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  Table of Contents    
CLINICO-PATHOLOGY CONFERENCE  
Year : 2015  |  Volume : 58  |  Issue : 4  |  Page : 500-505
A 36-year-old man with vomiting, pain abdomen, significant weight loss, hyponatremia, and hypoglycemia


1 Department of Pathology, Command Hospital (Air Force), Bengaluru, Karnataka, India
2 Department of Medicine, Command Hospital (Air Force), Bengaluru, Karnataka, India

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Date of Web Publication4-Nov-2015
 

   Abstract 

Diagnosis of Strongyloides stercoralis hyperinfection can be a challenge. The key to a timely diagnosis is to have a high index of suspicion. We present a rare case of a 36-year-old human immunodeficiency virus negative male patient, who was on multidrug therapy for lepromatous leprosy and was treated for type 2 lepra reactions with steroids in the past. The patient presented with vomiting and pain abdomen, persistent hyponatremia, and terminal hypoglycemia. He had features of malnutrition and had a rapid downhill course following admission. A diagnosis of S. stercoralis hyperinfection with sepsis and multiorgan failure, adrenal hemorrhage, and syndrome of inappropriate antidiuretic hormone secretion was established on a postmortem examination.

Keywords: Adrenal crisis, hyponatremia, leprosy, Strongyloides stercoralis hyperinfection

How to cite this article:
Mutreja D, Sivasami K, Tewari V, Nandi B, Nair G L, Patil SD. A 36-year-old man with vomiting, pain abdomen, significant weight loss, hyponatremia, and hypoglycemia. Indian J Pathol Microbiol 2015;58:500-5

How to cite this URL:
Mutreja D, Sivasami K, Tewari V, Nandi B, Nair G L, Patil SD. A 36-year-old man with vomiting, pain abdomen, significant weight loss, hyponatremia, and hypoglycemia. Indian J Pathol Microbiol [serial online] 2015 [cited 2019 Dec 16];58:500-5. Available from: http://www.ijpmonline.org/text.asp?2015/58/4/500/168871



   Clinical Protocol Top


A 36-year-old male presented with a history of pain abdomen of 1-day duration. Pain was acute, continuous, moderate in intensity and localized to the epigastric region without any radiation. There was associated vomiting for 2–3 days, nonprojectile, 6–8 episodes/day. The patient gave a history of gradual ill health for the past 1 month with generalized weakness, anorexia, lassitude, and significant weight loss of 12 kg. He was a known case of lepromatous leprosy on multidrug therapy (MDT) and had been treated 1-year prior to present illness, with steroids (prednisolone 50 mg/day) and thalidomide for type 2 lepra reactions.


   Clinical Examination and Investigations Top


Clinical evaluation revealed an ill looking thin individual in pain. He was afebrile, with pallor and bilateral pitting pedal edema. His body mass index was 16.3. Pulse and blood pressure were normal. Abdominal examination revealed epigastric tenderness without any palpable mass. Other systems were unremarkable. Investigations revealed hyponatremia, hypoalbuminemia, hypocholestremia, and hypotriglyceridemia [Table 1] with a mild normocytic normochromic anemia. Urine showed 1+ proteins. Prothrombin time and activated partial thromboplastin time were prolonged. Serum bilirubin and lactate dehydrogenase (LDH) were raised with mild transaminitis. Enzyme-linked immunosorbent assay for human immunodeficiency virus (HIV), hepatitis B and C were negative. Blood culture sent on day 1 was sterile.
Table 1: Laboratory Investigations*

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An urgent ultrasound abdomen revealed mild ascites with the presence of free fluid in subdiaphragmatic, paraaortic, hepatic and pelvic regions and a distended gall bladder with sludge.


   Course in the Hospital Top


The patient was managed with intravenous normal saline 1 L/day, oral salt 5 g 6 hourly, antacids and antiemetics. Laboratory investigations revealed persistent hyponatremia with serum sodium in the range of 108–112 mEq/L. The patient was planned to be worked up for an abdominal malignancy with possible dissemination. On day 3, an upper gastrointestinal (GI) endoscopy revealed intensely erythematous and friable gastric mucosa with yellowish deposits [Figure 1]a and [Figure 1]b and a biopsy was undertaken. Contrast enhanced computed tomogram (CECT) of the abdomen showed ascites, concentric thickening of the cecum, ileocecal wall and terminal ileum. Multiple subcentrimentric moderately enhancing mesenteric and paracecal lymph nodes without necrosis were also seen. The picture was suggestive of ileocecal tuberculosis [Figure 1]c. C-reactive protein was positive. On the 4th day after admission, the patient developed recurrent hypoglycemic episodes, with blood sugar levels ranging from 36 to 45 mg/dL, associated acidotic breathing, and hypotension. Arterial blood gas analysis revealed severe metabolic acidosis. A guided ascitic tap showed neutrophilic predominant exudative fluid. Patient developed desaturation, continued to deteriorate, and died on the 4th day after admission.
Figure 1: (a and b) Upper gastrointestinal endoscopy showing intensely erythematous and friable gastric mucosa with yellowish slough. (c) Contrast enhancing computed tomogram of abdomen showing cecal wall thickening and multiple subcentrimentric moderately enhancing mesenteric and paracecal lymph nodes without necrosis

