Unilateral Cauliflower Ear due to Leprosy or Trauma - ijl K Shilpa IInd et al (189-192).pdf · 190...

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The terminology Cauliflower ear refers to the appearance of external ear, sequelae to underlying primary inflammation/infection. Trauma and secondary infection or perichondritis being the most common cause of cauliflower ear, this article describes a case of unilateral cauliflower ear due to multibacillary leprosy. This case shows the importance of skin smears and the histopathological identification and classification of leprosy patients with unusual lesions. Keywords : Cauliflower ear, multibacillary (MB) leprosy, acid fast bacilli (AFB) http://www.ijl.org.in Case Report Received : Accepted : 10.03.2016 15.09.2016 Indian J Lepr 2016, 88 : 189-192 © Hind Kusht Nivaran Sangh, New Delhi 1 Dr K Shilpa, MBBS, MD(DVL), Assistant Professor 2 Dr B Leelavathy, MBBS, MD(DVL), Professor and Head 3 Dr DV Lakshmi, DVD, Junior Resident 4 Dr G Divya, MBBS, MD(DVL), Senior Resident Department of Dermatology, Venereology, Leprology, Bangalore Medical College and Research Institute, Bangalore. KR Market, Bengaluru-560002, Karnataka, India Corresponding author : Dr K Shilpa. e-mail: [email protected] Unilateral Cauliflower Ear due to Leprosy or Trauma - A Diagnostic Challenge 1 2 3 4 K Shilpa , B Leelavathy , DV Lakshmi , G Divya Introduction Cauliflower ear represents an end result of oto- hematoma caused by trauma to the auricle (pinna of the ear). It is commonly observed in wrestlers (el Bestar 2004). It is seen less commonly now, due to adequate head protection worn now, especially in contact sports. New cases of cauli- flower secondary to perichondritis are, however, being reported due to multiple and high trans- catilagenous ear piercing (Hanif et al 2001). The terminology “cauliflower ear” refers to the appearance of external ear, resulting due to underlying primary inflammation/infection to the external ear due totrauma and secondary infection, resulting in perichondritis. This is the most common cause of cauliflower ear. This article describes a case of unilateral cauliflower ear due to multibacillary leprosy. Case report A 50 year old male patient presented with asymptomatic swelling of right ear since last one year. It was spontaneous in onset and gradually increasing since then. There was no history of trauma, ear piercing, pain, fever, joint pain, skin lesion anywhere else in the body. The swelling of the auricle of external ear was suspected as perichondritis by the ENT surgeon and prescribed antibiotics (ciprofloxacilin) and oral steroids

Transcript of Unilateral Cauliflower Ear due to Leprosy or Trauma - ijl K Shilpa IInd et al (189-192).pdf · 190...

The terminology Cauliflower ear refers to the appearance of external ear, sequelae to underlying primary

inflammation/infection. Trauma and secondary infection or perichondritis being the most common cause of

cauliflower ear, this article describes a case of unilateral cauliflower ear due to multibacillary leprosy. This case

shows the importance of skin smears and the histopathological identification and classification of leprosy

patients with unusual lesions.

Keywords : Cauliflower ear, multibacillary (MB) leprosy, acid fast bacilli (AFB)

http://www.ijl.org.in

Case Report

Received : Accepted : 10.03.2016 15.09.2016

Indian J Lepr 2016, 88 : 189-192© Hind Kusht Nivaran Sangh, New Delhi

1 Dr K Shilpa, MBBS, MD(DVL), Assistant Professor2 Dr B Leelavathy, MBBS, MD(DVL), Professor and Head3 Dr DV Lakshmi, DVD, Junior Resident4 Dr G Divya, MBBS, MD(DVL), Senior Resident

Department of Dermatology, Venereology, Leprology, Bangalore Medical College and Research Institute, Bangalore.

KR Market, Bengaluru-560002, Karnataka, India

Corresponding author : Dr K Shilpa. e-mail: [email protected]

Unilateral Cauliflower Ear due to Leprosy or Trauma -A Diagnostic Challenge

1 2 3 4K Shilpa , B Leelavathy , DV Lakshmi , G Divya

Introduction

Cauliflower ear represents an end result of oto-

hematoma caused by trauma to the auricle (pinna

of the ear). It is commonly observed in wrestlers

(el Bestar 2004). It is seen less commonly now,

due to adequate head protection worn now,

especially in contact sports. New cases of cauli-

flower secondary to perichondritis are, however,

being reported due to multiple and high trans-

catilagenous ear piercing (Hanif et al 2001).

The terminology “cauliflower ear” refers to the

appearance of external ear, resulting due to

underlying primary inflammation/infection to

the external ear due totrauma and secondary

infection, resulting in perichondritis. This is the

most common cause of cauliflower ear. This

article describes a case of unilateral cauliflower

ear due to multibacillary leprosy.

