Cutaneous findings in patients with acromegaly and its relationship with concomitant endocrinopathies.

OBJECTIVES: Skin changes in acromegaly are often the first sign of the disease. The aim of this study was to describe the cutaneous findings in patients with acromegaly. In addition, a secondary aim was to investigate the possible association of these findings with remission status and concomitant endocrinopathies. DESIGN, PATIENTS, AND MEASUREMENTS: In this prospective multicenter study, 278 patients over the age of 18 years with acromegaly who were followed up in 14 different tertiary healthcare institutions were included. These patients, who were followed up by the Endocrinology Department, were then referred to a dermatologist for dermatological examination. The frequency of skin lesions was investigated by detailed dermatologic examination. Dermatological diagnosis is reached by clinical, dermatological and/or dermoscopic examination, and rarely skin punch biopsy examinations in suspicious cases. The possible association of the skin findings between remitted and nonremitted patients and with concomitant endocrinopathies were evaluated. RESULTS: The most common skin findings in patients with acromegaly in our study were skin tags (52.5%), cherry angiomas (47.4%), seborrhoea (37%), varicose veins (33%), acneiform lesions (28.8%), hyperhidrosis (26.9%) and hypertrichosis (18.3%). Hypertrichosis was significantly more prevalent in patients nonremitted (p: .001), while xerosis cutis was significantly more prevalent in patients remitted (p: .001). The frequency of diabetes mellitus and hypothyroidism was significantly higher in patients with varicose veins and seborrhoeic keratosis than those without. Additionally, the coexistence of hypothyroidism, hyperthyroidism and galactorrhea was significantly higher in patients with Cherry angioma than in those without Cherry angioma (p-values: .024, .034 and .027, respectively). The frequency of hypogonadism in those with xerosis cutis was significantly higher than in those without (p: .035). CONCLUSIONS: Cutaneous androgenization findings such as skin tag, seborrhoea, acne and acanthosis nigricans are common in patients with acromegaly. Clinicians should be aware that skin findings associated with insulin resistance may develop in these patients. It can be said that the remission state in acromegaly has no curative effect on cutaneous findings. Only patients in remission were less likely to have hypertrichosis. This may allow earlier review of the follow-up and treatment of acromegaly patients presenting with complaints of hypertrichosis. Additionally, it can be said that patients with skin findings such as cherry angioma may be predisposed to a second endocrinopathy, especially hypothyroidism. Including dermatology in a multidisciplinary perspective in acromegaly patient management would be beneficial to detect cutaneous findings earlier.

A singleton pregnancy with placental chorioangioma and hydrops fetalis complicated with mirror syndrome and ritodrine-induced side effects: a case report.

BACKGROUND: Ritodrine hydrochloride is a widely used beta-adrenergic agonist used to stop preterm labor in Taiwan. Many side effects causing maternal morbidity and mortality have been reported. We report a case complicated with ritodrine-induced side effects and mirror syndrome that was associated with placental chorioangioma. CASE PRESENTATION: A 36-year-old singleton pregnant woman at 25 6/7 weeks of gestation, with an undiagnosed placental chorioangioma, underwent tocolysis due to preterm uterine contractions. Her clinical condition deteriorated, attributed to mirror syndrome and adverse events induced by ritodrine. An emergency cesarean section was performed at 27 1/7 weeks of gestation, delivering an infant with generalized subcutaneous edema. A placental tumor measuring 8.5 cm was discovered during the operation, and pathology confirmed chorioangioma. Gradual improvement in her symptoms and laboratory data was observed during the postpartum period. Identifying mirror syndrome and ritodrine-induced side effects poses challenges. Therefore, this case is educational and warrants discussion. CONCLUSION: Our case demonstrates mirror syndrome induced by chorioangioma, which is rare, and ritodrine-induced side effects. The cessation of intravenous ritodrine and delivery are the best methods to treat maternal critical status due to fluid overload.

Rare complication - skin atrophy - after systemic conservative therapy of infantile hemangioma.

