active immunotherapy has been reported for centuries, di- rect contact ... cal paromomycin sulfate (twice/day) with intralesional meglumine antimonite for the ...
The interesting question remains, did the girl acquire the disease directly by frequently kissing her brother, which was reported by the mother and witnessed by the treating physician? CL is usually transmitted through the bite of an infected sandfly. Since deliberate scarification as a form of active immunotherapy has been reported for centuries, direct contact transmission is in principle possible, however, it is rarely reported [3, 4]. In fact, we did not find a single epidemiological study which has addressed whether or not leishmaniasis can be transmitted by direct contact. This problem is admittedly difficult to tackle, since most cases occur in endemic areas and the incubation period has such a great variability that even if direct transmission is considered clinically, a vector borne transmission can hardly be ruled out. As in our case, the skin around the lips often is macerated which would facilitate the entry of the parasite. Also, the specific location and long latency makes it conceivable that direct contact other than an insect bite transmitted the parasites. Clearly, more information is needed, but we see this correspondence as a starting point to stimulate future studies in that direction. j 1
Division of Dermatology, Tawam Hospital, P.O. Box 15258, Al Ain, Abu Dhabi, United Arab Emirates, Thomas Georg Berger, 2 Department of Histopathology, Tawam Hospital, Al Ain, Abu Dhabi, UAE
Lina HOUSSAMI1 Fadi HADDAD1 Hassan El TERAIFI2 Thomas G. BERGER1
1. Hepburn NC. Cutaneous leishmaniasis. Clin Exp Dermatol 2000; 25: 363-70. 2. Uthman MA, Satir, AA, Tabbara KS. Clinical and histopathological features of zoonotic cutaneous Leishmaniasis in Saudi Arabia. J Eur Acad Dermatol Venereol 2005; 19: 431-6. 3. Itani ZS, Moubayed AP, Huth F. Experimental inoculation of Leishmaniasis tropical from man to man. Arch Dermatol Res 1976; 256: 127-36. 4. Marsden PD, Almeida EA, Llanos-Cuentas EA, Costa JL, Megalhaes AV, Peterson NE, Cuba CC, Barreto AC. Leishmania braziliensis braziliensis infection of the nipple. Br Med J 1985; 290: 433-4. 5. Alrajhi AA, Ibrahim EA, De Vol EB, Khairat M, Faris RM, Maguire JH. Fluconazole for the treatment of cutaneous leishmaniasis caused by leishmania major. N Engl J Med 2002; 346: 891-5. 6. Shazad B, Abbaszadeh B, Khamesipour A. Comparison of topical paromomycin sulfate (twice/day) with intralesional meglumine antimonite for the treatment of cutaneous leishmaniasis caused by L. major. Eur J Dermatol 2005; 15: 85-7.
Brachioradial pruritus: report of a new case responding to gabapentin Brachioradial pruritus (BRP) is an enigmatic dermatosis characterized by persistent burning or stinging itch of the skin over the brachioradialis muscles. It was first reported in 1968 under the term «solar pruritus». Whereas chronic sun exposure has been considered to be the main cause of this disease [1,2], several recent reports suggest that BRP EJD, vol. 16, n° 3, May-June 2006
may be a manifestation of neuropathic pruritus, linked to spinal cord disease causing impingement of spinal nerves [3, 4]. The treatment of BRP is not well established. A new patient with BRP successfully treated with gabapentin is reported herein. A 54-year-old white man complained of severe, burning pruritus of two years’ duration localized to the neck, shoulders and the posterior-external aspect of both arms, spreading down to the elbows and the upper part of the forearms. Pruritus was intractable and worsened at night, waking the patient. Before visiting our department, the patient had consulted a dermatologist and tried various treatments, including oral antihistamines and creams, but these proved ineffective. Significant sun exposure was denied; moreover, the symptoms had no obvious relation with the summer season. Two skin biopsies taken from the arm had shown nonspecific findings consistent with a lesion induced by scratching. Routine laboratory workup (including CRP, transaminases, creatinin, IgE dosage) was within normal limits (except for slightly raised gamma-GT values). The patient’s past medical history was unremarkable (with the exception of surgery for biliary lithiasis). Traumatism to the spine or neck was not recalled. Clinically, the skin was unremarkable, save for the presence of sparse minute papular-follicular lesions due to scratching. On the basis of these findings, the diagnosis of BRP was made. X-ray examination showed sagittal rectitude of the spine with protrusive codiscarthrosis at the C5C6 and C6C7 levels. Because of the inefficacy of previous treartments, the patient was given, after informed consent, treatment with oral gabapentin, starting with 300 mg/d, increased to 3 × 400 mg/d over two months. This resulted in improvement of the pruritus, namely over the neck. Gabapentin was further increased to 3 × 600 mg/d; after two months, no further improvement was achieved, and the patient complained of diarrhea and sleepiness. After an additional two-month treatment, associated with an antipruritic cream containing 8% calamine and essential fatty acids, the patient reported significant (90%) improvement of pruritus; he could lead a normal life and sleep comfortably at night. Over the next two months he voluntarily decreased the treatment to 1200 and 600 mg/d before discontinuing it completely; pruritus developed again on the shoulders within a few days, although it was less severe. Gabapentin was increased to 1800 mg/d and the symptoms improved again. The patient was lost to further follow-up. BRP is rarely reported in the literature, but is probably under-recognized. Its pathogenesis remains unclear. Both solar radiation and cervical neuropathy have been incriminated. Very recently, familial cases have been reported, inherited probably in an X-linked, recessive pattern [5]. Treatment of BRP is difficult and not standardized. Capsaicin cream has reportedly given good results in some patients, but a controlled study failed to confirm this [1]. Sun protection, amitryptiline, acupuncture, cervical spine manipulation and application of ice packs or cold towels may produce relief. Gabapentin (C9H17NO2) is a novel agent that has been used successfully in the treatment of neuralgias (including postherpetic neuralgia and allodynia), neuropathies and various itching conditions, including uremic pruritus and BRP [6]. Its mechanism of action is unknown, although potentiation of inhibitory GABAergic transmission may be relevant. The effect of gabapentin on our patient’s pruritus was obvious, since its intensity was
