Disseminated histoplasmosis in primary Sjogren syndrome:A case report

BACKGROUND Sj?gren syndrome(SS)is a chronic and systemic autoimmune disease characterized by lymphocytic infiltration of the exocrine glands.And histoplasmosis is an invasive mycosis caused by the saprophytic dimorphic fungus H.capsulatum.In patients with primary SS(PSS),disseminated histoplasmosis(DH)is extremely rare.CASE SUMMARY We report a 37-year-old female patient admitted to our hospital with exacerbating fatigue,somnolence,and pancytopenia as the main symptoms.She was eventually diagnosed with DH based on pancytopenia,splenomegaly,and findings of bone marrow smears.The atypical clinical symptoms made the diagnosis process more tortuous.Unfortunately,she died of respiratory failure on the day the diagnosis was confirmed.CONCLUSION We present a rare and interesting case of DH in a PSS patient.This case updates the geographic distribution of histoplasmosis in China,and expands the clinical manifestations of DH in PSS,highlighting the significance of constantly improving the understanding of PSS with DH.

Intestinal histoplasmosis in immunocompetent adults

AIM: To present a retrospective analysis of clinical and endoscopic features of 4 cases of immunocompetent hosts with intestinal histoplasmosis(IH).METHODS: Four immunocompetent adults were diagnosed with IH between October 2005 and March 2015 at West China Hospital of Sichuan University. Clinical and endoscopic characteristics were summarized and analyzed retrospectively. GMS(Gomori methenamine silver), PAS(periodic acid-Schiff) and Giemsa staining technique were used to confirm Histoplasma capsulatum(H. capsulatum). The symptoms, signs, endoscopic presentations, radiographic imaging, pathological stain results and follow-up are presented as tables and illustrations.RESULTS: The cases were male patients, ranging from 33 to 61 years old, and primarily presented with nonspecific symptoms such as irregular fever, weight loss, abdominal pain and distention. Hepatosplenomegaly and lymphadenopathy were the most common signs. Endoscopic manifestations were localized or diffuse congestion, edema, ulcers, and polypoid nodules with central erosion involving the terminal ileum, ascendingcolon, transverse colon, descending colon, sigmoid colon and rectum, similar to intestinal tuberculosis, tumor, and inflammatory bowel disease. Numerous yeast-like pathogens testing positive for PAS and GMS stains but negative for Giemsa were detected in the cytoplasm of the histiocytes, which were highly suggestive of H. capsulatum.CONCLUSION: Immunocompetent individuals suffering from histoplasmosis are rarely reported. It is necessary that gastroenterologists and endoscopists consider histoplasmosis as a differential diagnosis, even in immunocompetent patients.

Gastrointestinal histoplasmosis complicating pediatric Crohn disease:A case report and review of literature

BACKGROUND Infection with Histoplasma capsulatum(H.capsulatum)can lead to disseminated disease involving the gastrointestinal tract presenting as diffuse abdominal pain and diarrhea which may mimic inflammatory bowel disease(IBD).CASE SUMMARY We report a case of 12-year-old boy with presumptive diagnosis of Crohn disease(CD)that presented with several months of abdominal pain,weight loss and bloody diarrhea.Colonoscopy showed patchy moderate inflammation characterized by erythema and numerous pseudopolyps involving the terminal ileum,cecum,and ascending colon.Histologic sections from the colon biopsy revealed diffuse cellular infiltrate within the lamina propria with scattered histiocytic aggregates,and occasional non-necrotizing granulomas.Grocott-Gomori’s Methenamine Silver staining confirmed the presence of numerous yeast forms suggestive of Histoplasma spp.,further confirmed with positive urine Histoplasma antigen(6.58 ng/mL,range 0.2-20 ng/mL)and serum immunoglobulin G antibodies to Histoplasma(35.9 EU,range 10.0-80.0 EU).Intravenous amphotericin was administered then transitioned to oral itraconazole.Follow-up computed tomography imaging showed a left lower lung nodule and mesenteric lymphadenopathy consistent with disseminated histoplasmosis infection.CONCLUSION Gastrointestinal involvement with H.capsulatum with no accompanying respiratory symptoms is exceedingly rare and recognition is often delayed due to the overlapping clinical manifestations of IBD.This case illustrates the importance of excluding infectious etiologies in patients with“biopsy-proven”CD prior to initiating immunosuppressive therapies.Communication between clinicians and pathologists is crucial as blood cultures and antigen testing are key studies that should be performed in all suspected cases of histoplasmosis to avoid misdiagnosis and inappropriate treatment.

Colonic Bleeding Due to Histoplasma and Mycobacterium Coinfection in Renal Transplant Patient

Introduction： Histoplasmosis is a rare infectious condition caused by the fungus Histoplasma capsulatum that can be presented from asymptomatic to severe forms. Tuberculosis, still an endemic infection in some developing countries, can also have variable clinical presentations. Both diseases involve the lungs mostly, but in immunocompromised patients, especially those with advanced HIV infection and transplant patients, disseminated forms are more frequently found. Gastrointestinal involvement is unusual, and digestive bleeding is an even rarer complication. Case presentation： We report the case of a 39-year-old female who was diagnosed with a Mycobacterium tuberculosis and Histoplasma capsulatum coinfection occurring 11 years after a living-donor-related renal transplant. The patient presented a severe gastrointestinal bleeding caused by an ulcer in the ascending colon. She improved after a combined treatment with tuberculostatic and fungicidal drugs. Conclusions： Simultaneous gastrointestinal involvement by histoplasmosis and tuberculosis, presenting as severe digestive bleeding, with minimal respiratory symptoms associated, make this an extremely rare case and a diagnostic challenge. Therefore, it is important to keep a high clinical suspicion of opportunistic infection, especially in immunocompromised patient who presents with LGB.