A case of infantile bullous pemphigoid successfully treated
with compound glycyrrhizin tablets
Dear Editor,
Bullous pemphigoid is an autoimmune blistering disorder that is
reported mostly in elderly individuals; it is exceptional in infants. When
the lesions are widespread, the administration of systemic prednisone,
immunosuppressants, or immunoglobulins may be necessary.1 However,
these medications may result in serious adverse effects. Here, we report
a case of severe BP that was cured with compound glycyrrhizin tablets.
A 6-month-old healthy girl, weighing 7 kg, presented with a
20-day history of generalized pruritic erythema and blistering. She
was born after an uncomplicated pregnancy and had not received any
vaccination within the previous 2 months. Examination revealed
extensive erythematous lesions in annular shapes and multiple tense
blisters or bullae on her face, trunk, and limbs (Figure 1A). The blisters
were noted particularly on the dorsal surfaces of her hands
(Figure 1B). The mucous membranes were not involved. Histological
examination revealed subepidermal fissures containing many eosinophils (Figure 2A). Direct immunofluorescence (DIF) of the skin revealed the linear deposition of immunoglobulin G (IgG) and
complement C3 along the dermoepidermal junction (Figure 2B,C). The
serum anti-BP180 antibody titer was 121.27 U/mL (normal range,
<9 U/mL) (Figure 3). Laboratory tests revealed a white blood cell
count of 11.30 × 109
/L (normal range, 3.5-9.5 × 109
/L), with 60.7%
lymphocytes (normal range, 20%-50%) and 8.3% eosinophils (normal
range, 0.4-8.0%). A diagnosis of BP was performed. Since her parents
refused systemic corticosteroid treatment, she was treated with oral
compound glycyrrhizin tablets (3.5 mg/kg/d) and topical moisturizing
cream. Two weeks later, the number of new blisters had obviously
decreased, and the erosions had re-epithelialized. Because the baby
responded well, the therapeutic schedule was continued without any
adjunctive therapy. Six weeks later, complete remission was observed
(Figure 1C). The anti-BP180 antibody titer dropped to 72.65 U/mL,
and the routine blood panel declined to normal. During the next
9 weeks, the baby remained healthy, and the anti-BP180 antibody
titer became normal. Subsequently, the medicine was tapered (1.7
mg/kg/d) and discontinued after 4 weeks. Presently, she has been
free of symptoms and rashes for 1 year.
Infantile BP (IBP) was first described in 1977,2 and since then, the
number of reported cases has increased. It has some unique characteristics, such as a peak prevalence at 3 to 4 months, uncommon mucosal
effects, and more generalized and favorable prognosis once appropriate
management is initiated. The pathogenesis is not clear, and the most
relevant theory is antigenic mimicking, in which a foreign antigen structurally resembling a self-antigen induces a cross-reactive antibody
response and influences immune system modulation.3
Compound glycyrrhizin tablets have a wide range of pharmacological actions, including anti-inflammatory, antiallergic, steroid, and
immunoregulation effects.4 They have been employed to treat immunoglobulin E (IgE)-induced allergic diseases, alopecia areata, vitiligo,
and psoriasis for several years. Previously, a 68-year-old male who
had erythrodermic psoriasis with BP was successfully treated by a
combination of methotrexate and compound glycyrrhizin tablets.5 To
the best of our knowledge, our report is the first instance of IBP cured
FIGURE 1 A, Extensive
erythematous lesions in annular
shapes and multiple tense
blisters or bullae on initial
treatment. B, The blisters were
noted particularly on the dorsum
of hands. C, Six weeks after the
therapy, complete remission was
achieved
Received: 25 February 2020 Revised: 2 May 2020 Accepted: 6 May 2020
DOI: 10.1111/dth.13550
Dermatologic Therapy. 2020;e13550. wileyonlinelibrary.com/journal/dth © 2020 Wiley Periodicals LLC. 1 of 3
https://doi.org/10.1111/dth.13550
by oral compound glycyrrhizin tablets. We hypothesize that the treatment might interfere with cells involved in the BP-specific immune
response, induce eosinophil apoptosis, and effectively downregulate
proinflammatory cytokines synthesized by T lymphocytes. Moreover,
the side effects of compound glycyrrhizin tablets are mild, and the
most common ones are edema and low-serum potassium levels, which
can be easily managed. In conclusion, we assume that this treatment
might be a safe and effective treatment strategy. Further reports are
needed to reach a consensus.
CONFLICT OF INTEREST
The authors declare no conflict of interest.
Kejia Xu1
Yi Wei2
Tingting Wang1
Li Ding1
Wei Li1
Lin Wang1
FIGURE 2 A, Subepidermal
fissures containing plenty of
eosinophils (hematoxylin and
eosin, ×100; bar = 100 m). B,C,
Linear deposition of
immunoglobulin G and
complement C3 along the
dermoepidermal junction (direct
immunofluorescence, ×200;
bar = 100 m)
FIGURE 3 Clinical timeline diagram for the time course of therapy
2 of 3 THERAPEUTIC HOTLINE: LETTER
Department of Dermatovenereology, West China Hospital, Sichuan
University, Chengdu, Sichuan Province, China
Department of Central Transportation, West China Hospital, Sichuan
University, Chengdu, Sichuan Province, China
Correspondence
Lin Wang, No. 37, Guo Xue Xiang, Wuhou District, Chengdu, Sichuan
Province, 610041, China.
Email: [email protected]
Kejia Xu and Yi Wei contributed equally to this article.
DOI 10.1111/dth.13550
REFERENCES
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Pediatrics. 1977;59(6):942-945.
3. Cinotti E, Douchet C, Perrot JL, Labeille B, Allombert-Blaise C,
Cambazard F. Bullous pemphigoid in an infant with milk protein allergy.
Br J Dermatol. 2013;169(1):191-192.
4. Shin YW, Bae EA, Lee B, et al. In vitro and in vivo antiallergic effects of
Glycyrrhiza glabra and its components. Planta Med. 2007;73(3):257-261.
5. Si X, Ge L, Xin H, Cao W, Sun X, Li W. Erythrodermic psoriasis with
bullous pemphigoid: combination treatment with methotrexate and
compound glycyrrhizin. Diagn Pathol. 2014;9:102.
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