Wednesday, January 24, 2007

Mystery from Micronesia

We received this note from a physician in Hawaii. Your thoughts will be appreciated.

"Two years ago, I was shown photos of a severe skin condition which afflicted more than 80 persons on the tiny atoll of Satowan in Chuuk. Now, more than 100 persons on Satowan are affected and about 10 on the neighboring atoll of Lekinioch.

Affected persons develop thick, pink plaques, usually on the arms and legs – this appearance resulted in the nickname “Spam.” While the condition is not life-threatening, the people with the condition are sometimes ostracized and made to feel shame and embarrassment. Anecdotally, the plaques are more common in those who work or play outdoors (particularly taro farmers) and may sometimes occur following skin trauma.

The only treatment used on the plaques has been surgical debridement. Dr. Bosco Buliche who practices in Weno, the capital of Chuuk, has seen and photographed cases on Satowan. He has posted 5 cases on the Pacific Island Healthcare Project website (through TAMC), including one with biopsy results, which showed only nonspecific inflammation – we could transport only a fixed specimen, unfortunately. Opinions from various practitioners on the cause of the condition have run the gamut from “island psoriasis” to mycobacterial infection.

So, two years later, there is still no answer. It would be wonderful to find someone with the interest and resources to investigate the problem. Suggestions will be most welcome."

Clinical Photos:



7 comments:

  1. This is not my area of expertise (I am not sure what is), but I suspect the answer here would be biopsies and perhaps culture of tissue. This would be an ideal project for a dermatology resident or even a medical student (from Hawaii?) The specimens could be brought back to U.H. or perhaps sent to a lab in the U.S. Deep fungal infection or atypical AFB.
    Here is a reference from PubMed:
    Australas J Dermatol. 1998 Aug;39(3):173-6.

    Mycobacterium marinum: chronic and extensive infections of the lower limbs in South Pacific islanders.

    Lee MW, Brenan J.

    Department of Dermatology, St Vincent's Hospital, Melbourne, Victoria,
    Australia.

    We report three adult cases of very chronic, extensive infection of the lower limbs due to Mycobacterium marinum. The patients were from South Pacific islands and, clinically, the widespread warty plaques resembled chromomycosis. One was associated with severe lymphoedema. All three patients gave a history of at least 20 years duration. The patients were otherwise well and not immunologically compromised. In all cases, the organism was identified on tissue cultures and was not seen on histopathology. The mycobacteria were sensitive to most antibiotics tested in vitro. The patients were treated with a combination of rifampicin and cotrimoxazole with good results.

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  2. Robert RudolphJanuary 25, 2007

    1. A mycobacterial infection
    2. sarcoid
    3. drug
    4. reaction to coral or something in the ocean
    5. reaction to some ingested worm from fish
    6. I have not a clue

    More biopsies are needed, along with PCR and/or bacterial cultures (for
    high and low temp organisms)

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  3. Clinically this is the delayed hypersensitivity reaction of mycobacterial, atypical mycobacterial or deep fungal infection. From John Brennan and Michael Lee's description this is indeed an atypical mycobacterial infection and Mycobacterium marinum or a variant would fit the bill.I can try to pass the images on to them for their opinion.

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  4. I agree with the earlier comments. We have seen similar cases in Alor Setar, Kedah, Malaysia where there are lots of paddy fields. Clinically they could go for either atypical mycobacterium infection or deep fungal infection esp chromomycosis.
    A deep incisional biopsy should be repeated for HPE and culture for mycobacterium infection and fungal infection as well.

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  5. I agree with my colleagues that the history and the picture is suggestive of chronic infection,possibly atypical mycobacterium infection.I have not seen such strange picture in my country.Further biopsies and other investigations are essential.Also epideomological study is urgently needed.
    khalifa sharquie

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  6. Although I am in Chennai in India at a conference ,such is the power of the Internet that I was able to contact Michael Lee in Australia and receive the following reply.

    Our `large` series of 3 cases were patients from South Pacific. The clinical photographs that you posted are identical to ours`.

    The organism is very difficult to grow and thus sensitivity to antibiotics may not be determined in laboratory.However fortunately there are reported cases including one of ours that responded to Minocycline or Cotrimoxazole as single agent.Needless to say long course will be required.The 2nd line is the traditional antiMb- Rifampicin and Ethambutol etc

    The extensive `psoriasiform` disseminated type which was the gist our paper is obviously not amenable to surgery.

    This organism grow optimally at 32C and not 37C.Thus the infection is confined to the skin.On the basis of this, heat therapy has been reported to be successful.However I cannot recall the details of how it is done from that paper. I hope this helps.

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  7. I had patient with similar type of lesion over the neck.culture didn't show any thing.I started her on minocycline as single drug she responded well.
    Dr.A.M.Jaaraaman

    ReplyDelete

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