Liesegang rings in a dermoid cyst of the floor of the mouth. Report of a case with cytologic findings

BACKGROUND: Eosinophilic, ring-shaped structures, referred to as Liesegang rings (LRs), have been identified in aspirates from many organs, usually in relation to cystic or inflammatory lesions, but never before in relation to a dermoid cyst. CASE: LRs were intermingled with squamous cells in aspirates from a cyst of the floor of the mouth. The patient, a 23-year-old man, presented with a sublingual mass that had recently grown. Histopathologic study showed a ruptured dermoid cyst with mural foci of granulomatous foreign body reaction mixed with numerous ringlike structures, LRs. CONCLUSION: To the best of our knowledge, this is the first reported case of LRs in a dermoid cyst. Pathologists should be aware of this association when diagnosing cystic lesions.     

Angular dermoid cysts in the endoscopic era: retrospective analysis of aesthetic results using the direct, classic method

The concept of the endoscopic approach was recently introduced for the removal of angular dermoid cysts. Therefore, it was considered necessary to evaluate the conventional direct incisional approach for angular dermoid cyst excision, with respect to long-term aesthetic results and complications. The need to change from the direct excisional technique to the endoscopic approach in hair-bearing areas was investigated. During a 25-year period, 95 children were surgically treated by a single surgeon (H.G.T.) for removal of angular dermoid cysts. Only 22 patients (23 percent) were available for reassessment. The follow-up periods after the excisional procedure were 1 to 12 years. Assessments were performed by a 15-member team of assessors, who scored the aesthetic results of the scars with comparative slides. In addition, a questionnaire was mailed to each family, to document the family members' perceptions of the aesthetic appearance of the scar. Twenty-eight families (29 percent) responded. The complications were determined through the medical records department. For 19 of 22 patients (86 percent), the scar was scored by 85 percent of the assessors as excellent or good. No assessor stated that a scar was unacceptable. In the family questionnaires, 26 of 28 families (93 percent) reported an excellent scar and two (7 percent) reported a fair scar. No family stated that the scar was unacceptable. The operative notes for the 95 patients revealed that only two cysts had ruptured during the surgical procedure (2 percent) and only one infection had occurred (1 percent). No other major complications were reported. It is concluded that the direct method for dermoid cyst excision is an excellent approach, with a low complication rate and a very high aesthetic success rate, when performed through a supra-eyebrow or infra-eyebrow incision.     

Branchial cleft anomaly, congenital heart disease, and biliary atresia: Goldenhar complex or Lambert syndrome?

The features of Goldenhar complex have been well-described and classically include branchial arch abnormalities, epibulbar dermoid and vertebral abnormalities. We have identified an infant with these features in association with complex congenital heart disease and intrahepatic biliary atresia. Although Lambert described an autosomal recessive disorder with an association of biliary atresia and branchial arch abnormalities, none of those cases had epibulbar dermoid. Diagnostic considerations in this case include inclusion of biliary atresia as a new feature in the expanding spectrum of the Goldenhar complex, versus Lambert syndrome with epibulbar dermoid.     

Acro-cardio-facial syndrome associated with neuroepithelial cyst: a case report

Acro-cardio-facial syndrome (ACFS) is a very rare genetic syndrome. Only 5 patients have been reported in the literature so far. A female neonate presented with limb abnormalities, cleft palate and congenital heart disease was diagnosed as ACFS. Her cranial magnetic resonance imaging revealed a huge cerebral neuroepithelial cyst. To our knowledge, this is the first case of ACFS in the literature associated with a neuroepithelial cyst in the brain.     

