Unilateral Distribution Research Articles

Hypohidrosis and metal allergy: Trigger factors for unilateral lichen planus.

Unilateral lichen planus (LP) is a rare clinical variant of LP. The etiology of unilateral LP has not been clarified, although various causes have been reported because of the characteristic distribution. We focused on the contribution of metal allergy and sweating in the development of unilateral LP in this case. To confirm the presence of metal allergy, patch tests with metal allergens were performed. To investigate the cause of the unilateral distribution in unilateral LP, the function of sweating and sweat leaking in the dermis was assessed by a thermoregulatory sweat test and immunohistochemical staining of dermcidin. The patch tests with SnCl2 , H2 PtCl6 , ZnCl2 and MnCl2 were positive. The thermoregulatory sweat test using the starch-iodine method (Minor test) with sweating provoked by heat stimulation revealed hypohidrosis of the affected area, whereas no skin lesions were observed on the dorsal hand and wrist where sweating was normal. Histopathological examination showed keratin plugging of the acrosyringium and lymphocytic infiltrations in the papillary and subpapillary dermis around the intraepidermal and intradermal eccrine duct in the affected area. The immunohistochemical staining of dermcidin confirmed sweat leakage in the subpapillary dermis in the affected area. The symptoms refractory to the topical steroids were markedly improved after removal of the dental metals. The coexistence of metal allergy and sweat leakage in the hypohidrotic area may be involved in the development of unilateral LP.

Evaluation of the frequency and characteristics of hypercementosis in the turkish population with cone-beam computed tomography.

The aim of this study was to evaluate the frequency and characteristics of hypercementosis in the Turkish population using cone-beam computed tomography (CBCT). A retrospective study was performed using CBCT (I-CAT Vision TM Imaging Science International) in 1263 patients admitted to the Faculty of Dentistry at the University of Dicle (Diyarbakir, Turkey) between January 2013 and January 2015. A total of 1152 patient and 29,606 teeth were evaluated with CBCT. The age, gender, location (left vs. right side, maxillary vs. mandibular teeth, incisors, premolars, and molars), and missing teeth were recorded for all patients. The Pearson Chi-squared test was used to determine the potential differences. Hypercementosis was observed with a frequency of 2.4%. Of these, 18 of 28 (64.3%) had only 1 tooth with hypercementosis and 10 (35.7%) had more than 1 tooth. It was identified in 42 of the 29,606 teeth examined (0.14%). The lesion was found in 2.47% (n = 14) of females and 2.38% (n = 14) of males. The lesion was detected in 28 molars and 14 premolars, but hypercementosis was not detected in incisors. Of the 42 teeth, 12 (28.6%) maxillary and 30 (71.4%) mandibular teeth were associated with hypercementosis. Of the 28 cases, 20 (59.0%) were unilateral and 8 (41.0%) were bilateral. Of the 20 unilateral cases, 12 (69.6%) were on the right and 8 (30.4%) were on the left side. In our study, the prevalence of hypercementosis has been found 2.4% in the Turkish population. Besides, the lesion has been found more in the mandible than the maxilla, in the molars than the premolars and in the unilateral distribution than the bilateral distribution.

Surgical management of venous compression causing trigeminal neuralgia

Venous compression is a rare cause of trigeminal neuralgia (TN). Vascular decompression of the trigeminal nerve root, as the only etiopathogenetic treatment of trigeminal neuralgia, is a well-known procedure that is extensively used at various neurosurgical clinics. As the number of interventions for TN increases, the absolute number of surgeries for eliminating venous compression of the trigeminal nerve also grows. Five hundred TN patients underwent surgery at the Neurosurgical Institute in the period from 2000 to 2015. The diagnosis was made based on the criteria of the International Classification of Headache Disorders, the 3rd edition, (ICHD-3) and the Burchiel classification. The study included 211 males and 289 females. The median age was 57 years. All patients had typical TN pain with a unilateral distribution. Two or more branches were affected in 90 patients; one branch was predominantly affected in 10 patients. The median disease duration was 16 months. The median visual analog scale (VAS) score was 8 (very severe persistent pain). In all cases, the patients underwent surgery using the retrosigmoid suboccipital approach. The median postoperative follow-up was 36 months. The aim of this work is to summarize the first experience of the Neurosurgical Institute in surgical treatment for venous compression of the Vth nerve root and suggest methods increasing the efficacy of vascular decompression in patients with this disease.

