Orbit, 2015; 34(4): 232–233 ! Informa Healthcare USA, Inc. ISSN: 0167-6830 print / 1744-5108 online DOI: 10.3109/01676830.2014.950297
C ASE REPORT
Caruncular Leishmaniasis—An Unusual Case Badhu Badri Prasad1, Arjana Shakya1, Lavaju Poonam1, and Karki Smriti2
Orbit Downloaded from informahealthcare.com by University New South Wales on 08/17/15 For personal use only.
1
Department of Ophthalmology and 2Department of Pathology, Bisheshwar Prasad Koirala Institute of Health Sciences, Dharan, Nepal
ABSTRACT Case: A 50-year-old patient presented with a left eye caruncular mass of 5-year duration. Examination revealed a fleshy mass originating from the left nasal upper and the lower lid, involving the nasal conjunctiva and the whole of the cornea. The mass was excised and histopathological examination revealed the presence of LD bodies, the hallmark feature of leishmaniasis. Conclusion: Ocular leishmaniasis is a potentially blinding disease and delay in diagnosis and treatment can cause irreversible damage to the eye and adnexae. Due to the uncommon occurrence of this disease, its diagnosis and treatment remains a challenge. Keywords: Caruncular leishmaniasis, caruncular mass, LD bodies
INTRODUCTION
The patient also complained of abdominal discomfort and distension for 2 months which was gradually progressive in nature. The patient had a history of Kala azar 10 years prior to presenting at the outpatient department for which he was given intravenous injections for a month in a regional hospital in India. On systemic examination, there was enlargement of the right central axillary lymph node. Abdominal examination revealed distension of the abdomen, hepatomegaly (liver palpable 2 cm below the right subcostal margin) with shifting dullness. On ocular examination, the right eye was normal. The visual acuity in the left eye was no perception of light, most probably due to secondary glaucoma. There was a fleshy caruncular mass encroaching on the medial two-thirds of the cornea and extending superiorly and inferiorly into the globe. The anterior chamber, as visible from the temporal one-third of the cornea, was of normal depth and quiet. The visible iris was normal and only the temporal margin of the pupil could be seen. Inner details could not be visualised. Ultrasonography (B-scan) of the left eye showed normal scan of the vitreous (the image of the scan could not be included as the patient lost the reports on his way back home) and the retina showed an
Ocular leishmaniasis (OL), a rare condition, is caused by different leishmania species. The predominant etiologic agent is L. donovani in southern Asia and eastern Africa.1 Clinical manifestations of OL include lid and skin ulcers, blepharitis, conjunctivitis, cataract, interstitial keratitis, anterior uveitis, glaucoma, and consequential loss of vision. OL, can simulate other conditions such as chalazion, lid or conjunctival tumors, dacryocystitis or non-specific uveitis.2,3 Here, we report a histologically diagnosed case of orbital leishmaniasis.
CASE REPORT A 50-year-old farmer from Bahadurgunj, Bihar, presented at the outpatient department of ophthalmology at BP Oirala Institute of earth science, with a caruncular mass in his left eye for 5 years. The mass was initially pea-sized, slowly progressive in nature and was not associated with pain nor bleeding. As the mass increased in size the patient experienced a gradual progressive diminution of vision.
Received 3 March 2014; Accepted 28 July 2014; Published online 2 June 2015 Correspondence: Arjana Shakya, Himalaya Apartment Hotel, Pulchowk-3, Lalitpur, P.O. Box 8975, Nepal. E-mail: [email protected]; [email protected]
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Carnucular Leishmaniasis enhancing soft tissue density lesion approximately 20 11 mm in size in the medial canthus of left orbit involving the medial rectus muscle indenting and abutting the globe. Preseptal swelling was also found. Five days after the presentation the patient underwent excisional biopsy of the left eye mass via an eye lid splitting incision. On examination the dermis of the mass showed dense inflammatory cell infiltrates, which were predominantly plasma cells intermingled with numerous extracellular histiocytes-laden LD bodies with few lymphocytes (Figure 1). On the basis of the histopathological reports, the diagnosis of orbital leishmaniasis was made and the patient was given oral miltefostine 50 mg BD (anti leishmaniasis drug) for 6 weeks. Ascites tapping, bone marrow aspiration and splenic aspiration were performed and subjected to laboratory and histopathological examination. None of these investigations showed features suggestive of leishmaniasis. The biopsy from the right central axillary lymph node showed reactive lymphadenitis. After the administration of oral miltefostine, the patient showed general improvement and was discharged. Three years later the patient came for
233
follow-up, complaining of a recurrence of growth in the left eye (Figure 2).