A review of 35 cases of asymmetric crying facies

A review of 35 cases of asymmetric crying facies: Congenital asymmetric crying facies (ACF) is caused by congenital hypoplasia or agenesis of the depressor anguli oris muscle (DAOM) on one side of the mouth. It is well known that this anomaly is frequently associated with cardiovascular, head and neck, musculoskeletal, respiratory, gastrointestinal, central nervous system, and genitourinary anomalies. In this article we report 35 ACF patients (28 children and 7 adults) and found additional abnormalities in 16 of them (i.e. 45%). The abnormalities were cerebral and cerebellar atrophy, mega-cisterna magna, mental motor retardation, convulsions, corpus callosum dysgenesis, cranial bone defect, dermoid cyst, spina bifida occulta, hypertelorism, micrognatia, retrognatia, hemangioma on the lower lip, short frenulum, cleft palate, low-set ears, preauricular tag, mild facial hypoplasia, sternal cleft, congenital heart defect, renal hypoplasia, vesicoureteral reflux, hypertrophic osteoarthropathy, congenital joint contractures, congenital hip dislocation, polydactyly, and umbilical and inguinal hernia. Besides these, one infant was born to a diabetic mother, and had atrial septal defect and the four other children had 4p deletion, Klinefelter syndrome, isolated CD4 deficiency and Treacher-Collins like facial appearance, respectively Although many of these abnormalities were reported in association with ACF, cerebellar atrophy, sternal cleft, cranial bone defect, infant of diabetic mother, 4p deletion, Klinefelter syndrome, isolated CD4 deficiency and Treacher-Collins like facial appearance were not previously published.     

Subcutaneous cystic granuloma caused byPhialophora verrucosa

A case of subcutaneous cystic granuloma caused byPhialophora verrucosa was reported. The patient was 21-year-old Japanese female with systemic lupus erythematosus (SLE) who had received prolonged steroid therapy. P. verrucosa has occasionally been isolated from patients as one of etiological agents of chromomycosis. There have been, however, no paper published to report an isolation of this fungus from subcutaneous cyst or abscess of a patient. This case is the first report of subcutaneous cystic granuloma induced clinically byP. verrucosa.     

Early cancer in congenital choledochal cyst

We report a case of 35-yr-old woman with early cancer in congenital choledochal cyst. She had a five-year history of intermittent right upper abdominal pain and intermittent jaundice. In this period she had a few abdominal ultrasonographies, but the cholelithiasis had not been found. Now, she was admitted to our hospital because she felt right upper abdominal pain with slight jaundice and subfebrile temperature four weeks ago. Abdominal ultrasonography showed enormous dilatation of the common bile duct, which was suspected as choledochal cyst. Computed tomography and endoscopic retrograde cholangiopancreatography revealed cystic dilatation of extrahepatic bile duct. An anomalous pancreaticobiliary junction was not found. The patient with congenital choledochal cyst was operated on. The excision of choledochal cyst was done with hepaticojejunostomy Roux-en-Y. There were no lymph nodes metastases. On the central part of choledochal cysts mucosa, it was shown a white plain area of thickness 0.3 cm and 0.8 cm in diameter. Histologically it was well-differentiated tubular adenocarcinoma, which was limited to the mucosa and which did not penetrate to other parts of the bile duct wall. From our knowledge, only small number cases of early cholangiocarcinoma in choledochal cyst were until now reported. Nearly eight years after the operation the patient feels very well, and has optimal working ability (Karnofsky 100%).     

Mixed heterotopic gastrointestinal and respiratory cyst of the lip: case report and review of the literature.

A congenital heterotopic gastrointestinal and respiratory cyst arising from the upper lip is reported. The lesion was excised directly with a satisfactory aesthetic result. Long-term follow-up is recommended because of a report of recurrence of this type of lesion after 13 years.     

Tailgut cyst associated with a carcinoid tumor: case report and review of the literature

We report the case of a 49-year-old woman who presented a tailgut cyst lined by a variety of epithelium including squamous, columnar and transitional. Fortuitously a microscopic carcinoid tumor expressing immunohistochemically neuroendocrine markers was identified in the cystic wall. Tailgut cysts are congenital abnormalities located in the presacrococcygeal area occurring usually in adult patients. Clinical diagnosis is difficult because they are often asymptomatic. Patients may present symptoms resulting from local mass effects or complications. The differential diagnoses include rectal duplication cysts, cystic sacrococcygeal teratomas, epidermal cysts, epidermoid cysts, anal duct or gland cysts. Magnetic resonance imaging has recently become the modality of choice to image the cyst. Malignant transformation is rare; 23 cases including 10 carcinoid tumors have been reported in the literature. To our knowledge, this is the eleventh case of carcinoid tumor arising in a tailgut cyst.     