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   Unit's Final Diagnosis Top


A 36-year-old male with possible gastric lymphoma with GI tuberculosis. Cause of death: Acute adrenal insufficiency with sepsis and multiorgan failure.


   Discussion on Clinical Protocol by Associate Professor Medicine Top


A young male, known case of multibacillary leprosy on MDT presented with ill health of 1-month duration with symptoms of generalized weakness, anorexia, lassitude, and significant weight loss of 12 kg. On admission, his main complaint was epigastric pain and vomiting. Clinical evaluation revealed features of severe malnutrition in the form of weight loss, edema, hypoproteinemia, and proteinuria. Deranged liver function, hyponatremia, and coagulopathy were also present. An ultrasound of the abdomen showed mild ascites with the distended gall bladder. A clinical differential diagnosis of acute gastritis, acute cholecystitis, and acute pancreatits in a setting of malnourishment were considered.

Severe malnourishment may have resulted from a possible protein-losing enteropathy, prolonged illness such as tuberculosis with associated poor intake. This patient also had severe persistent hyponatremia. Causes of hyponatremia are summarized in [Table 2]. Hyponatremia in this patient was attributed to prolonged vomiting or may have been dilutional due to fluid retention. It may have also resulted from syndrome of inappropriate antidiuretic hormone secretion (SIADH) in the setting of disseminated malignancy as the calculated plasma osmolality was low. The causes of SIADH are enumerated in [Table 3].
Table 2: Causes of hyponatremia

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Table 3: Causes of syndrome of inappropriate antidiuretic hormone secretion

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On day 3, an upper GI endoscopy showed features of gastroduodenitis. Endoscopic differential diagnoses of severe gastroduodenitis due to an infectious etiology, gastric lymphoma, and amyloidosis were considered.

Based on CECT findings, a possibility of disseminated tuberculosis with associated SIADH was thought of. The pointers in favor of tuberculosis were malnourishment, raised LDH, deranged liver function, gastric involvement on endoscopy, and abdominal imaging findings. Tubercular involvement of stomach is reported to be extremely rare and presents with gastric outlet obstruction.[1] Hence, an associated gastric lymphoma was considered. Features supporting the diagnosis of a lymphoma were the patient's presentation with B symptoms with elevated LDH. Amyloidosis was also a differential diagnosis in view of proteinuria, hypoproteinemia, liver involvement and evidence of adrenal crisis at the time of death. However amyloidosis is rare in the present era of MDT for leprosy, which is reported more often in the pre-MDT period.[2],[3] Persistent hyponatremia and terminal hypoglycemia were pointers towards adrenal insufficiency in the setting of a disseminated abdominal malignancy or sepsis. Further, multiorgan failure with sepsis was also considered in view of an exudative ascitic tap with neutrophil predominance. Lepromatous leprosy in the absence of fresh skin or nerve lesion or evidence of lepra reaction was unlikely to have contributed to death in this case.[4]


   Final Clinical Diagnosis Top


Gastric lymphoma with associated GI tuberculosis with a differential diagnosis of amyloidosis.

Terminal event: Sepsis with multiorgan failure.

Comorbidity: Lepromatous leprosy.


   Pathology Protocol by Associate Professor Pathology Top


Gross findings

At autopsy, the body of an emaciated young male with anasarca, marked facial puffiness, postauricular edema, pedal edema, sacral, and scrotal edema was seen. Rigor mortis was absent. There were bilateral pleural effusions and turbid peritoneal effusion.

The most significant findings were confined to the abdominal cavity. Small whitish plaques (1–5 mm) were seen on the omentum, the posterior surface of the pancreas, mesentry, and retroperitoneum. Stomach and small intestines showed mucosal congestion and erosions. Large intestines showed numerous multiple discrete ulcers [Figure 2]a, many of them showing overhanging edges [Figure 2]b with hemorrhagic slough on the surface and edematous intervening mucosa. Multiple enlarged mesenteric and retroperitoneal lymph nodes with yellowish cut surface measuring 1 to -2.5 cm in size were dissected. The right adrenal was distended and hemorrhagic [Figure 3]. The liver showed a nutmeg appearance [Figure 4]. Spleen showed tiny whitish nodularities on the surface [Figure 5]. Lungs, kidneys, testes, and brain, were unremarkable on gross.
Figure 2: (a) Formalin fixed specimen of cecum showing discrete ulcers, varying in size from 2 mm to 10 mm. (b) Cecum, mucosal surface showing ulcer with overhanging mucosa

