Case: Male, 35 years old, “progressive walking difficulty for 30 years with episodic limb convulsions for more than 10 years”, gradually developed walking difficulty, abnormal posture, inversion of the left foot, unclear speech, and progressive aggravation since the age of 5. He had progressive decline in academic performance and was unable to continue his studies in late elementary school. he was unable to walk and developed dysphonia after the age of 16. he developed seizures at the age of 25 and was unable to communicate at the age of 28. Physical examination: (data at age 12) slightly poor rotation of both upper limbs, increased tone in both lower limbs, hyperactive tendon reflexes, ankle clonus +, bilateral baroreflex signs +, scissor gait. (At 35 years of age) clear, speech incompetent, severe muscle atrophy of the extremities, flexion contracture of both upper limb joints, hypertonic lower limbs. Tendon reflexes of the extremities were not elicited, and bilateral pathological signs were not elicited. Family history: non-consanguineous parents, his sister (25 years old) had similar symptoms since the age of 4. MRI showed extensive atrophy of the cerebral cortex, cerebellum and brainstem, thinning of the corpus callosum, iron deposition in the basal ganglia area, cerebral white matter dystrophy and degeneration. FA2H gene testing revealed compound heterozygous mutations. Diagnosis: hereditary spastic paraplegia type 35 (SPG35) Brief evaluation: SPG35 is a rare and complex type of hereditary spastic paraplegia and a subtype of NBIA, which is inherited in an autosomal recessive manner. Its clinical features are childhood onset, characterized by spastic paraplegia with dysarthria, moderate mental retardation associated with cerebral white matter dystrophy, some patients may also Some patients may also have dystonia, optic atrophy, ataxia, and seizures. The disease is associated with mutations in the FA2H (fatty acid 2-hydroxylase) gene. This case is the first reported case of SPG35 in China, and only 10 cases have been reported worldwide before. We confirmed a 2nd case of SPG35 soon after this family, while several SPG35 families were confirmed by other research groups in China, suggesting that the actual number of SPG35 cases in China is more than the preestimated number, which may still be mainly due to the lack of awareness of the disease. We briefly summarized the typical clinical phenotype of SPG35: disseminated or recessive family history; onset at 2-6 years of age, progressive spastic paraplegia with moderate mental retardation; need for crutch-assisted walking starting around 12 years of age, with involvement of the upper extremities; need for a wheelchair starting around 15 years of age, with difficulty in articulation and communication; seizures starting around 20 years of age, followed by prolonged bed rest, and Loss of speech function was unable to communicate, and muscle atrophy was evident, showing signs of severe dystrophy. Extensive brain atrophy, white matter dystrophy, and iron salt deposits in the basal ganglia are typical imaging findings. Confirmation of the diagnosis requires FA2H genetic testing.