Angiolymphoid hyperplasia with eosinophilia (ALHE) is a rare condition affecting muscular arteries, typically of the head and neck. It was first described in 1969 by Wells and Whimster. They reported nine patients between the ages of 19 and 43, five women and four men, with single to multiple lesions in the head and neck region with blood eosinophillia in all patients and regional lymphadenopathy in four of nine patients. Previously it had been described as pseudo- or atypical pyogenic granuloma, subcutaneous angioblastic lymphoid hyperplasia with eosinophilia, and papular angioplasia[1, 5]. Initially thought to be related to Kimura's disease, a condition occurring in male Asians sharing some of the same clinical and histological features, ALHE is now considered a distinct entity[2, 5, 17, 18].
In approximately 50% of cases, a muscular artery is at the center of the lesion, as in our case report. Occasionally, in such cases, the differential diagnosis is traumatic pseudoaneurysm,  although in the latter condition, the lymphoid infiltrate and eosinophilic response is generally minimal.
Although the lymphoid infiltrate is a prominent component of ALHE, there are few data supporting a primarily lymphoproliferative process for this condition. In a study conducted by Jang et al, two of seven cases of ALHE showed positive result for PCR analysis of rearranged TCR-gene; however, all the cases were negative for heteroduplex-PCR. The conclusion of these authors was that the lymphoid reaction in ALHE is most likely reactive. However, more recently, Kempf et al  demonstrated T-cell gene rearrangements in 5 of 7 cases of ALHE and monoclonality was confirmed by automated high-resolution PCR fragment analysis. These authors raised the question of whether ALHE or a subset of ALHE represents either a true T-cell lymphoma of low-grade malignancy or a specific variant of reactive lymphoid hyperplasia . In our current case PCR analysis of the TCR-gene showed monoclonality between the peripheral T-cell lymphoma and the ALHE specimens.
Further support that ALHE is a monoclonal T-cell process is the finding of ALHE confirmed histologically in patients with synchronous or metachronous T-cell lymphoma. Adreae et al  reported a young girl with ALHE who subsequently developed peripheral T-cell lymphoma years after initial diagnosis. The current report demonstrates a second case, in which the ALHE developed months after the diagnosis of peripheral T-cell lymphoma. The finding of ALHE and peripheral T-cell lymphoma, and the demonstration of T cell gene rearrangements in both tissues, has not been previously documented in a single patient.
Although the current patient supports the concept that ALHE is, at least in some patients, reflected of a T-cell lymphoproliferative process, the finding of T-cell gene rearrangements indicated of monoclonality. According to Kempf et al , clonal lesional lymphocytes cannot be considered synonymous with malignant potency or overt malignancy, but it does shed a new light on the pathogenetic aspects of this disorder.
In conclusion, we report a case of ALHE developing after the diagnosis of peripheral T-cell lymphoma with T-cell gene rearrangements studies showing monoclonality in both the lymphoma and vascular lesions.