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PHOTO ESSAY
Year : 2020  |  Volume : 32  |  Issue : 1  |  Page : 80-82

Allergic conjunctival granuloma with Splendore–Hoeppli phenomenon


Department of Orbit, Oculoplasty and Ocular Oncology, Giridhar Eye Institute, Kochi, Kerala, India

Date of Submission12-Dec-2019
Date of Acceptance13-Dec-2019
Date of Web Publication17-Apr-2020

Correspondence Address:
Dr. Marian Pauly
Department of Orbit, Oculoplasty and Ocular Oncology, Giridhar Eye Institute, Ponneth Temple Road, Kadavanthra, Kochi - 682 020, Kerala
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/kjo.kjo_88_19

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  Abstract 

Allergic conjunctival granuloma (ACG) or Splendore–Hoeppli phenomenon (SHP) (asteroid bodies) is characterized by microorganisms (fungi, bacteria, and parasites) or biologically inert substances surrounded by radiating intensely eosinophilic material thought to be due to the deposition of antigen–antibody complexes and debris from the host inflammatory cells. Noninfective pathologies (hypereosinophilic syndrome and ACGs) demonstrate this phenomenon occasionally. The purpose of this case report is to report a case of ACG with SHP in a 59-year-old female with a presumed noninfectious etiology which completely resolved with surgical excision.

Keywords: Allergic conjunctival granuloma, multinucleated giant cells, Splendore–Hoeppli


How to cite this article:
Ananthakrishna S, Pauly M. Allergic conjunctival granuloma with Splendore–Hoeppli phenomenon. Kerala J Ophthalmol 2020;32:80-2

How to cite this URL:
Ananthakrishna S, Pauly M. Allergic conjunctival granuloma with Splendore–Hoeppli phenomenon. Kerala J Ophthalmol [serial online] 2020 [cited 2020 Aug 12];32:80-2. Available from: http://www.kjophthal.com/text.asp?2020/32/1/80/282664




  Introduction Top


Splendore–Hoeppli phenomenon (SHP) is a rare but very distinct pathological entity characterized by the histological appearance of an amorphous, acellular eosinophilic material surrounded by epithelioid histiocytes, multinucleated giant cells, lymphocytes, and eosinophils. It has been reported to occur primarily in infectious processes. Hypereosinophilic syndrome and allergic conjunctival granulomas (ACGs) are noninfectious disorders that may also be associated with SHP.[1],[2] The SHP associated with the conjunctiva is rarely reported in the medical literature.


  Case Report Top


A 59-year-old female with a history of swelling in the right eye upper lid of 2 months duration, gradually progressing in size, associated with pain and redness. She was on treatment with an alternate form of medicine for the same initially for 1.5 months. Since the symptoms were persisting, she presented to us. She was a known case of diabetes mellitus of 12 years' duration and bronchial asthma of 15 years' duration on treatment. She gave a history of lid edema and protrusion of the right eye 2 years back. She was treated for the same as myositic form of pseudotumor with intravenous corticosteroid injections.

On examination, her best-corrected visual acuity was 20/20, N6 both eyes. The anterior segment examination revealed apparent ptosis of the right upper lid with obliteration of the upper lid crease. Ill-defined reddish elevated lesion in superior bulbar conjunctiva extending laterally and medially was noted, the posterior extent of which was not visible [Figure 1] and [Figure 2]. Extraocular movements were full and painless. Intraocular pressure was 14 mm Hg. Fundus was within normal limits. The left eye was within normal limits.
Figure 1: Apparent ptosis of the right upper eyelid with increase in upper eyelid fullness

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Figure 2: Ill-defined reddish elevated lesion in superior bulbar conjunctiva

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Computerized tomography of the orbit was done which did not reveal any evidence of a space-occupying lesion [Figure 3].
Figure 3: Computerized tomography of the orbit-No evidence of space occupying lesion

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An excisional biopsy of the lesion was planned. She underwent excisional biopsy under local anesthesia which revealed epithelioid histiocytes, Langerhans giant cells, lymphocytes, and a diffuse infiltration of eosinophils in subepithelial stroma of the conjunctiva which was suggestive of allergic granuloma of the conjunctiva with SHP [Figure 4].
Figure 4: Epithelioid histiocytes, langerhans giant cells, lymphocytes and a diffuse infiltration of eosinophils in subepithelial stroma of the conjunctiva (H and E, ×10)

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Peripheral smear was negative for filarial parasite. Her absolute eosinophil count and S. IgE were within normal limits.