Progressive Cribriform and Zosteriform Hyperpigmentation along with Vitiligo

Background: Progressive cribriform and zosteriform hyperpigmentation (PCZH) is a disorder of pigmentation. Although several cases of PCZH have been reported, cases that associated with vitiligo have not been published in the past. Aim: We report the case to reveal the interesting mosaicism reflecting on the skin. Case Presentation: This case presents a phenomenon of the coexistence of hyperpigmentation and depigmentation arranged in unilateral and symmetric distribution in one patient. Conclusion: The aetiology of the pigmental disorders is still unknown. The linear nature of the pigmented bands probably reflects the clonal migration and proliferation of embryonic melanoblasts, so somatic mosaicism that develops during embryogenesis appears to be the underlying aetiology, which is leading to proliferation and migration of two mixed populations of melanocytes with different potential for pigment production.

Prevention of Herpes Zoster and its complications: From clinical evidence to real life experience

ABSTRACTHerpes zoster (HZ) is an acute viral illness characterized by a vesicular rash with unilateral distribution, which can also result in severe complications such as post-herpetic neuralgia (PHN), ophthalmic zoster, stroke or other neurological complications. The estimate incidence in Europe ranges between 2.0 and 4.6 cases per 1,000 person-years, with a sharp increase in >50 year-old subjects. Currently, treatment options for HZ are only partially effective in limiting the acute phase, while the management of complications is complex and often unsatisfactory. The total burden of the disease and the high costs related to its diagnostic and therapeutic management led researchers to develop a new preventive approach through a live attenuated virus vaccine. The currently available vaccine, with a high antigen content, is safe, well tolerated and reduces the incidence of HZ, PHN and the burden of illness. Several countries have introduced this vaccination, albeit with different recommendations and methods of financing. Taking into account the barriers to this immunization registered in some areas (difficulty of vaccine distribution, lack of physician recommendations, the cost of vaccine for patients, etc.), this group of Italian experts advocate that a common strategy able to guarantee a good compliance with this vaccination should be implemented. The same group addresses some practical questions concerning the use of zoster vaccine.

Prevention of herpes zoster and its complications: from the clinic to the real-life experience with the vaccine.

The erpes zoster is an acute viral illness characterized by a vesicular rash of unilateral distribution, which can eventually cause severe complications, such as post-herpetic neuralgia, ophthalmic zoster, stroke or other neurological complications. In Europe, an incidence of between 2.0 and 4.6 cases per 1000 person-years is estimated, with an increase after 50 years of age. Currently, the therapeutic options for are only partially effective in limiting the acute phase, while the management of complications is frequently complex and not satisfactory. The overall burden of the disease and the elevated costs associated with diagnosis and clinical and therapeutic management led to the development of a new preventive approach through a live attenuated virus vaccine. The vaccine now available decreases the incidence of the disease, post-herpetic neuralgia and the burden of illness. Moreover, the vaccine is safe and well tolerated and it seems to confer long-term protection. Based on the clinical results and evidence provided by the Health Technology Assessment, several countries introduced immunization although with different recommendations and methods of funding.

Bilateral Total Hip Arthroplasty as a Treatment for Ollier’s Disease of the Proximal Femur: a Case Report

Ollier’s disease is very rare condition that is characterized by the presence of multiple cartilaginous tumors in the long tubular bones which can lead to severe deformities and pathological fractures at early age. More commonly the lesions have a unilateral distribution. We report a case of an adult patient with medical history of Ollier’s disease involving both hips treated with a 2-stage bilateral total hip arthroplasty (THA) using a cement less ace tabular cup and a short cement less femoral stem, achieving excellent clinical and radiographic results.

