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Von Hippel Lindau Disease


Von Hippel Lindau disease (VHL) is an inherited mutation of the VHL gene, which causes tumors to form in areas of the body that contain large numbers of blood vessels. According to recent medical studies, one in every 32,000 children is affected by VHL.

 

Von Hippel Lindau (VHL) disease was first described at the beginning of the 20th century by Eugen von Hippel and Arvid Lindau. A mutation of the VHL gene can affect several organs of the body, and can be expressed differently in every patient and every family. The most common manifestations of VHL include cysts and tumors of the retina, brain, spinal cord, kidney, pancreas and inner ear.

 

Patients with VHL can present with cerebellar hemangioblastomas, retinal hemangioblastomas, pheochromocytomas, kidney cancer, pancreatic cysts and pancreatic neuroendocrine tumors.

It is very important that patients and families living with VHL have their care coordinated by a team of doctors and counselors with expertise in the various aspects of their care. Proper genetic testing, appropriate screening and appropriately timed therapies are all important factors of any treatment plan for VHL.

 

Pheochromocytoma
 

The adrenal glands are located on top of each kidney.  Each adrenal gland is divided into an outer cortex and an inner medulla. The adrenal cortex is responsible for making several key steroid hormones that are important in maintaining blood pressure and salt balance, among other things. The adrenal medulla is derived from nerve cells and is a source of the catecholamines, epinephrine (adrenaline) and norepinephrine (noradrenaline). 

 

A pheochromocytoma is a tumor that derives from the adrenal medulla and makes excess catecholamines. In VHL syndrome, these tumors tend to make more normetanephrine (a breakdown product of norepinephrine). Occasionally, a catecholamine-producing tumor arises from other nerve cells located outside of the adrenal glands; in these cases, the tumor is called a paraganglioma. For simplification, the term pheochromocytoma will be used to designate catecholamine-producing tumors that arise from both within and outside the adrenal gland.

 

A pheochromocytoma will be identified in about 10-15% of VHL patients. By definition, it occurs only in individuals with VHL type II; therefore, in families with VHL type II, it can be quite common. Furthermore, pheochromocytomas are more likely to occur in more than one location (e.g. both adrenal glands) in VHL syndrome.

 

Although a pheochromocytoma is a tumor, it is rarely malignant in VHL syndrome, meaning that it is unlikely to spread to other places. If detected early, pheochromocytomas are not difficult to deal with. However, if not treated, they may be potentially lethal because of the dangerously high blood pressures that can occur with physical stressors such as accidents, surgery or childbirth.

Symptoms of Pheochromocytoma
 

A pheochromocytoma can present with classic symptoms or it may have no symptoms at all. The symptoms may resemble what one feels in an emergency (“fight or flight”) situation:

  • High blood pressure, either persistent or intermittent
  • Sweating
  • Headache
  • Rapid or irregular heartbeats
  • Feelings of anxiety/panic/fear
  • Pallor (paleness)
  • Dizziness/lightheadedness with standing
  • Tremor
  • Weight loss

Hemangioblastomas
 

Between 60 to 80% of patients with VHL develop hemangioblastomas, which occur in the cerebellum, the spinal cord and the brainstem.The average age for developing these is 33 years, although cases have been seen in patients as young as nine. Although these are benign tumors, they can cause a large number of problems, with changes in brain or spinal cord function arising out of the size or number of hemangioblastomas, or because of bleeding into the hemangioblastomas. 

 

Symptoms
 

Symptoms of hemangiolastomas vary depending on their location.

Symptoms of tumors in the cerebellum include: 

  • Difficulty walking and with muscular coordination
  • Vertigo
  • Headaches
  • Double vision
  • Vomiting

Symptoms of hemangioblastomas in the spinal cord include:

  • Decreased sensation in the arms, legs and body
  • Weakness
  • Difficulty walking
  • Difficulties with bowel and bladder function.

Brainstem hemangioblastomas can cause decreased sensation, difficulty walking, difficulties swallowing, headaches and poor coordination.

 

Kidney Cancer & VHL
 

Kidney cancer affects up to 40% of patients with VHL and is the most common cause of disease-related death in patients with VHL. Kidney cancer can arise as a single tumor in one kidney or as multiple tumors in both kidneys. The key to treatment is early detection, which can be achieved with CT scanning at regular intervals. 

