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On-line version ISSN 1678-9946
Rev. Inst. Med. trop. S. Paulo vol.49 no.6 São Paulo Nov./Dec. 2007
LETTER TO THE EDITOR
São Paulo, July 11, 2007
Two persons met one of us to ask for medical advice. On the basis of the clinical picture, we established the diagnosis of Ekbom syndrome, fundamentally related to skin lesions. We judged it relevant to communicate these occurrences, particularly for the reason that, in spite of our vast experience in dealing with clinical and assistance practice, we had not until now come into contact with patients in such situation.
There is no doubt that this subject deserves specific commentaries, as it connotes speculation, doubts, and a wide range of opinions.
The first account of a complex set of symptoms later known as the restless legs syndrome (anxietas tibiarum) was given in 1685 by Sir Thomas Brown, an English physician. The same investigator wrote a monograph under the title "Letter to a Friend" describing a condition observed in children of Languedoc, characterized by the periodic extrusion of stiff "black hairs" observed on their lumbar region. These children were called "morgellons" in the local language. Even now, such fundamental aspects of both conditions, as etiology, clinical picture and therapy, are the object of doubts. The symptoms of the restless legs syndrome are at first felt when the patient is at rest: an unpleasant sensation in the legs (rarely in the arms) accompanied by uncontrollable movements which become less intense when there is voluntary activity, to return when this activity ceases. Serious sleep disturbance results3,4.
Both syndromes: the restless legs (Wittmaak - Ekbom) and the morgellons are now identified as two distinct entities, but there are some authors who still consider them as two aspects of the same entity because neuropsychiatric symptoms considered to be analogous can be observed in both cases. The persistence of such doubts is an indication of scarcity of information available about the causes of symptoms that, for lack of proper investigation, are inexplicable as to their origin and persist interfering with people's normal daily activities.
A better fate was not in prospect for those with morgellons disease. They sense the migration of mysterious "parasites" under their skin. Sometimes skin fragments together with structures defined as "fibers" are extruded. Such cases are often diagnosed as delusory parasitosis2. As yet their etiology has not been defined.
In the consultation room these patients describe the feeling that they have migrating parasites and, as evidence to prove their case, they bring for examination small boxes containing fragments of apparently normal skin in which can sometimes be found filaments which are not identified as pieces of cloth. The frequency of this occurrence led medical students to coin the expression "matchbox syndrome". Atrocious suffering is associated with the presence of such "parasites". Sometimes the diagnostic hypothesis of "larva migrans" is accepted and anthelminthic treatment is prescribed.
On consulting a dermatologist, the patients are usually referred to a psychiatrist. The paucity of convincing information about their causes has led to the acceptance of the hypothesis that the symptoms of the morgellons disease are a manifestation of Lyme disease, even though no epidemiological or laboratory data confirm such diagnostic assumption. There is a tendency to judge the accounts of patients as illogical, resulting from neuropsychiatric problems which do not correspond to a true clinical picture1. In view of such hypothesis, prophylactic measures against this syndrome would be pointless. A tendency has been recently observed to oppose the notion that the Wittmaack-Ekbom syndrome is exclusively the result of a neuropsychiatric disorder; various medical research institutions are showing interest in identifying its possible causative agents, devising new laboratory research techniques and eventually finding a suitable therapeutic project.
Basic data concerning the two cases observed are specified below:
Case 1 - EMLZ, feminine gender, 53 years old, plastic artist, residing in Ilha Bela (São Paulo State, Brazil).
She feels that there are "animalcules" everywhere in her body, which have been producing violent itching since an epoch she cannot precisely remember. She has lost weight (36 kg) since then. Treatment: several non-specified anthelminthics and topic thiabendazole. Clinical examination revealed intense destructive lesions in finger- and toenails, no measures having been taken to ascertain their origin. The patient suffers from diabetes insipidus and revealed some episodes worth mentioning, according to our opinion: bat and snake bites, impetigo secondary to insect bites, pre-myocardial infarction and Stevens-Johnson syndrome due to the use of thiabendazole, and reaction due to the use of tetanus serum. Lives in a region where African snails are abundant.
Case 2 - MCJ, feminine gender, 48 years old, psychologist, residing in Buenos Aires, Argentina.
Starting six months ago: pruritus, locally hardened scales in the skin, keratosis, pain with biting sensation in various regions of the body, which worsen when she is in a theater, reading or in bed. Treatments used: ivermectin, which was not tolerated, and thiabendazole, which caused the extrusion of "black things" from the skin. Clinical examination revealed leg varices and little hematomas or angiomas.
The first patient brought with her very small fragments of nails and dry skin, believed by her to be parasites (Fig. 1). The second patient also brought skin fragments identified by her as parasites (Fig. 2). Both were submitted to histopathological examination showing fragments of epidermis.
In times past, when we were still lacking in some important information about the Ekbom syndrome, we came into contact with two situations similar to those described here. One of these persons went as far as to make a sketch of the human body showing the path of the peculiar "things". At that time all this sounded as nonsensical arguments.
Possibly, people suffering from Ekbom syndrome try to solve their problems by consulting different medical specialists. However, it is not the subject of only one area of medical practice; it would make sense to consider it to be a multidisciplinary subject requiring the adoption of specific therapeutic measures. We became involved with this subject as medical practitioners dedicated to the treatment of infectious and parasitic diseases. Supposedly, their condition is produced by "parasites". In cases of serious skin lesions, one could think of consulting a dermatologist, although other medical practitioners could be aware of the problem. Concerning Ekbom syndrome, we think that courses and other events on this subject should be recommended, as it is necessary to expand the available information about this disease.
We are not personally concerned about further in-depth investigation about the cases mentioned. Indeed, we had no intention to do that. We only intended to record the facts verified. Moreover, it must be mentioned that our explanation to the patients about their condition and their mistaken attitude produced disenchantment and diffidence, the continuity of our assistance being dispensed with.
As a detail, considering that speculations as to the cause of Ekbom syndrome associate it to a neuropsychiatric disorder, it is opportune to say that one of the persons involved identified herself as a psychologist, but could not see the incoherence of her situation.
Basically, we had the intention to emphasize the importance of this problem which deserves a deeper evaluation, particularly aiming at finding a desirable therapy.
Vicente AMATO NETO
Juliane Gomes de Paula AMATO
Valdir Sabbaga AMATO
Cláudio Santos FERREIRA
Laboratório de Investigação Médica/Parasitologia
do Hospital das Clínicas, da Faculdade de Medicina USP.
Laboratório de Parasitologia do Instituto de Medicina Tropical
de São Paulo/Universidade de São Paulo.
1. AREHART-TREICHEL, J. - Strange list of symptoms perplexes patients, doctors. Psychiat. News, 41: 19-20, 2006.
2. KOO, J. & LEE, C.S. - Delusions of parasitosis. A dermatologist's guide to diagnosis and treatment. Amer. J. clin. Derm., 2: 285-290, 2001.
3. O'KEEFFE, S.T. - Delusions of infestation during delirium in a patient with restless legs syndrome. J. geriatr. Psychiat. Neurol., 8: 120-122, 1995.
4. SAVELY, V.R.; LEITÃO, M.M. & STRICKER, R.B. - The mystery of Morgellons disease. Amer. J. clin. Derm., 7: 1-5, 2006.
Prof. Dr. Vicente Amato Neto
Laboratório de Investigação Médica, Parasitologia
Av. Dr. Enéas de Carvalho Aguiar 470
05403-000 São Paulo, SP, Brazil
Tel./fax: +55 11 3061- 7042