Linear Psoriasis- A rare case report

Linear psoriasis is a rare form of disease which is very difficult to differentiate from inflammatory linear verrucous epidermal nevus. Sometimes clinical history, physical examination and histopathology analysis may not be sufficient to confirm the diagnosis. We report a case of25 year-old male presented with a linear plaque covered with silvery scales on left upper extremity extending from tip of the index to mid forearm for last 2 years. A skin biopsy was consistent with psoriasis, and the unilateral distribution in a linear pattern led to a diagnosis of linear psoriasis, which is a rare variant of psoriasis. Although histopathologically it can be difficult to distinguish from inflammatory linear verrucous epidermal nevus (ILVEN), linear psoriasis presents in adulthood and responds to conventional topical antipsoriatic therapies.

Lichen aureus: clinicopathological features in a Chinese series.

Lichen aureus (LA) is a rare variant of pigmented purpuric dermatosis. There are few Chinese cases of LA documented in the literature in English. We aimed to investigate the clinical and histopathological characteristics of LA in a series of Chinese patients. This was a retrospective monocentric study conducted in the Department of Dermatology, Peking Union Medical College Hospital. A total of 25 patients were included in the study; 15 male and 10 female patients. Unilateral distribution of the lesions was found in 88.0% of the patients, while bilateral distribution was found in 12.0%. A segmental distribution was observed in 12 patients. The most commonly involved areas were the upper extremities (n=10; 40.0%) and lower extremities (n=10; 40.0%). Fifteen cases (60%) presented with an inflammatory reaction pattern of a band-like lymphocytic infiltrate. Periadnexal inflammation and perineural inflammation were found in 12 patients and five patients, respectively. A segmental pattern of distribution of the lesion is very common in LA. Both upper limbs and lower limbs may be equally affected. Band-like lymphocytic infiltrate is the most common inflammatory reaction pattern. Periadnexal inflammation and perineural inflammation are relatively common in LA.

Anatomical Study of the Posterior Mandible: Lateral Lingual Foramina in Cone Beam Computed Tomography.

To evaluate the lateral lingual foramen (LLF), an aperture located in the posterior lingual mandible area, through cone beam computed tomography (CBCT) images. To evaluate the LLF presence through 100 CBCT images. Presence of LLF was compared between sex and age. Width and height were determined and the distance from the LLF to the mental foramen (LLF-MF) and that of the LLF to the lower mandible border (LLF-LBM) were obtained. LLF was observed in 39% of the examinations and 52 LLF were found and the most prevalent area of occurrence was at the second premolar area. The mean width was 1.26 ± 0.57 mm and the mean height was 1.82 ± 1.12 mm. The mean values of LLF-MF and LLF-LBM distances were 12.74 ± 7.7 and 5.82 ± 2.78 mm, respectively. LLF are asymmetrical structures with bilateral or unilateral distribution, located in the second premolar region close to the LBM.

Association Between Migraine and Diabetes in Pregnancy

Background: Migraine ranks as the eighth most disabling condition and one of the most common causes of headache in Pakistan. Objectives: In this study, we aimed to determine the association between migraine and diabetes in pregnant women. Patients and Methods: This cross-sectional study recruited 498 pregnant women, grouped into pregnant without diabetes (n = 300) and pregnant with diabetes (n = 198) according to the International Association of the Diabetes and Pregnancy Study criteria. Seventy-five women with known migraine were also recruited as positive controls. After confirming that the study subjects had headache at least once a month, the researcher filled out a comprehensive form based on the International Classification of Headache Disorders version II. Migraine disability score was used to assess severity. Fasting blood glucose levels were measured by using the enzymatic method. Data were presented as meanSD and frequencies, where applicable. Chi-square test and Spearman correlation test were performed. A P value of <0.05 was considered significant. Results: The headache prevalence during pregnancy was 69% in the women with diabetes and 64% in the women without diabetes. A positive unilateral distribution was observed in 51% of the cases and 36.3% of the controls (P< 0.01). However, 94.7% of the pregnant cohort reported not having experienced aura. Of the pregnant women with diabetes, 19% fell within the mild to moderate disability score as compared with the 10.3% of the pregnant women without diabetes (P < 0.01). High fasting blood glucose levels showed a significant association with headache scores (r = 0.144; P < 0.01). Conclusions: Headaches, particularly migraine without aura, are a common occurrence in pregnancy in our population. Migraine severity is positively associated with high blood glucose levels.

