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  IN THIS Article
 ::  Abstract
 :: Introduction
 :: Case Report
 :: Discussion
 :: Acknowledgments
 ::  References
 ::  Article Figures

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  Table of Contents     
CASE REPORT
Year : 2012  |  Volume : 58  |  Issue : 3  |  Page : 207-209

Hidradenitis suppurativa in a HIV-infected child


Department of Pediatrics, Regional Pediatric ART Centre, Lokmanya Tilak Municipal Medical College and General Hospital, Sion, Mumbai, India

Date of Submission10-Jun-2011
Date of Decision06-Oct-2011
Date of Acceptance06-Feb-2012
Date of Web Publication26-Sep-2012

Correspondence Address:
M Manglani
Department of Pediatrics, Regional Pediatric ART Centre, Lokmanya Tilak Municipal Medical College and General Hospital, Sion, Mumbai
India
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DOI: 10.4103/0022-3859.101403

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 :: Abstract 

Hidradenitis suppurativa (HS), a painful and chronic condition, commonly occurs in women and coincides with post-pubertal increase in sex hormones. A 13-year-old pre-pubertal HIV-infected male child presented to our clinic with a discharging right axillary lymph node swelling. The biopsy of the lesion showed features of HS. The patient was treated with oral antibiotics, oral steroids, and local antibiotic wash. Though the patient responded to this treatment, the clinical response was not adequate and the lesion recurred. Subsequently, the child was started on antiretroviral therapy (zidovudine, lamivudine, and nevirapine). Following these medications, the lesions healed and had not recurred till we last examined the child. Thus, this is a rare presentation of a known condition in an HIV-infected pre-pubertal male child, which did not respond to usual modalities of treatment and had to be treated with antiretroviral therapy.


Keywords: Child, hidradenitis suppurativa, HIV


How to cite this article:
Prabhu G, Laddha P, Manglani M, Phiske M. Hidradenitis suppurativa in a HIV-infected child. J Postgrad Med 2012;58:207-9

How to cite this URL:
Prabhu G, Laddha P, Manglani M, Phiske M. Hidradenitis suppurativa in a HIV-infected child. J Postgrad Med [serial online] 2012 [cited 2014 Oct 22];58:207-9. Available from: http://www.jpgmonline.com/text.asp?2012/58/3/207/101403



 :: Introduction Top


Hidradenitis suppurativa (HS) is a disabling chronic disorder of the terminal follicular epithelium in the apocrine gland-bearing skin. [1] It is rare in healthy individuals with a prevalence of 1-4% in the general population and is three times more commonly seen in women. [2],[3] Furthermore, HS is rare before puberty and most cases are seen during 20-40 years of age (range 11-50 years), and coincides with the increase in sex hormones. [4],[5],[6] Some of the common treatment modalities are: medical such as antibiotics, retinoids, intralesional steroids and hormonal manipulation, and surgical such as excision and radical therapy, and recently even biologics such as adalimumbab have also been tried. [7],[8]

HS is painful and can be disabling but rarely fatal, except when it progresses to an overwhelming systemic infection in an immune-compromised patient. There is very scant literature on the occurrence of HS in HIV-infected individuals, especially in children. We present a rare presentation of HS in an HIV-infected pre-pubertal male child responding inadequately to some of the conventional medical modalities of treatment. To the best of our knowledge, there is no case of HS in an HIV-infected child reported so far in global literature.


