Yuka Okura et al. 2022Food protein-induced enterocolitis syndrome caused by scallop

Food protein-induced enterocolitis syndrome (FPIES) is a non-immunoglobulin E (IgE)-mediated food allergy that predominantly affects children.
The reports of FPIES have been increasing recently; however, the detailed pathophysiological mechanism of FPIES is yet to be well understood.
Although any solid food can cause FPIES reaction, there is regional variation in common triggers.
However, to the best of our knowledge, children with FPIES caused by scallop (scallop-FPIES) have not been described so far.
We report two patients with scallop-FPIES, which may develop in association with dietary habits.
Written informed consent for publication of these case reports was obtained from both patients' guardians.

食物タンパク質による腸炎症候群(FPIES)は、小児に好発する非IgE介在性食物アレルギーである。
近年、FPIESの報告は増加傾向にあるが、その詳細な病態生理は未だ十分に解明されていない。
FPIESは、あらゆる固形食が原因となり得ますが、その誘因には地域差があります。
しかし、我々の知る限り、ホタテに起因するFPIES(scallop-FPIES)を発症した小児はこれまで報告されていない。
我々は、食生活に関連して発症する可能性のあるscallop-FPIESの患者2名を報告する。
なお、これらの症例報告の公表にあたっては、両症例の保護者から書面による同意を得ている。

Patient 1 is a now 4-year-old Japanese boy.
He was admitted to nursery school at the age of 11 months.
Starting from 13 months of age, he had four episodes of vomiting several times that began 2.5 h after eating the dishes provided at his nursery school.
His mother noticed that scallops were commonly served before all episodes.
He visited our hospital at the age of 15 months.
His scallop-specific IgE was <0.10 kUA/L.
He underwent the oral food challenge (OFC) with scallop at 17 months of age.
After 165 min of ingestion of 9 g scallop, he lethargically vomited six times in six hours.
Based on the positive OFC result, he was diagnosed with scallop-FPIES and was advised to avoid scallops.
Oral food challenges with scallop performed at the age of 28 months and 40 months were positive, respectively.
He is currently avoiding scallops.

患者1は、現在4歳の日本人男児である。
生後11カ月で保育園に入園した。
生後13カ月から,保育園の給食を食べてから2時間半後に数回の嘔吐を4回繰り返した.
母親は,すべてのエピソードの前にホタテがよく食べられていることに気づいた。
生後15カ月に当院を受診した。
ホタテ貝特異的IgEは0.10 kUA/L以下であった.
生後17カ月にホタテ貝の経口食物負荷試験(OFC)を施行した。
9gのホタテを165分間摂取後,6時間以内に6回嗜眠嘔吐した.
OFCの陽性結果に基づいてscallop-FPIESと診断され、ホタテを避けるよう助言された。
生後28ヶ月と40ヶ月に実施したホタテの経口食物負荷試験もそれぞれ陽性であった。
現在、ホタテを避けている。

Patient 2 is a now 3-year-old Japanese girl.
She suffered from egg yolk (EY)-associated FPIES (EY-FPIES) from the age of 9 months.
She was admitted to nursery school at 16 months of age. An OFC with EY performed at the age of 26 months was negative.
However, 29 months of age onward, she had three episodes of vomiting that began 2 h after eating the dishes served at her nursery school.
Her mother noticed that scallops were commonly contained in the dishes at nursery school and visited our hospital.
Her scallop-specific IgE was <0.10 kUA/L.
She underwent OFC with scallop when she was 33 months old.
After 140 min of ingestion of 8 g scallop, she lethargically vomited 10 times within 5 h. Based on the positive OFC result, she was diagnosed with scallop-FPIES and has been instructed to avoid scallop.
She is currently avoiding scallops.

患者2は、現在3歳の日本人女児である。
生後9ヶ月から卵黄関連FPIES(EY-FPIES)に罹患した。
生後16ヶ月で保育園に入園した。生後26ヶ月に実施されたEYによるOFCは陰性であった。
しかし,生後29カ月以降,保育園で出された料理を食べてから2時間後に3回の嘔吐を認めた。
母親が保育園の食卓にホタテがよく出てくることに気づき,当院を受診した.
ホタテ貝特異的IgEは0.10 kUA/L以下であった。
生後33カ月にホタテ貝を用いたOFCを施行した。
ホタテ貝8gを140分後に摂取し,5時間以内に10回嗜眠嘔吐したため,OFC陽性からホタテFPIESと診断し,ホタテを避けるように指導した.
現在、彼女はホタテを避けている。

We reported two children with scallop-FPIES who were diagnosed according to the diagnostic criteria of the international guideline of FPIES. Both patients presented with repeated vomiting after eating scallop dishes served at nursery schools. According to the household survey of the Statistics Bureau of Japan, Hokkaido prefecture is one of the largest scallop consumption areas in Japan. In Sapporo, the capital city of Hokkaido, where the patients are living, model menus for nursery lunches are proposed by the Bureau for the Future of Children in Sapporo. These menus included 10 kinds of scallop dishes (10-12 g of scallop for each menu) in the past 6 months. Thus, scallop is one of the most popular shellfishes among wide age groups.

Scallops are marine bivalve mollusks in the taxonomic family Pectinidae. Although a small number of children have been reported as having bivalve mollusk-associated FPIES caused by short-necked clam, clam, mussel, and oyster, scallop-FPIES has not been reported in children. To date, only a 53-year-old man with scallop-FPIES who remained symptomatic for 20 years has been reported.

About 30%-40% of patients with FPIES react to multiple foods. In bivalve mollusk-associated FPIES, the combination of short-necked clam and squid, mussel, and cod, and mussel, clam, and sole have been reported. However, there is no report of bivalve mollusk-associated FPIES which developed after recovering from EY-FPIES. The mother of patient 2 noticed that delayed episodes of vomiting after the lunch were similar to those of EY-FPIES and visited our hospital, leading to early diagnosis. In recent years, given the fact that EY-FPIES is increasing in Japan, attention should be paid for FPIES to different foods, which may occur even after achievement of tolerance in EY-FPIES.

In conclusion, scallop-FPIES may develop in association with dietary habits. Bivalve mollusk-associated FPIES could occur after recovering from FPIES to any food and needs attention.

アップロード日:2022年6月14日
最終更新日:2022年6月20日
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