Vancomycin-induced bullous dermatosis: a rare case report

Introduction:

Linear IgA bullous dermatosis (LABD) is a rare acquired skin blistering autoimmune disease. It can be diagnosed by confirming the presence of a linear band of IgA at the dermoepidermal junction on direct immunofluorescence microscopy. LABD can be characterized by vesicular lesions, diffuse blisters, or even as a mimicker of Steven–Johnson syndrome. LABD may be caused by tumours, infections, or drugs (amiodarone, furosemide, phenytoin, however, vancomycin is the potential inciting drug in most reports).

Case presentation:

The authors present here a case of a 61-year-old woman with a history of HTN. The patient had a discectomy 15 years ago, and also underwent a lumbar fusion surgery that resulted in complications with her discitis. Due to the complications from the surgery, intravenous treatment with vancomycin and meropenem was initiated. After a few days of treatment, the patient developed clear, tense, fluid-filled bullae over the upper extremities. Immunofluorescence microscopy is not available in our hospital. Therefore a diagnosis of vancomycin-induced LABD was proposed based on the clinical manifestation of the lesions and the coincidence with vancomycin administration. After 2 days of discontinuing the administration of vancomycin and applying local diprosone, the lesions started to regress and a full recovery was achieved on day 10.

Discussion and conclusion:

Even though drug-induced LABD is uncommon, its incidence has been steadily increasing in the last few years. LABD is a simple condition with a good prognosis and full recovery after the discontinuation of vancomycin

Introduction

Highlights

• Linear IgA bullous dermatosis can present as papules, tense vesicles, and bullae on erythematous or normal-appearing skin.

• Correct diagnosis is resulting in a good prognosis and full recovery after the discontinuation of vancomycin.

• We can use the Naranjo algorithm, or Adverse Drug Reaction Probability Scale, to assess whether there is a causal relationship between a specific unwanted clinical event and a drug by using a simple questionnaire to assign probability scores.

• Early and accurate diagnosis of the lesion can improve the prognosis and maintain the patient’s quality of life.

Linear IgA bullous dermatosis (LABD) is a rare acquired skin blistering autoimmune disease with an incidence rate of about 0.5–2.3 cases per million individuals per annum¹. It is diagnosed by confirming the presence of a linear band of IgA at the dermoepidermal junction on direct immunofluorescence microscopy². The clinical presentation varies from vesicular lesions, to diffuse blisters, or even manifests as a mimicker of Steven–Johnson syndrome³. LABD is usually idiopathic, but it may be induced by drugs, tumours, or infections². Drugs that have been implicated as the cause include amiodarone, furosemide, phenytoin, however, vancomycin (a glycopeptide antibiotic administered intravenously for suspected or proven infection with invasive Gram-positive including methicillin-resistant Staphylococcus aureus ⁴) is the potential .inciting drug in most reports².

Case report

A 61-year-old woman, with a history of HTN (17/9) and a surgical lumbar discectomy from 15 years prior, presented Lower Back Pain (8/10) and weakness in lower extremities. The patient underwent lumbar instrumentation and fusion surgery. A few days after surgery she developed discitis as a complication and intravenous antibiotics (vancomycin and meropenem) were administered. However, a few days after the administration of vancomycin, she noticed skin lesions on the 9th day of admission. Upon physical examination, clear fluid-filled bullae were detected, concentrated on the patient’s upper extremities (Fig. 1).

Figure 1.

Multiple tense clear fluid-filled bullae on the right upper extremities.

The diagnosis was linear IgA bullous dermatosis—particularly with the patient’s recent history of receiving vancomycin. Unfortunately, direct immunofluorescence studies are not available in our hospital to diagnose the lesions, so the only treatment choice was Diprosone cream and the discontinuation of vancomycin administration. The patient’s lesions started to improve after 2 days due to the discontinuation of vancomycin and she reached a full recovery after 10 days.

This case report has been reported in line with the SCARE Criteria (Fig. 2).

Figure 2.

After a few days of discontinuing vancomycin administration.