Hypodontia and ovarian cancer: A systematic review.

Hypodontia can be defined as the non-formation of one or more teeth during the developmental period. Mutation in several genes related to tooth formation has previously been correlated with cancer. Regarding the ovarian cancer, there are few studies that associate the presence of hypodontia with ovarian cancer. A systematic literature search was performed in PubMed and Scopus. In total, 385 patients were included in this study. Control group was present in 3 out of 4 studies (340 patients). Hypodontia was present in 56 out of 290 patients (incidence of 19.3%). Only in 2 out of 4 studies, the number of missing teeth was mentioned (47 teeth), while the majority of them were either maxillary second premolars or maxillary lateral incisors. Unilateral distribution of the missing teeth was present in 28 out of 46 patients, while bilateral distribution of the missing teeth was present in 18 out of 46 patients. The presence of ovarian cancer in the family medical history occurred in 12 out of 33 patients. Only 1 out of 4 studies examined the presence of genes with mutations in the included patients. Based on our findings, the lack of clinical studies was the principal obstacle to clarify the possible predictive value of hypodontia in the early prediction of patients with higher risk of ovarian cancer.

Unilateral proteus syndrome in a neonate: A very rare presentation

Proteus syndrome (PS) is a rare sporadic disorder with postnatal asymmetric overgrowth from any of the three germinal layers. The tissue overgrowth may present at birth but becomes more conspicuous with the progression of age. Though it apparently can affect any tissue, it commonly involves skin, bones, and central nervous system. The PS is difficult to diagnose as any part of the body is affected along with several complications. To overcome the diagnostic confusion, there are three general mandatory criteria with at least one of the three category signs designated as specific criteria according to First National Conference on PS. We diagnosed our case by connective tissue nevus in sole with three general criteria. Huge cerebriform nevus sebaceous as epidermal nevus was not found previously. A few neonatal PS cases were reported. All the lesions were unilaterally distributed. Our case was neonatal PS with unilateral distribution. We report this case because of its rarity.

Extensive unilateral nevus comedonicus without genetic abnormality

Nevus comedonicus is considered a genodermatosis characterized by the presence of multiple groups of dilated pilosebaceous orifices filled with black keratin plugs, with sharply unilateral distribution mostly on the face, neck, trunk, upper arms. Lesions can appear at any age, frequently before the age of 10 years, but they are usually present at birth. We present a 2.7-year-old girl with a very severe form of nevus comedonicus. She exhibited lesions located initially at the left side of the body with a linear characteristic, following Blascko lines T1/T2, T5, T7, S1 /S2, but progressively developed lesions on the right side of the scalp and left gluteal area.

Pathogenesis, Surgical Treatment, and Cure for SUNCT Syndrome

Short-lasting unilateral neuralgiform headache attacks with conjunctival injection and tearing (SUNCT) are characterized by attacks of moderate to severe stabbing pain, strictly unilateral, with periorbital or temporal distribution, associated with cranial autonomic symptoms, such as lacrimation and redness of the ipsilateral eye. To obtain mechanistic insights into the pathogenesis of SUNCT syndrome, more than 800 cases treated in our institution during the last 7 years were retrospectively reviewed. Two patients showed typical autonomic symptoms of SUNCT. Magnetic resonance imaging suggested potential compression of the trigeminal nerve by the intracranial artery in these cases and complete remission was achieved by microvascular decompression. Microvascular decompression provides an appropriate therapeutic choice if vascular compression of the trigeminal nerve is identified. From our 2 cases, we propose that, in some cases of SUNCT diagnosed previously, characteristic symptoms were induced by compression of the side surface of the first branch of the trigeminal nerve at the root exit zone by the intracranial artery.