DISCUSSION Ocular leishmaniasis is an ocular protozoal infection which is widespread in the world but is more common in developing countries.3 Due to the rarity of the disease and a varied presentation, ocular leishmaniasis poses a diagnostic and therapeutic challenge to the treating physician. In our case, the patients gave a past history of systemic leishmaniasis and a treatment history with an intravenous drug most likely to be a pentavalent antimonial for a month. Furthermore, the ocular mass biopsy showed the presence of LD bodies, leading us to the diagnosis of ocular leishmaniasis presenting as a caruncular mass, which later progressed to involve the conjunctiva and also encroached on the cornea, diminishing the patient’s vision. The presence of past history of systemic leishmaniasis and the histopathological examination of the ocular mass, led us to the diagnosis of ocular leishmaniasis. The hepatomegaly and radiologically seen enlargement of the spleen in this patient could be due to systemic leishmaniasis. Ascitis can also occur in systemic leishmaniaisis due to the presence of hypoalbuminenia or due to portal hypertension. There was absence of evidence supporting Leishmaniasis in the splenic biopsy and the bone marrow aspiration. This could have resulted from the prior administration of anti-leishmanial drug milefostine.
ACKNOWLEDGMENTS We acknowledge the faculty members, especially Professor Suman Rijal and the staff of the Tropical Medicine Unit of the Bisheshwar Prasad Koirala Institute of Health Sciences for systemic evaluation and treatment of the case. FIGURE 1. Histopathological photograph showing LD bodies stained in hematoxylin and eosin stain, magnification 4.
DECLARATION OF INTEREST The authors report no conflicts of interest. The authors alone are responsible for the content and writing of the paper.
REFERENCES
FIGURE 2. Photograph showing recurrence of mass in the left eye, 3 years after the initial excision biopsy. !
2015 Informa Healthcare USA, Inc.
1. Herwaldt BL. Leishmaniasis. In: Fauci AS, Kasper DL, Longo DL, Braunwald E, Hauser SL, Jameson JL, and Loscalzo J. editors. Harrison’s Principles of Internal Medicine. 17th Edition. New York: McGraw-Hill; 2008; p 1296. 2. Kumar PV, Roozilab MH, Lak P, Sadeghi E. Ocular leishmaniasis: a cause of blindness. Iranian Journal of medical science 1993;18:106–111. 3. ModarresZadeh M, Manshai K, Shaddel M, Oormazdi H. Ocular leishmaniasis. Iran J Ophthalmol 2006;19:1–5.
C ASE REPORT
Caruncular Leishmaniasis—An Unusual Case Badhu Badri Prasad1, Arjana Shakya1, Lavaju Poonam1, and Karki Smriti2
Orbit Downloaded from informahealthcare.com by University New South Wales on 08/17/15 For personal use only.
1
Department of Ophthalmology and 2Department of Pathology, Bisheshwar Prasad Koirala Institute of Health Sciences, Dharan, Nepal
ABSTRACT Case: A 50-year-old patient presented with a left eye caruncular mass of 5-year duration. Examination revealed a fleshy mass originating from the left nasal upper and the lower lid, involving the nasal conjunctiva and the whole of the cornea. The mass was excised and histopathological examination revealed the presence of LD bodies, the hallmark feature of leishmaniasis. Conclusion: Ocular leishmaniasis is a potentially blinding disease and delay in diagnosis and treatment can cause irreversible damage to the eye and adnexae. Due to the uncommon occurrence of this disease, its diagnosis and treatment remains a challenge. Keywords: Caruncular leishmaniasis, caruncular mass, LD bodies
INTRODUCTION
The patient also complained of abdominal discomfort and distension for 2 months which was gradually progressive in nature. The patient had a history of Kala azar 10 years prior to presenting at the outpatient department for which he was given intravenous injections for a month in a regional hospital in India. On systemic examination, there was enlargement of the right central axillary lymph node. Abdominal examination revealed distension of the abdomen, hepatomegaly (liver palpable 2 cm below the right subcostal margin) with shifting dullness. On ocular examination, the right eye was normal. The visual acuity in the left eye was no perception of light, most probably due to secondary glaucoma. There was a fleshy caruncular mass encroaching on the medial two-thirds of the cornea and extending superiorly and inferiorly into the globe. The anterior chamber, as visible from the temporal one-third of the cornea, was of normal depth and quiet. The visible iris was normal and only the temporal margin of the pupil could be seen. Inner details could not be visualised. Ultrasonography (B-scan) of the left eye showed normal scan of the vitreous (the image of the scan could not be included as the patient lost the reports on his way back home) and the retina showed an
Ocular leishmaniasis (OL), a rare condition, is caused by different leishmania species. The predominant etiologic agent is L. donovani in southern Asia and eastern Africa.1 Clinical manifestations of OL include lid and skin ulcers, blepharitis, conjunctivitis, cataract, interstitial keratitis, anterior uveitis, glaucoma, and consequential loss of vision. OL, can simulate other conditions such as chalazion, lid or conjunctival tumors, dacryocystitis or non-specific uveitis.2,3 Here, we report a histologically diagnosed case of orbital leishmaniasis.
