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CASE REPORT Table of Contents  
Ahead of print publication
Severe pulmonary-renal syndrome in honeybee sting envenomation – A case report


 Department of Internal Medicine, Postgraduate Institute of Medical Education and Research, Chandigarh, India

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Date of Submission01-May-2022
Date of Decision03-Jun-2022
Date of Acceptance05-Jul-2022
Date of Web Publication28-Sep-2022
 

  Abstract 


Honeybee stings with features of envenomation (either local or allergic and rarely systemic toxicity) are often seen in agriculture workers. An initial presentation with severe diffuse alveolar hemorrhage or pulmonary-renal syndrome is rare and only confined to a few case reports. Herein, we describe a case of a 45-year-old male who presented with multiple bee stings and subsequently developed acute kidney injury and pulmonary hemorrhage. He was managed with hemodialysis, invasive mechanical ventilation, red-cell transfusion, and pulse methylprednisolone. However, he developed cardiac arrhythmias with ventricular tachycardia and died.

Keywords: Bee sting, case report, diffuse alveolar hemorrhage, envenomation, honeybee, pulmonary-renal syndrome, renal failure


How to cite this URL:
Shaji A, Parvez M, Chirumamilla NK, Sharma N, Pannu AK. Severe pulmonary-renal syndrome in honeybee sting envenomation – A case report. Turk J Emerg Med [Epub ahead of print] [cited 2022 Dec 2]. Available from: https://www.turkjemergmed.org/preprintarticle.asp?id=357332





  Introduction Top


Bee sting and subsequent hypersensitivity reactions constitute frequent emergency department visits globally. Severe adverse events or life-threatening organ failure may occur, especially after multiple stings.[1] Renal dysfunction is not uncommon and usually has a good prognosis.[2] Diffuse alveolar hemorrhage (DAH) is rarely described with bee sting and is limited to only case reports.[3],[4],[5] Management is primarily intensive supportive care. Corticosteroid therapy has been tried. Herein, we describe a case of multiple bee stings complicated by renal dysfunction, pulmonary hemorrhage, and cardiac arrhythmias.


  Case Report Top


A 45-year-old male, farmer by occupation, had multiple bee stings while working on his farm. He applied homemade herbal preparations over the bite sites. Two days later, he started having a decreased urine output and facial puffiness. He had one episode of hemoptysis and presented to our institute's emergency department. He did not have other bleeding tendencies. There was no history of fever, dysuria, myalgia, or arthralgia. His past medical history was unremarkable, without any comorbidities. On physical examination, the pulse was 110 beats per min, blood pressure was 136 / 90 mm Hg, respiratory rate was 26 breaths per minute, and the oxygen saturation was 92% while the patient was breathing high-flow oxygen through a venturi mask. He had multiple sting marks on his arms, face, and trunk with surrounding erythema [Figure 1]. Pallor, periorbital puffiness, and edema over both shins were noted. Lung auscultation revealed inspiratory crackles diffusely scattered over both lung fields.
Figure 1: Bee sting marks on patient's back

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Laboratory evaluation showed anemia with a hemoglobin drop from 10.2 g/dL to 7.5 g/dL within 24 h, consistent with hemoptysis. His total leukocyte count was 13 × 10[9]/L and platelet count was 178 × 10[9]/L at presentation and did not show any falling trend. The coagulation profile was normal with an international normalized ratio of 1.1. He had acute kidney injury (AKI) with blood urea of 434.0 mg/dL, creatinine of 16.0 mg/dL, and metabolic acidosis (blood pH, 7.2; bicarbonate, 9 mmol/L). Serum bilirubin, liver enzymes, and creatinine kinase were grossly normal. Urine routine analysis showed albuminuria (2+ on dipstick) and no red cell or cast. A 24-h urine examination detected 1.4 g of protein. A chest X-ray revealed perihilar infiltrates in both lungs with peripheral sparing [Figure 2]. Hemodialysis was started for AKI; however, the dyspnea and oxygen saturation did not improve. The hemoglobin further decreased to 6 g/dL. A thoracic computed tomography revealed diffuse alveolar infiltrates sparing periphery in both lungs [Figure 3].
Figure 2: Chest X-ray showing perihilar infiltrates in both lungs with peripheral sparing