Combination therapy with 308-nm excimer laser, topical tacrolimus, and short-term systemic corticosteroids for segmental vitiligo: A retrospective study of 159 patients

Segmental vitiligo (SV) is characterized by a unilateral and localized distribution, early onset, and stable disease after rapid progression. And SV is often associated with poor response to various treatment modalities. We sought to evaluate the effectiveness of combination therapy with 308-nm excimer laser, topical tacrolimus, and short-term systemic corticosteroids for SV, and to search for factors associated with the treatment response. A retrospective interventional case-series study was performed on 159 patients with SV who were treated with the combination therapy for more than 3 months. The rate of 75% or more repigmentation was 50.3% after a median treatment duration of 12.1 months; 36.5% and 13.8% of the patients showed nearly complete (75%-99%) and complete (100%) repigmentation, respectively. Multivariable analysis showed the following to be independent factors withpoor response: disease duration longer than 12 months (odds ratio 0.372, 95% confidence interval 0.157-0.882, P= .025), poliosis (odds ratio 0.494, 95% confidence interval 0.247-0.988, P= .046), and plurisegmental subtype (odds ratio 0.175, 95% confidence interval 0.065-0.474, P= .001). This was a retrospective study. The combination therapy is effective for SV. Prolonged disease duration, poliosis, and plurisegmental subtype were shown to be independent prognostic factors of poor response in patients with SV.

SYMMETRY OF THEIBEXRIBBON OF ENHANCED ENERGETIC NEUTRAL ATOM (ENA) FLUX

The circular ribbon of enhanced energetic neutral atom (ENA) emission observed by the Interstellar Boundary Explorer (IBEX) mission remains a critical signature for understanding the interaction between the heliosphere and the interstellar medium. We study the symmetry of the ribbon flux and find strong, spectrally dependent reflection symmetry throughout the energy range 0.7-4.3?keV. The distribution of ENA flux around the ribbon is predominantly unimodal at 0.7 and 1.1?keV, distinctly bimodal at 2.7 and 4.3?keV, and a mixture of both at 1.7?keV. The bimodal flux distribution consists of partially opposing bilateral flux lobes, located at highest and lowest heliographic latitude extents of the ribbon. The vector between the ribbon center and heliospheric nose (which defines the so-called BV plane) appears to play an organizing role in the spectral dependence of the symmetry axis locations as well as asymmetric contributions to the ribbon flux. The symmetry planes at 2.7 and 4.3?keV, derived by projecting the symmetry axes to a great circle in the sky, are equivalent to tilting the heliographic equatorial plane to the ribbon center, suggesting a global heliospheric ordering. The presence and energy dependence of symmetric unilateral and bilateral flux distributions suggest strong spectral filtration from processes encountered by an ion along its journey from the source plasma to its eventual detection at IBEX.

Segmentally arranged hyperpigmented basaloid follicular hamartoma.

