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  Table of Contents    
CASE REPORT  
Year : 2021  |  Volume : 64  |  Issue : 4  |  Page : 827-830
Isolated gastrointestinal histoplasmosis: A rare diagnosis of pediatric chronic abdominal pain


1 Department of Paediatric Gastroenterology, AMRI Hospitals, Mukundapur, West Bengal, India
2 Department of Paediatrics, AMRI Hospitals, Mukundapur, West Bengal, India
3 Department of Histopathology, AMRI Hospitals, Mukundapur, West Bengal, India

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Date of Submission08-Apr-2020
Date of Decision22-May-2020
Date of Acceptance05-Oct-2020
Date of Web Publication20-Oct-2021
 

   Abstract 


Gastrointestinal (GI) histoplasmosis usually occurs as a part of disseminated histoplasmosis in immunocompromised or elderly subjects. Isolated histoplasmosis involving the GI tract in an immunocompetent host is very rare. It is also not considered as an etiology for chronic abdominal pain in children. Here we present an 8-year-old boy with abdominal pain and weight loss who underwent treatment for tuberculosis but on reinvestigation was diagnosed as GI histoplasmosis. He responded well to treatment and achieved good catch up growth.

Keywords: Abdominal pain, chronic, gastrointestinal, histoplasma, ileal ulcer

How to cite this article:
Acharyya BC, Acharyya S, Chakrabarty H. Isolated gastrointestinal histoplasmosis: A rare diagnosis of pediatric chronic abdominal pain. Indian J Pathol Microbiol 2021;64:827-30

How to cite this URL:
Acharyya BC, Acharyya S, Chakrabarty H. Isolated gastrointestinal histoplasmosis: A rare diagnosis of pediatric chronic abdominal pain. Indian J Pathol Microbiol [serial online] 2021 [cited 2021 Dec 1];64:827-30. Available from: https://www.ijpmonline.org/text.asp?2021/64/4/827/328548





   Introduction Top


Chronic abdominal pain is a common problem in children. In the majority the pain is functional and the basic investigations are not contributory. When red flag signs are present, suggestive of an organic pain it is essential to investigate the children appropriately and at times interesting and rare diagnosis may emerge. Here we present an 8-year-old immunocompetent boy who presented with organic chronic abdominal pain initially diagnosed as tuberculosis. Since he was unresponsive to antitubercular medicine, he was clinically suspected to have inflammatory bowel disease. But surprisingly the final diagnosis was isolated gastrointestinal histoplasmosis (GIH).


   Case Report Top


An 8-year-old boy presented with 1 year of colicky abdominal pain and 4 kg weight loss in 4 months. He was born in Afghanistan and has moved with his family to India 2 years ago. The pain was colicky occurred everyday, disturbed his sleep and aggravated by food intake. He had occasional nonbilious vomiting. He was constipated and passed motion once in 3–4 days. His appetite was poor and he had night sweats. He had no fever or respiratory symptoms. There was no history of jaundice, GI bleed, or surgery. He was investigated elsewhere and treated with antitubercular medicine for more than 9 months without much benefit.

On examination, the boy was below the 9th percentile for height and weight. He had pallor, mild digital clubbing, and no cyanosis, icterus, edema or significant lymphadenopathy. His abdomen was distended (no free fluid) with soft hepatomegaly (1 cm below RCM) and spleen was not palpable. The respiratory system was normal. USG was reported as multiple mesenteric lymphadenopathies, a few more than 2 cm in size. A CECT abdomen revealed thickened ileal and jejunal loops with mesenteric lymphadenopathy without matting or central necrosis. His Hb was 10 g/dL, TLC was 16,000/dL with neutrophil 70%, CRP was 55 mg/dL and ESR was 38 mm. Biochemical tests of the liver were normal except an albumin of 3.2 g/dL. Fecal Calprotectin was 1100 mcg/gm (normal up to 50 mcg/gm). Endoscopic evaluation was planned.

Upper GI endoscopy showed extensive white lesions suggestive of esophageal candidiasis, biopsy was not taken. Ileocolonoscopy revealed multiple, patchy nodular ulcers involving the entire mucosa from the sigmoid colon to caecum. The ileocecal valve was severely edematous with ulcerations and ileum showed large deep ulcers with surrounding edema and erythema [Figure 1]a and [Figure 1]b. Histopathology from colon and ileum showed marked cryptitis, chronic active inflammation with histoplasma capsulatum inside macrophages which was confirmed with GM (Grocott–Gomori methenamine) silver stain [Figure 2]a and [Figure 2]b. Treatment was initiated with oral Itraconazole to which he responded well with improvement of appetite and relief of pain. X-ray chest and HRCT of the chest were essentially normal. He was investigated thoroughly for any immunodeficiency status including immunoglubulin profile, NBT, HIV status, CD4, CD8 counts and ratio and all were normal.
Figure 1: (a-c) - Colonic Nodular Ulcer; Ileal Ulcers and post-treatment ileum and ileoileal anastomotic stoma

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Figure 2: (a-c) Histoplasma in the Ileal tissue section H and E stain, GM Silver stain and Histoplasma in Lymphnodes (GM stain)

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He presented one month later with 3 days history of abdominal distension, vomiting, and features of subacute intestinal obstruction. CECT abdomen revealed an ileal stricture. This did not respond to endoscopic balloon dilatation. He underwent laparoscopic ileal bypass surgery along with the removal of mesenteric lymph nodes. The histopathology of the nodes also showed Histoplasma [Figure 2c]. Liposomal Amphotericin B was initiated and continued for 10 days which was later switched over to oral Itraconazole for 6 months. He responded well to therapy. Post-treatment colonoscopy revealed normal mucosa [Figure 1c]. His weight and height increased to between 25th and 50th centile and he resumed attending school.


