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 Table of Contents  
CASE REPORT
Year : 2015  |  Volume : 2  |  Issue : 2  |  Page : 100-103

Toxic Epidermal Necrolysis Seen in a Human Immunodeficiency Virus Positive Pregnant Patient


1 Department of Obstetrics and Gynaecology, Specialist Hospital Jalingo, Taraba State, Nigeria
2 Department of Medicine, Specialist Hospital Jalingo, Taraba State, Nigeria
3 Department of Ophthalmology, Specialist Hospital Jalingo, Taraba State, Nigeria

Date of Submission21-Jan-2015
Date of Acceptance17-Feb-2015
Date of Web Publication20-May-2015

Correspondence Address:
Dr. Hajaratu U Sulayman
Department of Obstetrics and Gynaecology, Specialist Hospital Jalingo, Taraba State
Nigeria
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DOI: 10.4103/2384-5147.157437

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  Abstract 

This is a case report of a 27-year-old multipara with human immunodeficiency virus (HIV) infection in pregnancy. She was commenced on antiretroviral drugs: Nevirapine, Zidovudine, Lamivudine antiretroviral therapy, and co-trimoxazole but developed generalized maculopapular rash and eye lesions on the 6 th day of treatment. Toxic epidermal necrolysis was suspected. She was managed by a multi-disciplinary team of physicians and in a high dependency care setting and she recovered fully. This case is presented to highlight the possible occurrence of this condition among HIV-positive patients, the challenges of care and the importance for teamwork in such cases.

Keywords: Nevirapine, pregnancy, toxic epidermal necrolysis


How to cite this article:
Sulayman HU, Kwala WI, Attia RA, Aliyu ZY, Bushi MM. Toxic Epidermal Necrolysis Seen in a Human Immunodeficiency Virus Positive Pregnant Patient. Sub-Saharan Afr J Med 2015;2:100-3

How to cite this URL:
Sulayman HU, Kwala WI, Attia RA, Aliyu ZY, Bushi MM. Toxic Epidermal Necrolysis Seen in a Human Immunodeficiency Virus Positive Pregnant Patient. Sub-Saharan Afr J Med [serial online] 2015 [cited 2021 Jan 25];2:100-3. Available from: https://www.ssajm.org/text.asp?2015/2/2/100/157437


  Introduction Top


Cutaneous manifestations are common in patients with human immunodeficiency virus (HIV) infection, and in the initial stage, a transient maculopapular rash may appear. During the otherwise asymptomatic phase that follows, patients may develop seborrheic dermatitis, persistent genital ulcer disease, pruritic papular eruption, and/or a variety of scaling dermatoses. [1],[2],[3]

Toxic epidermal necrolysis (TEN) is said to be an immune-complex-mediated hypersensitivity reaction involving the skin and mucous membranes of the mouth, nose, eye, vagina, urethra, gastrointestinal and lower respiratory tract, which may progress to necrosis. [4],[5] The clinical manifestations include painful blistering of the skin/mucous membranes preceded by flu-like symptoms and high fever; the skin literally sloughs off. The etiological factors could be idiopathic in 50% of cases. Infections like herpes simplex virus; drugs like penicillins, sulfonamides, nevirapine, ibuprofen, and cocaine as well as malignancies have all been implicated. [1],[2],[3],[6],[7]

Toxic epidermal necrolysis (TEN) is extremely rare in pregnancy; the potential morbidity due to the immunologic effect of pregnancy is a cause for concern. Therefore, any case of TEN in pregnancy such as the one experienced by this patient deserves to be reported.


  Case Report Top


Mrs. A. I was a 27-year-old Gravida 3 para 2+0 housewife who presented at 24 weeks of gestation with a week's history of fever, generalized hyperpigmented rashes associated with painful blistering and sloughing of her skin.

She was HIV positive with a CD4 count of 128 cells/ml. Her anti-retroviral therapy (ART) combination consisted of nevirapine, zidovudine, and lamivudine and was also started on cotrimoxazole prophylaxis 6 days prior to presentation. She developed generalized blistering and sloughing of the skin around the face, lips, and throat leading to difficulty in breathing and eating.

Her husband was also HIV positive on ART. Their first child died at 5 years from complications of HIV. Second child was alive and HIV negative.

On examination, she was anxious, mildly pale, in respiratory distress, moderately dehydrated, and febrile (axillary temp 38.4°C). She had axillary and inguinal? lymphadenopathy, no pedal edema. Respiratory rate: 30 cycles/min, with bilateral basal crepitations. Pulse rate: 86 bpm and BP: 110/70 mmHg.

