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Saudi Journal of Kidney Diseases and Transplantation
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CASE REPORT  
Year : 2013  |  Volume : 24  |  Issue : 4  |  Page : 764-767
Esophageal histoplasmosis in a renal allograft recipient


1 Department of Nephrology, SMS Hospital and Medical College, Jaipur, India
2 Department of Pathology, S. R. Kalla Memorial Gastro and General Hospital, Jaipur, India
3 Department of Gastroenterology, S. R. Kalla Memorial Gastro and General Hospital, Jaipur, India
4 Department of Nephrology, S. R. Kalla Memorial Gastro and General Hospital, Jaipur, India

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Date of Web Publication24-Jun-2013
 

   Abstract 

Histoplasmosis is a progressive granulomatous disease caused by the intracellular dimorphic fungus Histoplasma capsulatum. We report a rare case of esophageal histoplasmosis in a renal allograft recipient. A 55-year-old male who received a live, unrelated renal allograft 20 years ago presented with complaints of recurrent fever for ten to 12 months, weight loss over six months, progressive dysphagia more for solids for five to six months and joint pain and swelling involving the bilateral metacarpo-phalangeal and proximal interphalangeal joints. Biopsy from the esophageal ulcers revealed dense inflammation infiltrated with lymphocytes and macrophages with clusters of strongly positive intracellular fungal spores with a clear area or "halo-like" zone suggestive of Histoplasma capsulatum invasion. The patient was treated with intravenous liposomal amphotericin B for ten days and later switched over to oral itraconazole. Repeated endoscopy revealed significant improvement of the lesions.

How to cite this article:
Sharma L C, Falodia J, Kalla K, Kalla M, Gupta J B, Gupta S S, Beniwal P, Singh M N, Malhotra V, Agarwal D. Esophageal histoplasmosis in a renal allograft recipient. Saudi J Kidney Dis Transpl 2013;24:764-7

How to cite this URL:
Sharma L C, Falodia J, Kalla K, Kalla M, Gupta J B, Gupta S S, Beniwal P, Singh M N, Malhotra V, Agarwal D. Esophageal histoplasmosis in a renal allograft recipient. Saudi J Kidney Dis Transpl [serial online] 2013 [cited 2019 Dec 14];24:764-7. Available from: http://www.sjkdt.org/text.asp?2013/24/4/764/113874

   Introduction Top


Histoplasmosis is a progressive granulomatous disease caused by the intracellular dimorphic fungus Histoplasma capsulatum. Although gastrointestinal involvement is common in autopsy series, [1],[2],[3] clinical presentation with gut symptomatology is uncommon (3-12%). [4],[5]

We report a rare case of esophageal histoplas mosis from a non-endemic area in a renal allograft recipient on long-standing immunosuppression.


   Case Report Top


A 55-year-old man who received a live, unrelated renal allograft 20 years ago, with stable allograft function, presented with complaints of recurrent fever for ten to 12 months and approximately 8 kg weight loss over six months, progressive dysphagia for solids for five to six months and joint pain and swelling involving the bilateral metacarpo-phalangeal and proximal interphalangeal joints. He was on immunosuppression with two drugs (Prednisolone 10 mg/d and Azathioprine 75 mg/d). On examination, he was moderately built and poorly nourished, weighing 40 kg and had polyarthritis involving the bilateral metacarpo-phalangeal and proximal interphalangeal joints; otherwise, the cardiovascular, respiratory and abdominal examinations were unremarkable.

The hemogram was unremarkable except for mild anemia (Hb 10.5 gm%) and serum creatinine was 1.2 mg/dL, which was stable during the last six to 12 months. The measured creatinine clearance was 33.5 mL/min. Chest X-ray and computerized tomography (CT) of the thorax did not reveal any opacity, pleural effusion or hilar or mediastinal lymphadenopathy. HIV, HBsAg and anti-HCV were all negative. Blood and urine cultures for both bacteria and fungus were sterile.

