accurate preoperative diagnostic localization is not always easy, particularly in centers with limited diagnostic tools.
Multiple myeloma can occasionally manifest with joint disease. We report the case of an individual with a progressive bilateral carpal syndrome and a symmetrical severe seronegative polyarthritis and joint swelling. Investigations revealed an erosive seronegative inflammatory arthritis in association with bilateral carpal tunnel syndrome, anaemia, hepatic impairment and nephrotic-range proteinuria. Synovial fluid cytology demonstrated plasmablasts and multinucleated cells with products of chondrolysis. The diagnosis of multiple myeloma (with secondary amyloidosis) was made on serum protein electrophoresis and bone marrow biopsy.
The relationship between myeloma and joint disease is discussed, highlighted by the presence in this case of all three pathogenic features associated with arthritis in myeloma patients- an erosive arthritis, carpal tunnel syndrome and an invasive tumoural arthritis.