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PubMed2002

Multiple basaloid follicular hamartomas associated with acrochordons, seborrhoeic keratoses and chondrosarcoma.

Basaloid follicular hamartoma is an uncommon neoplasm with distinctive histopathological findings. It presents as four distinctive clinical forms: a solitary papule, a localized plaque of alopecia, a localized linear and unilateral type, and generalized papules with associated alopecia and myasthenia gravis. Histologically, basaloid follicular hamartomas are characterized by thin branching strands and thick cords of basaloid or squamoid cells extending from a follicle into a loose, fibrillar, fibrocytic or mucinous connective tissue stroma. We report a case of long-standing, generalized basaloid follicular hamartomas associated with acrochordons, seborrhoeic keratoses, and a history of chondrosarcoma. In general, solitary tumours are sporadic; multiple tumours are inherited and frequently associated with a syndrome. Further surveillance is warranted to determine if the association of multiple basaloid follicular hamartomas and chondrosarcoma constitutes an inherited syndrome.

Acrodermatitis↗
PubMed2000

Gianotti-Crosti syndrome in two unrelated adults.

Two unrelated adult patients are described who both developed an exanthem with the characteristic morphology and distribution of the Gianotti-Crosti syndrome. The literature of adult cases of Gianotti-Crosti syndrome is reviewed with the suggestion that the syndrome may not be as uncommon as previously supposed.

Acrodermatitis↗
PubMed2003

Characterization of the vls antigenic variation loci of the Lyme disease spirochaetes Borrelia garinii Ip90 and Borrelia afzelii ACAI.

The vls locus of Borrelia burgdorferi B31 consists of 15 silent cassettes and one expression site (vlsE), and the presence of the encoding plasmid lp28-1 correlates with high infectivity. Recombination between the expression cassette and silent cassettes occurs in vivo, and this process may enable B. burgdorferi to evade the immune response. To determine the characteristics of the vls loci in other Borrelia strains, we have cloned and characterized the vls silent cassette loci of Borrelia garinii Ip90 and Borrelia afzelii ACAI, consisting of 11 vls silent cassettes and 14 vls silent cassettes respectively. The silent cassettes share 90-97% nucleotide sequence identity with one another within the Ip90 vls locus and 84-91% within the ACAI vls locus. In both organisms, the silent cassettes resemble the B31 Vls sequences in overall amino acid similarity (50-65%) and in the presence of six variable regions interspersed between six relatively invariant regions. The vlsE expression sites of these two strains have not been isolated, but transcripts of vlsE were detected by reverse transcriptase-polymerase chain reaction for both Ip90 and ACAI. In addition, the occurrence of sequence variation within the vlsE cassette region of these transcripts was verified. This study indicates that the vls loci present in B. garinii Ip90 and B. afzelii ACAI have characteristics similar to those found in B. burgdorferi B31.

Acrodermatitis↗
PubMed2001

Bullous lesions in Bazex syndrome and successful treatment with oral psoralen phototherapy.

A 59-year-old man presented with a psoriasiform dermatitis with associated bullae and destructive nail dystrophy of the hands and feet. He had lost 10 kg weight over 6 months and a mass in the neck was noted. He was provisionally diagnosed with Bazex syndrome (acrokeratosis paraneoplastica) and subsequent investigations revealed a squamous cell carcinoma in the right piriform fossa. His skin lesions were treated with oral psoralen and ultraviolet A phototherapy and this cleared the cutaneous changes, but the nail changes have persisted at 18 months follow up.

Acrodermatitis↗
PubMed2003

Truncal lesions do not exclude a diagnosis of Gianotti-Crosti syndrome.

A boy aged 2 years and 10 months had a pruritic eruption and low-grade fever for 1 week. Examination revealed discrete erythematous papules and papulovesicles on his buttocks and extensor surfaces of four limbs. Several papules were also present on his back. No lymphadenopathy and no hepatosplenomegaly were noted. Investigations failed to identify a single viral aetiology. Topical calamine lotion and a sedating oral antihistamine to be taken at night were prescribed. The truncal lesions subsided in 3 weeks. Complete remission of all lesions was seen after 6 weeks. Although most patients with Gianotti-Crosti syndrome only have the typical acrally distributed eruption, additional truncal lesions, if few in number, do not exclude the diagnosis.

Acrodermatitis↗
PubMedDate not supplied

Psoriasiform acral dermatitis: a peculiar clinical presentation of psoriasis in children.