The lesion completely disappeared and had no recurrence in the 6-month follow-up. She was symptom free in her last follow-up visit [Figure 5]a and [Figure 5]b.
Figure 5: (a) Patient at 6 months follow-up. (b) Patient at 6 months follow-up with complete resolution of conjunctival lesions

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  Discussion Top


SHP is characterized by an unusual histopathological pattern of amorphous eosinophilic material surrounded by granulomatous inflammation with an abundance of eosinophils. It has been postulated to be caused by both infectious (fungi, bacteria, and parasites) and noninfectious etiologies (hypereosinophilic syndrome and ACGs). This phenomenon was first discovered and published by Splendore in 1908.[3] Splendore initially observed the phenomenon surrounding fungal spores. This observation was later validated by Hoeppli in 1932, who noticed a similar fringe of eosinophilic material surrounding schistosoma eggs.[3]

The exact causation is unknown.[4],[5],[6] Investigations have shown that the amorphous, acellular material contains high levels of antigen–antibody complexes, namely IgM, IgG, and complement C3[4],[5],[6],[7] and a high concentration of eosinophil major basic protein.[8]

Ashton and Cook[4] noted that histologically, the sharply demarcated granuloma consisted of granular eosinophilic centers containing nuclear debris encircled by epithelioid cells along with giant cells and even more peripherally by an inflammatory infiltrate dominated by eosinophils. These observations are common to most reported cases, and the findings in our case are similar.[1],[2],[3],[4],[5],[6]

There is no established treatment for the SHP. The use of topical or systemic corticosteroids is controversial because this was found to be of no benefit in many patients, as demonstrated in a patient in whom the SHP developed under high-dose oral steroids.[9] There have been reports of a successful outcome after the use of topical cyclosporine A for 18 months[10] and tacrolimus eye drops.[11] Few studies have shown that excisional biopsy of the granuloma is the therapy of choice[12] which has been supported by our experience.

Our experience with this patient concludes that ACG/SHP should be considered as one of the differential diagnoses in patients with superior conjunctival nodules, especially in patients with allergic or hypersensitivity predisposition. Our case report adds to the noninfectious causes of SHP.

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.
Hussein MR. Mucocutaneous Splendore-Hoeppli phenomenon. J Cutan Pathol 2008;35:979-88.  Back to cited text no. 1
    
2.
Read RW, Zhang J, Albini T, Evans M, Rao NA. Splendore-Hoeppli phenomenon in the conjunctiva: Immunohistochemical analysis. Am J Ophthalmol 2005;140:262-6.  Back to cited text no. 2
    
3.
Hoeppli R. Histological observations in experimental schistosomiasis Japonica. Chin Med J 1932;46:1179-86.  Back to cited text no. 3
    
4.
Ashton N, Cook C. Allergic granulomatous nodules of the eyelid and conjunctiva. The XXXV Edward Jackson Memorial Lecture. Am J Ophthalmol 1979;87:1-28.  Back to cited text no. 4
    
5.
Lawson JM, Dart JK, McCartney AC. Conjunctival nodules associated with the Splendore-Hoeppli phenomenon. Arch Ophthalmol 1991;109:426.  Back to cited text no. 5
    
6.
Font RL, Hunts JH. Botryomycosis of limbal conjunctiva with Splendore-Hoeppli phenomenon. Arch Ophthalmol 1996;114:1006-8.  Back to cited text no. 6
    
7.
von Lichtenberg F, Smith JH, Cheever AW. The Hoeppli phenomenon in schistosomiasis. Comparative pathology and immunopathology. Am J Trop Med Hyg 1966;15:886-95.  Back to cited text no. 7
    
8.
Kephart GM, Andrade ZA, Gleich GJ. Localization of eosinophil major basic protein onto eggs of Schistosoma mansoni in human pathologic tissue. Am J Pathol 1988;133:389-96.  Back to cited text no. 8
    
9.
Pinto N, Hirst L, Whitby M. Conjunctival and episcleral Splendore-Hoeppli phenomenon. Clin Exp Ophthalmol 2008;36:865-7.  Back to cited text no. 9
    
10.
McGrath LA, Whitehead K, Lee GA. Topical ophthalmic use of cyclosporin A for Splendore-Hoeppli phenomenon. Clin Exp Optom 2014;97:184-6.  Back to cited text no. 10
    
11.
Foroutan A, Ghafourian A, Habibi A, Foroutan P, Ashrafkhorasani M, Alemzadeh SA. Successful treatment of allergic conjunctival granuloma by topical tacrolimus: A clinicopathologic case report. Indian J Ophthalmol 2018;66:578-80.  Back to cited text no. 11
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12.
Völcker HE, Enke P, Naumann GO. Eosinophilic, central necrotizing granuloma of the conjunctiva (Splendore-Hoeppli phenomenon). Clinical-histopathological study. Klin Monbl Augenheilkd 1985;186:254-8.  Back to cited text no. 12
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]



 

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