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Figure 3: Thoracic computed tomography showing diffuse alveolar infiltrates sparing periphery in both lungs

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The patient had systemic envenomation with AKI. His shortness of breath was initially attributed to fluid overload secondary to AKI. However, due to hemoptysis, progressive hemoglobin drop, and no improvement after multiple hemodialysis sessions, a possibility of DAH was kept. Chest imaging was also consistent with DAH. Testing for common etiologies for the pulmonary-renal syndrome, including antineutrophil cytoplasmic antibodies, antinuclear antibodies, complements (C3 and C4) levels, double-stranded DNA antibodies, and anti–glomerular basement membrane antibodies was noncontributory. Serologies for the human immunodeficiency virus, hepatitis B, and hepatitis C were negative. Workup for hemolysis (i.e. plasma hemoglobin, urine hemoglobin, G6PD enzyme deficiency, and schistocytes on the peripheral blood smear) was negative. After ruling out common etiologies for the immune-mediated pulmonary-renal syndrome, bee sting envenomation-related DAH was diagnosed.

For envenomation-related DAH, the patient was treated with pulse methylprednisolone pulse 1 g/day for 5 days. Hemodialysis was given along with packed red cell transfusions. For progressive respiratory failure and DAH, he required invasive mechanical ventilation. Topical mupirocin was applied over the skin lesions, and intravenous piperacillin-tazobactam was started. However, he developed ventricular tachyarrhythmia on Day 7 of hospitalization and died.

Written informed consent is present. The patient's brother was explained about the confidentiality, and the case information will be used for education purposes only.


  Discussion Top


Hymenoptera insects are characterized by having two pairs of wings. Within the hymenoptera order, the three families of stinging insects are the Apidae (honeybees and bumblebees), Vespidae (hornets, yellowjackets, and wasps), and Formicidae (ants). The honeybee and the bumblebee are the only two bees of medical importance. Bee sting envenomation can range from mild-to-severe clinical features and depends on the number of stings, atopy, age, and associated medical comorbidities.[1],[6] Most patients have local manifestations, which are usually self-limiting. Allergic features such as pruritus, angioedema, and diarrhea and anaphylactic reactions such as bronchoconstriction and shock are attributed to biogenic amines in the venom and result from immunoglobulin E-mediated hypersensitivity.[1] Systemic features include vomiting, diarrhea, myalgia, hemolysis, rhabdomyolysis, AKI, hypotension, vasculitis, and rarely, pulmonary hemorrhage.[7] Systemic envenomation is thought to be mediated by hyaluronidase in the venom, resulting in the cleavage of hyaluronic acid from the extracellular matrix.[6]

The pathogenesis of AKI is usually multifactorial. It includes ischemic acute tubular necrosis secondary to hypotension, toxin-induced direct tubular injury, pigment nephropathy due to hemolysis or rhabdomyolysis, vasculitis, glomerulonephritis, and thrombotic microangiopathy.[8],[9],[10] In the index patient, the AKI was likely due to tubular injury with or without glomerular involvement, as evident by the nonnephrotic range proteinuria; however, the histopathological examination could not be performed.

Bee sting-related DAH is rare. The proposed mechanisms include hypersensitivity to the bee venom and direct toxicity to alveolar capillaries manifesting as alveolocapillaritis or as a part of systemic vasculitis.[3],[4],[5] The onset is usually abrupt, i.e. within a week of the bee sting. Clinical presentation is similar to other autoimmune causes, such as cough, breathlessness, fever, and hemoptysis.[11] Chest imaging reveals diffuse bilateral alveolar opacities with peripheral sparing. Because fluid overload may mimic the clinic-radiological picture, the presence of associated advanced renal failure may delay the diagnosis. Management is usually supportive with ventilation and blood transfusion. Because prospective data are lacking, the role of steroids is limited to the case reports. A therapeutic benefit of plasma exchange is not described. Cardiac manifestations include myocardial ischemia, myocarditis, and arrhythmias (including conduction blocks and tachyarrhythmia).[12] These manifestations can occur up to a week after the sting, thus requiring vigilance, appropriate treatment, patient education, and follow-up. Unlike ours, the patients with systemic envenomation who present within 24–48 h of bee sting had favorable outcomes.[3],[4],[5]


