Eosinophilic esophagitis: a case report with a review of the literature

Eosinophilic esophagitis (EE) is an allergic inflammatory condition of the esophagus and is characterized by dense eosinophilic infiltration of the esophagus. There has been a dramatic increase in the diagnosis of EE in Western countries in recent years; however, in Japan, there are very few reports of EE. We present a rare case of EE in a 70-year-old Japanese woman, who had dysphasia for 2 years, but which worsened over a 6-month period. Laboratory examinations showed peripheral eosinophilia (1279/μl). Significant thickening of the esophageal wall was observed on computed tomography scan and many circular rings appeared when an esophagogastroduodenoscopy was carried out. From these circular rings, EE was suspected and a biopsy was then taken from the esophagus. As the histologic findings from the esophageal biopsy showed that >25 eosinophils existed per high-power field, the patient was diagnosed with EE. Oral corticosteroid (prednisolone 30 mg/day) therapy was administered and after 3 days of treatment her symptoms almost disappeared. EE needs to be considered as a differential diagnosis if patients with non-erosive reflux disease have dysphagia but do not respond to high-dose proton pump inhibitor therapy.     

Eosinophilic granulomatosis with polyangiitis complicated by cholecystitis: a case report and review of the literature

The objectives are to report an atypical case of eosinophilic granulomatosis with polyangiitis (EGPA) associated with cholecystitis and to review the main clinical features, therapy, and prognosis of it. We present a 49-year-old male with non-classic clinical manifestations of EGPA and EGPA-related cholecystitis. EGPA was diagnosed by histology of the gallbladder after cholecystectomy. In addition, 11 cases of EGPA-associated cholecystitis have been reported and were described in details in this article. Gallbladder involvement is very uncommon in EGPA. All cases reviewed showed multiple organs involved as well as obviously elevated eosinophilic granulocyte proportion with inflammatory index, although antineutrophil cytoplasmic antibody may be negative. All patients in this cohort that showed gallbladder involvement were eventually confirmed with EGPA by histology examination after cholecystectomy. The pathological change could be infiltration of inflammatory mononuclear cells of small- and medium-sized vessels. Of the cases, 91.7 % responded well to steroid and immunosuppressant therapy. Gallbladder involvement is a very rare comorbid condition in EGPA. However, it is an important symptom or secondary condition to alert physicians the diagnosis of EGPA. Moreover, timely diagnosis and correct administration in the early stage of this disease could obviously improve the prognosis.     

Eosinophilic appendicitis caused by Schistosoma japonicum: a case report and review of the literature

Parasitic appendicitis is uncommon. The authors reviewed the pathology of 4,130 appendices resected over the past 10 years (2000 to 2009). Only one case of eosinophilic appendicitis caused by Schistosoma japonicum was identified. The overall prevalence of schistosomal appendicitis was 0.024%. The case was a 61-year-old woman who presented with right lower quadrant abdominal pain. She had been a farmer in Chumphon and Surat Thani Provinces, which are endemic for schistosomiasis in Thailand. Physical, laboratory and ultrasound examinations were suggestive of acute appendicitis. She underwent emergency appendectomy. Intraoperative findings revealed a ruptured appendix with a fecalith in the appendiceal lumen. The histopathologic diagnosis was suppurative eosinophilic appendicitis with schistosomal ova in the mucosa, submucosa, muscular layer and vascular lumens, identified as S. japonicum eggs. The patient was treated for the parasite with praziquantal. We briefly review the clinicopathologic features and pathogenesis of schistosomal appendicitis.     

Chronic eosinophilic leukemia: a case report and review of literature

Chronic Eosinophilic Leukemia (CEL) is a rare type of chronic myeloproliferative disorder of unknown etiology with no available true incidence. The vaguely overlapping clinico — pathological picture of CEL with idiopathic hypereosinophilic syndrome (IHES) often adds to the diagnostic confusion. An evidence of genetic clonality of eosinophils or an increase in blast cells in the blood or bone marrow is mandatory for diagnosis of CEL while no specific diagnostic tests exist for IHES; making it an entity of exclusion. Till date, CEL is a rarely reported entity in India. We add yet another case of eosinophilic leukemia along with review of available literature.     

Granular acute lymphoblastic leukemia: a case report and literature review

Authors report a rare case of granular acute lymphoblastic leukemia (ALL) in a 45-year-old woman with a history of multiple myeloma. The patient's lymphoblasts contained large numbers of distinctive cytoplasmic granules closely mimicking heavily granulated myeloblasts in acute myeloid leukemia. These blasts were completely negative for myeloperoxidase but positive for acid phosphatase and Periodic Acid-Schiff reaction by cytochemical staining. Immunophenotype analysis by immunohistochemistry clearly demonstrated precursor B-cell phenotype. Granular ALL occurs approximately 2 to 7% in childhood populations but is extremely rare in adults. Such cases may cause problematic distinction from myeloid differentiation and lead to misdiagnosis of acute myeloid leukemia. Ten cases of adult granular acute lymphoblastic leukemia described to date in the literature were also reviewed.     

