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Anisakiasis Involving the Oral Mucosa

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Archives of Craniofacial Surgery

Copyright © 2017 The Korean Cleft Palate-Craniofacial Association

This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/

licenses/by-nc/4.0/) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.

www.e-acfs.org pISSN 2287-1152 eISSN 2287-5603

261

Case Report

Arch Craniofac Surg Vol.18 No.4, 261-263 https://doi.org/10.7181/acfs.2017.18.4.261

INTRODUCTION

Anisakis is a genus of parasite with alife cycle involving fish and marine mammals. Anisakis is also known as whale worm, her- ring worm, and seal worm [1]. Anisakiasis is a syndromic disease characterized by acute or chronic symptoms thatoccurs when the patient ingests raw or uncooked infected seafood. Anisakiasis is typically classified into gastric, intestinal, and ectopic anisakiasis, though 95% of cases are gastric in nature [2]. Ectopic or extragas- trointestinal anisakiasis is very rare and includes cases involving the oral mucosa, pleural cavity, subcutaneous tissue, and parame- trium. Herein, we report a case of anisakiasis involving the oral cavity.

Anisakiasis Involving the Oral Mucosa

Anisakis is a parasite with life cycles involving fish and marine mammals. Human infec- tion, anisakiasis, occurs with the ingestion of raw infected seafood and usually presents with acute or chronic gastrointestinal symptoms from esophageal or gastric invasion.

We report a rare caseinvolving the oral cavity. A 39-year-old male presented with oral and sub-sternal pain of one day duration after eating raw cuttlefish. Physical examina- tion revealed areas of erythema and edema with a central white foreign particle on the labial and buccal mucosa. With microscopic field we could remove the foreign mate- rial from the lesions. The foreign material was confirmed to be Anisakis. Anisakis was also removed from the esophagus by esophagogastroduodenoscopy. The patient was discharged the following day without complication. Anisakiasis is frequently reported in Korea and Japan, countries where raw seafood ingestion is popular. The symptoms of acute anisakiasis include pain, nausea, and vomiting and usually begin 2–12 hours after ingestion. The differential diagnosis includes food poisoning, acute gastritis, and acute pancreatitis. A history of raw seafood ingestion is important to the diagnosis of anisa- kiasis. Treatment is complete removal of the Anisakis to relieve acute symptoms and prevent chronic granulomatous inflammation.

Keywords: Anisakiasis / Squid Sang Kyu Choi,

Cheol Keun Kim, Soon Heum Kim, Dong In Jo

Department of Plastic and Reconstructive Surgery, Chungju Hospital, Konkuk University School of Medicine, Chungju, Korea

CASE REPORT

A 39-year-old male presented to our clinic complaining of oral and sub-sternal pain for one day. Oral mucosal symptoms began 30 minute after the ingestion of raw cuttlefish, and sub-sternal pain began 8 hours after ingestion. Before consulting at our clinic, white thread-like foreign material had beenremoved from his oral mucosa at a local hospital. However, the patient’s pain persisted, prompting his presentation at our clinic. Physical examination revealed prominent erythematous and edematous lesions on the labial and buccal mucosa. Each lesion was noted to have a central- ly-placed small white foreign particle penetrating the mucosa (Figs. 1, 2). The patient’s vital signs were stable. Laboratory exami- nations (complete blood count, white cell differential count, chemistry, electrolyte, and C-reactive protein) were unremark- able. Based on the patient’s history and physical examination, we suspected anisakiasis. We consulted the department of internal medicine for evaluation of the patient’s sub-sternal pain. With the

Correspondence: Dong In Jo

Department of Plastic and Reconstructive Surgery, Konkuk University School of Medicine, 82 Gukwon-daero, Chungju 27376, Korea

E-mail: cozarmd@kku.ac.kr

Received Sep 3, 2017 / Revised Oct 17, 2017 / Accepted Oct 18, 2017

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Archives of Craniofacial Surgery Vol. 18, No. 4, 2017

www.e-acfs.org

262

aid of microscopic field, we were able to remove the foreign mate- rial from the lesions with a micro-forceps. The foreign material was threadlike, with a length of 3–8 mm and a width of <1 mm.

In all, 8 foreign bodies were removed and were confirmed to be Anisakis larvae. Consultants from the internal medicine depart- ment performed esophagogastroduodenoscopy (EGD) and re- moved Anisakis larvae from the center portion of the esophagus (Fig. 3). The patient was discharged the following day without complication.

DISCUSSION

Anisakiasis is a zoonotic parasitic infection that occurs as a result of

the ingestion of raw or uncooked seafood such as a sea eel, banded rudderfish, and cuttlefish. It occurs most frequently in Northeast Asia (Korea, China, and Japan) because of the popularity of raw seafood ingestion [3]. However, a recent increase in the ingestion of raw seafood in Western countries has led to an increase in the number of cases of anisakiasis reported in these areas [2].

Anisakiasis begins with the accidental human ingestion of thethird stage larvae of marine mammal parasitic nematodes be- longing to the family Anisakidae. Family members Anisakis sim- plex and Pseudoterranova decipiens have been associated with human infection [2]. Anisakis simplex is the most common caus- ative parasite in reports from Northwest Asia and is most com- monly associated with gastric anisakiasis. On the other hand, Pseudoterranova decipiens is more common in European coun- tries and is more commonly associated with intestinal anisakiasis.