Subcutaneous Mycotic Cyst Caused by <Emphasis Type="Italic">Roussoella percutanea</Emphasis> in a UK Renal Transplant Patient

Fungi from more than 100 genera have been implicated in subcutaneous fungal infections, usually following traumatic inoculation of the etiologic agent. With the advent of molecular approaches to fungal identification and taxonomy, novel agents of subcutaneous mycoses are increasingly reported. In this manner, Roussoella percutanea, a novel species in Pleosporales, was described in 2014 from a subcutaneous mass in an immunocompetent male adult. Two additional cases involving renal transplant recipients were recently reported from patients resident in France and Germany, with several further cases discovered after analyses of historical culture collection isolates. Here, we describe a new case of subcutaneous R. percutanea infection, causing a mycotic cyst in a renal transplant patient resident in the UK. Although fungal infection was confirmed histologically, viable fungal isolates could not be recovered in culture from biopsy material and identification of the causative agent relied upon PCR amplification and sequencing of fungal rDNA genes. This is the fourth well-documented case of infection with R. percutanea in renal transplant patients, and the first reported from a patient resident in the UK. The current case illustrates the importance of molecular approaches for the identification of emerging fungal pathogens in culture-negative subcutaneous fungal infections.     

Congenital Cystic Malformation of the Bile Ducts: Report of a Case and Review of Related Literature

A 20-year-old woman had a cyst of the proximal part of the common bile duct and a cyst of the left hepatic duct; these lesions were diagnosed preoperatively by intravenous cholangiography and successfully operated upon. At the time of writing, she has been followed up for one year.Congenital defects in the biliary system are rare and, in a review of the literature, only two cases were found similar to this one. It is generally accepted that these lesions are congenital, but the exact pathogenesis is unknown.Alonso-Lej, Rever and Pessagno² reviewed the literature in 1959 and found 403 authentic congenital cysts of the hepatic ducts. The most common congenital defect is a single choledochal cyst of the lower end of the common bile duct. Pain, jaundice and tumour are the main symptoms.Until the advent of intravenous cholangiography, these lesions were seldom recognized preoperatively. Means of operative repair as well as complications and prognosis are reviewed.     

Phialophora verrucosa-induced subcutaneous phaeohyphomycosis. Fine needle aspiration findings

A 34-year-old woman on immunosuppressive therapy presented with a subcutaneous, cystic lesion on the dorsum of the right foot. Cytologic examination of material obtained by fine needle aspiration (FNA) revealed a mixture of acute and granulomatous inflammation as well as brown-pigmented fungi in the form of budding yeast, pseudohyphae and septate hyphae. The findings suggested subcutaneous phaeohyphomycosis (phaeomycotic cyst). Culture grew Phialophora verrucosa. The cytologic, histologic and cultural findings are given. This case demonstrates that phaeohyphomycosis can be diagnosed by FNA but that fungal culture is necessary to establish the identity of the etiologic agent. This appears to be the first case of P. verrucosa-induced subcutaneous phaeohyphomycosis reported in the Western Hemisphere.     

Echinococcal cyst of the subcutaneous tissue: a rare case report

Hydatid disease is an endemic zoonosis caused by the cestode Echinococcus granulosus. The most common affected organs are the liver and the lungs. Echinococcal cyst located in the subcutaneous tissue constitutes an extremely rare manifestation of the hydatid disease. Here we present a case report with a cystic mass in the subcutaneous tissue of the left gluteal area, diagnosed as being a hydatid cyst.     

