<i>“Strongyloides stercoralis</i> infestation in a severely malnourished (SAM) celiac disease child:” A rare case report from Western Rajasthan :Usha Verma, Vishakha Ashopa, Pooja Nareda, Eshank Gupta, Ekta Gupta, Prabhu Prakash, Tropical Parasitology

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Year : 2021 | Volume : 11 | Issue : 1 | Page : 60--63

“Strongyloides stercoralis infestation in a severely malnourished (SAM) celiac disease child:” A rare case report from Western Rajasthan

Usha Verma¹, Vishakha Ashopa¹, Pooja Nareda¹, Eshank Gupta¹, Ekta Gupta², Prabhu Prakash¹,
¹ Dr. S.N. Medical College, Jodhpur, Rajasthan, India
² Department of Orthodontics, AIIMS, Jodhpur, Rajasthan, India

Correspondence Address:
Prabhu Prakash
Dr. S.N. Medical College, Jodhpur, Rajasthan
India

Abstract

A case of Strongyloides stercoralis infection in severe protein energy malnutrition child with associated celiac disease (CD) is herein reported. The case was a 4-year-old, severely malnourished female admitted to the tertiary level hospital of Western Rajasthan, due to watery diarrhea, pain abdomen, and vomiting, not responding to treatment. The patient was HIV negative, nondiabetic, had no evidence of tuberculosis, Liver Function Test, Renal Function Test were normal was within normal limits. She had microcytic hypochromic anemia, hypoalbuminemia, and serum Antitissue transglutaminase (anti tTG IgA)was 301.35 U/ML which confirmed the diagnosis of CD. Stool parasitological examination revealed numerous rhabditiform larvae of threadworm “S. stercoralis.” After treatment with gluten free diet and Albendazole and Ivermectin, the patient recovered without evidence of S. stercoralis in follow-up stool samples.

How to cite this article:
Verma U, Ashopa V, Nareda P, Gupta E, Gupta E, Prakash P. “Strongyloides stercoralis infestation in a severely malnourished (SAM) celiac disease child:” A rare case report from Western Rajasthan.Trop Parasitol 2021;11:60-63

How to cite this URL:
Verma U, Ashopa V, Nareda P, Gupta E, Gupta E, Prakash P. “Strongyloides stercoralis infestation in a severely malnourished (SAM) celiac disease child:” A rare case report from Western Rajasthan. Trop Parasitol [serial online] 2021 [cited 2023 Mar 25 ];11:60-63
Available from: https://www.tropicalparasitology.org/text.asp?2021/11/1/60/315938

Full Text

Introduction

Celiac disease (CD) is an autoimmune disorder, occurs in genetically susceptible, triggered by gluten, leads to flattening of the small intestinal mucosa, subsequent nutrient malabsorption, and affects many organ systems.[1] It can involve people of all ages from middle infancy to old age.[2] Consuming gluten-containing foods can initiate a range of gastrointestinal (GI) symptoms like abdominal pain, diarrhea, flatulence, bloating, weight loss, and extra-intestinal signs such as anemia, osteoporosis, infertility, and nervous problems.[3] The “classical” malabsorption syndrome is characterized by diarrhea, steatorrhea, weight loss, and fatigue.[2]

The diagnosis of CD is based on a combination of symptoms, by detecting serum level of IgA-anti-tissue transglutaminase level, HLA, and duodenal histology. Severe Malnutrition can occur if the diagnosis is delayed.[2]

Treatment of choice is gluten-free diet or enzymes degrading gluten to be ingested with meals or genetically modified grains or immunotherapy.[2]

A clinical study, published in International Journal for Parasitology, infected CD patients on gluten-free diets with hookworms. Although the parasites did not cure anyone, they reduced the immune reaction when subjects ate food containing gluten.[3]

Some investigations show that various protozoa and helminths are connected with the main immune-mediated intestinal conditions including CD, inflammatory bowel diseases (IBD), and irritable bowel syndrome. CD is a digestive and autoimmune disorder that can damage the small intestine and characterized by a multitude GI and extra GI symptoms.[4]

Strongyloidiasis is an infection of an intestinal parasitic nematode, affects about 30–100 million people worldwide.[5],[6] Strongyloides stercoralis can exist in a nonparasitic, free-living state in the soil, so there are many potential sources of human infection with this pathogen.[7] It is endemic in areas where sanitary conditions are poor and where the climate is warm and humid.[8] The clinical manifestations of Strongyloidiasis vary greatly according to infection intensity and the immune-status of the patient.[9] This parasite should be ruled out in any patient from an endemic region who is to be treated with corticosteroids or immunosuppressive agents.[7] S. stercoralis can lead to death in cases of delayed diagnosis.[10]

In India, the true prevalence is still not known, possibly because the diagnosis is being delayed or missed. A significant increase of 15.5 cases per year from a North Indian tertiary hospital establishes that CD is increasingly being recognized.[11] The prevalence of CD in this north Indian community is 1 in 96.[12] In Northwest Rajasthan, Prevalence of 1.04% (1 in 96) among school children in Bikaner[13] but in S. stercoralis infection is low in non-HIV patients nondiabetic patients.

