Erythrodermic Atopic Dermatitis Associated with Dust Mite and Alternaria alternata in an Eight-Year-Old Child: A Case Report
Correspondence Address :
Dr. Ruchi Shah,
Reader, Department of Conservative Dentistry and Endodontics, KM Shah Dental College and Hospital, Sumandeep Vidhyapeeth, Vadodara-391760, Gujarat, India.
E-mail: ruh006@gmail.com
Atopic Dermatitis (AD) is a chronic inflammatory condition of the skin characterised by itching, recurrent lesions, and lichenification. Erythroderma, also known as generalised exfoliative dermatitis, is characterised by erythema that covers more than 90% of the body’s surface. These erythematous lesions are much more prone to infection. In the present case, an eight-year-old girl presented with a chief complaint of rashes all over the body with severe itching for two years and presented to the Allergy and Asthma Centre. She had a positive history of atopy and multiple septicaemia-related hospitalisations for the last two years. Skin biopsy led to the diagnosis of Spongiotic Dermatitis consistent with Erythroderma secondary to AD. Multiple allergies, including those to dust mites (DP der p2, p21, DF der f2) and Alternaria alternata (alt 1), were identified by Component-Resolved Diagnosis (CRD). Cyclosporin, Omalizumab (a monoclonal antibody against immunoglobulin E), and allergen-specific sublingual immunotherapy were given for the management and all gave excellent results in the patient. AD can be properly diagnosed and treated in its early stages, breaking the cycle that leads to severe erythroderma. To fully comprehend the molecular and immunological genesis of these allergic types, more research is required.
Allergic reaction, Chronic inflammation, Erythroderma, House dust mite, Immunotherapy
An eight-year-old girl presented to the Allergy and Asthma Centre with a chief complaint of on-and-off rashes all over the body with severe itching for two years. In the family, the mother had asthma and dust mite allergy from her childhood and was under medication for the same. Furthermore, the patient was living in a very old vintage home in a downtown area. The patient was relatively asymptomatic two years back, then she started experiencing rashes, especially on the antecubital fossa, lateral regions of the neck, knee joint, and ankle joint. The patient took multiple treatments from dermatologists and paediatricians, where steroid ointments (Omnacortil 0.1% Cream) and emollients (Sebamed) were prescribed, which helped in resolving the lesions, but they recurred. The patient also took Ayurvedic and Homoeopathic treatment for the same, but lesions recurred after their medications also. The patient also had a history of multiple hospitalisations due to septicaemia for the last two years. For the last month, the patient had continuous rashes and severe itching all over the body (90% of body surface area of exfoliative skin scaling, termed as erythroderma), maculopapular rash was present on the face, flexors, feet, and ankle area with multiple lymphadenopathies (Table/Fig 1).
Further, scratching of lesions led to erosions followed by painful fissures. Histological examination showed lamellate stratum corner with parakeretosis. There was presence of epidermal acanthoses with hypogranulosis and lymphocytic exocytosis with subcorneal collection of lymphocytes. The papillary dermis showed mild oedema with superficial perivascular infiltrate consisting lymphocytes with plenty eosinophils. Histopathological features were suggestive of spongiotic dermatitis consistent with erythroderma secondary to AD (Table/Fig 2). In blood reports, the absolute eosinophilic count was 3,000 (normal range 0 to 500 cells/μL), the total IgE count was 45011 IU/mL, and IgA, IgG, IgM, IgD levels were within the normal limits as per age.
Molecular diagnosis by CRD was strongly positive for house dust mites/Dermatophagoides pteronyssinus (DP, Der p2, NPC2 Family >50.00 kUA/L, Der p 21, >50 kUA/L, Der p 1, cysteine protease 45.99 kUA/L), Dermatophagoides farinae (DF, Der f 2, NPC2 family 49.65 kUA/L, der f1, cysteine protease 47.51 kUA/L), Moulds Alternaria alternata (Alt a1 family 45.92 kUA/L), Malassezia sympodialis (Mala s 6, 35.31 kUA/L), moderately positive for chickpea lentil, peas, wheat, sesame and mildly positive for casein (Table/Fig 3). Differential diagnosis, in this case, included other conditions that could lead to the exfoliative erythrodermic syndrome, erythrodermic psoriasis, lymphoma, leukaemia and cutaneous drug reaction, pityriasis rubra pilaris and pemphigus foliaceus which were ruled out after skin histological reports. Final diagnosis of Erythrodermic AD associated with house dust mites and Alternaria alternata majorly was confirmed.
As there was an increased probability of staphylococcal infection in such erosive rashes, hospitalisation was advised, to which the parents were resistive. The patient was given symptomatic and supportive treatment to relieve the acute symptoms with emollient (Emolene) and Clobetasol Propionate cream, and antibiotics (amoxicillin clavulanic acid 40 mg/Kg/day and linezolid 10 mg/Kg/dose TDS) for 21 days to reduce the chances of infection. Avoidance of food allergens was advised along with cyclosporine initially with 5 mg/Kg/day dose for three months initially and was tapered after six months to 3 mg/Kg/day, Ketotifen (H1 receptor blocker and mast cell stabiliser) (1 mg BD) for controlling the symptoms prescribed for three months initially and continued with same dose for an year. Inj.Omalizumab 150 mcg (Monoclonal anti-immunoglobulin E antibody) monthly for three months and sublingual immunotherapy for dust mite daily for four weeks initially followed by twice a week for an year, was started to stop the progression of allergy and the atopic march. Cyclosporins dose was tapered to 3 mg/kg/day and Ketotifen in same dose, that is 1 mg BD, were continued for a year. Immunotherapy is still continued after a year, will be modified depending on the symptoms of the patients.