BACKGROUND: Hemangiomas, also called infantile hemangiomas (IH) or hemangiomas of infancy are the most frequently seen benign vascular tumors of infancy. Different types of hemangiomas are described in the literature. The current approach is to assess the risk and, if needed, first line treatment is to initiate systemic propranolol. CASE PRESENTATION: A 3-month-old Caucasian female patient was brought as an outpatient. The main complaint was an infantile hemangioma in the facial area, which as per the parents' story appeared within a week of birth like a small reddish line and it rapidly grew. Systemic propranolol was proposed as a first-line treatment and the adverse effects were explained. The parents, afraid of the side effects, wanted to explore other possibilities such as topical timolol, however, since it had no effect, propranolol was initiated in the end. Hemangioma was completely reduced in size; however, a skin defect was detected. As per the dermatologist's counsel, topical cream was initiated. The skin defect was reduced but not fully healed. The child is still being monitored periodically. CONCLUSION: After successful treatment of hemangioma, we identified a skin defect, which was very similar to steroid-induced skin atrophy. However, we cannot attribute this to a single factor. The only thing that can be concluded is that the subject needs a thorough studying, since rate of infantile hemangioma is high, and pediatricians need a clear management strategy of how to approach skin atrophy after successfully treating the hemangioma itself.

Large, ulcerated infantile hemangioma of the chest wall complicated by life-threatening hemorrhage: Case report and literature review.

While ulceration is one of the most common infantile hemangioma (IH) complications, severe bleeding is a rare consequence, with a paucity of patients reported. We report a 5-month-old girl with a very large, mixed, partial segmental IH of the upper chest wall who, despite medical intervention, developed severe ulceration and multiple episodes of life-threatening bleeding that ultimately led to hemorrhagic shock. Experience in our patient and a review of six previous reports shows that severe bleeding is a risk when ulceration extends directly into an arterial feeding vessel that is often visible clinically. Other potential predictors for severe bleeding include large to very large IH size with extension of the tumor into underlying structures, segmental or partial segmental patterning, mixed and bulky morphology, and white discoloration as a sign of impending or worsening ulceration.

Paediatric hepatic angiosarcoma with consumptive hypothyroidism-an important diagnostic pitfall to avoid during evaluation of hepatic vascular tumours.

Hepatic angiosarcoma is an extremely rare primary malignant vascular tumour in children with very poor prognosis. Radiological diagnosis of hepatic angiosarcoma is challenging due to overlapping imaging features with other benign vascular hepatic tumours, particularly infantile hepatic haemangioma. Consumptive hypothyroidism is a condition that is almost exclusively associated with infantile hepatic haemangioma and has never been reported in angiosarcoma. We present a case of hepatic angiosarcoma in a 20-month-old girl, associated with consumptive hypothyroidism and, as a result, initially misdiagnosed as infantile hepatic haemangioma. Radiologists should be aware that consumptive hypothyroidism is not a reliable feature to use in excluding paediatric hepatic angiosarcoma. Biopsy should be performed in patients older than 1 year of age or with atypical imaging features.

Synovial hemangioma presenting with chronic painful infrapatellar mass: a case report.

BACKGROUND: Synovial hemangiomas are rare benign vascular anomalies surrounded by a synovial lining and were first described by Bouchut in 1856. These neoplasms can develop in the intra-articular region, resulting in effusions and knee pain. However, their cause remains unknown. Prompt diagnosis and intervention are critical to prevent chondral damage. Histopathological examination is used to achieve the diagnosis, which is often delayed because of a lack of specific clinical signs. This report describes a unique case in which a painful infrapatellar mass was diagnosed as a synovial hemangioma. The absence of typical magnetic resonance imaging (MRI) findings highlights the importance of arthroscopic excision for diagnosis and symptom relief. CASE PRESENTATION: A 20-year-old woman presented with persistent anterior left knee pain that became exacerbated when she climbed stairs. Despite previous pain management and physical therapy, she developed a painful lump beneath her patella that worsened over time. She had also undergone arthrocentesis, but this did not relieve her pain. Physical examination revealed a palpable, immobile 5-cm mass along the patellar tendon with limited knee flexion and extension and normal ligament stability. T1-weighted fat-saturated MRI of the left knee with gadolinium-based contrast revealed a lobulated intra-articular mass in Hoffa's fat pad that resembled a soft tissue chondroma. A biopsy of the mass was performed to provide histopathological evidence, confirming the benign nature of the mass. The subsequent excisional arthroscopy, combined with incision enlargement for mass removal, confirmed the histopathologic diagnosis of synovial hemangioma based on the presence of numerous dilated blood vessels and venous proliferation within sections of the synovium. Recovery was complete, and no residual tumor was detected on follow-up MRI after 1 year. CONCLUSION: This case study emphasizes the importance of arthroscopic excision over open surgery for patients with synovial hemangioma. The minimally invasive nature of arthroscopy combined with the well-encapsulated nature and location of the mass facilitates complete resection.