 

Ocular Manifestations of VHL
 

Retinal hemangioblastomas can be seen in up to 60% of patients with VHL. The median age of onset is 25 years, although they can occur as early as one year of age. Retinal hemangioblastomas can occur anywhere within the retina (visual portion of the eye). They can occur in both eyes, and in multiple sites within the same eye. 

 

Pancreatic Manifestations of VHL
 

Pancreatic cysts and serous cystadenomas arise in between 20 to 50% of patients with VHL. The more serious pancreatic neuroendocrine tumors arise in approximately 15% of patients with VHL. The average age pancreatic manifestations are diagnosed is around 35 years of age.

 

Pancreatic neuroendocrine tumors are generally silent, but can behave like true cancers, and have the potential to spread to other organs in the body. Spread of neuroendocrine tumors usually only occurs if they become larger than three centimeters (slightly more than one inch) in size.

 

Pancreatic cysts and cystadenomas are completely benign and generally do not cause difficulties for patients with VHL. In certain cases, the cysts can become large, and can press on surrounding pancreatic tissue or other organs in the body, causing blockage of the pancreas, pain or disruption of other organs.

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Clinical trials
Ensayo de fase 1a/2a, abierto y multicéntrico, para investigar la seguridad, tolerabilidad y actividad antitumoral de dosis repetidas de Sym015, una mezcla de anticuerpos monoclonales dirigida frente al receptor MET, en pacientes con tumores malignos sólidos en fase avanzada
Estudio fase IIIB, prospectivo, randomizado, abierto que evalúa la eficacia y seguridad de Heparina/Edoxaban versus Dalteparina en tromboembolismo venoso asociado con cáncer.
Tumores sólidos. Antiemesis Estudio fase III, multicéntrico, aleatorizado, doble ciego, con control activo para evaluar la seguridad y eficacia de Rolapitant en la prevención de náuseas y vómitos por la quimioterapia (NVIQ) en pacientes que reciben quimioterapia altamente emética (QAE). A phase III, multicenter, randomized, double blind, placebo controlled study of the safety and efficacy of Rolapitant for the treatment of Chemotherapy-induced nausea and vomiting in subjects receiving highly Emetogenic Chemotherapy (HEC)
Ensayo clínico en fase I de determinación de dosis del antiangiogénico multidiana Dovitinib (TKI258) más paclitaxel en pacientes con tumores sólidos.

10 Years of Age & Under

  • Complete general history and physical exam (particularly focusing on blood pressure measurement and neurological exam) annually
  • Ophthalmologic exam at diagnosis and annually
  • Abdominal ultrasound (US) at diagnosis and then every two years
  • CT abdomen (or MRI) using VHL-specific protocol only if abnormality detected on US or abnormal pheochromocytoma screening labs
  • MRI of the brain and spine only if neurologic symptoms

Screening for pheochromocytoma (typically with plasma metanephrines and plasma catecholamines) at diagnosis and then annually in patients with a family history of VHL type 2 or mutations known to be associated with VHL 2; consider every two-year testing in patients with a family history of VHL1 or in patients with mutations not associated with pheochromocytoma; screening should always be done in patients going to surgery for any reason.

Audiology examination at diagnosis and then every two years or as needed; obtain imaging (MRI and CT) of the internal auditory canals in patients with hearing loss, tinnitus, vertigo or unexplained balance difficulties.

 
10-15 Years of Age

  • Complete general history and physical exam (particularly focusing on blood pressure measurement and neurological exam) annually
  • Ophthalmologic exam at diagnosis and then every six months (during puberty)
  • Abdominal ultrasound (US) at diagnosis and then every year
  • CT abdomen (or MRI) using VHL-specific protocol only if abnormality detected on US or abnormal pheochromocytoma screening labs
  • MRI of the brain and spine at diagnosis and then annually

Screening for pheochromocytoma (typically with plasma metanephrines and plasma catecholamines) at diagnosis and then annually in patients with a family history of VHL type 2 or mutations known to be associated with VHL2; consider every two-year testing in patients with a family history of VHL1 or in patients with mutations not associated with pheochromocytoma; screening should always be done in patients going to surgery for any reason.

Audiology examination at diagnosis and then every two years or as needed; obtain imaging (MRI and CT) of the internal auditory canals in patients with hearing loss, tinnitus, vertigo or unexplained balance difficulties.