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Figure 3: Formalin fixed gross specimen of both adrenals, with hemorrhagic right adrenal (arrow)

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Figure 4: Formalin fixed gross specimen of liver showing normal capsule. Inset shows slice of liver with nutmeg appearance

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Figure 5: Formalin fixed gross specimen of spleen showing whitish nodules on surface (arrows)

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Microscopic findings

Unstained and stained smears from intestinal contents revealed numerous larval and adult forms of Strongyloides stercoralis [Figure 6]. Sections from stomach, intestines, and whitish plaques from omentum, mesentery, retroperitoneum, and posterior surface of pancreas showed larval forms of strongyloides with surrounding lymphoplasmacytic inflammation [Figure 7]a and [Figure 7]b. Small bowel showed transmural involvement with definite vascular permeation. Cecum showed flask shaped amoebic ulcers with numerous amoebic trophoziotes in addition to strongyloides [Figure 8]a and [Figure 8]b. Sections from lymph nodes showed strongyloides larvae in the subcapsular sinuses, while amoebic trophozoites and numerous lipofuscin laden macrophages were found in the medullary sinuses. Liver showed features of passive venous congestion and macrovesicular steatosis, more prominent in zone 3 [Figure 9]. Sections of spleen through the areas of whitish nodules showed hyalosclerosis [Figure 10]. Marked autolytic changes were seen in the gallbladder mucosa. Kidneys showed acute tubular necrosis. There was evidence of alveolar hemorrhages and hemosiderin laden macrophages in the lower lobes of both lungs. However, no larval forms could be demonstrated. Antemortem biopsies from stomach and duodenum also confirmed numerous strongyloides larvae. However, the results were available after the patient's demise [Figure 11]a and [Figure 11]b.
Figure 6: (a) Unstained preparation of intestinal contents showing strongyloides larva (×400). (b) MGG stained preparation of intestinal contents showing numerous larval forms (×100). Inset shows higher magnification of rhabditiform larva with well-defined esophagus

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Figure 7: (a) H and E stained section of small intestine showing adult and larval forms of strongyloides (×400). (b) H and E stained section of whitish plaque on mesentery showing larva with surrounding lymphoplasmacytic inflammation (×400)

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Figure 8: (a) H and E stained section through cecal ulcer showing adult strongyloides worm (thick arrow) and larval form (thin arrow) (×100). (b) Amoebic trophozoites within the ulcerated mucosa (H and E, ×1000)

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Figure 9: Section of liver normal showing centrilobular sinusoidal congestion. Inset shows micro and macrovesicular steatosis confined to zone 3. Portal tracts were within normal limits (H and E, ×100)

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Figure 10: Section of spleen through the whitish nodule showing hyalosclerosis (×400)

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Figure 11: Antemortem biopsies from stomach (a) and duodenum (b) numerous strongyloides larvae in crypts (H and E, ×400)

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   Final Diagnosis Top


S. stercoralis hyperinfection with sepsis, multiorgan failure, adrenal hemorrhage, and SIADH in a known case of lepromatous leprosy.

Clinicopathologic correlation

Postmoretm findings of absent rigor mortis, hepatic steatosis and lipofuscin laden macrophages in lymph nodes and spleen are added pointers towards the patient's malnourished state. Severe malnourishment may have resulted from protein-losing enteropathy secondary to strongyloides hyperinfection with associated ulcerative amoebic enteritis. This may have further lead to acquired immunodeficiency.

Hypoglycemia as a terminal event can be explained by adrenal crisis following adrenal hemorrhage (Waterhouse–Fredericksen syndrome). What triggered the adrenal hemorrhage remains unanswered; it could have been sepsis or the strongyloides hyperinfection per se.

Hyponatremia in our patient can be attributed in part to dilutional hyponatremia due to fluid retention and also to SIADH as evidenced by low calculated plasma osmolality. SIADH as a presentation of strongyloides hyperinfection syndrome has been reported in the past.[5],[6],[7] In our patient, the low calculated plasma osmolality, normal potassium levels and absence of any significant lung pathology or central nervous system pathology are suggestive of SIADH having been contributory to hyponatremia.