Case Report

A 4 years malnourished female child was suffering from abdominal pain, nausea, vomiting, weight loss, and her abdomen was distended. Suddenly she developed acute pain abdomen, diarrhea, and vomiting (intermittent bilious non bloody), not responded to treatment in the primary health center and she was referred to tertiary level hospital.

On admission, on general physical examination, head circumference was 44 cm, mid-upper arm circumference was 9.5 cm, height was 81 cm and weight was 6.920 kg.

On routine blood investigations, her hemoglobin was 8.7, had microcytic, hypochromic anemia, nondiabetic, HIV negative and her LFT, RFT was within the normal range. X-ray revealed few calcified foci in the chest and distended abdomen [Figure 1] bowel loops on flat plate abdomen [Figure 2] and on ultrasonography examination [Figure 3], prominent bowel loops with sluggish peristalsis [Figure 4] were noted. The patient was given symptomatic treatment but there was no relief in her symptoms. Her gastric aspiration was negative for mycobacterial infection (MTB) by CBNAAT testing and Acid Fast staining. Despite symptomatic treatment, she lost 1.5 kg weight and there was no relief in diarrhea. Her blood was sent for anti-tissue transglutaminase testing (TTG) and anti-tissue transglutaminase was positive (301.35 IU/ml) which confirmed the diagnosis of CD.

On stool macroscopic examination no visible parasite was seen, Wet examination of a stool specimen showed numerous larvae (10–15) of S. stercoralis [Figure 5] which were long thread-like in shape. The patient was advised Gluten-free diet and was treated with albendazole 10 ml OD HS stat along with symptomatic treatment. As the patient had severe malabsorption, she was referred to MTC ward where she was treated with Ivermectin and a high protein diet. On follow-up after 15 days, she continued to have negative stool microscopy for Strongyloides larvae and she was discharged healthy.{Figure 1}{Figure 2}{Figure 3}{Figure 4}{Figure 5}

Discussion

In the present case, the diagnosis was concluded both with serological testing for IgA and IgG antibodies to tissue transglutaminase positivity and clinical response to the gluten-free diet and microbiological findings by showing larvae on stool examinations.

A study done by M. Rostami Nejad et al. detected the prevalence of intestinal parasites in CD patients. Out of 25 celiac patients (61.2% female and 35.8% male), with a mean age of 41.66 years, 7 (28%) patients were infected with pathogen and nonpathogen parasites. The frequency rate of Giardia lamblia, Entamoeba coli, Endolimax nana, Blastosystis hominis, cryptosporidium parvoum were 3 (0.75%), 2 (0.5%), 1 (0.25%), 2 (0.5%), 2 (0.5%), respectively.[14]

A similar study was done by Ugur Korkmaz et al. in Turkey, detected S. stercoralis larvae within the duodenal crypts with CD from a 49 years old male patient. This case emphasizes the importance of a duodenal biopsy. This was the first case in the literature to show CD and S. stercoralis together in Turkey.[15]

According to Behera B et al., in both children and adults, untreated CD is the most common cause of malabsorption syndrome, and in these patients, some pathogenic parasites like Giardia lamblia, Ancylostoma duodenale, Entamoeba histolytica/dispar, Cyclospora cayetanensis, Hymenolepis nana, Cryptosporidium, Cyclospora, and Isospora belli more often colonized compared to healthy control.[16]

Many investigations showed the curative effects of controlled parasitic nematode infection on autoimmune disorders like IBD,[17],[18],[19] but the exact mechanism(s) of these effects are not clear. S. stercoralis, an intestinal nematode, is a common intestinal parasite in tropical areas, for example, Southeast Asia, Latin America, Sub-Saharan Africa, and many European countries.[20] As it can occasionally imitate IBD, particularly UC, it is important to be kept out.[21]

According to S. K. Agrawal, A 19 year male from a rural background with coeliac disease, without having any known immunosuppression (Antibodies to HIV, HBV, HCV were negative) presented with complaints of weight loss, chronic loose stools alternating with constipation, and pain abdomen since the last 7 months. On microscopic examination, several ova of helminths and cysts of protozoa were detected. In addition, larvae of S. stercoralis were also detected.[22]

The prevalence of CD thus was 1%, which was in concordance with screening studies using serological markers conducted in North Indian children.[4]

As a maximum of patients were observed in South Rajasthan to be of 5–10 years age group, hence related clinical symptoms were maximally observed in the same age group of celiac as well as the nonceliac group. Whereas children of <5 years age and >10 years age group, both celiac as well as nonceliac group, showed almost similar distribution of related clinical symptoms.[2]

A study done by Kumar Saurabh, detected G. lamblia and H. nana infestations, along with low immunoglobulin levels and the duodenal biopsy characteristically resembling to CD.[23]