 :: Case Report Top


A 13-year-old HIV-infected male child was referred to our centre in November 2008, with a history of right axillary discharging sinus [Figure 1]. The lesion had started as a swelling which gradually increased in size over a period of one month and ruptured to form a discharging sinus at the time of presentation. He was detected to be HIV-infected during family screening following the death of his father due to pulmonary tuberculosis (open type) a month prior to this episode. There was no history of similar lesions elsewhere on the body. On examination, the lesion was an indurated, tender, hyperpigmented nodular plaque 2 × 2 × 3 centimeters in size and showed serous discharge. No systemic abnormalities were observed in the child. There was no neurodevelopmental delay and the child was immunized for his age. The height and weight of the child were 135.5 centimeters and 24.5 kilograms respectively; both these were less than the fifth percentile for the age. The initial differential diagnoses based on the clinical presentation were scrofuloderma or hidradenitis suppurativa.
Figure 1: The lesion in the right axilla at presentation

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Investigations revealed hemoglobin of 12.7 g/dL (normal), the total white cell count of 13×10 3 /μL, with polymorphonuclear leucocytosis. Erythrocyte sedimentation rate was 20 mm at the end of one hour (normal). The chest X-ray did not reveal any abnormality and mantoux test was negative. Fine needle aspiration cytology was suggestive of mixed pyogenic inflammation. A culture of the aspirate grew methicillin-sensitive Staphylococcus aureus. However, it was negative for acid-fast bacilli and fungi. The BACTEC (rapid radiometric culture test) performed on the aspiration material too was negative for tuberculosis (TB). The skin biopsy from the site showed basketweave orthokeratosis, focal parakeratosis overlying an acanthotic epidermis, dense plasma cell infiltrate around the sweat glands in the dermis, and marked fibrosis in the deep dermis [Figure 2]; these findings were consistent with a diagnosis of HS. Scrofuloderma was ruled out as there were no features suggestive of granuloma. The CD4 count was 1054 cells/μL (27%) at the time of presentation, which dropped to 757 cells/μL in May 2009.
Figure 2: Histopathology of the punch biopsy of the lesion confirming HS. Arrow shows follicular plugging in the lesion

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The child was treated with local antibiotic washes, and with Amoxycillin and Clavulinic acid (40 mg/kg/day) for three courses of five days each along with a short course of oral prednisolone (2 mg/kg) for 10 days. Following this, the lesions improved but recurred; even the clinical improvement was inadequate. After three courses (last course was in May 2009) we started the child on antiretroviral therapy (ART) (Zidovudine/Lamivudine/Nevirapine) in July 2009. Following this therapy, the lesions healed and had not recurred at the last clinical examination in March 2011.


 :: Discussion Top


Thus, we highlight a rare presentation of HS based on the following features: (1) presentation in a pre-pubertal male patient; (2) association with HIV; and (3) inadequate response to conventional medical management but healing after ART. Of the 1170 HIV-infected children registered at our centre, this is the first case of HS. Furthermore, to the best of our knowledge, this is the first case of HS in a HIV-infected male child in the pre-pubertal age group.

HS, first described by Velpeau in 1839, is also known as Vemeuil's disease or acne inversa. It is an inflammatory skin disorder characterized by recurrent abscesses with pus-like discharge, difficult-to-heal open wounds, and scarring. [1] It commonly occurs on apocrine gland-bearing skin (such as in the groin, underarms and under the breast) and is a member of the follicular occlusion tetrad along with acne conglobata, dissecting cellulitis of the scalp, and pilonidal sinus. [9]

The exact etiology of HS both in healthy and immune-compromised individuals remains obscure. However, certain secondary factors such as genetic predisposition, onset of puberty, and cigarette smoking may play a role; though none of these were present in our patient. [4],[5],[10],[11] The diagnosis is primarily clinical and the consensus approach deems that three key elements are required to diagnose HS-typical lesion, characteristic distribution, and recurrence; these were present in our patient. As the disease become chronic, scars and contractures develop with persistent erythema; the patient's mobility may be restricted and the patient may not be able to fully raise the upper arm.