Dear Editor: Basaloid follicular hamartoma (BFH) is a rare malformation with characteristic histopathologic patterns. BFH consists of malformed hair follicles composed of anastomosing cords and strands of basaloid or squamoid cells1. Although BFH has been well described histologically, it occurs in the form of skin-colored papules, nodules, or plaques without specific clinical features1. Herein, we report a case of segmentally arranged hyperpigmented BFH in a 5-year-old girl. A 5-year-old girl presented with asymptomatic, firm, hyperpigmented, grouped papules and comedones arranged segmentally on the left lateral aspect of her nose, left retroauricular area, and posterior neck (Fig. 1A, B). The lesions appeared when she was 6 months old, and the affected area had increased since then. She had no medical or family history. A skin biopsy of the retroauricular area demonstrated multiple islands composed of basaloid and squamoid cells restricted to the upper dermis, with branching cords and anastomosing strands (Fig. 2A, B). Some anastomosing strands were connected to the epidermis. No significant clefting between the tumor and stroma was observed, nor were there any atypical cellular or mitotic features. Based on clinical and histological findings, a diagnosis of BFH was made. Given the child's age and her parents' preferences, we decided to observe the lesions rather than treat them surgically or by laser. Fig. 1 (A) Segmentally arranged, hyperpigmented, grouped papules and comedones on the left lateral aspect of the nose, (B) left retroauricular area, and posterior neck. Fig. 2 (A) Multiple islands restricted to the upper dermis with branching cords and anastomosing strands (H&E, ×100). (B) Basaloid cell proliferation admixed with squamoid cells surrounded by loose stroma and melanophages. No cleft between the ... The differential diagnosis of BFH, a benign adnexal tumor, includes infundibulocystic basal cell carcinoma (ICBCC), nevoid basal cell carcinoma syndrome (NBCCS), trichoepithelioma, and fibrofolliculoma. Of these, it is most important to rule out ICBCC, a malignant condition, to avoid unnecessary surgical excision. BFH is a superficial malformation of hair follicles and is composed of basaloid and squamoid cells, while ICBCC is composed of basaloid cells and can occasionally infiltrate into the deeper dermis, subcutaneous fat, or skeletal muscles. Additional characteristics of ICBCC include mitotic activity in the tumor cells and clefts within the stroma. Based on these histologic features, a diagnosis of BFH, rather than ICBCC, was made in this case. NBCCS is hereditary, unlike BFH, and consists of multiple basal cell carcinoma (BCC) that manifest at an early age, along with skeletal anomalies, jaw cysts, and ectopic calcification. Although the premalignant potential of BFH is currently undetermined, several cases of BCC within BFH have been reported2,3. Some evidence indicates that BFH is not a stable hamartomatous disease and that it might behave similarly to a sebaceous nevus, a lesion in which BCC can arise3. Twenty-nine cases of BFH have been reported thus far. In Korea, there have been several cases of skin-colored, localized or multiple, scattered BFH4,5. Our case is unique owing to its clinical features: hyperpigmented, segmentally, and linearly arranged lesions with unilateral distribution. Although BFH does not have well-recognized clinical features, a specific histologic pattern can be found, providing a diagnostic clue. Failure to differentiate BFH from BCC may result in aggressive surgical excision of a benign adnexal tumor. However, further studies are needed to elucidate the premalignant potential of hamartomatous lesions and the possible relationship between BFH and BCC.

Detection of Unknown sites of multiple enchondroma (Ollier′s Disease) mimicking like metastasis using bone scintigraphy

Ollier's disease characterized by multiple skeletal enchondroma is a rare noninherited disease of unknown etiology. Majority of the skeletal enchondroma are present in the metaphyses and diaphysis of tubular limb bones. Ollier's disease has a predilection for unilateral distribution. Malignant changes in Ollier's disease may occur in adult patients. Radionuclide bone scanning is one method used to assess lesions depicted on radiographs or magnetic resonance images that are presumed to be enchondromas. Furthermore, a bone scan may give a clue to the multifocality of the disease. We report a case of right first phalangeal enchondroma in a 23-year-old male, who underwent bone scintigraphy detected multifocal asymmetric right side involvement of radius, humerus, femur, and tibia which confirm a diagnosis of Ollier's disease.

Discussion of “On simulation and properties of the stable law” by L. Devroye and L. James

We contribute to the discussion of the paper by Devroye and James, by reviewing some of the most meaningful results that relate the unilateral stable distribution with the asymptotic behavior of the so-called Ewens-Pitman sampling model. Our focus is then on how these results have been exploited in the context of Bayesian nonparametric inference for species sampling problems.