   Discussion Top


GIH is an uncommon entity. It is reported as a part of disseminated histoplasmosis in 50–70% of cases due to hematogenous (borne by the macrophages) seeding of the GI tract.[1] GIH can affect both immunocompetent and immunocompromised individuals. Patients with acquired immune deficiency syndrome (AIDS),[2] are especially at risk. Acute pulmonary infection is commoner than the affection of any other organ in a healthy host.[3] Surprisingly index case had GI affection without any pulmonary infection.

The pathological spectrum of GI and hepatic histoplasmosis in adults and children had been characterized by Lamp et al. from 52 pathology specimens.[4] They concluded that though disseminated histoplasmosis is common in Immunodeficiency states GI and hepatic histoplasmosis can occur in both immunocompetent and immunocompromised patients, in children as well as in adults, and in endemic visa-vis nonendemic areas.

Various symptoms had been described in the GIH. In endemic areas, chronic unexplained diarrhea is a common manifestation.[5] Abdominal pain and weight loss are found in 30–50% of adults with GI involvement,[6] which were the two main complaints of the index case.

Endoscopic findings of GIH include erythema, plaque-like lesions, ulcers with raised margins and large deep irregular ulcers (both of these ulcers were present in the index patient). Child above had ulceration in colon and ileum. Ileal ulcers produced stricture once healing started. The lesions of GI tract histoplasmosis occur predominantly in the ileocecal region, like intestinal tuberculosis presumably because of an abundance of lymphoid tissue and that was the major site of involvement in this child.[5]

Pathologic examination and fungal cultures of GI tract tissue specimens are sensitive methods for diagnosing GI tract histoplasmosis.[5] Four pathologic patterns are commonly described: (1) no visible gross abnormalities but the presence of infected macrophages in the lamina propria, (2) collection of infected macrophages presenting as pseudopolyps or plaques, (3) ulceration with tissue necrosis, and (4) localized inflammation leading to stricture formation.[7],[8] GM methenamine silver nitrate staining (GM Silver stain) of the tissue specimen may be used to aid in diagnosis.[9] Although an invasive procedure is required to procure specimens, it provides a rapid and accurate diagnosis.[7] In our patient initial H and E stain showed the yeast in the colonic and ileal tissue which was reconfirmed by silver staining [Figure 2a-c]. Fungal culture was not needed.

A marked resemblance of clinical features of intestinal histoplasmosis with Crohn's disease and intestinal tuberculosis makes it difficult to differentiate clinically. Abdominal pain, chronic diarrhea, and weight loss might be present in all three. Endoscopicaly also histoplasmosis can produce deep ulcers like Crohn's disease but ulcers with nodular or raised margins are more pathognomic of Histoplasma. Ileocecal involvement in all three can confuse the issue more. Ultimately histopathology is the diagnostic tool. All three might have a granuloma. Tuberculosis will have caseating, complete, large (>400 μm), numbering more than 5 granulomas spreading to the submucosa. Crohn's Disease will have incomplete smaller (<200 μm) noncaseating, mucosal, pericryptal and usually less than 4 granulomas. In histoplasmosis, granulomas are like Crohn's disease but rarer. Visible Fungal bodies inside the macrophages confirm the diagnosis which is absent in the other two conditions.

Currently, recommended treatment in unstable patients with severe disease is with Amphotericin B for disseminated histoplasmosis. In stable patients such as our case, Itraconazole for 6 to 18 months is the treatment of choice.[10] For our child, treatment was started with Itraconazole but during the hospitalization for surgery, Liposomal Amphotericin B was given for 10 days. Then it was switched to Itraconazole for the next 6 months.