Medical management of HS is difficult and general measures include local hygiene, local antiseptic wash and prolonged course of oral antibiotics for a minimum of 3-6 months. Surgical intervention in typical post-pubertal HS is rarely required; however, in a pre-pubertal child it may be an effective intervention. [4] HS is not listed as one of the opportunistic infections (OI) in children and as per the clinical and immunologic staging our patient did not fit the criteria for initiating ART as per national (National AIDS Control Organization) guidelines for children. However, in view of child's non-response to the conventional treatment for HS, and assuming that the lesions were a result of impaired immunity, it was thought appropriate to start ART. This has a potential for improving the child's immunity by increasing the functionally normal CD4 cells and helping in combating the lesions better. Also, this was evident by the fact that the child responded well to this therapy. There was no further recurrence of the lesions suggesting thereby that initiating ART may be considered in children who do not respond to other modalities of treatment of HS.

Nonetheless, the case highlights an important rare presentation of HS in an HIV-infected child and also suggests management with ART in children with associated HS.


 :: Acknowledgments Top


We are grateful to our Dean, Dr. Sandhya Kamath for permitting us to publish this case report. We acknowledge the contribution and support of NACO, MDACS and UNICEF to our centre. We wish to thank Dr. H R Jerajani (Professor and Head, Dermatology, LTMG Hospital) for her help in diagnosing HS. We would like to acknowledge Dr. Maninder S Setia (Consultant Epidemiologist) for his editorial comments. We also thank our patient.

 
 :: References Top

1.Kurzen H, Kurokawa I, Jemec GB, Emtestam L, Sellheyer K, Giamarellos-Bourboulis EJ, et al. What causes hidradenitis suppurativa? Exp Dermatol 2008;17:455-6; discussion 457-72.  Back to cited text no. 1
[PUBMED]    
2.Jemec GB, Heidenheim M, Nielsen NH. The prevalence of hidradenitis suppurativa and its potential precursor lesions. J Am Acad Dermatol 1996;35:191-4.  Back to cited text no. 2
[PUBMED]    
3.Kelly AM, Cronin P. MRI features of hidradenitis suppurativa and review of the literature. AJR Am J Roentgenol 2005;185:1201-4.  Back to cited text no. 3
[PUBMED]    
4.Mengesha YM, Holcombe TC, Hansen RC. Prepubertal hidradenitis suppurativa: Two case reports and review of the literature. Pediatr Dermatol 1999;16:292-6.  Back to cited text no. 4
[PUBMED]    
5.Palmer RA, Keefe M. Early-onset hidradenitis suppurativa. Clin Exp Dermatol 2001;26:501-3.  Back to cited text no. 5
[PUBMED]    
6.Ngan V, Oakely A. Hidradenitis Suppurativa. DermNet NZ. DermNet NZ 13 April 2009. Available from: http//www. dermnetnz.org/acne/hidradenitis-suppurativa.html [Last cited on 2009 Jun 15].  Back to cited text no. 6
    
7.Miller I, Lynggaard CD, Lophaven S, Zachariae C, Dufour DN, Jemec GB. A double-blind placebo-controlled randomized trial of adalimumab in the treatment of hidradenitis suppurativa. Br J Dermatol 2011;165:391-8.  Back to cited text no. 7
[PUBMED]    
8.Slade DE, Powell BW, Mortimer PS. Hidradenitis suppurativa: Pathogenesis and management. Br J Plast Surg 2003;56:451-61.  Back to cited text no. 8
[PUBMED]    
9.Wiseman MC. Hidradenitis suppurativa: A review. Dermatol Ther 2004;17:50-4.  Back to cited text no. 9
    
10.Buimer MG, Wobbes T, Klinkenbijl JH, Hidradenitis suppurativa. Br J Surg 2009;96:350-60.  Back to cited text no. 10
    
11.Von Der Werth JM, Williams HC, Raeburn JA. The clinical genetics of hidradenitis suppurativa revisited. Br J Dermatol 2000;142:947-53.  Back to cited text no. 11
[PUBMED]    


    Figures

  [Figure 1], [Figure 2]

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Peter Riis Mikkelsen,Gregor B. E. Jemec
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