About 80% of her skin had vesiculobullous lesions, hyperpigmentation, and exfoliation with mucopurulent discharge and exfoliative dermatitis of the eyelids, and a mildly deranged visual acuity (5/6 in both eyes) [Figure 1] with swelling around the mouth and throat. The uterus was consistent with 24 weeks gestation [Figure 2].
Figure 1: On Admission

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Figure 2: Pregnant uterus on admission

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An impression of adverse drug reaction likely TEN probably due to nevirapine and/or co-trimoxazole in a HIV positive pregnant woman was made.

She was admitted in a high dependency ward, and all drugs were withdrawn.

She was immediately commenced on intravenous ceftriaxone 1 g daily for 5 days; intravenous metronidazole 500 mg 8 h for 5 days; intravenous dextrose saline 1 L 8 h for 5 days; intravenous dexamethasone 8 mg 8 h for 48 h. Oral lidocaine syrup three times daily for 7 days; intravenous methylprednisolone 125 mg 12 h for 48 h; and intravenous pentazocine 60 mg 6 h for 72 h was also prescribed. Intranasal 100% oxygen was given prn with the aid of a nasal catheter. Chloramphenicol eye ointment was administered four times daily. N G tube could not be passed due marked nasal mucosal edema. An indwelling urethral catheter was passed to monitor urine output.

She had barrier nursing care and daily dressing of the skin lesions with sofratulle (vaseline gauze).

Her vital signs were monitored closely. Investigation results: Full blood count: Packed cell volume - 23%, severe rouleaux formation, neutrophilic leukocytosis; U/E/CR, RBS and LFT were all normal. Ultrasound scan showed a singleton viable fetus at 24 weeks gestation. She had two units of blood transfused.

The respiratory distress and mucosal edema improved after 24 h. She started gradual sips of fluids and continued to make a remarkable recovery. She was discharged home after 2 weeks.

At a follow-up visit, she had improved [Figure 3]. CD4 count was 235 cell/ml, she was switched to another ART regimen of Zidovudine, Lamivudine, and at second follow-up, she had minimal ocular complications: Mild dryness of the eyes, minimal bilateral symblepharon, and left-sided superficial keratitis, these were managed by the ophthalmologist using artificial tears and ofloxacin eye drops. Her visual acuity became 6/6 (both eyes) thereafter.
Figure 3: Pregnant uterus with healed skin lesions on the first followup visit

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At 36 weeks Mrs. A. I went into spontaneous preterm labor and delivered a normal live male neonate weighing 2.8 kg. There were no complications. The patient chose infant formula feeding after due counseling. The baby and mother were seen two times postpartum and thereafter referred to the adult and pediatric ART Clinics, respectively, for follow-up [Figure 4] and [Figure 5].
Figure 4: Two weeks postpartum

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Figure 5: Mother and baby at 2 weeks postpartum

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  Discussion Top


Skin manifestations of adverse reactions to a variety of drugs occur more frequently in patients with HIV disease than in immunocompetent patients. In general, most skin diseases that occur in association with HIV disease respond well to standard treatment regimens. [1] Mrs. A. I had extensive skin and mucous membrane involvement and mild eye symptoms. Hers is also not a typical case as reactions to nevirapine occur at higher CD4 counts.

Treatment is usually supportive and should ideally be in a Burns center since it is a form of thermal injury. Fluids, analgesia, and oral care are of paramount importance. Ophthalmology consult is usual due to the formation of scar tissue inside the eyelids leading to corneal vascularization and impaired vision. Blindness can occur due to severe keratitis or panophthalmitis in 3-10% of cases. Cyclophosphamide, cyclosporine, intravenous immunoglobulins, and steroids are very useful treatment options. [2],[8]

Toxic epidermal necrolysis, Stevens-Johnson syndrome (SJS), and Lyell's disease are considered to be part of a spectrum of adverse cutaneous drug reactions with increasing severity and extensive skin detachment, ranging from SJS which has (<10% body surface area skin detachment accompanied by 1-5% mortality) to TENs (>30% skin detachment and 25-35% mortality). Both SJS and TENs are characterized morphologically by ongoing apoptotic keratinocyte cell death that results in the separation of the epidermis from the dermis. [1],[7],[9],[10],[11],[12] Mrs. A. I had more than 80% skin involvement hence making TENs more likely.

Human immunodeficiency virus-infected patients are at risk of developing cutaneous ADRs, especially TENs, SJS, and drug hypersensitivity syndrome. When no effective alternate therapy is available, drug re-challenge can be attempted with little morbidity or mortality if done according to rationalized protocols. [6] This was done in this patient according to the National Prevention of Mother To Child Transmission of HIV (PMTCT) guideline. HIV-infected patients with baseline CD4 <250 cells/µL can have severe Nevirapine-associated hepatitis. [13] Mrs A. I luckily had normal liver function test.