The patient returned after a week with a complete resolution of all symptoms and rashes (Table/Fig 4). The patient is under regular follow-ups. After one year patient was symptom free without any recurrence. The patient returned to her routine activities after the resolution.
The AD is an inflammatory chronic skin condition usually associated with a family history of atopy such as allergic rhinitis, asthma etc. Erythroderma presents as redness, lichenification, and scaling of the skin, with intense pruritus which leads to erosion and painful fissures which if left untreated can become a life-threatening condition (1). The predisposed areas affected are eyelids, face, neck, dorsa of feet and hands, flexors, wrist joint and or in severe 2condition can become generalised (2). These lesions are very prone to get colonised by Staphylococcus aureus which worsens the skin inflammation by forming a vicious circle of releasing continuous exotoxins and stimulating the T cells and macrophages further (3). The exact cause of this condition is yet not completely understood. The correlation between the genetic and environmental conditions, with the immunological reaction, gives an idea about their strong interaction (4). The diagnosis of erythrodermic AD is made based on clinical criteria proposed by Hanifin JM and Rajka G in which three of the four mentioned features should be presented. Pruritus, lichenification, chronically relapsing course and atopic history. Minor characteristic features present are Immediate (type I) skin reaction, elevated serum IgE level, early age of onset, cutaneous infection, cheilitis, recurrent conjunctivitis etc., (5).
Allergy associated with house dust mites in kids was 7.8% in a study in Indian children (6), while with Alternaria alternata was 17.9% in kids between 5 to 18 years of age (7). Evaluating IgE levels and skin prick test is the most common tests to determine sensitivity to allergens. Where 85% of cases show elevated IgE levels (2). The blood findings of increased IgE and absolute eosinophilic count was the key component after the clinical criteria which helped in early diagnosis and planning the strategy of treatment. A skin biopsy confirmed the lesion type. Similar case has been reported in a 21-year-old girl in Romania, with generalised erythematous eczematous skin lesions, flexural lichenifications accompanied by intense pruritus, painful fissures and erosions resulting from scratching. The main laboratory findings were- high serum eosinophilia (2,400/μL) and very high total IgE serum (11449 UI/L). Diagnosis of Erythrodermic AD with late onset-case presentation was made in this case (2). In a retrospective study conducted in New Delhi, India, 15% erythrodermic lesions are caused due to AD (8). In another study on Indian kids 12% cases of erythroderma were associated by AD (9).
A nine-year-old boy from Boston, Massachusetts, USA, who had widespread eczematous dermatitis and a positive history of atopy, was reported in a case similar to this one. With extremely positive ImmunoCAP specific IgE levels to dust mite, mouse, and cockroach, as well as various tree and grass pollens, his total serum IgE level was significantly raised at 4300 IU/mL. The prospect of allergen immunotherapy was explored with his family as a potential therapeutic option, along with emollient and topical corticosteroid therapy, cyclosporine, topical tacrolimus, and several other medications (10). In some cases of erythroderma, due to the unavailability of unaffected skin, a skin prick test cannot be perfomed. Thus, CRD or molecular diagnosis of allergy determines total serum IgE against purified native and recombinant allergic molecules. In CRD allergens are divided on basis of source (e.g., inhalant, nutritive, contact, hymenoptera venom), and the basis of protein molecules (storage proteins, profiling, a calcium-binding protein, serum albumin etc.,) (11). In the case presented, allergens were inhalant, nutritive type with protein type of tropomyosin, defense-like proteins and storage proteins. CRD can be used in two ways singleplex and multiplex-microarray assays (11),(12). The technique used in the case presented was multiplex assay as multiple allergens-specific IgE was required to be determined. Dermatophagoides pteronyssinus (DP, European house dust mite) and Dermatophagoides farinae (DF, American house dust mite) DP DF, are most commonly found in warm and moist areas, also commonly found in beds. Currently, 23 house dust mites allergens are known (Colloff, 2009) (13).
Alternaria alternata is a fungus seen routinely in humid indoor areas. Malassezia furfur (Pityrosporum ovale in hyphal form) is a type of yeast that is naturally found on the skin surfaces of humans and some other mammals (14). Short-term control comprises of supportive care such as fluid and electrolyte infusion, and systemic antibiotic administration (1),(15).
Further sublingual and oral combination immunotherapy for dust mites was initiated. Its successful management has been discussed previously (14). The evidence for the effectiveness of antigen specific immunotherapy for the treatment of AD was recently provided by a meta-analysis by Bae JM et al., (10). According to this study, Specific Immunotherapy (SIT) significantly reduced the risk of developing AD, with an Odds Ratio (OR) of 5.35 and a 95% Confidence Interval (CI) of 1.61-17.77. Furthermore, SIT significantly improved the condition of patients with severe AD (OR 6.42, 95%CI 1.31-7.48) (16). In these complicated situations, no one allergy treatment works. It’s a comprehensive plan for managing the body’s immunological response to enhance our patients’ quality of life (17),(18).
Emollients combined with topical corticosteroids are an efficient way to treat AD in children. The use of sophisticated medicines, such as immunomodulating systemic therapy, biologicals in the form of monoclonal anti-immunoglobulin E antibodies, and allergen-specific immunotherapy, as in the case described, may be necessary for severe AD. A deeper understanding of the disease’s pathophysiology is necessary to identify new therapeutic targets and enhance the quality of life for AD patients.
DOI: 10.7860/JCDR/2023/61808.17927
Date of Submission: Nov 23, 2022
Date of Peer Review: Feb 03, 2023
Date of Acceptance: Feb 28, 2023
Date of Publishing: May 01, 2023
AUTHOR DECLARATION:
• Financial or Other Competing Interests: None
• Was informed consent obtained from the subjects involved in the study? Yes
• For any images presented appropriate consent has been obtained from the subjects. Yes
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