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clearly related to treatment. This observation further highlights BRP as an expression of neuropathic pruritus, and upholds the usefulness of gabapentin for its treatment. j Dept. of Dermatology Ed. Herriot Hospital (Pav. R) 69437 Lyon cx 03, France
Jean KANITAKIS
1. Wallengren J. Brachioradial pruritus: a recurrent solar dermopathy. J Am Acad Dermatol 1998; 39: 803-6. 2. Wallengren J, Sundler F. Brachioradial pruritus is associated with a reduction in cutaneous innervation that normalizes during the symptom-free remissions. J Am Acad Dermatol 2005; 52: 142 -5. 3. Cohen AD, Masalha R, Medvedovsky E, Vardy DA. Brachioradial pruritus: a symptom of neuropathy. J Am Acad Dermatol 2003; 48: 825-8. 4. Goodkin R, Wingard E, Bernhard JD. Brachioradial pruritus: cervical spine disease and neurogenic/neuropathic pruritus. J Am Acad Dermatol 2003; 48: 521-4. 5. Wallengren J, Dahlenbeck K. Familial brachioradial pruritus. Br J Dermatol 2005; 153: 1016-8. 6. Winhoven S, Coulson I, Bottomley W. Brachioradial pruritus: response to treatment with gabapentin. Br J Dermatol 2004; 150: 786-7.
Livedo racemosa as a cutaneous manifestation of polycythemia vera In January of 2005, a 68-year-old woman visited our department complaining of discoloration of her foot. She had first noticed violaceous lesions of her toes 6 months previously. Then the skin change spread to her lower legs and became painful. She had been medicated for hypertension from 2002, and received intermittent phlebotomy as a therapy for polycythaemia vera (PV) which was diagnosed in 2003. Physical examination of both her lower legs revealed dark reddish erythema in a reticulate pattern (figure 1A). There were some infiltrated areas in the lesion. Clinical appearance of her lower legs was consistent with livedo racemosa (LR). Acrocyanosis was additionally seen in her bilateral toes (figure 1B). Raynaud’s phenomenon was only observed in the right big toe during the examination. Her face and both hands showed diffuse erythema and her conjunctiva were congested. Histological examination taken from the infiltrated lesion of LR showed eosinophilicstained vessels located in the subcutaneous fat tissues (figure 1C). High-power magnification disclosed a thrombus within a small vessel and secondary vasodilations around the vessel and in the dermis (figure 1D). Laboratory studies revealed the following values: haematocrit, 58% (normal 34-46); haemoglobin, 17.1 g/dL (normal 11.815.1); erythrocyte count, 871 × 104 /µL (normal 400-500); white blood cell count, 16,000 /µL (normal 4,000-9,600); platelet count, 55.4 × l04 /µL (normal 16-35); erythrocyte sedimentation rate, 1 mm/hr (normal 1-10); uric acid, 7.4 mg /dL (normal 2.6-5.8). Serum anticardiolipin antibodies, cryoglobulins and gammaglobulins were within normal limits. Taking clinical and histological findings into consideration, we supposed that LR in this patient was caused by the stasis condition of blood flow, complicated with PV. Treatment with aspirin, 100 mg daily, and sarpogrelate hydrochloride, 300 mg daily, was started. The pain was dramatically decreased, although the skin change was only slightly improved. One month later, a therapeutic
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A
B
C
D
Figure 1. Clinical features at first visit and histological findings of the biopsied specimen. (A) Dark reddish erythema in a reticulate pattern observed on her right lower leg. (B) Acrocyanosis in her bilateral toes. (C) Low-power micrograph illustrating the eosinophilic-stained vessels. (D) High-power magnification showing a thrombus within a small vessel and vasodilation around it.
phlebotomy of 400 ml was performed. This treatment dramatically resolved her skin symptoms including LR, Raynaud’s phenomenon, and acrocyanosis. Because there was an increased risk of hemorrhage, we immediately stopped the administration of sarpogrelate hydrochloride after the improvement of LR. Thereafter, she remained symptom-free for 4 months. Discussion
PV, a myeloproliferative disease, develops various clinical manifestations. Although it has been poorly mentioned in the literature, cutaneous lesions in PV display diverse morphology. Most frequent cutaneous changes are cyanosis and purple coloration of the face [1]. Aquagenic pruritus [2, 3] and erythromelalgia [4] have been also reported. These cutaneous manifestations are considered to be at least partly due to the microcirculatory disturbances resulting from the increased blood viscosity, secondary to an enlarged red cell volume, thrombocytosis, and occasional spontaneous platelet aggregation. LR is a symptomatic terminology, and can be observed in multiple pathological conditions. However, in our review of the literature published between 1975 and 2004, only 2 cases of PV with LR were found [1, 5]. The first case was a 75-year-old woman with LR and acrocyanosis in bilateral soles. Her cutaneous changes had completely resolved [5] with hydroxyurea in addition to aspirin and phlebotomy. EJD, vol. 16, n° 3, May-June 2006