Case report

A 50 year old male patient presented with

asymptomatic swelling of right ear since last one

year. It was spontaneous in onset and gradually

increasing since then. There was no history of

trauma, ear piercing, pain, fever, joint pain, skin

lesion anywhere else in the body. The swelling of

the auricle of external ear was suspected as

perichondritis by the ENT surgeon and prescribed

antibiotics (ciprofloxacilin) and oral steroids

Shilpa et al190

(prednisolone). However, there was no improve-

ment in the lesions, with the medications. An

incisional biopsy was done for histopathological

diagnosis and confirmation and the patient ref

erred for dermatologic consultation. Histopatho-

logy showed foci of mononuclear inflammatory

cell infiltrate in nerve bundle, perivascular and

periappendageal areas and a few foamy macro-

phages with the diagnosis of Hansen's disease.

On physical examination, there was diffuse

thickening of scaphoid and navicular fossa with

well defined, discrete papules over the helix of

right ear simulating a cauliflower appearance

(Fig 1). There was no tenderness of external earor

regional lymphadenopathy. On examination of

the peripheral nerves, there was thickening of

greater auricular nerve on the right side. No other

nerve thickening or involvement was observed

Fig 1 : Involvement of navicular and scaphoid

fossa of right external ear with relative sparing

of lobule with complete normal right ear.

Fig 2a : Lymphocytic infiltration in the dermis

(Blue arrow) with Langerhans giant cell

(red arrow) forming a well defined granuloma

Fig 2b : Lymphocytic inflammatory infiltrate

(Blue arrow) around the nerve bundle

(red arrow);

Fig 2c : Lymphocytic inflammatory infiltrate

(Blue arrow) around the adnexa – sebaceous

gland (red arrow) (H&E 10X)

Unilateral Cauliflower Eardue to Leprosy or Trauma - A Diagnostic Challenge 191

and there was no sensory or motor impairment

anywhere else in the body. Slit skin smear from

the affected ear showed acid fast bacilli with BI of

4+ on ZN staining under light microscopy.

Histopathological reports were reviewed with

Fite Faraco staining. Well defined lymphocytic

granulomas (Fig 2a), were observed with

perinueural (Fig 2b) and peri adnexal (Fig 2c)

inflammatory infiltrate. A special stain (Fite

Faraco) showed acid fast bacilli arranged in the

form of globi (Fig 3).

Based on these findings, a diagnosis of multi-

bacillary leprosy was made and patient was

started on multibacillary multidrug therapy, to

which he gradually responded.

Discussion

Leprosy is a chronic granulomatous infection

caused by Mycobacterium leprae which mainly

affects the skin and peripheral nerves. It has a

wide spectrum of presentation ranging from

tuberculoid to the lepromatous pole with

characteristic clinical and histological features

depending on the immune status of the host.

In lepromatous leprosy (LL), the classical

presentation includes, ill defined symmetrically

distributed anaesthetic macules, with infiltration

Fig 3 : Fite Faraco stain showing acid fast bacilli arranged in globi. (40X)

Shilpa et al192

and madarosis, combined with thickening of the

peripheral nerve trunks. Besides this, LL may

present in unusual ways that may cause confusion

in diagnosis and pose therapeutic problems. The

unusual presentations include localized lepro-

matous histoid leprosy (Job et al 1989), Lucio

leprosy, shiny infiltration of skin resembling

myxedema (Kaur et al 2005), lesions resembling

nodular subepidermal fibrosis (Sanchez 1965),

rarely, as urticarial wheals (Barman et al 2004a),

adenoma sebaceum (Barman et al 2004b),

erythema nodosum leprosum reaction mimicking

sweet's syndrome (Heng et al 2011). Sometimes

systemic involvement may be the only presen-

tation without skin or neurologic involvement

(Kerkerni et al 2011). In these studies, the

diagnosis was established on the basis of slit

skin smear examination and AFB positivity on ZN

staining (Barman et al 2013). A solitary lesion

with a high BI is a rare occurrence and has been

reported earlier by Sapkota et al (2009) as an

unusual presentation. In the present case, patient

had a single lesion and high BI similar to the above

case, a rarity yet possible occurrence in plethora

of LL spectrum. This case emphasizes the impor-

tance of skin smears and the histopathological

identification and classification of all patients

with unusual lesions.

Conclusion

In the post elimination era, the prevalence of

cases is decreasing, but unusual clinical presen-

tations are more commonly reported, which is a

reminder for keeping an open eye, a high degree

of suspicion, for detecting ad treating new cases

of leprosy. Cases as presently described is highly

infectious, hence early institution of anti-leprosy

treatment is mandatory to prevent transmission

of the disease, and adequate and effectively treat

them to prevent deformities.

References

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Subpolar lepromatous leprosy presenting as

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3. Barman KD, Goel K, Agarwal P et al (2013).

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tation: A case report. Indian J Lepr. 85: 27-31.

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flower ear. Egypt J Plast Reconstr Surg. 28: 23-7.

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How to cite this article : (2016). Ear due to Leprosy or Trauma - A Diagnostic Challenge. 88 : 189-192.

Shilpa K, Leelavathy B, Lakshmi DV and Divya D Unilateral Cauliflower Indian J Lepr.