Barium swallow revealed a narrowing at the mid- and lower esophagus [Figure 1], and subsequent esophagoscopy showed multiple ulcerated nodular lesions compromising the lumen at the mid- and lower esophagus [Figure 2]a. Biopsy from the esophageal ulcers revealed dense inflammation infiltrated with lymphocytes and macrophages, with clusters of strongly Periodic acid-Schiff PAS-positive intra-cellular fungal spores with a clear area or "halo-like" zone suggestive of Histoplasma capsulatum invasion [Figure 3].
Figure 1: Barium swallow showing narrowing at the mid- and lower esophagus.

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Figure 2:

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Figure 3: Biopsy from the esophageal ulcers revealed a large number of strongly PASpositive fungal spores with dense inflammation suggestive of Histoplasma capsulatum invasion.

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Considering the patient's long-standing fever, weight loss, polyarthritis and histopathology of esophageal ulcers demonstrating invasion of histoplasmosis, he was diagnosed to have disseminated histoplasmosis.

He was treated with intravenous liposomal amphotericin B for 10 days and later switched over to oral itraconazole. He had a remarkable improvement over the next five days, where his fever disappeared and his polyarthritis started regressing, with progressive gradual improvement in dysphagia. He underwent two sessions of esophageal dilatation. Repeated endoscopy revealed significant improvement in nodularity of the lesions [Figure 2]b.


   Discussion Top


Histoplasmosis presents clinically in different forms - asymptomatic infection; an acute or chronic pulmonary infection; mediastinal fibrosis or granulomas; and progressive disseminated histoplasmosis (PDH, which is typically seen in immunocompromised individuals who account for approximately 70% of the cases). [6] The spectrum of PDH ranges from an acute rapidly fatal course with diffuse interstitial or reticulonodular lung infiltrates causing respiratory failure, shock, coagulopathy and multiorgan failure, to a more subacute and chronic course with a focal organ distribution. The patients may present with fever, weight loss, hepatosplenomegaly, meningitis or focal brain lesions, ulcerations of the oral mucosa, gastrointestinal ulcerations and adrenal insufficiency. [6] Esophageal histoplasmosis can occur as a result of adjacent mediastinal adenitis during the course of acute infection or as a late consequence of progressive scarring in fibrosing mediastinitis or as a manifestation of progressive disseminated disease. Esophageal involvement in progressive disseminated histoplasmosis has been reported and is clinically distinct from the compressive syndromes resulting from mediastinitis. [7] These patients have underlying immunosuppression and present with dysphagia [8] or bleeding. [9]

Our patient had symptoms of dysphagia, and esophagoscopy revealed compromised lumen with nodular ulcerated lesion and histopathology that demonstrated strongly PAS-positive intracellular yeast cells in clusters with a clear area or "halo-like" zone around the organism, suggestive of histoplasmosis, which, to the best of our knowledge, is the first case to be reported in a renal transplant recipient. The basophilic cytoplasm of Histoplasma capsulatum retracts from the relatively thin cell wall, causing the halo-like zone around the organism. There was no evidence of mediastinal lymphadenopathy or fibrosis that could have caused the external compression to the esophagus. As histoplasmosis is uncommon in our country, serological tests for antigen and antibody detection are not widely available and hence, unfortunately, could not be performed.

Mortality without treatment in disseminated histoplasmosis is around 80%. [10] Studies have shown that early therapy with amphotericin B can reduce the mortality to <25%. [10],[11],[12],[13] In a recent study of AIDS patients, liposomal amphotericin B was more effective than the standard form of the drug in regard to time to resolution of fever and overall survival. [14] Thus, liposomal amphotericin B may be preferred for patients with severe or moderately severe disseminated histoplasmosis. Itraconazole is used for the treatment of disseminated histoplasmosis in patients with less-severe illness and also for maintenance therapy following an initial response to amphotericin B. [15] The duration of treatment for acute pulmonary histoplasmosis is six to 12 weeks, while that for PDH and chronic pulmonary histoplasmosis is ≥1 year. [6] Antigen levels in urine and serum should be monitored during and for at least one year after therapy for PDH. Stable or rising antigen levels suggest treatment failure or relapse, which constitute around 10-20% of the cases of PDH and around 80% of those with AIDS. [7]