It is possible that GIH is underdiagnosed. In view of the involvement of terminal ileum and caecum, the presence of nodular punched out ulcer, intraabdominal lymphadenopathy, and failure of antitubercular treatment, Inflammatory Bowel Disease was strongly considered for the present case, until the biopsy report unveiled the diagnosis. A true incidence or prevalence of GIH in immunocompetent pediatric population is not known but it is considered very rare. Isolated GIH is mostly reported in elderly adults with endstage renal disease[5],[6] and few immunocompetent hosts without any co-morbidities [Table 1].[11],[12],[13],[14],[15],[16],[17],[18],[19] To the best of our knowledge, this is the first reported case of Isolated GIH in an immunologically normal child.
Table 1: GI Histoplasmosis in immunocompetent hosts without any comorbidity

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Pediatricians should be aware of this entity and consider GIH as a differential for GI Tuberculosis or Inflammatory Bowel Disease. Empirical use of antitubercular medication without a definitive diagnosis should be condemned. It is also important to note that GIH has a good prognosis if treated properly, but if remains undetected can have fatal consequences especially in immunocompromised hosts.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Kahi CJ, Wheat JR, Allen SD, Sarosi GA. Gastrointestinal histoplasmosis. Am J Gastroenterol 2005;100:220-31.  Back to cited text no. 1
    
2.
Haggerty CM, Britton MC, Dorman JM, Marzoni Jr FA. Gastrointestinal histoplasmosis in suspected acquired immunodeficiency syndrome. West J Med 1985;143:244-63.  Back to cited text no. 2
    
3.
Goodwin RA Jr, Des Prez RM. State of the art: Histoplasmosis. Am Rev Respir Dis 1978;117:929-56.  Back to cited text no. 3
    
4.
Lamps LW, Molina CP, West AB, Haggitt RC, Scott MA. Pathologic spectrum of gastrointestinal and hepatic histoplasmosis. Am J Clin Pathol 2000;113:64-72.  Back to cited text no. 4
    
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Jain S, Koirala J, Castro-Pavia F. Isolated Gastrointestinal Histoplasmosis: Case Report and Review of the Literature. South Med J 2004;97:172-4.  Back to cited text no. 5
    
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Lee JT, Dixon MR, Murrell Z, Konyalian V, Agbunag R, Rostami S, et al. Colonic histoplasmosis presenting as colon cancer in nonimmunocompromised patient. Report of a case and review of the literature. Am Surgeon 2004;70:959-63.  Back to cited text no. 6
    
7.
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. 7
    
8.
Heneghan SJ, Li J, Petrossian E, Bizer LS. Intestinal perforation from gastrointestinal histoplasmosis in acquired immunodeficiency syndrome: Case report and review of the literature. Arch Surg 1993;128:464-6.  Back to cited text no. 8
    
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Bradsher RW. Histoplasmosis and blastomycosis. Clin Infect Dis 1996;22(Suppl 2):S102-11.  Back to cited text no. 9
    
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Wheat LJ, Freifeld AG, Kleiman MB, Baddley JW, McKinsey DS, Loyd JE, et al. Practice guidelines for the management of patients with histoplasmosis: Infectious diseases society of America. Clin Infect Dis 2007;45:807-25.  Back to cited text no. 10
    
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Parikh F, Londhe VA, Khude S, Kamat R, Sandhya A. Gastrointestinal histoplasmosis mimicking abdominal tuberculosis. J Assoc Physicians India 2009;57:76-8.  Back to cited text no. 11
    
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Yang B, Lu L, Li D, Liu L, Huang L, Chen L, et al. Colonic involvement in disseminated histoplasmosis of an immunocompetent adult: Case report and literature review. BMC Infect Dis 2013;13:143-6.  Back to cited text no. 12
    
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Badyal RK, Gupta R, Vaiphei K. Diffuse perforated necrotising amoebic colitis with histoplasmosis in an immunocompetent individual presenting as an acute abdomen. BMJ Case Rep 2013;2013:bcr2013009059.  Back to cited text no. 13
    
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Chaudhari D, Mckinney J, Hubbs D, Young M. Mediastinal histoplasmosis presenting as dysphagia: A case report with literature review. Clin J Gastroenterol 2013;6:315-8.  Back to cited text no. 14
    
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Wu Y, Zhuang J, Chen X. Histoplasmosis identified by colonoscopy: A case report. Baotou Med Coll J 2015;31:109-10.  Back to cited text no. 15
    
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Zhu L-L, Wang J, Wang Z-J, Wang Y-P, Yang J-L. Intestinal histoplasmosis in immunocompetent adults. World J Gastroenterol 2016;22:4027-33.  Back to cited text no. 16
    
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Kweyamba V, Apiyo M, Olika B, Kituuka O. A case of a 4-year-old boy with a mesenteric chylous cyst infected with histoplasma capsulatum. Case Rep Surg 2016;2016:4296059.  Back to cited text no. 17
    
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Sharma R, Lipi L, Gajendra S, Mohapatra I, Goel RK, Duggal R, et al. Gastrointestinal histoplasmosis: A case series. Int J Surg Pathol 2017;25:592-8.  Back to cited text no. 18
    
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Dang Y, Jiang L, Zhang J, Pan B, Zhu G, Zhu F, et al. Disseminated histoplasmosis in an immunocompetent individual diagnosed with gastrointestinal endoscopy: A case report. BMC Infect Dis 2019;19:992.  Back to cited text no. 19
    

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Correspondence Address:
Bhaswati C Acharyya
Consultant Pediatric Gastroenterologist and Hepatologist, AMRI Hospitals, 230 Barakhola Lane, Purba Jadavpur, Kolkata - 700 099, West Bengal
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/IJPM.IJPM_235_20

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