Urgent Treatment of Complicated Ulcerated Infantile Hemangioma with ß-Blockers via Telemedicine: A Case Report.

Introduction: Infantile hemangiomas (IH) exacerbated by ulceration invariably necessitate hospitalization, although simple IHs are sometimes managed remotely. Furthermore, according to international regulations, ß-blocker medication for such hemangiomas should be systemic and performed in a clinic, especially if there is infection and risk of bleeding. Case: War in Ukraine made it impossible to hospitalize and properly examine a patient with a complex ulcerated and infected IH, forcing us to administer ß-blocker timolol therapy only through telemedicine. Conclusions: Our case demonstrates the possibility of successful distant treatment of IH with ulcer using only a topical ß-blocker carried out remotely through telemedicine, which is critical in the context of the COVID-19 pandemic, war, hostilities, or natural disasters where inpatient treatment is not available.

[Stridor due to a subglottic hemangioma compressing the trachea]. / L'image du mois. Stridor secondaire à un hémangiome sous-glottique comprimant la trachée.

Subglottic haemangioma can cause stridor in young children, and sometimes be life-threatening. Larynx ultrasound is a useful, non-irradiating screening test, but the diagnosis must be confirmed by bronchial fibroscopy and injected chest CT scan. Nowadays propranolol is the first-line treatment. If treated early, the prognosis is excellent.

L'hémangiome sous-glottique peut être responsable d'un stridor chez le jeune enfant et, parfois, menacer le pronostic vital. L'échographie du larynx est un examen utile et non irradiant pour le dépistage, mais le diagnostic sera confirmé par une fibroscopie bronchique et un scanner thoracique avec injection de produit de contraste. Le traitement en première intention est le propranolol. Lors d'une prise en charge précoce, le pronostic est excellent.

Rheumatism as a cause of cardiac hemangioma: a rare case report and review of literature with special focus on etiology.

BACKGROUND: Cardiac hemangioma is a very rare benign tumor of the heart which accounts for 1-2% of all primary cardiac tumors. Multiple cardiac hemangiomas are even rarer with only three cases published in the literature. Pathologically it can be divided into cavernous hemangioma, capillary hemangioma, arteriovenous hemangioma, mixed-type hemangioma, and so on. At present, the etiology of cardiac hemangioma is not completely clear. In this study, we present multiple cardiac hemangiomas located in the right atrium and discuss the new unreported possible cause (rheumatism) of cardiac hemangioma. This is the fourth case of multiple cardiac hemangiomas in the medical literature and the first time to present rheumatism as the cause of cardiac hemangioma. CASE PRESENTATION: A 53-year-old man presented to the clinic with intermittent chest tightness and shortness of breath for 2 years. On echocardiography, multiple soft tissue masses in the right atrium were found. The patient had rheumatic heart disease with severe mitral stenosis and moderate tricuspid regurgitation. Two masses with a diameter of about 20 mm and 15 mm were seen in the right atrium. One mass was located on the inferior margin of the fossa ovalis and the other was adjacent to the inferior vena cava. Both masses were successfully removed surgically. The mitral valve replacement and tricuspid valve plasty were performed at the same time. The postoperative histopathology results confirmed the diagnosis of cavernous hemangioma. CONCLUSION: The occurrence of multiple hemangiomas in the heart is possible, especially in the presence of rheumatism. Rheumatism is one of the possible etiologies of cardiac hemangioma. Cardiologists and cardiac surgeons should be aware of its occurrence and should consider cardiac hemangioma as a differential diagnosis especially in rheumatic heart disease patients when they present with soft tissue cardiac masses for accurate management.

Skin sequelae in patients with infantile hemangioma: a systematic review.