Conclusion

Routine screening for CD must be carried out in all patients of chronic diarrhea with associated PEM, especially in the younger age group. Stool examination should be carried out to rule out associated parasitic infection. Finding of S. stercoralis in non-HIV CD patient is a rare infection in Western Rajasthan.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

References

1	Gautam A, Jain BK, Midha V, Sood A, Sood N. Prevalence of celiac disease among siblings of celiac disease patients. Indian J Gastroenterol 2006;25:233-5.
2	Sharma M, Mandot S. Prevalence and clinical profile of celiac disease among malnourished children in South Rajasthan, India. Int J Contemp Pediatr 2018;5:997-100.
3	Waffle V, et al. New Insight Into Celiac Disease; 2014.
4	Mohammadi R, Hosseini-Safa A, Ehsani Ardakani MJ, Rostami-Nejad M. The relationship between intestinal parasites and some immune-mediated intestinal conditions. Gastroenterol Hepatol Bed Bench 2015;8:123-31.
5	Bethony J, Brooker S, Albonico M, Geiger SM, Loukas A, Diemert D, et al. Soil-transmitted helminth infections: Ascariasis, trichuriasis, and hookworm. Lancet 2006;367:1521-32.
6	Olsen A, van Lieshout L, Marti H, Polderman T, Polman K, Steinmann P, et al. Strongyloidiasis–The most neglected of the neglected tropical diseases? Trans R Soc Trop Med Hyg 2009;103:967-72.
7	Burke JA. Strongyloidiasis in childhood. Am J Dis Child 1978;132:1130-6.
8	Hall A, Conway DJ, Anwar KS, Rahman ML. Strongyloides stercoralis in an urban slum community in Bangladesh: Factors independently associated with infection. Trans R Soc Trop Med Hyg 1994;88:527-30.
9	Foreman EB, Abraham PJ, Garland JL. Not your typical strongyloides infection: A literature review and case study. South Med J 2006;99:847-52.
10	Lim S, Katz K, Krajden S, Fuksa M, Keystone JS, Kain KC. Complicated and fatal Strongyloides infection in Canadians: Risk factors, diagnosis and management. CMAJ 2004;171:479-84.
11	Sood A, Midha V, Sood N, Kaushal V, Puri H. Increasing incidence of celiac disease in India. Am J Gastroenterol 2001;96:2804-5.
12	Makharia G, Verma AK, Amarchand R. Prevalence of celiac disease in the northern part of India: A community based study. J Gastroenterol Hepatol 2010;26:894-900.
13	Garg VK, Katewa V. Prevalence of celiac disease in school children in Bikaner region of North West Rajasthan. Int J Sci Res 2017;6:5 131-3.
14	Nejad MR, Rostami K, Nazemalhosseini E, Zali MR. Intestinal parasites in patients with celiac disease: A clinical study, 13^th international congress on infectious diseases Abstracts, poster presentations.
15	Korkmaz U, Duman AE, Gurkan B, Sirin G, Topcu Y, Dindar G, et al. Nonresponsive celiac disease due to Strongyloides stercoralis infestation. Intern Med 2012;51:881-3.
16	Behera B, Mirdha BR, Makharia GK, Bhatnagar S, Dattagupta S, Samantaray JC. Parasites in patients with Malabsorption syndrome: A clinical study in children and adults. Dig Dis Sci 2008;53:672-9.
17	Elliott DE, Weinstock JV. Helminth-host immunological interactions: Prevention and control of immune-mediated diseases. Ann N Y Acad Sci 2012;1247:83-96.
18	Ince MN, Elliott DE, Setiawan T, Metwali A, Blum A, Chen Hl, et al. Role of T cell TGF-β signaling in intestinal cytokine responses and helminthic immune modulation. Eur J Immunol 2009;39:1870-78.
19	Schnoeller C, Rausch S, Pillai S, Avagyan A, Wittig BM, Loddenkemper C, et al. A helminth immunomodulator reduces allergic and inflammatory responses by induction of IL-10-producing macrophages. J Immunol 2008;180:4265-72.
20	Moghadam KG, Khashayar P, Hashemi M. Gastrointestinal strongyloidiasis in immunocompromised patients: A case report. Acta Med Indones 2011;43:191-4.
21	Dave M, Purohit T, Razonable R, Loftus EV Jr. Opportunistic infections due to inflammatory bowel disease therapy. Inflamm Bowel Dis 2014;20:196-212.
22	Agrawal SK. Rising trend of seroprevalence of human amoebiasis in tertiary care hospital of north India PGIMER, Chandigarh, Chandigarh, 17^th international congress on infectious diseases. Int J Infect Dis 2016;45S: 1-477.
23	Saurabh K, Nag VL, Khera D, Elhence P. Giardiasis mimicking celiac disease in a patient of common variable immunodeficiency. Trop Parasitol 2017;7:125-7.