  Conclusion Top


Life-threatening adverse events after bee sting envenomation are infrequent; however, it is imperative to recognize severe features early, particularly if multiple stings are noted. DAH is rarely described as a bee sting. Management is primarily intensive supportive care and a delay in treatment results in a fatal outcome. Education of at-risk populations and prompt medical care are essential. Further studies on the role of steroids, immune modulators, and potential antivenom therapy are mandated.

Author contributions

1AS: collected patient data, drafted the manuscript

2MMP, 3NKC, 4NS: collected patient data

5AKP: collected patient data, drafted and revised the manuscript

The corresponding author is responsible for ensuring that the descriptions are accurate and agreed by all authors

Conflicts of interest

None declared.

Patient's consent

Written informed consent is present. The patient's brother was explained about the confidentiality, and the case information will be used for education purposes only.

Declaration of patient consent

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

Funding

None.



 
  References Top

1.
Pucca MB, Cerni FA, Oliveira IS, Jenkins TP, Argemí L, Sørensen CV, et al. Bee updated: Current knowledge on bee venom and bee envenoming therapy. Front Immunol 2019;10:2090.  Back to cited text no. 1
    
2.
Nair BT, Sanjeev RK, Saurabh K. Acute kidney injury following multiple bee stings. Ann Afr Med 2016;15:41-2.  Back to cited text no. 2
[PUBMED]  [Full text]  
3.
Fatma U, Metin A, Ozkan D. Anaphylaxis and diffuse alveolar haemorrhage following bee sting. Respir Case Rep 2013;2:10-3.  Back to cited text no. 3
    
4.
Mukhopadhyay A, Fong KF, Lim TK. Diffuse alveolar haemorrhage: A rare reaction to insect sting. Respirology 2002;7:157-9.  Back to cited text no. 4
    
5.
Lam SM. Acute pulmonary hemorrhage following a honeybee sting: A case report. J Microbiol Immunol Infect 1998;31:133-6.  Back to cited text no. 5
    
6.
Elieh Ali Komi D, Shafaghat F, Zwiener RD. Immunology of bee venom. Clin Rev Allergy Immunol 2018;54:386-96.  Back to cited text no. 6
    
7.
Hossen MS, Shapla UM, Gan SH, Khalil MI. Impact of bee venom enzymes on diseases and immune responses. Molecules 2016;22:25.  Back to cited text no. 7
    
8.
Silva GB Jr., Vasconcelos AG Jr., Rocha AM, Vasconcelos VR, Neto BJ, Fujishima JS, et al. Acute kidney injury complicating bee stings – A review. Rev Inst Med Trop Sao Paulo 2017;59:e25.  Back to cited text no. 8
    
9.
Grisotto LS, Mendes GE, Castro I, Baptista MA, Alves VA, Yu L, et al. Mechanisms of bee venom-induced acute renal failure. Toxicon 2006;48:44-54.  Back to cited text no. 9
    
10.
Ashley JR, Otero H, Aboulafia DM. Bee envenomation: A rare cause of thrombotic thrombocytopenic purpura. South Med J 2003;96:588-91.  Back to cited text no. 10
    
11.
Lally L, Spiera RF. Pulmonary vasculitis. Rheum Dis Clin North Am 2015;41:315-31.  Back to cited text no. 11
    
12.
Franco-Lugo D, Uribe-Vallarta DA, Bernal-Contreras KD, Vargas-Abonce VP, Soto-Pérez-De-Celis E, Rivera-Figueroa J, et al. Idioventricular rhythm and shock after a honeybee sting. Arch Cardiol Mex 2021;91:186-9.  Back to cited text no. 12
    

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Correspondence Address:
Ashok Kumar Pannu,
4th Floor, F Block, Nehru Hospital, Postgraduate Institute of Medical Education and Research, Chandigarh - 160 012
India
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/2452-2473.357332



    Figures

  [Figure 1], [Figure 2], [Figure 3]



 

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