A 10-year experience in the treatment of intraabdominal cerebrospinal fluid pseudocysts

The aim of this retrospective study was to assess a ten-year experience in the treatment of rare complications of ventriculoperitoneal shunting--intraabdominal cerebrospinal fluid pseudocysts. At this time there are no data about incidence, clinical course and treatment of these complications in Croatia. Cerebrospinal fluid (CSF) abdominal pseudocyst is an uncommon but important complication of ventriculoperitoneal shunts. Retrospective data were obtained from 5 children with abdominal CSF pseudocysts, treated between 1996 and 2007. The incidence of intraabdominal CSF pseudocysts in our study is 2.9%. All patients were girls ranged in age from 4 to 12 years old (mean 8.8 years). In most cases etiology of hydrocephalus was congenital, idiopathic. Abdominal pain and distension were the most frequent clinical finding (4/5). Although infection has been reported as responsible for pseudocyst formation, we did not found it in our series. Laparotomy with cyst wall excision and catheter replacement was performed in 2/5 cases, and only cyst fluid aspiration with catheter replacement in 3/5 cases. Recurrence of the abdominal cyst was observed in one girl who was in terminal stadium of anaplastic ependymoma. It is our opinion that only catheter replacement and cyst fluid evacuation, as one of the treatment modalities, may be successful, even in large CSF intraperitoneal pseudocysts.     

Proteus syndrome in 7 patients: clinical and genetic considerations.

The Proteus syndrome is a congenital hamartomatous disorder delineated in 1983. Because of its polymorphic appearance, the syndrome was named after the greek god Proteus whose name means much less than the polymorphous much greater than. Major clinical findings include hemi hypertrophy, macrodactyly, exostoses, scoliosis, epidermal nevi, haemangiomas, deeply rugated soles of the feet and a variety of deep and subcutaneous masses. We report on 7 new cases of Proteus syndrome. All reported cases have been sporadic. Therefore this syndrome could be due to the action of a dominant lethal gene surviving by mosaicism.     

Why are we shaped differently, and why does it matter?

Body fat distribution is an important predictor of metabolic abnormalities in obese humans. Dysregulation of free fatty acid (FFA) release, especially from upper body subcutaneous adipose tissue, appears to contribute substantially to these metabolic disturbances. Why different individuals preferentially store fat in upper vs. lower body subcutaneous fat or subcutaneous vs. visceral fat is not completely understood. Current evidence suggests that defects in regional lipolysis are not the cause of net fat retention in larger fat depots. Regional variations in the storage of fatty acids, both meal derived and direct reuptake, and storage of circulating FFAs that may help to explain why some depots expand at the expense of others have been reported. We review the quantitative data on regional lipolysis, meal, and FFA storage in adults to provide an overview of fat balance differences in adults with different fat distribution patterns.     

Intravenous pyogenic granuloma mimicking pleomorphic adenoma in a fine needle aspirate. A case report.

BACKGROUND: Intravenous pyogenic granuloma (IvPG) is a rare, benign lesion occurring usually as a subcutaneous mass in the neck or upper extremity. The cytologic features of IvPG have not been described before. CASE: A patient presented with a subcutaneous nodule on the lower border of the left parotid area. The clinical diagnosis was bronchial cleft cyst or lymphadenitis, and the fine needle aspiration diagnosis was pleomorphic adenoma. The tissue section, however, disclosed IvPG. CONCLUSION: Evaluation of subcutaneous nodules presenting cytologically as spindle cell lesions may be problematic, particularly in the neck and head region. Such lesions occurring in the parotid area may be interpreted as pleomorphic adenoma of the salivary gland.     

Fine needle aspiration cytology of subcutaneous cysticercosis of the breast. Case report and pathogenic discussion

A case of subcutaneous cysticercosis of the breast diagnosed by fine needle aspiration cytology is reported. The cytologic preparations showed the presence of many inflammatory cells mixed with spiked spherules resembling the rambutan tropical fruit. These spherules arose from the degenerated integument of the parasite found in the histologically excised cyst. They are suggestive of the presence of an encysted flat worm, but are not specific for cysticercosis, whose identification is based on the study of the cephalic extremity of the parasite.     

A composite forearm free flap for the secondary repair of the ruptured Achilles tendon.

The nasal dermal sinus cyst is one of many midline nasal masses that often pose diagnostic and treatment dilemmas for the plastic and reconstructive surgeon. The differential diagnosis of the midline nasal mass includes both congenital and acquired processes. A thorough understanding of its cause is crucial to treatment. A comprehensive discussion of the pathogenesis, diagnosis, sequelae, and surgical management, and a representative case analysis, of the nasal dermal sinus cyst is presented to delineate the role of open rhinoplasty in optimizing the care of this congenital nasal deformity.