 
15 Years of Age & Older

  • Complete general history and physical exam (particularly focusing on blood pressure measurement and neurological exam) annually
  • Ophthalmologic exam at diagnosis and annually
  • Abdominal ultrasound (US) at diagnosis and then every year from ages 15 to 20
  • MRI of the brain and spine at diagnosis and then annually (consider every two years in low risk patients)

CT abdomen (or MRI) using VHL-specific protocol starting at age 20 (or at diagnosis, if older than 20 years of age) and then every two years, alternating with abdominal US every other year; if abdominal US is not feasible, consider CT (or MRI) abdomen annually in high risk patients and every two years in low risk patients.

Screening for pheochromocytoma (typically with plasma metanephrines and plasma catecholamines) at diagnosis and then annually in patients with a family history of VHL type 2 or mutations known to be associated with VHL2; consider every two-year testing in patients with a family history of VHL1 or in patients with mutations not associated with pheochromocytoma; screening should always be done in patients going to surgery for any reason and during pregnancy.

 

Audiology examination at diagnosis and then every two years or as needed; obtain imaging (MRI and CT) of the internal auditory canals in patients with documented hearing loss, tinnitus, vertigo or unexplained balance difficulties.

 

Genetic Counseling
 

Genetic testing provides specific information on the presence or absence of mutations commonly associated with VHL. A blood sample is drawn and analyzed for the presence of abnormalities specific to VHL, and results should be interpreted in a consultation with a genetic counselor.

 

Anyone who has a parent with VHL has a 50% chance of having VHL. Genetic testing is a way to look at the VHL gene to see if it has any alterations. Genetic testing identifies who in a family is at risk for VHL, and who is not, so that screening and early detection can be focused on those who are at risk.

There is no current way to reverse the presence of the VHL mutation in patients. Nonetheless, early recognition and treatment of specific manifestations of VHL can substantially decrease complications and improve quality of life.

 

Pheochromocytoma
 

Once a pheochromocytoma is identified, it is usually treated with surgery. To prevent complications from a severe elevation of blood pressure, it is essential to take special medications for two to three weeks before surgery to prevent the effects of elevated catecholamine levels during surgery. 

 

Removal of a pheochromocytoma may require removal of the whole adrenal gland, although in some cases, the surgeon may be able to “spare” the cortex (outer layer) by removing only the tumor and the adrenal medulla. A laparoscopic approach (smaller incisions and quicker recovery) is used when feasible, although a conventional open adrenalectomy may be required. If both adrenal glands are removed for the treatment of bilateral pheochromocytoma, lifelong treatment with hormones normally produced by the adrenal cortex will be required.

 

Hemangioblastoma
 

If hemangioblastomas are small and/or slow-growing, they can potentially be observed. Larger, faster growing or symptom-causing hemangioblastomas can be removed via surgery, or can sometimes be treated with focused-beam or stereotactic radiation therapy.

 

Kidney Cancer & VHL
 

Generally it is recommended to remove suspicious kidney masses when they exceed 3 cm in size, or if they demonstrate rapid growth. In the past, patients with multiple tumors in both kidneys were fated to become dialysis dependent, as both kidneys required removal to prevent cancer progression. Recent treatment advances, including partial nephrectomy, radiofrequency ablation and cryoablation, have permitted safe and appropriate removal of tumors while saving enough normal kidney function to forestall or prevent the need for dialysis dependence.

 

If kidney cancer spreads beyond the kidney, it becomes very difficult to treat. A number of established treatments exist, including immunotherapy and chemotherapy, which help a limited number of individuals. New therapies are being developed with an emphasis on antiangiogenesis, or blood-vessel starving, and molecular targeting, taking advantage of specific tumor specific protein abnormalities to deliver therapy with higher effectiveness and lower toxicity.

 

Ocular Tumors
 

It is important that patients with retinal manifestations of VHL receive early diagnosis and treatment, as vision loss can be diminished or prevented.

Tumors found on the outer regions of the retina can be treated with laser photocoagulation (treatment with laser beams that heat up and destroy tumors), or cryotherapy (treatment with extremely cold probes that destroy the tumor). Vitrectomy, or surgical treatment, should be considered for patients with detachment and/or scarring of the retina.

Tumors in very delicate areas, like the optic disc, should be managed very carefully and usually without treatment due to the damage the treatment itself may induce on these critical regions of the eye.

 

Pancreatic Cysts & Cystadenomas
 

These tumors generally do not require treatment. In rare situations, when cysts become very large, their size can be diminished by evacuating fluid within the cyst.

Pancreatic neuroendocrine tumors require close follow up because of their potential for spread, which can result in death. Generally, if a known or suspicious pancreatic neuroendocrine tumor becomes larger than three centimeters, surgery is required to remove it.