Grossly, Anisakis simplex is smaller, thinner, and more brightly colored than Pseudoterranova decipiens [2]. Our patient was in- fected with Anisakis simplex. Usually, when Anisakis has been ingested, the third stage larvae invade the esophageal and gastric mucosa. Acute symptoms including nausea, vomiting, epigastric pain, and low grade fever typically begin 2–12 hours after the in- gestion of the infected seafood. In our case, larvae invaded the pa- tient’s oral mucosa immediately after ingestion of the infected cuttlefish, based on the onset of severe oral pain within 30 min- utes. Chronic symptoms of anisakiasi sare dyspepsia, diarrhea, Fig. 1. Gross photograph showing mucosal invasion by the Anisakis

larva (white arrows) seen as a white fragment in the lower labial mucosa.

Fig. 3. Gastroscopic finding showing a white Anisakis (red arrow) invading the esophageal mucosa.

Fig. 2. Gross photograph showing mucosal invasion by the Anisakis larva (white arrows) seen as a white fragment in the upper labial mucosa.

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Sang Kyu Choi et al. A case of oral anisakiasis

and epigastric pain lasting several weeks or months. Anaphylac- toid reactions to larval secretions have been rarely reported [2].

Because of the vague nature of the symptoms, the differential di- agnosis also includes food poisoning, acute gastritis, acute gastric ulcer, cancer, and other parasite infections. That is, severe pain oc- curred when sperm bags of a squid were stuck to the oral cavity after the ingestion of raw squid [4]. An eosinophilic leukocytosis is commonly found with laboratory testing; however, our patient’s laboratory results were normal. We suspect that this was because the infection was in a very acute phase. In patients with chronic anisakiasis such as intestinal anisakiasis, anti-anisakis IgG/IgA antibody titers are available [2]. But, first and foremost, obtaining a careful history regarding the ingestion of raw seafood is pivotal for establishing a diagnosis of anisakiasis [1-3].

Gastroscopic or surgical removal of the larvae provides defini- tive diagnosis and treatment at the same time. For direct visual- ization of the larvae in the gastric or esophagus mucosa, EGD should be used. Generally, the invading larvae will degenerate without developing into adult worms; however, this can lead to granuloma formation [5]. Therefore, complete removal of the lar- vae is the treatment of choice in patients with an acute infection.

The most direct method for removal is extraction of the larvae. If the larvae cannot be extracted because they are too deeply em- bedded in the mucosa, resection including the larvae and the mu- cosa is an alternative treatment.

Regarding prevention, public education about the risks of raw seafood ingestion is most important. The Food and Drug Ad- ministration recommends in United States that raw foods be blast frozen to ≤ –35℃ for 15 hours, be normally frozen to ≤ –20℃ for 7 days, or be heated to ≥ 70℃, since larvae often survive at tem- peratures as high as 50℃ [6]. Early evisceration of caught seafood can be recommended for preventing to migrate from the digestive tract of the captured organism to the muscle for several days.

However, early evisceration may not be adequate to prevent all

cases of anisakiasis because evisceration may not be performed completely. Also, in some fish infection of muscle is more promi- nent [7]. Discouraging the ingestion of raw seafood can also de- crease the incidence of anisakiasis.

Oral anisakiasis is a rare presentation of the parasitic infection anisakiasis. Severe oral pain can occur with one hour after the in- gestion of raw seafood. With the aid of microscopic field, this dis- ease can be detected and treated by a plastic surgeon.

CONFLICT OF INTEREST

No potential conflict of interest relevant to this article was reported.

PATIENT CONSENT

The patient provided written informed consent for the publica- tion and the use of their images.

REFERENCES

1. Hochberg NS, Hamer DH. Anisakidosis: perils of the deep. Clin In- fect Dis 2010;51:806-12.

2. Shimamura Y, Muwanwella N, Chandran S, Kandel G, Marcon N.

Common symptoms from an uncommon infection: gastrointestinal anisakiasis. Can J Gastroenterol Hepatol 2016;2016:5176502.

3. Im KI, Shin HJ, Kim BH, Moon SI. Gastric anisakiasis cases in Cheju- do, Korea. Korean J Parasitol 1995;33:179-86.

4. Shiraki Y, Kawai K, Kojima S, Suzuki Y, Ono K. Stinging in the oral cavity caused by ingestion of the sperm bags of a squid: a case report.

Pathol Int 2011;61:749-51.

5. Nakashima H, Akagi M, Miyabe S, Iwasawa H. Two unusual cases of a foreign body in the oral cavity caused by eating raw squid. Acta Oto- laryngol Suppl 1996;522:104-7.

6. Ahmed M, Ayoob F, Kesavan M, Gumaste V, Khalil A. Gastrointesti- nal anisakidosis: watch what you eat. Cureus 2016;8:e860.

7. Song SB, Lee SR, Chung HH, Han NS. Infection status of anisakid lar- vae in anchovies purchased from local fishery market near southern and eastern sea in Korea. Korean J Parasitol 1995;33:95-9.

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