 
   References Top

1.Goodwin RA Jr, Des Prez RM. State of the art: Histoplasmosis. Am Rev Respir Dis 1978; 117:929-56.  Back to cited text no. 1
    
2.Haggerty CM, Britton MC, Dorman JM, Marzoni FA Jr. Gastrointestinal histoplasmosis in suspected acquired immunodeficiency syndrome. West J Med 1985;143:244-6.  Back to cited text no. 2
    
3.Goodwin RA Jr, Shapiro JL, Thurman GH, Thurman SS, Des Prez RM. Disseminated histoplasmosis: Clinical and pathologic correlations. Medicine (Baltimore) 1980;59:1-33.  Back to cited text no. 3
    
4.Suh KN, Anekthananon T, Mariuz PR. Gastrointestinal histoplasmosis in patients with AIDS: Case report and review. Clin Infect Dis 2001; 32:483-91.  Back to cited text no. 4
    
5.Wheat LJ, Connolly-Stringfield PA, Baker RL, et al. Disseminated histoplasmosis in the acquired immune deficiency syndrome: Clinical findings, diagnosis and treatment, and review of the literature. Medicine (Baltimore) 1990;69: 361-74.  Back to cited text no. 5
    
6.Chadi A. Hage, L. Joseph Wheat. Histoplasmosis, Chapter 192 In: Harrison's principles of internal medicine, 17 th ed. In: Fauci AS, Kasper DL, Longo DL, eds. New York: McGraw Hill; 2008. p. 1244-6.  Back to cited text no. 6
    
7.Kahi CJ, Wheat LJ, Allen SD, Sarosi GA. Gastrointestinal histoplasmosis. Am J Gastroenterol 2005;100:220-31.  Back to cited text no. 7
    
8.Fucci JC, Nightengale ML. Primary esophageal histoplasmosis. Am J Gastroenterol 1997; 92:530-1.  Back to cited text no. 8
    
9.Forsmark CE, Wilcox CM, Darragh TM, Cello JP. Disseminated histoplasmosis in AIDS: An unusual case of esophageal involvement and gastrointestinal bleeding. Gastrointest Endosc 1990;36:604-5.  Back to cited text no. 9
    
10.Rubin HF, Yates JL, Brasher CA. The course and prognosis of histoplasmosis. Am J Med 1959;27:278-88.  Back to cited text no. 10
    
11.Sathapatayavongs B, Batteiger BE, Wheat J, Slama TG, Wass JL. Clinical and laboratory features of disseminated histoplasmosis during two large urban outbreaks. Medicine (Baltimore) 1983;62:263-70.  Back to cited text no. 11
    
12.Sarosi GA, Voth DW, Dahl BA, Doto IL, Tosh FE. Disseminated histoplasmosis: Results of long-term follow-up. A center for disease control cooperative mycoses study. Ann Intern Med 1971;75:511-6.  Back to cited text no. 12
    
13.Wilson DA, Muchmore HG, Tisdal RG, Fahmy A, Pitha JV. Histoplasmosis of the adrenal glands studied by CT. Radiology 1984;150: 779-83.  Back to cited text no. 13
    
14.Johnson PC, Wheat LJ, Cloud GA, et al. Safety and efficacy of liposomal amphotericin B compared with conventional amphotericin B for induction therapy of histoplasmosis in patients with AIDS. Ann Intern Med 2002; 137:105-9.  Back to cited text no. 14
    
15.Wheat J, Sarosi G, McKinsey D, et al. Practice guidelines for the management of patients with histoplasmosis. Infectious Diseases Society of America. Clin Infect Dis 2000;30:688-95.  Back to cited text no. 15
    

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Correspondence Address:
S S Gupta
Department of Nephrology, SMS Medical College and Hospital, Jaipur - 302 004
India
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DOI: 10.4103/1319-2442.113874

PMID: 23816727

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    Figures

  [Figure 1], [Figure 2], [Figure 3]

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