The prognosis of SJS/TEN can be predicted by a scoring system based on seven clinical and laboratory parameters. [2] Found on the genetic background of SJS/TEN, predictive tests can be used prior to starting high-risk medications. Treatment is still controversial, and further controlled studies are necessary. [14] Mrs A. I had a Scorten score of 4 hence her risk of dying was more than 58%. (Serum Glucose: 180 mg/dL, HCO 3 : 17 mEq/L, HR: 125 bpm, >80% BSA) [Table 1].
Table 1: Prognosis: SCORTEN Scale

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  Conclusion Top


Immune modulation with the use of high-dose short-term steroids, intravenous immune globulin, and cyclosporine remain attractive therapeutic options. Unequivocal efficacy data on these agents, however, remain limited. Our case illustrates the potential benefits of high dose short-term steroids use and raises the curious questions of the potential interaction between Nevirapine and Co-trimoxazole in increasing the risk for TENs akin to the well-validated cases of Lamotrigine and Valproic acid and SJS.

 
  References Top

1.
Pallangyo KJ. Cutaneous findings associated with HIV disease including AIDS: Experience from Sub Saharan Africa. Trop Doct 1992;22 Suppl 1:35-41-60.  Back to cited text no. 1
    
2.
Bastuji-Garin S, Fouchard N, Bertocchi M, Roujeau JC, Revuz J, Wolkenstein P. SCORTEN: A severity-of-illness score for toxic epidermal necrolysis. J Invest Dermatol 2000;115:149-53.  Back to cited text no. 2
    
3.
French LE, Trent JT, Kerdel FA. Use of intravenous immunoglobulin in toxic epidermal necrolysis and Stevens-Johnson syndrome: Our current understanding. Int Immunopharmacol 2006;6:543-9.  Back to cited text no. 3
    
4.
Ranario JS, Smith JL. Bullous lesions in a patient with systemic lupus erythematosus. J Clin Aesthet Dermatol 2014;7:44-9.  Back to cited text no. 4
    
5.
Kim DH, Yoon KC, Seo KY, Lee HS, Yoon SC, Sotozono C, et al. The role of systemic immunomodulatory treatment and prognostic factors on chronic ocular complications in stevens-johnson syndrome. Ophthalmology 2015;122:254-64.  Back to cited text no. 5
    
6.
Todd G. Adverse cutaneous drug eruptions and HIV: A clinician's global perspective. Dermatol Clin 2006;24:459-72, vi.  Back to cited text no. 6
    
7.
French LE. Toxic epidermal necrolysis and Stevens Johnson syndrome: Our current understanding. Allergol Int 2006;55:9-16.  Back to cited text no. 7
    
8.
Lonjou C, Borot N, Sekula P, Ledger N, Thomas L, Halevy S, et al. A European study of HLA-B in Stevens-Johnson syndrome and toxic epidermal necrolysis related to five high-risk drugs. Pharmacogenet Genomics 2008;18:99-107.  Back to cited text no. 8
    
9.
Tassaneeyakul W, Jantararoungtong T, Chen P, Lin PY, Tiamkao S, Khunarkornsiri U, et al. Strong association between HLA-B*5801 and allopurinol-induced Stevens-Johnson syndrome and toxic epidermal necrolysis in a Thai population. Pharmacogenet Genomics 2009;19:704-9.  Back to cited text no. 9
    
10.
Hung SI, Chung WH, Liu ZS, Chen CH, Hsih MS, Hui RC, et al. Common risk allele in aromatic antiepileptic-drug induced Stevens-Johnson syndrome and toxic epidermal necrolysis in Han Chinese. Pharmacogenomics 2010;11:349-56.  Back to cited text no. 10
    
11.
Magazine R, Chogtu B. Stevens Johnson syndrome and neurotoxic effects of metronidazole. Indian J Pharmacol 2014;46:565.  Back to cited text no. 11
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12.
Kakar R, Paugh H, Jaworsky C. Linear IgA bullous disease presenting as toxic epidermal necrolysis: A case report and review of the literature. Dermatology 2013;227:209-13.  Back to cited text no. 12
    
13.
Manosuthi W, Sungkanuparph S, Tansuphaswadikul S, Chottanapund S, Mankatitham W, Chimsuntorn S, et al. Incidence and risk factors of nevirapine-associated severe hepatitis among HIV-infected patients with CD4 cell counts less than 250 cells/microL. J Med Assoc Thai 2008;91:159-65.  Back to cited text no. 13
    
14.
Halevy S. Stevens-Johnson syndrome and toxic epidermal necrolysis - updates and innovations. Harefuah 2010;149:186-90, 193.  Back to cited text no. 14
    


    Figures

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

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