Eosinophilic gastroenteritis presenting as small bowel obstruction： A case report and review of the literature

Eosinophilic gastroenteritis is a rare disease of unknown etiology. It is characterized by eosinophilic infiltration of the bowel wall to a variable depth and symptoms associated with gastrointestinal tract. Recently, the authors experienced a case of eosinophilic gastroenteritis presenting as small bowel obstruction. A 51-year old woman was admitted to our hospital complaining of abdominal pain and vomiting. Physical examination revealed a distended abdomen with diffuse tenderness. Complete blood count showed mild leukocytosis without eosinophilia. Computed tomography confirmed a dilatation of the small intestine with ascites. An emergency laparotomy was performed for a diagnosis of peritonitis due to intestinal obstruction. Segmental resection of the ileum and end to end anastomosis were performed. Histologically, there was a dense infiltration of eosinophils throughout the entire thickness of ileal wall and eosinophilic enteritis was diagnosed. The patient recovered well, and was free from gastrointestinal symptoms at the time when we reported her disease.     

Eosinophilic meningoencephalitis due to Toxocara canis: case report and review of the literature

Toxocariasis is usually manifested as visceral larva migrans. Nervous system involvement is a rare complication. In this report, we describe one case of meningoencephalitis due to Toxocara canis and review the literature. We report a previously healthy two-year-old boy who was admitted after 24 hours of severe neurologic symptoms with marked eosinophilic pleocytosis in the cerebrospinal fluid and a single subcortical lesion detected by cerebral magnetic resonance imaging. Both serum and spinal fluid tested positive for T. canis. Initial treatment with thiabendazole was ineffective. After marked improvement in clinical and laboratory results were achieved using albendazole and corticosteroids, the child was discharged.     

Burkitt's leukemia with an atypical immunophenotype: report of a case and review of literature

Burkitt's leukemia (BL) constitutes a small but important fraction of acute leukemias in children. It is an aggressive type of leukemia that is responsive to high-intensity, short-duration chemotherapy with complete remission possible in 75% to 90% of cases. The recognition and proper designation of BL is important because treatment differs from that of precursor B-cell acute lymphoblastic leukemia (pre-B ALL). Burkitt's leukemia is separated by its typical morphologic features (blasts with typical French-American-British [FAB] L-3 morphology compared to FAB L-1/L-2 morphology in pre-B ALL) and a classic immunophenotype (blast positivity for CD45 [bright], CD20 [bright], CD10, CD19, surface immunoglobulin [SIg], Ig light chain restriction, and negative terminal deoxynucleotidyl transferase [TdT]) compared to pre-B ALL blasts (which are positive for CD45 [dim], CD10, CD19, and TdT and negative for CD20 and SIg). The diagnosis of Burkitt's leukemia is confirmed by the characteristic cytogenetic findings.The combination of Burkitt's morphology with precursor B-cell immunophenotype may present a diagnostic pitfall, resulting in delay of proper management.We describe such an atypical case in a 12-year-old girl and emphasize that correct classification and treatment starts with proper morphologic/immunophenotypic correlation, and the awareness of the overlapping features in some cases.     

Acute promyelocytic leukemia with myelofibrosis: A case report and literature review

Rationale:Acute promyelocytic leukemia (APL) with myelofibrosis (MF) is rare, and only 14 cases have been reported in the literature to date.Patient concerns:A 42-year-old woman was admitted to the hospital with easy bruising and excessive bleeding. With the remission of the primary disease during treatment, the degree of fibrosis did not decrease, but worsened progressively.Diagnosis:The woman was diagnosed with acute promyelocytic leukemia with secondary myelofibrosis.Interventions:All-trans retinoic acid (ATRA) was discontinued after 6 months of complete remission of APL. Arsenic trioxide (ATO) was discontinued because of supraventricular tachycardia 9 months after complete remission of APL.Outcomes:After withdrawal of ATRA for 2 months, the degree of fibrosis was significantly alleviated, and after withdrawal of ATRA for 8 months and ATO for 5 months, bone marrow biopsy showed no reticular fiber deposition.Lessons:In this case report and review of an additional 14 cases of APL with MF, we highlighted the importance of the degree of MF to be evaluated by bone marrow biopsy at the time of bone marrow aspiration when APL is suspected. If MF is present, the type of MF should be determined in a timely manner, and appropriate intervention measures should be taken accordingly.