   Brief Comment on Strongyloides Stercoralis Top


S. stercoralis hyperinfection is endemic in many tropical and subtropical areas, and usually associated with an immunocompromised state, seen to occur in human T-lymphotropic virus-1 and HIV infected, transplant recipients, malnourished individuals, and patients receiving corticosteroids.[8],[9] The true prevalence of strongyloidiasis in the Indian subcontinent is unknown as only case reports have been cited in the literature.[10],[11],[12]

The association of lepromatous leprosy and strongyloidiasis is extremely infrequent.[13],[14],[15] Hyperinfestation results due to the parasite's ability to persist and replicate within a host for years and internally autoinfecting the host without an obligatory external cycle [5],[8],[9] Of all the causes of hyperinfection syndrome, the strongest risk factor appears to be administered corticosteroids; with doses of oral prednisolone as low as 20 mg/day.[8],[12] Definitive diagnosis is based on detection of larvae in the stool or sputum samples. Serology is the investigation of choice for diagnosis and follow-up as stool microscopy has a low sensitivity due to the irregular shedding of the worm.[5],[8],[9] Prophylactic administration of ivermectin to all patients being exposed to steroids or immunosuppressive agents is recommended.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
   References Top

1.
Amarapurkar DN, Patel ND, Amarapurkar AD. Primary gastric tuberculosis –Report of 5 cases. BMC Gastroenterol 2003;3:6.  Back to cited text no. 1
    
2.
Ozaki M, Furuta M. Amyloidosis in leprosy. Int J Lepr Other Mycobact Dis 1975;43:116-24.  Back to cited text no. 2
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Gupta JC, Panda PK. Amyloidosis in leprosy. Lepr India 1980;52:260-6.  Back to cited text no. 3
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4.
Shen J, Liu M, Zhou M, Li W. Causes of death among active leprosy patients in China. Int J Dermatol 2011;50:57-60.  Back to cited text no. 4
    
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Hayashi E, Ohta N, Yamamoto H. Syndrome of inappropriate secretion of antidiuretic hormone associated with strongyloidiasis. Southeast Asian J Trop Med Public Health 2007;38:239-46.  Back to cited text no. 5
    
6.
Chowdhury DN, Dhadham GC, Shah A, Baddoura W. Syndrome of inappropriate antidiuretic hormone secretion (SIADH) in Strongyloides stercoralis hyperinfection. J Glob Infect Dis 2014;6:23-7.  Back to cited text no. 6
    
7.
Reddy TS, Myers JW. Syndrome of inappropriate secretion of antidiuretic hormone and nonpalpable purpura in a woman with Strongyloides stercoralis hyperinfection. Am J Med Sci 2003;325:288-91.  Back to cited text no. 7
    
8.
Greaves D, Coggle S, Pollard C, Aliyu SH, Moore EM. Strongyloides stercoralis infection. BMJ 2013;347:f4610.  Back to cited text no. 8
    
9.
Puthiyakunnon S, Boddu S, Li Y, Zhou X, Wang C, Li J, et al. Strongyloidiasis – An insight into its global prevalence and management. PLoS Negl Trop Dis 2014;8:e3018.  Back to cited text no. 9
    
10.
Tiwari S, Rautaraya B, Tripathy KP. Hyperinfection of Strongyloides stercoralis in an immunocompetent patient. Trop Parasitol 2012;2:135-7.  Back to cited text no. 10
[PUBMED]  Medknow Journal  
11.
Satyanarayana S, Nema S, Kalghatgi AT, Mehta SR, Rai R, Duggal R, et al. Disseminated Strongyloides stercoralis in AIDS: A report from India. Indian J Pathol Microbiol 2005;48:472-4.  Back to cited text no. 11
    
12.
Ghosh K, Ghosh K. Strongyloides stercoralis septicaemia following steroid therapy for eosinophilia: Report of three cases. Trans R Soc Trop Med Hyg 2007;101:1163-5.  Back to cited text no. 12
    
13.
De Souza JN, Machado PR, Teixeira MC, Soares NM. Recurrence of Strongyloides stercoralis infection in a patient with Hansen's disease: A case report. Lepr Rev 2014;85:58-62.  Back to cited text no. 13
[PUBMED]    
14.
Wambier CG, Lemos FB, Cappel MA, Bellissimo-Rodrigues F, Foss NT. Generalized serpiginous eruption during immunosuppressive treatment for leprosy reactive neuritis. PLoS Negl Trop Dis 2011;5:e1357.  Back to cited text no. 14
    
15.
Leang B, Lynen L, Tootill R, Griffiths S, Monchy D. Death caused by strongyloides hyperinfection in a leprosy patient on treatment for a type II leprosy reaction. Lepr Rev 2004;75:398-403.  Back to cited text no. 15
    

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Correspondence Address:
Dr. Deepti Mutreja
Department of Pathology, Command Hospital (Air Force), Bengaluru - 560 007, Karnataka
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0377-4929.168871

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    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7], [Figure 8], [Figure 9], [Figure 10], [Figure 11]
 
 
    Tables

  [Table 1], [Table 2], [Table 3]

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