CASE REPORT A 50-year-old farmer from Bahadurgunj, Bihar, presented at the outpatient department of ophthalmology at BP Oirala Institute of earth science, with a caruncular mass in his left eye for 5 years. The mass was initially pea-sized, slowly progressive in nature and was not associated with pain nor bleeding. As the mass increased in size the patient experienced a gradual progressive diminution of vision.
Received 3 March 2014; Accepted 28 July 2014; Published online 2 June 2015 Correspondence: Arjana Shakya, Himalaya Apartment Hotel, Pulchowk-3, Lalitpur, P.O. Box 8975, Nepal. E-mail: [email protected]; [email protected]
232
Orbit Downloaded from informahealthcare.com by University New South Wales on 08/17/15 For personal use only.
Carnucular Leishmaniasis enhancing soft tissue density lesion approximately 20 11 mm in size in the medial canthus of left orbit involving the medial rectus muscle indenting and abutting the globe. Preseptal swelling was also found. Five days after the presentation the patient underwent excisional biopsy of the left eye mass via an eye lid splitting incision. On examination the dermis of the mass showed dense inflammatory cell infiltrates, which were predominantly plasma cells intermingled with numerous extracellular histiocytes-laden LD bodies with few lymphocytes (Figure 1). On the basis of the histopathological reports, the diagnosis of orbital leishmaniasis was made and the patient was given oral miltefostine 50 mg BD (anti leishmaniasis drug) for 6 weeks. Ascites tapping, bone marrow aspiration and splenic aspiration were performed and subjected to laboratory and histopathological examination. None of these investigations showed features suggestive of leishmaniasis. The biopsy from the right central axillary lymph node showed reactive lymphadenitis. After the administration of oral miltefostine, the patient showed general improvement and was discharged. Three years later the patient came for
233
follow-up, complaining of a recurrence of growth in the left eye (Figure 2).
DISCUSSION Ocular leishmaniasis is an ocular protozoal infection which is widespread in the world but is more common in developing countries.3 Due to the rarity of the disease and a varied presentation, ocular leishmaniasis poses a diagnostic and therapeutic challenge to the treating physician. In our case, the patients gave a past history of systemic leishmaniasis and a treatment history with an intravenous drug most likely to be a pentavalent antimonial for a month. Furthermore, the ocular mass biopsy showed the presence of LD bodies, leading us to the diagnosis of ocular leishmaniasis presenting as a caruncular mass, which later progressed to involve the conjunctiva and also encroached on the cornea, diminishing the patient’s vision. The presence of past history of systemic leishmaniasis and the histopathological examination of the ocular mass, led us to the diagnosis of ocular leishmaniasis. The hepatomegaly and radiologically seen enlargement of the spleen in this patient could be due to systemic leishmaniasis. Ascitis can also occur in systemic leishmaniaisis due to the presence of hypoalbuminenia or due to portal hypertension. There was absence of evidence supporting Leishmaniasis in the splenic biopsy and the bone marrow aspiration. This could have resulted from the prior administration of anti-leishmanial drug milefostine.
ACKNOWLEDGMENTS We acknowledge the faculty members, especially Professor Suman Rijal and the staff of the Tropical Medicine Unit of the Bisheshwar Prasad Koirala Institute of Health Sciences for systemic evaluation and treatment of the case. FIGURE 1. Histopathological photograph showing LD bodies stained in hematoxylin and eosin stain, magnification 4.
DECLARATION OF INTEREST The authors report no conflicts of interest. The authors alone are responsible for the content and writing of the paper.
REFERENCES
FIGURE 2. Photograph showing recurrence of mass in the left eye, 3 years after the initial excision biopsy. !
2015 Informa Healthcare USA, Inc.
1. Herwaldt BL. Leishmaniasis. In: Fauci AS, Kasper DL, Longo DL, Braunwald E, Hauser SL, Jameson JL, and Loscalzo J. editors. Harrison’s Principles of Internal Medicine. 17th Edition. New York: McGraw-Hill; 2008; p 1296. 2. Kumar PV, Roozilab MH, Lak P, Sadeghi E. Ocular leishmaniasis: a cause of blindness. Iranian Journal of medical science 1993;18:106–111. 3. ModarresZadeh M, Manshai K, Shaddel M, Oormazdi H. Ocular leishmaniasis. Iran J Ophthalmol 2006;19:1–5.