Discussion

LABD, also known as linear IgA disease, is a rare, idiopathic or drug-induced autoimmune subepidermal, vesiculobullous eruption characterized by the linear deposition of IgA at the dermoepidermal junction^5,6.

The annual incidence rate is reported to be ~0.5–2 cases per million people⁶.

Two peaks in age of onset have been reported: one at 4.5 years of age and a second peak at 60 years of age with a slight female predominance⁷.

LABD can appear as papules, tense vesicles and bullae on erythematous or normal-appearing skin. The small blisters may be described as a “cluster of jewels” or “crown of jewels” and are most frequent in the childhood stage.

Drug-induced LABD, in particular vancomycin, may have erythema multiform-like findings or toxic epidermal necrolysis-like or morbilliform rash⁷.

Drug-induced LABD shows some different characteristics compared with the idiopathic variant. Drug-induced LABD patients were reported to have an absent of mucosal or conjunctival involvement, while up to 40% of patients with idiopathic LABD have mucosal involvement. Studies also show that patients with drug-induced LABD tend to be older than patients with idiopathic variant².

LABD may be associated with a number of various disorders such as: gluten-sensitive enteropathy^8,9, Ulcerative colitis, Crohn disease, gastric hypochlorhydria^2,7–15, systemic lupus erythematosus, dermatomyositis, thyrotoxicosis, autoimmune haemolytic anaemia, rheumatoid arthritis, glomerulonephritis^8,9,14–16, malignancies (including B-cell lymphoma, chronic lymphocytic leukaemia, and carcinoma of the bladder, thyroid, colon and oesophagus)^2,7–18, infections 20- (including varicella zoster virus, and upper respiratory infections)^19,20, and drugs.

Drugs that may be implicated include: Vancomycin (which is the most frequently implicated culprit)—followed by non-steroidal anti-inflammatory drugs and penicillins. Other drugs include: Captopril− Trimethoprim + sulphamethoxazole− Amiodarone – Furosemide – Ciclosporin – Glibenclamide – Lithium− Cephalosporins – Phenytoin− Sodium hypochlorite – statins—tea tree oil^2,5–21.

Vancomycin is a glycopeptide antibiotic administered intravenously for treatment of patients with suspected or proven invasive Gram-positive infections, including methicillin-resistant Staphylococcus aureus ²².

Drug-induced LABD is uncommon, but its incidence rate had a steady increase in recent years².

LABD can occur within 1 day to 1 month after the initiation of vancomycin¹³.

In our presented case, the patient had developed tense bullae over the upper extremities a few days after the treatment with vancomycin and meropenem, which arouses our suspicion of a drug-induced bullous disease.

The case is in accordance with vancomycin-induced LABD due to the absence of mucosal involvement, good general condition, negative Nikolsky’s sign, absence of pruritus and the chronology (onset after 9 days of Vancomycin initiation, improvement 2 days after discontinuation and complete resolution 10 days after termination of Vancomycin). This is also in accordance with vancomycin-induced LABD.

Due to the lack of skin biopsy techniques available in this hospital, and the non-availability of Immunofluorescence studies, we have made our diagnosis based on the chronological plausibility, a Naranjo score (5) and the characteristic clinical input.

The Naranjo Algorithm, or Adverse Drug Reaction Probability Scale, is a method which assesses the relationship between a drug and the possible adverse reactions²³.

In the case we presented the Naranjo algorithm was 5 (probable). The patient with Vancomycin-induced LABD recovered quickly without residual skin lesions.^23–25

Ethical approval

The ethical approval was obtained from the patient and the director of the Department of Neurosurgery.

Consent

Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request. As the patient is a child of two years of age, the consent was obtained from the patient’s parents.

Source of funding

None.

Author contribution

M.K., M.A.A., K.A., M.W., and N.S. did the surgery. N.S., A.J., M.A.A., K.A., and M.W. wrote the manuscript.

Conflicts of interest disclosure

The authors declare that there is no conflict of interest regarding the publication of this paper.

Research registration unique identifying number (UIN)

NA.

Guarantor

Mahmoud Wereekia.

Provenance and peer review

Not commissioned, externally peer-reviewed.