Recently an unusual chronic dermatosis, considered a new clinical entity and closely resembling psoriasis, has been described in the literature under the term psoriasiform acral dermatitis (PAD). It is characterized by cutaneous involvement of the digits without nail dystrophy. We describe three young patients, ages 6 to 8 years, in whom this condition was associated with psoriasis. Two children were affected by psoriasis vulgaris, while the third had a palmoplantar psoriasis. All laboratory investigations performed were within normal limits. Skin biopsy specimens taken from the fingers of two patients revealed the pathologic features of subacute spongiotic dermatitis. Histologic examination of a biopsy specimen taken from an erythematous squamous patch confirmed the clinical diagnosis of psoriasis in two patients. The dermatitis showed a fluctuating course in all three patients, with only a moderate to strong improvement with therapy with calcipotriol ointment (50 microg/g). During follow-up, two patients experienced marked spontaneous, persistent improvement, while the disease slightly worsened in the third. The children had features similar to those described in PAD, but were also suffering from psoriasis. Whether PAD is a distinctive entity or just a clinical manifestation of psoriasis in children is still an open question. We strongly believe this latter hypothesis, although further studies are needed to confirm it.

Acrodermatitis↗
PubMedDate not supplied

Gianotti-Crosti syndrome in an infant following immunization.

Gianotti-Crosti syndrome (GCS) is a distinctive, self-limiting acropapular or papulovesicular eruption. Typical clinical presentation is that of a monomorphic, nonpruritic eruption most commonly involving the face, neck, buttocks, and extremities. It tends to occur in childhood, though infrequently it has been reported in adults. We report GCS occurring in an infant following immunization.

Acrodermatitis↗
PubMedDate not supplied

A prospective case control study of the association of Gianotti-Crosti syndrome with human herpesvirus 6 and human herpesvirus 7 infections.

Gianotti-Crosti syndrome (GCS) is known to be associated with hepatitis B and Epstein-Barr virus (EBV) infections. Apart from a single case report based on serology alone, there are no published data on an association between GCS and human herpesvirus 6 (HHV-6) and human herpesvirus 7 (HHV-7) primary infections or reactivations. Our aim was to investigate the association between GCS and HHV-6 and HHV-7 infections. Ten patients diagnosed with GCS at a primary care practice over an 18-month period were recruited. Controls were age- and sex-matched patients with unrelated symptoms requiring venepuncture for other indications. Blood specimens were collected from patients and controls at presentation, and from patients 4 weeks later. Virologic evidence of HHV-6 and HHV-7 infection was sought in peripheral blood leukocytes and plasma using polymerase chain reaction (PCR) for viral DNA, reverse transcriptase polymerase chain reaction (RT-PCR) for HHV-6 U91 mRNA transcripts, and serology. Serology for EBV and hepatitis B virus was done. In contrast to the 10 controls, 2 patients (both infants) with clinically diagnosed GCS had evidence of active HHV-6 infection. This was demonstrated by detection of viral DNA in the absence of antibody in the acute plasma specimens and HHV-6 DNA viral loads of more than 5.3 log10 genome copies/5 microl in the whole blood specimens, a profile previously shown to be diagnostic of recent primary HHV-6 infection. None of the patients had evidence of recent EBV or hepatitis B infection. We conclude that primary HHV-6 infection may be associated with GCS in some infants.

Acrodermatitis↗
PubMed1999

Bazex syndrome mimicking a primary autoimmune bullous disorder.

Bazex syndrome is a paraneoplastic condition that is most frequently associated with squamous cell carcinoma of the upper aerodigestive tract. The lesions affect acral areas of the skin, including hands, feet, ears, nose, and, to a lesser extent, elbows and knees. Lesions mimic psoriasis and dermatitis. Paronychia and nail dystrophy are frequent. Bullous lesions have been reported only rarely. We report a patient with Bazex syndrome with predominantly bullous lesions that mimicked a primary autoimmune bullous disorder.

Acrodermatitis↗
PubMedDate not supplied

[Gianotti-Crosti syndrome in an infant following immunization].

Gianotti-Crosti syndrome (GCS), first described by F. Gianotti in 1955, was originally reported to be associated with hepatitis B virus infection in children. The typical clinical picture allows diagnosis of GCS at first glance. The pathogenesis is still unknown. Besides HBV infection, a large number of infectious agents, mostly viruses, have been described as a trigger of GCS. Here we report the occurrence of GCS in an infant five days after the fourth immunization against poliomyelitis, DTPa, Hib, hepatitis B and Streptococcus pneumoniae. Our data and review of the literature suggest that GCS is rarely associated with immunizations, especially when performed with inactivated vaccines.

Acrodermatitis↗