Adult localized Langerhans cell histiocytosis: A case report.

BACKGROUND: Langerhans cell histiocytosis (LCH) is a rare clonal proliferative disease of Langerhans cells with unknown pathogenesis. An increasing number of clinicians recognize that LCH has a wide clinical spectrum and a highly varied course. Adults rarely develop LCH. Here, we report a case of adult localized LCH. CASE SUMMARY: A 32-year-old woman presented with plaques and ulcers on the vulva and crissum, accompanied by pain that persisted for more than one year. Physical examination revealed a red-infiltrating plaque with ulcerations and exudates in the vulva and crissum. Pathological examination revealed a diffuse infiltration of lymphocytes, eosinophilic granulocytes, and histiocytoid cells in the superficial dermis. Proliferative histiocytoid cells showed mild atypia, partly with kidney-shaped nuclei. Immunohistochemical examination showed that the histiocytoid cells were positive for S100 protein and CD1 and weakly positive for CD68 (20% +), with a Ki-67 index of 30%. Laboratory tests did not reveal any other systemic damage. The patient was diagnosed with adult localized LCH and was prescribed oral prednisone (20 mg) once daily. The skin lesions gradually improved and are still being followed-up. CONCLUSION: Adult localized LCH is rare and must be differentiated from other common conditions.

Glomus tumor of the crissum: A case report and review of literature.

Glomus tumor is a rare mesenchymal neoplasm arising from the modified smooth muscle cells of the glomus body. Primary crissum glomus tumor is extremely rare without any published in the literature. In this article, we report the first case of primary crissum glomus tumor in an 80-year-old man with recurrent anal pain for 8 years, increased pain for 1 year. Rectal MRI for inflammatory lesions (sinus tract). Microscopic examination showed the tumor cells were arranged in sheets and nests, surrounding blood vessels and nerve bundles. At high magnification, the neoplastic cells show regular round shape with light eosinophilic and translucent cytoplasm. The cell boundary is clear, the nucleus is round and located in the center. The stroma of the tumor shows hyaline degeneration. Immunohistochemically, the tumor cells were positive for smooth muscle actin, h-caldesmon, Calponin, synaptophysin, Collagen IV and CD34, but completely negative for HMB45, S100, EMA, desmin, CgA and CD56. The histologic features and immunohistochemical profile supported a diagnosis of primary crissum glomus tumor. The patient was asymptomatic and disease free after the procedure.

Anal Canal Duplication Mimicking Recurrent Abscess: A Case Report and Review of the Literature.

Background: Anal canal duplication (ACD) is a very rare duplication of the gastrointestinal tract and is described as a secondary anal orifice along the posterior side of the normal anal canal. Early surgical removal is advisable, also in asymptomatic patients, because of the risk of inflammatory complications, such as recurrent crissum abscess, and malignant changes. Case presentation: A previously healthy 2-year-old boy was evaluated in the emergency department with fever. He complained of anal pain in the absence of incentive. Physical examination and ultrasound confirmed a diagnosis of perianal abscess. He was treated with incision and drainage of the abscess and intravenous antibiotics. Two months after his discharge from the hospital, he developed fever and had intervals discharge pus and pain in the same locations. Colorectal endoscopy revealed that there was no fistula opening at the rectal wall. Intraoperative fistulography showed a fistulous tract that was connected to a subcutaneous cavity. Excision of the fistulous tract and wide drainage of the deep postanal space were performed. The patient was referred to our hospital for further evaluation 6 months later. Physical examination showed a secondary anus that had not been noticed before. MRI showed an anal fistula between 1 and 3 o'clock, and preoperative fistulography revealed a 3-cm-long tubular structure without any connection with the rectum. The diagnosis of ACD was made by intraoperative examination with a metal catheter and the postoperative pathological analysis. The duplicated anal canal was resected completely via a perianal approach without any rectal injury. Histology showed a squamous epithelium in the distal end with some smooth-muscle fibers. After a follow-up of 8 months, the patient has been doing well. Conclusion: Recurrent crissum abscess should raise clinical attention to alimentary tract congenital malformations such as ACD. Prompt recognition of these unique presentations of ACD is needed, and complete excision through a perineal approach or posterior sagittal approach is recommended.

Isolation and characterisation of Chrysanthemum crassum SOS1, encoding a putative plasma membrane Na(+) /H(+) antiporter.

A full-length cDNA homologue of SOS1 (salt overly sensitive 1) was isolated from the salinity-tolerant species Chrysanthemum crassum and found to encode a Na(+) /H(+) antiporter, using degenerate PCR and RACE-PCR. The 3752-bp sequence comprised a 3438 bp open reading frame, encoding a 127-kDa protein with 12 transmembrane domains within its N terminal portion, and a hydrophilic cytoplasmic tail in its C-terminal portion. CcSOS1 appears to be a plasma membrane protein, and shares ∼62% identity at the peptide level with its Arabidopsis thaliana homologue. Expression of CcSOS1 in the roots of C. crassum was sensitive to salinity stress, while in the leaves CcSOS1 was down-regulated in the presence of abscisic acid. CcSOS1 transcript abundance was reduced in both roots and leaves of plants exposed to low temperature, while it was increased in leaves (but not in roots) after drought stress. CcSOS1 expression was not regulated in the presence of CaCl2 . A heterologous complementation assay in yeast suggested that CcSOS1 directs Na(+) efflux, mimicking the function of the endogenous NHA1 protein. Thus CcSOS1 appears to encode a salinity-inducible plasma membrane Na(+) /H(+) antiporter. This gene may be useful in transgenic approaches to improving the salinity tolerance of related ornamental species.

Clinic applicative value of MRI in the diagnosis of complex anal fistula / 重庆医学

Objective To evaluate the clinical value of MRI examination in the diagnosis and guidance of operation complexity a-nal fistula.Methods Preoperative MRI findings of 32 patients with complex anal fistula confirmed by operation were analyzed ret-rospectively,and compared with the operation results.Results The pre-operation MRI results of 32 patients with complex anal fis-tula revealed that there were 43 fistulas,10 anus week abscesses,58 orificium fistulas,comparing the MRI diagnosis with operation results,coincidence rate of fistula,anus week abscesses,orificium fistula was 100.0%,100.0%,89.3%,respectively.MRI manifes-tations of fistula were that T1 WI manifestations of fistula was or low signal,T2 WI and T2 WI fat suppression sequence manifesta-tions of fistula was high signal,fistula presented a tubular shape obviously as T1 WI enhancement scanning,internal opening repre-sented as intensive dot,complicated embranchment,two type signal of fistula appeared at the same time.MRI manifestations of ab-scesses were that circular,ellipse,irregular form and horseshoe-shape,T1 WI manifestations of abscesses was low signal,there was gas in abscess cavity of some patients;intestinal tube surrounding of some patients were involved by abscess cavity,and up through the edge of bladder to the perineum.Conclusion MRI examination could accurately display the number of complex anal fistula,fis-tula walking and branch,the position of internal opening,correlativity between fistula and surrounding muscles,status of abscess, which could provide guidance for anorectal surgery.