Infantile hemangioma (IH) is the most common vascular tumor in infancy. Although IHs can regress spontaneously, some problematic IHs still need treatment. However, either treated or untreated IHs may leave skin sequelae which can cause permanent disfigurement. Many studies evaluated the short-term efficacy of different kinds of treatment, but now, few studies are focusing on long-term skin sequelae. The objectives of our systemic review were to identify skin sequelae of IH thoroughly, determine specific factors associated with long-term IH sequelae, and learn how to improve these sequelae. We searched the following electronic databases: PubMed, EMBASE, Cochrane Library, and Clinicaltrials.gov. Three independent authors assessed the articles, and we reported this systemic review following PRISMA guidelines. Of 4448 articles initially identified, 62 underwent full-text review, and 17 met inclusion criteria. The overall rate of sequelae ranged from 5.3 to 93.5%. Factors associated with skin sequelae included patients' demographics, hemangioma characteristics, and treatment factors. What is Known: • Infantile hemangioma is the most common vascular tumor during infancy. • Infantile hemangiomas can regress spontaneously but either treated or untreated patients may leave permanent skin sequelae. What is New: • Skin sequelae in involuted Infantile hemangiomas are very common. • It is significant to prevent, recognize, and improve skin sequelae of infantile hemangiomas.

Occurrence of circumscribed choroidal hemangioma accompanied by polypoidal choroidal vasculopathy and branch retinal vein occlusion: a case report.

BACKGROUND: Circumscribed Choroidal hemangioma (CCH) is a kind of hamartoma that is caused by congenital vascular malformation. And, polypoidal choroidal vasculopathy (PCV) is an exudative maculopathy. There is no literature indicating that there is a correlation between the occurrences of CCH and PCV. CASE PRESENTATION: A 66-year-old male presented with decreased vision of his left eye for 4 years. Fundus photograph showed that the branches of blood vessels at the supratemporal retina were occluded in white lines, an orange lesion could be seen in the subnasal retina and mottled, yellowish white lesions were accompanied by punctate hard exudation in the macular in the left eye. The Fundus autofluorescence (FAF), fundus fluorescein angiography (FFA), indocyanine green angiography (ICGA) and Spectral domain optical coherence tomography (OCT) were done. There was a diagnosis of CCH, PCV and branch retinal vein occlusion accompanied with retinoschisis of the left eye. CONCLUSION: This article reports on a case of an elderly male Chinese patient with CCH and PCV accompanied by branch retinal vein occlusion with retinoschisis in the left eye. The common lesions are choroidal vascular abnormalities. Whether hypertension is related to CCH, PCV and branch retinal vein occlusion remains to be further studied.

Congenital hepatic hemangioma: an unusual case report of pulmonary hypertension.

BACKGROUND: Pulmonary hypertension (PH) in newborns is a rare but serious condition that often requires immediate intervention and quick diagnosis of the correct etiology to prevent mortality. Congenital hepatic hemangioma (CHH) is an example of an extrathoracic etiology of PH. CASE PRESENTATION: Herein, we report the case of a newborn with a giant liver hemangioma, who presented with an early onset of PH and was successfully treated with intra-arterial embolization. CONCLUSIONS: This case illustrates the importance of suspicion and prompt evaluation of CHH and related systemic arteriovenous shunts among infants with unexplained PH.

Aggressive vertebral hemangiomas in children.

BACKGROUND: Aggressive vertebral hemangiomas are rare tumors in children, usually occurring in the thoracic spine that can cause significant neurological morbidity. They are technically difficult to treat with significant risk of blood loss during surgery. METHODS: We describe a case of aggressive vertebral hemangioma managed in our institution. We performed a literature review of reported cases of aggressive vertebral hemangiomas in pediatric age group. We discuss the clinical presentation, diagnosis, and management of these lesions. RESULTS: We identified 23 cases of aggressive vertebral reported in children. Neurodeficit was the most common presentation, and the most common location was the thoracic spine. Surgery was the most common modality of treatment. All the patients reported in literature had improvement in their symptoms after treatment. CONCLUSION: Although technically challenging, aggressive vertebral hemangiomas have a good outcome after treatment. Treatment should be tailored to the individual patient. Further studies are needed to determine the optimum treatment strategy.

Hybrid lung lesions in children with segmental infantile hemangiomas, a new association?

Infantile hemangiomas (IHs) are the most common tumors of infancy and, in rare instances, can present in the setting of congenital structural anomalies or as part of syndromic disorders. In this study, we present three cases of children with segmental IHs born with concurrent pulmonary anomalies: congenital pulmonary airway malformations and bronchopulmonary sequestration. To date, no known association between these entities and hemangiomas has been described.

Clinical characteristics of infantile hemangiomas with aggressive, persistent, and destructive ulceration.

BACKGROUND/OBJECTIVES: Ulceration is a common complication of infantile hemangioma (IH). Severe, persistent ulceration occurs in a minority of patients. This study aims to characterize the clinical features of IH with aggressive ulceration (AU). METHODS: Multicenter retrospective study of clinical features of IH with AU. RESULTS: Thirty-five patients with AU were identified and included in the study. The majority of AU occurred in segmental IH (23/35, 65%). Segmental IH with AU were large (≥10 cm2 ; 16/23, 69%, p < .001) with a thin (<3 mm) superficial component (16/23, 69%, p < .001). Localized IH with AU had a thick (>3 mm) superficial component (11/12, 92%, p < .001). All diaper area IH with AU (9/35) were segmental with thin superficial component (100%, p = .02). IH with AU in the head/neck (10/35) were more commonly localized (67%) and mixed (62.5%), while segmental, thick superficial morphology was more common on trunk (9/35) and upper extremities (7/35). CONCLUSIONS: IH resulting in AU differ in clinical features by anatomic site. Those in the diaper area are nearly always segmental with thin superficial component, whereas other sites tend to be localized, mixed, with thick superficial component. These distinct phenotypes may prove useful in the clinical setting for physicians to identify patterns of IH ulceration with increased risk of aggressive, persistent ulceration.

Splenic surprise in a case of renal cell carcinoma: Unusual case or association?

Littoral cell angioma (LCA) is a rare benign tumor originating exclusively from the venous sinus lining cells of the splenic red pulp. These cells are unique in having a distinctive hybrid endothelial/histiocytic phenotype. Also, there are reports of the association of LCA with internal malignancies. We present a case report highlighting an unusual association of LCA with conventional renal cell carcinoma (RCC), masquerading as a metastatic lesion. Knowledge of such an association is necessary to avoid misdiagnosis and prevent potential overtreatment.

Infantile hemangiomatosis, a rare cause of high-output heart failure and pulmonary hypertension in the newborn baby: a case report.

Infantile hemangiomatosis is among the most common vascular tumours of childhood that is generally accepted as benign. Some cases may have multiple hemangiomas with organ involvement, especially of the liver. This case report will present the clinical and laboratory findings obtained during the treatment and follow-up of a 36-day-old female baby with hemangiomatosis with diffuse liver involvement, high-output heart failure, and pulmonary hypertension.

Multidisciplinary Treatment of a Giant Maxillofacial Congenital Hemangioma With Multiple Severe Complications.

Giant congenital hemangiomas are rare, especially when combined with multiple complications. This article presents a case of a giant congenital hemangioma of the maxillofacial region in a neonate with combined thrombocytopenia, coagulation dysfunction, and heart failure, which was eventually treated surgically after a multidisciplinary consultation and achieved a good outcome.

[The complex pediatric middle ear hemangioma]. / Das komplizierte kindliche Mittelohrhämangiom.

Hemangiomas in the pediatric middle ear are a rare occurrence. We present the case of a 3-year-old boy with unilateral hearing impairment and recurrent otitis media with effusion due to a hemangioma in the middle ear. This caused a venous sinus thrombosis, osteomyelitis, and mastoiditis. Interdisciplinary treatment was performed. This case shows that even benign lesions in the middle ear can cause severe, life-threatening complications due to the close proximity of important anatomic structures in the middle ear. Every case of asymmetric hearing loss should thus be examined in detail.

Kasabach-Merritt Syndrome: a case study of successful treatment with vincristine and propranolol.

Kasabach-Merritt syndrome is a rare condition, characterised by the presence of an enlarging vascular tumour associated with thrombocytopenia, microangiopathic haemolytic anaemia and consumptive coagulopathy. The syndrome manifests in infancy, with high morbidity and mortality rates. No standard guidelines have been established for the treatment of Kasabach-Merritt syndrome to date. To existing literature we add this report of a four-month-old female child with Kasabach-Merritt syndrome who was successfully treated with propranolol and vincristine. This drug combination helped reverse the severe thrombocytopenia as well as decrease in size of her haemangioma. Management of Kasabach-Merritt syndrome continues to be a challenge, with varying response to first line drugs. Early diagnosis and initiation of treatment in a closely monitored setting is essential to ensure good outcomes. Since this is a relatively rare condition and large studies are not feasible, documenting treatment experience for single cases or small series becomes even more important.